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幼年鸸鹋(新荷兰鸸鹋)III B型黏多糖贮积症(Sanfilippo B综合征)的病理和生化研究

Pathological and biochemical studies of mucopolysaccharidosis type IIIB (Sanfilippo syndrome type B) in juvenile emus (Dromaius novaehollandiae).

作者信息

Palmieri C, Giger U, Wang P, Pizarro M, Shivaprasad H L

机构信息

School of Veterinary Science, The University of Queensland, Gatton Campus, Gatton, QLD Australia.

Section of Medical Genetics, School of Veterinary Medicine, University of Pennsylvania, Philadelphia, PA, USA.

出版信息

Vet Pathol. 2015 Jan;52(1):160-9. doi: 10.1177/0300985814529314. Epub 2014 Apr 10.

Abstract

Mucopolysaccharidosis (MPS) type IIIB was diagnosed in 14 juvenile emus (Dromaius novaehollandiae), ages 3 weeks to 6 months, based on pathological and biochemical analyses. The animals had a history of neurological signs or sudden death; one of the birds with neurological signs and 3 others experienced acute hemoabdomen. Histopathologically, neuronal swelling and vacuolation in the cerebrum, cerebellum, brainstem, and spinal cord (80%-92%); retina (100%); autonomic ganglia of the intestine (71%); gizzard (50%); adrenal gland (27%); and ear (50%) were noted in affected but not healthy emus. Cytoplasmic vacuoles were also observed in the pancreas, liver, intestine, adrenal glands, and kidneys. The intracytoplasmic inclusions were periodic acid-Schiff and Luxol Fast Blue positive, consistent with a storage disease. Foamy macrophages infiltrated the liver, intestine, tunica media of the aorta, and spleen. By transmission electron microscopy, typical lamellated cytoplasmic bodies were detected in neurons of the brain and retina, while electron-dense bodies consistent with glycosaminoglycan inclusions were observed in hepatocytes and/or hepatic macrophages. The livers of the 2 affected emus studied contained large amounts of heparan sulfate, which is suggestive of MPS type III. Compared with normal controls, hepatic and serum α-N-acetylglucosaminidase activity was very low (<8% of control), while other enzyme activities were normal to increased in the 2 affected emus studied. Moreover, affected emus were homozygous for a 2-bp deletion in the NAGLU gene. This study characterizes the pathology of MPS type IIIB in emus, which is one of the rare inborn errors in birds, showing the homology of this condition to Sanfilippo syndrome in humans.

摘要

通过病理和生化分析,在14只3周龄至6个月大的幼年鸸鹋(Dromaius novaehollandiae)中诊断出IIIB型黏多糖贮积症(MPS)。这些动物有神经症状或猝死史;其中一只出现神经症状的鸟和另外3只经历了急性腹腔积血。组织病理学上,在患病但未患病的鸸鹋中,观察到大脑、小脑、脑干和脊髓(80%-92%)、视网膜(100%)、肠道自主神经节(71%)、砂囊(50%)、肾上腺(27%)和耳朵(50%)存在神经元肿胀和空泡化。在胰腺、肝脏、肠道、肾上腺和肾脏中也观察到细胞质空泡。胞质内包涵体对过碘酸希夫染色和卢氏固蓝染色呈阳性,与贮积病一致。泡沫巨噬细胞浸润肝脏、肠道、主动脉中膜和脾脏。通过透射电子显微镜观察,在大脑和视网膜的神经元中检测到典型的层状细胞质体,而在肝细胞和/或肝巨噬细胞中观察到与糖胺聚糖包涵体一致的电子致密体。所研究的2只患病鸸鹋的肝脏含有大量硫酸乙酰肝素,提示为III型MPS。与正常对照相比,所研究的2只患病鸸鹋的肝脏和血清α-N-乙酰葡糖胺酶活性非常低(<对照的8%),而其他酶活性正常或升高。此外,患病鸸鹋在NAGLU基因中存在一个2bp的缺失纯合子。本研究描述了鸸鹋IIIB型MPS的病理学特征,这是鸟类罕见的先天性疾病之一,显示了这种疾病与人类桑菲利波综合征的同源性。

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