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Aguiar, E. Ferreira, R. Pontes, A. Panzina, M. Paiva, A. Milheiro" "autores" => array:6 [ 0 => array:4 [ "nombre" => "I." "apellidos" => "Aguiar" "email" => array:1 [ 0 => "inesaguiarpereira@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "E." "apellidos" => "Ferreira" ] 2 => array:2 [ "nombre" => "R." "apellidos" => "Pontes" ] 3 => array:2 [ "nombre" => "A." "apellidos" => "Panzina" ] 4 => array:2 [ "nombre" => "M." "apellidos" => "Paiva" ] 5 => array:2 [ "nombre" => "A." "apellidos" => "Milheiro" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Anestesiología, Centro Hospitalario Vila Nova de Gaia/Espinho, E.P.E, Vila Nova de Gaia, Portugal" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dilemas en el manejo de glioblastoma espinal primario durante el embarazo" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 805 "Ancho" => 805 "Tamanyo" => 54277 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">T2-weighted sagittal cervicothoracic MRI showing the spinal GMB extending from C6 to T3.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Primary spinal glioblastoma (GBM) is an extremely rare entity<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> that develops in the glial cells and has a predilection for the cervical and thoracic spine.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a> Spinal GBMs are often low grade, with no predilection for sex.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The clinical presentation depends on the region of the spinal cord involved.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Prognosis is poor, despite intensive treatment.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> The association between spinal GBM and pregnancy has not hitherto been reported in the literature. In this case report, we describe the treatment of a woman diagnosed with primary spinal GBM during pregnancy, highlighting the dilemmas faced by anaesthesiologists, particularly in terms of the approach to caesarean section. The patient and her family gave their informed consent for the publication of this report.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 33-year-old woman, with 2 pregnancies and one delivery, with no medical history of note, was admitted at 20 weeks of gestation with a history of numbness and weakness in the arms, neck pain, muscle incoordination, urinary retention and alteration of intestinal motility for the preceding 2 and a half months. The neurological examination rated left upper limb strength at 3/5 and right at 4/5, bilateral lower limb strength at 4/5, and hypoesthesia as far as T2. Magnetic resonance imaging (MRI) showed an intramedullary lesion extending from C6 to T3 (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Three weeks later, a decompressive laminectomy was performed between C6 and T4, with subtotal removal of the lesion. The patient was carefully placed prone on a Wilson frame with a horseshoe-shaped headrest, under the supervision of a qualified obstetrician. After positioning the patient, and before the surgical incision, baseline transcranial somatosensory evoked potentials (SSEP) and motor evoked potentials (MEP) were measured to record her preoperative status. During surgery, multimodal monitoring included SSEP of the medial and posterior bilateral tibial nerves, MEP of the upper (C4–C7) and lower (L3–S1) extremities, and spontaneous bilateral electromyography (C4–C7; L3–S1). No changes were observed in SSEP and PEM of the upper extremities compared with baseline values. When the tumour was removed, an increase in the amplitude of PEM was observed in the lower extremities. Foetal well-being was ensured by maintaining haemodynamic stability and oxygenation of the mother. Surgery and anaesthesia were uneventful. The foetal heart rate was monitored immediate after surgery and found to be present and normal. Histopathology revealed primary spinal GBM, classified as grade IV by the World Health Organization.</p><p id="par0020" class="elsevierStylePara elsevierViewall">After the neurosurgical procedure, the patient's neurological status gradually deteriorated and she was unable to walk unaided due to bilateral weakness of the lower extremities. One week after surgery, she was transferred to a specialized centre for neuromotor and functional rehabilitation.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The case was discussed in a multidisciplinary meeting, where in accordance with the wishes of the patient and her family a caesarean section was scheduled for 28 weeks and 6 days of gestation to allow the start of chemotherapy and radiotherapy.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The preoperative evaluation showed a pregnant patient with a body mass index of 21.97<span class="elsevierStyleHsp" style=""></span>kg/m<span class="elsevierStyleSup">2</span> (weight<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>65<span class="elsevierStyleHsp" style=""></span>kg and height<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>172<span class="elsevierStyleHsp" style=""></span>cm), ASA (American Society of Anesthesiologists) physical status class III, who had recently undergone spinal surgery and presented residual intramedullary GBM that caused grade D tetraparesis on the AIS (American Spinal Injury Association Impairment Scale) scale: motor level C6 and C5 left and sensory level C7. An airway examination showed significantly limited neck mobility, but no other predictors of difficult airway. Laboratory studies showed anaemia, but no other abnormalities. The resting electrocardiogram was normal. Before surgery, the antenatal corticosteroid cycle was completed with dexamethasone to minimize the morbidity and mortality associated with prematurity, and infusion of magnesium sulphate was started to provide foetal neuroprotection.</p><p id="par0035" class="elsevierStylePara elsevierViewall">After discussing the strategy with all team members, it was decided to administer general anaesthesia. Monitoring included pulse oximetry, invasive blood pressure, 5-lead electrocardiogram, capnography, end-tidal sevoflurane concentration, anaesthetic depth, neuromuscular block, central and peripheral temperature, and urinary output.</p><p id="par0040" class="elsevierStylePara elsevierViewall">After intravenous administration of 10<span class="elsevierStyleHsp" style=""></span>mg metoclopramide and 50<span class="elsevierStyleHsp" style=""></span>mg ranitidine, rapid sequence induction was performed using bolus doses of propofol (2<span class="elsevierStyleHsp" style=""></span>mg/kg) and rocuronium (1.2<span class="elsevierStyleHsp" style=""></span>mg/kg). Intubation was performed with a video laryngoscope, after confirming total loss of response of the orbicularis oculi muscle with train of four (TOF) stimulation in the temporal branch of the facial nerve. Anaesthesia was maintained with 60% nitrous oxide and sevoflurane was titrated to maintain a bispectral index of between 40 and 60. The patient's temperature was maintained with a forced air heating blanket and heated intravenous fluids.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Pfannestiel and low-transverse uterine incisions were made, and an infant weighing 1340<span class="elsevierStyleHsp" style=""></span>g with an Apgar score of 5, 7, and 8 at min 1, 5, and 10 was born. After delivery of the child, anaesthesia was supplemented with 200<span class="elsevierStyleHsp" style=""></span>mcg of fentanyl. Intravenous infusion of 20<span class="elsevierStyleHsp" style=""></span>IU oxytocin in 500<span class="elsevierStyleHsp" style=""></span>ml of 0.9% saline was administered for 1<span class="elsevierStyleHsp" style=""></span>h. At the end of the procedure, the neuromuscular block was reversed using 200<span class="elsevierStyleHsp" style=""></span>mg sugammadex, and after ensuring a TOF ratio greater than 0.9, the patient was extubated without incident. Estimated blood loss was 900<span class="elsevierStyleHsp" style=""></span>ml. Total crystalloids was 1500<span class="elsevierStyleHsp" style=""></span>ml and urinary output was 1<span class="elsevierStyleHsp" style=""></span>ml/kg per hour. The patient was monitored in the intensive care unit for 24<span class="elsevierStyleHsp" style=""></span>h after surgery, where her neurological status and clinical parameters remained unchanged. She was transferred to an internal medicine service to start the treatment plan. On the sixth postpartum day, a follow-up MRI showed evidence of tumour progression, with leptomeningeal diffusion (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A combination of radiotherapy and chemotherapy with temozolomide was started, and the patient was discharged on the nineteenth postpartum day. Outpatient cancer treatment was maintained, but the patient's neurological and overall general status continued to deteriorate. Considering the clinical outlook and the poor prognosis associated with the disease, no additional treatment was considered: the patient died 9 months after the initial diagnosis.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">The baby remained hospitalized for 109 days, during which time he presented problems related to prematurity. He is currently being monitored by a neuropaediatrician on an outpatient basis, and continues to make progress despite his developmental delays.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">Spinal cord tumours account for about 2%–4% of all central nervous system tumours.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Primary spinal GBM is extremely rare, and accounts for only 1.5% of spinal cord tumours.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The most widely reported location of this tumour is the cervical and thoracic spine, with the cervical spine as the most affected segment, followed by the thoracic spine.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a> This neoplasm is more common in patients between 20 and 40 years old, with no predilection for sex.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The presence or absence of neurological findings will usually be determined by the location in the spinal cord and the local extent of the tumour.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">MRI is the standard technique for diagnosing intramedullary tumours.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Once the imaging diagnosis is suspected, surgical intervention is usually recommended to provide a definitive diagnosis, improve neurological functions and, if possible, remove the mass.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Although controversial, treatment for primary spinal GBM is usually multimodal and consists of surgery followed by radiation and adjuvant chemotherapy, since complete resection is rarely possible.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Studies show that early elective caesarean section before cancer treatment is started is an acceptable option for women in the final stage of the second trimester of pregnancy who present intracranial gliomas and progressive neurological signs.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In this case, the multidisciplinary team decided to perform neurosurgery during pregnancy followed by an elective caesarean section in order to administer postpartum radiation therapy and chemotherapy.</p><p id="par0075" class="elsevierStylePara elsevierViewall">The choice of anaesthesia for the caesarean section raised several questions which were discussed by the multidisciplinary team, weighing up the risks and benefits of general and neuraxial anaesthesia, and taking into account the wishes of the patient.</p><p id="par0080" class="elsevierStylePara elsevierViewall">The potential problems associated with spinal and epidural anaesthesia in this patient included sympathectomy with the possibility of spinal cord ischaemia, sensory changes due to residual tumour, changes in scarring and inflammation of the spinal cord, possible post-dural puncture neurological sequelae, and changes in spinal cord pressures.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5,6</span></a> Similarly, regional anaesthesia in a patient with “unstable” neurological injury would make it difficult, if not impossible, to determine the exact cause of possible postoperative neurological impairment.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Rapid induction of general anaesthesia was performed with rocuronium due to the succinylcholine-induced hyperkalemic response in patients with paraplegia and the availability of sugammadex for reversal. The orbicularis oculi muscle appeared to be ideal for monitoring the degree of neuromuscular block in this patient, since monitoring the paretic limb can lead to underestimation of the block and significant overdosage of the neuromuscular blocking agent.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">Despite severely limited neck mobility, there were no other predictors of difficult airway, and therefore the video laryngoscope seemed to be the optimal airway device for achieving tracheal intubation. The risks associated with the haemodynamic changes caused by aorto-cava compression of the uterus, general anaesthesia, and autonomic dysfunction were overcome by proper positioning with left uterine displacement, the use of invasive blood pressure monitoring, and immediate availability of vagolytic and vasoactive drugs. Similarly, dysregulation of the autonomic nervous system in patients with spinal cord injuries above T8 include poikilothermia, so every effort should be made to maintain perioperative normothermia.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">Primary spinal GBM usually carries a poor prognosis despite treatment. The overall survival for this malignancy tends to be 12–24 months.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Some authors argue that histological subtype, age at diagnosis, sex, and extent of excision are related to survival, while others report that postoperative radiation therapy is the only significant factor associated with prolonged survival.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> However, we offered advice based on the best of our knowledge, supported by the literature, to improve the outcome in this patient and her baby. The question remains as to whether waiting a few more weeks before the caesarean section would have prevented the delay in the baby's development, without affecting the mother's prognosis, although we feel that the wishes of the patient and her family should prevail.</p><p id="par0100" class="elsevierStylePara elsevierViewall">In conclusion, the management of pregnant patients with primary spinal GBM is difficult. For anaesthesiologists, it is not only an ethical but also an anaesthetic dilemma. Early caesarean section to allow cancer treatment should be considered individually, weighing up the potential benefits in terms of survival for the mother against the risks to the foetus. General anaesthesia for caesarean section is an option that was used safely in this patient. Regardless of the anaesthetic technique used, maintenance of oxygenation, normocarbia (normocapnia) and basal blood pressure are essential to provide placental perfusion and avoid foetal acidosis. Prognosis is poor, despite intensive treatment. More research is needed to provide guidelines and hopefully improve survival and improve quality of life.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Funding</span><p id="par0105" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflicts of interest</span><p id="par0110" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1375800" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1263655" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1375801" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1263654" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-12-07" "fechaAceptado" => "2020-02-21" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1263655" "palabras" => array:4 [ 0 => "Spinal glioblastoma" 1 => "Pregnancy" 2 => "Anaesthesia" 3 => "Caesarean delivery" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1263654" "palabras" => array:4 [ 0 => "Glioblastoma espinal" 1 => "Embarazo" 2 => "Anestesia" 3 => "Parto por cesárea" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Primary spinal glioblastoma (GBM) is a clinically rare entity with rapid progression and a dismal outcome despite aggressive treatment. In a pregnant woman, this malignancy is particularly dramatic because the potential benefits to the mother offered by standard GBM treatment must be balanced against the risks to the foetus. There is little guidance in the literature on how to manage pregnant patients with malignant neuraxial tumours and, to the authors’ knowledge, no reports have been published so far regarding this specific neoplasm in such population. This case report describes the management of a pregnant patient with a previously undiagnosed and rapidly progressive intramedullary GBM submitted to an elective caesarean delivery to allow subsequent onset of oncological treatment. Dilemmas faced by anaesthetists are discussed in hope to provide guidance for future decisions and optimize outcomes.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El glioblastoma espinal primario (GBM) es una entidad clínicamente rara, con progresión rápida y resultado inevitable, a pesar de su manejo intensivo. En una mujer embarazada, esta neoplasia es particularmente grave, ya que los beneficios potenciales para la madre que ofrece el tratamiento estándar para GBM deben sopesarse frente a los riesgos para el feto. Existen pocas directrices en la literatura sobre cómo manejar a las pacientes embarazadas con tumores neuroaxiales malignos y, en opinión de los autores, no se han publicado informes hasta la fecha sobre este neoplasma específico en dicha población. Este caso clínico describe el manejo de una paciente embarazada con GBM no diagnosticado previamente, con rápida progresión intramedular, a la que se realizó cesárea electiva para permitir el inicio de tratamiento oncológico. Debatimos los dilemas a los que se enfrentan los anestesistas, con esperanza de aportar directrices ante las futuras decisiones y de optimizar los resultados.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Aguiar I, Ferreira E, Pontes R, Panzina A, Paiva M, Milheiro A. Dilemas en el manejo de glioblastoma espinal primario durante el embarazo. Rev Esp Anestesiol Reanim. 2020;67:347–350.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 805 "Ancho" => 805 "Tamanyo" => 54277 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">T2-weighted sagittal cervicothoracic MRI showing the spinal GMB extending from C6 to T3.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 805 "Ancho" => 805 "Tamanyo" => 74992 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">T2-weighted sagittal cervicothoracic MRI showing progression of the GBM with leptomeningeal diffusion.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Primary spinal glioblastoma multiforme: a case report and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "C.X. 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Case report
Dilemmas in primary spinal glioblastoma management during pregnancy
Dilemas en el manejo de glioblastoma espinal primario durante el embarazo
I. Aguiar
, E. Ferreira, R. Pontes, A. Panzina, M. Paiva, A. Milheiro
Autor para correspondencia
Departamento de Anestesiología, Centro Hospitalario Vila Nova de Gaia/Espinho, E.P.E, Vila Nova de Gaia, Portugal