bluuebunny/crossref_metadata_2025 huggingface dataset:
Craniofacial Growth after latrogenic Cleft Palate Repair in a Fetal Ovine Model
Authors: Canady John W. , Thompson Sue Ann , Colburn Alex
Published: October 2018

Controversy exists over the impact of scar formation on craniofacial growth after cleft palate repair. The fetal ovine model presents an opportunity to study a group of animals with little or no scar using cephalometric studies of craniofacial growth after iatrogenic cleft palate repair. Grossly evident scar is formed in the palates of lambs repaired at 118 days or later in gestation, while those animals operated at 70 and 77 days' gestation exhibited no scar grossly and minimal scar histologically in the submucosa with normal nasal and oral mucosal surfaces. For this study, 15 lambs were studied: four were unoperated, three were operated at 70 days, one at 77 days, and seven had clefts produced and repaired at 118 to 133 days' gestation. The animals were euthanized at 1 month of age and the heads removed and frozen until analyzed. Computerized tomography of the heads was used to create voxel (volume pixel) data sets, and volume rendering and measurement software (Voxblast) was used to create a three-dimensional reconstruction of the skull. Palate measurements were obtained by selecting points on the upper deciduous premolars. A plane was set through the palate and upper deciduous premolars using standard points on the skull to maintain consistent visualization and point selection. The measurements were normalized to skull size measured by the distance between points on the right and left zygomatic bones. Using one-way analysis of variance, followed by a protected t test, no significant differences were found between the means of any measurement In the three treatment groups. Fetal palate repair, with or without scarring, resulted in normal craniofacial growth in the 1-month-old lamb.
A Peculiar Malformation of the Upper Lip: Report of Six Cases
Authors: Tange Ichiro
Published: October 2018

Six cases of a peculiar type of upper tip malformation have been observed by the author during the last 40 years. The malformations in this case series were remarkably similar, suggesting a single teratologic entity not been reported before. The six patients included 1-, 11-, 4-, and 6-month-old males, a 20 year-old male, and an 11-year-old female. Each was characterized by a crooked folded philtrum, a flat thin vermilion region of the upper lip, and high-positioned ala(e), that was unilateral or bilateral. The upper median incisors were separated occasionally by the thick lip frenulum without marked distortion of the nasomaxillary framework. The author speculates that these cases exhibited some disturbed penetration of the lateral nasal eminence (process) to the median part of the nasolabial region in their early embryologic development.
Dental Development in Apert Syndrome
Authors: Kaloust Scott , Ishii Kazuhiro , Vargervik Karin
Published: October 2018

Objective

Apert syndrome has been extensively studied and described. However, an area that has not been studied is the dental development of these Individuals. The purpose of this study was to evaluate the development of the dentition and compare it with that of unaffected children. There appears to be clinical observations Indicating delayed eruption of the permanent teeth in the Apert child.

Methods

This retrospective study examined all Apert syndrome patients from four craniofacial centers who had a panoramic radiograph taken before the age of 16 years. Thirty-six individuals, 19 boys and 17 girls ranging In age from 4.1 to 15.8 (mean = 9.3) years were examined. The seven left mandibular permanent teeth, second molar to central incisor, were rated on an eight-stage scale (A–H) using methods described by Demirjian and Goldstein (1976). The stage of each tooth was converted to the corresponding numeric value, and then all seven values were added to obtain a dental maturity score, which corresponded to a dental age, based on the sample of 4500+ normal children of the Demirjian et al. study. The dental age and chronologic age were compared using a paired t test. The correlation between chronologic age and length of delay was also determined.

Results

Thirty-one of 36 individuals had a dental age lower than their chronologic age. Compared to the normal sample, the mean developmental dental delay was 0.96 years (p < .001). The range in delay was 0.5 years advanced to 2.9 years delayed. There was a positive correlation to increased age with greater delay in dental development (p < .05).

Conclusion

Our study confirmed our clinical impressions: The results showed a mean developmental delay of 0.96 years, with a trend of Increasing delay with increased age. This positive correlation found between increased age and increased delay parallels the general growth of Apert children, with a greater degree of delay as the child grows older.
Residual Clefts in the Hard Palate: Correlation between Cleft Size and Speech
Authors: Lohmander-Agerskov Anette , Friede Hans , Söderpalm Ewa , Lilja Jan
Published: October 2018

Objective

This study was conducted to evaluate the relationship between size of residual clefts in the hard palate and speech.

Subjects

Fifteen 7-year-old children born with complete cleft lip and palate were investigated.

Methods

All of the children were treated according to a surgical regimen involving early soft palate repair and delayed hard palate closure. Measures were taken of the area, length, and maximal width of the residual cleft in the hard palate about a year before Its closure and correlated with a perceptual judgment of several speech variables.

Results

Significant positive correlations were obtained between the size of the cleft and two variables: weak pressure consonants and hypernasality. Nasal escape was very common among the patients, and almost half the children had retracted palatal or velar articulation of dental stop consonants. Neither of these two variables correlated with the size of the residual cleft.

Conclusion

Perceived oral pressure and, perhaps, resonance seem to be related to size of the opening of the residual cleft, whereas audible nasal escape and articulatory compensations are not, at least not the latter once established.
Comparison of 2-D Calculations from Periapical and Occlusal Radiographs versus 3-D Calculations from CAT Scans in Determining Bone Support for Cleft-Adjacent Teeth following Early Alveolar Bone Grafts
Authors: Rosenstein Sheldon W. , Long Ross E. , Dado Diane V. , Vinson Britt , Alder Marden E.
Published: October 2018

Objective

This investigation was conducted to determine the agreement between three-dimensional (3-D) calculations from CAT scans and two-dimensional (2-D) calculations from standard dental radiographs in evaluating bone support for cleft-adjacent teeth after primary bone grafting.

Design

This retrospective study utilized CAT scans and dental radiographs taken of the alveolar cleft in patients an average of 11 years after primary bone grafting.

Setting

The subjects were patients treated by the Cleft Palate Team at Children's Memorial Hospital and Loyola University Medical Center, Chicago, Illinois.

Patients

Fourteen UCLP patients (9 males, 5 females) agreed to participate In this study by undergoing CAT scan assessment of their alveolar cleft sites. They also had to have periapical or occlusal radiographs of the grafted cleft site taken within 6 months of the CAT scan.

Interventions

All patients underwent primary lip repair, placement of a passive palatal plate, primary alveolar bone grafting (mean age 6.4 months), and palatoplasty before 1 year of age. Major tooth movement through final orthodontics was completed by the time of the radiographic assessment.

Main Outcome Measures

CAT scan sections were reformatted and reconstructed to three-dimensionally calculate the percentage of root covered by bone support for the 15 teeth adjacent to the grafted cleft sites. Dental radiographs of the same teeth were also traced and digitized. Percentages of root supported by bone were also established using the dental radiographs by dividing the amount of root covered by bone, by the anatomic root length.

Results

A paired, two-sample t test revealed no significant differences between the two methods of assessment, while linear regression showed a statistically significant correlation between the CAT scan assessment and the percentages found on the radiographs.

Conclusions

Routine dental radiographs were able to estimate the total 3-D bone support for the roots of cleft adjacent teeth as determined by CAT scan to a statistically significant degree when groups where compared. The clinical significance for evaluation of individual cases was less impressive with a wide range of variability and a level of agreement that required acceptance of differences up to 25%.
A New Index for Assessing Surgical Outcome in Unilateral Cleft Lip and Palate Subjects Aged Five: Reproducibility and Validity
Authors: Atack Nikki E. , Hathorn Iain S. , Semb Gunvor , Dowell Tom , Sandy Jonathan R.
Published: October 2018

Objective

This study assessed the reproducibility, reliability, and predictive validity of a previously developed index by the authors for assessing surgical outcome in unilateral cleft lip and palate (UCLP) children aged 5.

Methods

Sixty randomly selected study models of 5- to 6-year-old complete UCLP subjects were obtained and the index was used to assess their surgical outcomes.

Results

Assessment of these study models using the new index demonstrated excellent intra-examiner agreement. The inter-examiner agreement was shown to be good. The corresponding longitudinal models at 16 to 18 years of 54 of the initial 5- to 6-year-old sample were also acquired. These subjects had undergone orthodontic treatment but not orthognathic surgery. The need for osteotomy amongst these models was assessed. Between 13% and 18% (depending on examiner) of 5-year-olds' models were scored in the groups likely to require orthognathic surgery. In the corresponding 16- to 18-year-olds' models, 9% were assessed as likely to benefit from an osteotomy. However, on an individual basis, it was not possible to predict future growth from study models at age 5.

Conclusions

This study has provided a reliable and reproducible index for assessing the outcome of surgery in UCLP subjects earlier than indices already available. True validation of the Index was not possible but it appears that it relies on face validity.
Comparison of Numeric Techniques in the Analysis of Cleft Palate Dental Arch Form Change
Authors: McAlarney Mona E. , Chiu Wei-Kwang
Published: October 2018

Objective:

Quantitative descriptions of form (size and shape) changes are significant to the understanding of the development, treatment planning, and prognosis of patients born with cleft lip and palate. This study compared the results of traditional dental arch form change measurements, such as width, depth, perimeter, and area, with four numeric methods: finite element scaling analysis, macroelement method, Euclidean distance matrix analysis, and conventional least-squares and resistant-fit theta rho Procrustes analyses.

Design:

Using tooth cusp landmarks on maxillary study casts, form change measurements of a male with complete bilateral cleft lip and palate at ages 2, 5, and 6 years were made comparing each age to the next older.

Results and Conclusions:

With the exception of the 2- to 5-year resistant-fit analysis, all numeric methods: 1) provide comparable results, 2) provide more detailed descriptions than do traditional methods, and 3) provide results that correlate well with the reported effects of increased lip pressure due to lip closure surgery. The use of finite-element scaling analysis on study casts is somewhat limited since: 1) there is more than one solution at teeth shared by many finite elements, 2) gross averaging of form change occurs within triangular elements, and 3) solutions can vary with the choice of element location. The use of the macroelement method circumvented the above finite element limitations with out compromising finite-element advantages. Procrustes results vary with the chosen superposition algorithm. The choice of the most appropriate Procrustes method required some a priori knowledge of form difference. The large number of results obtained by Euclidean distance matrix analysis and the nongraphic presentation of these results hamper quick interpretation but may be best suited for definitive statistical analysis. The graphic representation of both the magnitude and direction of: 1) landmark displacement in the Procrustes analyses (once size difference is eliminated), and 2) the rate of form change in the macroelement method provide an intuitive appreciation of how and where the casts differ.
Activities of Superior Pharyngeal Constrictor and Levator Veli Palatini Muscles Related to Respiration in Dogs
Authors: Kogo Mikihiko , Tanaka Susumu , Ishii Shouichiro , Hamaguchi Munehiro , Iida Seiji , Matsuya Tokuzo
Published: October 2018

Objective:

This study, using mongrel dogs, attempted to delineate the respiratory activity of two closure muscles of the velopharynx, the levator veli palatini (LVP) and superior pharyngeal constrictor (PC) muscles.

Method:

Fifteen mongrel dogs were given tracheotomies, with intratracheal tubes being connected to their respiratory circuits. Activities of the PC and LVP muscles were recorded electromyographically, and the effects of Pao2 and Paco2 were analyzed along with the effects of lung inflation intratracheally with O2 and N2.

Results:

Under hypoxic conditions, expiratory LVP activity was observed while PC activity diminished. The effects of lung inflation on both LVP and PC activities were also studied. The activities of both muscles were enhanced by lung inflation.

Conclusion:

It was thus suggested that the roles of both muscles during velopha-ryngeal closing movements are not the same.
Facial and Speech Relationships to Behavior of Children with Clefts across Three Age Levels
Authors: Richman Lynn C.
Published: October 2018

Objective:

This study was conducted to examine relationships of facial and speech ratings to behavior at three ages.

Design:

Multiple correlations to control for gender, IQ, and socioeconomic status (SES). Partial correlations between speech or facial ratings and behavior.

Setting:

A university hospital interdisciplinary cleft/craniofacial center.

Patients:

Sixty-five children with CP or CLP and no other anomalies seen at ages 6,9, and 12 years.

Interventions:

Speech ratings, facial ratings, hearing assessment, and SES ratings, were collected at ages 6,9, and 12.

Main Outcome Measure:

The Behavior Problem Checklist was used and internalizing problems were expected.

Results:

Less severe speech problems associated with behavioral inhibition were noted at age 9 years (p < .01). Greater facial disfigurement was correlated with greater inhibition at age 12 (p < .001).

Conclusions:

Longitudinal data indicates different associations of speech and facial variables were made to behavior at different ages (9 and 12). No significant relationships were identified at age 6.
Craniofacial Morphology in Patients with Kallmann's Syndrome with and without Cleft Lip and Palate
Authors: Mølsted Kirsten , Kjær Inger , Giwercman Aleksander , Vesterhauge Søren , Skakkebæk Niels Erik
Published: October 2018

Objective:

Kallmann's syndrome is characterized by the association of hypogonadotropic hypogonadism and anosmia or hyposmia. The principal endocrine defect of hypogonadotropic hypogonadism is a failure to secrete luteinizing hormone-releasing hormone (LHRH), resulting in underdevelopment of the pituitary gonadotropes and an inability to synthesize and release luteinizing hormone and follicle-stimulating hormone. The purpose of the present investigation was to describe the dentition and the craniofacial morphology in patients diagnosed with Kallmann's syndrome.

Design:

The sample consisted of 11 patients, 2 of whom also had bilateral cleft lip and palate. Radiographic investigations, including cephalometry, were performed. Comparisons were made to normal individuals and to cleft lip individuals without Kallmann's syndrome.

Results:

Dentition: tooth agenesis occurred more frequently in patients with Kallmann's syndrome. Craniofacial morphology: Increased mandibular inclination and mandibular angulation were seen in Kallmann patients. When clefting also occurred, extreme retrognathism of both maxilla and mandible was seen, a deviation which seemingly worsened during growth. The anterior cranial base and the sphenoid bone showed an altered morphology in one of the patients with Kallman's syndrome.

Conclusions:

An early diagnosis of Kallmann's syndrome is very important because the prognosis for endocrine treatment thereby improves, and therefore, it is recommended that the sense of smell be evaluated in patients with the craniofacial morphology described.
Effect of Alveolar Bone Grafting in the Mixed Dentition on Maxillary Growth in Complete Unilateral Cleft Lip and Palate Patients
Authors: Daskalogiannakis John , Ross R. Bruce
Published: October 2018

Objective:

This study was conducted to evaluate the effects on facial growth of alveolar bone grafting in the mixed dentition for patients with UCLP.

Design:

Retrospective cephalometric study.

Setting:

Craniofacial Treatment and Research Centre, The Hospital for Sick Children, Toronto, Ontario, Canada.

Patients:

The 58 patients participating in the study had a history of complete unilateral cleft lip and palate, all of which were repaired by the same plastic surgeon.

Interventions:

Twenty-one patients received an iliac-crest alveolar bone graft at a mean age of 10.3 years, while 37 did not receive an alveolar bone graft. Lateral cephalometric radiographs were obtained on all patients at two different times: at a mean age of 9.4 years (prior to bone grafting in the grafted group) and at a mean age of 15.2 years.

Main Outcome Measures:

All radiographs were traced and digitized by the same person, using cephalometric computer software. Superimposition and cephalometric analysis was undertaken to investigate the differences between the two groups in the 5.6-year experimental period. A two-way analysis of covariance was used for evaluation of the statistical significance of the results.

Results:

No statistically significant differences were found in 14 of the 15 cephalometric measurements performed. Harvold's maxillary unit length was statistically significantly shorter in the grafted group, although a lack of correlation with angular measurements and inherent problems with this specific measurement raise doubts in this finding.

Conclusion:

Mixed dentition bone grafting does not affect subsequent vertical and A-P development of the maxilla in complete unilateral cleft lip and palate patients during the first several postoperative years.
Structure of the Musculus Uvulae: Functional and Surgical Implications of an Anatomic Study
Authors: Huang Martin H. S. , Lee S. T. , Rajendran K.
Published: October 2018

Objective:

The role of the musculus uvulae in velopharyngeal function, its morphologic status in cleft palate, and its fate in palatoplasty procedures are subjects of controversy. The aims of this investigation were to re-examine this velar muscle to clarify its anatomic characteristics, to analyze its role in speech physiology, and to study the surgical implications of this information for cleft palate repair.

Methods:

Its attachments, morphology, and relations were examined in 18 fresh human adult cadavers by detailed dissection under 3.2× magnification and light microscopy.

Results:

The musculus uvulae was observed to be a paired midline muscle extending between the tensor aponeurosis anteriorly and the base of the uvula posteriorly along the nasal aspect of the velum. It had no attachments to the hard palate.

Conclusions:

These findings suggest that its action is to increase midline bulk on the nasal aspect of the velum, thus contributing to the levator eminence. It may also have an extensor effect on the nasal aspect of the velum, displacing it toward the posterior pharyngeal wall. Both of these actions would serve to maximize midline velopharyngeal contact. One clinical application of this anatomic information is that the muscle should be preserved in the dissection performed during intravelar veloplasty. Furthermore, it should be recognized that the musculus uvulae is invariably divided and reoriented incorrectly in the Furlow double opposing Z-plasty.
Measurement of Facial Soft Tissue Mobility in Man
Authors: Trotman Carroll-Ann , Stohler Christian S. , Johnston Lysle E.
Published: October 2018

Objective

The assessment of facial mobility is a key element in the treatment of patients with facial motor deficits. In this study, we explored the utility of a three-dimensional tracking system in the measurement of facial movements.

Methods and Results

First, the three-dimensional movement of potentially stable facial soft-tissue, headcap, and dental landmarks was measured with respect to a fixed space frame. Based on the assumption that the dental landmarks are stable, their motion during a series of standardized facial animations was subtracted from that of the facial and headcap landmarks to estimate their movement within the face. This residual movement was used to determine which points are relatively stable (≤1.5 mm of movement) and which are not (≥1.5 mm of movement). Headcap landmarks were found to be suitable as references during smile, cheek puff, and lip purse animations, and during talking. In contrast, skinbased landmarks were unsuitable as references because of their considerable and highly variable movement during facial animation. Second, the facial movements of patients with obvious facial deformities were compared with those of matched controls to characterize the face validity of three-dimensional tracking. In all instances, pictures that appear to be characteristic of the various functional deficits emerged.

Conclusions

Our results argue that tracking instrumentation is a potentially useful tool in the measurement of facial mobility.
Incidence of Cleft Lip and Palate and Risks of Additional Malformations
Authors: Hagberg Catharina , Larson Ola , Milerad Josef
Published: October 2018

Objective and Methods

Children with cleft lip and/or palate (n = 251) born between 1991 and 1995 in the county of Stockholm, Sweden, were studied with reference to incidence and rate ratios (RRs) of different types of clefts, gender, birth weight, mother's age, and length of pregnancy. Children who had clefts and additional malformations were compared with children who had clefts but no additional malformations.

Results

The incidence of clefts was 2.0/1000 live births, and it was higher among males than among females. The RR, an index of relative risk, was 1.58. The main groups, children with isolated cleft lip, children with cleft lip and palate, and children with isolated cleft palate, showed similar incidence values (0.6-0.7/1000 live births). Children with bilateral clefts had an incidence of 0.3/1000 live births. Additional malformations were found in approximately every sixth newborn with a cleft when children with Robin sequence were excluded. There was a tendency for newborns with bilateral clefts to have additional malformations (RR = 1.36; confidence interval = 0.74-2.49). Children with clefts and additional malformations had lower birth weight and were born earlier than children with clefts only.

Conclusion

Preterm cleft children with low birth weight should be screened for the presence of other birth defects.
A Cephalometric Evaluation of Craniofacial Morphology in Familial Dysautonomia
Authors: Mass Eliyahu , Brin Ilana , Belostoky Leon , Maayan Channa , Gadoth Natan
Published: October 2018

Objective

The purpose of this study was to delineate the craniofacial and dentoalveolar morphology of patients with familial dysautonomia (FD) in order to contribute to the understanding of the association between progressive sensory and autonomic neuropathy and the characteristic appearance of the dys-autonomic face.

Patients

The study group comprised 32 patients with FD (15 females and 17 males; mean age 10.8 years, SD 3.5 years, range 5.8-19.8 years).

Design

Lateral cephalograms from each patient were traced twice. The means of the two measurements were compared with homologous cephalo-metric normal values of ethnic-specific and classical norms from the literature.

Results

In some parameters, the craniofacial morphology of the FD group was significantly different from the classical norms. There was a pronounced retrognathism in the mandible and a steep mandibular plane angle. The skeletal features of FD patients more closely resembled those of their ethnic group, although they were more retrognathic, and the mandibular growth axis was more horizontal. The incisors of these patients were more retropositioned and retroclined than were those of their healthy counterparts.

Conclusions

The results suggest an insufficiency of the expected dentoal-veolar compensatory mechanism that usually helps to bridge skeletal discrepancies. It is postulated that the neuropathy is probably the important factor in the lack of this compensatory mechanism.
Median Cleft of the Lower Lip Associated with Lip Pits and Cleft of the Lip and Palate
Authors: Lekkas C. , Latief B.S. , Corputty J.E.M.
Published: October 2018

Objective

In this case report, we present an unusual combination of three congenital malformations: median cleft of the lower lip, lip pits, and unilateral cleft of the lip and palate without familial occurrence.

Conclusions

From an etiological point of view, this combination of malformations could have happened during the late embryogenic period. Why this combination is uncommon is not known.
Nasalance and Nasality in Low Pressure and High Pressure Speech
Authors: Watterson Thomas , Lewis Kerry E. , Deutsch Candace
Published: October 2018

Objective

This study compared nasalance measures and nasality ratings in low pressure (LP) and high pressure (HP) speech.

Subjects

The subjects for this study were 25 children ranging in age from 5 to 13 years. Twenty of the subjects were patients followed by a craniofacial team, and five had no history of communication disorder.

Results

The mean nasalance for the LP speech was 29.98% (SD, 16.16), and the mean nasalance for the HP speech was 30.28% (SD, 15.35). The mean nasality rating for the LP speech was 2.31, and the mean nasality rating for the HP speech was 2.59. Separate paired t tests revealed no significant difference between the LP or the HP speech for either the nasalance scores or the nasality ratings. The correlation coefficient between nasalance and nasality for the LP speech was r = 0.78, and for the HP speech r = 0.77. Using a cutoff of 26% for nasalance and 2.0 for nasality, Nasometer test sensitivity was 0.84 and test specificity was 0.88.

Conclusions

In general, clinicians may obtain valid measures of nasalance and/or ratings of nasality using either an LP stimulus or an HP stimulus. Sensitivity and specificity scores indicated that the Nasometer was reasonably accurate in distinguishing between normal and hypernasal speech samples.
Oral and Dental Health Status of Children with Cleft Lip and/or Palate
Authors: Paul Tahir , Brandt Ron S.
Published: October 2018

Objective

To ascertain the oral and dental health status of children with cleft lip and/or palate.

Design

Oral and dental examinations were carried out on 114 selected children with cleft lip and/or palate, using standard criteria and indices.

Setting

These children were examined at two cleft palate clinics in the United Kingdom.

Patients

All children examined were between the ages of 3 and 18 years.

Results

Sixty one (53.5%) of the subjects exhibited no evidence of previous caries experience, but as many as 20% exhibited active decay. The mean caries experience in the deciduous dentition (dmfs) was 2.3, and that in the permanent dentition (DMFS) was 0.9. Caries experience of the Caucasian children of the sample was lower (mean dmfs 1.9) than that of the Asian children (mean dmfs 5.5). The mean simplified debris index of the sample was 0.9, and the mean gingival bleeding index was 0.4. Children with cleft lip and palate had generally poorer oral and gingival health than those with isolated clefts of the lip or palate.

Conclusion

Twenty percent of the sample had active decay. These children had poor oral health in the surgically repaired anterior segment.
Spontaneous Cleft Palate in a Newborn Gorilla (Gorilla gorilla gorilla)
Authors: Siebert Joseph R. , Williams Bryan , Collins Darin , Winkler Linda A. , Swindler Daris R.
Published: October 2018

Objective

We report the first case of cleft palate in a newborn male gorilla (Gorilla gorilla gorilla).

Case History and Results

The full-term infant was born to clinically healthy, wild-caught parents and survived 5 days. Autopsy disclosed a unilateral cleft palate, moderate scalp hemorrhage (birth versus postnatal trauma), cerebral edema, and a sterile fibrin vegetation in the heart. The palate was also shorter and narrower than expected, and the biorbital breadth was reduced; otherwise, growth and development appeared normal. Standard cranial and intraoral radiographs and three-dimensional reconstructions of computerized tomographic (CT) scans provided thorough and noninvasive methods of studying the craniofacial complex and extracranial skeleton. By this technique, major findings were: intact premaxilla, interpremaxillary, and premaxillary/maxillary sutures; intramaxillary cleft with ipsilateral choanal atresia; mildly asymmetric inferior turbinates; and normal nasal septum and vomer.

Conclusions

Except for choanal atresia, cleft palate was not associated with other major craniofacial or extracranial anomalies in this case. Choanal atresia has been observed at times with cleft palate, but to our knowledge, the association has not been reported in nonhuman primates. Cleft palate, with or without cleft lip, has been recognized in a variety of nonhuman primates, including the lemur, marmoset, tamarin, squirrel monkey, and macaque. Some occurrences are spontaneous, while others are syndromic and/or arise from genetic or teratogenic influences. Each mode of presentation is poorly understood in nonhuman primates, but in this case, the absence of relevant environmental or parental history suggests that the occurrence was spontaneous. Anatomic studies of nonhuman primates are particularly valuable when they involve endangered species and will hopefully increase our understanding of the pathogenesis and etiology of congenital disorders, as well as other relationships between nonhuman primates and humans.
Etiopathogenesis of Isolated Robin Sequence
Authors: Marques Ilza Lazarini , Barbieri Marco Antonio , Bettiol Heloisa
Published: October 2018

Objective

To investigate the etiopathogenesis of isolated Robin sequence.

Design

A longitudinal and prospective study of children with isolated Robin sequence and no other associated syndromes or malformations.

Setting

The study was carried out at the Hospital de Reabilitacao de Anomalias Craniofaciais (formerly the Hospital for Research and Rehabilitation of Cleft Lip/Palate), University of São Paulo, Bauru, SP, Brazil, which provides care for patients with lip/palate lesions throughout Brazil.

Patients

Forty-three children were initially included in the study, seven of whom were later found to be cases of Stickler syndrome and excluded. The remaining 36 children presenting only the anomaly triad of microretrognathia, glossoptosis, and cleft palate were followed up from the first month of life to 4 years of age with repetitive clinical and ophthalmological examination.

Main outcome Measure

A family history of cleft lip/palate was determined on the basis of information provided by the parents and, when possible, the affected relative was submitted to physical examination.

Results

A family history of cleft lip/palate was observed in 27.7% of cases, one case having a younger brother with only cleft palate but no microretrognathia or glossoptosis. Six cases of isolated cleft palate and three cases of cleft lip with or without cleft palate were present in distant relatives. Complete U-shaped cleft palate (wide cleft) was the most frequent type of cleft, which was present in 75% of cases. Only one case of incomplete cleft palate was observed, but U-shaped; 25% of the patients presented complete V-shaped cleft palate.

Conclusions

We conclude that heredity could be a factor in the etiopathogenesis of isolated Robin sequence and suggest that cleft palate (usually complete and U-shaped) is the primary event in the determination of the triad of anomalies.
Psychological Issues in Craniofacial Care: State of the Art
Authors: Endriga Marya C. , Kapp-Simon Kathleen A.
Published: October 2018

This article is an extensive review of the psychological literature on cleft lip and palate and other craniofacial anomalies. Issues of parental acceptance, social competence, self-concept, emotional adjustment, and cognitive functioning are examined. Cumulative research suggests that many children with craniofacial conditions develop in a typical manner and do not experience psychological problems. However, a significant number of children (30% to 40% in most studies) experience difficulties with internalizing and/or externalizing problems, learning disorders, and social competence. A multifactorial model for examining the domains that are related to both psychological risk and protection is recommended. The need for multicenter studies that are prospective and clinically relevant is emphasized.
Medical Genetics on the Cleft Palate–Craniofacial Team: Understanding Parental Preference
Authors: Berk Nancy W. , Marazita Mary L. , Cooper Margaret E.
Published: October 2018

Objective

This study examined health care professionals' perceptions of parental preferences and preferences of parents of affected children (cleft lip, cleft palate, both, or other craniofacial anomaly) for prenatal testing with respect to oral-facial clefts and craniofacial anomalies.

Design

This was a descriptive study assessing and comparing questionnaire responses of two independent groups.

Participants

Healthcare professional participants comprised 570 U.S. members of the American Cleft Palate–Craniofacial Association. Parent participants comprised 97 parents of children seen at the University of Pittsburgh Cleft Palate–Craniofacial Center.

Main outcome Measures

A practitioner survey was developed to examine practitioner perception of parental desire for involvement, desire for knowledge, and reaction to learning of a birth defect. A parent survey was developed to correspond with the practitioner survey.

Results

Health professionals' perceptions of parental preferences often paralleled parental preference data. However, parental responses were found to be significantly different from practitioner perception of parental preferences on one question reflecting desire for involvement (x2 = 33.35, df=1, p = .001) and on one question reflecting desire for knowledge (x2 = 4.82, df =1, p = .03). For both questions, higher percentages of parents responded that they would want this involvement or information.

Conclusions

Findings are consistent with those reported In other studies of prenatal testing preferences and susceptibility. Results suggest that parents of affected children have a strong desire for information and involvement in prenatal testing and counseling decisions. Parents appear to value preparation in spite of acknowledging anxiety associated with prenatal information.
Long-Term Outcome Analysis of Two Treatment Methods for Cleft Palate: Combined Levator Retropositioning and Pharyngeal Flap versus Double-Opposing Z-Plasty
Authors: Lin Kant Y. , Goldberg David , Williams Christopher , Borowitz Kathleen , Persing John , Edgerton Milton
Published: October 2018

Objective

Two surgical techniques for repair of a cleft palate include levator retropositioning in combination with a pharyngeal flap and the Furlow double-opposing Z-plasty. This study compared morbidity and speech results from the use of these two methods in an effort to determine which was the superior technique.

Design

Patient records from 1986 to 1996 were retrospectively reviewed, and 10 patients with a cleft palate who underwent repair with a levator retropositioning and pharyngeal flap were compared to 14 patients who underwent a double-opposing Z-plasty repair. Postoperative complications including fistula formation, obstructive sleep apnea, and residual velopharyngeal insufficiency were recorded. Speech was assessed perceptually and through the use of nasometry.

Results

Both surgical techniques resulted in good speech In the majority of patients. Only two patients in the study, both In the Z-plasty group, had severe postoperative hypernasality. Two patients in the levator retropositioning and pharyngeal flap group developed severe postoperative obstructive sleep apnea, requiring additional surgery.

Conclusion

The levator retropositioning and pharyngeal flap technique was successful In achieving good speech results, but it also caused more serious postoperative complications when compared to the double-opposing Z-plasty technique.
The Organization and Delivery of Craniofacial Health Services: The State of the Art
Authors: Strauss Ronald P.
Published: October 2018

The dominant organizational structure providing care for cleft palate and other craniofacial conditions is the health care team. Various types of health care team organization are profiled, including intradisciplinary, multidisciplin-ary, and interdisciplinary teams. Effective team-based care delivery has the ability to address the fragmentation and dehumanization that can result when a variety of specialists and disciplines are required to provide assessment and technical care. A team's leadership and its hierarchy of professional authority can be expected to affect its ability to function effectively. Health reform and managed care are considered for their impact on the team and on the doctor-patient relationship. Trends in team regionalization, quality assurance, outcomes research, and consumer advocacy are reviewed. The cleft palate and craniofacial team is profiled as an organizational model that is being affected by the forces of health system change.
Comparison of Periodontal Disease in Patients with Clefts of Palate and Patients with Unilateral Clefts of Lip, Palate, and Alveolus
Authors: Schultes Günter , Gaggl Alexander , Kärcher Hans
Published: October 2018

Objective

Long-term health of the stomatognathic system as well as esthetic aspects are the therapeutic goals in patients with orofacial clefts. The aim of this study was to analyze the periodontal condition of patients with cleft palate (CP) and cleft lip, palate, and alveolus (CLP) and to determine the differing degrees and localization of periodontal disease.

Design

In 30 patients with unilateral cleft lip, palate, and alveolus and 30 patients with cleft palate (CP), periodontal lesions were identified and classified according to the Community Periodontal Index of Treatment Needs (CPITN) and pathological mobility of teeth was noted. The state of oral hygiene was recorded by the Approximal Plaque Index.

Results

In general, poor oral hygiene was found in all patients. Patients with CLP were classified as CPITN code 0 in 0%, codes 1 and 2 in 7%, code 3 in 43%, and code 4 in 50% of cases. Patients with cleft palate showed code 0 in 0%, codes 1 and 2 in 30%, code 3 in 45%, and code 4 in 25% of cases. In this study, periodontal disease was found in patients with cleft palate to a similar extent to that in the general population, whereas patients with cleft lip, palate, and alveolus had a predisposition to deep peridontal destruction of teeth adjacent to the cleft. The registration of pathological mobility of teeth and resulting loss of attachment corresponded to the periodontal disease classification according to the CPITN.

Conclusion

A critical periodontal situation was found in patients with unilateral cleft lip, palate, and alveolus. In contrast, patients with cleft palate exhibited a similar periodontal situation to that found in the general population, with additional damage that may be attributed to orthodontic treatment.
The Intrajudge Reliability of the Perceptual Rating of Cleft Palate Speech before and after Pharyngeal Flap Surgery: The Effect of Judges and Speech Samples
Authors: Keuning Kornelis H.D. , Wieneke George H. , Dejonckere Philippe H.
Published: October 2018

Objective

In this pilot study, the reliabilities of the perceptual ratings of four types of speech samples by six judges, with and without expertise in evaluating cleft palate speech, were studied.

Design

Pre- and postoperative tape recordings of 15 patients with cleft lip and palate who had undergone a superiorly based pharyngeal flap operation were selected. Five speech-language pathologists and one oral and maxillofacial surgeon perceptually rated the following variables on separate 100-mm visual analog scales: hypernasality, audible nasal emission, intelligibility, mis-articulations associated with velopharyngeal insufficiency, voice quality, and the presence or absence of hyponasality. These six variables were rated in four types of speech samples: reading of three sentences, repeating after the speech pathologist of three sentences, 10 sentences containing the aforementioned material, and the same 10 sentences in paired comparison. All speech samples were rerated after 3 months by the same judges.

Results

Judges differed largely in the range they used in their rating. Intrajudge reliability of .56 to .78 was found for ratings of hypernasality. No significant differences in intrajudge reliability were found for the ratings with the different types of speech samples. The intrajudge reliability of a judge with expertise was not necessarily higher than of a judge without this expertise.

Conclusions

The improvement in speech is most reliably assessed with speech samples in paired comparison. A speech-language pathologist with expertise in evaluating cleft palate speech does not guarantee a high intrajudge reliability of the rating.
Acoustic Airway Profiles in Unilateral Cleft Palate Patients
Authors: Kunkel Martin , Wahlmann Ulrich , Wagner Wilfried
Published: October 2018

Objective:

This study investigates the nasal airway in unilateral cleft palate patients by means of a noninvasive, objective diagnostic method that provides topographic information about the airway profile.

Design:

A consecutive sample of patients was measured.

Setting:

Cleft palate rehabilitation center of the University of Mainz, Germany.

Patients:

Forty-nine subjects were investigated: 34 full-grown patients with complete unilateral cleft lip and palate and 15 controls with subjective normal nasal patency.

Intervention:

A transnasal series of three acoustic measurements of nasal volume was performed per nostril; measurements were taken both before and after decongestion with 0.3 mg xylometazoline per nostril. Minimum cross-sectional area, nasal volume, and decongestion capacity were calculated for both the cleft side and the contralateral side and for both nasal sides in controls.

Results:

Pathologic obstructions (<0.4 cm2) were detected on the cleft side in 75% of patients but were detected in only 15% of patients on the contralateral side (p < .001). The valve area of the cleft side (0.32 ± 0.2 cm2) yielded significantly (p < .001) lower cross-sectional values compared with the contralateral side (0.56 ± 0.1 cm2). Total nasal volume was determined to be 35% smaller on the cleft side (p < .001). Significantly higher decongestion capacity was verified on the cleft side, thus indicating mucosal hypertropy.

Conclusion:

Despite a wide range of interindividual variability, we recognized a characteristic “descending W” airway pattern in cleft palate patients. Acoustic rhinometry seems to be a powerful tool for acquiring topographic information about the individual airway profile. It has proven helpful in visualizing the location and amount of pathologic obstructions, rendering it especially useful for preoperative investigation and quality control in corrective cleft nose surgery.
Effects of Maxillary Protraction Combined with Chin-Cap Therapy in Unilateral Cleft Lip and Palate Patients
Authors: Ishikawa Hiroyuki , Kitazawa Shinichi , Iwasaki Hiroshi , Nakamura Shinji
Published: October 2018

Objective

This study investigated the treatment effects of maxillary protraction combined with chin-cap therapy in complete unilateral cleft lip and palate patients at the deciduous and early mixed dentition stages.

Method

Twenty-six Japanese children (10 boys and 16 girls) with complete unilateral cleft lip and palate were examined. All had undergone pushback operations for palatal repair at approximately 18 months of age. Maxillary protraction began between 5 and 7 years of age and continued for 10 to 38 months. Lateral cephalograms were used to analyze skeletal changes during the first and second years of treatment, and the relationship between pretreatment midfacial morphology and forward displacement of the maxilla during the first year was investigated.

Results and Conclusions

During the first year of treatment, the mean increase in the ANB angle for all cases was 2.37°. The forward displacement of the maxilla varied considerably, from 0.23 mm to 3.03 mm. The treatment response was significantly smaller in the second year, and no benefit from treatment longer than 1 year was established. The amount of maxillary forward displacement was significantly correlated with the pretreatment posterior upper facial height. Patients with smaller posterior upper facial height showed a poorer treatment response, whereas patients with a greater posterior upper facial height responded better to treatment. Individual differences in maxillary growth acceleration may be related to growth inhibition associated with post-surgical scar tissue on the palates.
Three-Dimensional Bolton–Brush Growth Study Landmark Data: Ontogeny and Sexual Dimorphism of the Bolton Standards Cohort
Authors: Dean David , Hans Mark G. , Bookstein Fred L. , Subramanyan Krishna
Published: October 2018

Objective

The treatment of craniofacial reconstructive surgery patients may benefit from comparison to average referent three-dimensional landmark data. These data may be useful for diagnosis, treatment planning, prosthetic design, or outcomes assessment. With regard to subadult patients, we hypothesize that the pattern of ontogenetic shape change of same sex, same ethnicity, referent populations will show gross uniformity. We present a preliminary shape analysis of 50 three-dimensional landmarks derived from 317 Bolton–Brush Growth Study biorthogonal image pairs. We determine which landmarks can be collected from scanned radiographs reliably by four operators for the precisely locatable points, ontogenetic trends in landmark configuration shape change, and patterns of sexual dimorphism.

Participants

Participants were Bolton standards individuals (16 male and 16 female) who contributed biplane cephalograms seven or more times with annual or greater spacing between ages 3 and 18 years.

Design

After removing outliers, we searched for ontogenetic heterogeneity, including sexual dimorphism and within sex-specific Procrustes coordinate shape spaces.

Results

A cut-off of 4.3-mm interoperator error left 32 landmarks in our analysis. Three different approaches (principal component analysis, age-trend analysis, and principal components of age residuals) all found no patterns of individual variation around sex-specific average trends of shape change. Male shape change peaks at age 15, a correlate of the growth spurt.

Conclusions

Simultaneous frontal and lateral anatomic landmark identification improves three-dimensional localization reliability. Three-dimensional craniodental shape change from ages 8 to 18 within the Bolton standards presents little heterogeneity. Considerations of ethnicity aside, these may be initial grounds for use of these data as a normative referent.