Literature DB >> 37822

Ocular findings in metachromatic leukodystrophy. An electron microscopic and enzyme study in different clinical and genetic variants.

J Libert, F Van Hoof, D Toussaint, H Roozitalab, K R Kenyon, W R Green.   

Abstract

Histopathological studies of the eyes from three patients affected with the infantile form of metachromatic leukodystrophy (MLD) showed the storage of metachromatic complex lipids in the retinal ganglion cells, in the optic nerve and the ciliary nerves, as well as the storage of a mucopolysaccharide-like material in the nonpigmented epithelium of the ciliary body. The lesions were limited to the optic, ciliary, and sensory nerves in a fourth patient with the juvenile form of the disorder. These morphological aspects, which are probably related to differences in sulfatase A activities, may explain the variability of the ocular manifestations in metachromatic leukodystrophy. Seven children affected with infantile MLD or with mucosulfatidosis were examined by conjunctival biopsy. Typical lesions of the sensory nerves were obvious and allowed the diagnosis of the disease. However, it seemed impossible to separate the different forms by histopathological studies only. The tear enzymes were assayed in most of the cases and demonstrated a profound deficiency of arylsulfatase A, or of arylsulfatase A and B, in the classical MLD and in mucosulfatidosis, respectively.

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Year:  1979        PMID: 37822     DOI: 10.1001/archopht.1979.01020020157015

Source DB:  PubMed          Journal:  Arch Ophthalmol        ISSN: 0003-9950


  5 in total

Review 1.  Lacritin and other autophagy associated proteins in ocular surface health.

Authors:  Roy Karnati; Venu Talla; Katherine Peterson; Gordon W Laurie
Journal:  Exp Eye Res       Date:  2015-08-25       Impact factor: 3.467

2.  Thickening of the optic nerves in metachromatic leucodystrophy: A new MRI finding.

Authors:  Dylan Roi; Kshitij Mankad; Marios Kaliakatsos; Maureen Cleary; Adnan Manzur; Felice D'Arco
Journal:  Neuroradiol J       Date:  2016-02-25

Review 3.  Metachromatic leukodystrophy: a case of triplets with the late infantile variant and a systematic review of the literature.

Authors:  Asif Mahmood; Jay Berry; David A Wenger; Maria Escolar; Magdi Sobeih; Gerald Raymond; Florian S Eichler
Journal:  J Child Neurol       Date:  2009-12-28       Impact factor: 1.987

Review 4.  The efficacy of conjunctival biopsy as a screening technique in lysosomal storage disorders.

Authors:  M L Mazow
Journal:  Trans Am Ophthalmol Soc       Date:  1987

5.  New sites of ocular involvement in late-infantile metachromatic leukodystrophy revealed by histopathologic studies.

Authors:  I U Scott; W R Green; A K Goyal; Z de la Cruz; S Naidu; H Moser
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  1993-03       Impact factor: 3.117

  5 in total

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