Taiwanese Journal of Obstetrics & Gynecology 54 (2015) 336e338

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Research Letter

Urinothorax: An unusual complication of an oncologic gynecological surgery ~ izares Oliver a, b, Raquel Garcia-Simon a, b, *, Jesus Hergueta Herrera a, b, Silvia Can lez a, b, c Fernando Roldan Rivas a, b, Ernesto Fabre Gonza a b c

Department of Obstetrics and Gynaecology, Clinico Lozano Blesa Hospital, Zaragoza, Spain Instituto Aragon es de la Salud, Zaragoza, Spain Obstetrics and Gynaecology Department, University of Zaragoza, Zaragoza, Spain

a r t i c l e i n f o Article history: Accepted 6 August 2014

Urinothorax was first described in 1968 by Corriere et al [1] as the presence of urine in the pleural cavity due to retroperitoneal leakage of accumulated urine, known as a urinoma. Urine enters into the pleural space via diaphragmatic lymphatic or through an anatomical defect in the diaphragm [2,3]. Although classically it was defined as a transudate, on rare occasions it can present as an exudate [4,5]. Urinothorax often goes undiagnosed, but needs to be considered in patients with a pleural effusion of unknown etiology that is also associated with an obstructive uropathy. This unusual type of pleural effusion is frequently unilateral and ipsilateral to the obstructed urinary tract [6]. Other causes of urinothorax include trauma, surgical injury, malignancy of the urinary tract, renal transplantation, and kidney biopsy [7]. The importance of recognizing this entity lies in the fact that the condition is completely reversible when treatment is specifically directed to the correction of the primary cause and drainage of the urinoma. We report a case of a 56-year-old woman who developed a urinothorax following hysterectomy for endometrial cancer. A 56year-old female, with an unremarkable past medical history, consulted us because of postmenopausal bleeding. On physical examination we confirmed uterine bleeding. Ultrasonography demonstrated an 8 mm endometrium, which was biopsied, revealing endometrial adenocarcinoma. The extension study excluded the existence of a secondary disease. The patient underwent an uneventful abdominal hysterectomy and bilateral

* Corresponding author. Hospital Clinico Universitario Lozano Blesa, C/San Juan Bosco 15, 50009 Zaragoza, Spain. E-mail address: [email protected] (R. Garcia-Simon).

oophorectomy. In order to determine whether to practice a bilateral pelvic lymph node sampling or not, during surgery we asked the pathology service to inform us about the tumor infiltration in the myometrium. They confirmed to us that the tumor penetrated less than one-third of the thickness of the myometrium so we decided not to carry out the pelvic lymph node sampling. However, 36 hours after surgery the patient complained of dyspnea and epigastric pain. Physical examination revealed low oxygen saturation and absent breath sounds in the entire right hemithorax, consistent with a massive pleural effusion. Chest radiography demonstrated a massive right-sided pleural effusion (Figure 1). Due to the patient's recent surgery and diagnosis of endometrial cancer, she was treated with low molecular weight heparin and furosemide. A computerized tomography (CT) scan of the chest confirmed the presence of a right-sided pleural effusion. It also demonstrated the existence of perihepatic and perisplenic fluid collections in the more caudal images (Figure 2). There was no radiographic evidence of pulmonary embolism. Unfortunately, despite diuretic therapy with furosemide, the patient complained of increasing dyspnea and abdominal pain. She was transferred to the intensive care unit where serum electrolytes and liver function tests were normal, apart from a serum creatinine of 2.5 mg/dL. A repeat CT scan of the pelvis and abdomen demonstrated significant perihepatic, perisplenic, perigastric, and pelvic fluid. In addition, it showed bilateral pelvicalyceal dilatation due to an obstruction at the level of the left ureter, and the previously visualized right-sided urinoma. A chest tube inserted into the right fifth intercostal space drained 5800 cc of hematic pleural fluid over 12 hours. An analysis of the pleural fluid revealed a lactate dehydrogenase of 123 U/L, glucose of 109 mg/dL, pH of 7.24, urea of 0.37 mg/dL, and creatinine of 3.4 mg/dL. The ratio of the pleural fluid-to-serum creatinine was greater than one (1.48 mg/dL). Cultures from blood, urine, and pleural fluid were sterile. Cytology from the pleural fluid did not identify any metastatic cells from the patient's endometrial cancer. Re-operation confirmed partial obstruction at the level of the left distal ureter, probably caused during the abdominal hysterectomy during which we sutured the left uterine vessel, and a right-

http://dx.doi.org/10.1016/j.tjog.2014.08.007 1028-4559/Copyright © 2015, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. All rights reserved.

R. Garcia-Simon et al. / Taiwanese Journal of Obstetrics & Gynecology 54 (2015) 336e338

Figure 1. Chest radiograph demonstrating a right-sided pleural effusion.

sided urinoma. The urinoma was drained by percutaneous puncture. A double J catheter was placed to release the left ureteral obstruction. Following urological surgery, the patient's dyspnea and abdominal pain disappeared and serum creatinine levels returned to normal (0.8 mg/dL) within 24 hours. In consecutive radiographs, the right-sided pleural effusion progressively resolved. Ten days following hysterectomy, the patient was discharged from hospital. Pathological analysis of the resected endometrium 40 days after surgery reported an endometrial adenocarcinoma type 1, deration Internationale de Gyne cologie et d’Obste trique (FIGO) 1, Fe with infiltration of less than one-third of the thickness of the myometrium and absence of lymphatic or vascular invasion (TNM T1bNxMx). Therefore, the patient's disease was considered to be in complete remission, without the need for additional therapy. However, during physical examination, the patient was noted to have an output of urine from the vagina. A CT scan of the pelvis determined the existence of a small vesico-vaginal fistula. The fistula was sutured closed and the patient has remained asymptomatic since the closure of the fistula. Urinothorax is a rare cause of pleural effusion. When urine is found in the pleural space, it is usually in the setting of an obstructive uropathy [8,9]. The availability of sophisticated imaging techniques has increased awareness of this entity. Urinothorax occurs as a result of leakage of urine into the retroperitoneal space, with formation of a urinoma [8]. Urine reaches the pleural space by ascending through thoracic lymphatics or by direct transdiaphragmatic passage into the pleural cavity [10]. Although urinothorax has usually been reported as a complication of obstructive uropathy with hydronephrosis, it may also develop in association with malignancy, renal biopsy or transplantation, posterior urethral valves, percutaneous renal

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interventions, retroperitoneal fibrosis, or blunt renal trauma [2,3,11e13]. We have presented a case report in which the origin of urinothorax was not a urological disease. Urinothorax in our patient arose as a consequence of a gynecological disease, endometrial cancer, and its treatment with hysterectomy and oophorectomy. Only one other publication has reported a urinothorax that arose subsequent to a gynecological procedure. But, in that case, the pathology was nonmalignant. Urinothorax developed following a hysterectomy that was performed because of pelvic pain in a premenstrual woman [14]. Most cases of urinothorax that have been reported were unilateral and ipsilateral to the urinoma and the obstructive uropathy. However, a few publications have reported rare cases of urinothorax that were bilateral or contralateral to the urinothorax [9,15e18]. In our case, both the urinothorax and the urinoma were on the patient's right side. But, the obstructive uropathy was on the patient's left side. The left ureter was partially obstructed by sutures that were used to close the vaginal fornix after hysterectomy. The most commonly used radiographic examination for the detection of urinothorax is a chest radiograph. CT imaging of the pelvis and thorax is also indispensable for detecting pleural effusions and an underlying urinoma. The definitive diagnosis of an urinothorax is by thoracocentesis [15]. According to Light's criteria, pleural fluid is usually a transudate, characterized by low glucose, low pH, elevated lactate dehydrogenase, and low protein concentration [6,8,10]. However, the most important biochemical parameter is the ratio of pleural fluid-to-serum creatinine ratio, which is greater than one, and in most cases is greater than 10 [4,7,11,15,18,19]. Microbiological and cytopathological studies should be negative. In our case, the ratio of pleural fluid-to-serum creatinine was 1.48. Both routine microbiology cultures and cytology from pleural fluid were negative. Urinothorax management involves treating the cause of the urine leak and resolving urinary obstruction [11,20]. In conclusion, the diagnosis of urinothorax requires a high index of suspicion and should be considered whenever pleural effusion is found in the setting of urinary tract obstruction, or following a urologic or gynecologic procedure. Conflict of interest The authors declare that they have no conflicts of interest. Acknowledgments The authors would like to thank the following individuals: C. Leal and C. Gonzalez Moran for their contribution to the oncology gynecology section, A. Sanz for his help in promulgating the existence of this problem in San Sebastian, the staff who cared for this patient, and the Moya's brother and their family for being so patient. References

Figure 2. Computerized tomography which confirmed the presence of a right-sided pleural effusion.

[1] Corriere Jr JN, Miller WT, Murphy JJ. Hydronephrosis as a cause of pleural effusion. Radiology 1968;90:79e84. [2] Carcillo Jr J, Salcedo JR. Urinothorax as a manifestation of nondilated obstructive uropathy following renal transplantation. Am J Kidney Dis 1985;5: 211e3. [3] Kinasewitz GT. Transudative effusions. Eur Respir J 1997;10:714e8. [4] Garcia-Pachon E, Padilla-Navas I. Urinothorax: case report and review of the literature with emphasis on biochemical diagnosis. Respiration 2004;71: 533e6. [5] Mora RB, Silvente CM, Nieto JM, Cuervo MA. Urinothorax: presentation of a new case as pleural exudate. South Med J 2010;103:931e3. [6] Light RW. Transudative pleural effusions. Pleural diseases. 3rd ed. Baltimore: Williams & Wilkins; 1995.

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[7] Garcia-Pachon E, Romero S. Urinothorax: a new approach. Curr Opin Pulm Med 2006;12:259e63. [8] Light RW. Update: management of the difficult to diagnose pleural effusion. Clin Pulm Med 2003;10:39e46. [9] Sahn SA. Pleural effusions of extravascular origin. Clin Chest Med 2006;27: 285e308. [10] Light RW. The undiagnosed pleural effusion. Clin Chest Med 2006;27:309e19. [11] Salcedo JR. Urinothorax: Report of 4 cases and review of the literature. J Urol 1986;135:805e8. [12] O'Donnelli A, Schoenberger C, Weiner J, Tsou E. Pulmonary complications of percutaneous nephrostomy and kidney stone extraction. South Med J 1988;81:1002e5. [13] Nusser RA, Culhane RH. Recurrent transudative effusion with an abdominal mass. Chest 1986;90:263e4.

[14] Amro O, Webb-Smith F, Sunderji S. Urinothorax: a rare complication of total abdominal hysterectomy. Obstet Gynecol 2009;114:482e4. [15] Handa A, Agarwal R, Aggarwal AN. Urinothorax: an unusual cause of pleural effusion. Singapore Med J 2007;48:289e92. [16] Ralston MD, Wilkinson RH. Bilateral urinothorax identified by technetium99m DPTA renal imaging. J Nucl Med 1986;27:56e9. [17] Chanatry BJ, Gettinger A. Progressive respiratory insufficiency after cesarean section. Crit Care Med 1995;23:204e7. [18] Stark DD, Shanes JG, Baron RL, Koch DD. Biochemical features of urinothorax. Arch Intern Med 1982;142:1509e11. uzülgen IK, Og uzülgen AI, Sinik Z, Ko €ktürk O, Ekim N, Karaog lan U. An [19] Og unusual cause of urinothorax. Respiration 2002;69:273e4. [20] Maskell NA, Butland RJA. BTS guidelines for the investigation of a unilateral pleural effusion in adults. Thorax 2003;58:1008e17.

Urinothorax: An unusual complication of an oncologic gynecological surgery.

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