Evidence map›Paper›PMID 35488943›Full record

ArticleMetabolic brain disease2022

Does glutaric aciduria type 1 affect hearing function?

Dilek Demiral Özgedi K, Suna Tokgöz Yılmaz, Berrak Bilginer Gürbüz, H Serap Si Vri, Gonca Sennaroğlu

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Article in Metabolic brain disease, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

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0 citing papers in PubMed, 1 citations in OpenAlex.

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4 · The record

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5 · Who and what money

Authors and funding

5 authors at 3 institutions in 1 country.

Dilek Demiral Özgedi KHealth Sciences Faculty, Department of Speech Language Therapy, Lokman Hekim University, Söğütözü Mh. 2179 Cd. No: 6 Çankaya, 06510, Ankara, Turkey. dilek.demir10@yahoo.com.ORCID 0000-0003-2690-2750
Suna Tokgöz YılmazHealth Sciences Faculty, Audiology Department, Ankara University, Ankara, Turkey.ORCID 0000-0002-4656-099X
Berrak Bilginer GürbüzFaculty of Medicine, Department of Pediatrics, Metabolism Unit, Hacettepe University, Ankara, Turkey.ORCID 0000-0002-6197-0647
H Serap Si VriFaculty of Medicine, Department of Pediatrics, Metabolism Unit, Hacettepe University, Ankara, Turkey.ORCID 0000-0001-8260-9984
Gonca SennaroğluHealth Sciences Faculty, Audiology Department, Hacettepe University, Ankara, Turkey.ORCID 0000-0002-3337-2391
Hacettepe University · TRAnkara University · TRLokman Hekim Üniversitesi · TR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

This study aimed to evaluate audiological findings among patients with glutaric aciduria type 1 (GA-1). We used a large test battery for the audiological evaluation of 17 individuals with GA-1 (the study group) and 20 healthy individuals (the control group). Conventional audiometry (0.125-8 kHz), distortion product otoacoustic emissions (DPOAEs) (1, 1.5, 2, 3, 4, 6, and 8 kHz), contralateral suppression of otoacoustic emissions, and auditory brainstem response (ABR) ( 30, 50, 70 and 90 dB nHL) were measured for all participants (n = 37). Mild sensorineural hearing loss was found in 77.47% (n = 13) of the patients with GA-1, and normal hearing thresholds were seen in 23.53% (n = 4). There were three asymptomatic patients at the time of diagnosis [two developed mild mental motor retardation (MMR) and one developed severe MMR during the follow-up], one with a normal hearing threshold and two with mild hearing loss), and 14 symptomatic patients (three with normal hearing thresholds and 11 with mild hearing loss). Seven of the symptomatic patients diagnosed following an encephalopathic crisis required intensive care and showed significantly worse hearing thresholds than those without symptoms [20.86 ± 4.47 vs. 15.44 ± 3.96 decibel hearing level (dB HL), p = 0.039*], while five had mild-to-moderate hearing loss. Acute encephalopathic crisis had a negative effect on hearing function in the symptomatic patients. The emission and contralateral suppression amplitude values of the study group were significantly lower compared to the control group (p < 0.05). The I-V interpeak latency and absolute latencies of ABR waves I, III, and V of the study group were observed to be significantly prolonged and morphologically distorted compared to those of the control group (p < 0.05). Five patients had MMR, and three had moderate MMR; all eight had mild-to-moderate hearing loss. In addition, of the eight patients with mild MMR, four had mild hearing loss. In particular, the morphological findings of ABR waves were significantly worse in the patients with severe and moderate MMR (p < 0.05). There was a significant correlation between a macrocephaly history (12 patients) and hearing loss (p = 0.041*). Magnetic resonance imaging findings were evaluated in all the 17 patients with GA-1, and typical fronto-temporal atrophy and sylvian fissure enlargement were observed. Our findings support that GA-1 is associated with auditory impairment, primarily in symptomatic patients. Adequate audiological test battery evaluation is essential in this context, particularly for symptomatic patients with a history of encephalopathic crises.

Indexed as

HearingHearing LossAmino Acid Metabolism, Inborn ErrorsAudiometry, Pure-ToneAuditory ThresholdBrain Diseases, MetabolicGlutaryl-CoA DehydrogenaseHumansGlutaryl-CoA DehydrogenaseAuditory brainstem responseContralateral suppressionDistortion product otoacoustic emissionsGlutaric aciduria type 1Pure tone hearing thresholds

Identifiers

PMID35488943
OpenAlexW4225133886

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.