Literature DB >> 24763843

Melorheostosis of the hand affecting the c6 sclerotome and presenting with carpal tunnel syndrome.

Shalimar Abdullah, Noreen Fazlina Mat Nor, Nor Hazla Mohamed Haflah1.   

Abstract

Melorheostosis is a rare, progressive bone disease accompanied by hyperostosis and soft tissue fibrosis. While affected adults present with contracture and pain, children present with limb length discrepancy and deformity. We report the case of a 20-year-old woman with melorheostosis since childhood who presented with right hand deformity and numbness. Radiographs showed not only a combination of dense sclerosis and opacities, but also the classic 'flowing candle wax' appearance. Radiography can be used to identify melorheostosis, thus preventing unnecessary bone biopsies. Carpal tunnel release revealed the presence of a thickened flexor retinaculum and a degenerated median nerve distal to the retinaculum, but did not show hyperostosis. This case highlights the role of nerve decompression in melorheostosis and the importance of early identification of the disease to prevent unnecessary bone biopsies.

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Year:  2014        PMID: 24763843      PMCID: PMC4291949          DOI: 10.11622/smedj.2014060

Source DB:  PubMed          Journal:  Singapore Med J        ISSN: 0037-5675            Impact factor:   1.858


  9 in total

1.  Melorheostosis of the skeletally immature hand: a case report and long-term follow-up evaluation.

Authors:  R Rozencwaig; M R Wilson; G B McFarland
Journal:  J Hand Surg Am       Date:  1996-07       Impact factor: 2.230

Review 2.  Classical and unusual imaging appearances of melorheostosis.

Authors:  S Suresh; T Muthukumar; A Saifuddin
Journal:  Clin Radiol       Date:  2010-04-18       Impact factor: 2.350

3.  Melorheostosis. A report of the clinical, roentgenographic, and pathological findings in fourteen cases.

Authors:  C J Campbell; T Papademetriou; M Bonfiglio
Journal:  J Bone Joint Surg Am       Date:  1968-10       Impact factor: 5.284

4.  Melorheostosis of the ulna.

Authors:  Shalimar Abdullah; Gerry M H Pang; Nor Hazla Mohamed-Haflah; Jamari Sapuan
Journal:  J Chin Med Assoc       Date:  2011-10-20       Impact factor: 2.743

5.  Melorheostosis: a review of 23 cases.

Authors:  J Freyschmidt
Journal:  Eur Radiol       Date:  2001       Impact factor: 5.315

6.  Melorheostosis of the hand with complicating bony spur formation and bursal inflammation: diagnosis and treatment.

Authors:  S Ameen; L Nagy; U Gerich; S E Anderson
Journal:  Skeletal Radiol       Date:  2002-06-27       Impact factor: 2.199

7.  Soft tissue contractures from melorheostosis involving the upper extremity.

Authors:  D L Pruitt; P R Manske
Journal:  J Hand Surg Am       Date:  1992-01       Impact factor: 2.230

8.  Congenital carpal tunnel syndrome.

Authors:  T Barfred; T Ipsen
Journal:  J Hand Surg Am       Date:  1985-03       Impact factor: 2.230

9.  Melorheostosis in children. Clinical features and natural history.

Authors:  D Younge; D Drummond; J Herring; R L Cruess
Journal:  J Bone Joint Surg Br       Date:  1979-11
  9 in total

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