Bilateral hand dystonia following high-velocity thrust manipulation: a case report

Abstract

Thrust manipulation is one of the most commonly used techniques for managing musculoskeletal pain in clinical practice. This involves the application of a high-velocity, low-amplitude force directed to the joints with the intent of achieving joint cavitation. This current case report describes a female in her mid-20s who presented with excessive bilateral and involuntary hand muscle contractions after bilateral thrust manipulation. Dystonia appeared both at rest and during voluntary movements but was aggravated by actions such as finger flexion or spreading and disappeared during sleep. A diagnosis of complex regional pain syndrome (CRPS) type I combined with dystonia was made. Prednisolone administered between 2 and 5 weeks after symptom onset significantly reduced CRPS symptoms, but intramuscular botulinum toxin injection 5 weeks after symptom onset was ineffective at controlling her symptoms. Seven weeks after symptom onset, the patient was administered 2 mg trihexyphenidyl oral twice a day, 2.5 mg diazepam oral twice a day and 5 mg baclofen oral three times a day for 1 month and this significantly reduced dystonia, but complete resolution was not achieved. Clinicians should be aware that dystonia is a rare complication of thrust manipulation.

Introduction

Dystonia is defined as the involuntary contraction or spasm of antagonist muscles and overflow of extraneous muscles, resulting in twisting movements, impaired motor control and sustained abnormal postures.^1–3 Individuals with dystonia have difficulty controlling or predicting the movement of their bodies.^1–3 Although the cause of dystonia remains unknown in most cases, trauma precedes the onset of dystonia in some cases. ⁴ For example, central post-traumatic dystonia can occur with a delayed onset after traumatic brain injury. ⁴ Additionally, it has been increasingly recognized that peripheral trauma (i.e. outside the central nervous system) can also result in dystonia, with reports indicating that even minor peripheral trauma can cause dystonia. ⁴

Manipulation is a technique used by physical therapists, chiropractors and other healthcare practitioners to modulate the pain associated with various musculoskeletal disorders, increase joint range of motion, reduce soft tissue swelling or inflammation, and induce relaxation. ⁵ The thrust manipulation technique, which involves the application of a high-velocity, low-amplitude force directed to joints with the intent of achieving joint cavitation or an audible pop, is one of the most frequently applied manipulation methods. ⁶ Joint cavitation and the associated audible pop occur due to the rapid formation and collapse of gas bubbles within the synovial fluid of the joint. Occasionally, this force caused by thrust manipulation can cause traumatic injuries to musculoskeletal structures. ⁶ The occurrence of dystonia related to peripheral trauma caused by thrust manipulation has not been previously reported. This current case report describes a patient who developed dystonia as a complication of thrust manipulation therapy.

Case report

In February 2024, a female in her mid-20s presented to the Department of Rehabilitation Medicine, College of Medicine, Yeungnam University, Daegu, South Korea for the management of excessive and involuntary muscle contractions of the bilateral hands. The patient had no history of neurological, psychological, metabolic or renal disorders. She was not taking any medications. Her family and social history were unremarkable. Her primary complaints were myofascial pain syndrome in the bilateral upper trapezius and the muscles of both upper extremities. Two weeks prior to this current presentation, she had received manipulation therapy from a physical therapist at a local pain clinic to manage myofascial pain syndrome. During this treatment, high-velocity thrust manipulation of both hands was performed. Four hours after the manipulation therapy, the patient experienced immediate excessive and involuntary contractions of the hand muscles; with difficulty extending the fingers bilaterally. Additionally, severe pain presented in both hands (numeric rating scale [NRS]: 8, where 0 represents no pain and 10 represents the worst pain).

A neurological examination was normal except for dystonia of the bilateral hands, which presented as flexion of all fingers in the bilateral hands (see supplementary materials, Video 1). The dystonia appeared both at rest and during voluntary movements but was aggravated by actions such as finger flexion or spreading and disappeared during sleep. Hand and cervical radiographs captured 5 days after symptom onset revealed no specific anomalies. Magnetic resonance imaging (MRI) of the brain, cervical spine and hands, conducted 2 weeks after the onset of symptoms, revealed no abnormalities. Furthermore, no abnormal findings were observed on nerve conduction and electromyography tests performed 2 weeks after the onset of symptoms. The results of routine investigations, including haematological and biochemical screening, were all normal. The patient was therefore diagnosed with hand dystonia.

The patient further experienced continuous bilateral pain, which was aggravated by touch and the passive and active ranges of motion of the hands. In addition, she had bilateral allodynia and hand oedema. Based on the complex regional pain syndrome (CRPS) criteria proposed by the Budapest consensus group, the patient was diagnosed with type I CRPS combined with hand dystonia. ⁷ Two weeks after symptom onset, the patient was administered 60 mg prednisolone oral once a day for 2 weeks to control the symptoms of CRPS type I, tapering the dose over 1 week. ⁸ The prednisolone administration reduced hand pain to NRS 3 and the oedema on both hands was resolved. However, prednisolone had no effect on the dystonia. Five weeks after the onset of symptoms, to control hand dystonia, botulinum toxin was injected intramuscularly into the bilateral flexor digitorum profundus and superficialis (75 IU into each muscle, total 300 IU); ⁹ however, no symptom resolution was observed. Seven weeks after symptom onset, the patient was administered 2 mg trihexyphenidyl oral twice a day, 2.5 mg diazepam oral twice a day and 5 mg baclofen oral three times a day for 1 month. ¹⁰ At the 1-month follow-up after initiating oral medication, dystonia was still present but was significantly reduced. No adverse effects from the oral medications were observed and the patient’s adherence to oral medication was satisfactory.

This study was approved by the local institutional review board of Yeungnam University Hospital, Yeungnam University, Daegu, South Korea (no. 2024-07-019). Written informed consent to treatment and for publication of this case report was obtained from the patient. In addition, the patient provided a signed statement permitting the publication of the video for educational purposes. The reporting of this case report conforms to CARE guidelines. ¹¹

Discussion

This current case report presents a patient who developed bilateral hand dystonia combined with CRPS type I after undergoing bilateral thrust manipulations. This current patient’s hand dystonia seemed to have been caused by minor trauma during the bilateral trust manipulation procedure.

Peripheral post-traumatic dystonia is clinically diagnosed in patients with a history of precipitating trauma and consistent dystonic movement. ⁴ Brain and cervical spine MRI are necessary to exclude pathologies that can cause dystonia. A systematic review of 713 cases of movement disorders that developed after peripheral trauma demonstrated that 513 (72%) patients were diagnosed with dystonia. ¹² In addition, 214/513 (42%) patients had dystonia combined with CRPS. ¹² In patients with CRPS, dystonia is the most common movement disorder. ¹³ Mostly, dystonia developed focally at the injured site, while in 139/173 (19%) patients, dystonia spread to other body parts. ¹² However, spontaneous recovery was rare. ¹²

Although the mechanism of dystonia development after peripheral trauma has not been clearly demonstrated, peripheral trauma may alter sensory input and induce cortical and subcortical reorganization, which could account for the occurrence of dystonia. ¹⁴ A previous study conducted in rats demonstrated that peripheral tissue inflammation following repetitive motion injury induced the reorganization of the sensory and motor cortices. ¹⁵ Cortical reorganization could account for the development of dystonia. In addition, it has been proposed that peripheral nerve compression or injury may be associated with the occurrence of dystonia, which is similar to the mechanism of hemifacial spasms caused by vascular compression of the facial nerve. ¹² In addition, substance P, a neuropeptide released during neuroinflammation, has been shown to amplify nociceptive withdrawal reflexes. ¹³ This factor may have played a role in the flexion posture observed in patients with dystonia after peripheral trauma. ¹³ Furthermore, a previous report reported that the combination of genetic predisposition and peripheral trauma leads to dystonia. ¹⁵ In our opinion, this current case had undergone minor peripheral trauma during high-velocity thrust manipulation of both hands, which could have modified the sensory input via the peripheral and central nervous systems leading to a reorganization of the patient’s brain areas related to dystonia. Also, neuropeptides following peripheral trauma and genetic factors may have contributed to the development of hand dystonia in the current case.

In conclusion, this current report describes a patient who developed dystonia combined with bilateral CRPS of the hands after receiving high-velocity thrust manipulation. Clinicians should be aware of this potential complication. To the best of our knowledge, this is the first reported case of dystonia as a complication of manipulation therapy. Future research is warranted to elucidate the mechanisms underlying dystonia following high-velocity thrust manipulation.

Supplemental Material

sj-pdf-1-imr-10.1177_03000605241289284 - Supplemental material for Bilateral hand dystonia following high-velocity thrust manipulation: a case report

Supplemental material, sj-pdf-1-imr-10.1177_03000605241289284 for Bilateral hand dystonia following high-velocity thrust manipulation: a case report by Min Cheol Chang, Mathieu Boudier-Revéret, Jean-Paul Brutus and Seoyon Yang in Journal of International Medical Research

Supplemental Material

sj-mp4-2-imr-10.1177_03000605241289284 - Supplemental material for Bilateral hand dystonia following high-velocity thrust manipulation: a case report

Supplemental material, sj-mp4-2-imr-10.1177_03000605241289284 for Bilateral hand dystonia following high-velocity thrust manipulation: a case report by Min Cheol Chang, Mathieu Boudier-Revéret, Jean-Paul Brutus and Seoyon Yang in Journal of International Medical Research