Large retroperitoneal mass during pregnancy

Giménez-Bachs, J.M.; Villar-García, M.; Fernández-López, A.; Salinas-Sánchez, A.S.

doi:10.1016/j.acuroe.2024.01.004

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"apellidos" => "Giménez-Bachs" "email" => array:1 [ 0 => "gbjosem@sescam.jccm.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "M." "apellidos" => "Villar-García" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Fernández-López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "A.S." "apellidos" => "Salinas-Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Urología, Complejo Hospitalario Universitario de Albacete, Facultad de Medicina de Albacete, Universidad de Castilla-La Mancha, Albacete, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Radiología, Complejo Hospitalario Universitario de Albacete, Facultad de Medicina de Albacete, Universidad de Castilla-La Mancha, Albacete, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Masa retroperitoneal grande durante el embarazo" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 958 "Ancho" => 1340 "Tamanyo" => 133582 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0180" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "Abdominal ultrasound showing well delimitated complex cystic mass with echogenic homogeneous content and a high echoic mural nodule." ] ] ] "textoCompleto" => "We present the case of a retroperitoneal tumor in a pregnant woman, with atypical clinical presentation.A 34-year-old woman in the 25th week of pregnancy consulted for sudden right renal colic pain. Physical examination was normal. The laboratory tests showed anemia (hemoglobin 8.9g/dl, previously 11.8g/dl a month before). Abdominal ultrasound revealed a large complex cystic mass (20cm), well-circumscribed, and filled with low level internal echoes with some internal anechoic cystic areas (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).<elsevierMultimedia ident="fig0005"></elsevierMultimedia>Magnetic resonance imaging was performed, showing a 20-cm retroperitoneal multicystic tumor containing solid mural nodules, located along the right paracolic gutter, displacing the right colon medially. The tumor was well circumscribed, with thin-walled capsule (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), and compresses the right ureter generating moderate renal ectasia.<elsevierMultimedia ident="fig0010"></elsevierMultimedia>Because of the hemoglobin level reduction, surgical exeresis was decided, a large non-adhered cystic mass was removed without disruption of it. Grossly, the tumor was rounded, well-delimited and encapsulated, weighing 2450g and measuring 23.0cm in maximum diameter. It was widely cystic with liquid content of hemorrhagic aspect, showing a thin fibrous wall with several nodules of microcystic appearance and grayish coloration, the largest of them being 5cm (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).<elsevierMultimedia ident="fig0015"></elsevierMultimedia>Microscopically, the fibrous walls of the cystic spaces were lined by flattened to cuboidal, intestinal-type mucinous epithelium. No ovarian-like stroma or stromal invasion was seen. The solid nodule of hemorrhagic/necrotic aspect described above is formed by atypical epithelioid and spindle cells with abundant mitosis, expressing positivity for AE1/AE3, CK8/18 and vimentin. 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.The patient delivered at term without complications. Two years after surgery there is no signs of recurrence.Primary retroperitoneal mucinous cystic neoplasms (PRMCNs) are very uncommon epithelial tumors arising in the retroperitoneal space. Although they resemble ovarian mucinous neoplasms, PRMCNs happen in women with normal ovaries.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a>These tumors occur almost exclusively in women and usually present in premenopausal females, though they have also been described in postmenopausal females and men.<a class="elsevierStyleCrossRefs" href="#bib0015">3,4</a> Eight cases exist in the literature of PRMCNs diagnosed in pregnant women.<a class="elsevierStyleCrossRef" href="#bib0005">1</a>Wolf et al. identified 144 cases in the world literature between 1975 y 2015. Most (89%) of the cases occurred in women. The median age of all patients was 42 years old. Most of the tumors were malignant (53%), followed by benign (30%) and borderline lesions (17%).<a class="elsevierStyleCrossRef" href="#bib0020">4</a> Therefore, the incidence of the latter is rare.As with other retroperitoneal neoplasm, clinical symptoms and signs are very mild and nonspecific, even at large tumor dimensions.<a class="elsevierStyleCrossRef" href="#bib0020">4</a>There are not pathognomonic imaging features of PRMCN. This tumor usually manifests as a homogeneous, unilocular cystic mass; yet some have a solid component or a mural nodule.<a class="elsevierStyleCrossRef" href="#bib0025">5</a> PRMCN is found almost exclusively in the lateral retroperitoneal spaces.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> Retroperitoneal location can be difficult to establish and may sometimes only be inferred from the displacement of the ureters, kidneys and colon. Differentiating PRMCN from other retroperitoneal cystic lesions as cystic mesothelioma or cystic lymphangioma is difficult. Therefore, exploratory laparotomy with complete excision of the lesion is usually indicated for both diagnosis and treatment.<a class="elsevierStyleCrossRef" href="#bib0025">5</a>Pathogenesis of PRMCNs is poorly understood because epithelial cells do not normally exist in the retroperitoneum. There are four general theories about this: (1) ectopic ovarian tissue, (2) cystic teratomas, (3) remnants of the urogenital tract, and (4) coelomic epithelial metaplasia. Last theory is the most widely accepted which suggests tumors arise from invagination of the peritoneal mesothelial layer, with subsequent mucinous metaplasia and cyst formation.<a class="elsevierStyleCrossRefs" href="#bib0005">1,4,6</a>PRMCNs share histologic resemblance with mucinous ovarian tumors and can be classified into 3 types: benign mucinous cystadenoma, borderline mucinous cystadenoma, malignant mucinous cystadenocarcinoma.<a class="elsevierStyleCrossRef" href="#bib0005">1</a>PRMCN should be treated in the same way as a primary ovarian tumor of similar grade and stage. The treatment of choice is complete surgical removal of the tumor, being mandatory to avoid its rupture. Adjuvant chemotherapy and radiotherapy should be discussed with each patient individually.<a class="elsevierStyleCrossRefs" href="#bib0020">4,7</a>PRMCNs seem to be non-aggressive neoplasms but the prognosis cannot be determined. Survival in patients with benign and borderline cystadenoma is excellent.<a class="elsevierStyleCrossRefs" href="#bib0005">1,4,6</a> PRMCNs with malignant mural nodules are associated with worse prognosis.<a class="elsevierStyleCrossRef" href="#bib0040">8</a> The small number of registered cases, the insufficient amount of surveillance data, and mainly, the inability to identify patients at a high risk of recurrence support the need for effective registration and further study of these rare tumors.<a class="elsevierStyleCrossRef" href="#bib0045">9</a>Primary mucinous cystadenoma is a rare retroperitoneal cystic neoplasm with female predilection. Retroperitoneal cystic masses, which arise within the retroperitoneal space but outside the major organs within that compartment are uncommon, that is why the diagnosis of primary retroperitoneal mucinous cystic neoplasm is usually delayed." 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Actas Urológicas Españolas is an international journal dedicated to urological diseases and renal transplant. It has been the official publication of the Spanish Urology Association since 1974 and of the American Urology Confederation since 2008. Its articles cover all aspects related to urology. Actas Urológicas Españolas, governed by the peer review system (double blinded), is published online in Spanish and English. Consequently, manuscripts may be sent in Spanish or English and bidirectional free cost translation will be provided. It has high visibility and accessibility and is found in the most important data bases, Medline/Pubmed, Emabase, JCR, Ïndice Médico Español, Índice bibliográfico Español en Ciencias de la Salud (IBECS), Cuiden, Dialnet, Hinari, Scopus, Google Scholar.

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