bluuebunny/crossref_metadata_2025 huggingface dataset:
The Treatment of Craniosynostosis: An Ethical Perspective
Authors: Ward Christopher
Published: October 2018

The parents of children with craniofacial deformity have expectations that cannot always be reasonably met in a world of clinical uncertainty. In order to bridge the “reality gap,” the members of the cranofacial team must be open and honest in discussing the known harms and benefits of a proposed treatment, so that a relationship of trust evolves between the health care professional and the patient/parent. It is only through this trust that a truthful implementation of consent, within its moral framework, can be achieved. This, in turn, requires an analysis of the outcomes of the various options for treatment as well as evidence that the cranofacial team is able to provide high standards of care. All this leads to the ethical imperative of respecting the right of parents to make an informed choice and allowing them to see that their child is treated in a way that provides maximum benefit and minimum harm.
Temporal Characteristics of Velopharyngeal Function in Children
Authors: Leeper Herbert A. , Tissington M. Lynn , Munhall Kevin G.
Published: October 2018

Objective

This investigation was designed to examine the performance of children with normal speech on temporal aspects of aerodynamic tasks related to velopharyngeal closure.

Design

The investigation was a descriptive evaluation of variability in aerodynamic features related to velopharyngeal function during multiple repetitions of the word “hamper.”

Setting

Children without speech or velopharyngeal difficulties were seen in an experimental laboratory setting for the evaluation procedures.

Participants

Twenty-seven subjects were recruited for the experiment. Three subjects were rejected because of behavioral difficulties, and the remaining 24 subjects were subdivided into 4 groups of 6 children (3 males and 3 females) aged 3, 6, 9, and 12 years. The children, who were from local schools and day care centers, volunteered to participate in the experiment. All of the children had age-appropriate speech, language, and hearing abilities, as determined by screening tests administered by one of the examiners (L.T.).

Main Outcome Measures

Mean and variability of pressure-flow measures of peak intraoral air pressure and peak nasal airflow and the temporal measures accompanying each air pressure or airflow pulse were evaluated for the age groups of children examined in the experiment.

Results

The aerodynamic procedures employed to evaluate velopharyngeal closure during speech were reliable for use with young children. There was a numerical trend toward decreased duration of the temporal parameters with increasing age. Thus, children demonstrated durational values similar to those previously reported for normal-speaking adults. In general, peak oral air pressure and nasal airflow values were like those of previous investigations and demonstrated low variability across all age groups of children tested.

Conclusions

The data from the present investigation provide a preliminary base for comparison of temporal features of velopharyngeal closure for the aerodynamic evaluation of children with impaired velopharyngeal function.
Short-term Cost-effectiveness Analysis of Presurgical Orthopedic Treatment in Children with Complete Unilateral Cleft Lip and Palate
Authors: Severens Johan L. , Prahl Charlotte , Kuijpers-Jagtman Anne M. , Prahl-Andersen Birte
Published: October 2018

Objective

The aim of this article was to investigate cost-effectiveness in cleft palate treatment using cost-effectiveness of presurgical orthopedic treatment (PSOT) as an example.

Design

A three-center randomized clinical trial compared PSOT with non-PSOT for children with unilateral cleft lip and palate (UCLP, n=52).

Patients

The inclusion criteria for the trial were: complete UCLP, no other malformations, born at term, both parents Caucasian, trial entrance preferably within 2 weeks after birth, and informed consent by the parents.

Interventions

PSOT was performed by means of a passive plate according to Hotz and Gnoinski.

Main Outcome Measure

The short-term cost-effectiveness of PSOT was based on the time taken for the surgical lip closure procedure. Medical and nonmedical costs until surgical lip closure at 18 weeks of age were analyzed.

Results

The durations of the surgical lip closure procedures did not differ significantly (57.2 minutes for PSOT and 56.4 minutes for non-PSOT). The mean medical cost for PSOT treatment was US$852. The non-PSOT treatment group had a significantly different mean medical cost (US$304). Mean travel costs and indirect nonmedical costs were US$128 and US$231 for PSOT and US$79 and US$130 for non-PSOT, respectively.

Conclusions

The combination of a clinical trial and an economic evaluation makes it possible to relate effects to costs involved in treatment alternatives. Longer-term costs and effects will be incorporated into an extended cost-effectiveness analysis to determine the cost-effectiveness of PSOT.
Location and Distribution of Epithelial Pearls and Tooth Buds in Human Fetuses with Cleft Lip and Palate
Authors: Arnold W.H. , Rezwani T. , Baric I.
Published: October 2018

Objective

The purpose of this study was to establish the location and distribution of epithelial pearls and tooth buds in cleft palate fetuses, relative to the time of palate fusion.

Design

The facial skeletal structures, dental laminae, tooth buds, and epithelial pearls were examined in seven spontaneously aborted human fetuses, of which five had unilateral or bilateral cleft lip and palate or cleft palate. The sectioned fetuses were reconstructed by 3D-computer technology.

Results

Epithelial pearls were found in four of the investigated cases, of which one was a control specimen. They were located at the margins of the palatal shelves. In the cleft lip and palate cases, the cleft was found in the premaxilla between the first and second incisor tooth. The premaxilla was found to be hypoplastic in both bilateral cleft lip and palate cases and was totally absent in the unilateral cleft lip and palate case. The maxilla was hypoplastic in one case with unilateral cleft lip and palate. In all other specimens, it was developed symmetrically.

Conclusions

The results indicate that cleft lip and palate development may occur after the fusion of the frontonasal prominence with the maxillary prominence and the palatal shelves, as well as a nonfusion of the palatal shelves in the secondary palate.
HONC Measures in 4- to 6-Year-Old Children
Authors: Mra Zan , Sussman Joan E. , Fenwick Julie
Published: October 2018

Objective

To collect normative data using Horii's Oral Nasal Coupling Index (HONC) from 4- to 6-year-old children without cleft palate to be used in the evaluation of young children with cleft palate. In addition, to determine whether HONC values in children are similar to those of adults and thus show that the HONC ratio successfully normalizes nasal accelerometric signals across age, gender, and vocal intensity.

Design

Measurement of accelerometric and acoustic signals from novel nasal and nonnasal utterances, which the children repeated after the experimenter. Measurements also included four sustained [m] productions, which were used to calibrate correction factors used to equate nasal and oral signals during a sustained [m] production.

Setting

Laboratory at a state university.

Participants

Ten girls and 10 boys, aged 4 to 6 years, with normal speech, language, and hearing.

Results

Differences of 13 dB (HONC) were found to separate nasal from nonnasal sentences. No significant difference in HONC score was found across gender for nasal/nonnasal sentences and [m] productions. The correction factors generated during [m] calibration procedures did not differ between girls and boys.

Conclusions

Horii Oral Nasal Coupling Index differences between nasal and nonnasal utterances appear to be valid and reliable measures in both children and adults for detection of disorders of nasal resonance.
Conversational Skills of Preschool and School-Age Children with Cleft Lip and Palate
Authors: Chapman Kathy L. , Graham Karen Tecco , Gooch Janet , Visconti Colleen
Published: October 2018

Objective

The purpose of this study was to examine the conversational skills of preschool and school-age children with cleft lip and palate.

Design

The children were audio- and videotaped during interactions with an unfamiliar adult. In addition, standardized measures of speech and language were administered, and ratings of resonance were obtained. Comparisons were made between the children with cleft lip and palate and their same-age peers on measures of conversational participation and a standardized test of pragmatic skills.

Participants

Participants were 20 children with unilateral cleft lip and palate (10 preschoolers and 10 school-age children) recruited from the Craniofacial Team at Rainbow Babies and Children's Hospital, Cleveland (OH) and 20 noncleft peers matched for gender, age, and socioeconomic status.

Main outcome Measures

Separate comparisons were made for the preschool children with cleft lip and palate and their noncleft peers, and the school-age children with cleft lip and palate and their noncleft peers on eight measures of conversational assertiveness/responsiveness and the standardized tests of pragmatics. Next, each child with cleft lip and palate was classified for level of conversational participation.

Results

Paired t tests revealed no significant differences between the preschool and school-age children with cleft lip and palate and their noncleft peers in level of conversational participation. However, individual child comparisons revealed less assertive profiles of conversational participation for 50% of the preschool and 20% of the school-age children with cleft lip and palate.

Conclusions

Children with cleft lip and palate may show a less assertive style of conversational participation, at least during the preschool years. Therefore, craniofacial team evaluations should include examination of conversational competency, particularly for children who are demonstrating difficulty with other aspects of speech, language, or social development.
Psychosocial Adjustment in Twin Pairs with and without Hemifacial Microsomia
Authors: Maris Catherine L. , Endriga Marya C. , Omnell M. Lena , Speltz Matthew L.
Published: October 2018

Objective

To compare the psychosocial adjustment of children with and without hemifacial microsomia (HFM).

Design

This cross-sectional pilot study investigated psychosocial adjustment of twin pairs in which one twin was affected with HFM and the other was unimpaired. Data were analyzed using paired t tests.

Setting

The dental medicine clinic of a large urban children's hospital, serving a multistats area.

Participants

Participants were six twin pairs aged 9 to 15 years and their parents. Data were also collected from classroom teachers.

Main outcome Measures

Outcome measures included: the Child Behavior Checklist (CBCL), the Teacher Report Form (TRF), the Coopersmith Self-Esteem Inventory (SEI), and a structured self-concept interview (the Self-Interview).

Results

Children with HFM had significantly higher CBCL and TRF total behavior problem scores and lower SEI general self-esteem scores than their unimpaired twins. No differences were found in CBCL competence scores. Interview data revealed concerns among the HFM group related to appearance, negative social responses, and fear of hospital procedures. Children also reported positive aspects of coping with HFM.

Conclusions

The results of this study suggest that, similar to other craniofacial conditions, HFM is associated with an elevated risk for childhood psychosocial difficulties. Replication of specific findings with a larger sample is needed. Multicenter, collaborative studies are a crucial next step for this field.
A Teaching Device for Furlow Palatoplasty
Authors: Matthews Martha S.
Published: October 2018

The Furlow double-opposing z-plasty technique of palate repair has become a common treatment for cleft palate. The procedure is more complicated than other methods of palate closure and is difficult for the trainee to visualize; It Is even more difficult for the layperson without prior understanding of the concept of z-plasty to understand. A simple, inexpensive, portable model is described that shows the key elements of the repair and can be used for parent, patient, student, and resident teaching.
A Finite Element Model of the Soft Palate
Authors: Berry David A. , Moon Jerald B. , Kuehn David P.
Published: October 2018

Objective

As a step toward better understanding of normal and abnormal velar control, a finite element model of the soft palate was developed.

Design

A static two-dimensional midsagittal model of the velum was given physical dimensions to match that of a 10-year-old boy. Biomechanical properties of the tissues were inferred based on previous histologic studies. Velar movements were induced by the influence of three extrinisic velar muscles: the levator veli palatini, the palatoglossus, and the palatopharyngeus, which were simulated as external forces acting on the velar model.

Results and Conclusions

Velopharyngeal opened and closed positions were simulated as well as a variety of intermediate steps between the two configurations. Velopharyngeal closure was also simulated in a manner appropriate for both high and low vowels. Future extensions of the model will incorporate the muscles as an intrinsic component of the model and will include a full time-dependent implementation, including inertial effects. Future studies will compare model predictions with experimental data from the laboratory, including both kinematic data and velopharyngeal closure forces.
Brain Growth Rates in Craniosynostotic Rabbits
Authors: Cooper Gregory M. , Mooney Mark P. , Burrows Annie M. , Smith Timothy D. , Dechant Jason , Losken H. Wolfgang , Marsh Jeffrey L. , Siegel Michael I.
Published: October 2018

Objective

It has been suggested that abnormal brain morphology or growth rates may be a primary causal factor of craniosynostosis due, in part, to a lack of normal growth stretch and tension at the suturai margins. The purpose of the present study was to quantify cerebral hemisphere morphology and growth in a rabbit model of nonsyndromic coronal suture synostosis to determine whether cerebral dysmorphology is primary or secondary to synostosis in this model.

Design

Fifty-seven brains (114 hemispheres) were examined from 40 normal control rabbits and 17 rabbits with bilateral coronal suture synostosis ranging in age from 25 to 450 days postconception (synostosis occurs at approximately 23 days postconception in this model). The calvariae were removed, the brains were fixed in 10% paraformaldehyde, and in situ bilateral measurements of cerebral hemisphere length and cerebral hemisphere width were obtained using a Wild microscope with a camera lucida attachment and digital caliper. Regression analysis was used to compare cerebral cortex growth rates by age between the two groups.

Results

Cerebral hemisphere width and cerebral index regression line slopes had similar y intercepts (23 day postconception) with significantly (p < .05) diverging slopes over time. Normal rabbits increased more rapidly than synostosed rabbits. No significant (p > .05) differences were noted in regression line slopes between groups for cerebral hemisphere length by age or length by width.

Conclusions

Cerebral dysmorphologies are probably a compensatory, secondary (postsynostotic) event and not a primary causal factor of craniosynostosis in this rabbit model of human familial, nonsyndromic coronal suture synostosis.
The Efficacy of Perioperative Steroid Therapy in Pediatric Primary Palatoplasty
Authors: Senders Craig W. , Di Stephen M. , Brodie Hilary A. , Emery Brian E. , Sykes Jonathan M.
Published: October 2018

Objective

To determine if perioperative steroid therapy benefits pediatric primary palatoplasty patients.

Design

A prospective, double-blind, randomized trial with a placebo control.

Setting

An academic medical center.

Patients

Forty-five children undergoing primary repair of their cleft palate between 1989 and 1996 who were under the age of 48 months and without developmental delay or any associated syndrome.

Interventions

Intravenous dexamethasone sodium phosphate (0.25 mg/kg) or a placebo (5% dextrose in water) immediately preoperatively and once every 8 hours for two doses postoperatively.

Main outcome measures

Postoperative airway distress, fever, oral fluid intake, discharge eligibility, and palatal fistula formation.

Results

Perioperative steroid therapy significantly reduced the incidence of postoperative airway distress (p = .05) and postoperative fever (p = .02); postoperative oral fluid intake, discharge eligibility, and palatal fistula formation were not significantly affected.

Conclusions

Perioperative steroids effectively lower the risk of postoperative airway distress and postoperative fever in children undergoing the primary repair of their cleft palate. This finding favors a customary role for perioperative steroid therapy in pediatric primary palatoplasty.
A Multicenter Retrospective 3D Study of Serial Complete Unilateral Cleft Lip and Palate and Complete Bilateral Cleft Lip and Palate Casts to Evaluate Treatment: Part 1—The Participating Institutions and Research Aims
Authors: Berkowitz Samuel
Published: October 2018

Objective:

To compare the multicenter retrospective and prospective spaciotemporal (4D) serial analyses of complete unilateral (CUCLP) and complete bilateral (CBCLP) cleft lip and palate casts that had undergone different treatment procedures. The involved institutions are Miami Craniofacial Anomalies Foundation, South Florida Cleft Palate Clinic; University Hospital of Nijmegen Cleft Palate Center; Free University of Amsterdam Cleft Palate Center; Academic Hospital (Dijkzigt/Sophia) Rotterdam Cleft Palate Center; Center for Craniofacial Anomalies, University of Illinois College of Medicine; Cleft Palate Center, Sahigrenska University Hospital, Göteborg, Sweden; and Children's Memorial Medical Center, Northwestern University Cleft Palate Institute.

Design:

Using serial casts of the upper jaw and an electromechanical digitizer with special CadCam software (CadKey), the occlusal relationships and morphometric palatal growth changes that occur under the influence of presurgical orthopedics and various surgical procedures will be studied. It is anticipated that 3D geometric data extracted from serial casts will identify the important geometric palatal parameters present before cleft surgical closure, which will supply objective criteria for establishing a scientific basis for improved surgical therapy. This research study will test three hypotheses: (1) Conservative (varying the timing of surgical cleft closure according to the size of the cleft space) lip and palatal surgery will permit “catch-up” palatal growth and normalize palatal growth and development. (2) The amount of mucoperiosteal tissue relative to the size of the cleft space is important in determining the timing of palatal surgery, as it influences the degree of scarring and ultimately the palate's adult size and form. (3) Presurgical orthopedics (the use of appliances soon after birth) can stimulate palatal growth beyond its normal growth potential.

Results:

In a previous project and again after reviewing the data already collected during the first year of this study, it has been shown that incremental changes in size of palatal segments in CUCLP and CBCLP cases prior to surgery vary slightly. The CBCLP cases grow slightly faster than CUCLP cases before surgery, but growth of the CBCLP cases decreases in acceleration after surgery. Reasons for these differences will be determined when more cases are analyzed and subjected to biostatistical analysis.
Dentoalveolar Growth Inhibition Induced by Bone Denudation on Palates: A Study of Two Isolated Cleft Palates with Asymmetric Scar Tissue Distribution
Authors: Ishikawa Hiroyuki , Iwasaki Hiroshi , Tsukada Haruka , Chu Soowon , Nakamura Shinji , Yamamoto Katsuyuki
Published: October 2018

Objective:

This report presents two cases of isolated cleft palate with asymmetric distribution of postsurgical scar tissue determined by laser Doppler flowmetry. To determine the effect of mucoperiosteal denudation of the bone on maxillary alveolar growth, the analysis of dentoalveolar structures compared the affected side to the unaffected side of each case.

Method:

Two Japanese girls with isolated cleft palates were examined. Both subjects had undergone pushback operations (a modified version of the procedure of Wardill) for palatal repair at 18 months of age. Palatal blood flow was examined by laser Doppler flowmetry when the girls were 12 years old to determine the extent of postsurgical scar tissue over the denuded bone. To analyze the maxillary dentoalveolar structures three dimensionally, the whole surface of the upper dental cast was measured and recorded by an optical measuring device when the girls were 7 years old.

Results and Conclusions:

Analysis via flowmetry showed that the palatal scar tissue area was limited to the anterior tooth region on the right (unaffected) side but extended posteriorly to the premolar region on the left (affected) side in both subjects. The two girls had similar dentoalveolar structures, with the dental and alveolar arches deflected lingually at the deciduous molar area on the affected side. There were no differences in the buccolingual inclination of deciduous molars or in the vertical growth of the alveolar processes between the affected and unaffected sides. In both girls, bone denudation in the premolar region appeared to result in less than 3 mm of displacement of the teeth palatally, with no change in lingual inclination. Any effects of scar tissue on the vertical development of the alveolus were not substantiated.
Dentoalveolar Relations in Children Born with a Unilateral Cleft Lip and Palate (UCLP) in Western Australia
Authors: Johnson Nicola , Williams Alison C. , Singer Steven , Southall Peter , Atack Nikki , Sandy Jonathan R.
Published: October 2018

Objective

Our objective was to evaluate complete unilateral cleft lip and palate repair outcome in the Cleft Unit in Perth, Western Australia, by assessment of dentoalveolar relationships.

Design

This is a retrospective study.

Setting

Our subjects were individuals under the care of the cleft team in Perth, Western Australia.

Participants

All patients with unilateral cleft lip and palate and available 6-year casts who had been born since January 1, 1985, were identified from the cleft unit's database. The nature of the cleft was verified by examination of birth study models and photographs. A total of 54 such patients were identified.

Main outcome Measures

Main outcome measures were identified through dental arch relationship grading of study models using the 5 Year Old Study Model Index.

Results

Interexaminer and intraexaminer agreement kappa statistics revealed good to very good agreement using this index. The results indicate that the surgical outcome was graded as excellent, good, or fair for 77% of patients and poor or very poor for 23% of patients.

Conclusions

The results of the Western Australia study compare favorably to the overall U.K. outcome (the Clinical Standards Advisory Group study) but unfavorably to the results of some European centers, such as Oslo.
The Adult Unoperated Cleft Patient: Absence of Maxillary Teeth outside the Cleft Area
Authors: Lekkas C. , Latief B.S. , Ter Rahe S.P.N. , Kuijpers-Jagtman A.M.
Published: October 2018

Objective

The purpose of this study was to investigate the possible absence of teeth in the postcanine region of the upper jaw of the unoperated adult cleft patient.

Method

The study was performed on 266 dental casts of fully unoperated adult cleft patients. The patients were divided into four groups according to the type of the cleft: unilateral cleft lip and alveolus, unilateral cleft lip and palate, bilateral cleft lip and alveolus, and bilateral cleft lip and palate.

Results

No absence of permanent teeth in the canine and postcanine area of the upper jaw could be found.

Conclusion

The results are in contradiction with the established hypothesis that absence of teeth outside the cleft area of the maxilla is due to an unknown congenital factor. On the contrary, the findings support the hypothesis that surgery for the closure of the hard palate in early childhood is the most important etiological factor for the absence of teeth outside the cleft area in the early operated cleft patient. The superficial position of the tooth germs (at the time of the palatal surgery), especially those of the premolars, supports this hypothesis.
Radiographic Determination of the Position of the Maxillary Lateral Incisor in the Cleft Alveolus and Parameters for Assessing its Habilitation Prospects
Authors: Shashua Dafna , Omnell M. Lena
Published: October 2018

Objective

Develop patient selection criteria and guidelines for the diagnostic work-up for early secondary alveolar bone graft designed to accommodate a lateral incisor in the cleft.

Design

A literature review with clinical examples.

Interventions

For the majority of cleft lip and palate (CLP) patients, the timing of the secondary alveolar bone graft procedure is devised to accommodate the eruption of the maxillary canine. However, when the lateral incisor is present in the cleft area, a decision may be made to perform early grafting in order to salvage an additional tooth. Appropriate case selection for this intervention is the key for successful dental habilitation in the cleft area.

Results and Conclusions

Attempts to maintain the lateral incisor are recommended only if the tooth contributes to the comprehensive orthodontic and restorative patient care and when the presence of a lateral incisor with adequate size, shape, and proper position may be demonstrated. For optimal decision making, the position, form, and periodontal ligament support of the lateral incisor should be assessed. Evaluation of these parameters is based chiefly on radiographic imaging. Occlusal projections and traditional tomography may augment the information available from periapical and panoramic radiographs. However, each of these has benefits and drawbacks that require consideration.
Combined Bone Grafting and Delayed Closure of the Hard Palate in Patients with Unilateral Cleft Lip and Palate: Facilitation of Lateral Incisor Eruption and Evaluation of Indicators for Timing of the Procedure
Authors: Lilja Jan , Kalaaji Amin , Friede Hans , Elander Anna
Published: October 2018

Objective

To compare outcomes of bone grafting performed before eruption of the lateral incisor to outcomes of grafting performed before eruption of the canine and to evaluate the long-term results of bone grafting combined with delayed closure of the hard palate during mixed dentition.

Design

Seventy consecutive patients (52 men and 18 women) with complete unilateral cleft lip and palate were studied. All patients underwent bone grafting with simultaneous closure of the cleft in the hard palate at the stage of mixed dentition. The velum had been repaired in infancy. Mean age for the bone grafting procedure was 8.4 years. Bone grafting was performed to facilitate eruption of the lateral incisor in 43 (61%) of the patients and to facilitate eruption of the canine in the remaining 27 (39%) patients. Intraoral radiographs were used to evaluate the morphologic characteristics of the cleft and the stage of eruption of the permanent lateral incisor and canine before bone grafting. Mean follow-up time was 4.0 years (range, 1–10.1 years).

Results

The mean time for the surgery, which included bone grafting and repair of the residual cleft in the hard palate, was 109 minutes, and the mean amount of bleeding was 121 ml. The rate of dehiscence in the flap covering the alveolar bone graft was 14%, and the rate of total failure of bone grafting was 3%. An oronasal fistula developed in the hard palate of 13% of patients, but the fistula was of sufficient size to serve as an indication for reoperation in only 6%. The postoperative alveolar bony height in the cleft area was more than 75% of the normal height in 94% of patients. Closure of the cleft space in the dental arch was performed or planned to be achieved orthodontically in 91% of patients. When bone grafting was performed to facilitate eruption of the lateral incisor, the cleft space was closed orthodontically in 100% of patients. The optimal indicator for timing of the bone grafting procedure from an orthodontic point of view was when the permanent lateral incisor or the canine close to the cleft was covered by a thin shell of bone (i.e., 7–9 years of age).
Modification of the Koken Nasal Splint
Authors: Cobley T.D.D. , Orlando A. , Page K. , Mercer N.S.G.
Published: October 2018

Koken nasal splints have been found effective postoperatively in treating cleft-lip nose deformity. In our experience, their retention has been poor, so reducing the molding effect. This paper presents a modification designed to overcome the problem of retention.
Closure of Palatal Fistula with a Local Mucoperiosteal Flap Lined with Buccal Mucosal Graft
Authors: Honnebier M.B.O.M. , Johnson D.S. , Parsa A.A. , Dorian A. , Parsa F.D.
Published: October 2018

Objective

Oro-nasal fistula is a common complication of palatoplasty. Current methods for fistula repair utilize mucoperiosteal flaps or pedicled flaps. These procedures are often cumbersome and leave a raw nasal surface, which may increase the incidence of postoperative risks and problems. In addition, the recurrence rate of the fistula is as high as 34%. We propose a simple two-layer method of fistula repair to avoid recurrences.

Design

A standard mucoperiosteal flap is raised on the oral side. A buccal mucosal graft is harvested from the cheeks and sutured to the nasal side of the flap that is then inset into the fistula.

Setting

Patients were either referred to the senior author's private practice (four patients) or were patients who had previously been operated on by the senior author himself (three patients).

Subjects

Study subjects consisted of seven patients, four males and three females, ages 14 months to 8 years. All patients had previously undergone cleft palate repair, complicated by subsequent oro-nasal fistula formation.

Interventions

All patients underwent oro-nasal fistula repair under general anesthesia with a local mucoperiosteal flap lined with buccal mucosal grafts placed on the nasal side of the flap.

Results

In all cases, the fistula was completely closed at first attempt without complications. Patients were followed for a minimum of 2 years, without evidence of recurrence.

Conclusions

Our proposed surgical procedure for fistula closure using a standard mucoperiosteal flap lined with a buccal mucosal graft is a suitable alternative for the repair of postpalatoplasty oro-nasal fistulas. Further study and long-term follow-up is needed to establish this method as a new standard form of repair.
The Anchor of the Nasal Ala in Cleft Lip-Nose Patients: A Morphological Description and a New Surgical Approach
Authors: Morselli Paolo G.
Published: October 2018

Objective

The purpose of this article is to describe a morphological condition that is readily seen in both primary and secondary deformed noses of cleft patients and to present a new surgical technique to correct this specific deformity that afflicts the alar dome. This anomaly resembles a taut cord-like tissue that runs vertically from the pyriform aperture and is anchored onto the alar cartilage, thus impeding the cartilage's normal growth process. This new surgical technique releases the anchor and corrects the deformity of the nasal rim by using a triangular flap V-Y advancement technique.

Results

From 1994 to 1997, 88 cleft lip-nose cases were operated on by the author using the triangular flap V-Y advancement technique for releasing the anchor. Forty-two cases were primary repairs and 46 were secondary repairs.

Conclusions

By studying the normal and pathological anatomy of the nose and its proportions, the surgeon can obtain ideas for new surgical corrective techniques that can be used to restore anatomical balance and harmony between the nose and the face. Only in the last 15 to 20 years has the plastic surgeon begun to be interested in primary rhinoplasty in cleft lip-nose patients. Within this time, many different techniques have been proposed. All primary and secondary cases that have undergone this specific technique for release of the anchor have shown good results for restoring the height of the alar dome's affected side to that of the noninvolved side, thus establishing a more anatomically balanced nose.
Skull Base Factors in Relation to Hearing Impairment in Cleft Palate Children
Authors: Carrie S. , Sprigg A. , Parker A.J.
Published: October 2018

Objective

This investigation was performed to determine if an easily measurable, reproducible, bony parameter could be identified that might predict hearing loss in cleft palate children.

Subjects

In this prospective study performed at the Sheffield Children's Hospital (U.K.), 34 children with successfully repaired cleft palate who responded to a postal request for volunteers were assessed clinically, audiologically, and by lateral soft tissue neck radiography. Six children were excluded because of previous otologic surgery or poor quality radiographs. Twenty-six children who had the same series of investigations were randomly selected from routine otolaryngological outpatient clinics and acted as controls.

Methods

The sphenopalatine angle (SPA), which relates the facial and cranial components of the skull, was measured on each child's radiograph.

Results

The median SPA in the cleft palate group was significantly greater than in the control group (p = 0.01). In those cleft palate children with a hearing loss the sphenopalatine angle was smaller than in their normal hearing counterparts (p = 0.01). No significant difference was found in the SPA between the hearing loss and normal hearing controls. There was no significant difference in age ranges between the hearing and hearing loss subgroups in each of the two primary groups.

Conclusions

In this study, those cleft palate children with a smaller SPA have a greater incidence of hearing loss.
Comparison between Palatal Configurations in UCLP Infants with and without a Hotz Plate until Four Years of Age
Authors: Mishima Katsuaki , Mori Yoshihide , Sugahara Toshio , Minami Katsuhiro , Sakuda Masayoshi
Published: October 2018

Objective

To evaluate the efficacy of presurgical orthopedic treatment three dimensionally, we previously developed a novel measuring system and described the efficacy of the treatment until 18 months of age. The aim of this study was to compare palatal configuration in complete unilateral cleft lip and palate (UCLP) infants fitted with a Hotz plate to those without it until four years of age using the same measuring system.

Patients

Twenty complete UCLP infants were divided into two groups. A Hotz plate was used with 12 patients who first visited our clinic from January 1990 to March 1992 and was not used with an additional eight patients who first visited from April 1992 to July 1993.

Main outcome Measures

Palatal casts taken from birth until four years of age were measured three dimensionally and were compared between the two groups.

Results and Conclusions

The width of the palate was larger in the group with a Hotz plate than in the group without it at four years of age. No difference was observed between the anteroposterior distance of the palate in the two groups at four years of age. Similarly, the alveolar arch form did not differ between the two groups. The degree of curvature of the palatal surface was smaller in the group with a Hotz plate than in the group without it. These data were similar to data obtained at 18 months of age.
An Epidemiologic Study of Oral Clefts in Iran: Analysis of 1669 Cases
Authors: Rajabian Mohammad Hossain , Sherkat Mehdi
Published: October 2018

Objective

The objective of this study was to assess the epidemiology and some genetic aspects of oral clefting in Iran.

Design

The study was a 15-year cross-sectional (prevalence) study from August 1976 to September 1991.

Setting

The setting for the study was two plastic surgery departments, both mostly referral centers, and a maternity hospital in Iran.

Participants

The participants were 1669 consecutive surgical cases with oral clefts (79% between 1 day and 18 months of age), registered in two centers. In a parallel study, 19,369 livebirths born in a maternity hospital within the same period were surveyed to ascertain prevalence of clefting at birth. Normal children of this population were used as the control group. Samples were analyzed by prevalence of clefting at birth, type of oral cleft, side of clefting, sex ratio, associated malformations, clefts in relatives, and parental consanguinity.

Results

The prevalence of clefts was 1.03 per 1000 births. Cleft lip (without cleft palate) had a higher (34.9%) and cleft palate alone had a significantly lower prevalence (17.4%) than expected. Cleft lip (without cleft palate) was more common in females (53.6%) than in males (46.4%). The rate of associated malformations in cleft patients (7.73%) was higher than in controls (0.093%). Association between clefting and consanguinity was significant (45.8% in cases versus 43.1% in controls). Occurrence of noncleft congenital malformations among first-degree relatives of our cases (2.77%) was nearly two times that of controls (1.55%).

Conclusion

Our study reveals that the population incidence of oral clefts in Iran is much closer to Europian than Arab-African or South East Asian countries. However, significant variations in other epidemiologic and some genetic features were observed.
Are Infants with Orofacial Clefts at Risk for Insecure Mother-Child Attachments?
Authors: Maris Catherine L. , Endriga Marya C. , Speltz Matthew L. , Jones Karen , Deklyen Michelle
Published: October 2018

Objective

Several risk factors in the early lives of children with clefts are believed to interfere with their development of secure attachments to parents; however, this possibility has rarely been studied empirically. This study compared 12- and 24-month attachment classifications of infants with cleft palate (CP), infants with cleft lip and palate (CLP), and a comparison group of unimpaired infants (COMP).

Method

Twenty-two CP infants, 24 CLP infants, and 61 matched COMP infants were assessed at 12 and 24 months of age in an urban children's hospital. At both visits, mothers and infants participated in the Strange Situation, which was videotaped and subsequently coded for patterns of attachment behavior.

Results

CP infants displayed a lower rate of 12-month attachment security than infants in the CLP or COMP groups. By 24 months, no diagnostic group differences in attachment classification were found. Stable 12- to 24-month attachment classifications were less likely in the CP group (36.3%) than in the COMP (62.3%) group. CP infants who were insecure at 12 months were more likely to become secure by 24 months than were CLP or COMP group infants.

Conclusions

In contrast to previous theory and clinical speculation, the facial appearance of infants with CLP does not appear to affect the early mother-infant relationship adversely. The infancy period is marked by attachment instability for infants with CP, who demonstrated lower-than-expected rates of security at 12 months. However, these problems resolved in nearly all cases by 24 months of age. Most infants with clefts emerged from the first 2 years of life with secure maternal attachments.
Age-Related Changes in Intracranial Pressure in Rabbits with Uncorrected Familial Coronal Suture Synostosis
Authors: Fellows-Mayle Wendy K. , Mooney Mark P. , Losken H. Wolfgang , Dechant Jason , Cooper Gregory M. , Burrows Annie M. , Smith Timothy D. , Pollack Ian F. , Siegel Michael I.
Published: October 2018

Objective

Chronic, elevated intracranial pressure (ICP) in craniosynostotic infants may result in ocular and neurocapsular problems; however, not all infants exhibit elevated ICP. Clinical ICP studies are further confounded by small and heterogeneic samples, multiple-suture involvement, and varying surgical management protocols. The present study was designed to describe longitudinal changes in ICP in a large, homogenous sample of rabbits with uncorrected familial, nonsyndromic coronal suture synostosis.

Methods

Ninety-one rabbits were divided into four groups: (1) normal rabbits (n = 28), (2) rabbits with delayed-onset coronal suture synostosis (DOCS; n = 25), (3) rabbits with unilateral coronal suture synostosis (UCS; n = 12), and (4) rabbits with bilateral coronal suture synostosis (BCS; n = 26). ICP was measured at 24 and 42 days of age using a Codman epidural microtransducer.

Results

Rabbits with BCS had a significantly (p < .05) higher mean ICP at 25 days of age than rabbits in the other three groups by approximately 146%. However, by 42 days of age, mean ICP in normal control rabbits and rabbits with DOCS was significantly (p < .01) increased compared with their mean ICP values seen at 25 days of age, while mean ICP in BCS rabbits significantly (p < .01) decreased (by 32%) over the same time period. ICP in rabbits with UCS was between that seen in normal control rabbits and rabbits with BCS and did not significantly (p > .05) change over time.

Conclusions

These findings suggest that the degree of suture involvement may be related to early increases in ICP. Possible multifactorial explanations for intracranial decompression and compensation in the craniosynostotic rabbit model are discussed.
Dental Arch Relationships in Yorkshire Children with Unilateral Cleft Lip and Palate
Authors: Morris David O. , Roberts-Harry Dai , Mars Michael
Published: October 2018

Objective

To assess the dental arch relationships of children with a complete unilateral cleft lip and palate (UCLP), born consecutively between 1983 and 1987, who had undergone primary cleft repair in the West Yorkshire region of the United Kingdom. The treatment outcome of this UCLP sample was then compared with the results of a previously published intercenter European study.

Design

A retrospective study with standardized record collection and blind assessment.

Setting

Regional dental hospital providing secondary and tertiary health care to the local population.

Patients

35 UCLP patients whose primary repair had been performed within West Yorkshire.

Interventions

Dental casts obtained for each subject within 12 months of their 10th birthday.

Main outcome Measures

The West Yorkshire models were randomly mixed with an anonymous sample of 115 UCLP cases from other cleft centers in the U.K. The dental arch relationships of the 10-year-old models were assessed by applying the Goslon Yardstick.

Results

One-third of the West Yorkshire cleft sample were rated as belonging to Goslon group 2 (good result), another third to group 3 (fair result), and the remaining third to groups 4 and 5 (poor/very poor result). Numerically, the results were of a slightly higher standard than that previously published for other U.K. and European cleft centers but were inferior to the treatment outcome of two European centers. No statistically significant differences were found between the outcome results of the West Yorkshire group and the other cleft centers.

Conclusions

These results provide useful baseline data against which progress in achieving improved treatment outcome for West Yorkshire UCLP children can be determined by future research.
Lipoma of the Cleft Soft Palate: A Case Report of a Rare Congenital Anomaly
Authors: Mahabir Raman C. , Mohammad Jamal A. , Courtemanche Douglas J.
Published: October 2018

Objective

Congenital tumors of the oral cavity are extremely uncommon. The teratoid tumors (epiganthi, dermoid, and hairy polyps) account for almost all of the reported cases and are not infrequently associated with cleft of the soft palate. Of the remaining tumors in the pediatric cleft palate population, infantile lipoma of the oral cavity is exceedingly rare. A case of a congenital lipoma associated with a cleft of the soft palate is presented. The review of the literature, the description of the lesion, the diagnosis, and the management of this finding are outlined.
The Influence of Surgery and Orthopedic Treatment on Maxillofacial Growth and Maxillary Arch Development in Patients Treated for Orofacial Clefts
Authors: Kuijpers-Jagtman Anne M. , Long Ross E.
Published: October 2018

This paper describes maxillofacial growth and dental arch development in relation to different orthodontic and surgical interventions utilized in the management of orofacial clefts. It addresses presurgical orthopedic treatment in patients with unilateral cleft lip and palate, primary periosteoplasty, primary bone grafting, lip surgery, palatal surgery, pharyngeal flap surgery, maxillary protraction, and early secondary bone grafting. Difficulties associated with synthesizing previously published data and suggestions for approaching still unanswered questions in the next millennium are presented.
The Origin and Development of the Upper Lateral Incisor and Premaxilla in Normal and Cleft Lip/Palate Monkeys Induced with Cyclophosphamide
Authors: Wei Xin , Senders Craig , Owiti G. O. , Liu Xiaoming , Wei Zhen-Nian , Dillard-Telm Lisa , Mcclure Harold M. , Hendrickx Andrew G.
Published: October 2018

Objective

Cleft lip/palate (CLP) is a common human congenital defect in which the maxillary lateral incisors are often absent, malformed, and malpositioned. The present study was designed to examine the origin of the upper primary lateral incisor relative to the medial nasal process (MNP) and maxillary process (MP) fusion area and to the premaxillary/maxillary (incisive) suture in monkeys.

Method

Scanning electron microscopy, histology, skeletal staining, and drying techniques were used to study facial development in embryo and fetal monkey specimens. A teratogenic dose of cyclophosphamide was administered to pregnant monkeys prior to fusion of the MNP and MP and fetuses were examined for CLP.

Results

Formation of the anterior maxilla involved fusion of the MNP and MP at stages 14–18. At stages 18–20, the palatal portion of the MNP had formed the medial and lateral incisive mounds. By stage 22, the upper primary lateral incisor has formed within the MP, lateral to the MNP/MP fusion area and to the ossifying premaxilla. Ossification of the premaxilla begins in the MNP and subsequently spreads laterally across the MNP/MP fusion area into the MP. Accordingly, the lateral incisor undergoes a complex positional shift (mainly medial) relative to the incisive suture both prenatally and postnatally and is finally located medial to the suture. Examination of the cyclophosphamide-induced CLP fetuses showed that the lateral incisor is located lateral to the alveolar cleft and does not shift medial to the incisive suture.

Conclusion

Understanding the origin of the lateral incisor (the tooth closest to the cleft) and the shift after its formation provides clues to high incidence of malformations and ectopia of this incisor in cleft patients.
Congenital Heart Disease in a Patient with Microform Cleft Lip
Authors: Grech Victor , Lia Alexander , Mifsud Anton
Published: October 2018

Objective

Microform cleft lip (MCL), also known as congenital healed cleft lip, is a rare anomaly that is thought to be a spontaneously healed variant of the more common clefting of lip and palate. Although conventional clefting is known to be associated with congenital heart disease (CHD), MCL has not been similarly associated. We present one patient with MCL and CHD in the form of coarctation of the aorta, an association not previously described.

Conclusion

As is done in patients with cleft lip, cleft palate, or both, patients with MCL should be carefully examined for evidence of CHD and referred to a pediatric cardiologist if deemed advisable.