Neurocranial growth in the OIM mouse model of osteogenesis imperfecta

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

doi:10.1002/ar.25307

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Neurocranial growth in the OIM mouse model of osteogenesis imperfecta

Journal article

Open access

Peer reviewed

Neurocranial growth in the OIM mouse model of osteogenesis imperfecta

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

Anatomical record (Hoboken, N.J. : 2007), Vol.307(3), pp.581-591

03/2024

DOI: 10.1002/ar.25307

PMID: 37638403

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Abstract

Abstract Osteogenesis imperfecta (OI) is a disorder of type I collagen characterized by abnormal bone formation. The OI craniofacial phenotype includes midfacial underdevelopment, as well as neurocranial changes (e.g., macrocephaly and platybasia) that may also affect underlying nervous tissues. This study aims to better understand how OI affects the integrated development of the neurocranium and the brain. Juvenile and adult mice with OI (OIM) and unaffected wild type (WT) littermates were imaged using in vivo micro‐computed tomography (microCT). Virtual endocast models were used to measure brain volume, and 3D landmarks were collected from the cranium and brain endocasts. Geometric morphometric analyses were used to compare brain shape and integration between the genotypes. OIM mice had increased brain volumes (relative to cranial centroid size) only at the juvenile stage. No significant difference was seen in cranial base angle (CBA) between OIM and WT mice. However, CBA was higher in juvenile than in adult OIM mice. Brain shape was significantly different between OIM and WT mice at both stages, with OIM mice having more globular brains than WT mice. Neurocranial and brain morphology were strongly integrated within both genotypes, while adult OIM mice tended to have lower levels of skull‐brain integration than WT mice. These results suggest that neurocranial dysmorphologies in OI may be more severe at earlier stages of postnatal development. Decreased skull‐brain integration in adult mice suggests that compensatory mechanisms may exist during postnatal growth to maintain neurological function despite significant changes in neurocranial morphology.

Details

Title: Subtitle: Neurocranial growth in the OIM mouse model of osteogenesis imperfecta
Creators: 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
Lila A. Huston - University of North Texas Health Science Center
Courtney A. Miller - Department of Physiology and Anatomy University of North Texas Health Science Center Fort Worth Texas USA
Ashley T. Steele - University of North Texas Health Science Center
Lauren A. Gonzales - University of North Texas Health Science Center
Emma K. Handler - University of Iowa
Jason M. Organ - Indiana University – Purdue University Indianapolis
Rachel A. Menegaz - University of North Texas Health Science Center
Resource Type: Journal article
Publication Details: Anatomical record (Hoboken, N.J. : 2007), Vol.307(3), pp.581-591
DOI: 10.1002/ar.25307
PMID: 37638403
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: 08/28/2023
Date published: 03/2024
Academic Unit: Anatomy and Cell Biology
Record Identifier: 9984459413302771

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