A rare association of deformities with diplopodia, aplasia of the tibia and double fibula: A case report
© Khan et al; licensee BioMed Central Ltd. 2008
Received: 14 June 2007
Accepted: 07 April 2008
Published: 07 April 2008
The association of fibular duplication with metatarsal diplopodia is extremely rare with only a few cases reported in the medical literature.
We present a 4-month-old girl with left tibial agenesis with fibular duplication (mirror foot) and metatarsal diplopodia.
The case report highlights the need for an understanding of this rare congenital anomaly which may be seen only once in the working lifetime of an orthopaedic surgeon.
Diplopodia, which is an accessory tarsal or metatarsal bone with double fibula, is an extremely rare condition. It has to be differentiated from polydactyly, where accessory tarsal or metatarsal bones are not seen and is a relatively innocuous condition both in terms of diagnosis and management.
The equino-varus element of the foot deformity was corrected using serial casting. The child was kept under observation and the parents were advised about the possibility of a future above knee amputation being necessary.
Diplopodia (accessory tarsal or metatarsal bone) with double fibula is an extremely rare condition. It has to be differentiated from polydactyly, where accessory tarsal or metatarsal bones are not seen . The condition is known to be associated with congenital heart anomalies, mainly atrial septal defect . There was no congenital heart anomaly in our patient. Authors have reported a wide array of soft tissue anomalies along with the bony deformities in dissected specimens of these limbs [3, 4]. Duplication of the triceps surae muscles and of the extensor hallucis muscle is common .
Treatment is controversial. Initial treatment is conservative with plaster application to correct the equinovarus at the ankle. Following plaster applications, surgical removal of the supernumerary foot should be undertaken followed by reconstruction of the ankle and knee joints. If the limb length discrepancy is extreme, or if the deformity at the ankle is grotesque, amputation can be performed to limit further disability and improve the quality of life of the child .
Our case report highlights a rare congenital association between fibular duplication and metatarsal diplopodia. We feel that all babies with abnormal accessory toes should be evaluated for this particular anomaly and X-rays should be taken of the leg, rather than of the foot alone, to ensure a proper diagnosis.
We are thankful to the parents of our patient who kindly consented to allow us to publish this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- Karchinov K: Congenital diplopodia with hypoplasia or aplasia of the tibia. J Bone Joint Surg Br. 1973, 55: 604-611.PubMedGoogle Scholar
- Rivera RE, Hootnick DR, Gingold AR, Levinsohn EM, Kruger LM, Packard DS: Anatomy of a duplicated human foot from a limb with fibular dimelia. Teratology. 1999, 60 (5): 272-82. 10.1002/(SICI)1096-9926(199911)60:5<272::AID-TERA8>3.0.CO;2-E.View ArticlePubMedGoogle Scholar
- Narang IC, Mysorekar VR, Mathur BP: Diplopodia with double fibula and agenesis of tibia – a case report. Journal Bone Joint Surg (Br). 1982, 64 (2): 206-209.Google Scholar
- Igou RA, Kruger LM: Fibula dimelia in association with ipsilateral proximal focal femoral deficiency, tibial deficiency, and polydactyly. A case report. Clin Orthop Relat Res. 1990, 237-41. 258
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.