Anaplasmosis-Induced Hemophagocytic Lymphohistiocytosis: A Case Report and Review of the Literature

Scribner, Jacob; Wu, Benita; Lamyaithong, Andre; Arcega, Victor; Villanueva, Daphne-Dominique H

doi:10.1093/ofid/ofad213

Cited by 3 publications

(1 citation statement)

References 27 publications

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“…The following were excluded from the analysis: articles published in languages other than English; case reports not providing an appropriate amount of information, or where the diagnosis was not certain; and those reporting non-human subjects. The final number of articles included was 69, which resulted in a total number of 98 patients ( Figure 1 ) [ 15 , 16 , 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 , 66 , 67 , 68 , 69 , ...…”

Section: Methodsmentioning

confidence: 99%

Hemophagocytic Lymphohistiocytosis (HLH) in Patients with Tick-Borne Illness: A Scoping Review of 98 Cases

Jevtic,

da Silva,

Haylock

et al. 2024

Infectious Disease Reports

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Hemophagocytic lymphohistiocytosis (HLH) secondary to tick-borne infections is a rare but potentially life-threatening syndrome. We performed a scoping review according to PRISMA guidelines to systematically analyze the existing literature on the topic. A total of 98 patients were included, with a mean age of 43.7 years, of which 64% were men. Most cases, 31%, were reported from the USA. Immunosuppression was present in 21.4%, with the most common cause being previous solid organ transplantation. Constitutional symptoms were the most common, observed in 83.7% of the patients, while fever was reported in 70.4% of cases. Sepsis was present in 27.6%. The most common laboratory abnormalities in this cohort were thrombocytopenia in 81.6% of patients, while anemia, leukopenia, and leukocytosis were observed in 75.5%, 55.1%, and 10.2%, respectively. Liver enzyme elevation was noted in 63.3% of cases. The H-score was analyzed in 64 patients, with the mean value being 209, and bone marrow analysis was performed in 61.2% of patients. Ehrlichia spp. was the main isolated agent associated with HLH in 45.9%, followed by Rickettsia spp. in 14.3% and Anaplasma phagocytophilum in 12.2%. Notably, no patient with Powassan virus infection or Lyme borreliosis developed HLH. The most common complications were acute kidney injury (AKI) in 35.7% of patients, shock with multiple organ dysfunction in 22.5%, encephalopathy/seizure in 20.4%, respiratory failure in 16.3%, and cardiac complications in 7.1% of patients. Treatment included antibiotic therapy alone in 43.9%, while 5.1% of patients were treated with immunosuppressants alone. Treatment with both antibiotics and immunosuppressants was used in 51% of patients. Appropriate empiric antibiotics were used in 62.2%. In 43.9% of cases of HLH due to tick-borne disease, patients received only antimicrobial therapy, and 88.4% of those recovered completely without the need for immunosuppressive therapy. The mortality rate in our review was 16.3%, and patients who received inappropriate or delayed empiric therapy had a worse outcome. Hence, we suggest empiric antibiotic treatment in patients who are suspected of having HLH due to tick-borne disease or in whom diagnostic uncertainty persists due to diagnostic delay in order to minimize mortality.

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Section: Methodsmentioning

confidence: 99%

Hemophagocytic Lymphohistiocytosis (HLH) in Patients with Tick-Borne Illness: A Scoping Review of 98 Cases

Jevtic,

da Silva,

Haylock

et al. 2024

Infectious Disease Reports

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Tick-Borne Illnesses in Emergency and Wilderness Medicine

Sullivan,

Glose,

Sward

2024

Emergency Medicine Clinics of North America

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Hemophagocytic Lymphohistiocytosis Secondary to Anaplasmosis With Lyme Coinfection

Baum,

Argotsinger,

Taha

et al. 2024

Infect Dis Clin Pract

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Human granulocytic anaplasmosis and Lyme disease are tick-borne infections transmitted by the same vector, Ixodes scapularis, commonly isolated in areas within the Midwest region of the United States. Although typically associated with mild symptoms, both can progress to severe disease. Treatment of choice for a majority of tick-borne illness is doxycycline; however, options are limited in the setting of tetracycline allergy. Here, we present a complex case of human granulocytic anaplasmosis–induced hemophagocytic lymphohistiocytosis complicated by coinfection with Lyme encephalitis in a patient with a documented tetracycline allergy.

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Anaplasmosis-Induced Hemophagocytic Lymphohistiocytosis: A Case Report and Review of the Literature

Cited by 3 publications

References 27 publications

Hemophagocytic Lymphohistiocytosis (HLH) in Patients with Tick-Borne Illness: A Scoping Review of 98 Cases

Hemophagocytic Lymphohistiocytosis (HLH) in Patients with Tick-Borne Illness: A Scoping Review of 98 Cases

Tick-Borne Illnesses in Emergency and Wilderness Medicine

Hemophagocytic Lymphohistiocytosis Secondary to Anaplasmosis With Lyme Coinfection

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