Clinical and molecular analysis of haemoglobin H disease in Sardinia: haematological, obstetric and cardiac aspects in patients with different genotypes

Br J Haematol. 2007 Jan;136(2):326-32. doi: 10.1111/j.1365-2141.2006.06423.x. Epub 2006 Nov 27.

Abstract

In this study, 251 Sardinian patients (187 adults and 64 children) with haemoglobin (Hb) H disease were evaluated. Two-hundred and sixteen patients (86%) had the deletional type (- -/-alpha) and 36 (14%) patients had the non-deletional type (- -/alpha(ND)alpha). A clear genotype-phenotype correlation was found, with the non-deletional type more severe than the deletional type. Diagnosis of Hb H disease was incidental in about 60% of cases. Aplastic crises due to B19 parvovirus infection were found in five patients (2.1%), while 23 patients (9.6%) experienced one or more haemolytic crises. Nineteen patients with Hb H received sporadic red blood cell transfusions and three patients were repeatedly transfused. Forty-seven of 61 married women (77%) had 82 pregnancies. In children, mean serum ferritin was 87 +/-92 mug/l and in adults, was 192 +/- 180 mug/l in females and 363 +/- 303 mug/l in males. For the 98 male patients, a significant correlation was found between ferritin values and age (r2 = 0.33, P < 0.0001). In the Sardinian population, Hb H disease needs regular monitoring for early detection and treatment of possible complications, such as worsening of anaemia that may require red cell transfusion, cholelithiasis and iron overload.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Anemia, Aplastic / virology
  • Blood Transfusion
  • Chi-Square Distribution
  • Child
  • Child, Preschool
  • Echocardiography
  • Female
  • Ferritins / analysis
  • Gene Deletion
  • Genotype
  • Hemoglobin H / analysis
  • Hemoglobin H / genetics*
  • Humans
  • Infant
  • Infant, Newborn
  • Iron Overload / complications
  • Italien
  • Male
  • Middle Aged
  • Parvoviridae Infections / blood
  • Parvoviridae Infections / complications
  • Parvovirus B19, Human
  • Phenotype
  • Polymerase Chain Reaction
  • Pregnancy
  • Pregnancy Complications / blood
  • Pregnancy Complications / genetics*
  • Pregnancy Complications / therapy
  • Transferrin / analysis
  • alpha-Thalassemia / blood
  • alpha-Thalassemia / genetics*
  • alpha-Thalassemia / therapy

Substances

  • Transferrin
  • Ferritins
  • Hemoglobin H