The Synovitis, Acne, Pustulosis, Hyperostosis, And Osteitis (SAPHO) syndrome: A two-center study of 23 patients

Maatallah, Kaouther; Zouaoui, K.; Ferjani, H.; Dhahri, R.; Métoui, L.; Nessib, Dorra Ben; Slouma, M.; Triki, Wafa; Kaffel, Dhia; Hamdi, Wafa; Gharsallah, I.

doi:10.1016/j.ejr.2021.08.004

Cited by 1 publication

(4 citation statements)

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“…Three studies suggested beneficial effects of methotrexate on osteoarticular and cutaneous involvement. 19 26 54 Other studies suggested that using NSAIDs or corticosteroids alongside methotrexate may be more effective than methotrexate alone. 6 23 29 Overall, complete inefficacy of methotrexate was relatively uncommon (11/54 patients from 6 studies, 20.4%).…”

Section: Resultsmentioning

confidence: 99%

“…The cDMARDs methotrexate and sulfasalazine can correct proinflammatory and anti-inflammatory cytokine imbalances and have, therefore, successfully been used for the treatment of chronic arthritis and inflammatory skin diseases. 56 Several reports on the use of methotrexate for bone and skin involvement in SAPHO suggest efficacy, 19 26 54 while sulfasalazine delivered less promising results. 12 26 …”

Section: Discussionmentioning

confidence: 99%

“…12 26 Some patients refractory to NSAIDs experienced resolution of osteoarticular pain. 19 One patient had over 17 years sustained benefit as demonstrated by Physician Global Assessment scores. 54 Overall, cDMARDs were tolerated without severe side effects.…”

Section: Rmd Openmentioning

confidence: 98%

“…6 29 30 54 Three studies suggested beneficial effects of methotrexate on osteoarticular and cutaneous involvement. 19 26 54 Other studies suggested that using NSAIDs or corticosteroids alongside methotrexate may be more effective than methotrexate alone. 6 23 29 Overall, complete inefficacy of methotrexate was relatively uncommon (11/54 patients Single infusion (30 mg) Van Doornum et al 21 Single infusion per course (60 mg) Amital et al § 13 Infusion of 60 mg/day for three consecutive days Colina et al 15 Guignard et al 16 ¶ Solau-Gervais et al 15 16 20 Single infusion per course (180 mg)…”

Section: Antimicrobial Agentsmentioning

confidence: 99%

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Treatment and monitoring of SAPHO syndrome: a systematic review

Li,

Roberts,

Hedrich

2023

RMD Open

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Background and objectivesSynovitis acne pustulosis hyperostosis osteitis (SAPHO) is a rare heterogeneous disease of unknown aetiopathology. Externally validated and internationally agreed diagnostic criteria or outcomes and, as a result, prospective randomised controlled trials in SAPHO are absent. Consequently, there is no agreed treatment standard. This study aimed to systematically collate and discuss treatment options in SAPHO.MethodsFollowing ‘Preferred Reporting Items for Systematic Reviews and Meta-Analyses’ guidance, a systematic literature search was conducted using PubMed, Scopus and Web of Science databases. Prospective clinical studies and retrospective case collections discussing management and outcomes in SAPHO involving five or more participants were included. Articles not published in English, studies not reporting defined outcomes, and studies solely relying on patient-reported outcomes were excluded.ResultsA total of 28 studies (20 observational, 8 open-label clinical studies) reporting 796 patients of predominantly European ethnicity were included. Reported therapies varied greatly, with many centres using multiple treatments in parallel. Most patients (37.1%) received non-steroidal anti-inflammatory drugs alone or in combination. Bisphosphonates (22.1%), conventional (21.7%) and biological (11.3%) disease-modifying antirheumatic drugs were the next most frequently reported treatments. Reported outcomes varied and delivered mixed results, which complicates comparisons. Bisphosphonates demonstrated the most consistent improvement of osteoarticular symptoms and were associated with transient influenza-like symptoms. Paradoxical skin reactions were reported in patients treated with TNF inhibitors, but no serious adverse events were recorded. Most treatments had limited or mixed effects on cutaneous involvement. A recent study investigating the Janus kinase inhibitor tofacitinib delivered promising results in relation to skin and nail involvement.ConclusionsNo single currently available treatment option sufficiently addresses all SAPHO-associated symptoms. Variable, sometimes descriptive outcomes and the use of treatment combinations complicate conclusions and treatment recommendations. Randomised clinical trials are necessary to generate reliable evidence.

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Section: Resultsmentioning

confidence: 99%