Sleep breathing disorders in pediatric patients with spinal muscular atrophy 2

Bertran, Katalina; O, Sans Capdevila; Nascimiento, Andrés; Ortez, C.; Natera, D.; Riquer, Alex Iranzo de

doi:10.1016/j.sleep.2021.11.006

Cited by 9 publications

(11 citation statements)

References 15 publications

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“…18 but have limitations of subjectivity because nocturnal hypopnea can also be found in pediatric SMA patients without clinical symptoms. 19,20 Sleepdisordered breathing is associated with RRTIs in children with SMA, and long-term noninvasive ventilation can improve PSG scores and reduce the incidence of respiratory tract infection. 21 Our study used SMA children as samples and analyzed the association between PSG scores and the occurrence of RRTIs and ARF, which ensured the objectivity of the results and applicability to SMA disease.…”

Section: Discussionmentioning

confidence: 99%

Risk factors for recurrent respiratory tract infections or acute respiratory failure in children with spinal muscular atrophy

Guo

Meng

Cao

2022

Preprint

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Introduction: Assessment of and intervention for sleep-disordered breathing and malnutrition are related to respiratory management for preventing recurrent respiratory tract infections (RRTIs) and acute respiratory failure (ARF) in children with spinal muscular atrophy (SMA). However, the specific standard has not been clarified. Purpose: The study aimed to obtain the risk factors and the predictive index for RRTIs and/or ARF in children with SMA. Methods: In this retrospective study, the differences in clinical characteristics in patients with or without RRTIs and ARF were compared, and binary logistic regression analysis was carried out. The best cutoff points of the positive predictive index were obtained. Results: Type 1 (OR = 4.12, 95% CI 1.30-13.07, P =.016) and apnea hypopnea index (AHI) (OR = 1.14, 95% CI 1.05-1.24, P =.001) were risk factors, while body mass index z score (BMIz) (OR = 0.68, 95% CI 0.49-0.94, P =.018) and mean pulse oxygen saturation (MSpO ) (OR = 0.67, 95% CI 0.50-0.91, P =.010) were protective factors. The sensitivity and specificity of the standard of MSpO < 96% and AHI > 10 events/h or BMIz < -1 with the occurrence of RRTIs and/or ARF were 0.513 and 0.957, respectively. Conclusion: SMA Type 1, BMIz, AHI and MSpO should be used to estimate the risk for RRTI or ARF in children with SMA. MSpO < 96%, and AHI > 10 events/h or BMIz < -1 should be used as the intervention standard.

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Section: Discussionmentioning

confidence: 99%

Risk factors for recurrent respiratory tract infections or acute respiratory failure in children with spinal muscular atrophy

Guo

Meng

Cao

2022

Preprint

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“…This standard may be used as a reference for SMA, but the data were not obtained from SMA patients themselves. The questionnaires could be used to assess sleep disorders in SMA, 18 but have limitations of subjectivity because nocturnal hypoventilation can also be found in pediatric SMA patients without clinical symptoms 19,20 . Sleep‐disordered breathing is associated with RRTIs in children with SMA, and long‐term noninvasive ventilation can improve PSG scores and reduce the incidence of respiratory tract infection 21 .…”

Section: Discussionmentioning

confidence: 99%

“…The questionnaires could be used to assess sleep disorders in SMA, 18 but have limitations of subjectivity because nocturnal hypoventilation can also be found in pediatric SMA patients without clinical symptoms. 19 , 20 Sleep‐disordered breathing is associated with RRTIs in children with SMA, and long‐term noninvasive ventilation can improve PSG scores and reduce the incidence of respiratory tract infection. 21 Our study used SMA children as samples and analyzed the association between PSG scores and the occurrence of RRTIs and ARF, which ensured the objectivity of the results and applicability to SMA disease.…”

Section: Discussionmentioning

confidence: 99%

Risk factors for recurrent respiratory tract infections and acute respiratory failure in children with spinal muscular atrophy

Guo

Meng

Cao

2022

Pediatric Pulmonology

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Introduction: Assessment of and intervention for sleep-disordered breathing and malnutrition are related to the prevention of recurrent respiratory tract infections (RRTIs) and acute respiratory failure (ARF) in children with spinal muscular atrophy (SMA). However, specific standards for sleep-disordered breathing and malnutrition in the prevention of RRTIs and ARF have not been clarified. Purpose:The study aimed to identify the risk factors and predictive indices for RRTIs and/or ARF in children with SMA.Methods: In this retrospective study, the differences in clinical characteristics between patients with and without RRTIs and ARF were compared, and binary logistic regression analysis was carried out. The optimal cutoff points for positive predictors were obtained.Results: SMA type 1 (odds ratio (OR) = 5.21, 95% confidence interval (CI) 1.50-18.17, p = 0.010) and the apnea-hypopnea index (AHI) (OR = 1.12, 95% CI 1.01-1.24, p = 0.026) were risk factors, while the body mass index z score (BMIz) (OR = 0.65, 95% CI 0.46-0.91, p = 0.013) and mean pulse oxygen saturation (MSpO 2 ) (OR = 0.72, 95% CI 0.52-1.00, p = 0.049) were protective factors. A standard consisting of (i) MSpO 2 < 96% and (ii) AHI > 10 events/h and/or BMIz < -1 predicted the occurrence of RRTIs and/or ARF in the next year with a sensitivity of 0.513 and a specificity of 0.957. Conclusion:SMA type 1, BMIz, AHI and MSpO 2 should be used to estimate the risk of RRTI and/or ARF in children with SMA. MSpO 2 < 96% combined with AHI > 10 events/h or BMIz < -1 should be used as the intervention standard.

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“…19 Whilst in DMD the nocturnal features have been associated with the stages of progression of the disease, 7,13 in spinal muscular atrophy the prevalence of SDB has been defined by a relatively small number of studies, and its association with NHH has not been fully clarified. 20,21 In conclusion, the path to the Holy Grail of the perfect predictor to NHH is still quite long. A disease-specific approach for SDB in neuromuscular conditions is most likely going to be the key.…”

Section: Daytime Predictors Of Nocturnal Hypercapnic Hypoventilation ...mentioning

confidence: 96%

“…Whilst in DMD the nocturnal features have been associated with the stages of progression of the disease, 7,13 in spinal muscular atrophy the prevalence of SDB has been defined by a relatively small number of studies, and its association with NHH has not been fully clarified 20,21 …”

Section: Study Population Design Nhh Definition Daytime Predictor Of ...mentioning

confidence: 99%

Daytime predictors of nocturnal hypercapnic hypoventilation in children with neuromuscular disorders—The Holy Grail

Trucco

2022

Pediatric Pulmonology

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Sleep breathing disorders in pediatric patients with spinal muscular atrophy 2

Cited by 9 publications

References 15 publications

Risk factors for recurrent respiratory tract infections or acute respiratory failure in children with spinal muscular atrophy

Risk factors for recurrent respiratory tract infections or acute respiratory failure in children with spinal muscular atrophy

Risk factors for recurrent respiratory tract infections and acute respiratory failure in children with spinal muscular atrophy

Daytime predictors of nocturnal hypercapnic hypoventilation in children with neuromuscular disorders—The Holy Grail

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