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PubMed1994

The effect of dam's strain on the intrauterine craniofacial growth of mouse fetuses.

PURPOSE: The maternal effect is one of the important factors in mammalian growth in conjunction with the genetic effect. The present study investigated the prenatal maternal effect of a dam on the intrauterine craniofacial growth of a mouse fetus using embryo transfer and cephalometry. DDD/Qdj strain mouse embryos were transferred to four strains of recipient female mice (DDD/Qdj, C3H/Qdj, C57BL/Qdj, and DBA/1J Sea). Just after parturition cephalometric observation of the newborn offspring, which developed in the uteri of the four strains of dams, was performed and then the craniofacial size of the newborn offspring was calculated on the lateral cephalogram. Statistical analyses were performed to examine the correlation between the dam's weight and the craniofacial size of the newborn offspring, to test the significance of the effect of the dam's strain on the craniofacial size of the newborn offspring, and to evaluate the interstrain difference of the intrauterine craniofacial growth of the mouse fetuses. RESULTS: It was disclosed that there were a direct relation between the dam's weight and the craniofacial size of the newborn offspring, a significant effect of the dam's strain on the craniofacial size of the newborn offspring, and a significant interstrain difference in the craniofacial size of the newborn offspring after eliminating the effects of litter size and gestation period on the craniofacial size of the newborn offspring (DDD/Qdj > C3H/Qdj = C57BL/Qdj > DBA/1J Sea). CONCLUSION: Thus, it could be concluded that the four strains of dams affected differently the intrauterine craniofacial growth of the DDD/Qdj strain fetuses through each uterine condition, indicating that the dam's weight played an important role as one of the prenatal maternal effects on the intrauterine craniofacial growth of mouse fetuses.

Animals↗
PubMed2002

Analysis of craniofacial development in children with hypohidrotic ectodermal dysplasia.

Ectodermal dysplasias (ED) are a heterogeneous group of inheritable disorders characterized by abnormal development of embryologic ectoderm derivatives. The purposes of this study were to: 1) create baseline cephalometric norms for male children with ED; 2) assess craniofacial growth and development in hypohidrotic ED male children with severe hypodontia, compared with non-ED children with class I dental relationships; 3) compare the craniofacial morphology of titanium dental implant-treated ED males with non-implant-treated ED males; and 4) correlate the severity of hypodontia to craniofacial dysmorphology. Cephalometric radiographs of class I individuals and implant-treated and nontreated ED groups were used to evaluate craniofacial morphology. Traditional cephalometric landmarks and measurements were used to compare groups using the generalized estimate equation analysis. Age, gender, and the number of permanent maxillary teeth present had a significant (P =.01) explanatory relationship with the craniofacial measures when comparing untreated ED children to norms. Mean craniofacial differences between ED and non-ED children still existed when the explanatory effects of these variables were controlled, indicating dysmorphology in several craniofacial structures (e.g., cranial base, mandibular length). The number of missing maxillary permanent teeth was significantly related with craniofacial dysmorphology in the ED population. Craniofacial morphology did not differ significantly between implant-treated and nontreated ED children, suggesting that treatment with intraosseous dental implants, as applied in this population, did not rescue normal craniofacial growth and development.

Adolescent↗
PubMed2005

Community orthodontists and craniofacial care: results of a Washington state survey.

BACKGROUND: Little is known about community orthodontists' previous training in, experience with, or receptivity to caring for children with craniofacial disorders. OBJECTIVES: (1) To characterize the current level of participation by Washington state orthodontists in craniofacial care; and (2) to identify factors that promote or impede community orthodontists' involvement in caring for children with craniofacial conditions. DESIGN: Mail survey. METHODS: A 26-item questionnaire was designed and mailed to all active orthodontists in Washington state (N = 230). Question topics included practice characteristics, training and experience with craniofacial conditions, concerns related to public and private insurance, and communication with craniofacial teams. RESULTS: Of eligible respondents, 68% completed the survey. Most orthodontists' patient panels were made up of patients who either have private insurance or pay cash for services. On average, 2% of respondents' patients were Medicaid beneficiaries. Only 20% of respondents had seen more than three patients with cleft lip and/or palate in the past 3 years. Although a minority of orthodontists receive referrals from (27%) or are affiliated with (11%) craniofacial teams, most orthodontists perceived craniofacial care positively and were interested to learn more about craniofacial care and to accept additional patients with these conditions. CONCLUSIONS: Results of this survey can inform potential strategies to increase access to orthodontic care for children with craniofacial disorders. These would include developing an organized training, referral, and communication system between community orthodontists and state craniofacial teams and considering a case-management approach to facilitate this process.

Attitude of Health Personnel↗
PubMed2004

A survey of dentists in the United States regarding a specialty in craniofacial pain.

In an effort to explore whether a specialty for craniofacial pain is warranted, the American Academy of Craniofacial Pain (AACP) commissioned an opinion survey of dentists. The survey population (N=4000) was stratified by specialty, so that dentists in affected areas would be adequately represented: 500 orthodontists and dentofacial orthopedists, 500 oral and maxillofacial surgeons, 500 periodontists, 500 prosthodontists, and 2,000 general practitioners. A total of 930 dentists responded for a 23.2% response rate. The survey had multiple purposes: 1. to measure the percentage of craniofacial pain patients perceived in dental patient populations; 2. to determine whether each dentist prefers to treat the disorder or; 3. prefers to refer craniofacial pain patients to clinicians specializing in the disorder; and 4. whether dentists favor/oppose the formation of a craniofacial pain specialty. The respondents' perception of the prevalence of craniofacial pain among their patients was 13.9%. A majority of the responding dentists, 54.7%, are in favor of a craniofacial pain specialty. Overall, 65% of dentists treat craniofacial pain patients, although more than half, 55%, of all dentists also refer such patients. Even 43.6% of dentists who regularly treat craniofacial pain favor a specialty, while 76% of those who do not treat such patients favor the specialty. The data presented here advocate development of a dental specialty in craniofacial pain.

Attitude of Health Personnel↗
PubMed2004

Parental craniofacial morphology in orofacial clefting.

The parental craniofacial morphology in orofacial clefting (OFC) has been shown to differ from that of the non-cleft population when evaluated using conventional cephalometric analyses comprising a variety of linear, angular, and area measurements. In spite of this, the shape of the parental craniofacial complex is of greater importance in the search for the morphogenes involved in OFC. This retrospective case-control study employed three morphometric techniques [discriminant analysis of the principal components of shape (PCS), Euclidean distance matrix analysis (EDMA), and thin-plate spline analysis (TPS)] to localize the craniofacial skeletal shape differences between (a) the parents of children with OFC and a comparison group, (b) the parents of children with cleft lip and palate [CL(P)] and cleft palate (CP), and (c) the male and female parents of children with OFC. The postero-anterior (PA) cephalograms of 92 parents of children with non-syndromic OFC and 43 comparison group volunteers were scanned and digitized. The configurations of 24 reproducible landmarks were optimally superimposed using Procrustes algorithms to allow shape data to be derived using PCS, EDMA, and TPS. The parental craniofacial shape statistically significantly differed from that of the comparison group using PCS (P < 0.001) and EDMA (P = 0.001). However PCS, EDMA, and TPS differed in their localization of the shape differences, explainable by the different mathematical methods used by the individual techniques. Interestingly, the parental craniofacial shapes in CL(P) and CP were morphologically similar when tested using PCS (P = 0.03) and EDMA (P = 0.027). However, there was no shape-related sexual dimorphism in parental craniofacial morphology in OFC when tested using PCS (P = 0.35) and EDMA (P = 0.525). Thus, the parental craniofacial shape in OFC differs from the non-cleft population, the parental craniofacial shape does not differ between CL(P) and CP and there is no sexual dimorphism in the parental craniofacial morphology in OFC, as viewed on PA cephalograms.

Algorithms↗
PubMed2000

Endoscopic intracranial craniofacial and monobloc osteotomies with the aid of a malleable high-speed pneumatic drill: a cadaveric and clinical study.

Endoscopic techniques are now an accepted part of the surgical armamentarium and are used routinely in a number of aesthetic and reconstructive procedures. Endoscopic techniques are now being used commonly by plastic surgeons in forehead and face lifts. In both craniofacial surgery and in neurosurgery, the application of endoscopy potentially allows the surgical team to perform wide dissection of the dura mater in a minimally invasive fashion, thereby potentially reducing the risk of dural and brain injury. Also reduced by this surgical approach is potential injury to the major venous structures, such as the sagittal sinus, along with overall reduced bleeding. After an extensive laboratory study of 10 cadaveric dissections, the authors have refined a new endoscopic technique for completing an endoscopic intracranial craniofacial osteotomy. This study was conducted in the Department of Pathology at the University of Brno (Czech Republic), and was performed as a cooperative multicenter project between the University of Palermo, the Albert Einstein College of Medicine/ Montefiore Medical Center, the Hospital Infantil de Mexico, "Federico Gomez," and the Medtronic Midas Rex Institute, (Fort Worth, TX). During this cadaveric anatomic study and using small trephinations and skin incisions the authors were able to develop several different craniofacial and endoscopic monobloc procedures. To accomplish intracranial and facial osteotomies, a new malleable high-speed drill was designed for use in the endoscopic craniofacial approach. Using these newly developed cadaveric techniques and instrumentation, the authors performed two intracranial craniofacial procedures on children with congenital craniofacial anomalies. There would appear to be several significant advantages for the craniofacial patient as result of these new techniques: reduced surgical trauma, operative bleeding, surgical time, and hospitalization, along with a reduced risk of infection. It became quickly apparent, as a result of these cadaveric studies, that the learning curve for this endoscopic procedure is quite steep. Tutino M, Chico F, Tutino M, Goodrich JT, Ortiz Monasterio F. Endoscopic intracranial craniofacial and monobloc osteotomies with the aid of a malleable high-speed pneumatic drill: a cadaveric and clinical study.

Aged↗
PubMed2006

A zebrafish screen for craniofacial mutants identifies wdr68 as a highly conserved gene required for endothelin-1 expression.

BACKGROUND: Craniofacial birth defects result from defects in cranial neural crest (NC) patterning and morphogenesis. The vertebrate craniofacial skeleton is derived from cranial NC cells and the patterning of these cells occurs within the pharyngeal arches. Substantial efforts have led to the identification of several genes required for craniofacial skeletal development such as the endothelin-1 (edn1) signaling pathway that is required for lower jaw formation. However, many essential genes required for craniofacial development remain to be identified. RESULTS: Through screening a collection of insertional zebrafish mutants containing approximately 25% of the genes essential for embryonic development, we present the identification of 15 essential genes that are required for craniofacial development. We identified 3 genes required for hyomandibular development. We also identified zebrafish models for Campomelic Dysplasia and Ehlers-Danlos syndrome. To further demonstrate the utility of this method, we include a characterization of the wdr68 gene. We show that wdr68 acts upstream of the edn1 pathway and is also required for formation of the upper jaw equivalent, the palatoquadrate. We also present evidence that the level of wdr68 activity required for edn1 pathway function differs between the 1st and 2nd arches. Wdr68 interacts with two minibrain-related kinases, Dyrk1a and Dyrk1b, required for embryonic growth and myotube differentiation, respectively. We show that a GFP-Wdr68 fusion protein localizes to the nucleus with Dyrk1a in contrast to an engineered loss of function mutation Wdr68-T284F that no longer accumulated in the cell nucleus and failed to rescue wdr68 mutant animals. Wdr68 homologs appear to exist in all eukaryotic genomes. Notably, we found that the Drosophila wdr68 homolog CG14614 could substitute for the vertebrate wdr68 gene even though insects lack the NC cell lineage. CONCLUSION: This work represents a systematic identification of approximately 25% of the essential genes required for craniofacial development. The identification of zebrafish models for two human disease syndromes indicates that homologs to the other genes are likely to also be relevant for human craniofacial development. The initial characterization of wdr68 suggests an important role in craniofacial development for the highly conserved Wdr68-Dyrk1 protein complexes.

Animals↗
PubMed2003

Craniofacial anthropometric analysis in Down's syndrome patients.

Past investigations of Down's syndrome (DS) have indicated that there are marked abnormalities in the craniofacial morphology. The aim of this study was to establish the craniofacial anthropometric variables which discriminate DS group from healthy population and also to observe the changes occurring with growth. Using noninvasive method of craniofacial anthropometry, craniofacial pattern profile (CFPP) analysis (from twenty-five anthropometric measurements per person) was performed in 104 DS individuals and 365 healthy controls, aged seven to fifty-seven and divided into four age ranges. Z-scores were calculated for each variable and the variations in the craniofacial region have been identified by multivariate discriminative analysis. The results showed that three variables (head length (g-op), head circumference (OFC) and outer canthal distance (ex-ex) were responsible for 85.68% variability (p < 0.001). The analysis of z-scores showed that the majority of variables were in subnormal (under -2 SD) and normal range (from -2SD to +2SD), but none of them was in the supernormal range (over the +2SD). Some craniofacial characteristics are age-related. On the basis of craniofacial anthropometric traits it was possible to separate even 91.35% of DS patients from the healthy population. It could be concluded that these findings demonstrate the usefulness of application of CFPP in defining abnormal craniofacial dimensions in DS individuals.

Adolescent↗
PubMed1994

A comprehensive analysis of craniofacial trauma.

A review of the literature identified a need for a prospective study of the complete range of craniofacial trauma. The aims of this study were to determine the incidence, etiology, and mechanisms of craniofacial and associated injuries, enabling a greater understanding of their range and magnitude. Nine hundred fifty consecutive patients seen at an urban university hospital with any degree of craniofacial trauma were prospectively investigated. Craniofacial trauma was found to be very common at all ages. The causes were directly related to age, sex, and alcohol consumption, and determine the type and severity of injury. The commonest cause of soft-tissue injury was falls, whereas that of fractures was interpersonal violence. Falls accounted for most of the injuries in children and the elderly, whereas interpersonal violence was mainly responsible for those occurring in patients aged 15 to 50 years. Interpersonal violence mostly involved young male adults: fights occurring mainly between strangers who had consumed excessive amounts of alcohol. Women were usually assaulted by assailants known to them, their partners. Pedestrians showed a propensity to sustain cranial fractures, whereas motor vehicle occupants tended to sustain midfacial fractures and bicyclists mandibular fractures. Pedestrians incurred the severest injuries of all road users, and a significant proportion of road user collisions involved bicyclists. Sports were responsible for a significant proportion of craniofacial injuries in youths and young adults. Craniofacial soft-tissue injuries overall occurred most frequently on the forehead, nose, lips, and chin, and a method for their classification is proposed. The commonest craniofacial fracture was that of the nasal bones (45%), followed by cranial bones (24%), mandible (13%), zygoma (13%), orbital blow-out (3%), and maxilla (2%). The incidence of craniofacial trauma can be greatly reduced by improvements in interior home design, school education in alcohol abuse and handling potentially hostile situations (especially for men), improvement in automotive safety devices and compliance by motor vehicle occupants, and utilization of full-face helmets by bicyclists and motorcyclists.

Accidental Falls↗
PubMed1997

The role of the cranial base in facial growth: experimental craniofacial synostosis in the rabbit.

Craniofacial synostosis designates premature fusion in sutures of the cranial vault (calvarium). When craniofacial synostosis is associated with a syndrome (e.g., Apert, Crouzon), premature fusion of the cranial base has been postulated to occur as well. This study was designed to determine whether the primary growth disturbance in craniofacial synostosis is located at the cranial base (i.e., spheno-occipital synchondrosis) or the calvarial vault (i.e., coronal and sagittal sutures) or both. Sixty newborn New Zealand White rabbits were randomly assigned to six groups: (I) calvarial control, (II) cranial base control, (III) cranial base immobilization, (IV) coronal suture immobilization, (V) coronal and sagittal suture immobilization, and (VI) cranial base and coronal and sagittal suture immobilization. An anterior cervical microsurgical approach to the cranial base was used, while cranial vault sutures were exposed through a bicoronal scalp incision. All sutures were fused by periosteal abrasion and application of methyl cyanoacrylate. Cephalograms were taken at 30, 60, and 90 days postoperatively to assess craniofacial growth. Linear and angular measurements of facial, calvarial, and basicranial growth were subjected to multivariate analysis. Analysis indicated that (1) craniofacial length was shortened most significantly by cranial base fusion, (2) cranial base fusion and cranial vault fusion had an additive effect on craniofacial length restriction, (3) the anterior cranial base was significantly shortened by cranial base and cranial vault fusion (p < 0.05), (4) the posterior cranial base was shortened by cranial base fusion only (p < 0.05), and (5) cranial base fusion alone significantly flattened the cranial base angle (p < 0.05), whereas cranial vault fusion alone did not. These results suggest that cranial base fusion alone may account for many dysmorphic features seen in craniofacial synostosis. This model is consistent with the findings of other investigators and confirms both a primary directive and translational role of the cranial base in craniofacial growth.

Animals↗
PubMed2005

Engineering craniofacial scaffolds.

OBJECTIVE: To develop an integrated approach for engineering craniofacial scaffolds and to demonstrate that these engineered scaffolds would have mechanical properties in the range of craniofacial tissue and support bone regeneration for craniofacial reconstruction. EXPERIMENTAL VARIABLE: Scaffold architecture designed to achieve desired elasticity and permeability. Scaffold external shape designed to match craniofacial anatomy. OUTCOME MEASURE: Final fabricated biomaterial scaffolds. Compressive mechanical modulus and strength. Bone regeneration as measured by micro-CT scanning, mechanical testing and histology. SETTING: Departments of Biomedical Engineering, Oral/Maxillofacial Surgery, and Oral Medicine, Pathology and Oncology at the University of Michigan. RESULTS: Results showed that the design/fabrication approach could create scaffolds with designed porous architecture to match craniofacial anatomy. These scaffolds could be fabricated from a wide range of biomaterials, including titanium, degradable polymers, and degradable calcium phosphate ceramics. Mechanical tests showed that fabricated scaffolds had compressive modulus ranging 50 to 2900 MPa and compressive strength ranging from 2 to over 56 MPa, within the range of human craniofacial trabecular bone. In vivo testing of designed scaffolds showed that they could support bone regeneration via delivery of BMP-7 transduced human gingival fibroblasts in a mouse model. Designed hydroxyapatite scaffolds with pore diameters ranging from 400 to 1200 microns were implanted in minipig mandibular defects for 6 and 18 weeks. Results showed substantial bone ingrowth (between 40 and 50% at 6 weeks, between 70 and 80% at 18 weeks) for all scaffolds, with no significant difference based on pore diameter. CONCLUSION: Integrated image-based design and solid free-form fabrication can create scaffolds that attain desired elasticity and permeability while fitting any 3D craniofacial defect. The scaffolds could be manufactured from degradable polymers, calcium phosphate ceramics and titanium. The designed scaffolds supported significant bone regeneration for all pore sizes ranging from 300 to 1200 microns. These results suggest that designed scaffolds are clinically applicable for complex craniofacial reconstruction.

Animals↗
PubMed1995

The relationship between obesity and craniofacial structure in obstructive sleep apnea.

STUDY OBJECTIVE: To evaluate the interaction between craniofacial structure and obesity in male patients with obstructive sleep apnea (OSA). DESIGN: Retrospective analysis of a cohort of OSA patients. The relationships between neck circumference (NC), body mass index, apnea severity, and craniofacial and upper airway soft-tissue measurements from upright lateral cephalometry were examined. Patients were divided into groups; small to normal NC (group A), intermediate NC (group B), or large NC (group C). SETTING: A university teaching hospital and tertiary sleep referral center. PATIENTS: A consecutive series of patients with OSA who underwent polysomnography and lateral cephalometry. MEASUREMENTS AND RESULTS: Group A patients were less obese and had more craniofacial abnormalities such as a smaller mandible and maxilla and a more retrognathic mandible. Group B patients had both upper airway soft-tissue and craniofacial abnormalities. Group C patients were more obese with larger tongues and soft palates, and an inferiorly placed hyoid. Group C patients also had fewer craniofacial abnormalities than group A or B patients. There was no difference in airway size among the three groups. CONCLUSIONS: We conclude that there is a spectrum of upper airway soft-tissue and craniofacial abnormalities among OSA patients: obese patients with increased upper airway soft-tissue structures, nonobese patients with abnormal craniofacial structure, and an intermediate group of patients with abnormalities in both craniofacial structure and upper airway soft-tissue structures.

Adult↗