Cagnotti Giulia, Cantile Carlo, Chessa Stefania, Sacchi Paola, D'Angelo Antonio, Bellino Claudio
Department of Veterinary Science, University of Turin, Turin, Italy.
Department of Veterinary Sciences, University of Pisa, Pisa, Italy.
Front Vet Sci. 2020 Jun 23;7:348. doi: 10.3389/fvets.2020.00348. eCollection 2020.
Spinal muscular atrophy (SMA) is a motor neuron disease (MND) in humans and diverse animal species: canid, felid, and bovid. To date, bovine SMA has been reported in Brown Swiss, Holstein, Friesian, and Red Danish breed; it has been associated with a genetic mutation of the FVT1 gene, also known as 3-ketodihydrosphingosine reductase (KDSR). The aim of the present case series was to describe clinical presentation, pathological findings, and genetic analysis of five Blond d'Aquitaine calves diagnosed with SMA and to determine whether the mutation was associated with the disease. Five Blonde d'Aquitaine calves (three females and two males) from the same cow-calf operation farm were presented between June 2018 and February 2019 because unable to stand or walk unassisted since birth. Neurological examination aroused suspicion of a diffuse lesion affecting the peripheral nervous system in all calves. Findings from electromyographic investigations and muscle and nerve biopsies were consistent with a non-regenerative, chronic, active axonal neuropathy and marked neurogenic muscular atrophy and assumed to be associated with a neurodegenerative process. Histopathological examination of tissue samples from two animals revealed neuronal loss and several degenerated, shrunken, and hypereosinophilic neurons at the level of the ventral horn of the cervico-thoracic and the lumbo-sacral intumescence, diffuse loss of myelinated axons at the level of the ventral funiculi of all segments of the spinal cord, and moderate diffuse astrocytic reaction. These findings confirmed the diagnosis of SMA. No mutation of the FVT1 gene was found on genetic analysis. Further study into the causative gene mutation of SMA in Blonde D'Aquitaine calves is under way. Identification of a novel genetic mutation could improve our understanding of the disease in human medicine.
脊髓性肌萎缩症(SMA)是一种发生于人类以及多种动物物种(犬科、猫科和牛科动物)的运动神经元疾病。迄今为止,已在瑞士褐牛、荷斯坦牛、弗里西亚牛和丹麦红牛品种中报道了牛SMA;它与FVT1基因的基因突变有关,FVT1基因也被称为3-酮二氢鞘氨醇还原酶(KDSR)。本病例系列的目的是描述5头被诊断为SMA的阿基坦黄犊牛的临床表现、病理结果和基因分析,并确定该突变是否与疾病相关。2018年6月至2019年2月期间,来自同一奶牛犊牛养殖场的5头阿基坦黄犊牛(3头雌性和2头雄性)被送来就诊,因为它们自出生以来就无法独立站立或行走。神经学检查怀疑所有犊牛的外周神经系统受到弥漫性损伤。肌电图检查以及肌肉和神经活检的结果与非再生性、慢性、活动性轴索性神经病以及明显的神经源性肌萎缩一致,并假定与神经退行性过程有关。对两只动物的组织样本进行组织病理学检查发现,在颈胸段和腰骶膨大的腹角水平存在神经元丢失以及一些变性、萎缩和嗜酸性增强的神经元,脊髓所有节段腹侧索水平的有髓轴突弥漫性丢失,以及中度弥漫性星形细胞反应。这些发现证实了SMA的诊断。基因分析未发现FVT1基因的突变。对阿基坦黄犊牛SMA致病基因突变的进一步研究正在进行中。鉴定一种新的基因突变可能会增进我们对人类医学中该疾病的理解。