Palate Center Research Articles

A retrospective study on familial occurrence of cleft lip and/or palate

ObjectiveThe aim of this study was to assess whether familial occurrence has an influence on the state of patients with non-syndromic cleft lip and/or palate (NsCL/P). Materials and methodsA retrospective analysis was performed, using medical records of 425 patients with NsCL/P who underwent integrated treatment in the Cleft Lip and Palate Center, University of the Ryukyus Hospital between 1994 and 2010. No affected subjects had accompanying defects or findings suggestive of a syndromic diagnosis. ResultsOf the total of 425 participants with NsCL/P, 82 (19.29%) presented a positive history of cleft in their families and 343 (80.70%) presented a negative history. In the distribution of the cleft types, the frequencies of cleft lip only (CLO), cleft lip and alveolar (CLA) ridge, cleft lip and palate (CLP), and cleft palate only (CPO) except submucous cleft palate, were 10.97%, 18.29%, 56.09% and 14.63% in familial cases, and 12.82%, 23.32%, 39.06% and 24.78% in sporadic cases, respectively. There were statistical differences in the cleft types by chi-squared test (p=0.038), odds ratios and 95%CI (OR=0.84, 0.74, 1.99, 0.52; 95%CI, 0.39–1.79, 0.40–1.36, 1.22–3.24, 0.27–1.01, respectively). We also examined the distribution of the cleft side as well as the parental and neonatal status at delivery. However, we did not find any significant differences. ConclusionFamilial occurrence might have an influence on the cleft types.Further research is needed to define the reasons for this influence.

Left-Sided Predominance of Hypodontia Irrespective of Cleft Sidedness in a French Population

Individuals with oral clefts exhibit considerably more dental anomalies than do individuals without clefts. Our aim was to evaluate the prevalence of tooth agenesis in a sample composed of 124 children (81 boys and 43 girls, mean age 12.5 years) with clefts registered with the Cleft Palate Center in Strasbourg (France). Cleft types and dental agenesis were assessed by clinical and radiographic examination. Cleft types were divided into four groups according to the clinical extent of the cleft (cleft lip only [CL, 12.9%], cleft lip and alveolus [CLA, 4%], cleft lip and palate [CLP, 49.2%], and cleft palate only [CP, 33.9%]). Of the subjects 63% had evidence of hypodontia: maxillary lateral incisors (54%) and upper and lower premolars (32%) were the most common missing teeth. The number of dental ageneses associated with CP (54%) and CLP (79%) was significantly higher than that with CL (33%). All these anomalies were found in proportionately higher frequencies as the severity of the cleft increased, and we found left side predominance for hypodontia (p < .01) irrespective of cleft sidedness. Both right-sided and left-sided clefts were more frequently correlated with left-sided dental agenesis. This left-sided prevalence suggests that common signaling malfunctions might be involved, both in dental development anomalies and cleft genesis.

Longitudinal Study of Effect of Hotz's Plate and Lip Adhesion on Maxillary Growth in Bilateral Cleft Lip and Palate Patients

To investigate the effects of infant orthopedic treatment and lip adhesion on maxillary growth of patients with bilateral cleft lip and palate (BCLP). Prospective longitudinal study. The present study was conducted at the Cleft Lip and Palate Center, Harapan Kita Children and Maternity Hospital, Indonesia, and the Department of Oral and Maxillofacial Surgery, Kyushu University Hospital, Japan. The study sample consisted of 53 patients with complete BCLP and 10 noncleft patients with other diseases. Patients with BCLP were divided into three groups: H (-), 11 patients treated without Hotz's plate; H (+), 24 treated with Hotz's plate; and LA-H, 18 treated with lip adhesion and Hotz's plate. Serial dental casts were obtained from each BCLP child at the following four time points: first visit, labioplasty, palatoplasty, and 5 years of age. Each maxillary dental cast was scanned, and the linear and angular dimensions were measured. Lip adhesion showed a temporary negative effect. In all patients with BCLP, the surgeries affected the growth of the anterior arch width until the age of 5 years. Collapse of the premaxilla following labioplasty in the H (-) group affected the growth of dental arch length until the age of 5 years. Treatment using Hotz's plate prevented collapse of the premaxilla, and the growth of the arch length was comparable to that observed in the noncleft group.

Changing Face of Cleft Care

The organization and management of specialized cleft lip and palate centers in developing countries are challenging because of the lack of financial resources devoted to the national health care system. The treatment of cleft lip and palate is of low priority for health care and budgets; however, gradual progress is possible. As an example of how care might be improved in the developing world, we suggest guidelines to strengthen the local cleft lip and palate centers in Brazil based on the ideal geographic distribution of cleft centers around the country, to achieve the following objectives: first, avoid patient's migration; second, facilitate patient's adherence; third, focus on a global and continuous multidisciplinary treatment; and fourth, avoid indiscriminate opening of nonprepared cleft lip and palate centers in our country. This ideal scenario would put the cleft lip and palate health attention on the right path in Brazil.

Thermoplastic base plate modification of the nasoalveolar molding device: A case report

Nasoalveolar molding {NAM) is a nonsurgicalmethod of reshaping the gums, lip and nostrils before cleft lip and palate surgery,lessening the severity of the cleft. Surgery is performed after the molding is complete,approximately three to six months after birth. NAM is used mainly for children withlarge clefts,and has revolutionized cleft repair. In the past, fixing a large cleft required multiple surgeries between birth and age 18, putting the child at risk for psychologicaland socialadjustment problems. With nasoalveolar molding, the orthodontist and surgeon can improve a large cleft in the months before surgery. This helps the surgeon get a better shape of the nose and a thinner scar in only one surgery Although NAM is being used extensively at various cleft lip and palate centers , a serious disadvantage is gingival tissue inflammation of the infant due to the acrylic component of the device. We have substituted the acrylic with Thermoplastic baseplate which substantially reduced the gingivalinflammation and allergies associated with the use of acrylic NAM devices.

Initial size of cleft does not correlate with size and function of nasal airway in adults with unilateral cleft lip and palate

The noses of patients with clefts are often functionally inadequate. The aim of the present study was to evaluate the correlation between size of the maxillary cleft in infancy and size and function of the nasal airway in adults with unilateral cleft lip and palate (UCLP). This is a long-term follow up study including 53 patients with UCLP born between 1960 and 1987 and treated at the Cleft Lip and Palate Centre, Uppsala University Hospital, Sweden. Lip repair was performed at 3–4 months of age followed by either a one-stage or a two-stage palatal closure. The size of the cleft was measured on infant maxillary dental casts. Nasal minimum cross-sectional area (cm2) and volume (cm3) (acoustic rhinometry), air flow resistance (Pa s/cm3) (rhinomanometry), peak inspiratory flow (l/min) (peak nasal inspiratory flow) and number of identified odours (Scandinavian odor-identification test) were assessed in adulthood. The size of the maxillary cleft varied considerably at infancy. The size of the nasal airway and its function on the cleft side in adulthood were reduced compared with the non-cleft side, but no correlations were found between size of the initial cleft in infancy and size and function of the nasal airway in adulthood. In adults born with UCLP, therefore, size of the maxillary cleft in infancy does not seem to affect size and function of the nasal airway in adulthood.

The Americleft Study: An Inter-Center Study of Treatment Outcomes for Patients with Unilateral Cleft Lip and Palate Part 2. Dental Arch Relationships

To compare maxillomandibular relationships for individuals with nonsyndromic complete unilateral cleft lip and palate using the Goslon Yardstick for dental models. Retrospective cohort study. Five cleft palate centers in North America. A total of 169 subjects with repaired complete unilateral cleft lip and palate who were consecutively treated at the five centers. Ethics approval was obtained. A total of 169 dental models of patients between 6 and 12 years old with complete unilateral cleft lip and palate were assessed using the Goslon Yardstick. Weighted kappa statistics were used to assess intrarater and interrater reliabilities; whereas, analysis of variance and Tukey-Kramer analysis was used to compare the Goslon scores. Significance levels were set at p < .05. Intrarater and interrater reliabilities were very good for model ratings. One center that incorporated primary alveolar bone grafting showed especially poor Goslon scores that were significantly poorer than the remaining centers. The surgery protocols used by the other four centers did not include primary alveolar bone grafting but involved a number of different lip and palate closure techniques. Using the Goslon Yardstick assumptions, the center with the best scores would be expected to require end-stage maxillary advancement orthognathic surgery in 20% of its patients; whereas, the center with the worst scores would be likely to require this surgery in 66% of its patients. The Goslon Yardstick proved capable of discriminating among the centers' dental arch relationships. Possible explanations for the differences are discussed.

The Americleft Study: An Inter-Center Study of Treatment Outcomes for Patients with Unilateral Cleft Lip and Palate Part 3. Analysis of Craniofacial Form

To compare craniofacial morphology for individuals with nonsyndromic complete unilateral cleft lip and palate between the ages of 6 and 12 years. Retrospective cohort study. Four North American cleft palate centers. A total of 148 subjects with repaired complete unilateral cleft lip and palate who were consecutively treated at the four centers. The 148 preorthodontic lateral cephalometric radiographs were scanned, scaled, digitized, and coded to blind the examiners to radiograph origin. On each radiograph, 18 (angular and ratio) cephalometric measurements were performed. Measurement means, by center, were compared using analysis of variance and Tukey-Kramer analysis. Significant differences were found for sagittal maxillary prominence among the four centers. The most significant difference was seen between Center B (lowest SNA) and Center C (highest SNA). Similar differences were seen at the soft tissue level, with Center C showing a significantly larger ANB angle compared with Centers B and D. Center C was also shown to have statistically greater mean soft tissue convexity than Centers B, D, and E. The mean nasolabial angle in Center B was significantly more acute than in Centers C, D, and E. No statistically significant differences were seen for mandibular prominence, vertical dimensions, or dental inclinations. Significant differences were seen among the centers for hard and soft tissue maxillary prominence, but not for mandibular prominence, vertical dimensions, or dental inclinations. A modest but statistically significant (p < .001) negative correlation was found between Goslon scores and ANB angle (r = -.607).

The Americleft Study: An Inter-Center Study of Treatment Outcomes for Patients with Unilateral Cleft Lip and Palate Part 5. General Discussion and Conclusions

To summarize the Americleft study regarding treatment outcomes for patients with complete unilateral cleft lip and palate (CUCLP). Five cleft palate centers in North America. One hundred sixty-nine subjects, between the ages of 6 years and 12 years, with repaired CUCLP who were consecutively treated at the five centers. Study consisted of model comparisons assessing maxillomandibular relationship using the GOSLON Yardstick (169 patients from all 5 centers), soft and hard tissue craniofacial morphologic comparisons using lateral cephalometric analyses (148 patients from four of the centers), and nasolabial esthetics assessments (125 patients from four of the centers). Significant differences were found between the center with the best GOSLON scores and the remaining centers. These differences also corresponded to those found in the craniofacial morphologic cephalometric assessment. Sagittal maxillary prominence was found to be significantly better for the center with the best GOSLON scores, while no significant differences were seen among the centers for mandibular prominence, vertical dimensions, or dental inclinations. No differences were seen for nasolabial esthetics between the centers. Challenges experienced while undertaking the inter-center retrospective study are reviewed. Aspects of treatment that could potentially make the outcome of treatment less optimal included primary alveolar bone grafting and extensive treatment protocols. Differences in the outcomes identified between the centers were restricted to the maxilla, and no differences were identified for mandibular prominence, vertical dimensions, or dental inclinations.

The Americleft Study: An Inter-Center Study of Treatment Outcomes for Patients with Unilateral Cleft Lip and Palate Part 1. Principles and Study Design

The Americleft study is a North American initiative to undertake an intercenter outcome study for patients with repaired complete unilateral cleft lip and palate from five well-established North American cleft centers. Retrospective cohort study. Five cleft palate centers in North America. This is the first paper in a series of five that outlines the overall goals of the study and sets the basis for the clinical outcome studies that are reported in the following four papers. The five centers' samples and treatment protocols as well as the methods used for each study are reported. The challenges encountered and possible mechanisms to resolve them and reduce methodological error with intercenter studies are also reviewed.

Infant Orthopedics in Cleft Lip and Palate have No Effect on Maxillary Arch Dimensions in Children Younger than Age 6

Infant orthopedics has no effect on maxillary arch dimensions in the deciduous dentition of children with complete unilateral cleft lip and palate (Dutchcleft). Bongaarts CAM, van ’t Hof MA, Prahl-Andersen B, Dirks IV, Kuijpers-Jagtman AM. Cleft Palate Craniofac J 2006;43:665-72. Deborah A. Redford-Badwal, DDS, PhD Are there any differences at ages 4 and 6 years on maxillary arch dimensions in the deciduous dentition in children with unilateral cleft lip, alveolus, and palate who receive infant orthopedics before surgical closure of the soft palate versus those who do not receive orthopedics? The source of funding was not directly stated, rather it was reported that it was part of a Dutch intercenter study carried out in a collaboration among the Cleft Palate Centers of the University of Nijmegen, Academic Center of Dentistry in Amsterdam, and University Medical Center Rotterdam in Rotterdam Randomized controlled trial Level 1: Good-quality, patient-oriented evidence Not applicable

Clinical Study on Submucous Cleft Palate Patients in the Cleft Lip and Palate Center, Fujita Health University Hospital

Clinical Study on Submucous Cleft Palate Patients in the Cleft Lip and Palate Center, Fujita Health University Hospital

Epidemiological Analysis of Cleft Lip and/or Palate by Cleft Pattern

To analyze gender differences in cleft pattern by the clinical statistical study of Japanese patients with cleft lip and/or cleft palate. Cleft pattern modeling was used to analyze 782 patients with cleft lip and/or cleft palate (417 males and 365 females) who had been examined at the Cleft Lip and Palate Center, Aichi-Gakuin University Hospital, and whose details could be confirmed. Relationships between gender and cleft type were analyzed with chi-squared test. A comparison of gender differences by cleft type revealed that a greater percentage of males had milder cleft lip, cleft lip and palate, or cleft palate, whereas the percentage of females tended to be greater as cleft severity increased. Modeling of cleft patterns enables more detailed classification of cleft lip and palate, and can lead to a greater understanding of pathology.

The Effects of Unrepaired Cleft Palate on Early Language Development in Chinese Infants

To investigate the effects of unrepaired cleft palate on the development of vocabulary comprehension and expression of Chinese children aged 8 to 15 months. The parents or caregivers of 40 infants with unrepaired cleft palate were recruited from the Cleft Lip and Palate Center at the Stomatology School of Peking University. Vocabulary development was assessed using a parent report protocol, the Chinese Communicative Development Inventory: Infant, for Putonghua (PCDI: Infant). The scores for vocabulary comprehension and expression of infants with clefts were compared with typically developing infants in the same age group. All infants with unrepaired cleft palate performed below the typically developing infants in vocabulary comprehension, but there were no significant differences from 8 to 15 months of age. No significant differences were found from 8 to 13 months of age on the expressive vocabulary portion of the Chinese Communicative Development Inventory: Infant, for Putonghua. However, a significant difference appeared at 14 and 15 months, with infants with cleft palate performing at a significantly lower level than the typically developing infants. The development of vocabulary comprehension in Chinese infants younger than 14 months with unrepaired cleft palate is not significantly delayed. At 14 and 15 months, however, the development of vocabulary expression is delayed. The reasons may be found in the specific phonetic characteristics of Putonghua.

The morphology of the sella turcica in velocardiofacial syndrome suggests involvement of a neural crest developmental field

We described the morphology of the sella turcica in individuals with velocardiofacial syndrome (VCFS), also known as chromosome 22q11.2 deletion syndrome, and compared the morphology with that of a control group of individuals from the Oslo University Craniofacial Growth Archive. The aim was to measure the cranial base angles in individuals with VCFS and, if possible, to discover the developmental field that may be involved in the condition. The study included 33 patients with VCFS from the Copenhagen Cleft Palate Center, Denmark. The genotype was confirmed by fluorescence in situ hybridization. The morphology of the sella turcica was described and measurements of the cranial base angles were performed on lateral cephalometric radiographs. The VCFS individuals had larger deviations in the morphology of the sella turcica compared to individuals from the Oslo University Craniofacial Growth archive. The deviations were mostly in the posterior part of the dorsum sellae. Individuals with VCFS had increased cranial base angles. The results of this study combined with the information in the literature on the main defects in VCFS (palatal abnormalities, cardiac anomalies, thymic hypoplasia or aplasia, hypothyroidism, and posterior brain abnormality), suggest involvement of a specific developmental field.

Objective Assessment of the Nasal Airway in Unilateral Cleft Lip and Palate—A Long-Term Study

To objectively evaluate the nasal function in adults operated on for unilateral cleft lip and palate with one-stage or two-stage palate closure. The population consists of all unilateral cleft lip and palate patients born from 1960 to 1987 and treated at the Cleft Lip and Palate Center, Uppsala University Hospital, Sweden. The patients were treated according to the same protocol except for palate closure, which was performed in one stage until 1977 and in two stages thereafter. Eighty-three patients participated. Mean follow-up time after primary surgery was 32 years. An age-matched control group underwent the same examinations. Nasal minimum cross-sectional area (cm(2)) and volume (cm(3)) were assessed (acoustic rhinometry). Airflow resistance (Pa s/cm(3)) (rhinomanometry), peak inspiratory flow (L/min) (peak nasal inspiratory flow), and number of identified odors (Scandinavian Odor Identification Test) were determined. The cleft side of unilateral cleft lip and palate patients had significantly lower values for all parameters compared with controls (p < .001). No difference was found between one-stage and two-stage procedures in values for the cleft side. However, the nasal area and volume of the noncleft side were significantly larger in patients who underwent one-stage as compared with two-stage procedures (p < .05). The nasal airway of unilateral cleft lip and palate patients demonstrates a wide range of impairments that can be quantified by objective measurements. However, the measurements used did not differentiate between patients operated on with the one-stage and two-stage procedures except for values of the noncleft side.

Skull thickness in patients with clefts

The purpose was to analyze skull thickness in incomplete cleft lip (CL), cleft palate (CP), and combined cleft lip and palate (UCLP). Copenhagen School of Dentistry and Copenhagen Cleft Lip and Palate Centre. Patients with cleft lip, cleft palate, and combined cleft lip and palate and normal adult men. Four groups of patients comprised the study. One group of patients with CL (24 patients; 7 women, mean age 6; 17 men, mean age 7.1), one group of patients with UCLP (28 patients; 11 women, mean age 6.6; 17 men, mean age 6.7), one group of patients with CP (57 male patients aged 18-33), and one normal adult male control group. The CL and UCLP groups were compared. The CP group was compared with the normal adult male control group. CL women had a significantly thinner occipital bone compared with CL men (p = 0.027). Women with UCLP had significantly thicker occipital bone than the control women (incomplete CL) (p = 0.014). The study showed gender differences in skull thickness in different cleft types. It also demonstrated that particularly the occipital bone deviated in patients with UCLP, which may explain the considerable deviations in jaw shape and position, previously registered in patients with UCLP.

A Longitudinal Three-Center Study of Dental Arch Relationship in Patients with Bilateral Cleft Lip and Palate

To compare and evaluate longitudinally the dental arch relationships from 4.5 to 13.5 years of age with the Bauru-BCLP Yardstick in a large sample of patients with bilateral cleft lip and palate (BCLP). Retrospective longitudinal intercenter outcome study. Dental casts of 204 consecutive patients with complete BCLP were evaluated at 6, 9, and 12 years of age. All models were identified only by random identification numbers. Three cleft palate centers with different treatment protocols. Dental arch relationships were categorized with the Bauru-BCLP yardstick. Increments for each interval (from 6 to 9 years, 6 to 12 years, and 9 to 12 years) were analyzed by logistic and linear regression models. There were no significant differences in outcome measures between the centers at age 12 or at age 9. At age 6, center B showed significantly better results (p=.027), but this difference diminished as the yardstick score for this group increased over time (linear regression analysis), the difference with the reference category (center C, boys) for the intervals 6 to 12 and 9 to 12 years being 10.4% (p=.041) and 12.9% (p=.009), respectively. Despite different treatment protocols, dental arch relationships in the three centers were comparable in final scores at age 9 and 12 years. Delaying hard palate closure and employing infant orthopedics did not appear to be advantageous in the long run. Premaxillary osteotomy employed in center B appeared to be associated with less favorable development of the dental arch relationship between 9 and 12 years.

A Longitudinal Three-Center Study of Dental Arch Relationship in Patients With Bilateral Cleft Lip and Palate

To compare and evaluate longitudinally the dental arch relationships from 4.5 to 13.5 years of age with the Bauru-BCLP Yardstick in a large sample of patients with bilateral cleft lip and palate (BCLP).Retrospective longitudinal intercenter outcome study.Dental casts of 204 consecutive patients with complete BCLP were evaluated at 6, 9, and 12 years of age. All models were identified only by random identification numbers.Three cleft palate centers with different treatment protocols.Dental arch relationships were categorized with the Bauru-BCLP yardstick. Increments for each interval (from 6 to 9 years, 6 to 12 years, and 9 to 12 years) were analyzed by logistic and linear regression models.There were no significant differences in outcome measures between the centers at age 12 or at age 9. At age 6, center B showed significantly better results (p=.027), but this difference diminished as the yardstick score for this group increased over time (linear regression analysis), the difference with the reference category (center C, boys) for the intervals 6 to 12 and 9 to 12 years being 10.4% (p=.041) and 12.9% (p=.009), respectively.Despite different treatment protocols, dental arch relationships in the three centers were comparable in final scores at age 9 and 12 years. Delaying hard palate closure and employing infant orthopedics did not appear to be advantageous in the long run. Premaxillary osteotomy employed in center B appeared to be associated with less favorable development of the dental arch relationship between 9 and 12 years.

Parental Attitude towards the Provision of Nonsurgical Oral Health Care to Children with Oral Clefts: An Epidemiological Survey.

AimTo evaluate the attitudes of parents of 3 to 8 years old children with cleft lip and palate towards the provision of pediatric dental health care and assess the experience of dentistry in general dental practice.Materials and methodsA questionnaire was distributed to parents of 100 children in the age group of 3 to 8 years old with cleft lip and palate who visited the cleft lip and palate center in KS Hegde Medical Academy, Mangalore.ResultsOut of the hundred patients, 66% of the patients registered directly at the cleft center for their deformity. Only 20% of these children visited a local dental practitioner for their dental health care. 42% had neither undergone any dental treatment nor received any dietary or oral hygiene advices. Regarding the provision of the dental treatment, 74% wanted a pediatric dentist to provide dental check-up and treatment at the cleft center, whereas, 24% preferred a dental practitioner close to their home and 2% does not want treatment anywhere.ConclusionThe survey indicates that there is parental support for the pediatric dental assessment at the cleft center with subsequent arrangement of dental treatment with their local dental practitioners. The majority wanted a pediatric dentist to provide the dental check-up and treatment at the cleft center.