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首页> 外文期刊>Gynecologic Oncology Reports >Poorly differentiated high-grade urothelial carcinoma presenting as Paget's disease of the vulva with no overt urinary tract neoplasm detected
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Poorly differentiated high-grade urothelial carcinoma presenting as Paget's disease of the vulva with no overt urinary tract neoplasm detected

机译：低分化高级别尿路上皮癌，表现为外阴Paget病，未发现明显的尿路肿瘤

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Objective There are few reported cases of secondary (non-cutaneous) vulvar Paget's disease related to urothelial carcinoma (UC), with only 7 of them presenting initially with Paget's disease and up to a 13-year lapse from detecting a urinary tract neoplasm after the onset of symptoms. This is a case of Paget's disease of urothelial origin with no urinary tract neoplasm detected on initial presentation. Methods This is a 59-year-old African-American female who presented with worsening eczematous lesions for two years. She had no symptoms suggestive of UC. Initial biopsies showed poorly differentiated high-grade UC with pagetoid changes. Results Stains showed immunoreactivity for CK7, uroplakin III, p16 and p63 with negative CK20 expression. This staining pattern is characteristic of pagetoid urothelial intraepithelial lesion, now listed as secondary Paget's disease. Biopsies showed GATTA3 positivity suggestive of urothelial origin. Both GCDFP-15 and CEA were negative, which are normally expressed by Paget cells of the primary (cutaneous) type. A follow-up cystoscopy was unremarkable. The patient underwent a partial radical vulvectomy with bilateral lymphadenectomy for extensive disease. Final pathology confirmed infiltrating high-grade UC with overlying epidermis displaying pagetoid in-situ tumor component. Conclusion This is a rare case of secondary Paget's disease of urothelial origin where there was no concurrent UC nor did the patient present with symptoms suggestive of a urinary tract malignancy. In initial presentations of vulvar Paget's disease, it is important to be aware of the secondary classification because it warrants investigation of surrounding structures to rule out underlying malignancies that are or have not yet become clinically apparent. Highlights ? Rare case of secondary (non-cutaneous) vulvar Paget's disease of urothelial origin ? No concurrent urothelial malignancy detected at time of diagnosis ? Can have up to 14-year lapse between onset of Paget's disease to detection of urothelial carcinoma ? Warrants investigation of surrounding structures to rule out concurrent malignancy ? Closer surveillance may be required with routine cystoscopies.
机译：目的报道的继发性（非皮肤性）外阴Paget病与尿路上皮癌（UC）有关的报道很少，只有7例最初患有Paget病，并且从发现尿路肿瘤后到13年为止。症状发作。这是一例起源于尿路上皮的佩吉特氏病，初诊时未发现尿路肿瘤。方法这是一位59岁的非洲裔美国女性，她的湿疹病情恶化了两年。她没有暗示UC的症状。最初的活检显示分化较差的高级别UC，伴有页面状变。结果污渍对CK7，uroplakin III，p16和p63具有免疫反应性，而CK20表达阴性。这种染色模式是页面性尿路上皮上皮内病变的特征，现在被列为继发性Paget病。活检显示GATTA3阳性提示尿路上皮起源。 GCDFP-15和CEA均为阴性，通常由原发性（皮肤）类型的Paget细胞表达。膀胱镜检查无明显变化。患者因广泛疾病接受了部分根治性外阴切除术和双侧淋巴结切除术。最终病理证实浸润性高级UC并有覆盖表皮的原位肿瘤成分。结论这是尿道起源的继发性Paget病的罕见病例，既没有并发UC，也没有患者出现提示尿路恶性的症状。在外阴Paget病的最初表现中，重要的是要注意二级分类，因为它需要对周围结构进行研究，以排除潜在的临床上已经或尚未变得明显的恶性肿瘤。强调 ？尿路上皮继发性（非皮肤）外阴Paget病罕见病例？诊断时未同时发现尿路上皮恶性肿瘤吗？从佩吉特氏病发作到检测尿路上皮癌之间是否可以长达14年？对周围结构进行权证调查以排除并发恶性肿瘤？常规膀胱镜检查可能需要更严格的监测。

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