Hypomyopathic Dermatomyositis with Refractory Dermatitis Treated by Low-dose IL-2
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CASE REPORT
Hypomyopathic Dermatomyositis with Refractory Dermatitis Treated by Low-dose IL-2 Miao Miao
. Yuhui Li . Bo Huang . Jing He
. Zhanguo Li
Received: June 1, 2020 Ó The Author(s) 2020
ABSTRACT Hypomyopathic dermatomyositis (DM) presents with cutaneous lesions consistent with dermatomyositis but in the absence of clinically appreciable muscle weakness. The cutaneous manifestations are often refractory and more resistant to conventional therapy than concomitant muscle involvement. We present a 61-year-old hypomyopathic patient with DM who failed to respond to standard therapy but was successfully treated by low-dose interleukin-2 (IL-2) with no significant side effects. We conclude that low-dose IL-2 is a safe and effective treatment for hypomyopathic DM.
Digital Features To view digital features for this article go to https://doi.org/10.6084/m9.figshare.12562328. M. Miao Y. Li B. Huang J. He (&) Z. Li (&) Department of Rheumatology and Immunology, Peking University People’s Hospital, Beijing 100044, China e-mail: [email protected]. Li e-mail: [email protected] M. Miao Y. Li B. Huang J. He Z. Li Beijing Key Laboratory for Rheumatism Mechanism and Immune Diagnosis (BZ0135), Beijing, China Z. Li Peking-Tsinghua Center for Life Sciences, Peking University, Beijing, China
Keywords: Hypomyopathic dermatomyositis; Refractory dermatitis; Low-dose IL-2 Key Summary Points A patient with hypomyositis dermatomyositis presenting with refractory cutaneous lesions failed to respond to long-term standard therapy. Low-dose interleukin-2 (IL-2), which is an effective treatment for various autoimmune diseases, was also effective in this patient with intractable skin lesions. The clinical improvement by low-dose IL2 was associated to improvement of immune system imbalance by increasing Treg cell proliferation and decreasing the level of Th17 cells.
INTRODUCTION Hypomyopathic dermatomyositis (DM) is a rare form of dermatomyositis with autoimmune features based on serological characterization. The abnormal immunological distribution of circulating CD4? T cell subsets in hypomyopathic DM is similar to that found in classic DM. Previous studies have proven that Th17 cell
Dermatol Ther (Heidelb)
levels are increased and those of Treg cells are decreased in DM [1]. Low-dose interleukin-2 (IL2) is a proven effective treatment for DM, inducing Treg cell proliferation and suppressing Th17 cell level [1–3]. Consequently, immunoregulation driven by low-dose IL-2 represents a potential therapy to maintain selftolerance in autoimmune diseases [3]. However, to date, there has been no report of the effect of IL-2 treatment on hypomyopathic DM. Most patients with skin disease respond to the initial therapy, and some achieve sustained disease control either off all therapy or with low-dose maintenance therapy. Patients who fail to respond to conventional interventions or who relapse after an initial response have refractory disease and require the initiation of more aggressive therapies [4, 5]. Here, we describe a p
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