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Robotic wedge resection of a rare gastric perivascular epithelioid cell tumor: A case report

World Journal of Clinical Cases 2019年第23期7卷 4011-4019页

作者： Alessandra Marano Francesca Maione Yanghee Woo Luca Pellegrino Paolo Geretto Diego Sasia Mirella Fortunato Giulio Fraternali Orcioni Roberto Priotto Renato Fasoli Felice Borghi Department of Surgery General and Oncologic Surgery UnitSanta Croce e Carle HospitalCuneo 12100Italy Department of Surgery City of HopeDuarteCA 91010United States Department of Pathology Santa Croce e Carle HospitalCuneo 12100Italy Department of Radiology Santa Croce e Carle HospitalCuneo 12100Italy Department of Gastroenterology and Digestive Endoscopy Santa Croce e Carle HospitalCuneo 12100Italy

BACKGROUND perivascular epithelioid cell tumor(PEComa)is a rare mesenchymal neoplasm that can arise in many different organs with a broad spectrum of biological behavior,from indolent to aggressive progression.Only ten cases of gastric PEComas have been reported in the English literature,which were treated with endoscopic,laparoscopic,or open resections.Due to its rarity,the optimal surgical management and prognosis of this tumor are still uncertain.CASE SUMMARY We present a case of robotic wedge resection of a 6.5 cm bleeding lesion of the gastric fundus located 3 cm below the esophago-gastric junction in a 55-year-old man.Biopsy revealed a malignant tumor with epithelioid cells focally positive for muscle markers desmin and smooth muscle actin.In addition,histology revealed that the tumor was positive for HMB-45,melan-A(MART-1),microphthalmia transcription factor and negative for pan-cytokeratin AE1/AE3,CD34,p40,DOG-1,CD117(c-kit),S100,CD3,CD79a,caldesmon and myogenin.These markers suggested the possibility of a PEComa.The patient underwent a diagnostic laparoscopy via the da Vinci■Si^TM system and robotic wedge resection.Final pathology confirmed a malignant gastric PEComa with negative margins.At his 11-mo follow-up visit,the patient remained disease-free.CONCLUSION Gastric PEComa can be treated with a robotic R0 resection with acceptable postoperative and short-term oncological outcomes.

关键词： perivascular epithelioid cell tumor Stomach Robotic Surgery Minimally invasive Case report

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Retroperitoneal perivascular epithelioid cell tumours: A case report and review of literature

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World Journal of Clinical Cases 2019年第21期7卷 3524-3534页

作者： Zisis Touloumis Niki Giannakou Christos Sioros Argiro Trigka Maria Cheilakea Nikoletta Dimitriou John Griniatsos Department of Surgery Athens Medical GroupAthens 11525Greece Department of Pathology Athens Medical GroupAthens 11525Greece Emergency Department Konstantopouleio General HospitalN.Ionia 14233Greece Department of Surgery National and Kapodistrian University of AthensAthens 11527Greece

BACKGROUND The perivascular epithelioid cell tumour(PEComa)family of tumours mainly includes renal and hepatic angiomyolipomas,pulmonary lymphangioleiomyomatosis and clear cell“sugar”tumour of the lung.Several uncommon tumours with similar morphological and immunophenotypical characteristics arising at a variety of sites(abdominal cavity,digestive tract,retroperitoneum,skin,soft tissue and bones)are also included in the PEComa family and are referred to as PEComas not otherwise specified.CASE SUMMARY We present a 37-year-old female patient who underwent resection of an 8.5 cm×8 cm×4 cm retroperitoneal tumour,which eventually was diagnosed as PEComa of uncertain biological behaviour.Three years after the operation,the patient remains without any evidence of recurrence.A search was performed in the Medline and EMBASE databases for articles published between 1996 and 2018,and we identified 31 articles related to retroperitoneal and perinephric PEComas.We focused on sex,age,maximum dimension,histological and immunohistochemical characteristics of the tumour,follow-up and long-term outcome.Thirty-four retroperitoneal(including the present one)and ten perinephric PEComas were identified,carrying a malignant potential rate of 44%and 60%,respectively.Nearly half of the potentially malignant PEComas presented with or developed metastases during the course of the disease.CONCLUSION Retroperitoneal PEComas are not as indolent as they are supposed to be.Radical surgical resection constitutes the treatment of choice for localized disease,while mammalian target of the rapamycin(mTOR)inhibitors constitute the most promising therapy for disseminated disease.The role of mTOR inhibitors as adjuvant or neoadjuvant therapies needs to be evaluated in the future.

关键词： perivascular epithelioid cell tumour Retroperitoneum Mammalian target of the rapamycin inhibitors Tuberous sclerosis complex Case report Treatment

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Percutaneous ablation for perivascular hepatocellular carcinoma: Refining the current status based on emerging evidence and future perspectives

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World Journal of Gastroenterology 2018年第47期24卷 5331-5337页

作者： Tae Wook Kang Hyo Keun Lim Dong Ik Cha Department of Radiology and Center for Imaging Science Samsung Medical Center Sungkyunkwan University School of Medicine Department of Health Sciences and Technology SAIHST Sungkyunkwan University

Various therapeutic modalities including radiofrequency ablation, cryoablation, microwave ablation, and irreversible electroporation have attracted attention as energy sources for effective locoregional treatment of hepatocellular carcinoma(HCC); these are accepted non-surgical treatments that provide excellent local tumor control and favorable survival. However, in contrast to surgery, tumor location is a crucial factor in the outcomes of locoregional treatment because such treatment is mainly performed using a percutaneous approach for minimal invasiveness; accordingly, it has a limited range of ablation volume. When the index tumor is near large blood vessels, the blood flow drags thermal energy away from the targeted tissue, resulting in reduced ablation volume through a socalled "heat-sink effect". This modifies the size and shape of the ablation zone considerably. In addition, serious complications including infarction or aggressive tumor recurrence can be observed during follow-up after ablation for perivascular tumors by mechanical or thermal damage. Therefore, perivascular locations of HCC adjacent to large intrahepatic vessels can affect post-treatment outcomes. In this review, we primarily focus on physical properties of perivascular tumor location, characteristics of perivascular HCC,potential complications, and clinical outcomes after various locoregional treatments; moreover, we discuss the current status and future perspectives regarding percutaneous ablation for perivascular HCC.

关键词： Hepatocellular carcinoma perivascular Radiofrequency ablation Liver Cryoablation Microwave ablation Irreversible electroporation

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Glymphatic imaging and modulation of the optic nerve

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Neural Regeneration Research 2022年第5期17卷 937-947页

作者： Anisha Kasi Crystal Liu Muneeb A.Faiq Kevin C.Chan Department of Ophthalmology NYU Grossman School of MedicineNYU Langone HealthNew York UniversityNew YorkNYUSA Department of Radiology NYU Grossman School of MedicineNYU Langone HealthNew York UniversityNew YorkNYUSA Neuroscience Institute NYU Grossman School of MedicineNYU Langone HealthNew York UniversityNew YorkNYUSA Center for Neural Science College of Arts and ScienceNew York UniversityNew YorkNYUSA

Optic nerve health is essential for proper function of the visual system.However,the pathophysiology of certain neurodegenerative disease processes affecting the optic nerve,such as glaucoma,is not fully understood.Recently,it was hypothesized that a lack of proper clearance of neurotoxins contributes to neurodegenerative diseases.The ability to clear metabolic waste is essential for tissue homeostasis in mammals,including humans.While the brain lacks the traditional lymphatic drainage system identified in other anatomical regions,there is growing evidence of a glymphatic system in the central nervous system,which structurally includes the optic nerve.Named to acknowledge the supportive role of astroglial cells,this perivascular fluid drainage system is essential to remove toxic metabolites from the central nervous system.Herein,we review existing literature describing the physiology and dysfunction of the glymphatic system specifically as it relates to the optic nerve.We summarize key imaging studies demonstrating the existence of a glymphatic system in the optic nerves of wild-type rodents,aquaporin 4-null rodents,and humans;glymphatic imaging studies in diseases where the optic nerve is impaired;and current evidence regarding pharmacological and lifestyle interventions that may help promote glymphatic function to improve optic nerve health.We conclude by highlighting future research directions that could be applied to improve imaging detection and guide therapeutic interventions for diseases affecting the optic nerve.

关键词： aquaporin-4 cerebrospinal fluid glaucoma glymphatic system hydrocephalus imaging metabolic waste clearance optic nerve perivascular pharmacological and lifestyle interventions

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perivascular epithelioid cell neoplasm of the colon

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World Journal of Gastrointestinal Oncology 2010年第4期2卷 205-208页

作者： Hugh James Freeman Doug L Webber Department of Medicine(Gastroenterology) University of British ColumbiaVancouver BC V6T 1W5Canada Department of Pathology University of British ColumbiaVancouver BC V6T 1W5Canada

A 17-year-old female presented with rectal bleeding from an ulcerated sigmoid mass in 1994.Initial pathological evaluation revealed a rare clear cell neoplasm of the colon,possibly originating from kidneys,adrenals,lung or a gynecologic source as a metastatic lesion.Extensive imaging studies were negative,and over the next 15 years,she remained well with no recurrence.The original resected neoplasm was reviewed and reclassified as a perivascular epithelioid cell neoplasm (PEComa).Although the long-term natural history of PEComas requires definition,increased clinical and pathological awareness should lead to increased recognition of an apparently rare type of colonic neoplasm that likely occurs more often than is currently appreciated.

关键词： perivascular epithelioid cell neoplasm Carci noid tumor Colonic adenocarcinoma Clear cell tumor

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Cutaneous perivascular epithelioid cell tumors:A review on an infrequent neoplasm

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World Journal of Methodology 2016年第1期6卷 87-92页

作者： Mar Llamas-Velasco Luis Requena Thomas Mentzel Department of Dermatology Hospital Universitario de La Princesa Department of Dermatology Fundación Jiménez Díaz Department of Dermatopathology Dermatohis- topathologisches Gemeinschaftslabor

"perivascular epithelioid cutaneous" cell tumors(PEComa) are a family of mesenchymal tumors with shared microscopic and immunohistochemical properties: They exhibit both smooth muscle cell and melanocytic differentiation. Non-neoplastic counterpart of PEComa's cells are unknown, as well as the relationship between extracutaneous PEComa and primary cutaneous ones. We will review the clinical setting, histopathologic features, chromosomal abnormalities, differential diagnosis and treatment options for cutaneous PEComa.

关键词： perivascular epithelioid cell tumor Skin Cutaneous perivascular epithelioid cell tumors Clear cell myomelanocytic tumor

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Coincidence between malignant perivascular epithelioid cell tumor arising in the gastric serosa and lung adenocarcinoma

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World Journal of Gastroenterology 2015年第4期21卷 1349-1356页

作者： Sohsuke Yamada Atsunori Nabeshima Hirotsugu Noguchi Aya Nawata Hisae Nishii Xin Guo Ke-Yong Wang Masanori Hisaoka Toshiyuki Nakayama Department of Pathology and Cell Biology School of MedicineUniversity of Occupational and Environmental HealthKitakyushu 807-8555Japan Department of Urology School of MedicineUniversity of Occupational and Environmental HealthKitakyushu807-8555Japan Bio-information Research Center School of MedicineUniversity of Occupational and Environmental HealthKitakyushu 807-8555Japan Department of Pathology and Oncology School of MedicineUniversity of Occupational and Environ-mental HealthKitakyushu 807-8555Japan

A 4-mo history of both epigastralgia and back pain was presented in a 39-year-old male. Computed tomography showed right lung nodule and abdominal mass attached to the gastric wall, measuring approximately 30 mm and 70 mm in diameter. Since biopsy samples from the lung and abdomen revealed poorly differentiated adenocarcinoma and malignant tumor, clinicians first interpreted the abdominal mass as metastatic carcinoma, and a right lower lobectomy with following resection of the mass was performed. Gross examination of both lesions displayed gray-whitish to yellow-whitish cut surfaces with hemorrhagic and necrotic foci, and the mass attached to the serosa of the lesser curvature on the gastric body. On microscopic examination, the lung tumor was composed of a proliferation of highly atypical epithelial cells having abundant eosinophilic cytoplasm, predominantly arranged in an acinar or solid growth pattern with vessel permeation, while the abdominal tumor consisted of sheets or nests with markedly atypical epithelioid cells having pleomorphic nuclei and abundant eosinophilic to clear cytoplasm focally in a radial perivascular or infiltrative growth pattern. Immunohistochemically, the latter cells were positive for HMB45 or α-smooth muscle actin, but the former ones not. Therefore, we finally made a diagnosis of malignant perivascular epithelioid cell tumor(PEComa) arising in the gastric serosa, combined with primary lung adenocarcinoma. Furthermore, small papillary car-cinoma of the thyroid gland was identified. The current case describes the coincidence of malignant PEComa with other carcinomas, posing a challenge in distinction from metastatic tumor disease.

关键词： perivascular epithelioid cell tumor Malignant Gast

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Improving recognition of hepatic perivascular epithelioid cell tumor:Case report and literature review

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World Journal of Gastroenterology 2015年第17期21卷 5432-5441页

作者： Toshiya Maebayashi Katsumi Abe Takuya Aizawa Masakuni Sakaguchi Naoya Ishibashi Osamu Abe Tadatoshi Takayama Hisashi Nakayama Shunichi Matsuoka Kazushige Nirei Hitomi Nakamura Masahiro Ogawa Masahiko Sugitani Department of Radiology Nihon University School of MedicineItabashi-kuTokyo 173-8610Japan Department of Digestive Surgery Nihon University School of MedicineItabashi-kuTokyo 173-8610Japan Department of Gastroenterology and Hepatology Nihon University School of MedicineItabashi-kuTokyo 173-8610Japan Department of Pathology Nihon University School of MedicineItabashi-kuTokyo 173-8610Japan

A 58-year-old man presented with the chief complaint of abdominal bloating and was incidentally found to have a liver tumor.As diagnostic imaging studies could not rule out malignancy,the patient underwent partial resection of segment 3 of the liver.The lesion pathologically showed eosinophilic proliferation,in addition to immunohistochemical positivity for human melanoma black 45 and Melan-A,thereby leading to the diagnosis of a hepatic perivascular epithelioid cell tumor(PEComa).A PEComa arising from the liver is relatively rare.Moreover,the name ‘PEComa' has not yet been widely recognized,and the same disease entity has been called epithelioid angiomyolipoma(EAML),further diminishing the recognition of PEComa.In addition,PEComa imaging findings mimic those of malignant liver tumors,and clinically,this tumor tends to enlarge.Therefore,a PEComa is difficult to diagnose.We conducted a systematic review of PEComa and EAML cases and discuss the results,including findings useful for differentiating perivascular epithelioid cell tumors from malignant liver tumors.

关键词： Angiomyolipoma Tuberous sclerosis Melan-A perivascular epithelioid cell tumor Humanmelanoma black 45 Imaging

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Pancreatic perivascular epithelioid cell tumor: A case report with clinicopathological features and a literature review

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World Journal of Gastroenterology 2016年第13期22卷 3693-3700页

作者： Hui Jiang Na Ta Xiao-Yi Huang Ming-Hua Zhang Jing-Jing Xu Kai-Lian Zheng Gang Jin Jian-Ming Zheng Department of Pathology Changhai Hospital Second Military Medical University Department of General Surgery Changhai Hospital Second Military Medical University

perivascular epithelioid cell tumor(PEComa) of the pancreas is an unusual tumor deriving from mesenchyma. This paper described a case of pancreatic PEComa, which was initially suspected as neuroendocrine carcinoma by biopsy, and therefore surgical treatment was recommended due to undetermined diagnosis. Examination of the surgical specimen under a microscope showed that the tumor cell's morphology was epithelioid or spindle-shaped, and ranged in a nested pattern. Additionally, these cells had a large extent of acidophilic cytoplasm, no mitotic figures, and expressed HMB-45, melan-p, and smooth muscle actin immunohistochemically. Pathological examination indicated that PEComa originated from the pancreas, but symptoms related to tuberous sclerosis were absent. Since PEComa is extremely rare in the pancreas, it is likely to be ignored in differential diagnosis. In conclusion, our article highlighted the clinicopathological features of PEComa, and we conducted a literature review focusing on PEComa so as to deepen the understanding of this tumor type.

关键词： Pancreas perivascular epithelioid cell tumor HMB-45 Immunohistochemistry Clinicopathological feature

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Effect of Peroxynitrite (ONOO<sup>-</sup>) on the Function of Murine perivascular Adipose Tissue

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Pharmacology & Pharmacy 2019年第4期10卷 194-204页

作者： Azizah Binti Ugusman Alex Riddell Simon Kennedy Institute of Cardiovascular and Medical Sciences University of Glasgow Scotland UK Department of Physiology Faculty of Medicine National University of Malaysia Medical Centre Kuala Lumpur Malaysia

perivascular adipose tissue (PVAT) surrounds the exterior of blood vessels and releases numerous substances such as adiponectin which positively modulate blood vessel tone. In some cardiovascular diseases such as diabetes and high blood pressure, the function of PVAT changes and we speculate that oxidant stress may play a role in this change. PVAT has the ability to generate both superoxide and nitric oxide and these can combine rapidly under physiological conditions to form peroxynitrite (ONOO-). In disease states, the production of ONOO- may be increased and so its effect on the function of PVAT is of great interest. Consequently, we studied the effects of acute addition of the oxidant species ONOO- on vascular tone and production of adiponectin by mouse thoracic aortic PVAT. Murine PVAT was immunostained for nitrotyrosine, indicating that ONOO- is formed in the PVAT. Exogenous ONOO- significantly increased the anticontractile effect of PVAT via increased adiponectin content but had no effect on eNOS expression or phosphorylation. These results suggest that generation of ONOO- within PVAT may be an important regulatory mechanism which influences the activity of PVAT. The effect of chronic exposure to raised levels of ONOO- on PVAT function remains to be determined.

关键词： perivascular Adipose Tissue (PVAT) Peroxynitrite Nitric Oxide Adiponectin Anticontractile

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