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Prevalence of palatal and alveolar cysts in babies with cleft lip and palate.

OBJECTIVE: To evaluate the prevalence of palatal and alveolar cysts in babies with cleft lip and/or palate. DESIGN: Cross-sectional. SETTING: Hospital for Rehabilitation of Craniofacial Anomalies, University of São Paulo (HRAC-USP), Bauru, São Paulo, Brazil. PARTICIPANTS: Two hundred ninety-one Caucasian babies divided into four groups according to the type of cleft: cleft lip with or without cleft alveolus (70), complete unilateral cleft lip and palate (112), complete bilateral cleft lip and palate (56), and cleft palate (53). RESULTS: A low prevalence of palatal and alveolar cysts was observed among patients with the four different types of clefts, with no statistically significant difference between genders. The maxilla and the anterior area of the mouth were more affected than the mandible and the posterior area. CONCLUSIONS: The low prevalence of palatal and alveolar cysts in the four groups of babies with clefts included in this study may have been due to the high mean age of the sample.

Alveolar Process↗

Mandibular morphology in complete unilateral cleft lip and palate.

OBJECTIVE: The purpose of this study was to evaluate and compare mandibular morphology and spatial position in children with complete unilateral cleft lip and palate (UCLP) treated at two different cleft centers (Hannover and Brussels) following different surgical treatment protocols. PATIENTS: A total of 62 Caucasian children (40 boys, 22 girls) with nonsyndromic complete unilateral cleft lip and palate (UCLP) were evaluated by means of conventional cephalometric analysis at approximately the age of 10 years. Data of both cleft groups were compared with a control, noncleft group (n = 40) matched according to age and sex. INTERVENTIONS: The Hannover children with cleft (n = 36) underwent lip repair at a mean age of 5.83 +/- 1.16 months. The hard and soft palates were closed at a mean age of 29.08 +/- 4.68 and 32.25 +/- 4.29 months, respectively. The Brussels children with cleft (n = 26) were treated according to the Malek surgical protocol with soft palate repair at a mean age of 3.04 +/- 0.20 months and simultaneous lip and hard palate repair at a mean age of 6.15 +/- 0.68 months. RESULTS: Statistical analysis (analysis of variance with post hoc Tukey's test) showed a significant (p =.001) smaller mandibular ramus length (Co-Go) in the Brussels cleft group, compared with the control group. The Hannover-Brussels comparison data revealed that the S-N-B angle was significantly (p =.047) less in the Brussels cleft group. CONCLUSIONS: The influence of surgical procedures in patients with UCLP might not be restricted to the maxilla but could influence mandibular spatial position to the cranial base. Because of these positional changes of the mandible, both cleft groups showed facial balance.

Analysis of Variance↗

The prevalence of caries in children with a cleft lip and/or palate in Southern Vietnam.

OBJECTIVE: The aim of this study was to investigate the prevalence of caries in South Vietnamese children with a known oral cleft. SETTING: The children were screened at local dental hospitals in five different cities in southern Vietnam. PARTICIPANTS: One hundred fifty-four children aged 4 to 6, 11 to 13, and 14 to 16 years old with a cleft lip, cleft palate, or both were included in the study. OUTCOME MEASUREMENTS: For every age group, clefts were divided in four types. The dmft/DMFT was specified according to criteria of the World Health Organization (1997). RESULTS: The mean number of teeth affected by caries was 9.95 for 4- to 6-year-old children, 2.97 for 11- to 13-year-old children, and 4.93 for 14- to 16-year-old children, respectively. Four- to 6-year-old children with a unilateral or bilateral cleft lip and palate had significantly more caries and a higher dmft (decayed, missing, and filled teeth index) than children of the same age with only a cleft lip or a cleft lip and alveolus. CONCLUSIONS: Vietnamese children with an oral cleft have high numbers of teeth affected by dental caries, and special attention will be necessary for their oral health.

Adolescent↗

Early craniofacial morphology and growth in children with bilateral complete cleft lip and palate.

PURPOSE: Analysis of craniofacial morphology and growth in children with bilateral complete cleft lip and palate (BCCLP), compared with a control group with unilateral incomplete cleft lip (UICL), before any treatment as well as 20 months after lip closure. MATERIAL: The children were drawn from a group representing all Danish children with cleft born 1976 to 1981. Sixty-four children were included in the study (19 BCCLP and 45 UICL). The ages were 2 and 22 months at examinations 1 and 2, respectively. METHOD: The method of investigation was infant cephalometry in three projections. The craniofacial morphology was analyzed using linear, angular, and area variables. Growth was defined as the displacement vector from the coordinate of the corresponding landmark in the x-ray at examination 1 to its coordinate at examination 2, corrected for x-ray magnification. The growth of an anatomical region in a patient was assessed by investigating the growth pattern formed by a collection of individual growth vectors in that region. RESULTS: The BCCLP group differed significantly from the UICL group. The most striking findings in BCCLP were an extremely protruding premaxilla; markedly increased posterior maxillary width; increased width of the nasal cavity; short maxilla with reduced posterior height; short mandible; bimaxillary retrognathia; severe reduction in the size of the pharyngeal airway; and a more vertical facial growth pattern. CONCLUSION: Our findings indicate that a facial type including a wide and posterior short maxilla, short mandible, and bimaxillary retrognathia might be a liability factor that increases the probability of developing cleft lip and palate.

Case-Control Studies↗

Approaches of South African traditional healers regarding the treatment of cleft lip and palate.

OBJECTIVE: Due to the large number of Black South Africans known to consult with traditional healers and the influential role they play in the developing world, this study investigated the approaches of a group of 15 traditional healers toward the treatment of cleft lip and palate. DESIGN: An exploratory-descriptive, qualitative research design was employed, which involved the use of standardized, semistructured interviews, conducted with the assistance of an interpreter who was fluent in several African languages. SETTING: Interviews were conducted outdoors in places in which traditional healers usually consult with their patients. These areas were in the fields close to the traditional healers' homes or under a highway bridge in the Johannesburg inner city. PARTICIPANTS: Individual interviews were conducted with 15 African traditional healers who had been practicing in their callings for at least 1 year. RESULTS: Traditional healers interviewed had treated one to six persons with a cleft lip, cleft palate, or both. Most informants believed that clefts were caused by the ancestors, spirits, and witchcraft. A variety of plant and animal products were used to treat these conditions and were augmented by spiritual resources from the ancestors. All except one healer had undergone formal training, although they had received no specific training relative to cleft lip and palate. Most participants reported referring patients to Western health care practitioners who were referred to as modern doctors but did not receive reciprocal referrals from these professionals. Patients generally consulted with traditional healers because this approach was part of their culture. CONCLUSIONS: These findings have important implications for health care professionals as well as traditional healers in terms of cross-cultural consultation, collaboration, and information sharing with regard to cleft lip and palate; the potential use of traditional healers in primary health care and education; and further research.

Adult↗

Oral disease status of a sample of Jordanian people ages 10 to 28 with cleft lip and palate.

OBJECTIVE: To determine the levels of periodontal disease and dental caries in subjects with cleft lip and palate and to compare them with matched noncleft control subjects. DESIGN: A total of 32 subjects with cleft lip and palate, ages 10 to 28 years, and a similar number of noncleft control subjects were examined for plaque biofilm deposits, gingivitis, periodontitis, and dental caries by using the Silness and Löe plaque index (PI), Löe and Silness gingival index (GI), probing pocket depth (PPD), and the decayed-missing-filled-teeth (DMFT) index, respectively. SETTING: Faculty of Dentistry, Jordan University of Science and Technology, and Prince Rashed Hospital, Royal Medical Services, northern Jordan. RESULTS: Scores for PI, GI, PPD, and DMFT were significantly higher in subjects with cleft lip and palate than in control subjects. CONCLUSION: Subjects with cleft lip and palate are at an increased risk for dental caries and periodontal disease when compared with a noncleft population.

Adolescent↗

Contributions of PTCH gene variants to isolated cleft lip and palate.

OBJECTIVE: Mutations in patched (PTCH) cause the nevoid basal cell carcinoma syndrome (NBCCS), or Gorlin syndrome. Nevoid basal cell carcinoma syndrome may present with developmental anomalies, including rib and craniofacial abnormalities, and predisposes to several tumor types, including basal cell carcinoma and medulloblastoma. Cleft palate is found in 4% of individuals with nevoid basal cell carcinoma syndrome. Because there might be specific sequence alterations in PTCH that limit expression to orofacial clefting, a genetic study of PTCH was undertaken in cases with cleft lip and/or palate (CL/P) known not to have nevoid basal cell carcinoma syndrome. RESULTS: Seven new normal variants spread along the entire gene and three missense mutations were found among cases with cleft lip and/or palate. One of these variants (P295S) was not found in any of 1188 control samples. A second variant was found in a case and also in 1 of 1119 controls. The third missense (S827G) was found in 5 of 1369 cases and in 5 of 1104 controls and is likely a rare normal variant. Linkage and linkage desequilibrium also was assessed using normal variants in and adjacent to the PTCH gene in 220 families (1776 individuals), each with two or more individuals with isolated clefting. Although no statistically significant evidence of linkage (multipoint HLOD peak = 2.36) was uncovered, there was borderline evidence of significant transmission distortion for one haplotype of two single nucleotide polymorphisms located within the PTCH gene (p = .08). CONCLUSION: Missense mutations in PTCH may be rare causes of isolated cleft lip and/or palate. An as yet unidentified variant near PTCH may act as a modifier of cleft lip and/or palate.

Adenine↗

Phonetic description of babbling in Danish toddlers born with and without unilateral cleft lip and palate.

OBJECTIVE: To investigate prelinguistic vocalization sequences of 1-year-old children with and without cleft lip and palate. DESIGN: Prospective study. PARTICIPANTS: Thirty-eight children born with unilateral cleft lip and palate and 36 control children born without clefts. The cleft children had the lip, soft palate, and posterior part of the hard palate repaired at 4 months of age. The lip was closed ad modum Millard, the nose was corrected according to McComb, and the soft palate was closed with a posteriorly based vomer flap. METHODS: Data were obtained from a clinical visit during which the baby played with the mother. Video recordings were transcribed and analyzed concerning (1) the frequency of occurrence of vocalization sequences, (2) the frequency of occurrence of contoids and vocoids, (3) the contoids' place and manner of articulation, and (4) the percentage of children who entered the canonical babbling stage. RESULTS: No significant differences were observed between the cleft and control groups concerning frequency of occurrence of vocalization sequences, contoids, or vocoids. Structural differences between the groups seem to influence the contoid inventory, with a higher frequency of occurrence of nasal contoids and a smaller frequency of occurrence of alveolar contoids in the cleft group. Canonical babbling was achieved by most children in both groups, and no significant difference was found between the groups. CONCLUSIONS: Early closure of the soft palate seems to have a positive influence on the prelinguistic development of children with cleft palate.

Cleft Lip↗

Congenital isolated midline sinus of the upper lip: clinical and embryological approaches.

OBJECTIVES: Congenital sinuses or fistulas of the lip are uncommon malformations, yet true medial upper-lip fistulas (MULFs) are extremely rare. We present a new case of congenital upper-lip fistula located in the midline of the philtrum of an 8-month-old girl. INTERVENTION: Complete surgical removal was performed with a combined extra- and intraoral approach. Histological examination revealed that the fistula was lined by squamous epithelium with sebaceous and mucous glands and hair follicles. RESULTS: Several embryological hypotheses have been proposed concerning these anomalies. This article reexamines and discusses major embryological theories on pathogenesis of sinuses or fistulas of the upper lip. We propose that early ectodermal inclusion events may occur in the medial fusion area during formation of the intermaxillary process. This embryological approach is highly concordant with our recent hypothesis on nasal dermoid sinus cysts (NDSCs) pathogenesis, in which we proposed an embryological hypothesis with early ectodermal inclusion phenomenon in the midline suture area to explain NDSCs pathogenesis. CONCLUSIONS: Common early ectodermal inclusion phenomena could be involved in both NDSCs and MULFs pathogenesis.

Cutaneous Fistula↗

Presurgical nasoalveolar molding therapy for the treatment of bilateral cleft lip and palate: A preliminary study.

OBJECTIVE: To evaluate the outcome of presurgical nasoalveolar molding therapy in the treatment of patients with bilateral cleft lip and palate. DESIGN: A prospective study with blinded measurements. SETTING: The Cleft and Craniofacial Clinic at the University of Texas at Houston Medical School, Houston, Texas. PATIENTS: Eight patients with bilateral cleft lip and palate, treated between 2002 and 2004. INTERVENTIONS: The starting age for presurgical nasoalveolar molding therapy was 34.9 days and the average length of the therapy was 212.5 days. MAIN OUTCOME MEASURES: Measurements of intraoral and extraoral casts were performed, and statistical analyses were used to compare the differences between measurements before and after therapy. RESULTS: Intraoral measurements demonstrated that there was a statistically significant reduction of the premaxillary protrusion and deviation. There was also a significant reduction in the width of the larger cleft. Extraoral measurements revealed that there was a significant increase in the bi-alar width and in the columellar length and width. Moreover, there was a significant improvement in columellar deviation. Finally, the nostril heights of both sides were increased. CONCLUSION: The authors have quantitatively shown that presurgical nasoalveolar molding therapy has significant advantages in the treatment of bilateral cleft lip and palate patients. It improves the nasal asymmetry and deficient nasal tip projection associated with bilateral cleft lip and palate. It also forces the protruded premaxillary segment into alignment with the dental alveolar segments, improving the shape of the maxillary arch. As a result, the changes associated with presurgical nasoalveolar molding therapy help decrease the complexity of subsequent surgeries.

Acrylic Resins↗

Data-objective diagnosis of infant cleft lip, alveolus, and palate. Morphologic data guiding understanding and treatment concepts.

Maxillary plaster models of untreated infants with various cleft lip and palate forms have been studied under the Reflex Microscope. From the three- dimensional data processed by a PC, the specific dimensions of various cleft forms can be determined. Unexpected findings are that skeletal maxillary changes are present in infant velar clefts. The mid-sagittal maxillary length in particular becomes shorter with more severe clefting of the bony palate. From the morphologic point of view, Robin's sequence does not represent a specific entity, but usually a severe cleft of the hard palate. Unilateral clefts of lip, alveolus, and palate with a partial cleft lip reveal worse malformations than complete unilateral clefts. The morphologic changes recorded can be explained by a varying degree of lingual malposition. This reveals that displacements and malformations of infant cleft lip and palate are in fact secondary in nature. In correcting these secondary surface manifestations as soon as indicated by physiologic criteria one could hope for early normalization of sensation, perception, and function. Three-dimensional high-precision measurements are crucial for data-objective diagnoses, timing of therapy, choice of treatment, and in later evaluation of the results.

Alveolar Process↗

Cleft lip and palate in Tehran.

Seventy-nine cleft lip and/or palate births were isolated from 21,138 live births between January 1, 1983 and December 31, 1988 in one hospital in Tehran. Among these, 21 (26.58 percent) were cleft lip (CL), 45 (56.96 percent) were cleft lip and palate (CLP), and 13(16.45 percent) were cleft palate (CP). Chemical sulfur mustard gas was indicated as a major factor in 30 (37.97 percent) of the bilateral cleft lip and palate infants.

Cleft Lip↗

Evaluation of maxillary dental arch form in unilateral clefts of lip, alveolus, and palate from one month to four years.

This study evaluated the early changes of maxillary alveolar arches of operated unilateral cleft lip and palate patients. Dental casts were available at four age increments. Triangular flap cheiloplasty was carried out at an early age. Two-stage palatoplasty by vomer flap and soft palate closure took place later. Prior to lip repair, the alveolar arches were classified according to the relationship between greater and lesser segments. Almost a quarter had overlap of the alveolar segments with no contact between the alveolar ridges at the cleft site; some had no overlap with contact of the alveolar segments in the cleft region; almost a quarter had both overlap of the alveolar segments and contact; and almost half had no overlap of the segments and the alveolar ridges were not in contact at the cleft site. After lip repair, the arch relationships were examined and the percentage of patients in each of the four groups indicated a moulding effect of lip repair on the alveolar segments. This moulding effect caused the alveolar segments in most patients to be in contact at the cleft site. Most of these also had segment overlap. All patients were re-examined shortly after palatal repair. The trend for segment overlap and contact continued after palate surgery. However, when all patients were seen at age 4, percentages of patients in each group indicated that previous overlap of segments improved to a more desirable nonoverlapped relationship in approximately half of the patients. The other half continued to demonstrate arch collapse, in excess of what would be considered ideal ridge relationship.

Child, Preschool↗

Spheno-occipital synchondrosis in three-month-old children with clefts of the lip and palate: a radiographic study.

The literature has been focused on differences in craniofacial form and dimension in cleft lip and palate patients. No attention has been given to possible alterations in the spheno-occipital synchondrosis although the synchondrosis is regarded as an important maturity and growth center. The purpose of the present investigation was to examine if the spheno-occipital synchondrosis in children born with major complete clefts differs on lateral radiographs from that of children born with minor incomplete clefts. Fifty-seven three-month-old children with complete clefts of the lip, alveolus and palate and 42 three-month-old children with minor incomplete clefts of the lip were included in the study. The results of the comparison showed that children with complete major clefts had a broader spheno-occipital synchondrosis compared to children with a minor incomplete cleft of the lip, and that the distance from the superior part of the synchondrosis to the sella point was shorter in children with complete clefts. These findings could be related to a defect or a delay in maturity in the early development of the cartilaginous cranial base in children with major clefts.

Alveolar Process↗

P-A cephalometric analysis of nonoperated adult cleft lip and palate.

P-A cephalometric analysis was performed on the craniofacial morphology in 88 Brazilian men with nonoperated and operated cleft lip and palate. For the comparative study, these subjects were divided into the following four groups: (1) 31 nonoperated unilateral cleft lip and palate (UCLP), (2) 24 nonoperated bilateral cleft lip and palate (BCLP), (3) 16 operated UCLP, (4) 17 operated BCLP. Thirty Brazilian men without cleft lip and palate were used as control subjects. In comparison with the control subjects, nonoperated BCLP and UCLP showed remarkable facial deformity characterized by increased width of various facial parts. Facial morphology of surgically treated BCLP and UCLP, however, was very similar to that of noncleft subjects, apart from the immediate cleft region. There was no remarkable difference in the facial morphology between nonoperated BCLP and UCLP, except for the cleft width and the deviation of nasal septum base, while the only significant difference between operated BCLP and UCLP was in the cleft width.

Adolescent↗

Comparison of velopharyngeal growth patterns between cleft lip and/or palate patients requiring or not requiring pharyngeal flap surgery.

This investigation compares the patterns of velopharyngeal growth in cleft lip and/or palate patients. Those who had velopharyngeal competence and acceptable speech are compared with those who presented with velopharyngeal incompetence requiring pharyngeal flap surgery or prosthesis later. Lateral cephalograms of 30 cleft palate only (CPO), 35 unilateral cleft lip and palate (UCLP), and 20 bilateral cleft lip and palate (BCLP) children of the Lancaster Cleft Palate Clinic were studied. These records were taken at 6 month intervals during the first 2 postnatal years and annually thereafter up to 6 years of age. Soft tissue landmark points in the velopharyngeal region were digitized. Length and thickness of the soft palate and height and depth of the nasopharynx were measured. Evaluation of the growth curves of these four cephalometric variables indicated only two significant differences between children who later required pharyngeal flap surgery and those who did not. These differences were found in the growth in length of the soft palate of the CPO group and in the growth in depth of the nasopharynx of the BCLP group. Based on the present cephalometric data, it is impossible to predict at an early age those cleft lip and/or palate patients who will later require pharyngeal flaps.

Cephalometry↗

Microform cleft lip associated with a complete cleft palate.

Clefting of the lip with or without an associated cleft palate may be present in varying degrees of severity. The so-called microform cleft lip or forme fruste has been characteristically described as having the appearance of a repaired cleft lip. The following case describes a patient with microform cleft lip and a complete cleft of the hard and soft palates. To the best of our knowledge, this is the first report of such an association.

Cleft Lip↗

Longitudinal study on three-dimensional changes of facial asymmetry in children between 4 to 12 years of age with unilateral cleft lip and palate.

The purpose of the present study was to describe three-dimensional developmental changes of facial asymmetry in children with an operated complete unilateral cleft lip and palate (UCLP) and in children without craniofacial anomalies (controls). Using stereophotogrammetry, three-dimensional coordinates for 16 bilateral and 10 midsagittal facial landmarks were determined for the UCLP group (n = 33) and the control group (n = 63) on two occasions. In this mixed-longitudinal study, the children were 4 to 12 years of age. Facial asymmetry and left-right dominance was measured and resolved for transverse, vertical, and sagittal components. Significant effects were analyzed with multivariate analyses of variance (MANOVA). We concluded that individuals with complete unilateral cleft lip and palate, as well as individuals without craniofacial anomalies, show an increase, during growth, in the amount of facial asymmetry in the basal region of the nose. In the region that is related to the cleft, children with complete unilateral cleft lip and palate do not show changes in the amount of facial asymmetry between the occasions. Regarding facial left-right dominance and variation in dominance, no demonstrable growth changes take place in individuals with complete cleft lip and palate, nor in individuals without craniofacial anomalies.

Case-Control Studies↗