Guinamard Antoine, Clément Sylvain, Goemaere Sophie, Mary Alice, Riquet Audrey, Dellacherie Delphine
Univ. Lille, ULR 4072 - PSITEC - Psychologie : Interactions, Temps, Émotions, Cognition, Lille, France.
CHU Lille, Centre Régional de Diagnostic des Troubles d'Apprentissage, Lille, France.
Front Syst Neurosci. 2022 Aug 18;16:886427. doi: 10.3389/fnsys.2022.886427. eCollection 2022.
Developmental Cerebellar Anomalies (DCA) are rare diseases (e.g., Joubert syndrome) that affect various motor and non-motor functions during childhood. The present study examined whether music perception and production are affected in children with DCA. Sixteen children with DCA and 37 healthy matched control children were tested with the Montreal Battery for Evaluation of Musical Abilities (MBEMA) to assess musical perception. Musical production was assessed using two singing tasks: a pitch-matching task and a melodic reproduction task. Mixed model analyses showed that children with DCA were impaired on the MBEMA rhythm perception subtest, whereas there was no difference between the two groups on the melodic perception subtest. Children with DCA were also impaired in the melodic reproduction task. In both groups, singing performance was positively correlated with rhythmic and melodic perception scores, and a strong correlation was found between singing ability and oro-bucco-facial praxis in children with DCA. Overall, children with DCA showed impairments in both music perception and production, although heterogeneity in cerebellar patient's profiles was highlighted by individual analyses. These results confirm the role of the cerebellum in rhythm processing as well as in the vocal sensorimotor loop in a developmental perspective. Rhythmic deficits in cerebellar patients are discussed in light of recent work on predictive timing networks including the cerebellum. Our results open innovative remediation perspectives aiming at improving perceptual and/or production musical abilities while considering the heterogeneity of patients' clinical profiles to design music-based therapies.
发育性小脑异常（DCA）是罕见疾病（如乔伯特综合征），会影响儿童期的各种运动和非运动功能。本研究调查了DCA患儿的音乐感知和生成能力是否受到影响。16名DCA患儿和37名健康匹配对照儿童接受了蒙特利尔音乐能力评估量表（MBEMA）测试以评估音乐感知。音乐生成能力通过两项唱歌任务进行评估：音高匹配任务和旋律再现任务。混合模型分析表明，DCA患儿在MBEMA节奏感知子测试中表现受损，而在旋律感知子测试中两组之间没有差异。DCA患儿在旋律再现任务中也表现受损。在两组中，唱歌表现与节奏和旋律感知分数呈正相关，并且在DCA患儿中发现唱歌能力与口-颊-面部动作之间存在强相关性。总体而言，DCA患儿在音乐感知和生成方面均表现受损，尽管个体分析突出了小脑患者特征的异质性。这些结果从发育角度证实了小脑在节奏处理以及发声感觉运动环路中的作用。根据包括小脑在内的预测性定时网络的最新研究成果，讨论了小脑患者的节奏缺陷。我们的研究结果开辟了创新的补救方法，旨在提高音乐感知和/或生成能力，同时考虑患者临床特征的异质性以设计基于音乐的治疗方法。
