Objective:

We describe a patient who developed tardive dystonia after sulpiride treatment for over two decades. Few cases have been reported[1-3], thus we offer an additional case of a patient who developed lid-opening apraxia (LOA), OMD, and laterocollis.

Background:

Sulpiride, a benzamide and D2 antagonist, is not approved in North America or Australia, but is often used to treat psychosis, mild depression, anxiety, and vertigo in other countries[4]. Tardive dyskinesia following the use of DA receptor antagonists such as sulpiride is prevalent, but providers should also be aware of less common sequelae, such as oromandibular dystonia (OMD), Parkinsonism, and blepharospasm.

Method:

SS, a 68-year-old Japanese male, was seen via telemedicine in 2022 for second opinion. At 48, he was prescribed sulpiride 50 mg and mecobalamin 500 mg QD to temporarily relieve headaches. He subsequently developed insomnia and anxiety. Seven years later, headaches began at night so sulpiride was increased to 50 mg BID. Two years after starting the increased medication regimen, headaches recurred. Nine years later, SS began experiencing eye fatigue. Brain MRI was reportedly normal. A year later, eye fatigue progressed to eye strain, with eye opening. He was diagnosed with ptosis, but did not improve after corrective surgery. SS experienced tongue and jaw pain and tightness. Medications were weaned in 2021. Despite a lack of visible involuntary movements of the orbicularis oculi, he was diagnosed with blepharospasm. SS was also diagnosed with Meige Syndrome. Injection of botulinum toxin to the orbicularis oculi resulted in no improvement.

Results:

In consultation, SS reported jaw, tongue, and medial orbicularis oculi pain, heaviness in the neck and bilateral upper arms, and temporal-occipital headaches. He denied family history of movement disorders or exposure to toxins. On examination, the patient showed no signs of parkinsonism. He displayed LOA, hypertrophy of b/l SCM muscles (right greater than left) with right laterocollis. 

The diagnosis we suggest is segmental tardive dystonia involving LOA, OMD, and laterocollis due to long-term sulpiride use.

Conclusion:

This case study shows an uncommon presentation of tardive dystonia without retrocollis or blepharospasm. We caution prescribers to observe patients who take sulpiride for similar uncommon presentations of tardive dystonia.

References: 1. Imai, N., & Ikawa, M. (2011). Efficacy of Aripiprazole in Sulpiride-induced Tardive Oromandibular Dystonia. Internal Medicine, 50(6), 635–637.
2. Tsuji, S., Kikkawa, S., Horiguchi, J., Yamashita, H., Kagaya, A., Morinobu, S., & Yamawaki, S. (2002). Meige Syndrome with Apraxia of Lid Opening after the Discontinuation of
Sulpiride Treatment. Pharmacopsychiatry, 35(4), 155–156. doi:10.1055/s-2002-33198
3. Miller, L. G., & Jankovic, J. (1990). Sulpiride-induced tardive dystonia. Movement Disorders, 5(1), 83–84. doi:10.1002/mds.8700501191.
4. Caley, C F, and S S Weber. “Sulpiride: an antipsychotic with selective dopaminergic antagonist properties.” The Annals of pharmacotherapy vol. 29,2 (1995): 152-60.
doi:10.1177/106002809502900210

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MDS Abstracts - https://www.mdsabstracts.org/abstract/tardive-dystonia-manifesting-in-lid-opening-apraxia-oromandibular-dystonia-and-laterocollis-after-long-term-sulpiride-use/