Tracheal anomalies in Pfeiffer syndrome

Arch Otolaryngol Head Neck Surg. 2004 Nov;130(11):1298-302. doi: 10.1001/archotol.130.11.1298.

Abstract

Objective: To determine the types and frequency of airway anomalies in patients with Pfeiffer syndrome.

Design: Retrospective case series.

Setting: Academic tertiary care pediatric hospital.

Participants: Eleven patients with Pfeiffer syndrome, 6 of whom were severely affected, were identified. All were included in the study.

Main outcome measures: Presence of tracheal anomalies, need for tracheotomy, and length of life.

Results: The 6 severely affected patients had mutations in genes that code for fibroblast growth factor receptor 2 (S351C [3 patients]; C342S [2 patients]; and W290C [1 patient]). Five of these patients were diagnosed during bronchoscopy or tracheotomy as having a congenital tracheal cartilaginous sleeve. In 1 patient, supportive care was withdrawn at 2 weeks of life, and the patient died. The remaining 5 patients required tracheotomy because of severe upper airway obstruction. Three of these patients died (at ages 9 months and 7 and 15 years). Two are still alive at ages 23 and 18 months.

Conclusions: Patients with Pfeiffer syndrome manifest significant airway pathologic conditions. Upper airway obstruction is related to midface hypoplasia and secondary nasal obstruction. Tracheal anomalies have been infrequently reported.

MeSH terms

  • Acrocephalosyndactylia / genetics
  • Acrocephalosyndactylia / pathology*
  • Acrocephalosyndactylia / surgery
  • Adolescent
  • Bronchoscopy
  • Child
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Retrospective Studies
  • Trachea / abnormalities*
  • Trachea / pathology
  • Tracheotomy