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1.
Clin Nucl Med ; 47(5): e411-e413, 2022 May 01.
Artigo em Inglês | MEDLINE | ID: mdl-35195583

RESUMO

ABSTRACT: Pulmonary vasculitis is an uncommon type of vasculitis that may cause pulmonary artery stenosis with ensuing ischemic damage to lung tissue. 99mTc-macroaggregated albumin scintigraphy and iodine lung perfusion mapping are useful imaging modalities for assessment of lung perfusion but are not useful for assessing disease activity. On the other hand, 18F-FDG PET/CT is widely used for assessment of vasculitis activity but cannot provide perfusion information. We reported the clinical utility of dual-phase 18F-FDG PET/CT for pulmonary vasculitis in patients with SAPHO (synovitis-acne-pustulosis-hyperostosis-osteitis) syndrome.


Assuntos
Síndrome de Hiperostose Adquirida , Vasculite , Síndrome de Hiperostose Adquirida/diagnóstico por imagem , Fluordesoxiglucose F18 , Humanos , Pulmão , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada
2.
Clin Exp Dermatol ; 47(5): 978-980, 2022 May.
Artigo em Inglês | MEDLINE | ID: mdl-35089610

RESUMO

We report the use of ixekizumab in treating synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome, the first such report to our knowledge. The patient presented with palmoplantar pustulosis and sternoclavicular joint pain, which was markedly improved with ixekizumab treatment.


Assuntos
Acne Vulgar , Síndrome de Hiperostose Adquirida , Hiperostose , Osteíte , Sinovite , Acne Vulgar/tratamento farmacológico , Síndrome de Hiperostose Adquirida/diagnóstico , Síndrome de Hiperostose Adquirida/tratamento farmacológico , Anticorpos Monoclonais Humanizados , Humanos , Osteíte/diagnóstico , Osteíte/tratamento farmacológico , Sinovite/tratamento farmacológico
4.
Clin Nucl Med ; 47(3): 246-248, 2022 Mar 01.
Artigo em Inglês | MEDLINE | ID: mdl-34560692

RESUMO

ABSTRACT: A 66-year-old woman presented with anterior chest wall and knee joints pain and multiple skin lesions. 99mTc-MDP bone scintigraphy showed increased bone density in the sternal angle with abnormal bone metabolism. The patient was enrolled in a 68Ga-DOTA-FAPI-04 PET/CT tumor clinical trial, and it showed increased tracer uptake in the sternum and right knee joint. Subsequently, ultrasound of the right knee joint confirmed the existence of synovitis, and no malignant components were found in sternal biopsy. The patient's final diagnosis was synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. The case confirmed that 68Ga-DOTA-FAPI-04 imaging is helpful for evaluating the involved sites of SAPHO syndrome.


Assuntos
Síndrome de Hiperostose Adquirida , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada , Síndrome de Hiperostose Adquirida/diagnóstico por imagem , Idoso , Feminino , Compostos Heterocíclicos com 1 Anel , Humanos , Quinolinas , Tomografia Computadorizada por Raios X
6.
J Clin Rheumatol ; 28(2): e545-e551, 2022 03 01.
Artigo em Inglês | MEDLINE | ID: mdl-33843770

RESUMO

ABSTRACT: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare chronic disease with marked clinical and radiological heterogeneity. It is characterized by a combination of dermatological and osteoarticular manifestations. The treatment of SAPHO syndrome is not yet codified. It includes several therapeutic options such as anti-inflammatory drugs, bisphosphonates, antibiotics, conventional disease-modifying antirheumatic drugs, and biological treatment.This article aims to provide an updated review of the different pharmacological options for SAPHO syndrome. We also propose a therapeutic algorithm for the management of this disease.


Assuntos
Acne Vulgar , Síndrome de Hiperostose Adquirida , Hiperostose , Osteíte , Sinovite , Acne Vulgar/diagnóstico , Acne Vulgar/tratamento farmacológico , Síndrome de Hiperostose Adquirida/diagnóstico , Síndrome de Hiperostose Adquirida/tratamento farmacológico , Algoritmos , Humanos , Hiperostose/diagnóstico , Hiperostose/tratamento farmacológico , Hiperostose/etiologia , Osteíte/diagnóstico , Osteíte/tratamento farmacológico , Osteíte/etiologia
8.
J Proteomics ; 251: 104399, 2022 01 16.
Artigo em Inglês | MEDLINE | ID: mdl-34718201

RESUMO

SAPHO syndrome is an inflammatory disease invading the skin and bones, whose diagnosis has been difficult due to its low incidence and diversified manifestation. We investigated the serum proteomic profile of SAPHO patients to identify key proteins associated with SAPHO syndrome, trying to find clinical biomarkers or functional molecules for this rare disease. Blood samples from 8 SAPHO patients and 8 healthy controls were detected and analyzed using data independent acquisition (DIA) method to identify differentially expressed proteins (DEPs) specific to SAPHO. A total of 57 differentially expressed proteins were identified (p < 0.05, fold change >1.2), in which 27 proteins were upregulated and 30 downregulated. DEPs may participate in GO terms such as "lipid particle" and "Notch signaling pathway", as well as KEGG pathways including "complement and coagulation cascades" and "mTOR signaling pathway". The overexpression of inhibitors of the complement system (CFH and C4BP), were verified in a larger cohort (16 SAPHO patients, 8 AS patients and 24 healthy controls) with ELISA, and the combined diagnostic ability of CFH and C4BP was predicted by ROC curve with an AUC of 0.91, which may be molecular candidates for further study on diagnosis and pathology of this rare disease. SIGNIFICANCE: Our research provided the first insight into plasma proteomic profile for SAPHO patients,offering potential biomarkers for disease diagnosis. We found that inhibitors of complement system such as CFH and C4BP were up-regulated in SAPHO syndrome, which may play important roles in the pathogenesis of SAPHO syndrome.


Assuntos
Síndrome de Hiperostose Adquirida , Proteômica , Biomarcadores , Osso e Ossos , Proteínas do Sistema Complemento , Humanos , Proteômica/métodos
9.
J Int Med Res ; 49(12): 3000605211065314, 2021 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-34932408

RESUMO

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an umbrella term covering a constellation of bone lesions and skin manifestations, but has rarely been clarified in the clinic. We report a 28-year-old woman who had initial onset of SAPHO syndrome with involvement of the femur, and she experienced a tortuous diagnostic course. We also performed a literature review of SAPHO syndrome cases involving the femur and summarize several empirical conclusions by integrating previous findings with our case. Furthermore, we propose our perspective that ailment of the skin caused by infection of pathogens might be the first hit for triggering or perpetuating the activation of the immune system. As a result, musculoskeletal manifestations are probably the second hit by crosstalk of an autoimmune reaction. The skin manifestations preceding bone lesions can be well explained. Current interventions for SAPHO syndrome remain controversial, but drugs aiming at symptom relief could serve as the first preference for treatment. An accurate diagnosis and appropriate treatment can cure patients in a timely manner. Although the pathogenesis of SAPHO syndrome remains to be determined, physicians and surgeons still need to heighten awareness of this entity to avoid invasive procedures, such as frequent biopsies or nonessential ostectomy.


Assuntos
Acne Vulgar , Síndrome de Hiperostose Adquirida , Neoplasias Femorais , Hiperostose , Osteíte , Sinovite , Síndrome de Hiperostose Adquirida/diagnóstico , Síndrome de Hiperostose Adquirida/tratamento farmacológico , Adulto , Feminino , Humanos , Osteíte/diagnóstico por imagem , Osteíte/tratamento farmacológico
11.
Artigo em Chinês | MEDLINE | ID: mdl-34628822

RESUMO

SAPHO syndrome is a rare disease which affects the bones, joints, and skin. It is often misdiagnosed and treated mistakenly because of various clinical manifestations and general lack of awareness about the disease. The pathogenesis is inadequately understood, as a result, current therapy is empirical and aimed to control inflammatory process and alleviate pain. This paper summarizes the clinical manifestations and diagnosis and treatment scheme of SAPHO syndrome, and presents a case of patient with SAPHO syndrome who was treated in our department for bilateral tonsillectomy due to repeated pharyngalgia and fever for 10 years. Interestingly, the patient is getting better after the operation. The case is reported so as to provide reference for the diagnosis and treatment of SAPHO syndrome.


Assuntos
Síndrome de Hiperostose Adquirida , Tonsilite , Síndrome de Hiperostose Adquirida/diagnóstico , Doença Crônica , Humanos , Dor , Pele , Tonsilite/diagnóstico
12.
J Rheumatol Suppl ; 97: 34-38, 2021 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-34074664

RESUMO

Pustular psoriasis (PsO) is an uncommon variant of PsO that may present in a generalized or localized fashion with or without musculoskeletal or systemic inflammatory involvement.Generalized pustular PsO (GPP) presents as a widespread acute or subacute pustular eruption that may be familial and is often associated with severe flares and systemic inflammation. The palmoplantar pustulosis variant is localized to palms and soles, whereas acrodermatitis continua of Hallopeau is localized to the nail apparatus. Patients with pustular PsO may have overlapping plaque PsO and may develop psoriatic arthritis (PsA). Pustulosis is also a feature of both synovitis, acne, pustulosis, hyperostosis, osteomyelitis (SAPHO) syndrome and chronic non-bacterial osteomyelitis. At the 2020 Group for Research and Assessment of Psoriasis and Psoriatic Arthritis (GRAPPA) annual meeting, members were given an overview of the cutaneous features of pustular PsO, SAPHO, and recent insights into the genetics of GPP, leading to new targeted drug therapies and the development of validated endpoints.


Assuntos
Síndrome de Hiperostose Adquirida , Artrite Psoriásica , Doenças Musculoesqueléticas , Psoríase , Artrite Psoriásica/diagnóstico , Artrite Psoriásica/tratamento farmacológico , Humanos , Psoríase/diagnóstico , Psoríase/tratamento farmacológico , Pele
13.
Semin Arthritis Rheum ; 51(4): 957-961, 2021 08.
Artigo em Inglês | MEDLINE | ID: mdl-34176644

RESUMO

OBJECTIVE: A working group was established to develop a core domain set (CDS) for Chronic Nonbacterial Osteomyelitis (CNO) and Synovitis, Acne, Pustulosis, Hyperostosis, Osteitis (SAPHO) following the OMERACT filter 2.1. METHODS: A scoping review to identify disease-related manifestations was performed, followed by a special interest group (SIG) session at OMERACT2020 to begin the CNO/SAPHO CDS framework. RESULTS: Candidate items were identified from the scoping review and most fell under Life Impact and Pathophysiology Manifestation core areas. A SIG agreed on the need to develop a CDS for CNO and SAPHO (100%) and for children and adults (91%). CONCLUSION: Based on candidate items identified, qualitative research and Delphi surveys will be performed as next steps.


Assuntos
Acne Vulgar , Síndrome de Hiperostose Adquirida , Hiperostose , Osteíte , Osteomielite , Sinovite , Síndrome de Hiperostose Adquirida/diagnóstico , Adulto , Criança , Humanos , Osteomielite/diagnóstico , Opinião Pública
14.
Clin Med Res ; 19(3): 141-147, 2021 09.
Artigo em Inglês | MEDLINE | ID: mdl-33985979

RESUMO

Paraneoplastic arthritides are a group of immune-mediated inflammatory arthropathies associated with occult or manifest malignancy. Musculoskeletal spread of an underlying malignancy may also mimic many rheumatologic conditions. Distinguishing primary rheumatologic condition from paraneoplastic arthritides versus direct musculoskeletal spread of malignancy can be challenging especially in individuals with prior history of cancer and new musculoskeletal complaints. SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis) syndrome is an uncommon, although under recognized autoimmune disorder. Two musculoskeletal manifestations, namely inflammatory osteitis and hyperostosis of anterior chest wall with or without dermatologic manifestations, constitute a unifying feature of SAPHO syndrome. However, diagnosis of SAPHO syndrome is one of exclusion, and a wide variety of disorders including infections, malignancy (chondrosarcoma/osteosarcoma/metastasis), metabolic bone disorders (Paget's disease), osteoarthritis, seronegative spondyloarthropathy (spA) and osteonecrosis form part of a broad differential diagnosis. We present the case of a man, aged 72 years, with signs and symptoms of SAPHO syndrome and skin findings. Detailed history, radiological imaging, dermatology appearance, and role of immunohistochemical markers, especially staining for NKX3.1 protein with a novel antibody, led to a diagnosis of metastatic prostate adenocarcinoma. To our knowledge, this is the first case of metastatic adenocarcinoma of the prostate manifesting as SAPHO syndrome and cutaneous metastasis.


Assuntos
Síndrome de Hiperostose Adquirida , Carcinoma , Hiperostose , Osteíte , Síndrome de Hiperostose Adquirida/diagnóstico , Humanos , Masculino , Próstata
15.
BMC Microbiol ; 21(1): 161, 2021 05 30.
Artigo em Inglês | MEDLINE | ID: mdl-34053449

RESUMO

BACKGROUND: SAPHO syndrome is a group of symptoms consisting of synovitis, acne, pustulosis, hyperostosis and osteosis. There is no specific laboratory index assist in the diagnosis of SAPHO because of its highly heterogeneous clinical manifestations. Pathogenic microorganisms had been identified in biopsies of some SAPHO cases and particular gene mutations were also linked to the occurrence of SAPHO. It is largely unknown whether intestinal microbiome plays a role in pathogenesis of SAPHO. To explore the intestinal microbiome structure of SAPHO syndrome, fecal samples from 17 SAPHO patients and 14 healthy controls (HC) were collected for 16S rDNA sequencing. RESULTS: Our results showed that there was no significant difference in alpha indexes and beta diversity between SAPHO and HC samples, while there were 14 operational taxonomic units (OTUs) in the Wilcoxon rank-sum test and 42 OTUs in the MetagenomeSeq analysis showed significant difference in distribution between the SAPHO and HC groups, 3 of which in Firmicutes were also observed in the random forest analysis and used to construct a receiver operating characteristic curve to evaluate the diagnostic value, the area under the curve was 0.86. CONCLUSION: Fecal-associated microbiome in the SAPHO samples was characterized by the alteration in abundance of some nondominant species, and the 3 selected OTUs in Firmicutes could serve as candidate biomarkers for SAPHO syndrome diagnosis.


Assuntos
Síndrome de Hiperostose Adquirida/microbiologia , Bactérias/isolamento & purificação , Fezes/microbiologia , Microbioma Gastrointestinal , Adulto , Bactérias/classificação , Bactérias/genética , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Adulto Jovem
17.
Medicine (Baltimore) ; 100(12): e23940, 2021 Mar 26.
Artigo em Inglês | MEDLINE | ID: mdl-33761629

RESUMO

ABSTRACT: To identify the prevalence and clinical characteristics of Sjögren syndrome (SS) in a Chinese single-center cohort of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome.Patients diagnosed with SS were screened out from a cohort of 164 cases of SAHPO syndrome. Information regarding the patients' gender, age at onset, clinical features, laboratory tests, bone scintigraphy, and treatment was reviewed.Five patients were screened out. The prevalence of SS in SAPHO patients was 3.05% The mean onset age of SS was 48.0 ±â€Š12.0 years old and no apparent time order in the occurrence of SAPHO and SS was observed. Compared with the general SAPHO cohort, the 5 SS patients exhibited no significant difference in the SAPHO related clinical features or inflammatory markers, except for a higher prevalence of peripheral joints and bones involvement in bone scintigraphy. Objective evidence of dryness and positive salivary gland biopsy were found in all the patients. However, the positive rates of SSA and SSB antibody were only 20%. Anti-inflammatory treatment for SS was recorded in 3 patients (ESSDAI score: 3 in 2 patients; 12 in 1 patient) with extra-glandular manifestations, severe complications or poor response to the basic treatment.The prevalence of SS is higher in the SAPHO cohort than in the general Chinese population. Objective tests or biopsy might be more indicative than the antibody detection for SS diagnosis. Anti-inflammatory treatment should be prescribed in consideration of both the severity of SS and the demand for disease activity control of SAPHO.


Assuntos
Síndrome de Hiperostose Adquirida/epidemiologia , Síndrome de Sjogren/epidemiologia , Síndrome de Hiperostose Adquirida/imunologia , Adulto , Idade de Início , Anti-Inflamatórios/uso terapêutico , Biópsia , Comorbidade , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Prevalência , Estudos Retrospectivos , Glândulas Salivares/patologia , Índice de Gravidade de Doença , Síndrome de Sjogren/diagnóstico , Síndrome de Sjogren/tratamento farmacológico , Síndrome de Sjogren/imunologia , Tomografia Computadorizada por Raios X
19.
Z Rheumatol ; 80(5): 456-466, 2021 Jun.
Artigo em Alemão | MEDLINE | ID: mdl-33725179

RESUMO

The SAPHO syndrome is not a single entity but an inhomogeneous, nosologically heterogeneous complex of symptoms with unknown etiology and heterogeneous pathogenesis. Clinically subacute, recurrent or chronic disease processes and a common skin-bone association (skibo disease) can be found. Under the acronym SAPHO, chronically recurrent multifocal osteomyelitis (CRMO) is the most common disease that can occur in youth as well as adolescence. Spondylarthritis hyperostotica pustulo-psoriatica with the triad palmoplantar pustulosis, sternoclavicular hyperostosis and ossifying spinal manifestations is the most common SAPHO form found in adults. Abortive disease forms are the inflammatory anterior chest wall syndrome, extended sternoclavicular hyperostosis syndrome of the clavicle bone, acne CRMO and acne spondylarthritis. The SAPHO disease usually heals with a relatively favorable prognosis but there are also unfavorable courses with functional limitations. The diagnosis should be made based on clinical examination, imaging (x-ray, scintigraphy, magnetic resonance imaging) and/or histological bone biopsy analysis. Treatment should be interdisciplinary. Antibiotic treatment is obsolete. This article provides an overview of the SAPHO syndrome and a clinical-rheumatological imaging differentiation as well as classification of 35 cases at first presentation.


Assuntos
Síndrome de Hiperostose Adquirida , Osteomielite , Síndrome de Hiperostose Adquirida/diagnóstico por imagem , Síndrome de Hiperostose Adquirida/terapia , Adolescente , Adulto , Osso e Ossos , Humanos , Osteomielite/diagnóstico por imagem , Radiografia , Cintilografia
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