A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis

Myelofibrosis (MF) is a progressive disease characterized by accumulation of extracellular matrix (ECM) in the bone marrow (BM) which impairs normal hematopoiesis. While Janus kinase (JAK) inhibitors reduce spleen volume and provide symptomatic improvement, they do not resolve BM fibrosis and may c...

Full description

Saved in:
Bibliographic Details
Main Authors: Pankit Vachhani, Peter Tan, Anne-Marie Watson, Shang-Ju Wu, Ross Baker, Stanley Cheung, Sung-Eun Lee, Chih-Cheng Chen, Tsai-Yun Chen, Hui-Hua Hsiao, Jae Hoon Lee, Lucia Masarova, Shuh Ying Tan, Jana Baskar, Brett Charlton, Alison Findlay, Dieter Hamprecht, Wolfgang Jarolimek, Joanna Leadbetter, John Miller, Kristen Morgan, Amna Zahoor, Gabriela Hobbs
Format: Article
Language:English
Published: Ferrata Storti Foundation 2025-04-01
Series:Haematologica
Online Access:https://haematologica.org/article/view/12032
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850147136740524032
author Pankit Vachhani
Peter Tan
Anne-Marie Watson
Shang-Ju Wu
Ross Baker
Stanley Cheung
Sung-Eun Lee
Chih-Cheng Chen
Tsai-Yun Chen
Hui-Hua Hsiao
Jae Hoon Lee
Lucia Masarova
Shuh Ying Tan
Jana Baskar
Brett Charlton
Alison Findlay
Dieter Hamprecht
Wolfgang Jarolimek
Joanna Leadbetter
John Miller
Kristen Morgan
Amna Zahoor
Gabriela Hobbs
author_facet Pankit Vachhani
Peter Tan
Anne-Marie Watson
Shang-Ju Wu
Ross Baker
Stanley Cheung
Sung-Eun Lee
Chih-Cheng Chen
Tsai-Yun Chen
Hui-Hua Hsiao
Jae Hoon Lee
Lucia Masarova
Shuh Ying Tan
Jana Baskar
Brett Charlton
Alison Findlay
Dieter Hamprecht
Wolfgang Jarolimek
Joanna Leadbetter
John Miller
Kristen Morgan
Amna Zahoor
Gabriela Hobbs
author_sort Pankit Vachhani
collection DOAJ
description Myelofibrosis (MF) is a progressive disease characterized by accumulation of extracellular matrix (ECM) in the bone marrow (BM) which impairs normal hematopoiesis. While Janus kinase (JAK) inhibitors reduce spleen volume and provide symptomatic improvement, they do not resolve BM fibrosis and may cause or exacerbate anemia and thrombocytopenia. An anti-fibrotic therapy capable of normalising BM microenvironment and function remains a significant gap in the current treatment landscape. MF is associated with elevated expression of lysyl oxidases; enzymes responsible for maturation of the most abundant ECM proteins, collagen and elastin. PXS-5505 is a novel pan-lysyl oxidase inhibitor designed to exert an anti-fibrotic mode of action by preventing the cross-linking of collagen and elastin. PXS5505-MF-101 is a multi-center phase 1/2a study of PXS-5505 in MF patients which included a dose escalation phase (DEP) and a cohort expansion phase (CEP). Primary objectives were to determine the safety and tolerability of PXS-5505 and to define dosing for future studies. The DEP demonstrated that the highest dose tested, 200 mg BID, was safe and achieved robust systemic reduction of lysyl oxidase activity; this dose was therefore selected for the CEP. Twentyfour patients (median age 72 years) with relapsed or refractory MF were recruited into the CEP and 54% (13/24) completed 24 weeks of treatment. PXS-5505 was well tolerated and reached steady state concentrations by Day 28. Over the 24-week treatment period preliminary indications of clinical efficacy, including a reduction in BM collagen, were evident. Overall, these data support continued investigation of PXS-5505.
format Article
id doaj-art-fe053b40b57d433982b24e0088937fa0
institution OA Journals
issn 0390-6078
1592-8721
language English
publishDate 2025-04-01
publisher Ferrata Storti Foundation
record_format Article
series Haematologica
spelling doaj-art-fe053b40b57d433982b24e0088937fa02025-08-20T02:27:39ZengFerrata Storti FoundationHaematologica0390-60781592-87212025-04-01999110.3324/haematol.2024.287231A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosisPankit Vachhani0Peter Tan1Anne-Marie Watson2Shang-Ju Wu3Ross Baker4Stanley Cheung5Sung-Eun Lee6Chih-Cheng Chen7Tsai-Yun Chen8Hui-Hua Hsiao9Jae Hoon Lee10Lucia Masarova11Shuh Ying Tan12Jana Baskar13Brett Charlton14Alison Findlay15Dieter Hamprecht16Wolfgang Jarolimek17Joanna Leadbetter18John Miller19Kristen Morgan20Amna Zahoor21Gabriela Hobbs22Division of Hematology/Oncology, O'Neal Comprehensive Cancer Center, University of Alabama at Birmingham, BirminghamOne Clinical Research Pty Ltd, NedlandsLiverpool Hospital SW Area Pathology Service, LiverpoolNational Taiwan University Hospital and National Taiwan University College of MedicinePerth Blood Institute, Murdoch University, PerthICON Cancer Care, Kurralta ParkThe Catholic University of Korea, College of Medicine, SeoulChang-Gung Memorial Hospital, ChiayiCollege of Medicine, National Cheng Kung University, TainanKaohsiung Medical University, KaohsiungGachon University Gil Hospital, IncheonDepartment of Leukemia, The University of Texas MD Anderson Cancer Center, HoustonDepartment of Haematology, St Vincent's Hospital, MelbourneSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestSyntara Limited, Frenchs ForestMassachusetts General Hospital, Boston Myelofibrosis (MF) is a progressive disease characterized by accumulation of extracellular matrix (ECM) in the bone marrow (BM) which impairs normal hematopoiesis. While Janus kinase (JAK) inhibitors reduce spleen volume and provide symptomatic improvement, they do not resolve BM fibrosis and may cause or exacerbate anemia and thrombocytopenia. An anti-fibrotic therapy capable of normalising BM microenvironment and function remains a significant gap in the current treatment landscape. MF is associated with elevated expression of lysyl oxidases; enzymes responsible for maturation of the most abundant ECM proteins, collagen and elastin. PXS-5505 is a novel pan-lysyl oxidase inhibitor designed to exert an anti-fibrotic mode of action by preventing the cross-linking of collagen and elastin. PXS5505-MF-101 is a multi-center phase 1/2a study of PXS-5505 in MF patients which included a dose escalation phase (DEP) and a cohort expansion phase (CEP). Primary objectives were to determine the safety and tolerability of PXS-5505 and to define dosing for future studies. The DEP demonstrated that the highest dose tested, 200 mg BID, was safe and achieved robust systemic reduction of lysyl oxidase activity; this dose was therefore selected for the CEP. Twentyfour patients (median age 72 years) with relapsed or refractory MF were recruited into the CEP and 54% (13/24) completed 24 weeks of treatment. PXS-5505 was well tolerated and reached steady state concentrations by Day 28. Over the 24-week treatment period preliminary indications of clinical efficacy, including a reduction in BM collagen, were evident. Overall, these data support continued investigation of PXS-5505. https://haematologica.org/article/view/12032
spellingShingle Pankit Vachhani
Peter Tan
Anne-Marie Watson
Shang-Ju Wu
Ross Baker
Stanley Cheung
Sung-Eun Lee
Chih-Cheng Chen
Tsai-Yun Chen
Hui-Hua Hsiao
Jae Hoon Lee
Lucia Masarova
Shuh Ying Tan
Jana Baskar
Brett Charlton
Alison Findlay
Dieter Hamprecht
Wolfgang Jarolimek
Joanna Leadbetter
John Miller
Kristen Morgan
Amna Zahoor
Gabriela Hobbs
A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
Haematologica
title A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
title_full A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
title_fullStr A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
title_full_unstemmed A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
title_short A phase I / IIa trial of PXS-5505, a novel pan-lysyl oxidase inhibitor, in advanced myelofibrosis
title_sort phase i iia trial of pxs 5505 a novel pan lysyl oxidase inhibitor in advanced myelofibrosis
url https://haematologica.org/article/view/12032
work_keys_str_mv AT pankitvachhani aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT petertan aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT annemariewatson aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT shangjuwu aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT rossbaker aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT stanleycheung aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT sungeunlee aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT chihchengchen aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT tsaiyunchen aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT huihuahsiao aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT jaehoonlee aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT luciamasarova aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT shuhyingtan aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT janabaskar aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT brettcharlton aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT alisonfindlay aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT dieterhamprecht aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT wolfgangjarolimek aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT joannaleadbetter aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT johnmiller aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT kristenmorgan aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT amnazahoor aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT gabrielahobbs aphaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT pankitvachhani phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT petertan phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT annemariewatson phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT shangjuwu phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT rossbaker phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT stanleycheung phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT sungeunlee phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT chihchengchen phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT tsaiyunchen phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT huihuahsiao phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT jaehoonlee phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT luciamasarova phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT shuhyingtan phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT janabaskar phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT brettcharlton phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT alisonfindlay phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT dieterhamprecht phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT wolfgangjarolimek phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT joannaleadbetter phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT johnmiller phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT kristenmorgan phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT amnazahoor phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis
AT gabrielahobbs phaseiiiatrialofpxs5505anovelpanlysyloxidaseinhibitorinadvancedmyelofibrosis