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Fatal Hemophagocytic Lymphohistiocytosis in a Patient with Miliary Tuberculosis: a Case Report

ABSTRACT: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening hyperinflammatory syndrome induced by cytotoxic T-cells. Mostly, HLH is secondary to infections, malignancies, or autoimmune disorders. HLH triggered by miliary tuberculosis is rare and mortality rates are high. We report a case of a 58-year-old, Caucasian patient admitted to the ICU with respiratory failure. After extensive tests, the diagnosis of HLH was made. Despite aggressive treatment with antibiotics, etoposide, anakinra, and tocilizumab, our patient succumbed to the illness after 18 days in the ICU. Postmortem, a diagnosis of miliary tuberculosis was made, despite negative PCR and culture of mycobacteria during clinical course. Our case demonstrates the challenges of early diagnosis of HLH and the importance of considering miliary tuberculosis as a possible underlying trigger.

Supplementary Information

The online version contains supplementary material available at 10.1007/s42399-022-01232-y.

SUBMITTER: Schippers E

PROVIDER: S-EPMC9281255 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:BackgroundTuberculosis (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.Case presentationA 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 Mycobacterium tuberculosis (M. tuberculosis) 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 M. tuberculosis complex, respectively. mNGS of blood identified 48 DNA sequence reads corresponding to M. tuberculosis. 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.ConclusionsClinicians 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.

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Fatal Hemophagocytic Lymphohistiocytosis in a Patient with Miliary Tuberculosis: a Case Report

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