Eddou Hicham, Le Guyadec Thierry, Cremades Anne, Saint Blancard Pierre, Malfuson Jean Valère, Konopacki Joanna, de Revel Thierry
Service d'hématologie, Hôpital d'instruction des armées Percy, Clamart, France.
Ann Biol Clin (Paris). 2013 May-Jun;71(3):333-7. doi: 10.1684/abc.2013.0817.
Hyper-IgG4 syndrome is a rare entity characterized by fibro-inflammatory lesions of organs, an excess of IgG4 positive plasma cells in histology and high serum level IgG4. Many organs can be affected (pancreas, kidney, salivary glands) and the list continues to grow. The skin damage is rarely reported in the literature and is usually associated with other typical lesions of this syndrome. We report the case of a 53-year-old female followed since 2005 for lymphadenopathy, associated with axillary nodular skin lesions. The assessments made at that time had retained the diagnosis of pseudolymphoma with implementation of multi-line therapy. Six years later, and the persistence of the lesions, plasma cells marked by anti-IgG4 and the serum IgG4 has attached injuries to the syndrome hyper-IgG4. The patient is treated with low dose corticosteroids with a good and protacted response. Cutaneous pseudolymphoma could be a new presentation of the syndrome of hyper-IgG4 in the absence of any other injury usually associated with this entity. This case illustrates the interest for proposing a plasma cell labeling with anti-IgG4 in any case of cutaneous pseudolymphoma.
IgG4相关性疾病是一种罕见的疾病,其特征为器官的纤维炎症性病变、组织学上IgG4阳性浆细胞增多以及血清IgG4水平升高。许多器官均可受累(胰腺、肾脏、唾液腺等),受累器官的范围还在不断扩大。皮肤损害在文献中鲜有报道,且通常与该综合征的其他典型病变相关。我们报告一例53岁女性患者,自2005年起因淋巴结病就诊,伴有腋窝结节性皮肤损害。当时的评估诊断为假性淋巴瘤,并实施了多线治疗。6年后,病变持续存在,抗IgG4标记的浆细胞及血清IgG4水平升高,提示IgG4相关性疾病。患者接受低剂量糖皮质激素治疗,反应良好且持久。在无通常与该疾病相关的任何其他损害的情况下,皮肤假性淋巴瘤可能是IgG4相关性疾病的一种新表现形式。该病例表明,对于任何皮肤假性淋巴瘤病例,均有必要进行抗IgG4浆细胞标记。