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An investigation of the dentition of parents of children with cleft lip and palate.

OBJECTIVE: The aim of this investigation was to identify whether any dental features in parents of children with cleft lip and palate can be useful as predictors of clefting. METHODS: The dentition of 28 pairs of parents of children with complete unilateral (17) and bilateral (11) cleft lip and palate and 21 pairs of parents of noncleft children were evaluated. Clinical and radiographic examinations were carried out to identify abnormalities of tooth number and morphology. Study cast assessment was undertaken to evaluate incisor relationship, overjet, overbite, intercanine widths, and mesiodistal widths of individual teeth, and these data were subjected to statistical analysis. RESULTS: The prevalence of abnormalities of tooth number in parents of cleft children was similar to those reported elsewhere for general populations. In parents of children with unilateral clefts, there was no tooth-size asymmetry between teeth on the side corresponding to the child's cleft and noncleft sides, respectively. No differences were found in tooth widths and intercanine widths between parents of children with unilateral clefts and parents of children with bilateral clefts. The incisor relationship, overjet, overbite, and intercanine widths were found not to differ statistically between the parents of cleft children and parents of noncleft children. CONCLUSIONS: No predictors of clefting could be identified in the dentition of parents of children with cleft lip and palate.

Adult↗

Evaluation of primary dentition in cleft lip and palate children with and without natal/neonatal teeth.

OBJECTIVE: Natal/neonatal teeth are very common in children with complete unilateral and bilateral cleft lip and palate. The extraction of these teeth is the usual treatment in the Hospital for Rehabilitation of Craniofacial Anomalies. The objective of this study was to verify whether these teeth could be of the normal complement or whether they were supernumerary. DESIGN: The primary dentition of children with cleft lip and palate with and without natal/neonatal teeth was compared at the Hospital for Rehabilitation of Craniofacial Anomalies. SUBJECTS: The sample consisted of 55 children with complete unilateral and bilateral cleft lip and palate with natal/neonatal teeth and 54 without. RESULTS: No positive association between these groups and missing lateral incisor or supernumerary or complete dentition was found. CONCLUSION: The extraction of the natal/neonatal teeth did not alter the final complement of primary teeth, and these teeth could be the lateral incisor or supernumerary.

Anodontia↗

Dental health indices and caries associated microflora in children with unilateral cleft lip and palate.

OBJECTIVE: To investigate the dental health and caries related microflora of children with unilateral cleft lip and palate. STUDY GROUP: Sixty children with unilateral cleft lip and palate and matched controls. OUTCOME MEASURES: The decayed, missing, and filled teeth and surfaces in both the deciduous and permanent dentitions. The presence of developmental defects and plaque and gingivitis scores were also recorded. Plaque was collected from 25 of the children and their matched controls from three different sites, which were (1) the first approximal site distal to the cleft, (2) a contralateral anterior site, and (3) a remote site. It was cultured for Streptococcus mutans and lactobacilli. Plaque was collected from two sites in the matched controls. RESULTS: There was no significant difference in the caries, plaque, and gingivitis scores between the children with cleft palate and the controls. A greater number of enamel opacities were recorded in the control group, and there was a higher prevalence of enamel discoloration in the children with cleft lip and palate. There was no significant difference in the proportion of S. mutans or lactobacilli at the cleft site, compared with the unaffected site in the study group, although there was an anterior-posterior gradient in the proportion of S. mutans. There was no significant association between the stagnation area at the cleft site and the bacteria associated with dental caries.

Adolescent↗

Three-dimensional Z-plasty in the correction of the unilateral cleft lip nasal deformity.

OBJECTIVE: Numerous methods have been introduced for correction of the cleft lip nasal deformities, but no single procedure has given sufficiently satisfactory results to provide a surgical standard. Much effort has been put on restoring cartilaginous structures using alar cartilage modification and suspension. But even after the cartilage framework is repositioned well, redundant alar webbing is still unsightly and frequently conspicuous. This paper presents a procedure combining the usual open rhinoplasty technique and three-dimensional Z-plasty in an external approach to remove the alar web and to lengthen the columella at the same time. METHODS: Open rhinoplasty was performed in 26 consecutive patients with unilateral cleft lip nasal deformity from 1991 to 1996. We used an open rhinoplasty approach, which is a combination of the usual infracartilaginous incision on the noncleft side and a small triangular flap on the cleft side. RESULTS AND CONCLUSIONS: This external rhinoplasty incision can provide a wide surgical field for the handling of the whole cartilage framework. The three-dimensional Z-plasty utilizing redundant alar skin not only removes the alar web but also helps lengthen the columella and provides soft tissue to the nasal vestibule. This technique is easy, and the postoperative result is very consistent. Our open rhinoplasty approach has several advantages and can be a useful approach in the correction of the unilateral cleft lip nasal deformity.

Adolescent↗

Nasalance measures in Marathi consonant-vowel-consonant syllables with pressure consonants produced by children with and without cleft lip and palate.

OBJECTIVE: The purpose of this study was to ascertain whether there exist differential nasalance measures for consonant-vowel-consonant syllables consisting of different pressure consonants articulated by Marathi-speaking children with and without repaired cleft lip and palate. PARTICIPANTS: Ten Marathi-speaking children with repaired cleft palate between the ages of 5 and 12 years formed the experimental group. The control group consisted of 10 age- and sex-matched children with no clefts and no history of any hearing, speech, or voice disorder. METHODS AND MATERIALS: The Kay Elemetrics nasometer model 6200-2 (IBM-PC version) was used for obtaining nasalance measurements. Speech material consisted of eight Marathi monosyllabic words consisting of the pressure consonants in the initial position. The correlation between perceptual judgments of nasality and the instrumental measures was studied by computing the Phi coefficient. RESULTS: Results indicate that there exist differential mean nasalance scores for the syllables with different pressure consonants articulated by Marathi-speaking subjects with repaired cleft lip and palate and those without cleft lip and palate. Correlation coefficient computed between the instrumental measures and the perceptual judgments of nasality indicates moderate correlation between the two measures. CONCLUSIONS: Presence of differential nasalance scores may have diagnostic and therapeutic implications. Results also emphasize the need to evaluate nasal resonance in stimuli other than the traditional ones.

Analysis of Variance↗

Spectral analysis of prespeech sounds (spontaneous cries) in infants with unilateral cleft lip and palate (UCLP): a pilot study.

OBJECTIVE: The objectives of the present study were: (1) to analyze the cry features of infants with cleft lip and palate (UCLP) by means of spectral analysis, (2) to describe changes of the acoustic parameters from birth until 9 months of age, and (3) to compare these data with existing cry data of infants without cleft (control group). DESIGN: The study was designed on a interdisciplinary, prospective, and longitudinal basis. SETTING: Interdisciplinary study: (1) Institute of Anthropology at the Humboldt-University, Berlin; (2) Heidelberg University Hospital: Interdisciplinary Cleft Palate and Craniofacial Center. PATIENTS AND METHOD: The cry parameters of five patients with complete unilateral cleft lip, alveolar ridge, and hard and soft palate were analyzed from birth to 9 months of age. The patients were treated with the same protocol. At the age of 24 months, sensomotor development was assessed using the KIPHARD test. Perceptual judgment of speech, performed after 36 months of life, included nasal resonance, nasal emission of air, articulation disorders, and speech intelligibility. MAIN OUTCOME MEASURE: The cry parameters of fundamental frequency (F(0)), pitch period perturbation quotient (PPQ), and cry duration (Tsam) were analyzed. RESULTS: Contrary to the expectation that laryngeal parameters are not affected by vocal tract malformations, differences of cry parameters were found between the patients with UCLP and the non-cleft group. Particularly, the F(0) and its short-time variability (PPQ) were affected. CONCLUSIONS: The preliminary results of this study showed that F(0) and PPQ of spontaneous cries are influenced in patients with UCLP, and a cry analysis might become a noninvasive tool for early detection of an at-risk status for neuromuscular development and prediction of an at-risk status for later speech and language acquisition in infants with cleft lip and palate. Future research strategies are outlined.

Articulation Disorders↗

Chromosome 17: gene mapping studies of cleft lip with or without cleft palate in Chinese families.

OBJECTIVE: Involvement of loci on chromosome 17, including retinoic acid receptor alpha (RARA) in nonsyndromic oral clefts has been reported in Caucasian populations, although never investigated in Asian populations. The purpose of the present study was to investigate several loci on chromosome 17, including RARA, in Chinese families. PARTICIPANTS: Thirty-six multiplex families (310 individuals), ascertained through nonsyndromic cleft lip with or without cleft palate surgical probands from hospitals in Shanghai, China, participated in the present study. There were 23 families whose probands had cleft lip and cleft palate (CLP) and 13 with cleft lip alone (CL). RESULTS: Seventeen markers, spanning chromosome 17 and about 10 cM apart were assessed. Logarithm of odds ratio (LOD) scores (two point and multipoint), model-free linkage analyses, and allelic association tests (transmission/disequilibrium, Fisher's exact tests, and chi-square) were performed on the total family sample, families with CLP probands (CLP subgroup), and families with CL probands (CL subgroup). LOD scores from the two-point analyses were inconclusive. Multipoint analyses rejected linkage except for a few regions in the CL subgroup. However, positive results were found using the model-free linkage and association methods (p < .05). The markers with positive results varied across the CL and CLP subgroups. However, the RARA region and loci nearby yielded consistently positive results. CONCLUSION: Genetic variation within the RARA locus or nearby appears to be involved in the pathogenesis of nonsyndromic oral clefts in this population. Furthermore, based on the differing pattern of results in the CL versus CLP subgroups, it appears that the formation of CL and CLP is because of either differing alleles at the same genetic locus or different but related (and/or linked) genes that modify the severity and expression of oral clefting.

Alleles↗

Cephalometric and occlusal outcome in adults with unilateral cleft lip, palate, and alveolus after two different surgical techniques.

OBJECTIVE: To assess differences in the aesthetic and functional long-term results of one-stage and two-stage surgical and orthodontic treatment in patients with cleft lip, palate, and alveolus. DESIGN: Sixty adult patients who were operated on as children for unilateral cleft lip, palate, and alveolus were examined. In every patient the lip was closed using Tennison's technique. Thirty patients had soft and hard palate closure in two stages and 30 patients in a single stage. Lateral cephalometric and model analyses were conducted at a mean age of 18.4 years. RESULTS: In the model analysis, transverse narrowing was seen in all patients after two-stage operations and in three patients after one-stage operations. The deficit was more severe in the molar region in the two-stage group and nearly similar in the premolar and molar region in the one-stage group. A sagittal deficiency in the anterior maxilla was found in 26 patients after two-stage operations and in 16 patients after one-stage operations. In the lateral cephalometric analysis, the mean sella-nasion-point A angle in the one- and two-stage group was 78.2 degrees and 76.8 degrees, respectively. The ANB angle was normal in both groups. In both groups the inclination of the midface was low. There was a low posterior facial height. Minor scarring was seen in the single-stage group. CONCLUSION: A more severe impairment of growth of the maxilla in the sagittal and frontal plane was observed after two-stage operations on the cleft palate.

Adolescent↗

The effects of active infant orthopedics on occlusal relationships in unilateral complete cleft lip and palate.

OBJECTIVE: To evaluate the effects of active infant orthopedic treatment on dental arch relationships and determine the effect on maxillary growth in children born with unilateral complete cleft lip and palate (UCCLP). DESIGN: The GOSLON Yardstick was used to assess dental models taken on patients treated with and without active infant orthopedics. PATIENTS: Two groups of nonsyndromic Caucasian children born with UCCLP (total n = 40), all treated by the same surgeon and ranging from 5 to 10 years of age, were evaluated. INTERVENTIONS: One group had a Latham dentomaxillary alignment (DMA) appliance inserted at 5 to 6 weeks of age, after which a lip-nasal adhesion was performed at an average age of 3.5 months. This was followed by more definitive nasolabial repair at the average age of 5.9 months. Those patients treated without preoperative orthopedics underwent a lip-nasal adhesion at average age 1.5 months followed by nasolabial repair at average age 5.1 months. MAIN OUTCOME MEASURES: Randomized assessments using the GOSLON Yardstick were done independently at two separate times by three different examiners. Differences in GOSLON scores between the active orthopedic group and nonorthopedic group were evaluated by both categorical and continuous statistical analyses. RESULTS: The mean GOSLON score was 3.30 for the orthopedic group and 3.21 for the nonorthopedic group. There was no significant group difference in the modal scores of the two groups. CONCLUSIONS: This study showed that active infant orthopedics does not affect the dental arch relationships in preadolescent children with repaired UCCLP, compared with a similar group treated without orthopedic intervention at this center.

Activator Appliances↗

Treatment of scars with Er:YAG laser in patients with cleft lip: a preliminary report.

OBJECTIVE: Although one goal in cleft lip surgery is to avoid serious scarring, residual scars are often observed after primary surgery. The goal of this preliminary report was to present the standardized application of laser technology to reduce the appearance of residual scars. DESIGN: Ten patients with scarring after unilateral and bilateral cleft lip surgery were treated with a Dual Mode erbium yttrium-aluminum-garnet laser. This laser was recently introduced and had been used only for cosmetic applications, skin resurfacing, and correction of acne scars. RESULTS: An improvement in the clinical appearance of the laser-treated scars was observed after the first treatment, with continued improvement after the second laser session. The clinically observed improvements were corroborated by the patients' reports concerning satisfaction with the result. CONCLUSIONS: The combined mechanisms of ablation/coagulation and shrinking of the skin caused by the laser, in addition to observed patient satisfaction and low risk associated with the procedure, suggest that this treatment approach can be effective in the correction of residual scarring in patients with cleft lip.

Adult↗

An appraisal of three methods of rating facial deformity in patients with repaired complete unilateral cleft lip and palate.

OBJECTIVES: To evaluate the reliability of clinical assessment, two-dimensional color transparencies and three-dimensional imaging for evaluating the residual facial deformity in patients with repaired complete unilateral cleft lip and palate (UCLP) and compare the ratings of facial deformity made by health care professionals with those made by lay assessors. PATIENTS AND PARTICIPANTS Thirty-one randomly selected subjects aged 10 to 30 years with repaired complete UCLP. Five professionals and five laypersons evaluated each subject's residual cleft-related facial deformity using clinical assessment, two-dimensional color transparencies, and three-dimensional images. MAIN OUTCOME MEASURES: The facial deformity of the full face, lip, nose, and midface were scored using a 5-point ordinal scale on two occasions with a 1-month interval. Intra- and interexaminer agreements were calculated from weighted kappa statistics. Bootstrap permutation tests were used to detect any differences in agreement. RESULTS: Assessment of facial deformity showed good reproducibility across the three assessment media (kappa = 0.42 to 0.83, SE 0.08). Clinical assessment among lay assessors, however, was poor to moderate (kappa = 0.16 to 0.58, SE 0.07). For all assessors, there was no difference in the two nonclinical media relative to the standard clinical assessment for assessments of the full face (p =.377). For assessments of the lip or nose, transparency scores were in greater agreement with the clinical scores than were the three-dimensional assessment scores (p =.017 and.011, respectively). For rating the midface, the three-dimensional scores were in greater agreement with the clinical scores than were the color transparencies scores (p =.047). CONCLUSIONS: In comparison with lay assessors, clinical assessment among professionals was more reproducible. This was not so for nonclinical media. The equivalence of using the color transparencies and three-dimensional media relative to the clinical assessment depends on the region of the face being considered.

Adolescent↗

Genetics of cleft lip and palate revisited.

Cleft lip with or without cleft palate (CL/CP) is one of the most common structural birth defects with treatment including multiple surgeries speech therapy, and dental and orthodontic treatments over the first 18 years of life. Providing care for these patients and families includes educating patients and parents about the genetics of CL/CP, as well as meeting the immediate medical needs. Attempts at identifying susceptibility loci via family and case-control studies have proved inconsistent. It is likely that initial predictions of the complex interactions involved in facial development were underestimated. The candidate gene list for CL/P is getting longer and the need for an impartial systematic screening technique, to implicate or refute the inclusion of particular loci, is apparent. So we are faced with the question "Can this complex trait be too complex?" The aim of this review is to make the dentist aware of the differences between syndromic and non-syndromic cleft as well as understanding the etiological variation in cleft lip with and without cleft palate. This will aid the dentist in diagnosis and give proper genetic counseling to parents and patients of cleft lip and palate.

Adolescent↗

The use of overdentures in children with cleft lip and palate: a report of two cases.

The treatment of children with cleft lip and palate is complex. Advances in primary surgery and the advent of alveolar bone grafting have resulted in the reduction of prosthetic intervention. However, in patients where surgery is contraindicated or has been unsuccessful, an alternative treatment is required. Overdentures are a simple, conservative and reversible non-surgical alternative for children with cleft lip and palate. We present two cases with severe complete bilateral cleft lip and palate that were managed in this way.

Anodontia↗

A study of patients with Angle Class I unilateral cleft lip and palate (UCLP) using lateral roentgenographic cephalograms. A comparison between anterior cross-bite UCLP patients and anterior cross-bite non-UCLP, normal bite UCLP or normal bite non-UCLP patients.

In our previous study carried out in 1994, we found 49 patients with unilateral cleft lip and palate (UCLP) among 51 patients with cleft lip and palate who visited the Department of Orthodontics, Nihon University Dental Hospital at Matsudo. In particular, patients with cross-bite (anterior cross-bite) UCLP were predominant, accounting for 21 individuals (41%). In the present study, we reviewed 79 patients with malocclusion including 21 with anterior cross-bite UCLP, who represent the largest subpopulation among patients with cleft lip and palate. These 21 patients were compared with other UCLP patients showing normal bite with regard to morphological differences evident in lateral roentgenographic cephalograms, to establish a proper policy of orthodontic diagnosis and treatment of their condition. The anterior cross-bite UCLP group showed differences in the skeletal pattern and the denture to skeletal pattern from the other patient groups except for the mesio-distal relationship. The anterior cross-bite UCLP group had cross-bite of the concave type, while the normal bite UCLP group had malocclusion of the convex type accompanied by maxillary overdevelopment. Thus, it is suggested that normalization of reversed occlusion leads to favorable growth of the upper jaw in UCLP patients with anterior cross-bite.

Adolescent↗

Congenital unilateral lower lip palsy.

Thirty-nine cases of congenital facial asymmetry, in which one corner of the mouth does not dip downward symmetrically with the other, are presented. The author has termed this condition 'congenital unilateral lower lip palsy' (CULLP). CULLP was the most frequent condition in congenital facial anomalies seen in our clinic. Complaints were purely cosmetic. No functional disorders in pronouncing labial sounds or in suckling were noticed. As for pathogenesis, an insufficiency of the unilateral depressor labii inferior muscle is the most likely cause. An important point is that the lower lip, when drawn down, is normal, whereas the contralateral lip is paralysed. As far as facial asymmetry is concerned, CULLP is a minor deformity. Nevertheless, possible association with other anomalies must be seriously considered when examining patients, especially neonates. As for treatment, selective facial nerve blocking is successfully done in our clinic.

Abnormalities, Multiple↗

Tumor differentiation and tumor-host interactions as prognostic determinants in squamous cell carcinoma of the lip.

A series of 70 patients with the squamous cell carcinoma of the lip and followed-up for at least 5 years, was assessed on light microscopy and using histochemical staining for ANAE (acid-naphthyl acetate esterase) to demonstrate the morphological manifestations of tumor-host reactivity. The factors analysed include cancer differentiation (intrinsic malignancy) and stromal reactions (intensity of the immunocompetent cell infiltrate including the mast cells and the subpopulations, i.e. B- or T lymphocytes or mononuclear phagocytes). Differentiation of the lip cancer was shown to be directly (although not statistically significantly) related to the 5-year survival, as was also the intensity of the stromal immunocompetent cell infiltration. Cancer metastases were evidently the most powerful prognostic determinants, their development being influenced both by the intensity of the stromal immunocompetent cell infiltrate and cancer differentiation. B lymphocytes far outnumbered the T and MPS cells in all the infiltrates studied, the percentages of the latter two cell types, however, being inversely related to the intensity of the infiltrate. The cell composition in the infiltrates was seemingly without effect on the frequency of metastases and the 5-year survival, as was the stromal mast cell reaction, too. It was concluded that analysis of tumor-host relationships using a variety of morphological and immunohistochemical techniques may be of benefit in predicting the clinical course of lip cancer.

Adult↗

A comparative study of gonial angle and growth of the lower third molar in cleft lip and palate. An orthopantomographical study.

In children and young adults with cleft lip and palate, the gonial angle, the angle between the lower second molar and the mandibular plane, the angle between the longitudinal axes of the second and third molars as well as the timing of the development of the third molar were investigated on the right and left side from orthopantomograms. The results were compared with the corresponding results in a noncleft Finnish material. The series consisted of 100 randomly selected subjects aged from 10 to 24 years, 57 males and 43 females. One to three orthopantomograms were available for each subject, totalling 201. The results were examined in a cross-sectional and longitudinal material. The right and left gonial angles were not found to differ in the different cleft groups. Among subjects with cleft lip and palate the gonial angle was found to be larger in the different age groups than the corresponding values in the comparison material. In both materials, the gonial angle was found to decrease between the ages of 10 and 15 years. In males with cleft lip and palate the decrease in this angle, however, continued up to the age of 19. The anterior angle between the longitudinal axis of the lower second molar and the mandibular base line was about 10 degrees larger than in the comparison material, being slightly larger than that in the Angle's Class III occlusion type in the comparison material. When examined by degrees of development the development of the lower third molar was delayed approx. 0.9 years in males and 1.6 years in females compared with the corresponding times in the comparison material.

Adolescent↗

The embryological, developmental and functional importance in the repair of the nasal musculature to reduce the deformity of the cleft lip nose.

The latent ability of nasal growth to correct developmental deformities when caused by a deforming force has been shown in nasal dermoids where early surgery by removing the deforming elements allows subsequent growth of the nasal bones and cartilages to correct the abnormality. An embryological study into the nasal musculature has shown both an orbicularis and dilator system to be present. The correction of the abnormal musculature in the cleft lip nose by removing the deforming connections and realigning the muscles allows subsequent growth the possibility of producing normal development. Primary surgery in the cleft lip nose has not shown any detrimental growth patterns and correction of the nasal musculature should therefore be carried out at the time of the primary lip correction.

Cleft Lip↗