Document Detail


Trigonocephaly in rabbits with familial interfrontal suture synostosis: the multiple effects of premature single-suture fusion.
MedLine Citation:
PMID:  11066035     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Previous studies from our laboratory have characterized the craniofacial morphology and growth patterns of an inbred strain of rabbits with autosomal dominant coronal suture synostosis. A number of rabbit perinates from this colony have been collected sporadically over a 5-year period with premature interfrontal suture synostosis. The present study describes the very early onset of craniofacial dysmorphology of these rabbits and compares them to similar-aged normal control rabbits. A total of 40 perinatal New Zealand White rabbits were used in the present study. Twenty-one comprised the sample with interfrontal suture synostosis and ranged in age from 27 to 38 days postconception (term = 31 days) with a mean age of 33.53 days (+/-2.84 days). Nineteen rabbits served as age-matched, normal controls (mean age = 33.05 days +/-2.79 days). Lateral and dorsoventral radiographs were collected from each rabbit. The radiographs were traced, computer digitized, and 12 craniofacial measurements, angles, and indices were obtained. Mean measures were compared using an unpaired Student's t-test. All synostosed rabbits were stillborn or died shortly after birth. Grossly, these rabbits exhibited extreme frontal bossing, trigonocephaly with sagittal keeling, and midfacial shortening. No somatic anomalies were noted. Radiographically, rabbits with interfrontal suture synostosis had significantly (P < 0.05) narrower bifrontal widths, shorter cranial vault lengths, kyphotic cranial base angles, and different cranial vault indices (shapes) compared to controls. Results reveal severe and early pathological and compensatory cranial vault changes associated with premature interfrontal suture synostosis in this rabbit model. The 100% mortality rate noted in this condition may be related to the inheritance of a lethal genetic mutation or to neural compression from reduced intracranial volume. Results are discussed in light of current pathogenic hypotheses for human infants with premature metopic suture synostosis.
Authors:
M P Mooney; G M Cooper; A M Burrows; W Wigginton; T D Smith; J Dechant; R Mitchell; H W Losken; M I Siegel
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  The Anatomical record     Volume:  260     ISSN:  0003-276X     ISO Abbreviation:  Anat. Rec.     Publication Date:  2000 Nov 
Date Detail:
Created Date:  2000-11-20     Completed Date:  2000-11-30     Revised Date:  2007-11-14    
Medline Journal Info:
Nlm Unique ID:  0370540     Medline TA:  Anat Rec     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  238-51     Citation Subset:  IM    
Copyright Information:
Copyright 2000 Wiley-Liss, Inc.
Affiliation:
Department of Oral Medicine and Pathology, University of Pittsburgh, Pittsburgh, Pennsylvania 15261, USA. mpm4@pitt.edu
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MeSH Terms
Descriptor/Qualifier:
Animals
Animals, Inbred Strains
Animals, Newborn
Cephalometry
Cranial Sutures / abnormalities*,  radiography
Craniofacial Abnormalities / genetics,  pathology*,  radiography
Frontal Bone / abnormalities*,  pathology,  radiography
Rabbits / growth & development*
Synostosis / genetics,  pathology*,  radiography
Tomography, X-Ray Computed
Grant Support
ID/Acronym/Agency:
AR44172/AR/NIAMS NIH HHS; DE01830/DE/NIDCR NIH HHS

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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