Exploring the landscape of flagellate erythema as a rare cutaneous symptom of idiopathic inflammatory myopathy: a case series-based review.
- 1Department of Rheumatology, Medical University of Lodz, Zeromskiego 113, 90-549, Lodz, Poland. [email protected].
- 2Department of Rheumatology, Medical University of Lodz, Zeromskiego 113, 90-549, Lodz, Poland.
Abstract
Flagellate erythema is an exceptionally rare cutaneous manifestation of idiopathic inflammatory myopathies (IIM) with poorly understood clinical significance. We report two patients who developed zebra-like flagellate erythema: one with immune-mediated necrotising myopathy (IMNM) and one with anti-Mi-2-positive cancer-associated myositis (CAM). To place these observations in broader context, we performed a thorough literature search across PubMed, Scopus and Embase and identified 34 previously published cases of IIM-associated flagellate erythema. The most frequent underlying diagnosis was dermatomyositis (n = 14, 41.2%), followed by CAM (n = 9, 26.5%), amyopathic/clinically amyopathic dermatomyositis (n = 6, 17.6%), and juvenile dermatomyositis (n = 5, 14.7%). Anti-TIF1-γ antibodies were identified in 6 of 34 patients (17.6%) and anti-MDA5 antibodies in 7 of 34 (20.6%); malignancy was reported in 9 of 34 (26.5%), while a substantial proportion of cases were seronegative or lacked antibody testing. Muscle weakness or myalgia was present in most of the combined cohort, and a fatal outcome was reported in approximately one-fifth of cases. Our cases expand the clinical and serological spectrum of myositis-associated flagellate erythema, including the first report of this presentation in IMNM. While flagellate erythema has most often been reported with CAM or anti-TIF1-γ antibodies, the literature remains inconsistent, and current case-report-based evidence does not support a definitive association with any single serological or clinical subgroup. In our patients, flagellate erythema coincided with disease exacerbation; although this should be interpreted cautiously given the limited evidence, its occurrence may warrant careful evaluation for IIM flare and, where appropriate, malignancy screening.
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BibTeX
@article{opincrosiak2026exploring,
title = {{Exploring the landscape of flagellate erythema as a rare cutaneous symptom of idiopathic inflammatory myopathy: a case series-based review.}},
author = {Aleksandra Opinc-Rosiak and Milena Kaszubska and Aleksandra Nadel and Aleksander Sztajnert and Joanna Makowska and Anna Lewandowska-Polak},
journal = {Rheumatology international},
year = {2026},
month = oct,
doi = {10.1007/s00296-026-06328-5},
url = {https://europepmc.org/article/MED/42873265},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Exploring the landscape of flagellate erythema as a rare cutaneous symptom of idiopathic inflammatory myopathy: a case series-based review.
AU - Aleksandra Opinc-Rosiak
AU - Milena Kaszubska
AU - Aleksandra Nadel
AU - Aleksander Sztajnert
AU - Joanna Makowska
AU - Anna Lewandowska-Polak
PY - 2026
DA - 2026/10/14
JO - Rheumatology international
DO - 10.1007/s00296-026-06328-5
UR - https://europepmc.org/article/MED/42873265
AB - Flagellate erythema is an exceptionally rare cutaneous manifestation of idiopathic inflammatory myopathies (IIM) with poorly understood clinical significance. We report two patients who developed zebra-like flagellate erythema: one with immune-mediated necrotising myopathy (IMNM) and one with anti-Mi-2-positive cancer-associated myositis (CAM). To place these observations in broader context, we performed a thorough literature search across PubMed, Scopus and Embase and identified 34 previously published cases of IIM-associated flagellate erythema. The most frequent underlying diagnosis was dermatomyositis (n = 14, 41.2%), followed by CAM (n = 9, 26.5%), amyopathic/clinically amyopathic dermatomyositis (n = 6, 17.6%), and juvenile dermatomyositis (n = 5, 14.7%). Anti-TIF1-γ antibodies were identified in 6 of 34 patients (17.6%) and anti-MDA5 antibodies in 7 of 34 (20.6%); malignancy was reported in 9 of 34 (26.5%), while a substantial proportion of cases were seronegative or lacked antibody testing. Muscle weakness or myalgia was present in most of the combined cohort, and a fatal outcome was reported in approximately one-fifth of cases. Our cases expand the clinical and serological spectrum of myositis-associated flagellate erythema, including the first report of this presentation in IMNM. While flagellate erythema has most often been reported with CAM or anti-TIF1-γ antibodies, the literature remains inconsistent, and current case-report-based evidence does not support a definitive association with any single serological or clinical subgroup. In our patients, flagellate erythema coincided with disease exacerbation; although this should be interpreted cautiously given the limited evidence, its occurrence may warrant careful evaluation for IIM flare and, where appropriate, malignancy screening.
SN - 0172-8172
SN - 1437-160X
ER -