Hemophagocytic lymphohistiocytosis in a rheumatoid arthritis patient treated with infliximab.

Hemophagocytic lymphohistiocytosis (HLH) is a rare condition with high mortality. We report a case of a 74-year-old woman with rheumatoid arthritis who developed HLH secondary to pyelonephritis due to Escherichia coli infection following infliximab treatment. Bone marrow aspiration showed proliferation of histiocytes with hemophagocytosis. The patient died despite treatment with intravenous antibiotics intravenous methylprednisolone and intravenous immunoglobulin. Cytokine levels were measured and are discussed.

[1]  H. Ören,et al.  Etanercept for therapy‐resistant macrophage activation syndrome , 2008, Pediatric blood & cancer.

[2]  D. Valla,et al.  Thrombopénie sévère d’aggravation rapide comme seul signe d’une hémophagocytose chez un malade atteint de cirrhose avec infection léthale du liquide d’ascite par Escherichia coli , 2007 .

[3]  S. Eksborg,et al.  Severe bacteria‐associated hemophagocytic lymphohistiocytosis in an extremely premature infant , 2007, Acta paediatrica.

[4]  Y. Kirino,et al.  Successful treatment of refractory lupus-associated haemophagocytic lymphohistiocytosis with infliximab. , 2007, Rheumatology.

[5]  K. Kawa,et al.  Nationwide Survey of Hemophagocytic Lymphohistiocytosis in Japan , 2007, International journal of hematology.

[6]  M. Aricò,et al.  HLH‐2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis , 2007, Pediatric blood & cancer.

[7]  D. Valla,et al.  [Severe thrombopenia as single sign of hemophagocytosis in a patient with cirrhosis and lethal infection of ascitis fluid by Escherichia coli]. , 2007, Gastroenterologie clinique et biologique.

[8]  E. Ok,et al.  High-dose immunoglobulin therapy in renal transplant recipients with hemophagocytic histiocytic syndrome. , 2006, Journal of nephrology.

[9]  T. Miyamoto,et al.  Success with infliximab in treating refractory hemophagocytic lymphohistiocytosis , 2006, American journal of hematology.

[10]  W. Grossman,et al.  Hemophagocytic lymphohistiocytosis: Diagnosis, pathophysiology, treatment, and future perspectives , 2006, Annals of medicine.

[11]  Kuo-Hsuan Chang,et al.  Spontaneous Escherichia coli meningitis associated with hemophagocytic lymphohistiocytosis. , 2006, Journal of the Formosan Medical Association = Taiwan yi zhi.

[12]  L. Espinoza,et al.  Macrophage activation syndrome and etanercept in children with systemic juvenile rheumatoid arthritis. , 2004, The Journal of rheumatology.

[13]  K. Lassoued,et al.  Syndrome d’activation macrophagique associé à une septicémie à Escherichia coli : à propos d’un cas , 2003 .

[14]  A. Aouba,et al.  Haemophagocytic syndrome in a rheumatoid arthritis patient treated with infliximab. , 2003, Rheumatology.

[15]  R. Schneider,et al.  Macrophage activation syndrome following initiation of etanercept in a child with systemic onset juvenile rheumatoid arthritis. , 2003, The Journal of rheumatology.

[16]  K. Lassoued,et al.  [Hemophagocytosis associated with an Escherichia coli sepsis: a case report]. , 2003, La Revue de medecine interne.

[17]  D. Dickens,et al.  Etanercept in the treatment of macrophage activation syndrome. , 2001, The Journal of rheumatology.

[18]  R. Riley,et al.  Worsening of macrophage activation syndrome in a patient with adult onset Still's disease after initiation of etanercept therapy. , 2001, Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases.

[19]  Shotai Kobayashi,et al.  Autoimmune-associated hemophagocytic syndrome , 2004, Modern rheumatology.

[20]  Y. Murakawa,et al.  Reactive hemophagocytosis in systemic lupus erythematosus. , 1998, Internal medicine.

[21]  T. Kuribayashi,et al.  [Autopsy case of infection-associated hemophagocytic syndrome caused by E. coli-induced acute pyelonephritis]. , 1997, Nihon Naika Gakkai zasshi. The Journal of the Japanese Society of Internal Medicine.

[22]  K. Miyamoto A pathologic autopsy causing bacteria related hemophagocytosis syndrome from acute pyelonephritis by E. coli. , 1997 .

[23]  B. François,et al.  Syndrome hémophagocytaire réactionnel : une cause probablement sous-estimée de thrombopénie en réanimation , 1995 .

[24]  P. Vignon,et al.  [Reactive hemophagocytic syndrome: a probably underestimated cause of thrombocytopenia in intensive acre patients]. , 1995, Annales francaises d'anesthesie et de reanimation.

[25]  C. Dinarello,et al.  The role of interleukin-1 in disease. , 1993, The New England journal of medicine.

[26]  Y. Tsunematsu,et al.  Prognosis of children with virus-associated hemophagocytic syndrome and malignant histiocytosis: correlation with levels of serum interleukin-1 and tumor necrosis factor. , 1991, Acta haematologica.

[27]  Y. Mizuno,et al.  Histiocytic Hemophagocytosis in the Bone Marrow in Children with Sepsis and Disseminated Intravascular Coagulation , 1989, Acta paediatrica Japonica : Overseas edition.

[28]  R. Brunning,et al.  Bacteria‐associated hemophagocytic syndrome , 1984, Cancer.