SAPHO Syndrome Presenting With Severe Inflammatory Back Pain (Sacroiliitis) and Rare Retinol Associated Myopathy in an Iraqi Adolescent Male: A Case Report and Literature Review

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Authors: Farah Jaafar Mahdi, A L I Al-Shammari, Mustafa Majid Hameed, Marwah Sami Ahmed, Ibrahim Khalil, Zahraa Saeed Fahad, Ahmed Dahham Hasan, Nizar Abdulateef Jassim
Cited by: 1
References: 16

Abstract

SAPHO syndrome is a rare autoinflammatory disorder characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis. Although musculoskeletal and dermatologic manifestations are well recognized, extra-articular involvement remains uncommon, particularly muscular inflammation. We report the case of a 17-year-old male who presented with severe inflammatory low back pain and extensive nodulocystic acne for which he took escalating doses of isotretinoin. Clinical examination demonstrated positive sacroiliac stress tests, proximal muscle myopathy, and dermatologic evaluation confirmed acne conglobata. Laboratory testing revealed markedly elevated inflammatory markers. Magnetic resonance imaging of the sacroiliac joints showed bilateral asymmetric active sacroiliitis, fulfilling clinical criteria for Axial Spondyloarthritis. The infectious and autoimmune workup was negative. The patient responded favorably to isotretinoin withdrawal and immunosuppressive therapy, with sustained remission following steroid withdrawal. This case describes a rare and severe isotretinoin-associated inflammatory myopathy in SAPHO syndrome, highlighting the need to expand the differential diagnosis of myopathy in SAPHO syndrome. It is imperative to exercise caution when prescribing isotretinoin to patients with SAPHO syndrome.

Fields of Study & Subjects

Osteomyelitis and Bone Disorders Research Spondyloarthritis Studies and Treatments IgG4-Related and Inflammatory Diseases

Publication details

Journal / source
Clinical Case Reports
Publisher
Wiley
Volume
14
Issue
6
Pages
e72970–e72970
Publication date
1 June 2026
Language
EN