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Letter to the Director
Anesthetic management of neuromyotonia. Description of a case
Tratamiento anestésico de la neuromiotonía. Descripción de un caso
M.C. Bona Gracia
Corresponding author
carmen.bona.gracia@gmail.com

Corresponding author.
, P.E. Calderón Aguirre, C.D. Albendea Calleja, R. Murillo Pina, E. Montón Millán, T. Marín Abad
Anestesiología y Reanimación, Hospital de Alcañiz, Alcañiz, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Neuromyotonia&#44; also known as Isaac&#8217;s syndrome&#44; Isaac-Mertens syndrome&#44; or continuous muscle fibre activity syndrome&#44; is an immune-mediated peripheral motor neuron disorder characterised by continuous muscle activity at rest&#44; due to an alteration in voltage-dependent potassium channels &#40;VGKC&#41;&#46; It causes muscle stiffness more evident in distal than proximal muscles&#44; cramps&#44; myokymia and pseudomyotonia&#44; although it has also been associated with hyperhidrosis&#44; muscle hypertrophy and hyporeflexia&#44; less frequently<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a>&#46; These symptoms may occur during sleep or even under general anaesthesia<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a>&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">It is a rare disease&#44; with an incidence of less than one case per million population&#44; twice as frequent in men as in women&#46; It can appear from childhood to the sixth decade&#44; although it seems to be more frequent between 40 and 60 years of age<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a>&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">It may be hereditary or acquired in origin&#46; The hereditary form is associated with episodic ataxia type 1&#44; with autosomal dominant inheritance&#44; due to mutation of the potassium channel of the KCNA gene1<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a>&#46; Much more common is the acquired form&#44; with antibodies against VGKC channel proteins in peripheral nerves&#44; associated with autoimmune disorders such as myasthenia gravis&#44; but also with infections&#44; drugs&#44; toxins&#44; and in 20&#37; of cases with cancer&#44; mainly thymoma and small cell lung cancer<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a>&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">There is a more severe variant&#44; Morvan syndrome&#44; which also presents with personality and mood changes&#44; hallucinations&#44; memory loss and sleep disorders<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a> as it involves the central nervous system&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Diagnosis should be based on clinical history&#44; physical findings and an electromyogram &#40;EMG&#41; with motor unit discharges in the form of myokymic doublets&#47;triplets and fasciculations&#46; Anti-VGKC-complex antibodies are detected in only 35&#37;&#8211;40&#37; of cases with compatible clinical and EMG findings<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a>&#44; rising to 80&#37; in patients with neuromyotonia secondary to thymoma<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a>&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">This hyperexcitability of peripheral nerves benefits from treatment with antiepileptic drugs alone or in combination with muscle relaxants and analgesics&#46; Cannabinoids reduce muscle spasms and appear to be effective in these patients&#46; In more severe cases&#44; corticosteroids or immunosuppressants can be administered&#44; and in patients with Morvan syndrome&#44; plasmapheresis or intravenous immunoglobulin administration&#46; In the case of secondary neuromyotonia&#44; treatment of the cause is essential<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a>&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">We present the case of a 46-year-old man with symptoms of muscle pain in all four limbs&#44; generalised contractures and manipulative clumsiness of the left hand&#46; Following EMG&#44; a diagnosis of neuromyotonia with negative antibodies was made&#46; An extension study ruled out a triggering oncological aetiology&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">He underwent excision of submandibular adenopathy due to suspected lymph node tuberculosis&#46; He was receiving the following home treatment&#58; tetrahydrocannabinol&#47;cannabidiol nasal spray&#44; rescue oxycodone&#47;naloxone&#44; baclofen and lancosamide&#46; On the day of surgery&#44; midazolam 2&#8239;mg was administered as premedication and monitored with electrocardiography&#44; pulse oximetry and blood pressure&#46; Neuromuscular block was also monitored with TOF and anaesthetic depth with BIS&#46; After preoxygenation with 100&#37; O<span class="elsevierStyleInf">2</span>&#44; fentanyl 50&#8239;g&#44; propofol 170&#8239;mg and rocuronium 30&#8239;mg were administered and he was intubated without complications&#46; Maintenance of anaesthesia with perfusion of propofol at 8&#8239;mg&#47;kg&#47;h for a BIS between 40 and 60 and remifentanil at &#46;2&#8239;g&#47;kg&#47;min&#46; Intraoperatively he remained haemodynamically stable and the procedure was performed without complications within 30&#8239;min&#46; A fixed dose of sugammadex 200&#8239;mg was administered to move from a TOF 30&#37; to a TOF &#62;90&#37; and he was extubated without complications&#46; The postoperative period was uneventful until discharge&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The ideal anaesthetic management for these patients is not described&#59; there is little literature available on this subject due to the rarity of the disease&#46; This hyperexcitability&#44; as mentioned above&#44; remains during sleep&#44; during general anaesthesia and apparently also during locoregional anaesthesia&#46; Despite this&#44; 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&#44; relaxants and anticholinesterase drugs&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">The abnormal nerve discharge occurs at the neuromuscular junction&#44; so during general anaesthesia it may continue to occur unless neuromuscular relaxation with either depolarising or non-depolarising relaxants is used&#46; Kim et al&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> with their regional <span class="elsevierStyleSmallCaps">d</span>-tubocurarine test showed increased sensitivity&#44; with abolition of spontaneous muscle activity and delayed recovery similar to that occurring in patients with myasthenia gravis&#46; Neostigmine reversed the effect of the relaxant&#44; but more slowly&#46; This increased sensitivity could be explained by a down-regulation of acetylcholine receptors&#44; produced by a chronically high concentration of acetylcholine at the level of the neuromuscular plate&#46; 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"><span class="elsevierStyleSup">1</span></a>&#46; Moreover&#44; succinylcholine may precipitate the development of hyperkalaemia in patients with myopathies<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a>&#46; Atracurium could be considered a suitable relaxant because of its short duration and stable pharmacokinetics&#44; although today&#44; the wide availability of sugammadex makes rocuronium the muscle relaxant of choice&#44; drastically reducing the risk of recurarisation&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">As mentioned above&#44; halogenated anaesthetics should be avoided&#44; therefore&#44; total intravenous anaesthesia &#40;TIVA&#41;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> with propofol&#44; which also provides haemodynamic stability and reduces postoperative nausea and vomiting&#44; and short-acting opioids&#44; such as remifentanil&#44; for rapid recovery&#44; orientation and psychomotor and respiratory coordination&#44; seems indicated&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Although general anaesthesia does not predispose to clinical worsening&#44; some cases of myokymia of the laryngeal musculature leading to dyspnoea and hoarseness have been reported&#59; both muscle weakness and involuntary contractions at this level can lead to respiratory problems and increase the risk of bronchoaspiration and bronchospasm&#46; If this also involves the tongue and&#47;or the maxillary musculature it complicates airway management<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a>&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">It should be borne in mind that many of these patients are chronic drug users and may interact with relaxants&#44; altering their effect&#44; which again highlights the need for neuromuscular monitoring&#46; If they take baclofen&#44; we must be careful with opioids because they increase the risk of respiratory depression&#46;</p></span>"
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Original language: English
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