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PubMed1985

Toxic interaction of specific polychlorinated biphenyls and 2,3,7,8-tetrachlorodibenzo-p-dioxin: increased incidence of cleft palate in mice.

The induction of cleft palate in C57BL/6N mice is an extremely reproducible and sensitive indicator of 2,3,7,8-tetrachlorodibenzo-p-dioxin (TCDD) toxicity. This endpoint was used to look for potential interactions between two polychlorinated biphenyl (PCB) congeners and TCDD. Both 2,3,4,5,3',4'-hexachlorobiphenyl (HCB) and 2,4,5,2',4',5'-HCB are of relatively low toxic potency, but their biological properties differ. Pregnant mice were treated with TCDD and either HCB on gestation Days 10 through 13, and the fetuses examined for the presence of cleft palate and renal abnormalities on gestation Day 18. At a dose of TCDD which caused a low level of cleft palate, moderate hydronephrosis was observed. No renal or palatal anomalies were detected after 2,4,5,2',4',5'-HCB treatment, and the combination of this isomer with TCDD had no effect on the incidence of TCDD-induced cleft palate. 2,3,4,5,3',4'-HCB caused mild renal toxicity, but no cleft palate. However, treatment of pregnant mice with a combination of TCDD and 2,3,4,5,3',4'-HCB resulted in a 10-fold increase in the incidence of cleft palate. Thus, the toxicity of compounds such as TCDD may be enhanced by compounds of relatively low acute toxicity such as selected PCBs. The widespread environmental occurrence of such combinations suggests a need for further evaluation of the mechanism of this interaction.

Abnormalities, Drug-Induced↗

PubMed2002

Endoscopic partial adenoidectomy for children with submucous cleft palate.

OBJECTIVE: Children with submucous cleft palate who suffer from chronic nasal obstruction because of hypertrophic adenoids usually are not subjected to adenoidectomy because of the fear of postoperative velopharyngeal insufficiency. These patients present a therapeutic challenge because we are aware more than ever of the importance of normal nasal breathing and nocturnal respiration, especially during childhood. Our hypothesis was that transnasal endoscopic horizontal limited adenoidectomy may relieve nasal obstruction while preserving the function of the velopharyngeal valve. The objective of this study was to evaluate the efficacy of transnasal endoscopic horizontal partial adenoidectomy in patients with submucous cleft palate and adenoidal hypertrophy. SETTING: Patients were either referred to the outpatient clinic of the Palate Surgery Unit (seven patients) or were patients referred to the senior author's (Y.F.) private clinic. All the patients had been operated on by this senior author (Y.F.). PATIENTS: Ten children aged 3.5 to 13 years (six girls and four boys) with submucous cleft palate and hypertrophic adenoids were included in the study. All the patients were hyponasal and suffered nasal obstruction, loud snoring, and episodes of apnea. INTERVENTIONS: Endoscopic partial adenoidectomy was accomplished to open the lower third of the choanae. Nasal breathing was achieved in all the patients, and only mild snoring remained in two patients. The hyponasality disappeared and speech intelligibility normalized. Mild hypernasality developed in two patients but was still perceived as an overall improvement in speech. CONCLUSIONS: Transnasal endoscopic horizontal partial adenoidectomy may be an effective surgical method for relief of nasal obstruction while preserving velopharyngeal valve function in patients with submucous cleft palate who suffer from obstructive adenoids.

Adenoidectomy↗

PubMed2004

Evaluation of hearing thresholds in 3-month-old children with a cleft palate: the basis for a selective policy for ventilation tube insertion at time of palate repair.

Hearing thresholds in children with a cleft palate prior to cleft palate repair are not widely documented, and audiological criteria for short-term ventilation tube insertion do not exist. The aims of this prospective study are to estimate hearing thresholds in 40 children with a cleft palate by 3-month developmental age with auditory brainstem responses (ABRs) under natural sleep and to estimate a hearing threshold guideline for short-term ventilation tube insertion. Our results show a wide range of air conduction hearing thresholds using click ABRs (2-4 Hz), which ranged from 25 to 102 dBnHL in the left ear and from 25 to 80 dBnHL in the right ear with means of 53 and 49 and standard deviations of 17 and 13 respectively. The bone conduction thresholds ranged from 0 to 55 dBnHL with a mean of 26 and a standard deviation of 13. Eighty-three per cent of children had flat, type B, on high-frequency tympanograms, indicative of middle ear effusion. Thirty per cent of the infants had a cleft palate associated with a known syndrome. Currently, it is the authors' practice to use short-term ventilation tubes on a selective basis at the time of cleft palate repair when there is a conductive hearing loss of more than 55 dBnHL in the better ear as determined by ABR with type B high-frequency tympanograms. This threshold level takes into account electrophysiological and auditory pathway maturation discrepancies. With this as the guideline, between 28% and 35% of the children in this study would be eligible for surgery. This criterion still requires further validation.

Acoustic Impedance Tests↗

PubMed1998

Velopharyngeal function in nonsyndromic cleft palate: relevance of surgical technique, age at repair, and cleft type.

OBJECTIVE: The goal of this study was to determine the relative importance of surgical technique, age at repair, and cleft type for velopharyngeal function. DESIGN: This was a retrospective study of patients operated on by two surgeons using different techniques (von Langenbeck and Veau-Wardill-Kilner [VY]) at Children's Hospital, Boston, MA. PATIENTS: We included 228 patients who were at least 4 years of age at the time of review. Patients with identifiable syndromes, nonsyndromic Robin sequence, central nervous system disorders, communicatively significant hearing loss, and inadequate speech data were excluded. MAIN OUTCOME MEASURE: Need for a pharyngeal flap was the measure of outcome. RESULTS: Pharyngeal flap was necessary in 14% of von Langenbeck and 15% of VY repaired patients. There was a significant linear association (p = .025) between age at repair and velopharyngeal insufficiency (VPI). Patients with an attached vomer, soft cleft palate (SCP), and unilateral cleft lip/palate (UCLP) had a 10% flap rate, whereas those with an unattached vomer, hard/soft cleft palate (HSCP), and bilateral cleft lip/palate (BCLP) had a 23% flap rate (p = .03). Age at repair was critical for the unattached-vomer group (p = .03) but was not statistically significant for the attached-vomer group (p = .52). CONCLUSIONS: Surgical technique was not a significant variable either in aggregate or for the Veau types. Patients in the earliest repair group (8-10 months) were the least likely to require a pharyngeal flap. Early repair was more critical for HSCP and BCLP patients. There was no correlation between velopharyngeal insufficiency and Veau hierarchy. The attached vomer/levator muscle complex may be a more important predictor of surgical success than the anatomic extent of cleft.

Age Factors↗

PubMed1978

HLA phenotype frequencies in individuals with cleft lip and/or cleft palate.

The HLA types of 133 patients with cleft lip and/or cleft palate were determined. Caucasian patients with isolated cleft palate showed a possible association with HLA. Ten of 11 male patients had HLA--A2 as compared to one out of eight female patients (P less than 0.005). Caucasian males with cleft lip and cleft palate had a slight increased frequency of HLA--Aw24 (P = 0.07) and Mexican-American males with cleft lip and/or cleft palate showed an increase of antigen HLA-A28 (P = 0.07), though neither were statistically significant. Females with cleft lip and/or cleft palate from either racial group had no differences from the controls. The serum from 90 mothers of patients with cleft lip and/or cleft palate were reacted against their child's lymphocytes. Of these crossmatch tests, 12% were found to be positive. These preliminary results suggest that male patients with isolated cleft palate are worthy of further studies with respect of HLA associations.

Cleft Lip↗

PubMed1990

Patient compliance with cleft palate team regimens.

A cleft palate team's prescribed regimen requires prompt and continued compliance in order to meet the management objective and to justify the large investment of professional time. The purposes of this clinical investigation were to determine the rate of compliance with the recommendations made by a cleft palate team for its patients and to identify variables associated with compliance. A subject was defined as the person(s) who could best respond to questions concerning the management of the team's patient. Each subject was interviewed using a questionnaire. The mean patient compliance rate was 64.4%, and noncompliance with specific recommendations ranged from 12.5% to 100%. Using Jones and Caldwell's (1981) classification, 17.1% of the patients were classified as compliers, 78% as partial compliers, and 4.9% as noncompliers. The best predictor of compliance was a set of nine variables.

Cleft Palate↗

PubMed2003

Conditional inactivation of Tgfbr2 in cranial neural crest causes cleft palate and calvaria defects.

Cleft palate and skull malformations represent some of the most frequent congenital birth defects in the human population. Previous studies have shown that TGFbeta signaling regulates the fate of the medial edge epithelium during palatal fusion and postnatal cranial suture closure during skull development. It is not understood, however, what the functional significance of TGFbeta signaling is in regulating the fate of cranial neural crest (CNC) cells during craniofacial development. We show that mice with Tgfbr2 conditional gene ablation in the CNC have complete cleft secondary palate, calvaria agenesis, and other skull defects with complete phenotype penetrance. Significantly, disruption of the TGFbeta signaling does not adversely affect CNC migration. Cleft palate in Tgfbr2 mutant mice results from a cell proliferation defect within the CNC-derived palatal mesenchyme. The midline epithelium of the mutant palatal shelf remains functionally competent to mediate palatal fusion once the palatal shelves are placed in close contact in vitro. Our data suggests that TGFbeta IIR plays a crucial, cell-autonomous role in regulating the fate of CNC cells during palatogenesis. During skull development, disruption of TGFbeta signaling in the CNC severely impairs cell proliferation in the dura mater, consequently resulting in calvaria agenesis. We provide in vivo evidence that TGFbeta signaling within the CNC-derived dura mater provides essential inductive instruction for both the CNC- and mesoderm-derived calvarial bone development. This study demonstrates that TGFbeta IIR plays an essential role in the development of the CNC and provides a model for the study of abnormal CNC development.

Animals↗

PubMed2000

Von Langenbeck or Wardill procedures for primary palatal repair in patients with isolated cleft palate--speech results.

Sixty-six patients operated on for isolated cleft palate were evaluated as adults, and their speech was assessed clinically. Forty-four had undergone a von Langenbeck repair at the age of 7 months and 22 a Wardill repair at the age of 18 months. Speech was judged subjectively by the patients themselves, and perceptual assessment was made by three speech pathologists. The remaining speech problems, mainly hypernasality, were moderate or severe in 7 (16%) of the patients in the von Langenbeck group, and in 7 (32%) in the Wardill group. On the other hand, the patients in the Wardill group had fewer fistulas closed, and fewer velopharyngoplasties. There were no significant differences between the two methods regarding speech in adulthood. The Wardill method produced significantly more scar tissue clinically, which carries the potential risk of inhibition of facial growth. The present speech results will therefore be weighed against the outcome of maxillofacial growth for these patients.

Cleft Palate↗

PubMed2007

Closure of a large palatal fistula with maxillary segmental distraction osteogenesis in a cleft palate patient.

Various techniques have been proposed for the repair of palatal clefts. The first surgical closure technique that should be kept in mind is the use of adjacent mucosal flaps. When the palatal cleft is too large to close with adjacent mucosal flaps, distant flaps such as from the tongue or nasolabial region may be considered. This report presents a cleft patient who had previously undergone an unsuccessful palatal cleft repair with a tongue flap. The size of the large palatal fistula was reduced by approximating the segments to each other with maxillary anterior segmental distraction osteogenesis to make it more manageable using conventional mucosal flaps.

Adolescent↗

PubMed1987

A T-shaped musculomucosal buccal flap method for cleft palate surgery.

The primary aim of cleft palate surgery is not only to close the cleft palate but to push back the palate by repositioning the levator muscle to ensure that normal speech is obtained. Although the pushback operation using a mucoperiosteal flap is a readily effective method for velopharyngeal closure, postoperative fistula sometimes occurs, especially when the cleft palate is wide. Furthermore, postoperative maxillary deformity, possibly due to elevating the mucoperiosteal palatal flap, is extremely troublesome. For the purpose of pushing back the nasal mucosa, we applied the Kaplan buccal flap method, which is also applicable for reestablishing the levator muscle sling. The other buccal flap is covered on the hinged flap of the pared cleft margins. This results in far less disturbance of maxillary growth. We call it the T-shaped buccal flap method, and we have applied it in over 30 patients with various cleft palates and have obtained satisfactory results.

Cheek↗

PubMed1981

Alveolar reconstruction in adolescent patients with cleft palates.

Treatment of patients with cleft palates requires optimal interdisciplinary care. The efforts of the entire team treating the patient with cleft palate must be closely coordinated, even when the deformity is confined to the alveolus. In a significant number of patients with cleft palate, surgical reconstruction of the alveolus and adjacent soft tissue is a necessary adjunct to orthodontic, prosthetic, and facial soft tissue management. Surgical treatment and its complications are discussed, with specific case examples.

Adolescent↗

PubMed2001

Prevalence of aneuploidy and additional anatomic abnormalities in fetuses and neonates with cleft lip with or without cleft palate: a population-based study in Utah.

OBJECTIVE: To determine the prevalence of aneuploidy and additional major anatomic abnormalities in fetuses and neonates with cleft lip with or without cleft palate. METHODS: All cases of cleft lip with or without cleft palate (cleft lip/cleft palate) occurring in Utah from 1995 through 1999 were reviewed by using the Utah Birth Defect Network population-based surveillance system. All pregnancy outcomes are included (stillborn, live born, and termination) in this analysis. RESULTS: Of 263 cases of cleft lip/cleft palate, 72 (27.4%) were unilateral cleft lip, 112 (42.6%) were unilateral cleft lip and cleft palate, 12 (4.6%) were bilateral cleft lip, and 67 (25.5%) were bilateral cleft lip and cleft palate. Fifteen (5.7%) of the 263 fetuses and neonates were aneuploid. One (1.2%) with cleft lip (unilateral and bilateral combined) was aneuploid. Five (4.5%) of the fetuses and neonates with unilateral cleft lip and cleft palate were aneuploid compared with 9 (13.4%) of fetuses and neonates with bilateral cleft lip and cleft palate. In known or presumed euploid fetuses and neonates, additional sonographically occult major anatomic abnormalities occurred in 5 (7.0%) of 71 with unilateral cleft lip, 18 (16.8%) of 107 with unilateral cleft lip and cleft palate, 1 (8.3%) of 12 with bilateral cleft lip, and 12 (20.7%) of 58 with bilateral cleft lip and cleft palate. These abnormalities primarily involved the heart and the central nervous system. CONCLUSIONS: Amniocentesis for karyotype should be offered in all cases of cleft lip/cleft palate because of the risk of aneuploidy. Patients should be counseled that sonographically occult additional anatomic abnormalities might be present with all clefts.

Amniocentesis↗

PubMed1991

Hypodontia in patients with isolated cleft palate, its relationship to etiopathogenesis.

The authors studied the incidence of hypodontia in 200 patients with isolated cleft palate. These patients were divided into 3 groups according to the degree of malformation (complete cleft palate, incomplete cleft palate, soft palate cleft). In the 3 groups, hypodontia was studied on permanent teeth, with the exception of the 3rd molars. Compared with the healthy population, hypodontia in patients with cleft palate appeared at a significantly higher rate (25.5%). No difference was found between boys and girls. Hypodontia in the mandible appeared in 18%, in the maxilla in 10%. The upper jaw showed maximum incidence in complete cleft palate (21%), while in the mandible hypodontia was observed most frequently in soft palate clefts (37% patients). In most cases the dental germs of the 2nd lower premolar failed to develop. Our results correlate with hypotheses based on experimental studies on the etiopathogenesis of facial clefts.

Adolescent↗

PubMed2005

Delayed detection of cleft palate: an audit of newborn examination.

AIMS: To identify prevalence of delayed detection of cleft palate, and associated factors that could lead to improved identification at neonatal clinical examination. METHODS: Audit of hospital notes, parental questionnaire incorporating open ended questions, and telephone questionnaire of junior doctors in the referring hospitals incorporating fixed choice questions. RESULTS: Of 344 cleft palate patients without cleft lip or submucous cleft palate, the day the cleft was detected was recorded in 92%. Delayed detection, after the first day, was 28% overall, distributed as 37% with isolated cleft palate and 23% with syndromic cleft palate. Narrow V shaped clefts were more likely to be delayed in detection compared with broad U shaped clefts, as were soft palate clefts compared with hard palate clefts. Five with isolated cleft palates were not detected until after the first year. Babies born at home were unlikely to be detected on day 1. Symptoms were significantly increased in the delayed detection group for feeding problems and nasal regurgitation. A telephone questionnaire of trainee paediatricians in referring units revealed that digital examination was more commonly practised than visual inspection, and few recalled receiving specific instruction on examination of the palate. CONCLUSION: Delayed detection of cleft palate was not uncommon, and the features of those more likely to be missed suggested digital examination was related. Trainee doctors and midwives should be instructed to inspect visually using a light and tongue depressor, then digitally if submucous cleft palate is suspected.

Abnormalities, Multiple↗

PubMed2000

Linguistic development in cleft palate patients with and without compensatory articulation disorder.

INTRODUCTION: Cleft palate patients frequently show compensatory articulation disorder (CAD). CAD severely affects speech intelligibility and requires a prolonged period of speech intervention. CAD has been considered a phonologic disorder. Thus, it seems necessary to explore the relationship between CAD and language development. OBJECTIVE: To study the relationship between language development and the presence of CAD in cleft palate patients. MATERIALS AND METHODS: Cleft palate children with residual velopharyngeal insufficiency (VPI) after palatal closure, with and without CAD were studied. Only patients with an age ranging from 3 to 8 years were included in the study group. Twenty-nine cleft palate patients with residual VPI and CAD were included in the first group (active). The second group was assembled with 29 cleft palate patients with residual VPI without CAD, matched by age and sex (control). For evaluating language development, all patients were analyzed using the Situational-Discourse-Semantic (SDS) Model [13]. This Model is a valuable tool for conducting naturalistic observation and descriptive assessment of language development. The SDS Model provides a detailed description of three contexts (situational, discourse, and semantic) in ten levels of cognitive and linguistic organization. RESULTS: In all contexts considered by the model of cognitive and linguistic organization used for this study, i.e. SDS, a Fischer exact test demonstrated that patients with CAD showed a significantly higher frequency of language delay as compared with patients without CAD. None of the patients present with CAD showed an adequate level of language development. The degree of language delay was greater in the situational context as compared to the semantic and discourse contexts. CONCLUSIONS: Cleft palate patients present with CAD, demonstrated a significantly higher frequency of delay in language development as compared with cleft palate patients present with VPI without CAD. From the results of this paper, it seems that a detailed evaluation of all aspects of cognitive and linguistic organization should be performed in cleft palate patients, especially in patients present with CAD. Moreover, it seems that speech intervention in cleft palate patients with CAD should address not only the articulation process, but also specific aspects of language development.

Adaptation, Physiological↗

PubMed1988

Olfactory deficits in boys with cleft palate.

An odor identification task was used to determine whether individuals with cleft palate (with or without cleft lip) also have an increased prevalence of olfactory deficits. Olfactory responses of 35 affected subjects (7 to 22 years of age) were compared with those of 68 subjects of comparable age without cleft palates. Subjects were requested to identify the smell of ten common household odors. They selected their responses from an alphabetized list of the test odorants. After a practice trial, the set of odorants was presented five times in randomized sequences. The percentage of correct responses increased with age for prepubertal and pubertal subjects without cleft palates. Although the olfactory scores of girls without cleft palates continued to increase after puberty, this trend was absent in boys. On the average, the girls with cleft palates, compared with only three of 34 boys without cleft palates, had olfactory scores less than 60% correct. There was no evidence of heterogeneity in the magnitude or direction of the relationship between any of the subtypes of cleft palate and olfactory dysfunction. In this study, cleft palate is more strongly associated with olfactory deficits in boys than in girls, suggesting the possibility that the deficit may be a sex-influenced trait.

Age Factors↗

PubMed2003

Treatment of oralfacial clefts by state-affiliated craniofacial centers and cleft palate clinics.

BACKGROUND: Oralfacial clefting (OFC) disorders require expedient evaluation and treatment to obtain optimal outcome. In Florida, there is a statewide program targeted to the care of infants with OFC. We therefore sought to determine statewide referral and treatment patterns of children born with OFC identified through the Florida Birth Defects Registry. METHODS: Using data for 1996 and 1997 and ICD-9 CM codes 749.00 - 749.25, we identified 539 OFC cases. All cases were matched with the evaluation and treatment records of the statewide Children's Medical Services' (CMS) craniofacial centers (CFC) and cleft palate clinics (CPC). The likelihood of CMS contact was examined with respect to demographic and other descriptive data characterizing the OFC cases. RESULTS: 42% (227/539) of OFC cases were evaluated at or known to the CFC or CPC. Children with cleft lip and palate were more likely to have had contact than were those with cleft lip or cleft palate alone. The CFC and CPC programs were most likely to provide evaluation between age 2 months and 3 years. Of 12 counties with occurrences of more than 15 OFC cases, 2 had significantly lower contact rates, suggesting possible problems in accessibility or reporting of services. CONCLUSIONS: Statewide Birth Defect Registry data can be used in collaboration with statewide treatment programs to gain insight into referral patterns and provision of services. Factors influencing access to services and quality of care, though not addressed by this study, could be prospectively incorporated into such a project.

Abnormalities, Multiple↗

PubMedDate not supplied

[Case of cleft palate].

The present paper briefly outlines the pathogenesis of cleft palate and proposes a classification system for cleft palate since both are considered essential for the understanding and assessment of its sequelae. The Authors describe one case of cleft palate. Cleft palate patients present functional and aesthetic alterations in the maxillomandibular area that often require prosthetic correction. Given the features of these malformations therapy and rehabilitation should be programmed as soon as possible after birth and the plastic surgeon should be informed of the factors that cause the malformations and influence the growth of such patients.

Age Factors↗