bluuebunny/crossref_metadata_2025 huggingface dataset:
Facial Growth after Primary Periosteoplasty versus Primary Bone Grafting in Unilateral Cleft Lip and Palate
Authors: Sameshima Glenn T. , Banh Dan S. , Šmahel Zbynék , Melnick Michael
Published: October 2018

A cephalometric evaluation of long-term changes resulting from two different surgical techniques for the correction of unilateral cleft lip and palate (UCLP) was undertaken on a sample of 19 males with primary bone grafting, and 42 males with primary periosteoplasty from the Science Academy of the Czech Republic, Prague. Lateral cephalometric radiographs collected at approximately 10 and 15 years of age were traced, digitized, and analyzed using the finite-element method. Facial growth and development between the two groups were compared. Significant differences between the two groups were found in the upper face, the posterior part of the maxilla, the anterior bony chin, and nasal bone areas. The periosteoplasty group demonstrated significantly greater vertical development between anterior cranial base and the maxilla. The bony chin was larger and a greater inferior displacement of the posterior palate were found in the periosteoplasty group. An increased proclination in the nasal bone was observed in the bone graft group. This investigation both confirms the findings of a previous study of these sample populations, and provides additional important details regarding the differences between the groups.
Asymmetries of the Upper Lip and Nose in Noncleft and Postsurgical Unilateral Cleft Lip and Palate Individuals
Authors: Kyrkanides Stephanos , Bellohusen Ron , Subtelny J. Daniel
Published: October 2018

The purpose of this study was to retrospectively evaluate post-surgical asymmetries of the upper lip and nose in complete unilateral cleft lip and palate individuals using documentation collected over a period of years. Twenty-three complete unilateral cleft lip and palate subjects (UCLP) and 34 noncleft subjects who served as controls and had undergone orthodontic treatment were Included in the study. Nasal asymmetry, deviation of the tip of the nose, and the deviation of the midpoint of the vermilion border of the upper lip were compared to chronologic age and skeletal maturation in cleft subjects and controls. Comparison between cleft and control cases was also assessed. Results indicate that nasal asymmetry peaks at postpubertal growth stages in the control groups, and the difference between UCLP and controls decreases with time and maturation. The deviation of the nasal tip peaks during the pubertal growth spurt for both UCLP and controls, and the difference between UCLP and controls also decreases with time. The deviation of the midpoint of the vermilion border shows relatively small change with time and maturation, and there is no significant difference between UCLP and controls. In general, the facial asymmetries studied were not as great as expected, and the difference between UCLP and controls decreased with time, growth, and maturation.
Nasometric Values for Normal Spanish-Speaking Females: A Preliminary Report
Authors: Anderson Raquel T.
Published: October 2018

Most normative data available for assessing resonance through instrumentation have been collected with English-speaking individuals. The present study aimed at providing initial data on Spanish for use with the nasometer. Mean nasalance scores were obtained from 40 normal Puerto Rican Spanish-speaking females while they read three types of stimuli: (1) sentences containing nasal consonants, (2) a reading passage with both oral and nasal consonants, and (3) a reading passage with oral consonants. Results indicated significant differences In mean nasalance scores across the nasal sentences, as well as the two paragraph stimuli. In addition, a high degree of intersubject variability in the production of the target stimuli was evidenced. Comparisons with previous English normative data with similar reading stimuli are made and possible avenues for further research on the use of the nasometer with Spanish-speaking populations are discussed.
Aberrant Retropharyngeal Internal Carotid Artery in a Child with Spondyloepimetaphyseal Dysplasia and Cleft Palate
Authors: Fluke Matthew M. , Bronfin Daniel R. , Superneau Duane W. , Moses Michael H.
Published: October 2018

In a patient with spondyloepimetaphyseal dysplasia and cleft palate, the course of the internal carotid artery was aberrant. Prompt identification of this anomaly is critical, because operative fatality can occur if the defect is unrecognized. This case illustrates the occurrence of this vascular anomaly and focuses on options for radiologic identification and evaluation of the defect.
Cephalometric Comparison of Craniofacial Morphology between Primary Bone Grafted and Nongrafted Complete Unilateral Cleft Lip and Palate Adults
Authors: Suzuki Akira , Goto Keiya , Nakamura Norifumi , Honda Yasuo , Ohishi Masamichi , Tashiro Hideo , Fujino Hiroshi
Published: October 2018

The effect of primary bone grafts on craniofacial growth was evaluated in adult patients with complete unilateral cleft lip and palate (UCLP).The subjects were 18 UCLP patients with primary bone grafts and a control group of 36 UCLP patients without bone grafts. The former received primary transplantation of autogenous rib bone simultaneously with primary cheiloplasty by the same surgeon between 1963 and 1969. Frontal and lateral cephalograms taken after 16 years of age were traced. Skeletal landmarks were Identified on them, and their x,y-coordinates were digitized. The effects of gender and/or primary bone graft on the craniofacial morphology were tested by ANOVA using 21 angles, 8 breadths, 13 distances, and 2 ratios. Nasal cavity breadth was wider in the primary bone grafted group. However, the anterior maxilla in that group was more upward and retruded than that in the nongrafted group. The primary bone grafted group was divided into two subgroups based on the overjet of the incisors: group N = normal overjet; group C = anterior cross-bite. There was no difference in the maxilla between groups N and C. However, the mandible in group C was more anterior and superior than in group N. In conclusion, primary bone graft may impede maxillary horizontal and vertical growth to a certain degree, and severe anterior cross-bite in primary bone grafted subjects may be brought about by mandibular closure.
Orthodontic Treatment of a Cleft Palate Patient with Surgically Assisted Rapid Maxillary Expansion
Authors: Susami Takafumi , Kuroda Takayuki , Amagasa Teruo
Published: October 2018

Some adult cleft palate patients show severe maxillary transverse contraction and posterior crossbite. This case report demonstrates successful surgical-orthodontic treatment of such a patient. Surgically assisted rapid maxillary expansion (SA-RME) was completed prior to comprehensive orthodontic treatment. The osteotomy was performed on both the buccal and lingual aspects of the posterior maxillary alveolus. A Hyrax-type maxillary-expansion appliance was used, and the screw (0.2 mm, one quarter turn) was turned two or three times per day. Comprehensive orthodontic treatment was initiated after extraction of the mandibular first premolars and four third molars. The maxillary lateral incisors were also extracted after active orthodontic treatment. The amount of expansion achieved using SA-RME was greater at the posterior than at the anterior maxilla. Midpalatal suture opening occurred. After orthodontic treatment, occlusal stability was satisfactory. This case demonstrates the effectiveness of SA-RME in adult cleft palate patients with severe posterior crossbite.
Palatal Morphology and Type of Clefting
Authors: Kilpeläinen Pauli V.J. , Laine-Alava Maija T. , Lammi Seppo
Published: October 2018

In this study, the variation in palatal dimensions, particularly for height, width, and depth, were evaluated in subjects with different types of cleft palate using the moiré technique. The sample consisted of 95 subjects with cleft palate and 68 noncleft individuals. Effects of cleft type, gender, developmental stage of the dentition, missing teeth, and 11 skeletal and pharyngeal cephalometric variables on palatal dimensions were assessed using multiple-regression analyses. The typing of clefts had an effect on the all palatal dimensions. The palate was shallower in subjects with clefts involving the secondary palate than in other types of clefts or in none left individuals. The palatal width was decreased in subjects with cleft in the primary palate, and the palatal depth was affected by all cleft types except submucous cleft. Gender, developmental stage of the dentition, missing teeth, and some of the cephalometric variables also had a certain effect on the palatal dimensions.
Facial Growth in Patients with Unilateral Clefts of the Lip and Palate: A Two-Center Study
Authors: Roberts-Harry Dai , Semb Gunvor , Hathorn Iain , Killingback Norman
Published: October 2018

Two groups of 10-year-old patients with complete unilateral clefts of the lip and palate were examined in this study. Two centers; Bristol, U.K. and Oslo, Norway, who had different treatment regimens were used. The groups comprised 40 patients from Oslo and 32 from Bristol. The groups were matched, in proportion to the size of the groups, for age, sex, and presence of Simonart's bands. In Oslo, a Millard lip repaired was performed at 3 months of age with a von Langenbeck palatal repair at 18 months, no presurgical orthopedics was employed and there was no primary nasal correction. The Bristol center also repaired the lip at 3 months with a Millard type repair but also performed a radical nasal correction at the same time. The palate was repaired at 6 months with a Veau repair, and presurgical orthopedics using a pinned arch orthopedic plate was carried out. In addition, the volume of primary repairs per surgeon was much higher in Oslo, and a much stricter treatment protocol was used compared with Bristol. Lateral cephalograms obtained within 1 year of the child's tenth birthday were digitized, and the craniofacial morphology of the two groups was compared. Significant differences in maxillary growth and soft tissue profile were noted with a much more retruded mid-face and flatter nasiolabial angle in the Bristol group. The main factors for the better results in Oslo are suggested to be the absence of presurgical orthopedics, no radical nasal correction, the high volume of operations performed per surgeon, and the stricter protocol.
Major Residual Deformities in Cleft Patients: A New Anthropometric Approach
Authors: Vegter Florine , Mulder J. Wiebe , Hage J. Joris
Published: October 2018

Objective

A new method based on anthropometric ratios and inclinations was evaluated to confirm or reject whether residual deformities in cleft patients' faces differ significantly from faces without previous clefts.

Design, Setting, and Patients

From a retrospective, a selective review of our clinical flies, seven measurements were made on 156 photographs of the faces of 52 patients aged 19 to 25 years. Three ratios and two inclination angles were recorded for each patient.

Results

The primary hypothesis could be confirmed: the anthropometric ratios and angles used to express major residual deformities in cleft lip and nose patients differ significantly from normal values.

Conclusion

This new method allows for objective measurement and evaluation of both the stigmata and the outcome of their treatment.
Radiographic Assessment of Secondary Autogenous Alveolar Bone Grafting in Cleft Lip and Palate Patients
Authors: Kindelan Jeremy D. , Nashed Robert R. , Bromige Michael R.
Published: October 2018

Objective

To assess if a 4-point, radiographically based scale could be used between operators to reliably assess the success of secondary alveolar bone grafting.

Design

The study was retrospective with the clinicians blind to patient identity. Radiographs were examined twice by two clinicians with 1 week between assessments.

Setting

The research was carried out in a hospital-based orthodontic/oral and maxillofacial unit.

Patients

All patients who had secondary alveolar bone grafting in this unit between February 1992 and March 1995 were included In this study. There were 38 patients with a total of 48 grafted sites.

Interventions

The bone graft site was radiographed following orthodontic expansion prior to grafting. The radiograph was repeated postoperatively at a mean of 4 months after surgery.

Main Outcome Measure

The degree of bony fill in the cleft area was assessed using a 4-point scale: Grade 1 >75% bony fill; Grade 2 50–75% bony fill; Grade 3 < 50% bony fill; Grade 4 no complete bony bridge.

Results

Overall, 50% of grafts achieved grade 1, 23% were grade 2, 22% grade 3, and 5% grade 4. Levels of intra- and inter-observer agreement were highly variable (.33 to .72 kappa statistic).

Conclusions

The 4-point scale described could be used to assess the success of autogenous secondary alveolar bone grafting. It showed moderate to substantial intra-observer agreement, and fair-to-moderate inter-observer agreement.
Evaluation of the Results of Cleft Lip and Palate Surgical Treatment: Preliminary Report
Authors: Mortier P.B. , Martinot V.L. , Anastassov Y. , Kulik J.F. , Duhamel A. , Pellerin Ph. N.
Published: October 2018

Objective

The aim of this study was to present a measuring tool, in the form of a dual-rating grid, for preoperative appearance and postoperative results in the treatment of labionasal clefts.

Methods

This study was carried out on 43 children with partial unilateral clefts, operated on using a derivation of Millard's technique. The preoperative initial severity score (ISS) was the sum of points each corresponding to a precise anatomic anomaly. A severe cleft was given a rating of 6, and a mild cleft a rating of 1. The postoperative results score (PRS) was the sum of points corresponding to residual or acquired anatomic anomalies: a good result was given 0.5, and a poor result 3.5. Each score was a mark given separately and agreed on by two surgeons. To compare the rating grids, the correlation between the ISS and the PRS was calculated.

Results

Twenty-four patients had an ISS less than 5; 19 patients had an IGS more than or equal to 5. Marks for the PRS were between 0.5 and 3.5 with the majority at 1 to 1.5.

Conclusion

This study demonstrates the coherent connection between these two pre- and postoperative rating grids.
Three-Dimensional Study of Growth and Development of the Nose
Authors: Ferrario Virgilio F. , Sforza Chiarella , Poggio Carlo E. , Schmitz Johannes H.
Published: October 2018

Objective:

The aim of this study was to describe normal nasal growth in a large sample of boys and girls from 6 to 14 years of age, to compare nasal development at 14 years of age with its adult dimensions, and to evaluate differences in growth patterns between males and females.

Design:

Growth and development of the nose were analyzed through the three- dimensional facial morphometry method. One thousand thirteen examinations were performed on 402 children between 6 and 14 years of age and on 101 adults. Three-dimensional coordinates of five nasal landmarks were collected. Eleven parameters including nasal volume and external surface were calculated and averaged for age and sex.

Results:

Volume, surface, and linear distances were larger in males than in females, with the exception of the 11-to-12 age group, where a sharp female growth spurt was present. In males, the growth spurt was broader, and continued to 13 years of age at a nearly steady rate. Statistically significant gender differences could be found both in the adult group and before the adolescent growth spurt.

Conclusions:

Our findings are in general agreement with previous reports on craniofacial growth, which suggested a strong tendency in males for growth to continue for several years beyond that seen in females.
Examine Your Orofacial Cleft Patients for Gorlin-Goltz Syndrome
Authors: Lambrecht J. Thomas , Kreusch Thomas
Published: October 2018

The Gorlin-Goltz syndrome is characterized by tour primary symptoms: multiple nevold basal cell epitheliomas that usually undergo malignant transformation; jaw keratocysts that show constant growth; skeletal anomalies; and intracranial calcifications. A myriad of additional findings may also be noted. Among the most frequent are: palmar and plantar pits, a characteristic flattened facles and broad nasal root, frontal and parietal bossing, mandibular prognathia, hypertelorism, strabismus, dystrophia of the canthi, and clefts of the lip, alveolus, and/or palate. In this study, we review the literature and our 25 cases of Gorlin-Goltz syndrome patients, questioning their incidence of cleft formations (8.5%) as compared to the general population (0.1%). It is our contention that all patients who present with an orofacial cleft warrant deeper investigation as to the presence of additional signs indicative of Gorlin-Goltz syndrome. The nevi turn malignant with time, and thus, early diagnosis, follow-up, and treatment are imperative.
Research on Psychosocial Issues of Children with Craniofacial Anomalies: Progress and Challenges
Authors: Pope Alice W. , Speltz Matthew L.
Published: October 2018

No abstract available.
Presurgical and Postsurgical Mental and Psychomotor Development of Infants with Sagittal Synostosis
Authors: Speltz Matthew L. , Endriga Marya C. , Mouradian Wendy E.
Published: October 2018

Objective:

The current study compared the mental and psychomotor development of infants with nonsyndromic sagittal synostosis (SS) with a demographically matched comparison group without congenital defects. Within the SS group, we tested the hypothesis that age of cranial release would be inversely correlated with mental development.

Design:

The design was prospective and longitudinal. Participants were assessed at 4, 12, and 24 months of age.

Setting:

The study was conducted in a craniofacial clinic at an urban children's hospital.

Participants:

Participants were 19 infants with SS (consecutive craniofacial program referrals) and 19 demographically matched comparison infants recruited from the community. One infant with SS did not attend the 24-month assessment.

Main Outcome Measures:

Mental and Psychomotor indices from the Bayley Scales of Infant Development were the primary outcome measures. Subdomains of development were created using Kohen-Raz scoring procedures. All measures were determined a priori.

Results:

Repeated-measures MANOVAs revealed no statistically significant differences in the developmental trajectories of the two groups. None of the SS group infants received Mental Development Index (MDI) scores in the mentally retarded or borderline range of intellectual functioning (i.e., below 78). An inverse correlation (r = −.30) was found between the age at surgery and Bayley growth curve coefficients; however, this association was not statistically significant (p = .10, one-tailed).

Conclusions:

Results are consistent with previous studies of the mental and psychomotor development of infants with nonsyndromic craniosynostoses in relation to normative test data. The relation between surgery age and developmental outcome merits further study in a larger sample with a greater range of surgery ages.
Observed Social Interaction Patterns in Adolescents with and without Craniofacial Conditions
Authors: Kapp-Simon Kathleen A. , Mcguire Dennis E.
Published: October 2018

Objective:

This study examined social interactions of adolescents in a natural environment (school lunch room) to determine if there were identifiable differences in social behavior between children with and without craniofacial conditions (CFC).

Design:

This was an observational study comparing social interaction skills of children with CFC to peers without craniofacial conditions.

Setting:

The observations were conducted in the respective school lunch rooms of the adolescents with CFC.

Participants:

Clinical subjects were 13 adolescents (4 male) with various craniofacial conditions (5 cleft lip and palate) and 12 (4 male) peers without CFC present in the same lunch room.

Main Outcome Measures:

An unknown observer obtained 45 minutes of structured observational data on subject initiations, responses, nondirected comments, and extended conversations over two to three lunch room periods. Data was coded on the Epson HX-20 for type, frequency, and duration of social contact. Specific measures included: subject initiations and responses, peer initiations and responses, conversations events, and nondirected comments.

Results:

Statistically significant differences were found between CFC and comparison subjects (CS) on each social interaction variable measured. CS initiated more contacts, received positive responses more frequently, and engaged in longer conversations than CFC subjects ([F (1,24) = 14.1, p <.01; F (1,24) = 61.2, p <.001; F (1,24) = 5.50, p <.05]. CS were approached by and responded appropriately to peers more often [F (1,24) = 28.1, p <.001; F (1,24) = 43.2, p <.001]. Subjects with CFC were more likely to produce nondirected comments (N = 7, x = 0, p < .01).

Conclusions:

A significant number of children with CFC behaved differently than their peers in a natural, daily occurring situation. They were often at the periphery of the group, observers rather than participants in conversation.
Personality Attributions Based on Speech Samples of Children with Repaired Cleft Palates
Authors: Berry Leigh A. , Witt Peter D. , Marsh Jeffery L. , Pilgram Thomas K. , Eder Rebecca A.
Published: October 2018

Objective:

This study examined whether or not assumptions made about personality characteristics based on speech samples differed for children with repaired cleft palates (CP) versus unaffected children.

Design:

Audiotapes of speech samples were presented in random order to blind raters.

Patients/Participants:

The subjects were 20 children (10 females, 10 males) with repaired CP and 16 control (i.e., unaffected) children (8 females, 8 males). All children were 8 to 12 years of age, Caucasian, living in the St. Louis area, and lower-middle to upper-middle class. The raters were 20 (13 females, 7 males) 6th grade Caucasian students who attend a private school in the area.

Setting:

Raters heard tapes in a group setting, but with individual headphones, in their school's cafeteria.

Main Outcome Measure:

Each speech sample was rated (7-point Likert scale) by each student rater on a variety of personality characteristics based on the “Big Five” personality factors.

Results:

A factor analysis of the items revealed a two-factor solution, although the factors were highly negatively correlated. No significant differences were found between ratings for the CP sample and the control sample for either factor scale (ANOVA, p = .93; p = .67). Similarly, when the two factors were combined to form a single factor, no significant differences were found between the ratings for the CP sample and the control sample (ANOVA, p = .79).

Conclusions:

Overall, it does not appear that children differentially associated personality characteristics based on speech to children with repaired CP versus unaffected children, in the absence of visual input.
Comparison of Nasopharyngeal Growth between Patients with Clefts and Noncleft Controls
Authors: Wada Takeshi , Satoh Koichi , Tachimura Takashi , Tatsuta Unai
Published: October 2018

Objective:

This study was a comparison of the cephalometric growth characteristics of the nasopharyngeal structures between UCLP and noncleft controls.

Method:

Eighty patients with complete unilateral cleft lip and palate (UCLP group) and 82 noncleft controls (NCC group) were assigned to four develop mental stages (i.e., stage 1, at 4 years; stage 2, at 8 years; stage 3, at 12 years; and stage 4, at 17 years of age). Measurements on the anteroposterior and the vertical dimensions were derived from reference lines and points of nasopharyngeal structures on the lateral cephalograms.

Results:

The results showed that there were no growth differences between the two groups at any stages in the regions of cranial base and cervical vertebrae, and that growth of the posterior maxilla in the UCLP group was significantly less at any stage in both A-P and vertical dimensions than in the NCC groups. As well, the nasopharyngeal triangle (Ho-At-PMP) in the groups showed almost parallel increase with stage, though with short vertical dimension in the UCLP group, and the soft palate length in the UCLP group was significantly less at stages 2, 3, and 4 compared to that in the NCC group. The adequate ratio (soft palate length/pharyngeal depth) in the UCLP group tended to decrease and was significantly less at stage 4 compared to that in the NCC group.

Conclusions:

These results indicate that the growth of the cranial base and the upper cervical vertebrae is independent of the effect of clefts or of surgeries on clefts, and that the growth inhibition at the posterior maxilla results in morphologic disharmony of upper nasopharyngeal structures. This could be a potential factor for the reappearance of velopharyngeal incompetence at a later age.
Placement of Endosseous Implants in Grafted Alveolar Clefts
Authors: Kearns Gerard , Perrott David H. , Sharma Arun , Kaban Leonard B. , Vargervik Karin
Published: October 2018

Objective:

The purpose of this study was to determine the optimal timing for placement and to evaluate short- and long-term outcomes of endosseous implants in bone-grafted alveolar clefts.

Design:

Fourteen patients who underwent alveolar cleft bone grafting (ACBG) and closure of an oronasal fistula followed by restoration of the missing lateral incisor tooth using endosseous implants (EI) were studied. The oronasal fistulae were closed using local flaps, and the alveolus was grafted with fresh autogenous iliac marrow. Endosseous implants were placed a minimum of 4 months following ACBG. The average age at ACBG was 20.35 years (range, 12–65 yr), and at implant placement 22.2 years (range, 15–66 yr). It was necessary to regraft the alveolar cleft region in six patients to provide adequate bone volume for implant placement. Those who required alveolar regrafting had an increased mean interval between the initial ACBG and planned implant placement compared to the patients with adequate bone available for implant placement 26.4 months (range, 4–46 mo) versus 15.75 months (range, 4–36 mo).

Results:

Twenty-nine implants were placed in 14 patients, 9 outside of the cleft region and 20 in grafted alveolar clefts. Eighteen of 20 (90%) implants in the bone-grafted alveolar clefts have been successfully restored. The mean followup after implant placement was 39.1 months (range, 1–54 mo), and after restoration 28.5 months (range, 1–47 mo).

Conclusions:

Endosseous implants can be placed in bone-grafted alveolar clefts. Consideration should be given to the adequacy of graft volume and ridge morphology at the time of ACBG. The interval between ACBG and implant placement is important. The greater the interval beyond 4 months, the more likely there will be inadequate bone volume to accept an implant.
An Evaluation of a Noninvasive Method of Assessing Alveolar Bone Levels in an Experimental Model of Cleft Lip and Palate
Authors: Lawson Robert B. , Jones Malcolm. L.
Published: October 2018

Objective

The purpose of this study was to evaluate an ultrasound mucous-membrane-thickness-measuring device as a noninvasive, nonionizing alternative to radiography in the assessment of alveolar bone grafting.

Design

This was a prospective experimental study on porcine cadaver models.

Method

An ultrasound technique (Krupp SDM) was assessed ex vivo on three specially developed porcine cadaver models in comparison to radiography and ridge-mapping. Direct measurement of the mucosal thickness provided a standard for comparison. In each model, 30 measurement sites were identified for comparison of the techniques.

Results

All measurement techniques demonstrated clinically acceptable re-producibility. Of the clinical measurement techniques, radiography proved to be the most reliable, showing a small, nonsignificant statistical difference from direct measurement. Both the ultrasound technique and ridge-mapping showed significant tendencies to underestimate mucosal thickness that became greater with increasing mucosal thickness. At sites where mucosal thickness was less than 6 mm, the ultrasound technique underestimated mucosal thickness by 0.6 mm on average. At sites where mucosal thickness exceeded 6 mm, ultrasonic artifacts rendered the ultrasound technique unreliable.

Conclusion

The ultrasound technique could prove to be a useful clinical adjunct to radiography in the assessment of alveolar bone grafts, but in this particular application, care should be taken when using it to assess deeper alveolar defects.
Velopharyngeal Function in Nonsyndromic Cleft Palate: Relevance of Surgical Technique, Age at Repair, and Cleft Type
Authors: Marrinan Eileen M. , Labrie Richard A. , Mulliken John B.
Published: October 2018

Objective

The goal of this study was to determine the relative importance of surgical technique, age at repair, and cleft type for velopharyngeal function.

Design

This was a retrospective study of patients operated on by two surgeons using different techniques (von Langenbeck and Veau-Wardill-Kilner [VY]) at Children's Hospital, Boston, MA.

Patients

We included 228 patients who were at least 4 years of age at the time of review. Patients with identifiable syndromes, nonsyndromic Robin sequence, central nervous system disorders, communicatively significant hearing loss, and inadequate speech data were excluded.

Main Outcome Measure

Need for a pharyngeal flap was the measure of outcome.

Results

Pharyngeal flap was necessary in 14% of von Langenbeck and 15% of VY repaired patients. There was a significant linear association (p = .025) between age at repair and velopharyngeal insufficiency (VPI). Patients with an attached vomer, soft cleft palate (SCP), and unilateral cleft lip/palate (UCLP) had a 10% flap rate, whereas those with an unattached vomer, hard/soft cleft palate (HSCP), and bilateral cleft lip/palate (BCLP) had a 23% flap rate (p = .03). Age at repair was critical for the unattached-vomer group (p = .03) but was not statistically significant for the attached-vomer group (p = .52).

Conclusions

Surgical technique was not a significant variable either in aggregate or for the Veau types. Patients in the earliest repair group (8-10 months) were the least likely to require a pharyngeal flap. Early repair was more critical for HSCP and BCLP patients. There was no correlation between velopharyngeal insufficiency and Veau hierarchy. The attached vomer/levator muscle complex may be a more important predictor of surgical success than the anatomic extent of cleft.
Learning Disability, School Achievement, and Grade Retention among Children with Cleft: A Two-Center Study
Authors: Broder Hillary L. , Richman Lynn C. , Matheson Pamela B.
Published: October 2018

Objective

This study examined the prevalence of learning disability (LD), level of school achievement; and prevalence of grade retention by type of cleft and gender at two craniofacial centers.

Setting

The setting included two university-based craniofacial centers.

Design/Patients

Participants included 84 consecutively evaluated patients from one center who were matched by cleft type, age, and gender with 84 patients evaluated at the second center.

Outcomes

The outcomes included learning disability, school achievement, and grade retention.

Results

The results revealed that 46% of subjects with cleft had LD, 47% had deficient educational progress, and 27% had repeated a grade (excluding kindergarten) in school. Males with cleft palate only (CPO) had a significantly higher rate of LD than any other subject group. Males with CPO and females with cleft lip and palate (CLP) were more likely to repeat a grade in school than were females with CPO and males with CLP.

Conclusions

Children with cleft are at risk for learning disability, low school achievement, and grade retention.
Möbius Syndrome with Dental Involvement: A Case Report and Literature Review
Authors: Rizos Maria , Negrón Reinaldo J. , Serman Neill
Published: October 2018

The clinicopathological findings on a 17-year-old female with the Möbius/Moebius syndrome are reported. The signs and symptoms of this neuromus-cular condition include congenital bilateral or unilateral palsies of the facial and abducens cranial nerves and a broad scope of multisystem abnormalities. A case of unilateral deficiencies of cranial nerves VI and VII, congenital ectro-dactyly of toes, and multiple congenitally missing primary and permanent teeth is reported. A review of the literature reveals various ideas regarding the diversity of symptoms and the etiology of the syndrome. The purpose of this article is to report oral manifestations, such as congenitally missing teeth, associated with Möbius syndrome.
Nasalance Levels in the Speech of Normal Australian Children
Authors: Van Doorn Jan , Purcell Alison
Published: October 2018

Objective

Nasalance scores have been shown to depend on the regional dialect of English spoken. Australian cleft palate clinics are increasingly making use of the Nasometer as part of their evaluation of velopharyngeal inadequacy. There are, however, no normative data for Australian English available as reference information. The objective of this study, therefore, was to obtain comprehensive nasalance data for a large group of Australian children, aged 4 to 9 years, for two standard nasalance passages (Zoo Passage and Nasal Sentences) and to investigate any gender or age differences within that age range.

Participants

The participants were 245 children (123 female, 122 male) ranging in age from 4 years, 0 months, to 9 years, 3 months. The children were recruited from a variety of schools and preschools across the Sydney metropolitan region. The children all spoke Australian English, and their hearing, articulation skills, and speech resonance were within normal limits.

Method

Mean nasalance scores were obtained for two speech passages that are used as standards for Nasometer testing (Zoo Passage and Nasal Sentences). In addition, the nasalance data were analyzed for any gender and age dependence, using separate analyses of variance for each speech passage. Five consecutive age groups were used to examine age dependence (4-, 5-, 6-, 7-, and 8-year-old children).

Results

A mean score of 13.1 (SD, 5.9) was obtained for the Zoo Passage, and a mean of 59.6 (SD, 8.1) for the Nasal Sentences. The analysis of variance results indicated that, at a probability level of p < 0.01, there was no statistically significant age or gender dependence for either speech passage.

Conclusion

These normative nasalance data for children who speak Australian English will provide important reference information for clinicians who assess nasality disorders in cleft palate clinics in Australia.
Intraoral Pressure and Velopharyngeal Function
Authors: Mayo Robert , Warren Donald W. , Zajac David J.
Published: October 2018

Objective

The objective of this study was to determine the influence of velopharyngeal (VP) inadequacy on respiratory speech compensations.

Design

The pressure-flow technique was used to measure pressure, airflow, and timing variables associated with VP closure during the production of the initial plosive consonant /p/ in a series of the utterance “papa.”

Setting

The study was conducted in the speech and breathing laboratory of the UNC Craniofacial Center.

Participants

Eighty-two subjects with cleft lip and/or palate were assessed. The subjects were divided into two groups, those with adequate VP closure (VP size <.010 cm2) and those with inadequate VP closure (VP size >0.10 cm2). The adequate group was comprised of 62 subjects, and 20 subjects were categorized as inadequate.

Results

Peak intraoral pressure decreased in the inadequate group, but the difference was not significant. Nasal airflow increased (p < .01), but duration of the pressure pulse was the same for both groups. The area under the pressure curve decreased for the inadequate group (p = .04).

Conclusion

These data contrast with previously reported published data using /p/ in the utterance “hamper.” This suggests that phonetic context influences the compensatory response to velopharyngeal inadequacy. Additionally, while the findings are somewhat similar to studies that involved noncleft subjects whose oral airway was suddenly vented during the production of /p/, there is enough difference to suggest that learning also affects the compensatory outcome.
Scar Tissue Distribution on Palates and its Relation to Maxillary Dental Arch Form
Authors: Ishikawa Hiroyuki , Nakamura Shinji , Misaki Kouichi , Kudoh Motonori , Fukuda Hiroshi , Yoshida Shigemitsu
Published: October 2018

Objective

This study investigated the relationship between maxillary dental arch form and distribution of postsurgical scar tissue on previously denuded bone in isolated cleft palate patients.

Method

The palatal blood flow of 21 Japanese isolated cleft palate patients (6 males, 15 females) was examined by laser doppler flowmetry to determine the scar tissue areas. All had undergone pushback operations for palatal repair at around 18 months of age. Tissue blood flow was examined at a time ranging from 11 years, 5 months to 19 years, 9 months of age. To evaluate the maxillary dental arch form, dental casts obtained at the start of orthodontic treatment (a mean age of 8 years, 4 months) were analyzed.

Results/Conclusions

Scar tissue distribution in the 21 cases was classified into five types. Characteristic features in the maxillary dental arch form were found in each of the five types according to the extent of the scar tissue. It was evident that the severity of the maxillary dental arch constriction was closely related to the scar tissue distribution on palates.
Evaluation of Pharyngeal and Laryngeal Structure and Function in Patients with Oculo-Auriculo-Vertebral Spectrum
Authors: D'antonio Linda L. , Rice Reginald D. , Fink Sandra C.
Published: October 2018

Objective

This study described the occurrence and expression of pharyngeal and laryngeal anomalies in a population of patients with oculo-auriculo-vertebral spectrum (OAVS).

Design

Cross-sectional study using chart review, physical examination, and endoscopic evaluation of the pharynx and larynx, and perceptual and aerodynamic evaluation of speech for assessment of vocal tract function.

Setting

Academic tertiary referral center.

Patients

Chart review of 41 patients with a diagnosis of OAVS and clinical evaluation of 23 of the patients identified in the initial chart review, ranging in age from 9 months to 17 years.

Main outcome Measures

Data from chart review pertaining to airway and speech symptoms. Physical examination and endoscopic studies of pharyngeal and laryngeal structure and function and perceptual and aerodynamic evaluation of speech, resonance, and voice.

Results

Chart review showed a high occurrence of indicators of pharyngeal and laryngeal abnormalities such as velopharyngeal inadequacy and airway obstruction. Clinical evaluations documented a variety of abnormalities in pharyngeal and laryngeal structure and function as well as impairment in speech articulation, resonance, and voice. Presence and severity of pharyngeal and laryngeal abnormalities were not correlated with the severity of expression of the spectrum based on mandibular morphology.

Conclusions

Oculo-auriculo-vertebral spectrum appears to be associated with a high occurrence of structural and functional abnormalities of the pharynx and larynx that may contribute to increased risk of airway obstruction, communication impairment, and morbidity.
Quantitative Description of the Morphology of the Human Palate by a Mathematical Equation
Authors: Ferrario Virgilio F. , Sforza Chiarella , Schmitz Johannes H. , Colombo Anna
Published: October 2018

Objective

To derive a three-dimensional mathematical description of normal human hard tissue palatal size and shape.

Methods

The maxillary dental casts of 30 adolescents free from respiratory problems, who had a complete (28 teeth) permanent sound dentition with normal occlusion, were studied. The x, y, z coordinates of several standardized palatal and dental landmarks were obtained with a computerized three-dimensional digitizer. Palatal landmarks were used to derive a mathematical equation of palatal shape in the frontal and sagittal planes. Palatal width, length, frontal and sagittal heights, and sagittal slope, as well as dental arch transverse and anteroposterior dimensions, were computed.

Results

Neither the size nor the shape of the palate was significantly influenced by gender. Only the intercanine distance was larger (p < .025) in males than in females.

Conclusions

Data collected in the present investigation could represent a first database for the quantitative description of normal human palatal morphology in subjects with a complete permanent dentition.
Adverse Outcomes following Endoscopic Repair of a Fetal Cleft Lip Using an Ovine Model
Authors: Stelnicki Eric J. , Vanderwall Karen , Hoffman William Y. , Szabo Zoltan , Harrison Michael R. , Foster Rob , Longaker Michael T.
Published: October 2018

Objective

The purpose of this study was to determine if endoscopic techniques could be used to repair an epithelialized lip cleft with accuracy and with an outcome comparable to fetuses treated through an open hysterotomy.

Interventions and Results

In contrast to previous open fetal cleft lip repairs in the same model, none of the five fetuses reported here had a good aesthetic result. Although there was no evidence of scar histologically, the edges of the lip were poorly approximated. The epithelial lining and underlying dermis of the wound margins were notably inverted. The orbicularis oris muscle, which had been reapproximated, appeared thin and hypoplastic. Most of the vermilion elements were poorly aligned, and in one animal, there was a complete dehiscence of the repair.

Conclusions

In a more representative model of cleft lip that is not an acute lip wound, in utero endoscopic suture repair of the ovine lip gave a poor result using current technology. Only a meticulously performed, multilayered, open repair of a cleft appears to give a good cosmetic and functional outcome. Further studies to improve the endoscopic repair as our technology advances are therefore warranted.
Transmission of Mutans Streptococci between Mothers and Children with Cleft Lip and/or Palate
Authors: De Soet Johannes J. , Bokhout Birgitta , Buijs Jos F. , Van Loveren Cor , De Graaff Johannes , Prahl-Andersen Birte
Published: October 2018

Objective

The aim of this study was to investigate the transmission of Streptococcus mutans between children with cleft lip and/or palate and their mothers.

Design

Saliva samples of 21 mother-child pairs were collected and cultured on plates containing a selective growth medium for mutans streptocci. At least five separate colonies of each colony morphotype were isolated. A polymerase chain reaction (PCR) with randomly chosen primers was used to type the isolates.

Results

The number of morphotypes and PCR types was significantly lower in the children than in the mothers. Significant correlations were found between the number of morphotypes and PCR types, in the children as well as in the mothers. In only 38% of the mother-child pairs were the same PCR types found in mother and child.

Conclusions

This suggests that S. mutans had been transmitted from mother to child in one-third of the population studied. No correlations were found among the number of colony-forming units, the number of colony-colony-morphotypes, and the number of PCR types of the mothers and transmission. Similar PCR types in mother and child were found significantly more often in children who had more than one PCR type. The results indicate that transmission of S. mutans from mother to child is not frequent in children with oral cleft. This may have consequences for preventive treatment of cleft lip and/or palate children and their mothers.
Oral Health Status of Russian Children with Unilateral Cleft Lip and Palate
Authors: Turner Clara , Zagirova Asiya , Frolova Larisa , Courts Frank J. , Williams William N.
Published: October 2018

Objective

To evaluate the oral and dental health of Russian children who underwent Frolova primary palatoplasty.

Design

Eighty-nine children (62 boys and 27 girls; age range, 5 to 9 years) who had undergone repair of unilateral cleft lip and palate were included in this oral/dental evaluation. Factors such as stage of dentition; oral hygiene; carious, missing and restored teeth; and occlusion were recorded.

Results

Twenty-six of the 89 patients presented with a palatal fistula. Oral hygiene of patients without a palatal fistula was significantly better than that of patients with a fistula (p < .01). Forty-five percent of patients had eight or more decayed teeth. Significantly more patients with palatal fistulae had class II soft tissue facial profiles than those without palatal fistulae. Anterior crossbites were found in 48% of patients, unilateral posterior crossbites in 58%, and bilateral posterior crossbites in 12%.

Conclusions

The high percentage of poor oral hygiene and dental caries found in this group of patients is likely due to the general unavailability of dental hygiene products and the high cost of these products when available. In addition, it seems there is limited understanding by parents of the importance of dental hygiene and appropriate diet in preventing dental disease.
Progress toward Understanding Craniofacial Malformations
Authors: Nuckolls Glen H. , Shum Lillian , Slavkin Harold C.
Published: October 2018

Significant advances in the study of the human face have revealed the genetic and gene-environment bases of numerous common and rare craniofacial disorders. Classification of craniofacial malformations based on clinical phenotypes is sometimes quite different from the genetic findings of patients. Different mutations in a single gene can cause distinct syndromes, and mutations in different genes can cause the same syndrome. The extracellular signaling molecule SHH, fibroblast growth factor receptors, and transcription factors GLI3, MSX2, and TWIST are discussed as examples of molecules involved in interrelated signal transduction networks regulating craniofacial development. Progress in the understanding of normal and abnormal craniofacial development, through the study of morphoregulatory signaling pathways, has benefited from multifactorial approaches recommended 40 years ago at the National Institute of Dental Research–sponsored landmark Gatlinburg Conference. The utilization of biochemistry, protein structure analyses, tissue culture, and animal model systems for developmental genetics has resulted in remarkable scientific advances. The evolutionary conservation of morphoregulatory pathways has revealed the homology of genes associated with human craniofacial malformations and their counterparts that regulate the morphogenesis of fruit flies. The continued investments in basic, translational, and patient-oriented research regarding normal and abnormal craniofacial development will translate into substantial Improvements in the prevention, diagnosis, and treatment of craniofacial diseases and disorders.