Literature DB >> 7860732

Two missense mutations in the beta-globin gene can cause severe beta thalassemia. Hemoglobin Medicine Lake (beta 32[B14]leucine-->glutamine; 98 [FG5] valine-->methionine).

M B Coleman1, Z H Lu, C M Smith, J G Adams, A Harrell, M Plonczynski, M H Steinberg.   

Abstract

We studied the molecular basis of transfusion-dependent hemolytic anemia in an infant who rapidly developed the phenotype of beta thalassemia major. DNA sequence of one beta-globin gene of the proband revealed two mutations, one for the moderately unstable hemoglobin (Hb) Köln and another for a novel codon 32 cytosine-thymidine-guanine-->cytosine-adenine-guanine transversion encoding a leucine-->glutamine mutation. A hydrophilic glutamine residue at beta 32 has an uncharged polar side chain that could potentially distort the B helix and provoke further molecular instability. This new hemoglobin was called Hb Medicine Lake. Biosynthesis studies showed a deficit of beta-globin synthesis with early loss of beta-globin chains. An abnormal unstable hemoglobin, globin chain, or tryptic globin peptide was not present, demonstrating the extreme lability of this novel globin. Hb Medicine Lake mRNA was present, but an aberrantly spliced message was not. Absence of an abnormal beta-globin gene in the mother makes it likely that a de novo mutation occurred in the proband. The molecular pathogenesis of Hb Medicine Lake illustrates a mechanism whereby the phenotype of a genetic disorder, like the mild hemolytic anemia associated with a hemoglobinopathy, can be modulated by a coincident mutation in the same gene.

Entities:  

Mesh:

Substances:

Year:  1995        PMID: 7860732      PMCID: PMC295500          DOI: 10.1172/JCI117691

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  28 in total

1.  Primer-directed enzymatic amplification of DNA with a thermostable DNA polymerase.

Authors:  R K Saiki; D H Gelfand; S Stoffel; S J Scharf; R Higuchi; G T Horn; K B Mullis; H A Erlich
Journal:  Science       Date:  1988-01-29       Impact factor: 47.728

2.  Beta+-thalassemia in cis of a sickle cell gene: occurrence of a promoter mutation on a beta s chromosome.

Authors:  F Baklouti; R Ouazana; C Gonnet; A Lapillonne; J Delaunay; J Godet
Journal:  Blood       Date:  1989-10       Impact factor: 22.113

3.  Sickle cell anaemia in a septuagenarian.

Authors:  M H Steinberg
Journal:  Br J Haematol       Date:  1989-02       Impact factor: 6.998

4.  A new gene deletion in the alpha-like globin gene cluster as the molecular basis for the rare alpha-thalassemia-1(--/alpha alpha) in blacks: HbH disease in sickle cell trait.

Authors:  M H Steinberg; M B Coleman; J G Adams; R C Hartmann; H Saba; N P Anagnou
Journal:  Blood       Date:  1986-02       Impact factor: 22.113

5.  Abnormal processing of beta Knossos RNA.

Authors:  S H Orkin; S E Antonarakis; D Loukopoulos
Journal:  Blood       Date:  1984-07       Impact factor: 22.113

6.  Abnormal RNA processing due to the exon mutation of beta E-globin gene.

Authors:  S H Orkin; H H Kazazian; S E Antonarakis; H Ostrer; S C Goff; J P Sexton
Journal:  Nature       Date:  1982-12-23       Impact factor: 49.962

7.  [Hemoglobin Köln disease: familial hypochromic hemolytic anemia with hemoglobin anomaly].

Authors:  W Pribilla; P Klesse; K Betke; H Lehmann; D Beale
Journal:  Klin Wochenschr       Date:  1965-10-01

8.  Molecular analysis of the beta-thalassemia phenotype associated with inheritance of hemoglobin E (alpha 2 beta2(26)Glu leads to Lys).

Authors:  E J Benz; B W Berman; B L Tonkonow; E Coupal; T Coates; L A Boxer; A Altman; J G Adams
Journal:  J Clin Invest       Date:  1981-07       Impact factor: 14.808

9.  DNA sequencing with Thermus aquaticus DNA polymerase and direct sequencing of polymerase chain reaction-amplified DNA.

Authors:  M A Innis; K B Myambo; D H Gelfand; M A Brow
Journal:  Proc Natl Acad Sci U S A       Date:  1988-12       Impact factor: 11.205

10.  Hemoglobin Mississippi (beta 44ser----cys). Studies of the thalassemic phenotype in a mixed heterozygote with beta +-thalassemia.

Authors:  M H Steinberg; J G Adams; W T Morrison; D J Pullen; R Abney; A Ibrahim; R F Rieder
Journal:  J Clin Invest       Date:  1987-03       Impact factor: 14.808

View more
  3 in total

1.  Significance of each of three missense mutations, G484A, G667A, and G808A, present in an inactive allele of the human Lewis gene (FUT3) for alpha(1,3/1,4)fucosyltransferase inactivation.

Authors:  H Pang; Y Koda; M Soejima; H Kimura
Journal:  Glycoconj J       Date:  1998-10       Impact factor: 2.916

2.  Tertiary structural analysis of the elongated part of an abnormal hemoglobin, hemoglobin Pakse.

Authors:  Viroj Wiwanitkit
Journal:  Int J Nanomedicine       Date:  2006

3.  Molecular epidemiology, pathogenicity, and structural analysis of haemoglobin variants in the Yunnan province population of Southwestern China.

Authors:  Jie Zhang; Peng Li; Yang Yang; Yuanlong Yan; Xiaohong Zeng; Dongmei Li; Hong Chen; Jie Su; Baosheng Zhu
Journal:  Sci Rep       Date:  2019-06-04       Impact factor: 4.379

  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.