Sleep-disordered breathing: an under-recognized problem in infants with myelomeningocele defects regardless of timing of repair

Bendel-Stenzel, Ellen; Linabery, Amy M.; Jorgenson, Alissa; Ferrara, Thomas B; Spaulding, Alicen B

doi:10.1038/s41372-019-0433-7

Cited by 7 publications

(1 citation statement)

References 13 publications

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“…Sleep related breathing disorders (SRBD) are highly prevalent in patients with myelomeningocele with up to 81% involvement in a cohort who had undergone screening polysomnography [5]. These SRBD are manifested as obstructive sleep apnea, central sleep apnea, periodic breathing, hypoxemia, and sleeprelated hypoventilation [4,[6][7][8][9][10][11]. In a study of 20 infants (5 with prenatal repair and 15 with postnatal repair), Shellhaas et al found no difference in apnea hypopnea index between newborns who had prenatal and postnatal repairs [12].…”

Section: Introductionmentioning

confidence: 99%

Sleep-related breathing disorders in infants with spina bifida repaired prenatally and postnatally

Stark,

Wang,

Smith

et al. 2024

Journal of Clinical Sleep Medicine

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Purpose: Recent advances in prenatal repair of myelomeningocele (MMC) have improved outcomes involving different organ systems. There is limited data on respiratory outcomes following prenatal surgical repair. We hypothesize there is no difference in respiratory outcomes between spina bi da (SB) patients who have undergone prenatal versus postnatal repair.Methods: Retrospective study of 46 infants <1 year with SB seen at Children's Hospital Los Angeles from 2004-2022. Demographic data, timing of closure, neonatal course, Chiari II malformation (CIIM), ventriculoperitoneal shunt (VPS), polysomnography (PSG) results, and need for supplemental oxygen were collected. Unpaired t test and Chi-square Test were used to compare results between groups.Results: 31/46 had prenatal repair of MMC; average age at repair was 27 weeks post-conception (PCA).Average age at postnatal repair was 37 PCA. There was no difference in age at PSG. There was no difference in CIIM presence (p=0.61). 60% of patients with postnatal repair and 23% in the prenatal group underwent VPS placement (p=0.01).There was no difference in PSG ndings between the two groups: CAI (p=0.11), OAHI (p=0.64), average SpO 2 baseline (p=0.91), average SpO 2 nadir (p=0.17), average PETCO 2 baseline (p=0.87), average PETCO2 maximum (p=0.54). There were no signi cant differences in the proportion of patients on supplemental O 2 (p=0.25), CSA or OSA between groups.Conclusions: Patients with SB who've undergone closure of defect have persistent central apneas, obstructive apneas, and signi cant hypoxemia. There were no differences in the frequency or severity of sleep-disordered breathing in those with prenatal repair versus postnatal repair. What Is KnownThere is a high prevalence of sleep related breathing disorders in patients with spina bi da.Prenatal surgery has improved outcomes involving mobility and need for VP shunt. What Is NewFollowing prenatal closure of open neural tube defect, infants with spina bi da may have persistent central and obstructive apneas and signi cant hypoxemia.The timing of closure does not affect the presence and severity of the sleep related breathing disorder.

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Section: Introductionmentioning

confidence: 99%

Sleep-related breathing disorders in infants with spina bifida repaired prenatally and postnatally

Stark,

Wang,

Smith

et al. 2024

Journal of Clinical Sleep Medicine

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Sleep‐disordered breathing on respiratory polygraphy in neonates with spina bifida

Wachsmuth,

Bieli,

Grehten

et al. 2024

Pediatric Pulmonology

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IntroductionStudies have shown a high prevalence of sleep‐disordered breathing (SDB) in children with spina bifida. International standards for regular testing for SDB in this population are lacking. While there are studies investigating the prevalence of SDB in children with spina bifida, there are close to no studies in neonates.Aim and ObjectiveTo evaluate if routine respiratory polygraphy (RPG) testing is indicated for neonates with spina bifida and if yes, with what therapeutic consequence.MethodsWe conducted a retrospective cohort study of all neonates with spina bifida at the University (Children's) Hospital Zurich after fetal spina bifida repair born between 2017 and 2022, who had undergone at least 1 RPG evaluation during hospitalization on the neonatal ward. RPG were evaluated by a blinded group of experienced pediatric pulmonologists. Based on the neonatal RPG results and pediatric pulmonologist's recommendation for caffeine therapy the spina bifida cohort was divided into two groups. Neonatal baseline RPG and follow‐up RPG at the age of the 3 months were evaluated.Results48 neonates with RPG were included. Compared to the standard values in healthy neonates, the RPG results of this spina bifida cohort showed findings of SDB with central apnea and hypopnea. 22 (45.8%) neonatal RPG evaluations detected central SDB, prompting caffeine therapy. Follow‐up RPG conducted after 3 months showed significant improvement of SDB with (almost) no need for continuation of caffeine.ConclusionWe recommend the implementation of routine RPG testing in neonates with spina bifida to detect SDB and facilitate early targeted treatment.

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Sleep Disordered Breathing in Neurodevelopmental Disorders

Imran

Arganbright

Ingram

2021

Curr Sleep Medicine Rep

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Sleep-disordered breathing: an under-recognized problem in infants with myelomeningocele defects regardless of timing of repair

Cited by 7 publications

References 13 publications

Sleep-related breathing disorders in infants with spina bifida repaired prenatally and postnatally

Sleep-related breathing disorders in infants with spina bifida repaired prenatally and postnatally

Sleep‐disordered breathing on respiratory polygraphy in neonates with spina bifida

Sleep Disordered Breathing in Neurodevelopmental Disorders

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