Cargando…

Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain

The rarity of lipodystrophies implies that they are not well-known, leading to delays in diagnosis/misdiagnosis. The aim of this study was to assess the natural course and comorbidities of generalised and partial lipodystrophy in Spain to contribute to their understanding. Thus, a total of 140 patie...

Descripción completa

Detalles Bibliográficos
Autores principales: Fernández-Pombo, Antía, Sánchez-Iglesias, Sofía, Castro-Pais, Ana I., Ginzo-Villamayor, Maria José, Cobelo-Gómez, Silvia, Prado-Moraña, Teresa, Díaz-López, Everardo Josué, Casanueva, Felipe F., Loidi, Lourdes, Araújo-Vilar, David
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10687442/
https://www.ncbi.nlm.nih.gov/pubmed/38034001
http://dx.doi.org/10.3389/fendo.2023.1250203
_version_ 1785151979536777216
author Fernández-Pombo, Antía
Sánchez-Iglesias, Sofía
Castro-Pais, Ana I.
Ginzo-Villamayor, Maria José
Cobelo-Gómez, Silvia
Prado-Moraña, Teresa
Díaz-López, Everardo Josué
Casanueva, Felipe F.
Loidi, Lourdes
Araújo-Vilar, David
author_facet Fernández-Pombo, Antía
Sánchez-Iglesias, Sofía
Castro-Pais, Ana I.
Ginzo-Villamayor, Maria José
Cobelo-Gómez, Silvia
Prado-Moraña, Teresa
Díaz-López, Everardo Josué
Casanueva, Felipe F.
Loidi, Lourdes
Araújo-Vilar, David
author_sort Fernández-Pombo, Antía
collection PubMed
description The rarity of lipodystrophies implies that they are not well-known, leading to delays in diagnosis/misdiagnosis. The aim of this study was to assess the natural course and comorbidities of generalised and partial lipodystrophy in Spain to contribute to their understanding. Thus, a total of 140 patients were evaluated (77.1% with partial lipodystrophy and 22.9% with generalised lipodystrophy). Clinical data were collected in a longitudinal setting with a median follow-up of 4.7 (0.5-17.6) years. Anthropometry and body composition studies were carried out and analytical parameters were also recorded. The estimated prevalence of all lipodystrophies in Spain, excluding Köbberling syndrome, was 2.78 cases/million. The onset of phenotype occurred during childhood in generalised lipodystrophy and during adolescence-adulthood in partial lipodystrophy, with the delay in diagnosis being considerable for both cohorts. There are specific clinical findings that should be highlighted as useful features to take into account when making the differential diagnosis of these disorders. Patients with generalised lipodystrophy were found to develop their first metabolic abnormalities sooner and a different lipid profile has also been observed. Mean time to death was 83.8 ± 2.5 years, being shorter among patients with generalised lipodystrophy. These results provide an initial point of comparison for ongoing prospective studies such as the ECLip Registry study.
format Online
Article
Text
id pubmed-10687442
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-106874422023-11-30 Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain Fernández-Pombo, Antía Sánchez-Iglesias, Sofía Castro-Pais, Ana I. Ginzo-Villamayor, Maria José Cobelo-Gómez, Silvia Prado-Moraña, Teresa Díaz-López, Everardo Josué Casanueva, Felipe F. Loidi, Lourdes Araújo-Vilar, David Front Endocrinol (Lausanne) Endocrinology The rarity of lipodystrophies implies that they are not well-known, leading to delays in diagnosis/misdiagnosis. The aim of this study was to assess the natural course and comorbidities of generalised and partial lipodystrophy in Spain to contribute to their understanding. Thus, a total of 140 patients were evaluated (77.1% with partial lipodystrophy and 22.9% with generalised lipodystrophy). Clinical data were collected in a longitudinal setting with a median follow-up of 4.7 (0.5-17.6) years. Anthropometry and body composition studies were carried out and analytical parameters were also recorded. The estimated prevalence of all lipodystrophies in Spain, excluding Köbberling syndrome, was 2.78 cases/million. The onset of phenotype occurred during childhood in generalised lipodystrophy and during adolescence-adulthood in partial lipodystrophy, with the delay in diagnosis being considerable for both cohorts. There are specific clinical findings that should be highlighted as useful features to take into account when making the differential diagnosis of these disorders. Patients with generalised lipodystrophy were found to develop their first metabolic abnormalities sooner and a different lipid profile has also been observed. Mean time to death was 83.8 ± 2.5 years, being shorter among patients with generalised lipodystrophy. These results provide an initial point of comparison for ongoing prospective studies such as the ECLip Registry study. Frontiers Media S.A. 2023-11-16 /pmc/articles/PMC10687442/ /pubmed/38034001 http://dx.doi.org/10.3389/fendo.2023.1250203 Text en Copyright © 2023 Fernández-Pombo, Sánchez-Iglesias, Castro-Pais, Ginzo-Villamayor, Cobelo-Gómez, Prado-Moraña, Díaz-López, Casanueva, Loidi and Araújo-Vilar https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Endocrinology
Fernández-Pombo, Antía
Sánchez-Iglesias, Sofía
Castro-Pais, Ana I.
Ginzo-Villamayor, Maria José
Cobelo-Gómez, Silvia
Prado-Moraña, Teresa
Díaz-López, Everardo Josué
Casanueva, Felipe F.
Loidi, Lourdes
Araújo-Vilar, David
Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title_full Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title_fullStr Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title_full_unstemmed Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title_short Natural history and comorbidities of generalised and partial lipodystrophy syndromes in Spain
title_sort natural history and comorbidities of generalised and partial lipodystrophy syndromes in spain
topic Endocrinology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10687442/
https://www.ncbi.nlm.nih.gov/pubmed/38034001
http://dx.doi.org/10.3389/fendo.2023.1250203
work_keys_str_mv AT fernandezpomboantia naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT sancheziglesiassofia naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT castropaisanai naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT ginzovillamayormariajose naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT cobelogomezsilvia naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT pradomoranateresa naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT diazlopezeverardojosue naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT casanuevafelipef naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT loidilourdes naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain
AT araujovilardavid naturalhistoryandcomorbiditiesofgeneralisedandpartiallipodystrophysyndromesinspain