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Haemophagocytic lymphohistiocytosis after heart transplantation: a case report.

Artikel i vetenskaplig tidskrift
Författare Christian Danielsson
Kristjan Karason
Göran Dellgren
Publicerad i European heart journal. Case reports
Volym 4
Nummer/häfte 3
Sidor 1-4
ISSN 2514-2119
Publiceringsår 2020
Publicerad vid Institutionen för medicin, avdelningen för molekylär och klinisk medicin
Sidor 1-4
Språk en
Länkar dx.doi.org/10.1093/ehjcr/ytaa070
www.ncbi.nlm.nih.gov/entrez/query.f...
Ämnesord Case report, Haemophagocytic Lymphohistiocytosis, Haemophagocytic syndrome, Heart transplantation
Ämneskategorier Kardiovaskulär medicin

Sammanfattning

Haemophagocytic lymphohistiocytosis (HLH) is an uncommon but serious systemic inflammatory response with high mortality rates. It can be triggered by malignancy or infectious agents, often in the context of immunosuppression. Literature covering HLH in heart transplantation (HTx) is scarce.A 25-year-old male with a history of celiac disease underwent HTx at Sahlgrenska Hospital in 2011 due to giant cell myocarditis and was treated with tacrolimus, mycophenolate mofetil (MMF), and prednisolone. He developed several episodes of acute cellular rejections (ACR) during the first 3 post-HTx years, which subsided after addition of everolimus. In May 2017, the patient was admitted to the hospital due to fever without focal symptoms. He had an extensive inflammatory reaction, but screening for infectious agents was negative. Haemophagocytic lymphohistiocytosis was discussed early, but first dismissed since two bone marrow biopsies revealed no signs of haemophagocytosis. Increasing levels of soluble IL-2 were considered confirmative of the diagnosis. Even with intense immunosuppressant treatment, the patient deteriorated and died in progressive multiorgan failure within 2 weeks of the symptom onset.A 25-year-old HTx recipient with an extensive inflammatory response, fulfilled criteria for HLH, but the diagnosis was delayed due to normal bone marrow biopsies. A background with autoimmune reactivity and immunosuppressive therapy may have contributed to HLH, but the actual trigger was not identified. Haemophagocytic lymphohistiocytosis can occur in HTx recipients in the absence of malignancy, identifiable infectious triggers and signs of haemophagocytosis. Early diagnosis and intervention are likely to be of importance for a favourable outcome.

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