Ogata Ryo, Kido Takashi, Takeda Kazuaki, Nemoto Kazuki, Heima Riko, Takao Mami, Miyashita Ritsuko, Ozasa Mutsumi, Tokito Takatomo, Okuno Daisuke, Ito Yuya, Yura Hirokazu, Koga Tomohiro, Hashimoto Kunio, Takemoto Shinnosuke, Takazono Takahiro, Ishimoto Hiroshi, Sakamoto Noriho, Fukuda Kazumasa, Sasaki Yuka, Obase Yasushi, Ishimatsu Yuji, Yatera Kazuhiro, Izumikawa Koichi, Mukae Hiroshi
Department of Respiratory Medicine, Nagasaki University Hospital, Nagasaki 852-8501, Japan.
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan, Kitakyusyu 807-8556, Japan.
Microorganisms. 2023 Aug 24;11(9):2145. doi: 10.3390/microorganisms11092145.
Sarcoidosis is a systemic inflammatory disease characterized by noncaseating epithelioid cell granulomas. However, certain infections can exhibit similar histological findings. We present a case of a 69-year-old man who was initially diagnosed with sarcoidosis and later was confirmed, through 16S rRNA sequencing, to have disseminated infection. Acid-fast bacteria were detected in the bone marrow biopsy using Ziehl-Neelsen staining, but routine clinical tests did not provide a definitive diagnosis. The patient tested negative for HIV, anti-interferon-gamma antibodies, and genetic immunodeficiency disorders. He was treated with multiple drugs, including aminoglycosides and macrolides, but showed no improvement in fever and pancytopenia. However, these clinical signs responded favorably to steroid therapy. We reviewed 17 Japanese cases of infection. All cases were in males; 7/17 (41%) were HIV-negative; and 12/17 (71%) had a decreased CD4 count. Genetic analysis confirmed isolation, and macrolides were used universally. infection is challenging to identify and mimics other systemic inflammatory diseases such as sarcoidosis. There are no standard treatment protocols. Our case report and Japanese case review contribute to understanding this rare disease.
结节病是一种以非干酪样上皮样细胞肉芽肿为特征的全身性炎症性疾病。然而，某些感染也可表现出相似的组织学表现。我们报告一例69岁男性患者，最初被诊断为结节病，后来通过16S rRNA测序确诊为播散性感染。在骨髓活检中使用齐-尼氏染色检测到抗酸杆菌，但常规临床检查未能明确诊断。该患者HIV、抗干扰素-γ抗体及遗传性免疫缺陷疾病检测均为阴性。他接受了包括氨基糖苷类和大环内酯类在内的多种药物治疗，但发热和全血细胞减少未见改善。然而，这些临床症状对类固醇治疗反应良好。我们回顾了17例日本感染病例。所有病例均为男性；7/17（41%）为HIV阴性；12/17（71%）CD4计数降低。基因分析证实了分离，普遍使用大环内酯类药物。感染难以识别，可模仿结节病等其他全身性炎症性疾病。目前尚无标准治疗方案。我们的病例报告和日本病例回顾有助于对这种罕见疾病的理解。
