中华皮肤科杂志 ›› 2023, Vol. 56 ›› Issue (5): 415-420.doi: 10.35541/cjd.20220615

• 论著 • 上一篇    下一篇

国内首例慢性播散性副球孢子菌病

焦晓燕1    尹光文1    李冬芹1    李晓改2    孟中勤3    娄丽丽4    阴亚坤1    郑艳1   

  1. 1郑州大学第一附属医院皮肤性病科,郑州  450052;2郑州大学第一附属医院检验科微生物室,郑州  450052;3郑州大学第一附属医院病理科,郑州  450052;4郑州大学第一附属医院呼吸内科,郑州  450052
  • 收稿日期:2022-09-02 修回日期:2023-03-11 发布日期:2023-05-04
  • 通讯作者: 尹光文 E-mail:gwyin67@126.com

Chronic disseminated paracoccidioidomycosis: the first case reported in China

Jiao Xiaoyan1, Yin Guangwen1, Li Dongqin1, Li Xiaogai2, Meng Zhongqin3, Lou Lili4, Yin Yakun1, Zheng Yan1   

  1. 1Department of Dermatovenereology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China;2Department of Clinical Laboratory, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China;3Department of Pathology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China;4Department of Respiratory, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China
  • Received:2022-09-02 Revised:2023-03-11 Published:2023-05-04
  • Contact: Yin Guangwen E-mail:gwyin67@126.com

摘要: 【摘要】 报道国内首例慢性播散性副球孢子菌病。患者男,49岁,因皮肤丘疹、结节1年,口腔黏膜丘疹、溃疡2个月入院。皮肤科检查:左足水肿,左足底多发溃疡,表面结痂,左足第3、4趾间及第4、5趾间溃疡,基底呈颗粒状,伴点状出血、渗出;左足背、左足内侧及左膝多发丘疹、结节、斑块,中央溃疡、结痂;左腕部2个丘疹,上唇左侧1个丘疹,表面结痂;牙龈、颊黏膜、唇黏膜及上颚可见红色斑块伴溃疡、点状出血,皮损以左侧为主。浅表淋巴结彩超:双侧颈部及锁骨上窝淋巴结肿大,左侧为著。胸腹部计算机断层扫描图像示:双肺弥漫粟粒样结节影及条索状、云絮状、结节状高密度影,左侧肾上腺明显增粗。口腔、左下肢皮损组织真菌免疫荧光染色,可见酵母细胞。口腔黏膜、左下肢皮损组织病理:肉芽肿性炎,多核巨细胞内外可见酵母细胞,折射双膜,无芽、单芽或多芽;过碘酸希夫染色、六胺银染色阳性。左下肢皮损组织真菌培养:25 ℃、37 ℃沙氏葡萄糖琼脂培养基中培养阳性,均为菌丝相。口腔黏膜及肺泡灌洗液宏基因组学测序:巴西副球孢子菌。诊断:慢性播散性副球孢子菌病。予伊曲康唑胶囊400 mg/d口服,1个月后皮肤、黏膜皮损明显好转,肺部及左侧肾上腺影像学表现亦好转,3个月后伊曲康唑减量至200 mg/d。随访10个月,病情进一步好转。

关键词: 副球孢子菌病, 副球孢子菌, 皮肤, 口腔, 肺, 伊曲康唑

Abstract: 【Abstract】 To report the first case of chronic disseminated paracoccidioidomycosis in China. A 49-year-old male patient presented with papules and nodules of the skin for 1 year, and papules and ulcers on the oral mucosa for 2 months. Skin examination showed the edema of the left foot, multiple crusting ulcers on the sole of the left foot, ulcers with a granular base in the interdigital regions between the third and fourth toes as well as fourth and fifth toes of the left foot, accompanied by punctate hemorrhage and exudation; there were multiple papules, nodules, and plaques on the dorsum and medial side of the left foot and the left knee, with ulcers and crusts in the center; 2 papules were observed on the left wrist, and 1 papule on the left upper lip with a crusted surface; red plaques with ulcers and punctate hemorrhage were observed on the gingival mucosa, buccal mucosa, labial mucosa, and palate, and the lesions mainly occurred on the left side. Ultrasonography of superficial lymph nodes showed bilateral cervical and supraclavicular lymph node enlargement, which was more obvious on the left side. Computed tomography of the chest and abdomen showed diffuse miliary nodular shadows, and cordlike, cloudy flocculent and nodular high-density shadows in both lungs, as well as obvious thickening of the left adrenal gland in the abdomen. Yeast cells were observed by immunofluorescent staining of biopsy tissues from the oral mucosa and left lower limb. Histopathological examination of biopsy tissues from the oral mucosa and left lower limb showed granulomatous inflammation, and refractive double-membrane yeast cells could be observed inside or outside the multinucleated giant cells, without or with a single bud or multiple buds; periodic acid-Schiff staining and hexamine silver staining of the above biopsy tissues were positive. Fungal culture of the left lower limb lesion in Sabouraud dextrose agar medium at 25℃ and 37℃ both yielded fungal hyphae. Metagenomics sequencing of the oral mucosal tissue and alveolar lavage fluid indicated the infection with Paracoccidioides brasiliensis. The diagnosis of chronic disseminated paracoccidioidomycosis was confirmed. After 1-month oral treatment with itraconazole capsules at a dose of 400 mg/d, the lesions on the skin and oral mucosa markedly improved, and computed tomography imaging of the lung and left adrenal gland also showed obvious improvement. The dose of itraconazole was reduced to 200 mg/d after 3 months. The patient′s condition further improved during a 10-month follow-up.

Key words: Paracoccidioidomycosis, Paracoccidioides, Skin, Mouth, Lung, Itraconazole

引用本文

焦晓燕 尹光文 李冬芹 李晓改 孟中勤 娄丽丽 阴亚坤 郑艳. 国内首例慢性播散性副球孢子菌病[J]. 中华皮肤科杂志, 2023,56(5):415-420. doi:10.35541/cjd.20220615

Jiao Xiaoyan, Yin Guangwen, Li Dongqin, Li Xiaogai, Meng Zhongqin, Lou Lili, Yin Yakun, Zheng Yan. Chronic disseminated paracoccidioidomycosis: the first case reported in China[J]. Chinese Journal of Dermatology, 2023, 56(5): 415-420.doi:10.35541/cjd.20220615