American Journal of Case Reports最新文献

BACKGROUND Solitary fibrous tumors (SFTs) are rare mesenchymal neoplasms most commonly originating in the pleura but may also arise in extrapleural sites, including the head and neck region. Subcutaneous SFTs in the chin area are exceptionally rare; only a few cases have been reported. CD34 and Bcl-2 are commonly used immunohistochemical markers in the initial diagnostic workup, and their co-expression is strongly suggestive of an SFT. However, CD34 negativity can be misleading and complicate diagnosis. Accurate identification of SFTs is potentially difficult because of histologic variability and overlap with other spindle cell tumors. CASE REPORT We encountered a rare subcutaneous SFT in the chin of a 22-year-old woman who presented with facial asymmetry and a tender mass. Imaging revealed a well-circumscribed, contrast-enhancing lesion in subcutaneous soft tissue over the anterior mandible. Histologically, the tumor lacked typical staghorn vasculature but showed spindle cell proliferation within a fibrous stroma. Immunohistochemistry demonstrated strong nuclear STAT6 positivity; expression of CD99, Bcl-2, and SMA; and focal H-caldesmon staining. CD34, S100, and ALK1 displayed negative staining. Based on these findings, the patient was diagnosed with an SFT. CONCLUSIONS This case highlights the importance of considering SFT in the differential diagnosis of spindle cell tumors in the head and neck region. It also underscores the critical role of immunohistochemistry, particularly STAT6 staining, in distinguishing SFT from histologic mimics. Vigilant follow-up remains essential, especially in atypical or CD34-negative cases, given their potential for aggressive behavior.

BACKGROUND Bacterial purulent pericarditis is rare and can be fatal if not treated appropriately. Streptococcus anginosus can cause invasive and cryptogenic infections, and purulent pericarditis is an uncommon presentation. Alcohol abuse can be a risk factor for abscesses caused by this agent. Cell-free DNA testing is a noninvasive method that has great potential in cases of serious infections in which pathogens are not easily identifiable by traditional microbiological techniques. CASE REPORT A 27-year-old man reported alcohol abuse and was hospitalized for acute pericarditis without signs of severity. He developed cardiac tamponade on the fifth day of hospitalization, requiring emergency pericardiocentesis. A significant persistent pericardial effusion was observed. Videopericardiectomy revealed a large amount of fibrin and purulent secretion in the pericardial sac. Additional tests and cultures did not identify systemic disease or an etiological agent. A cell-free DNA assay identified S. anginosus. He was discharged after 4 weeks of broad-spectrum antimicrobial therapy. There was no progression to constrictive pericarditis. CONCLUSIONS We present a case of purulent bacterial pericarditis with a cryptogenic focus in a young adult patient with a history of alcohol, marijuana, and e-cigarette abuse. He developed cardiac tamponade but received rapid and appropriate in-hospital therapeutic support, with clinical recovery within a few weeks. Alcohol and smoking may have facilitated bacterial translocation from the oropharynx to the bloodstream and then to the pericardium. S. anginosus was identified only by molecular research.

BACKGROUND Philadelphia chromosome (Ph)-negative B-cell acute lymphoblastic leukemia (B-ALL) is usually treated with intensive chemotherapy, presenting challenges for Jehovah's Witness patients who refuse blood products due to religious beliefs. These regimens often lead to severe cytopenias that require transfusion support. New targeted therapies (eg, inotuzumab ozogamicin and blinatumomab) have become effective options with lower toxicity, particularly for older or frail patients; these benefits may extend to the Jehovah's Witness population. CASE REPORT A 44-year-old Jehovah's Witness man was diagnosed with Ph-negative B-ALL. His comorbidities included hypertension, atrial fibrillation, and a newly identified reduced ejection fraction of 30% to 35%, which precluded the use of anthracyclines. Due to his refusal of blood products and cardiac limitations, he was treated with a chemotherapy-free regimen consisting of inotuzumab induction followed by blinatumomab consolidation. Supportive care included epoetin alfa, romiplostim, iron, and vitamin supplementation. The patient tolerated induction well, with minimal cytopenias and no serious adverse effects. After 2 cycles of inotuzumab, he achieved complete morphologic remission and minimal residual disease (MRD) negativity according to ClonoSEQ. He is currently receiving consolidation with blinatumomab. CONCLUSIONS This is the first known reported case that demonstrates the feasibility and effectiveness of a chemotherapy-free induction strategy using inotuzumab and blinatumomab for frontline treatment of Ph-negative B-ALL in Jehovah's Witness patients. It shows that MRD negativity can be safely achieved without cytotoxic chemotherapy or transfusion support and supports the use of the ALLIANCE A041703 trial regimen as a treatment model for this unique and underserved patient group.

BACKGROUND The most common vascular lesions of the liver are hepatic hemangiomas (HH), which are generally asymptomatic and do not require medical intervention. However, when the lesions are large or multiple, they can be a significant clinical problem. This report describes a unique case of an elderly patient with isolated hepatic hemangiomatosis associated with the onset of consumptive coagulopathy and hemolytic anemia, ultimately resulting in liver failure and death. CASE REPORT A 74-year-old man presented with jaundice, progressive weight loss, fatigue, lower-limb edema, and recurrent epistaxis. Laboratory findings revealed normocytic anemia, thrombocytopenia, hyperbilirubinemia, hypoalbuminemia, and elevated liver enzymes. Abdominal ultrasound (US) and computed tomography (CT) demonstrated hepatomegaly with multiple hypoechoic and hypodense focal lesions, initially raising suspicion of metastatic disease. Colonoscopy and upper gastrointestinal endoscopy showed no evidence of malignancy. A core-needle biopsy of the hepatic lesions, complemented by positron emission tomography-computed tomography (PET-CT) and magnetic resonance imaging (MRI), confirmed the benign nature of the lesions, consistent with isolated hepatic hemangiomatosis. The patient was managed symptomatically and discharged home with recommendations for continued outpatient monitoring and urgent consultation in the event of any clinical deterioration. Unfortunately, after 3 months of relative symptoms improvement, he developed gastrointestinal bleeding and acute liver failure, which ultimately resulted in his death. CONCLUSIONS Hepatic vascular lesions are diagnostically challenging; definitive diagnosis requires correlation of clinical, imaging, and histopathological findings, as well as multidisciplinary consultation. The clinical course of hepatic hemangiomatosis ranges from benign to life-threatening, with potential complications such as hepatic failure or coagulopathy.

BACKGROUND The anion gap is a critical parameter in the clinical assessment of acid-base disorders. While metabolic acidosis with an elevated anion gap is commonly encountered, cases involving a negative anion gap are rare and have been reported in the context of hypoalbuminemia, severe hyperkalemia, bromide intoxication, and laboratory error. Notably, metabolic alkalosis as a cause of negative anion gap has been rarely described in the literature. CASE REPORT A 73-year-old woman with a 4-year history of interstitial pulmonary fibrosis and a 1-year history of coronary artery disease had been taking oral spironolactone for the past year. Six months before admission, torasemide was added to her regimen in combination with spironolactone. Five days prior to admission, she developed progressive dyspnea and respiratory failure. Initial investigations revealed hypokalemia, hyponatremia, metabolic alkalosis (HCO₃⁻=61.6 mmol/L), and a negative anion gap (-9.00 mmol/L), which remained negative after albumin correction (-6.35 mmol/L). Further evaluation identified loop diuretic overuse as the primary cause of severe metabolic alkalosis and negative anion gap. The application of targeted next-generation sequencing (t-NGS) successfully identified the infectious pathogen responsible for the patient's clinical deterioration, thereby guiding appropriate antimicrobial therapy. CONCLUSIONS This case illustrates the diagnostic and educational value of recognizing a negative anion gap as a rare but physiologically predictable artifact of severe chloride-depletion alkalosis, underscoring the importance of mechanism-based interpretation in complex acid-base disorders.

BACKGROUND Immune checkpoint inhibitor (ICI) myocarditis imposes immunotherapy discontinuation due to concerns of poor outcomes. We present a case of ICI cardiomyopathy for which ICI re-challenge was safely performed. According to the World Health Organization (WHO), only 13 cases of takotsubo cardiomyopathy (TTS) have been associated with ICI use. We report a rare case of this. CASE REPORT A 64-year-old woman with history of stage IV squamous cell carcinoma of the right lung presented with symptoms of heart failure and worsening dyspnea. Her symptoms started 2 weeks after her first round of chemotherapy with carboplatin, paclitaxel, and pembrolizumab. Electrocardiography (EKG) revealed right bundle branch block with ST elevations, troponin peaked at 424 ng/L, and proBNP 865 pg/mL. A transthoracic echocardiogram (TTE) showed left ventricle ejection fraction (LVEF) of 35% to 39% along with akinesis of all the mid- to apical left ventricle (LV) wall segments. CONCLUSIONS Not all ICI-related heart failure is myocarditis. This case highlights the utility of CMR and endomyocardial biopsy to aid diagnosis of TTS. TTS and ICI myocarditis may appear similar on CMR, with prominent edema, although the regional distribution may help finalize the diagnosis. Endomyocardial biopsies can be helpful to identify pro-inflammatory macrophages as possible mediators in the association between oncology treatment and development of TTS. This interesting case highlights the utility of further advanced cardiac testing before making the diagnosing of ICI myocarditis and potentially withholding life-saving cancer therapy.

BACKGROUND The PreserFlo MicroShunt is a subconjunctival drainage device increasingly used to manage open-angle glaucoma. While its safety profile is considered favorable compared with that of trabeculectomy, rare postoperative complications, including corneal surface pathology, can still be encountered. To our knowledge, corneal dellen has not been previously reported following PreserFlo implantation. CASE REPORT A 15-year-old boy with pathological myopia and Stickler syndrome underwent PreserFlo MicroShunt implantation for refractory secondary open-angle glaucoma. Two weeks postoperatively, he developed a temporal area of corneal stromal thinning consistent with dellen, located adjacent to an elevated bleb in the horizontal plane. Conservative therapy, including a switch from prednisolone acetate to loteprednol etabonate, intensive preservative-free lubrication, autologous serum drops, and bandage contact lens placement, was initiated. Full corneal re-epithelialization and recovery were achieved within 1 week, without compromising bleb function or intraocular pressure control. CONCLUSIONS Corneal dellen is a potential, although previously undocumented, complication of PreserFlo MicroShunt surgery, particularly when bleb elevation is prominent in areas not protected by the eyelid. Prompt recognition and targeted conservative therapy may allow for complete recovery while preserving device function and visual outcomes. Surgeons should monitor the ocular surface for this complication and adjust postoperative management accordingly.

BACKGROUND Scars, particularly those that are hypertrophic and retracting, are a major difficulty in dermatological and plastic surgery. Laser therapy offers a versatile approach to managing hypertrophic lip scars, addressing aspects of scar tissue, such as vascularity and collagen. CASE REPORT This case report describes a 43-year-old woman treated for a hypertrophic, retracting lip scar using fractional CO₂ laser (10 W, 1500 µs dwell time, D-pulse, 500 µm spacing, stack 1, double pass). Two sessions, 56 days apart, led to marked aesthetic and psychosocial improvement without complications. Post-treatment care included 7 days of antibiotic ointment, followed by sun protection and nightly silicone gel. Images were captured before and after the second treatment. During the procedure, the patient reported a perceived pain level of 3 on a scale of 1 to 5, indicating a moderate and tolerable level of discomfort. The Modified Vancouver Scar Scale (mVSS) chart indicated an overall improvement in scar characteristics, especially in pliability, vascularity, and pigmentation, with minimal changes in height; pain and pruritus levels remained unchanged from before the therapy. After treatments, the patient expressed extreme satisfaction with the results achieved. She reported being "extremely content and satisfied" with the improvement in her scar after laser treatment. No significant adverse effects were observed. The estimated reduced daily activity time was approximately 1 week after each session, with normal activities resumed shortly thereafter. CONCLUSIONS This case highlights the potential of CO₂ laser treatment in managing a complex hypertrophic and retracting scar, leading to notable esthetic improvement and a positive impact on the patient's emotional well-being.

BACKGROUND Achondroplasia is an autosomal dominant skeletal dysplasia and the most common genetic cause of dwarfism, characterized by shortened pedicles, thickened laminae, and reduced interpedicular distances that lead to congenital narrowing of the spinal canal. These anatomical abnormalities frequently result in lumbar spinal stenosis (LSS) requiring surgical management. Surgical treatment typically involves decompression of neural elements, with or without fusion, to restore canal patency and prevent postoperative deformity. However, adjusting the surgical approach to the unique anatomical constraints of achondroplasia remains critical for optimizing clinical outcomes. CASE REPORT We present a rare case of a 55-year-old woman with achondroplasia who presented with a 5-year history of progressively worsening low back pain, bilateral foot drop, and neurogenic claudication after walking short distances. Magnetic resonance imaging (MRI) demonstrated critical lumbar spinal stenosis at L2-L3, L3-L4, and L4-L5. She underwent multilevel laminectomy under general anesthesia with intraoperative fluoroscopic guidance and high magnification. Postoperatively, she was mobilized within 3 hours and discharged the same day without complications. At 4-week follow-up, the muscle strength of the anterior tibialis and quadriceps had improved to 4/5 on the MRC scale, patellar reflexes were normalized, and lower-limb sensation showed marked recovery. CONCLUSIONS This case highlights the importance of intraoperative precision and an appropriate surgical approach in managing lumbar spinal stenosis associated with achondroplasia. Decision-making, detailed preoperative planning with assessment of imaginary findings, intraoperative use of microsurgical techniques, and postoperative care are important in minimizing complications and optimizing clinical outcomes.

BACKGROUND Although current guidelines classify prolonged cardiopulmonary resuscitation (CPR) as a relative contraindication to thrombolytic therapy, this treatment may serve as a viable reperfusion strategy for patients with ST-segment elevation myocardial infarction (STEMI) who achieve return of spontaneous circulation (ROSC) when primary percutaneous coronary intervention (PCI) cannot be performed in a timely manner or is unavailable. This case series evaluated the safety and efficacy of thrombolytic therapy after ROSC in 12 patients with STEMI. CASE REPORT Twelve patients with STEMI (9 men and 3 women; mean age, 64.33 years) who had just returned to continuous spontaneous circulation via CPR received thrombolytic therapy at 3 hospitals (Hospital I, 1 patient; Hospital II, 9 patients; Hospital III, 2 patients) between April 2007 and February 2021. Electrocardiography showed anterior wall elevation in 66.7% and inferior wall elevation in 33.3% of patients; the ischemic site was independent of CPR duration (P=0.890). CPR duration was associated with a higher incidence of rib fractures (P=0.02) but not bleeding complications (P=0.160). Binary logistic regression analysis showed no correlation between CPR duration and grade of bleeding complications (odds ratio=1). Of the 8 long-term survivors, 1 had mild neurological sequelae. CONCLUSIONS Our findings support the safety and feasibility of post-ROSC thrombolysis as a therapeutic option for patients with STEMI after comprehensive clinical evaluation, particularly in resource-limited settings where primary PCI is unavailable. This approach achieves restoration of coronary perfusion and has a potential neuroprotective effect in survivors of cardiac arrest.