Secondary Hemophagocytic Lymphohistiocytosis in Post-COVID-19 Patients: A Report of Two Cases

Kalita, Pranjal; Laishram, Devina; Dey, Biswajit; Mishra, Jaya; Barman, Bhupen; Barman, Himesh

doi:10.7759/cureus.17328

Cited by 18 publications

(25 citation statements)

References 14 publications

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“…The diagnosis of sHLH has been previously described in COVID-19 (Núñez-Torrón et al 2021;Harris et al 2021;Swoboda et al 2021;Meazza Prina et al 2021;Bryce et al 2021;Prilutskiy et al 2020;Debliquis et al 2020;Prieto-Pérez et al 2020;Tholin et al 2020;Labro et al 2020;Lima et al 2020) and especially in patients after COVID-19 (Michaelis et al 2021;Naqvi and Bhutta 2021;Sangha et al 2021;Dandu 2021;Naous et al 2021;Kalita et al 2021;Kayaaslan et al 2021;Wiseman et al 2021) (Table 3). Nalbandian et al, defined post-acute COVID-19 syndrome as persistent symptoms and/or delayed or longterm complications of SARS-CoV-2 infection beyond four weeks after the onset of symptoms (Nalbandian et al 2021).…”

Section: Discussionmentioning

confidence: 82%

“…However, clinical, laboratory, and morphological findings appear controversial in patients recovering from COVID-19 (Bhattacharjee et al 2020). Table 3 shows current and previous cases of sHLH described after recovery from COVID-19, based on BM findings in living patients (Naous et al 2021;Naqvi and Bhutta 2021;Sangha et al 2021;Kayaaslan et al 2021;Kalita et al 2021;Michaelis et al 2021;Núñez-Torrón et al 2021;Wiseman et al 2021;Swoboda et al 2021;Debliquis et al 2020;Prieto-Pérez et al 2020;Dandu 2021;Harris et al 2021). Kayaaslan et al, showed that some patients may develop hemophagocytosis in the late COVID-19 period, even when recovering (Kayaaslan et al 2021).…”

Section: Discussionmentioning

confidence: 99%

“…Although COVID-19-related cytokine storm has been associated with the development of sHLH since the onset of the pandemic (England et al 2021), little is known about the clinical manifestations of sHLH that develop after a patient recovers from SARS-CoV-2 infection England et al 2021;Naous et al 2021;Naqvi and Bhutta 2021;Sangha et al 2021;Kayaaslant al. e 2021;Kalita et al 2021;Michaelis et al 2021;Núñez-Torrón et al 2021;Wiseman et al 2021). The majority of studies on histological findings of BM in patients with COVID-19 are postmortem (Núñez-Torrón et al 2021;Prilutskiy et al 2020;Swoboda et al 2021;Prieto-Pérez et al 2020;Bryce et al 2021).…”

Section: Introductionmentioning

confidence: 99%

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Hemophagocytic lymphohistiocytosis diagnosed by bone marrow trephine biopsy in living post-COVID-19 patients: case report and mini-review

et al. 2022

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Hemophagocytic lymphohistiocytosis (HLH) constitutes a life-threatening inflammatory syndrome. Postmortem histological findings of bone marrow (BM) from COVID-19 patients showed histiocytosis and hemophagocytosis and supported the hypothesis that secondary HLH (sHLH) may be triggered by SARS-CoV-2 infection. However, there are a limited number of sHLH cases in which trephine has been performed in living post-COVID-19 patients. Here we present a recent case and a mini-review of sHLH diagnosed by trephine biopsy in living patients after COVID-19. An 81-year-old man with a past medical history of hypertension, diabetes, ischemic stroke, was referred to the hospital to evaluate leukocytosis, pyuria, and elevation of inflammatory markers four weeks after recovering from COVID-19. Computed tomography of the abdomen did not reveal focal signs of infection or hepatosplenomegaly. The patient received intravenous meropenem and two packed red blood cell units. Leukocytes and C-reactive protein were gradually decreased. A BM biopsy was performed and the patient was discharged on cefixime. BM smear revealed severe anemia, lymphopenia, and dysplastic morphologic findings of erythroblasts, neutrophils, and megakaryocytes. Trephine biopsy revealed hypercellular marrow dyserythropoiesis, plasmacytosis, lymphocytosis, histiocytosis, hemophagocytosis, and the absence of granulomas or carcinoma. Immunohistochemistry documented a mixed population of T lymphocytes (CD3+) and B lymphocytes (CD20+). Strong positivity for CD68 confirmed histiocytosis. CD138 κ, λ staining proved polyclonal plasmacytosis. Perl's staining showed excess hemosiderin deposits. Based on our findings, we document sHLH in trephine BM biopsy of a living post-COVID-19 patient and persistent leukocytosis, underscoring the diagnostic value of trephine biopsy in preventing life-threatening conditions such as COVID-19.

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Section: Discussionmentioning

confidence: 82%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Hemophagocytic lymphohistiocytosis diagnosed by bone marrow trephine biopsy in living post-COVID-19 patients: case report and mini-review

et al. 2022

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“…The probability of HLH was determined by the H-score given by Fardet et al; our patient had a score of 256 points indicating a 99% probability of HLH (6). Previous case reports on post-COVID-19 HLH have relied mainly on H-score with scarce data on cytokine levels used in diagnosis for HLH (Table 2B) (8)(9)(10). The use of the H-Score for patients with COVID-19 has been questioned, and some authors recommend against using H-score.…”

Section: Discussionmentioning

confidence: 90%

“…There are case reports of SARS-CoV-2-induced sHLH diagnosed antemortem (11)(12)(13)(14)(15) and postmortem (1,(16)(17)(18)(19) while the illness is/was ongoing, as shown in Table 2B. However, the literature is sparse about COVID-19 recovered cases developing sHLH (8)(9)(10), as shown in Table 2B. A study focusing on postmortem bone marrow biopsies of patients with COVID-19 showed histiocytic hyperplasia with hemophagocytosis (1).…”

Section: Discussionmentioning

confidence: 99%

Case Report: Secondary Hemophagocytic Lymphohistiocytosis (sHLH) and Candida auris Fungemia in Post-acute COVID-19 Syndrome: A Clinical Challenge

et al. 2022

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Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection causes a disease (COVID-19) with multisystem involvement. The world is now entering a phase of post-COVID-19 manifestations in this pandemic. Secondary hemophagocytic lymphohistiocytosis (sHLH) is a life-threatening hyperinflammatory event triggered by viral infections, including SARS-CoV-2. Both Multisystem Inflammatory Syndrome-Adults (MIS-A) and Cytokine Storm Syndrome (CSS) are considered close differentials of sHLH and add to the spectrum of Post-acute COVID-19 syndrome (PACS). In this report, we presented the case of a middle-aged Asian man who was initially discharged upon recovery from severe COVID-19 infection after 17 days of hospitalization to a private institute and later came to our hospital 13 days post-discharge. Here, he was diagnosed with sHLH, occurring as an extension of CSS, with delayed presentation falling within the spectrum of PACS. The diagnosis of sHLH was made holistically with the HLH-2004 criteria. Our patient initially responded to intravenous immunoglobulin (IVIG) and dexamethasone, later complicated by disseminated Candida auris infection and had a fatal outcome. Though many cases of HLH during active COVID-19 and a few cases post COVID-19 recovery have been reported, based on H-score, which has limitations as a diagnostic tool. We report the first case report of post-COVID-19 sHLH using the HLH-2004 criteria, complicated by disseminated Candidemia, emphasizing that the care of patients with COVID-19 does not conclude at the time of hospital discharge. We highlight the importance of surveillance in the post-COVID phase for early detection of sHLH which may predispose to fatal opportunistic infections (OIs).

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Haematological sequelae in the post-acute phase of symptomatic SARS-CoV-2 infection

Bergamaschi,

Barteselli,

Calabretta

et al. 2023

Intern Emerg Med

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Secondary Hemophagocytic Lymphohistiocytosis in Post-COVID-19 Patients: A Report of Two Cases

Cited by 18 publications

References 14 publications

Hemophagocytic lymphohistiocytosis diagnosed by bone marrow trephine biopsy in living post-COVID-19 patients: case report and mini-review

Hemophagocytic lymphohistiocytosis diagnosed by bone marrow trephine biopsy in living post-COVID-19 patients: case report and mini-review

Case Report: Secondary Hemophagocytic Lymphohistiocytosis (sHLH) and Candida auris Fungemia in Post-acute COVID-19 Syndrome: A Clinical Challenge

Haematological sequelae in the post-acute phase of symptomatic SARS-CoV-2 infection

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