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Epithelioid Hemangioendothelioma with WWTR1-CAMTA1 Fusion in the Parotid Gland Presenting as Bell's Palsy. 腮腺上皮样血管内皮瘤与WWTR1-CAMTA1融合表现为贝尔麻痹。
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-06-24 eCollection Date: 2022-01-01 DOI: 10.1155/2022/5687190
Landon J Kunzelman, Shweta Agarwal, Nathan Boyd, Cory J Broehm

Epithelioid hemangioendothelioma is a rare tumor of endothelial differentiation most commonly arising in soft tissue, liver, and lung, following a variable clinical course. Most cases are characterized by a t(1;3)(p36;q23-25) resulting in WWTR1-CAMTA1 fusion. Only five epithelioid hemangioendothelioma have been previously reported arising in the salivary glands. None have presented as Bell's palsy. In the current case, a 37-year-old female presented with a longstanding complaint of pain and fullness in the right preauricular region and progressive episodes of Bell's palsy and facial nerve weakness. Surgical resection showed a tumor comprised of atypical cells with occasional intracytoplasmic vacuoles in a fibromyxoid stroma. Immunohistochemical stains demonstrated the neoplastic cells expressed ERG, CD31, and CD34, confirming vascular differentiation. Fluorescence in situ hybridization revealed a t(1;3)(p36;q25), confirming a diagnosis of epithelioid hemangioendothelioma. At 12-month follow-up, the patient has no evidence of disease.

上皮样血管内皮瘤是一种罕见的内皮分化肿瘤,最常见于软组织、肝脏和肺部,临床病程多变。大多数病例以t(1;3)(p36;q23-25)为特征,导致WWTR1-CAMTA1融合。只有五个上皮样血管内皮瘤在唾液腺中被报道过。没有一例表现为贝尔麻痹症。在本病例中,一名37岁女性长期主诉右侧耳前区疼痛和充盈,进行性贝尔氏麻痹和面神经无力。手术切除显示肿瘤由非典型细胞组成,纤维黏液样间质中偶有胞浆内空泡。免疫组化染色显示肿瘤细胞表达ERG、CD31和CD34,证实血管分化。荧光原位杂交显示t(1;3)(p36;q25),确认诊断为上皮样血管内皮瘤。随访12个月,患者无发病迹象。
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引用次数: 0
Ectopic Fetal Liver Tissue in the Placenta of a Twin Pregnancy: A Case Report and Review of Literature. 双胎妊娠胎盘异位胎儿肝组织1例报告及文献复习。
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-06-13 eCollection Date: 2022-01-01 DOI: 10.1155/2022/1966025
Andrei Leucă, Pieter Demetter, Amélie Boulay, Katherina Vanden Houte, Valérie Segers, Laurine Verset

Ectopic liver tissue represents a rare entity and is mostly attributed to events occurring during embryogenesis. Previous case reports documented the presence of fetal liver parenchyma within temporarily developed organs during pregnancy, such as the placenta or the umbilical cord. Moreover, the terminology of these benign findings varies from "ectopic liver" to "hepatocellular adenoma-like neoplasm" or "hepatocellular adenoma". Ancillary tests performed on these lesions have shown positive immunohistochemical staining for hepatocellular origin marker HepPar-1. Only one recent case report comprising molecular analysis showed no beta-catenin gain-of-function mutation. We report a case of ectopic liver in one placenta of a twin pregnancy, with an updated review of literature.

异位肝组织是一种罕见的实体,主要归因于胚胎发生期间发生的事件。以前的病例报告表明,胎儿肝实质存在于妊娠期间临时发育的器官内,如胎盘或脐带。此外,这些良性发现的术语从“异位肝”到“肝细胞腺瘤样肿瘤”或“肝细胞腺瘤”不等。对这些病变进行的辅助试验显示肝细胞起源标志物HepPar-1免疫组化染色阳性。只有一个最近的病例报告包括分子分析显示没有β -连环蛋白功能获得突变。我们报告一例异位肝在双胎妊娠的一个胎盘,与最新的文献回顾。
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引用次数: 0
Multiple Consecutive Cervicovaginal Cytology Specimens Confirm Persistent Colonization by Cokeromyces recurvatus: Case Report and Literature Review 多个连续的宫颈阴道细胞学标本证实了复发角酵母的持续定植:病例报告和文献复习
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-05-13 DOI: 10.1155/2022/2151926
Keng Lor, C. Hartley, B. Pritt, A. M. Kemp, Amy A. Swanson, C. Sturgis
The published literature on cervicovaginal cytology includes fewer than ten reported cases of Cokeromyces recurvatus identified in Pap test samples. We report a unique case of an asymptomatic 27-year-old female with persistent gynecologic tract colonization by C. recurvatus in which distinctive fungal microorganisms were identified in three samples collected over three consecutive years.
在已发表的宫颈阴道细胞学文献中,巴氏试验样本中发现的复发性Cokeromyces不到10例。我们报告一个独特的情况下,无症状的27岁女性与持续的妇科生殖道定植由C.复发,其中独特的真菌微生物在三个样本中被鉴定连续三年收集。
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引用次数: 0
Intradural Extramedullary Concurrent Schwannoma and Meningothelial Hyperplasia at C2-C3 Cervical Vertebrae: A Case Report and Review of Literature C2-C3颈椎硬膜内髓外并发神经鞘瘤及脑膜上皮增生1例报告及文献复习
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-05-05 DOI: 10.1155/2022/1087918
R. Rammal, Daniel F Marker, Rana Naous
Concomitant schwannomas and benign meningothelial proliferations, including meningothelial hyperplasia or meningioma, rarely occur at the same location outside the setting of neurofibromatosis. Herein, we present a rare case of concurrent schwannoma and benign meningothelial hyperplasia concomitantly occurring in the cervical spine of a 69-year-old male patient with no history of any genetic disorder.
伴随的神经鞘瘤和良性脑膜上皮增生,包括脑膜上皮增生或脑膜瘤,很少发生在神经纤维瘤病以外的同一部位。在此,我们报告一例罕见的神经鞘瘤和良性脑膜上皮增生同时发生在颈椎的69岁男性患者,无任何遗传疾病史。
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引用次数: 1
A Rare Case of Embryonal Rhabdomyosarcoma of the Uterine Cervix 子宫颈胚胎性横纹肌肉瘤1例
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-04-13 DOI: 10.1155/2022/8459566
Vishal Bahall, Lance De Barry, S. Sankar
Embryonal rhabdomyosarcoma (RMS) of the uterine cervix is an exceedingly rare mesenchymal tumor that accounts for less than 1% of all cervical cancers. This highly malignant tumor primarily affects adolescents and young adults. Due to the paucity of publications on this clinical entity, there are no clearly established treatment protocols. However, a multimodal approach to treatment that involves surgical intervention combined with adjuvant chemoradiotherapy appears to improve patient outcomes. Herein, we report a case of embryonal rhabdomyosarcoma of the uterine cervix in a 24-year-old female, who presented with an exophytic cervical mass and vaginal bleeding. Histopathology and immunohistochemistry confirmed embryonal rhabdomyosarcoma of the uterine cervix with extension into the lower uterine segment. This patient was successfully managed with a combination of neoadjuvant chemoradiotherapy, a total abdominal hysterectomy with bilateral salpingo-oophorectomy, and adjuvant chemoradiotherapy.
子宫颈胚胎横纹肌肉瘤(RMS)是一种极为罕见的间充质肿瘤,占所有宫颈癌的不到1%。这种高度恶性肿瘤主要影响青少年和年轻人。由于缺乏关于这种临床实体的出版物,没有明确建立的治疗方案。然而,包括手术干预和辅助放化疗在内的多模式治疗方法似乎可以改善患者的预后。在此,我们报告一例24岁女性的子宫胚胎性横纹肌肉瘤,她表现为外生性宫颈肿块和阴道出血。组织病理学和免疫组织化学证实子宫子宫颈胚胎性横纹肌肉瘤,并延伸至子宫下段。该患者成功地接受了新辅助放化疗、全腹子宫切除术和双侧输卵管卵巢切除术以及辅助放化疗。
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引用次数: 5
INI-1-Deficient Sinonasal Carcinoma: Case Report with Emphasis on Differential Diagnosis i -1缺失型鼻窦癌1例报告并强调鉴别诊断
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-03-30 DOI: 10.1155/2022/5629984
Anwaar M. Alsayed, Eman Aljufairi, Amjad O. Alshammari, Khalid A Alsindi, Omar A Sabra
SMARCB1-deficient sinonasal carcinoma is a newly described entity, with less than 100 reported cases. It is characterized by basaloid or rhabdoid morphology and is diagnosed by complete loss of nuclear SMARCB1 (INI-1). The morphologic appearance, specific immunophenotypic markers, and unique molecular make-up distinguish this entity from other various malignant neoplasms. We present a case of a 55-year-old male that presented with a large progressing palatine mass. Magnetic resonance imaging showed a heterogeneous mass involving the left maxillary space. The initial biopsy was diagnosed as undifferentiated carcinoma. Resection was performed, and immunohistochemical studies revealed a complete loss of INI-1, refining the diagnosis to SMARCB1-deficient sinonasal carcinoma. Diagnosis of SMARCB1-deficient sinonasal carcinoma should be considered in all undifferentiated sinonasal carcinomas. Immunohistochemistry or molecular studies are mandatory to confirm the diagnosis and exclude other morphologically similar entities.
smarcb1缺失型鼻窦癌是一种新发现的肿瘤,报道病例不足100例。它的特征是基底样或横纹肌样形态,并通过核SMARCB1 (ni -1)的完全缺失来诊断。形态学外观、特异性免疫表型标记和独特的分子组成将该实体与其他各种恶性肿瘤区分开来。我们提出一个病例55岁的男性,提出了一个大进展腭肿块。磁共振显示一非均匀肿块累及左侧上颌间隙。最初的活检诊断为未分化癌。手术切除后,免疫组化研究显示i -1完全缺失,明确诊断为smarcb1缺失型鼻窦癌。在所有未分化的鼻窦癌中,都应考虑smarcb1缺陷鼻窦癌的诊断。免疫组织化学或分子研究是强制性的,以确认诊断和排除其他形态相似的实体。
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引用次数: 2
Deep Seated Lobular Capillary Hemangioma (Pyogenic Granuloma) of the Colon: A Rare Case Requiring Surgery beyond Endoscopic Management 结肠深层小叶毛细血管瘤(化脓性肉芽肿):一个罕见的病例需要手术超越内镜管理
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-03-29 DOI: 10.1155/2022/5641608
Jae-Youn Park, Min-Jae Jung
Background Lobular capillary hemangiomas typically present as skin or oral mucosa lesions and have rarely been described in unusual sites, including the gastrointestinal tract. Most colonic lobular capillary hemangiomas, either asymptomatic or associated with GI bleeding, have been amenable to endoscopic treatment in literatures. Case Presentation. A 41-year-old woman presented with an incidental colonic mass during a systemic workup after adjuvant chemotherapy for HER2-positive breast cancer. Abdominal computed tomography revealed a deep seated colonic mass in the splenic flexure. An endoscopic strip biopsy was attempted for differential diagnosis of this lesion, but uncontrolled bleeding occurred, and an emergency surgery was eventually performed. Microscopic examination showed lobular capillary hemangioma involving full thickness of the colonic wall with mucosal ulceration. Conclusions Colonic lobular capillary hemangioma is a benign vascular proliferation but is a candidate in differential diagnosis of benign or malignant tumors. Furthermore, the exceptional case may be deep seated and require more invasive surgery, unlike most cases of colonic lobular capillary hemangioma that can be treated with endoscopic modality.
小叶毛细血管瘤通常表现为皮肤或口腔粘膜病变,很少出现在不寻常的部位,包括胃肠道。大多数结肠小叶毛细血管瘤,要么无症状,要么伴有消化道出血,在文献中都可以进行内镜治疗。案例演示。一名41岁的女性在her2阳性乳腺癌辅助化疗后的全身检查中出现偶然的结肠肿块。腹部计算机断层扫描显示脾脏屈曲处有一深层结肠肿块。为了鉴别诊断该病变,我们尝试了内镜下切片活检,但发生了无法控制的出血,最终进行了紧急手术。显微镜检查显示小叶毛细血管瘤累及全层结肠壁,粘膜溃疡。结论结肠小叶毛细血管瘤是一种良性血管增生,是良恶性肿瘤鉴别诊断的候选者。此外,特殊的病例可能是深层的,需要更多的侵入性手术,不像大多数结肠小叶毛细血管瘤可以通过内窥镜治疗。
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引用次数: 1
Enterocolic Lymphocytic Phlebitis Treated Preoperatively with Biologics and Immunosuppressive Agents 术前应用生物制剂和免疫抑制剂治疗小肠结肠淋巴细胞性静脉炎
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-03-07 DOI: 10.1155/2022/5120607
Soh Okano, T. Yao, Osamu Nomura, A. Nagahara, Toshiaki Hagiwara, Kiichi Sugimoto, Makoto Takahashi, K. Sakamoto
Enterocolic lymphocytic phlebitis is phlebitis of unknown etiology in which lymphocytes affect veins without arteries and shows evidence of systemic vasculitis in the intestinal wall and mesentery, mainly in the small intestine and colon. Although patients present with a variety of gastrointestinal symptoms and findings like those of inflammatory bowel disease or ischemic bowel disease, there are no specific findings for enterocolic lymphocytic phlebitis. As a result, a diagnosis tends to be made after surgery. There are few case reports of enterocolic lymphocytic phlebitis, and the impact of chronic courses and immunosuppressive drugs on enterocolic lymphocytic phlebitis is not well known. A 47-year-old man was treated with infliximab and steroids for unexplained ulceration and narrowing of the ileocecal area, which was suspected to be inflammatory bowel disease with atypical findings. Lymphocytic phlebitis was noted in the surgical specimen, and enterocolic lymphocytic phlebitis was diagnosed. No recurrence of enterocolic lymphocytic phlebitis was observed postoperatively. This disease should also be considered among patients with inflammatory bowel disease-like lesions that do not respond to infliximab or steroids.
小肠结肠淋巴细胞性静脉炎是一种病因不明的静脉炎,其中淋巴细胞影响静脉而不影响动脉,表现为肠壁和肠系膜的系统性血管炎,主要发生在小肠和结肠。虽然患者表现出多种胃肠道症状和表现,如炎症性肠病或缺血性肠病,但小肠结肠淋巴细胞性静脉炎没有特异性的表现。因此,诊断往往是在手术后做出的。小肠结肠淋巴细胞性静脉炎的病例报道很少,慢性病程和免疫抑制药物对小肠结肠淋巴细胞性静脉炎的影响尚不清楚。一名47岁男性因不明原因的溃疡和回盲区狭窄而接受英夫利昔单抗和类固醇治疗,怀疑为炎症性肠病,表现不典型。手术标本中发现淋巴细胞性静脉炎,诊断为肠结肠淋巴细胞性静脉炎。术后未见小肠结肠淋巴细胞性静脉炎复发。对英夫利昔单抗或类固醇无效的炎症性肠病样病变患者也应考虑此病。
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引用次数: 0
Recurrent Oncocytoma of the Lacrimal Sac 泪囊复发性瘤
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-02-28 DOI: 10.1155/2022/2955030
Faris Almutairi, Mazen S. Alsamnan, Azza M. Y. Maktabi, S. Elkhamary, H. Alkatan, Humoud AlOtaibi
Oncocytoma of the lacrimal sac is an extremely rare tumor. In this report, we present the case of an 82-year-old woman who presented with swelling in the region of the lacrimal sac. Systemic examination and ophthalmic examination of both eyes were unremarkable. Computed tomography of the brain and orbits revealed a mass lesion involving the right lacrimal sac with expansion of the related nasolacrimal duct. Neither bone destruction nor tissue invasion was observed. Right external dacryocystectomy and debulking of the tumor were performed. Histopathological examination of the surgical specimen showed oncocytic cells arranged in an adenomatous fashion, and a diagnosis of benign oncocytoma was made. Three years later, the same patient presented with a similar complaint that was pathologically proven to be a recurrent benign oncocytoma of the lacrimal sac.
泪囊的嗜瘤细胞瘤是一种极为罕见的肿瘤。在这个报告中,我们提出的情况下,82岁的妇女谁提出了肿胀的泪囊区域。双眼全身检查及眼科检查无明显差异。脑部及眶部电脑断层显示一肿块病变累及右侧泪囊及相关鼻泪管扩张。没有观察到骨破坏或组织侵犯。行右侧外泪囊切除术及肿瘤减体积术。手术标本的组织病理学检查显示癌细胞呈腺瘤状排列,诊断为良性癌细胞瘤。三年后,同一患者提出了类似的主诉,病理证实为复发性泪囊良性癌细胞瘤。
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引用次数: 0
Concurrent Primary Follicular Lymphoma and a Mature Cystic Teratoma of the Ovary: A Case Report and Review of Literature 并发原发性滤泡性淋巴瘤和卵巢成熟囊性畸胎瘤1例报告及文献复习
IF 0.6 Q4 PATHOLOGY Pub Date : 2022-02-25 DOI: 10.1155/2022/5896696
S. Hutspardol, Y. Li, V. Dubé, J. Delabie
Primary lymphoma concurrent with teratoma of the ovary is exceedingly rare. Based on our review of the literature, there are only 8 case reports describing concurrent primary diffuse large B-cell lymphoma and teratoma. Here, we report the first case of primary follicular lymphoma concurrent with mature ovarian cystic teratoma, which, to our knowledge, has not been described in the literature.
原发性淋巴瘤并发卵巢畸胎瘤极为罕见。根据我们的文献回顾,只有8例报告描述原发性弥漫性大b细胞淋巴瘤和畸胎瘤并发。在这里,我们报告第一例原发性滤泡性淋巴瘤并发成熟卵巢囊性畸胎瘤,据我们所知,这在文献中尚未被描述。
{"title":"Concurrent Primary Follicular Lymphoma and a Mature Cystic Teratoma of the Ovary: A Case Report and Review of Literature","authors":"S. Hutspardol, Y. Li, V. Dubé, J. Delabie","doi":"10.1155/2022/5896696","DOIUrl":"https://doi.org/10.1155/2022/5896696","url":null,"abstract":"Primary lymphoma concurrent with teratoma of the ovary is exceedingly rare. Based on our review of the literature, there are only 8 case reports describing concurrent primary diffuse large B-cell lymphoma and teratoma. Here, we report the first case of primary follicular lymphoma concurrent with mature ovarian cystic teratoma, which, to our knowledge, has not been described in the literature.","PeriodicalId":45638,"journal":{"name":"Case Reports in Pathology","volume":"7 1","pages":""},"PeriodicalIF":0.6,"publicationDate":"2022-02-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"79116616","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: 1
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Case Reports in Pathology
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