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一名患有血红蛋白病H的希腊男性患者同时存在阿格里尼奥血红蛋白和——地中海缺失:更严重的表型及文献综述

Concomitant Presence of Hb Agrinio and - -Med Deletion in a Greek Male Patient with Hemoglobinopathy H: More Severe Phenotype and Literature Review.

作者信息

Diamantidis Michael D, Pitsava Stefania, Zayed Omar, Argyrakouli Ioanna, Karapiperis Konstantinos, Chatzoulis Christos, Alexiou Evangelos, Manafas Achilles, Tsangalas Evangelos, Karakoussis Konstantinos

机构信息

Thalassemia and Sickle Cell Disease Unit, Department of Hematology, General Hospital of Larissa, 41221 Larissa, Greece.

Radiology Department, General Hospital of Larissa, 41221 Larissa, Greece.

出版信息

Hematol Rep. 2023 Aug 8;15(3):483-490. doi: 10.3390/hematolrep15030050.

DOI:10.3390/hematolrep15030050
PMID:37606495
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10443284/
Abstract

Hemoglobin (Hb) Agrinio is a rare non-deletional a-globin mutation observed almost exclusively in Greek, Spanish or other Mediterranean families. The clinical manifestations of a carrier of a single Hb Agrinio mutation (single heterozygosity) depend on the concomitant presence or absence of other mutations or variants in the beta, alpha or other modifying genes. We present a Greek patient harboring a Hb Agrinio variant plus the - -Med alpha deletional allele, having an infrequent severe form of alpha thalassemia, in contrast to the typical alpha thalassemic patient and requiring regular red blood cell (RBC) transfusions and chelation treatment. We also provide a concise literature review regarding alpha thalassemic hemoglobin variants and their molecular and clinical combinations. A phase 2, double-blind, randomized, placebo-controlled, multicenter clinical trial to determine the efficacy and safety of luspatercept (BMS-986346/ACE-536) for the treatment of anemia in adults with alpha thalassemia with the participation of our center is currently recruiting patients (NCT05664737).

摘要

血红蛋白(Hb)阿格里尼奥是一种罕见的非缺失型α-珠蛋白突变,几乎仅在希腊、西班牙或其他地中海家族中观察到。单一Hb阿格里尼奥突变携带者(单杂合子)的临床表现取决于β、α或其他修饰基因中其他突变或变异的伴存情况。我们报告了一名希腊患者,其携带Hb阿格里尼奥变异体以及--地中海α缺失等位基因,患有罕见的严重α地中海贫血,这与典型的α地中海贫血患者不同,需要定期进行红细胞(RBC)输血和螯合治疗。我们还提供了关于α地中海贫血血红蛋白变异体及其分子和临床组合的简要文献综述。一项由我们中心参与的2期、双盲、随机、安慰剂对照、多中心临床试验,旨在确定鲁司泊西(BMS-986346/ACE-536)治疗成人α地中海贫血贫血的疗效和安全性,目前正在招募患者(NCT05664737)。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3e6a/10443284/3872160aeecb/hematolrep-15-00050-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3e6a/10443284/a5c776f1d488/hematolrep-15-00050-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3e6a/10443284/3872160aeecb/hematolrep-15-00050-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3e6a/10443284/a5c776f1d488/hematolrep-15-00050-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3e6a/10443284/3872160aeecb/hematolrep-15-00050-g002.jpg

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Concomitant Presence of Hb Agrinio and - -Med Deletion in a Greek Male Patient with Hemoglobinopathy H: More Severe Phenotype and Literature Review.一名患有血红蛋白病H的希腊男性患者同时存在阿格里尼奥血红蛋白和——地中海缺失:更严重的表型及文献综述
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本文引用的文献

1
Homozygosity for the hyperunstable hemoglobin variant Hb Agrinio (HBA2:c.89T>C) leads to severe antenatal anemia: Eight new cases in three families.高不稳定血红蛋白变异体Hb Agrinio(HBA2:c.89T>C)的纯合性导致严重的产前贫血:三个家庭中的八例新病例。
Am J Hematol. 2022 Nov;97(11):E393-E395. doi: 10.1002/ajh.26687. Epub 2022 Sep 2.
2
Luspatercept for the treatment of anaemia in non-transfusion-dependent β-thalassaemia (BEYOND): a phase 2, randomised, double-blind, multicentre, placebo-controlled trial.芦可替尼治疗非输血依赖型β-地中海贫血(BEYOND)的贫血:一项 2 期、随机、双盲、多中心、安慰剂对照试验。
Lancet Haematol. 2022 Oct;9(10):e733-e744. doi: 10.1016/S2352-3026(22)00208-3. Epub 2022 Aug 22.
3
Α case of late diagnosis of compound heterozygosity for Hb Adana (HBA2:c.179G>A) in trans to an α+- thalassemia deletion: guilty or innocent.
1例Hb Adana(HBA2:c.179G>A)与α+地中海贫血缺失反式排列的复合杂合子迟发性诊断:有罪还是无罪。
Hippokratia. 2020 Jan-Mar;24(1):43-45.
4
A Phase 3 Trial of Luspatercept in Patients with Transfusion-Dependent β-Thalassemia.一项评估 luspatercept 治疗输血依赖型β地中海贫血患者的 3 期临床试验
N Engl J Med. 2020 Mar 26;382(13):1219-1231. doi: 10.1056/NEJMoa1910182.
5
Clinical Classification, Screening and Diagnosis for Thalassemia.地中海贫血的临床分类、筛查与诊断
Hematol Oncol Clin North Am. 2018 Apr;32(2):193-211. doi: 10.1016/j.hoc.2017.11.006.
6
First Cases of Hb Agrinio Described in Patients from the Republic of Macedonia.马其顿共和国患者中首次描述的阿格里尼奥血红蛋白病例。
Hemoglobin. 2017 Jul-Nov;41(4-6):308-310. doi: 10.1080/03630269.2017.1397016. Epub 2017 Dec 8.
7
Molecular basis of α-thalassemia.α地中海贫血的分子基础。
Blood Cells Mol Dis. 2018 May;70:43-53. doi: 10.1016/j.bcmd.2017.09.004. Epub 2017 Sep 21.
8
The α-thalassemias.α-地中海贫血症。
N Engl J Med. 2014 Nov 13;371(20):1908-16. doi: 10.1056/NEJMra1404415.
9
Study of three families with Hb Agrinio [α29(B10)Leu→Pro, CTG>CCG (α2)] in the Spanish population: three homozygous cases.
Hemoglobin. 2012;36(6):526-32. doi: 10.3109/03630269.2012.733988. Epub 2012 Oct 24.
10
Gene test review. Alpha-thalassemia.基因检测综述。α地中海贫血。
Genet Med. 2011 Feb;13(2):83-8. doi: 10.1097/GIM.0b013e3181fcb468.