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Long-term combination therapy with metformin and oxymetholone in a Fanconi anemia mouse model.
Dorrell, Craig; Peters, Alexander M; Zhang, Qingshuo; Balaji, Niveditha; Baradar, Kevin; Mochizuki-Kashio, Makiko; Major, Angela; Finegold, Milton; Liu, Chih-Wei; Lu, Kun; Grompe, Markus.
Afiliación
  • Dorrell C; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Peters AM; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Zhang Q; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Balaji N; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Baradar K; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Mochizuki-Kashio M; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
  • Major A; Department of Pathology, Texas Children's Hospital, Houston, Texas, USA.
  • Finegold M; Department of Pathology, Texas Children's Hospital, Houston, Texas, USA.
  • Liu CW; Department of Environmental Sciences and Engineering, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.
  • Lu K; Department of Environmental Sciences and Engineering, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.
  • Grompe M; Department of Pediatrics, Papé Family Pediatric Research Institute, Stem Cell Center, Pediatric Blood & Cancer Biology Program, Oregon Health & Science University, Portland, Oregon, USA.
Pediatr Blood Cancer ; 71(8): e31030, 2024 Aug.
Article en En | MEDLINE | ID: mdl-38733122
ABSTRACT
Fanconi anemia (FA) is a disease caused by defective deoxyribonucleic acid (DNA) repair that manifests as bone marrow failure, cancer predisposition, and developmental defects. We previously reported that monotherapy with either metformin (MET) or oxymetholone (OXM) improved peripheral blood (PB) counts and the number and functionality of bone marrow hematopoietic stem progenitor cells (HSPCs) number in Fancd2-/- mice. To evaluate whether the combination treatment of these drugs has a synergistic effect to prevent bone marrow failure in FA, we treated cohorts of Fancd2-/- mice and wildtype controls with either MET alone, OXM alone, MET+OXM, or placebo diet from age 3 weeks to 18 months. The OXM treated animals showed modest improvements in blood parameters including platelet count (p = .01) and hemoglobin levels (p < .05). In addition, the percentage of quiescent hematopoietic stem cell (HSC) (LSK [Lin-Sca+c-Kit+]) was significantly increased (p = .001) by long-term treatment with MET alone. The combination of metformin and oxymetholone did not result in a significant synergistic effect in any hematopoietic parameter. Gene expression analysis of liver tissue from these animals showed that some of the expression changes caused by Fancd2 deletion were partially normalized by metformin treatment. Importantly, no adverse effects of the individual or combination therapies were observed, despite the long-term administration. We conclude that androgen therapy is not a contraindication to concurrent metformin administration in clinical trials. HIGHLIGHTS Long-term coadministration of metformin in combination with oxymetholone is well tolerated by Fancd2-/- mice. Hematopoietic stem cell quiescence in mutant mice was enhanced by treatment with metformin alone. Metformin treatment caused a partial normalization of gene expression in the livers of mutant mice.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Oximetolona / Modelos Animales de Enfermedad / Quimioterapia Combinada / Anemia de Fanconi / Metformina Límite: Animals Idioma: En Revista: Pediatr Blood Cancer Asunto de la revista: HEMATOLOGIA / NEOPLASIAS / PEDIATRIA Año: 2024 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Oximetolona / Modelos Animales de Enfermedad / Quimioterapia Combinada / Anemia de Fanconi / Metformina Límite: Animals Idioma: En Revista: Pediatr Blood Cancer Asunto de la revista: HEMATOLOGIA / NEOPLASIAS / PEDIATRIA Año: 2024 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos