Ureteral diverticula are rare urological anomalies characterized by outpouchings of the ureteral wall. Malignancy arising from these structures is exceptionally rare, with only a few cases reported. We report a 57-year-old male with high-grade urothelial carcinoma arising from a ureteral diverticulum in the left distal ureter. The patient experienced painless gross hematuria, and computed tomography revealed a 3 cm undetermined mass near the left distal ureter. Multiple endoscopic diagnostic procedures were inconclusive, with the final diagnosis confirmed intraoperatively. The surgery involved resection of the mass, and intraoperative frozen section analysis revealed high-grade urothelial carcinoma. A segmental ureteral resection was performed with reconstruction using a Boari flap and Psoas hitch. Final histopathology confirmed locally advanced, high-grade pT3 urothelial carcinoma, and adjuvant chemotherapy was recommended. This case underscores the diagnostic and management challenges of upper tract urothelial carcinoma, especially in rare cases involving ureteral diverticula.
Cytomegalovirus (CMV) infection remains a common complication after kidney transplantation, particularly among high-risk donor-seropositive/recipient-seronegative (D+/R-) recipients. Valganciclovir is the standard first-line therapy; however, its use may be limited by hematological toxicity or impaired graft function. Maribavir is a recently approved antiviral agent with a distinct mechanism of action. We report four high-risk kidney transplant recipients in whom maribavir was introduced because continued valganciclovir therapy was difficult owing to cytopenia or renal dysfunction. After switching therapy, CMV viral loads decreased in all cases, and hematological abnormalities improved following discontinuation of valganciclovir. One patient developed a drug-induced skin eruption, while no other serious adverse events were observed. No CMV recurrence or graft dysfunction occurred during follow-up. These cases highlighted practical clinical considerations for managing CMV infection when standard antiviral therapy cannot be safely continued.
Ganglioneuroblastoma is a rare neuroblastic tumor derived from embryonic neural crest cells, composed of mature gangliocytes and immature neuroblasts. Although most neuroblastic tumors present in children and commonly involve the adrenal glands, adrenal ganglioneuroblastoma is rare in adults, with only 25 previously reported cases. A 20-year-old female presented with epigastric pain, and imaging revealed a 13 cm left adrenal mass with calcification and heterogeneous enhancement. Endocrinological tests were normal, except for a mildly elevated urinary metanephrine level. Open adrenalectomy was performed, and histopathological examination confirmed a mixed-type adrenal ganglioneuroblastoma with low proliferative activity (Ki-67, < 1%). Adjuvant therapy was not administered. The patient has remained recurrence-free for 39 months post-operatively. We report a rare case of primary adrenal ganglioneuroblastoma in an adult, emphasizing its diagnostic challenges, histopathological characteristics, and favorable post-operative course.
The role of metastasis-directed therapy in urothelial carcinoma remains uncertain, particularly in the immune checkpoint inhibitor era. We report a case of solitary pulmonary metastasis that developed after a favorable response to pembrolizumab and was managed with metastasectomy. A 65-year-old man with ureteral carcinoma underwent radical nephroureterectomy followed by adjuvant gemcitabine plus carboplatin. Three months later, a solitary pulmonary metastasis was detected and pembrolizumab was initiated. After initial enlargement, the lesion regressed and achieved a partial response after 13 cycles, followed by slight regrowth after 18 cycles, consistent with oligoprogression. Video-assisted thoracoscopic metastasectomy was performed 14 months after pembrolizumab initiation. Pembrolizumab was continued for 1 year postoperatively, with no recurrence observed 17 months after surgery. This case suggests that metastasis-directed therapy may be a reasonable option for carefully selected patients with oligoprogressive urothelial carcinoma during immune checkpoint inhibitor therapy.
Pheochromocytomas and paragangliomas are increasingly recognized as hereditary malignancies, with ~30%-40% of cases involving germline variants. We report a familial case of hereditary pheochromocytoma-paraganglioma syndrome caused by a novel variant in the SDHD gene. An 11-year-old boy was diagnosed with retroperitoneal paraganglioma. Pathological analysis confirmed the resected tumor as a pheochromocytoma and paraganglioma. SDHD sequencing from blood and tumor tissues revealed a c.74dupT single-nucleotide insertion variant, which was absent from all public databases. Another surgery was performed eight years later because of enlargement of the retroperitoneal mass and elevated plasma norepinephrine levels. His 17-year-old sister was later diagnosed with bilateral adrenal pheochromocytomas. Pathological analysis confirmed the resected tumors as pheochromocytoma and paraganglioma. She harbored the same genetic variant inherited from the children's asymptomatic father. Early identification of hereditary risks associated with pheochromocytomas and paragangliomas facilitates appropriate lifelong surveillance, timely treatment decisions, and informed genetic counseling.
Testicular malakoplakia is a very rare, chronic granulomatous inflammatory condition that frequently resembles testicular malignancy on imaging studies. Here, we report a case of testicular malakoplakia with characteristic magnetic resonance imaging (MRI) findings and present a brief review of the literature. A man in his 70s presented with left-sided scrotal pain. Ultrasonography revealed a well-defined hypoechoic intratesticular mass without internal vascularity, whereas computed tomography revealed enlargement of the left testis with a relative hyperattenuation area. MRI demonstrated low signal intensity on T2-weighted images, diffusion restriction, and a markedly high signal intensity on fat-suppressed T1-weighted images with contrast enhancement. Based on these findings, a malignant testicular tumor was suspected, and radical orchidectomy was conducted. Histopathological examination confirmed testicular malakoplakia with characteristic Michaelis-Gutmann bodies. This case highlights the importance of recognizing T1 shortening on fat-suppressed T1-weighted MRI as a potential imaging modality for malakoplakia.
Primary malignant neoplasms of the epididymis are rare. In this case report, we describe clear cell carcinoma associated with papillary cystadenoma of the epididymis (PCE). A 77-year-old man presented with a painless, enlarged left scrotum. Magnetic resonance imaging (MRI) revealed two different abnormal signals in the left epididymis. A left high inguinal orchiectomy and histopathological examination revealed a benign component consistent with PCE and a malignant component showing solid growth with cell nuclear enlargement, atypical mitotic figures, and invasion into surrounding tissues. The final diagnosis was malignant transformation to clear cell carcinoma of PCE. Although this epididymis tumor may follow an indolent course, the presence of areas with higher proliferative potential suggests surgical excision be recommended as definitive treatment.
Pediatric neurogenic lower urinary tract dysfunction (NLUTD) is commonly managed with anticholinergic agents and clean intermittent catheterization (CIC). However, treatment options are limited for patients who remain resistant to conventional therapy, particularly following spinal cord surgery. A 14-year-old boy with myelomeningocele underwent vertebral resection and spinal cord amputation. Despite CIC and solifenacin, he continued to experience daily urinary incontinence. 1 year postoperatively, his symptoms worsened and urinalysis became abnormal. Video-urodynamic study showed reduced bladder compliance and progression of bladder deformity. Vibegron 50 mg once daily was initiated. At 12 weeks, bladder capacity increased and compliance improved. Over 24 months, no urinary tract infections, incontinence episodes, or drug-related adverse events occurred. This case suggests that vibegron may be an effective, well-tolerated add-on therapy for pediatric patients with anticholinergic-resistant NLUTD after spinal cord surgery, improving bladder function and morphology while helping maintain quality of life.
Extragonadal germ cell tumors (EGCT) account for approximately 3%-7% of all germ cell tumors. Metachronous testicular tumors develop in 5%-7% of patients after EGCT treatment; however, bilateral testicular tumors have not been reported. A 30-year-old man underwent retroperitoneal tumor resection and was diagnosed as teratoma. A cystic lesion in the right testis without malignant features was managed by surveillance. Five years later, he presented with left testicular discomfort. Imaging revealed bilateral testicular tumors, and serum hCG was elevated (11.9 mIU/mL). Bilateral radical orchiectomy revealed pure seminoma (pT1) in the left testis and mixed germ cell tumor with seminoma and teratoma components (pT1) in the right testis. This is the first reported case of bilateral testicular tumors developing after EGCT treatment. In patients with EGCT, the potential presence of testicular germ cell neoplasia in situ should be considered, and long-term follow-up is recommended.
Tuberculosis is associated with an increased risk of bladder cancer; however, the simultaneous occurrence of urogenital tuberculosis and bladder cancer is rare. Primary prostate tuberculosis is relatively uncommon in patients with urogenital tuberculosis. A 68-year-old man presented with gross hematuria and irritative voiding symptoms. Cystoscopy revealed a bladder tumor. The PSA level was elevated (8.32 ng/mL), and MRI findings suggested prostate cancer. Chest radiography and CT indicated pulmonary tuberculosis, which was confirmed by sputum mycobacterial culture and PCR. Urine mycobacterial cultures revealed urinary tuberculosis. TURBT and prostate biopsy were performed after initiating treatment for tuberculosis and confirming sputum conversion. Histopathological examination revealed urothelial carcinoma of the bladder and prostate tuberculosis. This is the first reported case of concurrent bladder urothelial carcinoma and prostate tuberculosis. Patients with pulmonary tuberculosis may benefit from evaluation for urogenital tuberculosis, including prostatic involvement, and consideration of coexisting urothelial carcinoma.
Persistent perineal wetness in adults with normal standard imaging is uncommon and may indicate rare congenital anomalies such as urethral duplication. A man in his early 20s presented with a 3-year history of intermittent perineal wetness of unknown origin. Extensive gastrointestinal and urological investigations were unrevealing. Indigo carmine administration confirmed urinary leakage, prompting focused evaluation. Endoscopic assessment with meticulous perineal inspection under general anesthesia identified a tiny, non-inflamed perineal opening. Fistulography and cystoscopy demonstrated an accessory tract arising from the prostatic urethra and opening to the perineum, consistent with Effmann type IIA2 Y-type urethral duplication, extremely rare in adult-onset. Complete excision was achieved using a combined perineal and laparoscopic transabdominal approach with light-emitting catheter guidance, preserving urinary continence and erectile function. Symptoms resolved immediately without recurrence. This case highlighted key diagnostic pitfalls and demonstrates effective surgical management of adult-onset type IIA2 Y-type urethral duplication.
[This corrects the article DOI: 10.1002/iju5.70156.].
Reactive lymphoid hyperplasia (RLH) is a rare benign lymphoproliferative disorder that rarely involves the kidney and has not been reported in association with regional lymphadenopathy. A 70-year-old woman was incidentally found to have a left renal mass. Contrast-enhanced computed tomography revealed a 20-mm enhancing renal lesion with enlarged hilar lymph nodes, leading to a preoperative diagnosis of renal cell carcinoma with nodal metastasis (cT1aN1M0). Open radical nephrectomy with regional lymphadenectomy was performed. Histopathological analyses established a diagnosis of reactive lymphoid hyperplasia. No additional treatment was administered, and the patient has remained free of recurrence for 3 months. We report the first case of RLH accompanied by regional lymph node enlargement. Because RLH can closely mimic renal cell carcinoma on imaging, it should be considered in the differential diagnosis of renal masses when the clinical presentation is inconsistent with typical renal cell carcinoma.
[This corrects the article DOI: 10.1002/iju5.70178.].
The efficacy of neoadjuvant lenvatinib plus pembrolizumab for clear cell renal cell carcinoma with inferior vena cava tumor thrombus remains unclear. An 80-year-old man was diagnosed with clear cell renal cell carcinoma and tumor thrombus extending into the inferior vena cava. Following neoadjuvant lenvatinib plus pembrolizumab, the primary renal tumor shrank markedly, whereas the inferior vena cava tumor thrombus demonstrated only a slight reduction in size. Robot-assisted radical nephrectomy and lymphadenectomy were performed; however, conversion to open surgery was required because of an inferior vena cava laceration. Pathological examination revealed no residual viable cancer cells. The patient has remained free of recurrence for 1.5 years postoperatively. In this case, a pathological complete response was achieved following combination therapy of lenvatinib with pembrolizumab.
Mucinous tubular and spindle cell carcinoma (MTSCC) is a rare renal cell carcinoma subtype that is typically indolent but may exhibit aggressive behavior. No standard systemic therapy has been established for metastatic MTSCC. A 71-year-old woman underwent retroperitoneoscopic radical left nephrectomy for MTSCC (pT1b). Six months later, computed tomography (CT) and 18F-fluorodeoxyglucose positron emission tomography (PET)/CT revealed lung, bone, and adrenal metastases. Bone biopsy confirmed metastatic MTSCC, and the patient was classified as poor risk. Combination therapy with nivolumab and ipilimumab was initiated. After two cycles, PET/CT showed marked regression of metastatic lesions (SUVmax decreased from 21.7 to 4.3). RECIST evaluation demonstrated a partial response. Genomic profiling revealed microsatellite stability and a low tumor mutational burden, with no clinically actionable alterations identified on FoundationOne CDx. No disease progression was observed at 9 months. This case demonstrates the potential efficacy of combination immunotherapy in aggressive metastatic MTSCC.
Peritoneal carcinomatosis in patients with advanced bladder cancer is a rare but lethal event following robot-assisted radical cystectomy (RARC). We report a case of an unusual clinical course in which peritoneal dissemination of colon cancer was unexpectedly detected seven months after RARC. A 67-year-old man underwent two courses of neoadjuvant chemotherapy and subsequent RARC with extracorporeal urinary diversion for muscle-invasive bladder cancer. The pathological diagnosis of the RARC specimen was urothelial carcinoma in situ (ypTis). After 7 months, the patient developed small bowel obstruction and underwent laparotomy. Intraoperatively, numerous seeding nodules were observed in the pelvic cavities, involving the small intestine. Contrary to our expectation of bladder cancer recurrence, the pathological examination revealed adenocarcinoma, suggesting primary colorectal cancer. Peritoneal dissemination occurring shortly after RARC is often considered due to bladder cancer. However, the possibility of metastasis from other cancers must be taken into consideration.
Upper tract urothelial carcinoma with neuroendocrine differentiation (UC-NE) is extremely rare and generally associated with aggressive behavior and poor prognosis. Optimal treatment strategies remain unclear, particularly regarding the role of nectin-4-targeted therapy. A 61-year-old man was diagnosed with UC-NE of the renal pelvis. Laparoscopic nephroureterectomy revealed invasive UC-NE with lymphatic invasion (pT1, G2) and carcinoma in situ of the ureter (pTis, G1). Immunohistochemistry showed strong nectin-4 expression in the urothelial component but only weak to moderate expression in the neuroendocrine component. Ten months after surgery, para-aortic and bilateral pelvic lymph node recurrence developed. Treatment with enfortumab vedotin (EV) plus pembrolizumab achieved a complete response after 3 cycles, and remission was maintained with continued therapy. This case suggests that EV plus pembrolizumab may be effective for UC-NE and highlights the importance of evaluating nectin-4 and the tumor immune microenvironment when considering treatment strategies for this rare subtype.
Blunt trauma to the flaccid penis rarely causes laceration of the tunica albuginea of the corpora cavernosa. A 31-year-old male patient sustained blunt trauma to the flaccid penis during a basketball game, causing subcutaneous hematoma, hematuria, and urinary retention. Computed tomography demonstrated a laceration of the tunica albuginea of the corpora cavernosa. Immediate surgical exploration confirmed the laceration of the tunica albuginea and the partial disruption of the penile urethra. Both injuries were repaired, and a suprapubic catheter was inserted. On postoperative day 12, retrograde urethrography revealed no leakage; however, on postoperative day 24, leakage was observed at the injury site. The suprapubic catheter was maintained for 3 months. Final imaging confirmed urethral patency without leakage. We report a rare case of blunt trauma to the flaccid penis. Immediate repair was successful, and the patient recovered without stricture, although a compressive mechanism caused delayed urethral leakage.
Combination therapy with immune checkpoint inhibitors (ICIs) has become a standard treatment for metastatic renal cell carcinoma (mRCC). However, ICIs may also cause immune-related adverse events. We report a case of mRCC that developed fulminant immune-related enterocolitis. A 79-year-old man received ICIs therapy for mRCC and was urgently hospitalized because of worsening diarrhea. Despite the initiation of steroid therapy, circumferential intestinal necrosis developed, necessitating subtotal colectomy. The patient subsequently developed cytomegalovirus- and Epstein-Barr virus-associated enterocolitis and died from extensive intestinal necrosis caused by severe thrombosis secondary to disseminated intravascular coagulation. Persistent diarrhea during ICIs therapy requires prompt evaluation and specialist consultation, even when symptoms appear mild, as they may signal immune-related enterocolitis. Clinicians should also remain vigilant for infectious complications, which can exacerbate the severity of immune-related enterocolitis and lead to life-threatening outcomes.