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Case report
Haemodynamic management using non-invasive cardiac output monitoring for urgent craniotomy in fragile X syndrome: Case report
Manejo hemodinámico mediante monitor no invasivo de gasto cardiaco para craneotomía urgente en el síndrome X frágil: reporte de caso
Rosana Guerrero-Domíngueza,
Corresponding author
rosanabixi7@hotmail.com

Corresponding author at: Avda Ramón Carande, N° 11, 4°E, 41013 Sevilla, Spain.
, Daniel López-Herrera-Rodrígueza, Francisco Javier Beato-Lópezb, Ignacio Jiméneza
a Specialist Physician in Anaesthesiology and Resuscitation, Virgen del Rocío University Hospital, Seville, Spain
b Intern Resident Physician in Anaesthesiology and Resuscitation, Virgen del Rocío University Hospital, Sevilla, Spain
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considering that this approach only requires placement of a finger device that provides all the relevant information pertaining to the patient&#39;s haemodynamic status&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 38 year-old male patient with FXS&#44; severe mental retardation&#44; and refractory epileptic encephalopathy who suffered head injury following a tonic&#8211;clonic seizure&#44; with acute left frontoparietal subdural haematoma requiring emergent craniotomy&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Once monitoring with arterial blood pressure &#40;ABP&#41;&#44; electrocardiography &#40;EKG&#41;&#44; pulse oximetry&#44; bispectral index &#40;BIS&#41; and non-invasive cardiac output &#40;Nexfin<span class="elsevierStyleSup">&#174;</span> &#8211; BMEYE&#44; Amsterdam&#44; The Netherlands&#41; was established&#44; cefazoline 2<span class="elsevierStyleHsp" style=""></span>g and midazolam 5<span class="elsevierStyleHsp" style=""></span>mg were given IV through a peripheral venous line because the patient was uncooperative&#46; Propofol 120<span class="elsevierStyleHsp" style=""></span>mg&#44; remifentanil 0&#46;1<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;min and 50<span class="elsevierStyleHsp" style=""></span>mg of rocuronium were used for anaesthesia induction&#44; with subsequent successful endotracheal intubation with the help of Glidescope<span class="elsevierStyleSup">&#174;</span> videolaryngoscopy&#46; Mechanical ventilation was started and right internal jugular venous access was established under ultrasound guidance&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Propofol and remifentanil were used for anaesthesia maintenance&#46; Mannitol 25<span class="elsevierStyleHsp" style=""></span>g and furosemide 20<span class="elsevierStyleHsp" style=""></span>mg were required for lowering intracranial pressure &#40;ICP&#41; &#40;from 22<span class="elsevierStyleHsp" style=""></span>mmHg initially down to 4<span class="elsevierStyleHsp" style=""></span>mmHg after haematoma removal&#41;&#46; ICP was monitored using a subdural intracranial pressure sensor&#46; Goal directed therapy &#40;GDT&#41; was implemented during the intraoperative period based on cardiac output values obtained after the initial administration of 1000<span class="elsevierStyleHsp" style=""></span>mL&#44; increased to 4000<span class="elsevierStyleHsp" style=""></span>mL in response to signs of hypovolemia&#46; An infusion of 0&#46;1<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;min of noradrenaline was required in order to normalize extremely low initial values of systemic vascular resistance and to maintain mean arterial pressure at around 90<span class="elsevierStyleHsp" style=""></span>mmHg&#44; promoting adequate cerebral perfusion &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Haemodynamic stability was maintained&#44; vasoactive amine perfusion was removed and the patient was transferred to the intensive care unit with adequate sedation and analgesia&#44; and under mechanical ventilation&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">Described by Martin and Bell in 1943&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> FXS has typical physical and behavioural characteristics&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">1</span></a> Prevalence in males &#40;1&#58;3&#46;600&#41; is higher than in females &#40;1&#58;8&#46;000&#41;&#44; it is being associated with the X chromosome&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">1</span></a> It is caused by an abnormal expansion of the cytosine&#8211;guanine&#8211;guanine triplet &#40;CGG&#41; in the <span class="elsevierStyleItalic">FMR1</span> gene &#40;<span class="elsevierStyleItalic">Fragile X Mental Retardation 1 gene</span>&#41; on chromosome X&#40;Xq27&#46;3&#41;&#44; blocking the production of the <span class="elsevierStyleItalic">FMR1</span> gene protein &#40;FMRP&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The phenotypic characteristics of this syndrome may have significant anaesthetic implications&#44; among other findings&#44; because of craniofacial abnormalities&#46; Physical characteristics include macrocephaly&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">3&#44;6</span></a> hyperteolirsm&#44; strabismus&#44; prognathism&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> large prominent ears&#44; and postpubertal marcroorchidism&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">7</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Dental implantation is abnormal&#44; with abraded dental surfaces as well as large crowns that create severe bone&#8211;teeth discrepancies&#44;<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">8</span></a> limiting mouth opening and impairing the placement of the endotracheal tube&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Our patient had prognathism as well as abnormal dental implantation limiting mouth opening to 3<span class="elsevierStyleHsp" style=""></span>cm&#46; Because of an anticipated difficult airway&#44; we decided to use the Glidescope<span class="elsevierStyleSup">&#174;</span> videolaryngoscope&#44;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">9</span></a> with successful endotracheal intubation and minimum stimulus&#44; thus avoiding an increase in intracranial pressure&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">FXS patients have excess joint laxity&#44;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">7</span></a> flat feet&#44; pectus excavatum and scoliosis&#46; Patient positioning on the operating table with adequate support points is essential in order to prevent joint dislocations and gonadal compression&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In 80&#8211;90&#37; of FXS cases there is moderate to severe mental retardation&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> Autism&#44; hyperactivity&#44; agitation and anxiety are also frequent&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> hence the need for adequate sedation before induction&#44; because they are usually uncooperative&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">In 15&#8211;20&#37;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> of cases there are partial complex and generalized tonic clonic seizures that are usually benign<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">3</span></a> and disappear before 20 years of age&#46; The right premedication may reduce surgical stress by raising the convulsive threshold perioperatively&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">From the cardiovascular standpoint&#44; 80&#37; of cases may be associated with MVP with no previous episodes of chest pain or palpitations<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a> but which may give rise to intraoperative arrhythmias&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a> Occasionally&#44; there is also aortic root dilatation&#44;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">4</span></a> in which case it is advisable to use adequate monitoring in order to implement GDT and create better conditions for improved results in major surgery&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">10</span></a> Because our patient was transferred from another hospital as a vital emergency&#44; we were unable to confirm the presence of a MVP&#46; However&#44; given the high incidence of this disorder and the fact that symptoms become exacerbated by anaesthesia induction&#44; leading to cardiovascular collapse&#44;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">11</span></a> we decided to perform non-invasive cardiac output monitoring&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The main objective of the anaesthetic management was to prevent any reduction in left ventricular volume during systole in order to reduce mitral valve prolapse&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">11</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Reduced venous return and vascular resistance&#44; tachycardia and increased contractility are not well tolerated&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">11</span></a> During general anaesthesia&#44; the use of vasopressors is recommended in order to maintain ABP&#44; together with short-acting beta-blockers for heart rate control&#44; end-diastolic and systolic volume preservation&#44; and mitral prolapse control&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">11</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Management is different in cases associated with mitral regurgitation because maintenance of higher heart rates shortens diastolic time and reduces regurgitation volumes&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">12</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Pulse pressure variation &#40;PPV&#41; and systolic volume variation &#40;SVV&#41; are dynamic predictors of response to fluids in patients under mechanical ventilation&#46; Their measurement is usually invasive by means of the signal derived from the ABP curve&#46; Non-invasive CO monitoring using an inflatable cuff with an in-built photoelectric plethysmographic device provides continuous ABP measurement<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">13</span></a> based on the development of a pulsatile discharge from the arterial walls in the finger&#46;<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">13</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">SVV and PPV measurements without the need to use an intra-arterial catheter offers an advantage in emergency surgery&#44; particularly in neurosurgery&#44; where any delay in starting the intervention may determine worse clinical outcomes&#46; GDT allows for early detection of pathophysiological changes and individualized adjustment of intraoperative haemodynamic management&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">10</span></a> Invasive ABP monitoring with an intra-arterial catheter<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">13</span></a> is considered the gold standard&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">14</span></a> However&#44; non-invasive measurement of dynamic predictors of response to fluid replacement has been shown to have high specificity and sensitivity&#44;<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">13</span></a> with improved safety and comfort&#46;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">15</span></a> This system can be used on top of non-invasive ABP monitoring in haemodynamically stable patients under general anaesthesia&#44; with the benefit of providing beat-to-beat ABP measurements&#46;<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">14</span></a> However&#44; its concomitant use with the PICCO<span class="elsevierStyleSup">&#174;</span> transpulmonary thermodilution monitor for measuring cardiac output is controversial&#46;<a class="elsevierStyleCrossRefs" href="#bib0155"><span class="elsevierStyleSup">15&#44;16</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">In summary&#44; we discuss the anaesthetic management of patients with FXS&#44; given the low incidence of this disorders and the very few reports found in the anaesthesia literature&#46; We believe that non-invasive cardiac output monitoring is a new option for emergency neurosurgical procedures and in patients with heart disease&#44; considering that it shortens anaesthesia time and provides reliable parameters for GDT&#46; Moreover&#44; we believe that videolaryngoscopy is the first choice for managing a predictably difficult airway in which endotracheal intubation might result in a significant increase in ICP&#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="par0095" 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="par0100" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Right to privacy and informed consent</span><p id="par0105" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Funding</span><p id="par0110" class="elsevierStylePara elsevierViewall">We received no funding for our work&#46;</p></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflicts of interest</span><p id="par0115" class="elsevierStylePara elsevierViewall">There are no conflicts of interest&#46;</p></span></span>"
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            0 => "Presi&#243;n sangu&#237;nea"
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            2 => "Traumatismos cerebrovasculares"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Fragile X syndrome is an inherited form of mental retardation with a connective tissue component involving mitral valve prolapse&#46; The most frequent manifestations of fragile X syndrome are learning disability&#44; orofacial morphological alterations and macroorchidism&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The usefulness of advanced haemodynamic monitoring for goal-directed therapy is increasingly high during neurosurgical procedures&#46; Non-invasive cardiac output monitoring may be considered as a new alternative for emergency neurosurgical procedures&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Our aim was to detect haemodynamic changes in a syndromic fragile X patient&#44; given the usual concomitant presentation of cardiovascular disease&#44; such as mitral valve prolapse and dilated aortic root&#44; in an attempt at obtaining the best intraoperative and postoperative neurological outcomes without worsening cardiovascular function&#44; by means of individualized intra-operative goal directed therapy&#46; This type of non-invasive monitoring allows surgery to proceed without delay and provides excellent information of the haemodynamic status&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">This syndrome is relevant due to its anaesthetic implications and the paucity of cases published to date&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El s&#237;ndrome X fr&#225;gil es una forma hereditaria de retraso mental con una afectaci&#243;n de tejido conectivo que produce prolapso de la v&#225;lvula mitral&#46; Las manifestaciones m&#225;s frecuentes del s&#237;ndrome X fr&#225;gil son la dificultad en el aprendizaje&#44; alteraciones morfol&#243;gicas orofaciales y macroorquidismo&#46;</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">La utilidad de la monitorizaci&#243;n hemodin&#225;mica avanzada para terapia dirigida por objetivos es cada vez mayor durante los procedimientos neuroquir&#250;rgicos&#46; La monitorizaci&#243;n no invasiva de gasto cardiaco puede considerarse una nueva alternativa en los procedimientos neuroquir&#250;rgicos emergentes&#46;</p><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Nuestro objetivo fue detectar los cambios hemodin&#225;micos en un paciente sindr&#243;mico X fr&#225;gil que suelen presentar patolog&#237;a cardiovascular&#44; como prolapso mitral y dilataci&#243;n de la ra&#237;z a&#243;rtica&#44; intentando obtener los mejores resultados neurol&#243;gicos intraoperatorios y posoperatorios sin deteriorar la funci&#243;n cardiovascular individualizada por una terapia guiada por objetivos&#46; Este tipo de monitorizaci&#243;n no invasiva permite desarrollar la intervenci&#243;n quir&#250;rgica sin demora&#44; aportando gran informaci&#243;n del estado hemodin&#225;mico&#46; Este s&#237;ndrome es relevante debido a sus implicaciones anest&#233;sicas y los pocos casos publicados hasta la fecha&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Guerrero-Dom&#237;nguez R&#44; L&#243;pez-Herrera-Rodr&#237;guez D&#44; Beato-L&#243;pez FJ&#44; Jim&#233;nez I&#46; Manejo hemodin&#225;mico mediante monitor no invasivo de gasto cardiaco para craneotom&#237;a urgente en el s&#237;ndrome x fr&#225;gil&#58; reporte de caso&#46; Rev Colomb Anestesiol&#46; 2016&#59;44&#58;48&#8211;51&#46;</p>"
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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