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dc.contributor.authorRekeland, Ingrid Gurvin
dc.contributor.authorFosså, Alexander
dc.contributor.authorLande, Asgeir
dc.contributor.authorKtoridou-Valen, Irini
dc.contributor.authorSørland, Kari
dc.contributor.authorHolsen, Mari
dc.contributor.authorTronstad, Karl Johan
dc.contributor.authorRisa, Kristin
dc.contributor.authorAlme, Kine
dc.contributor.authorViken, Marte Katrine
dc.contributor.authorLie, Benedicte Alexandra
dc.contributor.authorDahl, Olav
dc.contributor.authorMella, Olav
dc.contributor.authorFluge, Øystein
dc.date.accessioned2021-06-22T10:46:26Z
dc.date.available2021-06-22T10:46:26Z
dc.date.created2020-09-22T12:06:18Z
dc.date.issued2020
dc.PublishedFrontiers in medicine. 2020, 7:162 1-15.
dc.identifier.issn2296-858X
dc.identifier.urihttps://hdl.handle.net/11250/2760597
dc.description.abstractIntroduction: Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is a disease with high symptom burden, of unknown etiology, with no established treatment. We observed patients with long-standing ME/CFS who got cancer, and who reported improvement of ME/CFS symptoms after chemotherapy including cyclophosphamide, forming the basis for this prospective trial. Materials and methods: This open-label phase II trial included 40 patients with ME/CFS diagnosed by Canadian criteria. Treatment consisted of six intravenous infusions of cyclophosphamide, 600–700 mg/m2, given at four-week intervals with follow-up for 18 months, extended to 4 years. Response was defined by self-reported improvements in symptoms by Fatigue score, supported by Short Form 36 (SF-36) scores, physical activity measures and other instruments. Repeated measures of outcome variables were assessed by General linear models. Responses were correlated with specific Human Leukocyte Antigen (HLA) alleles. Results: The overall response rate by Fatigue score was 55.0% (22 of 40 patients). Fatigue score and other outcome variables showed significant improvements compared to baseline. The SF-36 Physical Function score increased from mean 33.0 at baseline to 51.5 at 18 months (all patients), and from mean 35.0 to 69.5 among responders. Mean steps per 24 h increased from mean 3,199 at baseline to 4,347 at 18 months (all patients), and from 3,622 to 5,589 among responders. At extended follow-up to 4 years 68% (15 of 22 responders) were still in remission. Patients positive for HLA-DQB1*03:03 and/or HLA-C*07:04 (n = 12) had significantly higher response rate compared to patients negative for these alleles (n = 28), 83 vs. 43%, respectively. Nausea and constipation were common grade 1–2 adverse events. There were one suspected unexpected serious adverse reaction (aggravated POTS) and 11 serious adverse events in eight patients. Conclusion: Intravenous cyclophosphamide treatment was feasible for ME/CFS patients and associated with an acceptable toxicity profile. More than half of the patients responded and with prolonged follow-up, a considerable proportion of patients reported ongoing remission. Without a placebo group, clinical response data must be interpreted with caution. We nevertheless believe a future randomized trial is warranted.en_US
dc.language.isoengen_US
dc.publisherFrontiersen_US
dc.rightsNavngivelse 4.0 Internasjonal*
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/deed.no*
dc.titleIntravenous Cyclophosphamide in Myalgic Encephalomyelitis/Chronic Fatigue Syndrome. An Open-Label Phase II Studyen_US
dc.typeJournal articleen_US
dc.typePeer revieweden_US
dc.description.versionpublishedVersionen_US
dc.rights.holderCopyright 2020 The Authorsen_US
dc.source.articlenumber162en_US
cristin.ispublishedtrue
cristin.fulltextoriginal
cristin.qualitycode1
dc.identifier.doi10.3389/fmed.2020.00162
dc.identifier.cristin1832051
dc.source.journalFrontiers in medicineen_US
dc.source.407:162
dc.relation.projectNorges forskningsråd: 272680en_US
dc.identifier.citationFrontiers in medicine. 2020, 7, 162en_US
dc.source.volume7en_US


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Navngivelse 4.0 Internasjonal
Except where otherwise noted, this item's license is described as Navngivelse 4.0 Internasjonal