Sleep-Disordered Breathing among Newborns with Myelomeningocele

Shellhaas, Renée A.; Kenia, Payal V.; Hassan, Fauziya; Barks, John; Kaciroti, Niko; Chervin, Ronald D.

doi:10.1016/j.jpeds.2017.10.070

Cited by 23 publications

(11 citation statements)

References 13 publications

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“…Contrary to the hypothesis, similar proportions of unexplained deaths were noted regardless of shunt status, age, epilepsy, and gender. While MMC predisposes people to abnormal breathing during sleep [ 22 ], sleep apnea or brain MRI results were not available. It must be noted that patients with MMC are generally at higher risk for potential causes of sudden and sleep-related death, including cardiac arrhythmias, pulmonary emboli, anaphylaxis, and epilepsy.…”

Section: Discussionmentioning

confidence: 99%

Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study

Szymanski,

Adams,

Alkawaldeh

et al. 2023

PRM

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PURPOSE: This study aimed to analyze organ system-based causes and non-organ system-based mechanisms of death (COD, MOD) in people with myelomeningocele (MMC), comparing urological to other COD. METHODS: A retrospective review was performed of 16 institutions in Canada/United States of non-random convenience sample of people with MMC (born > = 1972) using non-parametric statistics. RESULTS: Of 293 deaths (89% shunted hydrocephalus), 12% occurred in infancy, 35% in childhood, and 53% in adulthood (documented COD: 74%). For 261 shunted individuals, leading COD were neurological (21%) and pulmonary (17%), and leading MOD were infections (34%, including shunt infections: 4%) and non-infectious shunt malfunctions (14%). For 32 unshunted individuals, leading COD were pulmonary (34%) and cardiovascular (13%), and leading MOD were infections (38%) and non-infectious pulmonary (16%). COD and MOD varied by shunt status and age (p < = 0.04), not ambulation or birthyear (p > = 0.16). Urology-related deaths (urosepsis, renal failure, hematuria, bladder perforation/cancer: 10%) were more likely in females (p = 0.01), independent of age, shunt, or ambulatory status (p > = 0.40). COD/MOD were independent of bladder augmentation (p = >0.11). Unexplained deaths while asleep (4%) were independent of age, shunt status, and epilepsy (p >= 0.47). CONCLUSION: COD varied by shunt status. Leading MOD were infectious. Urology-related deaths (10%) were independent of shunt status; 26% of COD were unknown. Life-long multidisciplinary care and accurate mortality documentation are needed.

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Section: Discussionmentioning

confidence: 99%

Causes of death among people with myelomeningocele: A multi-institutional 47-year retrospective study

Szymanski,

Adams,

Alkawaldeh

et al. 2023

PRM

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“… 51 However, no studies have consistently demonstrated an improvement in SDB after neurosurgical treatment in any children older than infancy. 4 , 51 …”

Section: Sleep-disordered Breathing In the Child With Spina Bifidamentioning

confidence: 99%

“…They found that infants with MMC had significantly higher AHI compared to control patients (34 events/hour vs 19 events/hour, respectively), with majority demonstrating a more predominant CSA index (10 events/hour vs 4 events/hour) and hypopnea index (21 events/hour vs 12 events/hour), versus OSA index (3 events/hour vs 2.5 events/hour). 51 …”

Section: Sleep-disordered Breathing In the Child With Spina Bifidamentioning

confidence: 99%

Impact of Spina Bifida on Sleep Quality: Current Insights

Gunnett,

Rocque,

Nourani

et al. 2023

NSS

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Spina bifida (SB) is one of the most common birth defects in children. The care for patients with SB continues to evolve, and there has been notable improvement in survival outcomes, degree of disability and quality of life for these children. However, patients with SB continue to remain at higher risk for sleep-related breathing disorders (SRBD), unexplained sudden death, and potential alterations in their sleep chronotype. Previous studies report on abnormalities in the spinal cord, brainstem function, and dysfunction of upper airway maintenance as the likely mechanisms behind SRBD that is commonly seen in SB. Most studies looking at prevalence of SRBD in SB have been retrospective studies. A recent prospective study identified a prevalence as high as 42% when a polysomnography (PSG) was completed on all patients regardless of symptomatology. Treatment options vary depending on the type and severity of SRBD and can range widely. Despite advances in care for patients with SB and SRBD, a subset of these patients with myelomeningocele (MMC) continue to experience sudden unexplained death. Studies continue to evaluate ways to stratify which of these patients may be at higher risk of this devastating outcome. Given that SRBD is potentially treatable, early assessment and intervention could become an integral part of a multidisciplinary treatment strategy to optimize long-term medical and neurodevelopmental outcomes for this patient population. By understanding the impact that SB may have on a patient’s sleep quality, their biological chronotype and their potential of developing SRBD, a provider may help to optimize the care a patient with SB receives from birth into adulthood.

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“…Literature suggests that a total AHI in excess of 5/h may normally occur in early infancy. [21][22][23][24] This may be due to a high frequency of both central events from immaturity of respiratory control and obstructive respiratory events secondary to physiological dynamic airway collapse. Daftary et al 24 reported a median AHI of 14.9/h with a significant contribution from central events in neonates.…”

Section: Definition Of Infant Osamentioning

confidence: 99%