Objective: To describe the results of bilateral globus pallidus internus (GPi) deep brain stimulation (DBS) in a patient with early-adulthood-onset sporadic focal subacute progressive action-induced dystonia of the abdominal muscles.

Background: Intramuscular injection of botulinum toxin is the first-line treatment for patients with focal and segmental dystonia. In some cases, when treatment failure occurs, DBS can be performed as a rescue therapy. Abdominal dystonia is a rare form of focal dystonia, with heterogeneous presentations and no defined underlying pathophysiology. Although botulinum toxin injection and pallidotomy have been reported as successful treatments, to our knowledge, this is the first case report of DBS improving focal abdominal dystonia.

Method: We describe a 42-year-old female patient with focal abdominal dystonia. After completing a swimming marathon training session, the patient reported an involuntary contraction of the abdomen, which caused a flexed posture and worsened over the following weeks. The contractions were induced by lower limb movements, such as walking or swimming, and resolved with rest. The patient had to stop swimming and, later, all physical activity. With disease progression, even walking short distances became a challenge. Imaging and laboratory investigations found no specific aetiology. Levodopa trial showed no benefit. Following electromyography assessment [figure1], patient received ultrasound-guided botulinum toxin injections [table1]. After the first injection, patient reported a moderate improvement, however, this benefit diminished progressively with subsequent injections. Three years after dystonia onset, in view of the patient’s poor quality of life, bilateral GPi DBS was performed [figure2]. Clinical improvement was assessed before DBS and 16 months after using Burke-Fahn-Marsden Dystonia Rating Scale-Movement (BFM-M) and Clinical Global Impression – Improvement (CGI-I); quality of life was assessed using SF-36 questionnaire.

Results: BFM-M scores improved from 6 to 2. SF-36 showed a significant improvement in quality of life [table2]. Patient reported “very much improved” at CGI-I. After surgery, patient did not need additional botulinum toxin injections. Side effects were self-limited and exclusively seen during monopolar review.

Conclusion: GPi DBS could be considered as a rescue treatment for patients with abdominal dystonia. Larger trials are necessary to confirm this hypothesis.

Figure 1: Electromyography recording

Table 1: Botulinum toxin injection schemes

Figure 2: DBS leads reconstruction

Table 2: SF-36 sub scores before and after DBS

References: 1. Fahmi A, Asadullah, Aji YK, Aprianto DR, Subianto H, Turchan A. Successful Relief of Abdominal Dystonia After Sequential GPi Pallidotomy with 2-Year Follow-Up. World Neurosurgery. 2020 Dec;144:68–70. doi:10.1016/j.wneu.2020.08.152
2. Rodrigues FB, Duarte GS, Prescott D, Ferreira J, Costa J. Deep brain stimulation for dystonia. Cochrane Movement Disorders Group, editor. Cochrane Database of Systematic Reviews. 2019 Jan 10;2020(10). doi:10.1002/14651858.CD012405.pub2

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MDS Abstracts - https://www.mdsabstracts.org/abstract/deep-brain-stimulation-for-focal-abdominal-dystonia/