A case of refractory langerhans cell histiocytosis complicated with hemophagocytic lymphohistiocytosis rescued by cord blood transplantation with reduced-intensity conditioning

Kana Washio, Michiko Muraoka, Kiichiro Kanamitsu, Megumi Oda, Akira Shimada

Research output: Contribution to journalArticle

2 Citations (Scopus)

Abstract

We diagnosed a female infant with Langerhans cell histiocytosis (LCH) who was refractory to conventional chemotherapy. She showed refractory inflammation that was complicated with hemophagocytic lymphohistiocytosis (HLH) during LCH chemotherapy; therefore, we changed the protocol to HLH2004 (dexamethasone, cyclosporine A and VP16). However, there were no signs of hematological recovery. We therefore performed cord blood transplantation with reduced-intensity conditioning, and she achieved complete remission for over 2 years. As salvage therapy for refractory LCH, hematopoietic stem cell transplantation may be a good therapeutic choice, especially when LCH is complicated with HLH.

Original languageEnglish
Pages (from-to)249-254
Number of pages6
JournalActa Medica Okayama
Volume71
Issue number3
Publication statusPublished - 2017

Fingerprint

Hemophagocytic Lymphohistiocytosis
Langerhans Cell Histiocytosis
Fetal Blood
Refractory materials
Blood
Chemotherapy
Transplantation
Salvaging
Stem cells
Drug Therapy
Salvage Therapy
Dexamethasone
Cyclosporine
Hematopoietic Stem Cell Transplantation
Recovery
Inflammation

Keywords

  • Hematopoietic stem cell transplantation (HSCT)
  • Hemophagocytic lymphohistiocytosis (HLH)
  • Langerhans cell histiocytosis (LCH)
  • Reduced-intensity conditioning (RIC)
  • Refractory

ASJC Scopus subject areas

  • Medicine(all)
  • Biochemistry, Genetics and Molecular Biology(all)

Cite this

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abstract = "We diagnosed a female infant with Langerhans cell histiocytosis (LCH) who was refractory to conventional chemotherapy. She showed refractory inflammation that was complicated with hemophagocytic lymphohistiocytosis (HLH) during LCH chemotherapy; therefore, we changed the protocol to HLH2004 (dexamethasone, cyclosporine A and VP16). However, there were no signs of hematological recovery. We therefore performed cord blood transplantation with reduced-intensity conditioning, and she achieved complete remission for over 2 years. As salvage therapy for refractory LCH, hematopoietic stem cell transplantation may be a good therapeutic choice, especially when LCH is complicated with HLH.",
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AU - Muraoka, Michiko

AU - Kanamitsu, Kiichiro

AU - Oda, Megumi

AU - Shimada, Akira

PY - 2017

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AB - We diagnosed a female infant with Langerhans cell histiocytosis (LCH) who was refractory to conventional chemotherapy. She showed refractory inflammation that was complicated with hemophagocytic lymphohistiocytosis (HLH) during LCH chemotherapy; therefore, we changed the protocol to HLH2004 (dexamethasone, cyclosporine A and VP16). However, there were no signs of hematological recovery. We therefore performed cord blood transplantation with reduced-intensity conditioning, and she achieved complete remission for over 2 years. As salvage therapy for refractory LCH, hematopoietic stem cell transplantation may be a good therapeutic choice, especially when LCH is complicated with HLH.

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KW - Hemophagocytic lymphohistiocytosis (HLH)

KW - Langerhans cell histiocytosis (LCH)

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