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Collagenous sprue in a 3-month-old infant
Keisuke Jimbo1, Yo Aoyagi, Masanori Tanaka
1Department of Pediatrics and Adolescent Medicine, Juntendo University Graduate School of Medicine, Tokyo, Japan.
Insights
Collagenous sprue (CS) is a rare malabsorption disorder. This case highlights successful treatment of infantile CS with steroids and immunomodulators, suggesting its consideration for infant diarrhea.
Area of Science:
- Gastroenterology
- Pediatric Pathology
- Immunology
Background:
- Collagenous sprue (CS) is a severe malabsorption disorder with an undefined etiology.
- Childhood-onset CS is exceptionally rare, with limited documented cases.
Observation:
- A 3-month-old infant presented with severe, intractable watery diarrhea.
- Endoscopy revealed an edematous duodenal mucosa, and biopsy showed severe villous atrophy with CD8-positive T cell infiltration.
- A thick subepithelial collagen band was identified in the duodenal biopsy.
Findings:
- The infant responded favorably to treatment with steroids and immunomodulators.
- Post-treatment, the subepithelial collagen deposits significantly diminished.
- This case represents one of the youngest documented instances of infantile CS.
Implications:
- CS should be considered in the differential diagnosis of intractable diarrhea in infants.
- Early diagnosis and treatment with immunomodulatory agents may lead to favorable outcomes in infantile CS.
- Further research into the etiology and pathogenesis of CS is warranted, particularly in pediatric populations.
Abstract:
Collagenous sprue (CS) is a severe malabsorption disorder, the etiology of which has not been well defined. Herein, we report the case of a 3-month-old infant with CS who responded to steroid and immunomodulator treatment and presented a thick subepithelial collagen band. A 3-month-old Japanese girl presented with severe watery diarrhea that lasted for 2 weeks. She was admitted to the referring hospital, but symptomatic improvement was not achieved with fasting and rehydration. Gastroduodenal endoscopy showed an edematous duodenal mucosal surface. Duodenal biopsy indicated severe villous atrophy with infiltration of mostly CD8-positive T cells; and deposition of subepithelial collagen was confirmed. The subepithelial collagen deposits, however, had disappeared after treatment. Historically, child-onset CS is extremely rare and this case is likely to be the youngest case of infantile CS. The present case suggests that CS should be considered as a differential diagnosis for intractable diarrhea, even in infants.
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