The Relationship of Apnoea and Stridor in Spina Bifida to Other Unexplained Infant Deaths

Wealthall, S. R.; Whittaker, G. E.; Greenwood, Nancy

doi:10.1111/j.1469-8749.1974.tb03458.x

Cited by 25 publications

(4 citation statements)

References 12 publications

(6 reference statements)

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“…A lack of timely diagnosis and management can lead to untreated SDB, with a risk for significant morbidity and mortality. Children with CM1 are at an increased risk for neurocognitive complications, including reduced intellectual quotient scores and behavioural difficulties, and may also rarely be at increased risk for adverse cardiorespiratory outcomes and death (31)(32)(33). Clinical sleep history and physical examination are not sufficiently sensitive or specific to establish a diagnosis of sleep apnea in patients with CM1 (34).…”

Section: Discussionmentioning

confidence: 99%

The Association between Sleep‐Disordered Breathing and Magnetic Resonance Imaging Findings in a Pediatric Cohort with Chiari 1 Malformation

Amin

Sayal

et al. 2015

Canadian Respiratory Journal

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Section: Discussionmentioning

confidence: 99%

The Association between Sleep‐Disordered Breathing and Magnetic Resonance Imaging Findings in a Pediatric Cohort with Chiari 1 Malformation

Amin

Sayal

et al. 2015

Canadian Respiratory Journal

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“…Complete recovery has been observed in some infants treated only with vigorous supportive care, including intubation, tracheostomy, gastrostomy, supplemental oxygen and cardiorespiratory monitoring (Wealthall et al 1974). A better understanding of the natural history of ventilatory dysfunction should lead to a more rational approach to treatment (Park et al 1983, Charney et al 1987.…”

Section: Central Ventilatory Dysfunction (Cvd)mentioning

confidence: 99%

Central Ventilatory Dysfunction in Myelodysplasia: An Independent Determinant of Survival

Hays

Jordan

McLaughlin

et al. 1989

Develop Med Child Neuro

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SUMMARY Central ventilatory dysfunction (CVD) is a significant complication of myelodysplasia with Arnold‐Chiari type II defect. The records of 616 patients with myelodysplasia were reviewed and CVD was documented in 35 cases (5·7 per cent): it was the single most common cause of death in this population. There was no significant association between CVD and level of lesion, severity of hydrocephalus, hemorrhage or infection. The effects of CVD may be variable, with some patients having spontaneous remission and others having severe, often fatal, disruption of ventilation. RÉSUMÉ Perturbation centrale de la ventilation dans les myélodysplasies: factuer indépendant de survie La perturbation centrale de la ventilation (CVD) est une complication significative des myélodysplasies avec Arnold‐Chiari de type II. Les dossiers de 616 sujets myélodysplasiques ont été analysés et la CVD retrouvée dans 35 cas (5,7 pour cent): il s'agissait de la cause unique de décès la plus habituelle dans cette population. II n'y avait pas de relation significative avec le niveau lésionnel, la gravité de 1'hydrocephalic une hémorragie ou une infection. Les effets de la CVD étaient variables, la remission spontanée ayant été observée chez certains sujets, d'autres ayant présenté une interruption grave, souvent mortelle, de la respiration. ZUSAMMENFASSUNG Zentrale Atemstörung bei Myelodysplasia: ein entscheidender Überlebensfaktor Die zentrale Atemstörung (CVD) ist eine signifikante Komplikation der Myelodysplasie mit dem Arnold‐Chiari Defekt Typ II. Die Krankenakten von 616 Patienten mit Myelodysplasie wurden durchgesehen und in 35 Fällen wurde eine CVD gefunden (5·7 Prozent): sie war die häufigste alleinige Todesursache bei dieser Gruppe. Es fand sich keine signifikante Beziehung zwischen CVD und Höhe der Läsion, Schwere des Hydrocephalus, Blutung Oder Infektion. Die Auswirkungen der CVD können unterschiedlich sein, bei einigen Patienten können spontane Remissionen eintreten und andere haben schwere, oft fatale Atempausen. RESUMEN Disfuncion ventilatoria central en la mielodisplasia: un determinate independia para la supervivencia La disfunción ventilatoria central (DVC) es una complicación significativa de la mielodisplasia con defecto de Arnold‐Chiari del tipo II. Se revisaron las historias de 616 pacientes con mielodisplasia y se halo una DVC en 35 casos (5·7 por ciento): era la causa más corriente de defuncion en esta población. No habia asociación significativa entre la DVC y el nivel de la lesión, la gravedad del hidrocéfalo, la hemorragia y la infección. Los efectos de la DVC pueden ser variables, habiendo pacientes con remisión espontanea, mientras que otros sufren una disrupcion de la ventilación grave y a menudo fatal.

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“…While some studies observed clinical symptoms or MRI findings that may have been associated with SDB in that specific cohort of patients, 31 , 34 , 38 others did not observe any correlations between clinical symptoms or MRI findings and likelihood of developing SDB or SDB severity. 47 …”

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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The Relationship of Apnoea and Stridor in Spina Bifida to Other Unexplained Infant Deaths

Cited by 25 publications

References 12 publications

The Association between Sleep‐Disordered Breathing and Magnetic Resonance Imaging Findings in a Pediatric Cohort with Chiari 1 Malformation

The Association between Sleep‐Disordered Breathing and Magnetic Resonance Imaging Findings in a Pediatric Cohort with Chiari 1 Malformation

Central Ventilatory Dysfunction in Myelodysplasia: An Independent Determinant of Survival

Impact of Spina Bifida on Sleep Quality: Current Insights

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