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Is it possible to diagnose mixed epithelial and stromal tumor of kidney prior to surgery? A case report. 是否有可能在手术前诊断肾上皮和间质混合瘤?一份病例报告。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241308688
Hiroki Nishiyama, Masaki Tominaga, Kozue Ito, Go Hasegawa, Kyohei Ishida, Noboru Hara, Tsutomu Nishiyama

We present a case in which renal mixed epithelial and stromal tumor (MEST) was considered in the differential diagnosis based on preoperative imaging findings. A 38-year-old woman was found to have a right renal tumor during an abdominal ultrasound examination conducted as part of a health checkup. Contrast-enhanced computed tomography revealed a 50 × 40-mm mass in the middle of the right kidney. The possibility of papillary renal cell carcinoma was considered. However, given the patient's age, sex, and characteristics of the mass, MEST was also considered a differential diagnosis. The patient underwent laparoscopic radical right nephrectomy. The tumor was diagnosed as MEST.

我们提出一个病例,其中肾脏混合上皮和间质瘤(MEST)被认为是鉴别诊断基于术前影像学结果。一名38岁的妇女在进行健康检查的腹部超声检查时发现右肾肿瘤。增强计算机断层扫描显示右肾中部有一个50 × 40毫米的肿块。考虑乳头状肾细胞癌的可能性。然而,考虑到患者的年龄、性别和肿块的特征,MEST也被认为是一种鉴别诊断。患者行腹腔镜根治性右肾切除术。肿瘤诊断为MEST。
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引用次数: 0
Pediatric eosinophilic granulomatosis with polyangiitis and intracardiac thrombus: A case report. 小儿嗜酸性肉芽肿合并多血管炎和心内血栓1例。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241309966
Katharine V Jensen, Nicholas Brochez, Christopher Spence, Joel Livingston, Michael Khoury, Jeanine McColl

Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic necrotizing vasculitis marked by eosinophilia and extravascular granulomas, predominantly affecting the respiratory tract. This report details a unique EGPA case in a 6-year-old girl with extensive cardiac involvement, featuring an atypical intracardiac mass suggestive of endomyocardial fibrosis and a concomitant thrombus. The clinical course unfolded in three phases: an initial prodrome with asthma; subsequent peripheral hypereosinophilia; and ultimately systemic vasculitis. Cardiac involvement, notably an intracardiac mass in the right ventricular apex extending into the interventricular septum, underscored the diverse nature of EGPA. The patient fulfilled sufficient criteria outlined by the American College of Rheumatology and the European Alliance of Associations for Rheumatology for an EGPA diagnosis, displaying hypereosinophilia, obstructive airway disease, and biopsy-confirmed inflammation predominantly characterized by extravascular eosinophils. Treatment included high-dose methylprednisolone and cyclophosphamide, which resulted in clinical improvement and inflammatory marker normalization. To halt right ventricular thrombus progression, therapeutic unfractionated heparin was initiated, and she was transitioned to warfarin, which resulted in complete resolution of the cardiac mass. This case highlights the necessity of a multidisciplinary approach for managing complex EGPA manifestations, particularly in pediatrics, and emphasizes the importance of timely intervention in mitigating the impact of cardiac complications associated with EGPA.

嗜酸性肉芽肿病合并多血管炎(EGPA)是一种罕见的系统性坏死性血管炎,以嗜酸性粒细胞增多和血管外肉芽肿为特征,主要影响呼吸道。本报告详细介绍了一例独特的EGPA病例,该病例发生在一名6岁女孩,心脏广泛受损伤,表现为非典型心内肿块,提示心内膜纤维化并伴有血栓。临床过程分为三个阶段:最初的前驱症状伴哮喘;随后的外周嗜酸性粒细胞增多;最后是系统性血管炎。心脏受累,特别是右心室心内肿块延伸至室间隔,强调了EGPA的多样性。该患者符合美国风湿病学会和欧洲风湿病协会联盟对EGPA诊断的充分标准,表现为嗜酸性粒细胞增多、气道阻塞性疾病和活检证实的炎症,主要以血管外嗜酸性粒细胞为特征。治疗包括大剂量甲基强的松龙和环磷酰胺,导致临床改善和炎症标志物正常化。为了阻止右心室血栓的进展,开始使用无分离肝素治疗,并改用华法林治疗,这导致心脏肿块完全消失。本病例强调了多学科方法治疗复杂EGPA表现的必要性,特别是在儿科,并强调了及时干预减轻EGPA相关心脏并发症影响的重要性。
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引用次数: 0
Stevens-Johnson syndrome with overlapping features of DRESS syndrome: A report of two cases. 具有DRESS综合征重叠特征的Stevens-Johnson综合征:附2例报告。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241307097
Laura D Chin, Michael L MacGillivary, Kerri S Purdy, Carly Kirshen

Stevens-Johnson syndrome and drug reaction with eosinophilia and systemic symptoms are severe cutaneous adverse reactions to drugs that are generally considered distinct entities. In addition to identifying the offending medication, distinguishing between these diagnoses is important, as they have differing treatment regimens and prognoses. Distinction between severe cutaneous adverse reactions, particularly in the early stages of disease, can be difficult, and overlapping conditions have been reported in the literature. We present two cases of severe cutaneous adverse reaction, one following initiation of carbamazepine and the other lamotrigine, with extensive mucosal involvement and epidermal detachment, initially diagnosed as Stevens-Johnson syndrome. Despite the use of cyclosporine and repeated doses of etanercept, both cases evolved to have significant edema of the face and extremities, palmar and plantar involvement, and rapid response to systemic corticosteroids, which is more in-keeping with drug reaction with eosinophilia and systemic symptoms. We aim to help clinicians gain awareness of Stevens-Johnson syndrome/drug reaction with eosinophilia and systemic symptoms overlap which may aid diagnosis and guide treatment.

史蒂文斯-约翰逊综合征和嗜酸性粒细胞增多的药物反应和全身症状是对药物的严重皮肤不良反应,通常被认为是不同的实体。除了识别有害药物外,区分这些诊断也很重要,因为它们有不同的治疗方案和预后。区分严重的皮肤不良反应,特别是在疾病的早期阶段,可能是困难的,文献中已经报道了重叠的情况。我们报告了两例严重的皮肤不良反应,一例是卡马西平,另一例是拉莫三嗪,伴有广泛的粘膜受累和表皮脱离,最初诊断为史蒂文斯-约翰逊综合征。尽管使用了环孢素和重复剂量的依那西普,但这两个病例都出现了面部和四肢明显水肿,手掌和足底受累,对全身皮质类固醇反应迅速,这更符合嗜酸性粒细胞增多和全身症状的药物反应。我们的目的是帮助临床医生了解史蒂文斯-约翰逊综合征/药物反应与嗜酸性粒细胞增多和全身症状重叠,这可能有助于诊断和指导治疗。
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引用次数: 0
Cutaneous involvement of light chain deposition disease: A case report. 轻链沉积病累及皮肤1例报告。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241307116
Saba Vafaei-Nodeh, Hamid Masoudi

Light chain deposition disease is a rare condition associated with plasma cell dyscrasia and other lymphoproliferative disorders in which there is overproduction and deposition of non-amyloid light chains in various organs, leading to organ dysfunction. It is well-established that the majority of patients with light chain deposition disease exhibit renal involvement. Although awareness of extrarenal manifestations is increasing, cutaneous involvement has rarely been reported. Herein, we present a case of light chain deposition disease with cutaneous manifestations in the absence of any renal disease. A biopsy of the skin revealed amorphous eosinophilic material within the superficial dermis. Using special stains, immunohistochemistry, and direct immunofluorescence, the deposits were confirmed to be kappa light chains.

轻链沉积病是一种罕见的与浆细胞病变和其他淋巴细胞增生性疾病相关的疾病,在这种疾病中,非淀粉样轻链在各器官中过量产生和沉积,导致器官功能障碍。目前已经证实,大多数轻链沉积病患者表现为肾脏受累。虽然对外源性表现的认识正在增加,但很少有皮肤受累的报道。在此,我们提出一个病例轻链沉积病与皮肤表现,没有任何肾脏疾病。皮肤活检显示真皮表层有无定形的嗜酸性物质。通过特殊染色、免疫组织化学和直接免疫荧光,证实沉积物为kappa轻链。
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引用次数: 0
Anaplastic lymphoma kinase1 positive inflammatory myofibroblastic tumor of the urinary bladder: A rare mesenchymal neoplasm with diagnostic and therapeutic implications. 膀胱间变性淋巴瘤激酶1阳性炎性肌纤维母细胞瘤:一种具有诊断和治疗意义的罕见间充质肿瘤。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241308992
Fnu Poombal, Ibrahim Mansoor, Randa M Abdellatif, Nada Shaker

Inflammatory myofibroblastic tumors (IMTs) are rare mesenchymal neoplasms characterized by spindle-cell morphology with accompanying inflammatory infiltrates. Originally described in 1939, these tumors can arise in various anatomic locations, with the urinary bladder being a rare site of occurrence but the most common within the genitourinary tract. IMTs typically present as polypoid masses or firm submucosal nodules, often with painless hematuria in bladder cases. Histopathologically, IMTs are composed of myofibroblasts with myxoid stroma and mixed inflammatory cells, predominantly lymphocytes and plasma cells. Immunohistochemically, these tumors commonly express anaplastic lymphoma kinase1 (ALK1), vimentin, smooth muscle actin (SMA), and cytokeratin, with ALK1 serving as a crucial marker for diagnosis. This report details the case of a 31-year-old female presenting with hematuria, found to have a soft tissue mass in the urinary bladder (5.0 × 3.0 cm). Imaging revealed a well-defined lesion with vascularity. Histopathological examination confirmed an IMT, with immunohistochemistry showing diffuse ALK1 positivity, patchy SMA staining, and variable desmin expression, consistent with the diagnosis. IMTs are generally considered neoplasms of intermediate malignant potential. While metastasis is exceedingly rare in bladder IMTs, local recurrence has been reported, particularly in cases of incomplete surgical resection. Recent advances highlight the role of ALK inhibitors in managing unresectable cases, enabling partial cystectomy in select patients. This article underscores the importance of achieving complete surgical excision and highlights the role of ALK expression in diagnosis and differentiation from other spindle-cell neoplasms. Further studies are needed to elucidate the molecular and clinical factors influencing prognosis and to refine treatment strategies for IMTs.

炎症性肌纤维母细胞瘤(IMTs)是一种罕见的间充质肿瘤,其特征为梭形细胞形态并伴有炎症浸润。这些肿瘤最初于1939年被描述,可以出现在不同的解剖部位,膀胱是一个罕见的发生部位,但最常见的是在泌尿生殖道内。imt通常表现为息肉样肿块或坚硬的粘膜下结节,膀胱病例常伴有无痛性血尿。组织病理学上,IMTs由肌成纤维细胞与黏液样基质和混合炎症细胞组成,主要是淋巴细胞和浆细胞。免疫组织化学上,这些肿瘤通常表达间变性淋巴瘤激酶1 (ALK1)、波形蛋白、平滑肌肌动蛋白(SMA)和细胞角蛋白,其中ALK1是诊断的关键标志物。本文报告一例31岁女性患者,以血尿为主要表现,发现膀胱内有软组织肿块(5.0 × 3.0 cm)。影像学显示病灶范围清晰,有血管。组织病理学检查证实为IMT,免疫组织化学显示弥漫性ALK1阳性,斑片状SMA染色,异蛋白表达,与诊断一致。imt通常被认为是具有中度恶性潜能的肿瘤。虽然膀胱imt的转移非常罕见,但有局部复发的报道,特别是在手术切除不完全的情况下。最近的进展强调了ALK抑制剂在治疗不可切除病例中的作用,使部分膀胱切除术成为可能。本文强调了实现完全手术切除的重要性,并强调了ALK表达在诊断和与其他梭状细胞肿瘤鉴别中的作用。需要进一步的研究来阐明影响预后的分子和临床因素,并完善IMTs的治疗策略。
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引用次数: 0
Corrigendum to "May-Hegglin anomaly associated nephropathy: Case series". “May-Hegglin异常相关肾病:病例系列”的更正。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-17 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241309485

[This corrects the article DOI: 10.1177/2050313X241302013.].

[这更正了文章DOI: 10.1177/2050313X241302013.]
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引用次数: 0
Acquired reactive perforating collagenosis secondary to Cabozantinib: A case report and literature review. 卡博赞替尼继发的获得性反应性穿孔性胶原病1例报告并文献复习。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-16 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241304961
Zahra Rehan, Andrea Haner, Amina Taleb, Nzechukwu Ikeri, Jori Hardin

Acquired perforating dermatoses (APD) encompass a group of skin conditions distinguished by transepidermal elimination of dermal components. Acquired reactive perforating collagenosis (ARPC), a subtype of APD, has been reported most commonly in association with diabetes mellitus, chronic renal failure, and medications. In this report, we identify a novel case of ARPC secondary to Cabozantinib treatment.

获得性穿孔性皮肤病(APD)包括一组皮肤状况,其特征是皮肤成分经表皮消除。获得性反应性穿孔性胶原病(ARPC)是APD的一种亚型,据报道最常与糖尿病、慢性肾功能衰竭和药物有关。在本报告中,我们发现了一例卡博赞替尼治疗后继发ARPC的新病例。
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引用次数: 0
Successful myomectomy during pregnancy for the indication of acute lower abdominal pain with red degenerative myoma or ovarian torsion? "Case report". 妊娠期子宫肌瘤切除成功对急性下腹痛伴红色退行性肌瘤或卵巢扭转的指征?“情况报告”。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-15 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241301605
Leta Hinkosa Dinsa, Amenu Diriba Eticha, Mathewos Tamene Terefe

Uterine fibroids are benign tumors, arising from uterine smooth muscle cells. They are one of the most common benign tumors of the female genital tract among childbearing women, occurring in 20%-50% of women of reproductive age. The association of uterine myomas with pregnancy is high. This occurrence is constantly increased, linked to the onset of pregnancies, leading to the incidence of myomas gradually rising with age, hence the emergence of ultrasound reveals during routine ultrasound examinations of pregnancies, that were previously asymptomatic. Myomectomy during the second trimester was done and pregnancy continued safely to the third trimester. After successfully repairing it, the pregnant woman followed her antenatal care at Wollega University Referral Hospital, Western Oromia, Ethiopia. We presented the 20-year-old primigravida lady with a complaint of lower abdominal pain and a slight fever. We found a non-pedunculated, attached to the uterus via a broad base) myoma measuring 4 × 4 cm on 2D pelvic ultrasound. We admitted the client and gave her conservative management after we put her on antibiotics. Despite antibiotics, the pain persists we decided to manage it through an operation after we counseled the client about the potential complications. Then we removed the myoma without manipulating the uterus and it was a successful myomectomy without complicating the fetomaternal conditions. Currently, fetomaternal conditions are safe, and the woman is following her antenatal care at Wollega University's Referral Hospital. In the majority of cases of women with fibroids, pregnancy comes with complications, according to literature data, The coexistence of the myoma with pregnancy: increased frequency of spontaneous abortions and premature births, increased risk of bearing ablation, higher incidence of maternal-fetal incommunicability, incorrect position of the fetus, more often performed cesarean surgery, higher probability of postpartum hemorrhage. Prenatal myomectomy can be safely performed in the first and second trimesters of pregnancy; however, it is recommended to deliver the baby via cesarean section, due to fear of intrauterine rupture of the uterus.

子宫肌瘤是良性肿瘤,起源于子宫平滑肌细胞。它们是育龄妇女中最常见的女性生殖道良性肿瘤之一,发生在20%-50%的育龄妇女中。子宫肌瘤与妊娠的相关性很高。这种情况不断增加,与怀孕的开始有关,导致肌瘤的发病率随着年龄的增长而逐渐上升,因此在常规妊娠超声检查中出现了以前无症状的子宫肌瘤。在妊娠中期进行子宫肌瘤切除术,妊娠安全持续到妊娠晚期。在成功修复后,这名孕妇在埃塞俄比亚西奥罗米亚的沃勒加大学转诊医院接受了产前护理。我们提出一位20岁的初产妇,主诉下腹疼痛和轻微发烧。我们在二维盆腔超声上发现一个无带梗的子宫肌瘤,通过一个4 × 4厘米的宽基肌瘤附着在子宫上。我们让病人入院并在给她使用抗生素后对她进行了保守治疗。尽管使用了抗生素,疼痛仍然存在,我们决定通过手术来控制它,在我们咨询了客户关于潜在的并发症。然后我们在没有操纵子宫的情况下切除了子宫肌瘤,这是一次成功的子宫肌瘤切除术,没有使胎儿和母体的状况复杂化。目前,母婴状况安全,该妇女正在沃勒加大学转诊医院接受产前护理。在大多数患有肌瘤的女性中,妊娠伴随着并发症,根据文献资料,肌瘤与妊娠并存:自然流产和早产的频率增加,子宫消融的风险增加,母胎不可通达的发生率更高,胎儿体位不正确,更常进行剖宫产手术,产后出血的概率更高。产前子宫肌瘤切除术可以安全地在怀孕的前三个月和中期进行;但是,由于担心宫内子宫破裂,建议通过剖宫产分娩。
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引用次数: 0
Negative patch test findings after initiation of upadacitinib: A case report. 开始upadacitinib后的阴性斑贴试验结果:1例报告。
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-15 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241304949
Meghan L McPhie, Melanie D Pratt, Mark G Kirchhof

Upadacitinib is a systemic Janus kinase inhibitor currently approved for the treatment of atopic dermatitis. Many patients with atopic dermatitis have a concomitant diagnosis of allergic contact dermatitis. The utility of Janus kinase inhibitors for the treatment of allergic contact dermatitis and the effect on patch testing results remains unclear. We present a case of a woman in her 30s with atopic dermatitis and allergic contact dermatitis who was patch test negative after initiating therapy with upadacitinib.

乌达帕替尼是一种全身性 Janus 激酶抑制剂,目前已被批准用于治疗特应性皮炎。许多特应性皮炎患者同时被诊断为过敏性接触性皮炎。Janus 激酶抑制剂在治疗过敏性接触性皮炎方面的作用及其对斑贴试验结果的影响仍不清楚。我们介绍了一例 30 多岁患有特应性皮炎和过敏性接触性皮炎的女性患者,她在开始使用达帕替尼治疗后,斑贴试验结果呈阴性。
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引用次数: 0
Sealing socket with custom healing abutment in case of immediate posterior implant placement: A case report. 在即刻后植入种植体的情况下,用定制的愈合基台密封种植窝:病例报告
IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL Pub Date : 2024-12-13 eCollection Date: 2024-01-01 DOI: 10.1177/2050313X241302654
Ghada Neji, Mohamed Tlili, Raki Selmi, Mootaz Mlouka, Mohamed Salah Khalfi, Faten Ben Amor

Replacing a hopeless molar with a dental implant is one of the most common treatment modalities in implant dentistry. However, immediate implant post-extraction in the posterior region requires managing the bone gap and ensuring primary closure of the socket. To address this anatomical consideration, several authors have proposed using a custom abutment to seal the socket after immediate implant placement, maintain soft tissue contour, and stabilize the surrounding tissues. For example, the "Sealing Socket Abutment" (SSA) enhances the anatomical emergence profile, which facilitates the biological, prosthetic, and aesthetic integration of the future supra-implant prosthesis. A 32-year-old female patient in good general health consulted the Outpatient and Implantology Department of the dental clinic in Monastir, Tunisia, for the extraction of the root tip of the left first mandibular molar (tooth number 36) and subsequent implant rehabilitation. The patient's medical history was unremarkable, with no known allergies, or chronic conditions. Follow-up appointments were scheduled to monitor the healing process and ensure successful integration of the implant. The aim of this paper is to present, through this clinical case report, the SSA abutment fabrication protocol using the direct technique and to discuss the advantages and limitations of this technique.

用种植牙代替没有希望的磨牙是种植牙科中最常见的治疗方式之一。然而,在后牙区立即拔除种植体需要控制骨间隙并确保窝的初级关闭。为了解决这一解剖学上的问题,一些作者建议在种植体即刻植入后使用定制的基台来密封牙槽,保持软组织轮廓,并稳定周围组织。例如,“密封槽基台”(SSA)增强了解剖涌现轮廓,促进了未来种植体上部假体的生物、假体和美学整合。一名健康状况良好的32岁女性患者向突尼斯Monastir牙科诊所的门诊和种植科咨询,要求拔除左侧第一下颌磨牙(牙号36)的根尖并随后进行种植康复。患者的病史一般,没有已知的过敏或慢性疾病。随访预约,以监测愈合过程,确保成功整合种植体。本文的目的是通过这个临床病例报告,介绍使用直接技术的SSA基台制作方案,并讨论该技术的优点和局限性。
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引用次数: 0
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SAGE Open Medical Case Reports
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