Abstract
Abdominal angioedema attacks are a frequent and typical symptom of hereditary angioedema (HAE) but very often generate diagnostic problems. The study presents laboratory and clinical findings of 7 patients with HAE 1/2 hospitalized due to severe attacks. In all cases, at admittance severe abdominal pain, flatulence, strong weakness, different grade of nausea/vomiting or diarrhoea and abundant free fluid in peritoneal cavity were present. In the history of all patients, recurrent 2 to 3 day long abdominal attack with ascites, were announced. Laboratory data done before the treatment showed elevated leukocytosis, hematocrit, serum glucose, high D-dimers and decreased value of APTT. All patients had an abdominal ultrasound examination, in 5 patients additional abdominal angio-CT was performed to exclude thromboembolic episode. The infusion of human C1 inhibitor concentrate was administered as causative treatment. Completely withdrawal of symptoms was noted up to72 hrs after infusion. In addition all laboratory parameters normalized as well as the free fluid in abdominal cavity disappeared, however, D-dimers serum level despite a decreasing tendency reached the normal range just after 2 weeks.
Highlights
Hereditary angioedema (HAE) is one of bradykinin dependent edema
During the course of both types of HAE, the attacks of angioedema are localized in the skin or mucous of the respiratory and gastrointestinal tract [4, 5]
The bradykinin angioedema attack differs from other swellings, including the most common histamine- dependent swelling, in longer duration, slower build-up phase, lack of urticaria, lack of pruritus and presence of prodromal symptoms [3]
Summary
Hereditary angioedema (HAE) is one of bradykinin dependent edema. Is inherited in an autosomal dominant manner. The bradykinin angioedema attack differs from other swellings, including the most common histamine- dependent swelling, in longer duration, slower build-up phase, lack of urticaria, lack of pruritus and presence of prodromal symptoms [3]. It might lead to hypovolaemia and shock Both abdominal and laryngeal attacks are life-threatening conditions. Surgical treatment of HAE abdominal attacks was the most common mistake as a result of lack of, or wrong differential diagnosis [4, 5]. The typical edema treatment, like anti- histamine drugs, systemic steroids or adrenaline in bradykinin dependent edema, is ineffective. It requires administration of human or recombinant C1-INH, a bradykinin receptor blocker or a kallikrein inhibitor. The aim of the study was the retrospective analysis of patients with HAE abdominal attacks who were hospitalized due to the severity of the attacks
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