Aseptic Meningitis as a Rare Manifestation of Sapho Syndrome

Hiwatani, Yasuhiro; Miwa, Hideto; Kondo, Tomoyoshi

doi:10.1212/01.wnl.0000334758.75290.d0

Cited by 8 publications

(9 citation statements)

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“…The main characteristic feature is lesions most frequently located on the anterior chest wall; however, the syndrome is occasionally associated with skull lesions that may result in recurrent headache episodes of unknown origin. There have also been reported cases of SAPHO syndrome with intracranial lesions, including cranial nerve palsy and thickening of the dura mater (1,2). We herein describe a case of SAPHO syndrome with repeated unilateral headaches and dura mater thickening.…”

Section: Introductionmentioning

confidence: 73%

“…Besides the anterior chest lesions, SAPHO syndrome is known to be frequently related to lesions involving the spine (7), articulatio sacroiliaca, os longum, and os plana, as well as peripheral arthritis (8). In addition, a limited number (four cases) of skull bone involvement have been reported (1,2,9,10). In all of these cases except for one with SAPHO syndrome leading to deafness (9), the patient suffered from headache.…”

Section: Discussionmentioning

confidence: 99%

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Recurrent Unilateral Headache Associated with SAPHO Syndrome

et al. 2014

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A 57-year-old woman was admitted with recurrent episodes of right frontal headache. Head magnetic resonance imaging (MRI) revealed extensive thickening and enhancement of the right frontal dura, muscle and fascia, as well as abnormal signal intensity and enhancement of bone marrow at the lesions. Synovitis-acnepustulosis-hyperostosis osteomyelitis (SAPHO) syndrome was diagnosed based on the patient's 8-year history of treatment of palmoplantar pustulosis and abnormal accumulations in the right temporal, sternum, and left medial clavicula on bone scintigraphy. SAPHO syndrome may be associated with skull lesions, which can contribute to the onset of repeated headache or dural thickening, thus these symptoms should be recognized as manifestations of this syndrome.

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Section: Introductionmentioning

confidence: 73%

Section: Discussionmentioning

confidence: 99%

Recurrent Unilateral Headache Associated with SAPHO Syndrome

et al. 2014

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“…SAPHO syndrome primarily presents as lesions on the skin and osteitis. However, recently reported neurological symptoms that overlap with SAPHO syndrome are headache as a result of SAPHO‐related inflammation, mixed‐type hearing loss and aseptic meningitis . Mechanisms underlying these symptoms are not completely understood, but are thought to be due to osteitis‐induced inflammation of the dura mater .…”

Section: Discussionmentioning

confidence: 99%

“…1,2 SAPHO syndrome is a neurocutaneous syndrome, but its pathophysiology is not well known. [3][4][5] Here we report on a patient who presented with refractory headache and polyneuritis (cranial nerves I, II and VII) overlapping with SAPHO syndrome.…”

Section: Introductionmentioning

confidence: 92%

“…Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome is a skin and bone disease characterized by skin lesions and osteitis . SAPHO syndrome is a neurocutaneous syndrome, but its pathophysiology is not well known . Here we report on a patient who presented with refractory headache and polyneuritis (cranial nerves I, II and VII) overlapping with SAPHO syndrome.…”

Section: Introductionmentioning

confidence: 95%

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Polyneuritis and refractory headache overlap with synovitis, acne, pustulosis, hyperostosis and osteitis syndrome

Matsuzono

Hishikawa

Ohta

et al. 2015

Neurology & Clinical Neurosc

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The primary clinical manifestations of synovitis, acne, pustulosis, hyperostosis and osteitis syndrome are skin lesions and osteitis. We describe a 53-year-old woman patient suffering from overlapping refractory headache and polyneuritis (cranial nerves I, II and VII). She complained of headaches, which recurred every month, followed by bilateral anosmia and left vision loss approximately 1 year later. Prednisolone therapy improved her symptoms; however, her refractory headache, pain in the right shoulder and left hip joint, and rash recurred 1 year after prednisolone withdrawal. Also, the patient presented with left sensorineural deafness and extensive acne on her body, and bone scintigraphy and bone biopsy showed synovitis, acne, pustulosis, hyperostosis and osteitis syndrome. After treatment, her symptoms improved without recurrence. Although synovitis, acne, pustulosis, hyperostosis and osteitis syndrome induced by polyneuritis is rare, it should be considered when clinicians encounter patients presenting with headache and polyneuritis of unknown cause.

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Diagnosing the SAPHO syndrome: a report of three cases and review of literature

et al. 2013

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SAPHO, an acronym for synovitis, acne, pustulosis, hyperostosis and osteitis, is a heterogeneous entity with myriad presentations and features overlapping with other entities. It is a differential in patients presenting with skin and bone symptoms, either singly or in combination. Often misdiagnosed radiologically as a malignancy or infection, the diagnosis is seldom thought of. We present three cases referred to us for evaluation of findings unrelated to the presenting symptoms. After evaluation, a (99)Tc bone scan was ordered, which showed the 'bull's head sign' in all the three cases, confirming the diagnosis. We review the literature for SAPHO. It has a few features which point to its diagnosis and can help us to distinguish it from other seronegative arthritis. The clinician should be aware of this entity and should not hesitate to order a (99)Tc bone scan. We conclude that SAPHO is not rare, but rather, it is underdiagnosed. High index of suspicion is necessary for diagnosis. A (99)Tc bone scan is diagnostic and should be ordered in patients having any of the presenting features of the syndrome. We put forward the suggestion of using (99)Tc bone scintigraphy to define a 'pre-MRI' stage of ankylosing spondylitis.

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Aseptic Meningitis as a Rare Manifestation of Sapho Syndrome

Cited by 8 publications

References 0 publications

Recurrent Unilateral Headache Associated with SAPHO Syndrome

Recurrent Unilateral Headache Associated with SAPHO Syndrome

Polyneuritis and refractory headache overlap with synovitis, acne, pustulosis, hyperostosis and osteitis syndrome

Diagnosing the SAPHO syndrome: a report of three cases and review of literature

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