Successful Treatment of Multifocal Histiocytic Sarcoma Occurring after Renal Transplantation with Cladribine, High-Dose Cytarabine, G-CSF, and Mitoxantrone (CLAG-M) Followed by Allogeneic Hematopoietic Stem Cell Transplantation

Abstract

Histiocytic sarcoma (HS) is a rare, aggressive malignancy. Lesions previously called HS were typically non-Hodgkin lymphomas, not HS. As such, chemotherapy directed at lymphoid neoplasms was frequently successful, but it is unclear if these regimens are ideal for HS. We present a 33-year-old African gentleman who underwent sequential renal transplants for glomerulonephritis. He subsequently developed HS of the upper airway and multiple cutaneous sites. The patient received cyclophosphamide, doxorubicin, vincristine, and prednisone (CHOP) followed by salvage ifosfamide, carboplatin, and etoposide (ICE) but had continuous progression of cutaneous involvement. Cladribine, high-dose cytarabine, G-CSF, and mitoxantrone (CLAG-M) yielded a partial response with near resolution of disease. Ultimately, the patient achieved a complete remission after myeloablative allogeneic hematopoietic stem cell transplant. HS occurring after solid organ transplant raises the possibility of HS as a potential posttransplant malignancy. The use of CLAG-M has not been reported in HS. In this case, histiocyte-directed chemotherapy with CLAG-M was superior to lymphoma-directed regimens.

Pediatric intracerebral histiocytic sarcoma with rhabdoid features: Case report and literature review

“…3,30,31 Myeloablative allogeneic hematopoietic stem cell transplantation may be reserved for relapsed histiocytic sarcoma in non-cerebral histiocytic sarcoma although attempts in cerebral histiocytic sarcomas have not yet been reported. 32 The present case is a rare case of pediatric cerebral histiocytic sarcoma, and is the first case showing BRAF V600Emutated intracerebral histiocytic sarcoma.…”

“…Radiation therapy combined with resection is recommended in non‐disseminated, localized disease . Myeloablative allogeneic hematopoietic stem cell transplantation may be reserved for relapsed histiocytic sarcoma in non‐cerebral histiocytic sarcoma although attempts in cerebral histiocytic sarcomas have not yet been reported …”

Targeted therapy of BRAF V600E‐mutant histiocytic sarcoma: A case report and review of the literature

“…HS tumour cells are derived from monocyte/macrophage lineages and arise sporadically (primary HS) or secondarily to other haematological malignancies, such as acute lymphoblastic leukaemia and non‐Hodgkin's lymphoma, or even after a kidney transplant, with a delayed onset ranging from 2 months to 17 years . The physiopathology of HS is not well defined and may be explained by three putative hypotheses: a trans‐differentiation from a haematolymphoid neoplasm, a dedifferentiation of a B‐cell progenitor followed by a redifferentiation into another lineage or a common progenitor which is able to differentiate into myeloid or lymphoid cell lineages …”

“…Lymphoma‐type chemotherapies (CHOP, ABVD, Bendamustine) are widely used in a few retrospectives studies and case reports. A few studies describe the use of autologous or allogeneic haematopoietic stem cell transplantation as a first‐line therapy or as a therapy for relapsed or refractory disease, but have resulted in inconsistent outcomes …”

“…A few studies describe the use of autologous or allogeneic haematopoietic stem cell transplantation as a first-line therapy or as a therapy for relapsed or refractory disease, but have resulted in inconsistent outcomes. 2,9,10 Recent genomic studies have identified a number of oncogenic drivers of the disease, including RAS-MAPK pathway activating mutations 11 and BRAF V600E mutations. 12 Our observation adds additional support for performing clinical trials assessing the use of targeted anti-BRAF V600E approaches, whenever the BRAF V600E mutation is detected.…”