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Hemoglobin H disease(HBH)

MedGen UID:
468531
Concept ID:
C3161174
Disease or Syndrome
Synonyms: ALPHA-THALASSEMIA, HEMOGLOBIN H TYPE; HBH; HEMOGLOBIN H DISEASE, DELETIONAL
SNOMED CT: Hemoglobin H disease (48553001)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Genes (locations): HBA1 (16p13.3); HBA2 (16p13.3)
 
Monarch Initiative: MONDO:0013512
OMIM®: 613978
Orphanet: ORPHA93616

Definition

Hemoglobin H disease is a subtype of alpha-thalassemia (see 604131) in which patients have compound heterozygosity for alpha(+)-thalassemia, caused by deletion of one alpha-globin gene, and for alpha(0)-thalassemia, caused by deletion in cis of 2 alpha-globin genes (summary by Lal et al., 2011). When 3 alpha-globin genes become inactive because of deletions with or without concomitant nondeletional mutations, the affected individual has only 1 functional alpha-globin gene. These people usually have moderate anemia and marked microcytosis and hypochromia. In affected adults, there is an excess of beta-globin chains within erythrocytes that will form beta-4 tetramers, also known as hemoglobin H (summary by Chui et al., 2003). Hb H disease is usually caused by the combination of alpha(0)-thalassemia with deletional alpha(+)-thalassemia, a combination referred to as 'deletional' Hb H disease. In a smaller proportion of patients, Hb H disease is caused by an alpha(0)-thalassemia plus an alpha(+)-thalassemia point mutation or small insertion/deletion. Such a situation is labeled 'nondeletional' Hb H disease. Patients with nondeletional Hb H disease are usually more anemic, more symptomatic, more prone to have significant hepatosplenomegaly, and more likely to require transfusions (summary by Lal et al., 2011). [from OMIM]

Clinical features

From HPO
Hepatomegaly
MedGen UID:
42428
Concept ID:
C0019209
Finding
Abnormally increased size of the liver.
Hemolytic anemia
MedGen UID:
1916
Concept ID:
C0002878
Disease or Syndrome
A type of anemia caused by premature destruction of red blood cells (hemolysis).
HbH hemoglobin
MedGen UID:
42397
Concept ID:
C0019026
Biologically Active Substance
Hemoglobin H (HbH) contains four beta-globin chains. It is normally not present at all in blood, but may make up about 1-40 percent of all hemoglobin in HbH disease, a subform of alpha thalassemia.
Reduced alpha/beta synthesis ratio
MedGen UID:
868731
Concept ID:
C4023136
Finding
A reduction in the ratio of production of alpha globin to that of beta globin. This is the major abnormality in the various forms of alpha thalassemia.
Splenomegaly
MedGen UID:
52469
Concept ID:
C0038002
Finding
Abnormal increased size of the spleen.

Professional guidelines

PubMed

Lin PC, Chang TT, Liao YM, Tsai SP, Chen YC, Hsu WY, Su HL, Zeng YS, Tseng YH, Chiou SS
Lab Med 2019 Apr 8;50(2):168-173. doi: 10.1093/labmed/lmy043. PMID: 30295867
Vichinsky E
Curr Med Res Opin 2016;32(1):191-204. Epub 2015 Nov 25 doi: 10.1185/03007995.2015.1110128. PMID: 26479125
Kemper AR, Knapp AA, Metterville DR, Comeau AM, Green NS, Perrin JM
J Pediatr 2011 May;158(5):780-3. Epub 2010 Dec 16 doi: 10.1016/j.jpeds.2010.10.042. PMID: 21167500

Curated

ACMG ACT, FA + High Barts Hb (>20-25% Barts) or FAB3), Alpha (a) Thalassemia: Hb H Disease

ACMG Algorithm, Hemoglobin FA + Barts: High Barts Alpha Thalassemia Screening Result (FAB3; FA + Barts > 20-25%), 2023

American College of Medical Genetics Newborn Screening ACT sheet, FA + Barts present (unquantified), FAB, Alpha Thalassemia, 2023

ACMG Algorithm, Hemoglobin FA + Barts: Unquantified Barts Alpha Thalassemia Screening Result (FAB), 2023

ACMG ACT, FA + Low/Moderate Barts Hb, FAB2, FAB1 Alpha Thalassemia: Silent carrier and alpha thalassemia trait, 2023

ACMG Algorithm, Hemoglobin FA + Barts: Low/Moderate Barts Alpha Thalassemia Screening Result (FAB2; FAB1), 2023

American College of Medical Genetics and Genomics, Newborn Screening ACT Sheet, FA + Barts Hb, Alpha (a) Thalassemia, 2012

American College of Medical Genetics and Genomics, Algorithm, Hb Screening (non-S), 2009

Recent clinical studies

Etiology

Musallam KM, Lombard L, Kistler KD, Arregui M, Gilroy KS, Chamberlain C, Zagadailov E, Ruiz K, Taher AT
Am J Hematol 2023 Sep;98(9):1436-1451. Epub 2023 Jun 26 doi: 10.1002/ajh.27006. PMID: 37357829
Lal A, Vichinsky E
Hematol Oncol Clin North Am 2023 Apr;37(2):327-339. doi: 10.1016/j.hoc.2022.12.004. PMID: 36907606
Lao TT
Best Pract Res Clin Obstet Gynaecol 2017 Feb;39:89-100. Epub 2016 Sep 23 doi: 10.1016/j.bpobgyn.2016.09.002. PMID: 28341055
Vichinsky EP
Cold Spring Harb Perspect Med 2013 May 1;3(5):a011742. doi: 10.1101/cshperspect.a011742. PMID: 23543077Free PMC Article
Muncie HL Jr, Campbell J
Am Fam Physician 2009 Aug 15;80(4):339-44. PMID: 19678601

Diagnosis

Musallam KM, Lombard L, Kistler KD, Arregui M, Gilroy KS, Chamberlain C, Zagadailov E, Ruiz K, Taher AT
Am J Hematol 2023 Sep;98(9):1436-1451. Epub 2023 Jun 26 doi: 10.1002/ajh.27006. PMID: 37357829
Lal A, Vichinsky E
Hematol Oncol Clin North Am 2023 Apr;37(2):327-339. doi: 10.1016/j.hoc.2022.12.004. PMID: 36907606
Lao TT
Best Pract Res Clin Obstet Gynaecol 2017 Feb;39:89-100. Epub 2016 Sep 23 doi: 10.1016/j.bpobgyn.2016.09.002. PMID: 28341055
Musallam KM, Rivella S, Vichinsky E, Rachmilewitz EA
Haematologica 2013 Jun;98(6):833-44. doi: 10.3324/haematol.2012.066845. PMID: 23729725Free PMC Article
Muncie HL Jr, Campbell J
Am Fam Physician 2009 Aug 15;80(4):339-44. PMID: 19678601

Therapy

Su JY, Chen Y, Chen HF, Tong JR, Wei YN, Huang LL, Deng L
Eur Rev Med Pharmacol Sci 2023 Feb;27(3):1027-1032. doi: 10.26355/eurrev_202302_31198. PMID: 36808349
Shash H
Medicina (Kaunas) 2022 Oct 21;58(10) doi: 10.3390/medicina58101496. PMID: 36295656Free PMC Article
Spencer-Chapman M, Luqmani A, Layton DM, Bain BJ
Am J Hematol 2018 Jul;93(7):963-964. Epub 2018 May 6 doi: 10.1002/ajh.25072. PMID: 29473200
Vichinsky E
Curr Med Res Opin 2016;32(1):191-204. Epub 2015 Nov 25 doi: 10.1185/03007995.2015.1110128. PMID: 26479125
Yin XL, Zhang XH, Wu ZK, Zhao DH, Zhou YL, Yu YH, Liu TN, Fang SP, Zhou TH, Wang L, Huang J
Acta Haematol 2013;130(3):153-9. Epub 2013 May 22 doi: 10.1159/000347177. PMID: 23711936

Prognosis

Musallam KM, Lombard L, Kistler KD, Arregui M, Gilroy KS, Chamberlain C, Zagadailov E, Ruiz K, Taher AT
Am J Hematol 2023 Sep;98(9):1436-1451. Epub 2023 Jun 26 doi: 10.1002/ajh.27006. PMID: 37357829
Leecharoenkiat K, Sornjai W, Khungwanmaythawee K, Paemanee A, Chaichana C, Roytrakul S, Fucharoen S, Svasti S, Smith DR
Dis Markers 2014;2014:340214. Epub 2014 Jun 15 doi: 10.1155/2014/340214. PMID: 25024506Free PMC Article
Vichinsky EP
Cold Spring Harb Perspect Med 2013 May 1;3(5):a011742. doi: 10.1101/cshperspect.a011742. PMID: 23543077Free PMC Article
Weatherall DJ
Blood Rev 2012 Apr;26 Suppl 1:S3-6. doi: 10.1016/S0268-960X(12)70003-6. PMID: 22631040
Kemper AR, Knapp AA, Metterville DR, Comeau AM, Green NS, Perrin JM
J Pediatr 2011 May;158(5):780-3. Epub 2010 Dec 16 doi: 10.1016/j.jpeds.2010.10.042. PMID: 21167500

Clinical prediction guides

Musallam KM, Lombard L, Kistler KD, Arregui M, Gilroy KS, Chamberlain C, Zagadailov E, Ruiz K, Taher AT
Am J Hematol 2023 Sep;98(9):1436-1451. Epub 2023 Jun 26 doi: 10.1002/ajh.27006. PMID: 37357829
Qiu Y, Wei S, Hou W, Lai K, Zhu H, Li W, Li Q, Yang Z, Shu Q, Chen P, Mo W
Hemoglobin 2023 Nov;47(2):52-55. Epub 2023 Jun 12 doi: 10.1080/03630269.2023.2219008. PMID: 37309066
Wiromrat P, Rattanathongkom A, Laoaroon N, Suwannaying K, Komwilaisak P, Panamonta O, Wongsurawat N, Nasomyont N
J Clin Densitom 2023 Jul-Sep;26(3):101379. Epub 2023 Apr 26 doi: 10.1016/j.jocd.2023.101379. PMID: 37147222
Paridar M, Azizi E, Keikhaei B, Takhviji V, Baluchi I, Khosravi A
Mol Biol Rep 2019 Oct;46(5):5041-5048. Epub 2019 Jul 4 doi: 10.1007/s11033-019-04955-9. PMID: 31273613
Leecharoenkiat K, Sornjai W, Khungwanmaythawee K, Paemanee A, Chaichana C, Roytrakul S, Fucharoen S, Svasti S, Smith DR
Dis Markers 2014;2014:340214. Epub 2014 Jun 15 doi: 10.1155/2014/340214. PMID: 25024506Free PMC Article

Recent systematic reviews

Musallam KM, Lombard L, Kistler KD, Arregui M, Gilroy KS, Chamberlain C, Zagadailov E, Ruiz K, Taher AT
Am J Hematol 2023 Sep;98(9):1436-1451. Epub 2023 Jun 26 doi: 10.1002/ajh.27006. PMID: 37357829
Kemper AR, Knapp AA, Metterville DR, Comeau AM, Green NS, Perrin JM
J Pediatr 2011 May;158(5):780-3. Epub 2010 Dec 16 doi: 10.1016/j.jpeds.2010.10.042. PMID: 21167500

Supplemental Content

Table of contents

    Clinical resources

    Practice guidelines

    • PubMed
      See practice and clinical guidelines in PubMed. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.
    • Bookshelf
      See practice and clinical guidelines in NCBI Bookshelf. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.

    Curated

    • ACMG ACT, 2023
      ACMG ACT, FA + High Barts Hb (>20-25% Barts) or FAB3), Alpha (a) Thalassemia: Hb H Disease
    • ACMG Algorithm, 2023
      ACMG Algorithm, Hemoglobin FA + Barts: High Barts Alpha Thalassemia Screening Result (FAB3; FA + Barts > 20-25%), 2023
    • ACMG ACT, 2023
      American College of Medical Genetics Newborn Screening ACT sheet, FA + Barts present (unquantified), FAB, Alpha Thalassemia, 2023
    • ACMG Algorithm, 2023
      ACMG Algorithm, Hemoglobin FA + Barts: Unquantified Barts Alpha Thalassemia Screening Result (FAB), 2023
    • ACMG ACT, 2023
      ACMG ACT, FA + Low/Moderate Barts Hb, FAB2, FAB1 Alpha Thalassemia: Silent carrier and alpha thalassemia trait, 2023
    • ACMG Algorithm, 2023
      ACMG Algorithm, Hemoglobin FA + Barts: Low/Moderate Barts Alpha Thalassemia Screening Result (FAB2; FAB1), 2023
    • ACMG ACT, 2012
      American College of Medical Genetics and Genomics, Newborn Screening ACT Sheet, FA + Barts Hb, Alpha (a) Thalassemia, 2012
    • ACMG Algorithm, 2009
      American College of Medical Genetics and Genomics, Algorithm, Hb Screening (non-S), 2009

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