Literature DB >> 3425320

Megaloblastic anemia and immune abnormalities in a patient with methionine synthase deficiency.

J Zittoun1, A Fischer, J Marquet, J L Pérignon, A Lagrue, C Griscelli.   

Abstract

We report a case of methionine synthase deficiency associated with cellular immune deficiency discovered in a 14-year-old boy. Principal findings were: developmental delay, recurrent upper and lower respiratory tract infections, megaloblastic anemia, discovered at 3 months of age, unresponsive to cyanocobalamin and poorly responsive to folinic acid. Biochemical studies showed: an abnormal deoxyuridine suppression test despite normal serum folate, cobalamin and transcobalamin levels; a normal intracellular uptake of these two coenzymes; and an absolute requirement of methionine for fibroblast growth, suggestive of defective methionine synthesis. An absence of methionine synthase activity in the patient's bone marrow and a profound depression of this activity in lymphocytes and liver were found. Hypergammaglobulinemia with variable lymphopenia, depressed lymphocyte transformation after lectin or recall-antigen stimulation, defective delayed-type hypersensitivity and decreased natural killer activity were noted as well. The patient died at the age of 14.

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Year:  1987        PMID: 3425320      PMCID: PMC7159520          DOI: 10.1111/j.1651-2227.1987.tb17284.x

Source DB:  PubMed          Journal:  Acta Paediatr Scand        ISSN: 0001-656X


  27 in total

1.  Studies on N5-methyltetrahydrofolate-homocystein methyltransferase in normal and leukemia leukocytes.

Authors:  R Peytremann; J Thorndike; W S Beck
Journal:  J Clin Invest       Date:  1975-11       Impact factor: 14.808

2.  Enzymatic activities on purine pyrimidine metabolism in nine mycoplasma species contaminating cell cultures.

Authors:  M Hamet; C Bonissol; P Cartier
Journal:  Clin Chim Acta       Date:  1980-04-11       Impact factor: 3.786

3.  Congenital methylmalonic aciduria--homocystinuria with megaloblastic anemia: observations on response to hydroxocobalamin and on the effect of homocysteine and methionine on the deoxyuridine suppression test.

Authors:  R Carmel; A A Bedros; J W Mace; S I Goodman
Journal:  Blood       Date:  1980-04       Impact factor: 22.113

4.  A simplified radioenzymatic assay for dihydrofolate reductase using [3H]dihydrofolate.

Authors:  S P Rothenberg; M P Iqbal; M Da Costa
Journal:  Anal Biochem       Date:  1980-03-15       Impact factor: 3.365

5.  Studies of a patient with megaloblastic anemia and an abnormal transcobalamin II.

Authors:  P A Seligman; L L Steiner; R H Allen
Journal:  N Engl J Med       Date:  1980-11-20       Impact factor: 91.245

6.  Homocystinuria and megaloblastic anemia responsive to vitamin B12 therapy. An inborn error of metabolism due to a defect in cobalamin metabolism.

Authors:  S Schuh; D S Rosenblatt; B A Cooper; M L Schroeder; A J Bishop; L E Seargeant; J C Haworth
Journal:  N Engl J Med       Date:  1984-03-15       Impact factor: 91.245

7.  Inactivation of methionine synthase by nitrous oxide.

Authors:  R Deacon; M Lumb; J Perry; I Chanarin; B Minty; M Halsey; J Nunn
Journal:  Eur J Biochem       Date:  1980-03

8.  Bone-marrow transplantation for inborn error of phagocytic cells associated with defective adherence, chemotaxis, and oxidative response during opsonised particle phagocytosis.

Authors:  A Fischer; P H Trung; B Descamps-Latscha; B Lisowska-Grospierre; I Gerota; N Perez; C Scheinmetzler; A Durandy; J L Virelizier; C Griscelli
Journal:  Lancet       Date:  1983-08-27       Impact factor: 79.321

9.  Effects of nitrous oxide-induced inactivation of cobalamin on methionine and S-adenosylmethionine metabolism in the rat.

Authors:  M Lumb; N Sharer; R Deacon; P Jennings; P Purkiss; J Perry; I Chanarin
Journal:  Biochim Biophys Acta       Date:  1983-04-20

10.  Effect of folate and cobalamin compounds on the deoxyuridine suppression test in vitamin B12 and folate deficiency.

Authors:  J Zittoun; J Marquet; R Zittoun
Journal:  Blood       Date:  1978-01       Impact factor: 22.113

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