Journal Information
Vol. 98. Issue 6.
Pages 483-485 (1 June 2023)
Vol. 98. Issue 6.
Pages 483-485 (1 June 2023)
Images in Paediatrics
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Remember bradykinin-induced angioedema — an unforgettable image
Recuerde el angioedema inducido por bradicinina: una imagen inolvidable
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1278
Nélia Santos Gaspara,b,
Corresponding author
, André Costa e Silvaa,c, Sylvia Jacoba, Diana Bordaloa,d
a Servicio de Inmunología y Alergia, Centro Hospitalar e Universitário de São João, Oporto, Portugal
b Servicio de Pediatría, Centro Hospitalar Médio Tejo, Torres Novas, Portugal
c Servicio de Pediatría, Unidad Local de Salud Alto Minho, Viana do Castelo, Portugal
d Departamento de Obstetricia y Ginecología, Faculdad de Medicina, Universidade do Porto, Oporto, Portugal
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A boy aged 4 years presented to the emergency department (ED) with exuberant facial swelling of 24 hours duration with progressive worsening. He had been evaluated earlier at a different ED, where he received oral antihistaminic and steroid drugs without subsequent improvement. The physical examination revealed pronounced oedema in the eyelids, lips and nose (Fig. 1). Due to a strong suspicion of bradykinin-induced angio-oedema, C1 inhibitor (C1-INH) was administered through the intravenous (IV) route (twice), followed by IV aminocaproic acid and subcutaneous icatibant (twice) due to a lack of response. A review of the medical history revealed a previous diagnosis of acquired angioedema due to C1-INH deficiency following several episodes of feet swelling. In a few hours, the patient exhibited slow but progressive improvement (Fig. 2). At present, the patient is in treatment with tranexamic acid for prophylaxis and on-demand icatibant.

Figure 1.

Initial presentation at the emergency department during the acute exacerbation.

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Figure 2.

The patient at discharge, after the administration of IV C1 inhibitor, IV aminocaproic acid and subcutaneous icatibant.

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Angio-oedema and C1-INH deficiency are potentially fatal diseases characterised by oedema of the skin or mucosae involving chiefly the skin, gastrointestinal tract and upper respiratory tract.1–3 Early treatment reduces the duration of the attack, so it is crucial to identify patients who could benefit from it (Fig. 3).1–3 In the case of our patient, the delay in the initiation of appropriate treatment could explain his delayed response, which underscores the importance of the identification and treatment of angio-oedema in order to prevent the associated morbidity and mortality.

Figure 3.

Management of patients with hereditary angioedema.

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Ethical considerations

The authors obtained the written informed consent of the legal guardians of the patient.

References
[1]
M. Maurer, M. Magerl, S. Betschel, W. Aberer, I.J. Ansotegui, E. Aygoren-Pursun, et al.
The international WAO/EAACI guideline for the management of hereditary angioedema - The 2021 revision and update.
Allergy., 77 (2022), pp. 1961-1990
[2]
A. Krogulska, D. Lewandowska, H. Ludwig, A. Dąbrowska, A. Kowalczyk.
Hereditary angioedema as a disease of different clinical courses and difficult diagnosis, particularly in children – a case report and literature review.
Adv Dermatol Allergol, 38 (2021), pp. 1118-1121
[3]
P. Busse, S.C. Christiansen, M.A. Riedl, A. Banerji, J.A. Bernstein, A.J. Castaldo, et al.
US HAEA Medical Advisory Board 2020 Guidelines for the Management of Hereditary Angioedema.
J Allergy Clin Immunol Pract., 9 (2021), pp. 1
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