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An investigation of hearing status o...

O'Grady, Gwendolyn M.

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An investigation of hearing status of children with infantile Pompe disease.

Record Type:	Electronic resources : Monograph/item
Title/Author:	An investigation of hearing status of children with infantile Pompe disease./
Author:	O'Grady, Gwendolyn M.
Description:	162 p.
Notes:	Source: Dissertation Abstracts International, Volume: 67-11, Section: B, page: 6282.
Contained By:	Dissertation Abstracts International67-11B.
Subject:	Health Sciences, Audiology. -
Online resource:	http://pqdd.sinica.edu.tw/twdaoapp/servlet/advanced?query=3243445
ISBN:	9780542991769

An investigation of hearing status of children with infantile Pompe disease.
O'Grady, Gwendolyn M.

An investigation of hearing status of children with infantile Pompe disease. - 162 p.

Source: Dissertation Abstracts International, Volume: 67-11, Section: B, page: 6282.

Thesis (Ph.D.)--University of Kansas, 2006.

Objective. The purpose of this study was to determine if hearing loss is a characteristic feature of infantile Pompe disease and to determine if hearing loss occurred or progressed during a one-year clinical trial with Enzyme Replacement Therapy (ERT).

ISBN: 9780542991769Subjects--Topical Terms:

1018138
Health Sciences, Audiology.

An investigation of hearing status of children with infantile Pompe disease.
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100 1 $a O'Grady, Gwendolyn M. $3 1923100
245 1 3 $a An investigation of hearing status of children with infantile Pompe disease.
300 $a 162 p.
500 $a Source: Dissertation Abstracts International, Volume: 67-11, Section: B, page: 6282.
500 $a Adviser: Judith E. Widen.
502 $a Thesis (Ph.D.)--University of Kansas, 2006.
520 $a Objective. The purpose of this study was to determine if hearing loss is a characteristic feature of infantile Pompe disease and to determine if hearing loss occurred or progressed during a one-year clinical trial with Enzyme Replacement Therapy (ERT).
520 $a Methods. The study was a retrospective chart review of audiologic results obtained from 23 infants and children with Pompe disease. All of the participants were undergoing a clinic trial with Enzyme Replacement Therapy (ERT). The original study protocol planned for audiologic assessment at baseline (prior to ERT), at 26 weeks post first ERT infusion, and at 52 weeks post first ERT infusion. The hearing testing included tympanometry, distortion product otoacoustic emissions (DPOAEs), behavioral hearing assessment (when the participant was physically able), or Auditory Brainstem Response (ABR) testing if the participant could not complete behavioral assessment. The participants were recruited and enrolled in the clinical trial from 2001 through 2006.
520 $a Results. Test results were interpreted as (1) normal hearing bilaterally, (2) adequate hearing, defined as normal hearing in at least one ear, (3) conductive hearing loss, or (4) sensorineural hearing loss. Results revealed that 14 of the 23 participants had normal hearing bilaterally, and 6 of 23 had adequate hearing. Thus, no auditory (re)habilitation was recommended for 20 of the 23 (87%) participants. Of the remaining three participants, one had a conductive hearing loss, one had a sensorineural hearing loss and the hearing status could not be determined in one participant. The results also revealed that middle ear dysfunction, as assessed with tympanometry, was more prevalent than expected in the general pediatric population.
520 $a Conclusion. The results of audiologic assessment indicate that cochlear hearing loss is not a salient feature of infantile Pompe disease and that hearing loss does occur nor progress during ERT in the majority of patients with infantile Pompe disease.
590 $a School code: 0099.
650 4 $a Health Sciences, Audiology. $3 1018138
650 4 $a Health Sciences, Pathology. $3 1017854
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790 1 0 $a Widen, Judith E., $e advisor
790 $a 0099
791 $a Ph.D.
792 $a 2006
856 4 0 $u http://pqdd.sinica.edu.tw/twdaoapp/servlet/advanced?query=3243445