Introduction Insulin autoimmune syndrome (IAS, Hirata’s disease) is a rare cause of endogenous hyperinsulinemic hypoglycemia classically linked to high-titer insulin autoantibodies and exposure to sulfhydryl (–SH)–containing drugs. IAS is strongly HLA-associated and is most frequently reported in East Asian populations. We report a case in which simultaneous exposure to two thiol-containing agents unmasked IAS in a non-Asian patient carrying HLA-DRB1*04:06. Case presentation A 60-year-old woman developed recurrent autonomic and neuroglycopenic episodes 2–4 hours after meals. Home medications included captopril and over-the-counter α-lipoic acid (ALA). During a symptomatic episode her plasma glucose was 38 mg/dL with markedly elevated serum insulin, C-peptide and proinsulin; mixed-meal testing reproduced a postprandial glucose peak followed by profound hypoglycemia accompanied by paradoxical hyperinsulinemia. Insulin autoantibodies were strongly positive (>150 U/mL) and HLA genotyping revealed DRB1*04:06. A supervised fast was non-diagnostic for insulinoma. Management comprised immediate discontinuation of both captopril and ALA (captopril replaced with losartan) and dietary measures (frequent small low-carbohydrate meals). Hypoglycemic episodes abated within two weeks and resolved by six weeks; fasting glucose, insulin and IAA titers had substantially improved by three months. Conclusion To our knowledge, this is the first reported case of IAS temporally associated with concurrent captopril and ALA exposure in a non-Asian carrier of HLA-DRB1*04:06. The case highlights the need to review both prescription drugs and supplements when investigating hyperinsulinemic hypoglycemia, that IAS can occur outside typical ethnic groups when permissive HLA alleles are present, and that prompt withdrawal of offending agents with conservative measures often leads to spontaneous remission.
Introduction Paraneoplastic pemphigus (PNP) is an uncommon autoimmune blistering disorder frequently associated with underlying malignancies. Renal cell carcinoma (RCC) is the most common primary renal cancer. It is associated with a wide range of paraneoplastic syndromes, but occasionally presents as a dermatological finding. Case presentation A 56-year-old woman with severe oral and cutaneous lesions was diagnosed with PNP associated with renal cell carcinoma (RCC). The patient was treated with 1 g of rituximab as an infusion, and after two weeks, another dose of 1 g of rituximab was administered in combination with steroids (prednisolone 0.5 mg/kg) until control, then gradually decreasing by 25% each month, until we reached 5 mg/day. The patient achieved complete clinical remission at 6 months; however, long-term outcomes remain unknown because she was subsequently lost to follow-up. Conclusion A comprehensive workup is crucial for diagnosing the underlying tumor associated with this phenomenon. Rituximab- and steroid-based therapies may be effective in controlling PNP symptoms.
Introduction Thyrotoxic periodic paralysis (TPP) is an uncommon endocrine emergency caused by rapid intracellular potassium redistribution during thyrotoxicosis. Carbohydrate exposure is a recognized precipitant, but direct observation of a first paralytic attack developing during supervised diagnostic glucose testing is unusual. Case Presentation A 29-year-old Indian man underwent a supervised oral glucose tolerance test (OGTT) for borderline fasting dysglycemia and episodic postprandial tremulousness. He had also experienced recent weight loss, heat intolerance, and palpitations but had no previous weakness. During the late phase of testing, progressive thigh heaviness evolved into symmetrical flaccid quadriparesis with lower-limb predominance, reduced reflexes, preserved sensation, and no respiratory involvement. Serial samples demonstrated a rapid transition from normal baseline potassium to severe hypokalemia, accompanied by hypophosphatemia and a marked insulin response. Normal acid-base status and suppressed urinary potassium excretion supported transcellular redistribution. Thyroid testing and antibody studies established previously undiagnosed Graves’ disease. Limited potassium replacement, magnesium supplementation, and propranolol produced complete neurological recovery without rebound hyperkalemia. Carbimazole therapy achieved progressive biochemical control, with no recurrent paralysis during 24 weeks of follow-up. Discussion Standardized glucose exposure and serial sampling documented the metabolic transition from normal potassium to paralytic hypokalemia. The episode supports an interaction between endogenous insulin stimulation and thyrotoxic skeletal-muscle susceptibility rather than hyperinsulinemia as an isolated cause. Conclusion Unrecognized thyrotoxicosis should be considered before carbohydrate provocation testing in patients with unexplained hyperadrenergic symptoms.
Anesthesia mumps, also known as acute postoperative sialadenitis, is a rare complication that can occur following general or regional anesthesia. The prevalence of this condition ranges from 0.16% to 0.2%. This case report presents a patient with recurrent bilateral parotid swelling after cesarean section (CS) under spinal anesthesia and its management. A 42-year-old G5P4 woman with gestational diabetes mellitus and antiphospholipid syndrome presented for elective cesarean delivery at 39 weeks and two days of gestation under spinal anesthesia. The patient developed bilateral facial swelling after five hours, which was more evident on the right parotid site. A review by an otolaryngologist confirmed the diagnosis of anesthesia mumps. Anesthesia mumps typically presents as painless swelling of the salivary glands. Salivary duct obstruction and hyperemia are believed to be common causes. This case highlights that anesthesia-induced mumps is a reversible and transient complication that may occur after short surgical procedures, such as cesarean sections. Early recovery was achieved following supportive conservative management, with hydration and analgesia.
Extramedullary plasmacytomas are an uncommon manifestation of multiple myeloma (MM) and may involve a wide range of soft-tissue sites. Superficial involvement of the back is particularly rare and may be mistaken for benign soft-tissue lesions. We report the case of a 59-year-old man with MM diagnosed in April 2024 who was treated with four cycles of bortezomib, cyclophosphamide, and dexamethasone (VCD), achieving remission with disappearance of serum and urine M-protein on immunofixation. In October 2025, the disease relapsed, with early progression despite treatment with daratumumab, prompting initiation of teclistamab therapy. During the course of his disease, he developed two progressively enlarging, painless indurations over the left upper and lower back. Ultrasound demonstrated well-circumscribed heterogeneous hypoechoic subcutaneous masses with internal arterial vascularity on Doppler imaging. Ultrasound-guided core biopsy revealed diffuse infiltration by monoclonal plasma cells expressing CD138 with lambda light-chain restriction, confirming secondary extramedullary plasmacytoma. This case highlights the importance of considering extramedullary plasmacytoma in the differential diagnosis of new superficial soft-tissue masses in patients with MM and underscores the complementary role of ultrasound and histopathological examination in establishing the diagnosis.
Introduction: Adjuvant endocrine therapy for breast cancer - particularly tamoxifen - is commonly associated with weight gain, increased adiposity, and metabolic dysfunction. Lifestyle measures often fail to reverse these effects. Evidence regarding glucagon-like peptide-1 (GLP-1) receptor agonists in this specific context remains limited. Reversal of tamoxifen-associated weight gain with liraglutide has been rarely reported. Case Presentation: A 58-year-old postmenopausal woman with stage IIA, hormone-receptor positive breast cancer experienced marked weight gain after 24 months of tamoxifen therapy, despite diet and exercise. She developed prediabetes, insulin resistance (assessed by Homeostatic Model Assessment of Insulin Resistance [HOMA-IR] 4.9), and dyslipidemia. Liraglutide, a glucagon-like peptide-1 (GLP-1) receptor agonist, was initiated and titrated to 3.0 mg/day. Over 12 months, she lost 18.5 kg, and her metabolic parameters, including HOMA-IR and glycated hemoglobin (HbA1c), normalized. Tamoxifen therapy was continued without adverse effects. Conclusion: This case suggests that liraglutide may reverse substantial tamoxifen-associated weight gain and related metabolic derangements in a breast cancer survivor. The observed improvement exceeded what is typically achieved with lifestyle measures alone. Formal studies are needed to define the role of GLP-1 receptor agonists in managing endocrine therapy–related metabolic complications.
Introduction Ewing sarcoma is a rare, aggressive malignancy of small round blue cells that typically arises from bone or soft tissue. Primary intracranial involvement is exceedingly uncommon, particularly in adults, and is associated with diagnostic challenges, high recurrence rates, and poor prognosis. Limited data exist regarding optimal management strategies for recurrent intracranial Ewing sarcoma in adult patients. Case Summary We report the case of a 24-year-old pregnant female who presented with progressive headaches, vomiting, and left parietal swelling. MRI revealed a ring-enhancing lesion in the left parietal region with surrounding edema. She underwent surgical resection, and histopathology demonstrated sheets of small round blue cells with strong CD99 positivity. Fluorescence in situ hybridization using an EWSR1 break-apart probe confirmed EWSR1 gene rearrangement; however, fusion partner identification was not available. Based on the morphologic and immunohistochemical features in conjunction with EWSR1 rearrangement, a diagnosis of EWSR1-rearranged round cell sarcoma consistent with Ewing sarcoma was made. Adjuvant therapy was delayed due to pregnancy. Despite subsequent multimodal treatment including surgery, VAC-IE chemotherapy, and radiotherapy, the patient developed local recurrence involving the parietal bone within one year and died four months after recurrence. Conclusion This case highlights the aggressive nature and high recurrence risk of primary intracranial Ewing sarcoma in adults. Diagnostic confirmation requires histopathological and molecular analysis, as radiological findings may mimic other central nervous system tumors. Early diagnosis, timely multimodal therapy, and close surveillance are critical; however, outcomes remain poor, underscoring the need for improved and individualized treatment strategies for adult intracranial Ewing sarcoma.
Introduction Congenital pulmonary airway malformation (CPAM) is a spectrum of cystic lung lesions caused by abnormal fetal airway development. It presents diagnostic and therapeutic challenges due to variable manifestations and potential complications. Surgical management aims to balance complete lesion removal with lung function preservation. This report highlights a rare case of CPAM coexisting with pectus carinatum, emphasizing individualized surgical planning. Case Presentation An 11-year-old boy, prenatally diagnosed with right lung CPAM, was asymptomatic until imaging revealed a large (10 × 8.7 × 9 cm), multiseptated cystic lesion in the right lower lobe consistent with Stocker-type I CPAM. Physical examination showed symmetric pectus carinatum (PC) without cardiopulmonary compromise. Pulmonary function tests indicated mild restrictive lung disease. The patient underwent right posterolateral thoracotomy with wedge resection of basal lung segments, preserving lung parenchyma. Histopathology confirmed CPAM. Postoperative recovery was uneventful, with no recurrence or complications during 44 weeks of follow-up. The PC showed favorable response to the orthotic treatment. Conclusion Wedge resection is a safe, effective alternative to lobectomy in selected older children with localized CPAM. Long-term follow-up with pulmonary function tests and imaging is essential. The rare association of CPAM with PC expands related anomalies and emphasizes the need for individualized, multidisciplinary management.
Background Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disorder characterized by high-spiking fever, arthritis, rash, and systemic inflammation. Because there is no single diagnostic test, diagnosis relies on compatible clinical features, fulfillment of classification criteria, and exclusion of infectious, malignant, and autoimmune conditions. Atypical cutaneous manifestations may further delay recognition, particularly in resource-limited settings. Case Presentation We report the case of a 22-year-old Ethiopian woman who presented with a five-month history of inflammatory polyarthritis, daily spiking fever, sore throat, and a persistent hyperpigmented scaling rash rather than the classic evanescent salmon-colored eruption. Laboratory evaluation demonstrated marked neutrophilic leukocytosis, elevated inflammatory markers, abnormal liver enzymes, and hyperferritinemia (1,675 μg/L). Echocardiography revealed mitral valvulitis with mild pericardial effusion. Infectious, autoimmune, and rheumatologic conditions, including acute rheumatic fever, rheumatoid arthritis, systemic lupus erythematosus, viral hepatitis, HIV infection, and other competing diagnoses, were excluded through clinical assessment and laboratory investigations. The patient fulfilled the Yamaguchi classification criteria for AOSD, while markedly elevated serum ferritin provided supportive evidence for the diagnosis. She was treated with intravenous pulse methylprednisolone followed by oral prednisolone and methotrexate, resulting in rapid resolution of fever, marked improvement in joint symptoms, fading of the skin lesions, and complete resolution of the pericardial effusion at one-month follow-up. Conclusion This case highlights the diagnostic challenges of AOSD when atypical dermatologic manifestations occur, particularly in resource-limited settings where infectious diseases are frequently prioritized in the differential diagnosis. Persistent hyperpigmented scaling lesions should not exclude consideration of AOSD when accompanied by characteristic systemic features. Although elevated serum ferritin is not diagnostic, it can provide valuable supportive evidence when interpreted alongside clinical findings and after exclusion of alternative diagnoses.
Background Troponin elevation is frequently observed in patients presenting with supraventricular tachycardia (SVT) and is commonly attributed to transient demand ischemia. However, its clinical significance remains uncertain, and alternative mechanisms such as coronary vasospasm may be underrecognized. Case Presentation We report the case of a 77-year-old male presenting with a first episode of SVT, successfully terminated with adenosine. Adenosine successfully restored sinus rhythm with partial regression of the ischemic ST-segment changes. Initial high-sensitivity troponin was elevated but demonstrated a significant rise on the following day, accompanied by new T-wave inversions in the lateral leads. Given the dynamic electrocardiographic changes and biomarker progression, coronary angiography was performed, revealing coronary vasospasm of the left anterior descending artery without obstructive coronary disease. The spasm resolved with intracoronary nitroglycerin administration, suggesting the diagnosis of coronary vasospasm. Conclusion Coronary vasospasm should be considered in patients with SVT and evolving troponin elevation, particularly when accompanied by dynamic electrocardiographic changes. Recognition of this mechanism is essential, as it has important diagnostic and therapeutic implications and requires a tailored management approach.
Introduction Overlap syndrome refers to the coexistence of more than one immune-mediated liver disease in a single patient. Primary sclerosing cholangitis–autoimmune hepatitis (PSC–AIH) overlap is a rare but clinically important entity, as delayed recognition may lead to progressive liver injury, cirrhosis, and liver failure. Differentiating this condition from isolated PSC or AIH remains challenging due to overlapping clinical, biochemical, and radiological features. Case Presentation A 20-year-old male presented with progressive jaundice, pruritus, acholic stools, and dark urine for one month. Laboratory evaluation revealed marked cholestasis with significantly elevated alkaline phosphatase and transaminases, hyperbilirubinemia, and coagulopathy, while viral and metabolic causes were excluded. Magnetic resonance cholangiopancreatography demonstrated characteristic intrahepatic biliary duct irregularities consistent with primary sclerosing cholangitis. However, disproportionately elevated alanine aminotransferase and serum immunoglobulin G levels prompted further evaluation. Liver biopsy revealed features of both diseases, including interface hepatitis with chronic portal inflammation and plasma cell infiltration, along with bile duct injury, ductopenia, and onion-skin fibrosis. Based on histology and diagnostic scoring, a diagnosis of PSC–AIH overlap syndrome was established. The patient was treated with ursodeoxycholic acid, corticosteroids, and azathioprine, resulting in marked clinical and biochemical improvement on follow-up. Conclusion This case highlights the importance of considering PSC–AIH overlap syndrome in young patients with cholestatic liver disease and unexpectedly elevated transaminases or immunoglobulin G levels. Early recognition and combined immunosuppressive and supportive therapy can lead to favorable outcomes and may prevent irreversible liver damage.
Introduction Ileosigmoid knotting (ISK) is a rare, life-threatening form of closed-loop bowel obstruction predominantly reported in volvulus-endemic regions, characterized by rapid progression to gangrene and high mortality. This report details a severe case managed with a specialized two-stage approach. Case Presentation A 62-year-old man presented with a 36-hour history of severe crampy abdominal pain, bilious vomiting, and obstipation. Physical examination revealed a distended, tender abdomen, with vitals demonstrating shock. Laboratory results showed a left shift on the complete blood count. Abdominal X-ray suggested obstruction. Management and Outcome Emergent exploratory damage control laparotomy revealed gangrene of the sigmoid colon and nearly the entire small bowel, sparing only 70 cm of the proximal jejunum and 10 cm of the distal ileum. Rapid resection without immediate restoration of continuity was completed within 30 minutes due to intraoperative shock. Temporary abdominal closure was achieved using a Bogota bag. Definitive care with jejunoileal end-to-end anastomosis and an end colostomy was completed in a second-look procedure 12 hours later, after adequate resuscitation. The patient was successfully transferred to an intensive care unit for specialized care. Conclusion This case underscores the utility of damage control surgery in managing complicated compound volvulus with hemodynamic instability and massive necrosis. Early clinical awareness is essential to prevent extensive gangrene, reduce mortality, and optimize survival in resource-constrained settings.
Securing the airway in patients with restricted cervical mobility is challenging because cervical movement should be minimized during tracheal intubation. We report the technical feasibility of a catheter-assisted modification of a standard endotracheal tube in a 45-year-old woman undergoing cervical spine surgery. A sterile flexible epidural catheter was passed through the tube lumen and redirected along its external surface. During direct laryngoscopy, gentle traction on the externalized catheter produced controlled anterior deflection of the tube tip, facilitating alignment with the laryngeal inlet. Tracheal intubation was successfully achieved on the first attempt without alternative airway devices. No device-related complications occurred during intubation, and postoperative follow-up showed only mild transient sore throat, with no other airway-related complications. This case demonstrates the technical feasibility of this catheter-assisted approach as a potential adjunct in selected patients. However, a single case cannot establish safety, comparative effectiveness, or generalizability, and further studies are required.
Introduction Rhabdomyolysis is characterized by skeletal muscle breakdown resulting in the release of intracellular contents into the circulation. Although trauma, medications, and metabolic disorders are common causes, viral infections are increasingly recognized as potential triggers. Case Presentation A 26-year-old previously healthy male presented with a 2-day history of abdominal pain, diarrhea, vomiting, bilateral thigh pain, and dark urine. Laboratory evaluation demonstrated severe rhabdomyolysis with a peak creatine phosphokinase (CK) level of 25,036 U/L, elevated aminotransferases, and preserved renal function. A stool gastrointestinal multiplex polymerase chain reaction (PCR) panel was positive for adenovirus and did not identify other enteric pathogens. Alternative etiologies, including significant recent exertion, medication-related muscle injury, toxin exposure, autoimmune myopathy, and inherited metabolic disorders, were considered but were not supported by the clinical history or disease course. The patient was treated with early intravenous isotonic fluid 1L every 6 hours and experienced progressive biochemical and clinical improvement without developing acute kidney injury. Discussion Adenovirus-associated rhabdomyolysis is an uncommon but increasingly recognized clinical entity in adults. The pathogenesis may involve direct viral muscle injury, immune-mediated inflammation, and contributory factors such as dehydration associated with gastrointestinal illness. Although adenovirus was considered the most likely precipitating factor in this case, a definitive causal relationship cannot be established. Conclusion This case highlights probable adenovirus-associated rhabdomyolysis in an immunocompetent young adult presenting predominantly with gastrointestinal symptoms and preserved renal function despite marked creatine kinase elevation. Early recognition, careful evaluation of alternative etiologies, and prompt supportive management are important for preventing complications and achieving favorable outcomes.
Background:Strongyloides stercoralis is an intestinal nematode with a unique autoinfective life cycle that enables decades-long persistence in the human host. Its gastrointestinal manifestations may resemble inflammatory bowel disease (IBD), creating a clinically important diagnostic pitfall. Misdiagnosis is particularly hazardous because corticosteroids or other immunosuppressive therapies may precipitate Strongyloides hyperinfection syndrome or disseminated disease. Case Presentation:We report a 52-year-old immunocompetent woman from northern Iran who presented with a 20-day history of persistent watery inflammatory diarrhea, severe hypokalemia (2.4 mEq/L), leukocytosis, and thrombocytosis. Serial stool examinations for ova and parasites were repeatedly negative for larvae; however, fecal leukocytes and erythrocytes, together with erosive duodenopathy, initially raised concern for IBD. Duodenal biopsy established the diagnosis by demonstrating marked eosinophilic infiltration of the lamina propria, reaching 45-50 eosinophils per high-power field, and rhabditiform larvae within the mucosal crypts. The larvae showed morphologic features supporting Strongyloides stercoralis, including a short buccal cavity/canal and a characteristic rhabditiform esophagus. Molecular testing, serology, and independent expert parasitologist confirmation were not performed, which represents a diagnostic limitation. The patient was treated with oral ivermectin plus adjunctive albendazole. The addition of albendazole was an individualized, non-standard clinical decision rather than a routine evidence-based recommendation. Her symptoms improved rapidly, and she remained asymptomatic at one-month follow-up. Conclusion:This case emphasizes the clinical and histopathological overlap between duodenal strongyloidiasis and IBD, particularly when stool parasitology is unrevealing. The novelty of this report lies primarily in its didactic value for clinicians in endemic regions: Strongyloides should remain in the differential diagnosis of IBD-like presentations with unexplained eosinophilic mucosal inflammation before immunosuppressive therapy is considered.
Background Hydatid cyst disease is a zoonotic parasitic infection caused by Echinococcus species and remains endemic in many regions worldwide, particularly in North Africa and the Mediterranean basin. The liver is the most commonly affected organ due to portal dissemination of the parasite. Although hepatic hydatid cysts are frequently encountered, vascular complications are uncommon, and portal vein invasion represents an exceptionally rare manifestation. This complication may lead to portal vein thrombosis, cavernous transformation, portal hypertension, and potentially life-threatening complications including anaphylaxis. Case Presentation We report the case of a 56-year-old male with a prior history of surgery for hepatic hydatid disease who presented with portal vein invasion mimicking portal vein thrombosis. Imaging studies revealed a hydatid cyst located in segment IV of the liver associated with portal venous obstruction and cavernomatous transformation. Color Doppler ultrasonography demonstrated absence of blood flow within the portal vein and collateral venous circulation consistent with portal cavernoma. Contrast-enhanced computed tomography clearly demonstrated communication between the multiloculated hydatid cyst and the portal vein, with multiple daughter vesicles extending from the portal trunk to the segmental portal branches causing vascular occlusion. Despite evidence of intraportal rupture, the patient did not develop anaphylactic shock or systemic allergic manifestations. Discussion Portal involvement in hepatic echinococcosis may occur through extrinsic compression, inflammatory invasion of the vascular wall, or direct rupture of the cyst into the portal circulation. Although all echinococcus larvae initially pass through the portal venous system, portal vein invasion remains exceedingly rare, likely due to the high pressure within the portal circulation. Chronic portal obstruction may result in extrahepatic portal vein obstruction, portal hypertension, and cavernous transformation. Radiologic imaging, particularly contrast-enhanced computed tomography, plays a crucial role in differentiating portal hydatid disease from conventional portal vein thrombosis by demonstrating intravascular daughter cysts and direct cysto-portal communication. Surgical management remains the treatment of choice and should address both the hydatid cyst and complications related to portal hypertension. Conclusion Portal vein invasion by hydatid cysts is a rare but serious complication of hepatic echinococcosis that may mimic portal vein thrombosis. Recognition of this entity is essential, particularly in endemic regions and in patients with previous hydatid surgery, in order to optimize diagnosis and management. Early identification through advanced imaging modalities may help prevent severe complications and improve clinical outcomes.
Introduction Splenic complications of Plasmodium falciparum malaria are rare but potentially life-threatening. While splenic rupture is more commonly reported, high-grade splenic injury with associated vascular compromise and infarction is less well-described and may present diagnostic and management challenges. Case Presentation A 32-year-old male presented with a three-day history of high-grade fever, chills, and progressive abdominal symptoms, including acute left upper quadrant pain radiating to the back. On examination, he was febrile with signs of peritonism, including involuntary guarding and rigidity. Laboratory investigations confirmed P. falciparum infection with severe anemia (Hgb 5.1 g/dL). A diagnostic abdominal tap yielded hemorrhagic fluid. Contrast-enhanced computed tomography (CT) demonstrated a high-grade splenic injury (AAST Grade IV) characterized by multifocal splenic infarctions, perisplenic hematoma, and involvement of the splenic hilum. Notably, no active contrast extravasation was identified. Despite this, the patient developed clinical deterioration with worsening hypotension, necessitating emergency splenectomy. Intraoperative findings confirmed a major splenic laceration with 2000 mL of hemolyzed blood in the peritoneum. The patient received intraoperative and postoperative transfusions. The postoperative course was uneventful, and the patient was discharged following appropriate vaccinations and antimalarial therapy. Conclusion High-grade splenic injury with infarction should be considered in malaria patients presenting with acute abdominal symptoms. Early recognition and prompt surgical intervention are critical, particularly in patients with clinical deterioration despite non-diagnostic imaging findings for active bleeding.
Ureteral endometriosis is a rare form of urinary tract endometriosis that may cause silent ureteral obstruction and irreversible renal damage. Mixed intrinsic and extrinsic ureteral involvement is uncommon, and right-sided disease is less frequently reported. We report the case of a 39-year-old woman with known endometriosis who presented with recurrent severe right flank pain. Imaging revealed right uretero-hydronephrosis associated with a right adnexal endometrioma and distal ureteral involvement. Diagnostic ureteroscopy could not be safely performed because of marked narrowing of the right ureteral meatus, and a double-J stent was initially placed. Renal scintigraphy subsequently demonstrated a nonfunctioning right kidney with preserved contralateral function. Definitive surgery was performed after recurrent symptoms and imaging progression documented persistent distal ureteral disease. The patient underwent laparoscopic right nephroureterectomy with open partial cystectomy and right salpingo-oophorectomy. Histopathological examination confirmed multifocal mixed intrinsic and extrinsic ureteral endometriosis associated with end-stage chronic nephritis and a right ovarian endometrioma. This case highlights the diagnostic difficulty of ureteral endometriosis, particularly when right-sided and mixed intrinsic-extrinsic disease is present, and emphasizes the importance of early functional assessment and multidisciplinary management to prevent irreversible renal loss.
Introduction Cannabinoid hyperemesis syndrome (CHS) is a debilitating disorder of chronic, heavy cannabis users characterized by cyclic severe nausea, vomiting, and abdominal pain that is classically relieved by hot baths. Standard antiemetics often fail, and dysregulation of endocannabinoid signaling with involvement of heat-sensitive TRPV1 channels has been proposed. Topical capsaicin - a TRPV1 agonist - has been reported in small series and case reports to reproduce the hot-water effect and rapidly relieve symptoms. Case presentation A 28-year-old man with daily high-potency cannabis use (∼2 g/day for 7 years) presented to the emergency department with 48 hours of intractable non-bilious vomiting (∼20 episodes/day), severe periumbilical cramping (8/10), and a history of similar episodic flares relieved by prolonged hot showers. Initial ED therapy (IV fluids, ondansetron, metoclopramide, pantoprazole) produced minimal benefit; labs showed hypokalemic, hypochloremic metabolic alkalosis and pre-renal azotemia, with otherwise unremarkable imaging and enzymes. After informed consent, ∼2 g of 0.1% topical capsaicin cream was applied across the abdomen. The patient experienced an acute burning sensation for ∼15–30 minutes; retching ceased within 10 minutes, pain fell to 2/10 by 30 minutes and resolved by 90 minutes, and he tolerated oral intake. Vital signs normalized and electrolytes/renal function returned to baseline at 48-hour follow-up. He was discharged with counseling on cannabis cessation and a capsaicin tube for prodromal use. Conclusion Topical capsaicin produced rapid, durable symptom resolution in this case of refractory CHS with only transient local discomfort. The effect is plausibly mediated by TRPV1 activation followed by peripheral desensitization, recapitulating the therapeutic hot-water response. Given consistent positive signals from case reports, series, and small pilot data, topical capsaicin is a low-risk, accessible adjunct for refractory CHS, but larger controlled studies are needed to define optimal dosing, duration, and long-term outcomes.
Background Hepatic alveolar echinococcosis (HAE), although rare, is a potentially life-threatening parasitic disease when left untreated. Following infection, the disease typically progresses through a prolonged asymptomatic latent phase, resulting in delayed clinical presentation in most patients. The radiological appearance of HAE may closely resemble that of primary hepatic malignancies, thereby creating significant diagnostic challenges. Case Presentation We report the case of a 49-year-old previously healthy man who presented with cholestatic liver enzyme abnormalities and imaging findings suggestive of biliary obstruction. Cross-sectional imaging revealed a calcified hilar hepatic mass with biliary dilatation and multiple heterogeneous liver lesions, strongly suggestive of perihilar cholangiocarcinoma. Endoscopic retrograde cholangiopancreatography (ERCP) with brush cytology was initially negative for malignancy. Subsequent magnetic resonance imaging (MRI) demonstrated an infiltrative multiloculated solid-cystic hepatic lesion involving the right hepatic lobe and hilar structures. Definitive diagnosis was ultimately established through liver biopsy and positive serology for alveolar echinococcosis. The patient was treated with albendazole followed by extended right hepatectomy, with favorable postoperative recovery. Conclusion HAE may closely mimic perihilar cholangiocarcinoma, exhibiting similar clinical and radiological characteristics. Maintaining a high index of suspicion and obtaining adequate tissue biopsies are essential for establishing an accurate diagnosis and ensuring timely, appropriate management.
