bluuebunny/crossref_metadata_2025 huggingface dataset:
A Comparative Study of Prespeech Vocalizations in Two Groups of Toddlers with Cleft Palate and a Noncleft Group
Authors: Willadsen Elisabeth , Enemark Hans
Published: October 2018

Objective

This study examined the prelinguistic contoid (consonant-like) inventories of 14 children with unilateral cleft lip and palate (C-UCLP) at 13 months of age. The children had received primary veloplasty at 7 months of age and closure of the hard palate was performed at 3–5 years. The results of this investigation were compared to results previously reported for 19 children with cleft palate and 19 noncleft children at the age of 13 months. The children with clefts in that study received a two-stage palatal surgery. This surgical procedure was formerly used at our center and included closure of the lip and hard palate at 3 months of age and soft palate closure at 22 months of age.

Design

Retrospective study.

Setting

The participants were videorecorded in their homes during play with their mothers. The videotapes were transcribed independently by three trained speech pathologists.

Patients

Fourteen consecutive patients born with C-UCLP and no known mental retardation or associated syndromes served as subjects.

Results

The children who received delayed closure of the hard palate demonstrated a significantly richer variety of contoids in their prespeech vocalizations than the cleft children in the comparison group. Both groups of subjects with clefts had significantly fewer plosives in their contoid inventory than the noncleft group, and there was no difference regarding place of articulation between the group that received delayed closure of the hard palate and the noncleft group.
Three-Dimensional Computer Morphometry of the Maxilla and Face in Infants with Pierre Robin Sequence-A Comparative Study
Authors: Bacher Margit , Bacher Ulrike , Göz Gernot , Pham Thinh , Cornelius Carl-Peter , Speer Christian P. , Goelz Rangmar , Arand Jörg , Wendling Fritz , Buchner Peter , Bacher Adelbert
Published: October 2018

Objective

To analyze the morphology of the maxillary crest in infants with Pierre Robin sequence using an anthropometric coordinate system and to compare the data with those of healthy infants.

Setting

The study was performed at a craniofacial center servicing a large geographic area.

Participants

The study involved eight infants aged 1–28 days (average, 7 days) with an established diagnosis of Pierre Robin sequence and six healthy infants aged 1–43 days (average, 22 days).

Main outcome Measures

Physical models of the maxilla and face obtained by alginate replication were analyzed by computer morphometry yielding the three-dimensional topology of the maxillary crest.

Results

The maxillary crest of children with Pierre Robin sequence shows an increased inclination relative to the transverse plane (30 ± 3.9 degrees) as compared with that of healthy infants (20 ± 2.9 degrees). The maxillary crest of the patients is shortened in the sagittal direction by comparison with healthy controls.

Conclusions

The increased inclination of the maxilla in infants with Pierre Robin sequence may aggravate the retroposition of the mandible and may thus be a pathogenetic factor contributing to the severe respiratory problems.
Analysis of Scar Tissue Distribution on Rat Palates: A Laser Doppler Flowmetric Study
Authors: Chu Soowon , Ishikawa Hiroyuki , Kim Takenori , Yoshida Shigemitsu
Published: October 2018

Objective

The aim of this study was to evaluate the effectiveness of laser Doppler flowmetry (LDF) to discriminate postoperative scar tissue distribution on the palate.

Methods

Nineteen male Wistar rats at the 20th postnatal day were divided into experimental and control groups. In the experimental group, lateral palatal mucoperiosteum was excised to form scar tissue on the palate. At the 11th postnatal week, changes in the palatal blood flow were recorded with LDF in both groups by occluding exposed common carotid arteries. Perfusion values of nonoccluded (stable) and occluded states and the ratio of occluded to stable states were compared for scar tissue and normal tissue areas, and also for the normal tissue areas. After the LDF measurements, India ink–injected specimens and tissue sections were prepared for histological observations.

Results and Conclusions

Scar tissue areas showed lower perfusion values both in the stable and occluded states, reflecting a lower vascular density in the scar tissue. The ratio of the occluded to stable states was higher in the scar tissue than in the normal tissue areas. In normal tissue areas, perfusion values of both the stable and occluded states appeared to vary, but the ratio did not vary among the areas. In the LDF study, the ratio of the occluded to stable states was considered to be the better parameter for discriminating scar tissue from normal tissue.
Secondary Cleft Lip Nasal Reconstruction: State of the Art
Authors: Cutting Court B.
Published: October 2018

Objective

This paper summarizes the state of the art in secondary cleft lip nasal reconstruction, distilled from the many papers written on the subject and from the author's experience with many of those procedures over the past 25 years.

Methods

The evaluation starts with the skeletal base and the need for LeFort 1 or alveolar bone grafting is discussed. The boney dorsum is next evaluated and a “monobloc” osteotomy considered. The cartilaginous dorsum follows and a “spreader-strut” graft is entertained. The tip cartilages are approached with either an open Potter or Dibbell preferred or replacement conchal graft if the tip has been destroyed by previous surgery. The skin envelope is then adjusted using methods described by Tajima, Dibbell, and Bardach.
Comparative Study of Mandibular Stability after Sagittal Split Osteotomies: Biocortical versus Monocortical Osteosynthesis
Authors: Fujioka Masaki , Fujii Tohru , Hirano Akiyoshi
Published: October 2018

Objective

A comparative study of clinical mandibular stability following bilateral sagittal split osteotomies by means of monocortical versus bicortical osteosynthesis was undertaken.

Design

This retrospective study utilized cephalometric radiographs, which were taken at 1 week and 6 months postoperatively.

Setting

The research was carried out at the Department of Plastic and Reconstructive Surgery of the Nagasaki University School of Medicine.

Patients

A total of 32 patients who underwent only sagittal split osteotomies and mandibular set back in our unit was included in this study. Of these patients, 17 patients were osteosynthesized monocortically, and 15 patients were osteosynthesized bicortically.

Main outcome Measures

Four measurements—gonial angle (GA), mandibular plane (MP), SNB, and percentage of upper face to total face height (percent upper to total face)—were completed to evaluate postoperative mandibular movement.

Result

Statistical analyses of cephalometric measurements (GA, MP, SNB, and percent upper face to total face height) showed that monocortical fixed mandibles were more changeable postoperatively on the GA and percent upper face to total face height, but MP and SNB showed no significant differences among the groups.

Conclusion

These findings suggested that the postoperative excessive shear force stress, produced by the compressive action of the masseter muscle, transformed the mandibular shape as the distal segment rotated clockwise and proximal segment rotated counterclockwise. Consequently, the mandible was bent at the miniplate. It was concluded that bicortical osteosynthesis was more rigid against this shearing stress than monocortical osteosynthesis.
Secondary Bone Grafting of Alveolar Clefts: A Densitometric Comparison of Iliac Crest and Tibial Bone Grafts
Authors: Sivarajasingam V. , Pell G. , Morse M. , Shepherd J. P.
Published: October 2018

Objective

To evaluate changes in the optical density of secondary alveolar cleft bone grafts obtained from two different donor sites over time and to determine whether one donor site gives a higher recipient bone density than the other.

Methods

A prospective study was performed evaluating 40 healthy patients with congenital cleft lip and palate undergoing secondary alveolar bone grafting, 20 (14 boys and 6 girls) having iliac crest and 20 (12 boys and 8 girls) receiving tibial bone grafts. Bone harvest and grafting was carried out by one operator (G.P.). Optical density of iliac and tibial grafts measured using a computerized densitometer, was compared at 6 days, 6 weeks, and 3 months. Due to interference from orthodontic appliances, optical density measurements for 16 subjects were not possible, and these patients were excluded from the study. The length of hospital stay postoperatively for both grafting procedures were recorded.

Results

A significant decrease in relative bone density was demonstrated during the 3-month postoperative period in both iliac and tibial bone graft groups (p < .05). The difference in densities between iliac crest and tibial groups were not significantly different at any of the time points (paired t test, p > .05). Subjects undergoing iliac crest grafts stayed an average of 5 days in the hospital postoperatively, compared with subjects with tibial grafts who stayed an average of 3 days postoperatively.

Conclusion

Optical density measurements of bone grafted into alveolar clefts, reported here for the first time, provide a valuable objective assessment of graft progress. Tibial and iliac crest grafts gave similar optical densities at recipient sites over the first 3 months. Iliac crest grafts required significantly longer postoperative stay; an important consideration in selecting donor sites for secondary bone grafting.
Cleft Lip and Palate Care in the United Kingdom—The Clinical Standards Advisory Group (CSAG) Study. Part 1: Background and Methodology
Authors: Sandy Jonathan R. , Williams Alison C. , Bearn David , Mildinhall Sue , Murphy Terrie , Sell Debbie , Murray John J. , Shaw William C.
Published: October 2018

Objective

To review the history of cleft care in the United Kingdom and the rationale and methodology of a national study.

Design

Descriptive overview to introduce this series of papers.

Setting

Fifty National Health Service cleft teams.

Patients/Participants

Two age cohorts, 5-year-olds and 12-year-olds, with nonsyndromic complete unilateral cleft lip and palate and their parents.

Main Outcome Measures

Facial development and appearance, quality of bone grafts, speech, oral health and patient/parent satisfaction.

Conclusions

The case for a national assessment of cleft care is made. The methodology of the cross-sectional outcome study is described, together with a survey of training for recently appointed clinicians.
Furlow Palatoplasty to Restore Velopharyngeal Competence following Tonsillectomy Avulsion of a Pharyngeal Flap
Authors: Gosain Arun K. , Remmler Daniel
Published: October 2018

Objective

We report the successful use of a Furlow palatoplasty to salvage velopharyngeal competence following iatrogenic avulsion of a pharyngeal flap that had been previously established to treat velopharyngeal insufficiency associated with a submucous cleft palate.

Intervention

A tonsillectomy, conducted by a surgeon unaffiliated with a cleft palate team, was used to remove enlarged tonsils that had developed after pharyngeal flap surgery and extended into the lateral ports causing nasal obstruction and hypernasality because of mechanical interference with port closure. A posttonsillectomy evaluation revealed avulsion of the pharyngeal flap, which was successfully treated using a Furlow palatoplasty.

Conclusions

To our knowledge, this is the first report of iatrogenic avulsion of a pharyngeal flap caused by tonsillectomy. Based on a review of the literature and this case experience, we would conclude that tonsillectomy should not be regarded as a routine procedure in patients previously treated with a pharyngeal flap. If required, it should be performed by a skilled otolaryngologist, preferably one affiliated with a multidisciplinary cleft palate team who is familiar with pharyngoplasty surgery. Finally, our experience would suggest that the Furlow palatoplasty is sufficiently robust to be used as a secondary salvage procedure to restore velopharyngeal sufficiency following iatrogenic avulsion of a pharyngeal flap.
Nasalance Measures in Cantonese-Speaking Women
Authors: Whitehill Tara L.
Published: October 2018

Objectives

To establish and evaluate stimulus materials for nasalance measurement in Cantonese speakers, to provide normative data for Cantonese-speaking women, and to evaluate session-to-session reliability of nasalance measures.

Participants and setting

One hundred forty-one Cantonese-speaking women with normal resonance who were students in the Department of Speech and Hearing Sciences, University of Hong Kong.

Procedures

Participants read aloud four speech stimuli: oral sentences, nasal sentences, an oral paragraph (similar to the Zoo Passage), and an oral-nasal paragraph (similar to the Rainbow Passage). Data were collected and analyzed using the Kay Nasometer 6200. Data collection was repeated for a subgroup of speakers (n = 28) on a separate day. Nasalance materials were evaluated by using statistical tests of difference and correlation.

Results

Group mean (standard deviation) nasalance scores for oral sentences, nasal sentences, oral paragraph, and oral-nasal paragraph were 16.79 (5.99), 55.67 (7.38), 13.68 (7.16), and 35.46 (6.22), respectively. There was a significant difference in mean nasalance scores for oral versus nasal materials. Correlations between stimuli were as expected, ranging from 0.43 to 0.91. Session-to-session reliability was within 5 points for over 95% of speakers for the oral stimuli but for less than 76% of speakers for the nasal and oral-nasal stimuli.

Conclusions

Standard nasalance materials have been developed for Cantonese, and normative data have been established for Cantonese women. Evaluation of materials indicated acceptable differentiation between oral and nasal materials. Two stimuli (nasal sentences and oral paragraph) are recommended for future use. Comparison with findings from other languages showed similarities in scores; possible language-specific differences are discussed. Session-to-session reliability was poorer for nasal than oral stimuli.
Social Anxiety in Chinese Adults with Oral-Facial Clefts
Authors: Berk Nancy W. , Cooper Margaret E. , Liu You-e , Marazita Mary L.
Published: October 2018

Objective

This study examined social anxiety and measures of psychosocial adjustment in Chinese adults with oral-facial clefts, their unaffected siblings, and age-matched controls.

Design

This cross-sectional study utilized a matched case-control study design.

Participants

Eighty-five adult cleft lip and cleft palate (CL/CP) subjects and 85 unaffected siblings (one adult sibling of each CL/CP subject) were recruited in Shanghai, China, from a larger CL/CP study. Eighty-five unaffected controls, gender- and age-matched to the CL/CP subjects, were recruited from Shanghai work units including factories, universities, and other institutions.

Outcome Measures

Social Avoidance and Distress Scale, Fear of Negative Evaluation, Rosenberg Self-Esteem Scale, Interpersonal Support Evaluation List.

Results

Affected adults reported significantly more social anxiety than unaffected siblings and controls. Affected adults also scored significantly lower on measures of self-esteem and social support than unaffected siblings and controls. Unaffected siblings and controls were not found to differ on any of these measures.

Conclusions

Findings suggest that individuals with oral-facial clefts may be more disadvantaged with respect to social affiliation and adaptation than unaffected adults. Cross-cultural research is essential in enabling us to determine whether similar trends exist across cultures.
The Effect of Neurocranial Surgery on Basicranial Morphology in Isolated Sagittal Craniosynostosis
Authors: DeLeon Valerie Burke , Zumpano Michael P. , Richtsmeier Joan T.
Published: October 2018

Objective

Isolated sagittal craniosynostosis produces a scaphocephalic neurocranium associated with abnormal basicranial morphology, providing additional evidence of the developmental relationship of the neurocranium and basicranium. Corrective surgical procedures vary, but the immediate impact of the surgical procedure is restricted to the neurocranium. This study addresses the secondary effects of neurocranial surgery on the cranial base.

Design

Three-dimensional (3-D) computed tomography (CT) scans were obtained for preoperative (n = 25) and postoperative (n = 12) patients with isolated sagittal synostosis. Landmark data from 14 landmarks on and around the cranial base were collected from 3-D CT reconstructions and analyzed using Euclidean distance matrix analysis. Subsamples of age-matched patients were used to identify basicranial differences in pre- and postoperative patients and to compare postoperative growth patterns identified in longitudinal data with preoperative growth patterns characterized in cross-sectional data.

Results

Statistically significant differences (p ≤ 0.10) were found in the morphology of the cranial base in preoperative and postoperative patients. The relative positions of the landmarks nasion, right asterion, and left asterion are similar in preoperative and postoperative patients. However, the position of these landmarks relative to the cranial base is different in the two groups, being positioned relatively more anteriorly in postoperative patients. In addition, we found that the cranial base angle, on average, neither increases nor decreases in the first postoperative year. These morphological differences are associated with divergent growth trajectories in the operated and unoperated cranial base.

Conclusion

Regardless of specific procedure, neurocranial surgery in sagittal synostosis patients affects growth patterns of the cranial base. The lack of change in the postoperative cranial base angle suggests that neurocranial surgery alleviates the occipital rotation and decreased cranial base angle described in the sagittal synostosis basicranium.
Nasal Airflow Patterns during the Velopharyngeal Closing Phase in Speech in Children with and without Cleft Palate
Authors: Dotevall Hans , Ejnell Hasse , Bake Björn
Published: October 2018

Objectives:

(1) To study the nasal airflow patterns during the velopharyngeal closing phase in speech produced by children with and without cleft palate. (2) To compare the nasal airflow patterns in bilabial, dental, and velar articulation in these children.

Design:

Prospective, cross-sectional study of a consecutive series of children with cleft palate referred for routine speech evaluation and controls.

Setting:

Sahlgrenska University Hospital, Göteborg, Sweden.

Participants:

Seventeen children with cleft lip and palate or cleft palate only and 22 controls aged 7 and 10 years.

Method:

Nasal airflow was transduced with a pneumotachograph attached to a nose mask and registered together with the acoustic speech signal. Sentences containing nasal-to-stop combinations in bilabial, dental, and velar articulatory positions were used.

Main Outcomes Measures:

The duration from peak to 5% nasal airflow, the maximum flow declination rate, and the nasal airflow at selected points in time during the transition from nasal-to-stop consonants.

Results:

In the cleft palate group, duration from peak to 5% nasal airflow was clearly longer than among the controls (p < .0001). The declination of airflow was slower (p < .006) and the rate of nasal airflow at the release of the stop consonant was higher (p < .004) in the cleft palate group. Differences between bilabial versus dental and velar articulation were found in the control group.

Conclusion:

Studies of the temporal and dynamic characteristics of the nasal airflow variations during speech appear potentially useful for the assessment of velopharyngeal function.
“Distraction” of Grafted Alveolar Bone in Cleft Case Using Endosseous Implant
Authors: Buis Jacques , Rousseau Paul , Soupre Veronique , Martinez Herve , Antoine Diner Patrick , Paule Vazquez Marie
Published: October 2018

Objective:

Presentation of a specific surgical technique of vertical alveolar ridge distraction performed on an alveolar bone graft 1 year after the primary grafting procedure designed to correct a labioalveolar cleft. The graft had not obtained the results desired and presented heavily scarred mucosal tissue.

Technique:

A dental implant placed within the graft once it has been entirely freed by osteotomies functions as an endless screw. To avoid vascular risk, the alveolar bone graft is detached as a whole along its total height. Consequently, no bone remains above it to provide support for a conventional alveolar distractor. An osteosynthetic miniplate, fastened in a horizontal position beneath the nasal mucosae above the graft, is used to replace the missing bony support and to stabilize the implant, which activates the distraction process.

Patient:

This study is based on our first clinical case with a follow-up of 18 months at present.

Result:

A vertical displacement of the entire graft was achieved, including its attached mucosal layer, which repositioned both bone and mucosa on a far better level in the cleft area. Thus, a normal length of abutment was obtained permitting prosthetic rehabilitation based on the same endosseous implant, which was left in place within the graft.

Conclusion:

This technique may prove particularly helpful in certain cases in which a primary alveolar bone grafting procedure has produced borderline results. In such cases, on the one hand, neither the reasonably satisfactory volume of the bone graft itself nor the poor quality of its scarred mucosal tissue argue in favor of a secondary grafting procedure. On the other hand, it is impossible to resort to currently available alveolar distracters since our choice of techniques leaves no bone support above the graft.
Analysis of Speech Characteristics in Children with Velocardiofacial Syndrome (VCFS) and Children with Phenotypic Overlap without VCFS
Authors: D'Antonio Linda L. , Scherer Nancy J. , Miller Laura L. , Kalbfleisch John H. , Bartley James A.
Published: October 2018

Objective:

To address two questions of theoretical importance regarding the profile and course of communication impairment associated with velocardiofacial syndrome (VCFS): (1) do speech characteristics of children with VCFS differ from a group of children with some of the phenotypic characteristics of VCFS who do not have the syndrome, and (2) do younger children with VCFS demonstrate speech patterns that differ from older children with VCFS?

Design:

Prospective, cross-sectional study comparing two groups of children at two age levels.

Patients:

Thirteen children with VCFS and eight children with some of the phenotypic features of VCFS who did not have the syndrome. Children ranged in age from 3 to 10 years.

Main Outcome Measure:

(1) Broad phonetic transcription of speech yielding measures of number of consonant types, Percent Consonant Correct, and percentage of glottal stops used; and (2) composite ratings of velopharyngeal function made from perceptual, aerodynamic, and endoscopic evaluations.

Results:

Younger children with VCFS demonstrated greater speech impairment than older children with VCFS or the children without VCFS, such as smaller consonant inventories, greater number of developmental errors, greater severity of articulation disorder, and higher frequency of glottal stop use. The relationship between ratings of velopharyngeal function and the speech variables analyzed was not straightforward.

Conclusions:

Some young children with VCFS demonstrated speech impairment that is qualitatively and quantitatively different from older children with VCFS or children without VCFS. This finding supports the hypothesis that some children with VCFS demonstrate a profile of speech production that is different from normal but also may be specific to the syndrome.
Palatal Development in Twirler Mice
Authors: Gong Siew-Ging , Eulenberg Rachel L.
Published: October 2018

Objective:

The development of the secondary palate of Twirler (Tw) mice was characterized, and a quantitative coronal plane evaluation of the width and length of the craniofacial regions of homozygous mutant embryos was conducted.

Results and Conclusions:

The secondary palatal shelves were retarded in growth and the timing of elevation from a vertical to horizontal position. The homozygous Tw embryos, when compared with their wild-type littermates, also had a significantly wider midface. It is not surprising, therefore, that the short palatal shelves in a wide midface resulted in cleftings of the secondary palate observed in all homozygous Tw mice. These findings are consistent with similar studies in humans and other murine models.
New Design of a Palatal Lift Prosthesis Combined with a Palatal Bar
Authors: Ueda Naoto , Sato Yasumori , Senoo Yasuhiro , Mori Toshiko , Moriguchi Takahiko , Nakagawa Hirofumi
Published: October 2018

Objective

In the past, palatal lift prostheses (PLPs) have been problematic with respect to stability. This stability problem has been addressed by designing a PLP that includes a palatal bar.

Structure

A PLP combined with a palatal bar is functionally characterized by a palatal bar that holds down the posterior palatal plate. By making this location the fulcrum, the force exerted by the soft palate on the lamina actually functions to stabilize the prosthesis instead of acting to overturn it.

Cases

This new prosthetic design was applied to patients with cleft palate and those who had undergone cerebral contrusion operations. The experimental group was composed of a total of 13 patients (7 men and boys, 6 women and girls) who ranged in age at the time of introduction of the prosthesis from 4 years 6 months old to 54 years 5 months. The period of follow-up after prosthetic insertion ranged from 2 months to 4 years 1 month. In all cases, the stability of the prosthesis was sufficiently maintained.

Conclusion

Because of increased stability, this technique can be applied to not only adult patients but also children in whom insertion during the deciduous dentition period can be difficult. The new PLP was also found to be effective in patients with cleft palate with a short clinical crown.
Mandibular Asymmetry and Antigonial Notching in Individuals with Unilateral Cleft Lip and Palate
Authors: Kyrkanides Stephanos , Richter Liubov
Published: October 2018

Objective

To determine whether asymmetric antigonial notching is associated with the development of mandibular and lower facial asymmetry in individuals with unilateral cleft lip and palate (UCLP).

Design

Retrospective mixed-longitudinal investigation including UCLP and nonaffected orthodontic patients.

Participants

All (24) available patients with UCLP treated in our clinic with complete longitudinal records, and 72 skeletal maturation matched noncleft orthodontic patients that served as controls. Subject records included hand-wrist, panoramic, frontal, and 45° oblique cephalometric radiographs.

Main outcome Measures

The development of mandibular asymmetry, measured on oblique cephalometric radiographs, as well as lower facial asymmetry, measured on frontal cephalometric radiographs, was monitored in individuals with UCLP in relation to antigonial notching asymmetry, assessed on panoramic and oblique cephalometric radiographs.

Results

First, no statistical differences were found in antigonial notching asymmetry between patients with UCLP and noncleft controls. Second, antigonial notching asymmetry, observed on panoramic and oblique cephalometric radiographs, significantly correlated with the attendant development of mandibular and lower facial asymmetry in individuals with UCLP. Third, the development of mandibular and lower facial asymmetry at pubertal and post-pubertal growth spurt stages significantly correlated with antigonial notching asymmetry, measured on panoramic radiographs, at prepubertal and pubertal growth spurt stages, respectively.

Conclusions

The degree of antigonial notching noted on panoramic radiographs can be used as an early indicator of developing mandibular and lower facial asymmetry in individuals with UCLP.
Assessing Intelligibility in Speakers with Cleft Palate: A Critical Review of the Literature
Authors: Whitehill Tara L.
Published: October 2018

Objective

A literature review was conducted in order to investigate three research questions: how is speech intelligibility being measured in speakers with cleft lip and palate? Is intelligibility adequately being distinguished from related measures such as acceptability? Has there been an increased understanding of intelligibility deficits in speakers with cleft lip and palate? Fifty-seven relevant articles published between 1960 and 1998 were included in the analysis.

Results

The results showed an increase in the number of articles that included a measure of intelligibility or a similar measure. Several concerns were raised as a result of the review, including the reliability and validity of measures being employed, adequate definition and differentiation of terms, and the need to determine speech and nonspeech variables contributing to reductions in intelligibility. Relevant literature on intelligibility from fields outside cleft lip and palate is reviewed, and a number of recommendations are made regarding the measurement of intelligibility in speakers with cleft lip and palate.
Facial Appearance and Attachment in Infants with Orofacial Clefts: A Replication
Authors: Coy Katherine , Speltz Matthew L. , Jones Karen
Published: October 2018

Objective

The current study sought to replicate an unexpected finding reported by Speltz et al. (1997): relatively unattractive infants at age 3 months were more likely than attractive infants to show secure maternal attachment at age 12 months, a finding unaffected by the diagnosis of cleft lip and palate (CLP), cleft palate only (CPO), or the absence of a cleft condition.

Design

We evaluated the effects of diagnosis (CLP, CPO, or no diagnosis) and age (3, 12, and 24 months) on facial attractiveness ratings derived from a modified Q-sort method.

Setting

Craniofacial clinic in an urban children's hospital.

Participants

Infants with CLP and CPO and typically developing infants without clefts (n = 126) and their mothers. Ratings were made by 13 adults unfamiliar with cleft conditions.

Main outcome Measures

Facial attractiveness ratings.

Results

Infants with CLP were rated as the least attractive at all time points. At ages 12 and 24 months, infants in the CPO group were rated as less attractive than typical infants but more attractive than infants in the CLP group. Typical infants and those with CLP—utnot CPO—received higher attractiveness ratings with age. As hypothesized, less attractive infants, regardless of diagnosis, were more likely to show secure attachment than were more attractive infants.

Conclusions

We tentatively conclude that the perceived vulnerability of young infants, as indexed by atypical or unattractive facial characteristics, engenders extraordinary protectiveness and responsiveness in some mothers, leading to a higher probability of secure attachment. A test of this hypothesis with a new sample of infants is recommended.
Cephalometric Analysis in Submucous Cleft Palate: Comparison of Cephalometric Data Obtained from Submucous Cleft Palate Patients with Velopharyngeal Competence and Incompetence
Authors: Park Susam , Omori Makoto , Kato Kogo , Nitta Naotugu , Kitano Ichiko , Masuda Tomomi
Published: October 2018

Objective

The purpose of this study was to investigate the relationship between craniofacial and nasopharyngeal morphology and velopharyngeal function in submucous cleft palate.

Design and Patients

Fifty-two lateral cephalometric radiographs of 46 sub-mucous cleft palate (SMCP) patients with velopharyngeal competence (24 patients) and incompetence (22 patients) at 4 and 7 years of age were studied. The patients had not received any surgical or orthodontic treatment prior to cephalography being performed.

Results

Significant differences were found between cephalometric variables (N-Ba, N-S-Ba angle) in children with velopharyngeal competence and incompetence. However, the results of our study showed that cephalometric data alone are not useful for predicting velopharyngeal function and can not serve as an absolute prognostic indicator.

Conclusion

There are many factors that can influence velopharyngeal function in SMCP patients. Cephalometric data did not demonstrate a strong relationship to velopharyngeal function.
Neonatal Permanent Jaw Constriction Because of Oral Synechiae and Pierre Robin Sequence in a Child with van der Woude Syndrome
Authors: Denion E. , Capon N. , Martinot V. , Pellerin P.
Published: October 2018

Objective

To report a newborn with van der Woude syndrome, Pierre Robin sequence, and oral synechiae. Pierre Robin sequence is a rare manifestation of van der Woude syndrome as are oral synechiae. We speculate that the oral synechiae may be causally related to the development of Pierre Robin sequence in this patient.
A Cephalometric Study of Facial Growth in van der Woude Syndrome
Authors: Kane Alex A. , Liao Yu-Fang , Lo Lun-Jou , Huang Chiung-Shing , Huang Li-Min , Chen Yu-Ray , Noordhoff M. Samuel
Published: October 2018

Objective

To test the hypothesis that maxillary growth and lower lip form in patients with van der Woude syndrome (VDW) is different from patients with nonsyndromic cleft lip and palate.

Design and Setting

Retrospective, case-control study at a tertiary cleft center, Chang Gung Memorial Hospital, Taipei, Taiwan.

Patients and Participants

Records of 53 patients with VDW, who presented for treatment during the years 1968 through 1998 were obtained. Twenty-three of 53 patients had received at least one lateral cephalogram during the course of their treatment. Of these 23, in 17 it was possible to find nonsyndromic case controls with identical cleft type, sex, and method of cleft palate repair, with year of birth matching within 1 year of the corresponding VDW patient. For these 17 pairs of VDW and nonsyndromic cleft controls, cephalogram acquisition dates were checked to see how well the corresponding pairs matched. A total of 43 pairs of cephalograms were deemed to be acceptably matched because the ages at acquisition differed by less than 15% of the VDW patient's age.

Main outcome Measures

Thirteen measurements were derived from the 11 standard lateral cephalometric landmarks recorded on each cephalogram. The data were classified into five groups according to age at time of cephalogram, and sets of paired nonsyndromic cleft and VDW measurements were tested for differences using a Wilcoxon signed rank sum test in two ways, first including all cleft types and then including only those patients with complete bilateral cleft lip and palate. A longitudinal growth analysis considering the movement of the skeletal A and B points was performed on the patients with complete bilateral cleft lip and palate.

Results

For the osseous measurements, anteroposterior maxillary length as described by the anterior nasal spine (ANS)-posterior maxillary point distance was statistically significantly shorter in the VDW patients of age 13 years and older, by 5.3 mm. Maxillary height, as described by the nasion-ANS distance was shorter in the VDW patients, closely approaching statistical significance in the age range 7 through 11 years. The lip soft tissue measurements showed significantly greater protrusion over several age ranges in the VDW patients. The longitudinal growth analysis showed a significantly more inferior vertical position of the B point in the controls.

Conclusions

This study demonstrates a few statistically significant differences in maxillary growth and lip conformation between VDW and matching controls. Small sample sizes for each age group hamper the ability to fully interpret or generalize the pattern of these differences.
Clinical and Biochemical Findings of a Patient with Thanatophoric Dysplasia Type I: Additional Finding of Dicarboxylic Aciduria
Authors: Okajima Kazuki , Asai Kiyofumi , Niwa Toshimitsu , Ohki Shigeru , Sobajima Hisanori , Tyson Jess , Malcolm Sue , Wada Yoshiro
Published: October 2018

Objective

A long-surviving thanatophoric dysplasia type I patient to age of 6 years is presented.

Results and Conclusions

Molecular studies revealed a heterozygous point mutation, S249C in the fibroblast growth factor receptor 3 gene. Most of the clinical course was similar to previous reports, including hearing loss and acanthosis nigricans. Abnormal urinary excretion of dicarboxylic acids and 3-hydroxydicarboxylic acids was observed. We hypothesize that this was a consequence of the FGFR3 mutation.
Palate Height: Another Indicator of Surgical Outcome in Unilateral Cleft Lip and Palate?
Authors: Kharbanda Om P. , Shaw William C. , Worthington Helen
Published: October 2018

Objective:

To determine whether palate height and maxillary arch depth are systematically related to the surgical center at which primary repair in unilateral cleft lip and palate (UCLP) was carried out.

Design:

A retrospective comparison based on study casts of consecutive cases of UCLP obtained at age 9 years from six different centers. The observer who conducted measurements was blinded to the source of individual records.

Setting:

The patients whose records were analyzed received all their surgical care in a national health service setting in six different northern European centers and regions.

Patients:

Patients were consecutively treated Caucasian children with non-syndromic complete UCLP born in the period 1976 to 1979.

Main Outcome Measures:

The main outcome measures for the original study were craniofacial form, dental arch relationships, nasolabial appearance, and speech. This report focuses on measurements of anterior maxillary arch depth and palate height.

Results:

Anterior arch depth and anterior palate height showed some variation among the centers. There was a tendency for anterior arch depth and palate height to also be reduced at centers at which patients showed unfavorable dental arch relationships.

Conclusions:

Anterior arch depth and palate height might be considered in future studies of surgical outcome and in their possible relationship to problems of articulation.
Isolated Soft Tissue Cleft Lip: The Influence on the Nasal Cavity and Supernumerary Laterals
Authors: Hansen Ken , Mehdinia Maryam
Published: October 2018

Objective:

The purpose of this investigation was to estimate the vertical deviation of the nasal floor and to register dental disturbances (i.e., hypo- and hyperdontia) in patients with an isolated soft tissue cleft lip.

Design:

Thirty patients with isolated cleft lips were randomly selected from the files of the Department of Maxillo-Facial Surgery and Jaw Orthopedics. As a matched control group, 30 patients were selected from the files of the Department of Orthodontics. Panoramic radiographs were analyzed and the lowest point of the floor of the left and right nasal cavity was measured to the nearest 0.5 mm. The presence of supernumerary (deciduous or permanent) or missing lateral incisors was recorded from the patient's journals.

Results:

The vertical position of the nasal floor differed significantly between the cleft and the noncleft side (p <0.001). Supernumerary, deciduous, and permanent laterals were recorded in 73% of the subjects in the cleft lip group (22 patients). In the control group, no differences were found between the position of the nasal floor on the left and right side and no supernumerary laterals were recorded. Missing laterals were not noted in any of the individuals investigated.

Conclusions:

Isolated cleft lips were in most cases associated with: (1) lowering of the nasal floor on the cleft side and (2) supernumerary primary or permanent lateral incisors on the cleft side. Panoramic radiographs can be used to detect differences in the vertical position of the floor of the nasal cavity in patients with isolated cleft lips and to diagnose supernumerary laterals.
Visualization of Individual Growth-Related Craniofacial Changes Based on Cephalometric Landmark Data: A Pilot Study
Authors: Lux Christopher J. , Starke Jens , Rübel Jan , Stellzig Angelika , Komposch Gerda
Published: October 2018

Objective:

An approach based on Euclidean distances between cephalometric landmarks is presented (1) to visualize and localize the individual shape changes of the complex craniofacial skeleton during growth and (2) to depict the individual dynamic behavior of developmental size and shape changes.

Patients and Method:

Growth-related craniofacial changes were investigated exemplarily for two male orthodontically untreated subjects from the Belfast Growth Study on the basis of lateral cephalograms at 7, 9, 11, 13, and 15 years. The interlandmark distances among seven skeletal cephalometric landmarks served as a database for the study. A modified Karhunen-Loèvedecomposition based on orthogonal modes and time-dependent scalar amplitudes was used to describe the growth process. The individual shape changes of the various craniofacial regions were visualized by allocation of colors to the respective distances, and overdrawn representations were reconstructed by means of multidimensional scaling.

Results and Conclusions:

This visualization technique allows anatomical regions to be characterized with respect to reduced or strengthened growth, compared with pure size changes. The clinically relevant mechanisms of craniofacial changes are visualized (e.g., shifts in the anteroposterior or vertical dimensions of the jaws in relation to cranial base and structural imbalances during development). In addition, overdrawing the effects of shape change on the skeletal structures gives a more readily comprehensible impression of the growth process. Taking account of the methodical limitations of this approach (e.g., the restrictions concerning the number of landmarks), the clinician may take advantage of this technique in orthodontic or surgical diagnostics to gain additional insight into the individual complex size and shape changes during development along with their dynamic behavior.
Speech Outcomes in Isolated Cleft Palate: Impact of Cleft Extent and Additional Malformations
Authors: Persson Christina , Elander Anna , Lohmander-Agerskov Anette , Söderpalm Ewa
Published: October 2018

Objective

The purpose of the study was to study the speech outcome in a series of 5-year-old children born with an isolated cleft palate and compare the speech with that of noncleft children and to study the impact of cleft extent and additional malformation on the speech outcome.

Design

A cross-sectional retrospective study.

Setting

A university hospital serving a population of 1.5 million inhabitants.

Subjects

Fifty-one patients with an isolated cleft palate; 22 of these had additional malformations. Thirteen noncleft children served as a reference group.

Interventions

A primary soft palate repair at a mean of 8 months of age and a hard palate closure at a mean age of 4 years and 2 months if the cleft extended into the hard palate.

Main outcome Measures

Perceptual judgment of seven speech variables assessed on a five-point scale by three experienced speech pathologists.

Results

The cleft palate group had significantly higher frequency of speech symptoms related to velopharyngeal function than the reference group. There were, however, no significant differences in speech outcome between the subgroup with a nonsyndromic cleft and the reference group. Cleft extent had a significant impact on the variable retracted oral articulation while the presence of additional malformations had a significant impact on several variables related to velopharyngeal function and articulation errors.

Conclusion

Children with a cleft in the soft palate only, with no additional malformations, had satisfactory speech, while children with a cleft palate accompanied by additional malformations or as a part of a syndrome should be considered to be at risk for speech problems.
Constriction of the Maxillary Dental Arch by Mucoperiosteal Denudation of the Palate
Authors: Kim Takenori , Ishikawa Hiroyuki , Chu Soowon , Handa Asako , Iida Junichiro , Yoshida Shigemitsu
Published: October 2018

Objective

This study examined the influence of two factors in the constriction of the maxillary dental arch by mucoperiosteal denudation of the palate: (1) inhibition of lateral growth and (2) medial inclination of teeth.

Method

Thirty-five male 20-day-old Wistar rats were divided into experimental and control groups. The experimental group had bilateral mucoperiosteum excised in the lateral one third of the palate. Methyl methacrylate resin–embedded frontal sections were prepared from both groups after alternate weekly injections of tetracycline and calcein in the dorsal subcutaneous area. The sections were observed and photographed under either a confocal laser scanning microscope, a fluorescence microscope, or both. Chronological changes in lateral palatal growth, maxillary dental arch width, and inclination of the upper first molars were examined up to 8 weeks after the operation. Paraffin-embedded frontal sections were also made and stained with Elastica van Gieson stain.

Results

The scar tissue formed on the rat palate by the mucoperiosteal denudation was tightly connected to the palatal bone and teeth. The intervals between the labeling lines of the experimental group were less definite during the first 2 weeks after the operation. Increments of palatal and maxillary dental arch widths were smaller in the experimental group than in the control group. The upper first molars in the control group gradually inclined laterally, whereas those in the experimental group inclined medially with age.

Conclusion

Medial inclination of teeth is a stronger influence than inhibition of lateral growth on constriction of the rat maxillary dental arch.
Tooth Eruption into the Newly Generated Bone Induced by Recombinant Human Bone Morphogenetic Protein-2
Authors: Kitamura Atsushi , Motohashi Nobuyoshi , Kawamoto Tatsuo , Baba Yoshiyuki , Suzuki Shoichi , Kuroda Takayuki
Published: October 2018

Objective

The purpose of this study was to observe tooth eruption pattern into the newly generated bone induced by recombinant human bone morphogenetic protein-2 (rhBMP-2).

Method

Three 12-week-old beagle dogs were used. Following wound healing of the extracted maxillary third deciduous incisor at the age of 13 weeks, a resection of maxillary alveolar bone (8 mm by 8 mm by 8 mm) was carried out, accompanied by extraction of the maxillary second incisor tooth germ. The rhBMP-2 (5 μg/100 μL or 10 μg/100 μL) was implanted in the resected area. A group that was not implanted after resection and a nonresected group were used as controls. Serial changes of the eruption pattern of the maxillary third incisor in the implanted area were observed radiographically. Oxytetracycline and calcein were employed as bone markers. Nine weeks after the implantation, the animals were sacrificed. The samples were observed histologically and analyzed using peripheral quantitative computerized tomography to acquire the bone mineral density (BMD).

Results

In the rhBMP-2 (5 μg/100 μL)-treated group, histological findings and the BMD of the newly generated bone were almost same as in the non-resected group. The maxillary third incisor erupted through the implanted area. However, in the rhBMP-2 (10 μg/100 μL)-treated group, more osteoinductive activity and significantly higher BMD were observed in the newly generated bone. The maxillary third incisor erupted mesially so as to avoid this implanted area.

Conclusion

Results showed that bone remodeling and tooth eruption through the newly generated bone appear to be dose dependent. Accordingly, proper dosage of rhBMP-2 should be determined for successful clinical application.