El-Bassyouni Hala T, Hassan Nagwa, Mahfouz Inas, Abd-Elnaby Azza E, Mostafa Mostafa I, Tosson Angie M S
Department of Clinical Genetics, National Research Centre, Cairo, Egypt.
Department of Nutrition, National Research Centre, Cairo, Egypt.
J Pediatr Genet. 2019 Dec;8(4):179-186. doi: 10.1055/s-0039-1695042. Epub 2019 Aug 4.
Prader-Willi syndrome (PWS) is a distinct neurodevelopmental disorder associated with the deletion within the chromosomal 15q11-q13 region or uniparental disomy of chromosome 15. The etiologic heterogeneity of PWS makes it very difficult to establish uniform diagnostic methods which would result in the detection of most affected individuals. The objective was to report the clinical criteria and oro-dental features in PWS, to report the effect of diet and laser acupuncture on PWS and highlighted an easy effective method for early diagnosis of individuals with PWS. The study included seventeen cytogenetically proven individuals with Prader-Willi syndrome. These patients were subjected to meticulous history taking, clinical examination including oro-dental examination, bone densitometry and neuropsychiatric evaluation. They received laser acupuncture sessions in addition to nutrition intervention. All cases had characteristic facies, hypotonia and various psychosocial difficulties. Other criteria of PWS were present in different percentages. Karyotyping revealed deletion 15q11-q13 in 6 patients, and fluorescence in situ hybridization (FISH) revealed a microdeletion in 15q11-q13 in the other 11 patients. To our knowledge, partial ankyloglossia, median grooved tongue and hypodontia have not previously been reported in PWS patients. Laser acupuncture sessions and diet were effective in weight decline for PWS patients. Our study emphasizes the importance of early detection of PWS, laser sessions, diet restriction and oro-dental examination in the follow up of patients with Prader Willi syndrome.
普拉德-威利综合征(PWS)是一种独特的神经发育障碍,与染色体15q11-q13区域的缺失或15号染色体单亲二体性有关。PWS的病因异质性使得建立统一的诊断方法非常困难,而这种方法本可以检测出大多数受影响的个体。目的是报告PWS的临床标准和口腔牙齿特征,报告饮食和激光针灸对PWS的影响,并强调一种早期诊断PWS个体的简便有效方法。该研究纳入了17例经细胞遗传学证实的普拉德-威利综合征患者。这些患者接受了详细的病史采集、包括口腔牙齿检查在内的临床检查、骨密度测定和神经精神评估。除营养干预外,他们还接受了激光针灸治疗。所有病例都有特征性面容、肌张力减退和各种社会心理问题。PWS的其他标准以不同百分比存在。核型分析显示6例患者存在15q11-q13缺失,荧光原位杂交(FISH)显示另外11例患者存在15q11-q13微缺失。据我们所知,部分舌系带过短、正中沟舌和牙齿发育不全此前尚未在PWS患者中报道。激光针灸治疗和饮食对PWS患者体重下降有效。我们的研究强调了早期检测PWS、激光治疗、饮食限制和口腔牙齿检查在普拉德·威利综合征患者随访中的重要性。