Adult onset Xanthogranuloma presenting as a solitary laryngeal localization: case report and review of literature.

IF 0.8 Q3 MEDICINE, GENERAL & INTERNAL Archive of clinical cases Pub Date : 2022-01-01 DOI:10.22551/2022.37.0904.10221
Gianluca Velletrani, Beatrice Francavilla, Valentina Rosati, Belen Padial, Lucia Anemona, Stefano Di Girolamo
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Abstract

Juvenile Xanthogranuloma (XG) is a rare disorder that belongs to the heterogeneous group of histiocytic neoplasms, characterized by a clonal expansion of non-Langerhans cell histiocytes that share a dermal macrophage phenotype. Although the head and neck region is the most common reported site of involvement by the Juvenile Xanthogranuloma family, laryngeal localization is extremely rare. We report a unique case of Adult Onset Xanthogranuloma with subglottic localization, presenting as a solitary laryngeal mass without other systemic or cutaneous lesions. A review of the previously described cases of laryngeal Xanthogranuloma has been performed, highlighting 7 cases of Juvenile Xanthogranuloma and only 3 cases of Adult Onset Xanthogranuloma. Despite the extreme rarity of laryngeal localization of XG, this histiocytic neoplasm should be considered as a differential diagnosis for laryngeal masses causing airway obstruction, even in the absence of other concomitant manifestations.

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成人发性黄色肉芽肿表现为喉部单发定位:病例报告及文献回顾。
幼年黄色肉芽肿(XG)是一种罕见的疾病,属于异质组织细胞肿瘤,其特征是非朗格汉斯细胞组织细胞的克隆扩增,具有真皮巨噬细胞表型。虽然头部和颈部是青少年黄色肉芽肿家族最常见的受累部位,但喉部定位极为罕见。我们报告一个独特的成人发病黄色肉芽肿声门下定位,表现为一个孤立的喉部肿块没有其他系统或皮肤病变。我们回顾了之前报道的喉黄色肉芽肿病例,其中7例为青少年黄色肉芽肿,只有3例为成人黄色肉芽肿。尽管XG的喉部定位极为罕见,但这种组织细胞性肿瘤应被视为引起气道阻塞的喉部肿块的鉴别诊断,即使没有其他伴随表现。
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