Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis

Deevyashali Parekh; Howard Lin; Akanksha Batajoo; Erin Peckham-Gregory; Vivekanudeep Karri; Whitney Stanton; Brooks Scull; Ryan Fleishmann; Nader El-Mallawany; Olive S. Eckstein; Zachary D. Prudowsky; Nitya Gulati; Jennifer E. Agrusa; Asra Z. Ahmed; Roland Chu; Matthew S Dietz; Stanton C Goldman; Michael D. Hogarty; Hamayun Imran; Stefanos Intzes; Jenny M Kim; Lisa M. Kopp; Carolyn Fein Levy; Philip Neff; Pallavi M. Pillai; Bryan A. Sisk; Deborah E. Schiff; Angela D Trobaugh-Lotrario; Kelly Walkovich; Kenneth L. McClain; Carl E. Allen

doi:10.1111/bjh.19376

Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis

Deevyashali Parekh, Howard Lin, Akanksha Batajoo, Erin Peckham-Gregory, Vivekanudeep Karri, Whitney Stanton, Brooks Scull, Ryan Fleishmann, Nader El-Mallawany, Olive S. Eckstein, Zachary D. Prudowsky, Nitya Gulati, Jennifer E. Agrusa, Asra Z. Ahmed, Roland Chu, Matthew S Dietz, Stanton C Goldman, Michael D. Hogarty, Hamayun Imran, Stefanos IntzesJenny M Kim, Lisa M. Kopp, Carolyn Fein Levy, Philip Neff, Pallavi M. Pillai, Bryan A. Sisk, Deborah E. Schiff, Angela D Trobaugh-Lotrario, Kelly Walkovich, Kenneth L. McClain, Carl E. Allen

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Abstract

Over 50% of patients with systemic LCH are not cured with front-line therapies, and data to guide salvage options are limited. We describe 58 patients with LCH who were treated with clofarabine. Clofarabine monotherapy was active against LCH in this cohort, including heavily pretreated patients with a systemic objective response rate of 92.6%, higher in children (93.8%) than adults (83.3%). BRAF^V600E+ variant allele frequency in peripheral blood is correlated with clinical responses. Prospective multicentre trials are warranted to determine optimal dosing, long-term efficacy, late toxicities, relative cost and patient-reported outcomes of clofarabine compared to alternative LCH salvage therapy strategies.

Original language	English
Pages (from-to)	1888-1893
Number of pages	6
Journal	British Journal of Haematology
Volume	204
Issue number	5
DOIs	https://doi.org/10.1111/bjh.19376
State	Published - May 2024

Keywords

Langerhans cell histiocytosis
clofarabine
myeloid neoplasia

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10.1111/bjh.19376

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Parekh, D., Lin, H., Batajoo, A., Peckham-Gregory, E., Karri, V., Stanton, W., Scull, B., Fleishmann, R., El-Mallawany, N., Eckstein, O. S., Prudowsky, Z. D., Gulati, N., Agrusa, J. E., Ahmed, A. Z., Chu, R., Dietz, MS., Goldman, SC., Hogarty, M. D., Imran, H., ... Allen, C. E. (2024). Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis. British Journal of Haematology, 204(5), 1888-1893. https://doi.org/10.1111/bjh.19376

@article{fe0907356b484c14b12a08bf68cdeff9,

title = "Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis",

abstract = "Over 50% of patients with systemic LCH are not cured with front-line therapies, and data to guide salvage options are limited. We describe 58 patients with LCH who were treated with clofarabine. Clofarabine monotherapy was active against LCH in this cohort, including heavily pretreated patients with a systemic objective response rate of 92.6%, higher in children (93.8%) than adults (83.3%). BRAFV600E+ variant allele frequency in peripheral blood is correlated with clinical responses. Prospective multicentre trials are warranted to determine optimal dosing, long-term efficacy, late toxicities, relative cost and patient-reported outcomes of clofarabine compared to alternative LCH salvage therapy strategies.",

keywords = "Langerhans cell histiocytosis, clofarabine, myeloid neoplasia",

author = "Deevyashali Parekh and Howard Lin and Akanksha Batajoo and Erin Peckham-Gregory and Vivekanudeep Karri and Whitney Stanton and Brooks Scull and Ryan Fleishmann and Nader El-Mallawany and Eckstein, {Olive S.} and Prudowsky, {Zachary D.} and Nitya Gulati and Agrusa, {Jennifer E.} and Ahmed, {Asra Z.} and Roland Chu and Matthew S Dietz and Stanton C Goldman and Hogarty, {Michael D.} and Hamayun Imran and Stefanos Intzes and Jenny M Kim and Kopp, {Lisa M.} and Levy, {Carolyn Fein} and Philip Neff and Pillai, {Pallavi M.} and Sisk, {Bryan A.} and Schiff, {Deborah E.} and Angela D Trobaugh-Lotrario and Kelly Walkovich and McClain, {Kenneth L.} and Allen, {Carl E.}",

note = "Publisher Copyright: {\textcopyright} 2024 British Society for Haematology and John Wiley & Sons Ltd.",

year = "2024",

month = may,

doi = "10.1111/bjh.19376",

language = "English",

volume = "204",

pages = "1888--1893",

journal = "British Journal of Haematology",

issn = "0007-1048",

number = "5",

}

Parekh, D, Lin, H, Batajoo, A, Peckham-Gregory, E, Karri, V, Stanton, W, Scull, B, Fleishmann, R, El-Mallawany, N, Eckstein, OS, Prudowsky, ZD, Gulati, N, Agrusa, JE, Ahmed, AZ, Chu, R, Dietz, MS, Goldman, SC, Hogarty, MD, Imran, H, Intzes, S, Kim, JM, Kopp, LM, Levy, CF, Neff, P, Pillai, PM, Sisk, BA, Schiff, DE, Trobaugh-Lotrario, AD, Walkovich, K, McClain, KL & Allen, CE 2024, 'Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis', British Journal of Haematology, vol. 204, no. 5, pp. 1888-1893. https://doi.org/10.1111/bjh.19376

TY - JOUR

T1 - Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis

AU - Parekh, Deevyashali

AU - Lin, Howard

AU - Batajoo, Akanksha

AU - Peckham-Gregory, Erin

AU - Karri, Vivekanudeep

AU - Stanton, Whitney

AU - Scull, Brooks

AU - Fleishmann, Ryan

AU - El-Mallawany, Nader

AU - Eckstein, Olive S.

AU - Prudowsky, Zachary D.

AU - Gulati, Nitya

AU - Agrusa, Jennifer E.

AU - Ahmed, Asra Z.

AU - Chu, Roland

AU - Dietz, Matthew S

AU - Goldman, Stanton C

AU - Hogarty, Michael D.

AU - Imran, Hamayun

AU - Intzes, Stefanos

AU - Kim, Jenny M

AU - Kopp, Lisa M.

AU - Levy, Carolyn Fein

AU - Neff, Philip

AU - Pillai, Pallavi M.

AU - Sisk, Bryan A.

AU - Schiff, Deborah E.

AU - Trobaugh-Lotrario, Angela D

AU - Walkovich, Kelly

AU - McClain, Kenneth L.

AU - Allen, Carl E.

PY - 2024/5

Y1 - 2024/5

N2 - Over 50% of patients with systemic LCH are not cured with front-line therapies, and data to guide salvage options are limited. We describe 58 patients with LCH who were treated with clofarabine. Clofarabine monotherapy was active against LCH in this cohort, including heavily pretreated patients with a systemic objective response rate of 92.6%, higher in children (93.8%) than adults (83.3%). BRAFV600E+ variant allele frequency in peripheral blood is correlated with clinical responses. Prospective multicentre trials are warranted to determine optimal dosing, long-term efficacy, late toxicities, relative cost and patient-reported outcomes of clofarabine compared to alternative LCH salvage therapy strategies.

AB - Over 50% of patients with systemic LCH are not cured with front-line therapies, and data to guide salvage options are limited. We describe 58 patients with LCH who were treated with clofarabine. Clofarabine monotherapy was active against LCH in this cohort, including heavily pretreated patients with a systemic objective response rate of 92.6%, higher in children (93.8%) than adults (83.3%). BRAFV600E+ variant allele frequency in peripheral blood is correlated with clinical responses. Prospective multicentre trials are warranted to determine optimal dosing, long-term efficacy, late toxicities, relative cost and patient-reported outcomes of clofarabine compared to alternative LCH salvage therapy strategies.

KW - Langerhans cell histiocytosis

KW - clofarabine

KW - myeloid neoplasia

UR - http://www.scopus.com/inward/record.url?scp=85188801716&partnerID=8YFLogxK

U2 - 10.1111/bjh.19376

DO - 10.1111/bjh.19376

M3 - Article

C2 - 38501389

AN - SCOPUS:85188801716

SN - 0007-1048

VL - 204

SP - 1888

EP - 1893

JO - British Journal of Haematology

JF - British Journal of Haematology

IS - 5

ER -

Clofarabine monotherapy in aggressive, relapsed and refractory Langerhans cell histiocytosis

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