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Oral pemphigoid masquerading as necrotizing ulcerative gingivitis in a child.

Nour J Musa1, Vijay Kumar, Lewis Humphreys

  • 1Department of Oral Diagnostic Sciences, School of Dental Medicine, University at Buffalo, NY 14214, USA. neiders@buffalo.edu

Journal of Periodontology
|June 27, 2002
PubMed
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Cicatricial pemphigoid, a rare autoimmune condition, can manifest in children as necrotizing ulcerative gingivitis (NUG). Direct immunofluorescence is crucial for diagnosing this childhood oral mucosal disease.

Area of Science:

  • Autoimmune diseases
  • Pediatric dermatology
  • Oral medicine

Background:

  • Cicatricial pemphigoid (benign mucous membrane pemphigoid) is a rare autoimmune blistering disease affecting mucosal tissues, predominantly in adults.
  • Childhood cases are exceptionally rare, with only nine reported instances, typically presenting as oral lesions.
  • This case highlights a unique presentation of childhood cicatricial pemphigoid mimicking necrotizing ulcerative gingivitis (NUG).

Observation:

  • A 9-year-old girl presented with persistent gingival bleeding and discomfort unresponsive to standard NUG treatments.
  • Initial clinical suspicion was NUG, leading to antibiotic and oral hygiene interventions.
  • Lack of improvement prompted further diagnostic investigations, including histopathology and immunofluorescence.

Findings:

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  • Hematoxylin and eosin (H&E) staining revealed non-specific ulceration and chronic inflammation.
  • Direct immunofluorescence demonstrated linear C3 deposition at the basement membrane zone.
  • These immunofluorescence findings were diagnostic of cicatricial pemphigoid.

Implications:

  • Cicatricial pemphigoid should be considered in the differential diagnosis of refractory ulcerative oral lesions in children.
  • Immunofluorescence is essential for accurate diagnosis, distinguishing it from other oral ulcerative conditions.
  • Early and accurate diagnosis is vital for appropriate management of this rare pediatric autoimmune disorder.