Co-occurrence between hereditary angioedema and multiple sclerosis: Therapeutic management of both diseases with fingolimod

Autor: Aigli G. Vakrakou, Dimitrios Tzanetakos, Eirini Giagkou, Maria-Eleptheria Evangelopoulos, Maria Anagnostouli, Elissavet Andreadou, Georgios Koutsis, Antonios Dimitrakopoulos, Elias Gialafos, John S. Tzartos, Evangelia Kompoti, George E. Fragoulis, Leonidas Stefanis, Constantinos Kilidireas
Rok vydání: 2022
Předmět:
Zdroj: Clinical Neurology and Neurosurgery. 216:107222
ISSN: 0303-8467
DOI: 10.1016/j.clineuro.2022.107222
Popis: Hereditary angioedema (HAE) related to C1 esterase-inhibitor deficiency activates the classic complement pathway and results to edematous crises. Although HAE is usually associated with multiple immunoregulatory disorders, neurologic manifestations are rare.We report on the case study of a 33-year-old man diagnosed with HAE (SERPIN1G gene mutation) and multiple sclerosis (MS), followed up for at least 6 years. After a first clinical attack of HEA with scrotal edema, MS disease exacerbation was observed. Treatment with glatiramer acetate could not prevent either MS or HAE clinical attacks with recurrent exacerbations been observed. Remission of MS and significant amelioration of HAE attacks were achieved under fingolimod treatment.Herein we provide long term evaluation of an extremely rare case of concomitant existence of HAE and MS and present the effects of MS current disease-modifying therapies in HAE attacks. Our case highlights the possible effect of fingolimod in immunoregulatory-mechanisms implicated in both diseases.
Databáze: OpenAIRE
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