Journal of Plastic, Reconstructive & Aesthetic Surgery
Volume 63, Issue 3 , Pages 390-397 , March 2010

Congenital duplication of lower extremity – A case report and review of the literature

  • Y. Osaki

      Affiliations

    • Department of Plastic Surgery, Kobe Children's Hospital, 1-1-1 Takakuradai, Suma, Kobe, Hyogo, Japan
    • ,
  • S. Nishimoto

      Affiliations

    • Department of Plastic Surgery, Kobe Children's Hospital, 1-1-1 Takakuradai, Suma, Kobe, Hyogo, Japan
    • Department of Plastic Surgery, Hyogo College of Medicine, 1-1 Mukogawacho, Nishinomiya, Hyogo, Japan
    • Corresponding Author InformationCorresponding author. Tel.: +81 798 45 6753; fax: +81 798 45 6975.
    • ,
  • T. Oyama

      Affiliations

    • Department of Plastic Surgery, Kobe Children's Hospital, 1-1-1 Takakuradai, Suma, Kobe, Hyogo, Japan
    • ,
  • Y. Yoshimura

      Affiliations

    • Department of Plastic Surgery, Kobe Children's Hospital, 1-1-1 Takakuradai, Suma, Kobe, Hyogo, Japan

Received 29 September 2008 ,Accepted 14 January 2009.

References 

  1. O'Rahilly R, Müller F. Developmental Stages in Human Embryos. Washington: Carnegie Institution of Washington Publication; 1987;
  2. Roberts DJ, Tabin C. The genetics of human limb development. Am J Hum Genet. 1994;55:1–6
  3. Haramis AG, Brown JM, Zeller R. The limb deformity mutation disrupts the SHH/FGF-4 feedback loop and regulation of 5′ HoxD genes during limb pattern formation. Development. 1995;121:4237–4245
  4. Laufer E, Nelson CE, Johnson RL, et al. Sonic hedgehog and Fgf-4 act through a signaling cascade and feedback loop to integrate growth and patterning of the developing limb bud. Cell. 1994;79:993–1003
  5. Kimura J, Ide H. Shh, Bmp-2 and Hoxd-13 gene expression in chick limb bud cells in culture. Dev Growth Differ. 1998;40:457–464
  6. Packard DS, Levinsohn EM, Hootnick DR. Extent of duplication in lower-limb malformations suggests the time of the teratogenic insult. Pediatrics. 1993;91:411–413
  7. Wolpert L. Vertebrate limb development and malformations. Pediatr Res. 1999;46:247–254
  8. Sessions SK, Ruth SB. Explanation for naturally occurring supernumerary limbs in amphibians. J Exp Zool. 1990;254:38–47
  9. Hanley EN, Stanitski CL. Incomplete congenital duplication of a lower extremity. A case report. J Bone Joint Surg Am. 1980;62:479–481
  10. McCredie J, Loewenthal J. Pathogenesis of congenital malformations. A hypothesis. Am J Surg. 1978;135:293–297
  11. Strecker TR, Stephens TD. Peripheral nerves do not play a trophic role in limb skeletal morphogenesis. Teratology. 1983;27:159–167
  12. Levinsohn EM, Hootnick DR, Packard DS. Consistent arterial abnormalities associated with a variety of congenital malformations of the human lower limb. Invest Radiol. 1991;26:364–373
  13. Packard DS, Levinsohn EM, Hootnick DR. Most human lower limb malformations appear to result from postspecification insults. Prog Clin Biol Res. 1993;383A:417–426
  14. Griffet J, Bastiani-Griffet F, Jund S, et al. Duplication of the leg–renal agenesis: congenital malformation syndrome. J Pediatr Orthop B. 2000;9:306–308
  15. Charite J, de Graaff W, Shen S, et al. Ectopic expression of Hoxb-8 causes duplication of the ZPA in the forelimb and homeotic transformation of axial structures. Cell. 1994;78:589–601
  16. Landauer W. Rudimentation and duplication of the radius in the duplicate mutant form of fowl. J Genet. 1956;54:199–218
  17. Hummel K. The inheritance and expression of disorganization, an unusual mutation in the mouse. J Exp Zool. 1958;137:389–423
  18. Cavalcanti DP. Unknown syndrome: abnormal facies, hypothyroidism, postaxial polydactyly, and severe retardation: a third patient. J Med Genet. 1989;26:785–786
  19. Kurtoglu S, Caksen H, Erdogan R, et al. Collodion baby concomitant with congenital hypothyroidism: a patient report and review of the literature. J Pediatr Endocrinol Metab. 1998;11:569–573
  20. Yanai N, Shveiky D. Fetal hydrops, associated with maternal propylthiouracil exposure, reversed by intrauterine therapy. Ultrasound Obstet Gynecol. 2004;23:198–201
  21. Van Groesen PJ, Beemer FA, Van de Kamp JH. Leg duplication and kidney agenesis: case report and pathogenic considerations. Genet Couns. 1990;1:265–272
  22. Weiner DS, Greenberg B, Shamp N. Congenital reduplication of the femur associated with paraxial fibular hemimelia. Report of a case. J Bone Joint Surg Am. 1978;60:554–555
  23. Kojima T, Hirakawa M, Hirase Y, et al. Complete congenital duplication with incomplete separation of a lower extremity. Plast Reconstr Surg. 1993;91:926–929
  24. Hootnick DR, Packard DS, Levinsohn EM. Congenital tibial aplasia with preaxial polydactyly: soft tissue anatomy as a clue to teratogenesis. Teratology. 1983;27:169–179
  25. Cornah MS, Dangerfield PH. Reduplication of the femur. Report of a case. J Bone Joint Surg Br. 1974;56-B:744–745
  26. Billett DM, Bear JN. Partial duplication of the lower limb. A case report. J Bone Joint Surg Am. 1978;60:1143–1145
  27. Hamanishi C, Ueba Y, Iwashita Y, et al. Diplopodia with reversed foot. Normal gait after operation at 8 years of age. Acta Orthop Scand. 1985;56:439–441
  28. Gould GaPW. Anomalies and Curiosities of Medicine. London: The Rebman Publishing Co.Ltd.; 1898;
  29. Zammit J. Posterior dichotomy: a three-legged boy. Br Med J. 1898;1:1528
  30. Deboo S. A three-legged person. Med Bull Bombay. 1943;11:339
  31. Smillie IS, Murdoch JH. Man with three legs. J Bone Joint Surg Br. 1952;34-B:630–635
  32. Norman WH. A child with three lower extremities. J Bone Joint Surg Am. 1964;46:1755–1758
  33. Srivastava KK, Garg LD. Reduplication of bones of lower extremity. J Bone Joint Surg Am. 1971;53:1445–1447
  34. Taniguchi K, Aoki Y, Kurimoto H, et al. Baby with a third leg. J Pediatr Surg. 1975;10:143–144
  35. Balog B, Skinner SR. Unilateral duplication of the femur associated with myelomeningocele. J Pediatr Orthop. 1984;4:488–490
  36. Weisselberg B, Ben-Ami T, Goodman RM. Partial duplication of the lower limb with agenesis of ipsilateral kidney–a new syndrome: report of a case and review of the literature. Clin Genet. 1988;33:234–239
  37. Viljoen DL, Kidson SH. Mirror polydactyly: pathogenesis based on a morphogen gradient theory. Am J Med Genet. 1990;35:229–235
  38. Bodurtha J, Coutinho M, Benator R, et al. Femoral duplication: a case report. Am J Med Genet. 1989;33:165–169
  39. Rivera RE, Hootnick DR, Gingold AR, et al. Anatomy of a duplicated human foot from a limb with fibular dimelia. Teratology. 1999;60:272–282
  40. Ganey TM, Carey TP, O'Neal ML, et al. Morphologic and radiographic characterization of fibular dimelia. J Pediatr Orthop B. 2000;9:293–305
  41. Brower JS, Wootton-Gorges SL, Costouros JG, et al. Congenital diplopodia. Pediatr Radiol. 2003;33:797–799
  42. Wada M, Kawabata H, Miki K, et al. A case with congenital duplication of the lower extremity at the groin. Kinki-Shoni-Seikeigeka. 2004;16:25–28
  43. Zhao L, Li MQ, Sun XT, et al. Congenital lumbosacral limb duplication: a case report. J Orthop Surg (Hong Kong). 2006;14:187–191
  44. Lilje C, Finger LJ, Ascuitto RJ. Complete unilateral leg duplication with ipsilateral renal agenesis. Acta Paediatr. 2007;96:464–465

PII: S1748-6815(09)00085-0

doi: 10.1016/j.bjps.2009.01.030

Journal of Plastic, Reconstructive & Aesthetic Surgery
Volume 63, Issue 3 , Pages 390-397 , March 2010

Article Tools

* Image Usage & Resolution