Assessment of impaired coordination between respiration and deglutition in children and young adults with ataxia telangiectasia

Maureen A. Lefton-Greif; Adrienne L. Perlman; Xuming He; Howard M. Lederman; Thomas O. Crawford

doi:10.1111/dmcn.13156

Assessment of impaired coordination between respiration and deglutition in children and young adults with ataxia telangiectasia

Maureen A. Lefton-Greif, Adrienne L. Perlman, Xuming He, Howard M. Lederman, Thomas O. Crawford

School of Medicine

Research output: Contribution to journal › Article › peer-review

8 Scopus citations

Abstract

Aim: This cross-sectional investigation aimed to assess the value of non-invasive measures of temporal respiratory–swallow coupling in individuals with ataxic swallowing. Method: Twenty participants (11 males, 9 females; range 9-21y) with ataxia telangiectasia were presented with water and pudding boluses. Their 193 swallows were compared with 2200 swallows from 82 age-matched healthy controls. The two components of airway protection during swallowing that were analyzed were: direction of peri-deglutitive airflow and duration of deglutitive inhibition of respiratory airflow (DIORA). Results: Safe expiratory patterns of peri-deglutitive airflow occurred significantly less often in participants with ataxia telangiectasia than in age-matched control participants (younger p<0.015 and older p<0.001). The frequency of an expiratory pattern of peri-deglutitive airflow increased with age in participants in the comparison group (p=0.006), but not in those with ataxia telangiectasia (p=0.234). With age, mean duration of DIORA decreased in controls (p<0.001) but was unchanged in participants with ataxia telangiectasia (p=0.164). Interpretation: Non-invasive quantitative measures of respiratory–swallow coupling capture temporal relationships that plausibly contribute to airway compromise from dysphagia. Changes in respiratory–swallow coupling observed with advancing age in control participants were not seen in participants with ataxia telangiectasia. Measures of perturbations may herald swallowing problems prior to development of pulmonary and nutritional sequelae.

Original language	English (US)
Pages (from-to)	1069-1075
Number of pages	7
Journal	Developmental medicine and child neurology
Volume	58
Issue number	10
DOIs	https://doi.org/10.1111/dmcn.13156
State	Published - Oct 1 2016

ASJC Scopus subject areas

Pediatrics, Perinatology, and Child Health
Developmental Neuroscience
Clinical Neurology

Access to Document

10.1111/dmcn.13156

Cite this

@article{115b92c51ce04ded8e7c675b7eb0527c,

title = "Assessment of impaired coordination between respiration and deglutition in children and young adults with ataxia telangiectasia",

abstract = "Aim: This cross-sectional investigation aimed to assess the value of non-invasive measures of temporal respiratory–swallow coupling in individuals with ataxic swallowing. Method: Twenty participants (11 males, 9 females; range 9-21y) with ataxia telangiectasia were presented with water and pudding boluses. Their 193 swallows were compared with 2200 swallows from 82 age-matched healthy controls. The two components of airway protection during swallowing that were analyzed were: direction of peri-deglutitive airflow and duration of deglutitive inhibition of respiratory airflow (DIORA). Results: Safe expiratory patterns of peri-deglutitive airflow occurred significantly less often in participants with ataxia telangiectasia than in age-matched control participants (younger p<0.015 and older p<0.001). The frequency of an expiratory pattern of peri-deglutitive airflow increased with age in participants in the comparison group (p=0.006), but not in those with ataxia telangiectasia (p=0.234). With age, mean duration of DIORA decreased in controls (p<0.001) but was unchanged in participants with ataxia telangiectasia (p=0.164). Interpretation: Non-invasive quantitative measures of respiratory–swallow coupling capture temporal relationships that plausibly contribute to airway compromise from dysphagia. Changes in respiratory–swallow coupling observed with advancing age in control participants were not seen in participants with ataxia telangiectasia. Measures of perturbations may herald swallowing problems prior to development of pulmonary and nutritional sequelae.",

author = "Lefton-Greif, {Maureen A.} and Perlman, {Adrienne L.} and Xuming He and Lederman, {Howard M.} and Crawford, {Thomas O.}",

note = "Funding Information: The authors wish to thank the participants and their families for contributing to this project. We also are grateful for contributions of Karen Rosquist, RN, and Jennifer Wright, RN, nurse coordinators for the Ataxia-Telangiectasia Clinical Center; Georgia Malandraki, Yihe Zu, and Christina Bronson-Lowe for their assistance with data analysis and reliability measures; and Will Crawford and Renee Allard for their assistance with illustrations. This work was supported by the National Institutes of Health grant NIDCD R24 DC008646-02 and the Johns Hopkins Institute for Clinical and Translational Research (ICTR), which is funded in part by grant number UL1 TR 000424-06 from the National Center for Advancing Translational Sciences (NCATS), a component of the National Institutes of Health (NIH), and the NIH Roadmap for Medical Research. The content is solely the responsibility of the authors and does not necessarily represent the official views of NIH, the ICTR, or NCATS. Additional support was received from Ataxia Telangiectasia Children's Project, Coconut Creek, FL, USA. The authors have stated that they had no interests that might be perceived as posing a conflict or bias. Publisher Copyright: {\textcopyright} 2016 Mac Keith Press",

year = "2016",

month = oct,

day = "1",

doi = "10.1111/dmcn.13156",

language = "English (US)",

volume = "58",

pages = "1069--1075",

journal = "Developmental medicine and child neurology",

issn = "0012-1622",

publisher = "Wiley-Blackwell",

number = "10",

}

TY - JOUR

T1 - Assessment of impaired coordination between respiration and deglutition in children and young adults with ataxia telangiectasia

AU - Lefton-Greif, Maureen A.

AU - Perlman, Adrienne L.

AU - He, Xuming

AU - Lederman, Howard M.

AU - Crawford, Thomas O.

N1 - Funding Information: The authors wish to thank the participants and their families for contributing to this project. We also are grateful for contributions of Karen Rosquist, RN, and Jennifer Wright, RN, nurse coordinators for the Ataxia-Telangiectasia Clinical Center; Georgia Malandraki, Yihe Zu, and Christina Bronson-Lowe for their assistance with data analysis and reliability measures; and Will Crawford and Renee Allard for their assistance with illustrations. This work was supported by the National Institutes of Health grant NIDCD R24 DC008646-02 and the Johns Hopkins Institute for Clinical and Translational Research (ICTR), which is funded in part by grant number UL1 TR 000424-06 from the National Center for Advancing Translational Sciences (NCATS), a component of the National Institutes of Health (NIH), and the NIH Roadmap for Medical Research. The content is solely the responsibility of the authors and does not necessarily represent the official views of NIH, the ICTR, or NCATS. Additional support was received from Ataxia Telangiectasia Children's Project, Coconut Creek, FL, USA. The authors have stated that they had no interests that might be perceived as posing a conflict or bias. Publisher Copyright: © 2016 Mac Keith Press

PY - 2016/10/1

Y1 - 2016/10/1

N2 - Aim: This cross-sectional investigation aimed to assess the value of non-invasive measures of temporal respiratory–swallow coupling in individuals with ataxic swallowing. Method: Twenty participants (11 males, 9 females; range 9-21y) with ataxia telangiectasia were presented with water and pudding boluses. Their 193 swallows were compared with 2200 swallows from 82 age-matched healthy controls. The two components of airway protection during swallowing that were analyzed were: direction of peri-deglutitive airflow and duration of deglutitive inhibition of respiratory airflow (DIORA). Results: Safe expiratory patterns of peri-deglutitive airflow occurred significantly less often in participants with ataxia telangiectasia than in age-matched control participants (younger p<0.015 and older p<0.001). The frequency of an expiratory pattern of peri-deglutitive airflow increased with age in participants in the comparison group (p=0.006), but not in those with ataxia telangiectasia (p=0.234). With age, mean duration of DIORA decreased in controls (p<0.001) but was unchanged in participants with ataxia telangiectasia (p=0.164). Interpretation: Non-invasive quantitative measures of respiratory–swallow coupling capture temporal relationships that plausibly contribute to airway compromise from dysphagia. Changes in respiratory–swallow coupling observed with advancing age in control participants were not seen in participants with ataxia telangiectasia. Measures of perturbations may herald swallowing problems prior to development of pulmonary and nutritional sequelae.

AB - Aim: This cross-sectional investigation aimed to assess the value of non-invasive measures of temporal respiratory–swallow coupling in individuals with ataxic swallowing. Method: Twenty participants (11 males, 9 females; range 9-21y) with ataxia telangiectasia were presented with water and pudding boluses. Their 193 swallows were compared with 2200 swallows from 82 age-matched healthy controls. The two components of airway protection during swallowing that were analyzed were: direction of peri-deglutitive airflow and duration of deglutitive inhibition of respiratory airflow (DIORA). Results: Safe expiratory patterns of peri-deglutitive airflow occurred significantly less often in participants with ataxia telangiectasia than in age-matched control participants (younger p<0.015 and older p<0.001). The frequency of an expiratory pattern of peri-deglutitive airflow increased with age in participants in the comparison group (p=0.006), but not in those with ataxia telangiectasia (p=0.234). With age, mean duration of DIORA decreased in controls (p<0.001) but was unchanged in participants with ataxia telangiectasia (p=0.164). Interpretation: Non-invasive quantitative measures of respiratory–swallow coupling capture temporal relationships that plausibly contribute to airway compromise from dysphagia. Changes in respiratory–swallow coupling observed with advancing age in control participants were not seen in participants with ataxia telangiectasia. Measures of perturbations may herald swallowing problems prior to development of pulmonary and nutritional sequelae.

UR - http://www.scopus.com/inward/record.url?scp=84985986994&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84985986994&partnerID=8YFLogxK

U2 - 10.1111/dmcn.13156

DO - 10.1111/dmcn.13156

M3 - Article

C2 - 27214374

AN - SCOPUS:84985986994

SN - 0012-1622

VL - 58

SP - 1069

EP - 1075

JO - Developmental medicine and child neurology

JF - Developmental medicine and child neurology

IS - 10

ER -

Assessment of impaired coordination between respiration and deglutition in children and young adults with ataxia telangiectasia

Abstract

ASJC Scopus subject areas

Access to Document

Other files and links

Fingerprint

Cite this