Medical Intensive Care Unit, Department of Respiratory and Critical Care Medicine, Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, China.
Institute of Respiratory Diseases, Sun Yat-Sen University, Guangzhou, China.
Front Immunol. 2021 Jun 10;12:676132. doi: 10.3389/fimmu.2021.676132. eCollection 2021.
Tuberculosis (TB) is a leading cause of morbidity and mortality in underdeveloped and developing countries. Disseminated TB may induce uncommon and potentially fatal secondary hemophagocytic lymphohistiocytosis (HLH). Timely treatment with anti-tuberculosis therapy (ATT) and downmodulation of the immune response is critical. However, corticosteroid treatment for TB-associated HLH remains controversial. Herein, we report a successful case of disseminated TB-associated HLH in a pregnant woman with Evans syndrome accompanied by a literature review.
A 26-year-old pregnant woman with Evans syndrome was transferred to the Third Affiliated Hospital of Sun Yat-Sen University because of severe pneumonia. She presented with cough, fever, and aggravated dyspnea. Nested polymerase chain reaction for () complex in sputum was positive. Sputum smear sample for acid-fast bacilli was also positive. Metagenome next-generation sequencing (mNGS) of the bronchoalveolar lavage fluid identified 926 DNA sequence reads and 195 RNA sequence reads corresponding to complex, respectively. mNGS of blood identified 48 DNA sequence reads corresponding to . There was no sequence read corresponding to other potential pathogens. She was initially administered standard ATT together with a low dose of methylprednisolone (40 mg/day). However, her condition deteriorated rapidly with high fever, acute respiratory distress syndrome, pancytopenia, and hyperferritinemia. Bone marrow smears showed hemophagocytosis. And caseating tuberculous granulomas were found in the placenta. A diagnosis of disseminated TB-associated HLH was made. Along with the continuation of four drug ATT regimen, therapy with a higher dose of methylprednisolone (160 mg/day) combined with immunoglobulin and plasma exchange was managed. The patient's condition improved, and she was discharged on day 19. Her condition was good at follow-up with the continuation of the ATT.
Clinicians encountering patients with suspected TB accompanied by unexplainable inflammation not responding to ATT should consider complications with HLH. Timely administration of ATT combined with corticosteroids may result in a favorable outcome.
结核病（TB）是不发达国家和发展中国家发病率和死亡率的主要原因。播散性结核病可能会引起罕见且潜在致命的继发性噬血细胞性淋巴组织细胞增生症（HLH）。及时进行抗结核治疗（ATT）和降低免疫反应至关重要。然而，皮质类固醇治疗与结核病相关的 HLH 仍然存在争议。在此，我们报告了一例成功的妊娠合并 Evans 综合征的播散性结核病相关 HLH 病例，并进行了文献复习。
一位 26 岁的妊娠合并 Evans 综合征的孕妇因严重肺炎转入中山大学附属第三医院。她表现为咳嗽、发热和呼吸困难加重。痰中嵌套聚合酶链反应（PCR）检测结核分枝杆菌复合体阳性。痰涂片抗酸杆菌阳性。支气管肺泡灌洗液的宏基因组下一代测序（mNGS）分别鉴定出 926 个 DNA 序列读段和 195 个 RNA 序列读段与结核分枝杆菌复合体相对应。血液 mNGS 鉴定出 48 个 DNA 序列读段与结核分枝杆菌相对应。没有与其他潜在病原体相对应的序列读段。她最初接受标准 ATT 联合低剂量甲基强的松龙（40mg/天）治疗。然而，她的病情迅速恶化，出现高热、急性呼吸窘迫综合征、全血细胞减少和铁蛋白血症。骨髓涂片显示噬血细胞现象。胎盘内可见干酪样结核肉芽肿。诊断为播散性结核病相关 HLH。随着四联抗结核方案的继续，给予更高剂量的甲基强的松龙（160mg/天）联合免疫球蛋白和血浆置换治疗。患者病情改善，第 19 天出院。继续 ATT 治疗后随访情况良好。
遇到疑似结核病且 ATT 治疗无效的不明原因炎症的患者，临床医生应考虑并发 HLH 的可能性。及时给予 ATT 联合皮质类固醇治疗可能会取得良好的结果。
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