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Silent aspiration in infants with Prader-Willi syndrome identified by videofluoroscopic swallow study.

Abstract Feeding intolerance in Prader-Willi syndrome (PWS) infants is well-recognized, but their swallow physiology is not well understood. Swallow dysfunction increases risks of respiratory compromise and choking, which have a high incidence in PWS. To investigate swallow pathology in PWS infants we undertook a retrospective review of videofluoroscopic swallow studies (VFSS) in infants with PWS seen at our institution. We hypothesize that VFSS will characterize swallow pathology suspected by clinical observation during a feeding evaluation and may help determine feeding safety in these infants.Retrospective review of 23 VFSS on 10 PWS infants (average age 9.7 ± 8.4 months; range 3 weeks-29 months). Logistic regression models evaluated associations between gender, genetic subtype, and growth hormone (GH) use on aspiration incidence. Polysomnographic (PSG) studies conducted on the same participant ±1 year from VFSS were examined to characterize respiratory abnormalities.There was a high rate of swallowing dysfunction (pharyngeal residue 71%, aspiration events 87%) and disordered sleep. All aspiration events were silent. There were no differences in rates of aspiration for gender, genetic subtype, or GH use.A high incidence of aspiration was identified indicating swallow dysfunction may frequently be present in infants with PWS. Comprehensive evaluation of feeding and swallowing is essential and requires a multidisciplinary approach. Providers should recognize risk factors for swallow dysfunction and consider a multidisciplinary approach to guide decision making and optimize feeding safety in PWS.
PMID
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Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start



 

PMID- 29390364
OWN - NLM
STAT- MEDLINE
DCOM- 20180212
LR  - 20180212
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 50
DP  - 2017 Dec
TI  - Silent aspiration in infants with Prader-Willi syndrome identified by
      videofluoroscopic swallow study.
PG  - e9256
LID - 10.1097/MD.0000000000009256 [doi]
AB  - Feeding intolerance in Prader-Willi syndrome (PWS) infants is well-recognized,
      but their swallow physiology is not well understood. Swallow dysfunction
      increases risks of respiratory compromise and choking, which have a high
      incidence in PWS. To investigate swallow pathology in PWS infants we undertook a 
      retrospective review of videofluoroscopic swallow studies (VFSS) in infants with 
      PWS seen at our institution. We hypothesize that VFSS will characterize swallow
      pathology suspected by clinical observation during a feeding evaluation and may
      help determine feeding safety in these infants.Retrospective review of 23 VFSS on
      10 PWS infants (average age 9.7 +/- 8.4 months; range 3 weeks-29 months).
      Logistic regression models evaluated associations between gender, genetic
      subtype, and growth hormone (GH) use on aspiration incidence. Polysomnographic
      (PSG) studies conducted on the same participant +/-1 year from VFSS were examined
      to characterize respiratory abnormalities.There was a high rate of swallowing
      dysfunction (pharyngeal residue 71%, aspiration events 87%) and disordered sleep.
      All aspiration events were silent. There were no differences in rates of
      aspiration for gender, genetic subtype, or GH use.A high incidence of aspiration 
      was identified indicating swallow dysfunction may frequently be present in
      infants with PWS. Comprehensive evaluation of feeding and swallowing is essential
      and requires a multidisciplinary approach. Providers should recognize risk
      factors for swallow dysfunction and consider a multidisciplinary approach to
      guide decision making and optimize feeding safety in PWS.
CI  - Copyright (c) 2017 The Authors. Published by Wolters Kluwer Health, Inc. All
      rights reserved.
FAU - Salehi, Parisa
AU  - Salehi P
AD  - Division of Endocrine, Seattle Children's Hospital.
FAU - Stafford, Holly J
AU  - Stafford HJ
AD  - University of Washington School of Nursing, University of Washington.
FAU - Glass, Robin P
AU  - Glass RP
AD  - Occupational Therapy Department, Seattle Children's Hospital.
AD  - Division of Rehabilitation Medicine, University of Washington.
FAU - Leavitt, Anne
AU  - Leavitt A
AD  - Division of Developmental Medicine.
FAU - Beck, Anita E
AU  - Beck AE
AD  - Division of Genetic Medicine.
FAU - McAfee, Amber
AU  - McAfee A
AD  - Division of Pulmonary and Sleep Medicine.
FAU - Ambartsumyan, Lusine
AU  - Ambartsumyan L
AD  - Division of Gastroenterology and Hepatology, Seattle Children's Hospital,
      University of Washington, Seattle, WA, USA.
FAU - Chen, Maida
AU  - Chen M
AD  - Division of Pulmonary and Sleep Medicine.
LA  - eng
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
SB  - AIM
SB  - IM
MH  - Child, Preschool
MH  - Deglutition Disorders/*physiopathology
MH  - Female
MH  - Fluoroscopy
MH  - Humans
MH  - Infant
MH  - Infant, Newborn
MH  - Male
MH  - Prader-Willi Syndrome/*physiopathology
MH  - Respiratory Aspiration/*diagnostic imaging/*physiopathology
MH  - Retrospective Studies
MH  - Risk Factors
MH  - Video Recording
EDAT- 2018/02/03 06:00
MHDA- 2018/02/13 06:00
CRDT- 2018/02/03 06:00
PHST- 2018/02/03 06:00 [entrez]
PHST- 2018/02/03 06:00 [pubmed]
PHST- 2018/02/13 06:00 [medline]
AID - 10.1097/MD.0000000000009256 [doi]
AID - 00005792-201712150-00114 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Dec;96(50):e9256. doi: 10.1097/MD.0000000000009256.