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一名23岁患有高IgM综合征的男性，表现为无症状的紫罗兰色面部斑块。

A 23-Year-Old Man with Hyper-IgM Syndrome Presenting with Asymptomatic Violaceous Facial Plaques.

作者信息

Barrios Mirelys, Nathan Neera, Trowbridge Ryan, Plovanich Molly, Nazarian Rosalynn M, Kroshinsky Daniela

机构信息

Department of Radiology, Brigham and Womens Hospital, Boston, Massachusetts, USA.

Department of Dermatology, Massachusetts General Hospital, Boston, Massachusetts, USA.

出版信息

Dermatopathology (Basel). 2020 Jan 22;6(4):246-250. doi: 10.1159/000503744. eCollection 2019 Oct-Dec.

DOI:10.1159/000503744
PMID:32083062
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7011746/
Abstract

Tumid lupus is a rare subtype of chronic cutaneous lupus that is characterized by urticaria-like photosensitive plaques. Unlike discoid lupus, it has minimal to no surface change and resolves without scarring. On pathological examination, it may be distinguished from other types of lupus by abundant interstitial mucin deposits. Herein, we describe a case of tumid lupus in a 23-year-old Kuwaiti male with hyper-IgM syndrome. To our best knowledge, this is the first report of tumid lupus in a patient with a primary immunodeficiency.

摘要

肿胀性狼疮是慢性皮肤型狼疮的一种罕见亚型，其特征为荨麻疹样光敏性斑块。与盘状狼疮不同，它几乎没有表面变化，消退后不留瘢痕。病理检查时，它可通过大量间质黏蛋白沉积与其他类型的狼疮相鉴别。在此，我们描述了一例患有高IgM综合征的23岁科威特男性的肿胀性狼疮病例。据我们所知，这是原发性免疫缺陷患者中肿胀性狼疮的首例报告。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/82ec/7011746/1cb15c7acf6e/dpa-0006-0246-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/82ec/7011746/c78d08924f2c/dpa-0006-0246-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/82ec/7011746/1cb15c7acf6e/dpa-0006-0246-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/82ec/7011746/c78d08924f2c/dpa-0006-0246-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/82ec/7011746/1cb15c7acf6e/dpa-0006-0246-g02.jpg

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Lupus erythematosus tumidus: a clinical and histological study of 25 cases.肿胀性红斑狼疮：25例临床与组织学研究
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Tumid lupus erythematosus: an intriguing dermatopathological connotation treated successfully with topical tacrolimus and hydroxyxhloroquine combination.肿胀性红斑狼疮：一种用外用他克莫司和羟氯喹联合治疗成功的有趣皮肤病理学表现。
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Activation-induced cytidine deaminase (AID) is required for B-cell tolerance in humans.激活诱导胞嘧啶脱氨酶 (AID) 对于人类 B 细胞耐受是必需的。
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Alopecia associated with papulonodular mucinosis (tumid) lesions of lupus erythematosus.红斑狼疮的丘疹结节性粘蛋白病（硬结性）皮损相关脱发。
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Comparison of histopathologic-clinical characteristics of Jessner's lymphocytic infiltration of the skin and lupus erythematosus tumidus: Multicenter study of 46 cases.皮肤淋巴细胞浸润症与肿胀性红斑狼疮的组织病理学-临床特征比较：46例多中心研究
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Clinical, immunologic and genetic analysis of 29 patients with autosomal recessive hyper-IgM syndrome due to Activation-Induced Cytidine Deaminase deficiency.29例因激活诱导胞苷脱氨酶缺乏导致的常染色体隐性高IgM综合征患者的临床、免疫学及遗传学分析
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