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The spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palate
  1. Joanna E MacLean1,2,3,
  2. David Fitzsimons2,3,
  3. Dominic A Fitzgerald2,4,
  4. Karen A Waters2,3,5
  1. 1Division of Respiratory Medicine, Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada
  2. 2Discipline of Paediatrics & Child Health, University of Sydney, Sydney, New South Wales, Australia
  3. 3Department of Respiratory Medicine, The Children's Hospital at Westmead, Westmead, New South Wales, Australia
  4. 4The Cleft Palate Clinic, The Children's Hospital at Westmead, Westmead, New South Wales, Australia
  5. 5Department of Physiology, University of Sydney, Sydney, New South Wales, Australia
  1. Correspondence to Dr Joanna E MacLean, Division of Respiratory Medicine, Department of Paediatrics, 4-590 Edmonton Clinic Health Academy (ECHA), 11405 87th Avenue, University of Alberta, Edmonton, AB T6G 1C9, Canada; Joanna.MacLean{at}ualberta.ca

Abstract

Objective To determine the prevalence of sleep-disordered breathing (SDB) symptoms and respiratory events during sleep in infants with cleft lip and/or palate (CL/P).

Design Prospective observational study.

Setting Cleft palate clinic, tertiary care paediatric hospital, before palate surgery.

Patients Consecutive newborn infants with CL/P.

Main outcome measures Demographics, clinical history, sleep symptoms, facial measurement and polysomnography (PSG; sleep study) data.

Results Fifty infants completed PSG at 2.7±2.3 months; 56% were male, and 30% had a clinical diagnosis of Pierre Robin sequence (PRS) or a syndrome. The majority of infants (75%) were reported to snore frequently or constantly, while 74% were reported to have heavy or loud breathing during sleep. The frequency of parent-reported difficulty with breathing during sleep was 10% for infants with isolated CL/P, 33% for those with syndrome, and 43% for PRS (χ2 16.1, p<0.05). All infants had an Obstructive–Mixed Apnoea–Hypopnoea Index (OMAHI) >1 event/h, and 75% had an OMAHI >3 events/h. Infants with PRS had higher OMAHI (34.3±5.1) than infants with isolated CL/P (7.6±1.2) or infants with syndromes (15.6±5.7, F stat, p<0.001). Multivariate analysis showed that PRS was associated with higher OMAHI (B 0.53±0.22, p=0.022), but the majority of the variance for SDB was unexplained (constant B 1.31±0.55, p=0.024).

Conclusions The results highlight that infants across the spectrum of CL/P have a high risk of SDB symptoms and obstructive respiratory events before palate surgery. Clinicians should enquire about symptoms of SDB and consider investigation with polysomnography in all infants with CL/P.

  • Syndrome
  • Sleep
  • Respiratory
  • Congenital Abnormalities

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