Infliximab Reverses Symptoms and May Protect from Developing Chronic Restrictive Ophthalmopathy in Children with Familial Orbital Myositis: A Case Report

Aimee Huynh, Owen M. Siggs, Brynn K. Wainstein, Paul E. Gray

Research output: Contribution to journalLetterpeer-review

Abstract

Background: Orbital myositis is a rare sporadic eye disease associated with extraocular eye muscle inflammation. To date, there have been two reports of familial orbital myositis (FOM), which demonstrate partially penetrant autosomal dominant inheritance. Cases: We report six new Australian cases of FOM, four of whom extend one of the reported pedigrees, as well as a separate mother and daughter manifesting orbital myositis, which constitutes a third report of familial occurrence. We can confirm that the disease has onset in childhood, appearing to go into remission in adult life, and that the inflammation is corticosteroid-responsive. However, one patient went on to develop permanent diplopia in upgaze. We also report two children suffering chronic pain and diplopia who demonstrated complete resolution of symptoms with the anti-TNF-α monoclonal infliximab. Conclusion: Uncontrolled FOM in childhood may result in permanent extraocular eye muscle damage, while TNF-α blockade provides an excellent steroid-sparing effect.

Original languageEnglish
Number of pages4
JournalOcular Immunology and Inflammation
Early online date27 May 2022
DOIs
Publication statusE-pub ahead of print - 27 May 2022

Keywords

  • Autosomal dominant
  • children
  • familial
  • infliximab
  • orbital myositis

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