Bladder paraganglioma is a rare neuroendocrine tumor (<0.5% of bladder neoplasms) that may be discovered incidentally or present with catecholamine-related symptoms. A 69-year-old man under oncologic surveillance for a renal cell carcinoma showed a bladder wall lesion enlarging from 4 × 5 mm to 8 × 6 mm over two years on CT. Cystoscopy demonstrated a small sessile lesion with intact mucosa. Complete transurethral resection was performed with continuous hemodynamic monitoring. Histology confirmed paraganglioma with synaptophysin and chromogranin A positivity, Ki-67 < 5%, and preserved SDHA/SDHB. Postoperative ¹⁸F-FDOPA PET/CT showed no residual or metastatic disease. Selected small incidental tumors with favorable pathology may be effectively managed by complete transurethral resection.
Background and Purpose Establishing a pathology-proven diagnosis of cholangiocarcinoma (CCA) remains challenging due to the limited sensitivity of conventional techniques. Endoscopic retrograde cholangiopancreatography (ERCP) with brush cytology and intraductal biopsies often fails to confirm malignancy. Sister Mary Joseph’s nodule (SMJN), a metastatic umbilical lesion typically associated with gastrointestinal or gynaecological malignancies, is a rare but potentially important diagnostic clue in patients with suspected CCA. Materials and methods We report a case series of five patients in whom the diagnosis of primary or recurrent CCA was ultimately established or clarified through the identification and histological assessment of SMJN. Clinical presentation, imaging findings, diagnostic procedures, and clinical outcomes were reviewed. Results In one patient, SMJN confirmed metastatic pCCA after repeated negative biliary brushings. In two patients, detection of SMJN revealed disseminated disease, thereby precluding further surgical or liver transplantation work-up. In two additional patients, SMJN represented metastatic recurrence following prior curative-intent treatment, including hepatic resection and liver transplantation. Conclusions SMJN is a rare manifestation of metastatic CCA but may provide an important diagnostic clue when conventional diagnostic modalities fail to establish malignancy. Careful physical examination, including inspection of the umbilical region, remains essential and may facilitate timely diagnosis and appropriate clinical decision-making.
Background: Bing-Neel syndrome (BNS) is a rare neurological complication of lymphoplasmacytic lymphoma/ Waldenstro & uml;m macroglobulinemia (WM), characterized by central nervous system (CNS) infiltration by malignant lymphoplasmacytic cells. Treatment guidelines are limited, and data on management after relapse on covalent Bruton tyrosine kinase inhibitors (BTKi) are scarce. Case presentation: We report the case of a 76-year-old woman with WM who developed BNS and achieved prolonged neurological response with ibrutinib. After 50 months of therapy, she experienced CNS relapse. Treatment with the non-covalent BTKi pirtobrutinib led to rapid clinical improvement and maintained hematological response. Conclusion: This case supports the potential efficacy of pirtobrutinib as salvage therapy for relapsed BNS after covalent BTKi exposure and adds to the limited real-world evidence in this rare clinical entity.
Treatment of advanced renal cell carcinoma has improved with immune checkpoint inhibitors; however, the role of deferred cytoreductive nephrectomy and biomarkers for immune-related adverse events remains unclear. We report a man in his seventies with metastatic renal cell carcinoma with sarcomatoid change treated with nivolumab plus ipilimumab followed by deferred cytoreductive nephrectomy. Despite grade 3 adrenal insufficiency, a durable complete response was maintained with nivolumab monotherapy. T-cell receptor repertoire analysis revealed decreased diversity and increased clonality after treatment, suggesting its potential as a biomarker of therapeutic response and immune-related toxicity.
Pseudoprogression (PsP) rarely occurs late after the initiation of immunotherapy and exceptionally in the adrenal glands. We report two unique cases of late isolated PsP in the adrenal glands of metastatic non-small cell lung cancer (mNSCLC), occurring 10 months after the initiation of nivolumab monotherapy. This provides proof of concept that late PsP can occur in the adrenal glands of mNSCLC patients treated with anti-PD-1. Clinicians should be aware of this potential diagnosis as an alternative to true tumor progression under immunotherapy, with important implications for the subsequent management. The neuro-immune interface may play a role in the development of PsP in the adrenal glands.
Neuroendocrine carcinoma (NEC) of the nasopharynx is a rare and highly heterogeneous malignant tumor, whose clinical characteristics remain poorly elucidated. Unlike conventional nasopharyngeal carcinomas, NECs are not typically associated with Epstein-Barr virus (EBV) infection and often follow an aggressive clinical course. Owing to the scarcity of reported cases and limited long-term follow-up data, their prognosis remains difficult to assess. In this report, we present a case of EBV-positive small-cell neuroendocrine carcinoma of the nasopharynx. The patient was treated with six cycles of chemotherapy using the Carilizumab-EP regimen (etoposide and cisplatin), followed by concurrent chemoradiotherapy. Significant remission was achieved after this comprehensive treatment, and the patient was subsequently discharged. This case contributes to the understanding of neuroendocrine tumors in the nasopharynx and provides relevant clinical data to inform future treatment strategies.
Background: Chemotherapy-refractory Philadelphia chromosome-negative B-cell acute lymphoblastic leukemia in adults who are ineligible for clinical trials remains difficult to cure, and real-world data on peri-transplant blinatumomab in Japanese adults are scarce. Methods: We conducted a single-center retrospective series of consecutive adult Japanese patients with chemotherapy-refractory, Philadelphia chromosome-negative B-cell acute lymphoblastic leukemia who received peri-transplant blinatumomab between 2019 and 2025. Results: Three trial-ineligible patients received blinatumomab as a bridge to allogeneic hematopoietic cell transplantation, with one also receiving post-transplant blinatumomab; all achieved complete remission, two attained pre-transplant minimal residual disease negativity, and all remain in continuous remission at 18, 57, and 76 months without relapse, graft failure, chronic graft-versus-host disease, or transplant-related mortality. Conclusions: Peri-transplant blinatumomab appears feasible in this setting and was associated with durable post-transplant remission and a favorable safety profile.
Metastatic triple-negative breast cancer (mTNBC) is associated with poor overall survival and limited treatment options especially in later lines of therapy and with tumors lacking PD-L1 tissue staining. We report on an Asian female diagnosed with stage IIA TNBC at age 40. She underwent bilateral mastectomy with an invasive ductal carcinoma on the left. Biopsies indicated CCR5 negativity in tumor cells but high CCR5 expression in leukocytes. Five percent of tumor cells and tumor infiltrating lymphocytes stained positive for PD-L1. She initially received adjuvant chemotherapy with dose-dense doxorubicin/cyclophosphamide followed by dose-dense paclitaxel. Subsequently an axillary lymph node dissection revealed a 0.7 cm implant. She was treated with capecitabine but was then diagnosed with lung metastases and underwent a right lung partial lobectomy. She then began therapy with atezolizumab and nab-paclitaxel, then added leronlimab, a CCR5 inhibitor, obtained through compassionate access. Subsequently she was diagnosed with brain metastases which were treated with parieto-occipital cortex stereotactic radiotherapy. The patient discontinued nab-paclitaxel but continued atezolizumab and leronlimab. Blood samples were analyzed for PD-L1 expression on cancer-associated macrophage-like cells and circulating tumor cells. These analyses showed that after receiving leronlimab the patient's PD-L1 expression increased significantly. > 60 months after starting leronlimab in combination with atezolizumab the patient remains alive with no evidence of disease. This case of disease-free survival in mTNBC is unexpected given the rapidity of disease recurrence during her standard-of-care adjuvant treatment. This case highlights the potential for CCR5 inhibition in combination with an ICI for treatment of mTNBC.
The management of recurrent or metastatic vulvar squamous cell carcinoma (VSCC) remains challenging due to the absence of standardized systemic therapies. While immune checkpoint inhibitors (ICIs) targeting the PD-1/PD-L1 axis are promising, their efficacy is traditionally linked to biomarkers such as PD-L1 expression or microsatellite instability-high (MSI-H) status. Here, we report an atypical case of a 23-year-old patient with advanced PD-L1-negative (CPS <1) and microsatellite stable (MSS) VSCC who achieved sustained partial remission following treatment with sintilimab (anti-PD-1), bevacizumab, and chemotherapy. Initially resistant to platinum-based chemotherapy, the patient exhibited rapid symptomatic relief and imaging-confirmed tumor reduction after two cycles of combination therapy, with durable response maintained through six cycles. This case challenges conventional biomarker paradigms, suggesting that ICIs combined with anti-angiogenic and cytotoxic agents may overcome resistance in PD-L1/MSS VSCC. In addition, we provide a comprehensive review of the current literature that the synergistic interplay between immune modulation, vascular normalization, and chemotherapy likely contributed to this outcome, underscoring the need for broader predictive frameworks beyond PD-L1 and MSI-H to optimize immunotherapy selection in rare gynecologic malignancies.
A 60-year-old postmenopausal woman presented to our hospital in April 2025 with a 2.2 cm vulvar mass. She had initially been evaluated for this mass in September 2024 but was subsequently transferred for a colonoscopy due to acute gastrointestinal symptoms, which led to a diagnosis of descending-sigmoid colon cancer. The patient underwent an immediate laparoscopic radical left hemicolectomy, during which the vulvar mass was overlooked. She subsequently completed 6 cycles of adjuvant XELOX chemotherapy; however, the vulvar mass progressively enlarged, leading to intermittent vaginal bleeding. A biopsy at our institution ultimately confirmed metastatic adenocarcinoma of lower gastrointestinal origin. This case highlights that for patients presenting with gynecological symptoms as the initial manifestation of occult colon cancer, screening for concurrent colon cancer should be considered. Furthermore, female patients with diagnosed colon cancer should be alerted to the possibility of vulvar metastases. For the treatment of such patients, there are currently no established guidelines and limited clinical evidence. Therefore, multidisciplinary discussion and individualized treatment are particularly crucial in managing these rare cases.
Autoimmune encephalitis is a condition where patients typically present with short-term memory deficits, psychiatric symptoms, and seizures, all with rapid progression. Recently, cases of limbic encephalitis associated with the use of new antibodies for cancer treatment have been reported, such as the monoclonal antibodies (mAb) IgG4 nivolumab and pembrolizumab, which block the binding of PD-1 to PD-L1 and PD-L2 ligands in the tumor microenvironment. We present the first case of limbic encephalitis related to the use of Pembrolizumab in Mexico.
Ureteral metastasis from prostate cancer is exceedingly rare and may mimic primary upper tract urothelial carcinoma, representing a major diagnostic pitfall. We report a 72-year-old man presenting with right flank pain and ureterohydronephrosis. Imaging revealed a proximal ureteral mass with a suspicious para-aortic lymph node, leading to nephroureterectomy. Histopathology demonstrated a grade group 5 prostatic adenocarcinoma, confirmed by subsequent prostate biopsies. Bone scintigraphy later revealed multiple metastases. The patient received combined hormonal therapy and chemotherapy. This case emphasizes the importance of histopathological confirmation and awareness of rare metastatic patterns to guide appropriate management.
Radiation-induced sarcomas are rare late complications of radiotherapy and represent aggressive mesenchymal tumors arising in previously irradiated tissues. They most commonly occur in the pelvis and thorax, while involvement of the colon is exceptional, particularly outside the rectum. A 48-year-old male with a history of gastric adenocarcinoma treated twelve years earlier with subtotal gastrectomy followed by adjuvant chemoradiotherapy according to the INT-0116 protocol presented with a five-month history of progressive abdominal pain, weight loss, hematemesis, melena, and recurrent vomiting. Abdominal computed tomography revealed an endoluminal mass in the transverse colon without evidence of distant metastasis. Colonoscopy demonstrated a large pedunculated tumor with necrotic borders occupying the entire colonic lumen. The patient underwent an extended right hemicolectomy with intracorporeal ileocolic anastomosis and was discharged on postoperative day five without complications.Histopathological examination revealed a high-grade inflammatory leiomyosarcoma with myogenic differentiation, extensive necrosis, and a high proliferative index. Surgical margins and lymph nodes were negative, and genetic testing showed no evidence of a hereditary cancer syndrome. The patient remains under close oncologic and colorectal surgical follow-up with a favorable clinical course after 2 years. This case highlights the importance of long-term surveillance in patients undergoing radiotherapy, as early recognition of radiation-induced sarcomas may allow curative surgical treatment despite their traditionally poor prognosis.
Introduction Clear cell carcinoma (CCC) of the uterus is a rare subtype of endometrial cancer, accounting for about 5% of cases and associated with poor prognosis. Limited data on CCC makes treatment challenging, with most guidelines based on retrospective studies. This case illustrates the use of radiation therapy for durable local control in a patient with oligometastatic uterine CCC. Case Presentation A 68-year-old woman was diagnosed with stage IA uterine CCC in March 2020. Following surgery, adjuvant chemotherapy, and brachytherapy, she developed an abdominal wall oligometastasis. Despite initial systemic treatment, her disease progressed with the appearance of a new lung nodule. She received definitive radiation therapy for the lung and abdominal wall oligometastases with complete resolution of both lesions on PET-CT scan. However, the patient developed systemic disease progression at distant sites 18 months later, for which she received further systemic therapy. Despite further disease progression, both irradiated sites (abdominal lesion and lung nodule) remained in complete remission with durable local control maintained for 25 months of follow-up. Conclusion Radiation therapy provided effective and durable local control in this case of oligometastatic uterine CCC. This suggests that definitive radiotherapy may be beneficial for managing oligometastatic CCC, though more research is needed to optimize treatment strategies. To our knowledge, there are no published reports describing the use of definitive radiotherapy for treating abdominal wall oligometastases from uterine CCC. This case is unique in demonstrating a durable complete response of this oligometastatic lesion following definitive external beam radiation (EBRT).
Anaplastic lymphoma kinase (ALK) rearrangements occur in 3-5% of non-small cell lung cancer (NSCLC), with the STRN-ALK fusion representing a rare variant with variable responses to ALK tyrosine kinase inhibitors (TKIs). Lorlatinib, a third-generation ALK TKI with broad activity against resistance mutations and superior CNS penetration, has not been previously reported in STRN-ALK fusion-positive NSCLC. We present a 52-year-old male never-smoker who initially complained of back pain and was found to have multiple thoracic lesions on a CT scan of the chest, pleural nodules and a pleural effusion, which was found to be consistent with exudative effusion and adenocarcinoma on cytology. He was diagnosed with stage IV lung adenocarcinoma with signet-ring cell features. Next-generation sequencing of tissue identified a STRN-ALK fusion along with pathogenic ARID1A and KMT2D mutations. The patient was initiated on lorlatinib 100 mg daily as first-line therapy. Treatment was complicated by manageable adverse events, including hyperlipidemia, peripheral neuropathy, first-degree AV block, and lower extremity edema. At nine-month follow-up, PET-CT demonstrated a partial response with resolution of bone metastases and a significant reduction in thoracic lesions. This represents the first reported case of STRN-ALK fusion-positive NSCLC treated with lorlatinib monotherapy, demonstrating the clinical efficacy and tolerability of third-generation ALK inhibition for this rare fusion variant, which has variable responses to earlier-generation TKIs in the literature. These findings support lorlatinib as an effective treatment option for STRN-ALK NSCLC, particularly given its superior CNS penetration, which is particularly relevant for preventing or treating brain metastases in patients with previously identified brain metastases.
Primary malignant melanoma of the cervix is an extremely rare malignant tumor, accounting for <0.5 % of all cervical malignancies. This article reports a case of a 66-year-old female patient who presented with "vaginal bleeding" and was diagnosed with primary malignant melanoma of the cervix via colposcopic biopsy and immunohistochemistry. The patient received dacarbazine chemotherapy. Through a systematic review of previous literature, this paper summarizes the clinicopathological features, diagnostic criteria, treatment strategies, and prognostic factors of this disease.
Superior vena cava (SVC) syndrome is an oncological emergency characterized by venous obstruction, most commonly due to malignancy, that leads to facial and upper extremity edema and dyspnea that worsen in the supine position. A 70-year-old man with SVC syndrome secondary to lung adenocarcinoma presented with severe orthopnea, generalized edema, and inability to tolerate the supine position. Because the peripheral and inguinal venous accesses were not feasible, distal femoral central venous catheterization (CVC) was performed under ultrasound guidance in a semi-sitting position, without complications. This facilitated the prompt initiation of chemotherapy, which stabilized the patient’s condition. This case demonstrates that in oncological emergencies, semi-sitting distal femoral CVC is a safe and practical alternative for patients with SVC obstruction who cannot tolerate the supine position.
Microsatellite-stable (MSS) or mismatch repair proficient (MMRp) colorectal cancer typically exhibits resistance to immune checkpoint blockade. Tumor mutational burden (TMB) and DNA repair defects may increase immunogenicity and sensitivity to novel immunotherapies. We present a patient with metastatic MSS colorectal adenocarcinoma, previously treated with multiple standard regimens, who demonstrated an exceptional and durable partial response to an investigational bispecific PD-1/ICOS antibody. XmAb23104 was initially given in combination with ipilimumab on a clinical trial (DUET-3) and subsequently via an expanded-access investigational new drug (IND) application. Genomic profiling revealed extremely high TMB and pathogenic BRCA1/2 mutations. He achieved a partial response which was sustained for >2 years without dose reductions. This case highlights a novel therapeutic opportunity for MSS CRC, in which high TMB and homologous recombination deficiency may sensitize tumors to bispecific checkpoint blockade. Further investigation of PD-1/ICOS bispecific antibodies in biomarker-selected MSS solid tumors is warranted.
Introduction: Acquired hemophilia A (AHA) is a rare autoimmune bleeding disorder that can cause severe hemorrhage in elderly patients. Solid tumors are considered one of the causes of this condition, and we report a case of AHA caused by colon cancer that led to death due to severe bleeding shortly after treatment intervention. Case Presentation: An 89-year-old, elderly male patient presented with significant subcutaneous hemorrhage and recurrent bloody stool. Colonoscopy revealed sigmoid colon cancer, and CT scans suggested metastatic lung cancer. Because the factor VIII inhibitor titer was high at approximately 100 BU/mL, prednisolone and cyclophosphamide were administered immediately after the diagnosis of AHA. While the inhibitor titer subsequently began to decrease, but the factor VIII activity level remained in the 1 % range and did not increase, so the patient's tendency to bleed became more pronounced; the bypassing hemostatic agent, a mixture of factors VIIa and X (Byclot®), was also administered. Despite being administered twice, hemostatic control was insufficient, and bleeding persisted intermittently. Furthermore, respiratory failure due to pulmonary emphysema rapidly worsened, and the patient died approximately 2 weeks after treatment intervention. Conclusion: In all three cases treated at our facility, including this patient, AHA develops after the cancer has progressed, making patients difficult to treat and resulting in a poor prognosis. In the future, it will be necessary to investigate a larger number of AHA patients with underlying solid tumors and consider the optimal treatment for each case.
