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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Intramedullary area filled with a well-defined&#44; sharp-contoured mass which has craniocaudal diameter of 33<span class="elsevierStyleHsp" style=""></span>mm with hypointensity in center and peripheric contrast enhancement on T1-weighted with contrast images at T4&#8211;T5 level&#46; Radiological preliminary diagnosis of the mass was ependymoma or metastasis&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Pancreatic cancer is a progressive and fatal disease&#46; There is an increase in incidence of pancreatic cancer along with the increase of the average life expectancy&#46; Although the prognosis is poor&#44; survival of patients is prolonged with surgical and adjuvant &#40;chemotherapy&#44; radiotherapy&#41; treatments&#46; In the follow-up of these patients&#44; there is need for screening of metastases to prevent poor quality of life and apply early treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The frequent sites of spinal metastases are vertebrae and epidural space&#46; Intramedullary involvement is very rare&#46; In large autopsy series of cancer patients&#44; the incidence of intramedullary spinal cord metastasis &#40;ISCM&#41; was found between 0&#46;9&#37; and 3&#46;5&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#8211;4</span></a> Recently&#44; studies indicate that the first choice of treatment modality should be early resection for suitable patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0235"><span class="elsevierStyleSup">5&#8211;7</span></a> In addition&#44; steroids and radiotherapy are advised for patients with poor general condition and advanced neurological deficits&#46; Although ISCM originating from various other primary tumors has also been reported&#44; to the best of our knowledge&#44; no reports have discussed ISCM originating from pancreatic adenocarcinoma&#46; Here&#44; we describe the first case of ISCM of pancreatic adenocarcinoma&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Case presentation</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 36-year-old woman presented with back pain&#44; difficulty in urinating&#44; and weakness in the lower extremities&#46; She had a history of Whipple operation 1 year ago&#46; She was histopathologically diagnosed as pancreatic adenocarcinoma after this operation&#46; She had a history of folfirinox&#44; Irinitekan&#44; Oxaliplatin&#44; 5-fluorouracil&#44; folinic acid treatment&#46; Albumin-bound paclitaxel and Gemcitabine therapy were applied as the patient had progression in the follow-up&#46; Primary tumor was stable after these chemotherapy regimens&#46; After the patient had started to complain about pain and numbness in the lower extremities for 2 weeks&#44; an intramedullary mass was detected at T4&#8211;T5 level on MR images &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; She was paraparetic &#40;2&#47;5&#41; and bilateraly hypoesthetic&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The patient was discussed in the neurooncology tumor board&#46; Surgical intervention was planned for intramedullary tumor&#46; Although the primary disease was expected to have a faster course and worse prognosis&#44; her stable condition after Whipple operation with adjuvant chemotherapy&#44; the absence of any distant metastasis in PET-CT at the time of diagnosis of intramedullary tumor&#44; and the progressive neurological deterioration were factors for decision period&#46; In addition&#44; the patient preferred surgical treatment because she did not have a sign&#47;symptom related to primary disease and she was unable to walk in a short time due to progressive neurological deficit&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient underwent T4&#8211;T5 total laminectomy&#44; and the dural sac was exposed&#46; The dural sac&#44; which was expanded and stretched&#44; was hung on the sides by making a liner incision in the midline&#46; The spinal cord was swollen and non-pulsatile&#46; Tumor tissue was reached by deepening through the medullary incision from the midline&#46; The lesion&#39;s color was dirty pink&#46; It was soft and had increased vascularity&#46; A sample was taken for frozen examination &#40;the result was adenocarcinoma metastasis&#41;&#46; En bloc removal was done&#46; Spillage of tumor fragments into subarachonid space was prevented by placing cottonoids below and above of the surgical area&#46; Somatosensory and motor evoked potentials were monitored during surgery&#44; and there was no change during surgery&#46; The pathological result of the mass was reported as adenocarcinoma metastasis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Postoperative motor score of the patient was examined as left lower limb global muscle strength 1&#47;5&#44; right lower limb proximal 2&#47;5&#44; distal 1&#47;5&#46; No mass was observed on postoperative T1-weighted images with contrast &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Physical therapy and rehabilitation program was started after surgery and she recovered mostly in 3 months with the help of rehabilitation &#40;She was &#8722;4&#47;5 paraparetic&#44; and was able to walk with support in last follow-up&#41;&#46; She survived for 6 months after surgery despite postoperative craniocervical palliative radiotherapy for leptomeningeal metastases&#46; The patient died because of distant disease&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Intramedullary primary spinal cord tumors mostly consist of gliomas and constitute 60&#8211;80&#37; of ependymomas&#46;<a class="elsevierStyleCrossRefs" href="#bib0250"><span class="elsevierStyleSup">8&#44;9</span></a> Metastases constitute 1&#8211;3&#37; of these tumors&#46; The prevalence of ISCM in cancer patients range from 0&#46;9&#37; to 2&#46;1&#37; in autopsy series&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#44;3</span></a> Lung cancer is the most common source of ISCM&#44; and constitutes 50&#37; of ISCMs&#46; Breast carcinoma is the second with 16&#37; incidence&#46; Prostate&#44; melanoma&#44; and renal cell carcinoma are among the less frequently reported sources&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#44;10&#44;11</span></a> The incidence of ISCM increases with improved survival in most cancer types due to advanced treatments and neuroimaging techniques&#46;<a class="elsevierStyleCrossRef" href="#bib0270"><span class="elsevierStyleSup">12</span></a> The number of published cases has increased in recent years&#46; According to a review conducted by Payer et al&#46; in 2015&#44; almost 60&#37; of all ISCM treated in last 3 years of a 22-year period&#44;<a class="elsevierStyleCrossRef" href="#bib0275"><span class="elsevierStyleSup">13</span></a> and the trend is still increasing&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Pancreatic cancer is found in approximately 27&#44;000 people each year in United States&#44; and about 338&#44;000 people in the world &#40;2&#37; of all cancer diagnosis&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0280"><span class="elsevierStyleSup">14&#8211;16</span></a> Patients with pancreatic cancer have one of the worst prognosis among patients with other types of cancer&#46; The overall median survival from diagnosis is 4&#46;6 months&#44; and median survival is 2&#46;8&#8211;5&#46;7 months despite all chemotherapy regimens&#46;<a class="elsevierStyleCrossRefs" href="#bib0295"><span class="elsevierStyleSup">17&#44;18</span></a> Park et al&#46; found that the incidence of central nervous system metastasis in patients with pancreatic cancer was 0&#46;57&#37;&#44; and 3 of 1229 patients had spinal metastases&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a> None of them was intramedullary&#46; However&#44; Kim et al&#46; reported a case of ISCM from a pancreatic neuroendocrine tumor which was a different pathology from our patient&#39;s&#46;<a class="elsevierStyleCrossRef" href="#bib0305"><span class="elsevierStyleSup">19</span></a> To our best knowledge&#44; this is the first published case report of ISCM originated from pancreatic adenocarcinoma&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Hematogeneous spread&#44; leptomeningeal seeding via cerebrospinal fluid&#44; parenchymal penetration through the veins penetrating the Virchow-Robin cavities&#44; direct extension from dura mater or nerve roots&#44; and spread through Batson&#39;s plexus are possible routes of tumor extension&#46;<a class="elsevierStyleCrossRefs" href="#bib0310"><span class="elsevierStyleSup">20&#8211;23</span></a> Hematogeneous spread is suggested mechanism of ISCM occurrence&#46;<a class="elsevierStyleCrossRef" href="#bib0310"><span class="elsevierStyleSup">20</span></a> Park et al&#46; found three out of four patients with cerebral metastasis also had lung metastasis in their study including 1&#44;229 patients with pancreatic cancer&#46; However&#44; only one of three patients with spinal metastasis had lung involvement&#46; Also&#44; spinal metastasis and pancreatic cancer were simultaneously diagnosed in these three patients&#46; So&#44; they stated that cerebral metastasis may occur hematogenously&#44; and spinal metastasis may be mediated by local spread through Batson&#39;s plexus&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a> We think that&#44; most favorable route for pancreatic cancer dissemination is local spread through venous plexus due to the proximity of primary tumor&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The prognosis of intramedullary metastasis has been repeatedly reported as extremely poor&#46; Several studies found a median survival of less than 12 months&#46;<a class="elsevierStyleCrossRefs" href="#bib0260"><span class="elsevierStyleSup">10&#44;24</span></a> There is no full consensus on ISCM treatment and the treatment schemes to be followed are still being discussed&#46; Previous studies are retrospective case series and do not give clear results&#46; Since ISCM is rare&#44; the amount of data to clarify treatment modalities is also insufficient&#46; Therefore&#44; the treatment scheme is arranged individually according to each case&#46; Resection for ISCM is an option in patients who have rapid neurological deterioration&#44; good preoperative The Karnofsky Performance Score &#40;KPS&#41; score&#44; radioresistant tumor or ISCM with unknown primary malignancy that requires histopathological diagnosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0275"><span class="elsevierStyleSup">13&#44;25&#8211;28</span></a> Kalayci et al&#46; analyzed 32 patients&#44; that have been published in different studies&#46;<a class="elsevierStyleCrossRef" href="#bib0270"><span class="elsevierStyleSup">12</span></a> They showed that there were no new postoperative neurological deficits&#46; They also found improvement in clinical conditions of patients and increase in survival &#40;from 5 months to 9&#46;4 months&#41;&#46; Gasser et al&#46;<a class="elsevierStyleCrossRef" href="#bib0355"><span class="elsevierStyleSup">29</span></a> and Wilson et al&#46;<a class="elsevierStyleCrossRef" href="#bib0350"><span class="elsevierStyleSup">28</span></a> reported that surgical treatment should be considered as a priority in choice&#46; Our patient survived for 6 months after surgery because of aggressive nature of pancreatic adenocarcinoma&#46; However&#44; surgery was our choice of treatment due to rapid neurological deterioration and good general condition of patient&#44; and stable primary tumor&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Strickland et al&#46; noted transient deterioration in neurological function postoperatively in 6 of 13 patients&#46; However&#44; 4 of them improved in 2 months&#46;<a class="elsevierStyleCrossRef" href="#bib0340"><span class="elsevierStyleSup">26</span></a> Our patient also had a decline in neurological function postoperatively&#46; Fortunately&#44; she recovered in 3 months with the help of rehabilitation&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Patients with no neurological deficits&#44; multiple metastases or low KPS scores are not good candidates for surgery&#46;<a class="elsevierStyleCrossRefs" href="#bib0260"><span class="elsevierStyleSup">10&#44;30&#44;31</span></a> If patients with ISCM are followed without treatment&#44; rapid neurological deterioration is observed due to local compression&#44; cord edema&#44; and infarction&#46;<a class="elsevierStyleCrossRefs" href="#bib0330"><span class="elsevierStyleSup">24&#44;32&#44;33</span></a> Although steroid and radiotherapy are the primary non-surgical treatments&#44; they are more effective when the neurological examination is intact and the tumor shows radiosensitivity&#46;<a class="elsevierStyleCrossRefs" href="#bib0370"><span class="elsevierStyleSup">32&#44;34&#44;35</span></a> Radiotherapy is preferable because it is not invasive&#44; slows tumor progression and provides neurological improvement&#44; nonetheless its effect is limited in radioresistant tumors like in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0390"><span class="elsevierStyleSup">36&#44;37</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In a study of 5 cases&#44; Shin et al&#46; reported clinical improvement in 4 patients after single fraction radiosurgery&#46; Median survival time has been reported as 5&#46;2 months &#40;2&#46;2&#8211;19&#46;4 months&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0400"><span class="elsevierStyleSup">38</span></a> In a study of 18 cases treated with fractionated radiotherapy&#44; Hashii et al&#46; reported clinical improvement in 10 of the patients&#46; Median survival was 4 months&#46;<a class="elsevierStyleCrossRef" href="#bib0405"><span class="elsevierStyleSup">39</span></a> Two studies&#59; involving 11 cases<a class="elsevierStyleCrossRef" href="#bib0410"><span class="elsevierStyleSup">40</span></a> and 6 cases<a class="elsevierStyleCrossRef" href="#bib0415"><span class="elsevierStyleSup">41</span></a> reported that following a short recovery period&#44; a rapid neurological deterioration observed within days after radiotherapy and steroid treatment&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Radiotherapy&#44; surgery or combined approaches will probably never be compared in a randomized trial due to the insufficient number of cases and data&#46; So&#44; it is necessary to evaluate each patient individually and propose the best treatment modality&#46; Also&#44; multidisciplinary approach &#40;neurosurgery&#44; radiation oncology&#44; and medical oncology&#41; should be taken into consideration to achieve better prognosis&#44; and quality of life for patients&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">To date&#44; there have been no reports of ISCM from pancreatic adenocarcinoma&#44; so no studies have discussed the optimal treatment for such cases&#46; Moreover&#44; a majority of pancreatic cancer patients do not respond to radiotherapy&#46;<a class="elsevierStyleCrossRef" href="#bib0420"><span class="elsevierStyleSup">42</span></a> Surgical treatment should be regarded as a significant therapeutic option&#44; such as in our case&#46; Although ISCM is a rare situation&#44; it is important to include it among the possibilities in differential diagnosis of patients suffering rapid and severe neurological deficits&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Conclusion</span><p id="par0075" class="elsevierStylePara elsevierViewall">To date&#44; there are no previous reports of ISCM from pancreatic adenocarcinoma&#46; We report the first case of ISCM originated from pancreatic adenocarcinoma that was removed successfully by microsurgical procedure&#46; Since it is known to be radioresistant&#44; the first treatment option for pancreatic adenocarcinomas should be surgery&#46; In patients with pancreatic adenocarcinoma and progressively developing neurological deficits&#44; ISCM should also be kept in mind in the differential diagnosis&#46; Survival and quality of life can be increased by early diagnosis and treatment in these patients&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Conflict of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">None&#46;</p></span></span>"
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    "fechaRecibido" => "2021-01-06"
    "fechaAceptado" => "2021-03-25"
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            2 => "Metastasis"
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            0 => "Adenocarcinoma de p&#225;ncreas"
            1 => "Intramedular"
            2 => "Met&#225;stasis"
            3 => "Cirug&#237;a"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Pancreatic cancer is a common gastrointestinal malignancy&#44; and is often associated with a poor prognosis&#46; Although liver is generally seen as a distant metastasis point&#44; it has been shown that it can metastasize to any organ&#44; especially the gastrointestinal tract&#44; and approximately 0&#46;3&#37; of metastases are observed in spinal cord&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case description</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We report a 36-year-old woman with a prior history of pancreatic adenocarcinoma who presented to us with a thoracic intramedullary lesion and recent onset of neurological deficits&#46; She underwent surgery with histological confirmation of a diagnosis of metastatic adenocarcinoma&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conclusion</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">To our knowledge there is no prior report of pure intramedullary spinal cord metastasis from a pancreatic adenocarcinoma in the literature&#46; We report the present patient in view of the rarity of intramedullary spinal cord metastasis and its clinical significance&#46; Although intramedullary metastases are rare&#44; they should be investigated in every patient with malignancy and progressive neurological deficit&#46; While its general prognosis is poor regardless of the type of treatment&#44; early diagnosis and treatment is important in terms of quality of life and survival&#46;</p></span>"
        "secciones" => array:3 [
          0 => array:2 [
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        "resumen" => "<span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Antecedentes</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El c&#225;ncer de p&#225;ncreas es una neoplasia maligna gastrointestinal com&#250;n y&#44; a menudo&#44; se asocia con un mal pron&#243;stico&#46; Aunque el h&#237;gado generalmente se ve como un punto de met&#225;stasis distante&#44; se ha demostrado que puede hacer met&#225;stasis a cualquier &#243;rgano&#44; especialmente al tracto gastrointestinal&#44; y aproximadamente el 0&#44;3&#37; de las met&#225;stasis se observan en la m&#233;dula espinal&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Descripci&#243;n del caso</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos una mujer de 36 a&#241;os con antecedentes de adenocarcinoma de p&#225;ncreas que se present&#243; con una lesi&#243;n intramedular tor&#225;cica y de reciente aparici&#243;n de d&#233;ficits neurol&#243;gicos&#46; Fue intervenida quir&#250;rgicamente con confirmaci&#243;n histol&#243;gica de diagn&#243;stico de adenocarcinoma metast&#225;sico&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusi&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Hasta donde sabemos&#44; no existe en la literatura ning&#250;n informe previo de met&#225;stasis intramedular pura de la m&#233;dula espinal de un adenocarcinoma pancre&#225;tico&#46; Presentamos el presente paciente en vista de la rareza de la met&#225;stasis intramedular de la m&#233;dula espinal y su importancia cl&#237;nica&#46; Aunque las met&#225;stasis intramedulares son raras&#44; deben investigarse en todo paciente con neoplasia maligna y d&#233;ficit neurol&#243;gico progresivo&#46; Si bien su pron&#243;stico general es malo independientemente del tipo de tratamiento&#44; el diagn&#243;stico y el tratamiento precoces son importantes en t&#233;rminos de calidad de vida y supervivencia&#46;</p></span>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Intramedullary area filled with a well-defined&#44; sharp-contoured mass which has craniocaudal diameter of 33<span class="elsevierStyleHsp" style=""></span>mm with hypointensity in center and peripheric contrast enhancement on T1-weighted with contrast images at T4&#8211;T5 level&#46; Radiological preliminary diagnosis of the mass was ependymoma or metastasis&#46;</p>"
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          "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Hematoxylin&#8211;eosin staining of a section from the intramedullary mass &#40;H&#38;E&#44; 40&#215;&#41;&#59; &#40;B&#41; High magnification &#40;H&#38;E&#44; 100&#215;&#41; view of the tumor with large pleomorphic epithelial cells and extensive necrosis&#59; &#40;C&#41; tumor cells shows focal nuclear positivity with CDX2 stain &#40;IHC&#44; 100&#215;&#41;&#59; &#40;D&#41; Tumor cells positive for cytokeratin &#40;IHC&#44; 100&#215;&#41;&#46;</p>"
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                            0 => "G&#46; Manzano"
                            1 => "B&#46;A&#46; Green"
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                      "doi" => "10.1038/sc.2008.51"
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:6 [
                            0 => "A&#46; Raco"
                            1 => "V&#46; Esposito"
                            2 => "J&#46; Lenzi"
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                            4 => "R&#46; Delfini"
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                          ]
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              "identificador" => "bib0245"
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                      "titulo" => "Functional outcome after surgical treatment of intramedullary spinal cord tumors&#58; experience with 78 patients"
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                        0 => array:2 [
                          "etal" => true
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                            0 => "I&#46;E&#46; Sandalcioglu"
                            1 => "T&#46; Gasser"
                            2 => "S&#46; Asgari"
                            3 => "A&#46; Lazorisak"
                            4 => "T&#46; Engelhorn"
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                  ]
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                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Fluorescein-guided resection of intramedullary spinal cord tumors&#58; results from a preliminary&#44; multicentric&#44; retrospective study"
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                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "F&#46; Acerbi"
                            1 => "C&#46; Cavallo"
                            2 => "K&#46;-M&#46; Schebesch"
                            3 => "M&#46;O&#46; Ak&#231;akaya"
                            4 => "C&#46; de Laurentis"
                            5 => "M&#46;K&#46; Hamamcioglu"
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Case Report
Intramedullary pancreatic adenocarcinoma metastasis: The first case in literature
Metástasis de adenocarcinoma de páncreas intramedular: primer caso en la literatura
Alican Tahtaa,
Corresponding author
atahta@medipol.edu.tr

Corresponding author.
, Ahmet Cetinkala, Elif Calisb, Cem Dinca
a Istanbul Medipol University, Faculty of Medicine, Department of Neurosurgery, Istanbul, Turkey
b Istanbul Medipol University, Faculty of Medicine, Department of Pathology, Istanbul, Turkey
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Intramedullary area filled with a well-defined&#44; sharp-contoured mass which has craniocaudal diameter of 33<span class="elsevierStyleHsp" style=""></span>mm with hypointensity in center and peripheric contrast enhancement on T1-weighted with contrast images at T4&#8211;T5 level&#46; Radiological preliminary diagnosis of the mass was ependymoma or metastasis&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Pancreatic cancer is a progressive and fatal disease&#46; There is an increase in incidence of pancreatic cancer along with the increase of the average life expectancy&#46; Although the prognosis is poor&#44; survival of patients is prolonged with surgical and adjuvant &#40;chemotherapy&#44; radiotherapy&#41; treatments&#46; In the follow-up of these patients&#44; there is need for screening of metastases to prevent poor quality of life and apply early treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The frequent sites of spinal metastases are vertebrae and epidural space&#46; Intramedullary involvement is very rare&#46; In large autopsy series of cancer patients&#44; the incidence of intramedullary spinal cord metastasis &#40;ISCM&#41; was found between 0&#46;9&#37; and 3&#46;5&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#8211;4</span></a> Recently&#44; studies indicate that the first choice of treatment modality should be early resection for suitable patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0235"><span class="elsevierStyleSup">5&#8211;7</span></a> In addition&#44; steroids and radiotherapy are advised for patients with poor general condition and advanced neurological deficits&#46; Although ISCM originating from various other primary tumors has also been reported&#44; to the best of our knowledge&#44; no reports have discussed ISCM originating from pancreatic adenocarcinoma&#46; Here&#44; we describe the first case of ISCM of pancreatic adenocarcinoma&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Case presentation</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 36-year-old woman presented with back pain&#44; difficulty in urinating&#44; and weakness in the lower extremities&#46; She had a history of Whipple operation 1 year ago&#46; She was histopathologically diagnosed as pancreatic adenocarcinoma after this operation&#46; She had a history of folfirinox&#44; Irinitekan&#44; Oxaliplatin&#44; 5-fluorouracil&#44; folinic acid treatment&#46; Albumin-bound paclitaxel and Gemcitabine therapy were applied as the patient had progression in the follow-up&#46; Primary tumor was stable after these chemotherapy regimens&#46; After the patient had started to complain about pain and numbness in the lower extremities for 2 weeks&#44; an intramedullary mass was detected at T4&#8211;T5 level on MR images &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; She was paraparetic &#40;2&#47;5&#41; and bilateraly hypoesthetic&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The patient was discussed in the neurooncology tumor board&#46; Surgical intervention was planned for intramedullary tumor&#46; Although the primary disease was expected to have a faster course and worse prognosis&#44; her stable condition after Whipple operation with adjuvant chemotherapy&#44; the absence of any distant metastasis in PET-CT at the time of diagnosis of intramedullary tumor&#44; and the progressive neurological deterioration were factors for decision period&#46; In addition&#44; the patient preferred surgical treatment because she did not have a sign&#47;symptom related to primary disease and she was unable to walk in a short time due to progressive neurological deficit&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient underwent T4&#8211;T5 total laminectomy&#44; and the dural sac was exposed&#46; The dural sac&#44; which was expanded and stretched&#44; was hung on the sides by making a liner incision in the midline&#46; The spinal cord was swollen and non-pulsatile&#46; Tumor tissue was reached by deepening through the medullary incision from the midline&#46; The lesion&#39;s color was dirty pink&#46; It was soft and had increased vascularity&#46; A sample was taken for frozen examination &#40;the result was adenocarcinoma metastasis&#41;&#46; En bloc removal was done&#46; Spillage of tumor fragments into subarachonid space was prevented by placing cottonoids below and above of the surgical area&#46; Somatosensory and motor evoked potentials were monitored during surgery&#44; and there was no change during surgery&#46; The pathological result of the mass was reported as adenocarcinoma metastasis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Postoperative motor score of the patient was examined as left lower limb global muscle strength 1&#47;5&#44; right lower limb proximal 2&#47;5&#44; distal 1&#47;5&#46; No mass was observed on postoperative T1-weighted images with contrast &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Physical therapy and rehabilitation program was started after surgery and she recovered mostly in 3 months with the help of rehabilitation &#40;She was &#8722;4&#47;5 paraparetic&#44; and was able to walk with support in last follow-up&#41;&#46; She survived for 6 months after surgery despite postoperative craniocervical palliative radiotherapy for leptomeningeal metastases&#46; The patient died because of distant disease&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Intramedullary primary spinal cord tumors mostly consist of gliomas and constitute 60&#8211;80&#37; of ependymomas&#46;<a class="elsevierStyleCrossRefs" href="#bib0250"><span class="elsevierStyleSup">8&#44;9</span></a> Metastases constitute 1&#8211;3&#37; of these tumors&#46; The prevalence of ISCM in cancer patients range from 0&#46;9&#37; to 2&#46;1&#37; in autopsy series&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#44;3</span></a> Lung cancer is the most common source of ISCM&#44; and constitutes 50&#37; of ISCMs&#46; Breast carcinoma is the second with 16&#37; incidence&#46; Prostate&#44; melanoma&#44; and renal cell carcinoma are among the less frequently reported sources&#46;<a class="elsevierStyleCrossRefs" href="#bib0220"><span class="elsevierStyleSup">2&#44;10&#44;11</span></a> The incidence of ISCM increases with improved survival in most cancer types due to advanced treatments and neuroimaging techniques&#46;<a class="elsevierStyleCrossRef" href="#bib0270"><span class="elsevierStyleSup">12</span></a> The number of published cases has increased in recent years&#46; According to a review conducted by Payer et al&#46; in 2015&#44; almost 60&#37; of all ISCM treated in last 3 years of a 22-year period&#44;<a class="elsevierStyleCrossRef" href="#bib0275"><span class="elsevierStyleSup">13</span></a> and the trend is still increasing&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Pancreatic cancer is found in approximately 27&#44;000 people each year in United States&#44; and about 338&#44;000 people in the world &#40;2&#37; of all cancer diagnosis&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0280"><span class="elsevierStyleSup">14&#8211;16</span></a> Patients with pancreatic cancer have one of the worst prognosis among patients with other types of cancer&#46; The overall median survival from diagnosis is 4&#46;6 months&#44; and median survival is 2&#46;8&#8211;5&#46;7 months despite all chemotherapy regimens&#46;<a class="elsevierStyleCrossRefs" href="#bib0295"><span class="elsevierStyleSup">17&#44;18</span></a> Park et al&#46; found that the incidence of central nervous system metastasis in patients with pancreatic cancer was 0&#46;57&#37;&#44; and 3 of 1229 patients had spinal metastases&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a> None of them was intramedullary&#46; However&#44; Kim et al&#46; reported a case of ISCM from a pancreatic neuroendocrine tumor which was a different pathology from our patient&#39;s&#46;<a class="elsevierStyleCrossRef" href="#bib0305"><span class="elsevierStyleSup">19</span></a> To our best knowledge&#44; this is the first published case report of ISCM originated from pancreatic adenocarcinoma&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Hematogeneous spread&#44; leptomeningeal seeding via cerebrospinal fluid&#44; parenchymal penetration through the veins penetrating the Virchow-Robin cavities&#44; direct extension from dura mater or nerve roots&#44; and spread through Batson&#39;s plexus are possible routes of tumor extension&#46;<a class="elsevierStyleCrossRefs" href="#bib0310"><span class="elsevierStyleSup">20&#8211;23</span></a> Hematogeneous spread is suggested mechanism of ISCM occurrence&#46;<a class="elsevierStyleCrossRef" href="#bib0310"><span class="elsevierStyleSup">20</span></a> Park et al&#46; found three out of four patients with cerebral metastasis also had lung metastasis in their study including 1&#44;229 patients with pancreatic cancer&#46; However&#44; only one of three patients with spinal metastasis had lung involvement&#46; Also&#44; spinal metastasis and pancreatic cancer were simultaneously diagnosed in these three patients&#46; So&#44; they stated that cerebral metastasis may occur hematogenously&#44; and spinal metastasis may be mediated by local spread through Batson&#39;s plexus&#46;<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">1</span></a> We think that&#44; most favorable route for pancreatic cancer dissemination is local spread through venous plexus due to the proximity of primary tumor&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The prognosis of intramedullary metastasis has been repeatedly reported as extremely poor&#46; Several studies found a median survival of less than 12 months&#46;<a class="elsevierStyleCrossRefs" href="#bib0260"><span class="elsevierStyleSup">10&#44;24</span></a> There is no full consensus on ISCM treatment and the treatment schemes to be followed are still being discussed&#46; Previous studies are retrospective case series and do not give clear results&#46; Since ISCM is rare&#44; the amount of data to clarify treatment modalities is also insufficient&#46; Therefore&#44; the treatment scheme is arranged individually according to each case&#46; Resection for ISCM is an option in patients who have rapid neurological deterioration&#44; good preoperative The Karnofsky Performance Score &#40;KPS&#41; score&#44; radioresistant tumor or ISCM with unknown primary malignancy that requires histopathological diagnosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0275"><span class="elsevierStyleSup">13&#44;25&#8211;28</span></a> Kalayci et al&#46; analyzed 32 patients&#44; that have been published in different studies&#46;<a class="elsevierStyleCrossRef" href="#bib0270"><span class="elsevierStyleSup">12</span></a> They showed that there were no new postoperative neurological deficits&#46; They also found improvement in clinical conditions of patients and increase in survival &#40;from 5 months to 9&#46;4 months&#41;&#46; Gasser et al&#46;<a class="elsevierStyleCrossRef" href="#bib0355"><span class="elsevierStyleSup">29</span></a> and Wilson et al&#46;<a class="elsevierStyleCrossRef" href="#bib0350"><span class="elsevierStyleSup">28</span></a> reported that surgical treatment should be considered as a priority in choice&#46; Our patient survived for 6 months after surgery because of aggressive nature of pancreatic adenocarcinoma&#46; However&#44; surgery was our choice of treatment due to rapid neurological deterioration and good general condition of patient&#44; and stable primary tumor&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Strickland et al&#46; noted transient deterioration in neurological function postoperatively in 6 of 13 patients&#46; However&#44; 4 of them improved in 2 months&#46;<a class="elsevierStyleCrossRef" href="#bib0340"><span class="elsevierStyleSup">26</span></a> Our patient also had a decline in neurological function postoperatively&#46; Fortunately&#44; she recovered in 3 months with the help of rehabilitation&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Patients with no neurological deficits&#44; multiple metastases or low KPS scores are not good candidates for surgery&#46;<a class="elsevierStyleCrossRefs" href="#bib0260"><span class="elsevierStyleSup">10&#44;30&#44;31</span></a> If patients with ISCM are followed without treatment&#44; rapid neurological deterioration is observed due to local compression&#44; cord edema&#44; and infarction&#46;<a class="elsevierStyleCrossRefs" href="#bib0330"><span class="elsevierStyleSup">24&#44;32&#44;33</span></a> Although steroid and radiotherapy are the primary non-surgical treatments&#44; they are more effective when the neurological examination is intact and the tumor shows radiosensitivity&#46;<a class="elsevierStyleCrossRefs" href="#bib0370"><span class="elsevierStyleSup">32&#44;34&#44;35</span></a> Radiotherapy is preferable because it is not invasive&#44; slows tumor progression and provides neurological improvement&#44; nonetheless its effect is limited in radioresistant tumors like in our patient&#46;<a class="elsevierStyleCrossRefs" href="#bib0390"><span class="elsevierStyleSup">36&#44;37</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In a study of 5 cases&#44; Shin et al&#46; reported clinical improvement in 4 patients after single fraction radiosurgery&#46; Median survival time has been reported as 5&#46;2 months &#40;2&#46;2&#8211;19&#46;4 months&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0400"><span class="elsevierStyleSup">38</span></a> In a study of 18 cases treated with fractionated radiotherapy&#44; Hashii et al&#46; reported clinical improvement in 10 of the patients&#46; Median survival was 4 months&#46;<a class="elsevierStyleCrossRef" href="#bib0405"><span class="elsevierStyleSup">39</span></a> Two studies&#59; involving 11 cases<a class="elsevierStyleCrossRef" href="#bib0410"><span class="elsevierStyleSup">40</span></a> and 6 cases<a class="elsevierStyleCrossRef" href="#bib0415"><span class="elsevierStyleSup">41</span></a> reported that following a short recovery period&#44; a rapid neurological deterioration observed within days after radiotherapy and steroid treatment&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Radiotherapy&#44; surgery or combined approaches will probably never be compared in a randomized trial due to the insufficient number of cases and data&#46; So&#44; it is necessary to evaluate each patient individually and propose the best treatment modality&#46; Also&#44; multidisciplinary approach &#40;neurosurgery&#44; radiation oncology&#44; and medical oncology&#41; should be taken into consideration to achieve better prognosis&#44; and quality of life for patients&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">To date&#44; there have been no reports of ISCM from pancreatic adenocarcinoma&#44; so no studies have discussed the optimal treatment for such cases&#46; Moreover&#44; a majority of pancreatic cancer patients do not respond to radiotherapy&#46;<a class="elsevierStyleCrossRef" href="#bib0420"><span class="elsevierStyleSup">42</span></a> Surgical treatment should be regarded as a significant therapeutic option&#44; such as in our case&#46; Although ISCM is a rare situation&#44; it is important to include it among the possibilities in differential diagnosis of patients suffering rapid and severe neurological deficits&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Conclusion</span><p id="par0075" class="elsevierStylePara elsevierViewall">To date&#44; there are no previous reports of ISCM from pancreatic adenocarcinoma&#46; We report the first case of ISCM originated from pancreatic adenocarcinoma that was removed successfully by microsurgical procedure&#46; Since it is known to be radioresistant&#44; the first treatment option for pancreatic adenocarcinomas should be surgery&#46; In patients with pancreatic adenocarcinoma and progressively developing neurological deficits&#44; ISCM should also be kept in mind in the differential diagnosis&#46; Survival and quality of life can be increased by early diagnosis and treatment in these patients&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Conflict of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">None&#46;</p></span></span>"
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    "fechaAceptado" => "2021-03-25"
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            2 => "Metastasis"
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            0 => "Adenocarcinoma de p&#225;ncreas"
            1 => "Intramedular"
            2 => "Met&#225;stasis"
            3 => "Cirug&#237;a"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Pancreatic cancer is a common gastrointestinal malignancy&#44; and is often associated with a poor prognosis&#46; Although liver is generally seen as a distant metastasis point&#44; it has been shown that it can metastasize to any organ&#44; especially the gastrointestinal tract&#44; and approximately 0&#46;3&#37; of metastases are observed in spinal cord&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case description</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We report a 36-year-old woman with a prior history of pancreatic adenocarcinoma who presented to us with a thoracic intramedullary lesion and recent onset of neurological deficits&#46; She underwent surgery with histological confirmation of a diagnosis of metastatic adenocarcinoma&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conclusion</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">To our knowledge there is no prior report of pure intramedullary spinal cord metastasis from a pancreatic adenocarcinoma in the literature&#46; We report the present patient in view of the rarity of intramedullary spinal cord metastasis and its clinical significance&#46; Although intramedullary metastases are rare&#44; they should be investigated in every patient with malignancy and progressive neurological deficit&#46; While its general prognosis is poor regardless of the type of treatment&#44; early diagnosis and treatment is important in terms of quality of life and survival&#46;</p></span>"
        "secciones" => array:3 [
          0 => array:2 [
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        "resumen" => "<span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Antecedentes</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El c&#225;ncer de p&#225;ncreas es una neoplasia maligna gastrointestinal com&#250;n y&#44; a menudo&#44; se asocia con un mal pron&#243;stico&#46; Aunque el h&#237;gado generalmente se ve como un punto de met&#225;stasis distante&#44; se ha demostrado que puede hacer met&#225;stasis a cualquier &#243;rgano&#44; especialmente al tracto gastrointestinal&#44; y aproximadamente el 0&#44;3&#37; de las met&#225;stasis se observan en la m&#233;dula espinal&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Descripci&#243;n del caso</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Presentamos una mujer de 36 a&#241;os con antecedentes de adenocarcinoma de p&#225;ncreas que se present&#243; con una lesi&#243;n intramedular tor&#225;cica y de reciente aparici&#243;n de d&#233;ficits neurol&#243;gicos&#46; Fue intervenida quir&#250;rgicamente con confirmaci&#243;n histol&#243;gica de diagn&#243;stico de adenocarcinoma metast&#225;sico&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusi&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Hasta donde sabemos&#44; no existe en la literatura ning&#250;n informe previo de met&#225;stasis intramedular pura de la m&#233;dula espinal de un adenocarcinoma pancre&#225;tico&#46; Presentamos el presente paciente en vista de la rareza de la met&#225;stasis intramedular de la m&#233;dula espinal y su importancia cl&#237;nica&#46; Aunque las met&#225;stasis intramedulares son raras&#44; deben investigarse en todo paciente con neoplasia maligna y d&#233;ficit neurol&#243;gico progresivo&#46; Si bien su pron&#243;stico general es malo independientemente del tipo de tratamiento&#44; el diagn&#243;stico y el tratamiento precoces son importantes en t&#233;rminos de calidad de vida y supervivencia&#46;</p></span>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Intramedullary area filled with a well-defined&#44; sharp-contoured mass which has craniocaudal diameter of 33<span class="elsevierStyleHsp" style=""></span>mm with hypointensity in center and peripheric contrast enhancement on T1-weighted with contrast images at T4&#8211;T5 level&#46; Radiological preliminary diagnosis of the mass was ependymoma or metastasis&#46;</p>"
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          "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Hematoxylin&#8211;eosin staining of a section from the intramedullary mass &#40;H&#38;E&#44; 40&#215;&#41;&#59; &#40;B&#41; High magnification &#40;H&#38;E&#44; 100&#215;&#41; view of the tumor with large pleomorphic epithelial cells and extensive necrosis&#59; &#40;C&#41; tumor cells shows focal nuclear positivity with CDX2 stain &#40;IHC&#44; 100&#215;&#41;&#59; &#40;D&#41; Tumor cells positive for cytokeratin &#40;IHC&#44; 100&#215;&#41;&#46;</p>"
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                            0 => "A&#46; Raco"
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                          "etal" => true
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                            0 => "I&#46;E&#46; Sandalcioglu"
                            1 => "T&#46; Gasser"
                            2 => "S&#46; Asgari"
                            3 => "A&#46; Lazorisak"
                            4 => "T&#46; Engelhorn"
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                      "titulo" => "Fluorescein-guided resection of intramedullary spinal cord tumors&#58; results from a preliminary&#44; multicentric&#44; retrospective study"
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                          "etal" => true
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                            1 => "C&#46; Cavallo"
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                            3 => "M&#46;O&#46; Ak&#231;akaya"
                            4 => "C&#46; de Laurentis"
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