Literature DB >> 24723233

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

C Palmieri1, U Giger2, P Wang2, M Pizarro3, H L Shivaprasad4.   

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.
© The Author(s) 2014.

Entities:  

Keywords:  emu; histology; mucopolysaccharidosis type IIIB; nervous system; storage disease; transmission electron microscopy; α-N-acetylglucosaminidase

Mesh:

Substances:

Year:  2014        PMID: 24723233     DOI: 10.1177/0300985814529314

Source DB:  PubMed          Journal:  Vet Pathol        ISSN: 0300-9858            Impact factor:   3.157


  7 in total

1.  Mucopolysaccharidosis IIIB (Sanfilippo syndrome B) in a commercial emu (Dromaius novaehollandiae) flock.

Authors:  Seiche C Genger; Keijiro Mizukami; Michael P Martin; Jeffrey R Applegate; H John Barnes; Urs Giger
Journal:  Avian Pathol       Date:  2017-10-10       Impact factor: 3.378

2.  Central nervous system pathology in preclinical MPS IIIB dogs reveals progressive changes in clinically relevant brain regions.

Authors:  Martin T Egeland; Marta M Tarczyluk-Wells; Melissa M Asmar; Evan G Adintori; Roger Lawrence; Elizabeth M Snella; Jackie K Jens; Brett E Crawford; Jill C M Wait; Emma McCullagh; Jason Pinkstaff; Jonathan D Cooper; N Matthew Ellinwood
Journal:  Sci Rep       Date:  2020-11-23       Impact factor: 4.379

3.  Sleep-related hypermotor epilepsy in a patient with mucopolysaccharidosis type III.

Authors:  Anna A Abramova; Hrayr P Attarian; Snezhana M Dolgova; Alexandra I Belyakova-Bodina; Elena V Iakovenko; Amayak G Broutian
Journal:  Sleep Sci       Date:  2021 Jan-Mar

4.  Temporospatial Development of Neuropathologic Findings in a Canine Model of Mucopolysaccharidosis IIIB.

Authors:  Tyler A Harm; Shannon J Hostetter; Ariel S Nenninger; Bethann N Valentine; N Matthew Ellinwood; Jodi D Smith
Journal:  Vet Pathol       Date:  2020-11-18       Impact factor: 2.221

5.  A model of mucopolysaccharidosis type IIIB in pigs.

Authors:  Qiang Yang; Xueyan Zhao; Yuyun Xing; Chao Jiang; Kai Jiang; Pan Xu; Weiwei Liu; Jun Ren; Lusheng Huang
Journal:  Biol Open       Date:  2018-10-26       Impact factor: 2.422

6.  Is the eye a window to the brain in Sanfilippo syndrome?

Authors:  Helen Beard; Glyn Chidlow; Daniel Neumann; Nazzmer Nazri; Meghan Douglass; Paul J Trim; Marten F Snel; Robert J Casson; Kim M Hemsley
Journal:  Acta Neuropathol Commun       Date:  2020-11-17       Impact factor: 7.801

7.  A deletion of IDUA exon 10 in a family of Golden Retriever dogs with an attenuated form of mucopolysaccharidosis type I.

Authors:  Kiterie M E Faller; Alison E Ridyard; Rodrigo Gutierrez-Quintana; Angie Rupp; Celia Kun-Rodrigues; Tatiana Orme; Karen L Tylee; Heather J Church; Rita Guerreiro; Jose Bras
Journal:  J Vet Intern Med       Date:  2020-08-12       Impact factor: 3.333

  7 in total

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