Mp. Mooney et al., DEVELOPMENT OF A STRAIN OF RABBITS WITH CONGENITAL SIMPLE NONSYNDROMIC CORONAL SUTURE SYNOSTOSIS .2. SOMATIC AND CRANIOFACIAL GROWTH-PATTERNS, The Cleft palate-craniofacial journal, 31(1), 1994, pp. 8-16
In the March 1993 issue of The Cleft Palate-Craniofacial Journal we re
ported a female rabbit born in our laboratory with complete bilateral
coronal suture (CS) synostosis. This follow-up study presents our atte
mpts to breed the animal and establish a strain of craniosynostotic ra
bbits. The second part of this study presents longitudinal somatic and
craniofacial growth data in offspring with coronal suture synostosis.
Serial growth data from 72 animals were collected for the present stu
dy. The sample consisted of 11 animals (10 offspring and the original
female) with complete nonsyndromic unilateral (plagiocephalic) or bila
teral (brachycephalic) CS synostosis, 19 animals with partial CS synos
tosis, and 42 unaffected control litter mates. At 10 days of age, all
animals had radiopaque amalgam markers placed on either side of the fr
ontonasal, coronal, anterior lambdoidal, and sagittal sutures. Body we
ights and serial lateral and dorsoventral head radiographs were taken
at 1.5 (10 days), 6, 12, and 18 weeks of age. All animals showed simil
ar body weights at 1.5 weeks of age, while completely synostosed anima
ls exhibited a slight (about 12%), but significantly (p < .001) lowere
d body weight by 18 weeks of age. Results revealed that by 1.5 weeks o
f age the completely synostosed animals already exhibited brachycephal
ic cranial vaults, mid-facial hypoplasia, and increased flattening of
the cranial base compared to unaffected siblings. This pattern continu
ed through 18 weeks of age, with the partially synostosed animals exhi
biting intermediate morphologies. Compensatory overgrowths were noted
primarily at the sagittal and frontonasal sutures, especially for comp
letely synostosed animals. Findings revealed that the craniosynostotic
rabbits followed predictable compensatory craniofacial growth pattern
s and exhibited secondary deformities similar to those reported for ca
ses of human coronal suture synostosis. Such findings support continue
d efforts at developing this model to help understand, in part, the et
iopathogenesis of this condition in human populations.