Background and Clinical Significance: Enterovesical fistulas are uncommon and are most often caused by inflammatory conditions. Cases caused by Meckel's diverticulum are exceptionally rare, with only a few cases reported in the literature. To our knowledge, no previous reports have described an ileovesical fistula due to Meckel's diverticulum coexisting with urothelial carcinoma. Case Presentation: A 74-year-old man was evaluated after presenting with fever and diagnosed with an ileovesical fistula. Since cystoscopy findings could not completely exclude bladder cancer, transurethral resection of the bladder tumor (TUR-Bt) was performed 1 month after the infection subsided. Histopathological examination confirmed the diagnosis of bladder cancer. Partial small intestine resection was performed 1 week after TUR-Bt to treat the ileovesical fistula. Histopathological examination of the resected specimen revealed perforation due to Meckel's diverticulitis. TUR-Bt was repeated 3 months after the initial surgery, and no residual tumor was detected. At 6 months postoperatively, no recurrence of bladder cancer or fistula was observed. Conclusions: This case highlights an extremely rare coexistence of ileovesical fistula due to Meckel's diverticulum and urothelial carcinoma. Given the potential for malignancy in or around fistulous lesions, careful diagnostic evaluation and appropriate management are essential.
Ureteral fibroepithelial polyps are quite rare in acute urology emergencies, and their management typically relies on an evidence-based approach. This report describes a unique case involving a 49-year-old woman who presented with flank pain, bleeding, left-sided hydronephrosis, and acute urinary retention. The diagnostic process involved CT scans, catheter insertion, and cystoscopy-leading to the removal of a large foreign body, which was confirmed as an autolysed infarcted ureteral polyp on histology. No recurrence or kidney obstruction was observed at follow-up. We have included a supplementary review of previously published cases and related literature.
A 58-year-old man was referred to our department for further evaluation of an incidental right intratesticular mass detected on abdominal and pelvic contrast-enhanced computed tomography performed for an unrelated condition. The mass measured approximately 13 mm and was not clearly palpable on physical examination. Scrotal magnetic resonance imaging showed a well-defined, lobulated, and multinodular mass within the right testis. As a malignant testicular tumor could not be excluded, a right high inguinal orchiectomy was performed for diagnostic and therapeutic purposes. Macroscopically, the excised specimen contained a solid yellow-white mass measuring 16×13×12 mm. Histopathological examination revealed proliferation of tumor cells with round nuclei and eosinophilic cytoplasm. Immunohistochemical staining showed that the tumor cells were positive for calretinin, focally positive for Melan-A, and negative for SALL4. Based on these findings, the diagnosis of a Leydig cell tumor of the testis was made. The patient's postoperative course was uneventful, and no evidence of local recurrence or distant metastasis was observed during 9 months of follow-up. Leydig cell tumors are rare, accounting for approximately 1-3% of all testicular tumors. To our knowledge, this case represents the 128th reported case of testicular Leydig cell tumor in Japan. We report this rare case and provide a review of the literature.
Caffeine is widely consumed and generally considered safe at customary doses. How-ever, high-dose preparations available online pose a risk of severe and potentially fatal intoxication. Although uncommon, lethal caffeine poisoning is associated with profound cardiovascular and neurological toxicity. A rare case of intentional acute caffeine intoxication with fatal outcome is presented. A 25-year-old woman ingested an estimated 60 tablets containing 200 mg of caffeine each, purchased online. She was admitted to hospital shortly after ingestion of the caffeine tablets with palpitations, agitation, dizziness, and repeated vomiting. On examination, she presented with arterial hypotension (90/60 mmHg) and marked sinus tachycardia (150 beats/min), accompanied by psychomotor agitation. Her blood caffeine concentration measured by means of high-performance liquid chromatography (HPLC) was 177 µg/mL. The patient's condition rapidly deteriorated, with the development of convulsive syndrome progressing to coma, extreme ventricular tachycardia, exotoxic shock, and toxic cardiomyopathy. Despite intensive care management, including mechanical ventilation and advanced cardiopulmonary resuscitation, the patient died several hours after admission. In conclusion, this case underscores the life-threatening potential of acute high-dose caffeine ingestion and highlights the risk associated with unrestricted access to concentrated caffeine products. Early recognition and aggressive management are crucial, yet may be insufficient in cases of massive overdose.
This study reports the case of a 67-year-old man with scrotal Paget's disease. A systematic analysis of the clinical features, diagnostic and therapeutic strategies, and prognosis management of this rare disease was performed in conjunction with a literature review. The patient had a prolonged disease course with a history of three previous lesion excisions and presented this time due to lesion enlargement accompanied by mucous discharge. Dermoscopic examination revealed a "red background with punctate vessels," and preoperative epidermal histopathology supported the diagnosis of Paget's disease. The surgical approach involved wide excision of the lesion, aiming to remove the lesion as completely as possible while preserving the morphology and function of the surrounding tissues. Intraoperative frozen section analysis showed positive margins at the 1, 2, 6, and 11 o'clock positions. After further excision, immunohistochemical staining confirmed cytokeratin 7 (CK7) (+), gross cystic disease fluid protein-15 (GCDFP-15) (+), consistent with a sweat gland origin, and podoplanin (D2-40) (+), indicating a potential risk of lymphatic invasion. Finally, the patient was followed up regularly as planned, and at the most recent follow-up, no signs of recurrence were observed, and treatment outcomes were satisfactory, thereby allowing the discontinuation of further follow-up examinations. This case report provides a valuable practical reference for the standardized diagnosis and treatment of this rare condition.
Ganglioneuroma (GN) is a rare benign tumor from neural crest tissue, composed of mature Schwann cells, ganglion cells, and nerve fibers. These tumors most often arise in the retroperitoneal region (52%) and posterior mediastinum (39%), while extra-adrenal lesions account for 51% of abdominal GN cases. This paper presents a 66-year-old female with paroxysmal atrial fibrillation found to have a left retroperitoneal mass on imaging. A 9.5-cm tumor was fully resected using robot-assisted laparoscopy. Pathology confirmed GN with ectopic inferior vena cava (IVC) invasion. This case highlights the need to consider retroperitoneal GN in the differential diagnosis of vascular structures. In addition, three-dimensional reconstruction is valuable for surgical planning, and robot-assisted surgery is preferred for complex cases.
Horseshoe kidney (HSK) is the most common renal fusion and malrotation anomaly, and ureteropelvic junction obstruction (UPJO) due to high ureteral insertion (HUI) is frequently associated with HSK. Bypass pyeloplasty (BP) is a non-dismembered surgical technique involving a side-to-side anastomosis between the ureter just distal to the UPJO and the dependent portion of the hydronephrotic renal pelvis, and is suited for HUI. Although several procedures have been suggested for UPJO in HSK patients, BP has not been discussed much. In this case report, we describe the detailed procedure for BP successfully performed in an HSK patient with UPJO due to HUI. The patient was a girl aged 1 year and 10 months who had experienced intermittent abdominal pain and occasional vomiting for 1 month. She was diagnosed with dilation of the left renal pelvis and renal calyces (Society for Fetal Urology classification grade 3-4) on ultrasonography and subsequently referred to our hospital for diagnosis of left intermittent hydronephrosis. Renal scintigraphy revealed the HSK, and the differential renal function (DRF) in the left kidney was 16.3%. At 1 year and 11 months, she underwent retrograde left ureteropyelography and pyeloplasty. The retrograde left ureteropyelography showed the HUI and obstruction of the left ureter at approximately 45 mm from the cephalic edge of the umbilicus. We performed a 3.0-cm left flank incision to expose the left renal pelvis and ureter through the retroperitoneal route. HUI of the left ureter and crossing of a white cord-like structure that compressed the left ureter and renal pelvis just distal to the UPJO were identified. We divided the white cord-like structure and performed BP between the ureter just distal to the UPJO and the dependent portion of the renal pelvis. The postoperative course was good, with improvement in the renal pelvis dilation and the DRF in the left kidney. There has been no recurrence for >6 years after surgery. We recommend BP as a physiological and suitable surgical approach that reduces the risk of impaired blood supply and postoperative anastomotic stenosis in HSK patients with UPJO due to HUI.
Post-transplantation lymphoproliferative disorder (PTLD) is an uncommon but serious complication of organ transplantation. Management often requires multiple treatment modalities and reduction in immunosuppressant treatment (RIT). Orbital involvement is rare, and optimal diagnostic and therapeutic approaches remain uncertain. Here, we report a case of localized orbital PTLD that was successfully treated with RIT alone. A 62-year-old man, who had undergone kidney transplantation 18 years prior, presented with left eye pain and proptosis. Imaging revealed an enhancing intraorbital mass extending to the optic nerve and adjacent intracranial structures. Transcranial biopsy confirmed B-cell-type PTLD. The lesion regressed with stepwise RIT alone without additional therapy. The patient showed clear clinical improvement and remained in remission for more than 13 months. This case suggests the potential role of a conservative, individualized, stepwise approach in selected patients with localized orbital PTLD, and may inform management strategies for other localized forms of PTLD.
Bladder involvement in neurofibromatosis type 1 is rare. We report a pediatric case of bladder plexiform neurofibroma treated with partial cystectomy, ileal augmentation cystoplasty, and ureteral reimplantation. Although low-pressure urinary tract reconstruction was achieved, the patient did not regain the ability to void spontaneously, requiring long-term suprapubic cystostomy and ureteral stenting. Five years later, the patient developed retroperitoneal malignant peripheral nerve sheath tumor. Despite subtotal resection and pazopanib therapy, the disease progressed and the patient died at 14 years of age. This case highlights persistent functional morbidity and later malignant progression during the long-term course of NF1-associated pelvic disease.
We present the first reported case of holmium laser enucleation of the prostate (HoLEP) in a patient with a mucosa-embedded nitinol-based "Butterfly" prostatic retractor device and a prior Excision and Primary Anastomosis urethroplasty for bulbar stricture. Using a 22Fr minimally invasive resectoscope (MiLEP), the device was fragmented with the laser and extracted in its entirety. Prostatic enucleation and morcellation were successfully and safely completed. Morcellation is feasible but requires caution due to device interference. This study demonstrates the feasibility of small-caliber MiLEP in patients with complex anatomy and implanted prostatic devices, expanding minimally invasive options for surgically challenging BPH cases.
Scrotal botulinum toxin type A (BoNT-A) injections ("scrotox") are increasingly used to relax the dartos and/or cremaster muscles for cosmetic purposes and for functional indications such as chronic scrotal pain/orchialgia, exaggerated cremasteric reflex, and cremasteric synkinesia. Evidence remains limited and dispersed across animal toxicology studies, urologic case reports, and pain-management trials. We conducted a systematic review to evaluate whether botulinum toxin exposure involving the scrotum, dartos, cremaster, spermatic cord/peritesticular region, or predefined regulatory male fertility datasets is associated with changes in male reproductive outcomes. We performed a systematic review in accordance with PRISMA 2020. PubMed/MEDLINE, Embase, and Scopus were searched from inception through January 5, 2026, with additional identification via reference screening and targeted retrieval of publicly available regulatory reproductive-toxicity documentation for onabotulinumtoxinA and prabotulinumtoxinA, the products for which relevant public regulatory datasets were available. Eligible studies included animal experiments and human clinical reports involving relevant anatomic exposure sites and reporting at least one fertility-relevant outcome (e.g., sperm parameters, spermatogenesis-related histology, reproductive hormones, or fertility indices). Given heterogeneity in exposure routes, dosing, and outcomes, meta-analysis was not planned; results were synthesized qualitatively. Risk of bias was assessed using SYRCLE (animal studies), Joanna Briggs Institute tools (case reports/series), and RoB 2.0 (randomized trials), and evidence levels were categorized using Oxford CEBM. The search identified 93 records; after deduplication, 75 underwent title/abstract screening, 15 were reviewed in full text, and 10 studies met inclusion criteria for qualitative synthesis. Evidence was heavily weighted toward animal data. Animal studies provided moderate-quality evidence suggesting dose- and site-dependent reproductive effects: repeated or higher-dose exposures delivered adjacent to the testes (particularly intracremasteric administration) were associated with impaired spermatogenesis and adverse testicular histopathology, whereas very low-dose systemic intramuscular exposure distant from the testes was reported to be neutral or potentially beneficial in an ageing model. Human evidence was uniformly low or very low quality for fertility inference because published case reports, case series, and clinical trials primarily evaluated pain relief, neuromuscular outcomes, or cosmetic satisfaction and did not prospectively measure semen parameters, endocrine function, thermoregulatory metrics, or fertility endpoints. Current evidence is insufficient to determine the reproductive safety of scrotal or peritesticular botulinum toxin injections in humans. Preclinical data raise concern for potential spermatotoxicity with higher-dose and/or anatomically proximal exposure, while human studies remain indirect and lack fertility-specific monitoring. Standardized dosing and technique reporting, incorporation of semen and hormonal outcomes, direct thermophysiological assessment, and longitudinal follow-up are needed. Until such data exist, clinicians should counsel men desiring future fertility that mechanistic risk is plausible and definitive human fertility outcomes have not been established.
Urothelial carcinoma with sarcomatoid differentiation (UCSD) is a rare and highly aggressive variant of urothelial carcinoma associated with poor prognosis. UCSD originating in the upper urinary tract is extremely uncommon, and its variable morphological presentation, along with the lack of established treatment guidelines, often poses diagnostic and therapeutic challenges. We herein report four cases of upper tract UCSD treated with radical nephroureterectomy. All tumors exhibited varying proportions (40%-90%) of sarcomatoid components with diverse histological patterns. Postoperatively, patients received adjuvant chemotherapy. Through this case report and review of the relevant literature, we aim to enhance the understanding of this rare entity and contribute to the discussion on optimal treatment strategies for upper tract UCSD.
Ectopic hepatocellular carcinoma (EHCC) with brain metastasis (BM) is exceptionally rare and carries a poor prognosis. We report a case initially presenting with tumor stroke syndrome, manifesting as sudden unconsciousness and unresponsiveness. Emergency intracranial hematoma evacuation was performed, and histopathology confirmed BM of hepatocellular carcinoma. Subsequent PET/CT localized the primary EHCC to the left lung lower lobe. This case underscores the critical role of radiological evaluation (CT/MRI for diagnosis/prognosis; PET/CT for primary lesion identification) in such scenarios. For patients with tumor stroke as the initial symptom, prompt suspicion of hemorrhagic malignant BM is essential, requiring emergent intervention and pathological confirmation. Notably, markedly elevated alpha-fetoprotein combined with extrahepatic FDG-avid masses on PET/CT strongly suggests EHCC.
We report a case of bladder pain syndrome (BPS) in which pain improved with clean intermittent catheterization (CIC). The patient wasa 26-year-old woman who had experienced urinary frequency since childhood. Two years prior to visiting our hospital, her symptoms worsened, leading her to seek medical attention for increased frequency and bladder pain. Cystoscopy revealed no Hunner's lesions, but a voiding diary indicated reduced bladder capacity. A trial of bladder hydrodistension was performed without symptomatic improvement. She was referred to our department for further evaluation and management. Pressure-flow study (PFS) showed reduced detrusor contractility with significant abdominal straining during voiding. Although there was no post-void residual, she experienced severe voiding pain, prompting initiation of CIC. While her symptoms improved, attempts to discontinue CIC resulted in pain recurrence, and she continued CIC for over two years. This case highlights the diagnostic value of PFS and the potential effectiveness of CIC in selected BPS patients.
Penile metastasis from prostate adenocarcinoma is an exceptionally rare clinical finding, occurring in <0.3% of cases. We report an 85-year-old male with a history of Gleason 4+3 acinar adenocarcinoma, managed conservatively with long-term bicalutamide monotherapy because of his clinical stability. Eight years after diagnosis, he developed a painful, ulcerated glans lesion, surgically excised via glansectomy. Histopathology revealed solid adenocarcinoma with angiolymphatic invasion, high Ki-67 index (20-70%), PSA and CDX2 positivity, and negative CK7/CK20/TTF-1/p63 staining, confirming prostatic origin. Despite the presence of metastasis, disease progression remained indolent, supporting the feasibility of individualized, conservative therapy in selected elderly patients. Literature review highlights venous or lymphatic spread as probable pathways, with prognosis varying widely. This case underscores the importance of considering secondary malignancy in penile lesions, utilizing histopathology and immunohistochemistry for definitive diagnosis, and tailoring management to patient comorbidities and preferences to preserve quality of life.
Upper tract urothelial carcinoma (UTUC) is a rare and aggressive malignancy, accounting for only 5-10% of all urothelial carcinomas (UCs). Lung, bone, liver, and distant lymph nodes are common sites of metastasis, while gastrointestinal metastasis is extremely rare. We present a case of a 63-year-old female who developed a descending colon lesion 19 months after left radical nephroureterectomy for high-grade ureteral UC. The diagnosis was established by computed tomography (CT), magnetic resonance imaging (MRI), colonoscopy, and biopsy, which excluded primary colorectal malignancy. First-line therapy consisted of six 21-day cycles of gemcitabine plus cisplatin, followed by two cycles of tislelizumab maintenance immunotherapy. Restaging with contrast-enhanced CT and positron emission tomography/computed tomography (PET/CT) demonstrated disease progression. Despite switching to second-line nab-paclitaxel, the patient rapidly deteriorated from tumor cachexia and ultimately succumbed to septic shock secondary to severe pulmonary infection. This represents the first reported case of descending colon metastasis from primary ureteral UC. It highlights the colon as a potential metastatic site where biopsy is essential for definitive diagnosis. Notably, although the patient initially responded to platinum-based therapy, the subsequent rapid progression underscores the need for vigilant monitoring and timely adjustment of therapeutic strategies in managing such high-risk presentations.
Urachal carcinoma is a rare and aggressive malignancy for which standardized management remains limited, particularly in patients with locally advanced and node-positive disease. We report the case of a 34-year-old woman with urachal adenocarcinoma involving the bladder dome and radiographically suspicious pelvic lymph nodes who underwent robot-assisted partial cystectomy with urachal resection and extended bilateral pelvic lymph node dissection. Because there was no clinical, radiologic, or intraoperative evidence of umbilical involvement, the umbilicus was preserved after preoperative counseling and intraoperative confirmation of a negative proximal margin. Final pathology demonstrated a 4.5 cm enteric-type urachal adenocarcinoma, pT3a pN2 (2/17), with lymphovascular invasion, perineural invasion, and negative surgical margins. Immunohistochemistry and DNA- and RNA-based next-generation sequencing showed microsatellite stability, mismatch-repair proficiency, low tumor mutational burden, and no actionable genomic alteration. Given the absence of an established adjuvant standard, the multidisciplinary tumor board selected adjuvant FOLFOX as a non-standard postoperative strategy based on the overall clinicopathologic context. The patient remained continent, experienced no postoperative complications or treatment-limiting toxicity, and showed normalization of carcinoembryonic antigen and carbohydrate antigen 19-9 levels. This case provides a carefully contextualized example of transparent surgical reasoning and restrained multidisciplinary management in a rare malignancy with limited prospective evidence.
Plasmablastic lymphoma (PBL) is an aggressive and uncommon variant of diffuse large B-cell lymphoma, most frequently associated with immunocompromised states. Although extranodal disease is characteristic, involvement of the urinary bladder is exceptionally rare and may mimic primary urothelial malignancy both clinically and histologically. We report the case of a 62-year-old man with a previous history of high-grade diffuse large B-cell lymphoma in remission who presented with acute gross haematuria. Imaging demonstrated bladder wall thickening with associated hydronephrosis. Transurethral resection of the bladder lesion, followed by detailed histopathological and immunohistochemical analysis, confirmed plasmablastic large B-cell lymphoma involving the bladder. This case underscores the importance of maintaining a broad differential diagnosis in patients presenting with haematuria, particularly those with a prior history of haematologic malignancy. A review of the literature highlights the rarity of bladder involvement and the diagnostic challenges associated with this entity.
Background: Polysubstance use, particularly the combination of opioids and stimulants, represents a growing public health concern due to its high risk of severe multisystem complications and mortality. Here, we present a case illustrating the lethal synergy of opioid-stimulant co-use. Methods: A 37-year-old male with chronic Hepatitis C and documented polysubstance use reported recent use of fentanyl, cocaine, methamphetamine, and cannabis. He presented with generalized weakness, left lower limb pain, tense edema, and anuria. Clinical assessment included monitoring of vital signs, physical examination, capillary blood gas analysis, extended laboratory panels (muscle and cardiac enzymes, electrolytes, and coagulation parameters), urinalysis, and Doppler imaging. Management over five days included intravenous hydration, diuretics, urinary alkalinization, electrolyte correction, anticoagulation, metabolic and vitamin therapy, hemodialysis, and comprehensive supportive care. Results: Laboratory evaluation revealed massive rhabdomyolysis (peak CK 161,050 U/L), severe hyperkalemia (K+ 8.4 mmol/L), metabolic acidosis, acute kidney injury with oligoanuria, and left-sided deep vein thrombosis. Despite intensive multidisciplinary interventions, the patient's repeated refusal of ongoing treatment critically contributed to a fatal outcome. Conclusions: This case underscores the high mortality risk associated with opioid-stimulant co-use and the crucial impact of treatment refusal. Clinicians and public health stakeholders should recognize the rapid progression of multisystem dysfunction in polysubstance users and prioritize early, aggressive interventions combined with patient engagement strategies to mitigate fatal outcomes.
Placenta percreta is the most severe form of abnormal placental invasion and is associated with significant maternal morbidity and mortality. Although bladder invasion may occur, gross hematuria is an uncommon and diagnostically challenging presentation. We report the case of a 35-year-old woman with a history of three prior cesarean sections who presented at 26 weeks of gestation with abdominal pain, spotting, acute urinary retention, and gross hematuria. Imaging suggested placenta previa with suspected placenta increta/percreta. Emergency cesarean delivery followed by hysterectomy was performed due to massive hemorrhage. Despite hysterectomy, persistent hematuria and hemodynamic instability continued. Cystoscopy revealed only nonspecific bladder wall hyperemia without an identifiable bleeding source. Definitive diagnosis and control of bleeding were achieved only after surgical re-exploration, which identified and ligated an actively bleeding intramural bladder vessel. This case highlights the limited diagnostic and therapeutic value of cystoscopy in placenta percreta-related hematuria. A normal or nonspecific cystoscopic examination should not exclude bladder invasion or delay surgical intervention in unstable patients.