Project ID CM-HD2026_26

ThemeCM-HD

Co Supervisor 1A Prof John Strouboulis Faculty of Life Sciences & Medicine, School of Cancer & Pharmaceutical Sciences, Comprehensive Cancer CentreEmail

Co Supervisor 1B Prof David Rees Faculty of Life Sciences & Medicine, School of Cancer & Pharmaceutical Sciences, Comprehensive Cancer CentreEmail

Third Supervisor Dr Barnaby Clark

Investigating the Role of Transcription Factor GATA1 in the Pathogenesis and Diagnosis of Diamond-Blackfan Anaemia

(a) Scientific basis
Mutations in ribosomal protein (RP) genes, coding for components of ribosomes (the protein production factories in cells), account for ~75% of Diamond-Blackfan Anaemia (DBA), a rare inherited syndrome characterised by severe anaemia due to a failure in the bone marrow to produce red blood cells. Two important questions arising are: (i) why do mutations in the ubiquitous RP genes specifically affect red blood cells, and (ii) what are the genetic mutations in the ~25% of undiagnosed cases?

We previously showed that GATA1, a key protein controlling all genes in red cells, contacts RP genes in red cells suggesting direct control by GATA1, which may help explain the anaemia caused by RP gene mutations. Moreover, possible mutations in GATA1 contact sites in RP genes, may account for (some of) the ~25% of undiagnosed cases. We thus abolished GATA1 contacts in two RP genes in red cells resulting in their downregulation. We also teamed up with the diagnostic lab at King’s College Hospital and the UK and French DBA Registries, to design and test a new diagnostic panel that includes all GATA1 contact sites in RP genes. In this PhD, the student will (i) characterise fully the cells with abolished GATA1 contacts in the two RP genes and (ii) analyse undiagnosed DBA samples using the new diagnostic panel.

(b) Techniques and skills:
– Cell culture, flow cytometry, molecular and cellular methods
– Polysome profiling by ultra-high performance liquid chromatography (uHPLC)
– Analysis of polysome-associated mRNAs (translatome) and RNA expression profiling by next generation sequencing (NGS)
– Proteomics by mass spectrometry
– Analysis of NGS and mass spectrometry data (bioinformatics)
– Electrophoretic mobility shift assays (EMSAs), CUT&RUN
– Gene editing by CRISPR/Cas9

(c) Overarching aims:
(i) detailed cellular, molecular and proteomic characterisation of cells with abolished GATA1 contacts in two RP genes (namely, RPS19 and RPL11);
(ii) screening of undiagnosed DBA samples for mutations in GATA1 contacts with RP genes, using a novel diagnostic panel;
(iii) Validation of prioritised mutations from (iii) using gene editing in an erythroid cell line.

(d) Objectives per year:
Year 1: Familiarisation with methods required for aim (i). Polysome profiling.
Year 2: Translatome, RNA expression profiling, proteomics; data analysis. Analysis by EMSAs/CUT&RUN of potential mutations from the diagnostic assay.
Year 3: Data analysis completion; first manuscript drafting. Gene editing of prioritised mutations from the diagnostic panel.
Year 4: Analysis of gene-edited cells; thesis writing; second manuscript drafting.

(e) Summary of a potential 3-month rotation project:
The student will culture RPS19 and RPL11 gene-edited cells and will check expression of the two genes at the RNA and protein levels. Will also prepare samples for polysome profiling.

Representative Publications

Co-Supervisor 1A

1. GATA1 and PU.1 Bind to Ribosomal Protein Genes in Erythroid Cells: Implications for Ribosomopathies, Amanatiadou EP, Papadopoulos GL, Strouboulis J, Vizirianakis IS, 2015, PLoS One, https://doi.org/10.1371/journal.pone.0140077
2. Reduced GATA1 levels are associated with ineffective erythropoiesis in sickle cell anemia, El Hoss S, Shangaris P, Brewin J, Psychogyiou ME, Ng, C, Pedler, L, Rooks, H, Gotardo EMF, Gushiken, LFC, Brito PL, Nicolaides KH, Conran N, Rees DC, Strouboulis J, 2025, Haematologica, https://doi.org/10.3324/haematol.2024.286010
3. Regulation of GATA1 levels in erythropoiesis, Gutierrez L, Caballero N, Fernandez-Calleja L, Karkoulia E, Strouboulis J, 2020, IUBMB Life, https://doi.org/10.1002/iub.2192

Co-Supervisor 1B

1. Males with sickle cell disease have higher risks of cerebrovascular disease, increased inflammation, and a reduced response to hydroxyurea. Di Mauro M, El Hoss S, Nardo-Marino A, Stuart-Smith S, Strouboulis J, Gibson JS, Rees DC, Brewin JN. 2023, Am J Hematol. doi: 10.1002/ajh.27074
2. The significance of spleen size in children with sickle cell anemia. Nardo-Marino A, Glenthøj A, Brewin JN, Petersen J, Braunstein TH, Kurtzhals JAL, Williams TN, Rees DC. 2022 Am J Hematol. doi: 10.1002/ajh.26703
3. A randomized, placebo-controlled, double-blind trial of canakinumab in children and young adults with sickle cell anemia. Rees DC, Kilinc Y, Unal S, Dampier C, Pace BS, Kaya B, Trompeter S, Odame I, Mahlangu J, Unal S, Brent J, Grosse R, Fuh BR, Inusa BPD, Koren A, Leblebisatan G, Levin C, McNamara E, Meiser K, Hom D, Oliver SJ.2022 Blood. doi: 10.1182/blood.2021013674.