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J Clin Densitom ; 21(1): 61-67, 2018.
Artigo em Inglês | MEDLINE | ID: mdl-27894728

RESUMO

Berardinelli-Seip congenital lipodystrophy (BSCL) is a rare autosomal recessive syndrome characterized by a difficulty storing lipid in adipocytes, low body fat, hypoleptinemia, and hyperinsulinemia. We report here laboratory, bone mineral density (BMD), and bone mineral content findings of 21 patients (24.1 ± 8.4 yr old, 14 females, 18 diabetics, 5.3% total body fat) with BSCL. The mean leptin was very low (0.91 ± 0.42 ng/mL), and the mean values of the Z-scores for all studied sites were positive, except for the 33% radius (Z-score -0.5 standard deviation [SD]). Twelve patients (57.1%) had a BMD Z-score higher than +2.5 SD in at least 1 site. There was no significant difference in the Z-scores between males and females. None of type 1 (AGPAT2) patients had Z-scores higher than +2.5 SD, and these patients had a smaller Z-score of BMD total body (0.26 SD vs 1.90 SD, p = 0.022) and of bone mineral content (1.59 SD vs 3.3 SD, p = 0.032) than type 2 (seipin) patients. Insulin, as well as HOMAIR (homeostasis model assessment), correlated positively with the BMD of all sites, except for the 33% radius. Z-Scores on this site (33% radius) were the smallest of all. More than half of our patients with BSCL have BMD Z-scores higher than +2.5 SD on at least 1 site, and this increase is more pronounced in the trabecular sites and in type 2 patients.


Assuntos
Densidade Óssea , Osso Esponjoso/diagnóstico por imagem , Insulina/sangue , Leptina/sangue , Lipodistrofia Generalizada Congênita/diagnóstico por imagem , Lipodistrofia Generalizada Congênita/fisiopatologia , Aciltransferases/genética , Adolescente , Adulto , Osso Esponjoso/fisiopatologia , Feminino , Cabeça do Fêmur/diagnóstico por imagem , Colo do Fêmur/diagnóstico por imagem , Subunidades gama da Proteína de Ligação ao GTP/genética , Homeostase , Humanos , Resistência à Insulina , Lipodistrofia Generalizada Congênita/genética , Vértebras Lombares/diagnóstico por imagem , Masculino , Rádio (Anatomia)/diagnóstico por imagem , Adulto Jovem
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