Gadolinium Chloride Rescues Niemann⁻Pick Type C Liver Damage.
Andrés D KleinJuan Esteban OyarzúnCristian CortezSilvana ZanlungoPublished in: International journal of molecular sciences (2018)
Niemann⁻Pick type C (NPC) disease is a rare neurovisceral cholesterol storage disorder that arises from loss of function mutations in the NPC1 or NPC2 genes. Soon after birth, some patients present with an aggressive hepatosplenomegaly and cholestatic signs. Histopathologically, the liver presents with large numbers of foam cells; however, their role in disease pathogenesis has not been explored in depth. Here, we studied the consequences of gadolinium chloride (GdCl₃) treatment, a well-known Kupffer/foam cell inhibitor, at late stages of NPC liver disease and compared it with NPC1 genetic rescue in hepatocytes in vivo. GdCl₃ treatment successfully blocked the endocytic capacity of hepatic Kupffer/foam measured by India ink endocytosis, decreased the levels CD68-A marker of Kupffer cells in the liver-and normalized the transaminase levels in serum of NPC mice to a similar extent to those obtained by genetic Npc1 rescue of liver cells. Gadolinium salts are widely used as magnetic resonance imaging (MRI) contrasts. This study opens the possibility of targeting foam cells with gadolinium or by other means for improving NPC liver disease. Synopsis: Gadolinium chloride can effectively rescue some parameters of liver dysfunction in NPC mice and its potential use in patients should be carefully evaluated.
Keyphrases
- induced apoptosis
- magnetic resonance imaging
- cell cycle arrest
- contrast enhanced
- end stage renal disease
- oxidative stress
- newly diagnosed
- ejection fraction
- genome wide
- chronic kidney disease
- prognostic factors
- type diabetes
- computed tomography
- gene expression
- cell death
- stem cells
- peritoneal dialysis
- cell proliferation
- adipose tissue
- metabolic syndrome
- drug delivery
- skeletal muscle
- copy number
- mesenchymal stem cells
- ionic liquid
- mouse model
- cell therapy
- transcription factor
- diffusion weighted imaging