Diagnosis and screening of patients with hereditary angioedema in primary care

Hereditary angioedema (HAE) is a rare autosomal dominant disease that commonly manifests with episodes of cutaneous or submucosal angioedema and intense abdominal pain. The condition usually presents due to a deficiency of C1 esterase inhibitor (C1-INH) that leads to the overproduction of bradykinin, causing an abrupt increase in vascular permeability. A less-understood and less-common form of the disease presents with normal C1-INH levels. Symptoms of angioedema may be confused initially with mast cell-mediated angioedema, such as allergic reactions, and may perplex physicians when epinephrine, antihistamine, or glucocorticoid therapies do not provide relief. Similarly, abdominal attacks may lead to unnecessary surgeries or opiate dependence. All affected individuals are at risk for a life-threatening episode of laryngeal angioedema, which continues to be a source of fatalities due to asphyxiation. Unfortunately, the diagnosis is delayed on average by almost a decade due to a misunderstanding of symptoms and general lack of awareness of the disease. Once physicians suspect HAE, however, diagnostic methods are reliable and available at most laboratories, and include testing for C4, C1-INH protein, and C1-INH functional levels. In patients with HAE, management consists of acute treatment of an attack as well as possible short- or long-term prophylaxis. Plasma-derived C1-INH, ecallantide, icatibant, and recombinant human C1-INH are new treatments that have been shown to be safe and effective in the treatment of HAE attacks. The current understanding of HAE has greatly improved in recent decades, leading to growing awareness, new treatments, improved management strategies, and better outcomes for patients.

[1]  T. Craig,et al.  Plasma‐derived C1‐INH for managing hereditary angioedema in pediatric patients: A systematic review , 2015, Pediatric allergy and immunology : official publication of the European Society of Pediatric Allergy and Immunology.

[2]  L. Bielory,et al.  Hereditary Angioedema and Gastrointestinal Complications: An Extensive Review of the Literature , 2015, Case reports in immunology.

[3]  K. Heimdal,et al.  A Nationwide Study of Norwegian Patients with Hereditary Angioedema with C1 Inhibitor Deficiency Identified Six Novel Mutations in SERPING1 , 2015, PloS one.

[4]  M. Maurer,et al.  Development and content validity testing of a patient-reported outcomes questionnaire for the assessment of hereditary angioedema in observational studies , 2015, Health and Quality of Life Outcomes.

[5]  M. Riedl Recombinant Human C1 Esterase Inhibitor in the Management of Hereditary Angioedema , 2015, Clinical Drug Investigation.

[6]  M. Margaglione,et al.  Characterization of patients with angioedema without wheals: the importance of F12 gene screening. , 2015, Clinical immunology.

[7]  R. Tachdjian,et al.  Current characteristics associated with hereditary angioedema attacks and treatment: the home infusion based patient experience. , 2015, Allergy and asthma proceedings.

[8]  T. Craig,et al.  Recombinant human C1 esterase inhibitor for the treatment of hereditary angioedema due to C1 inhibitor deficiency (C1-INH-HAE) , 2015, Expert review of clinical immunology.

[9]  H. Farkas,et al.  A novel assay to diagnose hereditary angioedema utilizing inhibition of bradykinin‐forming enzymes , 2015, Allergy.

[10]  N. Pescosolido Therapeutics and clinical risk management , 2015 .

[11]  G. V. Marcotte,et al.  Hereditary angioedema: imaging manifestations and clinical management , 2015, Emergency Radiology.

[12]  S. Cichon,et al.  Hereditary angioedema with F12 mutation: factors modifying the clinical phenotype , 2014, Allergy.

[13]  J. Bernstein,et al.  Diagnosis and Treatment of Bradykinin-Mediated Angioedema: Outcomes from an Angioedema Expert Consensus Meeting , 2014, International Archives of Allergy and Immunology.

[14]  T. Craig,et al.  Treatment of hereditary angioedema: a review (CME) , 2014, Transfusion.

[15]  K. Bork An evidence based therapeutic approach to hereditary and acquired angioedema , 2014, Current opinion in allergy and clinical immunology.

[16]  A. Bygum Hereditary angioedema - consequences of a new treatment paradigm in Denmark. , 2014, Acta dermato-venereologica.

[17]  M. Triggiani,et al.  Classification, diagnosis, and approach to treatment for angioedema: consensus report from the Hereditary Angioedema International Working Group , 2014, Allergy.

[18]  Erika F. H. Saunders,et al.  Depression and anxiety in patients with hereditary angioedema. , 2014, Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology.

[19]  C. Camargo,et al.  A consensus parameter for the evaluation and management of angioedema in the emergency department. , 2014, Academic emergency medicine : official journal of the Society for Academic Emergency Medicine.

[20]  R. Perricone,et al.  Psychological Correlates in Subjects with Hereditary Angioedema (HAE) , 2014 .

[21]  J. Scolapio,et al.  Gastrointestinal Manifestations, Diagnosis, and Management of Hereditary Angioedema , 2013, Journal of clinical gastroenterology.

[22]  Á. Szilágyi,et al.  Novel duplication in the F12 gene in a patient with recurrent angioedema. , 2013, Clinical immunology.

[23]  J. Bernstein,et al.  US Hereditary Angioedema Association Medical Advisory Board 2013 recommendations for the management of hereditary angioedema due to C1 inhibitor deficiency. , 2013, The journal of allergy and clinical immunology. In practice.

[24]  M. Maurer,et al.  Hereditary angioedema with C1 inhibitor deficiency: delay in diagnosis in Europe , 2013, Allergy, Asthma & Clinical Immunology.

[25]  Y. Tomino,et al.  Leukocytosis and high hematocrit levels during abdominal attacks of hereditary angioedema , 2013, BMC Gastroenterology.

[26]  R. Tachdjian,et al.  Use of Ecallantide in Pediatric Hereditary Angioedema , 2013, Pediatrics.

[27]  Hong-Yu Zhang,et al.  Clinical features of hereditary angioedema in Chinese patients: new findings and differences from other populations. , 2013, European journal of dermatology : EJD.

[28]  W. Lumry Overview of epidemiology, pathophysiology, and disease progression in hereditary angioedema. , 2013, The American journal of managed care.

[29]  J. Sheikh,et al.  A focused parameter update: hereditary angioedema, acquired C1 inhibitor deficiency, and angiotensin-converting enzyme inhibitor-associated angioedema. , 2013, The Journal of allergy and clinical immunology.

[30]  J. Dempster Management of hereditary angioedema. , 2013, Nursing standard (Royal College of Nursing (Great Britain) : 1987).

[31]  L. Buyantseva,et al.  Update on treatment of hereditary angioedema , 2013, Clinical and experimental allergy : journal of the British Society for Allergy and Clinical Immunology.

[32]  R. Lockey,et al.  WAO Guideline for the Management of Hereditary Angioedema , 2012, The World Allergy Organization journal.

[33]  H. Farkas,et al.  Short‐term prophylaxis in hereditary angioedema due to deficiency of the C1‐inhibitor – a long‐term survey , 2012, Allergy.

[34]  M. Cicardi,et al.  Hereditary angioedema with normal C1 inhibitor function: consensus of an international expert panel. , 2012, Allergy and asthma proceedings.

[35]  K. Bork,et al.  Fatal laryngeal attacks and mortality in hereditary angioedema due to C1-INH deficiency. , 2012, The Journal of allergy and clinical immunology.

[36]  T. Craig,et al.  Update on treatment of hereditary angioedema. , 2012, Asian Pacific journal of allergy and immunology.

[37]  D. Meyer‐Olson,et al.  Hereditary angioedema (HAE) in children and adolescents—a consensus on therapeutic strategies , 2012, European Journal of Pediatrics.

[38]  M. Riedl Hereditary angioedema therapies in the United States: movement toward an international treatment consensus. , 2012, Clinical therapeutics.

[39]  M. Cicardi,et al.  Hereditary angio-oedema , 2012, The Lancet.

[40]  Tom Bowen,et al.  International consensus and practical guidelines on the gynecologic and obstetric management of female patients with hereditary angioedema caused by C1 inhibitor deficiency. , 2012, The Journal of allergy and clinical immunology.

[41]  H. Longhurst,et al.  Many faces of angioedema: focus on the diagnosis and management of abdominal manifestations of hereditary angioedema , 2012, European Journal of Gastroenterology and Hepathology.

[42]  M. Triggiani,et al.  Evidence‐based recommendations for the therapeutic management of angioedema owing to hereditary C1 inhibitor deficiency: consensus report of an International Working Group , 2012, Allergy.

[43]  B. Chipps Nanofiltered C1 Inhibitor Concentrate for Treatment of Hereditary Angioedema , 2011, Pediatrics.

[44]  K. Bork,et al.  Risk of laryngeal edema and facial swellings after tooth extraction in patients with hereditary angioedema with and without prophylaxis with C1 inhibitor concentrate: a retrospective study. , 2011, Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics.

[45]  D. Johnston Diagnosis and Management of Hereditary Angioedema , 2011, The Journal of the American Osteopathic Association.

[46]  M. López-Trascasa,et al.  Consensus statement on the diagnosis, management, and treatment of angioedema mediated by bradykinin. Part I. Classification, epidemiology, pathophysiology, genetics, clinical symptoms, and diagnosis. , 2011, Journal of investigational allergology & clinical immunology.

[47]  D. Khan Hereditary angioedema: Historical aspects, classification, pathophysiology, clinical presentation, and laboratory diagnosis. , 2011, Allergy and asthma proceedings.

[48]  B. Zuraw The Pathophysiology of Hereditary Angioedema , 2010, The World Allergy Organization journal.

[49]  W. Lumry,et al.  The humanistic burden of hereditary angioedema: Impact on health-related quality of life, productivity, and depression. , 2010, Allergy and asthma proceedings.

[50]  William H. Yang,et al.  Icatibant, a new bradykinin-receptor antagonist, in hereditary angioedema. , 2010, The New England journal of medicine.

[51]  H. Farkas Pediatric hereditary angioedema due to C1-inhibitor deficiency , 2010, Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology.

[52]  P. Keith,et al.  2010 International consensus algorithm for the diagnosis, therapy and management of hereditary angioedema , 2010, Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology.

[53]  M. Gompels,et al.  HAE international home therapy consensus document , 2010, Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology.

[54]  B. Ritchie,et al.  Management of hereditary angioedema: 2010 Canadian approach , 2010, Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology.

[55]  L. Bouillet Hereditary angioedema in women , 2010, Allergy, Asthma, and Clinical Immunology : Official Journal of the Canadian Society of Allergy and Clinical Immunology.

[56]  T. Craig,et al.  Is there a need for clinical guidelines in the United States for the diagnosis of hereditary angioedema and the screening of family members of affected patients? , 2010, Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology.

[57]  M. Frank,et al.  Genius at work: Osler's 1888 article on hereditary angioedema. , 2010, The American journal of the medical sciences.

[58]  W. Osler Landmark Publication from The American Journal of the Medical Sciences: HEREDITARY ANGIO-NEUROTIC OEDEMA , 2010, The American journal of the medical sciences.

[59]  J. McGrath,et al.  Recurrent European missense mutation in the F12 gene in a British family with type III hereditary angioedema. , 2009, Journal of dermatological science.

[60]  J. Bernstein,et al.  Efficacy of human C1 esterase inhibitor concentrate compared with placebo in acute hereditary angioedema attacks. , 2009, The Journal of allergy and clinical immunology.

[61]  T. Craig,et al.  Frequency, timing, and type of prodromal symptoms associated with hereditary angioedema attacks. , 2009, Allergy and asthma proceedings.

[62]  H. Joller-jemelka,et al.  Functional C1-inhibitor diagnostics in hereditary angioedema: assay evaluation and recommendations. , 2008, Journal of immunological methods.

[63]  B. Zuraw Clinical practice. Hereditary angioedema. , 2008, The New England journal of medicine.

[64]  M. Frank 8. Hereditary angioedema. , 2008, The Journal of allergy and clinical immunology.

[65]  William H. Yang,et al.  Hereditary angiodema: a current state-of-the-art review, VII: Canadian Hungarian 2007 International Consensus Algorithm for the Diagnosis, Therapy, and Management of Hereditary Angioedema. , 2008, Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology.

[66]  H. Farkas,et al.  Management of Hereditary Angioedema in Pediatric Patients , 2007, Pediatrics.

[67]  M. Mohammadi,et al.  An evaluation of tests used for the diagnosis and monitoring of C1 inhibitor deficiency: normal serum C4 does not exclude hereditary angio‐oedema , 2007, Clinical and experimental immunology.

[68]  W. Sewell,et al.  Secondary systemic lupus erythematosus: an analysis of 4 cases of uncontrolled hereditary angioedema. , 2007, Clinical immunology.

[69]  A. Davis Mechanism of angioedema in first complement component inhibitor deficiency. , 2006, Immunology and allergy clinics of North America.

[70]  K. Bork,et al.  Missense mutations in the coagulation factor XII (Hageman factor) gene in hereditary angioedema with normal C1 inhibitor. , 2006, Biochemical and biophysical research communications.

[71]  K. Bork,et al.  Hereditary angioedema: new findings concerning symptoms, affected organs, and course. , 2006, The American journal of medicine.

[72]  A. Eckardt,et al.  Symptoms, Course, and Complications of Abdominal Attacks in Hereditary Angioedema Due to C1 Inhibitor Deficiency , 2006, The American Journal of Gastroenterology.

[73]  A. Eckardt,et al.  Hereditary angioedema involving the colon: endoscopic appearance and review of GI manifestations. , 2005, Gastrointestinal endoscopy.

[74]  M. López-Trascasa,et al.  Hereditary angioedema due to C1 inhibitor deficiency: patient registry and approach to the prevalence in Spain. , 2005, Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology.

[75]  M. Gompels,et al.  C1 inhibitor deficiency: consensus document , 2005, Clinical and experimental immunology.

[76]  K. Kragballe,et al.  Hereditary Angioedema. , 2018, The New England journal of medicine.

[77]  A. Tordai,et al.  Hereditary and acquired angioedema: Problems and progress: Proceedings of the third C1 esterase inhibitor deficiency workshop and beyond , 2004, Journal of Allergy and Clinical Immunology.

[78]  H. Griffiths,et al.  Normal complement C4 values do not exclude hereditary angioedema , 2004, Journal of Clinical Pathology.

[79]  M. Gompels,et al.  Misdiagnosis of hereditary angio‐oedema type 1 and type 2 , 2003, The British journal of dermatology.

[80]  M. Gompels,et al.  A multicentre evaluation of the diagnostic efficiency of serological investigations for C1 inhibitor deficiency. , 2002, Journal of clinical pathology.

[81]  W. Tremaine,et al.  Hereditary angioedema: a broad review for clinicians. , 2001, Archives of internal medicine.

[82]  M. Kazatchkine,et al.  A new type of acquired C1 inhibitor deficiency associated with systemic lupus erythematosus. , 2001, Arthritis and rheumatism.

[83]  A. D. De Schepper,et al.  CT of angioedema of the small bowel. , 2001, AJR. American journal of roentgenology.

[84]  K. Binkley,et al.  Clinical, biochemical, and genetic characterization of a novel estrogen-dependent inherited form of angioedema. , 2000, The Journal of allergy and clinical immunology.

[85]  K. Bork,et al.  Hereditary angioedema with normal C1-inhibitor activity in women , 2000, The Lancet.

[86]  A. Saincher A Canadian approach , 1999 .

[87]  A. Golik,et al.  Hereditary angioneurotic edema with severe hypovolemic shock. , 1993, Journal of clinical gastroenterology.

[88]  M. Cicardi,et al.  Hereditary and Acquired C1‐Inhibitor Deficiency: Biological and Clinical Characteristics in 235 Patients , 1992, Medicine.

[89]  B. Ferlazzo,et al.  Abdominal attack of hereditary angioedema associated with marked leucocytosis. A case report. , 1992, The Italian journal of gastroenterology.

[90]  A. Davis C1 inhibitor and hereditary angioneurotic edema. , 1988, Annual review of immunology.

[91]  C. Cochrane,et al.  Detection of active kallikrein in induced blister fluids of hereditary angioedema patients , 1980, The Journal of experimental medicine.

[92]  F. Rosen,et al.  Role of the second component of complement (C2) and plasmin in kinin release in hereditary angioneurotic edema (H.A.N.E.) plasma. , 1977, Transactions of the Association of American Physicians.

[93]  J. Atkinson,et al.  Hereditary angioedema: the clinical syndrome and its management. , 1976, Annals of internal medicine.

[94]  F. Rosen,et al.  Hereditary Angioneurotic Edema: Two Genetic Variants , 1965, Science.

[95]  R. R. Evans,et al.  A BIOCHEMICAL ABNORMALITY IN HEREDIATRY ANGIONEUROTIC EDEMA: ABSENCE OF SERUM INHIBITOR OF C' 1-ESTERASE. , 1963, The American journal of medicine.

[96]  E. Becker,et al.  Hereditary angioneurotic edema: II. Deficiency of inhibitor for serum globulin permeability factor and/or plasma kallikrein , 1962 .