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Case report
Severe cardiomyopathy secondary to pheochromocytoma: Usefulness of magnesium sulfate. Case report
Cardiomiopatia severa secundaria a feocromocitoma: utilidad del sulfato de magnesio. Reporte de un caso
Carlos Sanabriaa,b,
Corresponding author
carsana@yahoo.com

Corresponding author at: Calle 16 B 124-80 casa 9, Cali, Colombia.
, Marina Vendrellc
a MD mayúscula Anesthesiologist, Centro Médico Imbanaco, Cali, Colombia
b Department of Anesthesiology Universidad del Valle, Cali, Colombia
c MD mayúscula Anesthesiologist, Hospital Clinic, Barcelona, Spain
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hypertrophic or dilated myocardial compromise&#44; pulmonary edema&#44; and arrhythmias&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">2</span></a> Adequate handling of the acute crisis is based on the control of cardiac contractility with stabilization of rhythm&#44; rate&#44; and arterial pressure&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case</span><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 42-year-old female patient&#44; without important pathological antecedents&#44; who was admitted to the coronary unit with symptoms of anxiety&#44; polypnea&#44; sweating&#44; and abdominal pain and distension&#46; Upon admission&#44; she presented a labile arterial pressure between 180&#47;120 and 95&#47;60<span class="elsevierStyleHsp" style=""></span>mmHg&#44; a heart rate of 115<span class="elsevierStyleHsp" style=""></span>bpm&#44; a mitral systolic murmur&#44; and jugular engorgement&#46; The electrocardiogram showed evidence of sinus tachycardia&#44; and the transthoracic echocardiogram showed left ventricular dilation&#44; severe global hypokinesis&#44; a restrictive diastolic pattern with an E&#47;A ration of 3&#46;8<span class="elsevierStyleHsp" style=""></span>cm&#47;s&#44; an ejection fraction of 20&#37;&#44; systolic pressure of the pulmonary artery of 85<span class="elsevierStyleHsp" style=""></span>mmHg&#44; and severe tricuspid and mitral insufficiency &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1 and 2</a>&#41;&#46; Cardiac enzyme levels were normal&#44; as was the coronary arteriography&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig2"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Before the abdominal scanography that reported an image of a right suprarenal tumor with necrosis&#44; pheochromocytoma was suspected and the administration of doxazosin at 4<span class="elsevierStyleHsp" style=""></span>mg&#47;day and bisoprolol at 1&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; The plasma levels of catecholamine derivates &#40;metanephrine at 2063<span class="elsevierStyleHsp" style=""></span>pg&#47;ml &#40;normal<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>90&#41; and normetanephrine at 1291 &#40;normal<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>200&#41;&#41; and the function studies with images confirmed the diagnosis&#46; Despite the treatment&#44; for the first two weeks the instability of the arterial pressure and the same compromise of contractility persisted&#46; Therefore&#44; the alpha-blocker phenoxybenzamine was modified and the beta-blocker was suspended&#46; The echocardiogram in the following week revealed only minimal improvement of the ejection fraction to 25&#37;&#46; It was decided that intravenous magnesium sulfate would be initiated to improve contractility&#44; diastolic function&#44; and to reduce toxic effects of the intracellular calcium in the myocardium with a bolus injection of 40<span class="elsevierStyleHsp" style=""></span>mg&#47;kg and an infusion of 15<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;h&#46; One week later&#44; the day before the surgery&#44; though the ejection fraction continued at 25&#37;&#44; a great improvement was observed in the diastolic function&#44; the pulmonary systolic pressure was at 29<span class="elsevierStyleHsp" style=""></span>mmHg&#44; the E&#47;A ration was 1&#46;2<span class="elsevierStyleHsp" style=""></span>cm&#47;s&#44; and the mitral and tricuspid insufficiency was slight&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">On the day of the surgery&#44; the physicians proceeded to prepare for the laparoscopic resection of the pheochromocytoma&#46; Under sedation&#44; invasive arterial pressure monitoring was performed&#44; finding a mean arterial pressure of 105<span class="elsevierStyleHsp" style=""></span>mmHg and a heart rate of 100<span class="elsevierStyleHsp" style=""></span>bpm&#46; Induction was performed with 0&#46;2<span class="elsevierStyleHsp" style=""></span>mg&#47;kg etomidate&#44; 3<span class="elsevierStyleHsp" style=""></span>mcg&#47;kg fentanyl&#44; 0&#46;6<span class="elsevierStyleHsp" style=""></span>mg&#47;kg rocuronium&#44; and orotracheal intubation without cardiovascular complications&#46; The maintenance of anesthesia was with 3<span class="elsevierStyleHsp" style=""></span>mcg&#47;kg of fentanyl&#44; 10<span class="elsevierStyleHsp" style=""></span>mcg&#47;kg&#47;min rocuronium&#44; and desflurane at 4&#8211;5&#37; MAC&#44; as well as the infusion of magnesium at 15<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;h continued up to the moment of complete tumor resection&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Cardiac function was monitored with a transesophageal echocardiogram and a modified Swan-Ganz pulmonary artery catheter &#40;Edwards CCO&#47;SVO2 746HF8&#41;&#44; which allowed for a continuous estimation of cardiac output&#44; end-diastolic volume &#40;EDV&#41;&#44; and right ejection fraction&#46; The initial profile was&#58; cardiac output at 3&#46;4<span class="elsevierStyleHsp" style=""></span>l&#47;min&#44; EDV of 240<span class="elsevierStyleHsp" style=""></span>ml&#44; ejection fraction of 25&#37;&#44; central venous pressure at 22<span class="elsevierStyleHsp" style=""></span>mmHg&#44; systemic vascular resistance at 2850<span class="elsevierStyleHsp" style=""></span>dyn&#46;s&#47;cm<span class="elsevierStyleSup">5</span>&#44; pulmonary vascular resistance of 340<span class="elsevierStyleHsp" style=""></span>dyn&#46;s&#47;cm<span class="elsevierStyleSup">5</span>&#46; As such&#44; dobutamine was initiated at 5<span class="elsevierStyleHsp" style=""></span>mcg&#47;kg&#47;min&#46; The invasive monitoring permitted us to progressively determine the improvement in contractility until a final ejection fraction of 42&#37; was reached with a reduction of EDV to 160<span class="elsevierStyleHsp" style=""></span>ml and we could perform a titrated increase in blood volume while monitoring the central venous pressure&#44; which went from values of 22 to 6<span class="elsevierStyleHsp" style=""></span>mmHg&#46; Two moments of hypertensive crisis occurred at minutes 30 and 50 of the surgery &#40;mean arterial pressure of 122 and 160<span class="elsevierStyleHsp" style=""></span>mmHg&#44; respectively&#41; and were controlled with phentolamine in bolus injections of 2<span class="elsevierStyleHsp" style=""></span>mg each&#46; The surgical result was satisfactory with a surgical time of 120<span class="elsevierStyleHsp" style=""></span>min and bleeding of 350<span class="elsevierStyleHsp" style=""></span>cc&#46; Extubation was performed without cardiovascular or neuromuscular problems with adequate postsurgical progress and inotropic support that could be withdrawn after 24<span class="elsevierStyleHsp" style=""></span>h&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">To summarize&#44; this is the case of a female patient with a sub-acute presentation of severe heart failure&#44; lability in blood pressure values&#44; and severe abdominal pain with negative studies for coronary pathologies&#46; The abdominal scanography and elevated levels of catecholamine metabolites clarified the diagnosis that guided the slow improvement initiated with alpha-blockers and the introduction of magnesium sulfate&#46; The cardiovascular response to this drug was essential&#58; it improved cardiac contractility and arterial pressure thereby obtaining optimal physiological conditions that permitted the tumor resection with adequate results &#40;<a class="elsevierStyleCrossRefs" href="#fig0015">Figs&#46; 3 and 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">This patient with a pheochromocytoma tumor started with a crisis of severe dilated cardiomyopathy induced by catecholamines&#44; with a clinical presentation of left heart failure&#44; and abdominal symptoms secondary to acute tumor necrosis&#46; This is an infrequent presentation that is difficult to diagnosis&#46; As a result&#44; it has high morbimortality due to a late initiation of care&#46; The diagnostic bases were the absence of a history of cardiovascular disease with normal cardiac enzymes and coronary arteriography&#46; Cardiomyopathy in pheochromocytoma is produced by excessive levels of catecholamines&#44; with an overload of intracellular calcium as the main injury&#44; an ischemia-reperfusion type lesion with free radicals&#44; deterioration of the myocardial fiber&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">4</span></a> microvascular alteration&#44; and vasospasm&#46; Finally&#44; an increase in myocardial oxygen demand presents with a decrease in its contribution through coronary vasoconstriction&#46; The patient can present clinical signs of hypertrophic or dilated heart failure&#44; cardiogenic or non-cardiogenic pulmonary edema&#44; fatal arrhythmia and sudden death&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">This pathophysiology of the cells and adrenergic receptors has suggested magnesium sulfate as a possible therapy for the management of acute crises and surgery&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">6</span></a> Extensive literature exists telling of its efficacy in the management of hypertension and arrhythmias throughout the perioperative period to achieve an appropriate optimization of the patient&#46; In some cases&#44; it has been useful in situations where labetalol or sodium nitroprusside have not been beneficial&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">7</span></a> The optimization of this patient begins with alpha-adrenergic blockers &#40;doxazosin and phenoxybenzamine&#41;&#44; but faced with little improvement in myocardial contractility and instability of arterial pressure&#44; magnesium sulfate is introduced starting in the acute crisis and during the surgical process up until the complete removal of the flow from the pheochromocytoma&#44; ensuring a maximum level of magnesemia of 4&#46;5<span class="elsevierStyleHsp" style=""></span>mmol&#47;l&#46; This drug has been reported clinically in this context in case series in adults&#44;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">8</span></a> pediatric patients&#44;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">9</span></a> and pregnant patients&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">10</span></a> Magnesium is an ion that participates in essential enzymatic processes related to energy synthesis and metabolism&#46; It acts as a modulator and stabilizer of Na&#47;K currents in the plasmatic membrane by antagonizing calcium at the intracellular and vascular smooth muscle levels&#46; It also reduces the release of catecholamines from the adrenal medulla and in nerve endings and directly blocks their receptors&#46; As a consequence&#44; it blocks the systemic hemodynamic repercussions of catecholamines&#46;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">11</span></a> In addition&#44; it preserves myocardial cells during the ischemia-reperfusion process and is an anti-arrhythmic in the context of high concentrations of catecholamines&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">12</span></a> The clinical result is an improvement in myocardial&#44; systolic&#44; and diastolic function&#44; the stabilization of the heart rate&#44; progressive vasodilation of the vascular bed &#40;mainly arterial and minimal venous&#41; of target and peripheral organs&#46; In perioperative care&#44; it also decreases the adrenergic response to all stimuli such as orotracheal intubation and the surgical stimulus&#44; thereby achieving better perioperative hemodynamic control&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Echocardiographic monitoring during the crisis allowed us to give evidence of the response to the magnesium with sustained improvement of the restrictive pattern of the diastolic dysfunction &#40;change in E&#47;A ration from 3&#46;8 to 1&#46;2<span class="elsevierStyleHsp" style=""></span>cm&#47;s&#59; increase in ejection fraction from 20 to 25&#37;&#59; and reduction in systolic pressure of the pulmonary artery from 80 to 29<span class="elsevierStyleHsp" style=""></span>mmHg&#41;&#44; which&#44; in association with the dobutamine&#44; facilitated intraoperative hemodynamic management&#46; Intraoperatively&#44; the best monitor is transesophageal echography to establish overall cardiac compromise&#44; contractility&#44; and blood volume&#44; using the pulmonary artery catheter only in cases of severe cardiac compromise&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Recently&#44; cases of Takotsubo cardiomyopathy have been reported&#46; This is a syndrome characterized by transitory left ventricular dysfunction with apical ballooning&#44; and basal hyperkinesis simulating symptoms of myocardial infarction without significant coronary disease&#46; In cases of pheochromocytoma and Takotsubo cardiomyopathy&#44; the most frequent pattern is inverted with basal and midventricular akinesis and a hyperkinetic apex&#46; This syndrome is associated with emotional or physical stress augmented by catecholamines&#44; damage to cardiomyocytes&#44; and stunned heart&#44; which suggests a similarity in the pathophysiology of the two conditions&#8212;adrenergic overload&#8212;with Takotsubo syndrome having a more favorable prognosis than that of pheochromocytoma&#46; There are probably other neurohumoral agents secreted by the tumor chromaffin cells&#44; such as neuropeptide Y&#44; that can act synergistically with the catecholamines and worsen the myocardial injury and the patient&#39;s progress&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">13</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Magnesium sulfate has a good safety profile with easily managed toxicity and reversal&#46; It provides hemodynamic stability and facilitates cardiovascular control during the cardiomyopathy crisis and tumor removal&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">6&#44;13</span></a> representing a first-line option for reducing morbimortality in the management of pheochromocytoma&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Ethical disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Protection of human and animal subjects</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Confidentiality of data</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Right to privacy and informed consent</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; The corresponding author is in possession of this document&#46;</p></span></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">None</p></span></span>"
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            0 => "Cardiomyopathy&#44; hypertrophic"
            1 => "Cardiomyopathy&#44; dilated"
            2 => "Pheochromocytoma"
            3 => "Magnesium sulfate"
            4 => "Adrenal glands"
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            0 => "Cardiomiopat&#237;a hipertr&#243;fica"
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            2 => "Feocromocitoma"
            3 => "Sulfato de magnesio"
            4 => "Gl&#225;ndula suprarrenal"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">The following report on the perioperative anesthetic management of severe cardiomyopathy and resection of pheochromocytoma tumors offers a clinical and pharmaceutical experience with a good outcome for a high-risk pathology with little available world literature&#46; The female patient accesses emergency services in distress with tachycardia&#44; labile blood pressure&#44; dyspnea&#44; and severe abdominal pain&#46; Clinical studies reveal heart failure&#44; an adrenal mass&#44; and derivates of high levels of catecholamines in the blood&#44; which leads to the diagnosis of severe cardiomyopathy induced by pheochromocytoma&#46; The medical management for the acute crisis is performed with therapy in the intensive care unit&#44; antihypertensives and magnesium sulfate&#46; Once stabilized&#44; a laparoscopic tumor resection followed&#46; Her postoperative progress was adequate with a progressive resolution of symptoms&#46; Cardiomyopathy secondary to pheochromocytoma is a pathology with high morbimortality and low frequency and is produced by the action of great quantities of catecholamines released subacutely due to hemorrhagic tumor necrosis or manipulation of the pheochromocytoma&#46; It requires strict care in its acute crises and during surgery for its definitive resection&#46; This report shows our experience with the usefulness of magnesium sulfate as a contributory drug in the control of this pathology throughout the perioperative period due to its mechanism of action and pharmacodynamics&#46; Its easy availability in hospitals&#44; the good clinical results it produces&#44; and its scientific backing are important factors that make it a pharmacological option for pheochromocytoma&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El siguiente reporte de manejo anest&#233;sico perioperatorio de cardiomiopat&#237;a severa y resecci&#243;n de tumor de feocromocitoma&#44; ofrece una experiencia cl&#237;nica y farmacol&#243;gica con buen resultado&#44; de una patolog&#237;a de alto riesgo con poca literatura mundial&#46; La paciente ingresa al servicio de urgencias con angustia&#44; taquicardia&#44; tensi&#243;n arterial l&#225;bil&#44; disnea y dolor abdominal severos&#46; Sus estudios cl&#237;nicos revelan insuficiencia cardiaca&#44; masa suprarenal y derivados de catecolaminas elevados en sangre&#44; que hacen diagn&#243;stico de cardiomiopat&#237;a severa inducida por Feocromocitoma&#59; se realiza el manejo m&#233;dico de la crisis aguda con terapia en unidad de cuidado intensivo&#44; antihipertensivos y sulfato de magnesio y una vez estabilizada se lleva a resecci&#243;n tumoral laparoscopica&#46; Su evoluci&#243;n postoperatoria fue adecuada&#44; con resoluci&#243;n progresiva de los s&#237;ntomas&#46; La cardiomiopat&#237;a secundaria a feocromocitoma es una patolog&#237;a de alta morbimortalidad e inusual frecuencia&#44; producida por la acci&#243;n de grandes cantidades de catecolaminas liberadas de modo subagudo por necrosis tumoral hemorr&#225;gica o manipulaci&#243;n de feocromocitoma&#44; que requiere manejo estricto en su crisis aguda y en la cirug&#237;a de resecci&#243;n definitiva&#46; Este reporte muestra la experiencia de la utilidad del sulfato de magnesio&#44; como f&#225;rmaco coadyuvante en el control de esta patolog&#237;a durante todo el periodo perioperatorio&#44; por su mecanismo de acci&#243;n y farmacodinamia&#46; Su f&#225;cil accesibilidad hospitalaria&#44; buen resultado cl&#237;nico y soporte cient&#237;fico son factores importantes para ser considerado una opci&#243;n farmacol&#243;gica en Feocromocitoma&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Sanabria C&#44; Vendrell M&#46; Cardiomiopatia severa secundaria a feocromocitoma&#58; utilidad del sulfato de magnesio&#46; Reporte de un caso&#46; Rev Colomb Anestesiol&#46; 2016&#59;44&#58;58&#8211;62&#46;</p>"
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                      "autores" => array:1 [
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                          "etal" => false
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                            1 => "G&#46; Eisenhofer"
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                            3 => "K&#46; Pacak"
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                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
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                ]
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                      "titulo" => "Catecholamine-induced cardiomyopathy"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
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                  "contribucion" => array:1 [
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                      "titulo" => "Preoperative management of the pheochromocytoma patient"
                      "autores" => array:1 [
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos