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Pyostomatitis vegetans in childhood

H S Al-Rimawi1, M M Hammad, E A Raweily

  • 1Paediatric Department, Jordan University of Science and Technology, Irbid-Jordan.

European Journal of Pediatrics
|June 13, 1998
PubMed
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This study reports the youngest familial cases of pyostomatitis vegetans, a rare oral condition linked to inflammatory bowel disease. The findings suggest a potential genetic link in children presenting with this triad.

Area of Science:

  • Oral Medicine
  • Dermatology
  • Gastroenterology

Background:

  • Pyostomatitis vegetans is an oral eruption associated with inflammatory bowel disease (IBD).
  • It is rarely diagnosed in children and typically presents in adults.
  • The condition involves pustules, ulcers, and vegetations on oral mucosa and gingiva.

Observation:

  • This report details a sister and brother diagnosed with pyostomatitis vegetans at ages 5 and 7.
  • These represent the youngest reported cases of familial pyostomatitis vegetans.
  • Both siblings also presented with pyoderma gangrenosum and IBD.

Findings:

  • The familial occurrence in very young children suggests a potential hereditary component.
  • The triad of pyostomatitis vegetans, pyoderma gangrenosum, and IBD may indicate a specific genetic predisposition.

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  • Early-onset familial cases highlight the need for genetic evaluation in affected children.

    • Implications:

    • This case series expands the understanding of pyostomatitis vegetans in pediatric populations.
    • It underscores the importance of recognizing the association between oral lesions, skin conditions, and IBD in children.
    • Further research into the genetic basis of this rare triad is warranted.