Anesthetic management of neuromyotonia. Description of a case

Bona Gracia, M.C.; Calderón Aguirre, P.E.; Albendea Calleja, C.D.; Murillo Pina, R.; Montón Millán, E.; Marín Abad, T.

doi:10.1016/j.redare.2023.02.005

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Description of a case" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "240" "paginaFinal" => "241" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M.C. Bona Gracia, P.E. Calderón Aguirre, C.D. Albendea Calleja, R. Murillo Pina, E. Montón Millán, T. Marín Abad" "autores" => array:6 [ 0 => array:4 [ "nombre" => "M.C." "apellidos" => "Bona Gracia" "email" => array:1 [ 0 => "carmen.bona.gracia@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "P.E." "apellidos" => "Calderón Aguirre" ] 2 => array:2 [ "nombre" => "C.D." "apellidos" => "Albendea Calleja" ] 3 => array:2 [ "nombre" => "R." "apellidos" => "Murillo Pina" ] 4 => array:2 [ "nombre" => "E." "apellidos" => "Montón Millán" ] 5 => array:2 [ "nombre" => "T." "apellidos" => "Marín Abad" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Anestesiología y Reanimación, Hospital de Alcañiz, Alcañiz, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tratamiento anestésico de la neuromiotonía. Descripción de un caso" ] ] "textoCompleto" => "Neuromyotonia, also known as Isaac’s syndrome, Isaac-Mertens syndrome, or continuous muscle fibre activity syndrome, is an immune-mediated peripheral motor neuron disorder characterised by continuous muscle activity at rest, due to an alteration in voltage-dependent potassium channels (VGKC). It causes muscle stiffness more evident in distal than proximal muscles, cramps, myokymia and pseudomyotonia, although it has also been associated with hyperhidrosis, muscle hypertrophy and hyporeflexia, less frequently<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a>. These symptoms may occur during sleep or even under general anaesthesia<a class="elsevierStyleCrossRef" href="#bib0015">3</a>.It is a rare disease, with an incidence of less than one case per million population, twice as frequent in men as in women. It can appear from childhood to the sixth decade, although it seems to be more frequent between 40 and 60 years of age<a class="elsevierStyleCrossRef" href="#bib0010">2</a>.It may be hereditary or acquired in origin. The hereditary form is associated with episodic ataxia type 1, with autosomal dominant inheritance, due to mutation of the potassium channel of the KCNA gene1<a class="elsevierStyleCrossRefs" href="#bib0015">3,4</a>. Much more common is the acquired form, with antibodies against VGKC channel proteins in peripheral nerves, associated with autoimmune disorders such as myasthenia gravis, but also with infections, drugs, toxins, and in 20% of cases with cancer, mainly thymoma and small cell lung cancer<a class="elsevierStyleCrossRefs" href="#bib0010">2,4</a>.There is a more severe variant, Morvan syndrome, which also presents with personality and mood changes, hallucinations, memory loss and sleep disorders<a class="elsevierStyleCrossRefs" href="#bib0010">2,4</a> as it involves the central nervous system.Diagnosis should be based on clinical history, physical findings and an electromyogram (EMG) with motor unit discharges in the form of myokymic doublets/triplets and fasciculations. Anti-VGKC-complex antibodies are detected in only 35%–40% of cases with compatible clinical and EMG findings<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a>, rising to 80% in patients with neuromyotonia secondary to thymoma<a class="elsevierStyleCrossRef" href="#bib0025">5</a>.This hyperexcitability of peripheral nerves benefits from treatment with antiepileptic drugs alone or in combination with muscle relaxants and analgesics. Cannabinoids reduce muscle spasms and appear to be effective in these patients. In more severe cases, corticosteroids or immunosuppressants can be administered, and in patients with Morvan syndrome, plasmapheresis or intravenous immunoglobulin administration. In the case of secondary neuromyotonia, treatment of the cause is essential<a class="elsevierStyleCrossRefs" href="#bib0010">2,5</a>.We present the case of a 46-year-old man with symptoms of muscle pain in all four limbs, generalised contractures and manipulative clumsiness of the left hand. Following EMG, a diagnosis of neuromyotonia with negative antibodies was made. An extension study ruled out a triggering oncological aetiology.He underwent excision of submandibular adenopathy due to suspected lymph node tuberculosis. He was receiving the following home treatment: tetrahydrocannabinol/cannabidiol nasal spray, rescue oxycodone/naloxone, baclofen and lancosamide. On the day of surgery, midazolam 2 mg was administered as premedication and monitored with electrocardiography, pulse oximetry and blood pressure. Neuromuscular block was also monitored with TOF and anaesthetic depth with BIS. After preoxygenation with 100% O2, fentanyl 50 g, propofol 170 mg and rocuronium 30 mg were administered and he was intubated without complications. Maintenance of anaesthesia with perfusion of propofol at 8 mg/kg/h for a BIS between 40 and 60 and remifentanil at .2 g/kg/min. Intraoperatively he remained haemodynamically stable and the procedure was performed without complications within 30 min. A fixed dose of sugammadex 200 mg was administered to move from a TOF 30% to a TOF >90% and he was extubated without complications. The postoperative period was uneventful until discharge.The ideal anaesthetic management for these patients is not described; there is little literature available on this subject due to the rarity of the disease. This hyperexcitability, as mentioned above, remains during sleep, during general anaesthesia and apparently also during locoregional anaesthesia. Despite this, there is no contraindication for its use and it is even recommended for postoperative pain control and to avoid or minimise the use of opioids, relaxants and anticholinesterase drugs.The abnormal nerve discharge occurs at the neuromuscular junction, so during general anaesthesia it may continue to occur unless neuromuscular relaxation with either depolarising or non-depolarising relaxants is used. Kim et al.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> with their regional d-tubocurarine test showed increased sensitivity, with abolition of spontaneous muscle activity and delayed recovery similar to that occurring in patients with myasthenia gravis. Neostigmine reversed the effect of the relaxant, but more slowly. This increased sensitivity could be explained by a down-regulation of acetylcholine receptors, produced by a chronically high concentration of acetylcholine at the level of the neuromuscular plate. It seems reasonable to avoid the use of succinylcholine and halogenated drugs in these patients because of their increased susceptibility to malignant hyperthermia<a class="elsevierStyleCrossRef" href="#bib0005">1</a>. Moreover, succinylcholine may precipitate the development of hyperkalaemia in patients with myopathies<a class="elsevierStyleCrossRef" href="#bib0030">6</a>. Atracurium could be considered a suitable relaxant because of its short duration and stable pharmacokinetics, although today, the wide availability of sugammadex makes rocuronium the muscle relaxant of choice, drastically reducing the risk of recurarisation.As mentioned above, halogenated anaesthetics should be avoided, therefore, total intravenous anaesthesia (TIVA)<a class="elsevierStyleCrossRef" href="#bib0030">6</a> with propofol, which also provides haemodynamic stability and reduces postoperative nausea and vomiting, and short-acting opioids, such as remifentanil, for rapid recovery, orientation and psychomotor and respiratory coordination, seems indicated.Although general anaesthesia does not predispose to clinical worsening, some cases of myokymia of the laryngeal musculature leading to dyspnoea and hoarseness have been reported; both muscle weakness and involuntary contractions at this level can lead to respiratory problems and increase the risk of bronchoaspiration and bronchospasm. If this also involves the tongue and/or the maxillary musculature it complicates airway management<a class="elsevierStyleCrossRef" href="#bib0005">1</a>.It should be borne in mind that many of these patients are chronic drug users and may interact with relaxants, altering their effect, which again highlights the need for neuromuscular monitoring. If they take baclofen, we must be careful with opioids because they increase the risk of respiratory depression." 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Hirose" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1186/s40981-020-00321-y" "Revista" => array:5 [ "tituloSerie" => "JA Clin Rep" "fecha" => "2020" "volumen" => "6" "paginaInicial" => "14" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32062811" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0030" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:1 [ "referenciaCompleta" => "Recomendaciones para la anestesia de pacientes con Neuromiotonía [Accessed 4 December 2021]. 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ISSN: 2341-1929

Revista Española de Anestesiología y Reanimación (REDAR) -the Spanish Journal of Anesthesiology and Resuscitation- is the scientific body of the Spanish Society of Anesthesiology, Resuscitation and Pain Therapy. This is a monthly journal (10 issues) that publishes scientific articles of all areas within its specialty: clinical anesthesia, resuscitation-intensive medicine and critical care, diagnosis and treatment of acute and chronic pain, urgent care and emergencies and studies on basic science and related fields. The journal accepts work in both Spanish and English.

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