A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis

Shiraishi, Wataru; Hayashi, Shigenari; Iwanaga, Yasutaka; Murai, Hiroyuki; Yamamoto, Akifumi; Kira, Jun Ichi

doi:10.1016/j.jns.2014.12.020

Cited by 17 publications

(13 citation statements)

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“…Tonsillitis has been reported to be related to the onset and exacerbation of PPP [ 34 , 35 ]. In a case report by Shiraishi et al, a patient with SAPHO syndrome and hypertrophic pachymeningitis showed complete remission of both diseases after bilateral tonsillectomy [ 36 ]. Our findings provided new data for the hypothesis that SAPHO syndrome might be related to focal inflammation such as tonsillitis and sinusitis.…”

Section: Discussionmentioning

confidence: 99%

F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation

Sun

Cao

et al. 2018

J Orthop Surg Res

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BackgroundsWhole-body bone scintigraphy (WBBS) and MRI are widely used in assessment of patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. However, the value of F-18 fluorodeoxyglucose-positron emission tomography/computed tomography (18F-FDG PET/CT) in SAPHO syndrome was unclear. The aim of this study was to characterize the manifestation of SAPHO syndrome on 18F-FDG PET/CT and explore its relationship with clinical symptoms and WBBS.MethodsTwenty-six patients who suffered from SAPHO syndrome and had undergone whole-body 18F-FDG PET/CT were recruited in Peking Union Medical College Hospital from 2004 to 2016. Clinical manifestations and laboratory findings were recorded for all patients. Imaging data on 18F-FDG PET/CT and WBBS were collected and analyzed retrospectively.ResultsAll the 26 patients (20 females and 6 males) exhibited skeletal abnormalities on 18F-FDG PET/CT. Multiple skeletal lesions affecting the anterior chest wall or spine with low to moderate 18F-FDG uptake and coexistence of osteolysis and osteosclerosis presented as the typical features of SAPHO syndrome. Sixteen (61.5%) patients had abnormal 18F-FDG uptake outside the osteoarticular system. PET scan had moderate to substantial agreement with CT and WBBS in revealing lesions in the anterior chest wall and axial skeleton. Nonetheless, the correlation between increased 18F-FDG uptake and clinical symptoms was weak.ConclusionsSAPHO syndrome exhibits characteristic features on 18F-FDG PET/CT. It showed comparable capacity in revealing skeletal lesions with bone scintigraphy.Electronic supplementary materialThe online version of this article (10.1186/s13018-018-0795-0) contains supplementary material, which is available to authorized users.

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Section: Discussionmentioning

confidence: 99%

F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation

Sun

Cao

et al. 2018

J Orthop Surg Res

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“…40,42 Systemic manifestations such as fever and elevation of inflammatory markers are uncommon but occasionally reported. 7 Other extra-articular manifestations include inflammatory bowel disease, 43 pulmonary involvement, 44 venous thrombosis (most commonly affecting the subclavian vein), 45 dura mater hypertrophy, 46 and uveitis. 47…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: review and update

Liu

Tang

Cao

et al. 2020

Therapeutic Advances in Musculoskeletal

105

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a spectrum of heterogeneous diseases characterized by osteoarticular and dermatological manifestations. Osteitis and hyperostosis are core clinical manifestations in SAPHO syndrome, typically affecting multiple areas and possibly progressing to irreversible osteoarticular damage. Most patients with SAPHO have cutaneous involvement, mainly manifested as palmoplantar pustulosis and severe acne. Systemic manifestations are uncommon but occasionally reported. Epidemiological studies suggest the annual prevalence of SAPHO syndrome varies from 0.00144 in 100,000 in Japanese individuals to fewer than 1 in 10,000 in White individuals. The precise etiopathogenesis of SAPHO remains unclear, but it is generally considered an autoinflammatory syndrome that may be related to various etiologies, such as immune dysfunction, infection and genetic predisposition. Owing to the relapsing–remitting disease course, the goal of management is to improve clinical symptoms and prevent disease progression. Various treatments, including nonsteroidal anti-inflammatory drugs, conventional disease-modifying antirheumatic drugs, bisphosphonates, biologics, and antibiotics, are promising options for alleviating the disease.

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“…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%

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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A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis

Cited by 17 publications

References 9 publications

F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation

F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation

Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: review and update

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

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