Suspected thrombotic microangiopathy in a child with Epstein–Barr virus‐induced hemophagocytic lymphohistiocytosis

Cupit-Link, Margaret C; Kohorst, Mira A.; Tran, Cheryl L.; Bleesing, Jack J.; Galardy, Paul J.; Boyce, Thomas G.

doi:10.1002/pbc.27002

Cited by 2 publications

(3 citation statements)

References 8 publications

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“…In our systematic review of the literature, some patients were diagnosed with HUS and HLH simultaneously, 10,12,13,19 while others were diagnosed with HUS a couple of weeks after being diagnosed with HLH. 20 In the study of Akyüz et al, 3 a diagnosis of HLH was made on the 28th day after the diagnosis of aHUS.…”

Section: Discussionmentioning

confidence: 99%

“…TMA is a histopathologic term used to describe a variety of thrombotic vascular lesions that represents a group of diseases that include HUS, and thrombotic thrombocytopenic purpura (TTP). 9 In TTP, Santoriello et al 10 Inagaki et al 11 Ardalan et al 12 Esmaili et al 13 Singh et al 14 Bae et al 15 Wu et al 17 Fraga-Rodriguez et al 18 Akyüz et al 3 Cupit-Link et al 19 Chiang et al 20 Thaunat et al 16 Davies microvascular aggregation commonly occurs in the brain; however, in HUS platelet-fibrin thrombus mostly affects the kidney. Furthermore, the normal level of ADAMTS 13 in HUS is useful in differentiating HUS from TTP.…”

Section: Discussionmentioning

confidence: 99%

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Hemophagocytic Lymphohistiocytosis Associated With Hemolytic Uremic Syndrome in a Child: A Case Report and Systematic Literature Review

Taş

Gökçeoğlu

Yayla

et al. 2021

Journal of Pediatric Hematology/Oncology

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Hemophagocytic lymphohistiocytosis (HLH) is a lifethreatening disorder resulting from excessive activation and nonmalignant proliferation of T-lymphocytes and macrophages. Hemolytic uremic syndrome (HUS) is a disease characterized by microangiopathic hemolytic anemia, thrombocytopenia, and acute renal failure. Reports pertaining to the association between HLH and HUS are rarely published; however, we report on a 4-year-old boy who was diagnosed with both conditions and treated successfully with high-dose steroid and intravenous immunoglobulin. Differentiating HUS from HLH can be challenging because of their clinical similarities. Therefore, prompt diagnosis and immunosuppressive treatment are essential and life-saving to these patients.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Hemophagocytic Lymphohistiocytosis Associated With Hemolytic Uremic Syndrome in a Child: A Case Report and Systematic Literature Review

Taş

Gökçeoğlu

Yayla

et al. 2021

Journal of Pediatric Hematology/Oncology

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“…About half of the literature described cases of post‐ASCT HPS or TA‐TMA were related to an underlying infection, and in the remainder, no potential trigger could be identified (Ostronoff et al, ; Takagi et al, ; Laskin et al, ). Only a few cases of concurrent occurrence of secondary HPS and TMA in the non‐transplant setting have been described, also following viral, fungal or unknown infections (Chiang et al, ; Inagaki et al, ; Cupit‐Link et al, ).…”

Section: Course Of Clinical Events and Laboratory Parameters Post‐tramentioning

confidence: 99%

Engraftment syndrome, transplant‐associated thrombotic microangiopathy and hemophagocytic syndrome following autologous stem cell transplant in multiple myeloma

Singh

Kayal

Dubashi

et al. 2019

Transfusion Medicine

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Dear Sir,The most common toxicities for autologous stem cell transplantation (ASCT) in multiple myeloma (MM) are gastrointestinal and mucositis. Mortality is lower than 5% in well-selected patients. Engraftment syndrome (ES) is a common complication of ASCT, whereas transplant-associated thrombotic microangiopathy (TA-TMA) and hemophagocytic syndrome (HPS) are very rare, mostly occurring after allogeneic transplants, and often have a fulminant course. We describe a case of MM where the post-ASCT course was marked by ES, life-threatening TA-TMA and secondary HPS occurring concomitantly.A 55-year-old lady with MM stage IIIA received six cycles of induction chemotherapy with cyclophosphamide, bortezomib and dexamethasone (Reeder et al., 2010) and had very good partial response. In March 2017, she underwent ASCT by peripheral blood stem cell harvest performed using granulocyte-colony stimulating factor (G-CSF) mobilisation. CD34 was 2.01 × 10 6 kg −1 . Melphalan was given at 200 mg m −2 on D−1.G-CSF 5 μg kg −1 was started from D+1. Antibiotics were added on D+2 for febrile neutropenia and changed to meropenem, linezolid and liposomal amphotericin B for recurrent fever on D+8. Work-up for infection was negative. She developed grade 2 mucositis and diarrhoea. Neutrophil engrafted on D+10, and the platelet count was 20 × 10 9 L −1 with support. On D+11, fever recurred, with worsening of diarrhoea, weight gain (4% of baseline), mild dyspnoea, no skin rashes, new chest infiltrates and elevated creatinine and bilirubin over the next 48 h. Repeat work-up for infection was negative. ES was diagnosed, and G-CSF was discontinued. Diuretics and steroids (prednisone-1 mg kg −1 day −1 for 5 days) were initiated, colistin was added, and supportive treatment continued, to which she showed initial response. From D+17 to D+21, she deteriorated with recurring high fever, worsening diarrhoea, dyspnoea, tachypnoea, tachycardia, mild hypertension initially, mild gum/oral bleeding, persistent thrombocytopenia even with support, declining leucocyte count, anaemia, rising bilirubin and an episode of haemoptysis, haematuria and hypotension on D+21. Inotropes were initiated with the continuation of antibiotics and antifungals (liposomal amphotericin B), and steroids were restarted. Non-invasive ventilator (NIV) support was required

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Suspected thrombotic microangiopathy in a child with Epstein–Barr virus‐induced hemophagocytic lymphohistiocytosis

Cited by 2 publications

References 8 publications

Hemophagocytic Lymphohistiocytosis Associated With Hemolytic Uremic Syndrome in a Child: A Case Report and Systematic Literature Review

Hemophagocytic Lymphohistiocytosis Associated With Hemolytic Uremic Syndrome in a Child: A Case Report and Systematic Literature Review

Engraftment syndrome, transplant‐associated thrombotic microangiopathy and hemophagocytic syndrome following autologous stem cell transplant in multiple myeloma

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