Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita

Detalhes bibliográficos
Ano de defesa: 2022
Autor(a) principal: Madeira, Mayara Ponte
Orientador(a): Montenegro Junior, Renan Magalhães
Banca de defesa: Não Informado pela instituição
Tipo de documento: Tese
Tipo de acesso: Acesso aberto
Idioma: por
Instituição de defesa: Não Informado pela instituição
Programa de Pós-Graduação: Não Informado pela instituição
Departamento: Não Informado pela instituição
País: Não Informado pela instituição
Palavras-chave em Português:
Link de acesso: http://www.repositorio.ufc.br/handle/riufc/66933
Resumo: A new strain of human coronavirus (HCoV) spread rapidly around the world. Diabetes and obesity are associated with a worse prognosis in these patients. Patients with Congenital Generalized Lipodystrophy (CGL) often have diabetes that is difficult to control and require extremely high doses of insulin. There is no documentation in the literature of cases of COVID19 in patients with CGL. Thus, our objective was to assess the prevalence of SARS-CoV-2 infection and the association between the outcomes of this infection and clinical and metabolic characteristics in patients with CGL. This is a cross-sectional study carried out between July and October 2020. Clinical data collected were respiratory or other flu-like symptoms, need of hospitalization in the last three montHAS, CGL comorbidities, and medications in use. Cholesterol, triglycerides, glycohemoglobin A1c levels, anti-SARS-CoV-2 antibodies and nasopharyngeal swab for RT-qPCR were also obtained in all CGL patients. Pearson's chi-square test and Fisher's exact test were used to investigate the association between categorical variables. A significance level of 5% was adopted. We evaluated 22 out of 24 patients with CGL under follow-up at a specialized outpatient in Ceará, with a median age of 20 years (1 to 42 years). Eight of these patients (36.4%) had reactive anti-SARS-CoV-2 antibodies (SARSCoV-2+) and only one of them also had detectable RT-qPCR at the time of evaluation, with a median age of 13.5 years (1 to 37), of which 5 (62.5%) were women. Symptoms such as fever, malaise, nausea, diarrhea and chest pain were reported, but all children were asymptomatic. All patients had inadequate metabolic control, with no difference between those who had SARSCoV-2+ or not. Among reactive individuals, there was no difference between those subtypes of LGC, with mutations in the AGPAT2 (75%) and BSCL2 (25%) genes. No patient required hospitalization or died. In conclusion, this is the first description of clinical outcomes of SARSCoV-2 infection in patients with CGL. Despite the high cardiovascular risk and severe metabolic comorbidities in this rare disease, excellent evolution was observed in all cases. Such findings may be related to the predominance of young individuals or the absence of peripheral adipose tissue in CGL and its potential role as a viral reservoir. Although this is a significant Brazilian case series of LGC, further studies will be needed to confirm this association.
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spelling Madeira, Mayara PonteMontenegro Junior, Renan Magalhães2022-07-06T11:12:35Z2022-07-06T11:12:35Z2022-04-27MADEIRA M. P. Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita. 2022. Dissertação. (Mestrado em Ciências Médicas) - Faculdade de Medicina, Universidade Federal do Ceará, Fortaleza, 2022. Disponível em: http://www.repositorio.ufc.br/handle/riufc/66933. Acesso: 06 jul. 2022.http://www.repositorio.ufc.br/handle/riufc/66933A new strain of human coronavirus (HCoV) spread rapidly around the world. Diabetes and obesity are associated with a worse prognosis in these patients. Patients with Congenital Generalized Lipodystrophy (CGL) often have diabetes that is difficult to control and require extremely high doses of insulin. There is no documentation in the literature of cases of COVID19 in patients with CGL. Thus, our objective was to assess the prevalence of SARS-CoV-2 infection and the association between the outcomes of this infection and clinical and metabolic characteristics in patients with CGL. This is a cross-sectional study carried out between July and October 2020. Clinical data collected were respiratory or other flu-like symptoms, need of hospitalization in the last three montHAS, CGL comorbidities, and medications in use. Cholesterol, triglycerides, glycohemoglobin A1c levels, anti-SARS-CoV-2 antibodies and nasopharyngeal swab for RT-qPCR were also obtained in all CGL patients. Pearson's chi-square test and Fisher's exact test were used to investigate the association between categorical variables. A significance level of 5% was adopted. We evaluated 22 out of 24 patients with CGL under follow-up at a specialized outpatient in Ceará, with a median age of 20 years (1 to 42 years). Eight of these patients (36.4%) had reactive anti-SARS-CoV-2 antibodies (SARSCoV-2+) and only one of them also had detectable RT-qPCR at the time of evaluation, with a median age of 13.5 years (1 to 37), of which 5 (62.5%) were women. Symptoms such as fever, malaise, nausea, diarrhea and chest pain were reported, but all children were asymptomatic. All patients had inadequate metabolic control, with no difference between those who had SARSCoV-2+ or not. Among reactive individuals, there was no difference between those subtypes of LGC, with mutations in the AGPAT2 (75%) and BSCL2 (25%) genes. No patient required hospitalization or died. In conclusion, this is the first description of clinical outcomes of SARSCoV-2 infection in patients with CGL. Despite the high cardiovascular risk and severe metabolic comorbidities in this rare disease, excellent evolution was observed in all cases. Such findings may be related to the predominance of young individuals or the absence of peripheral adipose tissue in CGL and its potential role as a viral reservoir. Although this is a significant Brazilian case series of LGC, further studies will be needed to confirm this association.Uma nova cepa de coronavírus humano (HCoV) se disseminou rapidamente pelo mundo. Diabetes e obesidade estão associados a pior prognóstico nesses pacientes. Pacientes com Lipodistrofia Generalizada Congênita (LGC) geralmente têm diabetes de difícil controle e requerem doses extremamente altas de insulina. Não há documentação na literatura de casos de COVID-19 em pacientes com LGC. Assim, nosso objetivo foi avaliar a prevalência de infecção por SARS-CoV-2 e a associação entre os desfechos dessa infecção e características clínicas e metabólicas em pacientes com LGC. Este é um estudo transversal realizado entre julho e outubro de 2020. Os dados clínicos coletados foram sintomas respiratórios ou outros sintomas gripais, necessidade de internação nos últimos três meses, comorbidades e medicamentos em uso. Níveis sanguíneos de colesterol, triglicerídeos, glicohemoglobina A1c, anticorpos antiSARS-CoV-2 e swab nasofaríngeo para RT-qPCR também foram obtidos em todos os pacientes com LGC. O teste do qui-quadrado de Pearson e o teste exato de Fisher foram utilizados na investigação da associação entre variáveis categóricas. Foi adotado nível de significância de 5%. Foram avaliados 22 dentre 24 pacientes com LGC em seguimento em ambulatório especializado no Ceará, com mediana de idade de 20 anos (1 a 42). Oito desses pacientes (36,4%) tinham anticorpos anti-SARS-CoV-2 reativos (SARS-CoV-2+) e somente um deles também apresentou RT-qPCR detectável no momento da avaliação, com mediana de idade de 13,5 anos (1 a 37), dos quais 5 (62,5%) eram mulheres. Sintomas como febre, mal-estar, náusea, diarreia e dor no peito foram relatados, porém todas as crianças estavam assintomáticas. Todos os pacientes encontravam-se com controle metabólico inadequado, sem diferença entre aqueles que tiveram SARS-CoV-2 + ou não. Entre os indivíduos reativos não houve diferença entre aqueles ossubtipos de LGC, com mutações nos genes AGPAT2 (75%) e BSCL2 (25%). Nenhum paciente precisou de hospitalização ou evoluiu à óbito. Em conclusão, trata-se da primeira descrição de desfechos clínicos de infecção por SARS-CoV-2 em pacientes com LGC. Apesar do alto risco cardiovascular e das comorbidades metabólicas graves nessa rara enfermidade, foi observada excelente evolução em todos os casos. Tais achados podem estar relacionados ao predomínio de indivíduos jovens ou a ausência de tecido adiposo periférico na LGC e seu potencial papel como reservatório viral. Embora se trate de significativa casuística brasileira de LGC, novos estudos serão necessários para confirmar dessa associação.COVID-19Lipodistrofia Generalizada CongênitaSARS-CoV-2Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênitainfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/doctoralThesisporreponame:Repositório Institucional da Universidade Federal do Ceará (UFC)instname:Universidade Federal do Ceará (UFC)instacron:UFCinfo:eu-repo/semantics/openAccessORIGINAL2022_dis_mpmadeira.pdf2022_dis_mpmadeira.pdfapplication/pdf2574193http://repositorio.ufc.br/bitstream/riufc/66933/1/2022_dis_mpmadeira.pdfb815c253cdbbb9513a02b608bce9f05bMD51LICENSElicense.txtlicense.txttext/plain; charset=utf-82152http://repositorio.ufc.br/bitstream/riufc/66933/3/license.txtfb3ad2d23d9790966439580114baefafMD53riufc/669332022-07-06 08:24:09.224oai:repositorio.ufc.br:riufc/66933TElDRU7Dh0EgREUgQVJNQVpFTkFNRU5UTyBFIERJU1RSSUJVScOHw4NPIE7Dg08tRVhDTFVTSVZBIAoKQW8gY29uY29yZGFyIGNvbSBlc3RhIGxpY2Vuw6dhLCB2b2PDqihzKSBhdXRvcihlcykgb3UgdGl0dWxhcihlcykgZG9zIGRpcmVpdG9zIGF1dG9yYWlzIGRhIG9icmEgYXF1aSBkZXNjcml0YSBjb25jZWRlKG0pIMOgIFVuaXZlcnNpZGFkZSBGZWRlcmFsIGRvIENlYXLDoSwgZ2VzdG9yYSBkbyBSZXBvc2l0w7NyaW8gSW5zdGl0dWNpb25hbCBkYSBVRkMgLSBSSS9VRkMsIG8gZGlyZWl0byBuw6NvLWV4Y2x1c2l2byBkZSByZXByb2R1emlyLCBjb252ZXJ0ZXIgKGNvbW8gZGVmaW5pZG8gYWJhaXhvKSBlL291IGRpc3RyaWJ1aXIgbyBkb2N1bWVudG8gZGVwb3NpdGFkbyBlbSBmb3JtYXRvIGltcHJlc3NvLCBlbGV0csO0bmljbyBvdSBlbSBxdWFscXVlciBvdXRybyBtZWlvLiBWb2PDqiBjb25jb3JkYShtKSBxdWUgYSBVbml2ZXJzaWRhZGUgRmVkZXJhbCBkbyBDZWFyw6EsIGdlc3RvcmEgZG8gUmVwb3NpdMOzcmlvIEluc3RpdHVjaW9uYWwgZGEgVUZDIC0gUkkvVUZDLCBwb2RlLCBzZW0gYWx0ZXJhciBvIGNvbnRlw7pkbywgY29udmVydGVyIG8gYXJxdWl2byBkZXBvc2l0YWRvIGEgcXVhbHF1ZXIgbWVpbyBvdSBmb3JtYXRvIGNvbSBmaW5zIGRlIHByZXNlcnZhw6fDo28uIFZvY8OqKHMpIHRhbWLDqW0gY29uY29yZGEobSkgcXVlIGEgVW5pdmVyc2lkYWRlIEZlZGVyYWwgZG8gQ2VhcsOhLCBnZXN0b3JhIGRvIFJlcG9zaXTDs3JpbyBJbnN0aXR1Y2lvbmFsIGRhIFVGQyAtIFJJL1VGQywgcG9kZSBtYW50ZXIgbWFpcyBkZSB1bWEgY8OzcGlhIGRlc3RlIGRlcMOzc2l0byBwYXJhIGZpbnMgZGUgc2VndXJhbsOnYSwgYmFjay11cCBlL291IHByZXNlcnZhw6fDo28uIFZvY8OqIGRlY2xhcmEgcXVlIGEgYXByZXNlbnRhw6fDo28gZG8gc2V1IHRyYWJhbGhvIMOpIG9yaWdpbmFsIGUgcXVlIHZvY8OqKHMpIHBvZGUobSkgY29uY2VkZXIgb3MgZGlyZWl0b3MgY29udGlkb3MgbmVzdGEgbGljZW7Dp2EuIFZvY8OqIHRhbWLDqW0gZGVjbGFyYShtKSBxdWUgbyBlbnZpbyDDqSBkZSBzZXUgY29uaGVjaW1lbnRvIGUgbsOjbyBpbmZyaW5nZSBvcyBkaXJlaXRvcyBhdXRvcmFpcyBkZSBvdXRyYSBwZXNzb2Egb3UgaW5zdGl0dWnDp8Ojby4gQ2FzbyBvIGRvY3VtZW50byBhIHNlciBkZXBvc2l0YWRvIGNvbnRlbmhhIG1hdGVyaWFsIHBhcmEgbyBxdWFsIHZvY8OqKHMpIG7Do28gZGV0w6ltIGEgdGl0dWxhcmlkYWRlIGRvcyBkaXJlaXRvcyBkZSBhdXRvcmFpcywgdm9jw6oocykgZGVjbGFyYShtKSBxdWUgb2J0ZXZlIGEgcGVybWlzc8OjbyBpcnJlc3RyaXRhIGRvIHRpdHVsYXIgZG9zIGRpcmVpdG9zIGF1dG9yYWlzIGRlIGNvbmNlZGVyIMOgIFVuaXZlcnNpZGFkZSBGZWRlcmFsIGRvIENlYXLDoSwgZ2VzdG9yYSBkbyBSZXBvc2l0w7NyaW8gSW5zdGl0dWNpb25hbCBkYSBVRkMgLSBSSS9VRkMsIG9zIGRpcmVpdG9zIHJlcXVlcmlkb3MgcG9yIGVzdGEgbGljZW7Dp2EgZSBxdWUgb3MgbWF0ZXJpYWlzIGRlIHByb3ByaWVkYWRlIGRlIHRlcmNlaXJvcywgZXN0w6NvIGRldmlkYW1lbnRlIGlkZW50aWZpY2Fkb3MgZSByZWNvbmhlY2lkb3Mgbm8gdGV4dG8gb3UgY29udGXDumRvIGRhIGFwcmVzZW50YcOnw6NvLgogQ0FTTyBPIFRSQUJBTEhPIERFUE9TSVRBRE8gVEVOSEEgU0lETyBGSU5BTkNJQURPIE9VIEFQT0lBRE8gUE9SIFVNIMOTUkfDg08sIFFVRSBOw4NPIEEgSU5TVElUVUnDh8ODTyBERVNURSBSRVBPU0lUw5NSSU86IFZPQ8OKIERFQ0xBUkEgVEVSIENVTVBSSURPIFRPRE9TIE9TIERJUkVJVE9TIERFIFJFVklTw4NPIEUgUVVBSVNRVUVSIE9VVFJBUyBPQlJJR0HDh8OVRVMgUkVRVUVSSURBUyBQRUxPIENPTlRSQVRPIE9VIEFDT1JETy4gCk8gcmVwb3NpdMOzcmlvIGlkZW50aWZpY2Fyw6EgY2xhcmFtZW50ZSBvIHNldShzKSBub21lKHMpIGNvbW8gYXV0b3IoZXMpIG91IHRpdHVsYXIoZXMpIGRvIGRpcmVpdG8gZGUgYXV0b3IoZXMpIGRvIGRvY3VtZW50byBzdWJtZXRpZG8gZSBkZWNsYXJhIHF1ZSBuw6NvIGZhcsOhIHF1YWxxdWVyIGFsdGVyYcOnw6NvIGFsw6ltIGRhcyBwZXJtaXRpZGFzIHBvciBlc3RhIGxpY2Vuw6dhLgpSZXBvc2l0w7NyaW8gSW5zdGl0dWNpb25hbCBkYSBVRkMuCg==Repositório InstitucionalPUBhttp://www.repositorio.ufc.br/ri-oai/requestbu@ufc.br || repositorio@ufc.bropendoar:2022-07-06T11:24:09Repositório Institucional da Universidade Federal do Ceará (UFC) - Universidade Federal do Ceará (UFC)false
dc.title.pt_BR.fl_str_mv Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
title Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
spellingShingle Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
Madeira, Mayara Ponte
COVID-19
Lipodistrofia Generalizada Congênita
SARS-CoV-2
title_short Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
title_full Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
title_fullStr Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
title_full_unstemmed Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
title_sort Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita
author Madeira, Mayara Ponte
author_facet Madeira, Mayara Ponte
author_role author
dc.contributor.author.fl_str_mv Madeira, Mayara Ponte
dc.contributor.advisor1.fl_str_mv Montenegro Junior, Renan Magalhães
contributor_str_mv Montenegro Junior, Renan Magalhães
dc.subject.por.fl_str_mv COVID-19
Lipodistrofia Generalizada Congênita
SARS-CoV-2
topic COVID-19
Lipodistrofia Generalizada Congênita
SARS-CoV-2
description A new strain of human coronavirus (HCoV) spread rapidly around the world. Diabetes and obesity are associated with a worse prognosis in these patients. Patients with Congenital Generalized Lipodystrophy (CGL) often have diabetes that is difficult to control and require extremely high doses of insulin. There is no documentation in the literature of cases of COVID19 in patients with CGL. Thus, our objective was to assess the prevalence of SARS-CoV-2 infection and the association between the outcomes of this infection and clinical and metabolic characteristics in patients with CGL. This is a cross-sectional study carried out between July and October 2020. Clinical data collected were respiratory or other flu-like symptoms, need of hospitalization in the last three montHAS, CGL comorbidities, and medications in use. Cholesterol, triglycerides, glycohemoglobin A1c levels, anti-SARS-CoV-2 antibodies and nasopharyngeal swab for RT-qPCR were also obtained in all CGL patients. Pearson's chi-square test and Fisher's exact test were used to investigate the association between categorical variables. A significance level of 5% was adopted. We evaluated 22 out of 24 patients with CGL under follow-up at a specialized outpatient in Ceará, with a median age of 20 years (1 to 42 years). Eight of these patients (36.4%) had reactive anti-SARS-CoV-2 antibodies (SARSCoV-2+) and only one of them also had detectable RT-qPCR at the time of evaluation, with a median age of 13.5 years (1 to 37), of which 5 (62.5%) were women. Symptoms such as fever, malaise, nausea, diarrhea and chest pain were reported, but all children were asymptomatic. All patients had inadequate metabolic control, with no difference between those who had SARSCoV-2+ or not. Among reactive individuals, there was no difference between those subtypes of LGC, with mutations in the AGPAT2 (75%) and BSCL2 (25%) genes. No patient required hospitalization or died. In conclusion, this is the first description of clinical outcomes of SARSCoV-2 infection in patients with CGL. Despite the high cardiovascular risk and severe metabolic comorbidities in this rare disease, excellent evolution was observed in all cases. Such findings may be related to the predominance of young individuals or the absence of peripheral adipose tissue in CGL and its potential role as a viral reservoir. Although this is a significant Brazilian case series of LGC, further studies will be needed to confirm this association.
publishDate 2022
dc.date.accessioned.fl_str_mv 2022-07-06T11:12:35Z
dc.date.available.fl_str_mv 2022-07-06T11:12:35Z
dc.date.issued.fl_str_mv 2022-04-27
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
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dc.identifier.citation.fl_str_mv MADEIRA M. P. Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita. 2022. Dissertação. (Mestrado em Ciências Médicas) - Faculdade de Medicina, Universidade Federal do Ceará, Fortaleza, 2022. Disponível em: http://www.repositorio.ufc.br/handle/riufc/66933. Acesso: 06 jul. 2022.
dc.identifier.uri.fl_str_mv http://www.repositorio.ufc.br/handle/riufc/66933
identifier_str_mv MADEIRA M. P. Infecção por Sars-cov-2 em pacientes com lipodistrofia generalizada congênita. 2022. Dissertação. (Mestrado em Ciências Médicas) - Faculdade de Medicina, Universidade Federal do Ceará, Fortaleza, 2022. Disponível em: http://www.repositorio.ufc.br/handle/riufc/66933. Acesso: 06 jul. 2022.
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