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PMID: 4352462 Published · ppublish English Journal Article

Hemoglobin Abraham Lincoln, beta32 (B14) leucine leads to proline. An unstable variant producing severe hemolytic disease.

The Journal of clinical investigation ·Vol. 52 ·No. 7 ·1973-07-00 ·Pages 1746-55

Honig GR, Green D, Shamsuddin M, Vida LN, Mason RG, Gnarra DJ, Maurer HS

Abstract

An unstable hemoglobin variant was identified in a Negro woman with hemolytic anemia since infancy. A splenectomy had been performed when the patient was a child. The anemia was accompanied by erythrocyte inclusion bodies and excretion of darkly pigmented urine. Neither parent of the proposita demonstrated any hematologic abnormality, and it appeared that this hemoglobin variant arose as a new mutation. Erythrocyte survival in the patient was greatly reduced: the erythrocyte t(1/2) using radiochromium as a tag was 2.4 days, and a reticulocyte survival study performed after labeling the cells with L-[(14)C]leucine indicated a t(1/2) of 7.2 days. When stroma-free hemolysates were heated at 50 degrees C, 16-20% of the hemoglobin precipitated. The thermolability was prevented by the addition of hemin, carbon monoxide, or dithionite, suggesting an abnormality of heme binding. An increased rate of methemoglobin formation was also observed after incubation of erythrocytes at 37 degrees C. The abnormal hemoglobin could not be separated from hemoglobin A by electrophoresis or chromatography, but it was possible to isolate the variant beta-chain by precipitation with p-hydroxymercuribenzoate. Purification of the beta-chain by column chromatography followed by peptide mapping and amino acid analysis demonstrated a substitution of proline for beta32 leucine. It appears likely that a major effect of this substitution is a disruption of the normal orientation of the adjacent leucine residue at beta31 to impair heme stabilization.

MeSH Terms
Adult Amino Acids/analysis Anemia, Hemolytic Carbon Isotopes Carbon Monoxide Chemical Precipitation Chromatography, Ion Exchange Chromium Isotopes Electrophoresis Erythrocytes Female Half-Life Heme Hemoglobins, Abnormal/analysis Hot Temperature Humans Hydroxymercuribenzoates Inclusion Bodies Leucine Methemoglobin/biosynthesis Proline Reticulocytes Sulfites
Chemicals
Amino Acids Carbon Isotopes Chromium Isotopes Hemoglobins, Abnormal Hydroxymercuribenzoates Sulfites Heme Carbon Monoxide Methemoglobin Proline Leucine
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Honig G R
Green D
Shamsuddin M
Vida L N
Mason R G
Gnarra D J
Maurer H S
References (39)
39 references, click to expand
  1. Hemoglobin sabine beta 91 (f 7) leu to pro. An unstable variant causing severe anemia with inclusion bodies.
    N Engl J Med. 1969 Apr 3;280(14):739-45 PMID: 5773354
  2. Improved method for the determination of blood glutathione.
    J Lab Clin Med. 1963 May;61:882-8 PMID: 13967893
  3. Electrophoresis of hemoglobin on agar gels; frequency of hemoglobin D in a Negro population.
    Bull Johns Hopkins Hosp. 1959 Aug;105(2):77-88 PMID: 13671273
  4. Mild "unstable haemoglobin haemolytic anaemia" caused by haemoglobin Shepherds Bush(B74(E18) gly--asp).
    Nature. 1970 Mar 7;225(5236):939-41 PMID: 5415129
  5. Peptides obtained by tryptic hydrolysis of performic acid-oxidized ribonuclease.
    J Biol Chem. 1956 Apr;219(2):623-42 PMID: 13319284
  6. The unstable hemoglobins--molecular and clinical features.
    Prog Hematol. 1971;7(0):69-109 PMID: 4950839
  7. Hereditary non-spherocytic haemolytic anaemia with post-splenectomy inclusion bodies and pigmenturia caused by an unstable haemoglobin Santa Ana-beta-88 (F4) leucine--proline.
    J Med Genet. 1968 Dec;5(4):292-7 PMID: 5713642
  8. Reticulocyte survival in sickle cell anemia: effect of cyanate.
    Blood. 1972 Nov;40(5):733-9 PMID: 5078929
  9. Haemoglobin Köln (beta-98 valine--methionine): an unstable protein causing inclusion-body anaemia.
    Nature. 1966 May 28;210(5039):915-6 PMID: 5960324
  10. Characterization of Heinz bodies in unstable haemoglobin haemolytic anaemia.
    Nature. 1972 Nov 17;240(5377):150-2 PMID: 4564987
  11. A simple visual screening test for glucose-6-phosphate dehydrogenase deficiency employing ascorbate and cyanide.
    N Engl J Med. 1966 May 26;274(21):1162-7 PMID: 5934952
  12. Diseases due to abnormalities of hemoglobin structure.
    Annu Rev Med. 1970;21:157-78 PMID: 4912473
  13. Mixing technique for the oxygen-hemoglobin equilibrium and Bohr effect.
    J Appl Physiol. 1966 Nov;21(6):1898-902 PMID: 5929323
  14. Variations in the structure of human haemoglobin. With particular reference to the unstable haemoglobins.
    Br Med Bull. 1969 Jan;25(1):14-23 PMID: 5782754
  15. Estimation of small percentages of foetal haemoglobin.
    Nature. 1959 Dec 12;184(Suppl 24):1877-8 PMID: 13800165
  16. Pure native globin from human hemoglobin: preparation and some physico-chemical properties.
    Biochim Biophys Acta. 1958 Apr;28(1):221 PMID: 13535712
  17. Exchange of heme among hemoglobin molecules.
    Proc Natl Acad Sci U S A. 1966 Sep;56(3):974-8 PMID: 5230192
  18. Mechanisms of Heinz body formation and attachment to red cell membrane.
    Semin Hematol. 1970 Jul;7(3):341-54 PMID: 5425759
  19. Haemoglobin Southampton, 106 (G8) Leu leads to pro: an unstable variant producing severe haemolysis.
    Lancet. 1972 Dec 2;2(7788):1170-2 PMID: 4117593
  20. Three-dimensional Fourier synthesis of horse oxyhaemoglobin at 2.8 A resolution: the atomic model.
    Nature. 1968 Jul 13;219(5150):131-9 PMID: 5659637
  21. Abnormal haem binding and globin SH group blockade in unstable haemoglobins.
    Nature. 1968 Jun 29;218(5148):1214-7 PMID: 4968903
  22. Idiopathic Heinz body anaemia: Hb-Bristol (beta67 (E11) Val to Asp).
    Br J Haematol. 1970 Apr;18(4):435-46 PMID: 5420592
  23. Unstable haemoglobin disease caused by Hb Santa Ana- 88 (F4) Leu leads to Pro.
    Haematologia (Budap). 1970;4(2):141-55 PMID: 5522602
  24. Hemoglobin Savannah (B6(24) beta-glycine is greater than valine): an unstable variant causing anemia with inclusion bodies.
    J Clin Invest. 1971 Mar;50(3):650-9 PMID: 5545125
  25. Haemoglobin Genova: beta-28 (B10) leucine replaced by proline.
    Nature. 1967 May 27;214(5091):877-9 PMID: 6054966
  26. Abnormal human haemoglobins. Separation and characterization of the alpha and beta chains by chromatography, and the determination of two new variants, hb Chesapeak and hb J (Bangkok).
    J Mol Biol. 1966 Aug;19(1):91-108 PMID: 5967288
  27. Hemoglobin Riverdale-Bronx an unstable hemoglobin resulting from the substitution of arginine for glycine at helical residue B6 of the B beta polypeptide chain.
    Biochem Biophys Res Commun. 1968 Dec 30;33(6):1004-5 PMID: 5705748
  28. Familial haemolytic anaemia with erythrocyte inclusion bodies, bilifuscinuria and abnormal haemoglobin (haemoglobin Galliera Genova).
    Br J Haematol. 1965 Sep;11(5):511-7 PMID: 5830022
  29. Hemoglobin-Bibba or alpha-2-136Pro-beta 2, an unstable alpha chain abnormal hemoglobin.
    Biochim Biophys Acta. 1968 Jan 22;154(1):220-2 PMID: 5639009
  30. CHARACTERIZATION OF GENETIC VARIANTS OF BLOOD PROTEINS.
    Vox Sang. 1965 May-Jun;10:359-62 PMID: 14332941
  31. Three families with unstable hemoglobinopathies (Köln, Olmsted and Santa Ana) causing hemolytic anemia with inclusion bodies and pigmenturia.
    Am J Med. 1969 Mar;46(3):344-59 PMID: 5780360
  32. Haemoglobin Hammersmith (beta-42 (CDI) Phe replaced by ser).
    Nature. 1967 Nov 18;216(5116):663-5 PMID: 6082463
  33. Studies on the heterogeneity of hemoglobin. 13. Chromatography of various human and animal hemoglobin types on DEAE-Sephadex.
    J Chromatogr. 1968 Feb 20;32(4):723-7 PMID: 5645562
  34. On the mechanism of the dissociation of haemoglobin.
    J Mol Biol. 1967 Apr 28;25(2):253-73 PMID: 6034102
  35. Protein measurement with the Folin phenol reagent.
    J Biol Chem. 1951 Nov;193(1):265-75 PMID: 14907713
  36. Dependence of the oxygen affinity of blood on the presence of foetal or adult haemoglobin.
    Nature. 1970 Jul 25;227(5256):388-90 PMID: 5428441
  37. Molecular pathology of human haemoglobin.
    Nature. 1968 Aug 31;219(5157):902-9 PMID: 5691676
  38. Hemoglobin Philly (beta 35 tyrosine phenylalanine): studies in the molecular pathology of hemoglobin.
    J Clin Invest. 1969 Sep;48(9):1627-42 PMID: 5822575
  39. CONGENITAL HEINZ-BODY ANAEMIA. FURTHER EVIDENCE ON THE CAUSE OF HEINZ-BODY PRODUCTION IN RED CELLS.
    Br J Haematol. 1964 Jul;10:281-90 PMID: 14179483
Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1973-07-00
Pages
1746-55
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC302450
Subset
IM
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