Literature DB >> 817388

[Melorheostosis in adults. Apropos of 2 cases, 1 of them treated with diphosphonate (EHDP)].

J C Bied, C Malsh, P Meunier.   

Abstract

The authors report two cases of melorheostosis discovered in adults because of fairly intense pain and deformation of the affected limb. The radiological appearances were typical. Isotope bone scintigraphy showed strong hyperfixation in the affected bones. A histomorphometric study of a double iliac bone biopsy showed on the one hand, cortical hypertrophy, normal osteonic architecture, and normal lamellar texture in the pathological regions, and, on the other hand, the dynamic character of the healthy bone close the lesions. In one of the patients an original attempt at treatment with diphosphonate (1 200 mg/day of EHDP for 5 months) resulted in a clear improvement in the bone pain. The disease is considered to be a mesodermic dystrophy. It seems to be due to strong hyperactivity of the subperiosteal bone accretion, which, normally, persists only at a weak level in adults.

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Year:  1976        PMID: 817388

Source DB:  PubMed          Journal:  Rev Rhum Mal Osteoartic        ISSN: 0035-2659


  6 in total

1.  Leri's melorheostosis: three pediatric cases and a review of the literature.

Authors:  P Beauvais; C Fauré; J P Montagne; P L Chigot; P Maroteaux
Journal:  Pediatr Radiol       Date:  1977-10-28

2.  Melorheostosis and its treatment with intravenous zoledronic acid.

Authors:  Rosemary Jane Hollick; Alison Black; David Reid
Journal:  BMJ Case Rep       Date:  2010-04-05

3.  Successful treatment of pain in melorheostosis with zoledronate, with improvement on bone scintigraphy.

Authors:  Samy Slimani; Adlen Nezzar; Hachemi Makhloufi
Journal:  BMJ Case Rep       Date:  2013-06-21

4.  A Unique Case of Melorheostosis Presenting with Two Radiologically Distinct Lesions in the Shoulder.

Authors:  Ahmed A Elsheikh; Rohan S Pinto; Alpesh Mistry; Simon P Frostick
Journal:  Case Rep Orthop       Date:  2017-11-06

5.  Clinical improvement in a patient with monostotic melorheostosis after treatment with denosumab: a case report.

Authors:  Sarah Byberg; Bo Abrahamsen; Moustapha Kassem; Stuart Ralston; Peter Schwarz
Journal:  J Med Case Rep       Date:  2018-09-27

6.  Dripping Wax Bone Disease - Melorheostosis - A Rare Case Scenario.

Authors:  M Lokesh; Sundar Suriya Kumar; Saikrishna Gadde
Journal:  J Orthop Case Rep       Date:  2020-12
  6 in total

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