Literature DB >> 508619

Inherited lack of transcobalamin II in serum and megaloblastic anaemia: a further patient.

J F Burman, D L Mollin, N A Sourial, R A Sladden.   

Abstract

We have studied a patient, unrelated to the patients previously described, with inherited lack of the vitamin B12 binding protein Transcobalamin II. Severe haematological abnormalities were found within a few weeks of birth and responded to treatment with both vitamin B12 and folic acid. He was maintained in partial remission with such treatment until adolescence, except for a time in early childhood when folic acid alone was given and he suffered severe neurological deterioration. A the age of 18 years he was admitted to hospital because of convulsions; the deoxyuridine suppression test showed intracellular deficiency of B12 despite a normal serum B12 and normal haemoglobin concentration. His serum failed to promote the uptake of radioactive B12 by bone marrow cells, and analysis of serum B12 binding proteins demonstrated the lack of Transcobalamin II. Treatment with injections of 1000 micrograms of B12 three times weekly corrected the abnormality shown in the deoxyuridine suppression test; following this treatment, together with changes in anticonvulsive therapy, he remains healthy without occurrence of further convulsions, and is haematologically normal.

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Year:  1979        PMID: 508619     DOI: 10.1111/j.1365-2141.1979.tb03716.x

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  13 in total

1.  Positive newborn screen for methylmalonic aciduria identifies the first mutation in TCblR/CD320, the gene for cellular uptake of transcobalamin-bound vitamin B(12).

Authors:  Edward V Quadros; Shao-Chiang Lai; Yasumi Nakayama; Jeffrey M Sequeira; Luciana Hannibal; Sihe Wang; Donald W Jacobsen; Sergey Fedosov; Erica Wright; Renata C Gallagher; Natascia Anastasio; David Watkins; David S Rosenblatt
Journal:  Hum Mutat       Date:  2010-08       Impact factor: 4.878

Review 2.  Transcobalamin II deficiency: case report and review of the literature.

Authors:  Y Kaikov; L D Wadsworth; C A Hall; P C Rogers
Journal:  Eur J Pediatr       Date:  1991-10       Impact factor: 3.183

3.  Hereditary transcobalamin II deficiency: a 22 year follow up.

Authors:  P K Thomas; A V Hoffbrand
Journal:  J Neurol Neurosurg Psychiatry       Date:  1997-02       Impact factor: 10.154

4.  Long-term follow up of patients with transcobalamin II deficiency.

Authors:  P T Monagle; G P Tauro
Journal:  Arch Dis Child       Date:  1995-03       Impact factor: 3.791

5.  Standardization of nomenclature for transcobalamin II variants.

Authors:  M Fràter-Schröder; H J Porck; A W Eriksson; S P Daiger; L L Cavalli-Sforza
Journal:  Hum Genet       Date:  1982       Impact factor: 4.132

Review 6.  Cellular uptake of cobalamin: transcobalamin and the TCblR/CD320 receptor.

Authors:  Edward V Quadros; Jeffrey M Sequeira
Journal:  Biochimie       Date:  2013-02-14       Impact factor: 4.079

7.  Neurological involvement in hereditary transcobalamin II deficiency.

Authors:  P K Thomas; A V Hoffbrand; I S Smith
Journal:  J Neurol Neurosurg Psychiatry       Date:  1982-01       Impact factor: 10.154

8.  Expression of transcobalamin II mRNA in human tissues and cultured fibroblasts from normal and transcobalamin II-deficient patients.

Authors:  N Li; S Seetharam; D S Rosenblatt; B Seetharam
Journal:  Biochem J       Date:  1994-07-15       Impact factor: 3.857

Review 9.  Genetic patterns of transcobalamin II and the relationships with congenital defects.

Authors:  M Fràter-Schröder
Journal:  Mol Cell Biochem       Date:  1983       Impact factor: 3.396

10.  Update on transcobalamin deficiency: clinical presentation, treatment and outcome.

Authors:  Y J Trakadis; A Alfares; O A Bodamer; M Buyukavci; J Christodoulou; P Connor; E Glamuzina; F Gonzalez-Fernandez; H Bibi; B Echenne; I Manoli; J Mitchell; M Nordwall; C Prasad; F Scaglia; M Schiff; B Schrewe; G Touati; M C Tchan; B Varet; C P Venditti; D Zafeiriou; C A Rupar; D S Rosenblatt; D Watkins; N Braverman
Journal:  J Inherit Metab Dis       Date:  2013-12-05       Impact factor: 4.982

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