AME Case Reports最新文献

Background: Left ventricular free wall rupture (LVFWR) is a severe complication of acute myocardial infarction that frequently leads to cardiac tamponade and circulatory collapse. Prompt surgical repair and mechanical circulatory support are essential for patient survival. The Impella device provides left ventricular unloading and is increasingly used after cardiac surgeries. However, it carries the risks of bleeding and thromboembolism. Heparin-induced thrombocytopenia (HIT) may be an important contributor to thromboembolic risk. Diagnosing HIT in an acute postoperative setting using the Impella support is particularly challenging. Herein, we present a case of thromboembolism associated with HIT following Impella support after surgical repair of an LVFWR.

Case description: A 72-year-old man presented with chest pain. During preparation for percutaneous coronary intervention, the patient developed cardiac arrest and was resuscitated using extracorporeal membrane oxygenation (ECMO) and intra-aortic balloon pumping (IABP). Coronary angiography revealed left circumflex artery occlusion, and echocardiography revealed pericardial effusion. Surgical repair was performed using cardiopulmonary bypass. Intraoperative findings revealed left ventricular rupture in the circumflex artery area. Due to severe ventricular dysfunction, ECMO was continued, and IABP was initially chosen. On postoperative day (POD) 1, after stabilization of bleeding, the IABP was upgraded to Impella for ventricular unloading. ECMO was discontinued on POD 3, and Impella was maintained until POD 9. After Impella was removed, angiography revealed occlusion of both iliac arteries. Bilateral surgical thrombectomy was performed. A latex immunoturbidimetric assay confirmed HIT antibody positivity. Anticoagulation therapy was subsequently switched from heparin to argatroban.

Conclusions: This case illustrates the difficulty in diagnosing HIT in patients on Impella support in the acute postoperative period after cardiac surgery. Clinicians should be vigilant for Impella-associated thromboembolism and understand its possible mechanisms to ensure appropriate diagnosis and prevention.

Background: Chronic pulmonary aspergillosis (CPA) is often asymptomatic; however, respiratory symptoms may trigger detection of the disease. Some asymptomatic patients are diagnosed as having CPA after closer examination of abnormalities on chest computed tomography (CT). We report a rare case in which CPA was diagnosed after surgery for spontaneous pneumothorax.

Case description: An 86-year-old man with old pulmonary tuberculosis and autoimmune hepatitis on oral prednisolone treatment developed a right-sided pneumothorax with persistent air leak and worsening subcutaneous emphysema. Despite thoracic drainage, his condition deteriorated. Chest CT showed numerous cysts and cavities with a small nodule in the right lung and interstitial changes bilaterally, although the precise site of the air leak was unclear. Thoracoscopy revealed adhesions, exudative pleural effusion, and a fibrinous exudate consistent with acute empyema. A 10-mm grayish necrotic mass was found to be adherent to the right upper lobe and to obstruct a 5-mm fistula. To avoid spillage, this material was removed en bloc, and thorough debridement and irrigation were performed. Suspecting fungal infection, we performed a wide wedge resection including the fistula site. Although intraoperative pleural fluid cultures were negative, Aspergillus fumigatus was grown from the resected necrotic material, based on which we initiated 3 months' treatment with oral voriconazole. The patient recovered uneventfully without recurrence noted until the 2-year follow-ups. Histopathology revealed atelectasis-related sclerosis with nodular scarring and dry necrosis, while fungal growth was confirmed in the lung cavity wall, indicating localized pulmonary aspergillosis complicating a pneumothorax.

Conclusions: In pneumothorax patients with cystic changes in the lungs arising from old healed pulmonary tuberculosis, the potential presence of CPA should also be considered. Even with exposure of fungal colonies in the thoracic cavity during pneumothorax surgery, appropriate intraoperative and postoperative measures could allow even elderly steroid-treated patients to recover completely without recurrence of CPA.

Background: Primary retroperitoneal leiomyomas are exceptionally rare, accounting for approximately 1.2% of all retroperitoneal tumors. Those originating specifically from the levator ani muscle are even rarer. The contrast-enhanced ultrasound (CEUS) features of such tumors are poorly documented in the literature, often leading to diagnostic challenges and misdiagnosis. This case report aims to describe the comprehensive multimodal ultrasound findings of a large retroperitoneal leiomyoma arising from the levator ani and highlight the diagnostic value of integrating advanced ultrasound techniques.

Case description: A 31-year-old asymptomatic woman with a history of uterine fibroids was found to have a large pelvic mass during a routine health check-up. A comprehensive multimodal ultrasound examination was performed, utilizing transrectal double-plane probes, perineal pelvic floor probes, and high-frequency linear probes. Two-dimensional ultrasound revealed a well-defined, solid mass measuring 111 mm × 62 mm × 63 mm, originating from the left levator ani muscle with predominantly low echogenicity. Color Doppler flow imaging (CDFI) showed sparse internal vascularity. High-frame-rate transrectal CEUS demonstrated that the mass began enhancing at 18 seconds, peaked at 31 seconds with lower intensity compared to the adjacent levator ani muscle, and showed early washout, characterized as an "early enhancement, early washout, low enhancement" pattern. Strain elastography and shear wave elastography both indicated that the lesion tissue was softer than the ipsilateral levator ani muscle, with an elastic ratio of 1.16 and shear wave values of 15.73 kPa (mass) vs. 18.17 kPa (muscle). Based on these highly suggestive benign features, the patient underwent successful en bloc surgical resection via a perineal approach without preoperative biopsy. Histopathological examination confirmed the diagnosis of leiomyoma.

Conclusions: To our knowledge, this is the first reported case utilizing a combination of high-frame-rate CEUS and elastography for the preoperative evaluation of a retroperitoneal leiomyoma originating from the levator ani. This multimodal ultrasound approach, encompassing detailed anatomical mapping with specialized probes, dynamic contrast enhancement kinetics, and tissue stiffness assessment, proved crucial in accurately determining the tumor's origin and characterizing its nature. It can significantly improve diagnostic accuracy for complex pelvic masses, potentially reducing misdiagnosis and guiding optimal clinical management, including the decision to forgo biopsy in select cases with compelling benign imaging features planned for definitive surgery.

Background: The size of atrial septal defects (ASDs) is typically measured using echocardiography. In contrast, computed tomography (CT) images offer superior spatial resolution, and four-dimensional (4D) CT enables visualization of the defect hole during various cardiac phases. In this study, we compared the results of 4D CT measurements of defect sizes during atrial systole (As) and atrial diastole (Ad) in 7 of 18 cases of robot-assisted ASD closure (Robotic ASD closure) with measurements obtained by other methods.

Case description: Robotic ASD closure was performed in 18 patients between December 2019 and April 2025. Surgery was performed under general anesthesia, with a venous line from the right internal jugular vein and right femoral vein, and an arterial line on the right femoral artery to establish extracorporeal circulation. The procedure was conducted using the DaVinci Xi^® surgical system. Image analysis was performed using VINCENT^®, and the size of the defect was measured during As and Ad, and calculated using the formula: long radius × short radius × π. The mean age was 60.9 years (range, 34-82 years), and 10 (55.6%) patients were male. No residual shunt flow was detected on intraoperative or postoperative echocardiography. The defect size was measurable using 4D CT in 7 cases. The area of the defect was significantly different between As [65.9-207.2 mm², mean 134.1 mm², interquartile range (IQR) 58.9] and Ad (113.0-490.6 mm², mean 226.1 mm², IQR 94.2) (P=0.04).

Conclusions: Using 4D CT, it was possible to determine the morphology and area of ASD defects in As/Ad before surgery. This is useful for selecting surgical procedures and determining patch size and may contribute to reducing cardiac arrest and surgical time. To prove that 4D CT can accurately predict the size and morphology of the defects in As/Ad, further accumulation of cases and analyses are required.

Background: Small-cell prostate cancer is the most common type of treatment-related neuroendocrine prostate cancer. It typically originates from prostate adenocarcinoma following prolonged endocrine therapy, presenting as desmoplasia-resistant. At this stage, indicators such as prostate-specific antigen (PSA) and androgen receptor (AR) remain within normal ranges, conflicting with widespread metastatic lesions. Simultaneously, neuroendocrine indicators exhibit a significant elevation. Small-cell prostate cancer can secrete hormones, leading to ectopic endocrine syndrome.

Case description: In this context, we present a case wherein prostate adenocarcinoma transformed into small cell prostate cancer, accompanied by a rare paraneoplastic syndrome involving ectopic adrenocorticotropic-hormone (ACTH) production. The patient primarily presented with hypokalemia, edema, and hypertension. Following multi-line anti-tumor therapy and adrenal steroidogenesis inhibitors therapy, the patient's symptoms, including hypokalemia and edema, markedly improved. However, the patient ultimately succumbed to tumor progression. We present this case based on the CARE reporting checklist.

Conclusions: Our case referred to the treatment regimen for small-cell lung cancer, suggesting that the treatment regimen for small-cell lung cancer is effective for small-cell prostate cancer, but tumors can quickly become resistant. The efficacy of adrenal steroidogenesis inhibitors in treating ectopic ACTH syndrome (EAS) is demonstrated, underscoring that judicious treatment decisions can effectively prolong patient survival and enhance their quality of life. We present this case series based on a list of AME case series reports.

Background: Symphysis pubis diastasis (SPD) is a rare childbirth complication, involving a pubic symphysis separation of ≥10 mm, occurring in about 2.8% of women. Risk factors include fetal macrosomia, prolonged labor, and instrumental delivery. Symptoms include severe pelvic pain and mobility issues. Conservative treatments like physical therapy and pelvic bracing are first-line, with surgery reserved for refractory cases. Traditional Chinese medicine (TCM) manual therapy offers a non-invasive alternative with promising outcomes.

Case description: A 32-year-old primigravida experienced SPD after a prolonged labor, resulting in a 25 mm pubic symphysis separation. Initial treatment with analgesics and exercise therapy provided limited relief. She subsequently underwent TCM spinal correction therapy, which involved manual techniques to relax soft tissues, realign the pubic symphysis, and stimulate acupoints to improve qi and blood circulation. After two sessions, her pain significantly decreased, and a follow-up computed tomography (CT) scan showed a reduction in the symphysis gap to 10 mm. By 1 month post-treatment, she reported complete resolution of symptoms and resumed normal activities.

Conclusions: TCM spinal correction therapy could offer rapid and effective relief for postpartum SPD, providing a nonsurgical approach. This case highlights the potential of TCM in managing SPD, particularly for patients seeking conservative treatment options. Further research with larger sample sizes is recommended to validate these findings and explore the mechanisms underlying TCM's efficacy in SPD management.

Background: Thyrotoxic hypokalemic periodic paralysis (THPP) is a rare complication of hyperthyroidism, most often associated with Graves' disease. It is characterized by transient muscle weakness and hypokalemia due to an intracellular potassium shift. Although Graves' disease is also associated with an increased risk of thyroid nodularity and, in some cases, malignancy, the co-occurrence of THPP and papillary thyroid carcinoma (PTC) remains exceedingly rare. To our knowledge, this is only the second reported case documenting the simultaneous presence of all three conditions: THPP, Graves' disease, and PTC.

Case description: A 26-year-old male with a known history of Graves' disease presented with acute flaccid paralysis due to THPP. This episode led to thyroid imaging, which revealed thyroid nodules and cervical lymphadenopathy. Fine-needle aspiration (FNA) was suspicious for malignancy. The patient underwent total thyroidectomy with central and left neck dissections, with final pathology confirming multifocal PTC, including a widely invasive follicular variant, and regional lymph node metastases.

Conclusions: This case illustrates the rare triad of THPP, Graves' disease, and PTC. In this patient, an episode of THPP served as the clinical trigger that led to further thyroid evaluation and ultimately the diagnosis of an aggressive and multifocal thyroid malignancy. It emphasizes the need for thorough thyroid evaluation in patients with Graves' disease who present with THPP, as an underlying malignancy may be present. Early imaging, cytologic assessment, and surgical consultation should be considered.

Background: Multilocus pathogenic variants are increasingly recognized in neonates with complex phenotypes and have important implications for diagnosis and clinical management. Reporting such cases helps expand the phenotypic spectrum and improve clinical understanding of blended genetic disorders.

Case description: We describe a rare case of a male neonate born at 38+5 weeks of gestation with a birth weight of 1.73 kg, consistent with small-for-gestational-age status. The infant presented with perinatal asphyxia requiring 37 minutes of resuscitation, followed by hypotonia, poor feeding, a weak cry, and reduced responsiveness. Laboratory investigations revealed anemia, hyperbilirubinemia, and G6PD deficiency. He received supportive care in the neonatal intensive care unit (NICU) and was discharged in stable condition with planned follow-up. Trio-based whole-exome sequencing (trio-WES) identified compound heterozygous missense variants in MCM4 (c.1829G>A and c.2179G>C), compound heterozygous variants in RYR1 (c.11608+1G>A and c.13660-29G>A), and a hemizygous pathogenic variant in G6PD (c.482G>T, p.Gly161Val), each inherited from one parent. To our knowledge, this combination of variants has not been previously reported. At 18-month follow-up, the infant exhibited delayed motor and language development, unsteady gait, and had 14 hospital visits due to recurrent respiratory infections.

Conclusions: This case highlights the potential for blended phenotypes caused by multilocus variation and underscores the diagnostic value of trio-WES in neonates with unexplained multisystem involvement. Long-term follow-up and multidisciplinary management remain essential for such patients.

Background: Spontaneous intramural small bowel hematoma (SISBH) is a rare complication of long-term oral anticoagulation, particularly with vitamin K antagonists (VKAs). SISBH diagnosis is based on oral anticoagulation history associated with acute abdominal symptoms and suggestive radiological findings. Experience with this condition is still limited in the literature. This report highlights the clinical presentation and management of this rare condition, including the first known use of total-body positron emission tomography/computed tomography (PET/CT) scan in the diagnostic workup of SISBH.

Case description: A 53-year-old male on long-term oral anticoagulation with VKA for mechanical mitral valve replacement due to rheumatic heart disease presented to the emergency department for abdominal pain and nausea. Physical examination was remarkable for abdominal distension, increased bowel sounds and rebound tenderness, a presentation compatible with intestinal subocclusion. At admission, the international normalized ratio was 8.3, indicating VKA intoxication. Abdominal CT revealed significant jejunal parietal thickening, suggestive of SISBH. PET/CT findings were also consistent with a possible hematoma, although an inflammatory or neoplastic process could not be excluded. We opted for conservative treatment with temporary warfarin suspension and vitamin K infusion. The patient's intestinal subocclusion gradually improved, and he was discharged after seven days with full recovery.

Conclusions: SISBH, although rare, should be considered in the differential diagnosis of acute abdominal symptoms in patients receiving long-term oral anticoagulation, particularly with VKAs. Conservative treatment is the preferred initial option. A high index of suspicion, followed by prompt abdominal CT, is key to early diagnosis. PET/CT provided minimal diagnostic value beyond CT. Conservative management with anticoagulation reversal and supportive care is generally the primary therapeutic strategy adopted in the literature, demonstrating favorable short-term outcomes, as was the case with our patient.

Background: Internal carotid artery occlusion (ICAO) is uncommon in young adults and may present with mild or transient symptoms, posing challenges in determining the timing and necessity of endovascular intervention. While conservative management is often preferred in patients with minimal deficits, some may experience delayed infarct progression requiring escalation of care.

Case description: We present a 24-year-old male with no prior medical history who developed mild stroke symptoms during physical activity. Imaging revealed complete occlusion of the left internal carotid artery (ICA). Due to rapid symptom resolution and a low National Institutes of Health Stroke Scale (NIHSS) score, he was managed conservatively with antiplatelet therapy and discharged. Twelve days later, he returned with new-onset right-sided weakness and numbness. Imaging showed a new infarct and persistent ICAO with middle cerebral artery (MCA) involvement. Despite medical therapy, he deteriorated neurologically, and a large MCA infarct with midline shift was identified. Emergent mechanical thrombectomy of the first segment of the middle cerebral artery (M1) segment was performed. The patient showed meaningful improvement post-procedure and was discharged to rehabilitation. At follow-up, he remained ambulatory with assistance and functionally independent self-care but continued to have expressive aphasia and right-sided weakness.

Conclusions: This case demonstrates that young patients with ICAO and initially mild symptoms may still be at risk for infarct progression and delayed neurological deterioration. Mechanical thrombectomy can lead to favorable outcomes even when performed beyond traditional time windows. Our findings highlight the importance of close monitoring and re-evaluation in this population and support a flexible, individualized approach to intervention timing based on evolving clinical and radiographic evidence.