Refractory Subcutaneous Panniculitis-Like T-Cell Lymphoma with Hemophagocytic Syndrome Treated with Romidepsin and Allogeneic Hematopoietic Cell Transplantation.

Yuki Ishizawa, Tomomi Toubai, Tsubasa Ichikawa, Masahito Himuro, Mikiya Kato, Yusuke Nagano, Ryo Takahashi, Masashi Hosokawa, Yuka Hosokawa, Akane Yamada, Takuma Suzuki, Keiko Aizawa, Satoshi Ito, Daniel Peltier, Hisayuki Yokoyama, Kenichi Ishizawa
Author Information
  1. Yuki Ishizawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  2. Tomomi Toubai: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  3. Tsubasa Ichikawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  4. Masahito Himuro: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  5. Mikiya Kato: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  6. Yusuke Nagano: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  7. Ryo Takahashi: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  8. Masashi Hosokawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  9. Yuka Hosokawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  10. Akane Yamada: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  11. Takuma Suzuki: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  12. Keiko Aizawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  13. Satoshi Ito: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  14. Daniel Peltier: Division of Pediatric Hematology and Oncology, Department of Pediatrics, Herman B. Wells Center for Pediatric Research, Simon Cancer Center, Indiana University School of Medicine, Indianapolis, IN, USA.
  15. Hisayuki Yokoyama: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.
  16. Kenichi Ishizawa: Division of Hematology and Cell Therapy, Department of Internal Medicine III, Yamagata University Faculty of Medicine, Yamagata, Japan.

Abstract

Introduction: Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is a rare subtype of cutaneous T-cell lymphoma (CTCL) that when refractory or complicated by hemophagocytic syndrome (HPS) has a poor prognosis. Romidepsin is a histone deacetylase inhibitor, but its efficacy for SPTCL is unknown. The efficacy of allogeneic hematopoietic cell transplantation (allo-HCT) is also unclear. Herein, we report a case of refractory SPTCL with HPS that was successfully treated with romidepsin followed by consolidation with allo-HCT.
Case Presentation: A 26-year-old female presented with fever, generalized painful erythema, pancytopenia, and hemophagocytosis. F-fluorodeoxyglucose positron emission tomography/computed tomography (PET) showed diffuse PET-avid infiltration of the subcutaneous adipose tissue found to be SPTCL via skin biopsy. Her SPTCL was refractory to conventional chemotherapy but complete metabolic response was achieved after romidepsin. An allo-HCT was used for consolidation, and she remains in complete remission 3 years later.
Conclusion: Romidepsin with allo-HCT consolidation may be an effective approach for refractory SPTCL.

Keywords

References

  1. Ann Oncol. 2013 Oct;24 Suppl 6:vi149-54 [PMID: 23868906]
  2. J Clin Oncol. 2012 Aug 20;30(24):e221-5 [PMID: 22753921]
  3. Hematol Oncol. 2021 Jun;39 Suppl 1:46-51 [PMID: 34105822]
  4. Case Rep Oncol. 2022 Dec 12;15(3):1088-1094 [PMID: 36605220]
  5. Int J Immunopathol Pharmacol. 2021 Jan-Dec;35:20587384211009342 [PMID: 33845613]
  6. Front Immunol. 2021 Feb 24;12:641910 [PMID: 33732262]
  7. Blood. 2021 May 13;137(19):2646-2656 [PMID: 33512419]
  8. Oncologist. 2015 Sep;20(9):1084-91 [PMID: 26099743]
  9. Blood. 2008 Jan 15;111(2):838-45 [PMID: 17934071]
  10. Blood. 2024 Oct 31;144(18):1898-1909 [PMID: 38657272]
  11. Int J Hematol. 2019 Feb;109(2):187-196 [PMID: 30547418]
  12. Front Immunol. 2021 Jun 24;12:692219 [PMID: 34248986]
  13. Blood. 2005 May 15;105(10):3768-85 [PMID: 15692063]
  14. JAAD Case Rep. 2020 Oct 07;6(12):1245-1247 [PMID: 33294555]
  15. Blood. 2011 Oct 27;118(17):4577-84 [PMID: 21900192]

Word Cloud

Created with Highcharts 10.0.0SPTCLrefractoryRomidepsinallo-HCTSubcutaneousT-celllymphomaconsolidationpanniculitis-likesyndromeHPSefficacyhematopoieticcelltransplantationromidepsincompleteHemophagocyticAllogeneicIntroduction:raresubtypecutaneousCTCLcomplicatedhemophagocyticpoorprognosishistonedeacetylaseinhibitorunknownallogeneicalsounclearHereinreportcasesuccessfullytreatedfollowedCasePresentation:26-year-oldfemalepresentedfevergeneralizedpainfulerythemapancytopeniahemophagocytosisF-fluorodeoxyglucosepositronemissiontomography/computedtomographyPETshoweddiffusePET-avidinfiltrationsubcutaneousadiposetissuefoundviaskinbiopsyconventionalchemotherapymetabolicresponseachievedusedremainsremission3yearslaterConclusion:mayeffectiveapproachRefractoryPanniculitis-LikeT-CellLymphomaSyndromeTreatedHematopoieticCellTransplantation

Similar Articles

Cited By