bluuebunny/crossref_metadata_2025 huggingface dataset:
Cleft Lip with or without Cleft Palate: Effect of Family History on Reproductive Planning, Surgical Timing, and Parental Stress
Authors: Andrews-Casal Melanie , Johnston Dennis , Fletcher Jack , Mulliken John B. , Stal Samuel , Hecht Jacqueline T.
Published: October 2018

Objective

This study tested whether the presence of a family history of non-syndromic cleft lip with or without cleft palate (NSCLP) lessens the negative impact on reproductive planning, decreases treatment anxiety, and relieves parental stress when there is a recurrent cleft.

Design

Sixty-one participating families, retrospectively ascertained through their children with NSCLP, were divided into two groups: those with a family history of clefting and those in which only the proband was affected.

Setting

Each family completed a questionnaire either at their home or during a clinic visit.

Participants

Questionnaires were distributed to 117 families with an NSCLP child. Of these 117, 49 (42%) had another family member with NSCLP and 68 (58%) had no prior history of clefting. Sixty-one (52%) families returned the questionnaire and were categorized by family history.

Main outcome Measures

The Parenting Stess Index and the Hollingshead Index of Social Position were included with family and medical history questions in the 137-item questionnaire.

Results

The reproductive plans of the two groups were quite similar and were not dependent on the families' actual or perceived recurrence risks of NSCLP. The majority (85%) of the children with NSCLP underwent surgical closure at the recommended age. The two groups did not significantly differ on any score on the Parenting Stress Index, indicating a similar parental stress level between the groups.

Conclusions

These findings suggest that family history does not significantly influence reproductive plans, timing of cleft repair, or stress of parents of a child with NSCLP. Further, these results indicate a need for additional psychosocial study of this population.
Atrial Septal Defect in Cyanotic CL/Fr Mice
Authors: Kadowaki Shingo , Sakamoto Michiko , Kamiishi Hiroshi , Tanimura Takashi
Published: October 2018

Objective

It is widely known that some newborn CL/Fr mice with cleft lip and palate (CLP) also have cyanotic symptoms, which have been thought to depend on an atrial septal defect (ASD). In a previous study, we found that cyanotic mice tended to have more severe types of CLP. We hypothesize that the mechanical airway obstruction due to a poorly developed palatal shelf and unmoved tongue in CLP(+) mice might be related to the occurrence of cyanosis. The purpose of this study was to examine the relationships between ASD and cyanosis in CLP(+) newborns.

Method

The newborn hearts from CLP(-), noncyanotic CLP(+), cyanotic CLP(+), CL/Fr mice and ICR mice were examined histologically, and the incidence and size of ASD was determined on neonatal day (ND) 0. In CLP(-) newborns, similar procedures were performed from ND 1 to ND 4. Furthermore, in CLP(+) newborns, development of the palatal shelf was examined.

Results

While all the ICR mice had a well-developed atrial septum, and the incidence of ASD was 0%, about 80% of CL/Fr mice had ASD, irrespective of the presence or absence of CLP and cyanosis. On ND 0, the septum primum was significantly shorter in cyanotic CLP(+) mice than in CLP(-) mice. It also tended to be shorter in CLP(+) mice than in CLP(-) mice. Between the cyanotics and noncyanotics, there were no significant differences in the incidences of ASD and the rate of septal development. In CLP(-) mice, the septum primum developed well later and no ASD was observed on ND 4. Cyanotic newborns had significantly less developed palatal shelves than did noncyanotics.

Conclusions

Cyanosis may not be related to ASD and the rate of septal development, but may be related to the occurrence of CLP in this strain. Furthermore, we confirmed that some relationship exists between the development of the palatal shelf and cyanosis. The present study supports our hypothesis concerning the cause of cyanosis in CL/Fr mice.
Cleft Palate Formation in Fetal Br Mice with Midfacial Retrusion: Tenascin, Fibronectin, Laminin, and Type IV Collagen Immunolocalization
Authors: Singh G.D. , Johnston J. , Ma W. , Lozanoff S.
Published: October 2018

Objective

This study tested the hypothesis that altered craniofacial morphology does not affect the expression of extracellular matrix (ECM) molecules such as fibronectin (FN), laminin (LN), type IV collagen, and tenascin-C (TN) but is associated with failure of palatal shelf elevation and fusion concomitant with cleft palate formation.

Design

To test this hypothesis, a comparative immunohistological analysis of FN, LN, type IV collagen, and TN was undertaken on brachyrrhine (Br/Br) mice and normal (+/+) fetuses during secondary palate formation. Normal and Br/Br fetuses were collected at gestational days E13 and E14 (representing prefusion stages) and E15 and E18 (representing postfusion stages). Cryostat palatal sections (8 μm) were postfixed in methanol, washed, and stained with primary antibody. All sections were washed and coated with secondary antibody (swine-anti-rabbit IgG) and mounted with citifluor.

Results

Immunohistological analysis showed that LN and type IV collagen were located near the presumptive medial epithelial seam (MES) or edge (MEE) in +/+ or Br/Br fetuses, respectively. Fibronectin showed a homogeneous distribution at all stages in both groups of mice. In contrast, TN became localized below the presumptive MES or MEE in both groups of mice at E14. In +/+ animals at E15, TN dissipated and became confined to the oral basement membrane by E18. At E15 and E18 in cleft Br/Br mutants, TN stained beneath the MEE.

Conclusion

Although the distributions of ECM molecules are similar during normal and cleft palatogenesis, differences in TN expression are associated with cleft palate formation.
Reduced Need for Alveolar Bone Grafting by Presurgical Orthopedics and Primary Gingivoperiosteoplasty
Authors: Santiago Pedro E. , Grayson Barry H. , Cutting Court B. , Gianoutsos Mark P. , Brecht Lawrence E. , Kwon Soon Man
Published: October 2018

Objective

The purpose of this study was to evaluate if narrowing and approximation of the alveolar cleft through presurgical alveolar molding followed by gingivoperiosteoplasty (GPP) at the time of lip repair reduces the need for a bone-grafting procedure.

Design

This was a retrospective blind study of patients with unilateral or bilateral alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon. Alveolar bone formation was assessed prior to the eruption of the maxillary lateral incisor or canine by clinical examination, panoramic and periapical radiographs, and/or a dental CT scan. The criterion for bone grafting was inadequate bone stock to permit the eruption and maintenance of the permanent dentition.

Setting

This study was performed at the Institute of Reconstructive Plastic Surgery by the members of the Cleft Palate Team.

Patients

All patients with unilateral (n = 16) or bilateral (n = 2) alveolar clefts who underwent presurgical infant alveolar molding and GPP by a single surgeon from 1985 to 1988 were studied. The control population consisted of all alveolar cleft patients (n = 14) who did not undergo alveolar modeling or GPP during the same time period.

Interventions

Presurgical alveolar modeling was performed with an intraoral acrylic molding plate. This plate was modified on a weekly basis to align the alveolar segments and close the alveolar gap. The surgical intervention consisted of a modified Millard GPP.

Main outcome Measures

The primary study outcome measure was the elimination of the need for a secondary bone graft in patients who underwent presurgical alveolar molding and GPP.

Results

Of the 20 sites in the 18 patients who underwent GPP, 12 sites did not require an alveolar bone graft. Of the 8 sites requiring a bone graft, 4 presented minimal bony defects. All 14 patients in the control group required bone grafts.

Conclusions

In this series of 20 alveolar cleft sites treated with presurgical orthopedics and GPP, 60% did not need a secondary alveolar bone graft in the mixed dentition.
Articulation and Nasality Changes Resulting from Sustained Palatal Fistula Obturation
Authors: Pinborough-Zimmerman Judith , Canady Claudia , Yamashiro Duane K. , Morales Louis
Published: October 2018

Objective

The purpose of the study was to determine differences in articulation and nasality with obturation over time in children with a palatal fistula.

Design

Articulation and nasality were measured with the fistula open, immediately after obturation, and 4 to 7 weeks postobturation.

Setting, Patients, Participants

Subjects were 15 patients with a palatal fistula secondary to a repaired cleft palate who were seen through the Orofacial Program, Utah Department of Health, ranging in age from 4 years 6 months to 13 years 1 month.

Interventions

Acrylic palatal obturators were designed to provide coverage specific to the unique shape and location of each child's fistula. Obturators were cemented to molar teeth using wire clasps for control of usage.

Main outcome Measures

Measurements consisted of listener judgments of hypernasality, hyponasality, and nasal emissions; instrumental ratings of nasalance using the Nasometer 6200-2; and performance on a standardized articulation test.

Results

Significant improvement occurred only on nasal emission measures from the preobturation condition to immediate postobturation. However, significant improvement was found in articulation, listener judgments of hypernasality, nasal emissions, and Nasometric Nasal Sentence mean scores from the preobturation condition to 4 to 7 weeks postobturation and from the immediate postobturation condition to 4 to 7 weeks postobturation. No significant differences were found between conditions for listener judgments of hyponasality and Nasometric Zoo and Rainbow Passage scores. Obturation of the palatal fistula over a 4- to 7-week period resulted in no adverse effect on articulation ability, perceptual ratings of nasality, or instrumental ratings of nasalance.

Conclusions

Clinical management of patients with a palatal fistula can be enhanced with treatment using obturation over time. For subjects who continue to exhibit hypernasality immediately postobturation, sustained obturation is advocated prior to consideration of surgical intervention for treatment of a palatal fistula and/or velopharyngeal dysfunction.
An Anatomic Study of the Tensor Veli Palatini and Dilatator Tubae Muscles in Relation to Eustachian Tube and Velar Function
Authors: Barsoumian Raffi , Kuehn David P. , Moon Jerald B. , Canady John W.
Published: October 2018

In a gross anatomic study of 20 sides in 16 human head specimens, the tensor veli palatini, the dilatator tubae, and the tensor tympani muscles were studied. The tensor veli palatini was observed to insert onto the anterior one-third of the pterygoid hamulus, whereas the dilatator tubae rounded the middle one-third of the pterygoid hamulus without an insertion. Thus, the dilatator tubae, not the tensor veli palatini, could serve to tense the anterior velum. An insertion from the superior pharyngeal constrictor muscle onto the posterior one-third of the hamulus could provide a curbing function for the dilatator tubae muscle. Adipose tissue, located at the hamulus, could provide lubrication for the tendinous fibers of the dilatator tubae as they round the hamulus. The dilatator tubae was observed to attach to the hook of the eustachian tube and is accepted as the tubal dilator. Observed on 13 of 20 sides in 11 specimens, the bulk of the dilatator tubae remained distinct from the tensor veli palatini despite a connective tissue alliance and intermingling of some muscle fibers. On 5 of 20 sides in 5 specimens, fibers of the dilatator tubae intermingled extensively with the tensor veli palatini. Of the 20 dilatator tubae muscles dissected, 2 were observed to be deficient. The tensor veli palatini was observed to be continuous with the tensor tympani. Full color versions of the figures are available at the following website: http://www.shc.uiowa.edu/papers/tensor/ .
Craniofacial Morphology in Adult Patients with Unoperated Complete Bilateral Cleft Lip and Palate
Authors: Da Silva Filho Omar Gabriel , De Cássia Moura Carvalho Lauris Rita , Filho Leopoldino Capelozza , Semb Gunvor
Published: October 2018

Objective

This report is a retrospective study that compares the craniofacial morphology of adult subjects with unoperated bilateral complete cleft lip and palate (BCLP) with that of a noncleft group.

Methods

The study was performed on standardized lateral cephalograms obtained at the Hospital for Research and Rehabilitation of Cleft Lip and Palate, University of São Paulo, Brazil. The research group consisted of 28 subjects (20 males, 8 females) with unoperated BCLP, ranging in age from 15 to 41 years. The control group was matched to the cleft group with regard to gender and age. The findings were analyzed on the basis of the two-way analysis of variance (ANOVA) for cleft and gender.

Results

The most striking difference between the groups was the extremely prominent premaxilla in the cleft group that gave the BCLP face a very convex profile. The mandible exhibited a vertical growth pattern that resulted in a steep mandibular plane, an obtuse gonial angle and a long lower face height. The posterior face height was reduced. The cranial base dimensions were smaller, but there was no difference in cranial base angulation.

Conclusions

These findings confirm that in subjects with unoperated BCLP, the initial characteristics of the cleft malformation persist during growth.
Sensitivity of a Method for the Analysis of Facial Mobility. I. Vector of Displacement
Authors: Trotman Carroll-Ann , Faraway Julian J. , Silvester Kirsten T. , Greenlee Geoffrey M. , Johnston Lysle E.
Published: October 2018

Objective

(1) To determine which facial landmarks show the greatest movement during specific facial animations and (2) to determine the sensitivity of our instrument in using these landmarks to detect putatively abnormal facial movements.

Design

Movements of an array of skin-based landmarks on five healthy human subjects (2 men and 3 women; mean age, 27.6 years; range, 26 to 29 years) were observed during the execution of specific facial animations. To investigate the instrument sensitivity, we analyzed facial movements during maximal smile animations in six patients with different types of functional problems. In parallel, a panel was asked to view video recordings of the patients and to rate the degree of motor impairment. Comparisons were made between the panel scores and those of the measurement instrument.

Results

Specific regions of the face display movement that is representative of specific animations. During the smile animation, landmarks on the mid- and lower facial regions demonstrated the greatest movement. A similar pattern of movement was seen during the cheek puff animation, except that the infraorbital and chin regions demonstrated minimal movement. For the grimace and eye closure animations, the upper, mid-facial, and upper-lip regions exhibited the greatest movement. During eye opening, the upper and mid-facial regions, excluding the upper lip and cheek, moved the most, and during lip purse, markers on the mid- and lower face demonstrated the most movement. We used the smile-sensitive landmarks to evaluate individuals with functional impairment and found good agreement between instrument rankings based on the data from these landmarks and the panel rankings.

Conclusion

The present method of three-dimensional tracking has the potential to detect and characterize a range of clinically significant functional deficits.
Sensitivity of a Method for the Analysis of Facial Mobility. II. Interlandmark Separation
Authors: Trotman Carroll-Ann , Faraway Julian J.
Published: October 2018

Objective

This study demonstrates a method of quantifying facial movements based on distortions of the skin surface.

Design

Landmarks were identified on the faces of five healthy human subjects (2 men and 3 women; mean age, 27.6 years; range, 26 to 29 years), and the distortions were characterized by changes in the separation between 20 pairs of landmark distances during specific maximal facial animations: smile, lip purse, cheek puff, grimace, eye closure, and eye opening. Data were recorded with a video-based tracking system for a period of 3 seconds at a sampling rate of 60 Hz or frames per second. For each subject, we analyzed the change in the separation of 20 pairs of landmarks, of which the majority were bilaterally symmetrical and functionally active.

Results

Characteristic patterns of movement emerged for each animation. We found that smiling involved movements of the lateral orbital, circumoral, and chin regions; grimacing involved the inner orbital, lateral orbital, lateral nasal, and upper-lip regions; eye closure involved the inner orbital, lateral orbital, and, to a lesser degree, lateral nasal regions; eye opening involved the inner and lateral orbital regions; cheek puffing involved the cheek and lower-lip regions; and the lip purse animation involved the nasolabial, cheek, commissure, and lip regions.

Conclusion

This measurement of distortion provided a quantitative estimate of facial movement, and this approach is especially applicable to patients with unilateral problems in which the patient can serve as his or her own control.
Multiple Suture Synostosis and Increased Intracranial Pressure following Repair of Single Suture, Nonsyndromal Craniosynostosis
Authors: Hudgins Roger J. , Cohen Steven R. , Burstein Fernando D. , Boydston William R.
Published: October 2018

Objective

Increased intracranial pressure, frequently associated with closure of multiple cranial sutures, has been reported to occur in 36% of cases following correction of syndromal craniosynostosis. Although much less common, multiple suture closure may occur following repair of single suture, non-syndromal craniosynostosis and we present cases that concern two such children.

Results

Two children with nonsyndromal craniosynostosis, one metopic and one left-coronal, underwent fronto-orbital advancement at age 3 months. At age 19 months and at age 5 years, respectively, both patients re-presented with headaches, decrease in head circumference percentile, and acceptable cosmetic outcome. Both had computerized tomographic evidence of multiple closed cranial sutures and increased intracranial pressure (ICP) (determined by monitoring). Both patients improved following a cranial expansion procedure.

Conclusion

Delayed closure of multiple sutures and resultant increased ICP may occur following correction of nonsyndromal, single suture craniosynos-tosis. This may be more likely when the initial suture is contiguous with the facial sutures. Children should be followed for many years following cranio-synostosis repair with cranial, neurologic, and possibly funduscopic examinations as well as head circumference measurements to detect delayed closure of cranial sutures.
Intracranial Pressure in Single-Suture Craniosynostosis
Authors: Cohen Steven R. , Persing John A.
Published: October 2018

In this paper, we review the incidence of increased intracranial pressure in children with single-suture craniosynostosis. The major studies in this area are presented, along with their limitations. A rational treatment plan including multidisciplinary team management is recommended. All patients with proven synostosis should be followed closely, whether or not surgery is chosen. Continued clinical and basic science research are necessary to further clarify the ramifications of asymptomatic elevations of intracranial pressure in these patients.
Mental Development and Learning Disorders in Children with Single Suture Craniosynostosis
Authors: Kapp-Simon Kathleen A.
Published: October 2018

Objective

This study examined the global intellectual development and presence/absence of learning disorders in children with nonsyndromic metopic, sagittal, and unilateral coronal synostosis who had early surgery (under 1 year of age), late surgery (over 1 year of age), or no surgery to correct the synos-tosis across three time periods.

Design

The design was longitudinal. Children were assessed at initial team evaluation, 1 year after surgery or initial evaluation if surgery was not performed, and annually thereafter.

Participants

Participants included 84 consecutively evaluated patients. Seventy-two children were evaluated at Time 1 (T1), 8 months; 57 at Time 2 (T2), 21 months; and 39 at Time 3 (T3), 50 months. Data on learning disorders or mental retardation were available on 34 children who were school age.

Main Outcome Measures

The Mental Development Index from the Bayley Scales of Infant Development was the main outcome measure for T1 and T2. The General Cognitive Index from the McCarthy Scales of Children's Abilities was the main outcome measure for T3. Learning disorders were determined from school and clinical records.

Results

Repeated measures analyses of variance found no statistical differences in the development of children based on diagnosis or surgical status across time. Base rate of mental retardation at T1 was consistent with normative data; however, the incidence of retardation was two to three times the expected rate at T2 and T3. Learning disorders were present in 47% of school-age children who were not retarded. Chi-square analysis showed no significant differences between rates of retardation or learning disorders based on surgical status.

Conclusions

Most children with nonsyndromic craniosynostosis obtain developmental quotients within the normal range in infancy. Rates of retardation may increase relative to normative expectations as children mature. A high rate of learning disorders was identified. Results are preliminary due to sample size.
The Differential Diagnosis of Abnormal Head Shapes: Separating Craniosynostosis from Positional Deformities and Normal Variants
Authors: Huang Martin H.S. , Mouradian Wendy E. , Cohen Steven R. , Gruss Joseph S.
Published: October 2018

The correct differential diagnosis of an abnormal head shape in an infant or a child is vital to the management of this common condition. Establishing the presence of craniosynostosis, which warrants surgical correction, versus non-synostotic causes of head deformity, which do not, is not always straightforward. This paper deals with three groups of abnormal head shape that may cause diagnostic confusion: the spectrum of metopic synostosis; the dolicho-cephaly of prematurity versus sagittal synostosis; and the differential diagnosis of plagiocephaly. Special emphasis has been placed on the problem of posterior plagiocephaly, in the light of recent evidence demonstrating that lambdoid synostosis has been overdiagnosed. Metopic synostosis presents as a wide spectrum of severity. Although only severe forms of the disorder are corrected surgically, all cases should be monitored for evidence of developmental problems. The dolichocephalic head shape of preterm infants is non-synostotic in origin and is managed nonsurgically. The scaphocephalic head shape resulting from sagittal synostosis requires surgical intervention for correction. Posterior plagiocephaly may be due to unilambdoid synostosis or positional molding, which have very different clinical and imaging features. True lambdoid synostosis is rare. Most cases of posterior plagiocephaly are due to positional molding, which can usually be managed nonsurgically. Regardless of the suture(s) involved, all children with confirmed craniosynostosis should be monitored for increased intracranial pressure and developmental problems.
Maxillary Arch Dimensions in Bilateral Cleft Lip and Palate from Birth until Four Years of Age in Boys
Authors: Heidbuchel Kiki L.W.M. , Kuijpers-Jagtman Anne M. , Kramer Gem J.C. , Prahl-Andersen Birte
Published: October 2018

Objective

To describe the development of maxillary arch dimensions in children with bilateral cleft lip and palate (BCLP) during the first 4 years of life and to compare it with that in noncleft children.

Design

This was a retrospective, mixed-longitudinal study.

Setting

The study was conducted at the Cleft Palate Center of the University Hospital of Nijmegen.

Subjects

The sample consisted of 26 boys with BCLP who were born between 1976 and 1990 and treated at the University Hospital of Nijmegen. Data for a control group of 34 noncleft boys were collected at the University Hospital of Amsterdam.

Method

Palatal arch dimensions were digitized on dental casts. A comparison between BCLP and noncleft dimensions was made at fixed time intervals.

Results

At birth, anterior and posterior arch widths as well as arch depths were significantly larger in children with BCLP. After 7 months (lip closure), anterior arch width and arch depth diminished considerably in the cleft group. After 12 months (palatoplasty), a slight decrease in posterior arch width was observed, and arch depths showed slight catch-up growth. At 4 years of age, anterior arch width was significantly narrower and anterior arch depth was shorter in children with BCLP than in control subjects. Posterior arch width was significantly wider.

Conclusions

During the first 4 years of life, maxillary arch dimensions in children with BCLP show a unique development that is significantly different from that in noncleft children.
The Oral Health of Children with Clefts-A Review
Authors: Wong Fanny W. L. , King Nigel M.
Published: October 2018

A review of the studies of the caries prevalence and periodontal health of patients with cleft lip and palate (CLP) revealed that only five investigations of caries prevalence in CLP patients included children. One reported that CLP children did not have a higher caries prevalence in the permanent dentition, whereas more recent studies have reported a higher caries prevalence in both the primary and permanent dentitions of CLP children than in those of noncleft children. However, there is wide variation in the teeth examined and the method of presenting data on caries prevalence. Six papers have reported on the periodontal health of adult CLP patients, and only one has done so on that of children. The adult CLP patients had poorer oral hygiene and more gingivitis, but there is no conclusive evidence that they have a higher risk of developing periodontal disease. No data on the oral hygiene of CLP children were available, but it has been emphasized that they have significantly more gingivitis than noncleft children, especially in the maxillary anterior teeth.
Surgical Repositioning of the Premaxilla in Combination with Two-Stage Alveolar Bone Grafting in Bilateral Cleft Lip and Palate
Authors: Iino Mitsuyoshi , Sasaki Tomokazu , Kochi Shoko , Fukuda Masayuki , Takahashi Tetsu , Yamaguchi Tai
Published: October 2018

Objective

This paper introduces a surgical technique for premaxillary repositioning in combination with two-stage alveolar bone grafting for the correction of the premaxillary deformity of patients with bilateral cleft lip and palate (BCLP). The paper also reports on two patients with BCLP who underwent this surgical management.

Surgical Procedure

The operation is usually performed when the patient is 8 to 14 years of age. In the first stage of surgery, the side more accessible to the septopremaxillary junction is selected, and an osteotomy of the premaxilla and unilateral alveolar bone grafting are performed. Approximately 4 to 12 months after the first stage of surgery, contralateral alveolar bone grafting is carried out.

Conclusion

We have found that this surgical procedure is highly effective, because it ensures the blood supply to the premaxilla and minimizes the potential for surgical failure. Moreover, it affords wide exposure of the premaxillary bone surface, facilitating sufficient boney bridging and allowing for orthodontic tooth movement.
Incidence of Secondary Lip Surgeries as a Function of Cleft Type and Severity: One Center's Experience
Authors: Henkel Kai-Olaf , Gundlach Karsten , Saka Basam
Published: October 2018

Objective

This study was conducted to ascertain the frequency of surgeries typically required to adequately repair a cleft lip.

Material

All operations performed on patients with clefts of the lip (alveolus and palate) from 1968 to 1990 in the Rostock Cleft Center were evaluated.

Methods

Procedures were classified as primary labioplasties versus secondary revisions. Patients were also classified according to cleft type, the anatomical segment revised, and the reason for lip revision. Two indices were calculated. The revision index related the number of revisions to the number of primary labioplasties. The index of indication related the number of patients with revisions to the number of primary labioplasties.

Results

During the period of study, 712 primary labioplasties and 771 secondary revisions were performed. The overall revision index was 1.10. The index of indication varied from 0.28 for unilateral complete clefts of the lip to 0.67 for bilateral complete clefts of the lip. Revision of the lip was the most common secondary procedure, followed by nostril and columella revision, and revision of the oral vestibule.

Conclusions

The incidence of secondary surgery of the repaired cleft lip (index of indication) of about 35% is similar to previously reported data. However, the incidence varies with cleft type and is required more often with bilateral complete clefts of the lip.
Maxillary Dental Development in Complete Unilateral Alveolar Clefts
Authors: Solis Alex , Figueroa Alvaro A. , Cohen Mimis , Polley John W. , Evans Carla A.
Published: October 2018

Objective

This study was conducted to determine whether development of individual maxillary teeth in subjects with complete unilateral alveolar clefts was significantly different from that found in unaffected children.

Design

Retrospective, mixed longitudinal.

Setting

Craniofacial Center, university based.

Materials and Methods

A sample of 179 panoramic radiographs obtained during the mixed dentition from 79 subjects (47 males, 32 females) with complete alveolar clefts was analyzed. After visual evaluation of root development of the permanent maxillary teeth from radiographs, a score from 0 to 5 was assigned utilizing a predefined scoring system. Statistical analyses were performed between the cleft and unaffected reference groups available in the literature.

Results

The cleft side dentition was found to be significantly delayed in development relative to the noncleft side (p < .05). Compared to the reference group, the cleft side lateral incisor demonstrated a mean difference in development of 1.59 years followed by the canine (1.39 years), the central incisor (0.96 years), the first premolar (0.94), and the second premolar (0.78).

Conclusion

Teeth directly adjacent to the cleft site were shown to be the most delayed. The lateral incisors and canines were observed to be the most variable when compared to their corresponding antimeres. The information obtained from this study may assist the orthodontist in selecting the appropriate time to initiate orthodontic treatment in order to prepare the permanent dentition prior to alveolar bone grafting.
Effects of Irradiation and Methyl-Triazene on Craniofacial Development in Mouse Embryos: A Semiautomated Morphometric Analysis
Authors: Glineur R. , Van Sint Jan S. , Louryan S. , Philippson C. , De Maertelaer V. , Evrard L. , Rooze M.
Published: October 2018

Objective

The purpose of the present study was a 2D-semiautomated morphometric analysis of craniofacial growth in nuclear magnetic resonance imaged (NMRI) mouse embryos.

Methods

The NMRI mouse embryos were exposed in utero to either a single dose of 2 Gy X-irradiation on day 9 of gestation (113 embryos) or to 1.5 mg methyl-triazene administered orally to their pregnant mothers on gestational day 10.5 (124 embryos). An additional group of 108 embryos was used as controls. Digitized pictures of embryos from gestational days 14 to 17 were taken in lateral right view using a video system. Landmarks were located and digitized for computerized analysis of growth changes in relation to developmental stages of the face.

Results

The results revealed that the snout of control embryos lengthens during the developmental period considered. The snout of embryos previously submitted to methyl-triazene displayed micrognathia, and all treated fetuses exhibited macroscopic signs of microcephaly with a reduced mandible. The snouts of irradiated embryos appeared shortened at the 14-day stage and continued to shorten as development proceeded. A shortening of the midface was detected macroscopically in 83% of the cases.

Conclusion

The results of this morphometric analysis enabled us to trace the developmental progression of the induced dysmorphosis and to assess the differences compared with normal development.
Classic and Occult Submucous Cleft Palates: A Histopathologic Analysis
Authors: Stal Samuel , Hicks M. John
Published: October 2018

Objective

Although classic and occult submucous clefts have been well described with respect to clinical features and endoscopic findings, there is a paucity of information regarding the histologic characteristics of the midline palatal area and its role in the clefting process. This preliminary study describes the histopathologic features of classic and occult submucosal clefts.

Design

Twenty-eight patients with classic submucous cleft (12 patients) and occult submucous cleft (16 patients) were available for study. Histopathologic evaluation of two biopsies from each patient (muscular uvulae biopsy; levator muscle bundle biopsy) was performed.

Results

Submucosal fibrosis with relatively dense collagen deposition was noted in both the musculus uvulae and levator muscle bundle biopsy sites. Individual myocytes and myocyte fascicles were entrapped and encased by fibrotic tissue, resulting in disruption of the fascicular organization. Most fascicles were disrupted by intervening dense collagen bands within and interposed between the fascicles. The myocytes had an atrophic or hypoplastic appearance with cross-sectional diameters ranging from 42% to 61% (mean reduction, 53%) less than those expected for myocytes from normal palates. There was fascicular disorganization with intermixing of longitudinally and cross-sectionally oriented myocyte groups. Minor salivary glands were affected as well with increased collagen deposition between and within lobules of salivary gland tissue.

Conclusions

This histopathologic study indicates that significant fibrosis is present within submucosal cleft regions and is associated with myocyte atrophy or hypoplasia. The relatively dense fibrosis appears to be a well-organized, chronic process and is present in the absence of significant chronic inflammation. In addition, there is myocyte fascicular disorganization with a haphazard arrangement. This disorderly arrangement may imply that there is a failure of resorption of the epithelial/mesenchymal tissue during palatine shelf closure with retention of mesenchymal tissue that contains the insertion of the velopharyngeal musculature apparatus.
Infant TGF-Alpha Genotype, Orofacial Clefts, and Maternal Periconceptional Multivitamin Use
Authors: Shaw Gary M. , Wasserman Cathy R. , Murray Jeffrey C. , Lammer Edward J.
Published: October 2018

Objective

We previously demonstrated a strong association between periconceptional maternal cigarette smoking, infant transforming growth factor–alpha (TGFa) genotype, and risk of orofacial clefts. Because serum folate may be decreased by cigarette smoking and because maternal periconceptional use of multivitamins containing folic acid has been associated with a reduced risk of clefting, we explored whether a potential relation existed between infant TGFa genotype, maternal multivitamin use, and risk of orofacial cleft phenotypes.

Design

Data were derived from a population-based case–control study of fetuses and live-born infants among a cohort of 1987 to 1989 California births (n = 548,844). Information concerning periconceptional multivitamin use was obtained via telephone interviews with mothers of 731 (84.7% of eligible) orofacial cleft case infants, and of 734 (78.2%) nonmalformed control infants. DNA was obtained from newborn screening bloodspots and genotyped for the Taq1 polymorphism of TGFa. Among infants of interviewed mothers, genotypes were available for 571 (78.1%) case infants and 640 (87.2%) control infants.

Setting

The study encompassed all hospitals in selected California counties.

Main Outcome Measure

The main outcome measures were the risks of specific cleft phenotypes among infants with uncommon TGFa genotypes and whose mothers did not use multivitamins periconceptionally.

Results

Compared with infants homozygous for the common TGFa genotype and whose mothers used multivitamins, increased clefting risks were observed for infants with the A2 genotype (homozygous or heterozygous) and whose mothers did not use multivitamins. Risk estimates were 3.0 (1.4–6.6 [95% confidence interval]) for isolated cleft lip with or without cleft palate (CLP), 2.4 (0.69–11.6) for multiple CLP, 2.6 (0.97–7.7) for isolated cleft palate (CP), 4.2 (1.3–16.2) for multiple CP, and 8.1 (2.6–27.7) for “known-syndrome” clefts. Clefting risks for infants with the A2 genotype and whose mothers used multivitamins were substantially smaller, as were the risks for infants with the A1 genotype whose mothers did not use multivitamins.

Conclusion

These data provide preliminary evidence for a gene–nutrient interaction in risk of clefting.
A Retrospective Comparison of Craniofacial Form in Northern Irish Children with Unilateral Cleft Lip and Palate
Authors: Leonard Alan G. , Kneafsey Brian , Mckenna Stephen , Johnston Chris D. , Burden Donald J. , Stevenson Mike
Published: October 2018

Objective

This study evaluated the craniofacial form of a sample of Northern Irish children with unilateral cleft lip and palate (UCLP). The quality of the outcomes achieved was compared with the outcomes reported for the six centers involved in the European multicenter study (Mars et al., 1992; Mølsted et al, 1992).

Design

Retrospective analysis.

Patients

All children born with complete skeletal UCLP in Northern Ireland during the years 1983 to 1987.

Main outcome Measures

Cephalometric analysis was used to determine the craniofacial form and soft tissue profile. The quality of the dental arch relationships was independently assessed using the Goslon ranking system.

Results

The sample comprised 25 children with complete skeletal UCLP who had cephalometric radiographs and study casts recorded at a mean age of 9.4 years (range, 8 to 11 years). Cephalometric analysis revealed no important skeletal differences between the Northern Irish UCLP children and the published results from the six Eurocleft centers. The soft tissue profile of the Northern Irish UCLP children was significantly more convex than the soft tissue profile recorded for center D in the Eurocleft study. The Goslon ranking system revealed that 18 (72%) of the Northern Irish UCLP children had good or satisfactory dental arch relationships.

Conclusions

No clinically important differences were detected between the mean cephalometric skeletal parameters of the Northern Irish UCLP children and those published for the six cleft centers involved in the Eurocleft study. On average, the Northern Irish UCLP children were found to differ significantly from Eurocleft's center D in their soft tissue facial contour and sagittal lip profile. The quality of the dental arch relationships of the Northern Irish sample was between the best and the less good Eurocleft centers.
Electromyographic Investigation of Masticatory Muscles in Unilateral Cleft Lip and Palate Patients with Anterior Crossbite
Authors: Li Weiran , Lin Jiuxiang , Fu Minkui
Published: October 2018

Objective

To evaluate the characteristics of masticatory muscle activity in operated unilateral cleft lip and palate (UCLP) patients with anterior crossbite compared with normal individuals.

Subjects

Sixteen male and 13 female Chinese patients with UCLP and anterior crossbite. Fifteen male and 13 female Chinese individuals without cleft abnormalities served as a control group.

Design

Electromyographic activity of the masseter muscles and anterior temporalis muscles was recorded bilaterally in different mandibular positions using bipolar surface electrodes.

Results

Compared to noncleft controls, patients with UCLP demonstrated (1) higher activation levels of masseter and temporalis muscles in the rest position, (2) lower potential function of masseter and temporalis, (3) inharmonious activity of the masticatory muscles during mandibular border movement, (4) a higher asymmetry index of the masseter and temporalis muscles, and (5) longer silent periods of the two muscles.

Conclusions

The function of masticatory muscles is different in patients with UCLP with anterior crossbite. Muscle function should be considered when evaluating cleft patients for orthodontic treatment and orthognathic surgery.
Muscle Fiber Type Distribution in the Normal Human Levator Veli Palatini Muscle
Authors: Moon Jerald B. , Thompson Sue Ann , Jaeckel Elise , Canady John W.
Published: October 2018

Objective

This study examined the muscle fiber type distribution within the normal adult levator veli palatini muscle.

Methods

Levator veli palatini muscle tissue was harvested from the palates of 12 (seven female, five male) adult noncleft cadavers. Adjacent sections were stained for adenosine triphosphatase at pH 10.4 or 4.2. After mounting, magnifying, and photographing, Type I versus Type II fiber types were differentiated by the intensity of, or by the inhibition of, staining of matched fibers at each pH level. Type I fibers stained light at pH 10.4 and dark at pH 4.2, while Type II fibers stained light at pH 4.2 and dark at pH 10.4.

Main outcome Measures

The number of fibers counted for each specimen ranged from 60 to 616. The numbers of Type I and Type II stained fibers appearing in each muscle tissue sample were determined and expressed as a percentage of the total number of fibers identified. A few identified fibers could not be labelled as either Type I or Type II.

Results

The overall proportion of Type I fibers, averaged across all specimens, was 59.8%. Male specimens had 67.4% Type I fibers and 31.8% Type II fibers, while female specimens had 54.4% Type I fibers and 44.4% Type II fibers.

Conclusions

Observed fiber type distributions were similar to those reported for other articulatory muscles, but differed slightly from previously reported distributions for normal levator veli palatini. The distributions observed in this study provide a baseline against which to relate fiber type data from the levator veli palatini of cleft palates to the functional status of the velopharyngeal mechanism.
Survey of Dentists' Experience with Cleft Palate Children in Chile
Authors: Dabed Cynthia C. , Cauvi Doris L.
Published: October 2018

Objective

This study was designed to study the experience of dentists with children with palatal clefts seeking treatment in Santiago, Chile.

Design

A 13-item questionnaire was sent to 203 pediatric and general dentists treating children. Of the 141 dentists who voluntarily completed the questionnaire, 118 were selected for this study. These professionals worked in private and public health centers in the Santiago, Chile, metropolitan area.

Results and Conclusions

The results showed that a majority of dentists had treated a low number (1 to 3) of cleft palate children in the previous years, using a combination of preventive-curative and radical treatments. Although a majority reported that there were no differences in the treatment of the cleft and noncleft children, a majority had problems during the course of treatment. The results suggest the need for more current information about the care of cleft children.
A Comparison of Two Different Bone-Harvesting Techniques for Secondary Alveolar Bone Grafting in Patients with Cleft Lip and Palate
Authors: Mccanny C.M. , Roberts-Harry D.P.
Published: October 2018

Objective

To compare the outcome of the trephine with open hip surgery for alveolar bone grafting in cleft lip and palate surgery.

Design

The study was retrospective. The radiographs were assessed blindly and on two separate occasions 1 week apart.

Setting

University Teaching Hospital.

Patients, Participants

In one group (group A), a trephine was used; in the other (group B), open hip surgery was employed. Group A was comprised of 16 patients (nine with unilateral and seven with bilateral clefts of the lip and palate) and group B, 13 patients (eight unilateral and five bilateral). The prime entry criterion for inclusion in the study was that the canine tooth had erupted into the graft site.

Interventions

A long cone periapical radiograph was taken of the erupted canine tooth in the graft site.

Main Outcome Measure

The radiographs were graded from type I to type IV, as described by Bergland et al. (1986a). A comparison was also made of the eruption of the canine, postoperative morbidity, and length of stay in hospital for each group.

Results

There was no statistically significant difference in the interdental bone height (p = .61, Mann–Whitney U test). In group A, all patients had a satisfactory clinical outcome (type I or II), and in group B, only one patient had an unsatisfactory result (type III). The spontaneous eruption of the canine and the number of nights spent in the hospital were also similar for both groups. However, no patients in group A suffered postoperative complications, whereas three patients in group B reported either a limp or postoperative infection of the hip.

Conclusion

Both techniques produced satisfactory repair of the bony defect, but the open hip surgery resulted in greater postoperative morbidity.
Branchio-Oculo-Facial Syndrome with Cleft Lip and Bilateral Dermal Thymus
Authors: Bennaceur Sélim , Buisson Thierry , Bertolus Chloé , Couly Gérard
Published: October 2018

Objective

The objective of this study was to demonstrate that the branchio-oculo-facial (BOF) syndrome is a cervicocephalic neural crest maldevelopment.

Results

Using an embryologic study, we linked the clinical features and the level of the neural crest deficiency. We report here two cases of BOF syndrome with a particular branchial cleft presenting as bilateral supernumerary thymus glands on the surface of the skin; one of the cases was associated with tetralogy of Fallot. One patient underwent lip reconstruction at 4 months, combined with excision of bilateral auricular pits and superior labial fistula. The other patient had a surgical correction of the tetralogy of Fallot, and at 2 months, the two stages of the lip reconstruction were performed, combined with bilateral auricular pit excision. Both patients have shown normal developmental patterns to date.

Conclusion

The BOF syndrome must be considered as a neurocristopathy at different levels, with a tiny mesencephalo-prosencephalic lesion and a severe rhombencephalic lesion that includes seven consecutive hindbrain segments, from rhombomere 2 to rhombomere 8.