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October 2014 Volume 32, No.3 ISSN 1391-491X

THE SRI LANKAJOURNAL OF SURGERY

The College of Surgeons of Sri Lanka

Case reports

The College of Surgeons of Sri LankaThe College of Surgeons of Sri LankaThe College of Surgeons of Sri Lanka

October 2014 Volume 32, No.3 - Quarterly. ISSN 1391-491X e - journal ISSN 2279 2201

Mission: “To reach the highest standard of scientific surgical practice by dissemination of high quality scientific information and to foster and promote the growth of scientific surgery in Sri Lanka and in the region”

Kemal I. Deen Ajith P. Malalasekera Sivasuriya SivaganeshEditor-in- Chief Assistant Editor / E-journal

Ruvini Abeygunaratne Rohan Siriwardena

Aloka A. Pathirana Chandika Liyanage

Arjuna P. R. Aluvihare S. Mandika Wijeyaratne M. D. Lamawansa

Neville D. Perera F. Ranil Fernando

Naomal M. A. Perera Pradeep A. Fernando Serozsha A. S. Goonewardena

Suren C. Paul S. J. Stephen E. D. Rodrigo

C. S. Sinnatamby

Shamika M. Abeysekara Eardley Norton Dakshitha Wickramasinghe Editorial Assistant

Pramodh Chandrasinghe

The College of Surgeons of Sri Lanka Phone : 0094- 11 - 2682290No.6, Independence Avenue Fax : 0094- 11 - 2695080Colombo 07 Email : [email protected]

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THE COLLEGE OF SURGEONS OF SRI LANKA QUARTERLY ISSN 1391-49X

EDITORS

EDITORIAL BOARD

EMERITUS EDITORS

EDITORIAL OFFICE

Journal of

The College of Surgeons

of Sri Lanka.

October 2014 Volume 32, No.3

Contents

2

Editorial

Case reports

1

4

10

12

17

Management of a traumatic atlanto-occipital and atlanto-axial subluxation with fracture of

dens Y. Sisodia, D. Gautam, A.K. Tiwari, Y. Gautam

Ewing's sarcoma of the calcaneus boneA.K. Tiwari, Y. Sisodia, Y. Gautam

Cutaneous horn over an old burn scar at external occipital protuberanceR.K. Sharma, T.P. Singh, S.K. Pabari, T.S. Dhakad

A case of perforated gallbladder with liver abscessJ.C.Y. Gan, K.K. Chan

Midgut volvulus in an adultR. Soni, A. Sinha, A. Kunal, J. Verma, Pranav, A. Anand

Laparoscopic excision of choledochal cyst K.B. Galketiya, V. Pinto, R. Perera, R. Rohankumar,U. Jayasooriya, D.S.B. Gamage, S. Aluhivare

Rupture of an inferior pancreaticoduodenal artery aneurysmC.L. Fonseka, J. Arulchelvum, R. Cassim, P. de Silva, M. Wijerathna

Pancreatic pseudoaneurysm presenting as haematemesisT. Gowribahan, S. Mehan, L. Piyarisi

Complicated large multiple amoebic liver abscessesA. Goel, N.K. Dewanda

Primary hydatid cyst of head of pancreas communicating with common bile duct containing

stones D. Thakur, U. Somashekar, R. Kothari,V. Kumar, D. Sharma

Acute bilateral parotid gland enlargement S.K. Siddegowda

Double common bile duct S.K. Sahu, S. Kumar, J. Ray

Choledochal cyst with adenoma J. Gupta

A case of dengue fever with acute appendicitis: Not dengue fever mimicking appendicitisL. Kumarasena, P. Piranavan, S. Bandara, W.P.G. Pubudu, B. Jayasundara, A. de Silva

A foreign body in the recto sigmoid region resulting in colonic perforation and concurrent

acute appendicitisR. Gupta, T.A. Mala, A. Gupta, A.K. Gupta, S.A. Malla, R. Paul

24

27

30

33

R. Abeygunarate

6

22

35

8

14

19

42

Case reports

38

46

51

Malakoplakia of colonB.D. Dhaigude, R.N. Bharadwaj, M. Singh, S. Badhranavar

Undescended testis with congenital unilateral absence of the vas deferensV.D. Kulkarni, N.A. Deshpande, P.N. Deshpande, M. Phanse

Unusual presentation of urinary ascites diagnosed by laparoscopyV. Gaikwad, A.D. Chougale, S. Murchite, R.M. Kulkarni, J. Bagwan

Neonate with congenital mesoblastic nephroma surviving respiratory failure and

pulmonary embolism at laparotomyR. Ranawaka, A. Vithanage, P. Rathnayake, K. Hettiarachchi, N. Jayakody

Laparoscopic adrenalectomy for Conn syndrome K.A.P.R. Kumarasinghe, S.K. Kollure, R. Amerasena

Umbilical endometriosis-A simple but challenging diagnosis for surgeonsK.G.H. Jayathilake , S. Withana, R. Siriwardane, G. Mahendra, C. Liyanage

Post CABG (coronary artery bypass graft) surgical site infection by Nocardia M.N. Sumana, M. Shenoy, G.R. Manjunath

49

40

44

EDITORIAL

It is with great pleasure we present to you the first case report supplement for the Journal of the College of Surgeons of

Sri Lanka. This is a first in its history and is produced due to necessity as the number of good quality and interesting

submissions have exceeded our normal quota.

Case reports provide us a glimpse into the 'weird and sometimes the not so wonderful' that we encounter in surgical

pathology and practice. These add interest to our surgical practice and writing a case report enables junior medical staff

to commence their first steps to climb to a publication platform.

We would like to encourage more and more submissions to the journal and would like to thank all the authors for

submitting these interesting cases.

Ruvini Abeygunaratne

1The Sri Lanka Journal of Surgery 2014; 32(3): 1

CASE REPORTS

Management of a traumatic atlanto-occipital and atlanto-axial subluxation with fracture of dens

Y. Sisodia, D. Gautam, A.K. Tiwari, Y. Gautam M.B.S. Hospital, Kota, Rajasthan, India.

Keywords: Atlanto occipital subluxation; atlanto

occipital dislocation; atlanto axial subluxation; Goel

and Harms technique

Introduction

Atlanto-occipital subluxation of more than 2 mm

indicates a loss of major occipitocervical stabilizers. [1,

2] Occipitocervical dislocation is associated with a fatal

brain stem injury leading to cardiac and respiratory

arrest. Most authors advocate occipitocervical fusion,

for which various methods have been described viz.

Gallie fusion, Brooks-Jenkins fusion, Sonntag posterior

C1-C2 technique, C1-2 trans-articular screw technique,

Goel's technique modified by Harm and plate-screw -

rod construct.

We treated this patient with occipital plate linked by

rods to lateral mass screw of axis vertebra and atlas

vertebra to connecting rod by circlage wire in

association with bone graft.

Case report

A 14 year old, mentally challenged female came to the

emergency department of MBS Hospital, Kota (RAJ),

with a history of fall from stairs. On examination, she

was found to have right upper and lower limb paraplegia

(MRC grade 0). Babinski's sign, well sustained clonus,

exaggerated knee & ankle jerk were evident on right

side. Initial radiographs revealed atlanto-axial

subluxation with no visualisation of the dens. Non

contast computerised tomographic scan (NCCT) of the

cervical spine (Figure. 1 and 2) revealed an atlanto-axial

and atlanto-occipital subluxation with displaced Type 2

fracture of the dens. Magnetic resonance imaging

(MRI) revealed compression at C 1 – C 2 level.

Surgical technique: Original plan for the surgery was to

The Sri Lanka Journal of Surgery 2014; 32(3): 2-32

fix the occiput, C1 & C2 in reduced position by Goel's

Technique modified by Harm. [3] Two poly axial 3.5

mm pedicle screw were fixed in th the C2 vertebra. C1 Correspondence: Y. SisodiaE-mail: [email protected]

Figure 1. Magnetic resonance image showing fracture dislocation of the dens

Figure 2. Magnetic resonance image showing subluxation of atlas bone

lateral mass screws were being put but due to excessive

bleeding from C1 - C2 epidural space patient developed

hypotensive shock and developed premature ventricular

contractions and this step was abandoned. A 'T plate'

was fixed to the occiput and connected to C2 pedicle

screws by a connecting rod. A sub laminar 20 gauge

circlage wire was passed below the centre of posterior

arch of the C1 and tightened to cross link bar between

two connecting rods and bone graft was placed (Figure.

3).

Discussion

Oda et al[4] studied the biomechanics of five different

occipitocervical fixation constructs in cadaveric spines,

and found that screw fixation from the occiput to the

pedicle of C2 was the strongest. Transarticular screws

afforded greater stability than hooks and wires. The use

of wires relies on the integrity of the posterior elements

and is associated with a risk of injuring the dura when

they are passed through the occipital burr holes or under

the lamina of the spine. The siting of transarticular

screws is technically demanding. Currently, the best

method to achieve atlanto-axial fixation is the technique

first described by Goel and popularised by Harms. [3]

We would expect our construct to function

biomechanically in much the same way as Oda's

occipitoaxial pedicle screw fixation, but with the loss of

cervical rotation.


References

1. Anderson PA. Injuries to the occipital cervical articulation.

In: Clark RC, ed. The cervical spine, 3rd ed. Philladelphia:

Lippincott-Raven, 1998: 387-399.

2. White AA, Punjabi MM. Kinematics of spine. In : White

AA, Punjabi MM, eds. Clinical biomechanics of spine, 2nd

ed. Philladelphia: JB Lippincott, 1990.

3. Harms J, Melcher RP. Posterior C1-C2 fusion with

polyaxial screw and rod fixation. Spine 2001; 26:2467-71.

4. Oda I, Abumi K, Sell LC, et al. Biomechanical evaluation

of five different occipitoatlanto- axial fixation techniques.

Spine 1999; 24:2377-82.

Figure 3. Occipito-cervical cross linkage

Key points:

Stabilisation of the occiput to cervical spine was found to be strongest with pedicle screw fixation in

cadaveric spines.

Transarticular screws provided greater stability than hooks and wires.

The downside to this technique is the loss of cervical rotation.

CASE REPORTS

Ewing's sarcoma of the calcaneus bone A.K. Tiwari, Y. Sisodia, Y. Gautam M.B.S. Hospital, Kota, Rajasthan, India.

Keywords: Sarcoma; calcaneus; Ewings

Introduction

Ewing's sarcoma is a common primary malignant bone

tumour in 1st and 2nd decade of life, usually occurring

in long bones, pelvis and ribs, with only 3-5% of cases in

the bones of the hands and feet [1]. The prognosis is poor

and this cancer is known to metastasize to the lungs and

to other bones. Treatment recommendations include

multidrug chemotherapy, radiotherapy and surgical

resection for local control of the disease.

Case report

A 12 year old boy admitted to MBS Hospital, KOTA

(RAJ.) with painful swelling of his left ankle for 6

months and fever for 3 months. Local examination

revealed a firm tender swelling affecting the left ankle

with raised temperature of the joint and normal

surrounding skin.

Blood investigations revealed elevated erythrocyte

sedimentation rate (85 mm/hr). An X-ray of the ankle

(Figure 1) showed disuse osteopenia involving all the

tarsal bones and moth eaten type of bone destruction

involving calcaneus with sunray type of periosteal

reaction. Non-contrast computerised tomography

(Figure 2) of left foot and ankle revealed moth eaten

type of destruction in calcaneus. Furthermore, the

classical sun-ray appearance of periosteal reaction

along with soft tissue component is seen. Radiological

examination of chest and pelvis was normal. Biopsy of

the swelling showed Ewing's sarcoma which was

immunohistochemistry positive for CD99. A below

knee amputation was performed.

Discussion

Ewing's sarcoma is a primary malignant bone lesion


usually seen in the diaphysis of long bones and the flat

bones of young patients, in the age group of 5 to 20

years.

Clinical and laboratory features include local pain, soft-Correspondence: A.K. TiwariE-mail: [email protected]

Figure 1. Radiograph showing a "moth-eaten" calcaneus and osteopenia

Figure 2. The "sun-ray" appearance

CASE REPORTStissue swelling and erythema, occasionally

accompanied with fever, anaemia, leukocytosis, and

accelerated erythrocyte sedimentation rate [2].

In long bones, the tumour is seen as areas of lytic

destruction in the diaphysis. The periosteal reaction can

be lamellated, parallel, spiculated, perpendicular or

mixed. Characteristic is the “onion peel” appearance. In

the early stages of the disease, it needs to be

differentiated from osteomyelitis, since both may

produce periosteal reaction with bone destruction.

Ewing's sarcoma may involve the small bones of hands,

feet and even os calcis. Since 1921, Cook has reported

29 cases of Ewing's sarcoma of the calcaneum in the

literature [3].

Dahlin et al reported 165 cases of Ewing's sarcoma, of

these, only four cases occurred in the feet [4]. Reinus et

al reported 12 cases of Ewing's sarcoma involving bones

of hands and feet out of a total of 377 patients [5].

Conclusion

Ewing's sarcoma in an atypical location may be

misdiagnosed as osteomyelitis or cellulitis.

Early recognition of an atypical appearance and the


location of Ewing's sarcoma are necessary for its

adequate treatment.

References

1. Davies AM, Makwana NK, Grimer RJ, Carter SR: Skip

metastases in Ewing's sarcoma: a report of three cases.

Skeletal Radiol 1997, 26:379-384.

2. Pritchard DJ, Dahlin DC, Dauphine RT, Taylor WF,

Beabout JW: Ewing's sarcoma: A clinicopathological and

statistical analysis of patients surviving five years of longer. J

Bone Joint Surg 1975, 57:10-16.

3. Cook M A, Manfredi O L. Ewing's sarcoma of the hand: a

case report: Bulletin Hospital for Joint Diseases 1996; Vol 55,

N° 2: 75-7.

4. Dahlin DC, Coventry MB, Scanlon PW. Ewing's Sarcoma :

a critical analysis of 165 cases. J Bone Joint Surgery 1961 ;

43-A : 185-192.

5. Reinus WR, Gilula LA, Shirley SK et al. Radiographic

appearance of Ewing sarcoma of the hands and feet : report

from the Intergroup Ewing Sarcoma Study. Am J

Roentgenology 1985 ; 144 : 331-336.

Key points:

Atypical location of Ewing's sarcoma may mimic osteomyelitis or cellulitis and may be missed.

Early diagnosis is necessary for its adequate treatment.

CASE REPORTS

Cutaneous horn over an old burn scar at external occipital protuberance

R.K. Sharma, T.P. Singh, S.K. Pabari, T.S. Dhakad Government Medical College and A.G.H. Kota, India.

Keywords: Cutaneous horn; burns; scar

Introduction

Cutaneous horn is a conical, dense hyperkeratotic

protrusion composed of compact keratin that projects

above the surface of the skin. Cutaneous horns most

frequently occur in sun-exposed parts and are typically

found on the face and scalp, but may arise from any part

of the body. They are thought to result from underlying

benign, premalignant or malignant pathology, in 61.1%,

23.2% and 15.7% of cases respectively [1].

Histopathology, especially of the base of the lesion, is

necessary to rule out associated malignancy [2].

Case Report

A 67 year old male admitted with a painless horny

growth over the back of scalp. He had suffered a flame

burn of this region at the age of 2 months. He was not

concerned about burn scar and alopecia, until seven

years prior to presentation, when he noticed a growth

over the burn scar. The growth had increased in size

gradually and formed a flat hard scab. Whenever it

reached a size of 1 x 1 cm in 10 – 12 months, it was

repeatedly removed manually by patient. In last year,

the growth had increased rapidly and the horn projected

above the surface of skin to reach the present size.

On examination he had a whitish yellow curved horn

measuring 3 cm diameter at base and 8 cm in length,

over the external occipital protuberance. A burn scar of

10 x 15 cm with areas of hypopigmentation was present

over the back of the scalp (Figure 1). Base of the horn

was nontender. Cutaneous horn was excised along with

1 cm safe margin and split skin grafting was done

(Figure 2).

The histology revealed epidermis tissue which showed


plate-like growth with expanded and anastomosing rete

ridges, acanthosis and papillomatosis. Features were

suggestive of cutaneous horn overlying a seborrheic

keratosis.

Skin grafting uptake was good and there was no

recurrence of cutaneous horn up to 6 months follow up.

Discussion

Cutaneous horn is unusual cohesiveness of keratinized

material from the stratum corneum [3]. The important

consideration in these cases is not the horn, but the

underlying pathology which may be benign (seborrheic

keratosis, viral warts, histiocytoma, inverted follicular Correspondence: T.P.SinghE-mail: [email protected]

Figure 1. Cutaneous horn over external occipital protuberence.

Figure 2. Split skin grafting after excision of cutaneous horn.

CASE REPORTSkeratosis, verrucous epidermal nevus, molluscum

contagiosum,), premalignant (solar keratosis, arsenical

keratosis, Bowen's disease) or malignant (squamous

cell carcinoma, rarely, basal cell carcinoma, granular

cell tumor, sebaceous carcinoma, Kaposi's sarcoma) [4].

No clinical features reliably distinguish between benign

and malignant lesions. However, tenderness at the base

and lesions of larger size should raise suspicion of

malignancy [5].

Sun exposure is the most important etiological factor in

pathogenesis of the cutaneous horn [4]. Radiation

exposure and HPV-2 are other risk factors.

Surgical excision of the horn including the base with

appropriate margins and careful histological

examination to exclude malignancy remains the

treatment of choice.

Conclusion

The development of a cutaneous horn over an old burn

scar is rare. The main concern is underlying malignancy,


not the cutaneous horn itself. Thus histopathogy of the

base of the horn is an essential step in the management.

References

1. Yu RCH, Pryce DW, Macfarlane AW, Stewart TW: A

histopathological study of 643 cutaneous horns; Br J

Dermatol 1991, 124:449-452

2. Korkut T, Tan NB, Oztan Y. Giant cutaneous horn: A patient

report. Ann Plast Surg 1997; 39:654-5.

3. Mencıa-Gutierrez E, Gutierrez-Diaz E, Redondo-Marcos I

et al: Cutaneous horns of the eyelid: a clinicopathological

study of 48 cases; J Cutan Pathol 2004, 31:539-543.

4. Copcu E, Sivrioglu N, Culhaci N. Cutaneous horns: Are

these lesions as innocent as they seem to be? World J Surg

Oncol 2004; 2:18.

5. Larsen F, Ricotti C Cockerell JC. Cutaneous horn. E-

medicine (webmed) 2009. Available from: URL:

http://emedicine.medscape.com/article/10 565 68

Key points:

Exposure to sunlight is an important factor in the pathogenesis of a cutaneous horn.

Tenderness at the base of a horn and a large size should arouse suspicion of malignancy.

CASE REPORTS

A case of perforated gallbladder with liver abscess J.C.Y. Gan, K.K. Chan Department of General Surgery, Hospital Sultanah Aminah, Johor Bahru, Johor, Malaysia.

Keywords: Perforated gall bladder; liver abscess

Introduction

Perforation of the gallbladder resulting in an

intrahepatic abscess is rare. This condition is commoner

when the gallbladder is completely or partially

intrahepatic [1]. It is associated with a high morbidity

and mortality and requires accurate diagnosis and

aggressive treatment [2].

Case report

A 54 year old Chinese man with diabetes and

hypertension and who had a recent attack of acute

cholecystitis two weeks ago presented with intermittent

upper abdominal pain associated with fever, chills and

rigors for one week. During his prior hospitalization, he

was treated with intravenous antibiotics and was

discharged after four days with resolving symptoms.

On admission, he appeared septic with a temperature of

38.5◦C, blood pressure of 120/70 mmHg, a pulse rate of

140 beats per minute and a capillary blood sugar level

of13.1mmol/L. On examination he was not icteric. The

abdomen was tender with localized guarding at the right

hypochondrium. There were no palpable masses at the

right hypochondrium. His blood investigation revealed

a normal total white cell count and a serum amylase

level but the serum bilirubin (22umol/L) and alkaline

phosphotase (212 U/L) were mildly elevated.

Subsequently he had an urgent abdominal ultrasound

scan performed which showed a hypoechoic lseion in

segment V of the liver measuring 4.0 x 11.3 x 6.4cm

suggestive of an abscess with a a calculus within and

ruptured gall bladder empyema. He was resuscitated

with intravenous fluid, kept nil by mouth, and

intravenous co-amoxyclav 1.2 g 8 hourly commenced.

The abscess was further assessed with CECT abdomen

prior to percutaneous drainage which showed the


collection extending into segment V (Figure 1).

Percutaneous drainage was done under ultrasound

guidance within 24 hours of admission and it drained

about 100cc of pus. The catheter was placed within the

gall bladder. His blood culture grew Kleibsiella

pneumoniae which was sensitive to the antibiotic used.

His condition improved significantly following the

drainage. An ultrasound assessment was done on day 4

of admission which revealed a distended gallbladder

with multiple calculi within but the liver abscess had

reduced in size. He then underwent an open

cholecystectomy.

Intraoperatively, the gall bladder was densely adhered to

the surrounding structures especially at the abscess

cavity and thus partial cholecystectomy was performed.

Pus from the abscess cavity when cultured yielded

growth of Kleibsiella pneumoniae and Aeromonas

hydrophila which was sensitive to imipenem. The

patient was discharged five days later following the

surgery.

Discussion

This patient had type II gall bladder perforation which

was described according to classification by Niemeier

in 1934 [3,4]. The diagnosis is confirmed by

Figure 1. Gall bladder empyema with multiple gall stonesCorrespondence: J.C.Y. GanE-mail: [email protected]

radiological imaging e.g. ultrasonography or computed

tomography with constrast study (CECT). Upon

making the diagnosis, the decision for treatment is based

mostly on the clinical features of the patient including

patient's fitness for surgery at the time of diagnosis and

the severity of the disease. Our patient underwent

percutaneous gallbladder drainage (PTGBD) as it has

been proven to be effective and relatively safe with

lower risk of morbidity and mortality compared to

emergency cholecystectomy [5]. By performing a

percutaneous drainage prior to definitive surgery in fact

provides the opportunity for optimization of a patient's

condition including the comorbidity and the possibility

of cholecystectomy via laparoscopic approach.

References

1. Peer A1, Witz E, Manor H, Strauss S. Intrahepatic abscess

due to gallbladder perforation. Abdom Imaging. 1995 Sep-


Oct;20(5):452-5.

2. Derici H, Kara C, Bozdag AD. Diagnosis and treatment of

gallbladder perforation. World J Gastroenterol.

2006;12(48):7832–36.

3. Anderson BB, Nazem A. Perforations of the gallbladder

and cholecystobiliary fistulae: a review of management and a

new classification. J Natl Med Assoc 1987; 79:393–399.

4. Ibrarullah M, Saxena R, Sikora SS, Kapoor VK, Kaushik

SP. Unusual gallbladder perforation– –definition of a new

type. Indian J Gastroenterol 1992; 11:170.

5. C-C Huang, H-C lo, Y-M Tzeng et al. Percutaneous

transhepatic gall bladder drainage: a better initial therapeutic

choice for patients with gall bladder perforation in the

emergency department. Emerg Med J. 2007 December;

24(12):836-840.

Key points:

Perforation of the gallbladder causing an intrahepatic abcess is rare.

Performing a percutaneous drainage helps to optimise a patient’s condition prior to definitive surgery.

CASE REPORTS

Midgut volvulus in an adult R. Soni, A. Sinha, A. Kunal, J. Verma, Pranav, A. Anand Department of General Surgery, VMMC and Safdarjung Hospital, New Delhi, India

Keywords: Intestinal malrotation; volvulus; Whirlpool

sign; Ladd procedure

Introduction

Midgut malrotation is a congenital anomaly referring to

either lack of or incomplete rotation of the fetal intestine

around the axis of the superior mesenteric artery during

foetal development [1] .

It has been estimated to affect approximately 1 in 500

live births [2]. Sixty four to eighty percent of cases

present primarily during the neonatal period as acute

intestinal obstruction due to volvulus of small intestine.

The presentation of intestinal malrotation in adults is

rare and occurs in approximately 0.2 to 0.6 % adult

population. [3]

Case report

A 19 year old female patient presented complaining of

pain in the upper abdomen and vomiting for 10 days.

Vomitus was bilious, contained undigested food

particles and was non projectile. On examination the

patient was mildly dehydrated, afebrile and

hemodynamically stable. The abdomen was moderately

distended with significant tenderness in the central,

epigastric and right hypochondrial regions. There was

an ill defined lump palpable in the right upper abdomen.

Routine hematological investigations were normal with

slight altered renal functions and electrolyte imbalance.

Chest and abdominal radiographs were within normal

limits. Ultrasonography of the abdomen showed

rotation of bowel loops along the axis of superior

mesenteric vein and artery in the second and third part of

duodenum, with mesenteric lymphadenopathy and mild

free fluid in pelvis. Contrast-enhanced computerised

tomography (CT) was obtained which demonstrated

features of malrotation with swirl appearance of bowel

loops starting from the second part of the duodenum


with twisting of the intervening mesenteric vessels

involving the superior mesenteric arteries and vein (

whirlpool sign ). Patient was resuscitated to restore

normal renal function. Elective laparotomy was done

which revealed two clockwise rotation of small bowel

over the third part of the duodenum, with a narrow root

of mesentry, Ladd's bands extending from posterior

peritoneum, crossing over the duodenum (and

compressing it) to the caecum and ascending colon

without any evidence of bowel ischemia. Multiple

dilated venous channels were seen in relation to the

superior mesenteric vein. There were multiple enlarged

mesenteric lymph nodes. Standard Ladd's procedure

was performed after derotating the bowel

anticlockwise. Appendectomy was also performed. At

the end of the procedure the small bowel loops were

lying to the right of the vertebral column while the large

bowel was to the left side of the vertebral column. One

large mesenteric lymph node was excised for biopsy.

Biopsy of the lymph node revealed reactive picture with

sinus histiocytosis. Patient had an uneventful post

operative recovery and was discharged on fifth post-

operative day.

Correspondence: R. SoniE-mail: [email protected]

Figure 1. Contrast-enhanced CT scan of abdomen showing “ whirlpool sign” (arrow) suggestive of midgut volvulus.

Discussion

The term volvulus is derived from the Latin word

volvere, which means to turn or roll. Midgut volvulus is

a life-threatening complication of small bowel

malrotation. It occurs early in life and is rare in adults. It

is the most common cause of bowel obstruction in an

adult with malrotation [4]. Malrotation is the

incomplete rotation of the bowel during embryological

development. As a result, the mesenteric root which

extend from the duodenal-jejunal junction ( ligament of

Treitz) to the ileal-caecal junction is shortened. The

incomplete rotation will cause the cephalic portion of

the superior mesenteric vein to lie to the left of the

superior mesenteric artery.

The ligament of Treitz is located in these cases more

inferiorly and to the right of its normal position .This

short mesenteric attachment of the small bowel can

permit rotation of the small bowel around the axis of the


superior mesenteric artery, thereby resulting in midgut

volvulus [5]. Clinical presentation of midgut volvulus

is usually nonspecific. An abrupt onset of signs and

symptoms of small-bowel obstruction in a patient

without previous abdominal surgery or other obvious

causes (hernias), preceded by colicky epigastric or

periumbilical pain several days before, should raise

suspicion for this entity. Preoperative diagnostic

workup inc ludes p l a in abdomina l x - r ay,

ultrasonography (US), abdominopelvic CT scan and

more recently, multidetector CT (MDCT) angiography.

References

1. T. Clark gamblin et. al adult malrotation: a case report and

review of the literature ; current surgery; Volume 60, Issue 5,

September–October 2003, Pages 517–520.

2. Emanuwa et al : Midgut malrotation first presenting as

acute bowel obstruction in adulthood: a case report and

literature review. World Journal of Emergency Surgery 2011,

6:22

3. Shantanu kumar sahu et al : Adult intestinal malrotation

presenting as midgut volvulus ; case report . Journal of

surgical arts, ISSN: 1308-0709 ; Vol:5 No: 1 2012

4. F. Janssens et al : Midgut volvulus in an adult patient ;

JBR–BTR, 2003, 86: 74-76

5. Berdon W.E.: Midgut volvulus with whirlpool signs. AJR,

1999, 172:1689-1690.

Figure 2.: (left) Showing twisted bowel and mesentery, multiple enlarged mesenteric lymph nodes. (right) anticlock wise derotation of bowel.

Key points:

Midgut volvulus should be suspected in adults presenting with abrupt onset of abdominal pain and signs of

intestinal obstruction in a virgin abdomen.

The imaging modality of choice is the CT scan and MDCT angiography, which can demonstrate the rotated

small bowel and mesentery, providing simultaneously information of intestinal ischaemia.

Early diagnosis and immediate operative intervention are key factors associated with a better prognosis for

this group of patients.

CASE REPORTS

Laparoscopic excision of choledochal cyst K.B. Galketiya, V. Pinto, R. Perera, R. Rohankumar,U. Jayasooriya, D.S.B. Gamage, S. AluhivareTeaching Hospital Peradeniya, Kandy, Sri Lanka.

Keywords: Choledochal cyst; laparoscopy

Introduction

Choledochal cysts are cystic dilatations of the

extrahepatic and/or intrahepatic bile ducts. They may

present with right hypochondrial pain, obstructive

jaundice and cholangitis and carry a substantial risk of

malignant transformation [1,2]. Surgical resection and

reconstruction with a Roux-en-Y hepatico jejunostomy

is the treatment of choice. This may be done by open or

minimal access. Laparoscopic resection reduces the

morbidity of open access resulting in lesser post

operative pain and respiratory problems, faster

recovery, shorter hospital stay and reduced adhesion

formation compared with open operation [1,2,3,4,5].

Case Report

A 36 year old female presented with right hypochondrial

pain and recurrent cholangitis of eighteen months'

duration. Physical examination was unremarkable.

Abdominal ultrasound scan revealed a dilated common

bile duct and hepatic ducts ; a computerised tomogram

revealed a type IV choledochal cyst (Figure-1)

Surgical resection by laparoscopy was undertaken

because of the risk of choledochal cyst malignancy in

this symptomatic patient. The patient was informed of

the possibility of conversion to open surgery if deemed

necessary.

Surgery was performed under general anaesthesia with

the patient in the supine position on the operating table.

The head end was elevated and tilted towards right. Five

ports were used (Figure 2).The ascending colon, hepatic

flexure and proximal transverse colon were mobilized

to expose the duodenum.The duodenum was

Kocherized to expose inferior vena cava and aorta. The

liver was retracted via the epigastric port and the


choledochal cyst was dissected between the common

hepatic duct and the confluence of the bile and

pancreatic ducts.The cyst was dissected off the hepatic

artery and portal vein and removed en-bloc with the gall

bladder. The proximal division was at the proximal

common hepatic duct. The specimen was retrieved

through a 5cm incision. A roux-en-Y jejunal loop was

constructed extra corporeally while hepatico-

jejeunostomy was performed laparoscopically. Total

operating time was six hours ; three hours spent for

resection and rest for reconstruction. She required

narcotic analgesics during the first twenty four hours

and discharge from hospital on the sixth post operative

day.

Discussion

Excision of choledochal cyst and roux-en-Y hepatico-

jejunostomy has two major concerns of complete cyst

excision and leak proof anastomosis. Open surgery

requires a substantial incision with siginificant potential

for post-operative complications. Laparoscopic

excision involving anastomosis with intra-corporeal

suturing is a surgical challenge.

Correspondence: K.B.GalketiyaE-mail: [email protected]

Figure 1.Computerised tomogram image showing the choledochal cyst (arrow)

The patient presented had a successful total

laparoscopic resection and reconstruction. A mini

incision was used to retrieve the specimen and to

construct the roux loop.

Extracorporeal roux-en-Y construction could shorten

the operative time [3], since this step, in our hands, took


up to two hours. During surgery the display of the

anatomy was very clear by the magnification and the

ability to zoom in with the camera. This allowed precise

dissection with minimal blood loss facilitated by the use

of bipolar diathermy and ultrasonic dissection.

References

1. Hwang DW, Lee JH, Lee SY, Song DK, Hwang JW, Park

KM, Lee YJ, Early experience of laparoscopic complete en

bloc excision for choledochal cysts in adults.Surg Endosc

2012 Nov;26(11):3324-9

2. NT Liem,Laparoscopic surgery for choledochal cysts.

Journal of Hepatobiliary Pancreatic Sciences 2013 june

;10:487-491

3. ST Tang ,Y Yang ,Y Wang, YZ Mao,SW Li, QS Tong,GQ

Cao,ZX Zhao, Laparoscopic chledochal cyst excision and

hepatico jejunostomy. Surg Endosc 2011 Feb;25(2):416-22.

4. Lü SC, Shi XJ, Wang HG, Lu F, Liang YR, Luo Y, Ji WB,

Zhao ZM, Technical points of total laparoscopic choledochal

cyst excision.Chin Med J (Engl) 2013 Mar;126(5):884-7.

5. Diao M, Li L, Cheng W, Role of laparoscopy in treatment of

choledochal cysts in children.Pediatr Surg Int 2013

Apr;29(4):317-26

Figure 2. Port sites and mini-incision covered by adhesive plaster

Key points:

Laparoscopic excision of a choledechol cyst involving anastomosis with intra-corporeal suturing is a

surgical challenge.

Minimally invasive surgery may be beneficial in reducing postoperative recovery.

CASE REPORTS

1 1 1 2 1C.L. Fonseka , J. Arulchelvum , R. Cassim , P. de Silva , M. Wijerathna 1 University Surgical Unit, The National Hospital of Sri Lanka.2 Department of Radiology, The National Hospital of Sri Lanka.

K e y w o r d s : Vi s c e r a l a r t e r y ; a n e u r y s m ;

pancreaticoduadenal artery; rupture

Introduction

Visceral artery aneurysms (VAAs) and visceral artery

pseudo-aneurysms are rare with a reported incidence of

0.01 to 0.2% at autopsy [1]. Rupture of such aneurysms

is potentially lethal as the diagnosis is often delayed.

Although recently developed imaging technology has

been able to identify these aneurysms effectively, late

diagnosis and inappropriate interventions can lead to

disastrous consequences. Of all VAAs, aneurysms of the

pancreatico-duodenal artery (PDA) are rare, accounting

for less than 2% of all VAAs [2]. We report a case of a

ruptured PDA aneurysm which presented as an 'acute

abdomen'.

Case

A 52 year old previously healthy female was admitted to

the casualty surgical unit with sudden onset severe

epigastric pain associated with sweating and feeling

faint. On examination, she was pale, and in shock

associated with generalized abdominal tenderness and

guarding. Her haemoglobin value was 6.3g/dl. An

urgent electrocardiogram showed minor ischaemic

changes, probably attributable to anaemia and

tachycardia. She was resuscitated with intravenous

crystalloids and blood transfusions to stabilize her

haemodynamic status. A focused abdominal sonogram

revealed fluid in the peritoneum. At emergency

laparotomy, a retroperitoneal haematoma at the

peripancreatic area was found. The stabilized clot was

not disturbed; the abdomen was re-sutured. Subsequent

contrast enhanced computed tomography of the

abdomen demonstrated a ruptured VAA (Figure 1), and

selective angiography revealed a pseudo-aneurysm


arising from the inferior PDA which was embolized

successfully (Figure 2,3).

A post procedure angiogram showed complete sealing

of the aneurysmal inferior PDA resulting in uneventful

recovery.

Correspondence: C.L. FonsekaE-mail: [email protected]

Rupture of an inferior pancreaticoduodenal artery aneurysm

Figure 1. Possible ruptured and /or leaking aneurysm (1.5 x 1.8cm) posterior and inferior to the head of the pancreas with possible dissection and thrombus formation.

Figure 2. Selective angiogram demonstrating the visceral artery aneurysm.

Discussion

Of VAAs, aneurysms of the pancreatico-duodenal

arteries (PDA) are most rare. These aneurysms are

associated with a mortality of 70%. While 22% of

visceral artery aneurysms present as emergencies, 8.5%

result in death [1,2]. PDAs are often associated with

pancreatitis, trauma, cholecystitis, and post-Whipple

operation. Rarely, these aneurysms are a sequel of

vasculitis. Such aneurysms are often pseudo-aneurysms

secondary to inflammation, infection, or trauma. True

aneurysms of the pancreatico-duodenal arcades are rare,

and may be congenital or acquired secondary to

fibromuscular dysplasia or atherosclerosis. The most

common clinical presentation of a non-ruptured

aneurysm is vague abdominal pain [3]. This may be

related to extrinsic compression on the duodenum or

pancreatic and biliary ducts. Patients may present with

gastrointestinal bleeding, probably secondary to rupture

of the aneurysms into the duodenum and / or the

pancreatic duct. A few patients may present with acute

abdominal pain and shock following rupture [4], which

was the presentation of our patient. Symptomatic

aneurysmal rupture is an indication for urgent

intervention.

Trans-catheter embolization has been the preferred

method of intervention over surgical resection or

ligation of the aneurysm. Depending on the location of

the VAA, a surgical approach may prove to be difficult


[5,6]. Furthermore, endovascular coil embolization has

been shown to have excellent long-term outcomes in the

treatment of these aneurysms [7].

Conclusion

For ruptured PDA aneurysms, surgery is associated with

high mortality since the point of bleeding is usually not

identified at operation. Hence, during laparotomy when

a haematoma in close relation to a ruptured visceral

artery in an unfavourable location is found, it is best to

treat it with interventional radiological procedures.

References

1. Huang Y-K, Hsieh H-C, Tsai F-C, Chang SH, Lu MS, Ko

PJ. Visceral artery aneurysm: risk factor analysis and

therapeutic opinion. Eur J Vasc Endovasc Surg

2007;33(3):293–301.

2. Ferrero E, Ferri M, Viazzo A, Robaldo A, Carbonatto P,

Pecchio A, Chiecchio A, Nessi F. Visceral artery aneurysms,

an experience on 32 cases in a single center: treatment from

surgery to multilayer stent. Ann Vasc Surg. 2011

Oct;25(7):923-35.

3.Guijt M, Delden O M van, Koedam N A, Keulen E van,

Reekers J A. Rupture of true aneurysms of the

pancreaticoduodenal arcade: treatment with transcatheter

arterial embolization. Cardiovasc Intervent Radiol.

2004;27(2):166–168.

4.Rekha Kallamadi, Marc A. deMoya, Sanjeeva P. Kalva.

Inferior Pancreaticoduodenal Artery Aneurysms in

Association with Celiac Stenosis/Occlusion. Semin Intervent

Radiol. 2009 Sep; 26(3): 215–223.

5. Bageacu S, Cuilleron M, Kaczmarek D, Porcheron J. True

aneurysms of the pancreaticoduodenal artery: successful non-

operative management. Surgery. 2006 May;139(5):608-16.

6. Cano-Valderrama O, Gallego-Béuter JJ, Giner

M.Endovascular therapy as a treatment for ruptured

pancreaticoduodenal artery aneurysms. Minim Invasive Ther

Allied Technol. 2011 Sep;20(5):296-300.

7. Tulsyan N, Kashyap VS, Greenberg RK, Sarac TP, Clair

DG, Pierce G, Ouriel K.The endovascular management of

visceral artery aneurysms and pseudoaneurysms.J Vasc Surg.

2007 Feb;45(2):276-83.

Figure 3. Another view of the selective angiogram demonstrating the inferior pancreatico-duodenal artery prior to embolization.


Key points:

Ruptured visceral artery aneurysm needs to be suspected in a patient presenting with haemorrhagic shock,

when an obvious cause is not found.

It needs prompt investigation even if the patient subsequently becomes clinically stable after a state of shock.

Regaining haemodynamic stability provides a window of opportunity which should be used effectively.

Relatively less invasive trans-arterial embolization gives easier access to surgically inaccessible sites with

aneurysmal bleeds, and it needs to be arranged urgently, as rebleeding is associated with a poor prognosis.

CASE REPORTS

Pancreatic pseudoaneurysm presenting as haematemesis T. Gowribahan, S. Mehan, L. Piyarisi Surgical unit, Sri Jayewardenepura General Hospital, Nugegoda, Sri Lanka

Keywords: Pancreas; pseudoaneurysm; haematamesis;

pseudocyst

Introduction

Pancreatic pseudoaneurysm is a permanent

communication of a pancreatic or peri pancreatic artery

with a pseudocyst [1]. Diagnosis remains challenging

due to the rarity of this condition and non specific

symptomatology[2]. Pancreatitis with pseudocyst

formation is the most common cause of pancreatic

pseudoaneurysm, although they are known to occur

following biliopancreatic resection for malignancy,

pancreatic transplant and blunt or penetrating

abdominal trauma[3].

Case Report

A 34 year old male presented with a history of epigastric

pain, episodes of haematemesis and malaena of three

months duration. Previously he had been admitted twice

for acute on chronic pancreatitis, caused by heavy

alcohol intake over 10 years. His other systems revealed

no significant complaints. Examination revealed severe

pallor, a BMI of 15, and mild epigastric tenderness

without any palpable abdominal masses. Investigation

revealed a haemoglobin of 4.4 g/dl, ultrasonography

showed pancreatic enlargement with pancreatic

calcification, and no evidence of pseudocyst or

peripancreatic collection. To find out the cause of

gastrointestinal tract bleeding both upper and lower

gastrointestinal endoscopy were performed after

optimizing the patient. In the absence of any evidence of

upper gastrointestinal bleeding, CT abdomen with

mesenteric angiogram was performed. It revealed a

2cm × 2.5cm aneurysmal filling in the uncinate process

of the pancreas. Surgical exploration was planned with

all precautions and preparations. At exploration

fibrosed pancreas was found with fragile bloods vessels.


The aneurysm was indentified in the body of the

pancreas and was explored through the aneurysmal

cavity, with the feeder artery controlled and ligated.

Pancreatic aneurysmal cavity was anastomosed with the

posterior wall of the stomach. Patient's post op recovery

was uneventful with one day of ICU observation.

Following the clinic visit after three months he remains

symptom free and his haemoglobin remains in the

normal range.

Discussion

Most patients with visceral artery pseudoaneurysm are

asymptomatic, but life threatening bleeding can occur.

High index of suspicion is needed to diagnose this

condition when patients present with non specific

symptoms. Competent interventional radiological

procedures are required to confirm and plan out the

management [4] .The wide range of treatment options

available range from minimally invasive endovascular

coil embolization[5] to open surgery. Whatever the

minimally invasive procedures attempted, preparation

for exploration should be made due to the high risk of

complications. Haemorrhage is the a main

complication, with a mortality of 13%-40%[6]. It is

almost always fatal if left unattended. Mortality rate

following surgical treatment of pancreatic

pseudoaneurysm primarily depends on the anatomical

location, with pancreatic head aneurysm mortality

being as high as 43% but only 16% in the body or tail of

the pancreas [ 2].

Conclusion

Pancreatic pseudoaneurysm is rare with non specific

symptoms, and may lead to fatal complications. High

index of suspicion is needed to diagnose the condition

especially in patients having a history of pancreatitis. In

first world countries these are managed by

interventional radiological techniques. In the Sri

Lankan context a general surgical unit with experience Correspondence: T. GowribahanE-mail: [email protected]

and team approach can successfully manage the

condition.

Reference

1. Wolstenholme JT. Major gastrointestinal hemorrhage

associated with pancreatic pseudocyst. Am J Surg. Apr

1974;127(4):377-81.

2. Tessier DJ, Stone WM, Fowl RJ, Abbas MA, Andrews JC et

al. Clinical features and management of splenic artery

pseudoaneurysm: case series and cumulative review of

literature. J Vasc Surg. Nov 2003;38(5):969-74.

3. James CA, Emanuel PG, Vasquez WD, et al. Embolization

of splenic artery branch pseudoaneurysm after blunt

abdominal trauma. J Trauma. May 1996;40(5):835-7.


4. Hyare H, Desigan S, Nicholl H, et al. Multi-section CT

angiography compared with digital subtraction angiography

in diagnosing major arterial hemorrhage in inflammatory

pancreatic disease. Eur J Radiol. Aug 2006;59(2):295-300.

5. Radeleff B, Noeldge G, Heye T, et al. Pseudoaneurysms of

t h e c o m m o n h e p a t i c a r t e r y f o l l o w i n g

pancreaticoduodenectomy: successful emergency

embolization. Cardiovasc Intervent Radiol. Jan-Feb

2007;30(1):129-32.

6. Bender JS, Levison MA. Massive hemorrhage associated

with pancreatic pseudocyst: successful treatment by

pancreaticoduodenectomy. Am Surg. Oct 1991;57(10):653-

5.

Key points:

Pancreatic pseudo aneurysm is rare with non specific symptoms and may lead to fatal complications.

Clinical suspicion should be raised in doubtful cases.

A surgical team based management is the preferred choice due to resource limitations in Sri Lanka.

CASE REPORTS

Complicated large multiple amoebic liver abscesses A. Goel, N.K. DewandaGovernment Medical College and associated group of hospitals, Kota, Rajasthan, India.

Keywords: Abscesses; liver; amoebic

Introduction

Amoebiasis is largely a disease of tropical and

developing countries and a leading cause of diarrhoeal

disease worldwide. Most common form of

extraintestinal amoebiasis is amoebic liver abscess with

right upper quadrant pain and fever [1].

Late diagnosis or presentation of amoebic abscess may

lead to perforation in about 2% of patients and amoebic

peritonitis resulting in high mortality rates [2].

We report a rare case of large multiple amoebic liver

abscesses involving almost the whole liver and rupture

of both right and left lobe abscesses which were

managed successfully.

Case report

A 29 yr old male presented with abdominal pain and

fever for 15-20 days, with loss of appetite for 5 days.

Physical examination revealed a pulse rate of 110/min, 0blood pressure of 110/70 mmHg, temperature of 37.5 C,

respiratory rate of 34/min, with decreased air entry in

right side chest.

On abdominal examination there was tenderness and

guarding in the right upper quadrant with hepatomegaly

extending 5 cm below right costal margin.

Investigations showed Hb 9.0gm%, TLC 14300/mm3,

total bilirubin 1.4mg% (normal range, 0.2 – 1.0 mg%),

aspartate aminotransferase (AST) 209 IU/L (normal

value, <40 IU/L), alanine aminotransferase (ALT) 132

(normal value, <40 IU/L), alkaline phosphatase (ALP)

406 IU/L (normal range, 100 – 306 IU/L). HIV and

HBsAg serology were non-reactive.

Ultrasound examination showed multiple liver


abscesses in both lobes of the liver, largest being 7.5 ×

6.5 cm in size.

The patient tested positive for amoebic antibody by

ELISA test. No pathogenic organism was detected on

stool examination.

Patient was started on intravenous metronidazole.

Ultrasound guided aspiration was done and typical

anchovy sauce like pus was drained (Figure 1) which

was sterile on culture for bacteria. Repeat aspirations

were done on second and fourth day.

His condition suddenly deteriorated on fifth day with

signs of tachycardia, tachypnoea and generalized

guarding. A rupture of an abscess was suspected. Urgent

computerised tomographic (CT) scan was done.

It revealed multiple liver abscesses occupying almost

the whole of the liver, largest being 10 × 8 cm in the right

lobe (Figure 2) with a right pleural effusion. Abscesses

in the right and left lobe were near the capsule with

suspicion of rupture.

Urgent exploratory laparotomy was performed. It

revealed multiple liver abscesses with the two largest

Correspondence: A. GoelE-mail: [email protected] Figure 1. Anchovy sauce like pus of amoebic liver abscess

abscess cavities in the right and left lobes ruptured into

the peritoneum. One litre of anchovy sauce like pus was

drained from the abscesses and peritoneal cavity.

All the cavities were drained and lavage done. Drains

were placed in ruptured cavities and one drain was

placed in the pelvis.

A right intercostal drainage tube was placed, which

drained 700 ml of serous fluid.

Post-operatively the patient was given intravenous

piperacillin + tazobactum and metronidazole with

supportive care.

Patient was discharged after a month of hospital stay.

Discussion

Ten percent of the world's population harbors

Entamoeba histolytica in their colon, 10% of them may

develop invasive amoebiasis and 1–10% of these

patients develop amoebic liver abscesses (ALA) [3].

Eighty percent of ALAs present as a single abscess in the

right lobe of liver [1].

In patients with multiple liver abscesses aspiration may

be needed to differentiate between amoebic and


pyogenic abscess. Aspiration has also been indicated in

patients showing lack of clinical improvement in 48-72

hours, left lobe abscess, thin rim of liver tissue around

the abscess(<10mm). Surgical intervention is indicated

in large abscesses with poor yield on needle aspiration

and complicated abscesses [4].

The preferred drug for the treatment of extraintestinal

amoebiasis is metronidazole. It is completely absorbed

orally and has a cure rate of 90% in most cases [5].

About 2-7% of ALAs are complicated by perforation.

Perforation sites mostly include pleuropulmonary

(72%), subphrenic (14%) and peritoneal cavity (10%).

Non-ruptured ALAs have a mortality rate of 4.2 – 4.8%.

When ALAs rupture the mortality reaches 23-42% [6].

Early diagnosis and prompt intervention are essential to

reduce mortality.

References

1. Sowjanya S. Mohan, Natalie C. Klein, Burke A. Cunha.

Fever of unknown origin: Multiple Amoebic Liver Abscesses

in patient with hepatitis C. Infect Dis Clin Prac 2006; 14(4):

232-234.

2. Ken J G, Van Somenberg E, Casola G et al. Perforated

Amoebic Liver Abscess: Successful percutaneous treatment.

Radiology 1989, 170: 195-197.

3. WHO Meeting: Amoebiasis and its control. Bull World

Health Organ; 1985; 63: 417-26.

4. M P Sharma, Vineet Ahuja. Amoebic liver abscess. JIACM

2003; 4(2): 107-11.

5. LeBolt SA, Jurado R, Healy GR, Shulman JA.

Hepatocellular carcinoma simulating amebic liver abscess:

report of a case and analysis of current diagnostic methods.

Am J Gastroenterol. 1985; 80:639-642.

6. Mang X Y, Wu Jx. Perforated amoebic liver abscess clinical

analysis of 110 cases. South Med J 1994; 87(10): 985-990.

Figure 1. CT Scan showing huge multiple liver abscesses and rupture of left lobe abscess


Key points:

10% of the world’s population harbours Entamoeba histolytica in their colon.

10% will develop invasive amoebiasis.

1-10% will develop amoebic liver abscesses.

Preferred antibiotic is metronidazole.

Aspiration of the abscess is the treatment of choice, if small, and surgical drainage advocated for an abscess

that does not respond to antibitoics, or a large abscess.

CASE REPORTS

Primary hydatid cyst of head of pancreas communicating with common bile duct containing stones

D. Thakur, U. Somashekar, R. Kothari,V. Kumar, D. Sharma Department of Surgery, Government NSCB Medical College, Jabalpur (MP), India.

Keywords: Echinococcosis; pancreas; obstructive

jaundice

Introduction

Primary pancreatic involvement by Echinococcus

granulosis is extremely rare [1]. Hydatid cyst of

pancreatic head containing bile and stones with a biliary

communication has never been reported.

Case detail

A 28 year female presented with dull pain in the upper

abdomen for 1 year and distension/ fullness in abdomen

and itching all over the body for 1 month. Examination

showed icterus and a palpable gall bladder. Serum

bilirubin was 4 mg/DL, serum alkaline phosphatase was

700 IU/L, Alanine transaminase was 103 IU/L and

Aspartate transaminase was 201 IU/L. Magnetic

Resonance Cholangio-Pancreaticography (MRCP)

showed a thick walled cystic lesion with some solid

component, measuring 8 X 7 X 6.1 cm at lower end of

Common Bile Duct, with internal membranes and

calcifications (Figure 1). Common Bile Duct (23mm),

Common Hepatic Duct (13mm) and Intra Hepatic

Biliary Radicals were dilated. Gall bladder showed

multiple calculi. A provisional diagnosis of Hydatid cyst

of head of pancreas was made. Hydatic serology was not

done due to its non-availability. Patient was given 400

mg of Albendazole, once a day for 3 weeks and then was

taken up for surgery.

On exploration a large cystic mass was found in the head

of pancreas, which was not fixed to surrounding

structures. Intra-operative fine needle aspiration

showed frank bile, causing suspicion of choledochal

cyst. Pancreatico-duodenal resection was done (Figure

2). Resected specimen was cut open after the procedure


and revealed a cystic lesion containing hydatid cyst

(Figure 3). It also showed a large biliary communication

Correspondence: D. SharmaE-mail: [email protected]

Figure 1. MRCP showing thick walled cystic lesion with some solid component, measuring 8 X 7 X 6.1 cm at lower end of CBD Duct, with internal membranes and calcifications; with dilated CBD and Intra Hepatic Biliary Radicals

Figure 2. Pancreatico-duodenal resection specimen showing cystic mass in the head region

with a few stones and bile contained within the cyst

(Figure 4). Histopathological examination confirmed

the diagnosis. Post-operative recovery was uneventful.

Discussion

Pancreatic hydatid disease is quite rare. Logically,

hydatid disease should be considered in the differential

diagnosis of all cystic masses in the pancreas, even if

Casoni and indirect hemagglutination tests are negative,

especially in geographic regions where the disease is

endemic. Familiarity with the imaging findings of

hydatid disease is helpful in suspecting the diagnosis

and thereby preventing potential complications [2].

Standard treatment is resection for cysts in body or tail,

while a cystectomy with drainage or pancreatico-

duodenal resection is needed for lesions in the head of

pancreas [3, 4].

A review of literature failed to reveal a previous

publication showing pancreatic head hydatid cyst


containing bile and stones with a biliary

communication.

References

1. Hamamci EO , Besim H, Korkmaz A. Unusual locations of

hydatid disease and surgical approach. ANZ J Surg. 2004

May;74(5):356-60.

2. Yuksel M , Demirpolat G, Sever A, Bakaris S, Bulbuloglu

E, Elmas N. Hydatid disease involving some rare locations in

the body: a pictorial essay. Korean J Radiol. 2007 Nov-

Dec;8(6):531-40.

3. Shah OJ , Robbani I, Zargar SA, Yattoo GN, Shah P, Ali S, et

al. Hydatid cyst of the pancreas. An experience with six cases.

JOP. 2010;11(6):575-81.

4. Khiari A , Mzali R, Ouali M, Kharrat M, Kechaou MS,

Beyrouti MI. Hydatid cyst of the pancreas. Apropos of 7

cases. Ann Gastroenterol Hepatol (Paris). 1994 May-

Jun;30(3):87-91. [Article in French] Figure 3. Resected specimen cut open to show hydatid cyst.

Figure 4. Hydatid cyst cut open showing a large biliary communication with a few stones within the cyst.

Key points:

Hydatid disease should be considered in all cystic masses of the pancreas, although rare. This is essential in

areas where the disease has a high prevalence.

CASE REPORTS

Acute bilateral parotid gland enlargement S.K. SiddegowdaGujarat University, India.

Keywords: Upper gastrointestinal endoscopy, general

anaesthesia, parotid swelling, corrosive injury

Introduction

Bilateral parotid swelling after upper gastrointestinal

procedures is a very rare occurrence. It is a self limiting

transient event following anaesthesia [1],

oesophagoduodenoscopy, and bronchoscopy [2]. The

cause for this condition is not known due to rarity of the

condition. The possible causes are retention of

secretions causing blockage of the parotid ducts,

dehydration, parasympathetic stimulation during

oesophageal intubation, venous congestion due to

excessive coughing and straining during procedure.

Knowledge of this event and the self limiting nature of

the problem is important for the treating physician. We

report two cases where bilateral parotid swelling

following upper gastrointestinal endoscopy.

Case no 1.

A 32 year old female patient admitted with corrosive

ingestion underwent diagnostic upper gastrointestinal

endoscopy. The patient did not receive any sedation or

local anesthetic spray before the procedure. The

procedure went smoothly, without any excessive cough

or any untoward incident. There was only mild

erythema of esophageal mucosa, stomach was normal

suggestive of grade I esophageal injury during

endoscopy. Few minutes following the procedure,

patient started complaining of swelling and discomfort

of the left parotid. Soon after the symtoms were present

on the right side as well. There was no respiratory

distress, swelling or crepitus in the neck. There was no

change in voice or any other systemic symptoms.

Enlargement of both parotid glands was not associated

with erythema or local rise of temperature and

tenderness (Figure 1.). There was no involvement of


Correspondence: S.K. SiddegowdaE-mail: [email protected]

Figure 1. Photographs showing left and right parotid gland enlargement.

Figure 2. No parotid swelling after 4 hours.

submandibular salivary glands. Patient was closely

monitored for any respiratory distress and laryngeal

edema. Warm compressions were given and patient kept

nil by mouth. Serum amylase level was 90U/L

immediately after the event. Ultrasonography of the

neck showed bilateral swelling of parotid with no duct

abnormality. Over a period of 2 hours the swelling

started to resolve. At the end of 4hours of the procedure

the swelling totally disappeared and the patient was

allowed oral liquids. There were no other complaints

during the hospital stay. The patient did not have any

problems during follow up period for 2 months.

Case no 2.

A 19 year old female patient who developed a stricture

following corrosive ingestion was undergoing

oesophageal dilatation regularly. In this admission

upper gastrointestinal endoscopy & dilatation was not

possible hence we took her for dilatation of oesophagus

under anesthesia. Her past medical history was

insignificant and there was no history of allergy. The

preoperative hematological and biochemical reports

were within normal limits. Under general anesthesia

patient was put in supine position and the esophagus was

dilated serially upto 12 mm under fluoroscopic

guidance using Savary Guillard dilators. Ryle's tube was

inserted under scope guidance. Duration of the

procedure was 30 minitues. After extubation, a bilateral

swelling of the parotid region was noted. The swelling

increased in size and hardness (Figure 3) .We confirmed

that the patient had no surgical emphysema, hematoma

or tracheo-bronchial injury. Patient received

chlorpheniramine and hydrocortisone intravenously.


Figure 3. Photograph showing bilateral parotid enlargement.

Figure 3. Swelling of both parotd gland disappeared after 4 hours.

The swelling reduced in size gradually over 3 hours, and

disappeared after about 4 hours (Figure 4). Ultrasound

scan of the neck showed generalized swelling of both

parotids with their ducts being normal. Serum amylase

level was 106 U/L in immediate post procedure period.

Patient was discharged with Ryle's tube in situ after 2

days. Thereafter she was on regular follow up every

month for upper gastrointestinal endoscopy &

dilatation.

Disscussion

Acute and chronic swelling of the salivary glands is

known to occur in various disorders including mumps,

postoperative parotitis, tuberculosis, amyloidosis,

a u t o i m m u n e d i s e a s e s a n d m a l i g n a n c i e s .

Noninflammatory parotid enlargement is seen more

often in association with malnutrition, obesity, and liver

disease [3]. Transient bilateral parotid swelling

following general aneasthesia (Aneasthesia mumps),

upper gastrointestinal endoscopy [4,8-9], lidocaine

spray [5], midazolam [6], Sengstaken Blakemore tube

insertion has been reported. Although the exact

mechanism is not known, possible aetiological factors

are, a) parasympathetic stimulation during the

procedure causing parotid vasodilation and transient

enlargement, b) retention of secretions causing a

blockage of the salivary ducts, c) dehydration may play

a role in causing the secretions to be thick and may

predispose salivary-duct obstruction, d) retrograde

passage of air due to a loss of muscle tone around

Stenson's orifice, e) head positioning during the

procedure may be responsible for this event [7]. Some

authors are of the opinion that it maybe due to an adverse

drug reaction such as to atropine or suxamethonium

[10,12]. However, no common drug has been used in all

of the reported cases. Matsuki et al. [11] and Attas et al.

[12] proposed that coughing and straining can also

produce salivary gland swelling and may lead to venous

congestion of the salivary glands. Bonchek [10] and

Strowbridge [13] proposed that most likely explanation

to be instrumentation of the upper airway or

oesophagus stimulates a reflex arc, with the afferent

stimulus coming from the tongue, mouth or pharynx,

and intense parasympathetic stimulation resulting in

vasodilation and transient enlargement of the glands. All

the three namely parotid, submandibular and

submaxillary salivary glands may be enlarged, but

parotid is the most commonly reported in literature.

There is no change in the serum amylase during the

parotid enlargement. In both our patient's serum

amylase was normal. Warm compression and

reassurance is all that is required for this unusual event.

Bilateral salivary gland enlargement is a transient

benign and self limiting condition but sudden

appearance of swelling after procedure is alarming to

both patient and physician [14]. The treating doctor

should be aware of this condition as he should not panic

of this rare and scary event.

References

1. Serin S, Kaya S, Kara C et al. A case of anesthesia mumps.

Anesth Analg 2007; 104: 1005

2. Blackford RW. Recurrent swelling of parotid and

submaxillary gland following bronchoscopy. Ann Otol

Rhino Laryngol 1974; 53: 54-64.

3. Roberts HJ. Difficult diagnosis – A guide to the

interpretation of obscure illness. Philadelphia: W.B.

Saunders; 1958: 19, 158, 207, 402, 412


4. Bahadur S, Fayyaz M, Mehboob S. Salivary gland swelling

developing after endoscopy: anesthesia mumps. Gastrointest

Endosc2006; 63: 345–347

5. Sagar J, Chavan VA, Shah DK. Acute parotiditis and facial

nerve palsy – unusual side effects of Xylocaine spray.

Endoscopy 2007; 39 Suppl 1: E189

6. Gültuna S, Usküdar O, Yüksel I et al. Transient parotitis

after conscious sedation for endoscopy. Endoscopy 2009; 41

Suppl 2: E53

7. Adachi Y, Suzuki K, Itagaki T, Obata Y, Doi M, Sato S. Two

cases of anesthesia mumps in the upper parotid gland after

general anesthesia in the lateral position. Masui

2008;57:1150-2.

8. Shields HM, Soloway RD, Long WB et al. Bilateral

recurrent parotid gland swelling after endoscopy.

Gastroenterology 1977; 73: 164–165

9. Ziccardi V, Molloy PJ. Parotid swelling after endoscopy.

Gastrointest Endosc 1992; 38: 520.

10.Bonchek LI. Salivary gland enlargement during induction

of anaesthesia. JAMA 1969; 209: 1716-8.

11. Matsuki A, Wakayama S, Oyama T. Acute transient

swelling of the salivary glands during and following

endotracheal anaesthesia. Anaesthesist 1975; 24: 125-8.

12. Attas M, Sabawala PB, Keats AS. Acute transient

sialadenopathy during induction of anaesthesia.

Anaesthesiology 1968; 29: 1050-2.

13. Strowbridge MNF. Acute salivary gland enlargement

following instrumentation of the upper airway. J R Army Med

Corps 1987; 133: 163-5.

14. Vinu Jamwal, Vijant Singh Chandail.Transient Swelling

of Parotid Gland after Upper GI Endoscopy - “Anaesthesia

Mumps”: Time to Stay Cool Journal of Digestive Endoscopy

2012; 3(2):30-32

Key points:

Sudden bilateral salivary gland enlargement following ,various procedures like, endoscopy, bronchoscopy,

intubation, upper GI procedures like variceal banding and esophageal dilatation can occur rarely .

The treating physician should be aware of this condition as this is often self limiting and requires no

intervention.

CASE REPORTS

Double common bile duct S.K. Sahu, S. Kumar, J. Ray Department of Surgery, Himalayan Institute of Medical Sciences, India.

Keywords: Double common bile duct, cholangitis,

cholangiogram

Introduction

A rare congenital anomaly in which two common bile

ducts exist is known as double common bile duct

(DCBD). One usually has normal drainage into the

duodenum and the other usually named accessory

common bile duct (ACBD) opens in to a different part of

the upper gastrointestinal tract (stomach, duodenum,

ductuspancreaticus or septum in the common bile duct).

The first description of this rare congenital anomaly in a

male cadaver was by Vesalius in 1543 [1].

Case

A 70 year old male presented to the surgical clinic with a

history of on and off pain in the right upper quadrant of

the abdomen which radiated to the back for the last 7-8

months. Pain was aggravated by eating fatty food. There

was no history of vomiting, jaundice or fever during

these attacks. General examination was unremarkable

with no evidence of pallor or jaundice. Abdominal

examination showed tenderness over the right

hypochondrium. Routine haematological examination

showed a haemoglobin 1of 1.6gm/dl, total leukocyte

count of 14000/mm3 with differential leukocyte count

showing 86% neutrophils. Renal function tests were

within normal limit. Liver function test showed total

bilirubin of 2.6mg/dl with direct bilirubin of 1.8mg/dl,

serum glutamic pyruvic transaminase (SGPT) of

40µkat/L, serum glutamic oxaloacetic transaminase

(SGOT) of 35IU/L and serum alkaline phosphatase

(ALP) of 250 IU/L. Ultrasonography of the abdomen

was done which showed chlolelithiasis with

cholecystitis. Common bile duct was normal.

Open cholecystectomy with per operative needle


cholangiogram was planned before exploring the biliary

tract for suspected choledocholithiasis. Per operative

cholangiogram showed a double common bile duct with

the accessory bile duct opening into the second part of Correspondence: S.K. SahuE-mail: [email protected]

Figure 1. Per operative cholangiogram showing a double common bile duct with the accessory bile duct opening into the second part of duodenum via the common ampulla of vater.

Figure 2. Per operative cholangiogram showing a double common bile duct.

the duodenum via the common ampulla of vater as the

main duct. No stones were detected in the biliary tree.

Choledochotomy which had been planned earlier was

abandoned. (Figure 1 and Figure 2). Patient had an

uneventful post operative recovery.

Discussion

Double common bile duct is a very rare anomaly in the

western world, since Teilum identified only 24 cases in

western literature until 1986 while Yamashita reviewing

Japanese literature from 1968 to 2002 and found 47

patients with this anomaly. The first case detected on

ERCP was reported by Y. Horsmans in 1966. The ratio

of male to female is approximately 1:1.6.[2,3,4]

The pathogenicity of this congenital abnormality may

be a result of inadequate or abnormal recanalisation of

the common bile duct or real duplication due to the

presence of double anlage. The embryonic development

of the liver, gallbladder system and biliary tree starts

around the third week of gestation, when the primodial

liver, designated as the hepatic diverticulum, is formed

as an outgrowth of the endoderm in the distal part of the

anterior foregut. As the hepatic diverticulum grows, its

cells penetrate the mesenchyma of the ventral

mesogastrium, dividing into a ventral and a dorsal bud.

The primitive gallbladder is formed from the ventral

bud (pars cystica). The dorsal bud (pars hepatica)

divides in turn to the left and right liver lobe. As the liver

and biliary tree develops inseparably, the stem of the

hepatic primordium becomes the bile duct. The definite

lumen of the bile tree is developed by recanalisation of

the epithelium. The anomalies of biliary tree drainage

may be due to the disruption of the diverticulum

hepaticum. [5,6,7] On reviewing the literature a series of

confusing and contradictory embrolological

classification based on the anatomical appearance of the

entity have been reported by different authors. No

clinical significance was attributed to these anatomical

classifications. Finally, this anomaly is classified with

regard to the point of drainage of the accessory common

bile duct by Yamashita et al in 2002. According to

Yamashita based on the incidence of ectopic opening of

the accessory common bile duct the classification of

positions are stomach; second part of the duodenum;

first part of the duodenum and the main pancreatic duct.

Based upon the Yamashita 2002 classification lots of


clinical conditions were associated with this rare entity.

Gastric cancer has been reported in patients with

accessory common bile duct (ACBD) opening in the

stomach where as gallbladder cancer and ampullary

cancer is associated with ACBD openings in the second

portion of the duodenum and pancreatic duct. [3,8,9]

Double common bile duct has been described in co-

existence with multiple diverticuli of the first portion of

the duodenum, absence of the gall-bladder, congenital

duodenal obstruction and annular pancreas, anomaly of

pancreatic and common bile duct junction, congenital

biliary atresia of extrahepaticbile ducts, congenital cysts

of the non-ectopic common bile duct and

cholangiocarcinoma in the duplicated bile duct. Gall

bladder carcinoma and cholangiocarcinoma of

extrahepatic biliary tree are also reported in association

with this entity. [3,10,11,12,13] Common bile duct

duplication can clinically manifest as ascholangitis,

pancreatitis, cholecystitis or hepatic abscess as a result

of reflux of pancreatic or intestinal content in to the

common bile duct. Precise preoperative recognition of

this anomaly is very difficult but can prevent surgeons

from impairing this anomalous bile duct sometimes

discovered at operation accidentally. Magnetic

resonance cholangiography as well as ERCP could

reveal the existence of this anomaly preoperatively.

[3,14] Treatment of double common bile duct depends

on the co-existence of anomalous pancreaticobiliary

junction in which the pancreatic and biliary ducts are

joined outside the duodenal wall forming a long

common channel and concomitant gastric or biliary

system cancer. In cases without cancer, the resection of

accessory common bile duct is recommended. When

anomalous pancreaticobiliary junction is present, the

separation of the flow of bile and pancreatic juice into

the gastrointestinal tract should also be performed to

prevent cancer in the biliary system. [15,16]

References

1. Vesalius A. De humanicorporisfabricalibriseptem. Lib. 5,

Cap. 8, 1543, p. 624.

2. Teilum D. Double common bile duct. Case report and

review. Endoscopy 1986; 18: 159-161.

3. Yamashita K, Oka Y, Urakami A, Iwamoto S, Tsunoda T,

Eto T. Double common bile duct: a case report and a review of

the Japanese literature. Surgery 2002; 131: 676-681.


11. Kanematsu M, Imaeda T, Seki M, Goto H, Doi H,

Shimokawa K. Accessory bile duct drainage into the stomach.

A case report and review. Gastro-intestRadiol, 1992, 17: 27-

30.

12. Yamataka A, Yana T, Hosoda Y, Segawa O., Kobayashi H,

Miyano T. A case of biliary atresia with duplication of the

common bile duct. J PediatrSurg, 2001, 36 (3): 506-507.

13. Yamamato T, Nakahira A., Kubo S, Uenishi T. et al.

Education and imaging. Hepatobiliary and pancreatic:

cholangiocarcinoma in a double bile duct. J Gastro-

enterolHepatol, 2006, 21: 1859.

14. Taourel P, Bret PM, Reinhold C, Barkun AN, Atri M.

Anatomic variants of the biliary tree: diagnosis with MR

cholangiopancreatography. Radiology 1996; 199: 521-527.

15. Tashiro S, Imaizumi T, Ohkawa H, Okada A, Katoh T,

Kawaharada Y, Shimada H, Takamatsu H, Miyake H, Todani

T. Pancreaticobiliarymaljunction: retrospective and

nationwide survey in Japan. J HepatobiliaryPancreatSurg

2003; 10: 345-351.

16. Djuranovic SP, Ugljesic MB, Mijalkovic NS, Korneti VA,

Kovacevic NV, Alempijevic TM, Radulovic SV, Tomic DV,

Spuran MM. Double common bile duct: A report of a case.

World J Gastroenterol 2007; 13(27): 3770-3772.

4. Horsmans Y, De Grez T, Lefebvre V, Witterwulghe M.

Double common bile duct with ectopic drainage of the left

lobe into the stomach. A case report and review of the

literature. ActaGastroenterolBelg, 1996, 59 (4): 256-257.

5. Kodama T, Iseki J, Murata N, Futagawa S, Sugiura M.

Wada T. Duplication of common bile duct - a case report. Jpn J

Surg, 1980, 10 (1): 67-71.

6. Nakagawara G, Kobayashi T. Duplication of extra-hepatic

biliary duct (duplication of common hepatic duct, duplication

of cystic duct, duplication of common bile duct).

RyoikibetsuShokogunShirizu, 1996, 9: 111-113.

7. Bennion RS, Thompson JE Jr, Tompkins RK. Agenesis of

the gallbladder without extrahepatic biliary atresia. Arch Surg

1988; 123: 1257-1260.

8. Goor DA, Ebert PA. Anomalies of the biliary tree. Report of

a repair of an accessory bile duct and review of the literature.

Arch Surg 1972; 104: 302-309.

9. Saito N, Nakano A, Arase M, Hiraoka T. A case of

duplication of the common bile duct with anomaly of the

intrahepatic bile duct. Nippon GekaGakkaiZasshi 1988; 89:

1296-1301.

10. Lawinksi M. A case of congenital obstruction of the

duodenum coexisting with duplication of the common bile

duct and annular pancreas. Patol Pol 1964; 15: 95-98.

Key points:

Double common bile duct is a rare entity which is very difficult to diagnose clinically and may present with

nonspecific symptoms.

Management depends on the existence of anomalous pancreaticobiliary junction or concomitant gastric or

biliary system cancer.

CASE REPORTS

Choledochal cyst with adenoma J. GuptaDepartment of General Surgery, I.G.M.C, Shimla, Himachal Pradesh

Keywords: Choledocha l cys t ; roux-en-y ;

hepatojejunostomy

Abstract

Choledochal cyst usually presents during first decade.

Unresected choledochal cyst is associated with

increased risk of cholangiocarcinoma. We report a 46

year female who was incidentally diagnosed to be

having choledochal cyst with adenoma.

Introduction

A choledochal cyst is defined as isolated focal or

combined congenital dilatation of extrahepatic or

intrahepatic biliary tree. Around 50% of the patients

usually presents during the first decade of life. Females

are more commonly affected than males (4:1).

Choledochal cyst with adenoma is a rare entity.

Case report

A forty six year old female who was investigated for

menorrhagia at the gynaecology outpatient department

underwent an ultrasound scan USS of the abdomen and

pelvis. A grossly dilated common bile duct (CBD) with a

possible intra luminal polyp was detected and the

patient was referred to the surgical unit. She had no

history of jaundice, abdominal pain or fever. On

examination she was not icteric, pale or cachectic. There

were no stigmata of chronic liver disease. Abdominal

examination was unremarkable. Complete blood count,

renal function test, serum electrolytes and liver function

tests including serum bilirubin and alkaline phosphatase

were normal. In the ultrasonograph the dilated segment

was 6.0 x 2.4 cm in size and the lower part of the CBD

was normal. Inside the dilated segment there was a soft

tissue pedunculated mass of 1.3 x0.8 cm in size

(Figure1). Intrahepatic biliary radicle and gall bladder

w e r e n o r m a l . M a g n e t i c R e s o n a n c e


cholangiopancreaticography (MRCP) showed

choledochal cyst with small pedunculated polyp (Fig.2

and 3) with non visualized distal end. Rest of the biliary

system was normal. Patient was diagnosed to be having

type Ib choledochal cyst with polyp and was scheduled

for excision of the CBD with reconstruction.

Intraoperative findings confirmed the observations

made on imaging. Excision of cyst with Roux –en y

hepatojejunostomy was done. Post operative period was

uneventful. Inside the choledochal cyst was a

pedunculated polyp. On histological evaluation the cyst

was lined by a benign columnar epithelium, with

Correspondence: J. GuptaE-mail: [email protected]

Figure 2. MRCP showing pedunculated polyp in choledochal

Figure 1. USG showing polyp in CBD

thickening and fibrosis of the wall. Polyp showed

features of ademomatous polyp with no evidence of

dysplasia.

Discussion

Choledochal cyst (CDC) was initially described by

Douglas in 1852. Anatomical classification proposed by

Alenso et al was revised by Todani et al in 1977 [1].

Type I is the commonest type commonest occurring in

75% of cases. Classic symptom complex of abdominal

pain, mass and jaundice is uncommon and is observed in

only 20-30% patients. There may be recurrent

abdominal pain with minimal jaundice, which may not

be readily apparent as in our case. Imaging techniques

help in the diagnosis of choledochal cyst. USS helps in

diagnosis of 93% of paediatric and 72% of the adult

p o p u l a t i o n . M a g n e t i c r e s o n a n c e

cholangiopancreaticography is the non invasive

procedure of choice at present. CDC is associated with

abnormal pancreatic and bile duct union (anomalous

biliopancreatic junction) in 90% of cases. Differential

diagnosis is biliary stricture. In contrast to type I cyst

obstructing lesion will often cause elevated alkaline

phosphate and bilirubin. Various complication of CDC

are cholangitis, gall stones, jaundice, pancreatitis,

portal hypertension and cholangiocarcinoma. Various

malignancies are cholangiocarcinoma, gall bladder


carcinoma, adenoma and bile duct sarcoma. Benign

tumours of extrahepatic biliary tree rare , they account

for only 6% [3]. Bile duct adenoma, is a rare neoplasm

of the extrahepatic biliary tree and very few cases have

been reported in a choledochal cyst[4]. Although this

tumor is benign, it is considered to have a malignant

potential[5]. In MRCP a stone as well as tumour will

appear as dense shadow. A stone will be detected in a

dependent part while a polyp can be in the non

dependent area. Lifetime risk of cholangiocarcinoma in

CDC paediatric population is 2.3 % . In older untreated

patients the reported incidence is as high as 75 %.

Speculated etiological mechanism of carcinogenesis is

bile stasis, reflux of pancreatic juices mixed with bile,

changes in bile acids, superinfection or inflammation.

There is pathological evidence of hyperplasia-dysplasia

and cancer in patients with pancreaticobiliary

malignancy. Hyperplastic cells have increased cellular

proliferation markers cyclooxygenase-2, vascular

endothelium growth factor and k-ras mutation. Excision

o f g a l l b l a d d e r , C D C w i t h R o u x - e n - Y

hepaticojejunostomy is the the most commonly used

surgical procedure.

References

1. Todani T, Watanabe Y, Narusue M, et al. Congenital bile

duct cysts : classification, operative procedures and review of

37 cases including cancer arising from choledochal cyst. Am J

Surg1977;134: 263-69.

2. Parikh PY, Lilemoe KD. Choledochal cysts and benign

biliary strictures. In: Maingot's Abdominal operations. 12th

ed 1032

3. Dowdy GS, Olin WG, Shelton EL et al. Benign tumors of

the extra-hepatic biliary ducts: report of 3 cases and review of

literature. Arch. Surg. 1962; 85 : 503–13.

4. Aggarwal S, KumarS, kumar A, et al. Extrahepatic bile duct

adenoma in a patient with choledochal cyst. J Gastroenterol

and Hepatol 2003; 18 : 351–55

5. Davies W, Chow M, Nagorney D. Extra-hepatic biliary

cystadenomas and cystadenocarcinomas. Report of seven

cases and review of the literature. Ann. Surg. 1995; 222

Figure 3. MRCP showing filling defect in non dependent part


Key points:

Lifetime risk of cholangiocarcinoma in a paediatric choledochal cyst population is 2.3%

In older untreated patients the reported incidence is as high as 75 %.

Ultrasound is used in the diagnosis, but magnetic resonance cholangiopancreaticography is the non invasive

procedure of choice.

CASE REPORTS

A case of dengue fever with acute appendicitis: Not dengue fever mimicking appendicitis

L. Kumarasena, P. Piranavan, S. Bandara, W.P.G. Pubudu, B. Jayasundara, A. de SilvaNational Hospital of Sri Lanka

Keywords: Dengue; appendicitis

Introduction

Dengue fever (DF) is endemic in Sri Lanka with more

than 44,000 reported cases in 2012. It is a common cause

for an acute febrile illness and clinical profile ranges

from self limiting fever to dengue shock syndrome.

Other than for the febrile illness, DF may rarely present

as apparent acute abdominal surgical emergency such as

acute cholecystitis, acute pancreatitis, and acute

appendicitis (AA). Published literature provides limited

number of reports of acute abdominal problems

mimicking DF. [1-4] Acute appendicitis is a common

condition which can get complicated with perforation,

peri-appendicial abscess formation, peritonitis and

rarely death. [5] We report the experience with an

adolescent presented as having AA which was

confirmed peri-operatively as well as histologically,

who also had evidence of dengue hemorrhagic fever

(DHF) during post-operative period. To the best of our

knowledge this is the first case of simultaneous AA and

DHF, which created a diagnostic and management

dilemma.

Case report

A twelve and a half year old boy was admitted to a

medical casualty unit with fever and abdominal pain of

one day duration. He was then referred to the surgical

team with suspicion of AA. At surgical evaluation the

patient had right sided lower abdominal pain and fever

for 24 hours. He denied of migrating abdominal pain. He

had anorexia, nausea and vomiting but no urinary 0symptoms. He was febrile (100.5 F), had a regular pulse

rate of 96 beats per minute and was haemodynamically

stable. There was no flushing of his body. There was


evidence of localized peritonism at right iliac fossa with

guarding and rebound tenderness. His urine analysis

was normal and white cell count (WCC) was 7.2 × 106/l

with 79% neutrophils and 18% lymphocytes. Platelet

count was 195 × 109/l. With clinical evidence of AA

supported by an Alvarado score of 7/10, patient

underwent open appendicectomy. Surgical procedure

was uncomplicated and the appendix was found to be

inflamed. He was given diclofenac Na 25mg

suppository at recovery, followed by regular oral

paracetamol and subcutaneous morphine for pain relief.

Intravenous cefuroxime and metronidazole was

continued for a maximum of three doses. Initial 12 hours

of the post-operative period was uncomplicated and the

child was commenced on oral feeding. Twenty four

hours after appendicectomy, the patient had evidence of

increased localized peritonism which gradually

progress to be generalized. He developed a tachycardia

of 110 beats per minute but the blood pressure was

stable. There was no skin rashes noted. Repeated WCC

was 3.4 × 106/L with 78% neutrophils and 18%

lymphocytes. Platelet count was 147 × 109/L. C-

reactive protein (CRP) level was 23mg/L (normal value

<6). Due to worsening peritonism with a suspicion of

inadvertent bowel injury or ligature slip the patient had

re-exploration through the previous incision 36 hours

after appendicectomy. The second exploration was

negative and there was no excessive bleeding from the

incision. Abdomen was closed after a peritoneal lavage.

Subsequently the patient developed further

thrombocytopenia and neutropenia around 72 hours

from the onset of initial complain, leading to the

possible diagnosis of DHF. At this stage the diagnosis

was confirmed with a positive Dengue NS1 antigen test.

By fifth day of fever he had the maximum

thrombocytopenia of 46 × 109/L without bleeding

manifestations. He had elevated liver transaminase Correspondence: B. JayasundaraE-mail: [email protected]

levels. The patient was managed at critical care setting

with inputs from both medical and surgical teams. He

did not require transfusion of blood products. From

sixth day, clinical and biochemical parameters did

improve and the patient was discharged on day 12, with

normal WCC, platelet count and liver enzyme levels.

Histological evaluation confirmed AA with transmural

neutrophil infiltration of the appendix.

Discussion

Although self limiting abdominal pain and

manifestations like nausea and/or vomiting, diarrhoea

are seen in dengue infection, DF presenting as acute

abdomen is rare. Published literature cumulatively

report 21 cases of DF presenting as AA, and six patients

have undergone appendicectomy. [1-4] Microscopic

evaluation of these specimens showed lymphocytic

infiltration, lymphoid hyperplasia or normal

microscopy. None of these patients had histological

evidence of acute appendicitis and this patient is

probably the first to have microscopic evidence of

supurative AA. Exact mechanism of acute abdomen in

DF is not known. Wu et al postulated cholestasis,

endotoxemia, microangiopathy, ischaemia and

reperfusion injury as possible pathogenic mechanisms

for acute acalculous cholecystitis in DF [6]. With the

limited number of cases reported, it is still early to apply

similar postulations for apparent AA in DF.

Diagnosis of AA and management decision making is

mainly on clinical judgment. Clinical diagnosis of

appendicitis is also supported by raised inflammatory

markers. Alvarado provided a scoring system which

counted elevated WCC and left shift in addition to

clinical parameters, and most surgeons adapt this

system. [7] Despite the general expectation of elevated

serum markers like raised WCC and CRP, there are

reports of gangrenous appendicitis with normal serum

markers. [8, 9]

This patient highlights the dilemma created by AA with

DF on patient management for both surgical and

medical teams. Suspicion of DF was delayed until the

second exploration as patient had classical features of

appendicitis which was apparent at first surgery.

Previous cases of acute abdomen with DF which were

surgically intervened, required substantial amount of

blood/ blood product transfusion. [1, 3] The index


patient did not develop such complications. It was

unclear whether this patient had simultaneous

appendicitis and dengue fever as two different

presentations of dengue virus, or as a dual pathology.

Previous authors, who have managed the cases of DF

mimicking AA, have commented on the need to

differentiate the disease early to avoid unnecessary

surgical interventions. We highlight the occurrence of

true intra-abdominal inflammatory process of

appendicitis in the index case, which could have been a

rare presentation in DF either as a part of dengue virus

infection or as a concurrent dual pathology.

Acknowledgements

We acknowledge the valued contribution of University

medical team for their input in the patient management,

Dr Janakie Fernando and her team for the histological

assessment of the resected specimen and Dr Disna de

Silva for her advices in the preparation of the article.

References

1. Khor BS, Liu JW, Lee IK, Yang KD. Dengue hemorrhagic

fever patients with acute abdomen: clinical experience of 14

cases. American Journal of Tropical Medicine and Hygiene

2006; 74: 901–4.

2. Premaratna R, Bailey MS, Ratnasena BGN, de Silva HJ.

Dengue fever mimicking acute appendicitis. Transactions of

Royal Society of Tropical Medicine and Hygiene 2007; 101:

683–5.

3. Shamim M. Frequency, Pattern and Management of Acute

Abdomen in Dengue Fever in Karachi, Pakistan. Asian

Journal of Surgery 2010; 33: 107–13.

4. Kang YJ, Choi SY, Kang IJ et al. Dengue fever mimicking

acute appendicitis: a case report. Infection and Chemotherapy

2009; 41: 236-9.

5. Simpson J, Samaraweera AP, Sara RK, Lobo DN. Acute

appendicitis – a benign disease? Annals of Royal College of

Surgeons of England 2008; 90: 313–6.

6. Wu KL, Changchien CS, Kuo CM et al. Dengue fever with

acute acalculous cholecystitis. American Journal of Tropical

Medicine and Hygiene 2003; 68: 657–60.

7. Alvarado A. A practical score for the early diagnosis of

acute appendicitis. Annals of Emergency Medicine. 1986; 15:

557-64.

8. Vaughan-Shaw P, Richardson C, Lewis M. White cell count

and C-reactive protein measurement in patients with possible

appendicitis. Annals of Royal College of Surgeons of England

2011; 93: 183


9. Vaughan-Shaw PG, Rees JR, Bell E et al. Normal

inflammatory markers in appendicitis: evidence from two

independent cohort studies. Journal of Royal Society

Medicine Short Reports 2011; 2: 43.

Key points:

Always consider acute appendicitis, when appropriate, with presentation of other disease

Alvarado provided a scoring system which counted elevated total leucocyte count and left shift in addition to

clinical parameters, and most surgeons adapt this system.

CASE REPORTS

A foreign body in the recto sigmoid region resulting in colonic perforation and concurrent acute appendicitis

1 1 1 1 2 3R. Gupta , T.A. Mala , A. Gupta , A.K. Gupta , S.A. Malla , R. Paul1 Department of Paediatric Surgery, SPMCHI, SMS Hospital, Jaipur Rajasthan, India.2 S.S Medical College, Rewa (M.P) India.3 Jaipur Rajasthan, India.

Keywords: Foreign body; recto sigmoid; pen,

perforation; acute appendicitis.

Abstract

A child with a foreign body lodged in the rectoaigmoid

region resulting in colonic perforation and concurrent

acute appendicitis is presented. During clinical

evaluation, there were no signs of generalized

peritonitis; abdominal sonography was performed to

evaluate for acute appendicitis. An erect abdominal x-

ray showed a radio opaque shadow in the right iliac

fossa. At laparotomy, the tip of a pen was seen to

perforate the sigmoid colon and impinge on the base of

the appendix which was grossly inflamed.

Subsequently, the pen was removed from within the

colon and the colon was repaired in two layers. An

appendicectomy was performed.

Introduction

Foreign bodies in the rectum cause complications and

present a challenge to clinical management. They

should be treated expeditiously.

Case report

A 9-year-old male child presented with complaints of

pain in the right iliac fossa and anorexia for three days.

On clinical examination, he was hemodynamically

stable and afebrile, with marked tenderness in the right

iliac fossa and rebound tenderness. Rectal examination

was unremarakable. There were no signs of generalized

peritonitis. Laboratory investigations revealed a total

leukocyte count of 11,900 /cu mm with 80%

polymorphonucleocytosis. Abdominal ultrasonography

seemed to support the diagnosis of acute appendicitis.


Furthermore, an erect abdominal x-ray showed a radio

opaque shadow in the right iliac fossa [Figure 1]. There

was no free air. A clinical diagnosis of acute appendicitis

was made and patient was scheduled for

appendicectomy – the presence of a colonic foreign

body was not suspected.

During exploration of the abdomen through a Lanz

incision, we found the tip of a pen to be present at the

incision and formal laparotomy was performed by

elongating the incision. The pen was seen perforating

the rectosigmoid colon and touching the base of the

appendix which was grossly inflamed [Figure 2]. The

perforation, which was sealed by the pen, was one cm in

length, and did not leak faecal content. The pen was

carefully removed from the site of perforation which

was repaired in two layers. The remainder of the bowel

was inspected to look for any other perforation. The

appendix, which was found to be grossly inflamed, was

excised and submitted for histopathological

examination. The abdomen was closed in layers.

Correspondence: T.A. MalaE-mail: [email protected] Figure 1. showing radio opaque shadow in the right iliac fossa

Postoperative recovery in this patient was uneventful.

On further enquiry, the patient denied either swallowing

or anal introduction of the pen. Histopathological report

of appendectomy specimen confirmed acute

appendicitis [Figure 3]. He was subsequently referred

for psychometric assessment.

Discussion

The majority of reports of foreign bodies within the

rectum are from the West [1-8], varying in age from 16

to 80 years [1]. There is a bimodal age distribution,

observed in the twenties for anal erotism or forced

introduction through anus, and in the elderly, mainly for

prostatic massage or disimpaction of faeces. Reports

have been more common in males compared with

females [1.2]. Our case involved a 9-year old boy and is

unusual. In general, an object is inserted into the rectum

by the patient or partner for sexual gratification which

may not be retrievable. Abdominal and rectal pain,

rectal bleeding and urinary symptoms are the common

presenting symptoms. In our case, complaints were pain

in the right iliac fossa along with anorexia reflecting on

acute appendicitis.

Impacted rectal foreign bodies commonly reported were

plastic or glass bottles, vegetables, such as, cucumber

and carrot, wooden and rubber objects [2]. In our case

the foreign body was a ballpoint pen about 15cms in

length. Richter also reported ballpoint pen rectal

impaction in his study [8]. Although the abdominal x-

ray revealed a radio-opaque shadow in the right lower


quadrant of the abdomen, our attention was diverted by

the clinical status of acute appendicitis, completely

missing the thought of a possible foreign body

impaction. A majority (90%) of cases without

perforation are treated by transanal retrieval of the

foreign body [1,2,6,7]. Laparotomy is only required

where a foreign body cannot be retrieved by

conventional trans-anal methods and in the case of

perforation. To the best of our knowledge, there has not

been a report of acute appendicitis, proven by

histological examination, with a concurrent foreign

body penetration of the rectosigmoid colon in the world

literature.

References

1. Biriukov IuV, Valkov OV, An VK, Elu B, Dodina AN.

Treatment of patients with foreign body rectum (Russian)

Khirurgiia. 2000;7:41–3. [PubMed]

2. Subbotin VM, Davidov MI, Abdrashitov RR, Rylov IuL,

Sholin NV. Foreign bodies in rectum. Vestn Khir Im I I Grek.

2000;159:91–5. [PubMed]

3. Gaponov VV, Foreign bodies in the rectum and colon

(Russian) Klinicheskaia Khirugiia. 1992;2:37–40.

4. Neprasova P, Treska V, Simanek V. Injury of rectum with

porcelain cup (Czech) Rozhledy V Chirurgi i .

2001;80:128–30. [PubMed]

5. Batho G, Szanto L. Foreign bodies in rectum at our

department during last ten years. (Hungarian) Magyar

Sebeszet. 2000;53:180–2. [PubMed]

6. Petrolito E, Bracchitta S, Calabrese C, Riolo G, Donati A,

Pecorella G. Foreign bodies and injuries of the rectum

(Italian) Minerva Chirurgica. 1989;44:867–71. [PubMed]

7. Kouraklis G, Miaiakos E, Dovas N, Karatzas G, Gogas J.

Management of foreign bodies of the rectum: Report of 21

cases. J R Coll Edin. 1997;42:246–7.

8. Richter RM, Littman L. Endoscopic extraction of an

unusual colonic foreign body. Gastrointest Endosc 1975; 22:

40

Figure 2. The tip of a pen seen perforating the sigmoid colon.

Key points:

Presentation of acute appendicitis with a foreign body perforation, a rare occurence by a foreign body.

CASE REPORTS

Malakoplakia of colonB.D. Dhaigude, R.N. Bharadwaj, M. Singh, S. BadhranavarDepartment of General Surgery, Pad. Dr. D.Y Patil Medical College Pimpri, Pune, India

Keywords: Gastrointestinal system; macrophages;

histocytes ; genitourinary system.

Introduction

Malakoplakia, first described by Michaelis and

Gutmann in 1902 [1], occurs mainly in the genito-

urinary tract (60% of cases), and less frequently in the

gastrointestinal tract (10% of cases) [2]. Although the

pathogenesis of malakoplakia is not completely

understood, the disease may be considered the result of

an impairment of the mononuclear phagocyte and

immunoregulatory effector system [3]. The foci of

malakoplakia characteristically consist of proliferation

of histiocytes with the typical cytoplasmic inclusions

known as Michaelis-Gutmann bodies that probably

correspond to lysosomes. This rare condition may be

associated with inflammatory and infectious diseases,

immunosuppressive therapy such as steroids and with

tumours such as colorectal carcinoma [4].

Case report

An 80 year old patient presented with intermittent pain

in right side of abdomen for two years. Bowel and

bladder habits were normal. He had diabetes mellitus

and hypertension for ten years. There was no history of

use of immunosuppressant medication. On examination

there was a non-tender intra-abdominal lump 8x5 cms.

in diameter and palpable in right lumbar region.

Computerised tomographic scan of the abdomen

revealed right colonic inflammatory pathology with

extension into the right anterior pararenal space and

abscess formation in the retroperitoneum along the

anterolateral aspect of the right psoas and quardratus

lumborum muscles ; ultrasound guided needle

aspiration confirmed the abscess. Staining was negative

for acid fast bacilli.


At laparotomy we found an 8 x 8 x 5 cm firm to hard

mass involving the ascending colon and right half of

transverse colon (Figure 1). A right hemicolectomy was

performed to remove the mass and part of the abdominal

wall involved by the inflammatory mass. An ileocolic

anastomosis was performed to complete the procedure.

Furthermore, the retroperitoneal space posterior to

ascending colon was fluctuantand revealed 150 ml of

frank pus from right paravertebral, retrocolic space. Pus

culture revealed Klebseilla oxytoca. Histopathological

examination of the resected bowel revealed mixed

inflammatory infiltrates in colonic wall showing sheets

of histiocytes containing Michaelis-Gutmann

lymphocytes, plasma cells and polymorphonuclear

cells. Antimicrobial agents including piperacillin and

tazobactum, metronidazole and gentamicin were given

to the patient for five days. On post operative day seven,

this patient developed anastomotic leakage, and despite

re-exploration and repeat reconstruction, he succumbed

to a cardiac arrest ten days after operation.

Discussion

Malakoplakia is a chronic inflammatory disorder. It is

characterized by tumour like collections of sheets of

large histiocytes with abundant granular eosinophilic

Correspondence: M.SinghE-mail: [email protected] Figure 1

cytoplasm showing per-iodic acid Schiff (PAS) positive

rounded bodies called Michaelis-Gutmann bodies.

These primarily represent phagolysosomes. In 1903, it

was named 'Malakoplakia' by Von Hansemann. The

pathogenesis of malakoplakia remains obscure. It is a

rare condition that occurs predominantly in the

genitourinary tract [2] and less frequently in the

gastrointestinal tract [4] - the rectosigmoid region is

most commonly affected. The age of onset ranges from

6 weeks to 88 years, with a peak incidence in childhood

and a second peak at an older age. Colonic malakoplakia

has been reported immunodeficient states, such as

lymphoproliferative diseases, severe infection, drug

addiction, hepatic cirrhosis, and diabetes mellitus,

either in isolation or in association with colonic

tumours.

In our case, the right side of colon was involved with an

associated psoas abscess. This is an unusual site for

malakoplakia with only one case being reported to date

which involved the ascending colon [5]. It seems likely

that malakoplakia had developed as a result of altered

tissue immune response against normal bowel flora.

Diabetes mellitus was also one of the risk factors. In

patients with malakoplakia, the disease process is

usually progressive and refractory to treatment until

underlying immunosuppresion is corrected. It may be

the reason, along with diabetes, for this patient's

anastomotic failure.


Many different therapeutic modalities have been

described in the literature. The goals of treatment were

to improve the killing capability of macrophages by

attempts to elevate cyclic guanisine monophophate (C-

GMP) levels in blood, elimination of underlying

infectious causes with concurrent control of underlying

immunosuppressant pathology in those with

widespread disease. Surgical excision of localized

lesions is usually curative. Antimicrobial drugs should

be used in such patients to penetrate macrophages and

destroy undigested bacteria.

References

1. Michaelis L, Gutmann C. Ueber Einschlu¨sse in

Blasentumoren. Z Klin Med 1902; 47:208-215.

2. Ben Amna M, Hajri M, Oumaya C, et al. Genito-urinary

malakoplakia. Report of 10 cases and review of the literature.

Ann Urol. 2002; 36:388-3913.

3. Mc Clure. Malakoplakia. J Pathol 1983;140: 275-230.

4. Pillay K, Chetty R. Malakoplakia in association with

colorectal carcinoma: A series of four cases. Pathology. 2002;

34:332-335.

5. Van Furth R, Van't Wout JW, Wertheimer RP, et al.

Ciprofloxacin for treatment of malakoplakia. Lancet 1992;

339:148-9.

Key points:

Malakoplakia is a chronic inflammatory condition.

Rare, and predominantly effects the genitourinary tract.

Colonic malakoplakia has been reported in immunodeficient states such as lymphoproliferative diseases,

severe infection, hepatic cirrhosis and diabetes.

Surgical excision of localized lesions is usually curative with treatment of underlying immunosupression.

CASE REPORTS

Undescended testis with congenital unilateral absence of the vas deferens V.D. Kulkarni, N.A. Deshpande, P.N. Deshpande, M. PhanseDepartment of Surgery, Y.C.M.Hospital, Pimpri Pune, India.

Keywords: Vas deferens; cryptorchidism; cystic

fibrosis.

Abstract

Congenital absence of the vas deferens (CAVD) is a

condition in which the vasa deferentia, fail to form

properly prior to birth. It may either be unilateral

(CUAVD) or bilateral (CBAVD). Congenital absence of

vas deferens is responsible for upto 2% of male

infertility. The disease is considered to be a primary

genital form of cystic fibrosis. It contributes towards 6%

of obstructive azoospermia cases. Upto 80% of patients

with CAVD have mutations accociated with Cystic

Fibrosis Transmembrane Conductance (CFTR) gene.

Upper urinary tract malformations are noted in upto

20% of cases. This category of patients have a normal

frequency of CFTR mutations and considered a distinct

clinical entity. We hereby report a rare case of CUAVD

with ipsilateral renal agenesis with cryptorchidism.

Introduction

Disturbances in the development of human embryo are

responsible for the absence of vas deferens. Vas

deferens is derived from the mesonephric duct while the

testis and the globus major of the epididymis arise from

the genital ridge. The extent of abnormalities depends

on the time at which mesonephric duct stops

developing. If it occurs early in embryonic life (around

the fourth week) the ureter and kidney are absent [1]. If

the disturbances happen beyond fourth week, the kidney

and ureter are present, although their development could

be abnormal [2].

Case History

A 12 year old child presented with non-descent of the

right testis since birth. He did not have associated

abdominal pain. On examination, the right testis was


neither palpable in the scrotum nor found in the right

inguinal region. The left testis was normal in size and the

cord was palpable on scrotal examination. There was no

report of hospital admissions in the past.

Ultrasonography of the abdomen revealed an absent

right kidney, the left kidney being normal in size and

shape. The right testis was detected, by ultrasound, in

the right inguinal region. Intravenous pyelography

confirmed the above findings (Figure 1). Renal function

tests were normal. Sweat sodium estimation was in the

normal range. The patient received an orchidopexy after

informed consent was obtained from his parents. Intra-

operatively the right testis was in inguinal canal ( Figure

2). On evaluating cord structures, the vas deferens was

absent. Following orchidopexy, he was discharged on

the second day.

Discussion

Abnormalities in the vas deferens depend on the time at

which the mesonephric duct stops developing. Renal

agenesis associated with CUAVD occurs if the

Correspondence: V.D. KulkarniE-mail: [email protected]

Figure 1. Intravenous pyelography depicting right renal agenesis

mesonephric duct stops developing by the fourth week

[1]. The vas deferens is a cord like structure which can

be palpated easily in the normal scrotum. CAVD is a

distinct clinical entity. The diagnosis is easily made on

scrotal examination [3]. A lower frequency of CFTR

mutations has been detected in patients with CUAVD

[4]. Between 11% and 26% of patients with CAVD have

renal agenesis [5]. The association of CAVD with renal

agenesis in cryptorchidism is very low. CFTR mutations

are detected in men with CBAVD and, with a lesser

frequency in men with CUAVD, it appears that a normal

amount of functional CFTR protein is required to ensure

proper development of the vas deferens. However the

lack of CFTR mutations detected in CAVD with renal

anomalies also suggest that the mesonephric duct plays

a role in the development of the vas deferens. CAVD is

the cause of male infertility and hence genetic

counseling plays a major role in patients with CAVD [5]


Conclusions

CUAVD associated with renal anomalies and

cryptorchidism is a rare diagnosis. CUAVD may be

associated with cystic fibrosis and evaluation of the

same needs to be considered in all such patients. CAVD

can cause infertility in men.Genetic counseling plays a

major role in patients with CAVD because of its intricate

relations with CFTR gene.

References

1 Mickle, J., Milunsky, A., Amos, J.A. and Oates, R.D.

Congenital unilateral absence of the vas deferens: a

heterogeneous disorder with two distinct subpopulations

based upon aetiology and mutational status of the cystic

fibrosis gene. Hum.Reprod. 1995; 10:1728-1735.

2 Holsclaw, D.S., Perlmutter, A.D., Jockin, H. and

Shwachman, H. Genital abnormalities in male patients with

cystic fibrosis. J. Urol. 1971; 106: 568–574.

3 Marc De Braekeleer and Claude Ferec. Mutations in the

cystic fibrosis gene in men with congenital bilateral absence

of the vas deferens. Molecular Human Reproduction 1996;

2(9):669-677.

4 Casals, T, Bassas, Ll, Ruiz Romero, J et al. Extensive

analysis of 40 infertile patients with congenital absence of vas

deferens: in 50% of cases only one CFTR allele could be

detected. Hum. Genet. 1995; 95:205-211.

5 Schelgel, P.N., Shin, D. and Goldstein, M. Urogenital

anomalies in men with congenital absence of the vas deferens.

J. Urol. 1996; 155:1644-1648.

Figure 2. Right testis in the inguinal canal

Key points:

CUAVD can be associated with renal anomalies and cryptorchidism is a rare diagnosis.

CAVD can be a cause of infertility in males.

CASE REPORTS

Unusual presentation of urinary ascites diagnosed by laparoscopy V. Gaikwad, A.D. Chougale, S. Murchite, R.M. Kulkarni, J. BagwanDepartment of Surgery, Padmashri Dr D Y Patil Hospital and Research Institute, Kadamwadi, Kolhapur, India

Keywords: Urinary; ascitis; laparoscopy

Case report

A 40 year old female was referred for acute renal failure.

Her chief complaints were oliguria and distension of

abdomen with lower abdominal pain of three days. In

the past, this patient had renal tuberculosis with right

sided pyelonephritis (Positive TB polymerase chain

reaction qualitative test on urine sample by DNA

amplification for Mycobacterium Tuberculosis

Complex, MTC ) and right sided hydronephrosis with

placement of a double J stent and had taken anti-

tuberculosis medication for 6 months. The stent was

removed 3 months later. On general examination she

was anaemic, her abdomen was distended with flank

fullness and there was a fluid thrill. Haematological

investigation revealed a haemoglobin of 5.3 g/dl, serum

urea-90 mg/dl, and serum creatinine- 4.5 mg/dl. She had

normal liver biochemistry tests and normal serum

electrolytes and carbohydrate antigen (CA-125) was

29.38 U/ml (Normal- 0.0-35 U/ml). Urine analysis

showed pyuria and bacteriuria and ascitic fluid analysis

showed 95% lymphocytosis, however, urine and ascitic

fluid were negative for acid fast bacilli. An ultrasound

examination showed moderate to gross ascites, gross

right hydronephrosis and a thin right renal cortex with

mild compensatory hypertrophy of the left kidney, and

right adnexal cysts. The urinary bladder was

catheterized on admission and treatment commenced

for renal tuberculosis with acute renal failure. Gradually

her urine output improved to about 2000ml per day.

After satisfactory recovery and reduction in her

abdominal girth, her urinary catheter was removed.

However, in the ensuing days she developed ascitis

which required repeated paracentesis. Since no

definitive cause of ascitis could be ascertained a


diagnostic laparoscopy was planned. Laparoscopy

surprisingly revealed perforation of the urinary bladder

at its dome which was comfirmed when the tip of a

Foley catheter was visualized in the peritoneal cavity

(Figure 1). At mini laparotomy, we found a “thimble

bladder” with perforation which was closed in two

layers along with right salphingo-oophorectomy which

was performed for a concomitant right ovarian cyst.

Post op recovery was uneventful.

Discussion

Ascites may occur due to such conditions as liver

cirrhosis, tuberculous peritonitis, pyogenic peritonitis,

congestive heart failure, urinary ascites, biliary ascites

and pancreatic ascites. The cause for intractable ascites

is often difficult to diagnose by conventional laboratory

examinations and results in a diagnostic challenge for

clinicians. [3, 4]

Laparoscopy as an alternative to exploratory

Correspondence: V. GaikwadE-mail: [email protected]

Figure 1. The tip of a Foley catheter protruding through the urinary bladder.

laparotomy has been used in the past in the evaluation of

ascites when the cause is unclear. In a number of cases,

where the clinical picture is confusing and standard tests

fail to disclose the source of the fluid collection,

diagnostic laparoscopy is a reliable method.

Laparoscopy may visualize peritoneal deposits of

tuberculosis or tumour with the added advantage of

biopsy under direct vision even in the elderly and infirm

[5,6].

Conclusion

Laparoscopy is a valuable means of assessing the

peritoneal cavity in patients with unexplained ascites


when the primary cause remains unclear. With a careful

and standardized technique of entry, complications are

rare, the diagnosis can be accurately made with selective

biopsy specimens and appropriate treatment can then be

instituted without delay, which is particularly important

in patients where diagnosis becomes difficult to made

for origin of ascites.

References

1. Ho AM, Roth P, Cowan WD. Gaseous distension of the

urinary bag indicating bladder perforation during

laparoscopic pelvic procedures. Int J Gynecol Obstet. 1996;

55:297–8.

2. Greenberger NJ. Gastrointestinal Disorders: a Pathological

Approach. 4th ed. Chicago: Year Book Medical Publishers,

1990:365-7.

3. Bender MD. Disease of the peritoneum, mesentery and

diaphragm. In: Sleisenger MH, Fordtran JS, eds.

Gastrointestinal Disease: Pathophysiology, Diagnosis,

Management. 4th ed. Philadelphia: WB Saunders,

1989:1935-51

4. Uthman IW, Bizri AR, Shabb NS, Khury MY, Khalifeh MJ.

Peritoneal sarcoidosis: case report and review of the

literature. Semin Arthritis Rheum 1999; 28:351-4.

5. Roskos M, Popp MB. Laparoscopic diagnosis and

management of malignant ascites. Surg Laparosc Endosc

Percutan Tech 1999; 9:365-8.

6. Chu CM, Lin SM, Peng SM, Wu CS, Liaw YF. The role of

laparoscopy in the evaluation of ascites of unknown origin.

Gastrointest Endosc 1994; 40:285-9.

Figure 2. Urinary ascites with a right sided ovarian cyst at laparoscopy

Key points:

Laparoscopy is a valuble means of examining the peritoneal cavity. Providing a magnified view of organs

and pathology.

CASE REPORTS

Neonate with congenital mesoblastic nephroma surviving respiratory failure and pulmonary embolism at laparotomy

1 2 1 1R. Ranawaka¹, A. Vithanage , P. Rathnayake , K. Hettiarachchi , N. Jayakody1. Sirimavo Bandaranayake Specialised Children's Hospital, Peradeniya, Sri Lanka2.Teaching hospital Kandy, Sri Lanka

Keywords: Congenital mesoblastic nephroma,

pulmonary embolism, disseminated intravascular

coagulation

Introduction

Congenital mesoblastic nephroma (CMN) first

described by Bolande et al in 1967 [1] is the commonest

neonatal tumour of renal origin comprising of 3-10% of

all childhood tumours [2]. Common modes of

presentation include abdominal mass, antenatal

diagnosis and features of para-neoplastic syndrome [3].

There are two main histological varieties; classic type

where nephroureterectomy is curative and atypical

(cellular) type with unpredictable natural history [2,4].

We report a two day old developing severe cardio-

respiratory failure secondary to CMN, surviving

emergency tumour debulking laparotomy that was

complicated by pulmonary embolism and disseminated

intravascular coagulation (DIC). Subsequent

nephroureterectomy with total tumour clearance was

performed. To our knowledge there are no reports of

CMN with similar presentation in the world literature.

Case

An eighteen hour old baby with antenatal diagnosis of

“intraabdominal tumour” was transferred on

developing laboured breathing. Within hours she

required ventilatory support and cardiac inotropes.

Ultra sound scan detected a large intra-abdominal mass

displacing other viscera. The left kidney was not

identified. Computerised tomography (CT) scan was

not accessible.

During emergency laparotomy a bosselated


encapsulated tumour occupying almost the whole

abdomen with the consistency of an “oily sponge cake”

seen originating from left kidney. The liver, bladder,

uterus and Fallopian tubes were normal.

Suddenly, generalised bleeding and bradycardia

developed suggesting pulmonary air embolism leading

to DIC. Blood components were transfused and, the

operative field was filled with isotonic saline to

facilitate evacuation of pulmonary embolus. Surgery

was terminated and tumour remnant over-sutured.

Resected tumour mass weighed 350grams.

Approximately the same volume of tumour was left in-

situ.

Tumour markers were done as advised by the oncologist

(serum βHCG: <0.1miu/ml(<1miu/ml), α fetoprotein:

776.1iu/ml(<25iu/ml). Histology revealed degenerated

tumour tissue and undifferentiated spindle cells with no

b l a s t e m a l o r e p i t h e l i a l e l e m e n t s .

Immunohistochemistry was focally positive for

vimentin and, negative for SMA,PCK,CD10 and

MyoD1. The final diagnosis of CMN was made(4).

Subsequent CT scan of abdomen showed tumour

remnant compressing the aorta and inferior vena cava

(Figure 1).

At re-laparotomy, well encapsulated tumour arising

from the lower pole of left kidney with firm and fibrotic

consistency found. Rest of the viscera were normal.

Left nephroureterectomy with total tumour resection

was performed. Tumour weighed 400grams (Figure2).

Histology confirmed classic type CMN confined within

excision margins. She recovered completely and

remains well three months after surgery. Surveillance

USS is planned in three month's time. Correspondence: R.RanawakaE-mail: [email protected]

Discussion

Pulmonary embolism could occur in surgery during

exposure of a large vascular surface. The manifestations


range from trivial reduction in platelet count and

prolongation of clotting time to fulminant DIC [5]. The

management includes correction of DIC with

transfusion of blood components and, evacuation of

pulmonary air embolus by filling the surgical field with

isotonic saline while continuing ventilation.

Initial surgery leaving a residual tumour bulk could

theoretically be considered as tumour upstaging.

Adjuvant therapy has a place in metastatic CMN but is

not described for classic type. Since complete tumour

clearance was confirmed histologically, surveillance for

tumour recurrence with regular imaging was planned.

The rare experience of successful tumour excision after

complicated surgery in a neonate with no proper pre-

operative imaging and histology would be informative

to surgeons who experience similar surgical

emergencies.

References

1. Bolande PP, Brough AJ, Izant RJ, Congenital mesoblastic

nephroma of infancy. Pediatrics 1967;40: 272–279

2. dos Santos LG, de Sousa J, de Carvalho R et al, Cellular

congenital mesoblastic nephroma: case report. Jornal

Brasileiro de Nefrologoa 2011;33:88-90

3. Minesh K, Horacio O, Michael AS, Congenital Mesoblastic

Nephroma Presenting with Neonatal Hypertension. Journal

of Perinatology 2005; 25:433–435

4. Argani P, Fritsch M, Kadkol S et al, Detection of the ETV6-

NTRK3 chimeric RNA of infantile fibrosarcoma/cellular

congenital mesoblastic nephroma in paraffin- embedded

tissue: application to challenging pediatric renal stromal

tumors. Modern Pathology 2000; 13:29-36

5. Levi M, Ten Cate H, Disseminated intravascular

coagulation. New England Journal of Medicine 1999;

341:586–92.

Figure 1. CT scan of abdomen after initial tumour debulking laparotomy

Figure 2. Specimen after total resection

Upper pole of Left Kidney

Key points:

Pulmonary embolism could occur in surgery during exposure of large vascular surfaces.

Management includes filling the surgical field with isotonic saline while continuing ventilation and

correction of DIC.

CASE REPORTS

Laparoscopic adrenalectomy for Conn syndrome 1 1 2K.A.P.R. Kumarasinghe , S.K. Kollure , R. Amerasena

1 Department of Surgery, National Hospital Sri Lanka.2 Department opf Anaesthesia, National Hospital Sri Lanka.

Keywords: Adrenalectomy, laparoscopy, Conn's

adenoma

Introduction

Since its introduction in the 1990s, laparoscopic or

retroperitoneoscopic adrenalectomy has become the

'gold standard' in the resection of adrenal tumours,

except for tumours with signs of malignancy [1,2]. The

view of the operative field obtained during a

transperitoneal laparoscopic approach is superior to that

obtained by open approach.

Case Report

A 36 year old male with an episode of severe headache

and short term memory impairment was admitted to

medical casualty. On admission his blood pressure was

260/170mmHg with papilloedema. An urgent non

contrast computerised tomograghy (NCCT) of brain

revealed bleeding into choroid plexus (Figure 1).

Neurosurgically he was managed conservatively. His

blood pressure was controlled on losartan, nifedipine

and prazocin.

Further investigations revealed hypokalaemia (

2.6µmol/l) with elevated serum aldosterone levels

(20.1ng/dl). Serum renin level was markedly reduced

(0.29ng/ml/hr). His urinary VMA level was normal

(9.98). CT abdomen detected a lesion measuring 1×1

cm, in the right adrenal gland which is consistent with an

adenoma (Figure 2). Diagnosis of Conn's adenoma was

made. The patient was referred to our surgical unit.

After preoperative evaluation of the patient,

laparoscopic adrenalectomy was planned. Surgery was

performed under general anaesthesia with invasive

monitoring.

Patient was put on lateral decubitus position, four port


entry made into the peritoneal cavity (Figure 3).

Liver was retracted using a fan retractor. Lateral

peritoneal reflection of the right triangular ligament was

divided with ultrasonic shears. Liver retracted medially

to provide exposure to right kidney and adrenal gland.

Gerotas fascia over the midportion of the kidney

opened, perinephric fat dissected over its superior pole.

Lateral edge of the inferior vena cava (IVC) identified

which formed the medial margin of the dissection. The

adrenal gland was identified by its orange yellow colour

[3]. Short adrenal vein identified and divided between

haemostatic clips. Adrenal gland was gradually

dissected and direct adrenal branch from aorta and

branch from inferior phrenic artery divided with

ultrasonic shears. IVC retracted medially and extension

of the gland behind it dissected out.

Endobag extraction of specimen via a 5cm incision

done. There was no measurable blood loss and the

Correspondence: K.A.P.R. KumarasingheE-mail: [email protected] Figure 1. CT brain showing bleeding into choroid plexus.

procedure took 80 minutes. Histology confirmed

adrenal adenoma in right adrenal gland.

He was discharged on 4th post operative day. On

discharge his blood pressure control was maintained

only on losartan.

Discussion

In 80% of cases primary hyperaldosteronism is caused

by an aldostrone secreting adenoma in one adrenal

gland, a condition referred to as conn syndrome. In most

cases adenomas are solitary and small (<2cm in

diameter) [4].

Laparoscopic adrenalectomy is the commonest surgical

approach currently in use. It is associated with reduced

wound, chest and gastrointestinal complications,

decreased hospital stay, shorter recovery time and

improved patient satisfaction compared with open

surgery [5].

Unilateral laparoscopic adrenalectomy is an effective

therapy in patients with clear evidence of unilateral or

asymmetrical bilateral disease. A subtotal resection is

favoured in the case of a typical Conn's adenoma. In

10–30% of patients who undergo an adrenalectomy,

hypertension persists despite adequate diagnostic


work-up and treatment [1].

Currently in Sri Lanka laparoscopic equipment and

expertises are limited to few centres. Therefore open

adrenalectomy is more commonly perfomed. With

adequate facilities and surgical and anaesthetic

expertises, laparoscopic adrenalectomy provides a

revolutionary change over the conventional open

method.

References

1. Norman S. Williams, Christopher J.K. Bulstrode, P. Ronan

O'Connel: Baily and Love. 25th edition. Arnold; 2008

2. Matthew J. Mellon, Amanjot Sethi, Chandru P. Sundaram.

Laparoscopic Adrenalectomy: Surgical techniques. Indian

Journal of Urology. 2008; Oct-Dec24(4): 583-589

3. Carol E.H.Scott- Conner, David L. Dawson: Operative

Anatomy. 3rd edition. Philadelphia: Lippincott Williams &

Wilkins; 2009

4. Vinay Kumar, Abdul K. Abbas, Nelson Fausto, Richard N,

Mitchell. Robbins: Basic Pathology. 8th edition. Elsevier;

2009

5. Saba Balasubramanian, Barney Harrison. Investigation and

management of adrenal disease. Surgery (Oxford). 2011;

459-464

Figure 2. CT abdomen showing an adrenal adenoma on the right side

Figure 3. Port position


Key points:

Laparoscopic adrenalectomy is the commonest surgical approach currently in use.

Unilateral laparoscopic adrenalectomy is effective in patients with unilateral or asymmetrical bilateral

disease.

Subtotal resection is favoured in typical cases of Conn’s disease.

CASE REPORTS

Umbilical endometriosis-A simple but challenging diagnosis for surgeons 1 1 1 2 1K.G.H. Jayathilake , S. Withana , R. Siriwardane , G. Mahendra , C. Liyanage

1 Department of Surgery, Faculty of Medicine, University of Kelaniya, Sri Lanka.2 Department of Pathology, Faculty of Medicine, University of Kelaniya, Sri Lanka.

Keywords: Umbilical; endometriosis; umbilical lump;

extrapelvic endometriosis

Introduction

Endometriosis is a challenging but common

gynaecological condition which is characterized by the

presence of endometrial glands and stroma outside the

uterus [ ]. It presents mainly with involvement of the 1

pelvic organs. Extrapelvic presentations in almost all

parts of the body have been reported and account for

about one percent of the total cases [1]. However,

spontaneous umbilical endometriosis nodules or

endometriosis secondary to surgery accounts for only

0.5% to 1% of all endometriosis cases [2].

Case presentation

A 49 year-old parous woman who presented with an

umbilical lump for two years complained of cyclical

monthly pain and bluish discolouration. There was no

history of bleeding or discharge from the site. She had

undergone laparoscopic hysterectomy and left sided

salpingo-oophorectomy four years back due to

endometriosis causing severe dysmenorrhea.

At the time of presentation she had a 1 × 1.5 cm purplish

nodule at the umbilicus (Figure 1). It was not tender and

there were no signs of infection. There was a 1cm well

healed surgical scar below the umbilicus.

Fine needle aspiration cytology of the umbilical nodule

confirmed the diagnosis as endometriosis at the

umbilicus. Following a discussion with the patient and

clear explanation about the possibility of recurrence and

the need to remove the umbilicus, the nodule was

completely excised with the umbilicus and the defect

was repaired. As the patient was not concerned about the

cosmetic outcome, umbilical reconstruction was not


carried out. The histology confirmed the diagnosis of

endometriosis (Figure 2).

Discussion

Umbilical endometriosis can be primary or secondary to

a surgical scar. Villar, in 1886, reported the first case of

umbilical endometriosis. Hence, this condition is also

called Villar's nodule [ ]. Similar to our own case 1

umbilical endometriosis at the site of the laparoscopic

entry is one of the commonest. The primary umbilical

endometriosis is known to be associated with severe

pelvic endometriosis compared to those with scar

endometriosis [3].

In this patient, cyclical pain with a classical purplish

nodule clinched the diagnosis; but clinical diagnosis of

umbilical endometriosis is, at times, difficult with

varying presentations in colour, character of pain and

size. Hence, malignant melanoma, paraumbilical

hernia , granuloma, pr imary or metas ta t ic

adenocarcinoma (Sister Joseph nodule), nodular

melanoma, and cutaneous endosalpingosis should be

considered.Correspondence: K.G.H. JayathilakeE-mail: [email protected]

Figure 1. Umbilical lump with bluish discolouration.

Surgical excision is the treatment of choice for this

condition. If a wide excision and complete removal of

the umbilicus is anticipated, it should be discussed with

the patient, and reconstruction of the umbilicus can be

offered [4].


In our patient, endometriosis was previously diagnosed

and treated by hysterectomy and left sided salpingo-

oophorectomy. However, in a newly diagnosed similar

case the chances of coexisting pelvic endometriosis

should be sought and treated with special concern for

fertility. Pelvic endometriosis is a common condition,

but the diagnosis of primary umbilical endometriosis

requires a high degree of suspicion.

References

1. Sreelakshmi K, Muralidhar V P, Mary M; Umbilical

laparoscopic scar endometriosis. J Hum Reprod Sci. 2011

Sep-Dec; 4(3): 150–152.

2. Latcher JW; Endometriosis of the umbilicus. Am J Obstet

Gynecol. 1953;66:161–8.

3. Agarwal A, Fong Y F; Cutaneous endometriosis, Singapore

Med J 2008; 49(9): 704-709

4. Bagade P, Guirgus MM; Menstruating from the umbilicus

as a rare case of primary umbilical endometriosis: A case

report. J Med Case Reports. 2009;3:9326

Figure 2. Haematoxylin and eosin section (20x10) showing tortuous glands surrounded by dense cellular stroma lying in a collagenous background. The glands are lined by tall columnar epithelium that resembles the endometrial glandular epithelium. The sub-epithelium is composed of small round cells that resemble the endometrial stroma.

Key points:

Umbilical endometriosis can be primary or secondary to a surgical scar.

malignant melanoma, paraumbilical hernia, granuloma, primary or metastatic adenocarcinoma (Sister

Joseph nodule), nodular melanoma and cutaneous endosalpingosis should be considered in the differential

diagnosis.

Surgical excision and reconstruction of the umbilicus is the treatment of choice.

CASE REPORTS

Post CABG (coronary artery bypass graft) surgical site infection by Nocardia

M.N. Sumana, M. Shenoy, G.R. ManjunathJSS Medical College and Hospital, Mysore, India.

Keywords: Nocardia; coronary artery bypass graft

Introduction

Surgical site infection is the second most common

nosocomial infection. Wound infection is an important

cause of morbidity and occasional mortality after

coronary artery bypass graft surgery (CABG). Common

organisms encountered are Staphylococcus aureus,

Staphylococcus epidermidis, Enterococcal species,

Streptococcus pneumonia, and Pseudomonas

aeruginosa [1]. In certain cases of delayed CABG

wound healing, Candida species and occasionally

rapidly growing atypical Mycobacterial species are

isolated. Only one case of Nocardia asteroides

mediastinitis complicating CABG surgery has been

reported in the literature available by Susan Verghese et

al [4]. Nocardia occurs worldwide and the annual

incidence is 0.375 cases per 100000 persons as

estimated by three continents- North America, Europe

and Australia. This case report may be following

transcutaneous inoculation during CABG surgery from

the hospital environment or solution. The rarity of

Nocardia causing sternal wound infection after CABG

surgery prompted reporting of this case.

Case report

A 65 year old male patient reported to Surgical OPD,

with history of incomplete healing of wound on the

Chest wall for more than 2months duration after CABG

surgery. He underwent surgery 2 ½ months ago. He is a

known diabetic and a hypertensive under good control.

On examination, wound dehiscence was noticed at the

lower end of the CABG surgical scar with scanty

discharge. The discharge was sent to Microbiology lab

for Culture and sensitivity.


The sample was inoculated on to MacConkey agar and

blood agar, and incubated aerobically at 37 degree

Celsius overnight. A gram stained smear was also

prepared from the sample and observed under the

microscope. It showed gram positive branching

filaments. The smear was completely decolorized with

alcohol and re-stained with modified Zeihl-Nelson's

stain using 1% sulfuric acid as decolourizer and

observed under the microscope. It showed pink acid fast

branching thin filaments and tentatively identified as

Nocardia species.

The culture plates did not show any growth after

overnight incubation and they were further incubated

for another day. Next day when the plates were

examined, MacConkey's agar showed no growth but

blood agar showed growth of white, small, dry colonies

(Figure 1). Culture smear made showed gram positive

thin branching filaments. Modified Zeihl-Neelson's

stain using 1% sulfuric acid as decolourizer showed

pink acid fast filamentous branching bacteria (Figure 2).

The bacteria grown were identified as Nocardia species.

The biochemical reactions showed following results. It

failed to hydrolyse casein, xanthine and tyrosine and

Correspondence: M. ShenoyE-mail: [email protected] Figure 1. Showing dry white colonies on blood agar.

was urease negative. It was identified as Nocardia

asteroids. The sensitivity of the organism was done on

Mueller-Hinton blood agar. It was sensitive to

cotrimaxazole, penicillin, amoxicillin, gentamycin,

erythromycin, amikacin, ciprofloxacin and ofloxacin. A

repeat sample collected from the patient to confirm also

yielded the growth of same organism. The patient was

s tar ted on t r imethopr im (20mgm/kg) and

sulfamethoxazole (100mgm per kg) each day in two

divided doses for 15 days and later the dosage reduced to

5mgm per kg and 25mgm per kg respectively for 4

months. After 15 days of treatment the wound showed

signs of healing and after a month it was completely

healed.

Discussion

Post-operative wound infections like sternal wound

infections, donor site infections and mediastinitis form

serious complications after cardiac surgery. Common

causes of post-operative wound infection include

Staphylococcus aureus both MRSA (Methicillin

resistant Staphylococcus aureus) and MSSA

(Methicillin sensitive Staphylococcus aureus),

Staphylococcus epidermidis both MSSE (Methicillin

sensitive Staphylococcus epidermidis) and MRSE

(Methicillin resistant Staphylococcus epidermidis) [1].

Other less common causes could be Enterococcus,

Pseudomonas, Acenetobacter and members of

Enterobacteriaciae.

Bhatia JY et al from Hinduja hospital, Mumbai have

studied six hundred and fifteen patients. Of these 116

(18.86%) developed SSI, involving sternum 75%, leg

21.3%, and forearm sites 3.44%. Organisms isolated at


sternum site were MSSE, MRSA, and MRSE [1].

Softah et al have reported post CABG wound infection

due to Staphylococcus aureus in 33% of their patients

and Staphylococcus epidermidis in 31% of their patients

and mixed infections in 11.7% of patients [2].

Preeti N. Malani et al from Michigan have reported that

Candida species are an uncommon cause of sternal

wound infections. Mediastinitis due to Candida is a rare

but serious complication that usually occurs in the first

few days to weeks after the surgical procedure and is

associated with a mortality rate of >50%. They report on

a series of 11 patients who developed deep sternal

wound infections due to Candida albicans. These

infections were characterized by a chronic, indolent,

and recurrent course [3].

Susan Verghese et al have reported a case of Nocardia

asteroides mediastinitis complicating coronary artery

bypass surgery [4].

Thaler F et al have reported mediastinitis due to

Nocardia asteroids after cardiac transplantation [5].

References

1.Bhatia JY Pandey K, Rodrigues C, Mehta A, Joshi

VR.Rheumatology, PD Hinduja National Hospital and

Medical Research Centre, Mumbai - 400 016,

India.“Postoperative wound infection in patients undergoing

coronary artery bypass graft surgery: a prospective study with

evaluation of risk factors”. Indian J Med Microbiol. 2003 Oct-

Dec; 21(4):246-51.

2. A. Softah, MD; A. Bedard, MD; P. Hendry, MD; R.G.

Masters, MD;W. Goldstein, MD; M. Brais, MD; W. Keon,

MD. WOUND INFECTION IN CARDIAC SURGERY.

Annals of Saudi Medicine, Vol 22, No's 1 -2, 2002 P105-07

3. Preeti N. Malani, Shelly A. McNeil, Suzanne F. Bradley,

and Carol A. Kauffman University of Michigan Medical

School, Ann Arbor, Michigan, Candida albicans Sternal

Wound Infections: A Chronic and Recurrent Complication of

Median Sternotomy Clin Infect Dis. (2002) 35 (11): 1316-

1320. doi: 10.1086/344192.

4. Susan Verghese*, VM Kurian**, CF Maria***, P

Padmaja***, SJ Elizabeth***, KM Cherian+ “Nocardia

asteroides Mediastinitis Complicating Coronary Artery

Bypass Surgery”. JAPI VOL. 51 OCTOBER 2003

Figure 2. Showing branching acid fast filamentous bacteria


5. Thaler F, Gotainer B, Teodori G, Dubois C, Loirat P.

Mediastinitis due to Nocardia asteroides after cardiac

transplantataion. Intensive Care Med 1992;18:127-8.

Key points:

Nocardia and Candida species are an uncommon cause of sternal wound infections.

Infections occur early after CABG and are associated with mortality greater than 50 percent

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