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Chronic bullous disease of childhood and ulcerative colitis
J Handley1, M Shields, J Dodge
1Department of Dermatology, Royal Victoria Hospital, Belfast, North Ireland.
Insights
A rare case of chronic bullous disease of childhood (CBDC) in a patient with ulcerative colitis (UC) resolved after rectal stump removal. This suggests a potential link between these conditions.
Area of Science:
- Pediatric Dermatology
- Gastroenterology
- Immunodermatology
Background:
- Ulcerative colitis (UC) is a chronic inflammatory bowel disease.
- Chronic bullous disease of childhood (CBDC) is a rare autoimmune blistering disorder.
- Colectomy with rectal stump preservation is a surgical option for UC.
Observation:
- A 12-year-old girl with UC developed typical features of CBDC, including oral ulcers and skin lesions.
- The patient's mucocutaneous symptoms were resistant to standard immunosuppressive therapies.
- Surgical resection of the rectal stump led to a striking remission of CBDC symptoms.
Findings:
- The clinical, histologic, and immunopathologic findings were characteristic of CBDC.
- Treatment resistance highlighted the need for alternative therapeutic strategies.
- Rectal stump resection induced significant improvement in mucocutaneous manifestations.
Implications:
- This case suggests a potential etiologic association between CBDC and UC.
- Rectal stump management may play a role in managing associated dermatologic conditions in IBD patients.
- Further research is warranted to explore the link between inflammatory bowel disease and autoimmune blistering disorders.
Abstract:
A 12-year-old girl with ulcerative colitis (UC) treated by colectomy with rectal stump preservation, salazopyrin, and systemic steroids developed persistent mouth ulcers, cutaneous targetlike lesions, papulopustules, and bullae. The clinical, histologic, and immunopathologic features were typical of chronic bullous disease of childhood (CBDC). Although resistant to combined immunosuppressive therapy with low-dose prednisolone, cyclosporin, and thalidomide, striking remission of the mucocutaneous symptoms resulted with surgical resection of the diseased rectal stump. An etiologic association between CBDC and UC is postulated.