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Aggressive osteoblastoma with a secondary aneurysmal bone cyst treated with denosumab. 地诺单抗治疗侵袭性成骨细胞瘤伴继发性动脉瘤性骨囊肿。
IF 0.9 Q4 Medicine Pub Date : 2021-08-29 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211034710
Karlton Wong, Jomjit Chantharasamee, Scott Nelson, Mark A Eckardt, Kambiz Motamedi, Francis J Hornicek, Arun S Singh

Osteoblastomas and aneurysmal bone cysts (ABC) are rare benign bone tumors that make up about 1%-2% of primary bone malignancies, typically occurring in young patients with a median age of 20 years, most commonly effecting the axial skeleton. ABCs may develop independently as primary lesions, or secondary to other bony lesions including osteoblastomas, chondroblastomas, and giant cell tumors. Treatment of unresectable or extensive osteoblastomas can be challenging. In 2013, the Food and Drug Administration (FDA) approved denosumab for the treatment of giant cell tumors of the bone due to its efficacy in these morbid bony lesions. Various case reports have shown that osteoblastomas can respond to denosumab. Furthermore, numerous ABC case reports have described the efficacy of denosumab in these situations. We herein describe a unique case of a young patient with an aggressive osteoblastoma and secondary ABCs who was successfully treated with denosumab.

成骨细胞瘤和动脉瘤性骨囊肿(ABC)是罕见的良性骨肿瘤,约占原发性骨恶性肿瘤的1%-2%,通常发生在中位年龄为20岁的年轻患者中,最常见于中轴骨骼。abc可以独立发展为原发性病变,也可以继发于其他骨病变,包括成骨细胞瘤、成软骨细胞瘤和巨细胞瘤。不可切除或广泛的成骨细胞瘤的治疗是具有挑战性的。2013年,美国食品和药物管理局(FDA)批准了denosumab用于治疗骨巨细胞瘤,因为它对这些病态的骨病变有疗效。各种病例报告显示成骨细胞瘤对地诺单抗有反应。此外,许多ABC病例报告描述了denosumab在这些情况下的疗效。我们在此描述一个独特的情况下,一个年轻的患者与侵袭性成骨细胞瘤和继发性abc谁是成功地用地诺单抗治疗。
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引用次数: 5
Choriocarcinoma metastatic to the skin: A rare occurrence associated with dismal outcome. 绒毛膜癌转移到皮肤:一种罕见的发生与令人沮丧的结果。
IF 0.9 Q4 Medicine Pub Date : 2021-08-20 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211039724
Mousa ElKhaldi, Rakan Radi, Maysa Al-Hussaini

Germ cell tumors (GCTs) are a histologically heterogeneous group of tumors that arise from the primitive germ cell of the embryonic gonad. Choriocarcinoma is a variant of GCTs that is prone to hematogenous metastasis to the liver, lung, and brain. Cutaneous metastasis in choriocarcinoma is rarely encountered with only a few cases reported in literature. We report the case of a 28-year-old male presenting with lower back pain that, upon further work-up, was diagnosed with pure choriocarcinoma of the testes. Around 9 months after his initial presentation, he developed a cutaneous back lesion. Microscopic examination confirmed the presence of choriocarcinoma composed of mononuclear cytotrophoblasts which interweave with multinucleated syncytiotrophoblasts. The patient passed away 3 weeks after the onset of cutaneous metastasis.

生殖细胞肿瘤(gct)是一种组织学异质性的肿瘤,起源于胚胎性腺的原始生殖细胞。绒毛膜癌是gct的一种变体,容易发生肝、肺和脑的血液转移。绒毛膜癌的皮肤转移是罕见的,文献报道的病例很少。我们报告的情况下,一个28岁的男性提出腰痛,经进一步检查,被诊断为纯绒毛膜癌的睾丸。初次就诊后约9个月,患者出现背部皮肤病变。显微镜检查证实绒毛膜癌由单核细胞滋养细胞与多核合胞滋养细胞交织而成。患者于发生皮肤转移后3周死亡。
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引用次数: 0
Olaratumab's failure in soft tissue sarcoma. 奥拉拉单抗在软组织肉瘤中的失败。
IF 0.9 Q4 Medicine Pub Date : 2021-07-18 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211034115
Maroun Bou Zerdan, Aram H Bidikian, Ibrahim Alameh, Clara El Nakib, Hazem I Assi

Soft tissue sarcomas remain one of the rarest malignancies with numerous subtypes that go undiagnosed. The PDGFRα antagonist Olaratumab (Lartruvo) was withdrawn from the market due to disappointing findings in the phase III studies. We share our experience with this medication in a tertiary care center in the Middle East and North Africa region. Monitor the effect of Olaratumab on sarcomas when it was used prior to its withdrawal, and compare our findings with the literature. We performed a retrospective analysis of adult patients with advanced-/metastatic soft tissue sarcomas treated with at least two cycles of Olaratumab at a tertiary care center in Lebanon during the period from January 1, 2017 to December 31, 2018. Fifteen patients were included in the study. The mean age was 49 with a range of 26-75 years. The median duration of the use of Olaratumab was 21.3 months with a range of 7.3-37 months. The average number of number of cycles received per patient was four. Five patients were deceased. Median PFS was 7.87 months (95% CI 5.28-10.45), and mean OS was 12.26 months (95% CI 8.47-16.05) Median OS was 9.8 months (95% CI 6.07-13.53). Olaratumab has been withdrawn from the market, and it is currently being investigated as part of the phase II ANNOUNCE 2 trial. Our experience from a tertiary care center shows results similar to those reported in the literature. The immunogenicity and heterogeneity of soft tissue sarcomas pose a challenge to the treatment of soft tissue sarcomas, but they also allow a wide array of possible management solutions.

软组织肉瘤仍然是最罕见的恶性肿瘤之一,有许多亚型未被诊断。PDGFRα拮抗剂Olaratumab (Lartruvo)因III期研究结果令人失望而退出市场。我们在中东和北非地区的一家三级保健中心分享我们使用这种药物的经验。监测停药前使用奥拉拉单抗对肉瘤的影响,并将我们的发现与文献进行比较。我们对2017年1月1日至2018年12月31日期间在黎巴嫩一家三级医疗中心接受至少两个周期奥拉拉单抗治疗的晚期/转移性软组织肉瘤成年患者进行了回顾性分析。15名患者参与了这项研究。平均年龄49岁,年龄范围26-75岁。使用Olaratumab的中位持续时间为21.3个月,范围为7.3-37个月。每位患者接受的平均周期数为4次。5名患者死亡。中位PFS为7.87个月(95% CI 5.28-10.45),平均OS为12.26个月(95% CI 8.47-16.05),中位OS为9.8个月(95% CI 6.07-13.53)。Olaratumab已退出市场,目前正在作为II期ANNOUNCE 2试验的一部分进行研究。我们在三级护理中心的经验显示了与文献报道相似的结果。软组织肉瘤的免疫原性和异质性对软组织肉瘤的治疗提出了挑战,但它们也允许广泛的可能的管理解决方案。
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引用次数: 0
A rare case of chordoma presenting as a Pancoast tumor. 一例罕见的脊索瘤表现为Pancoast肿瘤。
IF 0.9 Q4 Medicine Pub Date : 2021-07-02 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211029493
Philip T Sobash, Krishna Vedala, Daniel Alfano, Heather Pinckard-Dover, Jason L Muesse, Raman Desikan

The notochord is the defining structure of all chordate embryos. It is a midline structure ventral to the ectoderm, neural plates, and neural arch. Remnants of the notochord ultimately give rise to the nucleus pulposus. The function of the notochord is to organize the surrounding structures. Chordoma is a rare malignant bone tumor arising from remnants of the notochord. These tumors are indolent and can present as incidental or locally advanced involving adjacent structures. These tumors typically present at the skull base and sacral spine but more rarely can be seen on the cervical and thoracic spine. Rare cases of chordoma invading the brachial plexus have been recorded. Surgical resection is the mainstay of treatment for chordomas. We would like to discuss a novel presentation of a chordoma as a Pancoast tumor, and aim to highlight the clinical importance of accurate diagnosis and planning therapy along with poor prognosis of incomplete surgical resection.

脊索是所有脊索动物胚胎的决定性结构。它是外胚层、神经板和神经弓腹侧的中线结构。脊索的残余最终形成髓核。脊索的功能是组织周围的结构。脊索瘤是一种罕见的恶性骨肿瘤,起源于脊索的残余。这些肿瘤是无痛的,可表现为偶发或局部进展,累及邻近结构。这些肿瘤通常出现在颅底和骶骨,但很少出现在颈椎和胸椎。有罕见的脊索瘤侵袭臂丛的病例记录。手术切除是脊索瘤的主要治疗方法。我们将讨论脊索瘤作为Pancoast肿瘤的一种新表现,并旨在强调准确诊断和计划治疗的临床重要性,以及不完全手术切除的不良预后。
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引用次数: 0
Extragonadal germ cell tumor, a report of two cases presenting in the gastrointestinal tract. 生殖道外生殖细胞瘤2例报告。
IF 0.9 Q4 Medicine Pub Date : 2021-06-30 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211029487
Mousa Elkhaldi, Ahamd Moayad Naser, Yazan AlHalaseh, Maysa Al-Hussaini

Germ cell tumors are a heterogeneous group of tumors that can present primarily as gonadal tumors in either a localized or metastatic pattern. Rarely these tumors can initially present at extra-gonadal locations, including the gastrointestinal tract. We report two young male patients who presented with nonspecific gastrointestinal symptoms caused by a mass lesion involving the duodenum. Pathologically, both were confirmed to be germ cell tumors; an unfamiliar initial presentation of germ cell tumors. In both cases, evidence of pre-existing gonadal tumor in the form of a testicular mass and a burned-out tumor with microlithiasis, in the first and second cases, respectively was detected following the confirmed diagnosis of extra-gonadal germ cell tumor. Each patient's clinical course and outcome emphasizes the importance of a high index of suspicion, timely diagnosis, and appropriate management.

生殖细胞肿瘤是一类异质性肿瘤,主要表现为局部或转移性性腺肿瘤。这些肿瘤最初很少出现在性腺外部位,包括胃肠道。我们报告了两名年轻男性患者,他们表现出由十二指肠肿块病变引起的非特异性胃肠道症状。病理证实为生殖细胞瘤;一种不熟悉的生殖细胞肿瘤的初始表现。在这两个病例中,在确诊为性腺外生殖细胞肿瘤后,分别在第一个和第二个病例中发现了以睾丸肿块和烧伤肿瘤合并微石症的形式存在的性腺肿瘤的证据。每位患者的临床过程和结果都强调了高度怀疑、及时诊断和适当管理的重要性。
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引用次数: 2
Undifferentiated pleomorphic sarcoma in oropharyngeal mucosa of patients with multiple basal cell carcinomas. 多发性基底细胞癌患者口咽黏膜中的未分化多形性肉瘤。
IF 0.9 Q4 Medicine Pub Date : 2021-06-30 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211026483
Andrea Dekanić, Marko Velepič, Margita Belušić Gobić, Ita Hadžisejdić, Nives Jonjić

Malignant mesenchymal tumors of oropharyngeal mucosa are rare. Those with fibroblastic and histiocytic differentiation in the skin are called atypical fibroxanthoma (AFX) and in the soft tissue undifferentiated pleomorphic sarcoma (UPS). Here we present a case of an older patient with a history of multiple basal cell carcinomas and recently with a rapidly growing polypoid lesion in the mucosa of posterior oropharyngeal wall with AFX/UPS morphology. The differential diagnosis, histological pitfalls of this poorly characterized mesenchymal lesions, and the challenges associated with treatment are discussed.

口咽粘膜恶性间质瘤十分罕见。在皮肤中,具有成纤维细胞和组织细胞分化的恶性间质瘤被称为非典型纤维黄瘤(AFX),在软组织中被称为未分化多形性肉瘤(UPS)。在此,我们介绍了一例老年患者的病例,该患者曾有多发性基底细胞癌病史,最近口咽后壁粘膜出现快速生长的息肉样病变,形态为AFX/UPS。本文讨论了这种特征不清的间质病变的鉴别诊断、组织学陷阱以及治疗方面的挑战。
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引用次数: 0
Breast erythema and nodular skin metastasis as the first manifestation of breast implant-associated anaplastic large cell lymphoma. 乳房红斑和结节性皮肤转移是乳房植入物相关间变性大细胞淋巴瘤的首要表现。
IF 0.9 Q4 Medicine Pub Date : 2021-06-30 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211028498
Noellie Ducastel, Ioana-Mariana Cimpean, Ivan Theate, Olivier Vanhooteghem

Anaplastic large cell lymphoma (BIA-ALCL) associated with rough textured breast implants was first reported in 1997. It is a non-Hodgkin's lymphoma originating from a T lymphocyte which occurs on average 10.9 years after placement of the breast implant. BIA-ALCL mainly manifests as a periprosthetic seroma or a mass adjacent to the implant. To our knowledge, we describe the first case of BIA-ALCL with initial presentation by indurate erythematous plates located in both breasts and the progressive appearance of several asymptomatic metastatic nodular lesions that have been appearing on the right arm some weeks later.

间变性大细胞淋巴瘤(BIA-ALCL)与粗糙的乳房植入物相关,于1997年首次报道。这是一种起源于T淋巴细胞的非霍奇金淋巴瘤,发生在乳房植入物平均10.9年后。BIA-ALCL主要表现为假体周围的血肿或假体附近的肿块。据我们所知,我们描述了第一例BIA-ALCL,最初表现为双乳硬化性红斑板,几周后在右臂出现了几个无症状的转移性结节病变。
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引用次数: 2
Intraosseous ancient Schwannoma: A rare case in the mandible and a literature review. 骨内古老神经鞘瘤:下颌骨一例罕见病例并文献复习。
IF 0.9 Q4 Medicine Pub Date : 2021-06-27 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211026480
Neda Kardouni Khoozestani, Maziar Motiee-Langroudi, Ali Salehi, Paniz Ranji

Schwannoma, otherwise known as neurilemmoma, is a benign tumor that originates from Schwann cells. Ancient Schwannoma is a long-standing Schwannoma with degenerative and xanthomatous changes. The Head and neck region is one of the most common sites for nerve sheath tumors but Involvement of the jaw is a considerably infrequent event. We report a rare case of intraosseous ancient Schwannoma of the mandible in a 24-year-old male patient which was initially misdiagnosed with carcinoma. The clinical features, radiographic findings, and treatment plan are discussed. Furthermore, a thorough literature review demonstrated nine published cases of intra-osseous ancient Schwannoma with their summarized features. Recognition of various histopathologic features of ancient Schwannoma is essential to prevent over-treatment.

神经鞘瘤,又称神经鞘瘤,是一种起源于雪旺细胞的良性肿瘤。古代神经鞘瘤是一种长期存在的神经鞘瘤,伴有退行性和黄瘤性变化。头颈部是神经鞘肿瘤最常见的部位之一,但累及颌骨是相当罕见的事件。我们报告一个罕见的病例骨内古老的神经鞘瘤下颌骨在24岁的男性患者最初被误诊为癌。本文讨论了其临床特征、影像学表现和治疗方案。此外,我们对9例已发表的骨内古神经鞘瘤进行了全面的文献回顾,并总结了其特征。认识各种组织病理特征的古老神经鞘瘤是必不可少的,以防止过度治疗。
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引用次数: 6
Histological scores and tumor size on stage II in adrenocortical carcinomas. 肾上腺皮质癌II期的组织学评分和肿瘤大小。
IF 0.9 Q4 Medicine Pub Date : 2021-06-27 eCollection Date: 2021-01-01 DOI: 10.1177/20363613211026494
Rui Caetano Oliveira, Maria João Martins, Carolina Moreno, Rui Almeida, João Carvalho, Paulo Teixeira, Miguel Teixeira, Edgar Tavares Silva, Isabel Paiva, Arnaldo Figueiredo, Maria Augusta Cipriano

Adrenocortical carcinomas (ACC) are aggressive tumors with a poor prognosis. Histological scores are advised for the diagnosis, however, there are borderline cases that may be misjudged as adrenocortical adenomas (ACA). The three main scores used are: Weiss Modified System (WMS), Reticulin Algorithm (RA), and Helsinki Score (HS). We intend to compare the accuracy of the three scores in ACC diagnosis and to identify predictive factors of overall survival (OS). Retrospective study (2004-2016) at Centro Hospitalar e Universitário de Coimbra of the adrenal tumors, classified as ACC or ACA, with a history of posterior tumor relapse/metastases, without lesions in the contralateral adrenal gland: 13F and 6M, with a median age of 51 ± 12.41 years. Nodules' median size was 9.20 ± 6.2 cm. Patients had a median OS of 52 ± 18.6 months, with 57.9% and 46.3%, at 3 and 5 years. Seven patients had local recurrence and nine had metastases. Thirteen cases were in stage II. The WMS and the HS allowed a diagnosis of ACC in 15 cases and the RA defined ACC in 17 cases. All cases had, at least, focal disruption of the reticulin framework. More than 5 mitosis/50 HPF was associated with worse OS: 49.67 ± 21.43 versus 108.86 ± 14.02 months (p = 0.026). In patients with stage II, tumor size ⩾10 cm was associated with worse OS: 19.25 ± 7.15 versus 96.11 ± 16.7 months (p = 0.007), confirmed by multivariate analysis (p = 0.031). The correct diagnosis of ACC is a pathologist responsibility. The RA seems the most accurate. Any loss of the reticulin framework should raise awareness for malignancy. In patients on stage II, a size ⩾10 cm is a predictor of worse prognosis.

肾上腺皮质癌(ACC)是侵袭性肿瘤,预后差。组织学评分是诊断的建议,然而,也有可能被误诊为肾上腺皮质腺瘤(ACA)的边缘性病例。使用的三种主要评分是:Weiss Modified System (WMS)、Reticulin Algorithm (RA)和Helsinki Score (HS)。我们打算比较三种评分在ACC诊断中的准确性,并确定总生存期(OS)的预测因素。回顾性研究(2004-2016年)在Universitário de Coimbra中心医院进行的,分类为ACC或ACA,有肿瘤后侧复发/转移史,对侧肾上腺无病变的肾上腺肿瘤:13F和6M,中位年龄51±12.41岁。结节的中位大小为9.20±6.2 cm。患者在3年和5年的中位OS为52±18.6个月,分别为57.9%和46.3%。7例局部复发,9例转移。13例为II期。WMS和HS诊断ACC 15例,RA诊断ACC 17例。所有病例至少有局灶性网状结构破坏。超过5次有丝分裂/50 HPF与较差的OS相关:49.67±21.43 vs 108.86±14.02月(p = 0.026)。在II期患者中,肿瘤大小小于10 cm与更差的OS相关:19.25±7.15与96.11±16.7个月(p = 0.007),经多变量分析证实(p = 0.031)。正确诊断ACC是病理学家的责任。RA似乎是最准确的。网状蛋白框架的任何缺失都应提高对恶性肿瘤的认识。在II期患者中,小于10厘米是预后较差的预测因子。
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引用次数: 1
A case report of imiquimod topical therapy as treatment for cutaneous metastasis of breast cancer. 咪喹莫特局部治疗乳腺癌皮肤转移1例报告。
IF 0.9 Q4 Medicine Pub Date : 2021-06-24 eCollection Date: 2021-01-01 DOI: 10.1177/2036361320975748
Anthony L Nguyen, Esther G Chong, Joanne Lee, Saied Mirshahidi, Hamid Mirshahidi

Cutaneous metastasis of breast cancer carries a poor prognosis, invokes a poor quality of life, and increases mortality by raising one's risk of bleeding and infection. Currently, options for treatment are systemic chemotherapy, surgical resection and radiation. These treatments are invasive and can have toxic side effects. A 50-year-old African-American woman with stage IV breast cancer with cutaneous metastasis to the left anterior chest and left supraclavicular area was successfully treated with topical imiquimod. She experienced improvement in appearance and symptoms within several months of starting treatment, resulting in near resolution of her cutaneous metastasis. Imiquimod is currently approved for several cutaneous conditions and has the potential to treat cutaneous metastasis of breast cancer.

乳腺癌的皮肤转移预后差,生活质量差,并通过增加出血和感染的风险增加死亡率。目前,治疗的选择是全身化疗、手术切除和放疗。这些治疗是侵入性的,可能有毒副作用。一位50岁的非裔美国女性患有IV期乳腺癌,并伴有左前胸和左锁骨上区域的皮肤转移,我们成功地用局部咪喹莫特治疗了乳腺癌。在开始治疗的几个月内,她的外观和症状有所改善,导致她的皮肤转移几乎消失。咪喹莫特目前被批准用于几种皮肤疾病,并有可能治疗乳腺癌的皮肤转移。
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引用次数: 3
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Rare Tumors
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