Table 1.

Partial list of couplonopathies

Protein Skeletal muscle Cardiac muscle 
 Gene Disease References Gene Disease References 
DHPR CACNA1S MHS5 Monnier et al., 1997, 2002; Pirone et al., 2010; Toppin et al., 2010  CACNA1C Conduction and other diseases Venetucci et al., 2012  
RyR RYR1 MHS1 Durham et al., 2007; MacLennan and Zvaritch, 2011  RYR2 CPVT1, conventional Durham et al., 2007; Priori and Chen, 2011; Venetucci et al., 2012  
EHS and ER Capacchione and Muldoon, 2009  CPVT1, unconventionale Zhao et al., 2015  
CCD and related CMs Durham et al., 2007; Dowling et al., 2014  
Calsequestrin CASQ1 CAM Rossi et al., 2014  CASQ2 CPVT2 Faggioni and Knollmann, 2012  
Unknowna Kraeva et al., 2013a  
Muscle weakness, MH-like EHSb Paolini et al., 2007; Protasi et al., 2009; Canato et al., 2010; Olojo et al., 2011  
Triadin TRDN Muscle weaknessb Shen et al., 2007; Oddoux et al., 2009  TRDN CPVT5 Roux-Buisson et al., 2012  
Junctin and Junctate ASPH -c Divet et al., 2007  ASPH -b,c Yuan et al., 2007; Hong et al., 2008  
JP-45 JSRP1 Muscle weaknessb,f Gouadon et al., 2006; Yasuda et al., 2013  -d   
Protein Skeletal muscle Cardiac muscle 
 Gene Disease References Gene Disease References 
DHPR CACNA1S MHS5 Monnier et al., 1997, 2002; Pirone et al., 2010; Toppin et al., 2010  CACNA1C Conduction and other diseases Venetucci et al., 2012  
RyR RYR1 MHS1 Durham et al., 2007; MacLennan and Zvaritch, 2011  RYR2 CPVT1, conventional Durham et al., 2007; Priori and Chen, 2011; Venetucci et al., 2012  
EHS and ER Capacchione and Muldoon, 2009  CPVT1, unconventionale Zhao et al., 2015  
CCD and related CMs Durham et al., 2007; Dowling et al., 2014  
Calsequestrin CASQ1 CAM Rossi et al., 2014  CASQ2 CPVT2 Faggioni and Knollmann, 2012  
Unknowna Kraeva et al., 2013a  
Muscle weakness, MH-like EHSb Paolini et al., 2007; Protasi et al., 2009; Canato et al., 2010; Olojo et al., 2011  
Triadin TRDN Muscle weaknessb Shen et al., 2007; Oddoux et al., 2009  TRDN CPVT5 Roux-Buisson et al., 2012  
Junctin and Junctate ASPH -c Divet et al., 2007  ASPH -b,c Yuan et al., 2007; Hong et al., 2008  
JP-45 JSRP1 Muscle weaknessb,f Gouadon et al., 2006; Yasuda et al., 2013  -d   

Corresponding proteins of skeletal and cardiac muscle are listed on the same row. CAM, calsequestrin aggregate myopathy; CMs, congenital myopathies related to CCD (multiminicore disease, congenital myopathies with cores and rods, central nuclear myopathy, and congenital fiber type disproportion); EHS, exertional environmental heat stroke; ER, exertional rhabdomyolysis.

a

A Calsequestrin 1 variant is predicted to reduce SR Ca store but showed no disease phenotype in carriers.

b

Disease phenotype observed in null or knockout animals.

c

Not associated with a disease phenotype in humans. See references for studies on animal models.

d

JP-45 is not present in cardiac muscle (Zorzato, F., personal communication).

e

Unconventional for being accompanied by EADs instead of DADs.

f

JP-45 variants found in the Swiss MH population.

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