Approach to hemophagocytic syndromes.

Hemophagocytic lymphohistiocytosis (HLH) is a potentially fatal hyperinflammatory condition. It may occur as a primary (genetic) condition due to mutations in genes important in the cytolytic secretory pathway that cause perforin and granzymes to induce apoptosis in target cells. Primary HLH is divided into familial HLH (FHLH1-5), in which HLH is the only manifestation of disease, and other genetic causes in which HLH is one of several clinical manifestations. The identical clinical findings may arise secondary to infectious, rheumatologic, malignant, or metabolic conditions. Whether primary or secondary, HLH therapy needs to be instituted promptly to prevent irreversible tissue damage. It is helpful to think of HLH as the severe end of the spectrum of hyperinflammatory disorders when the immune system starts to damage host tissues (immunopathology). Therefore, no single clinical feature alone is diagnostic for HLH, and it is important that the entire clinical presentation be considered in making the diagnosis. This article contains a discussion of the genetic background, clinical presentation, diagnostic dilemmas, and features that are helpful in making the diagnosis of HLH, along with a discussion of common problems in its management.

[1]  P. Newburger,et al.  Hemophagocytic lymphohistiocytosis with MUNC13‐4 gene mutation or reduced natural killer cell function prior to onset of childhood leukemia , 2011, Pediatric blood & cancer.

[2]  K. McClain,et al.  Hemophagocytic lymphohistiocytosis associated with precursor B acute lymphoblastic leukemia , 2011, Pediatric blood & cancer.

[3]  S. Imashuku Treatment of Epstein-Barr Virus-related Hemophagocytic Lymphohistiocytosis (EBV-HLH); Update 2010 , 2011, Journal of pediatric hematology/oncology.

[4]  S. Davies,et al.  Reduced-intensity conditioning significantly improves survival of patients with hemophagocytic lymphohistiocytosis undergoing allogeneic hematopoietic cell transplantation. , 2010, Blood.

[5]  S. Gupta,et al.  Primary and secondary hemophagocytic lymphohistiocytosis: clinical features, pathogenesis and therapy , 2010, Expert review of clinical immunology.

[6]  A. Filipovich,et al.  Histiocytic disorders: recent insights into pathophysiology and practical guidelines. , 2010, Biology of blood and marrow transplantation : journal of the American Society for Blood and Marrow Transplantation.

[7]  G. Janka,et al.  Hemophagocytic Lymphohistiocytosis: When the Immune System Runs Amok , 2009, Klinische Padiatrie.

[8]  A. Filipovich Hemophagocytic lymphohistiocytosis (HLH) and related disorders. , 2009, Hematology. American Society of Hematology. Education Program.

[9]  V. Breakey,et al.  Hemophagocytic lymphohistiocytosis onset during induction therapy for precursor B-cell acute lymphoblastic leukemia. , 2008, Journal of pediatric hematology/oncology.

[10]  L. Castillo High elevated ferritin levels and the diagnosis of HLH/Sepsis/SIRS/MODS/MAS , 2008, Pediatric blood & cancer.

[11]  K. Nichols,et al.  Successful use of the anti‐CD25 antibody daclizumab in an adult patient with hemophagocytic lymphohistiocytosis , 2008, American journal of hematology.

[12]  C. Kozinetz,et al.  Highly elevated ferritin levels and the diagnosis of hemophagocytic lymphohistiocytosis , 2008, Pediatric blood & cancer.

[13]  W. Grossman,et al.  Unrelated donor hematopoietic cell transplantation for hemophagocytic lymphohistiocytosis , 2008, Bone Marrow Transplantation.

[14]  H. Gadner,et al.  Frequency and spectrum of central nervous system involvement in 193 children with haemophagocytic lymphohistiocytosis , 2008, British journal of haematology.

[15]  William M. Lee,et al.  Soluble CD163 from activated macrophages predicts mortality in acute liver failure. , 2007, Journal of hepatology.

[16]  Eric O Long,et al.  Defective cytotoxic lymphocyte degranulation in syntaxin-11 deficient familial hemophagocytic lymphohistiocytosis 4 (FHL4) patients. , 2007, Blood.

[17]  A. Fischer,et al.  Immunotherapy of Familial Hemophagocytic Lymphohistiocytosis With Antithymocyte Globulins: A Single-Center Retrospective Report of 38 Patients , 2007, Pediatrics.

[18]  D. Teachey,et al.  Use of rituximab in conjunction with immunosuppressive chemotherapy as a novel therapy for Epstein Barr virus-associated hemophagocytic lymphohistiocytosis. , 2007, Journal of pediatric hematology/oncology.

[19]  T. Wada,et al.  Immunophenotypic analysis of Epstein–Barr virus (EBV)‐infected CD8+ T cells in a patient with EBV‐associated hemophagocytic lymphohistiocytosis , 2007, European journal of haematology.

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

[21]  A. Ramanan,et al.  The diagnostic significance of soluble CD163 and soluble interleukin-2 receptor alpha-chain in macrophage activation syndrome and untreated new-onset systemic juvenile idiopathic arthritis. , 2007, Arthritis and rheumatism.

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

[23]  S. Imashuku SYSTEMIC TYPE EPSTEIN-BARR VIRUS-RELATED LYMPHOPROLIFERATIVE DISEASES IN CHILDREN AND YOUNG ADULTS: Challenges for Pediatric Hemato-Oncologists and Infectious Disease Specialists , 2007, Pediatric hematology and oncology.

[24]  L. Moretta,et al.  Analysis of natural killer-cell function in familial hemophagocytic lymphohistiocytosis (FHL): defective CD107a surface expression heralds Munc13-4 defect and discriminates between genetic subtypes of the disease. , 2006, Blood.

[25]  A. Fischer,et al.  Hematopoietic Stem Cell Transplantation in Hemophagocytic Lymphohistiocytosis: A Single-Center Report of 48 Patients , 2006, Pediatrics.

[26]  Y. Lau,et al.  Cytotoxic therapy for severe avian influenza A (H5N1) infection , 2006, The Lancet.

[27]  J. Henter,et al.  Histiocytic Disorders of Children and Adults: Clinical aspects and therapy of hemophagocytic lymphohistiocytosis , 2005 .

[28]  M. Kurrer,et al.  Soluble hemoglobin–haptoglobin scavenger receptor CD163 as a lineage‐specific marker in the reactive hemophagocytic syndrome , 2005, European journal of haematology.

[29]  K. Sullivan,et al.  Healing hemophagocytosis. Discussion , 2005 .

[30]  K. Sullivan,et al.  Healing hemophagocytosis. , 2005, Clinical immunology.

[31]  G. Janka,et al.  Familial hemophagocytic lymphohistiocytosis , 1983, European Journal of Pediatrics.

[32]  R. Egeler,et al.  Treatment of hemophagocytic lymphohistiocytosis with HLH-94 immunochemotherapy and bone marrow transplantation. , 2002, Blood.

[33]  L. Sung,et al.  The role of infections in primary hemophagocytic lymphohistiocytosis: a case series and review of the literature. , 2001, Clinical infectious diseases : an official publication of the Infectious Diseases Society of America.

[34]  M. Kato,et al.  Requirement for etoposide in the treatment of Epstein-Barr virus-associated hemophagocytic lymphohistiocytosis. , 2001, Journal of clinical oncology : official journal of the American Society of Clinical Oncology.

[35]  A. Fischer,et al.  Perforin gene defects in familial hemophagocytic lymphohistiocytosis. , 1999, Science.

[36]  K. Sullivan,et al.  Defective Natural Killer Cell Function in Patients with Hemophagocytic Lymphohistiocytosis and in First Degree Relatives , 1998, Pediatric Research.

[37]  隆夫 吉原,et al.  化学療法と同種骨髄移植により3年以上初回寛解を維持しているfamilial erythrophagocytic lymphohistiocytosis (FEL) の1例 , 1991 .

[38]  A. Öst,et al.  Incidence in Sweden and Clinical Features of Familial Hemophagocytic Lymphohistiocytosis , 1991, Acta paediatrica Scandinavica.

[39]  H. Macmahon,et al.  FAMILIAL ERYTHROPHAGOCYTIC LYMPHOHISTIOCYTOSIS. , 1963, Pediatrics.