Peripheral Chemoreceptor Function in Children With Myelomeningocele and Arnold-Chiari Malformation Type 2

Gozal, David; Arens, Raanan; Omlin, K. J.; Jacobs, Robert A.; Keens, Thomas G.

doi:10.1378/chest.108.2.425

Cited by 41 publications

(22 citation statements)

References 22 publications

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“…This malformation affects the brain stem and is known to influence both O 2 and CO 2 peripheral chemoreceptor function in children with SB (Gozal et al 1995;Petersen et al 1995). Future studies should include assessment of ventilatory reserve during exercise, so that a distinction can be been made between the different causes of pulmonary limitations, e.g.…”

Section: Peak Oxygen Uptakementioning

confidence: 99%

Limiting factors in peak oxygen uptake and the relationship with functional ambulation in ambulating children with Spina Bifida

et al. 2008

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The objective of this study is to interpret the outcomes of peak oxygen uptake (VO 2peak ) in children with SB and explore the relationship between VO 2peak and functional ambulation using retrospective cross-sectional study. Twenty-three ambulating children with SB participated at Wilhelmina's Children's Hospital Utrecht, the Netherlands. VO 2peak was measured during a graded treadmill-test. Eschenbacher's and Maninna's algorithm was used to determine limiting factors in reaching low VO 2peak values. Energy expenditure during locomotion (both O 2 rate and O 2 cost) and percentage of VO 2peak and HR peak were determined during a 6-min walking test (6MWT). Differences between community and normal ambulators were analyzed. VO 2peak , VO 2peak /kg, HR peak , RER peak and VE peak were significantly lower compared to reference values, with significant differences between normal and community ambulators. Limiting factors according to the algorithm were mostly ''muscular and/or deconditioning'' (47%) and ventilatory ''gasexchange'' (35%). Distance walked during 6MWT was 48.5% of predicted distance. Both O 2 rate and O 2 cost were high with significant differences between normal and community ambulators [17.6 vs. 21.9 ml/(kg min) and 0.27 vs 0.43 ml/ (kg m)]. Also %HR peak and %VO 2peak were significantly higher in community ambulators when compared to normal ambulators (resp. 97.6 vs. 75% and 90.2 vs. 55.9%). VO 2peak seems to be mostly limited by deconditioning and/ or muscular components and possible ventilatory factors. For both peak values and functional ambulation, community ambulators were significantly more impaired than normal ambulators. High energy expenditure, %VO 2peak and %HR peak reflect high level of strain during ambulation in the community ambulators. Future exercise testing in children with SB should include assessment of ventilatory reserve. Exercise training in ambulatory children should focus on increasing both VO 2peak and muscular endurance, as well as decreasing energy cost of locomotion.

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Section: Peak Oxygen Uptakementioning

confidence: 99%

Limiting factors in peak oxygen uptake and the relationship with functional ambulation in ambulating children with Spina Bifida

et al. 2008

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“…Outros [3][4][5]7,14,24 consideram que os sinais e sintomas estariam relacionados à desorganização estrutural do tronco encefálico, baseando-se em evidências clínicas e anatomopatológicas. Gozal et al 35 , sugerem que os quimiorreceptores periféricos de portadores da MChII seriam anormais, tendo como consequência os distúrbios respiratórios associados ao sono, frequentemente observados nestes pacientes. Mori e Nishimura 36 estudando potenciais evocados auditivos e somatosensitivos, observaram lenta maturação do tronco encefálico com sinais de deterioração, provavelmente resultando do estiramento de nervos cranianos baixos.…”

Section: Discussionunclassified

Malformação de Chiari do tipo II sintomática

Salomão

Bellas

Leibinger

et al. 1998

Arq. Neuro-Psiquiatr.

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RESUMO -Os autores analisam uma série de 17 crianças portadoras de mielodisplasias que desenvolveram sinais e sintomas da malformação de Chiari do tipo II. De acordo com a idade, dois grupos ficaram bem definidos: Grupo I, crianças no primeiro ano de vida, em que predominaram sinais e sintomas de comprometimento do tronco encefálico e nervos cranianos bulbares (n=13); Grupo II, composto por crianças com idade superior a um ano, em que as principais manifestações foram dor cervical e sinais cerebelares (n=4). O resultado do tratamento cirúrgico nos dois grupos foi distinto: enquanto a mortalidade no Grupo I atingiu 46,1%, nenhum paciente do Grupo II veio a falecer. O tratamento inicial consistiu na instalação ou revisão de derivação ventricular, sendo a descompressão crânio-vertebral reservada àqueles que não se beneficiaram com esses procedimentos. Os autores enfatizam a necessidade do imediato reconhecimento e tratamento do quadro, de modo a se obter resultados satisfatórios.PALAVRAS-CHAVE: mielomeningocele, malformação de Chiari do tipo II, malformação de ArnoldChiari, desconpressão de fossa craniana posterior, hidrocefalia, paralisia de cordas vocais. Symptomatic Chiari type II malformationABSTRACT -The Chiari type II malformation is the leading cause of death in infants with myelomeningocele. The authors report 17 cases of symptomatic Chiari type II malformation occurring in two distinct age dependent population. In Group I, 13 neonates and infants in the first year of life presented with cranial nerve and brain stem dysfunction characterized by vocal cord paralysis, apnea, dysphagia and laryngeal stridor. In Group II, 4 patients developed signs and symptoms after the first year of life. In this group, the presentation was more insidious and included neck pain and cerebellar manifestations. The surgical treatment consisted initially in shunt implantation or revision and when there was no improvement, posterior fossa decompression was performed. The response to the surgical treatment differed considerably between the two groups: older patients improved promptly after surgery and there was no casualties; in newborn and infants, especially those under 6 months of age, the mortality rate was 46.1%. The authors stress that prompt diagnosis and surgical intervention should be performed in order to produce a favorable outcome.

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“…The presence of CM-II, myelomeningocele, and associated hydrocephalus causes compression of the respiratory nuclei in the medulla and impingement of the lower cranial nerves controlling vocal cords and bulbar muscles involved in pharyngeal airway patency. 8,10,15 Furthermore, children with myelomeningocele have dysfunction of not only the central chemoreceptors, but also the peripheral chemoreceptors causing arousal deficits to respiratory stimuli. 9,18,20,22 These patients appear to have normal breathing during wakefulness but exhibit prolonged expiratory apnea with cyanosis during sleep that manifests mainly as an obstructive problem.…”

Section: Mechanism Of Sdb In Patients With Myelomeningocelementioning

confidence: 99%

“…Many of these abnormalities have the potential to alter respiratory pattern and reflex, leading to sleepdisordered breathing (SDB). 8,10,13 The patient with myelomeningocele may manifest respiratory abnormalities, such as apnea or hypopnea, that lead to life-threatening conditions.…”

mentioning

confidence: 99%

Sleep-disordered breathing in patients with myelomeningocele

Patel¹,

Rocque²,

Hopson³

et al. 2015

PED

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OBJECT A paucity of literature examines sleep apnea in patients with myelomeningocele, Chiari malformation Type II (CM-II), and related hydrocephalus. Even less is known about the effect of hydrocephalus treatment or CM-II decompression on sleep hygiene. This study is an exploratory analysis of sleep-disordered breathing in patients with myelomeningocele and the effects of neurosurgical treatments, in particular CM-II decompression and hydrocephalus management, on sleep organization. METHODS The authors performed a retrospective review of all patients seen in their multidisciplinary spina bifida clinic (approximately 435 patients with myelomeningocele) to evaluate polysomnographs obtained between March 1999 and July 2013. They analyzed symptoms prompting evaluation, results, and recommended interventions by using descriptive statistics. They also conducted a subset analysis of 9 children who had undergone polysomnography both before and after neurosurgical intervention. RESULTS Fifty-two patients had polysomnographs available for review. Sleep apnea was diagnosed in 81% of these patients. The most common presenting symptom was “breathing difficulties” (18 cases [43%]). Mild sleep apnea was present in 26 cases (50%), moderate in 10 (19%), and severe in 6 (12%). Among the 42 patients with abnormal sleep architecture, 30 had predominantly obstructive apneas and 12 had predominantly central apneas. The most common pulmonology-recommended intervention was adjustment of peripheral oxygen supplementation (24 cases [57%]), followed by initiation of peripheral oxygen (10 cases [24%]). In a subset analysis of 9 patients who had sleep studies before and after neurosurgical intervention, there was a trend toward a decrease in the mean number of respiratory events (from 34.8 to 15.9, p = 0.098), obstructive events (from 14.7 to 13.9, p = 0.85), and central events (from 20.1 to 2.25, p = 0.15) and in the apnea-hypopnea index (from 5.05 to 2.03, p = 0.038, not significant when corrected for multiple measures). CONCLUSIONS A large proportion of patients with myelomeningocele who had undergone polysomnography showed evidence of disordered sleep on an initial study. Furthermore, 31% of patients had moderate or severe obstructive sleep apnea. Myelomeningocele patients with an abnormal sleep structure who had undergone nonoperative treatment with peripheral oxygen supplementation showed improvement in the apnea-hypopnea index. Results in this study suggested that polysomnography in patients with myelomeningocele may present an opportunity to detect and classify sleep apnea, identify low-risk interventions, and prevent future implications of sleep-disordered breathing.

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Peripheral Chemoreceptor Function in Children With Myelomeningocele and Arnold-Chiari Malformation Type 2

Cited by 41 publications

References 22 publications

Limiting factors in peak oxygen uptake and the relationship with functional ambulation in ambulating children with Spina Bifida

Limiting factors in peak oxygen uptake and the relationship with functional ambulation in ambulating children with Spina Bifida

Malformação de Chiari do tipo II sintomática

Sleep-disordered breathing in patients with myelomeningocele

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