Literature DB >> 448532

Late-onset nonketotic hyperglycinemia and spinocerebellar degeneration.

G S Steiman, M Yudkoff, P H Berman, B Blazer-Yost, S Segal.   

Abstract

Investigation of a 15-year old boy with progressive optic atrophy and spinocerebellar degeneration revealed elevated plasma, cerebrospinal fluid, and urine glycine concentrations. During an oral glycine loading test, the patient's plasma glycine concentration rose to a higher level than control values, although the initial rate of rise was slower; there was no concomitant rise in the plasma serine concentration. An oral serine loading test resulted in a prompt rise of both glycine and serine serum concentrations. The renal glycine clearance was elevated, and the renal tubular glycine reabsorption was diminished. These findings of decreased intestinal uptake and increased renal tubular glycine clearance suggest that a generalized derangement of glycine entry into cells may account for the phenotypic manifestations of the disorder.

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Year:  1979        PMID: 448532     DOI: 10.1016/s0022-3476(79)80211-5

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  9 in total

1.  Treatment of late-onset nonketotic hyperglycinaemia: effectiveness of imipramine and benzoate.

Authors:  E J Wiltshire; N K Poplawski; J R Harrison; J M Fletcher
Journal:  J Inherit Metab Dis       Date:  2000-02       Impact factor: 4.982

2.  Benzoate treatment and the glycine index in nonketotic hyperglycinaemia.

Authors:  J L K Van Hove; K Vande Kerckhove; J B Hennermann; V Mahieu; P Declercq; S Mertens; M De Becker; P S Kishnani; J Jaeken
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

Review 3.  Non-ketotic hyperglycinaemia: clinical and biochemical aspects.

Authors:  K Tada; K Hayasaka
Journal:  Eur J Pediatr       Date:  1987-05       Impact factor: 3.183

4.  Variant non ketotic hyperglycinemia is caused by mutations in LIAS, BOLA3 and the novel gene GLRX5.

Authors:  Peter R Baker; Marisa W Friederich; Michael A Swanson; Tamim Shaikh; Kaustuv Bhattacharya; Gunter H Scharer; Joseph Aicher; Geralyn Creadon-Swindell; Elizabeth Geiger; Kenneth N MacLean; Wang-Tso Lee; Charu Deshpande; Mary-Louise Freckmann; Ling-Yu Shih; Melissa Wasserstein; Malene B Rasmussen; Allan M Lund; Peter Procopis; Jessie M Cameron; Brian H Robinson; Garry K Brown; Ruth M Brown; Alison G Compton; Carol L Dieckmann; Renata Collard; Curtis R Coughlin; Elaine Spector; Michael F Wempe; Johan L K Van Hove
Journal:  Brain       Date:  2013-12-11       Impact factor: 13.501

5.  Difficulties in assessing the effect of strychnine on the outcome of non-ketotic hyperglycinaemia. Observations on sisters with a mild T-protein defect.

Authors:  E A Haan; D M Kirby; K Tada; K Hayasaka; D M Danks
Journal:  Eur J Pediatr       Date:  1986-09       Impact factor: 3.183

Review 6.  Hereditary spastic paraparesis in adults associated with inborn errors of metabolism: a diagnostic approach.

Authors:  F Sedel; B Fontaine; J M Saudubray; O Lyon-Caen
Journal:  J Inherit Metab Dis       Date:  2007-10-22       Impact factor: 4.982

7.  Nystagmus and subnormal electroretinographic response in nonketotic hyperglycinemia.

Authors:  S Hayasaka; T Setogawa; S Hara; S Ishizawa; K Hayasaka; K Tada
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  1987       Impact factor: 3.117

8.  GLRX5-associated [Fe-S] cluster biogenesis disorder: further characterisation of the neurological phenotype and long-term outcome.

Authors:  Bindu Parayil Sankaran; Sachin Gupta; Michel Tchan; Beena Devanapalli; Yusof Rahman; Peter Procopis; Kaustuv Bhattacharya
Journal:  Orphanet J Rare Dis       Date:  2021-11-03       Impact factor: 4.123

9.  Cerebrospinal fluid amino acids glycine, serine, and threonine in nonketotic hyperglycinemia.

Authors:  Michael A Swanson; Kristen Miller; Sarah P Young; Suhong Tong; Lina Ghaloul-Gonzalez; Juanita Neira-Fresneda; Lisa Schlichting; Cheryl Peck; Linda Gabel; Marisa W Friederich; Johan L K Van Hove
Journal:  J Inherit Metab Dis       Date:  2022-04-06       Impact factor: 4.750

  9 in total

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