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PMID: 14630445 Published · ppublish English Case Reports Letter Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Late presentation of dyskeratosis congenita as apparently acquired aplastic anaemia due to mutations in telomerase RNA.

Lancet (London, England) ·Vol. 362 ·No. 9396 ·2003-11-15 ·Pages 1628-30

Fogarty PF, Yamaguchi H, Wiestner A, Baerlocher GM, Sloand E, Zeng WS, Read EJ, Lansdorp PM, Young NS

Abstract

Aplastic anaemia in adults is usually acquired, but rarely constitutional types of bone marrow failure can occur late in life. We assessed two families with onset of pancytopenia in adults and detected two novel point mutations in the telomerase RNA gene (TERC) in each family. This gene is abnormal in some kindreds with dyskeratosis congenita. Individuals in our families with mutated TERC did not have physical signs of dyskeratosis congenita, and their blood counts were nearly normal, but all had severely shortened telomeres, reduced haemopoietic function, and raised serum erythropoietin and thrombopoietin. Bone marrow failure of variable severity due to dyskeratosis congenita, historically characterised by associated physical anomalies and early pancytopenia, may be present in otherwise phenotypically normal adults, and can masquerade as acquired aplastic anaemia.

MeSH Terms
Adult Anemia, Aplastic/etiology Dyskeratosis Congenita/diagnosis,genetics Female Hematopoietic Stem Cells/ultrastructure Humans Male Middle Aged Pancytopenia/etiology Pedigree Point Mutation RNA/genetics Telomerase/genetics
Chemicals
telomerase RNA RNA Telomerase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Fogarty Patrick F
Hematology Branch, National Heart, Lung, and Blood Institute, 10 Center Drive, MSC 1652, Bethesda, MD 20892-1652, USA.
Yamaguchi Hiroki
Wiestner Adrian
Baerlocher Gabriela M
Sloand Elaine
Zeng Weihua S
Read Elizabeth J
Lansdorp Peter M
Young Neal S
Article Info
Journal
Lancet (London, England)
Abbr.
Lancet
ISSN
1474-547X
Published
2003-11-15
Pages
1628-30
Language
English
Region
England
NLM ID
2985213R
Subset
IM
Grants
PHS HHS · R01A129524 · United States
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