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Spontaneous regression of an orbital Langerhans cell histiocytosis after biopsy: A case report. Spontaneous regression of an orbital Langerhans cell histiocytosis. 眼眶朗格汉斯细胞组织细胞增多症活检后自发性消退1例报告。眼眶朗格汉斯细胞组织细胞增多症的自发消退。
IF 0.9 Q4 Medicine Pub Date : 2022-10-18 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221135987
Safia Yahiaoui, Asma Ghorbel, Khadija Ben Zid, Semia Zarraa, Lina Kchaou, FidaNoubigh, Wael Kaabia, Fadoua Bouguerra, Alia Mousli, Rim Abidi, Amani Yousfi, Boujelbene Nadia, Chiraz Nasr

Langerhans histiocytosis or Langerhans cell histiocytosis (LCH) is a rare benign pathology representing less than 1% of orbital tumors. It can cause either localized or generalized lesions, leading to the destruction of hard and soft tissues. Eosinophilic granuloma is the most benign form and the predominant clinical presentation of LCH. We report a case of eosinophilic granuloma with orbital involvement in an 18-year-old male patient. Orbital radiotherapy was initially planned, but finally it was not performed due to a spontaneous regression of the lesion after the incisional biopsy. The presented case supports an expectant attitude given the possibility of a spontaneous regression after the biopsy, especially in small lesions. However, long-term follow-up is essential given the risk of recurrence.

朗格汉斯组织细胞增生症(LCH)是一种罕见的良性病理,占眼眶肿瘤的不到1%。它可以引起局部或全身性病变,导致硬组织和软组织的破坏。嗜酸性肉芽肿是LCH最良性的形式和主要的临床表现。我们报告一个18岁男性患者眼眶受累的嗜酸性肉芽肿病例。最初计划眼眶放射治疗,但最终因切口活检后病变自发消退而未进行。鉴于活检后自发消退的可能性,特别是在小病变中,所提出的病例支持期待的态度。然而,考虑到复发的风险,长期随访是必要的。
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引用次数: 0
A case of initially metastasizing pleomorphic adenoma of parotid gland. 腮腺多形性腺瘤初转移1例。
IF 0.9 Q4 Medicine Pub Date : 2022-10-03 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221130155
Sungchul Ko, Kye Hoon Park, Ji-Hye Lee, Ki Nam Park

Metastasis of pleomorphic adenoma (PA) is rare and usually presented as a locoregional recurrence developed many years after excision of the primary tumor although the PA is the most common neoplasm in the parotid gland. We described a case of a 48-year-old male with a parotid tumor with multiple enlarged ipsilateral lymph nodes which suggested a malignancy. The tumors had been neither evaluated nor excised and preoperative evaluation revealed benign PA in both lesions. After the complete surgical excision, the final pathology was notable for benign PA with metastasis to regional lymph nodes. At 1 year follow up he was clinically and radiographically free of disease. This implies that pleomorphic adenoma can occur as initially metastasis to regional lymph node even though benign neoplasm.

多形性腺瘤(PA)是腮腺中最常见的肿瘤,但它的转移是罕见的,通常表现为原发肿瘤切除多年后的局部复发。我们描述了一个病例48岁的男性腮腺肿瘤与多个扩大的同侧淋巴结提示恶性肿瘤。肿瘤既没有评估也没有切除,术前评估显示两个病变均为良性PA。完全手术切除后,良性PA转移到区域淋巴结的最终病理是显著的。在1年的随访中,他的临床和放射学无疾病。这表明多形性腺瘤即使是良性肿瘤,也可以在最初转移到区域淋巴结时发生。
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引用次数: 2
Very low-dose radiation therapy for management of inflammatory myofibroblastic tumor of the lung. 极低剂量放疗治疗肺炎性肌成纤维细胞瘤。
IF 0.9 Q4 Medicine Pub Date : 2022-07-15 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221106270
Nimeh Najjar, Hamel Patel, Scott Steinberg, Brett Baskovich, Stephanie Rothweiler, Bradford Hoppe

Inflammatory myofibroblastic tumor (IMT) is an uncommon chest pathology. Treatment primarily focuses on surgical resection for diagnostic and therapeutic purposes. However, there are instances in which alternative therapies with steroids, chemotherapy, or radiation are necessary. We discuss a case of recurrent IMT for which very low dose radiation proved an effective treatment.

炎症性肌纤维母细胞瘤(IMT)是一种罕见的胸部病理。治疗主要集中在手术切除诊断和治疗目的。然而,在某些情况下,类固醇、化疗或放疗等替代疗法是必要的。我们讨论了一例复发性IMT,非常低剂量的辐射证明是有效的治疗。
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引用次数: 1
Supratentorial cortical ependymoma: A systematic literature review and case illustration. 幕上皮质室管膜瘤:系统的文献回顾和病例说明。
IF 0.9 Q4 Medicine Pub Date : 2022-07-08 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221112432
Joshua A Cuoco, Andrew C Strohman, Brittany M Stopa, Michael S Stump, John J Entwistle, Mark R Witcher, Adeolu L Olasunkanmi

Cortical ependymomas are currently not considered a subgroup of supratentorial ependymomas; however, there is a growing body of literature investigating the natural history of these lesions compared to supratentorial ependymomas. We performed a systematic literature review of cortical ependymomas with a focus on the natural history, clinical characteristics, and clinical outcomes of these lesions as compared to supratentorial ependymomas. Our search revealed 153 unique cases of cortical ependymomas. The mean age on presentation was 21.2 years. Males and females comprised 58.8% (90/153) and 41.2% (63/153) of cases, respectively. The most common presenting symptom was seizure activity occurring in 44.4% of the cohort (68/153). The recently recognized C11orf95-RELA fusion was identified in 13.7% of the cohort (21/153) and 95.5% of cases (21/22) reporting molecular characterization. World Health Organization grades 2 and 3 were reported in 52.3% (79/151) and 47.7% (72/151) of cases, respectively. The frontal lobe was involved in the majority of cases (54.9%, 84/153). Gross total resection was achieved in 80.4% of cases (123/153). Tumor recurrence was identified in 27.7% of cases (39/141). Mean clinical follow-up was 41.3 months. Mean overall survival of patients who expired was 27.4 months whereas mean progression-free survival was 15.0 months. Comparatively, cortical ependymomas with C11orf95-RELA fusions and supratentorial ependymomas with C11orf95 RELA fusions exhibited differing clinical outcomes. Further studies with larger sample sizes are necessary to investigate the significance of RELA fusions on survival in cortical ependymomas and to determine whether cortical ependymomas with C11orf95-RELA fusions should be classified as a distinct entity.

皮质室管膜瘤目前不被认为是幕上室管膜瘤的一个亚组;然而,与幕上室管膜瘤相比,越来越多的文献研究了这些病变的自然历史。我们对皮质室管膜瘤进行了系统的文献回顾,重点是与幕上室管膜瘤相比,皮质室管膜瘤的自然史、临床特征和临床结果。我们的研究发现了153例独特的皮质室管膜瘤。平均发病年龄为21.2岁。男性占58.8%(90/153),女性占41.2%(63/153)。最常见的症状是发作活动,发生率为44.4%(68/153)。最近确认的C11orf95-RELA融合在13.7%的队列(21/153)中被确定,95.5%的病例(21/22)报告了分子特征。世界卫生组织2级和3级分别占52.3%(79/151)和47.7%(72/151)。大多数病例累及额叶(54.9%,84/153)。总切除率为80.4%(123/153)。肿瘤复发率为27.7%(39/141)。平均临床随访41.3个月。过期患者的平均总生存期为27.4个月,而平均无进展生存期为15.0个月。相比之下,C11orf95-RELA融合的皮质室管膜瘤和C11orf95-RELA融合的幕上室管膜瘤表现出不同的临床结果。为了进一步研究RELA融合对皮质室管膜瘤存活的意义,并确定是否应将具有C11orf95-RELA融合的皮质室管膜瘤作为一个独立的实体进行分类,还需要更大样本量的进一步研究。
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引用次数: 1
Remission of a Primary, Recurrent Thoracic Ewing Sarcoma in a 74-year-old woman. 一名 74 岁女性的原发性复发性胸腔尤文肉瘤得到缓解。
IF 0.9 Q4 ONCOLOGY Pub Date : 2022-07-04 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221110836
Robert M Tungate, Kristi Lara, Dakshesh Patel, Alexander Fedenko, James Hu

Ewing sarcoma is a primitive neuroectodermal tumor which seldom presents with primary disease in people over age 40 and outside of the appendicular or axial skeleton. We examine a case of primary thoracic Ewing Sarcoma diagnosed initially by CT-guided biopsy in a woman at the age of 74 years. The disease progressed after initial combined modality therapy consisting of neoadjuvant chemotherapy, surgical resection, and adjuvant radiation therapy and two additional courses of multiagent chemotherapy. After relapse of her disease, subsequent second- and third-line systemic agents which included chemotherapy and targeted agents were given with disease stabilization achieved now over 30 months from initial diagnosis. To our knowledge, this is the first report of a primary pulmonary Ewing sarcoma diagnosed in a patient greater than 70 years of age in whom multiple remissions have been achieved with tolerable toxicities.

尤文肉瘤是一种原始神经外胚层肿瘤,很少在 40 岁以上人群中出现原发性疾病,也很少发生在阑尾或轴向骨骼以外的部位。我们研究了一例原发性胸腔尤文肉瘤病例,患者是一名 74 岁的女性,最初通过 CT 引导活检确诊。在接受了包括新辅助化疗、手术切除、辅助放疗和两个额外的多药化疗疗程在内的初始联合治疗后,病情出现进展。疾病复发后,她接受了包括化疗和靶向药物在内的二线和三线系统药物治疗,现在距离最初诊断已超过 30 个月,病情趋于稳定。据我们所知,这是首例在 70 岁以上患者中诊断出原发性肺尤文肉瘤并在可耐受毒性反应的情况下实现多次缓解的报告。
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引用次数: 0
Colonic schwannoma: A case of unusual presentation and outcome. 结肠神经鞘瘤:一个不寻常的表现和结果。
IF 0.9 Q4 Medicine Pub Date : 2022-06-24 eCollection Date: 2022-01-01 DOI: 10.1177/20363613221110837
Salsabil Nasri, Mohamed Hedi Mraidha, Mehdi Ben Abdelkrim, Sabri Youssef, Fehmi Hamila, Mohamed Amine Elghali

Background: Schwannomas are mesenchymal tumors arising from neural sheath cells and whose diagnosis is based on immunohistochemistery. The digestive and especially colonic location of this tumor is rare. Commonly described in elderly patient, their malignancy is unusual.

Case report: We report the case of a 23-year-old girl, with learning disability, operated in emergency for acute peritonitis. Peroperatively, we discovered a peritonitis secondary to a bulky perforated cecal tumor. We performed a right colectomy and an ileostomy. The posterior margin of the resection was macroscopically involved. The diagnosis of colonic schwannoma was confirmed with immunopathological examination of the surgical specimen. Surgical exploration 4 months later and morphological investigations during 2 years showed stability of the tumoral residue in the right iliac fossa. However, we noticed, on CT scanning control 2 years postoperatively, the appearance of a mesenteric recurrence. Exploratory laparotomy confirmed the unresectability of this mesenteric mass and showed the presence of multiple parietal nodules whose biopsies revealed their neurofibromatous nature. The 4 year follow-up of the patient didn't reveal any complication rather than need to right nephrostomy due to ureter compression by the primary tumoral residue.

Discussion: Colonic schwannoma is a rare disease, commonly described in uncomplicated stages. The learning disability of our patient had favorized the absence of declared symptoms and promoted the evolution of the cecal swhannoma until its perforation. This complication was not described before. Local recurrence has been also rarely reported in the literature. Association with neurofibroma may be hereditary in a context of neurofibromatosis or sporadic.

Conclusion: Colonic schwannoma may have polymorphic presentation mimicking malignant tumor in such cases. Not treated in time, it can lead to severe complications, such as tumoral perforation. Surgical resection remains the mainstay treatment. The slow evolutionary genius of schwannoma, even when incompletely resected by necessity, confirms its good prognosis.

背景:神经鞘瘤是起源于神经鞘细胞的间充质肿瘤,其诊断基于免疫组织化学。这种肿瘤很少发生在消化道,尤其是结肠。常见于老年患者,恶性肿瘤不常见。病例报告:我们报告一例23岁的女孩,有学习障碍,在急性腹膜炎急诊手术。手术中,我们发现一个腹膜炎继发于一个巨大的盲肠穿孔肿瘤。我们做了右结肠切除术和回肠造口术。切除后缘在宏观上受累。手术标本的免疫病理检查证实了结肠神经鞘瘤的诊断。4个月后手术探查和2年形态学检查显示右侧髂窝肿瘤残留稳定。然而,我们注意到,在术后2年的CT扫描对照中,出现肠系膜复发。剖腹探查证实了这个肠系膜肿块的不可切除性,并显示存在多个肠壁结节,活检显示其神经纤维瘤性质。患者4年随访未发现任何并发症,无需因原发肿瘤残留压迫输尿管而行右肾造口术。讨论:结肠神经鞘瘤是一种罕见的疾病,通常分期简单。本例患者的学习障碍倾向于没有明确的症状,并促进盲肠鱼鞘瘤的发展直至其穿孔。这种并发症以前没有被描述过。文献中也很少报道局部复发。与神经纤维瘤的关联在神经纤维瘤病或散发性的情况下可能是遗传性的。结论:结肠神经鞘瘤可能具有类似恶性肿瘤的多形态表现。如果不及时治疗,它会导致严重的并发症,如肿瘤穿孔。手术切除仍是主要的治疗方法。神经鞘瘤的缓慢进化天赋,即使在必要时不完全切除,也证实了其良好的预后。
{"title":"Colonic schwannoma: A case of unusual presentation and outcome.","authors":"Salsabil Nasri,&nbsp;Mohamed Hedi Mraidha,&nbsp;Mehdi Ben Abdelkrim,&nbsp;Sabri Youssef,&nbsp;Fehmi Hamila,&nbsp;Mohamed Amine Elghali","doi":"10.1177/20363613221110837","DOIUrl":"https://doi.org/10.1177/20363613221110837","url":null,"abstract":"<p><strong>Background: </strong>Schwannomas are mesenchymal tumors arising from neural sheath cells and whose diagnosis is based on immunohistochemistery. The digestive and especially colonic location of this tumor is rare. Commonly described in elderly patient, their malignancy is unusual.</p><p><strong>Case report: </strong>We report the case of a 23-year-old girl, with learning disability, operated in emergency for acute peritonitis. Peroperatively, we discovered a peritonitis secondary to a bulky perforated cecal tumor. We performed a right colectomy and an ileostomy. The posterior margin of the resection was macroscopically involved. The diagnosis of colonic schwannoma was confirmed with immunopathological examination of the surgical specimen. Surgical exploration 4 months later and morphological investigations during 2 years showed stability of the tumoral residue in the right iliac fossa. However, we noticed, on CT scanning control 2 years postoperatively, the appearance of a mesenteric recurrence. Exploratory laparotomy confirmed the unresectability of this mesenteric mass and showed the presence of multiple parietal nodules whose biopsies revealed their neurofibromatous nature. The 4 year follow-up of the patient didn't reveal any complication rather than need to right nephrostomy due to ureter compression by the primary tumoral residue.</p><p><strong>Discussion: </strong>Colonic schwannoma is a rare disease, commonly described in uncomplicated stages. The learning disability of our patient had favorized the absence of declared symptoms and promoted the evolution of the cecal swhannoma until its perforation. This complication was not described before. Local recurrence has been also rarely reported in the literature. Association with neurofibroma may be hereditary in a context of neurofibromatosis or sporadic.</p><p><strong>Conclusion: </strong>Colonic schwannoma may have polymorphic presentation mimicking malignant tumor in such cases. Not treated in time, it can lead to severe complications, such as tumoral perforation. Surgical resection remains the mainstay treatment. The slow evolutionary genius of schwannoma, even when incompletely resected by necessity, confirms its good prognosis.</p>","PeriodicalId":46078,"journal":{"name":"Rare Tumors","volume":" ","pages":"20363613221110837"},"PeriodicalIF":0.9,"publicationDate":"2022-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/7a/8c/10.1177_20363613221110837.PMC9237920.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40468230","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
Extraskeletal myxoid chondrosarcoma: A case series and review of the literature 骨外黏液样软骨肉瘤1例并文献复习
IF 0.9 Q4 Medicine Pub Date : 2022-01-01 DOI: 10.1177/20363613221079754
Michael P. Fice, Linus Lee, Pavan Kottamasu, Abdullah Almajnooni, Matthew R. Cohn, Charles A. Gusho, S. Gitelis, Alan T. Blank
Background Extraskeletal myxoid chondrosarcoma (EMC) is a rare malignant soft tissue sarcoma (STS) that accounts for less than 3% of all soft tissue tumors. The conventional treatment for primary EMC is wide local excision with or without radiation therapy. Materials and Methods This study was a retrospective review of all EMC cases treated within a single institution between 1992 and 2019. EMC was diagnosed using a combination of histologic morphology and immunostaining, with confirmatory fluorescent in situ hybridization. Overall survival (OS) and disease-specific survival (DSS) were defined using Kaplan–Meier analysis. Results Fifteen patients were evaluated, including 11 males and four females. The average age at presentation was 51.7 ± 20.4 years and the mean follow-up time was 61.5 months (range, 5–286 months). The average resected tumor size at largest dimension was 7.14 cm (range, 2.4–18.7). Twelve of fifteen (80%) patients underwent wide local excision, and nine of the twelve (75%) underwent local radiation therapy. The 1-, 5-, and 10-year OS was 80% (95% CI, 59.8–100), 72% (95% CI, 48.5–95.5), and 72% (95% CI, 48.5–95.5), respectively. The 1-, 5-, and 10-year DSS was 92.3% (95% CI, 77.8–100), 83.1% (95% CI, 61.5–100), and 83.1% (95% CI, 61.5–100), respectively. At last follow-up, 11 patients were alive and ten (90.9%) were disease free. Conclusions Extraskeletal myxoid chondrosarcoma is a very rare STS most often seen in males and in the extremities. Our cohort was too small to provide meaningful statistical analysis; however, we observed lower rates of local recurrence in patients treated with radiation.
背景骨外黏液样软骨肉瘤(EMC)是一种罕见的恶性软组织肉瘤(STS),在所有软组织肿瘤中所占比例不到3%。原发性EMC的传统治疗方法是广泛的局部切除,无论是否进行放射治疗。材料和方法本研究对1992年至2019年间在一家机构接受治疗的所有EMC病例进行了回顾性审查。采用组织形态学和免疫染色相结合的方法,结合荧光原位杂交对EMC进行诊断。总生存期(OS)和疾病特异性生存期(DSS)采用Kaplan-Meier分析进行定义。结果15例患者接受了评估,其中男性11例,女性4例。出现时的平均年龄为51.7±20.4岁,平均随访时间为61.5个月(范围为5-286个月)。最大尺寸的平均切除肿瘤大小为7.14cm(范围2.4-18.7)。15名患者中有12名(80%)接受了广泛的局部切除,12名患者中的9名(75%)接受了局部放射治疗。1年、5年和10年OS分别为80%(95%CI,59.8-100)、72%(95%CI,48.5-95.5)和72%(95%可信区间,48.5-9.5)。1年、5年和10年DSS分别为92.3%(95%置信区间,77.8-100)、83.1%(95%可信区间,61.5-100)和83.1%(95%CI,61.5-100)。在最后的随访中,11名患者存活,10名(90.9%)无疾病。结论骨外黏液样软骨肉瘤是一种非常罕见的STS,最常见于男性和四肢。我们的队列太小,无法提供有意义的统计分析;然而,我们观察到接受放射治疗的患者局部复发率较低。
{"title":"Extraskeletal myxoid chondrosarcoma: A case series and review of the literature","authors":"Michael P. Fice, Linus Lee, Pavan Kottamasu, Abdullah Almajnooni, Matthew R. Cohn, Charles A. Gusho, S. Gitelis, Alan T. Blank","doi":"10.1177/20363613221079754","DOIUrl":"https://doi.org/10.1177/20363613221079754","url":null,"abstract":"Background Extraskeletal myxoid chondrosarcoma (EMC) is a rare malignant soft tissue sarcoma (STS) that accounts for less than 3% of all soft tissue tumors. The conventional treatment for primary EMC is wide local excision with or without radiation therapy. Materials and Methods This study was a retrospective review of all EMC cases treated within a single institution between 1992 and 2019. EMC was diagnosed using a combination of histologic morphology and immunostaining, with confirmatory fluorescent in situ hybridization. Overall survival (OS) and disease-specific survival (DSS) were defined using Kaplan–Meier analysis. Results Fifteen patients were evaluated, including 11 males and four females. The average age at presentation was 51.7 ± 20.4 years and the mean follow-up time was 61.5 months (range, 5–286 months). The average resected tumor size at largest dimension was 7.14 cm (range, 2.4–18.7). Twelve of fifteen (80%) patients underwent wide local excision, and nine of the twelve (75%) underwent local radiation therapy. The 1-, 5-, and 10-year OS was 80% (95% CI, 59.8–100), 72% (95% CI, 48.5–95.5), and 72% (95% CI, 48.5–95.5), respectively. The 1-, 5-, and 10-year DSS was 92.3% (95% CI, 77.8–100), 83.1% (95% CI, 61.5–100), and 83.1% (95% CI, 61.5–100), respectively. At last follow-up, 11 patients were alive and ten (90.9%) were disease free. Conclusions Extraskeletal myxoid chondrosarcoma is a very rare STS most often seen in males and in the extremities. Our cohort was too small to provide meaningful statistical analysis; however, we observed lower rates of local recurrence in patients treated with radiation.","PeriodicalId":46078,"journal":{"name":"Rare Tumors","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43245665","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 4
Pelvic retroperitoneal pleomorphic hyalinizing angiectatic tumor: A case report and review of literature. 盆腔腹膜后多形性透明化血管扩张瘤1例并文献复习。
IF 0.9 Q4 Medicine Pub Date : 2022-01-01 DOI: 10.1177/20363613221103751
Mehdi Salehipour, Mohammad Hossein Anbardar, Bita Geramizadeh, Hamed Jafari, Ali Zare, Ali Adib

Pleomorphic hyalinizing angiectatic tumor (PHAT) is a rare tumor of the soft tissue, usually located in lower extremities. There are rarely tumors reported in other anatomic locations. Herein, we report retroperitoneal PHAT in a male patient. A 41-year-old man was referred to our clinic due to an incidentally found retroperitoneal mass. Computed tomography (CT) scan showed a solid hypoechoic lesion containing fat component and calcified elements measuring about 80*72*45 mm in the right lower quadrant (RLQ) of the abdomen. Magnetic resonance imaging (MRI) showed circumscribe lesion measuring about 60 x 48 mm with partial enhancement and fat component. In pelvic exploration, a large mass was found that had encased the right external iliac artery and vein. Therefore, the mass and its surrounding iliac vessels were excised and removed en block. Then, the external iliac vessels were reconstructed with Gortex graft. No recurrence was found in 1 month and 3 months post-operation follow up. We report a pelvic retroperitoneal PHAT as a rare location of this tumor. It seems that PHAT must be considered in differential diagnosis in patients with soft tissue tumors in the pelvic cavity.

摘要多形性透明化血管扩张瘤(PHAT)是一种罕见的软组织肿瘤,通常位于下肢。在其他解剖部位很少有肿瘤的报道。在此,我们报告一位男性患者腹膜后PHAT。一位41岁的男性因偶然发现腹膜后肿块而被转介到我们诊所。CT示腹部右下象限(RLQ)一实性低回声病灶,含脂肪成分和钙化元素,大小约80*72*45 mm。磁共振成像(MRI)显示病灶范围约60 x 48 mm,局部增强,脂肪成分。在盆腔探查中,发现一个大肿块包裹了右侧髂外动脉和静脉。因此,肿块及其周围的髂血管被切除并整体切除。然后用Gortex移植物重建髂外血管。术后1个月及3个月随访均无复发。我们报告盆腔腹膜后PHAT是一个罕见的肿瘤位置。因此,在盆腔软组织肿瘤患者的鉴别诊断中必须考虑到PHAT。
{"title":"Pelvic retroperitoneal pleomorphic hyalinizing angiectatic tumor: A case report and review of literature.","authors":"Mehdi Salehipour,&nbsp;Mohammad Hossein Anbardar,&nbsp;Bita Geramizadeh,&nbsp;Hamed Jafari,&nbsp;Ali Zare,&nbsp;Ali Adib","doi":"10.1177/20363613221103751","DOIUrl":"https://doi.org/10.1177/20363613221103751","url":null,"abstract":"<p><p>Pleomorphic hyalinizing angiectatic tumor (PHAT) is a rare tumor of the soft tissue, usually located in lower extremities. There are rarely tumors reported in other anatomic locations. Herein, we report retroperitoneal PHAT in a male patient. A 41-year-old man was referred to our clinic due to an incidentally found retroperitoneal mass. Computed tomography (CT) scan showed a solid hypoechoic lesion containing fat component and calcified elements measuring about 80*72*45 mm in the right lower quadrant (RLQ) of the abdomen. Magnetic resonance imaging (MRI) showed circumscribe lesion measuring about 60 x 48 mm with partial enhancement and fat component. In pelvic exploration, a large mass was found that had encased the right external iliac artery and vein. Therefore, the mass and its surrounding iliac vessels were excised and removed en block. Then, the external iliac vessels were reconstructed with Gortex graft. No recurrence was found in 1 month and 3 months post-operation follow up. We report a pelvic retroperitoneal PHAT as a rare location of this tumor. It seems that PHAT must be considered in differential diagnosis in patients with soft tissue tumors in the pelvic cavity.</p>","PeriodicalId":46078,"journal":{"name":"Rare Tumors","volume":"14 ","pages":"20363613221103751"},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/51/60/10.1177_20363613221103751.PMC9150221.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10252651","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
Primary leiomyosarcoma of the colon with synchronous liver metastasis 原发性结肠平滑肌肉瘤伴同步肝转移
IF 0.9 Q4 Medicine Pub Date : 2022-01-01 DOI: 10.1177/20363613221080549
D. Massaras, E. Kontis, K. Stamatis, E. Zampeli, D. Myoteri, Elias Primetis, E. Pantiora, G. Fragulidis
Leiomyosarcomas (LMS) are mesenchymal tumors of smooth muscle origin. Approximately 20% of leiomyosarcomas are found in the GI tract, and account for 1–2% of GI malignancies. Within the gastrointestinal tract, the small intestine is the most common site of presentation followed by the colon. They are often diagnosed incidentally during abdominal pain investigation, and they usually present in the fifth decade of life. In the past, the diagnostic differentiation between leiomyosarcomas of gastrointestinal tract and gastrointestinal stromal tumors (GISTs) was very challenging as they share common microscopic appearance. Nowadays, these tumors are diagnosed by immunohistochemical methods as they are positive for smooth muscle markers actin (SMA) and desmin, negative for GIST markers such as receptor tyrosine kinase (KIT), CD34, DOG1, and negative for the schwannoma marker S100 protein. In the current literature, most cases of intestinal leiomyosarcomas are localized in the small intestine, and there are no reports of synchronous liver metastases at the time of presentation of the primary tumor. Herein, we present a patient who was admitted in our department, with the diagnosis of primary leiomyosarcoma of the colon and synchronous liver metastasis.
平滑肌肉瘤(LMS)是起源于平滑肌的间质肿瘤。大约20%的平滑肌肉瘤发生在胃肠道,占胃肠道恶性肿瘤的1-2%。在胃肠道内,小肠是最常见的发病部位,其次是结肠。通常是在腹痛调查中偶然发现的,通常出现在50岁左右。由于胃肠道平滑肌肉瘤与胃肠道间质瘤(gist)在显微镜下具有相同的外观,因此过去的诊断鉴别非常具有挑战性。目前,这些肿瘤通过免疫组织化学方法诊断,因为它们的平滑肌标志物肌动蛋白(SMA)和desmin呈阳性,GIST标志物如受体酪氨酸激酶(KIT)、CD34、DOG1呈阴性,神经鞘瘤标志物S100蛋白呈阴性。在目前的文献中,大多数肠道平滑肌肉瘤的病例局限于小肠,在原发肿瘤出现时没有同步肝转移的报道。在此,我们报告一位在我科住院的病人,诊断为原发性结肠平滑肌肉瘤并同步肝转移。
{"title":"Primary leiomyosarcoma of the colon with synchronous liver metastasis","authors":"D. Massaras, E. Kontis, K. Stamatis, E. Zampeli, D. Myoteri, Elias Primetis, E. Pantiora, G. Fragulidis","doi":"10.1177/20363613221080549","DOIUrl":"https://doi.org/10.1177/20363613221080549","url":null,"abstract":"Leiomyosarcomas (LMS) are mesenchymal tumors of smooth muscle origin. Approximately 20% of leiomyosarcomas are found in the GI tract, and account for 1–2% of GI malignancies. Within the gastrointestinal tract, the small intestine is the most common site of presentation followed by the colon. They are often diagnosed incidentally during abdominal pain investigation, and they usually present in the fifth decade of life. In the past, the diagnostic differentiation between leiomyosarcomas of gastrointestinal tract and gastrointestinal stromal tumors (GISTs) was very challenging as they share common microscopic appearance. Nowadays, these tumors are diagnosed by immunohistochemical methods as they are positive for smooth muscle markers actin (SMA) and desmin, negative for GIST markers such as receptor tyrosine kinase (KIT), CD34, DOG1, and negative for the schwannoma marker S100 protein. In the current literature, most cases of intestinal leiomyosarcomas are localized in the small intestine, and there are no reports of synchronous liver metastases at the time of presentation of the primary tumor. Herein, we present a patient who was admitted in our department, with the diagnosis of primary leiomyosarcoma of the colon and synchronous liver metastasis.","PeriodicalId":46078,"journal":{"name":"Rare Tumors","volume":" ","pages":""},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48957682","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 2
Giant teratoma of the pancreas expanding to the mediastinum: Rare tumor and literature review. 胰腺巨大畸胎瘤扩散至纵隔:罕见肿瘤及文献回顾。
IF 0.9 Q4 Medicine Pub Date : 2022-01-01 DOI: 10.1177/20363613221147470
Nikolaos Tasis, Antonia A Prountzopoulou, Evangelia Skafida, Maria Arnaouti, Theodoros Tsirlis, Aris Plastiras, Dimitrios K Manatakis, Niki Arnogiannaki, Dimitrios P Korkolis

Pancreatic mature cystic teratomas are very rare with limited cases found in the literature. These lesions raise a diagnostic challenge and complicate the surgical approach not only because of their anatomic position but also because of their ever-growing size. An elusive diagnosis, usually leads to the operative theatre where surgical resection takes place. We present a rare case of a large pancreatic cystic teratoma extending into the mediastinum in a 29-year-old woman which was succesfully managed with en-bloc distal pancreatectomy and spleenectomy.

胰腺成熟囊性畸胎瘤是非常罕见的,在文献中发现的病例有限。这些病变不仅由于其解剖位置,而且由于其不断增长的大小,对诊断提出了挑战,并使手术入路复杂化。一个难以捉摸的诊断,通常导致手术切除进行手术室。我们报告一个罕见的病例,一个巨大的胰腺囊性畸胎瘤延伸到纵隔,在一个29岁的女性成功地管理与整体远端胰腺切除术和脾切除术。
{"title":"Giant teratoma of the pancreas expanding to the mediastinum: Rare tumor and literature review.","authors":"Nikolaos Tasis,&nbsp;Antonia A Prountzopoulou,&nbsp;Evangelia Skafida,&nbsp;Maria Arnaouti,&nbsp;Theodoros Tsirlis,&nbsp;Aris Plastiras,&nbsp;Dimitrios K Manatakis,&nbsp;Niki Arnogiannaki,&nbsp;Dimitrios P Korkolis","doi":"10.1177/20363613221147470","DOIUrl":"https://doi.org/10.1177/20363613221147470","url":null,"abstract":"<p><p>Pancreatic mature cystic teratomas are very rare with limited cases found in the literature. These lesions raise a diagnostic challenge and complicate the surgical approach not only because of their anatomic position but also because of their ever-growing size. An elusive diagnosis, usually leads to the operative theatre where surgical resection takes place. We present a rare case of a large pancreatic cystic teratoma extending into the mediastinum in a 29-year-old woman which was succesfully managed with en-bloc distal pancreatectomy and spleenectomy.</p>","PeriodicalId":46078,"journal":{"name":"Rare Tumors","volume":"14 ","pages":"20363613221147470"},"PeriodicalIF":0.9,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/3b/bb/10.1177_20363613221147470.PMC9806373.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10494337","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
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Rare Tumors
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