The First Case of Multifocal Osteonecrosis in Behcet’s Disease

  • Seyedeh Tahereh Faezi Rheumatology Research Center, Tehran University of Medical Sciences
  • Mohammad Nejadhosseinian Mail Joint reconstruction research center , Imam Khomeini hospital , Tehran university of medical science , Tehran , IR Iran
  • Farhad Shahram Rheumatology Research Center, Tehran University of Medical Sciences
  • Nahid Sadighi Tehran University of Medical Sciences
  • Masoumeh Banihashemian Tehran University of Medical Sciences
  • Pedram Paragomi Department of Internal Medicine, University of Pittsburgh Medical Center, Pittsburgh, PA, USA
  • Fereydoun Davatchi Rheumatology Research Center, Tehran University of Medical Sciences
Keywords:
Behcet’s Disease; Osteonecrosis; Avascular Necrosis of Bone

Abstract

Background: Osteonecrosis is the death of bone tissue due to the interruption of the blood supply. It can occur in autoimmune disease due to multiple mechanisms. It occurs rarely in some kind of multisystem disease such as Behcet’s disease (BD). The aim of this article is to present a case of BD with multifocal osteonecrosis.

Case Presentation: A 26-year-old woman with oral aphthosis, panuveitis, and retinal vasculitis was diagnosed to have BD, and oral treatment of prednisolone (0.5 mg/kg/day) together with azathioprine (2 mg/kg/day) and intravenous (IV) cyclophosphamide (CYC) (0.75 g/m2) was administered for her. Prednisolone was tapered to 15 mg/day after 3 months. Because of unresponsiveness of eye involvement to traditional immunosuppressive therapy, infliximab (5 mg/kg) was started for her, and IV CYC was discontinued. 4 months after initial therapy, she complained of her right shoulder pain. Regarding shoulder pain, magnetic resonance imaging (MRI) showed osteonecrosis of the right shoulder. After the diagnosis of osteonecrosis, prednisolone was discontinued by herself. 3 months later, she complained of both knees, both hips, and right ankle pain. According to the previous diagnosis of osteonecrosis of her right shoulder, MRI was done for all involved joints, which revealed osteonecrosis of all of them. After the diagnosis of multifocal osteonecrosis, the following tests were done that all results were normal: anticardiolipin antibody (ACA) [immunoglobulin G (IgG), immunoglobulin M (IgM)], lupus anticoagulant (LA), anti-beta-2 glycoprotein 1 (antiB2GP1) (IgG, IgM), coagulation tests, and lipid profiles.

Conclusions: Bone infarction should be considered in the differential diagnosis of patients with joint pain and without the evidence of arthritis; however, it rarely occurs in patients with BD.

Author Biographies

Seyedeh Tahereh Faezi, Rheumatology Research Center, Tehran University of Medical Sciences

Associate Professor, Dr. Shariati Hospital, Rheumatology Research Center, Tehran University of Medical Sciences, Tehran, Iran

Mohammad Nejadhosseinian, Joint reconstruction research center , Imam Khomeini hospital , Tehran university of medical science , Tehran , IR Iran

Orthopedic Surgeon, Department of Orthopedics, Joint reconstruction research center, Imam Khomeini Hospital, Tehran University of Medical Science, Tehran, Iran

Farhad Shahram, Rheumatology Research Center, Tehran University of Medical Sciences

Professor of Rheumatology, Dr. Shariati Hospital, Rheumatology Research Center, Tehran University of Medical Sciences, Tehran, Iran

Nahid Sadighi, Tehran University of Medical Sciences

Associate Professor of Radiology, Imam Khomeini Hospital, Tehran University of Medical Sciences, Tehran, Iran

Masoumeh Banihashemian, Tehran University of Medical Sciences

MD, Department of Radiology, Yas Women's Hospital, Tehran University of Medical Sciences, Tehran, Iran

Fereydoun Davatchi, Rheumatology Research Center, Tehran University of Medical Sciences

Professor of Rheumatology, Dr. Shariati Hospital, Rheumatology Research Center, Tehran University of Medical Sciences, Tehran, Iran

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Published
2020-05-07
How to Cite
1.
Faezi ST, Nejadhosseinian M, Shahram F, Sadighi N, Banihashemian M, Paragomi P, Davatchi F. The First Case of Multifocal Osteonecrosis in Behcet’s Disease. J Orthop Spine Trauma. 4(4):80-83.
Section
Case Report