- 期刊
Solitary Angiokeratoma of the Tongue-A Case Report and Review of the Literatures
舌部孤立性血管角化瘤－病例報告及文獻回顧
摘要
Angiokeratoma (AK) is a rare, cutaneous disorder that is often associated with a number of metabolic disorders. Despite the rare occurrence of solitary oral AK, when found in the oral cavity, it most commonly affects the tongue. Here, we present a rare case of a solitary AK of the tongue base affecting a 50-year-old male with a history of left gingival squamous cell carcinoma who underwent tumor-wide excision and free fibular flap reconstruction in February 2018. During the six-month postoperative follow-up interval, a 2 × 2 cm mass along the left posterior tongue border and base speckled with red spots was observed upon physical examination. An incisional biopsy was performed and histopathology showed hyperparakeratosis, acanthosis and dilated vascular channels lying directly in the subepithelial region. Immunohistochemistry revealed that the endothelial cells of the vascular channels were positive for CD31 as well as CD34 but negative for D2-40. Subsequently, the patient underwent tumor-wide excision of the mass and the final pathologic diagnosis was compatible with a solitary AK of the tongue. Reviews of the currently available literature of AK which have been described in the oral cavity indicate that etiological factors include trauma, chronic irritation, high venous pressure or vascular malformations. If lesions are seen elsewhere, it may be related to a systemic disease such as Fabry's disease or fucosidosis. This is a rare case and should be differentiated from malignant tumors in the oral cavity. Only three other cases of solitary AK of the tongue have been reported in English literature to date.
並列摘要
孤立性血管角化瘤是一種罕見的皮膚疾病，通常與許多代謝性疾病相關。儘管在口腔中很少發現孤立性血管角化瘤，但如在口腔發生，最好發於舌頭。本病例為一名50歲男性，患有左側齒齦上皮鱗狀細胞癌的病史，於2018年2月進行了左側改良型頸部淋巴廓清術、腫瘤廣泛性切除和左側腓骨游離皮瓣重建。在術後六個月的回診檢查發現左後舌緣和底部有一2×2公分且表面呈紅斑點狀之腫塊。病理切片檢查發現組織部分有角化不全和上皮下區域血管擴張。免疫組織化學染色顯示，血管的內皮細胞對CD31和CD34呈陽性，但對D2-40呈陰性。隨後患者接受了腫塊廣泛性切除，最終的病理診斷與舌部孤立性血管角化瘤相符。現有的文獻回顧顯示，舌部孤立性血管角化瘤的病因包括創傷、慢性刺激、高靜脈壓或血管畸形。若病灶呈現在身體的其它部位，則可能與全身性疾病，例如法布瑞氏症或岩藻糖代謝異常有關。此罕見的情況，應與口腔惡性腫瘤的診斷區別。迄今為止，英語文獻中僅記載了另外三例舌部孤立性血管角化瘤。