Page 170 - MI-2-3

Microbes & Immunity

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 Moura * , Raquel Faria 2,3,4 , and Ernestina Santos 1,2,3,4
1,2
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

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
*Corresponding author: a recurrent headache, which had persisted for several years, and diffuse arthralgias.
João Moura A computed tomography-angiogram demonstrated the narrowing of the left
(moura.neuro@chporto.min-saude.pt) carotid canal. The patient had an erythrocyte sedimentation rate of 19 mm/s, and
Citation: Moura J, Faria R, her cerebrospinal fluid contained 20 leukocytes/µL (13 mononuclear) and 0.43 g/L of
Santos E. Migraine-like headache proteins. Bone scintigraphy showed radiotracer uptake in the frontal bones and spine.
due to frontal osteitis and meningitis
in a delayed diagnosis of SAPHO The patient’s son had previously been diagnosed with SAPHO syndrome; therefore,
syndrome: A case report and the possibility of a disorder in the same spectrum was considered. Corticotherapy
literature review. Microbes & followed by sulphasalazine resolved the symptoms. This case illustrates an atypical
Immunity. 2025;2(3):162-167.
doi: 10.36922/mi.4667 cranial and meningeal involvement of the SAPHO autoinflammatory process. Our
findings underscore the importance of considering immune-mediated disorders in
Received: August 27, 2024
the differential diagnosis of headaches.
Revised: September 12, 2024
Accepted: October 18, 2024 Keywords: Migraine; Headache; Frontal osteitis; SAPHO syndrome; Autoinflammatory
Published online: November 28, syndrome
2024
Copyright: © 2024 Author(s).
This is an Open-Access article
distributed under the terms of the 1. Background
Creative Commons Attribution
License, permitting distribution, The distinction between primary and secondary headache disorders is pivotal in clinical
and reproduction in any medium, practice. Several immune-mediated diseases occur with headache, particularly migraine.
1
provided the original work is
properly cited. Synovitis, acne, pustulosis, hyperostosis, and osteomyelitis (SAPHO) syndrome is a
polygenic autoinflammatory disorder associated with conditions involving the central
Publisher’s Note: AccScience
Publishing remains neutral with nervous system, including headache. 2
regard to jurisdictional claims in
published maps and institutional This report presents the case of a patient with SAPHO syndrome, featuring headache
affiliations. as the predominant clinical feature, highlighting the multifaceted nature of headache

Volume 2 Issue 3 (2025) 162 doi: 10.36922/mi.4667