Literature DB >> 217298

Juvenile amaurotic idiocy (neuronal ceroid lipofuscinosis) and lymphocyte fingerprint profiles.

R J Baumann, W R Markesbery.   

Abstract

Lymphocytes from 3 children with a form of juvenile amaurotic idiocy (characterized by retinal blindness, progressive dementia, and extrapyramidal motor disturbance) were studied by electron microscopy. Numerous fingerprint profiles (FP) were found in vacuolated lymphocytes from all 3 patients and an asymptomatic younger sibling of 1 patient who subsequently became symptomatic. We propose that the combination of this clinical picture and vacuolated lymphocytes with FP is sufficiently distinctive for clinical and research purposes until the biochemical defect is discovered. Wider utilization of ultrastructural study of lymphocytes should increase the number of children diagnosed and allow detection of asymptomatic patients.

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Year:  1978        PMID: 217298     DOI: 10.1002/ana.410040609

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  5 in total

1.  Blood lymphocytes in neuronal ceroid lipofuscinosis.

Authors:  A Simonati; N Rizzuto
Journal:  Ital J Neurol Sci       Date:  1988-06

2.  Fingerprint profiles in lymphocytic vacuoles of mucopolysaccharidoses I-H, II, III-A, and III-B.

Authors:  H H Goebel; K Ikeda; F Schulz; U Burck; A Kohlschütter
Journal:  Acta Neuropathol       Date:  1981       Impact factor: 17.088

3.  Blood film examination for vacuolated lymphocytes in the diagnosis of metabolic disorders; retrospective experience of more than 2,500 cases from a single centre.

Authors:  G Anderson; V V Smith; M Malone; N J Sebire
Journal:  J Clin Pathol       Date:  2005-12       Impact factor: 3.411

4.  Ultrastructural pathology of human lymphocytes in lysosomal disorders: a contribution to their morphological diagnosis.

Authors:  K Ikeda; H H Goebel; U Burck; A Kohlschütter
Journal:  Eur J Pediatr       Date:  1982-03       Impact factor: 3.183

5.  Homozygous TBC1 domain-containing kinase (TBCK) mutation causes a novel lysosomal storage disease - a new type of neuronal ceroid lipofuscinosis (CLN15)?

Authors:  Stefanie Beck-Wödl; Klaus Harzer; Marc Sturm; Rebecca Buchert; Olaf Rieß; Hans-Dieter Mennel; Elisabeth Latta; Axel Pagenstecher; Ursula Keber
Journal:  Acta Neuropathol Commun       Date:  2018-12-27       Impact factor: 7.801

  5 in total

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