tetano
Editor, Senior Moderator
Pathogens
. 2023 Mar 2;12(3):403.
doi: 10.3390/pathogens12030403.
Mild COVID-19 in an APECED Patient with Chronic Inflammatory Demyelinating Polyneuropathy (CIDP) and High Titer of Type 1 IFN-Abs: A Case Report
Mariella Valenzise[SUP] 1 [/SUP], Simone Foti Randazzese[SUP] 1 [/SUP], Fabio Toscano[SUP] 1 [/SUP], Fortunato Lombardo[SUP] 1 [/SUP], Giuseppina Salzano[SUP] 1 [/SUP], Cristina Pajno[SUP] 2 [/SUP], Malgorzata Wasniewska[SUP] 1 [/SUP], Antonio Cascio[SUP] 3 4 [/SUP], Maureen A Su[SUP] 5 [/SUP]
Affiliations
Abstract
Autoimmune-Poly-Endocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED), caused by mutations in the Autoimmune Regulator (AIRE) gene, is an autosomal recessive multi-organ autoimmunity syndrome usually defined by high serum titers of type I Interferon Autoantibodies (Type 1 IFN-Abs). These antibodies have recently been found in individuals in the general population who develop life-threatening Coronavirus Disease 2019 (COVID-19), but the significance of pre-existing Type 1 IFN-Abs in APECED patients with COVID-19 remains unclear. Previous reports of COVID-19 outcomes in APECED patients have been divergent, and protective roles have been proposed for female sex, age <26 years, and immunomodulatory medications including intravenous immunoglobulin (IVIg). We report the case of a 30-year-old male APECED patient who experienced a Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2) infection with mild symptoms of fatigue and headache without respiratory distress and did not require hospitalization. He received a stress dose of hydrocortisone for adrenal insufficiency and continued on his baseline medications, including subcutaneous administration of Immunoglobulins (SCIgs) for chronic inflammatory demyelinating polyneuropathy (CIDP). Mild COVID-19 in a 30-year-old male patient with APECED and pre-existing Type 1 IFN-Abs was unexpected. Younger age and management of autoimmunity may have played a role.
Keywords: APECED; COVID-19; case report; corticosteroids; subcutaneous immunoglobulins; type 1 IFN-Abs.
. 2023 Mar 2;12(3):403.
doi: 10.3390/pathogens12030403.
Mild COVID-19 in an APECED Patient with Chronic Inflammatory Demyelinating Polyneuropathy (CIDP) and High Titer of Type 1 IFN-Abs: A Case Report
Mariella Valenzise[SUP] 1 [/SUP], Simone Foti Randazzese[SUP] 1 [/SUP], Fabio Toscano[SUP] 1 [/SUP], Fortunato Lombardo[SUP] 1 [/SUP], Giuseppina Salzano[SUP] 1 [/SUP], Cristina Pajno[SUP] 2 [/SUP], Malgorzata Wasniewska[SUP] 1 [/SUP], Antonio Cascio[SUP] 3 4 [/SUP], Maureen A Su[SUP] 5 [/SUP]
Affiliations
- PMID: 36986325
- DOI: 10.3390/pathogens12030403
Abstract
Autoimmune-Poly-Endocrinopathy-Candidiasis-Ectodermal Dystrophy (APECED), caused by mutations in the Autoimmune Regulator (AIRE) gene, is an autosomal recessive multi-organ autoimmunity syndrome usually defined by high serum titers of type I Interferon Autoantibodies (Type 1 IFN-Abs). These antibodies have recently been found in individuals in the general population who develop life-threatening Coronavirus Disease 2019 (COVID-19), but the significance of pre-existing Type 1 IFN-Abs in APECED patients with COVID-19 remains unclear. Previous reports of COVID-19 outcomes in APECED patients have been divergent, and protective roles have been proposed for female sex, age <26 years, and immunomodulatory medications including intravenous immunoglobulin (IVIg). We report the case of a 30-year-old male APECED patient who experienced a Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2) infection with mild symptoms of fatigue and headache without respiratory distress and did not require hospitalization. He received a stress dose of hydrocortisone for adrenal insufficiency and continued on his baseline medications, including subcutaneous administration of Immunoglobulins (SCIgs) for chronic inflammatory demyelinating polyneuropathy (CIDP). Mild COVID-19 in a 30-year-old male patient with APECED and pre-existing Type 1 IFN-Abs was unexpected. Younger age and management of autoimmunity may have played a role.
Keywords: APECED; COVID-19; case report; corticosteroids; subcutaneous immunoglobulins; type 1 IFN-Abs.