Dramatic improvement of refractory Isaacs’ syndrome after treatment with dronabinol

Meyniel, Claire; Ollivier, Yolaine; Hamidou, M.; Péreón, Yann; Derkinderen, Pascal

doi:10.1016/j.clineuro.2010.11.006

Cited by 5 publications

(6 citation statements)

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“…The mechanism is presumed to be decreased release of acetylcholine by activation of potassium channels and inhibition of calcium channels. There may be an immunomodulatory role as well . The treatment of malignancies, if identified, may have little positive effect on the clinical severity of the neuromyotonia, although case reports have also noted improvement with chemotherapy or surgical resection …”

Section: Managementmentioning

confidence: 99%

Isaacs syndrome: A review

2015

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Isaacs syndrome is a peripheral nerve hyperexcitability (PNH) syndrome that presents as continuous motor activity. Clinical findings include cramps, fasciculations, and myokymia. Electrodiagnosis plays a key role in diagnosis by demonstrating after-discharges on nerve conduction studies, and fasciculation potentials, myokymic discharges, neuromyotonic discharges, and other types of abnormal spontaneous activity on needle examination. Etiopathogenesis involves the interaction of genetic, autoimmune, and paraneoplastic factors, which requires a broad-ranging evaluation for underlying causes. Initial treatment is symptomatic, but immune therapy is often needed and can be effective. The purpose of this review is to describe the syndrome and its pathogenesis, assist the reader in evaluating patients with suspected Isaacs syndrome and distinguishing it from other disorders of PNH, and suggest an approach to management, including both symptomatic and immunomodulating therapy.

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Section: Managementmentioning

confidence: 99%

Isaacs syndrome: A review

2015

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“…Autoantibodies in IS reduce the number of VGPCs those control nerve excitability, and neurotransmitter releasing imbalance consequently occurs. Disrupted neural voltage potential and related imbalance of neurotransmitter release are held to account for spontaneous muscle fibre activities, spasticity, and pain [3]. Sodium channel blocking agents such as carbamazepine are assumed as good treatment options to compensate the reduction of functional potassium channels [1].…”

Section: Discussionmentioning

confidence: 99%

“…Cannabinoids are also regarded as novel therapeutic agents in IS. Clinical improvement was shown with the cannabinoid dronabinol in IS [3]. CB1 receptors are G-protein coupled receptors located at the presynaptic terminal and widely spread in both central and peripheral nervous system.…”

Section: Discussionmentioning

confidence: 99%

“…Carbamazepine, phenytoin, and gabapentin are pointed out as appropriate treatment options to control the symptoms of neuromyotonia [2]. Without decent evidencing, psychotropics like dronabinol, antidepressants, and benzodiazepines are also offered in the treatment [3,4]. Nevertheless, precise therapeutic mechanisms of these drugs in the treatment have not been decently argued.…”

Section: Introductionmentioning

confidence: 99%

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Polysubstance use disorder as a probable self-medication in Isaacs’ syndrome

Balcıoğlu

Kırlıoğlu

Aydin

2017

Psychiatry and Clinical Psychopharmacology

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Isaacs' syndrome (IS) is an autoimmunological hyperexcitability syndrome of the peripheral motor nerves, manifesting with progressive muscle stiffness, involuntary continuous muscle twitching, muscle pain and cramping, sweating, and decreased reflexes. We report a 31-yearold man who was suffering from muscle twitches and stiffness in lower extremities and previously diagnosed with IS in his age of 16 through electrophysiological studies and the shown presence of autoantibodies against voltage-gated potassium channels. Without any adherence to the prescribed treatment, he had been using synthetic cannabinoids and opioids for 10 years. He admitted lessened complaints by using them. Current literature offered cannabinoid receptor agonists not only for symptomatic relief in IS, but also potential modulator effects on both potassium channels and autoimmunity. Opioids were also recognized with their analgesic and antispastic effects in the management of IS. This report aimed to discuss possible medicinal effects and therapeutic mechanisms of aforementioned psychoactive molecules on the symptomatology of IS.

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“…Gabapentin and acetazolamide were also reported effective in case reports [68][69][70]. Other drugs less commonly used include antiepileptics such as valproate, lamotrigine, and lacosamide, as well as dronabinol, a synthetic cannabinoid [71]. Cramp-fasciculation syndrome was reported to not necessarily need immunomodulation as the symptoms often resolve with symptomatic treatment, or even spontaneously [72].…”

Section: Diagnosis and Treatmentmentioning

confidence: 99%

Autoimmune neuromyotonia

Comperat

Pégat

Honnorat

et al. 2022

Current Opinion in Neurology

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Purpose of reviewAutoimmune neuromyotonia encompasses a group of rare immune-mediated neurological disorders frequently associated with anti-contactin-associated protein-like 2 (CASPR2) antibodies and featuring clinical and electrical signs of peripheral nerve hyperexcitability (PNH). We aim to summarize the current knowledge on immune-mediated neuromyotonia, focusing on clinical presentations, pathophysiology, and management. Recent findingsNeuromyotonia is a major feature of several autoimmune neurological syndromes characterized by PNH with or without central neurological system involvement. Experimental and clinical evidence suggest that anti-CASPR2 antibodies are directly pathogenic in autoimmune neuromyotonia patients. SummaryNeuromyotonia, a form of PNH, is a major feature in several syndromes associated with anti-CASPR2 antibodies, including cramp-fasciculation syndrome, Isaacs syndrome, Morvan syndrome, and autoimmune limbic encephalitis. Diagnosis relies on the identification of motor, sensory, and autonomic signs of PNH along with other neurological symptoms, anti-CASPR2 antibody-positivity, and of characteristic electroneuromyographic abnormalities. Paraneoplastic associations with thymoma are possible, especially in Morvan syndrome. Patients usually respond to immune-active treatments, including steroids, intravenous immunoglobulins, plasma exchanges, and rituximab.

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Dramatic improvement of refractory Isaacs’ syndrome after treatment with dronabinol

Cited by 5 publications

References 4 publications

Isaacs syndrome: A review

Isaacs syndrome: A review

Polysubstance use disorder as a probable self-medication in Isaacs’ syndrome

Autoimmune neuromyotonia

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