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[睾丸鞘膜高分化乳头状间皮瘤:一例报告]

[Well-differentiated papillary mesothelial tumour of the tunica vaginalis: A case report].

作者信息

Fang Yangyi, Li Qiang, Huang Zhigao, Lu Min, Hong Kai, Zhang Shudong

机构信息

Department of Urology, Peking University Third Hospital, Beijing 100191, China.

Department of Urology, Beijing Zhongguancun Hospital, Beijing 100190, China.

出版信息

Beijing Da Xue Xue Bao Yi Xue Ban. 2024 Aug 18;56(4):741-744. doi: 10.19723/j.issn.1671-167X.2024.04.031.

DOI:10.19723/j.issn.1671-167X.2024.04.031
PMID:39041574
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11284478/
Abstract

The mesothelium, which consists of a monolayer of mesothelial cells, extends over the surface of the serosal cavities (pleura, pericardium, peritoneum and tunica vaginalis). Mesothelial tumours of the tunica vaginalis is rare compared with those arise from pleura or peritoneum. According to World Health Organization 2022 Classification of Urinary and Male Genital Tumours (5th edition), mesothelial tumours of the tunica vaginalis were categorized into adenomatoid tumour, well-differentiated papillary mesothelial tumour (WDPMT) and mesothelioma. Since WDPMT of tunica vaginalis was rare, there was no consensus concerning the treatment of it. In this case report, a 29-year-old man who had endured intermittent right scrotal pain for 8 months, aggravating scrotal pain for 2 weeks was admitted. No symptoms, such as frequent, urgent, or painful urination were shown. Physical examination revealed the enlargement and tenderness of right scrotum, with no signs of lifting pain. The most recent scrotal ultrasonography before surgery revealed right hydrocele with maximum depth of 4 centimeters and poor blood flow of right testis. Under the circumstance of patient' s chronic history of testicular hydrocele, he underwent an emergency operation of right scrotal exploration and hydrocelectomy under epidural anesthesia. After opening the vagina tunic cavity, spot-like bleeding was observed on the right testicle, epididymis and vaginalis surface. The vaginalis was obviously thickened and the inner and outer walls were smooth. The post-operative histopathology revealed a grayish-brown tissue with a thickness of 0.3-0.5 cm, smooth inner and outer walls, and a suspected WDPMT with a diameter of 1. 5 cm. Immunohistochemical staining showed positive for Calretinin, BAP1, WT-1, CK5/6, D2-40 and P16,which confirmed the diagnosis of WDPMT. To sum up, the purpose of this case report was to raise awareness of a rare disease WDPMT, which was usually asymptomatic and could be diagnosed by pathology and immunohistochemistry. The disease should be differentiated from testicular torsion, epididymitis, orchitis and oblique inguinal hernia in symptoms, and from malignant mesothelioma and adenomatoid tumour in pathology. Because of the rarity of the cases, there was no unified standard for the treatment of WDPMT at present. The common treatment methods reported in literature included orchidectomy and vaginectomy. Due to the lack of understanding of this disease, postoperative follow-up was still recommended for at least 5 years.

摘要

间皮由单层间皮细胞组成,覆盖在浆膜腔(胸膜、心包膜、腹膜和鞘膜)表面。与起源于胸膜或腹膜的间皮肿瘤相比,鞘膜的间皮肿瘤较为罕见。根据世界卫生组织《2022年泌尿系统及男性生殖器官肿瘤分类》(第5版),鞘膜的间皮肿瘤分为腺瘤样瘤、高分化乳头状间皮肿瘤(WDPMT)和间皮瘤。由于鞘膜的WDPMT较为罕见,对于其治疗尚无共识。在本病例报告中,一名29岁男性因间歇性右侧阴囊疼痛8个月、加重2周入院。未出现尿频、尿急或尿痛等症状。体格检查发现右侧阴囊增大且有压痛,无提睾痛体征。术前最新的阴囊超声检查显示右侧鞘膜积液,最大深度为4厘米,右侧睾丸血流不佳。在患者有睾丸鞘膜积液慢性病史的情况下,他在硬膜外麻醉下接受了右侧阴囊探查及鞘膜切除术的急诊手术。打开鞘膜腔后,在右侧睾丸、附睾及鞘膜表面观察到点状出血。鞘膜明显增厚,内外壁光滑。术后组织病理学显示为一个灰褐色组织,厚度为0.3 - 0.5厘米,内外壁光滑,有一个直径为1.5厘米的疑似WDPMT。免疫组化染色显示钙视网膜蛋白、BAP1、WT - 1、CK5/6、D2 - 40和P16呈阳性,确诊为WDPMT。综上所述,本病例报告的目的是提高对罕见疾病WDPMT的认识,该疾病通常无症状,可通过病理学和免疫组化进行诊断。该疾病在症状上应与睾丸扭转、附睾炎、睾丸炎和腹股沟斜疝相鉴别,在病理上应与恶性间皮瘤和腺瘤样瘤相鉴别。由于病例罕见,目前WDPMT的治疗尚无统一标准。文献报道的常见治疗方法包括睾丸切除术和鞘膜切除术。由于对该疾病认识不足,仍建议术后至少随访5年。

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本文引用的文献

1
Epididymal Adenomatoid Tumour: A Case Report.附睾腺瘤样瘤:一例报告
Cureus. 2023 Oct 23;15(10):e47505. doi: 10.7759/cureus.47505. eCollection 2023 Oct.
2
Well-differentiated Papillary Mesothelial Tumour of the Tunica Vaginalis Testis - A Rare Lesion, but one Pathologists Should Know About Two Patient Reports and a Review of the Literature.睾丸鞘膜的高分化乳头状间皮瘤——一种罕见病变,但病理科医生应该了解:两例患者报告及文献复习。
Int J Surg Pathol. 2023 Sep;31(6):1126-1131. doi: 10.1177/10668969221129888. Epub 2023 Jan 9.
3
The 2022 World Health Organization Classification of Tumours of the Urinary System and Male Genital Organs-Part A: Renal, Penile, and Testicular Tumours.2022 年世界卫生组织泌尿系统和男性生殖器官肿瘤分类 - 第 A 部分:肾脏、阴茎和睾丸肿瘤。
Eur Urol. 2022 Nov;82(5):458-468. doi: 10.1016/j.eururo.2022.06.016. Epub 2022 Jul 16.
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Testicular compartment syndrome: An unexpected diagnosis in a case of testicular pain.睾丸间隔综合征:一例睾丸疼痛病例中的意外诊断
Urol Case Rep. 2021 Aug 23;39:101823. doi: 10.1016/j.eucr.2021.101823. eCollection 2021 Nov.
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Clinicopathological characteristics and outcomes in men with mesothelioma of the tunica vaginalis testis: analysis of published case-series data.睾丸鞘膜间皮瘤患者的临床病理特征和结局:已发表病例系列数据的分析。
J Cancer Res Clin Oncol. 2021 Sep;147(9):2671-2679. doi: 10.1007/s00432-021-03533-6. Epub 2021 Feb 9.
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Hum Pathol. 2019 Oct;92:48-58. doi: 10.1016/j.humpath.2019.07.009. Epub 2019 Aug 1.
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Well-differentiated papillary mesothelioma of tunica vaginalis testis of unknown malignant potential: Sonographic appearance.睾丸鞘膜未知恶性潜能的高分化乳头状间皮瘤:超声表现
J Clin Ultrasound. 2018 Jun;46(5):364-367. doi: 10.1002/jcu.22538. Epub 2017 Oct 9.
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Well-Differentiated Papillary Mesothelioma of the Tunica Vaginalis: Case Report and Systematic Review of Literature.睾丸鞘膜高分化乳头状间皮瘤:病例报告及文献系统综述
Clin Genitourin Cancer. 2016 Aug;14(4):e435-9. doi: 10.1016/j.clgc.2016.03.007. Epub 2016 Mar 12.
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Scrotal mesothelioma.阴囊间皮瘤
Can J Urol. 2014 Feb;21(1):7163-5.
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Mod Pathol. 2010 Aug;23(8):1165-72. doi: 10.1038/modpathol.2010.113. Epub 2010 Jun 4.