Successful treatment of co‐existent <scp>SAPHO</scp> syndrome and hidradenitis suppurativa with adalimumab and methotrexate

Genovese, Giovanni; Caorsi, Roberta; Moltrasio, Chiara; Marzano, Angelo Valerio

doi:10.1111/jdv.15849

Cited by 19 publications

(24 citation statements)

References 6 publications

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“…The use of steroid-sparing agents in refractory cases, including MTX, sulfasalazine, or azathioprine, was only reported as beneficial for noncutaneous symptoms [88,95]. Anti-TNFα agents were ultimately used in refractory cases, including infliximab [94,96,97], etanercept [94], or adalimumab [80,94,[98][99][100]. The response was usually rapid for both cutaneous and osteoarticular symptoms.…”

Section: Resultsmentioning

confidence: 99%

Phenotypes and Pathophysiology of Syndromic Hidradenitis Suppurativa: Different Faces of the Same Disease? A Systematic Review

et al. 2020

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Background: There is growing evidence that (certain) hidradenitis suppurativa (HS) comorbidities comprise syndromes including HS as a key cutaneous manifestation. These apparently autoinflammatory syndromes and their diagnostic delay might have detrimental effects on affected patients. Methods: A systematic review was performed on the databases MEDLINE, EMBASE, and CENTRAL utilizing a standardized extraction form according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Results: Sixty-four eligible articles on syndromic HS were retrieved. The identified syndromes included already described ones (pyoderma gangrenosum-acne-suppurative hidradenitis, pyogenic arthritis-pyoderma gangrenosum-acne-suppurative hidradenitis, psoriatic arthritis-pyoderma gangrenosum-acne-suppurative hidradenitis, pyoderma gangrenosum-acne vulgaris-hidradenitis suppurativa-ankylosing spondylitis, synovitis-acne-pustulosis-hyperostosis-osteitis) and further novel symptom constellations. Cutaneous signs, including HS lesions, usually precede signs from other organs. The cutaneous signs of a considerable proportion of patients appear refractory to conventional treatment, and monotherapy with biologics does not suffice to sustain remission. Conclusion: The results are subsequently discussed with focus on the pathophysiology and treatment of the detected syndromes. The dermatologist’s role in the precise diagnosis and early treatment administration of HS is pivotal. The purpose of the treatment should be the effective prevention or delay of the autoinflammatory march and its irreversible consequences.

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Section: Resultsmentioning

confidence: 99%

Phenotypes and Pathophysiology of Syndromic Hidradenitis Suppurativa: Different Faces of the Same Disease? A Systematic Review

et al. 2020

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“…6 However, this study reviewed mostly adult patients and did not report pediatric data separately, 6 whereas our review included only pediatric studies. [13][14][15][16][17][18][19][20][21][22][23][24][25][26][27] In addition to anti-TNF-α , anti-IL-1 may be effective against HS as elevated TNF-α and IL-1β levels have been detected in HS lesions. 28 IL-1β and TNF-α levels in HS lesions were elevated 31-fold and 5-fold, compared to healthy skin.…”

Section: Discussionmentioning

confidence: 99%

Biologic Use in Pediatric Patients With Hidradenitis Suppurativa: A Systematic Review

et al. 2021

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Background: There is currently at least 1 biologic (adalimumab) approved in North America for treatment of Hidradenitis Suppurativa in the pediatric population. However, no reviews or clinical trials have specifically analyzed the effectiveness and safety data of biologic use in this population. The objective of this systematic review is to identify and summarize the outcomes of biologic therapy in pediatric patients with HS. Methods: MEDLINE and EMBASE databases were used to conduct the search on Sept 18, 2020. Results: The 15 included studies consisted of 26 patients, with the mean age of 15 ± 2.3 years. Females accounted for 53.8% ( n = 14/26) of cases. The mean duration of HS prior to biologic initiation was 3.5 ± 2.9 years, with the majority having Hurley Stage II. The 26 patients received 34 biologics in total: 85.3% treated with TNF alpha inhibitors (adalimumab n = 17, infliximab n = 10, etanercept n = 1, unspecified n = 1), 5.9% with IL-12/23 inhibitors (ustekinumab n = 2), 5.9% with IL-1 inhibitors (i.e., anakinra n = 2) and 2.9% received IL-23 inhibitors (i.e., guselkumab n = 1) biologics. Of the 26 patients, 23.1% ( n = 6/26) experienced complete resolution (CR), 73.1% ( n = 19/26) experienced partial resolution (PR), and 3.8% ( n = 1/26) had no resolution outcomes reported. The time to resolution of HS lesions after biologic initiation ranged from 10 days to 11.5 months (mean: 5.1 months). No adverse events were reported in the studies. Conclusion: Although anti-TNF alpha were the most common biologics used for HS in pediatric cases, large-scale trials specific to pediatric patients with HS are needed to confirm these findings.

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“…To our knowledge there are only few cases reporting a favourable effect of ADA in the treatment of HS associated to SAPHO syndrome [17][18][19]. ADA is considered the first-choice biologic agent in moderate/severe HS after failure of conventional treatments and in refractory SAPHO syndrome [8,17].…”

Section: Discussionmentioning

confidence: 99%

SAPHO syndrome: the supposed trigger by isotretinoin, the efficacy of adalimumab and the specter of depressive disorder: a case report

et al. 2020

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Background SAPHO (synovitis, acne, pustolosis, hyperostosis and osteitis) syndrome is a rare autoinflammatory chronic disorder, presenting with non-infectious osteitis, sterile joint inflammation and skin manifestations including palmoplantar pustolosis and severe acne. It could be often misdiagnosed for its heterogeneous clinical presentation. Treatment is challenging and, due to the rarity of this syndrome, no randomized controlled clinical trials have been conducted. Empirical treatments, including non-steroidal anti-inflammatory drugs (NSAIDs), corticosteroids, antibiotics and bisphosphonates and disease-modifying anti-rheumatic drugs (DMARDs) could be quite effective. Anti-tumor necrosis factor-alpha (anti-TNF-α) agents and interleukin-1 (IL-1) antagonists have shown promising results in refractory patients. Isotretinoin, commonly used for severe acne, has been rarely described as possible trigger of osteo-articular manifestations, in particular sacroiliitis. Case presentation The case of a boy, affected by acne fulminans and depression, who presented with sacroiliitis after a 10-week treatment with isotretinoin is presented. After SAPHO diagnosis, NSAIDs therapy was started but the onset of bilateral gluteal hidradenitis suppurativa required the switch to a TNF-α antagonist (Adalimumab) with the achievement of a good control of the disease. Despite specific therapy with sertraline, the patient continued to complains severe depression. Conclusions Our case reports a temporal association between the onset of osteo-articular symptoms and the introduction of isotretinoin, as previously described. However, this timeline is not sufficient to establish a causal role of this drug into the pathogenesis of sacroiliitis. At this regard, further studies are required. The occurrence of hidradenitis suppurativa during SAPHO course supported the introduction of TNF-α blockers with a favourable result, as reported in a few cases in literature. The association between SAPHO syndrome and depressive mood disorders is already reported. Our patient experienced severe depression whose trend seems to be independent from the course of the main disease. Currently, it is not clarified if depression could be considered reactive to the underling disease or if it forms an integral part of the autoinflammatory disorder.

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Successful treatment of co‐existent SAPHO syndrome and hidradenitis suppurativa with adalimumab and methotrexate

Cited by 19 publications

References 6 publications

Phenotypes and Pathophysiology of Syndromic Hidradenitis Suppurativa: Different Faces of the Same Disease? A Systematic Review

Phenotypes and Pathophysiology of Syndromic Hidradenitis Suppurativa: Different Faces of the Same Disease? A Systematic Review

Biologic Use in Pediatric Patients With Hidradenitis Suppurativa: A Systematic Review

SAPHO syndrome: the supposed trigger by isotretinoin, the efficacy of adalimumab and the specter of depressive disorder: a case report

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