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PubMed1991

Managing the cleft lip and palate patient.

We have attempted to elucidate, first through an examination of the basic science of clefting and then through a description of the roles of the various members of the cleft palate team, an understanding of the multiplicity of problems faced by this heterogeneous group of children with cleft lip or palate. With an incidence of cleft of 1 in 700 births, all practicing pediatricians will at some time be faced with a patient with a cleft. Pediatric management begins in the hospital nursery by ruling out possible associated anomalies (e.g., congenital heart disease or urinary tract) or syndromes. At the same time, cleft palate nursers may be needed to overcome feeding problems. Simultaneously, counseling for the family begins with a positive attitude toward outcome and an initial explanation of the schedule of corrective procedures, i.e., lip repair at 3 months of age and palate repair at about 1 year. The counseling should incorporate an understanding of the cause of clefting, both its genetics and teratogenetics, and then proceed to noting the actual recurrence risks of 1% for a new cleft and 16% if there is a positive family history in a first-degree relative. The recurrence risks in recognized syndromes may follow mendelian patterns. Later, the pediatrician can help the family deal with multiple ear infections and the likely need for tympanostomy tubes, anticipating the need for tubes in a way that might facilitate placement at the time of anesthesia for the lip or the palate repairs. An understanding of the speech and language difficulties that may be encountered in later infancy may relieve parental anxiety. Later, after palate repair, knowledge of velopharyngeal incompetence may avert premature hypernasal speech problems caused by adenoidectomy, which should be avoided if at all possible. The pediatrician needs to be aware of the tooth malformations that accompany clefting of the alveolus as well as the increased susceptibility to caries, so that the family may be directed to early intervention by the pediatric dentist. With support by pediatrics of the efforts of pediatric dentistry, the child is then in optimal condition for orthodontics. Continually keeping the child's emotional adjustment in mind, the pediatrician can encourage the child and family, knowing that with palatal expansion techniques plus possible bone graft surgery to the cleft alveolus at about 9 years of age, the child may be orthodontically corrected to very near normal.(ABSTRACT TRUNCATED AT 400 WORDS)

Child↗

PubMed2003

The effect of orthognathic surgery on taste function on the palate and tongue.

PURPOSE: Perceived taste intensity and taste quality identification on localized regions of the palate and tongue were examined for 9 patients before orthognathic surgery and again at 1 to 2 and 6 to 9 months after surgery. Taste function would be at risk on the palate after maxillary Le Fort I osteotomy (LFI) and on the tongue after mandibular sagittal split osteotomy (SSO) because of potential damage to peripheral nerves conducting afferent chemosensory information from these regions. PATIENTS AND METHODS: Three patients had LFI and SSO, 1 had LFI only, and 5 had SSO only. Patients rated taste intensity (using a 10-point fixed-interval scale) and identified taste quality of 4 solutions (NaCl, sucrose, citric acid, and quinine.HCl) brushed with a cotton-tipped applicator on each of 6 oral locations (left and right soft palate, left and right anterior and posterolateral tongue). RESULTS: Perceived taste intensity of NaCl, sucrose, and citric acid was reduced on average to 34% of presurgery values on the palate for patients who underwent LFI, but the taste intensity of quinine was not affected. LFI also affected the ability to correctly identify the quality of tastants applied to the palate: Patients made 38% correct quality identifications postsurgery compared with 91% presurgery. Perceived taste intensity of quinine.HCl placed on the tongue was reduced to 72% of its presurgery value after SSO, but the taste intensities of NaCl, sucrose, and citric acid were not affected. Correct quality identifications of sucrose, citric acid, and quinine.HCl were reduced to 75% at 2 months post-SSO compared with 96% presurgery and at 6 months postsurgery, but identification for NaCl was 96% before and after surgery. CONCLUSION: In the patients studied, taste function on the palate was significantly decreased for 6 to 9 months after LFI, likely a result of impairment of function of the greater superficial petrosal branch of the facial nerve. Lingual taste function, reduced at 1 to 2 months after SSO, likely due to impaired chorda tympani nerve function, improved by 6 to 9 months. Palatal and lingual neurosensory testing can be used to identify reversible sequelae of oral maxillofacial surgery.

Adolescent↗

PubMed2001

Prenatal diagnosis of cleft palate by three-dimensional ultrasound.

Prenatal diagnosis of cleft palate is very important to prenatal consultation and management after birth. To examine if three-dimensional (3-D) ultrasound (US) is an accurate diagnostic method for clinical use, we analyzed our experience in detecting cleft palate by 3-D US. From June 1996 to January 2000, 21 fetuses with facial clefts were scanned by 2-D US, as well as by 3-D US. The coronal and oblique planes were reconstructed by 3-D US to detect the cleft palate. In addition, level II US was performed to find any possibly associated anomalies. All the scans were recorded on optic disks for final analysis. In our study, the gestational age when prenatal diagnosis was made by US initially was between 20 and 34 weeks. The accuracy for prenatal diagnosis of cleft lip with or without cleft palate by 3-D US was 100%, which was superior to that by 2-D US (p < 0.05). In addition, we proposed a novel method to evaluate the cleft palate systemically by 3-D US. In conclusion, from our study, fetuses with cleft lip combined with or without cleft palate can be easily differentiated by 3-D US. The reconstruction of coronal and oblique planes by 3-D US is a powerful tool for detecting cleft palate.

Cleft Lip↗

PubMed1993

Mandibular growth in patients with cleft lip and/or cleft palate--the influence of cleft type.

A total of 229 adult white patients with cleft lip or palate were evaluated, through cephalometric radiographs, to determine cleft-type influence on mandibular structure and spatial position. The sample includes adult patients with unilateral complete cleft lip and alveolus (n = 50), complete cleft lip and palate (n = 118), and isolated cleft palate (n = 61). A group of 65 subjects with normal occlusion and without cleft lip or palate was used as a control group. All groups were matched according to sex. When compared, the cleft groups and the normal group showed that the mandibular structure was significantly different. The mandible in the cleft groups displayed shorter mandibular ramus and body length, without difference between the cleft types. Mandibular position relative to cranial base was similar in the cleft groups with involvement of the palate, and differed significantly from that observed in the group with cleft lip and alveolus and in the subjects without cleft. Palatal clefts induce a significant downward and backward rotation of the mandible associated with a more obtuse gonial angle. Thus a small mandible is a structural characteristic of all cleft types studied. However, mandibular shape and spatial position are strongly influenced in clefts that involve the palate.

Adolescent↗

PubMed1987

Mucoperiosteal migration after palatal surgery in beagle dogs. A longitudinal radiographic study.

In this study, the effect of palatal surgery on the mucoperiosteum was investigated radiographically. 33 beagle dogs were used, divided into 3 experimental groups, a control group and a sham operated group. At the age of 6, 16 or 25 weeks in the experimental groups, a soft tissue defect was created in the median region of the palate by excising a standardized elliptical mucoperiosteal flap. This defect was closed according to the Von Langenbeck technique. Metallic implants were placed in the mucoperiosteum. Dorso-ventral radiographs were taken until the age of 37 weeks. Increments of distances between these mucoperiosteum markers were calculated. Wound contraction in the denuded areas, recorded as the approaching of the opposite metallic implants followed the same pattern for all experimental groups and was restricted to the first 2 experimental weeks. In all experimental groups, the anterior and posterior overall palatal width diminished during the early wound healing compared to the control group. The largest initial effect was found in the anterior region of the palate. The margins of the mucoperiosteal tissue in the median region of the palate moved towards the denuded areas during the first postoperative week. Palatal surgery as performed did not influence antero-posterior distances on the palatal mucoperiosteum.

Animals↗

PubMed2000

Reconstruction of human hard-palate mucosal epithelium on de-epidermized dermis.

Artificial hard-palate mucosa equivalents were reconstructed using keratinocytes derived from normal human hard-palate and de-epidermized dermis. Reconstructed hard-palate mucosal epithelium formed in three-dimensional culture was compared to native hard-palate mucosal epithelium and reconstructed oral buccal mucosal epithelium with regard to keratin expression. Artificial hard-palate mucosal epithelium reconstructed in medium with delipidized serum showed a differentiation pattern similar to that of hard-palate epithelium in vivo. The present study also confirmed that keratinocytes derived from hard-palate mucosa are intrinsically different from those of nonkeratinizing oral surfaces.

Cell Communication↗

PubMed2003

Collagen XI sequence variations in nonsyndromic cleft palate, Robin sequence and micrognathia.

Cleft palate is a common birth defect, but its etiopathogenesis is mostly unknown. Several studies have shown that cleft palate has a strong genetic component. Robin sequence consists of three of the following four findings: micrognathia, glossoptosis, obstructive apnea, and cleft palate. While cleft palate is mainly nonsyndromic, about 80 percent of Robin sequence cases are associated with syndromes. Mutations in genes coding for cartilage collagens II and XI, COL2A1, COL11A1 and COL11A2, have been shown to cause chondrodysplasias that are commonly associated with Robin sequence, micrognathia or cleft palate. We therefore analyzed a cohort of 24 patients with nonsyndromic Robin sequence, 17 with nonsyndromic cleft palate and 21 with nonsyndromic micrognathia for mutations in COL11A2. A total of 23 Robin sequence patients were also analyzed for mutations in COL2A1 and COL11A1. We detected two disease-associated mutations in patients with Robin sequence, an Arg to stop codon mutation in COL11A2 and a splicing mutation in COL11A1. Two putatively disease-associated sequence variations were found in COL11A1 in Robin sequence patients, one in COL11A2 in a patient with micrognathia and one in COL2A1 in two patients with Robin sequence. The results showed that sequence variations in these genes can play a role in the etiology of Robin sequence, cleft palate and micrognathia but are not common causes of these phenotypes.

Cleft Palate↗

PubMed2000

Psychological study of cleft palate children with or without cleft lip by kinetic family drawing.

To clarify how cleft palate children with or without cleft lip perceive their family, we applied the Kinetic Family Drawing (KFD) technique to 60 cleft palate children (with or without cleft lip). The children were aged 7-9 years (in the 1-3 grade of elementary school) and attended the Department of Oral Surgery, Nagoya University Hospital, between 1990 and 1997. Controls consisted of 100 normal elementary school children of the same age. Major findings were: (1) Compared with normal children, cleft lip and palate boys and cleft palate only girls drew their self-images significantly more often in region D (lower right) and in region A (upper left), respectively. (2) Cleft palate children with or without cleft lip felt anxiety and fear toward their family, rarely viewing their homes as a restful place; this suggested insecurity. Uniquely dynamic relations were also suggested in their homes. (3) Cleft palate children with or without cleft lip often perceived their father or mother as persons easily acceptable in society. Rather remote psychological distance between parents and cleft palate children with or without cleft lip was suggested.

Activities of Daily Living↗

PubMed2005

Cleft lip and palate versus cleft lip only: are they distinct defects?

Cleft lip defects are usually regarded as a single entity, with the assumption that an accompanying cleft palate represents the more severe form. The authors linked data from the Medical Birth Registry of Norway with medical records from two centralized centers to provide a population-based data set. They assessed the distribution of cleft lip only and cleft lip with cleft palate by covariate. Among 1.8 million Norwegian livebirths between 1967 and 1998, there were 1,572 cases of cleft lip with cleft palate and 1,122 cases with cleft lip only. Seventeen percent of those with cleft lip and palate had another defect compared with 9% of those with cleft lip only. For boys, the risk was greater for cleft lip and palate than for cleft lip only (odds ratio=2.4 vs. 1.8, p<0.001 for difference). The risk of cleft lip only, but not of cleft lip and palate, was increased for twins (odds ratio=1.6 vs. 1.1, p=0.11) and infants whose parents were first cousins (odds ratio=2.7 vs. 0.7, p=0.07). Although cleft lip with cleft palate may simply represent a more severe form of the defect, epidemiologic assessments of cleft lip should, when possible, include separate analyses of these two groups.

Abnormalities, Multiple↗

PubMed1996

Assessing third-year medical students' knowledge of and exposure to cleft palate before and after plastic surgery rotation.

The purpose of this study was to evaluate third-year medical students' knowledge of and exposure to cleft palate before and after a rotation in plastic surgery. A 26-item questionnaire was administered to 37 students before and after their 12-week rotation that included the evaluation and management of patients with cleft palate. The questions addressed various aspects of the disorder, including identification of the members of a cleft palate team. Students were also asked about their clinical and academic exposure to cleft palate. When the students' responses to the 19 basic information items about cleft palate and associated problems were compared before and after the rotation, positive changes were documented for 14 of the 19 questions, minimal negative changes for 4, and no change for 1 question. The overall percentage of students indicating any type of clinical or academic exposure to cleft palate increased significantly after the rotation. Findings from this study suggest that an undergraduate clinical rotation in plastic surgery can increase the knowledge of and exposure to cleft palate. It was clear that supplemental learning can occur in other settings outside the classroom through interactions between faculty and students without additional curricular time.

Cleft Palate↗

PubMed1980

Velopharyngeal insufficiency due to hypoplasia of the musculus uvulae and occult submucous cleft palate.

With the help of nasopharyngoscopy, it was possible to delineate specific morphologic changes in the palates of patients with velopharyngeal insufficiency, without an overt cleft and without the triad of symptoms of submucous cleft palate, visible through the oral cavity. Such malformations are part of the broad spectrum of the faulty midline mesodermal fusion of the palate. This anomaly is aptly called occult submucous cleft palate, because it can only be detected by viewing the functioning palate from the nasal surface. The musculus uvulae is either absent or deficient and is frequently associated with some degree of muscular diastasis that does not involve the oral surface. Like the cleft of the secondary palate, the submucous cleft palate often occurs as part of a generalized syndrome of multiple malformations.

Abnormalities, Multiple↗

PubMed1982

Early cleft palate repair and speech outcome.

Speech production and age at palatal repair were investigated in 80 cleft palate children. Children whose palates were repaired prior to the onset of speech production demonstrated significantly better speech than those whose palates were repaired between 12 and 27 months of age. The supposition that earlier palatal repair results in more normal speech development was, in fact, demonstrated in these cases. Rather than using chronologic age alone as the deciding factor in determining timing of initial palatal repair, the stage of each child's phonemic development should be considered if maximum speech potential is to be achieved and if speech development is to parallel normal noncleft peers. Determining this stage of development through early speech and language evaluations, beginning at 6 months of age, thus becomes an essential component in the habilitation of children with cleft palate. Continued research is needed to ensure against giving the obtainment of early speech normalcy disproportionate emphasis over craniofacial growth considerations. To this end, continued cooperative research between surgeons and speech pathologists is imperative in order to base these important decisions on substantiated findings.

Child, Preschool↗

PubMed1991

Growth status of children treated for unilateral cleft lip and palate.

Cephalometric distances, angles, and proportions were evaluated for 32 children 5 to 8 years of age treated for unilateral cleft lip and palate. The children were age and sex matched with untreated controls with normal skeletal relationships. The unilateral cleft lip and palate sample was treated by the same surgeon and orthodontist using the same techniques and appliances. Measures of overall facial proportions, facial convexity, and prognathism were not significantly different between the two groups. The primary group differences pertain to the posterior aspect of the maxilla, which is vertically short in the unilateral cleft lip and palate sample. Horizontally, the maxilla of the unilateral cleft lip and palate children was significantly longer, producing a steeper palatal plane. In addition, the zygoma and orbits of unilateral cleft lip and palate children were somewhat retruded; the posterior cranial base and total mandibular length also were longer in the unilateral cleft lip and palate children.

Analysis of Variance↗

PubMed1997

Two-dimensional cephalometric analysis of the effects of subperiosteal palatal soft-tissue expansion in growing cats.

The feasibility and possible effects of palatal soft-tissue expansion in palatal repair were studied. A prospective longitudinal animal experiment was performed in 75 growing cats assigned to 5 groups. In 31 cats, a midline defect was made, and bipediced flaps were raised at the age of 8 weeks (stimulated Langenbeck operation) in order to create palatal scars. At the age of 14 weeks, custom-made tissue expanders were inserted palatally in 61 animals. Tissue expansion was performed by weekly inflation in 33 cats (16 without and 17 with scars) for an 8-week period. The remaining 28 cats (14 without and 14 with scars) served as sham groups. A control group was formed by 14 animals (without scars and without tissue expanders). Soft-tissue gain and its effects on maxillofacial growth and development were measured in the midsagittal plane on tracings from standardized lateral radiographs. The effects of the experimental interventions were evaluated for 8 weeks after removal of the tissue expanders. Not all the cats yielded results at all time periods. This study showed that soft-tissue expansion of palatal mucoperiosteum is feasible. The surgically induced scars did not cause significant differences between the different groups in the midsagittal plane, and the data from both expansion and sham groups could be pooled. Significant soft-tissue gain was achieved by the tissue-expansion technique. Iatrogenic side effects were significant anteroposterior growth retardation at the level of the bony palate and an increase in vertical growth of the anterior nasomaxillary height and the posterior skull height during active tissue expansion. After removal of the tissue expanders, some accelerated growth was found in the tissue expansion in the scarred tissue group, with initial correction of the abnormal growth at the cranial base level. It is concluded that palatal soft-tissue expansion is possible in growing cats. This technique, however, impaired maxillofacial growth and development.

Analysis of Variance↗

PubMed1997

Bilateral buccal flaps with double opposing Z-plasty for wider palatal clefts.

Described is a technique that has evolved from the challenges of closure of larger cleft palate defects and that we are now using in preference over other techniques to repair a wide variety of clefts. Soft-palate closure and muscular sling reconstruction are accomplished using a modified Furlow technique. An associated cleft of the hard palate and the gaps produced by posterior displacement of the reconstructed soft palate are closed by adding tissue, buccal flaps, rather than by closure under tension or leaving residual raw surfaces. Palate lengthening is achieved both by the Z-plasty effect and by the interposition of buccal flaps between the hard and soft palate. Seventy-six palates have been repaired using this procedure. There were three postoperative fistulas.

Cleft Palate↗

PubMed2002

Sleep-related obstructive disordered breathing in cleft palate patients after palatoplasty.

Sleep-disordered breathing is frequently associated with children presenting congenital midface defects. Because of structural and functional anomalies in the upper airway, children with cleft palate, especially after surgery, may carry a higher risk of developing sleep-disordered breathing. However, the presence of such sleep-disordered breathing in older cleft palate children has not been emphasized. The aim of this comparative overnight cardiorespiratory sleep study was to evaluate cleft palate patients according to sleep-disordered breathing. A group of 43 cleft palate children (17 girls and 26 boys; mean age, 12.1 +/- 3.8 years) was compared with a control group of 20 randomly selected, noncleft children matched for age, sex, and body mass index. None of the patients suffered from manifest sleep-disordered breathing. Cleft palate patients had a statistically significantly higher respiratory disturbance index and snoring index, but no increased apnea index. The data suggest that cleft palate patients having undergone primary closure of the palate demonstrate microsymptoms of nocturnal upper airway obstruction.

Adolescent↗

PubMed2006

Clinical and epidemiologic studies of nonsyndromic cleft lip and palate in china: analysis of 4268 cases.

The aim of our study was to review clinical and epidemiologic characteristic of the nonsyndromic cleft lip and cleft palate over a period of 10 years at West China Stomatological Hospital, Sichuan University. Four thousand two hundred sixty-eight nonsyndromic cleft lip and cleft palate cases were retrospectively analyzed according to the following variables: general information, cleft type, maternal age, familial history, as well as associated malformations, et al. In these cases, 1075 of which were cleft lip, 1985 were cleft lip with cleft palate; 1208 were isolated cleft palate. There were more men than women in the cleft lip and cleft lip with cleft palate group, while there were more women than men in the isolated cleft palate group. There were significantly more unilateral cleft lip cases than cleft lip on both sides; the cases of cleft lip on the left side were more than that of cleft lip on the right side. The cases with inherited history accounted for 6.68% of all the cases. Most infection cases occurred among the deleterious factors and the complications experienced during the first 3 months after the pregnancy. In all of the 14 twin cases, only 1 of the babies was affected. There were totally 152 cases with other associated malformations. Patients with cleft lip born in November to January were less than those born in the other three quarters of the year. Patients of A blood group composed a higher proportion than the control group. Our data may provide references for appropriate resource use, cleft lip and cleft palate prevention programs, and counseling programs with China-specific data.

Adult↗

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↗