Morphological variability in the inner ear of mice with osteogenesis imperfecta.

Autor: Huston LA; Department of Physiology and Anatomy, University of North Texas Health Science Center, Fort Worth, Texas, USA.; University of the Incarnate Word School of Medicine, San Antonio, Texas, USA., Husain TS; Department of Physiology and Anatomy, University of North Texas Health Science Center, Fort Worth, Texas, USA.; Arkansas College of Osteopathic Medicine, Chad, Arkansas, USA., Moore JC; 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., Organ JM; Department of Anatomy, Cell Biology & Physiology, Indiana University School of Medicine, Indianapolis, Indiana, USA., Menegaz RA; Department of Physiology and Anatomy, University of North Texas Health Science Center, Fort Worth, Texas, USA., Handler EK; Department of Anatomy and Cell Biology, University of Iowa, Iowa City, Iowa, USA., Gonzales LA; Department of Physiology and Anatomy, University of North Texas Health Science Center, Fort Worth, Texas, USA.
Jazyk: angličtina
Zdroj: Anatomical record (Hoboken, N.J. : 2007) [Anat Rec (Hoboken)] 2024 Mar; Vol. 307 (3), pp. 592-599. Date of Electronic Publication: 2023 Jul 29.
DOI: 10.1002/ar.25298
Abstrakt: 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.
(© 2023 The Authors. The Anatomical Record published by Wiley Periodicals LLC on behalf of American Association for Anatomy.)
Databáze: MEDLINE
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