Singapore Med J 2014; 55(4): e54-e56; http://dx.doi.org/10.11622/smedj.2014060
Melorheostosis of the hand affecting the c6 sclerotome and presenting with carpal tunnel syndrome
Abdullah S, Mat-Nor NF, Mohamed-Haflah NH
Correspondence: A/Prof Nor Hazla Mohamed Haflah, hazla1971@yahoo.com
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.
Keywords: biopsies, carpal tunnel syndrome, hand, melorheostosis
Singapore Med J 2014; 55(4): e54–e56; http://dx.doi.org/10.11622/smedj.2014060
REFERENCES
1. Suresh S, Muthukumar T, Saifuddin A. Classical and unusual imaging appearances of melorheostosis. Clin Radiol 2010; 65:593-600.
http://dx.doi.org/10.1016/j.crad.2010.02.004 |
| |
2. Ameen S, Nagy L, Gerich U, Anderson SE. Melorheostosis of the hand with complicating bony spur formation and bursal inflammation: diagnosis and treatment. Skeletal Radiol 2002; 31:467-70.
http://dx.doi.org/10.1007/s00256-002-0542-8 |
| |
3. Freyschmidt J. Melorheostosis: a review of 23 cases. Eur Radiol 2001; 11:474 - 9.
http://dx.doi.org/10.1007/s003300000562 |
| |
4. Abdullah S, Pang GM, Mohamed-Haflah NH, Sapuan J. Melorheostosis of the ulna. J Chin Med Assoc 2011; 74:469-72.
http://dx.doi.org/10.1016/j.jcma.2011.08.019 |
| |
5. Landi A, Leti Acciaro A, Della Rosa N, Pellacani A. Carpal Tunnel Syndrome: Rare causes. Berlin Heidelberg: Springer, 2007: 95-100.
http://dx.doi.org/10.1007/978-3-540-49008-1_13 |
| |
6. Pruitt DL, Manske PR. Soft tissue contractures from melorheostosis involving the upper extremity. J Hand Surg Am 1992; 17:90-3.
http://dx.doi.org/10.1016/0363-5023(92)90120-E |
| |
7. Barfred T, Ipsen T. Congenital carpal tunnel syndrome. J Hand Surg 1985; 10:246-8.
http://dx.doi.org/10.1016/S0363-5023(85)80114-3 |
| |
| 8. Campbell CJ, Papademetriou T, Bonfiglio M. Melorheostosis. A report of the clinical, roentgenographic, and pathological findings in fourteen cases. J Bone Joint Surg Am 1968; 50:1281-304. |
| |
9. Rozencwaig R, Wilson MR, McFarland GB JR. Melorheostosis of the skeletally immature hand: a case report and long term follow-up evaluation. J Hand Surg Am 1996; 21:703-6.
http://dx.doi.org/10.1016/S0363-5023(96)80033-5 |
| |
| 10. Younge D, Drummond D, Herring J, Cruss RL. Melorheostosis in children. Clinical features and natural history. J Bone Joint Surg Br 1979; 61-B:415-8 |
