In our center, oral surgeons discuss presurgical orthodontic treatments and the preferable timing for alveolar bone grafting (BG) with orthodontic dentists.
We evaluated the results of BG retrospectively.
Objects and methods: Among cases given BG in 2007-2010, we focused on 27 unilateral cleft lip alveolar (UCLA) cases and 58 unilateral cleft lip alveolar and palate (UCLP) cases, and investigated the gender distribution, age at surgery, cleft width, presence and eruption of lateral incisors in the cleft side, eruption and root formation of canines in the cleft and non-cleft sides at surgery, weight of transplanted bone, and marginal bone level obtained, and compared them between the UCLA and UCLP groups.
Results: 1. There were 13 males and 14 females with UCLA, and 35 males and 23 females with UCLP. Age at surgery was 118.4+/-20.5 (92-171) months in the UCLA group, and 119.1+/-14.7 (89-168) months in the UCLP group. There was no significant difference in gender distribution or age at surgery between both groups.
2. Cleft width (at alveolar crest and nasal floor) was 5.7+/-2.3 and 12.1+/-4.5mm in the UCLA group, and 7.3+/-2.7 and 14.6+/-3.9mm in the UCLP group. Weight of transplanted bone was 2.1+/-1.0g in the UCLA group, and 2.5+/-1.0g in the UCLP group. Thus, there were significant differences between both groups (p < 0.05).
3. At surgery, canines erupted in 8 UCLA and 5 UCLP cases in the cleft side. There was a significant difference between both groups (p < 0.05).
4. Canine root formation was more than half full in 14 UCLA and 11 UCLP cases in the cleft side, in 13 UCLA and 12 UCLP cases in the non-cleft side. Canine root formation was significantly faster in the UCLA group than the UCLP group in both sides (p < 0.05).
5. For the evaluation of marginal bone level, Enemark's level of more than 2 was obtained in 96.3% of UCLA and 98.3% of UCLP cases. There was no significant difference between both groups.
Conclusion: In this study, significant differences were detected in cleft width, weight of transplanted bone, eruption of canines in the cleft side, and root formation of canines in the cleft and non-cleft sides at surgery between both groups, but marginal bone formation was excellent in both groups.

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We have performed modified two-stage palatoplasty since November 2003. In the first operation of two-stage palatal repair, not only the soft palate but also the posterior half of the hard palate is closed simultaneously using modified Furlow's method. The purpose of this study was to evaluate retrospectively the speech outcome at 4 and 5 years of age for patients treated by this procedure.
The subjects were 39 consecutively treated patients with unilateral and bilateral cleft lip and palate, who underwent the first operation of two-stage palatal repair at 18 to 25 months of age (mean age: 20.4) from November 2003 to August 2008. Eighteen of the 39 patients (46.2%) wore a palatal plate postoperatively.
Velopharyngeal competency was competent in 87.2% at 4 years of age and in 89.7% at 5 years of age. The incidence of articulation disorders was 59.0% at 4 years of age and 56.4% at 5 years of age. At the age of 4 years, palatalized misarticulation was observed most frequently, followed by glottal stop, nasopharyngeal misarticulation, lateral misarticulation and pharyngeal fricatives, at 50.0%, 35.7%, 7.1%, 3.6% and 3.6%, respectively. At the age of 5 years, the same tendency was seen: palatalized misarticulation 48.0%, glottal stop 32.0%, lateral misarticulation 16.0% and pharyngeal fricatives 4.0%.
Velopharyngeal competency was relatively good and the incidence of articulation disorders was low in the previous reports of domestic centers adopting two-stage palatoplasty. It is thought that narrowing of the remaining oro-nasal communication in the hard palate is facilitated because the palatal closure at the first operation of two-stage palatoplasty is performed on the posterior half of the hard palate from the soft palate, thus reducing adverse effects for articulation.

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We investigated velopharyngeal function in 25 patients with submucous cleft palate who presented for assessment by the Shizuoka Children's Hospital cleft palate team during the 35 years from 1977 to 2012. These patients had had good velopharyngeal function at preschool age and had not undergone surgery.
We found that:
1) About 1/3 of patients maintained good velopharyngeal function; the remainder showed aggravation or variability of function.
2) There was no significant correlation between the coexistence of other diseases and changes in velopharyngeal function.
3) The average time of deterioration of velopharyngeal function was age 9 years and 7 months, but the time varied greatly (from ages 6 to 16 years) among individuals.
These results suggested the necessity of long-term follow-up.

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Beckwith-Wiedemann Syndrome (BWS) is a common overgrowth syndrome that involves abdominal wall defects, macroglossia, and gigantism. BWS is caused by an imprinted gene within the chromosome 11p15.5 region. A patient was referred to our center for speech disorder and macroglossia. Speech training and conservative treatment were performed. An imprinted gene was analyzed in the BWS patient. We detected hypomethylation of KvDMR1 in this patient. We report on the imprinted gene and mechanism of BWS development.

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A clinico-statistical analysis of associated congenital anomalies was made on 661 patients with cleft lip and/or palate, who were examined at our department from January 1967 to December 2011. The following results were obtained:
1. The number of patients with other associated congenital anomalies and/or syndrome was 82, which was a 12.4% incidence rate.
2. Of the associated congenital anomalies，congenital heart disease was observed in 27 cases (4.1%), anomalies of extremities in 18 cases (2.7%), auricular anomalies in 17 cases (2.6%), and anomalies of eyes and palpebral conjunctiva in 12 cases (1.8%).
3. In the patients with associated congenital anomalies, the number of associated congenital anomalies in a patient averaged 1.3 with a maximum of 6.
4. Of 30 cases (4.5%)were diagnosed as a syndrome, Pierre Robin syndrome was observed in 17 cases and the first and second branchial arch syndrome and Treacher Collins syndrome in 2.

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We report a rare neglected case of an adult with unilateral cleft lip and palate. The patient was a 20-year-old man who presented to our department with complaints of palate formation and tooth alignment. Cheiloplasty alone had been carried out at a certain university three months after birth, but subsequent medical treatment had been neglected for a long time. We observed an unoperated cleft palate with oronasal fistula around the alveolar cleft and secondary deformities of the lip and nose. Hypernasality and slight articulatory disorder were noted. We planned a primary palatoplasty with alveolar bone grafting and secondary repair of the lip and nose in consideration of postoperative orthodontic treatment. After the operation, we noted modification of the uvula, but progress was good. Speech training and orthodontic treatment are currently being continued. Since this case was an adult, we considered it to be much more important to close the alveolar cleft rather than active acquisition of velopharyngeal dysfunction. Therefore, we modified the design: the amount of push-back was minimized, the exposed area of the bone at the front of the palate which arises after the operation was made as small as possible, and the bone graft part with oronasal fistula was covered with sufficient lateral gingival mucoperiosteal flap in conjunction with a palatal flap. Comparatively good recovery was obtained and the patient and his family are satisfied.

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Tracheal stenosis of newborn infants and children is often noted in subglottic cricoid cartilage. A cleft lip alveolus patient with subglottic tracheal stenosis exhibited reduced airway narrowing following a 3-month period which allowed developmental and weight gains to occur. The patient was a female infant, one month old, with no familial or past history, born at 39 weeks gestation, weighing 2290 g, Apgar score 10/10. There were no respiratory and heart symptoms of note, but a diagnosis of left cleft lip and alveolus was made, and suckling instruction was commenced.
A plan was made to perform cheiloplasty at 7 months and 5960 g post weight-gain. The preoperative chest radiograph was normal, but endo-tracheal intubation to deliver anesthesia was prevented by subglottic resistance. The surgery was postponed due to resultant airway edema post extubation, and concerns over possible subglottic tracheal stenosis. The patient was discharged on following day, and the respiratory status was satisfactory, allowing a growth period, following post radiographic findings of subglottic development. Cheiloplasty was performed at 1 year 3 months old, at weight 7505 g, with intubation, without resistance on induction of anesthesia. At 8 months post-surgery, the respiratory status was normal and the patient was progressing well, with only mild subglottic stenosis. The pre-operative assessment was problematic, however, so a further growth period was allowed to ensure the surgery was successfully performed.

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A seminar on 5-Year Olds' Index hosted by the Japancleft Committee, Japanese Cleft Palate Association took place at the Sanjo Conference Hall, the University of Tokyo on July18-19, 2012. The purpose of the seminar was to provide the opportunity to have an exercise in assessing dental arch relation in patients aged 5 years with unilateral cleft lip and palate. Dr. Nikki Atack and Prof. Jonathan Sandy who had developed the index were invited from the University of Bristol as the lecturers at the seminar. The number of participants was over 80 from various special fields such as plastic surgery, oral surgery, pediatric dentistry and orthodontics. A total of 355 dental models were collected from participating institutions and were employed for evaluation training. As a result of intra-examiner agreement and agreement with Gold standard, almost all delegates obtained an agreement score of 0.80 or above, which would be very suitable and able to assess 5 year olds' models. Although the models collected were not selected based upon critical inclusion criteria, the result of assessment of the Japanese models was very analogous to that of UK Pre-CSAG (Clinical Standard Advisory Group). We hope that the present seminar would be helpful for the improvement of treatment outcomes in patients with cleft lip and palate.

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