An Unusual Cause of Foot Pain in a Child:Osteochondrosis of the IntermediateCuneiformOzkan Kose, MD,1 Bahtiyar Demiralp, MD,2 Murat Oto, MD,2 and Ali Sehirlioglu, MD2

Osteochondrosis of the intermediate cuneiform is a rare entity that may cause foot pain and limping in chil-dren. We report a case of osteochondrosis of the intermediate cuneiform in a child who underwent a spon-taneous recovery after conservative treatment. Level of Clinical Evidence: 4 (The Journal of Foot & AnkleSurgery 48(4):474–476, 2009)

Key Words: apophysitis, avascular necrosis, gait, magnetic resonance image, osteochondritis,osteonecrosis

Limping in a child is a frequent reason for admission to

a foot and ankle surgeon and, in some cases, this patient pres-

ents on an emergency basis. The differential diagnosis of

a limp is extensive and includes acute trauma, infection,

neoplasm, inflammatory musculoskeletal disorders, congen-

ital defects, as well as neuromuscular and developmental

disorders (1). Pedal osteochondroses can also cause enough

pain to induce an antalgic gait (2). In regard to the pedal os-

teochondroses, the most commonly affected sites include the

tarsal navicular and the second metatarsal head, specifically

referred to as Kohler’s disease and Freiberg’s infraction,

respectively. Other sites in the foot that are susceptible to

osteochondrosis include the calcaneal apophysis, a condition

referred to as Sever’s disease, and apophysitis of the fifth

metatarsal base, which is referred to as Iselin’s disease (3).

Despite the prevalence of these more common forms of pedal

osteochondrosis, involvement of a cuneiform bone, in partic-

ular the intermediate cuneiform, is a rare event (2, 4–11).

Because of its apparent rarity, we decided to report the

following case of osteochondrosis of the intermediate cunei-

form in a young patient.

Case Report

A 4-year-and-8-month-old girl presented to the outpatient

clinic complaining of intermittent aching pain in the right

Address correspondence to: Ozkan Kose, MD, Diclekent Bulvarı, AtaslarSerhat Evleri B.Blok D:19, Peyas mh, Kayapinar, Diyarbakir, Turkey.E-mail: drozkankose@hotmail.com.

1Diyarbakir Education and Research Hospital, Orthopaedics and Trauma-tology Clinic, Diyarbakir, Turkey.

2Gulhane Military Medical Academy, Department of Orthopaedics andTraumatology, Ankara, Turkey.

Financial Disclosure: None reported.Conflict of Interest: None reported.Copyright � 2009 by the American College of Foot and Ankle Surgeons1067-2516/09/4804-0012$36.00/0doi:10.1053/j.jfas.2009.02.010

474 THE JOURNAL OF FOOT & ANKLE SURGERY

midfoot, associated with a limping gait. These symptoms

had been present for a duration of 3 months. Her parents

observed that the complaints were worse with prolonged

physical activity and walking long distances. There was no

history of antecedent trauma, penetrating injury to the foot,

or recent infection before the onset of her pain. Her past

medical history was unremarkable. Physical examination

revealed bony tenderness over the dorsum of her right

mid-foot, and there was pain on passive movements of her

mid-tarsal joints. The range of ankle dorsiflexion and plantar-

flexion, as well as inversion and eversion, were within normal

limits. Moreover, her pedal neurovascular status was grossly,

clinically normal. Radiographs of her right foot revealed

increased density and marginal irregularity localized to the

intermediate cuneiform (Fig 1). Based on these findings,

a presumptive diagnosis of osteochondrosis of the interme-

diate cuneiform was made; conservative therapy with an

arch support, acetaminophen, and rest were initiated; and

magnetic resonance image (MRI) scans as well as blood labo-

ratory studies were requested.

Routine laboratory studies, including a complete blood

count, erythrocyte sedimentation rate (ESR), C-reactive

protein (CRP), albumin, alkaline phosphatase, total bilirubin,

blood urea nitrogen, calcium, cholesterol, creatinine, glucose,

phosphorus, serum glutamic-oxaloacetic transaminase, total

protein, and uric acid were all within normal limits. The

MRI revealed loss of signal intensity on the sagittal and axial

T1-weighted images (Fig 1). Follow-up evaluation approxi-

mately 4 weeks after the initial visit indicated that the initial

therapeutic measures had been helpful, and the diagnosis of

osteochondrosis of intermediate cuneiform was considered

to be accurate. The decision was made to continue the

supportive treatments and to monitor the patient’s progress

over time. She continued to progress well and, at the time of

the final follow-up examination approximately 4 years after

the initial admission, she was completely free of signs and

FIGURE 1 A, Anteroposterior and

oblique views of the foot. B, Axial

and sagittal T1-weighted MRI ofthe foot (white arrows show the

intermediate cuneiform bone).

symptoms of osteochondrosis and had long before resumed

normal physical activity. Radiographs of her right foot at

that time revealed complete resolution of the disease (Fig 2).

Discussion

Osteochondroses are a group of idiopathic syndromes that

are characterized by abnormal enchondral ossification that

usually affects children and adolescents. Involvement of

several bones or the portions of bones has been documented

(12), and repetitive microtrauma is commonly cited as a cause

of disruption of enchondral ossification with resultant osteo-

chondrosis (13). Although the condition is generally referred

to with terms such as osteochondritis and avascular necrosis,

we believe that the precise etiology remains obscure and that

osteochondrosis is perhaps the most precise nomenclature

because it implies both vascular disruption with resultant os-

teonecrosis, articular collapse, and joint inflammation

secondary to articular incongruity.

Antalgic gait, avoidance of weightbearing on the affected

foot, compensatory postural out-toeing on the involved side,

and tenderness on palpation of the affected part are major

clinical findings associated with pedal osteochondrosis (6,

9). Direct radiography may be unremarkable at the beginning

of the disease, although in the patient who we described,

plain film changes were evident in the intermediate cunei-

form after just 3 months of symptomatology. Eventually,

decreased size, marginal irregularity, and increased density

of the involved bone are noted as a result of osteonecrosis

and structural collapse (4, 5). Technetium-99 m bone scans

have been shown to reveal increased radionuclide uptake in

area of the involved bone (7), and this is particularly true

with the small bones of the foot where decreased uptake is

not easily identified. Currently, the gold-standard imaging

technique entails identification of reduced signal intensity

on T1-weighted MRI scans (10). Although it is rather limited,

the list of differential diagnoses for this lesion entails

a number of serious conditions, including osteomyelitis,

eosinophilic granuloma, and Ewing’s sarcoma (7). In the

case described in this report, the ESR and CRP were normal,

FIGURE 2 Anteroposterior and oblique views of the foot with

normal radiographic findings at the final follow-up.

475VOLUME 48, NUMBER 4, JULY/AUGUST 2009

and there were no localized erythema and increased local

warmth associated with the area of pedal pain. Hence, osteo-

myelitis was not considered to be a likely cause of the

patient’s symptoms. Furthermore, the MRI scans were

strongly suggestive of osteonecrosis. Careful physical exam-

ination and evaluation of both imaging and laboratory

studies, along with periodic follow-up examinations, are, in

our opinion, necessary to make the diagnosis of intermediate

cuneiform osteochondrosis.

Based on our experience with the patient described in this

report, osteochondrosis of the intermediate cuneiform

appears to be a self-limited disorder, which can resolve

with time and without pathological sequelae. Physical

activity restriction and pain relief appear to be sufficient ther-

apeutic interventions (3), and biomechanical support by

means of foot orthoses, padding and strapping can also be

useful. If the pain is severe and unresponsive to analgesics,

a short-leg walking cast may be used for symptom relief

(7). One of the important points of the treatment, we feel,

is to reassure the parents that the condition is likely to be

self-limiting and to resolve with time (2, 7, 10). We were

only able to identify one reported case that required treatment

by means of decompression with multiple drill holes (8).

In conclusion, a thorough history, careful physical exami-

nation, evaluation of both laboratory and imaging studies and

a high index of suspicion are necessary to make the diagnosis

of osteochondrosis of the intermediate cuneiform, and to be

satisfied with a course of watchful waiting. Failure to

consider this rare entity, and to patiently provide supportive

therapy, may lead the clinician to treat this self-limited

disorder with potentially harmful invasive interventions.

Periodic reevaluation is an important facet of the conserva-

tive management of this disorder, and therapeutic adjustment

476 THE JOURNAL OF FOOT & ANKLE SURGERY

may be indicated if the patient’s progress warrants doing so.

In this report, we have described a very rare case of pedal os-

teochondrosis, namely one that involved the intermediate

cuneiform.

References

1. Herring JA. The limping child. In Tachdjian’s Pediatric Orthopaedics,

pp 83–94, edited by JA Herring, WB Saunders, Philadelphia, 2002.

2. Mubarak SJ. Osteochondrosis of the lateral cuneiform: another cause of

a limp in a child. A case report. J Bone Joint Surg Am 74(2):285–289,

1992.

3. Fallat LM, Morgan JH. Osteochondroses of the foot and ankle. In

McGlamry’s Comprehensive Textbook of Foot and Ankle Surgery, pp

2088, edited by AS Banks, MS Downey, DE Martin, SJ Miller, Williams

& Wilkins, Lippincott, Philadelphia, 2001.

4. Meilstrup D. Osteochondritis of the internal cuneiform, bilateral. Am J

Radiol 58:329–330, 1947.

5. Hicks BT. Osteochondritis of the tarsal second cuneiform bone. Br J Ra-

diol 26(304):214–215, 1953.

6. Zimberg J, Levitt JC, Brahim F. Osteochondrosis of the medial cunei-

form. A case report. J Am Podiatr Med Assoc 75(10):538–539, 1985.

7. Leeson MC, Weiner DS. Osteochondrosis of the tarsal cuneiforms. Clin

Orthop Relat Res 196:260–264, 1985.

8. Chew JT, Low CK, Mitra AK. A case report on bilateral avascular

necrosis of the medial cuneiforms. Ann Acad Med Singapore 24:

453–454, 1995.

9. Vaquero Martin J, Vicente-Herrera E, Pereiro de Lamo J, Vidal

Fernandez C. Osteochondritis of the medial cuneiform. J Pediatr Orthop

B 8(1):69–71, 1999.

10. Watmough PJ, Tselentakis G, Day JB. Avascular necrosis of the inter-

mediate cuneiform bone. J Pediatr Orthop B 12:402–405, 2003.

11. Buchman J. Osteochondritis of the internal cuneiform. J Bone Joint Surg

Am 15:225–232, 1933.

12. Siffert RS. Classification of the osteochondroses. Clin Orthop Relat Res

158:10–18, 1981.

13. Douglas G, Rang M. The role of trauma in the pathogenesis of the osteo-

chondroses. Clin Orthop Relat Res 158:28–32, 1981.