The spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palate

MacLean, Joanna E.; Fitzsimons, David; Fitzgerald, Dominic A.; Waters, Karen A.

doi:10.1136/archdischild-2012-302104

Cited by 89 publications

(78 citation statements)

References 48 publications

(33 reference statements)

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“…In achondroplasia, we reported a very weak correlation between clinical signs and PSG results 6 . One particularly interesting study compared sleep data in 12 infants with Sleep-disordered breathing is also very frequent in case of cleft palate, with severity increasing from isolated cleft to syndromic cleft to PierreRobinsequence 9 ( fig.1 and fig. 2).…”

Section: Introductionmentioning

confidence: 99%

The particular case of sleep-disordered breathing in syndromic patients

Fauroux

Amaddeo

2015

J Dentofacial Anom Orthod

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This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.Classical childhood obstructive sleep apnea syndrome (OSAS) is a relatively frequent pathology, affecting 1% to 2% of children between the ages of 3 and 6 years 10 . Adenotonsillar hypertrophy is the primary cause. It is associated with nocturnal and diurnal signs, of variable severity. Nocturnal signs may include snoring, apneas, night-sweats, nycturia, parasomnia, agitated sleep and sometimes abnormal sleeping posture with the head in hyperextension. Diurnal signs comprise difficulty in awakening, irritability, hyperactivity, attention and memory disorder, asthenia, daytime somnolence, headache or vomiting, breakfast anorexia, oral breathing or growth disorder (only seen INTRODUCTIONThe particular case of sleep-disordered breathing in syndromic patients KEY WORDSObstructive sleep apnea syndrome, genetic diseases, rare diseases, upper airway deformity, continuous positive airway pressure (CPAP)

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Section: Introductionmentioning

confidence: 99%

The particular case of sleep-disordered breathing in syndromic patients

Fauroux

Amaddeo

2015

J Dentofacial Anom Orthod

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“…Infants with cleft lip and or palate have a higher incidence of airway obstruction and sleep disordered breathing. 7 Even in the presence of a single underlying anatomic defect as cleft lip, it is prudent to look for possible coexistent etiologies contributing to OSA which could be treatable. Our patient also had laryngomalacia and recurrent respiratory infections.…”

Section: Discussionmentioning

confidence: 99%

Vallecular Cyst as a Cause of obstructive Sleep apnea in an infant

Don

Magee

et al. 2013

Journal of Clinical Sleep Medicine

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A 3-month-old baby was diagnosed with obstructive sleep apnea (OSA) on polysomnography (PSG) with a high apnea hypopnea index (AHI). On further investigations he was found to have a vallecular cyst that was successfully treated. We discuss the clinical presentation of vallecular cysts and the importance of polysomnography in identifying this rare condition. REpoRt oF CaSEA 3-month-old baby with a history of incomplete right cleft lip and alveolus was referred by the Craniofacial Surgery team for PSG for evaluation prior to surgical closure. Patient was born full term via C-section for fetal decelerations with an uneventful perinatal course. He had stridor, snoring, and gasping during sleep since birth. A PSG was performed during daytime nap. He slept for 64 minutes with an arousal/awakening index of 5.6 per hour. He was found to have 45 obstructive apneas, 2 obstructive hypopneas, and 4 mixed apneas (AHI 64.8/h, baseline oxygen saturation 94% to 96%, desaturation index 89/h, SpO 2 nadir 75%, P ET CO 2 highest 47 torr). Due to OSA severity, he was admitted for further investigation.On physical exam, he was well developed, noticeably stridulous, with high arched palate and right incomplete lip with alveolar cleft. He had a pectus excavatum deformity with mild subcostal retractions but good bilateral air entry and clear breath sounds.Chest x-ray incidentally showed a right middle lobe infi ltrate, and he was found to be positive for metapneumovirus. He was treated for presumed aspiration pneumonia, and refl ux management was initiated for clinical evidence of refl ux. He was noted to have laryngomalacia on bedside laryngoscopy performed by the otorhinolaryngologist. He was discharged home on oxygen supplementation at half liter per minute (Lpm) via nasal cannula. He had a subsequent hospitalization for RSV bronchiolitis within two weeks of discharge.Subsequently, on outpatient follow-up with the otorhinolaryngologist, full offi ce laryngoscopy revealed a vallecular cyst (Figure 1) cyst was successfully marsupialized. Six weeks later, a repeat PSG during daytime nap was performed; it showed marked improvement of his obstructive apneas, and reduction of AHI from 64.8 to 7.2 (baseline oxygen saturation 96% to 98%; desaturation index 12/h; SpO 2 nadir 76%, P ET CO 2 highest 44 torr). His need for supplemental oxygen was reduced to 0.25 Lpm. He had his cleft lip successfully repaired at 9 months of age, and a follow-up PSG during daytime nap was completely normal (AHI 0.2/h; baseline oxygen saturation 95% to 97%; desaturation index 1.2/h; SpO 2 nadir 91%, P ET CO 2 highest 45 torr).

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“…A prospective study of 50 infants found a mean obstructive AHI of 7.6/h in those with non-syndromic CLP. 62 One of the difficulties in determining the prevalence of and risk factors for OSAS in the CLP population is the inherent heterogeneity of presentation in these children.…”

Section: Cleft Lip/palatementioning

confidence: 99%