Oral verruciform xanthoma is an uncommon mucocutaneous lesion with low recurrence rate. Until 2018, less than 500 cases have been published in the English literature, and only 4 including the present case were found recurrent. In this case we present a middle-age male patient with verruciform xanthoma on palatal gingiva, and the recurrence lesion was noticed six months after total excision and extraction of teeth in relation. The lesion was in consistent with all previous reports, which verruciform xanthoma was described with sessile or pedunculated manifestation with a rough pebbled surface. Due to the low prevalence, verruciform xanthoma was seldom taken as provisional diagnosis, instead , the similar feature of hyperkeratotic facade usually leads to misdiagnosis as squamous papilloma, verrucous hyperplasia and early carcinoma. Histologically, our case presents characteristically with abundance of the lipid-laden macrophages within the connective tissue papillae, which is positively highlighted in immunohistochemical stain of anti-CD68. Though the etiology was unclear, most of the authors believed it is an unusual reaction or immune response secondary to chronic inflammation and relates to damage of epithelial cells. In this case, the patient meets several potential etiological factors such as the chronic inflammatory state developed by periodontitis, and the constant trauma especially due to alcohol consumption, betel quid chewing and cigarette smoking. In conclusion, we believe the present case is valuable for correct diagnosis and treatment of verruciform xanthoma.
口腔疣狀黃瘤為發生率極低且不易復發之上皮粘膜病灶,至2018年底,僅少於500件病例被發表於英文期刊,而復發之病例報告截至今時更是僅有4例。本病例報告一中年男性患者罹患左上顎顎側牙齦之疣狀黃瘤,且於病灶切除及拔除患齒六個月後再復發之案例。口腔疣狀黃瘤之特徵為具有粗糙鵝卵石表面的無柄或帶蒂表現。由於其流行率低,疣狀黃瘤甚少於臨床診斷中被鑑別出來,反而常因其過度角化外觀的相似特徵而被誤診為鱗狀乳頭狀瘤,疣狀增生或早期癌。在組織學上,我們的病例符合疣狀黃瘤之代表性特徵-於結締組織乳頭內可見富含脂質之巨噬細胞,並在抗CD68的免疫組織化學染色中再次印證。儘管致病機轉尚不清楚,大多數學者認為口腔疣狀黃瘤是肇始於慢性炎症的一種不尋常免疫反應,且與上皮細胞的損傷有關。本案例中,患者尤其符合上述幾種潛在病因,例如由牙周炎引起之慢性炎症狀態,以及因飲酒,嚼食檳榔和吸煙引起的持續上皮損傷。