Adamantinoma of the Tibia: A Case Report

 

What to Learn from this Article?

Slow growing by locally aggressive tumor of tibia. differential diagnosis and management


Case Report |  Volume 4 | Issue 1 | JOCR Jan-Mar 2014 | Page 15-17 | Anoumou M N, Kouame M, Koffi E K, Varango G

DOI: 10.13107/jocr.2250-0685.140


Authors: Anoumou M N[1],Kouame M[1], Koffi E K[2], Varango G[1]

[1]Department of Traumatology and Orthopaedic surgery, University Hospital of Treichville. Côte d’Ivoire.

[2]Department of  Anatomy and pathology, University Hospital of Treichville. Côte d’Ivoire.

Address of Correspondence:

Dr Anoumou N. Michel, Department of Traumatology and Orthopaedic surgery, University Hospital of Treichville. Côte d’Ivoire. Email: anoumoumichel@yahoo.com


Abstract

Introduction: Adamantinoma is a bone tumor which is commonly showed in mandible. Its occurrence in long bones constitutes a rare affection and a difficult histological diagnosis This rare malignant tumor of mesenchymal and epithelial origin was discovered in the tibia of a male patient.

Case Report: The authors report a case of adamantinoma of the tibia in a 36- years old. He presented with slowly progressing leg swelling that evolved over 2 years without significant clinical manifestation. Complementary examination and investigation did not help in differential diagnosis and confirmation was only possible after histological examination. There was increase in size of lesion with increased pain and patie3nt presented with pathological fracture. Local extension of the tumor indicated an amputation of leg. The histological exam of the amputation specimen confirmed the results of the initial biopsy by showing cells tumorales in favour of an adamantinoma. After a good medium-term follow up, the patient died secondary to lung metastases. The authors discuss their method of diagnosis and therapeutics in front of such tumor.

Conclusion: Adamantinoma is characterized by a slow clinical development marked by the prevalence of lung metastases and local recurrences. After the diagnosis, there must be a complete and sufficient surgery to avoid a local recurrence or metastasis.

Keywords: Adamantinoma, Tibia, Tumor.


Introduction

We report a case of a young female soccer player affected by congenital medial bilateral malleolus pseudoarthrosis and os subfibulare. Congenital pseudoarthrosis is the failure of the bones to fuse prior or at birth.  The etiology is still unknown, although frequency is high in subjects affected by neurofibromatosis or correlated syndromes, so it has been suggested that these congenital disorders may be the cause of congenital pseudoarthrosis.

Case Report

Our patient, a 16-year-old female, high level soccer player, was referred to us following a right ankle sprain during a match. She reported no medical history of tibia-tarsus joint injuries or disease. Pain, swelling and functional impairment were noted immediately after the accident. Standard radiographs in the Emergency Department revealed a displaced fracture of the medial malleolus and the presence of os subfibularis (Fig. 1-2). The patient was transferred to our Traumatology and Orthopedic Department to undergo malleolus ostheosynthesis. Before surgery swelling, functional impairment and intense pain at the medial malleolus level were confirmed. However, there was no ankle relaxation, instability or pronation pain; furthermore the flexion-extension was preserved with slight pain. Twenty-four hours later a considerable remission of symptoms was evident: increase of the R.O.M. and reduction in the swelling and the post-traumatic edema. A radiograph on the left ankle to compare with that of the right ankle was necessary to overcome the discrepancy between the instrumental diagnosis and the clinical examination. The radiographic results of both medial malleoli were comparable although on the left the os subfibularis was absent. (Fig 3). Since the diagnosis of fracture by the association between the radiographs and the symptomatology was doubtful, a bilateral CT was performed. The scan revealed a medial bilateral malleolus pseudoarthrosis and an accessory right subfibularis nucleus (Fig. 4-5-6). The patient did not have any history of trauma in the past and also the bilateral nature of the disease forced us to think in terms of congenital etiology. The patient was discharged from hospital with the diagnosis of “second degree right ankle sprain in patient affected by congenital medial bilateral malleolus pseudoarthrosis”. A therapeutic-rehabilitative program was prescribed for the ankle sprain and unnecessary surgery was avoided. After 30 days there was an almost complete remission of pain. At a follow-up of six months the patient was completely asymptomatic and gradually began competitive activity.

Discussion

Any discrepancy between clinical diagnosis and radiological findings should be carefully evaluated. The classification most commonly used to evaluate pseudarthrosis is that described by Boyd as it includes clinical examination, radiological signs and findings prognosis. Nevertheless this patient did not fit any type. The presence of os subfibularis lead the formation of os tibialis and secondary pseudarthrosis during developmental phase of the epiphysis. [7] The radiographs revealed a different morphology between medial and lateral malleolus. Accessory growing nucleus generally have smooth and defined borders as were observed in the peroneal malleolus. On the contrary, the radiographs of the medial malleolus showed jagged borders and spurs, consistent with pseudarthrosis. [8,9] Heterotypical ossification can occur in any malleolus. It is not known if they are present as an anatomical variation or as the consequence of chronic microtrauma. The radiographs show them as two different entities, but they may represent one anatomical entity [10], which may undergo repeated stress during sports activities until avulsion occurs. [11] As shown with this patient, the chance finding should not induce the physician to prescribe competitive sport cessation. Generally os-accessory appears between the age of 7 and 10 years. Os tibial is present in 20% of healthy ankles, os fibular in 1%. Ogden [11], evaluating 103 patients, observed possible bilateral accessory ossification often diagnosed following an accident leading to ankle sprain. By evaluating the bone borders, a differential diagnosis between accessory nucleus and pseudarthrosis is possible. [12]. Although uncommon, several cases of stress fractures in young athletes have been described in literature. [13] The Salter-Harris [14] and the Ogden classifications were consulted but they did not describe our case properly. The total absence of café-au-lait spots and neurofibromas excluded the presence of Neurofibromatosis type 1. The patient’s brother’s and mother’s radiographs did not show any pathological features. Unfortunately the father’s radiographs were unavailable. The absence of family correlations lead to the conclusion that this was probably just a sporadic case.

Conclusion

Congenital pesudoarthrosis of medial malleolus is very rare may be due to disturbance in epiphyseal development. These may be confused with traumatic fractures as seen in i=our case and may lead to inadvertent surgery. Careful assessment of bone borders and clinical examination of instability will lead to accurate diagnosis. Conservative management will be enough in these cases and presence of psudoarthrosis may not justify cessation of sports activity.

References

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5. Ogden JA, Ganey TM, Ogden DA. The histopathology of injury to the accessory malleolar ossification center. J Pediatr Orthop. 1996; 16: 61-62.
6 Grogan DP, Love SM, Ogden JA. Congenital malformation of the lower extremities. Orth Clin of North Am. 1987; 18: 537-554.

7. Love SM, Ganey T, Ogden JA. Postnatal epiphyseal development: the distal tibia and fibula. J Pediatr Orthop. 1990; 10:298-305.
8. Haraguchi N, Kato F, Hayashi H. New radiographic projections for avulsion fractures of the lateral malleolus. J Bone Joint Surg Br. 1998; 80: 684-688.
9. Rogers LF, Poznanski AK. Imaging of epiphyseal injuries. Radiology. 1994; 191: 297-308.
10. Ogden JA, Lee J. Accessory ossification patterns and injuries of the malleoli. J Pediatr Orthop. 1990; 10: 306-316.
11. Ishii T, Miyagawa S, Hayashi K. Traction apophysitis of the medial malleolus. J Bone Joint Surg Br. 1994; 76: 802-806.
12. Mandalia V, Shivshanker V. Accessory ossicle or intraepiphyseal fracture of lateral malleolus: are we familiar with these? Emerg. Med. 2005; 22;149-152.
13. Shabat S, Sampson KB, Mann G, et al. Stress fractures of the medial malleolus—review of the literature and report of a 15-year-old elite gymnast. Foot Ankle Int. 2002; 23: 647-650.

14. Salter R, Harris W. Injuries involving the epiphyseal plate. J Bone Joint Surg Am. 1993; 45: 587-622.


 

How to Cite This Article: Anoumou M N, Kouame M, Koffi E K, Varango G. Adamantinoma of the Tibia: A Case Report. Journal of Orthopaedic Case Reports 2014 Jan-Mar ;4(1): 15-17. Available from: http://www.jocr.co.in/wp/2014/01/06/2250-0685-140-fulltext/


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