Department of Pathology, College of Veterinary Medicine, The University of Georgia, Athens, GA, USA.
Department of Pathology, College of Veterinary Medicine, and Department of Basic Sciences, AU/UGA Medical Partnership, The University of Georgia, Athens, GA, USA.
Adv Exp Med Biol. 2021;1348:325-335. doi: 10.1007/978-3-030-80614-9_15.
Though soft tissue disorders have been recognized and described to some detail in several types of domestic animals and small mammals for some years, they remain uncommon. Because of their low prevalence, not much progress has been made not only in improved diagnosis but also in our understanding of the biochemical basis and pathogenesis of these diseases in animals. Ehlers-Danlos syndrome (EDS) described in dogs already in 1943 and later in cats has only minor impact on the well-being of the dog as its effects on skin of these animals are rather limited. The involved skin is thin and hyperextensible with easily inflicted injuries resulting in hemorrhagic wounds and atrophic scars. Joint laxity and dislocation common in people are less frequently found in dogs. No systemic complications, such as organ rupture or cardiovascular problems which have devastating consequences in people have been described in cats and dogs. The diagnosis is based on clinical presentation and on light or electron microscopic features of disorganized and fragmented collagen fibrils. Several case of bovine and ovine dermatosparaxis analogous to human Ehlers-Danlos syndrome type VIIC were found to be caused by mutations in the procollagen I N-proteinase (pnPI) or ADAMTS2 gene, though mutations in other sites are likely responsible for other types of dermatosparaxis. Cattle suffering from a form of Marfan syndrome (MFS) were described to have aortic dilatation and aneurysm together with ocular abnormalities and skeletal involvement. As in people, mutations at different sites of bovine FBN1 may be responsible for Marfan phenotype. Hereditary equine regional dermal asthenia (HERDA), or hyperelastosis cutis, has been recognized in several horse breeds as affecting primarily skin, and, occasionally, tendons. A mutation in cyclophilin B, a chaperon involved in proper folding of collagens, has been identified in some cases. Warmblood fragile foal syndrome (WFFS) is another Ehlers-Danlos-like disorder in horses, affecting primarily Warmbloods who present with skin fragility and joint hyperextensibility. Degenerative suspensory ligament desmitis (DSLD) affects primarily tendons and ligaments of certain horse breeds. Data from our laboratory showed excessive accumulation of proteoglycans in organs with high content of connective tissues. We have identified increased presence of bone morphogenetic protein 2 (BMP2) in active foci of DSLD and an abnormal form of decorin in proteoglycan deposits. Our most recent data obtained from next generation sequencing showed disturbances in expression of genes for numerous proteoglycans and collagens.
虽然软组织疾病在一些家畜和小型哺乳动物中已经被认识和详细描述了几年,但它们仍然很少见。由于它们的发病率低,不仅在改善诊断方面没有取得多大进展,而且我们对这些疾病在动物中的生化基础和发病机制的了解也没有取得多大进展。1943 年在狗身上和后来在猫身上描述的埃勒斯-当洛斯综合征(EDS)对狗的健康影响很小,因为它对这些动物的皮肤影响有限。受累的皮肤薄而超伸展,容易造成损伤,导致出血性伤口和萎缩性疤痕。在人身上常见的关节松弛和脱位在狗身上则较少发现。在猫和狗身上没有描述过像人埃勒斯-当洛斯综合征 VIIC 那样的器官破裂或心血管问题等全身性并发症,这些并发症会产生毁灭性的后果。诊断基于临床表现和胶原纤维不规则和碎片化的光或电子显微镜特征。已经发现几种类似于人类埃勒斯-当洛斯综合征 VIIC 型的牛和绵羊的皮肌营养不良是由前胶原 I N-蛋白酶(pnPI)或 ADAMTS2 基因突变引起的,尽管其他部位的突变可能导致其他类型的皮肌营养不良。患有马凡氏综合征(MFS)形式的牛被描述为主动脉扩张和动脉瘤,同时伴有眼部异常和骨骼受累。与人一样,牛 FBN1 不同部位的突变可能导致马凡氏表型。遗传性马地区域性皮肤脆弱症(HERDA)或皮肤过度弹性症,已在几个马品种中被识别为主要影响皮肤,偶尔也影响肌腱。在一些病例中,已鉴定出参与胶原正确折叠的伴侣蛋白环孢素 B 的突变。热血脆弱驹综合征(WFFS)是马的另一种类似埃勒斯-当洛斯的疾病,主要影响热血马,表现为皮肤脆弱和关节过度伸展。退行性悬韧带黏液囊炎(DSLD)主要影响某些马品种的肌腱和韧带。我们实验室的数据显示,在富含结缔组织的器官中,糖胺聚糖过度积累。我们已经在 DSLD 的活跃病灶中发现了骨形态发生蛋白 2(BMP2)的过度表达,并在糖胺聚糖沉积物中发现了一种异常形式的核心蛋白聚糖。我们最近从下一代测序获得的数据显示,许多糖胺聚糖和胶原基因的表达发生了紊乱。