Int J Case Rep Images 2021;12:101219Z01PS2021. Soares et al. 1 www.ijcasereportsandimages.com CASE REPORT PEER REVIEWED | OPEN ACCESS Vaginal ureterocele: Rare finding in complicated pyelonephritis Paulo Silveira Campos Soares, Vinícius Henrique Almeida Guimarães, Ana Letícia Freitas-Silva, André Rezek Rodrigues, Vitor Augusto Alves Cobo, Rolf Carvalho Lara, Paulo Ricardo Monti ABSTRACT unusual and challenging anatomical findings, such as the pyelocalyceal bilateral duplicity and a rare infected Introduction: Pyelonephritis is a urinary tract infection ectopic ureterocele with vagina implantation. These that can frequently be complicated by anatomical challenging findings required transvaginal drainage abnormalities such as in ectopic ureteroceles, pathologies and heminephrectomy, what shows that anatomical which are rare in the literature. rare abnormalities can be hidden in a typical clinical presentation of pyelonephritis and demand an intense Case Report: A 23-year-old woman with a clinical radiologic investigation and unusual treatments. history, physical and laboratory examinations compatible with pyelonephritis was admitted. Ultrasonography Keywords: Nephrectomy, Pyelonephritis, Ureterocele revealed a ureterocele and hydronephrosis of the upper collecting system of the left kidney. We evoked a How to cite this article complicated pyelonephritis and the patient underwent a computed tomography (CT) scan that confirmed bilateral Soares PSC, Guimarães VHA, Freitas-Silva AL, pyelocalyceal and ureteral duplicity, pyonephrosis, Rodrigues AR, Cobo VAA, Lara RC, Monti PR. and implantation of the upper ureter of the left kidney Vaginal ureterocele: Rare finding in complicated into the vaginal dome, forming a left ureterocele. The pyelonephritis. Int J Case Rep Images patient was placed on antibiotics and the ureterocele 2021;12:101219Z01PS2021. was drained through the vaginal canal. After twelve days of hospitalization, she underwent an open partial nephrectomy of the upper portion of the left kidney. On Article ID: 101219Z01PS2021 the third postoperative day, she was asymptomatic and was discharged. ********* Conclusion: Despite the typical clinical picture of complicated pyelonephritis, this case stands out for the doi: 10.5348/101219Z01PS2021CR Paulo Silveira Campos Soares1, Vinícius Henrique Almeida Guimarães2, Ana Letícia Freitas-Silva2, André Rezek Rodri- INTRODUCTION gues1, Vitor Augusto Alves Cobo1, Rolf Carvalho Lara1, Paulo Ricardo Monti1 Pyelonephritis is a frequent urinary tract infection, Affiliations: 1Department of Urology, Clinical Hospital of the with global estimates ranging from 10.5 million to 25.9 Federal University of the Triangulo Mineiro, Uberaba, Brazil; million cases annually. Each year, pyelonephritis causes 2Institute of Health Sciences, Federal University of Triangulo 4000 deaths in the United States and its treatment costs Mineiro, Uberaba, Brazil. USD 2 billion [1]. Corresponding Author: Paulo Silveira Campos Soares, However, despite being common in the population Department of Urology, Clinical Hospital of the Federal and well known by the medical community, urinary tract University of the Triangulo Mineiro, Uberaba, Brazil; infections, especially pyelonephritis, can be challenging Email: [email protected] in some cases with an underlying anomalous anatomy because these rare anomalies can make the diagnosis and treatment substantially more difficult [2]. Received: 17 February 2021 Accepted: 24 April 2021 Among these rare anomalies, we highlight ureterocele, Published: 17 May 2021 a congenital condition formed by the dilatation of International Journal of Case Reports and Images, Vol. 12, 2021. ISSN: 0976-3198 Int J Case Rep Images 2021;12:101219Z01PS2021. Soares et al. 2 www.ijcasereportsandimages.com the distal segment of the ureter. It occurs in 1 in every 5000 to 12,000 births and is frequently diagnosed in childhood. Thus, in adults, this condition usually presents as an incidental radiological finding and an orthotopic insertion. When uncommonly symptomatic, ureterocele is related to lithiasis or its own prolapse [3, 4]. However, we describe a rare case of an adult woman diagnosed with complicated pyelonephritis who was found to have several rare anatomical abnormalities, which were challenging to treat. CASE REPORT A 23-year-old married woman was admitted to the hospital for a 5-day progressive, severe colicky pain in the suprapubic region, irradiating to the left labia majora. This pain was associated with dysuria, fever, and hyporexia. Two days earlier, the pain began to irradiate to the left flank with a progressive increase in intensity and concentrated diuresis with a fetid odor. The patient’s general condition was stable, with intense pain on deep palpation of the left hemiabdomen and Giordano's sign present on the left. She was tachycardic (HR = 110 bpm), eupneic, dehydrated (+1/+4), pale (+1/+4), febrile (T = 37.9°C), and hypotensive (BP = 90/50 mmHg). Laboratory tests confirmed an acute infectious process with marked neutrophilia (32,000 leukocytes with 29% neutrophils) and a positive urine culture for multi-sensitive Escherichia coli. Ultrasonography of the kidneys (Figure 1), urinary tract, and pelvis showed a duplicated left kidney collecting system, hydronephrosis of the upper collecting system with probable pyogenic content, and a left ureterocele with thickened content. To better evaluate the case, the patient underwent contrast-enhanced computed Figure 1(A–C): Ultrasonography showing the left kidney, left tomography (CT) of the whole abdomen and pelvis, kidney pelvis, and ureterocele in the left ureter. We can see the hydronephrosis of the upper collecting system with probable which demonstrated bilateral pyelocalyceal and ureteral pyogenic content, pyelocaliceal, and ureteral duplicity and the duplicity, left pyonephrosis, and anomalous implantation ureterocele with thickened content. of the upper left ureter into the vagina, forming a ureterocele with a probable pyogenic content (Figure 2). Tapering of the renal parenchyma of the left kidney's upper unit was also associated with hydronephrosis, without contrast uptake. The magnetic resonance and radioisotope renography weren’t available in the moment. Speculum examination confirmed the presence of a ureteral meatus inserted into the vagina (Figure 3). Thus, we could schematically represent the main anatomical alterations of the patient (Figure 4). She was started on ceftriaxone 1 g every 12 h, and ureterocele drainage was performed through an intravaginal scalpel incision in the wall facing the ureterocele, with drainage of approximately 100 mL of purulent content. After 12 days of hospitalization, the clinical picture had significantly improved and an open upper left polar nephrectomy with closure of the ureteral Figure 2(A–B): Contrast-enhanced computed tomography in coronal section showing pyonephrosis, ureteral dilatation with stump was performed. The patient evolved satisfactorily a tortuous pathway, and a ureterocele and vaginal insertion of and was discharged on the third postoperative day. the ureter of the upper unit of the left kidney. International Journal of Case Reports and Images, Vol. 12, 2021. ISSN: 0976-3198 Int J Case Rep Images 2021;12:101219Z01PS2021. Soares et al. 3 www.ijcasereportsandimages.com DISCUSSION The most common pyelonephritis symptoms are systemic inflammation (fever, chills, headache, sweating, and malaise), bladder inflammation (dysuria, urgency, altered urination, and polyuria), gastrointestinal symptoms (anorexia, hyporexia, nausea, vomiting, and abdominal pain), and renal pain symptoms (pain in the lumbar region and flank ipsilateral to the affected kidney) [1, 2]. In this case, we immediately suspected pyelonephritis based on clinical symptoms and laboratory tests [1, 5]. However, this case is peculiar due to the infrequent findings on imaging exams. Bilateral duplication of the collecting system occurs in 0.07–0.11% of the population [6] and ureterocele occurs in 1 among 5000 to 12,000 autopsies [7]. However, the rarest finding is the insertion of the left ureter into the vagina forming a ureterocele, which is described in very few case reports. This rare insertion may be diagnosed due to urinary incontinence. Still, as the patient did not report any history of incontinence, but she answered about similar attacks of pain and fever, previous finding in occasional gynecology Figure 3: Colposcopy exam showing the ureteral meatus vaginal examination or recurrent urinary tract infection inserting into the vagina. (UTI) during childhood, even when directly questioned. It is believed that the ureteral meatus in the vagina was not adequately formed, preventing urine outflow [8]. The clinical management and the drainage of the infectious content of the ureterocele were fundamental for the patient's recovery. Even with the scarce literature on the approach of vaginal ectopic ureterocele with infected content in adult patients, drainage was necessary due to the purulent collection in the ectopic ureterocele. Drainage of the ureterocele with bladder insertion ureter is usually done by the transurethral endoscopic approach [9], but vaginal drainage was chosen for the only possibility of access. A partial open nephrectomy was indicated for pyonephrosis with destruction of the left kidney's upper unit. Complete duplicity of the ureter may become a challenge for urologists in pyelonephritis management due to anatomical variations. If the renal function in one segment is severely impaired, heminephrectomy