AME Case Reports最新文献

Background: This study highlights an unusual and previously unreported adverse event (AE) following the minimally invasive treatment of pancreatic walled-off necrosis (WON). The standard treatment for WON currently involves primary drainage via an ultrasound-guided endoscopic, typically transgastric, approach. This method is associated with lower mortality and morbidity rates compared to traditional surgery. However, emerging AEs from these procedures may necessitate the involvement of a multidisciplinary team. Our case highlights the potential for gastrovesical fistula development as a rare AE following endoscopic drainage. Treatment for our patient prioritized individualized and non-surgical strategy, although surgical revision was also considered.

Case description: A 42-year-old male presented with a large symptomatic pancreatic WON refractory to conservative management, necessitating transgastric drainage. Despite the gradual evacuation of the WON contents, treatment was complicated by stent-related issues, including inadvertent bladder penetration. Rather than surgical correction, a collaborative approach among urology, gastroenterology, and surgery teams was employed, focusing on conservative treatment strategies. This approach successfully resolved the fistula, leading to the patient's full recovery.

Conclusions: Given the increasing use of endoscopic transluminal drainage in (peri)pancreatic collections, it is crucial to be aware of all potential AEs. To our knowledge, this is the first documented case of gastrovesical fistula following drainage of WON. Early recognition and a multidisciplinary approach are vital to manage this event.

Background: Intraductal papillary mucinous neoplasm of the bile duct (IPMN-B) is a neoplastic disease of the bile duct with papillary hyperplasia and mucus secretion, which originates from the duct epithelium and rarely complicates with fistula formation.

Case description: The patient was admitted to the hospital due to abdominal pain and yellow skin. Laboratory results showed alanine aminotransferase 299 U/L, total bilirubin 350 µmol/L, computerized tomography showed severe dilatation of common bile duct and extrahepatic bile duct. Magnetic resonance cholangiopancreatography showed the intra- and extrahepatic bile ducts were markedly dilated, and the signal of the bile ducts was uneven. Endoscope identified a large amount of mucus above the papilla that flowing out from the fistula. Further cholangiography through the fistula showed significant dilatation of the extrahepatic bile duct. SpyGlass examination showed a large amount of gelatinous mucus in the bile duct lumen and "fish-scaly or coral" changes in the mucosa of the right anterior branch bile duct, hepatic hilum as well as lower common bile duct. IPMN-B with choledochoduodenal fistula was diagnosed. The patient was then discharged with nasal biliary drainage and biliary stenting, oral antipyretic and hepatoprotective drugs. The patient's biliary obstruction and symptoms of infection improved with medical treatment but recured. Unfortunately, the patient died 10 months after his first visit.

Conclusions: SpyGlass has advantages in identifying the nature and extent of lesions, providing important references for diagnosis and treatment. Endoscopic intervention relieves biliary obstruction to some extent in patients with high operative risk or reluctance to undergo surgery.

Background: Primary adenocarcinomas of the vulva, particularly those from anogenital mammary-like glands, are exceedingly rare. These typically encompass a spectrum of cancers, including ductal, lobular, mixed ductal-lobular, tubular, and mucinous types, predominantly with invasive growth patterns.

Case description: A 73-year-old woman reported an 8-year history of a slowly enlarging vulvar mass, the mass skin surface color of the tumor was normal and there was no ulceration, redness, heat and pain, no skin itching and abnormal secretions. Examination revealed a 3 cm cyst-like nodule on the left vulva. Surgical excision was performed. Pathology showed a 2.5 cm nodular mass with a soft, mucinous texture. Microscopic analysis revealed a uniform cell distribution with papillary and solid patterns. Immunohistochemical staining showed estrogen receptor (ER), progesterone receptor (PR), epithelial membrane antigen (EMA), pan-cytokeratin [CK(pan)], GATA-binding protein 3 (GATA3) expression, among others, with a Ki67 index around 10%, suggesting a specific tumor profile-encapsulated papillary carcinoma, originating from the vulva anogenital mammary-like glands.

Conclusions: This article presents a unique case of anogenital mammary-like gland adenocarcinoma-encapsulated papillary carcinoma. It closely mirrors the morphology, immunohistochemistry, and biological behavior of its breast counterpart. Due to its slow progression and localized, encapsulated nature, the treatment approach for this carcinoma differs from other vulvar adenocarcinomas, aligning more with carcinoma in situ management similar to breast encapsulated papillary carcinoma.

Background: With regard to the treatment of massive pulmonary embolism (MPE) with circulatory and respiratory collapse and thrombolytic contraindications, current guidelines and researches usually give the priority to veno-arterial extracorporeal membrane oxygenation (V-A ECMO). However, the objective of this clinical case report is to highlight the effective use of veno-venous extracorporeal membrane oxygenation (V-V ECMO) in a 35-year-old pregnant woman with MPE complicated by hemorrhage, persistent hypoxia and multiple cardiac arrests.

Case description: A 35-year-old pregnant woman with gestational mellitus suddenly presented with complaints of nausea, vomiting and dyspnea after going to the toilet, combined with increasing heart rate (HR) of 150 bpm, decreasing pulse oxygen saturation (SpO₂) of 94%, larger right heart and the growing D-dimer at 11.2 µg/mL, who was considered as the pulmonary embolism. Unpredictable cardiac arrest occurred repeatedly before and after the cesarean section. Although cardiopulmonary resuscitation (CPR) was started timely and successfully, the maintenance of blood pressure still depended on high-dose pressor drugs, even terribly, the oxygenation was unstable under the assistance of mechanical ventilation with pure oxygen. Thus, V-V ECMO supporting was commenced following by gradual recovering in haemodynamics and respiratory function. And the diagnosis of MPE was ascertained again through computed tomographic pulmonary angiography (CTPA) and pulmonary angiography. Directing at the pathogeny, thrombolysis infusion catheters and anticoagulant therapy were initiated after bilateral uterine artery embolism for postpartum haemorrhage, later the patient discharged from hospital after recovery and had a good prognosis.

Conclusions: V-V ECMO could be effective for some patients with MPE who suffer from successful CPR after cardiac arrest while still combined with severe hypotension and refractory hypoxemia.

Background: Wilson's disease (WD) is a rare genetic disorder characterized by copper accumulation in the body, leading to a spectrum of health issues, such as liver disease, neurological disturbances, and psychiatric disorders. In recent years, there has been increasing recognition that WD can also result in osteoarticular defects. Research has shed light on the potential of WD to cause these findings, which in some instances, can progress to osteoarthritis and persistent pain. However, the exact pathophysiological process through which WD leads to osteochondral defects remains unclear.

Case description: We present a case of a 30-year-old male diagnosed with WD exhibiting musculoskeletal symptoms. The patient's medical history revealed chronic intermittent knee pain. Radiographic and magnetic resonance imaging (MRI) studies revealed a substantial osteochondral lesion with high-grade chondral fissuring. This report reviews the proposed pathophysiology of orthopedic pathology in WD, offers an updated literature review, and provides clinical recommendations for management. Treatment options including nonsurgical options and surgery are discussed.

Conclusions: This case underscores the significance of identifying the orthopedic manifestations of WD, even in the absence of classic signs and symptoms. Any WD patient suspected of having osteoarticular defects should be thoroughly evaluated, with a low threshold for initiating imaging studies. Moreover, treatment plans should be tailored to the patient's specific presentation, emphasizing the importance of individualized patient care. This case highlights key findings in WD and provides important insights, particularly on the clinical relevance of osteoarticular defects in WD, the potential application of nonsurgical and surgical treatments, and the importance of individualized patient care in the management of WD.

Background: Granulomatosis with polyangiitis (GPA) is a systemic autoimmune disease characterized by necrotizing granulomatous vasculitis of the small- and medium-sized vessels. Classical GPA affects the upper and lower airways and kidneys. It commonly affects the nose and paranasal sinuses, middle and inner ear, and subglottic region of the larynx. Therefore, otolaryngological involvement is common in GPA and can sometimes be the initial presentation. In rare cases, otologic signs and symptoms can be the initial manifestations of this disease, including recurrent otitis media, otitis media with effusion, and sensorineural hearing loss.

Case description: In the present study, we describe an atypical case of GPA of a 22-year-old female. The patient presented with severe and complicated otitis media with hearing loss and polyneuropathy (facial nerve paralysis and trigeminal nerve impairment), in addition to nasopharyngeal and parotid infiltration, in the absence of other otolaryngologic and systemic manifestations of GPA.

Conclusions: The diagnosis of GPA can be challenging due to its wide range of clinical manifestations. Otologic signs and symptoms are rare in this disease and can sometimes be the initial manifestations. Early diagnosis and treatment are important to prevent serious and permanent complications of the disease. Otolaryngologists should have high index of suspicion to systemic diseases such as GPA.

Background: Reasonable nutritional intervention is very important to promote wound healing and rehabilitation in patients with radical esophagectomy for esophageal cancer. This report aims to summarize the experience of nutritional and continuous nursing intervention in a patient who underwent radical resection of esophageal cancer after liver transplantation, by testing a comprehensive approach to optimize nursing plans in similar clinical practice. We hope that the implementation of home enteral nutrition can improve the nutrition status and quality of life of postoperative patients.

Case description: A patient with liver transplantation was admitted to The Fourth Hospital of Hebei Medical University for postoperative care. The nursing intervention were subsequently summarized and analyzed. In July 2023, the patient successfully underwent radical resection for esophageal cancer. Following the operation, the patient received regular medication and on-site nutritional intervention with the consent of her family. At discharge, the prealbumin, albumin, total protein and hemoglobin values of the patient were low, and body weight was 91 kg. The patient's nutritional risk screening (NRS2022) score was 5 points, and the Patient-Generated Subjective Global Assessment (PG-SGA) score was 4 points. After discharge, the patient continued to receive family enteral nutrition treatment, dietary guidance and psychological nursing. A follow-up review conducted 4 weeks after discharge showed improvements in the patient's NRS2022, albumin, total protein, hemoglobin, and body weight.

Conclusions: Strengthening postoperative nutritional intervention are vital for promoting rehabilitation in patients who undergo radical resection of esophageal cancer after liver transplantation.

Background: Intravenous leiomyomatosis (IVL) is a special type of uterine leiomyoma and is rare. Intravenous lipoleiomyomatosis (LPL) is a rare subtype of IVL, distinguished by the presence of adipose tissue. Although histologically benign, this disease exhibits aggressive biological behavior such as local invasion and high recurrence rate. The disease initially presents with no obvious clinical features, and cardiac symptoms may only appear in the later stages. Diagnosis primarily relies on imaging studies, and due to its rarity and atypical clinical presentation, imaging diagnosis can be challenging, leading to misdiagnosis and missed diagnosis. Previously, there was no report on the imaging findings of this disease.

Case description: This article reports a case of a 52-year-old patient who presented with lower abdominal discomfort due to IVL, and who underwent surgical resection and had a good recovery.

Conclusions: This is the first time we report the imaging features of a disease of intravenous LPL with an extension of the inferior vena cava (IVC), and its characteristic imaging features [ultrasound shows a mass with high echogenicity, computed tomography (CT) shows low-density signal similar to fat, magnetic resonance imaging (MRI) shows high signal on T1-weighted (T1W) image and low signal on T1W with fat-suppression (T1FS)] can lead to an accurate preoperative diagnosis and guide clinical treatment.

Background: Papillary thyroid cancer (PTC) is the most common type of well-differentiated endocrine malignancy. Generally, thyroid nodules with multiple oncogenic mutations are uncommon with an occurrence which may be related to more aggressive biological behavior of tumors. RET/PTC rearrangement, RAS, and BRAF mutations are considered to be mutually exclusive in PTC. Concomitant RET/PTC, RAS, or BRAF mutations have been documented, although the impact of these mutations for tumor growth and survival is debated.

Case description: Here we present a rare case of woman 46 years old with a neck mass and thyroid nodule classified as TIR5 on cytological examination. We found contemporary BRAF p.(Val600Glu) [p.(V600E); c.1799T>A] and NRAS p.(Gln61Arg) [p.(Q61R); c.182A>G] mutations in morphologically different areas within the same lobe (the right one); The two lesions show different morphology. The mutated BRAF lesion showed morphological characteristics compatible with classic papillary carcinoma. The mutant NRAS lesion shows morphological features compatible with follicular variant papillary carcinoma. To the best of our knowledges, this is the first time that such mutations, which are normally mutually exclusive, have been detected at the same time.

Conclusions: The finding of synchronous mutations is a rare occurrence suggesting for intratumoral heterogeneity (ITH) even in PTC. Patients with multiple mutations have a clinical worse prognosis, generally characterized by an aggressive thyroid cancer, which may influence the surgical treatment, chemotherapy, and BRAF V600E mutation-targeting therapy.

Background: Subungual exostosis is a type of heterotopic ossification, which often has unclear margins. Therefore, marginal resection may cause recurrence and wide resection is sometimes required to achieve a complete cure. However, wide resection may cause postoperative nail deformity and revision of this deformity is generally difficult. The primary treatment of subungual exostosis is surgical treatment, and there have been no comprehensive reports on the efficacy of adjunctive treatments. Although postoperative electron beam irradiation has been successfully used after heterotopic ossification excision to prevent recurrence, there are no reports on the use of this procedure following subungual exostosis resection.

Case description: Herein, we report a case of refractory subungual exostosis that developed as a result of chronic irritation and inflammation caused by an ingrown nail and recurred after initial resection. We performed marginal resection of the lesion to preserve the nail matrix and nail bed as possible, a two-stage skin grafting procedure, and electron-beam irradiation to prevent recurrence.

Conclusions: Excellent results were achieved both in terms of complete cure and cosmetic appearance, suggesting that electron-beam irradiation following refractory subungual exostosis excision may help prevent its recurrence. We expect a further study including many cases of subungual exostosis treated with postoperative electron-beam irradiation to be conducted.