D V Nair Lal, Sivanesan Senthil Kumar, Kumar Devika S
Pediatrics, Saveetha Medical College and Hospital, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, IND.
Research and Development, Saveetha Medical College and Hospital, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, IND.
Cureus. 2024 Sep 13;16(9):e69356. doi: 10.7759/cureus.69356. eCollection 2024 Sep.
Introduction Autism spectrum disorder (ASD) is a lifelong disorder affecting children quite early in life, manifested as delays in communication and stereotypic behaviors. To date, it is diagnosed clinically using the Diagnostic and Statistical Manual-5 (DSM-V) criteria due to the lack of biomarkers that can specifically denote the disorder. The role of synucleins in this context has been considered due to the increasing evidence of neurodegeneration in many autistic children. Synucleins are a group of soluble neuronal proteins primarily expressed in the central nervous system. They are of three types: α-, β-, and ɣ-synuclein. α-synuclein is involved in vesicle trafficking and release of neurotransmitters. There is no uniformity in the scientific community regarding their levels of autism, with few studies showing increasing levels and others to the contrary. Hence, the present study was conceived to analyze the levels of α-synuclein and β-synuclein in autistic children and to correlate with the disease severity. Objectives The main objective of the study was to assess the levels of α- and β-synuclein in autistic children of 2-8 years of age and to identify the correlation between the severity of core symptoms of autism and α- and β-synuclein levels. It is intended to assess the possibility of using α- and β-synuclein/their ratio as a biomarker of the severity of autism. Materials and methods Plasma levels of α-synuclein and β-synuclein were measured in 160 ASD children and 40 healthy age and sex-matched children by ELISA. Their symptom severity was assessed with CARS-2 ST and the Indian Scale of Autism Assessment (ISAA). Values of α- and β-synuclein were analyzed for correlation with the severity rating of ASD. Cut-off values of α-synuclein and β-synuclein that discriminate the presence of autism and its severity were assessed using Jamovi 2.4.14 software. Results The results show that α-synuclein levels were significantly reduced (5.02 ± 0.586; range: 3.13-6.0 ng/ml) when compared with healthy controls (29.47 ± 18.62 ng/ml; range: 22.39- 36.56) with p < 0.001, and β-synuclein levels were elevated (1424 ng/ml ± 122; range: 1229-1616 ng/ml) when compared to control, though not significantly. Plasma levels of α-synuclein significantly correlate with disease severity with good diagnostic accuracy (86%), but β-synuclein levels did not correlate with severity. The fold changes of synucleins, especially the fold decrease in levels of α-synuclein, were discriminative for the diagnosis and severity with good sensitivity (93.6%), specificity (74.3%), positive predictive (92.6%), and negative predictive values (76.5%). The fold increase in β-synuclein did not have any significance in predicting the severity of autism. Conclusion The present study showed that α-synuclein and β-synuclein were associated with ASD and can be used to assess its severity. A fold decrease in α-synuclein was found to have good discriminating value in differentiating the severity of autism. It may be of use especially in mild and high-functioning autism, when clinically distinguishing them may be difficult.
引言
自闭症谱系障碍(ASD)是一种终身性疾病,在儿童早期就会对其产生影响,表现为沟通延迟和刻板行为。迄今为止,由于缺乏能够特异性指示该疾病的生物标志物,临床上使用《精神疾病诊断与统计手册》第五版(DSM-V)标准进行诊断。鉴于许多自闭症儿童中神经退行性变的证据越来越多,突触核蛋白在这种情况下的作用受到了关注。突触核蛋白是一组主要在中枢神经系统中表达的可溶性神经元蛋白。它们有三种类型:α-、β-和γ-突触核蛋白。α-突触核蛋白参与囊泡运输和神经递质释放。科学界对于它们在自闭症中的水平尚无统一认识,少数研究显示其水平升高,而其他研究则相反。因此,本研究旨在分析自闭症儿童中α-突触核蛋白和β-突触核蛋白的水平,并将其与疾病严重程度相关联。
目的
本研究的主要目的是评估2至8岁自闭症儿童中α-和β-突触核蛋白的水平,并确定自闭症核心症状的严重程度与α-和β-突触核蛋白水平之间的相关性。旨在评估将α-和β-突触核蛋白/其比值用作自闭症严重程度生物标志物的可能性。
材料与方法
通过酶联免疫吸附测定法(ELISA)测量了160名自闭症谱系障碍儿童和40名年龄、性别匹配的健康儿童血浆中α-突触核蛋白和β-突触核蛋白的水平。使用儿童孤独症评定量表第二版(CARS-2 ST)和印度自闭症评估量表(ISAA)评估他们的症状严重程度。分析α-和β-突触核蛋白的值与自闭症谱系障碍严重程度评分的相关性。使用Jamovi 2.4.14软件评估区分自闭症存在及其严重程度的α-突触核蛋白和β-突触核蛋白的临界值。
结果
结果显示,与健康对照组(29.47±18.62 ng/ml;范围:22.39 - 36.56)相比,α-突触核蛋白水平显著降低(5.02±0.586;范围:3.13 - 6.0 ng/ml),p<0.001;与对照组相比,β-突触核蛋白水平升高(1424 ng/ml±122;范围:1229 - 1616 ng/ml),但差异不显著。α-突触核蛋白的血浆水平与疾病严重程度显著相关,诊断准确性良好(86%),但β-突触核蛋白水平与严重程度无关。突触核蛋白的倍数变化,尤其是α-突触核蛋白水平的倍数降低,对诊断和严重程度具有判别意义,敏感性良好(93.6%)、特异性(74.3%)、阳性预测值(92.6%)和阴性预测值(76.5%)。β-突触核蛋白的倍数增加在预测自闭症严重程度方面没有任何意义。
结论
本研究表明,α-突触核蛋白和β-突触核蛋白与自闭症谱系障碍相关,可用于评估其严重程度。发现α-突触核蛋白的倍数降低在区分自闭症严重程度方面具有良好的判别价值。尤其在轻度和高功能自闭症中,当临床上难以区分时,它可能会有所帮助。