Pyoderma gangrenosum induced by adalimumab in a seropositive rheumatoid arthritis patient: a paradoxical effect of adalimumab?

Tan, Yvonne; Kavaklieva, Svetlana; Wood, Fiona

doi:10.1093/rheumatology/keab194

Cited by 8 publications

(8 citation statements)

References 7 publications

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“…HS can also be a main or secondary element of certain syndromes highlighted through their unique phenotypes, 5 known as autoinflammatory diseases (AID). AID manifests with recurrent sterile inflammation, while high autoantibody serum levels or antigen-specific lymphocytes are lacking.…”

Section: Discussionmentioning

confidence: 99%

“…3 Moreover, pustular lesions and PG could be explained as "paradoxical" reactions to the previous adalimumab treatment or its switch to a new biologic treatment. 4,5 Despite these promising results, long term follow-up for these patients and controlled clinical studies can determine if treatment with IL-17A inhibitors can result in long-term remission. 6 Moreover, this case underlines the role of the dermatologist in diagnosing such symptoms and leading a multidisciplinary approach for these patients.…”

Section: Discussionmentioning

confidence: 99%

“…Such syndromes include the triad of PG, acne and HS (PASH) alone or in combination with PA (PAPASH) or psoriatic arthritis (PsAPASH). In addition, HS can also be a feature of other syndromes such as the SAPHO syndrome, 5 which appear recalcitrant to treatment, even after the use of various biologics, such as anti-TNF and anti-IL-1. Here we describe a patient with a novel phenotypic variant of the syndromes described, who responded to treatment with the IL-17A inhibitor secukinumab.…”

Section: Introductionmentioning

confidence: 99%

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Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

et al. 2021

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Syndromic hidradenitis suppurativa (HS) is a form of symptom constellations, which differs from the familial and genetic form and comprises predominantly osteoarticular manifestations. Many forms include pyoderma gangrenosum and acne (PASH), pyogenic arthritis (PAPASH), spondyloarthritis (PASS) and psoriatic arthritis (PsAPASH) and are categorized in the autoinflammatory syndromes. Anti-TNF-α and anti-IL-1a blockade are between the therapeutic approaches that improve skin symptoms and prevent permanent osteoarticular damage. This case report refers to the successful treatment of a mixed phenotype of the aforementioned symptoms using the IL-17A inhibitor secukinumab after initial treatment with adalimumab. The therapy improved both cutaneous and reported osteoarticular symptoms. Different approaches for these recalcitrant HS syndromes are essential in order to achieve long-term remission for those patients.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

et al. 2021

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“…In PG-related diseases, the precipitating factors should be treated with caution, and patients alerted to its complications. There are also reports in the literature that drugs used to treat PG may paradoxically lead to its occurrence ( 24 ), which brings confusion and illustrates its complexity.…”

Section: Clinical Featuresmentioning

confidence: 99%

Practical aspects of the diagnosis and management of pyoderma gangrenosum

Chen

2023

Front. Med.

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Pyoderma gangrenosum (PG) is a rare autoinflammatory ulcerative neutrophilic skin disease. Its clinical presentation is a rapidly progressing painful skin ulcer with ill-defined borders and surrounding erythema. The pathogenesis of PG is complex and not fully understood. Clinically, patients with PG often have various systemic diseases, the most common being inflammatory bowel disease (IBD) and arthritis. Due to the lack of specific biological markers, diagnosing PG remains difficult, which easily resulting in misdiagnosis. Some validated diagnostic criteria have been applied in clinical practice that facilitate its diagnosis. The treatment of PG currently consists mainly of immunosuppressive and immunomodulatory agents, especially biological agents, which have bright prospects for PG therapy. After the systemic inflammatory response is controlled, the problem of wounds becomes the main contradiction in PG treatment. Surgery is not controversial for PG, increasing evidence shows that with adequate systemic treatment, the benefits of reconstructive surgery for patients are increasing.

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“…At first the possibility of a paradoxical reaction to anti-TNFα treatment was considered, since adalimumab is one of the most promising therapeutic choices for PG, rarely it also may induce inflammatory immune-mediated skin manifestations. 31,32 PG has been recently described as part of some genetic autoinflammatory conditions such as PAPA (pyogenic arthritis, PG and acne), PASH (PG, acne and suppurative hidradenitis), and PAPASH (pyogenic arthritis, acne, PG and suppurative hidradenitis), these are all characterized by cutaneous involvement with a neutrophilic infiltrate, an inflammasome impairment, and an overproduction of IL-1. 33 In our patient canakinumab induced a rapid and satisfactory response, with bone pain and PG resolution, inflammatory markers normalization, and an almost complete radiological bone lesions recovery.…”

Section: F I G U R Ementioning

confidence: 99%

Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with pyoderma gangrenosum

et al. 2022

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Background Chronic recurrent multifocal osteomyelitis (CRMO) is a bone inflammatory disorder characterized by osteolytic, usually multiple, symmetric lesions. Diagnosis is one of exclusion, and no standardized therapies are available. Presumed deregulation of the interleukin (IL)‐1β axis, as observed in 2 monogenic autoinflammatory conditions such as Majeed syndrome (LPIN2 mutations) and deficiency of IL‐1 receptor antagonist (IL1RN mutations) with CRMO‐like bone involvement, suggests the blockade of IL‐1 as potentially useful also in this condition, even if scarce data are available. Case presentation We report the case of a 13‐year‐old girl affected by a multidrug‐resistant and pyoderma gangrenosum‐complicated CRMO treated with canakinumab, a human monoclonal antibody targeting IL‐1β. Conclusion In this young patient pyoderma gangrenosum and CRMO showed a rapid and satisfactory response to canakinumab, although over time a decreased efficacy in controlling bone disease was observed.

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Pyoderma gangrenosum induced by adalimumab in a seropositive rheumatoid arthritis patient: a paradoxical effect of adalimumab?

Cited by 8 publications

References 7 publications

Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

Case Report: PsAPSASH syndrome: an alternative phenotype of syndromic hidradenitis suppurativa treated with the IL-17A inhibitor secukinumab

Practical aspects of the diagnosis and management of pyoderma gangrenosum

Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with pyoderma gangrenosum

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