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Longitudinal study of the development of obstruent correctness from ages 3 to 5 years in 108 Danish children with unilateral cleft lip and palate: a sub-study within a multicentre randomized controlled trial.

108 例丹麦单侧唇腭裂患儿 3 ~ 5 岁obstruent正确性发展的纵向研究: 一项多中心随机对照试验内的子研究。

  • 影响因子:1.96
  • DOI:10.1111/1460-6984.12508
  • 作者列表:"Jørgensen LD","Willadsen E
  • 发表时间:2020-01-01

BACKGROUND:Speech-sound development in preschoolers with unilateral cleft lip and palate (UCLP) as a group is delayed/disordered, and obstruents comprise the most vulnerable sound class. AIMS:To evaluate the development of obstruent correctness (PCC-obs) and error types (cleft speech characteristics (CSCs) and developmental speech characteristics (DSCs)) from ages 3-5 and to investigate possible predictors (error types, velopharyngeal dysfunction (VPD) and gender) of PCC-obs at age 5 in two groups of children with UCLP. METHODS & PROCEDURES:Subgroup analysis was conducted within a multicentre randomized controlled trial (RCT) of primary surgery (Scandcleft Project). A total of 125 Danish children with UCLP received lip and soft palate repair around 4 months of age and early hard palate closure at 12 months (EHPC group) or late hard palate closure at 36 months (LHPC group). Audio and video recordings of a naming test were available for 108 children at ages 3 and 5, and recordings were transcribed phonetically by blinded raters. OUTCOMES & RESULTS:PCC-obs scores increased significantly from ages 3-5 in both groups, but with small effect sizes in the EHPC group that had higher scores at age 3 than the LHPC group. DSCs decreased in both groups whereas CSCs only decreased in the LHPC group that had more CSCs at age 3 than the EHPC group. The frequency of CSCs at age 3 was a significant predictor of PCC-obs scores at age 5 in both groups. DSCs significantly improved the logistic regression model in the EHPC group, whereas VPD and gender did not significantly improve the model in either group. CONCLUSIONS & IMPLICATIONS:Although PCC-obs developed significantly from ages 3 to 5, children with UCLP as a group did not catch up to typically developing Danish children at age 5. Furthermore, the LHPC group at age 5 did not reach the 3-year level of the EHPC group, which means that delaying hard palate closure until age 3 is detrimental to obstruent development. Both CSCs and DSCs at age 3 were important predictors of PCC-obs at age 5 and should be considered when determining need for intervention.


背景: 单侧唇腭裂 (UCLP) 为群体的学龄前儿童语音发育延迟/紊乱,obstruct患者构成最脆弱的声音类别。 目的: 评估obstruent正确性 (PCC-obs) 和错误类型 (分裂语音特征 (CSCs) 和发育语音特征 (DSCs)) 的发展从 3-5 岁开始,调查两组UCLP患儿 5 岁时PCC-obs的可能预测因素 (错误类型、腭咽闭合功能障碍 (VPD) 和性别)。 方法和步骤: 在一项初次手术的多中心随机对照试验 (RCT) 中进行亚组分析。共有 12 5 名患有UCLP的丹麦儿童在 4 个月左右接受了唇和软腭修复,12 个月时接受了早期硬腭关闭 (EHPC组) 或 36 个月时硬腭关闭晚期 (LHPC组)。录音和录像命名test可供 108 名儿童年龄 3 和 5,录音转录语音通过盲评分员. 结局和结果: 两组PCC-obs评分从 3-5 岁开始显著增加,但EHPC组的效应量较小,3 岁时的评分高于LHPC组。两组DSCs均降低,而仅在 3 岁时CSCs多于EHPC组的LHPC组CSCs降低。3 岁时CSCs的频率是两组 5 岁时PCC-obs评分的显著预测因子。DSCs显著改善了EHPC组的logistic回归模型,而VPD和性别未显著改善两组的模型。 结论和意义: 尽管PCC-obs在 3 ~ 5 岁显著发展,但UCLP患儿在 5 岁时没有赶上典型发育中的丹麦儿童。此外,5 岁时的LHPC组没有达到EHPC组的 3 年水平,这意味着延迟硬腭关闭到 3 岁对obstruent发育是有害的。3 岁时的CSCs和DSCs都是 5 岁时PCC-obs的重要预测因子,在确定干预需求时应予以考虑。



作者列表:["Denadai R","Seo HJ","Lo LJ"]

METHODS:BACKGROUND:The anterior oronasal fistulae neighboring the alveolar cleft could persist or reappear after the alveolar reconstruction with cancellous bone grafting. The persistent symptomatic anterior oronasal fistulae need to be repaired, but surgery remains a challenge in cleft care. Surprisingly, this issue has rarely been reported in the literature. The purpose of this long-term study was to report a single surgeon experience with a therapeutic protocol for persistent symptomatic anterior oronasal fistula repair. METHODS:This is a retrospective study of consecutive patients with Veau type III and IV clefts and persistent symptomatic anterior oronasal fistulae managed according to a therapeutic protocol from 1997 to 2018. Depending on fistula size, patients were treated with local flaps associated with an interpositional graft or two-stage tongue flaps (small/medium or large fistulae, respectively). The surgical outcomes were classified as "good" (complete fistula closure with no symptoms), "fair" (asymptomatic narrow fistula remained), or "poor" (failure with persistent symptoms). RESULTS:Forty-four patients with persistent symptomatic anterior oronasal fistulae were reconstructed with local flaps associated with interpositional fascia or dermal fat grafting (52.3%) or two-stage tongue flaps (47.7%). Most of patients (93.2%) presented "good" outcomes, ranging from 87% to 100% (local and tongue flaps, respectively). Three (6.8%) patients presented symptomatic residual fistula ("poor" outcomes). CONCLUSIONS:For the repair of persistent symptomatic anterior oronasal fistulae, this therapeutic protocol provided satisfactory outcome with low fistula recurrence rate.

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作者列表:["Cleary B","Loane M","Addor MC","Barisic I","de Walle HEK","Matias Dias C","Gatt M","Klungsoyr K","McDonnell B","Neville A","Pierini A","Rissmann A","Tucker DF","Zurriaga O","Dolk H"]

METHODS:OBJECTIVE:Methadone is a vital treatment for women with opioid use disorder in pregnancy. Previous reports suggested an association between methadone exposure and Pierre Robin sequence (PRS), a rare craniofacial anomaly. We assessed the association between gestational methadone exposure and PRS. DESIGN/SETTING:This case-malformed control study used European Surveillance of Congenital Anomalies population-based registries in Ireland, the Netherlands, Italy, Switzerland, Croatia, Malta, Portugal, Germany, Wales, Norway and Spain, 1995-2011. PATIENTS:Cases included PRS based on International Classification of Disease (ICD), Ninth Edition-British Paediatric Association (BPA) code 75 603 or ICD, Tenth Edition-BPA code Q8708. Malformed controls were all non-PRS anomalies, excluding genetic conditions, among live births, fetal deaths from 20 weeks' gestation and terminations of pregnancy for fetal anomalies. An exploratory analysis assessed the association between methadone exposure and other congenital anomalies (CAs) excluding PRS. Methadone exposure was ascertained from medical records and maternal interview. RESULTS:Among 87 979 CA registrations, there were 127 methadone-exposed pregnancies and 336 PRS cases. There was an association between methadone exposure and PRS (OR adjusted for registry 12.3, 95% CI 5.7 to 26.8). In absolute terms, this association reflects a risk increase from approximately 1-12 cases per 10 000 births. A raised OR was found for cleft palate (adjusted OR 5.0, 95% CI 2.7 to 9.2). CONCLUSIONS:These findings suggest that gestational methadone exposure is associated with PRS. The association may be explained by unmeasured confounding factors. The small increased risk of PRS in itself does not alter the risk-benefit balance for gestational methadone use. The association with cleft palate, a more common CA, should be assessed with independent data.

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作者列表:["Jahanbin A","Eslami N","Torkamanzadeh N"]

METHODS::Orthopedic treatment to improve deficient maxillary growth of cleft lip and palate patients is an important part of treatment. The success of this treatment is strongly dependent on the time of initiation of therapy. There has been a large controversy in the available literature regarding the skeletal age of these patients. The aim of the present study was to compare the skeletal age of cleft lip and palate patients with normal individuals.37 unilateral and 14 bilateral cleft lip and palate patients and 47 healthy individuals participated in this cross-sectional study. The patients were classified into 8 to 10 and 11 to 14-year-old individuals. Cervical vertebral maturational stage of participants was evaluated in the lateral cephalometry. The skeletal age of cleft lip and palate patients was compared with normal controls. Chi-square was used for statistical analysis. There was not a significant difference in the skeletal developmental stage of unilateral and bilateral cleft compared to their normal peers according to their age and sex. Also, significant difference in skeletal maturational stage of cleft lip and palate patients was not found between boys and girls (P = 0.8). Similarly, no significant difference was found in the skeletal age of the 3 studied groups without considering the age and sex of participants (P = 0.5). Regarding the similar skeletal maturational stage of cleft lip and palate patients with normal controls in our study, their maxillofacial orthopedic treatment can be initiated at similar time to normal peers.

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