Louis L. Sarliève

Centre National de la Recherche Scientifique

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P. Mandel

Université Libre de Bruxelles

G. Rebel

ASML (Netherlands)

Nenad Nešković

University of Osijek

G. Vincendon

Institut de Chimie

Catherine Fressinaud

Centre Hospitalier Universitaire d'Angers

Jack Puymirat

Université Laval

Véronique Ferret-Sena

Escola Superior de Saúde Egas Moniz

Jean‐Pierre Delaunoy

Hôpital Civil, Strasbourg

Dominique Baas

Université Claude Bernard Lyon 1

Keith Langley

Institut National de Recherche en Santé Publique

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Centre National de la Recherche Scientifique

100

Inserm

Institut de Chimie

Université de Strasbourg

Sorbonne Université

Laboratoire de Neurobiologie Cellulaire et Moléculaire

University of California, Los Angeles

Hôpitaux Universitaires de Strasbourg

Universidad Autónoma de Madrid

Université Paris Cité

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Phospholipid composition in late infantile neuronal ceroid lipofuscinosis

European Journal of Clinical Investigation (2000)

Luc‐André Granier Keith Langley Claude Leray Louis L. Sarliève

Neuronal ceroid lipofuscinosis (NCL) is a relatively common group of inherited neurodegenerative disorders characterised by the accumulation of autofluorescent lipopigments (ceroid) similar to lipofuscin. Because of this property, studies have concentrated on fatty acid metabolism and lipid peroxidation.In the present study, the fatty acid composition of phosphatidylcholine (PC) and phosphatidylethanolamine (PE) and the molecular species compositions of diacylglycerophosphocholine (diacyl GPC), diacylglycerophosphoethanolamine (diacyl GPE) and alkenylacyl GPE (plasmalogens) were investigated in cultured skin fibroblasts from three patients with a confirmed diagnosis of the late infantile form of the disease (LINCL, CLN2) and three healthy age-matched controls.Relatively minor differences in the fatty acid compositions of PC and PE were observed between patients and controls. However, dimethyl acetals of plasmalogens were found to be 40% higher in the patients compared to in the controls. Control and LINCL fibroblasts displayed only slight differences in the molecular compositions of diacyl GPE and diacyl GPC. In contrast, compared with normal cells, LINCL fibroblasts had higher levels of alkenylacyl GPE species containing both 18 : 1 and polyunsaturated fatty acids, but lower levels of species with 16 : 0 or 18 : 0 in the sn-1 position.The molecular composition of PC and PE subclasses in skin fibroblasts of healthy subjects and patients suffering from LINCL is here described for the first time. While few differences are noticeable in the fatty acid composition of PC and PE and the molecular species distribution of diacylGPC and diacylGPE, the alkenylacyl GPE (or ethanolamine plasmalogens) were found to differ significantly between patients and healthy controls.

Lipofuscin

Phosphatidylethanolamine

Plasmalogen

10.1046/j.1365-2362.2000.00757.x

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Citations (18)

Investigations on myelination in vitro. Regulation by thyroid hormone in cultures of dissociated brain cells from embryonic mice.

Journal of Biological Chemistry (1979)

Narayan R. Bhat Louis L. Sarliève G. S. R. Subba Rao Ronald A. Pieringer

Cultures of dissociated brain cells from embryonic mice were used to study the influence of thyroid hormone on myelination in vitro. Synthesis of myelin-associated lipids such as cerebrosides and sulfatides was used as an index for myelination. An experimental design, in which the cells were grown on medium containing serum from a thyroidectomized calf, was employed to demonstrate the direct effect of L-3,5,3'-triiodothyronine (T3Y on the biosynthesis of myelin lipids. The cells grown in the presence of hypothyroid calf serum which contained very low levels of thyroid hormones (T4 (thyroxine), 1.2 microgram/ml; T3, less than 25 ng/100 ml) compared to normal serum (T4, 5.8 microgram/ml; T3, 110 ng/100 ml) showed a diminished synthesis of myelin-associated glycolipids. This reduced activity could be restored to normal by including T3 (13 ng/ml) in the medium.

Microgram

10.1016/s0021-9258(19)83519-6

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Citations (105)

Neuronal and Glial Cell Cultures, a Tool for Investigation of Ganglioside Function

Advances in experimental medicine and biology (1980)

P. Mandel H. Dreyfus A.N.K. Yusufi Louis L. Sarliève Jacques Robert

Ganglioside

Lipid raft

Cell membrane

10.1007/978-1-4684-7844-0_45

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Citations (40)

Urinary glycolipids in Fabry's disease. Their examination in the detection of atypical variants and the pre-symptomatic state.

PubMed (1969)

Michel Philippart Louis L. Sarliève A Manacorda

Fabry's disease

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Some properties of brain PAPS-cerebroside sulphotransferase in Jimpy and Quaking mice.

PubMed (1972)

Louis L. Sarliève Nenad Nešković G. Rebel P. Mandel

Cerebroside

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Citations (42)

Expression of Thyroid Hormone Receptor Isoforms in the Oligodendrocyte Lineage

Neurochemical Research (2004)

Louis L. Sarliève Ángeles Rodríguez-Peña Keith Langley

Thyroid hormone receptor

Remyelination

10.1023/b:nere.0000021235.83952.9a

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Citations (38)

SULPHOLIPID METABOLISM IN DEVELOPING CHICKEN RETINA¹

Journal of Neurochemistry (1978)

H. Dreyfus Joanne Pieringer A.A. Farooqui S. Harth G. Rebel

Abstract— Glycolipid analysis of chicken retina and brain indicated the presence of cerebroside, cerebroside 3‐sulphate and sulphogalactosylglycerolipid In retina, the ratio of cerebroside to cerebroside 3‐sulphate was approximately half compared to brain. During chicken retina ontogenesis the ratio of cerebroside 3‐sulphate to sulphogalactosylglycerolipid increased rapidly and in the adult animal, the amount of cerebroside 3‐sulphate was 14 times higher than that of sulphogalactosylglycerolipid. The activity of PAPS: cerebroside sulphotransferase and arylsulphatase A in developing chicken retina indicated that the general ontogenic profiles of retinal PAPS: cerebroside sulphotransferase and arylsulphatase A were similar to those obtained for the brain. Both the enzymes showed the highest activity just before hatching. The significance of occurrence of sulpholipids in retina is discussed.

Cerebroside

10.1111/j.1471-4159.1978.tb07048.x

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Citations (17)

Announcement

Developmental Neuroscience (1989)

N Baumann Robert J. Desnick Stefano Di Donato Antonio Federico H. Galjaard

The aim of the meeting is to stress the discussion between clinicians and biologists on the biochemical pathogenesis of the late-onset neurometabolic disorders, to evidentiate clinical signs for an early diagnosis of the adult forms and the presence of minor clinical signs in carriers.The topics include: a general introduction to clinical, biochemical and neuropathological aspects of lysosomal and peroxisomal disorders with late onset; Gaucher; Fabry; Niemann-Pick; metachromatic

10.1159/000111919

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Citations (0)

Transient expression of 3,5,3′‐triiodothyronine nuclear receptors in rat oligodendrocytes: In vivo and in vitro immunocytochemical studies

Journal of Neuroscience Research (1994)

F. Besnard Min Luo M Miehe J. H. Dusssault Jack Puymirat

Abstract It is generally accepted that the action of thyroid hormones is mediated through specific nuclear receptors. Recent studies have demonstrated the homology of the thyroid receptor with the cellular product of the oncogen v‐erbA. So far, two genes have been identified and classified as α and β subtypes. In this study, the expression of nuclear triiodothyronine (T 3 ) receptors (NT 3 RS) was examined in secondary cultures containing 85–90% oligodendrocytes (OL) prepared from newborn rat brain primary cultures enriched in OL. These cultures, which are able to produce myelin membranes, were examined by double immunolabelling with a monoclonal antibody (2B3) raised against purified rat liver NT 3 Rs and with antibodies against two maturation markers of OL: an early marker, galactocerebroside (GC), and myelin basic protein (MBP), which is expressed later than GC. 2B3 recognized three nuclear proteins with the same molecular weights as β, α1, and α2 sybtypes with different capacities for binding T 3 . In 5‐day ‐old OL secondary cultures (25 days, total time in culture), 2B3‐NT 3 R immunoreactivity was located in 77% of morphologically immature OL (GC) + cells, whereas only 44% of morphologically mature OL were immunoreactive. Only 35% of the MBP + cells co‐expressed NT 3 Rs. In the corpus callosum of devloping rat brain, at all ages studied from 7–60 days postnatal, the total absence of NT 3 Rs in dark OL (morphologically mature), confirmed by ultrastructural immunocytochemistry, indicates an even more dramatic decrease during maturation. Furthermore, the percentage of medium OL (less mature) stained by 2B3 is reduced by approximately half in 60‐ compared to 20‐day‐old rat brain. It is of interest to note that the in vitro observation with maturation markers mirrors the in vivo decrease of NT 3 R expression during development. It is intersting that NT 3 Rs are absent in vivo before the critical period of active myelination. These data indicate the presence of a nuclear T 3 binding protein in the nuclei of OL at the time of myelination both in vitro and in vivo. The transient expression of these NT 3 Rs during active myelination argues in favour of a direct effect of thyroid hormones on OL. © 1994 Wiley‐Liss, Inc.

Galactocerebroside

Thyroid hormone receptor

10.1002/jnr.490370304

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Citations (16)

Molecular species of choline and ethanolamine glycerophospholipids in rat brain myelin during development

Lipids (1994)

Claude Leray Louis L. Sarliève H. Dreyfus R. Massarelli Luciano Binaglia

The composition of the molecular species of various phospholipid subclasses was examined in myelin isolated from brain of 15-, 21- and 90-day-old rats. The molecular species of diacylglycerophosphocholine (PtdCho), diacylglycerophosphoethanolamine (PtdEtn) and plasmenyl-ethanolamine (PlsEtn) were quantified by high-performance liquid chromatography (HPLC) after phospholipase C treatment and dinitrobenzoyl derivatization. In rat brain myelin, each phospholipid subclass showed a specific pattern of molecular species that changed during development. PtdCho contained large amounts of saturated/monounsaturated and disaturated species and low amounts of saturated/polyunsaturated species. During brain development, the levels of saturated/monounsaturated molecular species increased whereas those of the disaturated and saturated/polyunsaturated species decreased. PtdEtn were characterized by their low levels of disaturated species and a high content of saturated/monounsaturated and saturated/polyunsaturated species, of which those containing fatty acids of the n-3 series decreased, whereas those containing fatty acids of the n-6 series did not change during brain development. The levels of saturated/monounsaturated species increased in PtdEtn. No disaturated molecular species could be detected in PlsEtn. This alkenylacyl subclass contained large amounts of saturated/polyunsaturated, saturated/monounsaturated and dimonounsaturated molecular species. During development, the levels of saturated/polyunsaturated molecular species decreased while those of the two others increased. The data indicated that myelin sheaths undergo phospholipid changes during brain development and maturation.

Glycerophospholipids

Lipidology

Choline

Neurochemistry

Brain Development

10.1007/bf02537095

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Citations (15)