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Letter to the Editor
Epstein–Barr virus associated smooth-muscle tumor
Tumor de músculo liso asociado al virus de Epstein-Barr
Anna Aguilara, Tulio Mateo Silvab, Arnau Monfortec,
Corresponding author
arnau.monforte@vallhebron.cat

Corresponding author.
a Department of Internal Medicine, Hospital Universitari Vall d’Hebron, Spain
b Department of Pathology, Hospital Universitari Vall d’Hebron, Spain
c Department of Infectious Diseases, Hospital Universitari Vall d’Hebron, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 30-year-old man attended the emergency room presenting a ten-day course of progressive paresthesia from toes to pelvis&#44; inability to walk and urinate in the last 48<span class="elsevierStyleHsp" style=""></span>h&#46; He was born in Pakistan and he was diagnosed with HIV infection in the past&#46; He presented a disseminated <span class="elsevierStyleItalic">Mycobacterium avium complex</span> &#40;MAC&#41; infection which was treated&#44; but he abandoned both antimycobacterial and antirretroviral treatment &#40;ART&#41;&#44; until two weeks before the present episode&#46; He was asymptomatic with 30 CD4 T-lymphocyte cells&#47;mm<span class="elsevierStyleSup">3</span> and three million of HIV RNA copies&#47;ml&#44; and he restarted ART&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">At the time of admission the physical examination revealed a urinary retention&#44; a motor balance of 3 out of 5 in both legs and hypoesthesia up to T8 level&#46; Urgent magnetic resonance imaging was performed under the suspicion of spinal cord compression &#40;AIS D&#41;&#44; showing two epidural lesions&#44; one of them with severe compressive effect over the spinal cord at T6-T7 level&#44; and the other one at T4 level&#46; Intravenous steroids bolus at an initial dose of 500<span class="elsevierStyleHsp" style=""></span>mg of methylprednisolone per day were started under a suspected neoplastic etiology&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">In the following 24<span class="elsevierStyleHsp" style=""></span>h the patient showed improvement in leg strength&#44; however&#44; the symptoms worsened after 48<span class="elsevierStyleHsp" style=""></span>h with decrease in motor balance to 1 over 5 &#40;AIS C&#41; and progression of sensory impairment up to T6&#46; The patient was taken for urgent decompressive surgery with a bilateral T7 laminectomy and T4 left-hemilaminectomy&#44; visualizing a granulomatous tissue with low level of vascularization&#46; The patient presented a very good postoperative course and started rehabilitation&#46; The pathologist informed a moderately cellular proliferation composed of medium-sized elongated nuclei with fine granular chromatin and round nuclei with granular chromatin&#44; and eosinophilic-pale elongated cytoplasm&#46; There was diffuse EBV-encoded small RNA &#40;EBER&#41; expression&#44; which was conclusive of a smooth-muscle tumor associated with EBV &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The result was discussed with Oncology and Radiotherapy in committee&#44; precluding the need for a systemic treatment other than immune recovery&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Smooth-muscle tumor &#40;SMT&#41; associated with Epstein&#8211;Barr virus &#40;EBV&#41; was firstly reported in 1995 in young PWH&#46; Demonstration of EBV infection by in situ hybridization within smooth-muscle tumor cells remains critical for the final diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Its pathogenesis is unknown&#44; but some hypotheses may suggest EBV infection of mesenchymal cells&#46; There the EBV may have an oncogenic role&#44; as has been described in B lymphocytes &#40;lymphoma&#41; and epithelial cells &#40;nasopharyngeal carcinoma&#41;&#46; The multicentricity of these tumors appears to be related to various EBV infectious events rather than a malignant dissemination of the tumor&#46; Therefore they are considered non-malignant and they have better prognosis than other aggressive mesenchymal tumors&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Surgery could be necessary to control compressive symptoms&#44; but the recovery of immunity is the most effective measure to control viral replication and tumor growth&#44; even without additional systemic treatment&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3&#44;4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">This case shows an emergency due to this non-malignant tumor&#44; which is surprising regarding the rapid onset of the symptoms&#46; In advanced HIV infection&#44; CD4 T-lymphocytes decrease which can drive to opportunistic infections and replication of some potentially oncogenic viruses&#46; However&#44; after the reintroduction of ART a rapid decrease in HIV-RNA and increase in CD4 T-lymphocytes count could produce an inflammatory cascade known as inmune reconstitution inflammatory syndrome &#40;IRIS&#41; in locations where there is an infection&#46; Such presentation has been described in many mycobacterial&#44; fungal and viral infections in advanced inmunosupresion&#44; including human herpesvirus-8 &#40;HHV-8&#41; and EBV&#46; Therefore&#44; the EBV involvement in this tumor could explain its rapid occurrence after the reintroduction of ART due to an associated IRIS&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The differential diagnosis was challenging&#46; A thoracic epidural lesion in advanced HIV infection suggested an infectious or a malignant cause of a spinal cord compression&#46; The absence of vertebral involvement was inconsistent with tuberculosis but the patient had already been diagnosed in the past with a disseminated MAC infection&#44; which was not completely treated&#46; Disseminated neoplastic disease is a common cause of thoracic spinal cord compression&#44; being lymphoproliferative disorders well described in advanced HIV infection&#44; mainly related to EBV infection&#46; In this case&#44; a surgical biopsy revealed the diagnosis&#44; and the patient presented a favorable clinical outcome&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion&#44; SMT should be suspected in a severely immunocompromised host in association with EBV replication&#44; as it could potentially be cured by restoration of the immunity&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0040" class="elsevierStylePara elsevierViewall">Written informed consent was obtained from the patient for publication of this case and accompanying images&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0045" class="elsevierStylePara elsevierViewall">This work has not received any funding&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare they have no conflict of interests&#46;</p></span></span>"
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ISSN: 00257753
Original language: English
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