Journal of Japanese Cleft Palate Association
Online ISSN : 2186-5701
Print ISSN : 0386-5185
ISSN-L : 0386-5185
Volume 41, Issue 1
Displaying 1-9 of 9 articles from this issue
  • Akemi HIKAGE, Kanji NOHARA, Chihiro SUGIYAMA, Nobukazu TANAKA, Etsuko ...
    2016Volume 41Issue 1 Pages 1-7
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    Introduction: In those with cleft palate, it is well known that velopharyngeal function and the form of the palate affect the onset of articulation disorders. However, some patients with cleft palate develop misarticulation, while others do not, regardless of the severity of velopharyngeal incompetence and morphological defect of the palate. These findings suggest that some factors besides velopharyngeal function and the palatal form affect the onset of articulation disorders. It has been reported that some individuals without cleft palate develop articulation disorders if they have impaired motor coordination, but for those with cleft palate, there are no reports about the relationship between the onset of misarticulation and coordinated motion. The purpose of this study was to clarify this relationship in speakers with cleft palate.
    Subjects and methods: Sixty-one individuals with operated cleft palate, who visited Osaka University Dental Hospital to be followed up or receive speech therapy from November 2011 to April 2012, were enrolled as subjects. The parents of these subjects were asked to complete the Developmental Coordination Disorder Questionnaire (DCDQ). The subjects were divided into two groups based on whether they had articulation disorders or not, and we subsequently compared the DCDQ score (including the three subdivision factors: Control during Movement, Fine Motor and Handwriting, and General Coordination) between these two groups.
    Results: As for the total DCDQ score, in the group without misarticulation, 7 subjects showed a score under the standard value and the other 25 demonstrated a score equal to or higher than it. On the other hand, in the group with misarticulation, 14 subjects showed a score under the standard value and the other 15 demonstrated a score equal to or higher than it. There was a significant difference between the two groups (p<0.05). However, there was no significant difference between the two groups with regard to the subdivision factors of Control during Movement and Fine Motor and Handwriting. As for the subdivision factor of General Coordination, in the group without misarticulation, 5 subjects showed a score under the standard value and the other 27 demonstrated a score equal to or higher than it. On the other hand, in the group with misarticulation, 13 subjects showed a score under the standard value and the other 16 demonstrated a score equal to or higher than it. There was a significant difference between the two groups in this regard (p<0.05).
    Conclusions: These results suggest that not only local factors related to articulation, including velopharyngeal function and the form of the palate, but also impaired motor coordination cause the onset of articulation disorders in those with cleft palate.
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  • —Long-Term Follow-Up of 40 Individuals with Cleft Lip and Palate—
    Tomoe KIMURA, Akiko SATO, Reiko YOROZUYA, Masashi SATO, Fumio OHKUBO, ...
    2016Volume 41Issue 1 Pages 8-16
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    The purpose of this study was to investigate the speech outcomes of adult patients with repaired cleft lip and palate (CLP) via retrospective analysis of long-term, longitudinal follow-up data. The subjects were 40 patients (21 males and 19 females). Ten had a bilateral CLP, and 30 had a unilateral CLP. They had undergone a modified V-Y pushback method for primary palate repair at 1 year of age by multiple plastic surgeons on the Showa University Cleft Palate Team (SCPT) from 1978 to 1995. Perceived velopharyngeal function (VPF) and presence of articulation disorder were assessed by three experienced speech-language-hearing therapists using the Japanese Assessment of Cleft Palate Speech and the Japanese Assessment of Articulation. The age at speech evaluation was distributed among five periods: 3 to 5 years (I), 6 to 7 years (II), 9 to 12 years (III), 13 to 18 years (IV), and over 19 years (V). The results were as follows:
    1. Of the 40 patients, good/normal VPF was judged in 27 (67.5%) at I but in 19 (47.5%) at V. In contrast, slight VPD was judged in 6 (15.5%) at I and in 18 (45.0%) at V. 37 patients (92.5%) were considered as having velopharyngeal competence for daily conversation at V.
    2. A change in VPF at II or later was found in 31 patients (77.5%). The tendency of VPF deterioration was frequent at III and IV, but improvement was frequent at II and V.
    3. Normal articulation was judged in 18 (45.0%) at I. Articulation disorder was judged in 9 at II or later, but all the subjects acquired normal articulation again at V.
    4. 21 of 22 (95.5%) with articulation disorders had received speech therapy from I to II, and half of the subjects achieved normal articulation at III or later.
    5. Palatalized articulation was most frequent, with 11 at I and 6 at V.
    6. The number of patients judged with normal articulation was 28 (70.0%) at V.
    It is not possible to predict VPF at adulthood only from perceived VPF at childhood. From the results of this study, speech re-evaluation was considered to be necessary at age around 10, 16, and 20 at completion of treatment regardless of VPF at childhood.
    Acquisition of normal articulation in individuals with repaired CLP continued until adulthood after speech therapy at childhood. For this reason, continued follow-up and speech intervention should be effective for achieving normal articulation.
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  • Misuzu MATSUDA, Mihoko NAKANII, Yoshiko NISHIO, Mikihiko KOGO
    2016Volume 41Issue 1 Pages 17-23
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    【Purpose】 To identify the experiences of treatment among children with a cleft lip or palate who have undergone multiple surgeries, and to use the results as basic data to improve the treatment environment of patients and their families.
    【Subjects】 The study involved 12 subjects, 8 junior and 4 senior high school students. Seven and 5 were male and female, respectively, and 10 and 2 had a cleft palate and lip, respectively. The total number of surgeries ranged from 3 to 7.
    【Methods】 Semi-structured interviews were conducted to investigate the experiences of children who have undergone multiple surgeries, and their statements were analyzed using a qualitative and inductive approach, while defining “experiences” as impressive events and mental and physical conditions when undergoing surgery.
    【Results】 The experiences of the children were classified into 8 categories 《the continued presence of anxiety and a fear of surgery》 《expectations of recovery》 《postoperative pain and distress due to various limitations》 《management by medical professionals and support from others encouraged the children to undergo surgery》 《considerations for the mother feeling remorse》 《difficulty in dealing with bullying and teasing》 《not regarding the congenital deformity as special》 and 《being aware of the inevitable necessity of further surgeries.》
    【Discussion】 Healthcare providers must consider the effects of the initial experience of surgery on future events in children to provide detailed explanations and care, suggesting the need to effectively use preparation and distraction techniques. Also, the children received the impression that they had undergone unwanted surgery as they discovered that their mothers had a sense of guilt, suggesting the importance of providing support to help mothers accept their child's disease soon after children receive their diagnosis. It is also important to provide support to help children increase their self-care ability and describe their conditions in their own words, in order to cope with bullying.
    【Conclusions】 Healthcare providers must understand the characteristics of children with a cleft lip or palate requiring multiple surgeries, and improve the treatment environment to help them develop positive attitudes toward surgery.
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  • —Diagnosis and clinical manifestations—
    Rei OMINATO, Takanori KOBAYASHI, Yasumitsu KODAMA, Takahiro KOYAMA, Yu ...
    2016Volume 41Issue 1 Pages 24-30
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    We evaluated the characteristics of 84 patients with submucous cleft palate who visited the Oral and Maxillofacial Surgery clinic at the Niigata University Medical and Dental Hospital between 1982 and 2012. The characteristics included gender, body weight at birth, age at diagnosis, chief complaint, occasion of diagnosis, other congenital anomalies, Calnan's triad, our treatment strategy, and mother's age at delivery. We diagnosed submucous cleft palate from soft palate muscle diastasis.
    The results were as follows:
    1) The subjects enrolled in this study were 42 males and 42 females.
    2) The age at diagnosis ranged from 9 days postpartum to 49 years with an average of 4.6 years.
    3) The most common chief complaint was speech symptoms in 59 patients (70.2%).
    4) The most common occasions for diagnosis were pediatric clinics in 26 patients (31.0%) and dental clinics in 21 patients (25.0%).
    5) The most common congenital anomaly was mental retardation in 28 patients (33.3%).
    6) As to Calnan's triad of submucous cleft palate, all three findings were present in 62 patients (73.8%).
    7) We suggested that palatoplasty, to normalize the primary anatomic abnormality, was the first step in improving velopharyngeal function. There were 60 patients (71.4%) in the surgically treated group, and 24 patients (28.6%) in the non-surgically treated group.
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  • Kazuaki NISHIMURA, Shoko KOCHI, Kaoru IGARASHI
    2016Volume 41Issue 1 Pages 31-38
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    A clinicostatistical investigation was carried out on patients with cleft lip and/or palate in the Clinics for Maxillo-Oral Disorders, Tohoku University Hospital during the 20-year period from 1995 through 2014, and the following results were obtained.
    1. The total number of patients with cleft lip and/or palate was 1,428. The overall male to female ratio was 1 : 0.93.
    2. Regarding area of residence, 65.4% of patients were from Sendai city and Miyagi prefecture, a proportion which has increased in recent years.
    3. The distribution of cleft type was as follows: cleft lip and palate 40.0%, cleft lip and alveolus 30.0%, cleft palate 30.0%. Cleft lip and palate was more common in males than in females (P<0.01). Cleft palate was more common in females than in males (P<0.01).
    4. The peak age of the first visit was 0 years. The mean age of the first visit was 3 years, 11 months.
    5. The total number of primary patients with cleft lip and/or palate was 897 (male: 475, female: 422). The overall male to female ratio was 1 : 0.89. Regarding area of residence, 74.2% of primary patients were from Sendai city and Miyagi prefecture. Primary cleft lip and palate was more common in males than in females (P<0.01). Primary cleft palate was more common in females than in males (P<0.01). The mean age of the first visit in primary cases was 6 months.
    6. The number of patients with other associated congenital anomalies was 182, which was an incidence rate of 20.3%.
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  • Tsuyoshi TANABE, Kikuo KOZONO
    2016Volume 41Issue 1 Pages 39-43
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
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    False median cleft lip is often associated with dysplastic brain, epilepsy, and metabolic endocrine abnormalities. The condition is often associated with a poor life expectancy. However, several recent reports indicated good results from invasive surgical treatments. When general anesthesia is possible in cooperation with the pediatric department, surgery is implemented for epilepsy treatment and improvement of the patient's general condition. In this study, we reported our experience with a patient who underwent palatoplasty for a false median cleft lip and developed frequent epileptic seizures immediately after extubation, which caused glossoptosis and difficulty in mask ventilation. The patient was reintubated and required intensive care treatment.
    A 1-year 6-month-old male patient had had epileptic seizures from birth and false median cleft lip. Rhinocheiloplasty was performed at 4 months old. At the age of 1 year 10 months when palatoplasty was performed, he developed apnea after the operation. He was reintubated and required intensive care treatment, and subsequently underwent tracheostomy to keep his breathing stable.
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  • Yuka SHIGEMURA, Koichi UEDA, Takaya SENO, Mina MORIYA
    2016Volume 41Issue 1 Pages 44-50
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
    JOURNAL RESTRICTED ACCESS
    Background: Robin sequence is an etiologically nonspecific complex that could occur with various syndromes of known or unknown origin or in isolation. The disorder is derived from an underdevelopment of the mandible, which causes problems such as secondary glossoptosis and airway obstruction. The result brought by dyspnea is a dangerous problem. We experienced a case of mandibular distraction in a 6-year-old with serious airway obstruction.
    Case Report: The patient was the second of female twins delivered at 34 weeks. Her respiration was managed by tracheal intubation from birth. Tracheostomy was performed to correct her airway obstruction at 2 months of age. No further surgical treatments were performed on her micrognathia for the next 6 years, during which time no significant growth or improvement was observed in the affected area. When the 6-year-old patient was brought in for consultation, her Robin sequence was confirmed. She was suffering from Goldenhar syndrome. Head and neck CT showed the following: (1) hypoplasia and deficit of the mandible, (2) severe glossoptosis, and (3) airway constriction resulting from marked hyperplasia of granulation tissue surrounding the trachea incision site. 3D models were created for preoperative consideration before performing bilateral mandibular body osteotomy distraction.
    Results: Distraction was started at 1mm/day from postoperative day 4. On the final distraction (postoperative day 18), the patient's ability to make a sound was checked, when the tracheotomy hole was closed. On postoperative day 23, the patient's cannula was switched to a speech cannula for the first time and language training started.
    Conclusion: Although the next goal is extubation, much time will be required to train the vocal cords and respiratory muscles. It will not be an easy process because the patient's organs and muscles have not been used for six years; it will take a number of years to reach extubation, and we believe it must be done carefully. The patient may need additional distractions in the future.
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  • Hideto IMURA, Hiroo FURUKAWA, Katsutoshi KUBO, Teruyuki NIIMI, Toko HA ...
    2016Volume 41Issue 1 Pages 51-55
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
    JOURNAL RESTRICTED ACCESS
    Lipoma is a nonepithelial benign tumor composed of mature fat cells. The occurrence of lipoma in the head and neck region is about 2–8%. However, the occurrence of lipoma in the alveolar cleft region is rare. This report describes a case of lipoma in the alveolar cleft region. The patient was a nine-year-old male with unilateral cleft lip and palate. Cheiloplasty was operated at the age of 5 months and palatoplasty was operated at the age of 20 months. Panorama X-ray images taken at the age of nine years before the bone-grafting operation showed the alveolar cleft. Through CT, a low-density area was observed in the cleft alveolar region. It was diagnosed as unilateral alveolar cleft and tumor in the cleft region, thus resection of the tumor, alveolar plasty and bone grafting were performed. The pathological diagnosis was lipoma. The lipoma was successfully resected and the bone grafting operation was performed. The patient's progress remains fine 2 years after the operation.
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  • Chisato SAKUMA, Hideto IMURA, Teruyuki NIIMI, Yukiko MORITA, Toko HAYA ...
    2016Volume 41Issue 1 Pages 56-60
    Published: April 25, 2016
    Released on J-STAGE: May 13, 2016
    JOURNAL RESTRICTED ACCESS
    We report a case in which a Hotz plate improved respiratory disturbance.
    The patient was a 22-day-old male baby at the first examination. We diagnosed him as trisomy 21 with left cleft lip and palate. He had inspiratory stridor and insufficient oral intake, leading to him being fed 600 ml/day from a nasal tube. When we examined him the second time, he had cyanosis, retractive breathing and apnea. We decided that he needed hospital treatment, and he was admitted to our hospital. We used a Hotz plate in his mouth. We started to measure his arterial oxygen saturation SpO2 24 hours a day immediately after his admission. His arterial oxygen saturation SpO2 went down frequently after one day from the start of using the plate in his mouth. After 15 hours he could keep putting his tongue forward with the help of the plate and his respiration improved gradually. In addition, he could drink milk from a special feeding bottle designed for cleft lip and palate patients. However, he still could not take enough nutrition through oral intake only, so he was also placed under tube feeding. He was discharged as he showed improvement in respiration and sucking. He is 10 months old now and his respiration is stable. Recently, he started to eat baby food. We considered that the Hotz plate guided his tongue forward and kept it there, and the plate may have reduced his airway obstruction. Because the Hotz plate can greatly change the intra-oral condition, it might induce cyanosis temporarily. Therefore, it is necessary to pay close attention to respiration and to adjust the shape of the plate carefully depending on the case.
    We concluded that the respiration was likely improved by the Hotz plate in this case.
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