Objective:

(1) To investigate the effect of probiotic bacterium Lactobacillus paracasei SD1 on the level of salivary mutans streptococci and lactobacilli, and (2) the oral persistence of L. paracasei SD1 in orthodontically treated nonsyndromic cleft lip and palate patients.

Design:

Double-blinded, randomized, placebo-controlled study.

Participants:

A total of 30 orthodontically treated nonsyndromic cleft lip and palate patients (aged 19.22 ± 3.66 years): 15 in the intervention group (A) and 15 in the control group (B).

Interventions:

Milk with or without L. paracasei SD1. Average daily consumption of milk in both groups was 50 mL for 4 weeks. Main Outcome Measures: Salivary mutans streptococci and lactobacilli were enumerated using a quantitative differential culture at baseline and once a week after the end of the administration period for 4 weeks. The persistence of L. paracasei SD1 was traced using arbitrarily primed polymerase chain reaction of the DNA fingerprinting.

Results:

A statistically significant reduction in mutans streptococci counts occurred in group A, in contrast to group B (P < .001). A significant increase of lactobacilli numbers was found in group A (P < .001), and L. paracasei SD1 could be detected up to 4 weeks following cessation of dosing.

Conclusions:

The probiotic milk powder containing L. paracasei SD1 could reduce mutans streptococci counts and was apparently able to colonize the oral cavity of the orthodontically treated cleft lip and palate patients. However, the potentially beneficial influence of the probiotic milk on the complex oral microflora justifies further studies with a larger group of volunteers.

Objective Comparison of the effects of vomerine flap (VF) closure of the hard palate at the time of lip repair with non-closure of the hard palate in subjects with unilateral cleft lip and palate (UCLP).

Design:

Retrospective, single-blinded, cohort study.

Setting:

Study model sets of 40 consecutive, non-syndromic, infants with complete UCLP operated on between 1988 and 1998.

Patients:

All subjects were operated on by a single consultant plastic surgeon immediately before and after the unit's change of protocol (1993), when VF closure of the hard palate was incorporated at the time of lip repair. Subjects were divided into two groups: VF ( n = 18) and non-VF (n = 22), which acted as a control group. Each subject had maxillary impressions taken before lip repair at 3 months (VF mean age = 11.7 weeks; non-VF mean age = 13.4 weeks) and before palate repair at 6 months (VF mean age = 22.8 weeks; non-VF mean age = 24.0 weeks). Main Outcome Measures: Seven predetermined landmarks and four maxillary dimensions were computed following single-blinded analysis using a reflex microscope.

Results:

Repeatability tests showed good measurement precision. The operator measurement errors were 0.00018 mm in a horizontal plane (X and Y) and 0.00028 in the vertical plane (Z). The VF group showed significant changes in the alveolar cleft width. There were no statistically significant changes in any arch-form variable between the VF and non-VF groups.

Conclusion:

The decrease of alveolar arch gap width at palate repair (6 months) in the VF group was significantly more than the decrease observed in the non-VF group, and there was no significant decrease in the, anterior and posterior arch width or anteroposterior length of the hard palate in the VF group compared with the non-VF group.

Objective : To assess whether individuals born to consanguineous parents had a higher frequency of nonsyndromic orofacial clefts compared with those with no parental consanguinity. Design : A prespecified plan for a search strategy, inclusion/exclusion criteria, and data extraction from studies reporting consanguinity in relation to nonsyndromic orofacial clefts (NSOFC) was carried out. Papers reporting observational studies with control populations were included, without language restrictions, and these reports were assessed for quality. Sensitivity analyses using subgroups, homogeneity evaluation, and assessment of publication bias were carried out, and meta-analyses of extracted data were performed. Results : Sixteen studies fulfilled the selection criteria and were included in the meta-analyses. There were statistically significant relationships between consanguinity and NSOFC for all 16 studies combined (P = .0003), with odds ratio (OR) = 1.83 and 95% confidence interval (CI) = (1.31, 2.54); 10 case-control studies (P = .006), with OR = 2.06 and 95% CI = (1.23, 3.46); six cross-sectional studies (P = .03), with OR = 1.34 and 95% CI = (1.02, 1.76); first cousins consanguineous marriages (P = .04), with OR = 1.40 and 95% CI = (1.01, 1.93); cleft palate alone (P = .01), with OR = 1.89 and 95% CI = (1.14, 3.13); and cleft lip with or without cleft palate cases (P = .002), with OR = 1.56 and 95% CI = (1.18, 2.07). Conclusion : Although there was a high level of study heterogeneity, the evidence is consistent in suggesting that consanguinity is a risk factor for NSOFC, with an overall OR of 1.83 (95% CI, 1.31 to 2.54), implying that there was almost twice the risk of a child with NSOFC being born if there was parental consanguinity.

Objective :  While sex-determining region Y-Box 2 (SOX2) mutations are typically recognized as yielding ocular and central nervous system abnormalities, they have also been associated with other craniofacial defects. To elucidate the genesis of the latter, Sox2 hypomorphic (Sox2(HYP)) mice were examined, with particular attention to secondary palatal development. Results :  Clefts of the secondary palate were found to be highly penetrant in Sox2(HYP) mice. The palatal clefting occurred in the absence of mandibular hypoplasia and resulted from delayed or failed shelf elevation. Conclusions :  Sox2 hypomorphism can result in clefting of the secondary palate, an effect that appears to be independent of mandibular hypoplasia and is thus expected to result from an abnormality that is inherent to the palatal shelves and/or their progenitor tissues. Further clinical attention relative to SOX2 mutations as a basis for secondary palatal clefts appears warranted.

Objective :  To determine whether children with repaired cleft palate and adequate velopharyngeal closure exhibit abrupt laryngeal engagement during stop plosive-vowel transitions as compared with children without cleft palate. Design :  A prospective group design was used with convenience sampling of patients at a university craniofacial center. Participants :  Participants were 25 children (15 boys, 10 girls) with repaired cleft palate (mean age = 10.9 years, standard deviation = 1.8 years) and 20 children (10 boys, 10 girls) without cleft palate (mean age = 10.8 years, standard deviation = 1.8 years). All children with cleft palate had adequate velopharyngeal closure as determined by aerodynamic testing. Main Outcome Measures :  (1) Peak oral airflow was determined during the release of /t/ in the word "two" during a counting task. (2) An index of laryngeal engagement defined as the ratio of the maximum oral airflow declination to peak oral airflow was calculated during the release of /t/. (3) Sound pressure level was determined during the vowel of the word "two." Results :  Children with cleft palate exhibited significantly more negative laryngeal engagement ratios (i.e., more abrupt adduction) (P = .002) and greater sound pressure level (P = .049) than controls. There was a significant negative relationship between laryngeal engagement and sound pressure level for all children (r = -.428, P = .003). Conclusions :  Children with repaired cleft palate and adequate velopharyngeal function appear to use a strategy of abrupt laryngeal adduction during stop plosive-vowel transitions. This strategy-perhaps learned even prior to palate surgery-may help to achieve either adequate sound pressure level and/or velopharyngeal closure.

Background :  In spite of studies reporting a relatively high frequency of additional conditions in children with a cleft lip and/or cleft palate (CL/P), almost no research has focused on this clinically important subgroup. The objective of this study was to compare psychosocial adjustment in children with CL/P with and without an additional condition. Design :  Cross-sectional data based on routine psychological assessments at age 10 years, with comparisons to national reference groups. Setting :  Centralized treatment (country). Participants :  Two hundred five children with CL/P (participation rate: 80.1%) from three consecutive birth cohorts. Outcome measures :  The Strengths and Difficulties Questionnaire (self-report and parent report) and the Child Experience Questionnaire (self-report). Results :  Eighty-one children (39.5%) were identified as having at least one condition in addition to the cleft. These children reported significantly more psychosocial difficulties than children with a cleft alone. Differences between specific conditions were minor. Children with a cleft alone (n = 124) reported mean scores that were comparable to those reported by the reference group. There were no differences in adjustment between children with a visible versus a non-visible cleft. Conclusions :  The present study highlights the need for research to be conducted in children with CL/P who have additional conditions to provide better knowledge and clinical care for a potentially vulnerable subgroup of children and their parents.

Objective : The purpose of this experiment was to evaluate the use of RPL and LAHSHAL coding systems by individuals not specialized in craniofacial abnormalities to code cleft lip and palate (CLP). The effectiveness of system use by referring services and the electronic medical record (EMR) applicability of other CLP systems was evaluated by literature review and testing data. Design and Participants : The RPL and LAHSHAL systems were presented together to a sample of medical students (n = 28) and neonatal intensive care unit nurses (n = 24) from Loma Linda University. Following the presentation, a test assessing the application of each system was administered. A second assessment of the medical students (n = 23) 2 weeks after the initial presentation evaluated system retention. Scores were compared using t test (P ≤ .05). Results : Both the medical students and nurses used RPL more accurately than LAHSHAL in the first assessment (76.9% versus 45.2%, P < .001; 46.6% versus 22.5%, P < .001). Medical students again used RPL more accurately at the 2-week assessment (72.2% versus 43.7%, P < .001). Accuracy of use within each system was not significantly different between the two assessments. Conclusion : Our test results and literature review indicate that, when compared with LAHSHAL, RPL is more easily and accurately used by representatives not specialized in abnormalities in CLP; therefore, RPL may be a more effective system for nonspecialist health care providers to improve the accuracy of referrals and simple EMR documentation.

Objectives :  To bring a neonatal classification system of unilateral cleft lip and palate and to correlate this classification with the distribution of the permanent lateral incisor and maxillary growth. Design :  Retrospective with longitudinal follow-up. Setting :  Tertiary. Patients :  A total of 112 individuals with treated unilateral cleft lip and palate and 30 controls. Main Outcome Measures :  Unilateral cleft lip and palate neonatal casts were classified anatomically in four categories, in which Class 1 corresponds to a maxillary arch with a narrow alveolar cleft; Class 2 corresponds to a balanced form; Class 3 corresponds to a wide cleft and short maxilla; and Class 4 corresponds to a wide cleft and long maxilla. The classification was correlated with the distribution of the permanent lateral incisor. Maxillary growth was evaluated using a cephalometric analysis after the age of 10 years. Results :  Clinical classification of unilateral cleft lip and palate found 10 cases of Class 1 (8.9%), 34 cases of Class 2 (30.4%), 46 cases of Class 3 (41.1%), and 22 cases of Class 4 (19.6%). The permanent lateral incisor was most often present in narrower clefts (Classes 1 and 2); whereas, large clefts (Classes 3 and 4) were relatively more frequently associated with an agenesis of the permanent lateral incisor (P = .019). Maxillary growth impairment was most severe in Class 3, with a mean sella-nasion-A point angle at 71.9° ± 4.6° (P < .001). Conclusions :  Using the cleft width, arch form, and shape of the nasal septum, unilateral cleft lip and palate can be classified into four different classes at birth, which can all give information about permanent lateral incisor agenesis and maxillary growth.

Objectives :  The aim of this study was to characterize three-dimensional (3D) facial morphological variation of children with cleft lip and palate compared to an age- and sex-matched control group. Design :  Cross-sectional, case-controlled observational morphometric study. Materials and Methods :  Subjects were 103 children aged 8 to 12 years old with cleft lip and palate-40 with unilateral cleft lip and palate, 23 with unilateral cleft lip and alveolus, 19 with bilateral cleft lip and palate, and 21 with isolated cleft palate (ICP)-and 80 sex- and age-matched control subjects. Subjects were scanned using 3D stereophotogrammetry. Thirty-nine landmarks were digitized for each scan, and the x, y, and z coordinates for each landmark were extracted. Linear and angular facial measurements were computed. Multivariate analysis of covariance was undertaken to detect significant differences in facial morphometry between the groups. Results :  Statistically significant differences (P < .05) were observed between all groups. The greatest morphometrical impact was seen in those groups where both lip and palate were affected and repaired; the group with ICP was the most similar to the control subjects. Conclusions :  Significant differences were seen in 3D soft-tissue measurements, mainly in the nasolabial region, between the cleft groups where the lip is affected and the ICP and control groups.

Objective :  The main objective of the study is to identify practical and cultural factors influencing the mental health of mothers of children with an orofacial cleft in Benin and to compare it with a sample of Swiss mothers in the same conditions. Method :  Thirty-six mothers of children with an orofacial cleft in Benin and 40 mothers of children with an orofacial cleft in Switzerland were interviewed about practical and emotional aspects concerning their child and their own lives. Then, they completed the Perinatal Postraumatic Stress Questionnaire and the Beck Depression Inventory. Results :  Mothers in Benin had significantly higher posttraumatic stress and depression symptoms compared with mothers in Switzerland. Depression symptoms were higher in Beninese mothers coming from urban areas, in Beninese mothers with few or no other children, and in Beninese mothers whose child was operated on at a more advanced age. Discussion :  This study stressed the importance of cultural differences in perceptions of orofacial clefts in order to provide appropriate care to patients and their families. In particular, wide campaigns of information should help parents to understand the cleft origin and the medical staff in small dispensaries to provide adequate support and care. This may diminish anxiety concerning the child's short- and long-term prognosis. Creation of a Beninese parental support group for children with clefts and their families could be another way to provide information and support where multidisciplinary care is not available.