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Case and Research Letter
Large retroperitoneal mass during pregnancy
Masa retroperitoneal grande durante el embarazo
J.M. Giménez-Bachsa,
Corresponding author
gbjosem@sescam.jccm.es

Corresponding author.
, M. Villar-Garcíab, A. Fernández-Lópezb, A.S. Salinas-Sáncheza
a Servicio de Urología, Complejo Hospitalario Universitario de Albacete, Facultad de Medicina de Albacete, Universidad de Castilla-La Mancha, Albacete, Spain
b Servicio de Radiología, Complejo Hospitalario Universitario de Albacete, Facultad de Medicina de Albacete, Universidad de Castilla-La Mancha, Albacete, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present the case of a retroperitoneal tumor in a pregnant woman&#44; with atypical clinical presentation&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 34-year-old woman in the 25th week of pregnancy consulted for sudden right renal colic pain&#46; Physical examination was normal&#46; The laboratory tests showed anemia &#40;hemoglobin 8&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;dl&#44; previously 11&#46;8<span class="elsevierStyleHsp" style=""></span>g&#47;dl a month before&#41;&#46; Abdominal ultrasound revealed a large complex cystic mass &#40;20<span class="elsevierStyleHsp" style=""></span>cm&#41;&#44; well-circumscribed&#44; and filled with low level internal echoes with some internal anechoic cystic areas &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Magnetic resonance imaging was performed&#44; showing a 20-cm retroperitoneal multicystic tumor containing solid mural nodules&#44; located along the right paracolic gutter&#44; displacing the right colon medially&#46; The tumor was well circumscribed&#44; with thin-walled capsule &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#44; and compresses the right ureter generating moderate renal ectasia&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Because of the hemoglobin level reduction&#44; surgical exeresis was decided&#44; a large non-adhered cystic mass was removed without disruption of it&#46; Grossly&#44; the tumor was rounded&#44; well-delimited and encapsulated&#44; weighing 2450<span class="elsevierStyleHsp" style=""></span>g and measuring 23&#46;0<span class="elsevierStyleHsp" style=""></span>cm in maximum diameter&#46; It was widely cystic with liquid content of hemorrhagic aspect&#44; showing a thin fibrous wall with several nodules of microcystic appearance and grayish coloration&#44; the largest of them being 5<span class="elsevierStyleHsp" style=""></span>cm &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Microscopically&#44; the fibrous walls of the cystic spaces were lined by flattened to cuboidal&#44; intestinal-type mucinous epithelium&#46; No ovarian-like stroma or stromal invasion was seen&#46; The solid nodule of hemorrhagic&#47;necrotic aspect described above is formed by atypical epithelioid and spindle cells with abundant mitosis&#44; expressing positivity for AE1&#47;AE3&#44; CK8&#47;18 and vimentin&#46; Based on the morphology and immunophenotype the diagnosis of a primary retroperitoneal mucinous neoplasm of borderline malignancy with foci of intraepithelial carcinoma and mural nodule of sarcomatoid carcinoma was made&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The patient delivered at term without complications&#46; Two years after surgery there is no signs of recurrence&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Primary retroperitoneal mucinous cystic neoplasms &#40;PRMCNs&#41; are very uncommon epithelial tumors arising in the retroperitoneal space&#46; Although they resemble ovarian mucinous neoplasms&#44; PRMCNs happen in women with normal ovaries&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">These tumors occur almost exclusively in women and usually present in premenopausal females&#44; though they have also been described in postmenopausal females and men&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> Eight cases exist in the literature of PRMCNs diagnosed in pregnant women&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Wolf et al&#46; identified 144 cases in the world literature between 1975 y 2015&#46; Most &#40;89&#37;&#41; of the cases occurred in women&#46; The median age of all patients was 42 years old&#46; Most of the tumors were malignant &#40;53&#37;&#41;&#44; followed by benign &#40;30&#37;&#41; and borderline lesions &#40;17&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Therefore&#44; the incidence of the latter is rare&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">As with other retroperitoneal neoplasm&#44; clinical symptoms and signs are very mild and nonspecific&#44; even at large tumor dimensions&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">There are not pathognomonic imaging features of PRMCN&#46; This tumor usually manifests as a homogeneous&#44; unilocular cystic mass&#59; yet some have a solid component or a mural nodule&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> PRMCN is found almost exclusively in the lateral retroperitoneal spaces&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Retroperitoneal location can be difficult to establish and may sometimes only be inferred from the displacement of the ureters&#44; kidneys and colon&#46; Differentiating PRMCN from other retroperitoneal cystic lesions as cystic mesothelioma or cystic lymphangioma is difficult&#46; Therefore&#44; exploratory laparotomy with complete excision of the lesion is usually indicated for both diagnosis and treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Pathogenesis of PRMCNs is poorly understood because epithelial cells do not normally exist in the retroperitoneum&#46; There are four general theories about this&#58; &#40;1&#41; ectopic ovarian tissue&#44; &#40;2&#41; cystic teratomas&#44; &#40;3&#41; remnants of the urogenital tract&#44; and &#40;4&#41; coelomic epithelial metaplasia&#46; Last theory is the most widely accepted which suggests tumors arise from invagination of the peritoneal mesothelial layer&#44; with subsequent mucinous metaplasia and cyst formation&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4&#44;6</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">PRMCNs share histologic resemblance with mucinous ovarian tumors and can be classified into 3 types&#58; benign mucinous cystadenoma&#44; borderline mucinous cystadenoma&#44; malignant mucinous cystadenocarcinoma&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">PRMCN should be treated in the same way as a primary ovarian tumor of similar grade and stage&#46; The treatment of choice is complete surgical removal of the tumor&#44; being mandatory to avoid its rupture&#46; Adjuvant chemotherapy and radiotherapy should be discussed with each patient individually&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;7</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">PRMCNs seem to be non-aggressive neoplasms but the prognosis cannot be determined&#46; Survival in patients with benign and borderline cystadenoma is excellent&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4&#44;6</span></a> PRMCNs with malignant mural nodules are associated with worse prognosis&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> The small number of registered cases&#44; the insufficient amount of surveillance data&#44; and mainly&#44; the inability to identify patients at a high risk of recurrence support the need for effective registration and further study of these rare tumors&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Primary mucinous cystadenoma is a rare retroperitoneal cystic neoplasm with female predilection&#46; Retroperitoneal cystic masses&#44; which arise within the retroperitoneal space but outside the major organs within that compartment are uncommon&#44; that is why the diagnosis of primary retroperitoneal mucinous cystic neoplasm is usually delayed&#46;</p></span>"
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