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Airway obstruction due to a rudimentary premaxilla in holoprosencephaly

F V Mehendale1, B C Sommerlad

  • 1St. Andrew's Centre for Plastic Surgery, Broomfield Hospital, Essex, London, England.

The Cleft Palate-Craniofacial Journal : Official Publication of the American Cleft Palate-Craniofacial Association
|June 2, 2001
PubMed

Insights

Two infants with holoprosencephaly experienced nasal obstruction from a mobile premaxilla. Early identification and management of this rudimentary structure are crucial for infant airway patency.

Area of Science:

  • Craniofacial anomalies
  • Pediatric surgery
  • Neonatal respiratory distress

Background:

  • Holoprosencephaly (HPE) is a congenital disorder affecting brain development, often associated with midline facial clefts.
  • Nasal airway obstruction is a significant concern in infants, impacting feeding and respiratory function.
  • The premaxilla's role in neonatal airway dynamics is not well-established, particularly in HPE cases.

Observation:

  • Two infants diagnosed with holoprosencephaly and primary cleft palate presented with severe nasal airway obstruction.
  • A previously undescribed cause was identified: a highly mobile, rudimentary premaxilla acting like a ball-valve.
  • This mobile premaxilla was attached to a soft tissue stalk, leading to intermittent airway blockage.

Findings:

  • The rudimentary premaxilla's ball-valve action was the primary cause of nasal obstruction in these infants.
  • Intranasal examination revealed the mobile nature and anatomical position of the premaxilla.
  • Conservative management, including surgical stabilization, was considered to address the obstruction.

Implications:

  • Highlights a novel cause of nasal airway obstruction in infants with holoprosencephaly.
  • Emphasizes the critical need for thorough intranasal examination in affected neonates.
  • Suggests preserving and potentially stabilizing the rudimentary premaxilla to ensure airway patency and improve infant outcomes.
Abstract

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