Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial

Background: Indolent systemic mastocytosis is a clonal mast cell disease that results in an increase in mast cells in the skin, bone marrow, and other organ systems. IL-6 has been shown to promote mast cell maturation, proliferation, and reactivity in vitro. Serum levels of IL-6 correlate with sever...

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Main Authors: Hirsh D. Komarow, MD, Jing Wang, MS, Robin Eisch, RN, Linda Scott, CRNP, Erica H. Brittain, PhD, Dean D. Metcalfe, MD, MS
Format: Article
Language:English
Published: Elsevier 2025-08-01
Series:Journal of Allergy and Clinical Immunology: Global
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Online Access:http://www.sciencedirect.com/science/article/pii/S2772829325000992
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author Hirsh D. Komarow, MD
Jing Wang, MS
Robin Eisch, RN
Linda Scott, CRNP
Erica H. Brittain, PhD
Dean D. Metcalfe, MD, MS
author_facet Hirsh D. Komarow, MD
Jing Wang, MS
Robin Eisch, RN
Linda Scott, CRNP
Erica H. Brittain, PhD
Dean D. Metcalfe, MD, MS
author_sort Hirsh D. Komarow, MD
collection DOAJ
description Background: Indolent systemic mastocytosis is a clonal mast cell disease that results in an increase in mast cells in the skin, bone marrow, and other organ systems. IL-6 has been shown to promote mast cell maturation, proliferation, and reactivity in vitro. Serum levels of IL-6 correlate with severity of disease and risk of progression of systemic disease. Objective: We conducted a double-blind placebo-controlled study to assess safety and efficacy for the use of sarilumab in improving the quality of life for those with indolent systemic mastocytosis. ClinicalTrial.gov registration NCT03770273. Methods: A double-blind trial randomized 16 participants. The primary analysis compared the arms on the Mastocytosis Quality of Life Questionnaire (MC-QoL) index at 16 weeks, adjusting for the baseline MC-QoL. Mean baseline MC-QoL was 47.8. Results: The difference between the arms in the primary analysis was not statistically significant, with the results favoring the placebo arm; mean absolute MC-QoL improvement in the placebo arm was 14.7 relative to 8.9 in the sarilumab arm. The estimated treatment effect from regression analysis was a 6.0-unit advantage of MC-QoL in the placebo arm (P = .40; 95% confidence interval, −8.9, 20.8), limiting a possible sarilumab advantage to at most 9 units of MC-QoL. Similar conclusions were observed for other quality-of-life indices. Conclusions: Results in this small trial did not support using sarilumab to treat this population and highlights the importance of double-blind randomized studies.
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spelling doaj-art-1be37af411064cff8e03eba84b4b77c72025-08-20T03:42:40ZengElsevierJournal of Allergy and Clinical Immunology: Global2772-82932025-08-014310049810.1016/j.jacig.2025.100498Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trialHirsh D. Komarow, MD0Jing Wang, MS1Robin Eisch, RN2Linda Scott, CRNP3Erica H. Brittain, PhD4Dean D. Metcalfe, MD, MS5Laboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, Md; Corresponding author: Hirsh D. Komarow, MD, Laboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bldg 10, Room 6D44A, 10 Center Dr, Bethesda, MD 20892-1960.Clinical Monitoring Research Program Directorate, Frederick National Laboratory for Cancer Research, Frederick, MdLaboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, MdLaboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, MdBiostatistics Research Branch, Division of Clinical Research, NIAID, NIH, Bethesda, MdLaboratory of Allergic Diseases, National Institute of Allergy and Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, MdBackground: Indolent systemic mastocytosis is a clonal mast cell disease that results in an increase in mast cells in the skin, bone marrow, and other organ systems. IL-6 has been shown to promote mast cell maturation, proliferation, and reactivity in vitro. Serum levels of IL-6 correlate with severity of disease and risk of progression of systemic disease. Objective: We conducted a double-blind placebo-controlled study to assess safety and efficacy for the use of sarilumab in improving the quality of life for those with indolent systemic mastocytosis. ClinicalTrial.gov registration NCT03770273. Methods: A double-blind trial randomized 16 participants. The primary analysis compared the arms on the Mastocytosis Quality of Life Questionnaire (MC-QoL) index at 16 weeks, adjusting for the baseline MC-QoL. Mean baseline MC-QoL was 47.8. Results: The difference between the arms in the primary analysis was not statistically significant, with the results favoring the placebo arm; mean absolute MC-QoL improvement in the placebo arm was 14.7 relative to 8.9 in the sarilumab arm. The estimated treatment effect from regression analysis was a 6.0-unit advantage of MC-QoL in the placebo arm (P = .40; 95% confidence interval, −8.9, 20.8), limiting a possible sarilumab advantage to at most 9 units of MC-QoL. Similar conclusions were observed for other quality-of-life indices. Conclusions: Results in this small trial did not support using sarilumab to treat this population and highlights the importance of double-blind randomized studies.http://www.sciencedirect.com/science/article/pii/S2772829325000992Mastocytosismast cellstryptaseanti–IL-6sarilumab
spellingShingle Hirsh D. Komarow, MD
Jing Wang, MS
Robin Eisch, RN
Linda Scott, CRNP
Erica H. Brittain, PhD
Dean D. Metcalfe, MD, MS
Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
Journal of Allergy and Clinical Immunology: Global
Mastocytosis
mast cells
tryptase
anti–IL-6
sarilumab
title Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
title_full Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
title_fullStr Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
title_full_unstemmed Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
title_short Treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
title_sort treatment of indolent systemic mastocytosis with sarilumab is not supported in a randomized trial
topic Mastocytosis
mast cells
tryptase
anti–IL-6
sarilumab
url http://www.sciencedirect.com/science/article/pii/S2772829325000992
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