Evidence map›Paper›PMID 37460606›Full record

ArticleAnnals of hematology2023

Cytopenia: a report of haplo-cord transplantation in twin brothers caused by a novel germline GATA1 mutation and family survey.

Xing-Hua Sun, Qin Liu, Sheng-Nan Wu, Wu-Hen Xu, Kai Chen, Jing-Bo Shao, Hui Jiang

Abstract readCase Reports
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In one paragraph

Article in Annals of hematology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.3field-weighted citation impact, top 30% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 1 citations in OpenAlex.

  1. Article
  2. GATA1 in Normal and Pathologic Megakaryopoiesis and Platelet Development.Advances in experimental medicine and biology · 2024
    Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors at 1 institution in 1 country.

Xing-Hua Sun *Department of Hematology and Oncology, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, No. 24, Lane 1400, Beijing West Road, Jing'an District, Shanghai, 200040, China.
Qin Liu *Department of Hematology and Oncology, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, No. 24, Lane 1400, Beijing West Road, Jing'an District, Shanghai, 200040, China.
Sheng-Nan WuCentral Laboratory, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China.
Wu-Hen XuCentral Laboratory, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China.
Kai ChenDepartment of Hematology and Oncology, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, No. 24, Lane 1400, Beijing West Road, Jing'an District, Shanghai, 200040, China. shaojb@shildren.com.cn.ORCID http://orcid.org/0000-0002-9684-1511
Jing-Bo ShaoDepartment of Hematology and Oncology, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, No. 24, Lane 1400, Beijing West Road, Jing'an District, Shanghai, 200040, China.
Hui JiangDepartment of Hematology and Oncology, Shanghai Children's Hospital, School of Medicine, Shanghai Jiao Tong University, No. 24, Lane 1400, Beijing West Road, Jing'an District, Shanghai, 200040, China.
Shanghai Jiao Tong University · CN

Funding

Clinical Research Plan of SHDC SHDC2020CR4089Excellent Youth Talent Training Plan of Shanghai Children's Hospital 2021YQ01Research Foundation of Shanghai Municipal Health Commission 20194Y0112
6 · The paper itself

Abstract

Cytopenia due to the abnormal regulation of GATA1 could manifest as varying degrees of thrombocytopenia and/or anemia and more severely in male children than in female children. Here, we describe the case of pancytopenic and transfusion-dependent twin brothers at our center whose bone marrow puncture revealed low bone marrow hyperplasia. Whole-exome sequencing revealed that the twins had a new germline GATA1 mutation (nm_002049: exon 3:c.515 T >C:p.F172S), which confirmed the diagnosis of GATA1 mutation-related pancytopenia. The mutation was inherited from their mother, who was heterozygous for the mutation. Sanger sequencing verified the pathogenicity of the mutation. Further family morbidity survey confirmed that GATA1 mutation-related pancytopenia is an X-linked recessive genetic disorder. We developed haploid hematopoietic stem cell transplantation programs for twins, with the father as the only donor, and finally, the hematopoietic reconstruction was successful. Although they experienced acute graft-versus-host disease, hemorrhagic cystitis, and a viral infection in the early stage, no abnormal manifestations or transplant-related complications were observed 3 months after transplantation. Through hematopoietic stem cell transplantation technology for one donor and two receptors, we eventually cured the twins. The p.F172S variant in the new germline GATA1 mutation may play an essential role in the pathogenesis of GATA1 mutation-related cytopenia.

Indexed as

AnemiaGraft vs Host DiseaseHematopoietic Stem Cell TransplantationPancytopeniaThrombocytopeniaAnemia, Dyserythropoietic, CongenitalChildGATA1 Transcription FactorGenetic Diseases, X-LinkedHumansMaleMutationSiblingsGATA1 protein, humanGATA1 Transcription FactorAllogeneic hematopoietic stem cell transplantationCytopeniaFamily surveyGATA1Haploid

Identifiers

PMID37460606
OpenAlexW4384497953

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.