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Idiopathic unilateral complete oculomotor nerve palsy: a case report of diagnostic quandary. 特发性单侧完全性眼球运动神经麻痹:诊断难题的病例报告。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-30 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002514
Sagun Ghimire, Shikher Shrestha, Dinuj Shrestha, Kajan Ranabhat, Suman Bhattarai, Kritick Bhandari, Rikesh Kc

Introduction and importance: When compared to other cranial nerve palsies idiopathic unilateral oculomotor nerve palsy with pupillary sparing is one of the least noted neurological conditions. Moreover, there lies a series of diagnostic dilemmas to come into a final diagnosis resulting in several array of clinical investigations. Hence, there is a delay in prompt management.

Case summary: An elderly female without any known comorbidities presented with the complaint of headache, dizziness and dropping of left eyelid. Several arrays of diagnostic workups was done to come to a diagnosis, but even with rigorous laboratory investigations and radiological examinations, a common working diagnosis could not be made. Hence with a diagnosis of exclusion after proper neurological and neuro-ophthalmological examination, idiopathic unilateral common oculomotor nerve palsy was identified for which improvement with steroids was noted in the patient.

Discussion: Idiopathic unilateral complete oculomotor nerve palsy is considered as a diagnosis of exclusion when all the diagnostic parameters fail to signify and positive results. The vague symptomatic presentation of the disease condition further compels the treating physician to carry out several panels of laboratory to radiological investigations. But if identified in time the treatment modality is straightforward.

Conclusion: The diagnostic quandary in timely identification of such disease conditions needs a pertinent diagnostic guideline so as to avoid the unwanted panel of investigations.

简介和重要性:与其他颅神经麻痹相比,特发性单侧眼球运动神经麻痹伴瞳孔缺失是最不引人注意的神经系统疾病之一。此外,要做出最终诊断还存在一系列诊断难题,需要进行一系列临床检查。病例摘要:一位没有任何已知合并症的老年女性主诉头痛、头晕和左眼睑下垂。为了确诊,医生做了一系列的诊断工作,但即使进行了严格的实验室检查和放射检查,也无法得出共同的诊断结果。因此,在进行了适当的神经学和神经眼科检查后,确定了特发性单侧眼球运动神经麻痹为排除性诊断:讨论:特发性单侧完全性眼球运动神经麻痹被认为是一种排除性诊断,因为所有诊断指标都无法显示阳性结果。该病的症状表现模糊不清,这进一步迫使主治医生进行多项实验室和放射学检查。但如果能及时发现,治疗方法就会很简单:结论:及时发现这类疾病的诊断难题需要一个相关的诊断指南,以避免不必要的检查。
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引用次数: 0
Mucosal infection with Tsukamurella species following nasal septum procedure: a rare case report. 鼻中隔手术后粘膜感染冢霉菌:一例罕见病例报告。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-30 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002515
Alamjeet K Sidhu, Shreya Khandelwal, Francis V Dominic Savio, Simranjeet Bedi, Yashash D Pathak

Introduction and importance: Tsukamurella species are rare, aerobic, gram-positive bacteria known to cause infections, primarily in immunocompromised individuals. This case report presents a rare instance of a mucosal infection caused by Tsukamurella species following a nasal septum procedure in an immunocompetent patient.

Case presentation: A 51-year-old man with a history of multiple hereditary exostosis, allergic rhinitis, and recent nasal fracture repair presented with persistent fevers and low back pain. Postoperatively, he developed sinus pain and small oral lesions, initially treated with antibiotics for presumed sinusitis. Despite treatment, his fever persisted, leading to an emergency department visit. Laboratory tests indicated sepsis, but a CT scan of the sinuses showed no sinusitis. Despite broad-spectrum antibiotics, the patient's fever continued. On admission day 9, nasal endoscopy and culture identified Tsukamurella species. The patient was treated with augmentin, fluconazole, and levofloxacin, leading to the resolution of symptoms and discharge with ongoing treatment.

Clinical discussion: Tsukamurella species are uncommon pathogens that are often associated with bacteremia in immunocompromised individuals. This case highlights the diagnostic challenges and the importance of considering unusual pathogens in postprocedural infections, even in immunocompetent patients. Accurate identification and appropriate management are critical in improving outcomes for patients with Tsukamurella infections.

Conclusion: This case underscores the need for vigilance in diagnosing rare infections like Tsukamurella, even in immunocompetent individuals. The successful resolution with combination therapy highlights the importance of appropriate antibiotic selection in managing such infections.

简介和重要性:月金杆菌是一种罕见的需氧革兰氏阳性细菌,已知可引起感染,主要是在免疫力低下的人群中。本病例报告了一例免疫功能正常的患者在鼻中隔手术后由月金杆菌引起粘膜感染的罕见病例:病例介绍:一名 51 岁的男性患者有多发性遗传性外生殖器炎、过敏性鼻炎病史,近期进行过鼻骨骨折修复术,并伴有持续发热和腰痛。术后,他出现鼻窦疼痛和口腔小损伤,最初因推测为鼻窦炎而使用抗生素治疗。尽管进行了治疗,但他仍持续发烧,于是到急诊科就诊。实验室检查显示他患有败血症,但鼻窦 CT 扫描显示他没有鼻窦炎。尽管使用了广谱抗生素,但患者仍持续发烧。入院第 9 天,鼻内窥镜检查和培养发现了冢霉菌。患者接受了增强素、氟康唑和左氧氟沙星治疗,症状得到缓解,经持续治疗后出院:临床讨论:月金杆菌是一种不常见的病原体,通常与免疫力低下者的菌血症有关。本病例凸显了在手术后感染中考虑不常见病原体的诊断难度和重要性,即使是免疫功能正常的患者也不例外。准确的识别和适当的处理对于改善月金雷菌感染患者的预后至关重要:本病例强调,即使是免疫功能正常的患者,在诊断月金雷菌等罕见感染时也需要保持警惕。综合疗法成功解决了这一问题,突出了在治疗此类感染时选择适当抗生素的重要性。
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引用次数: 0
Predictive models and biomarkers for survival in stage III breast cancer: a review of clinical applications and future directions. III 期乳腺癌生存预测模型和生物标志物:临床应用和未来方向综述。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-30 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002517
Emmanuel Ifeanyi Obeagu, Getrude Uzoma Obeagu

Stage III breast cancer, characterized by locally advanced tumors and potential regional lymph node involvement, presents a formidable challenge to both patients and healthcare professionals. Accurate prediction of survival outcomes is crucial for guiding treatment decisions and optimizing patient care. This publication explores the potential clinical utility of predictive tools, encompassing genetic markers, imaging techniques, and clinical parameters, to improve survival outcome predictions in stage III breast cancer. Multimodal approaches, integrating these tools, hold the promise of delivering more precise and personalized predictions. Despite the inherent challenges, such as data standardization and genetic heterogeneity, the future offers opportunities for refinement, driven by precision medicine, artificial intelligence, and global collaboration. The goal is to empower healthcare providers to make informed treatment decisions, ultimately leading to improved survival outcomes and a brighter horizon for individuals facing this challenging disease.

III 期乳腺癌的特点是局部晚期肿瘤和潜在的区域淋巴结受累,这给患者和医护人员都带来了严峻的挑战。准确预测生存结果对于指导治疗决策和优化患者护理至关重要。本刊物探讨了预测工具的潜在临床效用,包括遗传标记、成像技术和临床参数,以改善 III 期乳腺癌的生存结果预测。整合这些工具的多模式方法有望提供更精确和个性化的预测。尽管存在数据标准化和基因异质性等固有挑战,但在精准医疗、人工智能和全球合作的推动下,未来仍有改进的机会。我们的目标是让医疗服务提供者能够做出明智的治疗决策,最终改善患者的生存状况,为面临这一挑战性疾病的患者带来更光明的前景。
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引用次数: 0
Curriculum and program evaluation in medical education: a short systematic literature review. 医学教育中的课程和项目评估:简短的系统文献综述。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-30 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002518
Himayat Ullah, Sarwat Huma, Ghulam Yasin, Muhammad Ashraf, Qazi Tahir-Ud-Din, Hossam Shabana, Junaid Sarfraz

Medical education is constantly evolving worldwide and facing various challenges. To cope with these, continuous and fruitful evaluation of an educational program is the need of the day. This study aims to know the purpose of evaluation, various theories related to program evaluation, and different models of curriculum and program evaluation. This will help educationists evaluate their programs fruitfully and effectively according to their needs and objectives. Different search engines including Medline's PubMed interface, Google Scholar, and Cochrane Review databases using keywords, curriculum evaluation, evaluation models, and evaluation strategies in education, were searched without any date restrictions, and 20 full-text articles were selected for review and data extraction. While reviewing the literature it was found that most of the modern educational program and curriculum evaluation models are based on the reductionist, system, and complexity theories of evaluation. The experimental/quasi-experimental model is based majorly on the linear approach and reductionism, but its drawback is that it is impractical for the whole curriculum and sometimes ethically unfavorable. Kirkpatrick's model, Philips' model, the CIPP model, and the logic model are based on the system and complexity theory and are more practical in medical education. Each of these models has its advantages and limitations. In this review, the authors discussed the important distinctive features of these evaluation theories and models and their applicability and usefulness in evaluating different programs and curricula.

医学教育在全球范围内不断发展,面临着各种挑战。为了应对这些挑战,必须对教育项目进行持续而富有成效的评估。本研究旨在了解评估的目的、与项目评估相关的各种理论以及课程和项目评估的不同模式。这将有助于教育工作者根据自己的需求和目标,对其课程进行富有成效的评估。我们使用不同的搜索引擎,包括 Medline's PubMed 界面、Google Scholar 和 Cochrane Review 数据库,使用关键字、课程评价、评价模式和教育评价策略进行搜索,没有任何日期限制,并选择了 20 篇全文文章进行审查和数据提取。在查阅文献时发现,现代教育项目和课程评价模式大多基于还原论、系统论和复杂性评价理论。实验/准实验模式主要以线性方法和还原论为基础,但其缺点是对整个课程不切实际,有时在伦理上也不利。柯克帕特里克模型、飞利浦模型、CIPP 模型和逻辑模型以系统论和复杂性理论为基础,在医学教育中更为实用。这些模式各有其优势和局限性。在这篇综述中,作者讨论了这些评价理论和模型的重要特色,以及它们在评价不同项目和课程时的适用性和实用性。
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引用次数: 0
Effects of oral montelukast on pulmonary function and clinical symptoms in acute asthma exacerbations: a randomized, double-blind, placebo-controlled trial. 口服孟鲁司特对哮喘急性加重期肺功能和临床症状的影响:随机、双盲、安慰剂对照试验。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-30 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002507
Masoud Aliyali, Siavash Abedi, Ali Sharifpour, Erfan Ghadirzadeh, Mahdie Fattahi, Jamshid Yazdani Charati, Hossein Mehravaran

Introduction: Montelukast is a leukotriene receptor antagonist that helps treat chronic asthma and allergic rhinitis by reducing inflammation and bronchoconstriction. However, oral montelukast's effectiveness in managing acute asthma attacks has yet to be completely identified.

Methods: This randomized, double-blind, placebo-controlled trial investigated the efficacy of oral montelukast in acute exacerbations of asthma. Seventy patients between 18 and 65 years of age with a primary diagnosis of asthma attack were included in the study and were randomly assigned to receive 10 mg of montelukast orally daily or placebo. Symptoms, signs, forced expiratory volume in 1 second (FEV1), and peak expiratory flow rate (PEFR) were evaluated.

Results: Our findings showed a statistically significant difference between montelukast and placebo regarding FEV1 (78.05 ± 7.84 vs. 72.05 ± 12.00, P = 0.016), PEFR (322.86 ± 28.95 vs. 290.86 ± 44.21, P = 0.003), and wheezing (P = 0.022) on the fifth day of treatment. Additionally, FEV1 and PEFR values were compared in two subgroups of patients, ICS users (ICSU) and non-ICS users (NICSU), in both the montelukast and placebo groups. In the montelukast group, while PEFR improved significantly for day 5 in both the ICSU (P = 0.007) and NICSU (P = 0.027) subgroups, FEV1 only improved in the ICSU (P = 0.009) subgroup compared to placebo.

Conclusion: The present study demonstrated that oral montelukast administered in acute asthma exacerbation could lead to better values of PEFR and FEV1 on pulmonary function and improvement of wheezing in terms of symptoms.

简介孟鲁司特是一种白三烯受体拮抗剂,可通过减轻炎症和支气管收缩来治疗慢性哮喘和过敏性鼻炎。然而,口服孟鲁司特对控制哮喘急性发作的疗效尚未完全确定:这项随机、双盲、安慰剂对照试验研究了口服孟鲁司特对哮喘急性发作的疗效。研究纳入了 70 名年龄在 18 岁至 65 岁之间、主要诊断为哮喘发作的患者,他们被随机分配到每天口服 10 毫克孟鲁司特或安慰剂。研究人员对患者的症状、体征、1秒内用力呼气容积(FEV1)和呼气峰流速(PEFR)进行了评估:结果:我们的研究结果表明,在治疗的第五天,孟鲁司特和安慰剂在 FEV1(78.05 ± 7.84 vs. 72.05 ± 12.00,P = 0.016)、PEFR(322.86 ± 28.95 vs. 290.86 ± 44.21,P = 0.003)和喘息(P = 0.022)方面的差异具有统计学意义。此外,还比较了孟鲁司特组和安慰剂组两个亚组患者的 FEV1 和 PEFR 值,即 ICS 使用者(ICSU)和非ICS 使用者(NICSU)。在孟鲁司特组中,虽然 ICSU(P = 0.007)和 NICSU(P = 0.027)亚组患者第 5 天的 PEFR 都有显著改善,但与安慰剂相比,只有 ICSU(P = 0.009)亚组患者的 FEV1 有所改善:本研究表明,在哮喘急性加重期口服孟鲁司特可提高肺功能的 PEFR 和 FEV1 值,并改善喘息症状。
{"title":"Effects of oral montelukast on pulmonary function and clinical symptoms in acute asthma exacerbations: a randomized, double-blind, placebo-controlled trial.","authors":"Masoud Aliyali, Siavash Abedi, Ali Sharifpour, Erfan Ghadirzadeh, Mahdie Fattahi, Jamshid Yazdani Charati, Hossein Mehravaran","doi":"10.1097/MS9.0000000000002507","DOIUrl":"10.1097/MS9.0000000000002507","url":null,"abstract":"<p><strong>Introduction: </strong>Montelukast is a leukotriene receptor antagonist that helps treat chronic asthma and allergic rhinitis by reducing inflammation and bronchoconstriction. However, oral montelukast's effectiveness in managing acute asthma attacks has yet to be completely identified.</p><p><strong>Methods: </strong>This randomized, double-blind, placebo-controlled trial investigated the efficacy of oral montelukast in acute exacerbations of asthma. Seventy patients between 18 and 65 years of age with a primary diagnosis of asthma attack were included in the study and were randomly assigned to receive 10 mg of montelukast orally daily or placebo. Symptoms, signs, forced expiratory volume in 1 second (FEV1), and peak expiratory flow rate (PEFR) were evaluated.</p><p><strong>Results: </strong>Our findings showed a statistically significant difference between montelukast and placebo regarding FEV<sub>1</sub> (78.05 ± 7.84 vs. 72.05 ± 12.00, <i>P</i> = 0.016), PEFR (322.86 ± 28.95 vs. 290.86 ± 44.21, <i>P</i> = 0.003), and wheezing (<i>P</i> = 0.022) on the fifth day of treatment. Additionally, FEV<sub>1</sub> and PEFR values were compared in two subgroups of patients, ICS users (ICSU) and non-ICS users (NICSU), in both the montelukast and placebo groups. In the montelukast group, while PEFR improved significantly for day 5 in both the ICSU (<i>P</i> = 0.007) and NICSU (<i>P</i> = 0.027) subgroups, FEV<sub>1</sub> only improved in the ICSU (<i>P</i> = 0.009) subgroup compared to placebo.</p><p><strong>Conclusion: </strong>The present study demonstrated that oral montelukast administered in acute asthma exacerbation could lead to better values of PEFR and FEV<sub>1</sub> on pulmonary function and improvement of wheezing in terms of symptoms.</p>","PeriodicalId":8025,"journal":{"name":"Annals of Medicine and Surgery","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2024-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11444550/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142364124","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Axillary lesion in a young adult ends up to a peculiar diagnosis: primitive neuro-ectodermal tumor (PNET) of ulnar nerve: a rare case report. 一名年轻成年人的腋窝病变最终被确诊为尺骨神经原始神经外胚层肿瘤(PNET):罕见病例报告。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-26 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002505
Mohammadreza Emamhadi, Nooshin Zaresharifi, Zoheir Reihanian, Anita Khalili, Mohammad Taghi Ashoobi, Sama Noroozi Guilandehi, Iraj Baghi, Alireza Mehrvarz

Introduction and importance: Primitive neuro-ectodermal tumor (PNET) is a highly aggressive tumor composed of small round blue cells, mostly developing in children and young adults. Being a member of Ewing's Sarcoma Family of Tumors (ESFT); it has been discussed in two subcategories of central and peripheral PNET. PNETs of peripheral nerves are very uncommon pathologic findings, as to the best of our knowledge only 12 well-documented cases have been yet reported.

Case presentation: A 30-year-old male presented with progressive paresthesia of his right hand's little finger and painless swelling of the right axilla. Magnetic resonance (MR) neurography demonstrated a heterogeneous, high-signal, round mass within the right axilla fossa in proximity to the medial aspect of brachial plexus branches. The clinical and radiological study failed to an accurate diagnosis, thus surgical resection of the tumor was done for tissue evaluation. Histopathologic study of the lesion revealed a neoplasm comprising sheets of small, round, blue cells (Hematoxylin and Eosin stain), which immunohistochemically consisted with the diagnosis of PNET.

Clinical discussion: The differential diagnosis of axillary fossa masses, focusses on peripheral nerve tumors like Schwannoma and PNET. MR neurography aids in evaluation, but tissue diagnosis remains crucial. Treatment involves surgical resection, chemotherapy, and radiotherapy tailored to individual patients.

Conclusion: Although pPNET is not apparently the first differential diagnosis coming to mind when encountering a rapidly growing mass in the axillary fossa with peripheral nerve origin, its highly malignant behavior, makes it crucial to be considered in the differential diagnoses.

导言和重要性:原始神经外胚层瘤(PNET)是一种由小圆形蓝细胞组成的侵袭性极强的肿瘤,多发于儿童和青壮年。作为尤文肉瘤家族肿瘤(ESFT)的一员,它被分为中枢性和周围性 PNET 两个亚类进行讨论。外周神经的 PNET 是非常罕见的病理结果,据我们所知,目前仅有 12 例记录详实的病例被报道过:一名 30 岁的男性因右手小指进行性麻痹和右侧腋窝无痛性肿胀而就诊。磁共振(MR)神经影像学检查显示,右侧腋窝内靠近臂丛神经分支内侧有一异质、高信号、圆形肿块。临床和放射学检查均未能得出准确诊断,因此对肿瘤进行了手术切除以进行组织评估。病变组织病理学检查显示,肿瘤由成片的小圆形蓝色细胞组成(血红素和伊红染色),免疫组化诊断为 PNET:腋窝肿块的鉴别诊断主要集中在周围神经肿瘤,如许旺瘤和 PNET。磁共振神经显像有助于评估,但组织诊断仍然至关重要。治疗包括手术切除、化疗和放疗,因人而异:虽然当腋窝出现迅速生长的周围神经源性肿块时,pPNET显然不是首先想到的鉴别诊断,但其高度恶性的行为使其成为鉴别诊断中必须考虑的因素。
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引用次数: 0
Urachal carcinoma initially presenting only as a liver metastasis, a diagnostic and therapeutic challenge: first case in literature. 最初仅表现为肝转移的尿道癌,诊断和治疗的难题:文献中的首个病例。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-26 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002499
George Bashour, George Hneino, Zain Aldin Zaher, Ali Dway, Georges Michael, Zuheir Alshehabi

Introduction: Urachal carcinomas are uncommon malignant neoplasms comprising only 0.01% of all adult cancers. Most patients were aged from 58 to 64 years at diagnosis with 60 years being the median. It usually metastasizes to the lungs (22%), bones (22%), and liver (16%).

Presentation: We report a case of a 71-year-old female patient who initially presented with two large liver masses and a small nodule on the anterior side of the bladder. The symptoms were nonspecific with abdominal discomfort. The final diagnosis of urachal adenocarcinoma was finalized with a biopsy of the bladder mass. The patient initially received six doses of FOLFOX6 without improvement and then Gem-Carbo, showing improvement after six doses. Finally, the patient received two doses of FOLFIRI-B with no response and kept deteriorating and died after 19 months of treatment.

Discussion: About 90% of patients are symptomatic and hematuria is the most typical presenting symptom at diagnosis. The low incidence and the histopathologic similarities to adenocarcinoma from various sources pose a difficulty in recognizing the tumor. Our study presents the only case of a urachal carcinoma first manifesting with abdominal mass resulting from liver metastasis with no prior symptoms of urological origins. Also, our study presents the first attempt of using FLOFIRI-B to treat metastatic UraC.

Conclusion: This case highlights the necessity for clinicopathological correlation to make the correct diagnosis and the challenges in the treatment which urges the need for further research to identify more effective treatment strategies for this rare cancer.

导言:尿道癌是一种不常见的恶性肿瘤,仅占所有成人癌症的 0.01%。大多数患者确诊时年龄在 58 至 64 岁之间,中位数为 60 岁。它通常会转移到肺部(22%)、骨骼(22%)和肝脏(16%):我们报告了一例 71 岁女性患者的病例,她最初表现为两个大的肝脏肿块和膀胱前侧的一个小结节。症状为非特异性腹部不适。通过对膀胱肿块进行活检,最终确诊为膀胱腺癌。患者最初接受了六次 FOLFOX6 治疗,但病情未见好转,随后接受了 Gem-Carbo,六次治疗后病情有所好转。最后,患者接受了两剂 FOLFIRI-B,但无反应,病情持续恶化,治疗 19 个月后死亡:讨论:约 90% 的患者无症状,血尿是确诊时最典型的症状。该肿瘤发病率低,组织病理学上与各种来源的腺癌相似,这给识别该肿瘤带来了困难。在我们的研究中,仅有一例尿道癌首先表现为肝转移引起的腹部肿块,且之前没有任何泌尿系统症状。此外,我们的研究还首次尝试使用 FLOFIRI-B 治疗转移性尿道癌:本病例强调了临床病理相关性对做出正确诊断的必要性,以及治疗过程中面临的挑战,这促使我们需要进一步研究,为这种罕见癌症找到更有效的治疗策略。
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引用次数: 0
Managing neurofibromatosis type I and vision impairment in a resource-limited setting: a case study and multidisciplinary approach. 在资源有限的环境中管理 I 型神经纤维瘤病和视力障碍:案例研究和多学科方法。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-26 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002482
Pratik Adhikari, Nabin Bhujel

Background: Neurofibromatosis type I (NF1) is a genetic disorder characterized by the development of multiple benign tumors along nerves in the skin, brain, and other parts of the body. It is associated with a range of clinical manifestations, including skin lesions, neurofibromas, and ocular abnormalities, which can significantly impact a patient's quality of life. Management of NF1 is particularly challenging in resource-limited settings due to limited access to diagnostic and therapeutic resources.

Clinical presentation: A 62-year-old woman with a known history of NF1 presented with progressive visual impairment. Her condition began in childhood with multiple hyperpigmented skin macules, which developed into numerous cutaneous tumors over time. Examination revealed numerous neurofibromas, café-au-lait spots, and axillary freckling. Significant visual impairment was caused by large fibromas on her eyelids. Histological analysis confirmed benign nerve tissue tumors.

Clinical discussion: The management strategy in this resource-limited setting focused on regular monitoring, patient education, symptomatic treatment, and multidisciplinary care. Despite the limitations, the patient's condition was managed effectively through these adapted strategies. The importance of genetic testing for confirmation and further management was noted but not performed due to resource constraints.

Conclusion: This case highlights the complexities of managing NF1 in resource-limited settings, emphasizing the need for adaptable management approaches. Multidisciplinary care and patient education were crucial in improving the patient's quality of life. This case underscores the importance of early diagnosis and intervention to prevent complications like visual impairment.

背景:I 型神经纤维瘤病(NF1)是一种遗传性疾病,其特征是沿着皮肤、大脑和身体其他部位的神经发生多发性良性肿瘤。它与一系列临床表现有关,包括皮肤损伤、神经纤维瘤和眼部异常,会严重影响患者的生活质量。由于诊断和治疗资源有限,在资源有限的环境中,NF1 的治疗尤其具有挑战性:临床表现:一名 62 岁的女性患者,已知患有 NF1,并伴有进行性视力障碍。她的病症始于童年时期的多发性色素沉着性皮肤斑丘疹,随着时间的推移发展成许多皮肤肿瘤。检查发现了许多神经纤维瘤、咖啡斑和腋窝雀斑。眼睑上的大纤维瘤导致视力严重受损。组织学分析证实为良性神经组织肿瘤:在这种资源有限的情况下,治疗策略侧重于定期监测、患者教育、对症治疗和多学科护理。尽管条件有限,但通过这些调整后的策略,患者的病情得到了有效控制。我们注意到了基因检测对确诊和进一步治疗的重要性,但由于资源有限,我们没有进行基因检测:本病例凸显了在资源有限的环境中管理 NF1 的复杂性,强调了采用适应性管理方法的必要性。多学科护理和患者教育对于改善患者的生活质量至关重要。本病例强调了早期诊断和干预对预防视力损伤等并发症的重要性。
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引用次数: 0
CAR-T-cell therapy in meningioma: current investigations, advancements and insight into future directions. 脑膜瘤的 CAR-T 细胞疗法:当前研究、进展和未来方向展望。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-22 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002491
Shree Rath, Murtaja Satea Shafeea, Abbas F Abdul Hussein, Ahmed Shamil Hashim, Sara Hassanaien, Santiago Pastrana-Brandes, Bipin Chaurasia

Meningiomas, the most common tumors of the central nervous system (CNS), present significant challenges in treatment, particularly for atypical and anaplastic subtypes where standard therapies often fall short of therapeutic expectations. Chimeric antigen receptor (CAR) T-cell therapy, a groundbreaking immunotherapy approach, has demonstrated great success in hematological malignancies but faces obstacles in solid tumors, including CNS tumors like glioblastomas. This article provides a comprehensive review of the efficacy of CAR-T therapy in meningiomas, highlighting the tumor's immunogenic potential and the challenges associated with applying this therapy in clinical practice. Through an extensive literature review, the study explores potential antigens for CAR-T targeting in meningiomas, shedding light on the tumor-immune microenvironment interactions. Challenges such as tumor heterogeneity, blood-brain barrier penetration, off-target effects, and tumor recurrence are discussed, alongside potential strategies to overcome these obstacles. The study also investigates recent advancements in CAR-T therapy, including the identification of novel target antigens and the development of engineering approaches to enhance therapeutic efficacy. Furthermore, the article highlights the importance of ongoing research efforts in exploring the tumor-immune dynamics in meningiomas and underscores the urgent need for clinical trials to validate the safety and efficacy of CAR-T therapy in this context. By addressing these challenges, CAR-T therapy holds the promise of revolutionizing meningioma treatment, offering new hope for patients suffering from this disease.

脑膜瘤是中枢神经系统(CNS)最常见的肿瘤,它的治疗面临巨大挑战,尤其是非典型性和无弹性亚型,标准疗法往往达不到预期治疗效果。嵌合抗原受体(CAR)T细胞疗法是一种开创性的免疫疗法,在血液恶性肿瘤中取得了巨大成功,但在实体瘤(包括胶质母细胞瘤等中枢神经系统肿瘤)中却面临障碍。本文全面回顾了CAR-T疗法在脑膜瘤中的疗效,强调了这种肿瘤的免疫原性潜力以及在临床实践中应用这种疗法所面临的挑战。通过广泛的文献综述,该研究探讨了脑膜瘤中CAR-T靶向的潜在抗原,揭示了肿瘤与免疫微环境的相互作用。研究讨论了肿瘤异质性、血脑屏障穿透、脱靶效应和肿瘤复发等挑战,以及克服这些障碍的潜在策略。研究还探讨了CAR-T疗法的最新进展,包括新型靶抗原的鉴定和提高疗效的工程方法的开发。此外,文章还强调了正在进行的探索脑膜瘤中肿瘤-免疫动态的研究工作的重要性,并强调了在这种情况下验证 CAR-T 疗法安全性和有效性的临床试验的迫切需要。通过应对这些挑战,CAR-T疗法有望彻底改变脑膜瘤的治疗,为该病患者带来新的希望。
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引用次数: 0
Clinical outcomes following hospitalization for COVID-19 in patients with cardiac sarcoidosis in the United States: a propensity-matched analysis from national inpatient sample database from April 2020 to December 2021. 美国心脏肉样瘤病患者因 COVID-19 住院后的临床结果:2020 年 4 月至 2021 年 12 月全国住院病人样本数据库的倾向匹配分析。
IF 1.7 Q2 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-08-22 eCollection Date: 2024-10-01 DOI: 10.1097/MS9.0000000000002474
Sebastian Mactaggart, Raheel Ahmed, Asma Riaz, Shehroze Tabassum, Kamleshun Ramphul, Maham Bilal, Yumna Jamil, Mansimran Singh Dulay, Alexander Liu, Mushood Ahmed, Hiroyuki Sawatari, Prince K Pekyi-Boateng, Alessia Azzu, Athol Wells, Vasilis Kouranos, Anwar Chahal, Rakesh Sharma

Background: The highly arrhythmogenic nature of cardiac sarcoidosis (CS) leads to high morbidity and mortality, the rates of which may be higher in COVID-19 patients. This study aimed to evaluate the outcomes of CS patients admitted to hospitals with COVID-19.

Methods: The study utilised the 2020-2021 National Inpatient Sample database, examining primary COVID-19 cases in adults aged older than or equal to 18 years. Those with CS were identified using ICD-10 code "D86.85" and compared with and without propensity matching (1:10) to those without CS for baseline characteristics and primary outcomes of acute kidney injury (AKI), use of mechanical ventilation, cardiac arrest and mortality.

Results: In total, 2 543 912 COVID-19 cases were identified. Before propensity matching, CS patients were more likely to be younger (58.0 vs. 64.0 years, P<0.01), male (64.0% vs. 52.6%, P=0.011), of Black ethnicity (60.0% vs. 15.9%, P<0.01), exhibit higher Charlson Comorbidity Index (CCI) scores (3.00 vs. 1.00, P<0.01) and had a higher incidence of in-hospital cardiac arrest (aOR 2.649, 95% CI 1.366-5.134, P=0.004). After propensity matching (CS, N=95; non-CS, N=875), those with CS were at a statistically significant reduced risk of AKI (aOR 0.484, P=0.01); however, the outcomes of death, cardiac arrest, mechanical ventilation, length of stay (LOS) and healthcare costs did not reach significance.

Conclusion: In a propensity-matched cohort admitted with COVID-19, CS patients had a reduced risk of AKI, but comparable LOS, rates of cardiac arrest, mechanical ventilator use, and mortality. Future research is warranted to develop evidence-based guidelines for managing COVID-19 in patients with CS.

背景:心脏肉样瘤病(CS)具有高度致心律失常性,导致高发病率和高死亡率,COVID-19 患者的发病率和死亡率可能更高。本研究旨在评估因 COVID-19 而入院的 CS 患者的预后:研究利用了 2020-2021 年全国住院病人抽样数据库,对年龄大于或等于 18 岁的成年人中的 COVID-19 初诊病例进行了调查。使用ICD-10代码 "D86.85 "确定了CS病例,并就基线特征以及急性肾损伤(AKI)、使用机械通气、心脏骤停和死亡率等主要结果与无CS病例进行了倾向匹配(1:10)比较:共发现 2 543 912 例 COVID-19 病例。在倾向匹配前,CS 患者更可能更年轻(58.0 岁 vs. 64.0 岁,PPP=0.011)、更可能是黑人(60.0% vs. 15.9%,PPP=0.004)。经过倾向匹配后(CS,95人;非CS,875人),CS患者发生AKI的风险在统计学上显著降低(aOR 0.484,P=0.01);然而,死亡、心脏骤停、机械通气、住院时间(LOS)和医疗费用等结果均未达到显著性:结论:在使用 COVID-19 的倾向匹配队列中,CS 患者发生 AKI 的风险较低,但住院时间、心脏骤停发生率、机械通气使用率和死亡率相当。未来需要开展研究,为 CS 患者管理 COVID-19 制定循证指南。
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引用次数: 0
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Annals of Medicine and Surgery
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