A case of SAPHO syndrome with the lesions limited to the skull

Oguro, Eri; Kato, Yasuhiro; Takamatsu, Hyota; Narazaki, Masashi; Kumanogoh, Atsushi

doi:10.1093/rap/rkaa034

Cited by 3 publications

(4 citation statements)

References 6 publications

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“… 2‐7 A painful bulge on the head was described in five cases 2‐6 . Enhanced swelling in the cephalic soft tissue was observed on brain MRI 2‐6 ; while skull involvement was demonstrated as a lesion with Gd enhancement 2,3,5‐7 or increased 99m Tc‐tracer uptake, 2‐4,6,7 and enhanced dura mater was detected in five cases 2‐6 . Cervical involvement was showed in two cases 4 including our case.…”

Section: Discussionmentioning

confidence: 58%

“…As far as we know, there are six reported cases of secondary headache associated with SAPHO syndrome (Table 1). 2‐7 A painful bulge on the head was described in five cases 2‐6 . Enhanced swelling in the cephalic soft tissue was observed on brain MRI 2‐6 ; while skull involvement was demonstrated as a lesion with Gd enhancement 2,3,5‐7 or increased 99m Tc‐tracer uptake, 2‐4,6,7 and enhanced dura mater was detected in five cases 2‐6 .…”

Section: Discussionmentioning

confidence: 97%

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Cranial osteomyelitis associated with SAPHO syndrome: An underdiagnosed cause of chronic headache

Sugie¹,

Yasaki²

2022

Neurology & Clinical Neurosc

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Synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is characterized by pustular skin lesions and aseptic osteoarticular lesions. 1 The recognition of this syndrome by neurologists is important because it rarely induces skull lesion that may result in unexplained headache.Here, we describe a case of chronic headache with SAPHO syndrome. We further review previous cases and highlight a specific symptom of the headache that can be attributed to SAPHO syndrome. | C A S E REP ORTA 50-year-old woman with a 10-year history of palmoplantar pustulosis (PPP) presented with an occipital headache. Five years prior, she had idiopathic cervical spondylitis with severe neck pain. The bacterial culture of the vertebral tissue was negative, and she was treated with nonsteroidal anti-inflammatory drugs (NSAIDs); however, there was a sequela of dull pain. At 5 months before admission, she developed an occipital headache with tenderness. Although her personal doctor diagnosed her with neuralgia and prescribed NSAIDs, her symptoms persisted.On admission, she had red pustules on the palms and soles (Figure 1A). A slight bulge with tenderness was observed in the right occipital region. The culture tests of blood and cerebrospinal fluid were negative. Brain magnetic resonance imaging (MRI) with gadolinium (Gd) administration showed enhanced swelling in the subcutaneous and muscular tissues of the cephalic region that corresponded to the bulging locus (Figure 1B-a, b). Technetium-99m ( 99m Tc)-hydroxymethylene diphosphonate scintigraphy (Figure 1C) demonstrated increased uptake in the parieto-occipital bone, sternoclavicular joints with "bullhead sign," and the fifth cervical

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Section: Discussionmentioning

confidence: 58%

Section: Discussionmentioning

confidence: 97%

Cranial osteomyelitis associated with SAPHO syndrome: An underdiagnosed cause of chronic headache

Sugie¹,

Yasaki²

2022

Neurology & Clinical Neurosc

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“…Three cases 8,11,12 reported in the literature underwent pathological examinations, which indicated chronic inflammation of the bone marrow accompanied by lymphocyte infiltration, but no malignant cells were found. Osteolysis in SAPHO syndrome appears radiographically similar to infection and malignancy.…”

Section: Discussionmentioning

confidence: 99%

Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome with cranial bone involvement: Case report and literature review

Hou

Liu

et al. 2023

Int J of Rheum Dis

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoimmune inflammatory disease characterized by osteoarticular and dermatological manifestations. The most common osteoarticular manifestations involve the anterior chest wall, axial skeleton, and long bones. Cranial bone involvement is less reported in SAPHO syndrome. We herein present three cases of SAPHO syndrome with cranial bone involvement, and review the previous literature on similar manifestations. It was revealed that SAPHO syndrome could lead to cranial bone involvement, which could involve the dura mater, leading to hypertrophic pachymeningitis, but the outcome is usually good. Janus kinase inhibitors may be a potential treatment option.

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“…Osteitis and osteophytes are the core clinical manifestations of SAPHO syndrome, and when typical bone lesions are located at characteristic target sites, (e.g., sternoclavicular joints (93.0%), costovertebral joints (87.0%), sternoclavicular joints (84.5%), spine (33%), and sacroiliac joints (16.6%), among others [ 3 ]), the diagnosis of SAPHO syndrome is not difficult. However, if atypical sites are involved in patients with dermatologic disease, (e.g., mandible (2–10%) [ 4 ], skull (4.5%) [ 5 ] etc. ), making the diagnosis is often challenging.…”

mentioning

confidence: 99%

Is palmoplantar pustulosis combined with hip involvement a SAPHO syndrome?

Sun,

Meng,

Chen

et al. 2024

pdia

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A case of SAPHO syndrome with the lesions limited to the skull

Cited by 3 publications

References 6 publications

Cranial osteomyelitis associated with SAPHO syndrome: An underdiagnosed cause of chronic headache

Cranial osteomyelitis associated with SAPHO syndrome: An underdiagnosed cause of chronic headache

Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome with cranial bone involvement: Case report and literature review

Is palmoplantar pustulosis combined with hip involvement a SAPHO syndrome?

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