bluuebunny/crossref_metadata_2025 huggingface dataset:
Facial Clefting and Psychiatric Diseases: A Follow-Up of the Danish 1936–1987 Facial Cleft Cohort
Authors: Christensen Kaare , Mortensen Preben Bo
Published: October 2018

Objective

This study assesses whether patients with facial cleft have an increased risk of psychiatric diseases.

Design

Through the Danish Facial Cleft Database, patients with facial cleft born between 1936 and 1987 in Denmark were identified, and the admission pattern for these patients with facial cleft was available for the period 1969 through 1993 through the Danish Psychiatric Central Registry.

Participants

A total of 6,462 patients with facial cleft followed up for a total of 127,068 person-years.

Main outcome Measure

Hospitalization for psychiatric diseases. The expected number of admissions for the cleft population was calculated by multiplication of the observed person-years with admission rates for Denmark stratified for sex, 1-year age group, and 1-year calendar period.

Results

A total of 284 patients with facial cleft (4.4%) were hospitalized for psychiatric diseases. The relative risk of hospitalization was 1.65 (95% confidence interval 1.3 to 2.0) for patients with isolated cleft palate (CP) and 1.15 (95% confidence interval 0.99 to 1.29) for patients with cleft lip ± cleft palate (CL[P]). The overall risk estimates were above unity for both CP and CL(P) in all major diagnosis groups, neurosis and autism (for CL[P]) being the only exceptions. The excess risk was not accounted for by patients with known associated anomalies/syndromes.

Conclusion

The risk of hospitalized mental disorders in general is increased in patients with CP but not to any substantial degree in patients with CL(P). Both groups had an increased risk of mental retardation and substance abuse, but the risk for schizophrenia or bipolar illness was not statistically significantly increased, compared with the background population. Further, our data provide no evidence that the psychosocial stressors associated with CL(P) and its treatment have any substantial impact on the risk for hospitalized mental illness.
Perceptual Evaluation of Speech and Velopharyngeal Function in Children with and without Cleft Palate and the Relationship to Nasal Airflow Patterns
Authors: Dotevall Hans , Lohmander-Agerskov Anette , Ejnell Hasse , Bake Björn
Published: October 2018

Objectives

The aim was to study the relationship between perceptual evaluation of speech variables related to velopharyngeal function and the pattern of nasal airflow during the velopharyngeal closing phase in speech in children with and without cleft palate.

Participants

Fourteen children with cleft lip and palate or cleft palate only and 15 controls aged 7 and 10 years. All were native Swedish speakers.

Method

Three experienced listeners performed a blinded perceptual speech evaluation. Nasal airflow was transduced with a pneumotachograph attached to a nasal mask. The duration from peak to 5% nasal airflow, maximum flow declination rate, and nasal airflow at selected points in time during the transition from nasal to stop consonants in bilabial and velar articulatory positions in sentences were estimated. The analysis was focused on the perceptual ratings of “velopharyngeal function” and “hypernasality.”

Results

A strong association was found between ratings of “velopharyngeal function” and “hypernasality” and the pattern of nasal airflow during the bilabial nasal-to-stop combination /mp/. Both the sensitivity and specificity were 1.00 for the bilabial temporal airflow measure in relation to ratings of “velopharyngeal function.” The nasal airflow rate during /p/ in /mp/ had a sensitivity of 1.00 and specificity of 0.92 to 0.96 in relation to ratings of “hypernasality.”

Conclusion

Assessment of the nasal airflow dynamics during the velopharyngeal closing phase in speech presents quantitative, objective data that appear to distinguish between perceptually normal and deviant velopharyngeal function with high sensitivity and specificity.
Temporal Characteristics of Aerodynamic Segments in the Speech of Children and Adults
Authors: Zajac David J. , Hackett Alyssa M.
Published: October 2018

Objectives

The primary purpose of this study was to determine the temporal characteristics of aerodynamic segments in the normal speech of children and adults without cleft palate. A secondary objective was to determine the withinspeaker variability of the segments.

Method

Speakers consisted of 46 children aged 6 to 8 years, 41 older children aged 11 to 12 years, and 41 adults aged 18 to 37 years (total n = 128) who repeated the word “hamper” during continuous utterances. The pressure-flow method was used to determine the duration of six segments of the oral air pressure and nasal airflow pulses associated with the /mp/ sequence. Descriptive statistics, including coefficients of variation (COV), were computed for each segment as a function of age and sex of the speakers. Analysis of variance (ANOVA) procedures were used to determine the effects of age, sex, or both on the temporal variables.

Results

ANOVAs indicated statistically significant main effects (p < .008) for age on five of the six temporal measures and for sex on three of the six measures. Five of the six COVs were also statistically significant for age. There were no statistically significant interactions between speaker age and sex for any measure.

Conclusions

The results indicate distinct patterns of timing for aerodynamic segments of speech produced by children and adults. Overall, adults exhibited less temporal variability than children. The generally longer and more variable segments produced by children suggest diagnostic and treatment implications relative to speakers with velopharyngeal dysfunction.
Gross Unilateral Abnormalities of the Velum and Pharynx
Authors: Mehendale Felicity V. , Sommerlad Brian C.
Published: October 2018

Objective

Velopharyngeal incompetence because of gross unilateral velopharyngeal hypoplasia is rare, particularly in patients with no significant hemifacial microsomia or facial asymmetry. We describe the abnormal anatomy and treatment of three patients with gross congenital velopharyngeal asymmetry.

Results and Conclusions

Following surgery, all three patients showed a good improvement in velopharyngeal function and speech. Nasendoscopy and lateral videofluoroscopy were important in confirming the abnormal anatomy and pathology and in evaluating the degree of movement of the affected side of the velum and pharyngeal walls as well as the size and location of the defect. Muscles from the normal side were radically dissected and mobilized across the midline to reconstruct the hypoplastic hemivelum. Mucosal lengthening was achieved by suturing the normal mucosa to the mucosa of the hypoplastic hemivelum and the lateral pharyngeal wall or by insetting a posterolateral pharyngeal flap (modified Moore pharyngoplasty) into the nasal surface of the hypoplastic side.
Extensive Facial Clefting in a Patient with Goltz Syndrome: Multidisciplinary Treatment of a Previously Unreported Association
Authors: Ascherman Jeffrey A. , Knowles Sean L. , Troutman Kenneth C.
Published: October 2018

Objective

Goltz syndrome is a rare, X-linked dominant, multisystem disorder found almost exclusively in female patients. Although the cutaneous features predominate in most reports, characteristic abnormalities are also frequently present in the musculoskeletal system and facial region. We report a female infant born with a severe form of Goltz syndrome that included an extremely wide facial cleft, an abnormality not previously reported in a patient with this disorder. Her management demonstrates the advantages of a multidisciplinary approach to effectively care for patients with severe craniofacial abnormalities.
Change in Levator Veli Palatini Muscle Activity for Patients with Cleft Palate in Association with Placement of a Speech-Aid Prosthesis
Authors: Tachimura Takashi , Nohara Kanji , Fujita Yoshinori , Wada Takeshi
Published: October 2018

Objective:

The purpose of this study was to examine whether a speech-aid prosthesis normalizes the activity of the levator veli palatini muscle for patients with cleft palate who exhibit velopharyngeal incompetence.

Design:

Each subject was instructed to produce repetitions of /mu/, /u/, /pu/, /su/, and /tsu/ and to blow with maximum possible effort. Electromyographic (EMG) activity of the levator veli palatini muscle was recorded with and without a hybrid speech-aid prosthesis in place.

Participants:

The participants were five patients with repaired cleft palate who were routinely wearing a hybrid speech-aid prosthesis.

Results:

With the prosthesis in place, the mean value of levator activity changed positively in relation to oral air-pressure change during blowing. Differences in levator activity in relation to speech samples were similar to those in normal speakers. With the prosthesis in place, levator activity for speech tasks was less than 50% of the maximum levator activity for all subjects. The findings were similar to those reported previously for normal speakers.

Conclusion:

Placement of the prosthesis changed EMG activity levels of the levator veli palatini muscle to levels that are similar to normal speakers. It is possible that, with the increase in the differential levator activity between speech and a maximum force task, the velopharyngeal mechanism has a greater reserve capacity to maintain velopharyngeal closure compared with the noprosthesis condition.
Effects of Cheiloplasty on Maxillary Dental Arch Development in Infants with Unilateral Complete Cleft Lip and Palate
Authors: Huang Chiung-Shing , Wang Wein-I , Liou Eric Jein-Wein , Chen Yu-Ray , Chen Philip Kao-Ting , Noordhoff M. Samuel
Published: October 2018

Objective:

To identify and analyze quantitatively the development of the maxillary dental arch before and after cheiloplasty.

Design:

Prospective, longitudinal study of maxillary dental arch development at age of 1, 3, 6, and 12 months.

Setting:

All patients were treated at a university hospital craniofacial center.

Patients:

Twenty-seven infants with nonsyndromic, unilateral complete cleft lip and palate.

Intervention:

Millard's rotation-advancement cheiloplasty was performed between the ages of 3 and 4 months.

Results:

The anterior portion of the nonclefted segment (I-G), anterior ridge length of the nonclefted segment (I-C), and anterior ridge length of the clefted segment (L-C′) continuously increased from 1 to 12 months of age. The anterior cleft width (G-L), anterior arch depth (I⊥CC), anterior basal angle (∠GC-CC′), and anterior arch curature angle (∠GIC) continuously decreased after the cheiloplasty.

Conclusions:

Cheiloplasty could mold the anterior portion of the maxillary dental arch palatally by exerting continuous pressure.
Relationship between Primary Palatal Form and Maxillofacial Growth in Japanese Children with Unilateral Cleft Lip and Palate: Infancy to Adolescence
Authors: Honda Yasuo , Suzuki Akira , Nakamura Norifumi , Ohishi Masamichi
Published: October 2018

Objective:

The purpose of this retrospective study was to evaluate the relationship between the severity of maxillary defects and maxillofacial growth from infancy to adolescence in children with complete unilateral cleft lip and palate (UCLP).

Subjects:

Thirty-one Japanese children with UCLP who had received cheiloplasty, palatoplasty, and orthodontic treatments at the Kyushu University Dental Hospital were selected.

Materials and Methods:

A three-dimensional laser scanner was used to measure maxillary dental casts taken at cheiloplasty. Surface areas and volumes of the palatal tissue were calculated. Lateral cephalographs that were taken both at cheiloplasty and 15 years of age or over (average age 16.5 years) were traced and digitized. Angular and linear measurements were calculated from the x, y-coordinates. Using correlation analyses, defects in the palatal tissue were compared with maxillofacial morphology both at cheiloplasty and in adolescence or maxillofacial growth during that time span.

Results:

(1) The surface areas or volumes of the palatal tissue were significantly correlated with the following maxillofacial measurements at infancy and adolescence: Or-depth; A-depth; PNS-height; ANS-height; N-Ba; S-N; S-Ba; N to P.P; A'-Ptm’; Cd-Pog; Cd-Gn; Cd-G; and the N-S-Ba, S-N-A, and A-N-B angles. (2) They were also significantly correlated with the following maxillofacial growth measures from infancy to adolescence: A-depth, S-Ba, and the N-S-Ba and S-N-A angles. Only the N-S-Ba angle showed a negative correlation.

Conclusion:

The patient who had more palatal tissue at cheiloplasty showed better maxillofacial growth.
Cervical Root Resorption in Two Patients with Unilateral Complete Cleft of the Lip and Palate
Authors: De Moor Roeland J.G. , De Vree Hilde M. , Cornelis Carol , De Boever Jan A.
Published: October 2018

Objective:

Case reports on cervical root resorption in patients with complete cleft of the lip and palate are scarce. Moreover, cervical root resorption is sometimes diagnosed several years after a combined orthodontic and surgical therapy in patients with cleft. In this article a review of the literature is presented, and a description and the management of the cervical lesion is provided. An additional objective of this report is to advocate for alveolar bone grafting before the age of 12 years while the vulnerable cervical region of the teeth adjacent to the cleft defect is still protected by a thick layer of bone.
Photogrammetric Assessment of the Soft Tissue Profile in Unilateral Cleft Lip and Palate
Authors: Bearn David R. , Sandy Jonathan R. , Shaw William C.
Published: October 2018

Objective

To describe the soft tissue profile of children with unilateral cleft lip and palate from profile photographs and assess the reliability of this method.

Design

Retrospective analysis of prospectively collected data.

Setting

A national study in the United Kingdom.

Subjects

Caucasian children born in the United Kingdom between April 1, 1982, and March 31, 1984, and aged between 12 and 14 years at data collection. A cleft side and noncleft side profile photograph was available for each of 175 children.

Method

Seven angular measurements were made using Dentofacial Planner Plus software, and the profile appearance was rated by an expert panel.

Results

Reliability for repeated measurement of the same photograph was acceptable for all but li-sms-pgs (labiomental fold). Similar levels of reliability were found when comparing measurements from the cleft side and non-cleft side photograph. Logistic regression showed that the variables nst-sn-ls (nasolabial angle), sss-ns-pgs (maxillary prominence), and gs-prn-pgs (facial convexity) were associated with the profile score.

Conclusions

Soft tissue profile analysis from photographs is reliable and robust under a range of conditions. Soft tissue profile measurements are associated with panel ratings of profile.
Ethical and Scientific Decision Making in Distraction Osteogenesis
Authors: Shaw W.C. , Mandall N.A. , Mattick C.R.
Published: October 2018

Objective

To propose a prospective registry for distraction osteogenesis research.

Results

The search strategy identified 82 reports, of which 11 were review articles, 17 were animal studies, 5 were theoretical models, and 49 were case reports/series.

Conclusions

So far, the literature concerning distraction osteogenesis does not allow reliable choices to be made on the most appropriate form of distraction osteogenesis in different clinical decisions or whether it is superior to osteotomy or nontreatment. A prospective registry is proposed to hasten critical appraisal of distraction osteogenesis.
Position of the Hyoid Bone in Cleft Lip, Alveolus, and Palate: Variation of Normal Anatomy or Sign Accompanying the Malformation?
Authors: Kaduk Wolfram M.H. , Grabowski Rosemarie , Gundlach Karsten K.H.
Published: October 2018

Objective

To compare the hyoid bone position in patients with clefts of lip, alveolus, and palate with orthodontic patients with no cleft.

Design

Retrospective cephalometric long-term study.

Patients

Lateral cephalograms of 23 patients with a complete unilateral cleft were compared with those of 24 orthodontic patients with no cleft with an Angle class 1 occlusion. Twenty-five of the patients were girls, and 22 were boys. The age of the patients with cleft at the beginning of the study was 8.1 ± 0.7 years and at the end 14.8 ± 0.6 years. The patients without cleft were 8.4 ± 0.8 years old at the beginning and 14.4 ± 0.6 years at the end of the study.

Main Outcome Measures

Cephalometric analysis was used to determine the skull growth pattern as well as the positions of the head and hyoid bone twice in all 47 persons.

Results

With increasing age of the patients, the skull growth pattern changed from vertical to horizontal growth, but no significant differences were found between patients with and without cleft. Head position did not differ significantly between patients with and without cleft. However, hyoid bone position differed significantly. The hyoid in patients with cleft was located more caudally and more anteriorly than in patients without cleft.

Conclusion

The hyoid bone in a subset of north German adolescents with complete unilateral cleft lip and palate was found to be caudal and anterior, compared with a control group.
Phonological Development of Toddlers with Unilateral Cleft Lip and Palate who were treated with and without Infant Orthopedics: A Randomized Clinical Trial
Authors: Konst Emmy M. , Rietveld Toni , Peters Herman F.M. , Prahl-Andersen Birte
Published: October 2018

Objective

To investigate the phonological development of toddlers from 2 to 3 years of age with complete unilateral cleft lip and palate (UCLP) treated during the first year of life with and without infant orthopedics (IO).

Design

In a randomized clinical trial (Dutchcleft), two groups of children were followed up: one treated with IO (IO group) and another that did not receive IO (non-IO group). Phonological skills were analyzed at 2, 2.5, and 3 years of age using a system for assessing phonological development of Dutch children (Fonologische Analyse van het Nederlands: FAN). The analysis included number of acquired consonants, order of phonological development, use of phonological processes, and occurrence of nasal escape.

Patients

Criteria for inclusion were complete UCLP, no soft tissue bands, no other malformations, parents fluent in Dutch, birth weight of a minimum of 2500 g, and gestation time of a minimum of 38 weeks.

Interventions

IO treatment based on a modified Zurich approach was started within 2 weeks after birth and used until soft palate closure at 12 months of age. Children in the non-IO group visited the clinic for an extra check-up at 6 weeks as well as before and after lip repair and soft palate closure. All other interventions were the same across groups.

Results

Phonological development of most 2.5-year-old IO children was normal or delayed. Most children in the non-IO group followed an abnormal developmental pattern. At age 3, the children in the IO group had acquired more initial consonants. There were no group differences in the use of phonological processes or the occurrence of nasal escape.

Conclusions

Children treated with IO during their first year of life followed a more normal path of phonological development between 2 and 3 years of age.
Aerodynamic and Cephalometric Analyses of Velopharyngeal Structure and Function following Re-Pushback Surgery for Secondary Correction in Cleft Palate
Authors: Nakamura Norifumi , Ogata Yuko , Sasaguri Masaaki , Suzuki Akira , Kikuta Rumiko , Ohishi Masamichi
Published: October 2018

Objective

The goal of this study was to clarify the efficacy of and indication for re-pushback surgery as secondary treatment for cleft palate.

Participants

Fifteen patients treated by re-pushback surgery involving intravelar veloplasty (IVV) with buccal mucosal grafting on the nasal surface and followed up more than 6 months were enrolled in this study.

Main Outcome Measures

Pre- and postoperative velopharyngeal functions were analyzed by perceptual voice analysis, blowing ratio, and nasalance scores during phonation of /i/ and /tsu/. Cephalometric analysis was used to evaluate the relationship between velopharyngeal structure and the outcome of re-pushback surgery. Control data were obtained from the longitudinal files of normal 10-year-old children in Kyushu University Dental Hospital.

Results

Eight of 15 patients obtained complete velopharyngeal closure (complete group), five patients improved remarkably (improved group), and no effective result was seen in two patients (ineffective group). Nasality disappeared or remarkably improved after the operation in 13 patients. Effective surgical results were found in 86.7% of the patients. Partial flap necrosis was seen in two patients in whom re-pushback surgery was performed using mucosal palatal flaps instead of mucoperiosteal flaps. Preoperative velar length and the length/depth ratio of the re-pushback group were significantly smaller than the controls, but there was no difference after the operation. Furthermore, the preoperative length/depth ratio of the complete group (ranged more than 100%) was significantly greater than those of the other two groups (ranged less than 100%).

Conclusion

Re-pushback surgery by IVV with free mucous grafting on the nasal surface was effective in managing velopharyngeal incompetence secondarily, improving velopharyngeal structure and function.
Midface Distraction to Alleviate Upper Airway Obstruction in Achondroplastic Dwarfs
Authors: Elwood Eric T. , Burstein Fernando D. , Graham Leroy , Williams Joseph K. , Paschal Michelle
Published: October 2018

Objective

The use of midface distraction in patients with achondroplasia and upper airway obstruction secondary to midface hypoplasia has not been reported. In this report, we review the treatment of two patients with severe midface hypoplasia and obstructive sleep apnea secondary to achondroplasia using midface distraction osteogenesis.

Design, Setting, and Patients

Two patients with achondroplastic dwarfism and midface hypoplasia with airway obstruction were treated in a tertiary referral center for craniofacial disorders.

Results and Conclusions

Both patients had their tracheostomies decannulated after midface distraction of 25 mm. Midface distraction osteogenesis is useful to alleviate upper airway obstruction from midface hypoplasia seen in achondroplasia.
Nerve Growth Factor Receptor Immunolocalization during Human Palate and Tongue Development
Authors: Ono Takashi , Fischer-Hansen Birgit , Nolting Dorrit , KjÆr Inger
Published: October 2018

Objective

To investigate the temporospatial pattern of nerve growth factor receptor (NGFR) immunolocalization during human palatal closure.

Materials

Human palate and tongue tissues from 33 embryos/fetuses, 9 to 22 weeks of fertilization age.

Methods

Tissues were divided according to developmental stage and palatal development (before, during, and after closure) and then subjected to decalcification, paraffin embedding, serial sectioning, survey staining, and p75NGFR immunohistochemical staining.

Results

Specific temporospatial patterns of p75NGFR reactivity were observed; reactivity was intense in the soft tissue palatal shelves before and during palatal closure and was weaker in the palate after palatal closure. In the tongue, intense reactivity was seen throughout 9 to 22 weeks.

Conclusion

The observed patterns suggest that p75NGFR may enable the visualization of physiological events in palatal closure during normal human development.
Early Craniofacial Morphology and Growth in Children with Nonsyndromic Robin Sequence
Authors: Hermann N. V. , Kreiborg S. , Darvann T. A. , Jensen B. L. , Dahl E. , Bolund S.
Published: October 2018

Purpose

Craniofacial morphology and growth comparisons in children with untreated nonsyndromic Robin Sequence (RS) and a control group with unilateral incomplete cleft lip (UICL) in which the lip was surgically closed at 2 months of age.

Material

The 52 children (7 RS and 45 UICL) included in the study were drawn from a group representing all Danish cleft children born 1976 through 1981. The ages of the children were 2 and 22 months at the time of examination 1 and 2, respectively.

Method

The method of investigation was three-projection cephalometry. Craniofacial morphology was analyzed by means of linear, angular, and area variables. Growth at a specific anatomical location in a patient was defined as the displacement vector from the coordinate of the corresponding landmark at examination 1 to its coordinate at examination 2.

Results

The most striking findings in the RS group were markedly increased posterior maxillary width, increased width of the nasal cavity, short maxilla with reduced posterior height, short mandible, bimaxillary retrognathia, and severe reduction in size of the pharyngeal airway. The amount of facial growth was similar in the two groups; however, a tendency toward a more vertical growth direction was observed in the RS group.

Conclusion

Facial morphology in children with RS differed significantly from that of children with UICL at both 2 and 22 months of age. The magnitude of facial growth was similar in the two groups, whereas a tendency toward a more vertical facial growth direction was observed in the RS group.
Influence of Lip Repair on Craniofacial Morphology of Patients with Complete Bilateral Cleft Lip and Palate
Authors: da Silva Filho Omar Gabriel , Valladares Neto José , Capelloza Filho Leopoldino , de Souza Freitas JoséAlberto
Published: October 2018

Objective

The aim of this study was to compare two groups of adult male patients with complete bilateral cleft lip and palate (BCLP) on the basis of lateral cephalometric radiographs.

Patients

The first group of adult male patients with complete BCLP was comprised of 13 unoperated patients with an average age of 21 years. The second group was comprised of 14 patients with an average age of 21 years 7 months, who had been operated only on the lip prior to 2 years of age.

Design

The following measurements were evaluated: angle and length of cranial base; maxillary spatial positioning and length; mandibular spatial positioning; morphology and length; maxillomandibular relationship; vertical facial length; dental positioning; interdental arch relationship; and soft profile.

Results

The results suggest that lip repair has a significant influence on certain areas of the craniofacial complex, mainly the premaxilla and the upper incisors.

Conclusions

The most significant findings consequent to lip repair consisted of reduction of the premaxillary anterior projection and lingual tipping of the upper incisors. Retropositioning of the premaxilla, especially in the alveolar part, is a desired effect of lip repair in complete BCLP. Such effect on the projected premaxilla is usually beneficial, except when the exceedingly severe lip pressure, unfavorable growth pattern, or both retropositions the midface profile beyond acceptable sagittal limits.
Dental Anomalies of the Permanent Lateral Incisors and Prevalence of Hypodontia outside the Cleft Area in Complete Unilateral Cleft Lip and Palate
Authors: Lourenço Ribeiro Luciana , Teixeira das Neves Lucimara , Costa Beatriz , Ribeiro Gomide Marcia
Published: October 2018

Objective

To determine in complete unilateral cleft lip and palate (UCLP) subjects the characteristics (location, shape) of the cleft-side lateral incisor. The presence of a supernumerary tooth at the cleft side and the prevalence of hypodontia outside the cleft area were evaluated. A comparison was made of the shape of the cleft side lateral incisor to its contralateral incisor.

Setting

Hospital for Rehabilitation of Craniofacial Anomalies (HRCA), Sao Paulo, Brazil.

Patients

Orthopantomograms of 203 subjects with UCLP and without syndromes were chronologically selected from the HRCA data bank, within an age range of 5 to 10 years.

Outcome Measure

Orthopantomograms were analyzed by the same observer according to established criteria.

Results

There were no statistically significant differences between sexes for any of the criteria studied. The cleft-side lateral incisor was present in 50.2%, and it was more commonly located at the distal side (76.5%). The congenital absence of the cleft-side lateral incisor was observed in 49.8% of the sample, and its antimere was congenitally missing in 10.9%, this difference being statistically significant. The most commonly missing tooth outside the cleft area was the maxillary second premolar.

Conclusions

The high prevalence of hypodontia of the permanent lateral incisor in the cleft side showed that the cleft could play an important role in this absence. There were different patterns for the presence of the cleft-side lateral incisor.
Management of Infants with Pierre Robin Sequence
Authors: Wagener Silke , Rayatt Sukh S. , Tatman Andy J. , Gornall Peter , Slator Rona
Published: October 2018

Objective

Several methods of treating babies with Pierre Robin sequence have been described since the condition itself was first documented in 1923. The main aim of treatment has been to relieve upper airway obstruction. Treatment methods used range from positioning of the baby to invasive surgery. The aim of this article was to describe the assessment, treatment, and monitoring methods used for babies referred with Pierre Robin sequence (PRS).

Setting/Patients

From December 1995 to May 2000, 22 consecutive patients were admitted to Birmingham Children's Hospital with PRS. Their airway and nutritional status were assessed and continuously monitored.

Interventions

Treatment concentrated on the relief of airway obstruction with a nasopharyngeal airway (NPA) and nutritional support of the babies until they grew out of their respiratory and feeding difficulties.

Main Outcome Measures

Outcome measures were oxygen saturation, growth of the babies, and the need for surgery.

Results

All babies were managed successfully with an NPA and nutritional support. No baby required surgery, and the majority showed good weight gain.

Conclusion

Relieving airway obstruction by NPA is an effective and safe treatment for babies with PRS until they have grown out of their respiratory and feeding difficulties. It avoids the need for surgery and can be used on neonatal wards using the monitoring described.
Cephalometric Analysis of the Consolidation Phase following Bilateral Pediatric Mandibular Distraction
Authors: Hopper Richard A. , Altug Ayse T. , Grayson Barry H. , Barillas Ingrid , Sato Yuki , Cutting Court B. , McCarthy Joseph G.
Published: October 2018

Objective

The goal of the consolidation phase of mandible distraction is to maintain the improvement in maxillomandibular form and relationship while the generated tissue ossifies. During this period, external deforming forces can act on the healing generated bone. The purpose of this study was to describe the potential cephalometric changes that occur following pediatric bilateral mandibular distraction using external devices.

Design

Retrospective lateral superimposition cephalometric analyses.

Participants

Thirty-five cases of pediatric mandible distraction were reviewed. Seven of these cases were included in the study after exclusion criteria were applied. These cases represented a group with severe congenital dysmorphology and a mean device activation of 26.5 mm.

Main Outcome Measures

Changes in pogonion position, symphyseal plane rotation, mandible length, and mandible length relative to maxillary length during the 18 to 36 days of activation, the eight weeks of consolidation, and the 1-year period following removal of the distraction device were measured.

Results

All patients demonstrated variable changes in position of the mandible during the consolidation phase. The most common were retrusion of pogonion, a decrease in mandible length, and a clockwise rotation of the symphyseal plane. In some cases the changes that occurred during consolidation were greater than those that occurred on 1-year follow-up.

Conclusions

The consolidation phase of distraction osteogenesis is a dynamic phase and should not be assumed to be static. Multicenter use of this cephalometric technique would help to identify potential risk factors associated with postactivation changes.
Increased Fistula Risk following Palatoplasty in Treacher Collins Syndrome
Authors: Bresnick Stephen , Walker John , Clarke-Sheehan Noreen , Reinisch John
Published: October 2018

Objective

Patients with Treacher Collins syndrome have abnormal vascular supply to the palate, yet it is unknown whether there are increased postoperative healing problems following palatoplasty. This study investigated the correlation between Treacher Collins syndrome and postoperative palatal fistula formation.

Design

Retrospective chart review was performed.

Patients

Children undergoing palatoplasty at Childrens Hospital Los Angeles from 1987 to 2000 were evaluated. Ten children with Treacher Collins syndrome, 92 children with other syndromes and cleft palate, and 458 nonsyndromic patients with isolated cleft palate were studied.

Interventions

All children were treated with a one-stage, double-reversing Z-plasty cleft palate repair.

Main Outcome Measures

Outcome measures included intraoperative observations of surgical anatomy and postoperative clinic follow-up of fistula formation. Palatal fistula rates between patients with Treacher Collins syndrome, other syndromes, and no syndrome were compared with chi-square analysis.

Results

Children with Treacher Collins syndrome had significantly greater palatal fistula rates (50%) than children with other syndromes (8.7%) or no syndrome (4.1%). Treacher Collins patients demonstrated large palatal fistulas and poor flap vascularity.

Conclusions

Children with Treacher Collins syndrome and cleft palate have significantly higher palatal fistula risk than other children with cleft palate when double-reversing Z-plasty palate repair is performed. Our findings suggest that children with Treacher Collins syndrome and cleft palate may have poor vascularity to palatal flaps created during palatoplasty. Furthermore, we recommend that surgeons performing palatoplasty minimize the dissection of mucoperiosteal flaps around the greater palatine arterial pedicle and utilize closure techniques creating the least vascular disruption of palatal tissue.
Are Facial Expressions Reproducible?
Authors: Johnston D. J. , Millett D. T. , Ayoub A. F. , Bock M.
Published: October 2018

Objectives

To determine the extent of reproducibility of five facial expressions.

Design

Thirty healthy Caucasian volunteers (15 males, 15 females) aged 21 to 30 years had 20 landmarks highlighted on the face with a fine eyeliner pencil. Subjects were asked to perform a sequence of five facial expressions that were captured by a three-dimensional camera system. Each expression was repeated after 15 minutes to investigate intrasession expression reproducibility. To investigate intersession expression reproducibility, each subject returned 2 weeks after the first session. A single operator identified 3-dimensional coordinate values of each landmark. A partial ordinary procrustes analysis was used to adjust for differences in head posture between similar expressions. Statistical analysis was undertaken using analysis of variance (linear mixed effects model).

Results

Intrasession expression reproducibility was least between cheek puffs (1.12 mm) and greatest between rest positions (0.74 mm). The reproducibility of individual landmarks was expression specific. Except for the lip purse, the reproducibility of facial expressions was not statistically different within each of the two sessions. Rest position was most reproducible, followed by lip purse, maximal smile, natural smile, and cheek puff. Subjects did not perform expressions with the same degree of symmetry on each occasion. Female subjects demonstrated significantly better reproducibility with regard to the maximal smile than males (p = .036).

Conclusions

Under standardized conditions, intrasession expression reproducibility was high. Variation in expression reproducibility between sessions was minimal. The extent of reproducibility is expression specific. Differences in expression reproducibility exist between males and females.
An Investigation of the Relationship between Associated Congenital Malformations and the Mental and Psychomotor Development of Children with Clefts
Authors: de Veye Henriëtte F. N. Swanenburg , Beemer Frits A. , Mellenbergh Gideon J. , Wolters Wim H. G. , Heineman-de Boer Josien A.
Published: October 2018

Objective

This research studied the relationship between associated congenital malformations and the mental and psychomotor development of children with clefts.

Design

The study was cross-sectional.

Setting

The study was conducted in a university hospital for children.

Participants

The sample consisted of 148 children with cleft lip, cleft palate, or both.

Main Outcome Measures

The children were assessed by a clinical geneticist at the age of 18 months. The children's level of development was determined by means of the Dutch version of the Bayley Scales of Infant Development.

Results

One-third of the total sample had associated malformations. Children with an isolated cleft lip showed the least. Children with an isolated cleft palate showed the highest percentage of minor malformations that are minor yet possibly worrisome. The total group achieved a mean developmental index (DI) on the mental scale of 98.9 with SD of 20.9. The motor scale showed a mean DI of 104.9 and SD of 24.7. Analysis of variance (ANOVA) showed that on the mental scale, the three main effects (diagnosis, evaluation, and sex) were significant at the 5% level. On the motor scale, only the main effect “evaluation” was significant.

Conclusions

This study demonstrated that children with associated congenital malformations might be disadvantaged with respect to their development. These malformations occurred most frequently with the cleft lip and palate and cleft palate only subgroups. More research, especially concerning the cleft palate only subgroup is needed because they are most at risk.
Characteristics of Nonverbal Behavior in Patients with Cleft Lip and Palate during Interpersonal Communication
Authors: Adachi Tomoaki , Kochi Shoko , Yamaguchi Tai
Published: October 2018

Objective

This study examined characteristics of nonverbal behavior that patients with cleft lip and palate (CLP) presented during interpersonal communication.

Design

This was a case-control design comparing nonverbal behavior of adult women with CLP with females without CLP.

Participants

Subjects were 20 adult women with CLP and 20 noncleft control women matched for age and educational experience.

Main Outcome Measures

Subject gestures and facial expressions were videotaped during interviews and analyzed with a computer-based kinematic measurement system.

Results

The clinical group displayed significantly fewer head movements and a lower smile frequency than the control group. Furthermore, head and hand movements and smiles were less coordinated or congruent for the subjects with CLP than for the comparison group.

Conclusions

Even slight facial disfigurement could have a harmful effect on communication behavior in female patients with CLP.
Sphenoethmoidal Encephalocele: A Case Report
Authors: Acherman D. S. , Bosman D. K. , van der Horst C. M. A. M.
Published: October 2018

Objective

This article documents the characteristics and treatment of an infant patient with a sphenoethmoidal encephalocele. An extracranial, transpalatal approach was used to eliminate the encephalocele.
A Randomized Prospective Clinical Trial of the Effect of Infant Orthopedics in Unilateral Cleft Lip and Palate: Prevention of Collapse of the Alveolar Segments (Dutchcleft)
Authors: Prahl Charlotte , Kuijpers-Jagtman Anne M. , van't Hof Martin A. , Prahl-Andersen Birte
Published: October 2018

Objective

To study the effect of infant orthopedics (IO) on maxillary arch form and position of the alveolar segments.

Design

Prospective two-arm randomized, controlled trial in parallel with three participating academic cleft palate centers. Treatment was assigned by means of a computerized balanced allocation method.

Setting

Cleft palate centers of Amsterdam, Nijmegen, and Rotterdam, the Netherlands.

Patients, Participants

Infants with complete unilateral cleft lip and palate and no other malformations.

Interventions

One group (IO+) wore passive maxillary plates during the first year of life; the other group (IO−) did not. All other interventions were the same.

Main Outcome Measure(s)

The presence of contact and/or overlap (collapse) between the maxillary segments at maxillary casts made shortly after birth, at 15, 24, 48, 58, and 78 weeks. Survival experience of contact and collapse with time as well as the frequencies of different arch forms and severity of collapse were evaluated.

Results

Comparable arch forms with no contact or overlap of the maxillary segments were seen at birth in both groups. With time the frequency of collapse increased, with no significant differences between groups. No significant group differences were found with respect to the survival experience of contact and collapse or for the severity of collapse at the end of the observational period.

Conclusions

Infant orthopedics does not prevent collapse and can be abandoned as a tool to improve maxillary arch form.
Effects of Subglottal Air Pressure on Velopharyngeal Muscle Activity in Dogs
Authors: Kogo Mikihiko , Iida Seiji , Senoo Hitomi , Ishii Shouichiro , Hamaguchi Munehiro , Enomoto Akifumi , Matsuya Tokuzo
Published: October 2018

Objective

To analyze the effects of airflow in the larynx on activity of the levator veli palatini and pterygopharyngeal muscles.

Design

Ten adult beagle dogs were anesthetized with sodium pentobarbital. In each dog, two tracheal tubes were inserted subsequent to tracheotomy, one in the direction of the vocal folds and the other toward the lungs for respiration. In the first of three experiments, the effect of artificial airflow on electromyographic activity of the levator and pterygopharyngeal muscles was studied. In the second experiment, the effect of air pressure beneath the vocal folds on the activity of these muscles was studied. For the third experiment, the larynx was isolated surgically without cutting the bilateral superior laryngeal nerves and the effect of airflow through it examined.

Results and Conclusion

Both outward airflow and higher pressure enhanced expiratory activity of the levator and pterygopharyngeal muscles. Receptors in the subglottal area play major roles in this enhancement. Furthermore, an increase in air pressure during expiration enhances closure of the velopharynx.
Language Skills of Young Children with Unilateral Cleft Lip and Palate following Infant Orthopedics: A Randomized Clinical Trial
Authors: Konst Emmy M. , Rietveld Toni , Peters Herman F. M. , Kuijpers-Jagtman Anne Marie
Published: October 2018

Objective

To investigate the effects of infant orthopedics (IO) on the language skills of children with complete unilateral cleft lip and palate (UCLP).

Design

In a prospective randomized clinical trial (Dutchcleft), two groups of children with complete UCLP were followed up longitudinally: one group was treated with IO based on a modified Zurich approach in the first year of life (IO group); the other group did not receive this treatment (non-IO group). At the ages of 2, 2½, 3, and 6 years, language development was evaluated in 12 children (six IO and six non-IO). Receptive language skills were assessed using the Reynell test. Expressive language skills of the toddlers were evaluated by calculating mean length of utterance (MLU) and mean length of longest utterances (MLLU); in the 6-year-olds, the expressive language skills were measured using standardized Dutch language tests.

Patients

The participants had complete UCLP without soft tissue bands or other malformations.

Results

IO did not affect the receptive language skills. However, the expressive language measures MLU and MLLU were influenced by IO. At age 2½ and 3 years, the IO group produced longer utterances than the non-IO group. In the follow-up, the difference in expressive language between the two groups was no longer significant.

Conclusions

Children treated with IO during their first year of life produced longer sentences than non-IO children at the ages of 2½ and 3 years. At 6 years of age, both groups presented similar expressive language skills. Hence, IO treatment did not have long-lasting effects on language development.
Craniofacial Morphology and Growth Comparisons in Children with Robin Sequence, Isolated Cleft Palate, and Unilateral Complete Cleft Lip and Palate
Authors: Hermann N. V. , Kreiborg S. , Darvann T. A. , Jensen B. L. , Dahl E. , Bolund S.
Published: October 2018

Objective

Comparison of early craniofacial morphology and growth in children with nonsyndromic Robin Sequence (RS), isolated cleft palate (ICP), and unilateral complete cleft lip and palate (UCCLP).

Subjects

One hundred eight children with cleft: 7 with RS, 53 with ICP, and 48 with UCCLP were included in the study. The children were drawn from the group of all Danish children with cleft born 1976 through 1981.

Method

Three-projection infant cephalometry.

Results

The craniofacial morphology in the RS, ICP, and UCCLP groups had some common characteristics: a wide maxilla with decreased length and posterior height, wide nasal cavity, short mandible, bimaxillary retrognathia, and reduced pharyngeal airway. The shortest mandible was found in RS followed by ICP and UCCLP; the pharyngeal airway was reduced in RS and ICP, compared with UCCLP; and the maxillary complex and nasal cavity were wider in UCCLP than in the other groups. The amount of facial growth in all three groups was similar; however, the direction was more vertical in UCCLP than in RS and ICP.

Conclusion

Except for a shorter RS mandible, the facial morphology of infants with RS and ICP was similar, as was the amount of facial growth and the growth pattern. The differences in facial morphology can be ascribed to the difference in the primary anomaly. The amount of facial growth was similar in the three groups; however, the growth pattern showed a more vertical direction in UCCLP than in RS and ICP. It is hypothesized that the mandibular retrognathia in RS represents the outer end of that of the ICP distribution.