Hereditary angioedema

Hereditary angioedema (HAE)
Other namesHereditary angioneurotic edema (HANE), familial angioneurotic edema
Swollen right hand during a hereditary angioedema attack.
SpecialtyHematology
SymptomsRecurrent attacks of severe swelling
Usual onsetChildhood
DurationAttacks last a few days (without treatment)
TypesType I, II, III
CausesGenetic disorder (autosomal dominant)
Diagnostic methodMeasuring C4 and C1-inhibitor levels.
Differential diagnosisIntestinal obstruction, other types of angioedema
Preventionplasma kallikrein inhibitors, C1 inhibitor concentrates
TreatmentSupportive care, medications
MedicationC1 inhibitor, ecallantide, icatibant, sebetralstat
Prognosis25% risk of death if airway involved (without treatment)
Frequency~1 in 50,000

Hereditary angioedema (HAE) is a rare disease that results in recurrent attacks of severe swelling. HAE affects approximately 1 in 50,000 people. The condition is typically first noticed in childhood.

The swelling most commonly affects the arms, legs, face, intestinal tract, and airway. If the intestinal tract is affected, abdominal pain and vomiting may occur. Swelling of the airway can result in its obstruction and can cause trouble breathing. Without treatment, this can cause a risk of death in about 25% of people. With treatment, outcomes are generally good. Without preventive treatment, attacks typically occur every two weeks and last for a few days.

There are three main types of HAE. Types I and II are caused by a mutation in the SERPING1 gene, which encodes the C1 inhibitor protein, and type III, now called HAE with normal C1 inhibitor (HAE-nl-C1INH). Types I and II affect females and males equally, while HAE with normal C1 inhibitor affects females more often than males.

Six known mutations are described in the literature under HAE with normal C1 inhibitor. The result is increased levels of kallikrein activity and bradykinin, which promotes swelling. The condition may be inherited in an autosomal dominant manner or occur as a spontaneous mutation.

Known common HAE triggers are:

  • Stress
  • Minor trauma
  • Anxiety
  • Surgery
  • Ailments such as colds/flu/other viral infections

People with HAE have also reported exposure to cold and activities that cause mechanical trauma (gardening, hammering, shoveling) can also be triggers. Attacks can often occur without any obvious preceding events or a known trigger.

Diagnosis of HAE types I and II is based on measurement of C4 and C1-inhibitor levels or genetic testing.

Management of HAE involves efforts to prevent attacks and the treatment of attacks if they occur. During an attack, supportive care such as intravenous fluids and airway support may be required. Preventative treatments include C1-inhibitor concentrates and plasma kallikrein inhibitors. Acute treatment options include C1-inhibitor concentrates, bradykinin receptor antagonists like icatibant, and plasma kallikrein inhibitors such as ecallantide and sebetralstat.

The condition was first described in 1888 by Canadian physician William Osler.