Literature DB >> 22511070

An unusual case of adult onset progressive heterotopic ossification suggesting a variant form of fibrodysplasia ossificans progressiva.

J A S B Jayasundara1, G L Punchihewa, D S de Alwis.   

Abstract

Progressive heterotopic ossification (HO) is a rare disease of genetic inheritance. Fibrodysplasia ossificans progressiva (FOP) is an identified debilitating subcategory in which anomalous ossification usually begins in childhood. Congenital big toe anomalies and specific patterns of progression of ossification confirm the classic disease. Adult onset disease is extremely rare. The mechanism of disease progression is still unclear, and there is no consensus on the treatment modalities. We report a 47-year-old man with adult-onset progressive HO around the bilateral pelvic and shoulder girdles and thoracolumbar spine, which suggested a variant form of FOP. Although surgical excision is considered counterproductive in FOP, our patient showed improvement in his shoulder movement following surgery. Other management strategies, including surgery around the hips, indomethacin prophylaxis, irradiation and bisphosphonate therapy, did not improve his range of movement or disease progression.

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Year:  2012        PMID: 22511070

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


  4 in total

1.  Fibrodysplasia Ossificans Progressiva: A Report of Four Cases.

Authors:  Mohamed Elamin; Ashraf Almutasim Ibrahim; Abdalla Omer
Journal:  Cureus       Date:  2022-03-22

Review 2.  Challenges in the treatment of fibrodysplasia ossificans progressiva.

Authors:  Kardelen Gencer-Atalay; Ekim Can Ozturk; Ilker Yagci; Pinar Ata; Kenan Delil; Zerrin Ozgen; Gulseren Akyuz
Journal:  Rheumatol Int       Date:  2018-10-20       Impact factor: 2.631

3.  Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature.

Authors:  Esmée Botman; Jan Coen Netelenbos; Thomas Rustemeyer; Linda J Schoonmade; Jakko A Nieuwenhuijzen; Bernd P Teunissen; Marieke Visser; Pieter Raijmakers; Adriaan A Lammertsma; Max Dahele; Marelise Eekhoff
Journal:  Front Endocrinol (Lausanne)       Date:  2020-02-12       Impact factor: 5.555

4.  When Limb Surgery Has Become the Only Life-Saving Therapy in FOP: A Case Report and Systematic Review of the Literature.

Authors:  Esmée Botman; Sanne Treurniet; Wouter D Lubbers; Lothar A Schwarte; Patrick R Schober; Louise Sabelis; Edgar J G Peters; Annelies van Schie; Ralph de Vries; Zvi Grunwald; Bernard J Smilde; Jakko A Nieuwenhuijzen; Marieke Visser; Dimitra Micha; Nathalie Bravenboer; J Coen Netelenbos; Bernd P Teunissen; Pim de Graaf; Pieter G H M Raijmakers; Jan Maerten Smit; Elisabeth M W Eekhoff
Journal:  Front Endocrinol (Lausanne)       Date:  2020-08-21       Impact factor: 5.555

  4 in total

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