Tuberculosis-Associated HLH in an 8-Month-Old Infant: A Case Report and Review

Hauch, Holger; Skrzypek, S.; Woessmann, Wilhelm; Lehmberg, Kai; Ehl, Stephan; Speckmann, Carsten; Schneck, Emmanuel; Koerholz, Dieter; Jux, Christian; Neuhäuser, Christoph

doi:10.3389/fped.2020.556155

Cited by 4 publications

(3 citation statements)

References 55 publications

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“…The number of criteria fulfilled by patients ranged from two to eight, with five being the most common. Table S3 provides an overview of each of the criteria for every patient in this study [ 3 , 4 , 5 , 6 , 7 , 10 , 13 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 ...…”

Section: Resultsmentioning

confidence: 99%

“…The survival rate in this study population was 55%, similar to earlier reviews on TB-HLH [ 5 , 6 , 10 ]. Compared to some reviews on special patient populations, this differs, though these reviews entail only a few patients, with reviews on neonates and infants with TB-HLH, where 6/9 (67%) patients survived [ 39 ], and in patients under 20 years of age, where 9/11 (82%) patients survived [ 22 ].…”

Section: Discussionmentioning

confidence: 99%

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Tuberculosis-Associated Hemophagocytic Lymphohistiocytosis: A Review of Current Literature

Fauchald,

Blomberg,

Reikvam

2023

JCM

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Hemophagocytic lymphohistiocytosis (HLH) is a condition of immune dysregulation and hyperinflammation, leading to organ failure and death. Malignancy, autoimmune conditions, and infections, including Mycobacterium tuberculosis (TB), are all considered triggers of HLH. The aim of this study was to review all reported cases of TB-associated HLH in English literature, and to summarize the epidemiology, diagnostics, treatment, and mortality in patients with concomitant HLH and TB. A systematic review of described cases with TB-associated HLH, via a structured literature search in the medical database PubMed, is presented. Additional articles were included through cross-referencing with existing review articles. Articles were reviewed based on a predetermined set of criteria. A total of 116 patients with TB-associated HLH were identified with a male:female ratio of about 3:2. The age at presentation ranged from 12 days to 83 years. Malignancy, autoimmunity, and renal failure were the most common comorbid conditions. Most patients received both tuberculostatic and specific immunomodulating treatment, which was associated with a 66% (48/73) survival rate compared to 56% (15/27) in those receiving only tuberculostatic treatment, and 0% (0/13) in those receiving only immunomodulating treatment. The survival rate was 55% overall. The overlapping presentation between disseminated TB and HLH poses challenging diagnostics and may delay diagnosis and treatment, leading to increased mortality. TB should be considered as a potential trigger of HLH; clinicians’ knowledge and awareness of this may result in the appropriate investigations needed to ensure diagnosis and proper treatment.

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Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Tuberculosis-Associated Hemophagocytic Lymphohistiocytosis: A Review of Current Literature

Fauchald,

Blomberg,

Reikvam

2023

JCM

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“…Most of them were in critical situation at the time of HPS onset. Some patients required mechanical ventilation and extracorporeal membrane oxygenation (ECMO) support and prolonged hospital stay for survival (7)(8)(9)(10).…”

Section: Discussionmentioning

confidence: 99%

Pediatric isoniazid-resistant tuberculosis of the bone marrow manifesting as hemophagocytic syndrome: A case report

Zheng

Yang

et al. 2022

Front. Pediatr.

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Hemophagocytic syndrome (HPS) is a critical syndrome of ineffective hyperinflammatory immune response resulting in infiltration of lymphocytes and histiocytes in various organs. Causes can be hereditary or due to malignancy, autoimmune disease, or infection. HPS due to Mycobacterium tuberculosis is rare as only a handful of cases are reported, and they are mostly associated with severe disseminated tuberculosis (TB). We reported a 9-year-old boy with tuberculosis of the bone marrow accompanied with hemophagocytic syndrome. The patient presented with manifestation of HPS and had no respiratory symptoms or risk factors for TB but was later diagnosed of isoniazid-resistant TB in the bone marrow. He had a good outcome after receiving anti-TB drugs and corticosteroids on time. This case highlights that bone marrow might be a shelter for Mycobacterium tuberculosis. Concurrent testing for drug susceptibility in TB cases with an uncommon manifestation is recommended even for first episodes. Early diagnosis and etiological confirmation of the infection origin and appropriate treatment are essential to improve survival in this otherwise life-threatening condition.

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Paradoxical upgrading reaction following treatment of disseminated tuberculosis-associated haemophagocytic lymphohistiocytosis in an infant without HIV: a case report and review of the literature

Chidambaram

Maya

Sugumar

et al. 2022

Paediatrics and International Child Health

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Tuberculosis-Associated HLH in an 8-Month-Old Infant: A Case Report and Review

Cited by 4 publications

References 55 publications

Tuberculosis-Associated Hemophagocytic Lymphohistiocytosis: A Review of Current Literature

Tuberculosis-Associated Hemophagocytic Lymphohistiocytosis: A Review of Current Literature

Pediatric isoniazid-resistant tuberculosis of the bone marrow manifesting as hemophagocytic syndrome: A case report

Paradoxical upgrading reaction following treatment of disseminated tuberculosis-associated haemophagocytic lymphohistiocytosis in an infant without HIV: a case report and review of the literature

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