Dystonia is a neurological disorder characterized by abnormal involuntary muscle contractions, classified as hypokinetic and hyperkinetic muscular movements, resulting in abnormal postures and twisting and repetitive movements of body parts [1,2]. It is caused by the co-contraction of agonist and antagonist muscles [3] leading to decreased spatial and temporal somatosensory discrimination and abnormal sensory motor integration [1,4], which significantly affect the quality of life to a greater extent [5]. Dystonia is classified into primary and secondary dystonia and further into focal, multifocal, segmental, hemi-dystonia, and generalized dystonia. Primary dystonia is caused by a functional disturbance within the basal ganglia system without any neurostructural pathologies and underlying causes. Conversely, secondary dystonia has underlying causes such as head injury or the use of drugs [1,3].

In 2021, a study revealed that tremor disorders were the third most common tertiary movement disorders in clinics in India [6]. Studies have reported that adult-onset focal forms of primary dystonia were more common than primary generalized dystonia, with a mean annual prevalence of 87.6 per 100,000 people and high incidence in women. The most prevalent types are cervical dystonia and blepharospasm [7]. In the field of acupuncture, tremors, tics, muscle spasms, rigidities, and impairments in limb flexion and extension are caused by the internal wind produced by liver imbalance affecting the tendons and ligaments of the body [8]. To the best of our search knowledge, few studies have explored dystonia with its pathophysiology and allopathic management. However, no studies have reported the role of acupuncture for dystonia; thus, this is the first study emphasizing the effect of acupuncture on idiopathic generalized dystonia.

A 26-year-old male patient with a moderate build and dystonic gait visited our outpatient department with complaints of frequent involuntary muscle contractions on the right upper limb, emaciation, and twisting of the right upper, arm, wrist, metacarpal, and interphalangeal joints. For the past 5 years, he also had difficulty communicating verbally and moving the right upper and lower limbs and altered facial expressions. No demonstrable psychiatric and cognitive symptoms of heterogenicity were found. He had good social cognition without evidence of auditory and visual impairments. Symptom severity was assessed using the global dystonia severity rating scale (GDS) and was rated 5.5 as moderate dystonia [9].

The symptoms were provoked by exercises, physical and mental stress, hypersensitivity to temperature swifts such as intake of chilled and diuretic foods, prolonged sun exposure which caused gross profuse sweating, and gradual weight loss. The medical and family history was not significant for any comorbidities such as endocrine, metabolic, and neurological diseases. He was taking anticholinergic medication benzhexol (trihexyphenidyl hypochloride) to alleviate tremors and muscular spasms and promote muscle relaxation and vitamin B complex from the onset of the condition at the time of admission to our hospital.

He consented to treatment modalities and the use of medical data in research and its publication.

On observation, the patient had myoclonus of the right hand, hypertrophy of forearm muscles, increased muscle rigidity, and twitching on the face, neck, and right extremities, and loss of dexterity without any sensory impairment. He also had subtle facial spasms, chewing difficulty, and slurred speech. The patient had no history of trauma, head injury, acute illness, or exposure to toxins before the onset of dystonia symptoms. Results of investigations such as complete blood count, renal function tests, liver function tests, and urine analysis were all within the normal limits, and results of ultrasonography of the abdomen and pelvis were normal. Magnetic resonance imaging of the brain did not detect apparent abnormalities. After ruling out all likely causes, the patient was diagnosed with idiopathic generalized dystonia. Based on Traditional Chinese Medicine (TCM) pulse diagnosis, left-hand middle position in a deeper aspect was wiry, and the left-hand proximal positions of both superficial and deep aspects were weak, denoting liver yin and kidney yin deficiency.

The patient received needling at acupuncture points selected based on TCM’s physiology and pathophysiology of the affected organs. The details of the acupuncture points [8] used in this study and needling details such as number (unilateral or bilateral), depth, and direction of needling in each point are provided in Table 1. The patient was informed about the procedure and sensations of needle insertion and the response sought. Needling was performed using 0.25 × 13 mm size stainless steel sterile filiform needles (Tx-Pinpai; Fangbang Industrial Park). The needles were left for 20 minutes without manual or electrical stimulation. The patient received a total of three courses of acupuncture (one course consists of 20 minutes of acupuncture/day daily for 15 days followed by no intervention for 15 days) for 3 months. No other concurrent treatment was given along with acupuncture. Needling was performed by a Doctor of Medicine scholar in acupuncture and energy medicine who is institutionally qualified and a registered medical practitioner with > 3 years of clinical experience.

Table 1 . Details of the acupuncture points used in this study.

Acupuncture point	Location	Needling			Rationale
		Direction	Unilateral/bilateral	Depth (cun)
KI-3 (Taixi)	In the depression between the medial malleolus and Achilles tendon	Perpendicular	Unilateral: right side	0.3	Earth point stabilizes kidney qi and nourishes kidney yin
GB-34 (Yanglingquan)	In the depression anteroinferior to the head of the fibula	Perpendicular	Unilateral: right side	1	He-sea point regulates the coordination of movements and harmonizes liver qi
LR-3 (Taichong)	On foot dorsum, between the 1st and 2nd metatarsal bones, proximal to the metatarsophalangeal joint	Slightly oblique	Unilateral: right side	0.5	Strengthens liver yin, promotes the smooth flow of liver qi, and extinguishes the interior wind
GB-20 (Fengchi)	At the lower border of the occipital bone, in the depression between the origin of the sternocleidomastoid and trapezius muscle	Anteriorly toward the nose	Unilateral: right side	0.5	Extinguishes liver wind, subdues liver yang, opens the channel, nourishes the marrow, and clears the brain
GV-20 (Baihui)	At the junction of line connecting the apices of the ears and midline	Transversely posterior	Single needle on the meridian	0.5	Calms the mind, benefits the brain, and nourishes the sea of the marrow
GV-14 (Dazhui)	On the midline, below the spinous process of C7 vertebra	Perpendicular	Single needle on the meridian	0.5	Meeting point of all yang meridians, clears the mind, and eliminates internal wind

KI, kidney; GB, gall bladder, LR, liver; GV, governor vessel..

The intensity of myoclonus reduced on the very first day of treatment, as well as the frequency of tremors. The patient felt energetic throughout the day, and tremors vanished from day 5 after needling. The needling session continued for another 10 days. The pulse was assessed on day 15 on completion of the first course of intervention, and improvement in liver qi was found. Moreover, the patient was meticulously followed for the next 3 months with 15 days of intervals and 15 days of needling alternatively (completing three courses of the intervention) and did not report the null incidence of tremors then and thereafter. At 1 month follow-up, myoclonus did not recur (after the fourth course of intervention) even without acupuncture with a 3.8 rating in the GDS, and details are mentioned in Table 2 and Fig. 1. The patient was satisfied with the positive prognosis such as the reduction in tremors and improvement in the strength and stability of the limbs without any additional medical drug support.

Figure 1. Timeline of myoclonus improvement. GDS, global dystonia severity rating scale.

Table 2 . Comparison of baseline and post-intervention assessments using the global dystonia severity rating scale (GDS).

Body area	Assessment before intervention	Assessment after the completion of the follow-up
1 Eyes and upper face	2	1
2 Lower face	4	2
3 Jaw and tongue	5	4
4 Larynx	4	4
5 Neck	8	6
6 Shoulder and proximal arm (right)	9	4
Shoulder and proximal arm (left)	5	2
7 Distal arm and hand including elbow (right)	9	6
Distal arm and hand including elbow (left)	5	2
8 Pelvis and proximal leg (right)	6	6
Pelvis and proximal leg (left)	3	3
9 Distal leg and foot including knee (right)	9	8
Distal leg and foot including knee (left)	6	5
10 Trunk	2	0
Global score	77	53
Rating (global score/ 14)	5.5	3.8

Dystonia can be treated using modalities such as physical and supportive therapy, pharmacologic therapy, chemodenervation with botulinum toxin, and peripheral/central surgery [10]. Occupational and physical therapies aid in limiting mounting contractures and mobilizing frozen joints [11]. Dopamine modulators, anticholinergic drugs, baclofen, and muscle relaxants are used for long-term treatment. A study showed that deep-brain stimulation (DBS) is more effective for generalized dystonia [12]. Bilateral DBS of the globus pallidus was found to reduce the severity of dystonia and functional disability and improve the quality of life [13].

According to TCM diagnosis, the patient had a liver energy imbalance. In this case, the selection of acupuncture points, detailed case history, and analysis of the pulse of the patient reduced the myoclonus and stabilized the limbs. Thus, correcting the liver qi flow and eliminating the liver wind help reduce the symptoms of the patient. As the liver controls the sinews (tendons, cartilages, and ligaments of the limbs), its imbalance causes muscles and tendon disabilities leading to contractions, spasms, and muscular weakness. Poor diet, emotional stress, excessive physical exercise, hemorrhage, and fluid and electrolyte loss can lead to an imbalance in the liver. In this case, poor diet, emotional disturbance of long-term sadness, and grief depleted the liver yin.

This liver and kidney yin deficiency resulted in (internal wind) liver wind causing myoclonus. A Chinese saying connotes that sudden rigidity is caused by wind. In the later stage, liver yin deficiency develops empty heat that affects the mental, emotional, and behavioral aspects of a person, eventually affecting the quality of life. Pathophysiologically, liver yin deficiency affects other vital organs leading to kidney and heart yin deficiencies and liver yang rising. In such severe cases, it can also lead to convulsion, unconsciousness, and hemiplegia [14]. Based on our search knowledge, no studies have focused on TCM-based acupuncture intervention for dystonia. Thus, this is the first paper emphasizing the benefits of acupuncture for the active management of myoclonus in generalized dystonia. However, a study revealed that acupuncture is feasible and safe and provides subjective symptomatic benefits as an adjunct treatment for chronic cervical dystonia [15]. In this study, the selected acupuncture points aided in reducing myoclonus purely based on pulse analysis. However, the same acupuncture points cannot be implemented for all dystonic conditions because each individual has different body constitutions and thereby different organ-based elemental energy imbalances. To prove this finding, more studies with large sample sizes are needed to determine the effectiveness of the same set of acupuncture points on individuals with dystonia.

In conclusion, this case study presents that acupuncture interventions for three months is effective in reducing the incidence of myoclonus in a patient with generalized dystonia. However, further clinical studies are recommended to confirm the results of this study.

Conceptualization: JA, VK, MRM, NP, AM. Data curation: JA, VK, NP, AM. Formal analysis: MRA. Investigation: JA, VK, NP. Methodology: JA, VK, MRM, NP, AM. Project administration: VK, NP. Resources: AM. Software: AM. Supervision: VK. Validation: MRM, NP. Visualization: VK, NP. Writing – original draft: MRM. Writing – review & editing: JA, VK, NP, AM.

The authors have no conflicts of interest to declare.

None.

Written informed consent was obtained from the participant. As this is a single case study, the institutional ethics committee exempted it for review.