Dimitriadis Dimitrios, Terzi Irene, Dragoumani Ioulia, Liapis Konstantinos, Papazoglou Dimitrios
Second University Department of Internal Medicine, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece.
Department of Haematology, University Hospital of Alexandroupolis, Democritus University of Thrace, Alexandroupoli, Greece.
Eur J Case Rep Intern Med. 2025 Jun 18;12(7):005590. doi: 10.12890/2025_005590. eCollection 2025.
Tumour necrosis factor-alpha (TNF-alpha) inhibitors increase susceptibility to granulomatous infections, including both (MTB) and nontuberculous mycobacteria. We describe a complex case of sequential disseminated (MAC) and central nervous system MTB infection in a patient treated with adalimumab, complicated by hemophagocytic lymphohistiocytosis (HLH).
A 65-year-old man on long-term adalimumab for psoriasis presented with prolonged fever, hepatosplenomegaly, cytopenia and elevated inflammatory markers. Bone marrow aspiration revealed hemophagocytosis and liver and bone marrow biopsy revealed granulomatous inflammation. Polymerase chain reaction (PCR) testing of bronchoalveolar lavage (BAL) fluid identified MAC, supporting a diagnosis of disseminated MAC-associated HLH. The patient responded to triple MAC therapy (azithromycin, ethambutol, rifampicin), intravenous immunoglobulin and low-dose corticosteroids, with rapid clinical improvement. Three months later, he was readmitted with fever and altered mental status. Brain magnetic resonance imaging showed meningeal thickening. Lumbar puncture revealed cerebrospinal fluid pleocytosis, hypoglycorrhachia and elevated protein. PCR detected MTB complex deoxyribonucleic acid and a rifampicin resistance gene, prompting the initiation of a four-drug antituberculosis regimen (isoniazid, pyrazinamide, levofloxacin, ethambutol) and high-dose dexamethasone. The patient improved and was discharged after a month of hospitalization, remaining clinically stable at 1-year follow-up.
This case highlights the risk of sequential or overlapping MAC and MTB infections in patients receiving TNF-alpha inhibitors, the potential for HLH as a serious complication, and the diagnostic value and limitations of BAL PCR testing. Vigilant screening and multidisciplinary management are essential in such high-risk populations.
The increased risk of opportunistic infections associated with long-term tumour necrosis factor-alpha inhibitor use necessitates ongoing infection screening.Hemophagocytic lymphohistiocytosis triggered by mycobacterial infections requires prompt recognition and targeted treatment.Polymerase chain reaction results should be interpreted with caution in complex clinical scenarios, as deoxyribonucleic acid detection may indicate colonization rather than active infection.
肿瘤坏死因子-α（TNF-α）抑制剂会增加机体对肉芽肿性感染的易感性，包括结核分枝杆菌（MTB）和非结核分枝杆菌感染。我们描述了1例在用阿达木单抗治疗的患者中先后发生播散性鸟分枝杆菌复合群（MAC）感染和中枢神经系统MTB感染的复杂病例，该病例并发噬血细胞性淋巴组织细胞增生症（HLH）。
1例65岁因银屑病长期使用阿达木单抗的男性患者，出现长期发热、肝脾肿大、血细胞减少及炎症指标升高。骨髓穿刺显示噬血细胞现象，肝脏和骨髓活检显示肉芽肿性炎症。支气管肺泡灌洗（BAL）液的聚合酶链反应（PCR）检测发现MAC，支持播散性MAC相关HLH的诊断。患者接受三联MAC治疗（阿奇霉素、乙胺丁醇、利福平）、静脉注射免疫球蛋白和小剂量糖皮质激素治疗后，临床症状迅速改善。3个月后，患者因发热和精神状态改变再次入院。脑部磁共振成像显示脑膜增厚。腰椎穿刺显示脑脊液细胞增多、脑脊液低糖和蛋白升高。PCR检测到MTB复合群脱氧核糖核酸及利福平耐药基因，于是开始四联抗结核治疗方案（异烟肼、吡嗪酰胺、左氧氟沙星、乙胺丁醇）及大剂量地塞米松治疗。患者病情改善，住院1个月后出院，在1年的随访中临床症状保持稳定。
该病例突出了接受TNF-α抑制剂治疗的患者发生先后或重叠的MAC和MTB感染的风险、HLH作为严重并发症的可能性，以及BAL PCR检测的诊断价值和局限性。在这类高危人群中，进行警惕性筛查和多学科管理至关重要。
长期使用肿瘤坏死因子-α抑制剂导致机会性感染风险增加，因此需要持续进行感染筛查。由分枝杆菌感染引发的噬血细胞性淋巴组织细胞增生症需要及时识别并进行针对性治疗。在复杂的临床情况下，应谨慎解读聚合酶链反应结果，因为脱氧核糖核酸检测可能表明为定植而非活动性感染。
