Table 2.

Status of approved treatments and experimental therapies for LSDs with selected bibliography

Therapy Target organelle In vitro POC In vivo POC Clinical trials Regulatory approval References 
Enzyme replacement (ERT) Lysosome Brady, 2006b; Neufeld, 2011  
Bone marrow transplantation (BMT) Lysosome N/A Krivit, 2002; Brady, 2006a  
Substrate reduction therapy (SRT) Golgi Platt and Butters, 2004; Platt and Jeyakumar, 2008; Cox, 2010  
Enzyme enhancement therapy (EET) ER/lysosome − In progress − Okumiya et al., 2007; Fan, 2008  
Gene therapy (GT) Nucleus In progress − Gritti, 2011; Tomanin et al., 2012  
Stop codon read-through Nucleus − − − Brooks et al., 2006  
Calcium modulation therapy (CMT) ER − − Lloyd-Evans et al., 2008  
Enhanced exocytosis therapy (ExT) Exosome − − − Strauss et al., 2010; Medina et al., 2011  
Chaperone therapy by HSp70 (CT) Lysosome − − − Kirkegaard et al., 2010  
Proteostasis regulation therapy (PRT) ER − − − Balch et al., 2008; Mu et al., 2008  
Cholesterol removal using cyclodextrin in NPC1 disease Lysosome − − Davidson et al., 2009; Ward et al., 2010; Aqul et al., 2011  

POC, proof of concept.

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