prenatal diagnosis of a huge foetal immature sacrococcygeal … · 2014-03-02 · case reor page 1...

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For citation purposes: Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-cygeal teratoma: our experience of a rare case and review of the literature. OA Case Reports 2013 Jan 31;2(1):10.

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Prenatal diagnosis of a huge foetal immature sacrococcygeal teratoma: our experience of a rare case and review

of the literatureM Varras1*, G Diakakis1, I Monselas1, CH Akrivis2

increased blood flow into the tumour. Pulsed Doppler showed the resist-ance index of flow velocity wave-forms on the tumoural arteries to be 0.51. Amnioreduction of 740 cc amni-otic fluid was performed under ultra-sonographic examination. Caesarean section was performed at 33 weeks’ gestation due to profuse polyhydram-nios via an upper vertical incision in the uterus. After the stabilization of the newborn, tumour resection was successfully performed on the first day after delivery. Grossly, in the sur-gical specimen the tumour measured 18 cm in its maximum diameter and weighted 1 500 g. ConclusionPrenatal diagnosis and ultrasono-graphic follow-up are needed for the good prognosis of sacrococcygeal ter-atomas. Prenatal intervention should be considered when the foetus devel-ops hydrops for foetal salvage or in cases with placentomegaly to avoid the maternal risk of mirror syndrome.

IntroductionTeratomas are the most common perinatal tumours developed from multipotent stem cells, are usually extragonadal and contain tissues of ectodermal, endodermal and meso-dermal origin, foreign to the ana-tomic site, in which they arise1–3. Ter-atomas account for 25%–52% of all congenital tumours and have a yearly incidence of one in 20 000 to one in 40 000 live births3,4–7. Sacrococcy-geal teratomas have an incidence of 35 000–40 000 live births and have usually a female predominance8–10. They are classified as mature, imma-ture or malignant based on their histopathologic appearance and

behaviour6. Sacrococcygeal terato-mas arise from the Hensen’s nodule located on the anterior surface of the sacrum or coccyx and may be cystic, multilocular or cystic, and solid and variable in size11,12.

The morbidity and mortality of the sacrococcygeal teratomas are low when they are diagnosed prenatally and the surgical management is per-formed during the neonatal period8,9. The prognosis is usually poor when they are diagnosed prior to 30 weeks’ gestation and are predominantly solid with high vascularisation due to high-output cardiac failure, development of hydrops fetalis, premature delivery and intrauterine foetal demise. Arteri-ovenous shunting through the tumour causes the high-output cardiac failure. The causes for the prematurity are usually the uterine distension from the neoplasm and the polyhydramnios, while the intrauterine foetal demise is secondary to the vascular steal syn-drome and the increased metabolic demands of a huge neoplasm7–9,13–15. In addition, the foetus is at risk of chronic foetal anaemia due to haem-orrhage within the tumour16. The suc-cessful management of sacrococcy-geal teratomas depends on the mode of delivery and the classification of the tumour7,9,13. Delivery of the foetus through a caesarean section prevents dystocia, tumour rupture and pro-fuse tumoural bleeding14,15. The risk of sacrococcygeal teratoma rupture is increased during the ablative tumour resection and particularly when the diameters of the neoplasm exceed 10 cm. The middle sacral artery usu-ally derives the most blood supply of these neoplasms. The progno-sis is influenced by the presence of

AbstractIntroductionSacrococcygeal teratomas are rare germ cell tumours associated with high perinatal and postnatal mor-tality and morbidity. The purpose of our study was to report a case of a foetal huge immature sacrococcygeal teratoma diagnosed prenatally and managed successfully in the early neonatal period with surgical resec-tion of the tumour. Case reportA foetal mass with solid and cystic components in the sacral region of the foetus measuring 42 × 34 mm was detected during the foetal anat-omy ultrasound scan, in a 28-year-old Greek woman, gravida 2, para 1. The foetal karyotyping showed nor-mal number of chromosomes 13, 18 and 21 (46 ΧY). Polyhydramnios was seen, at 31 weeks of gestation, and the patient was admitted to our hospital owing to a risk of prema-ture labour. Ultrasound examination showed the sacrococcygeal mass to be enlarged with maximum diam-eter of 20 cm. There were no signs of hydrops as demonstrated by the absence of ascites, pleural or peri-cardial effusion or placentomegaly. Colour Doppler ultrasound exami-nation showed extensive vasculari-sation within the mass, suggesting

*Corresponding authorEmail: [email protected] Third Department of Obstetrics and

Gynecology, “Elena Venizelou” General Maternity Hospital, Athens, Greece

2 Department of Obstetrics and Gynecology “G. Chatzikosta” General State Hospital, Ioannina, Greece

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immature elements in the neoplasm and there is a benign behaviour in cases with only neuroectodermal components2,15. It is estimated that at delivery 4%–20% have malignant tissue17,18. Severe clinical consequen-ces and frequent recurrences are observed when the neoplasms are dis-covered after the age of two months or the surgeons fail to perform proper coccygectomy or when they contain foci of yolk sac tumour, even an optimal surgical resection2,19–23. Gilcrease et al.24 suggested that micro-scopic foci of yolk sac tumour can be identified in low-grade immature ter-atomas as well and there is a possibil-ity for malignant recurrences in these cases2. In severe cases, sacrococcygeal teratomas with hydrops are associ-ated with maternal risk for develop-ment of mirror syndrome, which is a severe form of preeclampsia and is associated with placentomegaly25.

We report a case of a huge sacro-coccygeal teratoma diagnosed pre-natally at 21 weeks’ gestation by two-dimensional ultrasound scan, with foetal delivery via caesarean section at 33 weeks because of pro-fuse polyhydramnios and successful tumour resection management on the first day after caesarean section.

Case report In a 28-year-old Greek woman, gravida 2, para 1, during the foetal anatomy ultrasound scan at 21+2 weeks of gesta-tion, a foetal mass in the sacral region was detected containing both solid and cystic elements and measuring 42 × 34 mm. The neoplasm had incre-ased vascularity. Foetal biometry was normal for gestational age: bipa rietal head diameter (BPD): 50.00 mm, head circumference (HC): 187.00 mm, cis-terna magma: 4.8 mm, transverse cer-ebellar diameter: 21 mm, abdominal circumference (ΑC): 160.00 mm, femur length (FL): 32 mm, humeral length: 33.00 mm, HC/AC: 1.17. The amount of amniotic fluid was normal and the umbilical cord had three vessels. Foetal nuchal fold was measured to

4.5 mm (normal upper rate 6.00 mm). The spine appeared normal and the morphology of the cerebellum as well. The estimated foetal weight was 368 g by ultrasounds.

No other abnormalities were detected. A diagnosis of sacrococcy-geal teratoma was made. Foetal kary-otyping was recommended.

However, during the first trimes-ter ultrasound scan for nuchal trans-lucency thickness measurement at 12 weeks of gestation, the neoplasm was not detected. The foetal crown–rump length was 62 mm and the nuchal translucency thickness was 1.4 mm. No signs of cardiac dysfunc-tion, such as tricuspid regurgitation and/or absent or reverse flow in the ductus venosus during atrial contrac-tion were found in the foetus during the first trimester ultrasound scan.

A repeat scan at 24+5 weeks was performed for amniocentesis and foe-tal karyotyping and for checking the development of the foetus. The foetal biometry showed: BPD: 70.73 mm, HC: 235.56 mm, ΑC: 202.15 mm, FL: 48.14 mm. The amount of amniotic fluid was normal. The estimated foetal weight was 807 g by ultrasounds. The foetal heart rate was 167 beats per minute. The neoplasm contained both solid and cystic elements and measured 82 × 63 × 75 mm. The normal number of chromosomes 13, 18 and 21 was found (46 ΧY). The mutation DF508 for cystic fibrosis was not detected.

Polyhydramnios was seen, at 31 weeks of gestation, and the patient was admitted to our hospital owing to a risk of premature labour. The patient stated that she had regu-lar follow-up in a medical centre of another city throughout the previ-ous gestational weeks. The foetal biometry showed: BPD: 91.40 mm, HC: 304.00 mm, ΑC: 299.00 mm, FL: 64.20 mm. The ultrasonogra phic age of the foetus corresponded to 33+5 weeks. The foetus was with cephalic presentation. The estima ted foetal weight was 2314 g by ultrasounds. Ultrasound examination showed the

sacrococcygeal mass to be enlarged; the lesion was with solid and cystic components and maximum diameter of 20 cm (Figures 1 and 2). Polyhy-dramios was found. There were no signs of hydrops as demonstrated by the absence of ascites, pleural or per-icardial effusion or placentomegaly. Middle cerebral artery peak systolic velocity was normal. M-mode ultra-sound examination showed that nei-ther heart ventricle was dilated and the fractional shortening of both ven-tricles were found normal. The infe-rior vena cava was not enlarged and the preload index of the inferior vena cava was normal. The heart rate was 150 beats per minute. Colour Dop-pler ultrasound examination showed extensive vascularisation within the mass, suggesting increased blood

Figure 1: Axial view of the foetus demonstrated the sacrococcygeal teratoma (31-week gestational age).

Figure 2: Midline sagittal view of the foetus showed the sacrococcy-geal teratoma, consisting of cystic and solid components (31-week gestational age).

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flow into the tumour (Figure 3). Pulsed Doppler showed the resist-ance index (RI) of flow velocity waveforms on the tumoural arter-ies to be 0.51 (Figure 4). The patient was hospitalized and monitored with serial non-stress tests. Corticosteroid therapy was initiated for pulmonary maturation and indomethacin for preventive tocolysis. No cervical dila-tation was found. Three days later amnioreduction of 740 cc amniotic fluid was performed under the ultra-sonographic examination. Consulta-tion by paediatricians and paediatric surgeons was given to the parent and it was concluded that postpartum surgery should be performed.

Some days later, the risk of continu-ing with the pregnancy was explained and, following counselling, a caesarean delivery was performed at 33 weeks of gestation through a midline verti-cal incision in the abdominal wall and upper vertical incision in the uterus as well (Figure 5). A male infant weigh-ing 4 000 g was delivered with Apgar score of 5–6 at one minute and 7–8 at five minutes (Figure 6). The new-born underwent emergency surgical

resection of the tumour on the first day after delivery at the Department of Paediatric Surgery. Postoperatively, the baby did well. At the time of writ-ing he was over nine months of age and continued to do well.

PathologyMacroscopic examinationGrossly, the surgical specimen of soft tis sues had dimensions 20 × 16 × 13 cm, weight 1 500 g and consisted of fibro-adipose tissue and part of striated muscle fibres (Figure 7). A relatively circumscribed neoplasm with dimen-sions 18 × 14 × 12 cm, which was partially covered by a fusiform epi-dermal piece measuring 17 × 13 cm, occupied the surgical specimen. The tumoural sections showed partially a cystic conformation consisting of a cystic area underneath the skin with dimensions 12 × 8 × 3 cm and some smaller cystic areas with maximum diameters between 0.8 and 2.00 cm, which were full of mucous materi-als. Solid neoplasmatic areas were seen between the cystic areas; the larger solid neoplasmatic area had dimensions 5 × 4 × 3.5 cm and was in continuity with the cystic cavity, which had maximum diameter of 12 cm. They had yellowish/whit-ish hue and elastic consistency with places of important haemorrhagic infiltration and necrosis. At the periphery of the tumour by one pole,

Figure 3: Colour Doppler ultra-sonography showed extensive vascu-larity of the immature sacrococcygeal teratoma suggesting increased blood flow into the tumour.

Figure 4: Pulsed Doppler ultrasono-graphy showed the resistance index (RI) of flow velocity on the tumoural arteries to be 0.51 (31-week gesta-tional age).

Figure 5: Delivery via caesarean section was performed in the foetus with the huge sacrococcygeal tumour.

Figure 6: The premature infant was born at 33 weeks of gestational age. The appearance of the huge sacrococ-cygeal tumour was shown in the new-born lying in the right lateral decubitus position immediately after the caesar-ean section. The combined weight of the tumour and baby was 4 000 g.

Figure 7: The surgical specimen after the initial resection measured 20 × 16 × 13 cm; its weight was 1 500 g.

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opposing to the epidermal covering, there was recognised a continuous fractured part of cartilage and coccyx bone measuring 1.5 × 0.7 cm.

Microscopical examinationHistological type of the epidermal piece, subcutaneous adipose tissue and striated muscular fibres with localisation of extragonadal tumour from germ cells. Teratoma consist-ing of immature tissues of embryonic type and partially mature tissues of adult type, with representation of the three blastic layers with prevalence of the ectoderm (50%), notable pres-ence of the endoderm (30%) and the mesoderm (20%).

The ectoderm was represented by prevalence of cerebral substance under the form of mature neuro-glia and neurons, oligodendroglial cells, ependymal tubules and many cavities invested by ependyma and chorioid plexus. Characteristic was the noticeable presence (20% of the total volume) of immature neuro-epithelium with mild to severe cellu-larity, which was composed of small and average size cells with scarce cytoplasm, thin-walled nucleus without evident nucleoli, absence of notable nuclear atypia with high mitotic activity (15–50 mitotic divisions per 10 optic fields ×40) and with formation of nodules, tubules and focally papules. In addi-tion, recognised were sites and cavities with investment of mela-nochrostic epithelium, neuronal fas-ciae and agglomerations of ganglia cells and cavities with epidermal investment, presence of chorion in developmental phase and adnexal particles and spheres of squamous non- keratinized epithelium.

The endoderm was represented with multiple cavities invested by res-piratory type epithelium, other coated with cylindrical or transitional type epithelium with squamous metaplasia, cavities invested with gastric epithe-lium and colonic mucosa, other cavi-ties lined with serosal paramesonefric

type or mucosal of endocervical type agglomerations of seromucinous glan-dules. Characteristics were: (i) the presence of multiple individual islets of foetal hepatic parenchyma in vari-ous stages of development and foetal pancreatic tissue and foetal gastroin-testinal structures in various stages of development; (ii) the emergence of multiple bi-layer clear-cell glandules with sub-nuclear or extra-nuclear vacuoles, focally contiguous with high cellularity aggregations of asteroid/ roundish cells without marked nuc-lear atypia and low mito tic activ-ity (3–7 mitotic divisions/10 optic fields ×40); (iii) few cystic cavities and communicating cavernous spaces with clear-celled investment or invest-ment from highly eosinophilic cells; (iv) aggregations of follicles of thyroid gland in growth phase.

The mesoderm was represented from the meninge, the smooth mus-cle fasciae in developmental phase and organising in smooth muscle layer, the striated muscle fibres of embryonal type, the multiple sites of immature mesenchyma, the areas of embryonal and mature adipose tissue, the islets of immature and mature hyaline cartilage, the islets of neo-formed coarse bone tissue and the osteoid with mild hyperpla-sia of the osteoblasts. Multiple foci of extramedullary haematopoiesis and important haemorrhagic infiltration in some areas, and ischaemic type necrosis (20%) and focal deposits of calcium salts were also observed.

Extension of infiltrative type was observed in the consecutive striated muscle fibres and in the co-excised portion of cartilage and bone tissue and beyond, in the form of cerebral substance and cavities lined by chori-oid plexus or clear cells. Also, expan-sion of the tumour was observed in the form of cavities coated with res-piratory epithelium type or muci-nous gastric type or colonic cells up to the reticular dermis of the skin and without expansion to the skin. Additionally, within the tumour,

an encapsulation of portions of large venous and arterial vascular branches and growth of islets of foe-tal liver parenchyma and glia in the wall of the middle venous vases of the area of the co-excised cartilage tissue were found. The operative margins showed neoplastic expan-sion to the fibro-adipose and dense fibrous connective tissue. There was no tumoural infiltration in the two tiny nodes with maximum diameter of 0.4 and 0.5 cm respectively, which were found within the co-excised fibro-adipose tissue.

ImmunohistochemistryThe immunohistochemical exami-nation showed expression of kera-tin 8.18 in the epithelial structures and expression of GFAP in the glia. Also, expression of synaptophysin in the neurons and expression of EMA in the investment tubules and the lumen of the glandular structures including clear-cell glandules were found. In addition focal expression of Glypican 3 in the clear-cell glandules was detected. Expression of AFP was not observed in the epithelial struc-tures including the few clear-cell glandules. The expected expression of AFP and Glypican 3 in the islets of foetal liver was observed as well. The cell proliferation marker Ki-67/MIB-1 was detected heterogeneously in the epithelial structures (5%) and in the immature foci of the immature neuroepithelium (15%–20%).

DiscussionIn our case, the tumour was an extragonadal teratoma of the sacro-coccygeal area of the immature type with a maximum diameter of 18 cm and maturity level 3. In the tumour there was representation of all the three blastic layers with prevalence of ectoderm elements, mostly neu-roectoderm, important representa-tion of the endoderm and visible representation of the mesoderm. Infiltrative type extension of the neoplasm in the adjacent striated

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muscle fibres and co-excised portion of cartilage/coccyx bone tissue and also beyond, in the adipose tissue was observed. The surgical margins showed extension of the tumour in the fibro-adipose tissue.

The gradation of immaturity of the extragonadal immature tera-tomas in the paediatric population in accordance to the criteria refer-ring to the ovarian teratomas does not reflect the biological behaviour of the tumour, which is determined by the presence or absence of yolk sac tumour elements. The immu-nohistochemical detection of AFP and Glypican 3 is expected in islets of foetal liver, as in our case, with-out being a yolk sac tumour com-ponent. Detection of Glypican 3 in clear-cell glandules with parallel EMA expression and absence of AFP expression does not document a yolk sac tumour diagnosis of intes-tinal type. Elements of other germ cell tumours, including the yolk sac tumour, were not recognised in the tumour of our case.

Recurrences after resections of sacrococcygeal teratomas have been described and half of the recurrences are malignant. As the tumour is che-mosensitive, follow-up with serum AFP levels and physical examina-tion for at least three years is recom-mended postoperatively7,18,26. The AFP levels are high in foetuses and return to normal at the ninth postpartum month. Therefore, any rapid postop-erative increase in AFP levels suggests tumoural recurrence7,18,26. In our case, there was no evidence of recurrence until the ninth month of the follow-up period despite the diagnosis of imma-ture sacrococcygeal teratoma.

Urologic complications of the con-genital sacrococcygeal teratomas have been described such as uri-nary retention due to bladder outlet compression of tumour followed by secondary renal deterioration, olig-ohydramnios and pulmonary hypo-plasia. Compression or infiltration of the sacral nerves by the tumour

and surgical trauma to hypogastric nerves during tumour resection cause damage to the innervation of the lower urinary tract in sacrococ-cygeal teratoma11,14,27–31. In addition, neuropathic bladder, vesicoureteric reflux, incontinence, recurrent uri-nary tract infections and impo-tence are possible complications after resection of sacrococcygeal teratomas10,29,32,33.

The prenatal diagnosis of the sac-rococcygeal teratomas can be easily made with conventional two-dimen-sional ultrasound scan12. In the first trimester these neoplasms usually escape the ultrasonographic diagno-sis because of their small size12,13. The advantages of the three-dimensional ultrasound scan are the definition of the total volume of the neoplasm, its extension to the pelvis and the pos-sible skeletal involvement for a better surgical excision12. Rapidly growing huge tumours with predominantly solid components are often recog-nised in cases with an immature his-tology as in our case or malignant histology. Also, in these cases a high vascularisation during the colour Doppler examination is usually recog-nised17,34,35. In our case, RI of the blood flow was 0.51 and it is in accordance with the other literature reports, in which the blood flow RI is lower in the immature or malignant than in mature sacrococcygeal teratomas but not as low as in adult women with ovarian cancer (cut-off levels <0.40)17,34,35. The differential diagnosis at mid-gesta-tion of a sacral cystic mass includes meningomyelocele and urogenital anomalies12. Although chromosomal anomalies have been found in cases with sacrococcygeal teratomas, foe-tal karyotyping is not usually rec-ommended because tumoural cells can desquamate into the amniotic fluid and in case with normal foetal chromosomes, foetal karyotyping by amniocentesis may result in mosai-cism. Therefore, in these cases, the foetal karyotyping should be supple-mented with foetal blood sampling12.

In our case the foetal karyotyping was normal. No other foetal abnormalities were found in our patient; the only complication was the polyhydram-nios from the 31st week to delivery by caesarean section. There were no ultrasonographic findings of foetal hyprops or abnormal foetal echocar-diographic findings. In patients with sacrococcygeal teratomas greater than 5 cm, caesarean section is rec-ommended usually with upper verti-cal uterine incision to avoid dystocia or trauma during normal delivery18,26. In specific surgical centres, foetal interventions have been performed with some success including in utero open foetal surgery, radiofrequency tumoural ablation, laser vessel abla-tion and alcohol sclerosis in an attempt to increase survival in foe-tuses with hydrops, while the mothers are still in good health25.

ConclusionSacrococcygeal teratomas are rare germ cell tumours associated with high perinatal and postnatal mortality and morbidity. Prenatal intervention should be considered when the foetus develops hydrops for foetal salvage or in cases with placentomegaly to avoid the maternal risk of mirror syndrome.

ConsentWritten informed consent was obtained from the patient for publi-cation of this case report and accom-panying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

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