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

Acierno, Sabrina; Angrisani, Francesca; Marino, Achille; Caporali, Roberto; Cimaz, Rolando; Giani, Teresa

doi:10.1111/1756-185x.14425

Cited by 12 publications

(7 citation statements)

References 36 publications

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“…We describe a case of pyoderma gangrenosum in a 16-years-old girl that arose after the administration of the TNF-α inhibitor adalimumab for CRMO. Some of this case details have been previously reported by our rheumatology colleagues ( 8 ). Here, we illustrate the case from a dermatologic perspective, including clinical and histopathological description of PG lesions, and speculate on the possible nature of PG, whether consider it as a drug reaction or a cutaneous manifestation in the setting of CRMO, possibly exacerbated by the anti-TNF therapy.…”

Section: Introductionmentioning

confidence: 68%

“…However, due to persistent skeletal involvement confirmed by magnetic resonance imaging (MRI), anti TNF-α agent adalimumab was introduced at the dosage of 40 mg every 2 weeks subcutaneously. Cutaneous lesions appeared after 2 months of the newly introduced adalimumab therapy and were reported at first as reddish papules, that suddenly enlarged and ulcerated ( 8 ).…”

Section: Case Presentationmentioning

confidence: 99%

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Pyoderma gangrenosum following anti-TNF therapy in chronic recurrent multifocal osteomyelitis: drug reaction or cutaneous manifestation of the disease? A critical review on the topic with an emblematic case report

et al. 2023

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Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory disease, clinically characterized by chronic and recurrent episodes of osteoarticular inflammation, that generally presents in children and adolescents. From a dermatological point-of-view, CMRO can be associated with skin rashes mainly including psoriasis, palmoplantar pustulosis and acne. Pyoderma gangrenosum (PG) is a rare immune-mediated inflammatory skin disease classified within the spectrum of neutrophilic dermatoses that, in some cases, has been reported as cutaneous manifestation in CMRO patients. This paper presents a 16-year female patient diagnosed with CMRO, who presented PG lesions located on the lower leg, that arose after the administration of the tumour necrosis factor (TNF)-α inhibitor adalimumab. Cases of PG have been reported in patients being treated with certain medications, including TNF-α antagonists, leading to classified them in a setting aptly termed “drug-induced PG.” In this paper, we discuss the co-occurrence of PG and CRMO, in the light of recent evidence on the pathogenesis of both diseases and giving ample space to a literature review on drug induced PG. In our case, it is plausible that PG could be considered a cutaneous manifestation of CRMO, although the mechanisms underlying this intriguingly relationship remain to be fully unraveled.

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Section: Introductionmentioning

confidence: 68%

Section: Case Presentationmentioning

confidence: 99%

Pyoderma gangrenosum following anti-TNF therapy in chronic recurrent multifocal osteomyelitis: drug reaction or cutaneous manifestation of the disease? A critical review on the topic with an emblematic case report

et al. 2023

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“…Four of them were completely healed [145]. Cases of complete resolution of skin lesions after the use of canakinumab have also been reported in the literature [146,147]. Anakinra, a recombinant, non-glycosylated form of IL-1 receptor antagonist used in a dose of 100 mg once a day from 8 weeks to 10 months, was administered only to a few patients described in case reports [148].…”

Section: Il-1 Antagonistsmentioning

confidence: 99%

The Pathophysiology and Treatment of Pyoderma Gangrenosum—Current Options and New Perspectives

Łyko,

Ryguła,

Kowalski

et al. 2024

IJMS

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Pyoderma gangrenosum (PG) is an uncommon inflammatory dermatological disorder characterized by painful ulcers that quickly spread peripherally. The pathophysiology of PG is not fully understood; however, it is most commonly considered a disease in the spectrum of neutrophilic dermatoses. The treatment of PG remains challenging due to the lack of generally accepted therapeutic guidelines. Existing therapeutic methods focus on limiting inflammation through the use of immunosuppressive and immunomodulatory therapies. Recently, several reports have indicated the successful use of biologic drugs and small molecules administered for coexisting diseases, resulting in ulcer healing. In this review, we summarize the discoveries regarding the pathophysiology of PG and present treatment options to raise awareness and improve the management of this rare entity.

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“…Lastly, given the implication of the IL-1 pathway in the pathogenesis of CNO, modulation of this pathway has been proposed as a promising therapy. However, treatment of sporadic CNO with anti-IL-1 therapy has shown mixed results (80)(81)(82).…”

Section: Management Of Autoinflammatory Bone Diseasesmentioning

confidence: 99%

Pediatric autoinflammatory bone disorders—a mini review with special focus on pathogenesis and inborn errors of immunity

Hetrick

Oliver

2023

Front. Pediatr.

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Autoinflammatory bone disorders are a group of diseases characterized by sterile osteomyelitis. This includes chronic nonbacterial osteomyelitis and the monogenic forms, Majeed syndrome and deficiency of the interleukin-1 receptor antagonist. These disorders result from innate immune system dysregulation and cytokine imbalance that triggers inflammasome activation causing downstream osteoclastogenesis and excessive bone remodeling. In this review, we will summarize the immunopathogenesis of pediatric autoinflammatory bone diseases with a special focus on the genetics and inborn errors of immunity, while briefly touching on the clinical manifestations and management of each disease as well as areas for future research.

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

Cited by 12 publications

References 36 publications

Pyoderma gangrenosum following anti-TNF therapy in chronic recurrent multifocal osteomyelitis: drug reaction or cutaneous manifestation of the disease? A critical review on the topic with an emblematic case report

Pyoderma gangrenosum following anti-TNF therapy in chronic recurrent multifocal osteomyelitis: drug reaction or cutaneous manifestation of the disease? A critical review on the topic with an emblematic case report

The Pathophysiology and Treatment of Pyoderma Gangrenosum—Current Options and New Perspectives

Pediatric autoinflammatory bone disorders—a mini review with special focus on pathogenesis and inborn errors of immunity

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