SEARCHCASE_REPORTpediatric ophthalmology, pediatrics

case_report·pediatric ophthalmology, pediatrics, gastroenterology, dermatology, immunology·PMC10191521

Treatment of Pyoderma Gangrenosum in Pediatric Inflammatory Bowel Disease

JPGN Reports · 4 authors, 1 centre

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TL;DR

A 17-year-old female with ulcerative colitis presented with severe pyoderma gangrenosum of the left lower leg that was refractory to prior antibiotics and topical corticosteroids.

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This case report describes a 17-year-old female with ulcerative colitis diagnosed at age 10, who presented at age 17 with a large left lower leg ulcerating lesion measuring approximately 15 x 7 cm, characterized by a violaceous border, purulent exudate, and necrosis, consistent with severe pyoderma gangrenosum confirmed histopathologically. Laboratory studies showed marked elevations of erythrocyte sedimentation rate (>130 mm/h) and C-reactive protein (>40 mg/L). The patient had been treated at age 13 for what was diagnosed as a venous stasis ulcer with multiple courses of antibiotics and topical corticosteroids without resolution. A literature review of 12 published studies identified 19 pediatric IBD-associated PG cases (13 with Crohn's disease, 6 with ulcerative colitis).

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case_report

SPECIALTY

pediatric ophthalmology

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ORIGINAL PAPER

PMC10191521

Treatment of Pyoderma Gangrenosum in Pediatric Inflammatory Bowel Disease

JPGN Reports

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ABSTRACT

Abstract Pyoderma gangrenosum (PG) is a rare, necrotizing dermatologic condition associated with neoplastic and immune dysregulatory states, including adult and pediatric inflammatory bowel disease (IBD). Over the last decade, the elucidation of inflammatory mediators in PG has led to a plethora of localized and systemic corticosteroid sparing therapies including antibiotics, antiinflammatory, and immunomodulatory agents. Herein, we describe the case of a 17-year-old female with ulcerative colitis in clinical remission, who presented with a long-standing, large, deep, and painful lower extremity PG lesion. Following failed attempts both at local and at systemic therapies, her PG was successfully treated with the tumor necrosis factor-alpha (TNF-α) monoclonal antibody adalimumab, and the lesion remains in remission after four years of subcutaneous anti-TNF therapy. This case serves as the basis for our presenting a review of the pathogenesis, diagnostic criteria, differential diagnosis, therapies and treatment outcomes for pediatric IBD-associated PG. Our experience adds to earlier reports suggesting anti-TNF-α biologic therapy is most likely to achieve long-term resolution of IBD-associated PG in children and adolescents with severe lesions or who failed other treatments. Keywords: adalimumab, corticosteroids, immunomodulators, infliximab, inflammatory bowel disease, tumor necrosis factor-α

CITATION EXPORT

Katherine Vaidy, Rebecca Winderman, Simon S Rabinowitz et al.. Treatment of Pyoderma Gangrenosum in Pediatric Inflammatory Bowel Disease. JPGN Reports. (2020). https://doi.org/10.1097/PG9.0000000000000008

AUTHORS

4 · 1 CENTRES

Katherine Vaidy, Rebecca Winderman, Simon S Rabinowitz, Steven M Schwarz

1From the Division of Gastroenterology, Hepatology and Nutrition, Children’s Hospital at Downstate, SUNY-Downstate Health Sciences University, Brooklyn, NY.