AME Case Reports最新文献

Background: Giant pulmonary bullae (GPB) are a rare manifestation of emphysema that often requires procedural intervention. Operative management of GPB has been shown to significantly improve quality of life and pulmonary function test (PFT) parameters. These improvements have been shown to persist for years, with a gradual decline associated with the progression of emphysema. For those with GPB who have had a pneumothorax, a future contralateral pneumothorax is more common in those with contralateral blebs. We present the unique case of a patient with GPB who required contralateral bullectomy 40 years after his first.

Case description: A 63-year-old man with a history of left-sided bullectomy in 1986 who was lost to follow-up presented in 2021 with progressive dyspnea on exertion. Computed tomography (CT) demonstrated a giant bulla occupying >50% of the right thorax, causing ipsilateral atelectasis and contralateral tracheal shift. Also present was a right-sided pneumothorax and a left-sided hilar mass. Biopsy of this mass was negative for malignancy, and it self-resolved on subsequent imaging. He re-established as an outpatient, and four months later was directly admitted from clinic for hypoxic respiratory failure. Follow-up CT imaging demonstrated minimal interval change over 1.5 years. He continued outpatient follow-up, and by 2023, had reduced his smoking and participated in pulmonary rehab. PFT demonstrated a moderately severe obstructive ventilatory defect with evidence of heterogeneity of ventilation but remained stable from 1/2023 to 8/2024. His dyspnea on exertion and oxygen requirement (4 L/min) were stable in this interval. He presented in 8/2024 for elective video-assisted thoracic surgery (VATS) with right upper lobe bullectomy and right middle lobe wedge resection. At follow-up 4 months later, his O₂ requirement had reduced to 2.5 L/min with activity. He endorsed significant subjective improvement in his breathing. Follow-up PFT demonstrated improvement in 6-minute walk test (6MWT), forced expiratory volume in 1 second (FEV1), forced vital capacity (FVC), total lung capacity (TLC), and diffusing capacity of the lungs for carbon monoxide (DLCO).

Conclusions: Bullectomy remains an effective treatment for dyspnea in patients with GPB. Prophylactic management of contralateral blebs remains an ongoing debate, though primarily in the context of pneumothorax. Advancements in medical and surgical management of chronic obstructive pulmonary disease (COPD) have expanded the length of time patients should be closely monitored for consideration of procedural intervention for GPB.

Background: Hemophilia is an X-linked recessive inherited bleeding disorder caused by genetic defects in specific proteins involved in blood coagulation. It is classified into two main types: hemophilia A and hemophilia B. The primary clinical characteristic of hemophilia is prolonged spontaneous and/or traumatic bleeding, which most commonly occurs within the musculoskeletal system. However, spontaneous splenic rupture in adults with hemophilia A has been reported infrequently. This rare but life-threatening condition necessitates prompt diagnosis and intervention. This study aims to evaluate the safety and efficacy of splenic artery embolization (SAE) in patients with hemophilia A who present with spontaneous splenic rupture and maintain hemodynamic stability.

Case description: This case report describes a 20-year-old male with hemophilia A [factor VIII (FVIII) activity at 5%] who presented with intermittent epigastric distension and discomfort with a 3-day history. Imaging studies confirmed the presence of splenic rupture with hemoperitoneum. Emergency SAE was performed, successfully achieving hemostasis. Both preoperatively and postoperatively, the patient received FVIII replacement therapy along with other blood products. As a result, the patient's FVIII level increased to 31%, hemoglobin stabilized at 100 g/L, and the patient was discharged after 1 week.

Conclusions: This case underscores the efficacy of SAE as a minimally invasive and effective treatment for spontaneous splenic rupture in patients with hemophilia A, particularly in hemodynamically stable individuals.

Background: Giant mediastinal ectopic thyroid tissue is a rare entity that presents a diagnostic and therapeutic challenge for clinicians investigating mediastinal masses. Most cases are asymptomatic and incidentally detected on imaging. When symptomatic, presentations are typically influenced by the mass's size and anatomical location. In this report, we present a right hemithoracic benign follicular goiter in an asymptomatic patient that was removed through a right posterolateral thoracotomy.

Case description: A 39-year-old male is discovered to have an incidental mediastinal mass on a chest radiograph following a road traffic accident. Chest computed tomography (CT) scan revealed a heterogeneously enhancing hyper-vascular mediastinal mass in the right paratracheal location with areas of cystic degeneration and calcification. His thyroid gland was normally located in the neck and was not anatomically a part of the mediastinal mass. A CT-guided biopsy of the mass was positive for thyroglobulin and thyroid transcription factor 1 (TTF-1). The patient's thyroid function tests, including thyroid-stimulating hormone (TSH) and free thyroxine (T4), were within the normal range. During surgery, performed via a right posterolateral thoracotomy, the mass was found to be adherent to lung tissue, posterior mediastinal pleura, right main bronchus, and superior vena cava. A complete surgical excision of the mass was achieved, and the patient underwent an uneventful recovery. The mass was an encapsulated multinodular 15 cm × 12 cm × 9 cm round dark red soft tissue with a firm consistency. Histopathology exhibited thyroid tissue with nodular hyperplasia and cystic changes.

Conclusions: Giant mediastinal ectopic thyroid tissue necessitates surgical excision for both diagnostic and therapeutic reasons. The preoperative assessment of giant mediastinal ectopic thyroid tissue location and size aids in directing the surgical approach for mass excision.

Background: Ondansetron, a 5-hydroxytryptamine 3 (5-HT3) receptor antagonist, is used as an antiemetic medication for the management of nausea and vomiting. Since ondansetron may cause QT-interval prolongation, the Food and Drug Administration (FDA) issued a warning about its use, particularly in individuals who are at risk for cardiac arrhythmias. We report a case of cardiac arrest and cardiomyopathy following a single intravenous (IV) ondansetron dose with successful complete recovery after taking proper clinical interventions. Our findings would therefore raise awareness to take preventive measures upon administering ondansetron, whether in patients who are medically free or those who are at risk for developing cardiac arrhythmias.

Case description: We report a 43-year-old female patient with a negative medical and psycho-social history who had cardiac arrest due to ondansetron administration, along with hypomagnesemia, hypocalcemia, and lower-limit serum potassium level, which resulted in ventricular fibrillation, torsade de pointes, and cardiomyopathy. We also raised the suspicion of anaphylaxis to ondansetron, where certain investigations were done accordingly. The patient required cardiopulmonary resuscitation (CPR) and intensive care unit (ICU) admission for further management of electrolyte imbalance, left ventricular (LV) dysfunction, and suspected anaphylaxis. Certain interventions were done, which resulted in significant improvement and complete patient recovery.

Conclusions: Our case emphasizes the need to be cautious when administering ondansetron, and electrolyte imbalance is considered a risk factor for cardiac arrhythmias following ondansetron administration. It is then imperative that clinicians consider ondansetron-associated cardiac arrest and cardiomyopathy, take preventive measures, and remain vigilant for any unfavorable incidents that may arise.

Background: Foreign body ingestion (FBI) is common among children, with coins being the most frequently ingested object. Nevertheless, radiographic differentiation can become difficult when two juxtaposed coins resemble the contour of a button battery (BB).

Case description: This paper describes an 8-year-old boy who presented with drooling, vomiting, and absolute dysphagia. Chest radiographs revealed a radiopaque object in the upper esophagus with a double-rim or "halo" sign, raising concern for a BB. Due to the patient's symptoms and radiologic uncertainty, we performed urgent esophagoscopy under general anesthesia. Intraoperative findings revealed two stacked coins lodged in the upper esophagus. The surrounding esophageal mucosa displayed circumferential necrosis and eschar formation, although no perforation was present. Postoperative computed tomography angiography (CTA) was performed as a precaution and confirmed no vascular injury. We managed the patient conservatively where he then was discharged after 2 weeks in stable condition. Upon follow-up imaging, there were no complications, and we confirmed mucosal healing.

Conclusions: This case highlights the diagnostic challenge posed by stacked coins, which may radiographically mimic BB. Although inert, stacked coins can still cause significant esophageal injury, particularly when impacted for an unknown duration. In symptomatic cases, urgent intervention remains essential regardless of object type. Lateral imaging and careful interpretation can help avoid unnecessary alarm or delayed management in similar scenarios.

Background: Adenomyoepithelioma (AME) of the breast is a rare tumor composed of epithelial and myoepithelial cells, typically exhibiting benign behavior. However, malignant transformation (MAME), local recurrence, and distant metastases have been reported. Polymorphous adenocarcinoma (PmA), an exceptionally rare breast carcinoma, can mimic MAME morphologically, complicating diagnosis and management. Due to their rarity, standardized management guidelines remain undefined highlighting the need for further research and comprehensive evaluation.

Case description: This study presents nine cases of AME diagnosed at a single institution diagnosed with AME, including one initially misdiagnosed with PmA but later reclassified as MAME. The patients aged 31-61 years with a varied clinical presentation including palpable masses (n=3), lesions detected on mammography (n=4), microcalcifications (n=1), and nipple discharge (n=1). Imaging findings revealed predominantly solid masses with lobulated contours on ultrasound (US) and variable enhancement patterns on magnetic resonance imaging (MRI). Immunohistochemical (IHC) analysis, including p63, CK5/6, and calponin, was essential for confirming diagnoses. The highlight case involved a 54-year-old woman initially diagnosed with PmA based on histopathological evaluation. However, upon retrospective review, the lesion was reclassified as MAME due to predominant myoepithelial differentiation. The patient received surgical excision and radiation therapy and remained disease-free after 78 months of follow-up. AME typically presents as a unilateral, centrally located mass, with recurrence rates of 11.1% reported. MAME, characterized by high mitotic rates and cellular atypia, requires aggressive management, while PmA, a low-grade carcinoma, often exhibits perineural invasion and triple-negative status.

Conclusions: This case series underscores the diagnostic challenges and importance of accurate differentiation between AME, MAME, and PmA, as misdiagnosis can significantly impact treatment and prognosis and highlighting the importance of IHC and molecular profiling. Given the potential for recurrence and metastasis, complete excision with negative margins remains the mainstay of treatment. This series contributes to the limited literature on these rare tumors, emphasizing the critical role of meticulous pathological evaluation and multidisciplinary approaches to improve patient outcomes.

Background: Overlap syndrome (OS) refers to a condition where a patient meets the classification criteria for at least two connective tissue diseases (CTDs), either simultaneously or at different points in times. The spectrum of OS includes mixed CTDs such as rheumatoid arthritis (RA) and systemic sclerosis (SSc). This syndrome may occur alongside other medical conditions, such as Hashimoto's thyroiditis and interstitial lung disease (ILD), complicating its diagnosis and management.

Case description: This report describes the case of a 48-year-old Malay woman who visited a primary care clinic, presenting with a year-long history of intermittent joint pain, swelling, morning stiffness, and skin thickening involving her hands and arms. On physical examination, swelling was observed in the proximal interphalangeal joints, with sclerodactyly extending to the upper arms. Initially, in primary care, she was diagnosed with RA and suspected SSc before being referred to a rheumatologist, where the diagnosis of OS was ultimately confirmed. Treatment with disease-modifying anti-rheumatic drugs and analgesics was initiated, leading to symptom improvement. However, she returned to primary care with symptoms of fatigue, dry skin, constipation, and dry cough which already persisted for 6 months. Thyroid function tests and anti-thyroid peroxidase antibodies confirmed the diagnosis of Hashimoto's thyroiditis. Spirometry showed a restrictive lung pattern, and high-resolution computed tomography indicated early ILD changes. She was managed by multidisciplinary teams, including primary care, rheumatology, endocrinology, respiratory medicine and radiology, and her condition improved.

Conclusions: Managing OS in primary care is challenging due to multiple related conditions that complicate diagnosis and management. Primary care physicians play a vital role in early detection, timely referral, and coordinating multidisciplinary care for these complex conditions.

Background: Stress fractures are fatigue-induced injuries that typically affect weight-bearing bones of the lower limb. While often associated with mechanical overuse, atypical sites of fracture-such as the patella-should raise suspicion for underlying systemic pathology. Primary hyperparathyroidism (PHPT), characterised by autonomous parathyroid hormone overproduction, can lead to cortical bone resorption and fragility, predisposing patients to such fractures.

Case description: A 45-year-old physically active woman presented to her general practitioner with non-specific right knee pain and no history of trauma. Clinical examination was unremarkable, and initial management was conservative. Four weeks later, she developed acute pain and functional impairment. Urgent radiography revealed a displaced transverse patellar stress fracture, which was surgically managed with open reduction and internal fixation using tension band wiring. Routine follow-up in primary care prompted biochemical investigations that revealed elevated serum calcium (2.9 mmol/L) and parathyroid hormone (122 pmol/L), confirming a diagnosis of PHPT. Imaging identified a solitary parathyroid adenoma, and she underwent successful parathyroidectomy. Eight months later, she experienced hardware-related skin complications requiring surgical removal of the tension band. She made a full recovery, with resolution of hypercalcaemia and no recurrence of symptoms.

Conclusions: This case highlights the importance of considering endocrine causes in patients presenting with stress fractures at uncommon sites. The diagnostic journey from a seemingly benign musculoskeletal complaint to the discovery of PHPT underscores the pivotal role of multidisciplinary coordination across primary care, orthopaedics, and endocrinology.

Background: Patients with clonal cytopenia of undetermined significance (CCUS) who undergo hip replacement surgery are at risk of serious complications such as bleeding, infection, and anemia. There are certain difficulties in clinical treatment. Such cases have not been reported in international medical journals.

Case description: A 35-year-old man with CCUS for 4 years developed bilateral avascular necrosis of the femoral head 1 year after hormone therapy for the blood disorder. The patient required arthroplasty surgery due to hip pain and functional limitations. The patient's persistent severe deficiency of platelets, red blood cells, and granulocytes limited the operation. Before surgery, the multidisciplinary team formulated the blood transfusion treatment plan for correcting blood system abnormalities, the treatment plan for basic internal medicine diseases, the minimally invasive manipulation anesthesia plan, the surgical plan, the postoperative refined care and the rapid recovery plan. We completed bilateral hip arthroplasty surgery for patients and had good recovery of hip joint function during the 6-month follow-up after surgery (Harris Hip Score =90 points).

Conclusions: CCUS patients have a higher operation risk when perform the total hip arthroplasty. Under the collaborative treatment of the multidisciplinary team, we successfully completed the patient's surgery and achieved a good surgical outcome.

Background: Intra-articular corticosteroid (IACS) injections are generally considered safe for diabetic patients due to minimal systemic absorption. However, rare complications such as diabetic ketoacidosis (DKA) may still occur. We present a rare case of DKA following IACS, highlighting the need for heightened awareness among clinicians. While IACSs are frequently used for localized joint inflammation and pain relief, systemic complications such as DKA are exceedingly rare. To date, only a handful of case reports have documented this association, making this case a valuable addition to the limited literature.

Case description: We present a case of 76-year-old gentleman who presented to emergency department with DKA on a background of type 1 diabetes mellitus. A thorough history and extensive investigations identified that three days prior to this presentation, the patient had received an IACS injection in his right hip for pain management. The patient was treated for DKA according to the hospital protocol and was reviewed by the Rheumatology and Endocrinology teams who concluded that the probable cause in this case was steroid administration.

Conclusions: The patient made a full recovery with no long-term sequelae. This case underscores the importance of recognizing that even localized steroid therapy may have systemic metabolic consequences in diabetic patients. Clinicians should consider proactive monitoring of blood glucose and ketones following IACS injection, particularly in patients with type 1 diabetes.