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Titolo:
A RABBIT MODEL OF HUMAN FAMILIAL, NONSYNDROMIC UNICORONAL SUTURE SYNOSTOSIS - I - SYNOSTOTIC ONSET, PATHOLOGY, AND SUTURAL GROWTH-PATTERNS
Autore:
MOONEY MP; SIEGEL MI; BURROWS AM; SMITH TD; LOSKEN HW; DECHANT J; COOPER G; KAPUCU MR;
Indirizzi:
UNIV PITTSBURGH,DEPT ANAT & HISTOL,619 SALK HALL PITTSBURGH PA 15261 UNIV PITTSBURGH,DEPT ANTHROPOL PITTSBURGH PA 15261 UNIV PITTSBURGH,DEPT PLAST & RECONSTRUCT SURG PITTSBURGH PA 15261 UNIV PITTSBURGH,DEPT ORTHODONT PITTSBURGH PA 15261 UNIV PITTSBURGH,CLEFT PALATE CRANIOFACIAL CTR PITTSBURGH PA 15261 SLIPPERY ROCK UNIV,DEPT PHYS THERAPY SLIPPERY ROCK PA 00000 CHILDRENS HOSP,DEPT PEDIAT PLAST SURG PITTSBURGH PA 15213
Titolo Testata:
Child's nervous system
fascicolo: 6, volume: 14, anno: 1998,
pagine: 236 - 246
SICI:
0256-7040(1998)14:6<236:ARMOHF>2.0.ZU;2-Z
Fonte:
ISI
Lingua:
ENG
Soggetto:
UNILATERAL CORONAL SYNOSTOSIS; CRANIAL SYNOSTOSIS; CRANIOSYNOSTOSIS; INSIGHTS; BIOLOGY; SKULL; PROGRESSION; DEFECTS; CLOSURE; PALATE;
Keywords:
PLAGIOCEPHALY; ANIMAL MODEL; CRANIOSYNOSTOSIS; RABBITS; GROWTH; MORPHOLOGY;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Science Citation Index Expanded
Science Citation Index Expanded
Science Citation Index Expanded
Citazioni:
92
Recensione:
Indirizzi per estratti:
Citazione:
M.P. Mooney et al., "A RABBIT MODEL OF HUMAN FAMILIAL, NONSYNDROMIC UNICORONAL SUTURE SYNOSTOSIS - I - SYNOSTOTIC ONSET, PATHOLOGY, AND SUTURAL GROWTH-PATTERNS", Child's nervous system, 14(6), 1998, pp. 236-246

Abstract

Poswillo has stated, ''The more severe anomalies of the calvaria, such as plagiocephaly, Crouzon [syndrome], and Apert syndrome still defy explanation, in the absence of an appropriate animal system to study''(p. 207). This two-part study reviews data from a recently developed colony of New Zealand white rabbits with familial, nonsyndromic unilateral coronal suture synostosis. Part 1 presents pathological findings and compensatory sutural growth data from 109 normal rabbits and 82 craniosynostotic rabbits from this colony. Synostotic foci, onset, and progression were described in the calvariae from 102 staged (fetal days21, 25, 27, 33; term = 30 days) fetuses (39 normal, 63 synostosed). Calvarial suture growth patterns from 10 to 126 days of age were assessed from serial radiographs obtained from 89 rabbits (70 normal rabbitsand 19 rabbits with unicoronal suture synostosis) with amalgam bone marker implants. Perinatal results revealed that by fetal day 25 the synostotic focal point in synostotic rabbits consistently originated from the endocortical surface of the calvaria in the middle of the coronal suture at a presumed high-tension, interdigitating zone. Histological analysis revealed hyperostotic osteogenic fronts on the affected side compared with the unaffected side. Postnatal sutural growth data revealed a predictable pattern of plagiocephaly (contralateral coronal sutures growing more than ipsilateral sutures and ipsilateral frontonasal and anterior lambdoidal sutures growing more than contralateral sutures), which resulted in early cranial vault deformities and a double ''S'' shape torquing towards the affected side. The advantages and disadvantages of these rabbits as a model for human familial, nonsyndromicunicoronal suture synostosis are discussed, especially in light of recent cytokine and genetic findings from human craniosynostotic studies.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 07/08/20 alle ore 06:49:30