Cladribine (2-chlorodeoxyadenosine) in frontline chemotherapy for adult Langerhans cell histiocytosis: A single-center study of seven cases

Adam, Zdeněk; Szturz, Petr; Vaníček, Jiří; Moulis, Mojmír; Pour, Luděk; Krejčí, Marta; Hájek, Roman; Mayer, Jiřı́

doi:10.3109/0284186x.2012.716164

Cited by 19 publications

(22 citation statements)

References 24 publications

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“…Differences in disease activity assessment, which can be challenging in a‐LCH, may also play a role. Moreover, two additional small series have been published since the initiation of the present study, and their results were consistent with our findings (Schini et al , 2013; Adam et al , 2013). Schini et al reported on five patients treated with 2CdA monotherapy, of whom four experienced a response.…”

Section: Discussionsupporting

confidence: 94%

Long‐term efficacy and safety of 2CdA (cladribine) in extra‐pulmonary adult‐onset Langerhans cell histiocytosis: analysis of 23 cases from the French Histiocytosis Group and systematic literature review

et al. 2020

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Summary Langerhans cell histiocytosis (LCH) is a rare protean disease that usually affects children. Few data are available for management of adult‐onset cases. A complete picture of the efficacy and safety of 2CdA (2‐chlorodeoxyadenosine, cladribine) is lacking. We report a retrospective multicentre study of 23 adult LCH (a‐LCH) patients who received single‐agent 2CdA and a systematic literature review. All had previously received systemic therapy (vinblastine, n = 19). Response to 2CdA was evaluable in 22 cases. Overall response rate (ORR) was 91%. Complete response (CR) occurred in 11 cases (50%). Nine patients (39%) developed grade 3–4 neutropenia and/or severe infection. A literature review yielded 48 additional cases. A pooled analysis confirmed our findings (ORR: 88%, CR: 49%). CRs were rare with cumulative dose <50 mg/m2. Disease progression rates were 20% and 30% at two and five years, respectively. Partial response (PR) to 2CdA was predictive of disease progression. Among eight re‐treated patients, five went into CR, two in PR, and one died. Single‐agent 2CdA is effective in reactivated a‐LCH, including at intermediate doses. Toxicity, significant but acceptable, warrants infectious prophylaxis. Complete responders may enter prolonged remission. Further studies are needed to determine 2CdA sequencing with other agents (vinblastine, cytarabine).

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Section: Discussionsupporting

confidence: 94%

Long‐term efficacy and safety of 2CdA (cladribine) in extra‐pulmonary adult‐onset Langerhans cell histiocytosis: analysis of 23 cases from the French Histiocytosis Group and systematic literature review

et al. 2020

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“…For historical reasons, principally misdiagnosis of non-Hodgkin lymphomas as HS, lymphoma-directed therapy such as CHOP or CHOP-like regimens has been used despite a lack of data for superiority over histiocyte-directed therapies [ 3 ]. Cladribine, cytarabine, and vinblastine all have documented activity in Langerhans histiocytosis but have not been reported to be active in histiocytic sarcoma [ 20 – 26 ]. Application of CLAG-M to this patient yielded a partial response that was maintained with vinblastine as a bridge to allogeneic HCT.…”

Section: Discussionmentioning

confidence: 99%

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

Tomlin

Orosco

Boles

et al. 2015

Case Reports in Hematology

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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.

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“…No drug or regimen has been proved superior to the others. Cladribine (2-CdA) is a promising agent in this setting as previously reported also with a review of previous studies (8). Cladribine has good bioavailability in the CNS.…”

Section: Discussionmentioning

confidence: 73%

Treatment of Multifocal Multisystem BRAF Positive Langerhans Cell Histiocytosis with Cladribine, Surgery and Allogenic Stem Cell Transplantation

Gabalec

Šimkovič

Zavřelová

et al. 2017

Acta Med. (Hradec Kralove, Czech Repub.)

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Langerhans cell histiocytosis (LCH) is a very rare disease in adults and as well a very rare cause of sellar expansion. The clinical presentation can be heterogeneous, from a single bone lesion to potentially fatal, widespread disease. We describe the difficulties with the diagnosis and treatment of LCH as well as successful treatment with cladribine chemotherapy and allogeneic stem cell transplantation.

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Cladribine (2-chlorodeoxyadenosine) in frontline chemotherapy for adult Langerhans cell histiocytosis: A single-center study of seven cases

Cited by 19 publications

References 24 publications

Long‐term efficacy and safety of 2CdA (cladribine) in extra‐pulmonary adult‐onset Langerhans cell histiocytosis: analysis of 23 cases from the French Histiocytosis Group and systematic literature review

Long‐term efficacy and safety of 2CdA (cladribine) in extra‐pulmonary adult‐onset Langerhans cell histiocytosis: analysis of 23 cases from the French Histiocytosis Group and systematic literature review

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

Treatment of Multifocal Multisystem BRAF Positive Langerhans Cell Histiocytosis with Cladribine, Surgery and Allogenic Stem Cell Transplantation

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