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Case report
Anaesthetic management of thyroid storm in a patient with Friederich’s ataxia. A case report
Manejo anestésico de una tormenta tiroidea en un paciente afecto de ataxia de Friederich. A propósito de un caso
M. Sneyers Closa
Corresponding author
msc9319@gmail.com

Corresponding author.
, A. Pérez Requena, S. Sánchez García, J. Sistac Ballarín
Servicio Anestesiología, Reanimación y Terapéutica del Dolor del Hospital Universitari Arnau de Vilanova, Lleida, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Friedreich&#39;s ataxia &#40;FA&#41; is an autosomal recessive disease that affects the corticospinal and pyramidal tracts&#46; It has a prevalence of 2&#8722;4&#58;100&#44;000 population&#46; FA is a neurodegenerative disorder characterized by the appearance of ataxia&#44; dysarthria&#44; oculomotor dysfunction&#44; kyphoscoliosis&#44; muscle weakness&#44; and spasticity&#46; It can also involve the heart&#44; with hypertrophic cardiomyopathy&#44; sometime with conduction abnormalities&#44; being the most frequent alteration&#46; Half of all patients with FA may present supraventricular and ventricular arrhythmias&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Amiodarone is a class III antiarrhythmic drug with a half-life of up to 100 days that is indicated for the management of supraventricular and ventricular arrhythmias refractory to other treatments&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">In the thyroid&#44; amiodarone inhibits conversion of T4 to T3&#46; Given its high iodine content&#44; amiodarone has a toxic effect on thyroid epithelial cells&#44; and in 3&#37; of cases this can lead to hyperthyroidism or thyrotoxicosis &#40;usually prevalent in iodine-deficient regions&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Amiodarone-induced thyrotoxicosis is classified as type I or type 2&#58; Type 1 occurs in patients with pre-existing thyroid disease and causes hyperthyroidism secondary to increased production and release of thyroid hormone&#59; type II occurs in patients with no thyroid dysfunction&#46; Amiodarone-induced thyroiditis causes follicular disruption and leads to the release of thyroid hormones&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In these circumstances&#44; amiodarone should be discontinued and treatment with antithyroid drugs and corticosteroids started&#46; Discontinuing amiodarone can occasionally worsen thyrotoxicity and cause serious arrhythmias&#46; Total thyroidectomy should be considered in patients that are refractory to antithyroid drugs&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">We report the case of a patient with FA undergoing total thyroidectomy due to failure of medical management of hyperthyroidism secondary to amiodarone-induced thyrotoxicosis&#44; who presented intraoperative thyroid storm&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0030" class="elsevierStylePara elsevierViewall">Our patient was a 26-year-old man&#44; allergic to cephalosporins&#44; with a history of FA with structural cardiac involvement &#40;non-obstructive hypertrophic cardiomyopathy&#41; diagnosed in 2013&#46; Since 2018&#44; he had been admitted several times due to decompensation secondary to atrial fibrillation&#47;atrial flutter &#40;AFib&#47;AFL&#41; with rapid ventricular response and impaired ventricular function despite treatment with amiodarone and several electrical cardioversion &#40;ECV&#41; procedures&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">He developed significant hyperthyroidism while taking amiodarone&#44; so the drug was discontinued and methimazole &#40;15 mg&#47;8 h&#41; and prednisone &#40;60 mg&#47;24 h&#41; were started&#46; One month later&#44; he consulted for a new episode of AFL with 2&#58;1 conduction that persisted despite 2 ECVs and more than 36 h of beta-blocker treatment &#40;propranolol 0&#46;1 mg&#47;kg&#41; and digoxin &#40;1 mg&#47;24 h&#41; to slow the heart rate&#46; The symptoms were consistent with a thyroid storm&#44; so it was decided to start antithyroid treatment and plasmapheresis&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Symptoms improved during his hospital stay&#44; and the treatment administered normalised his heart rate to 80&#8211;100 bpm&#46; Despite this improvement&#44; it was impossible to normalise his thyroid hormones levels &#40;TSH&#42; &#60; 0&#46;01&#44; T4l&#42; 46&#46;1&#59; T3l&#42; 6&#46;07&#41; despite high-dose antithyroid drugs &#40;propylthiouracil 150 mg&#47;8 h&#44; Lugol&#39;s solution 5&#37; 5 drops&#47;6 h&#41; and corticosteroids &#40;prednisone 60 mg&#47;24 h&#41;&#44; so he was scheduled for surgery&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Preoperative blood tests were only remarkable for abnormal thyroid function &#40;T4 48&#46;7&#44; T3 6&#46;91&#41;&#46; ECG&#58; AFib at 103 bpm&#46; The echocardiogram showed concentric hypertrophic cardiomyopathy with moderate-to-severe systolic dysfunction due to global hypocontractility&#46; Severe left atrial dilatation&#46; Moderate central functional MR&#46; Mild PHT&#46; EF 30&#37;&#8211;32&#37;&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">We decided to perform total intravenous anaesthesia &#40;TIVA&#41;&#46; The patient was pre-medicated with 40 mg IV pantoprazole&#44; 240 mg IV hydrocortisone and 1 mg IV midazolam&#46; Prior to induction&#44; awake left radial artery cannulation was performed for in-depth haemodynamic monitoring&#46; The patient was preoxygenated for 5 min with 100&#37; FiO2&#44; and IV induction was performed with 16 mg etomidate&#44; 100 mcg fentanyl&#44; and 30 mg rocuronium&#46; A Glidescope was used for intubation&#44; which was uneventful&#46; Following this&#44; an oesophageal temperature sensor and a nasogastric &#40;NG&#41; tube were placed&#46; TIVA was maintained with propofol &#40;6&#8722;8 mg&#47;kg&#47;h&#41; and remifentanil &#40;0&#46;05&#8722;0&#46;15 mcg&#47;kg&#47;min&#41; monitored with bispectral index &#40;BIS&#41; and TOF &#40;train-of-four&#41;&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">All the patient&#39;s vital signs remained stable and similar to baseline for the first 100 min of the intervention&#44; except for fluctuations in BIS&#58; BIS 35&#8211;55&#59; temperature 36&#46;3&#8211;36&#46;7&#176;&#59; HR 90&#8211;100 bpm&#59; SAP 100&#8211;120&#44; DAP 55&#8211;75&#59; SpO2 98&#37;&#8211;100&#37;&#46; Approximately 100 min after anaesthesia induction we observed a gradual increase in body temperature &#40;reaching a peak of 38&#46;7 &#176;C&#41; accompanied by an increase in heart rate &#40;AFib with rapid ventricular response of about 140 bpm&#41; and moderate hypotension &#40;minimum blood pressure&#58; 74&#47;30&#41;&#46; These changes could not be attributed to anaesthesia or any specific surgical manipulation&#46; Lung auscultation revealed bibasal crackles&#44; absent prior to surgery&#46; After calculating the Burch-Wartofsky Point Scale score &#40;65&#41;&#44; we concluded that this was a new episode of thyroid storm&#44; and started support measures&#58; temperature control &#40;cold compresses&#44; 1 g IV paracetamol&#41; and heart rate control &#40;IV bolus of 30 mg esmolol &#43; continuous infusion 0&#46;3 mg&#47;kg&#47;min&#41;&#46; We also administered IV boluses of phenylephrine as required &#40;total dose&#58; 350 mcg&#41;&#46; Antithyroid drugs &#40;propylthiouracil 250 mg and thiamazole 30 mg&#41; were administered via the NG tube&#46; These measures allowed us to keep the patient stable enough to complete the surgery&#46; At the end of the intervention&#44; we decided not to extubate the patient&#44; and he was transferred to the postoperative critical care unit &#40;PCCU&#41; for monitoring&#46; In the PCCU&#44; his postoperative evolution was good&#44; and he was extubated 4 h after the end of the surgery&#46; Continuous infusion of esmolol was gradually decreased until it could be withdrawn prior to extubation&#46; Antithyroid treatment with propylthiouracil 250 mg&#47;6 h and thiamazole 30 mg&#47;6 h&#44; via the NG tube was maintained&#46; No new events were observed during his stay in the PCCU&#44; and he was discharged to the ward 24 h after surgery&#46; The NG tube was removed on the ward&#44; but the oral antithyroid treatment initiated in the PCCU was maintained &#40;propylthiouracil 250 mg&#47;6 h PO and thiamazole 30 mg&#47;6 h PO&#41;&#46; No new episodes of instability were observed during his stay on the ward&#46; The patient was discharged 5 days later&#44; and scheduled for a follow-up visit in the Endocrinology and Cardiology outpatient departments 1 month later&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0060" class="elsevierStylePara elsevierViewall">In this patient&#44; the challenges and potential complications &#40;thyroid storm&#41; involved in the perioperative management of a hyperthyroid patient were further complicated by the patient&#8217;s high baseline morbidity&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Ideally&#44; prior to surgery&#44; patients should be in a euthyroid state in order to reduce the probability of thyroid storm&#44; and with it&#44; the risk of perioperative complications&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> Thyroid storm is an endocrinological emergency associated with high morbidity and mortality&#46; Early diagnosis and treatment are vital to improve survival&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> but diagnosis is often difficult&#44; so various scales have been developed to help in this task&#46; The best known is the Burch-Wartofsky Point Scale &#40;BWPS&#41; &#40;see <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> that not only takes into account the dysfunction of several organs &#40;thermoregulatory dysfunction&#44; tachycardia&#44; altered mental state&#44; presence of congestive heart failure&#44; gastrointestinal and&#47;or hepatic dysfunction&#41;&#44; but also rates the severity of the decompensation and the possible precipitating factors&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0070" class="elsevierStylePara elsevierViewall">With regard to perioperative management&#44; there is no clear guidance in the literature on the choice between general anaesthesia or locoregional anaesthesia&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In our patient&#44; we opted for general anaesthesia in the belief that it would guarantee greater haemodynamic stability and greater control of haemodynamic factors&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#8211;10</span></a> Therefore&#44; in addition to basic monitoring&#44; we also performed invasive radial artery blood pressure monitoring&#44; BIS and TOFF monitoring&#44; and monitored temperature with an oesophageal probe&#46; This in-depth monitoring allowed us to rapidly diagnose the early signs of thyroid storm and immediately start treatment&#44; which&#44; according to the literature&#44; are essential for survival&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">According to existing guidelines&#44; management of thyroid storm is based on 5 strategies&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6&#44;10</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">The first is to treat the symptoms&#44; including hyperthermia&#44; with paracetamol and not NSAIDs&#44; since these are thought to increase T3 levels by interfering with the binding of T3 to serum carrier proteins&#46; We administered 1 g IV paracetamol and applied physical measures &#40;cold compresses&#41; to control temperature&#46; The second strategy involves treating the cardiovascular&#44; neurological and&#47;or hepatic alterations&#46; Our patient only presented cardiovascular alterations&#44; in the form of atrial fibrillation with rapid ventricular response &#40;HR 140 bpm&#41;&#44; leading to haemodynamic instability &#40;BP 74&#47;30&#41;&#46; Guidelines recommend the use of beta-blockers&#44; preferably B1-selective&#44; short-acting drugs &#40;esmolol&#44; as in our case&#44; or landoliol&#41; for heart rate control&#46; For blood pressure control&#44; non-catecholamine sympathomimetic agonists such as phenylephrine are recommended&#44; since hyperthyroidism patients are thought to have increased sensitivity to catecholamines&#46; For this reason&#44; as soon as symptoms appeared we administered several boluses of phenylephrine &#40;a total of 350 mcg IV&#41; combined with continuous infusion of esmolol &#40;0&#46;3 mg&#47;kg&#47;min&#41;&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">The third strategy is to treat thyrotoxicosis&#44; which is our case was achieved with propylthiouracil &#40;250 mg via NG tube&#41; and thiamazole &#40;30 mg via NG tube&#41;&#46; Both these antithyroid drugs reduce thyroid hormone synthesis&#44; but propylthiouracil should be the first choice because it acts on both the thyroid gland and other peripheral organs&#44; and is therefore believed to reduce T3 levels more rapidly&#46; The fourth strategy consists of avoiding triggering stimuli&#44; while the fifth is definitive treatment &#40;thyroidectomy&#41;&#44; which is beyond the remit of the anaesthesiologist&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">We believe that early identification of the clinical picture and rapid implementation of the 5 treatment strategies are the key to increasing the patient&#8217;s likelihood of surviving a thyroid storm&#46;</p><p id="par0095" class="elsevierStylePara elsevierViewall">This is why both close monitoring and skills in the use of scales such as the BWPS will allow the anaesthesiologist to rapidly diagnose the early signs of a thyroid storm&#44; even in the presence of diffuse symptoms&#46;</p><p id="par0100" class="elsevierStylePara elsevierViewall">Regarding treatment&#44; temperature control and haemodynamic management were effective management strategies&#46; Although antithyroid drugs were not immediately effective in resolving the symptoms&#44; they were useful in achieving a good postoperative evolution&#46; However&#44; the most effective measure was completion of the intervention&#44; as this ensured that patient was no longer exposed to possible triggering stimuli&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interests</span><p id="par0105" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A 26-year-old patient with Friederich&#39;s ataxia with hypertrophic obstructive cardiomyopathy undergoing total thyroidectomy due to persistent amiodarone-induced thyrotoxicosis &#40;despite high doses of antithyroid drugs and corticosteroids&#41;&#44; presented an intraoperative episode suggestive of thyroid storm&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Thyroid storm is an endocrine emergency that is associated with high morbidity and mortality&#46; Early diagnosis and treatment&#44; which is of vital importance to improve survival&#44; includes symptomatic treatment&#44; treatment of cardiovascular&#44; neurological&#44; and&#47;or hepatic manifestations and thyrotoxicosis&#44; measures to suppress or avoid triggering stimuli&#44; and definitive treatment&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Paciente de 26 a&#241;os afecto de ataxia de Friederich con una miocardiopat&#237;a hipertr&#243;fica obstructiva sometido a una tiroidectom&#237;a total por tirotoxicosis secundaria a amiodarona persistente &#40;a pesar de elevadas dosis de antitiroideos y corticoides&#41; que intraoperatoriamente present&#243; un episodio sugestivo de tormenta tiroidea&#46;</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">La tormenta tiroidea es una emergencia endocrinol&#243;gica que asocia una elevada morbimortalidad&#46; Para mejorar la supervivencia es de vital importancia un diagn&#243;stico y tratamiento precoz que incluya&#58; un tratamiento sintom&#225;tico&#59; el tratamiento de las manifestaciones cardiovasculares&#44; neurol&#243;gicas y&#47;o hep&#225;ticas y de la tirotoxicosis&#59; y as&#237; como suprimir o evitar est&#237;mulos desencadenantes y practicar un tratamiento definitivo&#46;</p></span>"
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                  \t\t\t\t"><span class="elsevierStyleBold">Thermoregulatory dysfunction &#40;Temperature &#176;C&#41;</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">- 37&#46;2&#8722;37&#46;7</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t"><span class="elsevierStyleBold">Cardiovascular</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t">- 120&#8722;129 bpm&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">Atrial fibrillation</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">- Severe &#40;seizures&#44; coma&#41;</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t"><span class="elsevierStyleBold">Precipitant history</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">- Absent</span>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span><span class="elsevierStyleItalic">- Present</span>&nbsp;\t\t\t\t\t\t\n
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Original language: English
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