Abstract

Abstract Two 12 year old boys presented to pediatric urology with chief complaints of genital swelling who were eventually diagnosed with Crohn’s disease. Patient 1 presented with swelling, erythema and discomfort of his penis. He did not respond to topical hydrocortisone or oral antibiotics. With history of short stature he was referred to pediatric GI. He had oral ulcerations and perianal skin tag, but denied abdominal pain or diarrhea. Weight was at the 3rd percentile height was at the 2nd percentile. He had Hgb of 12.2, and iron saturation of 8 with normal ESR and CRP. ASCA IgA was normal but IgG was elevated at 66 (normal <20). Endoscopy revealed erythema with linear ulcerations in the stomach and aphthous ulcerations of the colon with erythema and edema of the ileocecal valve with inability to intubate it. Pathology demonstrated mild chronic active gastritis and mild chronic active colitis with granulomas. Magnetic resonance enterography (MRE) showed mild enhancement at the ileocecal area. Budesonide improved the penile erythema but did not change the edema. He was then started on methotrexate and infliximab with no improvement of the edema. Topical tacrolimus did not resolve edema. At age 15 he had surgical resection of the indurated subcuticular penile tissues with pathology revealing fibrotic stroma with lymphoplasmacytic inflammation and rare granulomas. He has recovered well from his penile procedure with a dramatic improvement in the cosmetic appearance, with no recurrence of the penile edema. Patient 2 had scrotal edema, erythema, and discomfort. A scrotal ultrasound demonstrated moderate to severe scrotal edema without epididymitis, orchitis, or abscess. He was prescribed a dose of prednisone and diphenhydramine. Swelling improved but he presented a month later with recurrent scrotal edema and pain and was prescribed doxycycline for a presumed diagnosis of cellulitis along with recommendations for ice, compression and elevation. He returned to the ER the next day due to serous seepage from the scrotum. Upon follow up in urology, a pediatric gastroenterology referral was performed as a family history of Crohn’s disease in the mother, maternal aunt, and maternal grandmother was noted. Labs revealed a slightly elevated ESR of 17, elevated CRP of 7.3 (normal <0.5), negative ASCA, and mildly elevated fecal calprotectin of 193. Endoscopy revealed aphthous ulcerations in the duodenal bulb and patchy erythema of the terminal ileum. Pathology revealed mild reflux esophagitis, mild reactive changes in the stomach, mild chronic duodenitis, and mild active ileitis. MRE was normal. He was treated with budesonide and mesalamine with subsequent improvement in the scrotal edema. Patients presenting as genital edema as their initial Crohn’s presentation is rare and it is important for providers to be aware of this rare manifestation. Epitheliod granuloma in the bowel. Epitheliod granuloma in penile tissue.

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