AccScience Publishing / MI / Online First / DOI: 10.36922/mi.4667
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CASE REPORT

Migraine-like headache due to frontal osteitis and meningitis in a delayed diagnosis of SAPHO syndrome: A case report and literature review

João Moura1,2* ,  Raquel Faria2,3,4 ,  Ernestina Santos1,2,3,4
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1 Department of Neurology, Hospital de Santo António, Centro Hospitalar Universitário de Santo António, Porto, Portugal
2 Unit of Multidisciplinary Research in Biomedicine, Instituto de Ciências Biomédicas Abel Salazar, University of Porto, Porto, Portugal
3 Clinical Immunology Unit, Hospital de Santo António, Centro Hospitalar Universitário de Santo António, Porto, Portugal
4 Laboratory for Integrative and Translational Research in Population Health, University of Porto, Porto, Portugal
MI, 4667 https://doi.org/10.36922/mi.4667
Submitted: 27 August 2024 | Accepted: 18 October 2024 | Published: 28 November 2024
© 2024 by the Author(s). This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution 4.0 International License ( https://creativecommons.org/licenses/by/4.0/ )
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Abstract

Synovitis, acne, pustulosis, hyperostosis, and osteomyelitis (SAPHO) syndrome is a rare polygenic autoinflammatory disorder that is associated with headaches of different etiological origins. Herein we report the case of a patient with SAPHO syndrome and provide a literature review. The patient was a 64-year-old female who complained of a recurrent headache, which had persisted for several years, and diffuse arthralgias. A computed tomography-angiogram demonstrated the narrowing of the left carotid canal. The patient had an erythrocyte sedimentation rate of 19 mm/s, and her cerebrospinal fluid contained 20 leukocytes/μL (13 mononuclear) and 0.43 g/L of proteins. Bone scintigraphy showed radiotracer uptake in the frontal bones and spine. The patient’s son had previously been diagnosed with SAPHO syndrome; therefore, the possibility of a disorder in the same spectrum was considered. Corticotherapy followed by sulphasalazine resolved the symptoms. This case illustrates an atypical cranial and meningeal involvement of the SAPHO autoinflammatory process. Our findings underscore the importance of considering immune-mediated disorders in the differential diagnosis of headaches.

Keywords
Migraine
Headache
Frontal osteitis
SAPHO syndrome
Autoinflammatory syndrome
Funding
None.
Conflict of interest
E. Santos is on the advisory board for Biogen, Genzyme, Bayer, Merck, Alexion, and Novartis, and has received a grant from Roche. The authors certify that there is no conflict of interest regarding the publication of this paper.
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Microbes & Immunity, Electronic ISSN: 3029-2883 Print ISSN: 3041-0886, Published by AccScience Publishing
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