| Antimuscarinics |
Clinical trial (randomized, double-blind, placebo) |
31 adults (Dystonia) |
Trihexyphenidyl |
Improved dystonia |
(Burke et al., 1986) |
|
Clinical trial (open label) |
23 (children) 52 (adults) dystonia |
Trihexyphenidyl ethopropazine |
Improved dystonia |
(Fahn, 1983) |
|
Animal study |
Dyt1 knockin mice |
Trihexyphenidyl |
Normalized DA release |
(Downs et al., 2019) |
|
Animal study |
Dyt1 knockin mice |
Trihexyphenidyl VU0255035 (M1 antagonist) |
Normalized corticostriatal plasticity |
(Maltese et al., 2014) |
|
Animal study |
dtsz hamster |
Trihexyphenidyl tropicamide |
Improved dystonia |
(Hamann et al., 2017) |
| Nicotinic receptors |
Case reports |
2 adults (hemidystonia) |
Nicotine (lozenges and transdermal patch) |
Improved dystonia |
(Lees, 1984) (Vaughan et al., 1997) |
|
Animal study |
Dyt1 knockin mice |
AZD1446 (non-desensitizing nChR agonist) |
No effect on DA release/overflow |
(Zimmerman et al., 2017) |
| mGluR5 receptors |
Animal study |
Dyt1 knockin mice |
ADX48621 (mGluR5 NAM) |
Normalized corticostriatal plasticity |
(Sciamanna et al., 2014) |
| A2A adenosine receptors |
Animal study |
Dyt1 knockin mice |
KW6002 (A2A receptor antagonist) |
Normalized Corticostriatal plasticity |
(Napolitano et al., 2010) |
|
Animal study |
Dyt11 knockin mice |
SCH 58261 (A2A receptor antagonist) |
Normalized Corticostriatal plasticity |
(Maltese et al., 2017) |
|
Animal study |
dtsz hamster |
CPA (N(6)-cyclopentyladenosine (A1A receptor agonist) CGS 21680 (A2A receptor agonist) |
Improved dystonia |
(Richter and Hamann, 2001) |
|
Animal study |
dtsz hamster |
Non-selective adenosine receptor antagonists A1A receptor antagonists |
Worsened dystonia |
(Richter and Hamann, 2001) |
| Endocannabinoid receptors (CB1) |
Animal study |
dtsz hamster |
WIN 55,212–2 (CB1 agonist) |
Improved dystonia |
(Richter and Loscher, 1994) |
|
Animal study |
dtsz hamster |
WIN 55,212–2 (CB1 agonist) |
Improved dystonia |
(Richter and Loscher, 2002) |
|
Clinical trial (randomized, double-blind, Placebo controlled) |
15 (primary dystonia) |
Nabilone (CB1/CB2) agonist |
No improvement |
(Fox et al., 2002) |
|
Clinical trial (randomized, placebo controlled) |
9 (cervical dystonia) |
Dronabinol (CB1/CB2) agonist |
No improvement |
(Zadikoff et al., 2011) |
|
Clinical trial (open-label) |
5 (undefined dystonia) |
Cannabidiol (CB1/CB2) partial agonist |
Improved dystonia |
(Consroe et al., 1986) |
| NMDA receptors |
Animal study |
dtsz hamster |
NVP-AAM077 (NR2A antagonist) |
Improved dystonia |
(Avchalumov et al., 2014) |
|
Animal study |
dtsz hamster |
Ro 25–6981 (NR2B antagonist) |
Worsened dystonia |
(Richter, 2003a) |
| SV2A modulators |
Animal study |
dtsz hamster |
Piracetam Levetiracetam |
Improved dystonia |
(Loscher and Richter, 2000) |
|
Animal study |
dtsz hamster |
Brivaracetam Seletracetam |
Improved dystonia |
(Hamann et al., 2008) |
|
Clinical trial |
7 (cranial and oromandibular dystonia) |
Levetiracetam |
No improvement |
(Park et al., 2017) |
|
Clinical trial |
10 (generalized/Segmental dystonia) |
Levetiracetam |
No improvement |
(Hering et al., 2007) |
|
Case report |
1 (generalized dystonia) |
Levetiracetam |
Significant improvement |
(Sullivan et al., 2005) |
|
Case report |
1 (Meige’s syndrome) |
Levetiracetam |
Significant improvement |
(Yardimci et al., 2006) |
| EIF2α signaling |
Animal study |
Dyt1 knockin mice |
Salubrinal (eIF2α Dephosphorylation inhibitor) |
Rescued ER function and abnormal torsinA localization |
(Rittiner et al., 2016) |
| BDNF signaling |
Animal study |
Dyt1 knockin mice |
ANA-12 (TrkB inhibitor) |
Normalized Corticostriatal plasticity |
(Maltese et al., 2018) |
