Myelofibrosis is characterized by aberrant JAK/STAT signaling, with approved therapy including the JAK inhibitor ruxolitinib. Preclinical evidence implicates BCL-2 family proteins in MF pathogenesis and therapeutic response. Here, we evaluated baseline and on-treatment expression of BCL2, BCL2L1 (encoding BCL-xL), and MCL1 in 19 myelofibrosis patients receiving ruxolitinib. Quantitative PCR fold-change (FC) values, relative to healthy donors, revealed reduced baseline BCL2 (mean FC 0.15) and MCL1 (0.32) expression, with BCL2L1 showing a non-significant trend toward upregulation. Baseline BCL2 and BCL2L1 expression was significantly higher in patients achieving spleen response (responders; n = 7) compared to non-responders (BCL2: 0.30 vs 0.07, p = 0.0130; BCL2L1: 2.73 vs 0.52, p = 0.0096). Logistic regression confirmed both as independent predictors of response. We derived a combinatorial score (CS = FCBCL-2 * FCBCL2L1), which outperformed individual genes in response prediction, as confirmed in logistic regression analysis (OR, 7.5; p = 0.0028). ROC-defined cutoff (0.06) stratified patients by likelihood of response (OR 3.3, p = 0.0037). Longitudinal analysis showed no significant overall change in gene expression on ruxolitinib, but responders exhibited BCL2 down-regulation at loss of response (p = 0.0419). Overall, these preliminary findings suggest that BCL2 and BCL2L1 expression, individually and via a simple CS, predict response to ruxolitinib in myelofibrosis. While limited by small sample size and retrospective design, our data support prospective validation and exploration of BCL-2 pathway modulation as a therapeutic strategy.

Combinatorial BCL2/BCL2L1 expression predicts clinical response to ruxolitinib in myelofibrosis / Coltro, Giacomo; Videschi, Viola; Gesullo, Francesca; Violi, Federica; Balliu, Manjola; Vannucchi, Alessandro M.; Guglielmelli, Paola. - In: BIOMARKER RESEARCH. - ISSN 2050-7771. - ELETTRONICO. - 13:(2025), pp. 151.1-151.5. [10.1186/s40364-025-00865-0]

Combinatorial BCL2/BCL2L1 expression predicts clinical response to ruxolitinib in myelofibrosis

Coltro, Giacomo;Videschi, Viola;Gesullo, Francesca;Violi, Federica;Balliu, Manjola;Vannucchi, Alessandro M.;Guglielmelli, Paola
2025

Abstract

Myelofibrosis is characterized by aberrant JAK/STAT signaling, with approved therapy including the JAK inhibitor ruxolitinib. Preclinical evidence implicates BCL-2 family proteins in MF pathogenesis and therapeutic response. Here, we evaluated baseline and on-treatment expression of BCL2, BCL2L1 (encoding BCL-xL), and MCL1 in 19 myelofibrosis patients receiving ruxolitinib. Quantitative PCR fold-change (FC) values, relative to healthy donors, revealed reduced baseline BCL2 (mean FC 0.15) and MCL1 (0.32) expression, with BCL2L1 showing a non-significant trend toward upregulation. Baseline BCL2 and BCL2L1 expression was significantly higher in patients achieving spleen response (responders; n = 7) compared to non-responders (BCL2: 0.30 vs 0.07, p = 0.0130; BCL2L1: 2.73 vs 0.52, p = 0.0096). Logistic regression confirmed both as independent predictors of response. We derived a combinatorial score (CS = FCBCL-2 * FCBCL2L1), which outperformed individual genes in response prediction, as confirmed in logistic regression analysis (OR, 7.5; p = 0.0028). ROC-defined cutoff (0.06) stratified patients by likelihood of response (OR 3.3, p = 0.0037). Longitudinal analysis showed no significant overall change in gene expression on ruxolitinib, but responders exhibited BCL2 down-regulation at loss of response (p = 0.0419). Overall, these preliminary findings suggest that BCL2 and BCL2L1 expression, individually and via a simple CS, predict response to ruxolitinib in myelofibrosis. While limited by small sample size and retrospective design, our data support prospective validation and exploration of BCL-2 pathway modulation as a therapeutic strategy.
2025
13
1
5
Goal 3: Good health and well-being
Coltro, Giacomo; Videschi, Viola; Gesullo, Francesca; Violi, Federica; Balliu, Manjola; Vannucchi, Alessandro M.; Guglielmelli, Paola
File in questo prodotto:
File Dimensione Formato  
s40364-025-00865-0.pdf

accesso aperto

Tipologia: Pdf editoriale (Version of record)
Licenza: Creative commons
Dimensione 915.63 kB
Formato Adobe PDF
915.63 kB Adobe PDF

I documenti in FLORE sono protetti da copyright e tutti i diritti sono riservati, salvo diversa indicazione.

Utilizza questo identificatore per citare o creare un link a questa risorsa: https://hdl.handle.net/2158/1467137
Citazioni
  • ???jsp.display-item.citation.pmc??? ND
  • Scopus ND
  • ???jsp.display-item.citation.isi??? 0
social impact