Morphological variability in the inner ear of mice with osteogenesis imperfecta

Lila A. Huston; Tooba S. Husain; Jacob C. Moore; Jason M. Organ; Rachel A. Menegaz; Emma K. Handler; Lauren A. Gonzales

doi:10.1002/ar.25298

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Morphological variability in the inner ear of mice with osteogenesis imperfecta

Journal article

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Morphological variability in the inner ear of mice with osteogenesis imperfecta

Lila A. Huston, Tooba S. Husain, Jacob C. Moore, Jason M. Organ, Rachel A. Menegaz, Emma K. Handler and Lauren A. Gonzales

Anatomical record (Hoboken, N.J. : 2007), Vol.307(3), pp.592-599

03/2024

DOI: 10.1002/ar.25298

PMID: 37515586

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Abstract

Abstract Osteogenesis imperfecta (OI) is known to cause hearing loss in ~60% of the affected human population. While OI‐related pathologies have been studied in the middle ear, the development of cochlear pathologies is less well understood. In this study, we examine OI‐related pathologies of the cochlea in a mouse model of OI to (1) document variation between OI and unaffected mice, and (2) assess the intrusion of the otic capsule onto the cochlea by analyzing differences in duct volumes. Juvenile and adult OIM C57BL/6mice were compared to unaffected wildtype (WT) mice using three‐dimensional models of the cochlea generated from high resolution micro‐CT scans. Two‐tailed Mann–Whitney U tests were then used to investigate duct volume differences both within and between the OI and WT samples. Areas of higher ossification were observed at the cochlear base in the OI sample. OI mice also had significant intraindividual differences in duct volume between right and left ears (4%–15%), an effect not observed in WT mice. WT and OI duct volumes showed a large degree of overlap, although the OIM volumes were more variable. Our findings indicate that OIM mice are likely to exhibit more asymmetry and variation in cochlear volume despite minor differences in sample cochlear volumes, possibly due to bony capsule intrusion. This suggests a potential mechanism of hearing loss, and a high potential for cochlear and otic capsule alteration in OIM mice.

Details

Title: Subtitle: Morphological variability in the inner ear of mice with osteogenesis imperfecta
Creators: Lila A. Huston - University of North Texas Health Science Center
Tooba S. Husain - University of North Texas Health Science Center
Jacob C. Moore - Department of Physiology and Anatomy University of North Texas Health Science Center Fort Worth Texas USA, Edward Via College of Osteopathic Medicine Monroe Louisiana USA
Jason M. Organ - Indiana University – Purdue University Indianapolis
Rachel A. Menegaz - University of North Texas Health Science Center
Emma K. Handler - University of Iowa
Lauren A. Gonzales - University of North Texas Health Science Center
Resource Type: Journal article
Publication Details: Anatomical record (Hoboken, N.J. : 2007), Vol.307(3), pp.592-599
DOI: 10.1002/ar.25298
PMID: 37515586
NLM abbreviation: Anat Rec (Hoboken)
ISSN: 1932-8486
eISSN: 1932-8494
Grant note: DOI: 10.13039/100017787, name: Health Science Center, University of North Texas; DOI: 10.13039/100006733, name: Indiana University; DOI: 10.13039/100007114, name: Ralph W. and Grace M. Showalter Research Trust Fund
Language: English
Electronic publication date: 07/29/2023
Date published: 03/2024
Academic Unit: Anatomy and Cell Biology
Record Identifier: 9984455612902771

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