Long-term follow-up of mandibular dental arch changes in patients with complete non-syndromic unilateral cleft lip, alveolus, and palate.

BACKGROUND: Treatment of cleft lip and palate (CLP) requires a comprehensive interdisciplinary approach and long-term follow-up. Only a few studies are available that reported on changes after treatment, which showed that in particular the transverse dimension, in patients with CLP is prone to changes after treatment. However, those studies did not pay attention to concomitant changes in the mandibular arch that occur after treatment. OBJECTIVES: To evaluate mandibular transverse dental arch dimensions and interarch transverse changes in patients with complete non-syndromic unilateral cleft lip, alveolus, and palate (CUCLAP) up to five years after treatment. MATERIAL AND METHODS: Retrospective longitudinal study in 75 consecutive patients with CUCLAP directly after comprehensive treatment (T0), two (T2), and 5 years after treatment (T5). Great Ormond Street, London and Oslo (GOSLON) scores were available for all patients. Three-dimensional scans of all dental casts were made. Inter premolar and intermolar distances between the mandibular contralateral teeth were measured. The modified Huddart Bodenham (MHB index) was applied to assess the transverse interarch relationship. Paired t-tests and ANOVA were used to analyze transverse and interarch transverse changes. Linear regression analysis was done to define contributing factors. RESULTS: Paired t-tests showed a significant decrease of the mandibular inter first and second premolar distances (p < 0.05) and an increase of the inter second molar distance, whilst the MHB Index deteriorated at all time points for all segments and for the total arch score (p < 0.05). Linear regression showed no significant contributing factors on the decrease of the transverse distances. However, inter arch transverse relationship was significantly affected by age at the end of treatment, missing maxillary lateral incisor space closure, and the GOSLON Yardstick score at the end of treatment (p < 0.05), especially during the first two years after treatment. CONCLUSIONS: Changes occurred in the mandibular arch expressed as changes in the transverse dimensions and interarch relationship measured by the MHB Index. A younger age at the end of treatment, space closure for a missing maxillary lateral incisor and a higher GOSLON score at the end of treatment negatively influence the interarch transverse deterioration especially in the first two years after treatment. For the transverse dimensional changes in the mandibular arch such influencing factors could not be determined.

Pattern of Morphological Variability in Unrepaired Unilateral Clefts With and Without Cleft Palate May Suggest Intrinsic Growth Deficiency.

In individuals with cleft lip and palate (CLP) an iatrogenic effect of operations on subsequent maxillary growth is well-known. Much less is known about the association between occurrence of CLP and intrinsic growth deficiency of the maxillofacial complex. The aim of this study was to compare morphological variability in subjects with unilateral cleft lip and alveolus/palate and unaffected controls using geometric morphometric methods. The research hypothesis was that if subjects with unrepaired unilateral CLP have intrinsic growth deficiency, the pattern of their craniofacial growth variation may differ from that in unaffected individuals. Lateral cephalograms were available of three groups of the same ethnic background (Proto-Malayid): (a) non-syndromic unrepaired unilateral complete cleft lip, alveolus, and palate (UCLP), N = 66, mean age 24.5 years (b) non-syndromic unrepaired unilateral complete cleft lip and alveolus (UCLA), N = 177, mean age 23.7 years, and (c) NORM (N = 50), mean age 21.2 years without a cleft. Using geometric morphometrics shape variability in groups and shape differences between groups was analyzed. Principal component analysis (PCA) was used to examine shape variability, while differences between groups and sexes were evaluated with canonical variate analysis. Sexual dimorphism was evaluated with discriminant function analysis (DA). Results showed that in comparison to NORM subjects, shape variability in UCLA and UCLP is more pronounced in the antero-posterior than in vertical direction. Pairwise comparisons of the mean shape configurations (NORM vs. UCLA, NORM vs. UCLP, and UCLA vs. UCLP) revealed significant differences between cleft and non-cleft subjects. The first canonical variate (CV1, 68.2% of variance) demonstrated that differences were associated with maxillary shape and/or position and incisor inclination, while in females, the CV1 (69.2% of variance) showed a combination of differences of "maxillary shape and/or position and incisor inclination" and inclination of the cranial base. Shape variability demonstrated considerable differences in subjects with UCLA, UCLP, and NORM. Moreover, in subjects with a cleft, within-sample variability was more pronounced in the antero-posterior direction, while in non-cleft subjects, within-sample variability was more pronounced in the vertical direction. These findings may suggest that subjects with unilateral clefts have intrinsic growth impairment affecting subsequent facial development.

Morphological variability in unrepaired bilateral clefts with and without cleft palate evaluated with geometric morphometrics.

In subjects with orofacial clefts, there is an unresolved controversy on the effect of congenital maxillary growth deficiency vs. the effect of surgical intervention on the outcome of treatment. Intrinsic growth impairment in subjects with orofacial clefts can be studied by comparing facial morphology of subjects with untreated cleft and unaffected individuals of the same ethnic background. Bilateral cleft lip and palate is the most severe and least prevalent form of the orofacial cleft. The aim of this study was to compare facial morphology in subjects with unrepaired complete bilateral clefts and unaffected controls using geometric morphometrics. Lateral cephalograms of 39 Indonesian subjects with unrepaired bilateral complete cleft lip and alveolus (mean age: 24 years), or unrepaired bilateral complete cleft lip, alveolus, and palate (mean age: 20.6 years) and 50 age and ethnically matched controls without a cleft (25 males, 25 females, mean age: 21.2 years) were digitized and traced and shape variability was explored using principal component analysis, while differences between groups and genders were evaluated with canonical variate analysis. Individuals with clefts had a more pronounced premaxilla than controls. Principal component analysis showed that facial variation in subjects with clefts occurred in the anteroposterior direction, whereas in controls it was mostly in the vertical direction. Regression analysis with group, sex, and age as covariates and principal components from 1 to 6 as dependent variables demonstrated a very limited effect of the covariates on the facial shape variability (only 11.6% of the variability was explained by the model). Differences between cleft and non-cleft subjects in the direction of facial variability suggest that individuals with bilateral clefts can have an intrinsic growth impairment affecting facial morphology later in life.

Subphenotyping and Classification of Cleft Lip and Alveolus in Adult Unoperated Patients: A New Embryological Approach.

OBJECTIVE: Previously, a new embryological classification was introduced subdividing oral clefts into fusion and/or differentiation defects. This subdivision was used to classify all subphenotypes of cleft lip with or without alveolus (CL±A). Subsequently, it was investigated whether further morphological grading of incomplete CLs is clinically relevant, and which alveolar part is deficient in fusion/differentiation defects. DESIGN: Observational cohort study. SETTING: Three hundred fifty adult unoperated Indonesian cleft patients presented themselves for operation. Cephalograms, dental casts, and intraoral and extraoral photographs-eligible for the present study-were used to determine morphological severity of CL±A. PATIENTS: Patients with unilateral or bilateral clefts of the primary palate only were included. MAIN OUTCOME MEASURES: Clefts were classified-according to developmental mechanisms and timing in embryogenesis-as fusion and/or differentiation defects. Grades of incomplete CLs were related to the severity of alveolar clefts (CAs) and hypoplasia, and permanent dentition was used to investigate which alveolar part is deficient in fusion/differentiation defects. RESULTS: One hundred eight adult patients were included. All subphenotypes-96 unilateral and 12 bilateral clefts-could be classified into differentiation (79%), fusion (17%), fusion-differentiation (2%), or fusion and differentiation (2%) defects. The various grades of incomplete CLs were related to associated CAs and hypoplasia, and all alveolar deformities were located in the premaxillae. CONCLUSIONS: This study showed that all CL±A including the Simonart bands can be classified, that further morphological grading of incomplete CLs is clinically relevant, and that the premaxilla forms the deficient part in alveolar deformities.

Width and elevation of the palatal shelves in unoperated unilateral and bilateral cleft lip and palate patients in the permanent dentition.

Patients with cleft left lip and palate (CLP) normally require extensive surgery from an early age up to the end of adolescence. These surgeries affect the growth of the maxillofacial complex. The degree to which the cleft itself affects growth of the maxillofacial complex remains poorly understood. By analysing the width and elevation of the palatal shelves in unoperated adolescents and adults with unilateral and bilateral cleft lip and palate (UCLP and BCLP, respectively) and a non-cleft control group, it is possible to gain more insight into the real intrinsic growth potential of the maxillary structures. In this study, dental casts of the full permanent dentition of individuals with unrepaired UCLP (n = 68) and BCLP (n = 13) and non-cleft controls (n = 24) from the same area of Indonesia were digitized three-dimensionally. Maxillary arch width in the canine, premolar and molar regions, and the width and elevation of the palatal shelves were measured. Results showed that in patients with UCLP, the width of the palatal shelves on the cleft side in all regions, and on the non-cleft side in the canine/first premolar region, was significantly smaller compared with the control group. BCLP subjects showed similar deviations. In the UCLP group, the palatal shelves were rotated cranially and positioned more vertically. In the BCLP group, the palatal shelves were inclined by almost 10 ° more than the control group. The width of the palatal shelf and width of the maxillary arch positively correlated in the canine and first premolar regions for both the cleft and non-cleft side in patients with UCLP, and in the canine region for patients with BCLP. This means that the wider the palatal shelf, the wider the maxillary arch. The elevation of palatal shelves correlated with the maxillary arch width in all regions in patients with UCLP, and only in the premolar region in the control group. Thus, the wider the arch width, the smaller the elevation angle (the maxillary shelves are less vertical). No correlations between palatal shelf elevation and maxillary arch width were found in the BCLP group. This shows that the intrinsic growth potential in patients with UCLP and BCLP is affected by a smaller palatal shelf width and larger elevation of the shelves. These deviations may result in a wider cleft.

Maxillary arch width in unoperated adult of the unilateral cleft lip and alveolus patients.

BACKGROUND: The transversal measurement on the dento-alveolar fragments of the maxilla, in the unoperated adult patients with unilateral cleft lip and alveolus are rare and have not been well documented and archived in the scientific journals. The effect of the congenital malformation is obvious in the Unilateral Cleft Lip and Alveolus (UCLA) patients and is well documented. OBJECTIVES: The aim of this study is to investigate whether there are any influences of the final transversal development of the maxilla based on the location and extent of the defect in patients with UCLA. METHODS: 168 adult unoperated UCLA patients (study group) and 24 non cleft patients (control group) participated in this study. Plaster of Paris (POP) casts were made of the maxillary dentition and mesurement were taken using coordinate measuring machine (CCM) (Zeiss Numere, Carl Zeiss, Stuttgart, Germany). RESULT: The mean values of maxillary transverse measurement of first premolar through the second molar of the UCLA and control group patients showed no statistically significant difference. The inter-canine distance was significantly smaller in the UCLA patients than non-cleft patients CONCLUSIONS: The maxillary inter-canine distance in untreated UCLA patients were significantly different from that of non cleft patients. There is narrow and constricted maxillary growth in UCLA patients causing maxillary morphological changes and cosmetic facial changes.