Resolution of a steroid-resistant, hypereosinophilic immune diathesis with mepolizumab and concomitant amelioration of a mixed thrombotic microangiopathy.

TitleResolution of a steroid-resistant, hypereosinophilic immune diathesis with mepolizumab and concomitant amelioration of a mixed thrombotic microangiopathy.
Publication TypeJournal Article
Year of Publication2018
AuthorsJ Mulvey J, Magro C, Chadburn A
JournalBlood Cells Mol Dis
Volume69
Pagination38-42
Date Published2018 03
ISSN1096-0961
KeywordsAdult, Antibodies, Monoclonal, Humanized, Biomarkers, Biopsy, Bone Marrow, Disease Susceptibility, Drug Resistance, Eosinophils, Humans, Immunoglobulins, Intravenous, Male, Steroids, Thrombotic Microangiopathies, Treatment Outcome
Abstract

The anaphylatoxins produced by an unbridled complement cascade in atypical hemolytic uremic syndrome (aHUS) can alter the leukocyte environment in tissues and peripheral blood, causing clinically significant eosinophilia. While the membrane attack complex and C5a anaphlatoxin can be suppressed with anti-C5 biologics, the production of C3a is still capable of driving a destructive hypereosinophilic syndrome in spite of anticomplement therapy. The side-effects of glucocorticoids in treating hypereosinophilic syndrome limit their therapeutic index in long-term treatment and this behooves the use of alternative strategies. While use of the anti-IL-5 antibody, mepolizumab, has been reported for treatment of primary hypereosinophilic syndromes off-label, its usage in the setting of complement-driven thrombotic microangiopathy has not been attempted. We report mepolizumab's rapid resolution of a glucocorticoid-resistant hypereosinophilic syndrome that caused multi-organ dysfunction in a patient with a complex immune diathesis. The patient's long standing TTP/aHUS disease activity, shown to have direct correlation with his eosinophil count, improved with anti-IL-5 therapy, suggesting a reciprocal enhancement between the conditions.

DOI10.1016/j.bcmd.2017.04.008
Alternate JournalBlood Cells Mol Dis
PubMed ID28648630
Related Faculty: 
Amy Chadburn, M.D. Cynthia M. Magro, M.D.

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