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Colon perforation in hyperimmunoglobulin E syndrome

E H Hwang1, J T Oh, S J Han

  • 1Department of Pediatric Surgery and Pathology, Yonsei University College of Medicine, Seoul, Korea.

Journal of Pediatric Surgery
|October 10, 1998
PubMed
Summary
This summary is machine-generated.

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Colon perforation is a rare complication of hyperimmunoglobulin E syndrome. This case report details a successful surgical intervention for a child experiencing this rare condition, highlighting the importance of prompt treatment for improved outcomes.

Area of Science:

  • Gastroenterology
  • Immunology
  • Pediatric Surgery

Background:

  • Hyperimmunoglobulin E syndrome (HIES) is a rare primary immunodeficiency characterized by recurrent infections, eczema, and elevated serum IgE levels.
  • Gastrointestinal complications, such as colon perforation, are exceptionally rare in HIES patients.
  • Literature review indicates only one previously reported case of colon perforation associated with HIES.

Observation:

  • An 8-year-old girl with a known diagnosis of HIES presented with symptoms suggestive of abdominal catastrophe.
  • Physical examination revealed panperitonitis, indicating widespread inflammation of the abdominal lining.
  • Diagnostic imaging confirmed a spontaneous colon perforation as the cause of peritonitis.

Findings:

  • The patient underwent emergency surgery involving resection of the perforated colon segment.

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  • A double-barrel colostomy was performed as part of the surgical management.
  • Histopathological examination confirmed the colon perforation, ruling out other potential causes.

    • Implications:

    • This case adds to the limited literature on gastrointestinal manifestations of HIES.
    • Prompt surgical management, including resection and colostomy, can lead to favorable outcomes in pediatric patients with HIES-related colon perforation.
    • Further research is warranted to understand the underlying mechanisms linking HIES and gastrointestinal perforation.