Infliximab for a girl with refractory pyoderma gangrenosum

Abstract

Pyoderma gangrenosum is a rare chronic ulcerative noninfectious disease of the skin. Half of patients are complicated with other autoimmune diseases, most commonly inflammatory bowel disease, Takayasu disease, and rheumatoid arthritis. It has been reported that approximately 4% of them were childhood-onset. The conventional treatments of pyoderma gangrenosum were described as systemic corticosteroids and cyclosporine. The combination of corticosteroids with immunosuppressants such as tacrolimus, mycophenolate mofetil has been reported as steroid-sparing modalities. We herein reported a girl, 12 years of age, having pyoderma gangrenosum refractory to the conventional combination of systemic prednisolone with cyclosporine, but successfully treated with infliximab, the anti-TNFalpha monoclonal antibody. Rapid improvement of pyoderma gangrenosum was seen within three doses of infliximab infusion. All skin lesions eventually healed completely and new skin ulcers were never coming out again. The dramatical improvement suggested that infliximab should be considered for patients with refractory pyoderma gangrenosum though further experiences and investigations are required to determine the mechanism of infliximab.

Pyoderma gangrenosum with increased levels of serum cytokines

“…Moreover, neutrophilic dermatoses including PG have been reported to occur after therapeutic administration of G‐CSF . Significant upregulation of IL‐6 and IL‐8 has also been previously reported in PG . IL‐6 is a multifunctional cytokine that has a wide range of biological activities in various target cells, regulating immune response and acute phase reactions.…”

“…While the etiology of PG is still largely unknown, the disease is considered to be a part of the wider spectrum of neutrophilic dermatoses. Prior reports suggest that particular cytokines, such as granulocyte colony‐stimulating factor (G‐CSF), may be involved in the pathogenesis of these neutrophilic dermatoses . Here, we report a case of PG in which clinical symptoms were correlated with serum levels of G‐CSF and interleukin (IL)‐6.…”

Treatment of Pyoderma Gangrenosum in Pediatric Inflammatory Bowel Disease

“…Thirty-one references were identified, of which 9 were eliminated as lacking a discussion of specific clinical cases. Ten references [1][2][3][4][5][6][7][8][9][10] were excluded because they did not satisfy all of the above inclusion criteria. Twelve published studies met all search criteria and described a total of 19 patients (13 females, 6 males).…”