Display options
Share it on

J Pediatr Hematol Oncol. 2021 Nov 01;43(8):e1164-e1167. doi: 10.1097/MPH.0000000000001986.

t(1;22)(p13;q13) Acute Megakaryoblastic Leukemia Complicated by Hepatic Fibrosis: Antifibrosis Therapy?.

Journal of pediatric hematology/oncology

Jianhua Feng, Alex W K Leung, Frankie W T Cheng, Grace K S Lam, Terry T W Chow, Margaret H L Ng, Winnie C W Chu, Natalie P H Chan, Chi-Kong Li

Affiliations

  1. Departments of Paediatrics.
  2. Department of Paediatrics, The First Affiliated Hospital of Wenzhou Medical University, Wenzhou, PR China.
  3. Anatomincal & Cellular Pathology.
  4. Diagnostic Radiology and Organ Imaging, Prince of Wales Hospital, The Chinese University of Hong Kong, Hong Kong SAR.

PMID: 33122587 DOI: 10.1097/MPH.0000000000001986

Abstract

BACKGROUND: There is no established effective treatment for patients with t(1;22)(p13;q13) acute megakaryoblastic leukemia (AMKL) and hepatic fibrosis.

OBSERVATION: Here we report the outcomes of 2 t(1;22)(p13;q13) AMKL patients with hepatic fibrosis. One patient died from liver failure despite the control of leukemia. The other patient was successfully treated with reduced-intensity chemotherapy and antifibrosis therapy with tretinoin and α-tocopheryl acetate, the hepatic fibrosis resolved and leukemia was in remission for 3 years.

CONCLUSIONS: Reduced-intensity chemotherapy plus antifibrosis therapy with tretinoin and α-tocopheryl acetate could be a treatment option for these patients with t(1;22)(p13;q13) AMKL and hepatic fibrosis.

Copyright © 2020 Wolters Kluwer Health, Inc. All rights reserved.

Conflict of interest statement

The authors declare no conflict of interest.

References

  1. Carroll A, Civin C, Schneider N, et al. The t(1;22) (p13;q13) is nonrandom and restricted to infants with acute megakaryoblastic leukemia: a Pediatric Oncology Group Study. Blood. 1991;78:748–752. - PubMed
  2. Bernstein J, Dastugue N, Haas OA, et al. Nineteen cases of the t(1;22)(p13;q13) acute megakaryblastic leukaemia of infants/children and a review of 39 cases: report from a t(1;22) study group. Leukemia. 2000;14:216–218. - PubMed
  3. Zhai XW, Cheng FW, Lee V, et al. Improved survival outcome of childhood acute myeloid leukemia with intensified chemotherapy in Chinese children. Pediatr Hematol Oncol. 2011;28:269–278. - PubMed
  4. NOPHO-DBH AML (2012) Protocol: research study for treatment of children and adolescents with acute myeloid leukaemia 0-18 years. ClinicalTrials.gov Identifier:NCT01828489. - PubMed
  5. Inaba H, Zhou Y, Abla O, et al. Heterogeneous cytogenetic subgroups and outcomes in childhood acute megakaryoblastic leukemia: a retrospective international study. Blood. 2015;126:1575–1584. - PubMed
  6. Kinjo T, Inoue H, Kusuda T, et al. Chemokine levels predict progressive liver disease in Down syndrome patients with transient abnormal myelopoiesis. Pediatr Neonatol. 2019;60:382–388. - PubMed
  7. Garel D, Mielot F, Gaulard P, et al. Acute megakaryocytic leukemia (AMKL) with major myelofibrosis in an infant. Diagnosis by liver biopsy and response to treatment. Nouv Rev Fr Hematol. 1991;33:5–8. - PubMed
  8. Becroft DM, Zwi LJ. Perinatal visceral fibrosis accompanying the megakaryoblastic leukemoid reaction of Down syndrome. Pediatr Pathol. 1990;10:397–406. - PubMed
  9. Li J, Fan R, Zhao S, et al. Reactive oxygen species released from hypoxic hepatocytes regulates MMP-2 expression in hepatic stellate cells. Int J Mol Sci. 2011;12:2434–2447. - PubMed
  10. Xia JR, Liu NF, Zhu NX. Specific siRNA targeting the receptor for advanced glycation end products inhibits experimental hepatic fibrosis in rats. Int J Mol Sci. 2008;9:638–661. - PubMed
  11. Hisamori S, Tabata C, Kadokawa Y, et al. All-trans-retinoic acid ameliorates carbon tetrachloride-induced liver fibrosis in mice through modulating cytokine production. Liver Int. 2008;28:1217–1225. - PubMed
  12. Wang H, Dan Z, Jiang H. Effect of all-trans retinoic acid on liver fibrosis induced by common bile duct ligation in rats. J Huazhong Univ Sci Technolog Med Sci. 2008;28:553–557. - PubMed
  13. Ye Y, Dan Z. All-trans retinoic acid diminishes collagen production in a hepatic stellate cell line via suppression of active protein-1 and c-Jun N-terminal kinase signal. J Huazhong Univ Sci Technolog Med Sci. 2010;30:726–733. - PubMed
  14. Bendia E, Benedetti A, Baroni GS, et al. Effect of cyanidin 3-O-beta-glucopyranoside on hepatic stellate cell proliferation and collagen synthesis induced by oxidative stress. Dig Liver Dis. 2005;37:342–348. - PubMed
  15. Brown KE, Poulos JE, Li L, et al. Effect of vitamin E supplementation on hepatic fibrogenesis in chronic dietary iron overload. Am J Physiol. 1997;272:G116–G123. - PubMed
  16. Pietrangelo A, Gualdi R, Casalgrandi G, et al. Molecular and cellular aspects of iron-induced hepatic cirrhosis in rodents. J Clin Invest. 1995;95:1824–1831. - PubMed
  17. Okuno M, Moriwaki H, Imai S, et al. Retinoids exacerbate rat liver fibrosis by inducing the activation of latent TGF-beta in liver stellate cells. Hepatology. 1997;26:913–921. - PubMed
  18. Zhou TB, Drummen GP, Qin YH. The controversial role of retinoic acid in fibrotic diseases: analysis of involved signaling pathways. Int J Mol Sci. 2012;14:226–243. - PubMed
  19. Nobili V, Manco M, Devito R, et al. Lifestyle intervention and antioxidant therapy in children with nonalcoholic fatty liver disease: a randomized, controlled trial. Hepatology. 2008;48:119–128. - PubMed
  20. Lavine JE, Schwimmer JB, Van Natta ML, et al. Effect of vitamin E or metformin for treatment of nonalcoholic fatty liver disease in children and adolescents: the TONIC randomized controlled trial. JAMA. 2011;305:1659–1668. - PubMed

Publication Types