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Improved grip myotonia in a patient with myotonic dystrophy type 1 following electroacupuncture therapy: A CARE-compliant case report.

ABSTRACT: RATIONALE:Myotonic dystrophy type 1 (DM1) is an autosomal-dominant disorder associated with a short life expectancy and various symptoms, including grip myotonia. Even though grip myotonia decreases quality of life, activities of daily living (ADLs), and work performance, very few interventions provide symptomatic relief. PATIENT CONCERNS:In this case report, we present a patient with DM1 and gradually worsening grip myotonia. A 35-year-old woman developed grip myotonia at age 27. She had no underlying diseases or family history of relevant conditions, including DM1. She was unresponsive to medication for several years. DIAGNOSIS:Her symptoms gradually worsened, and she was finally diagnosed with DM1 via genetic, neurologic, and laboratory testing in a tertiary hospital at age 32. She tried several medication therapies; however, she stopped medication at age 34 due a perceived poor response and several adverse events. INTERVENTION:At the age of 35, she underwent 29 sessions (10?minutes per session) of electroacupuncture therapy on TE9 acupuncture point with 120?Hz electrical stimulation over 3 months. OUTCOMES:After 3 months, relaxation time after maximal voluntary isometric contraction decreased from 59 to 2?seconds with treatment. Her Michigan Hand Outcomes Questionnaire score improved (total score, 66.6-75.9; ADL sub-score, 59.7-73.6; function sub-score, 70-90; satisfaction sub-score, 75-91.7). Her Measure Yourself Medical Outcome Profile 2 score also improved from 4.33 to 2. There were no serious adverse events. LESSONS:Electroacupuncture is a potential treatment modality and produced an immediate antimyotonic effect, and cumulative long-term treatment effect, in a patient with DM1 and grip myotonia. Other notable treatment outcomes included improving relaxation time, hand function, ADLs, and overall satisfaction. Electroacupuncture is a potential treatment modality for patients with DM1 and grip myotonia. Further prospective clinical studies are warranted to confirm this hypothesis.

SUBMITTER: Yoon SH

PROVIDER: S-EPMC7489697 | biostudies-literature | 2020 Sep

REPOSITORIES: biostudies-literature

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Improved grip myotonia in a patient with myotonic dystrophy type 1 following electroacupuncture therapy: A CARE-compliant case report.

Yoon Sang-Hoon SH Baek Jang-Hyun JH Leem Jungtae J

Medicine 20200901 37

<h4>Rationale</h4>Myotonic dystrophy type 1 (DM1) is an autosomal-dominant disorder associated with a short life expectancy and various symptoms, including grip myotonia. Even though grip myotonia decreases quality of life, activities of daily living (ADLs), and work performance, very few interventions provide symptomatic relief.<h4>Patient concerns</h4>In this case report, we present a patient with DM1 and gradually worsening grip myotonia. A 35-year-old woman developed grip myotonia at age 27. ...[more]

PMID: 32925721

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Project description:Importance:Patients with myotonic dystrophy (MD) have an increased risk of malignancy including uveal melanoma. This case series further explores the association between these 2 diseases. Objective:To describe a cohort of patients with uveal melanoma associated with MD, including a case of iris melanoma, and MD-associated uveal melanoma in relatives. Design, Setting, and Participants:Retrospective case series at 3 tertiary referral centers (Wills Eye Hospital, Philadelphia, Pennsylvania; Mayo Clinic, Rochester, Minnesota; and Moorfields Eye Hospital, London, England), between January 1, 2000, and August 31, 2017. The study included 6 patients with MD and uveal melanoma. Main Outcomes and Measures:Melanoma response to treatment and development of metastatic disease. Results:There were 6 patients, 4 men and 2 women, with MD and uveal melanoma. The mean patient age at melanoma diagnosis was 47 years (median, 43 years; range, 30-67 years), and the tumor involved the choroid in 5 patients (83%) and iris in 1 patient (17%). The diagnosis of MD was known since young adulthood in 2 patients (33%) and was discovered in adulthood in 4 patients (67%). The main clinical features of MD included muscle weakness (n = 5; 83%), myotonia (n = 4; 67%), polychromatic cataract (n = 4; 67%), complications with general anesthesia (n = 4; 67%), myalgia (n = 3; 50%), cardiac arrhythmia (n = 2; 33%), and frontal baldness (n = 2; 33%). Genetic testing revealed MD type 1 (4 of 4 tested patients), and 2 patients demonstrated positive family history of MD with classic clinical features and preferred no testing. Melanoma treatment included plaque radiotherapy (n?=?4; 67%), photodynamic therapy (n?=?1; 17%), and declined treatment (n?=?1; 17%). At follow-up of 6, 6, 41, 42, and 87 months (5 patients), findings included melanoma regression (4 of 5 tumors), melanoma recurrence (1 of 5 tumors), and no metastatic disease (5 of 5 patients). Conclusions and Relevance:Six adult patients with MD demonstrated uveal melanoma involving the choroid or iris, emphasizing the association between these 2 diseases. Further research seems warranted to explore the pathogenesis of uveal melanoma in MD. These findings support the consideration of ophthalmic examination for uveal melanoma in patients with MD.

Dataset Information

Improved grip myotonia in a patient with myotonic dystrophy type 1 following electroacupuncture therapy: A CARE-compliant case report.

Publications

Improved grip myotonia in a patient with myotonic dystrophy type 1 following electroacupuncture therapy: A CARE-compliant case report.

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