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      Two cases of Paget’s disease of scrotum in biological brothers

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          Abstract

          This article reports two cases of scrotum Paget’s disease in two biological brothers who were admitted and treated in our hospital in 2013. They are very rare cases. The present article discusses the potential management of Paget’s disease and the importance of long-term follow-up.

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          Most cited references 20

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          Paget's disease of the vulva: Diagnosis and follow-up key to management; a retrospective study of 50 cases from Queensland.

          To review the clinical features, diagnosis, management, and outcomes for the 50 cases of Paget's disease (PD) of the vulva referred to Queensland Centre for Gynaecological Cancer between 1986 and 2009. Vulvar PD cases from QCGC were reviewed and analyzed using the computer software Statistical Package for the Social Sciences (SPSS) 11.0. Paget's disease (PD) of the vulva is uncommon. Of the 50 patients, 2 have died of their PD, 1 patient that had coexisting PD died of squamous cell vulva cancer, and 11 died of unrelated causes. The mean age at diagnosis was 67.6 years (range, 31 to 91). All cases were Caucasian. Time from onset of symptoms to diagnosis averaged 21 months. Not until a biopsy was performed was the diagnosis made. The most common presenting complaint was pruritis (27 cases, 54%). There was no identifiable "favored" site on the vulva for PD. Positive groin lymph nodes were found in 4 of the 10 cases who underwent node biopsy. Two who had poorly differentiated carcinoma in the nodes and PD died of disease within a year of diagnosis, one is alive three years later. The fourth case had coincidental PD and vulvar squamous cell carcinoma with squamous carcinoma groin nodes. Initial treatment was surgical. The prognosis for primary extra-mammary PD of the vulva confined to the epidermis (IEP) is excellent. Early diagnosis and long term follow-up are the keys to successful management. The status of disease at the margins of surgical specimens does not reliably equate to patient long term outcomes. Copyright © 2011 Elsevier Inc. All rights reserved.
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            Extramammary Paget's disease of the penis and scrotum: excision, reconstruction and evaluation of occult malignancy.

            We describe treatment and reconstruction in patients after surgery for extramammary Paget's disease of the penis and scrotum. We also investigated whether this disease causes an increased risk of undiagnosed visceral malignancy. We reviewed the databases at our institution from 1996 to 2000 and identified 6 men 67 to 87 years old (mean age 76). In addition, we reviewed the literature on the clinical and pathological features of this disease. In our 6 patients scrotal involvement was present in 83% and penile extramammary Paget's disease was present in 33%. Each man underwent wide local excision and large skin defects were immediately reconstructed with split-thickness skin grafts. In 1 case extramammary Paget's disease had spread to the superficial inguinal nodes. At a mean followup of 29 months there has been no local recurrence and internal malignancy has not been diagnosed. Our literature review revealed 13 patients with penoscrotal extramammary Paget's disease and visceral malignancy, including 12 (92%) with malignancy of the genitourinary system. Extramammary Paget's disease of the penis and scrotum is a rare disease that can be managed by excision and immediate reconstruction with skin grafting or a local skin flap. Disease may spread to the regional lymph nodes. Although genitourinary cancer may accompany penoscrotal extramammary Paget's disease, an extensive search for cancer of the thorax or abdomen may be unnecessary because only 1 reported case of colon cancer has been associated with penile or scrotal extramammary Paget's disease.
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              Extramammary Paget's disease of scrotum--report of 25 cases and literature review.

              We evaluate the clinical manifestations, management, and prognostic characteristics of scrotal extramammary Paget's disease (EMPD). The study comprised 25 patients with scrotal EMPD at our institute from January 1982 to February 2005, with all available clinical and pathological data reviewed. Of these 25 patients, 1 received radiotherapy and 24 received local wide excisions. In 24 operated patients, 7 had local recurrence and/or metastasis of groin lymph node. Five of the 7 with recurrence had a positive surgical margin postoperatively and they received a second local extensive excision. One of the 7 with recurrence and metastasis of the groin lymph node had a second local extensive excision with groin lymphadenectomy, and the last one who only had metastasis of the groin lymph node had a groin lymphadenectomy. Four of 13 patients with dermal invasion by Paget's cell had metastasis. None of the other 12 patients without dermal invasion had metastasis. However, there was no statistical metastasis rate difference (P = 0.096) between the patients with dermal invasion by Paget's cell and without. There was no statistical difference (P = 0.947) in mean delay time from onset of symptoms to diagnosis between the 2 groups either. The follow-up duration varies from 17 to 243 months (mean 119 + 86.2 months). One patient with stage D died of EMPD of the scrotum. We found that EMPD of the scrotum is usually a slow progressive disease, mainly seen in elderly patients, and has a good prognosis when there is noninvasive disease. The primary treatment for EMPD of the scrotum is wide surgical excision. The key to decreasing tumor recurrence, however, is a precise, preoperative histological examination to define the range of the lesion. Copyright 2010 Elsevier Inc. All rights reserved.
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                Author and article information

                Journal
                Ther Clin Risk Manag
                Ther Clin Risk Manag
                Therapeutics and Clinical Risk Management
                Therapeutics and Clinical Risk Management
                Dove Medical Press
                1176-6336
                1178-203X
                2015
                27 May 2015
                : 11
                : 885-887
                Affiliations
                [1 ]Department of Urology, First affiliated Hospital, Medical College, Zhejiang University, Hangzhou, People’s Republic of China
                [2 ]Department of Urology, second affiliated Hospital of Wenzhou Medical University, Wenzhou, People’s Republic of China
                Author notes
                Correspondence: Liping Xie, Department of Urology, First Affiliated Hospital of Zhejiang University, Hangzhou 310003, People’s Republic of China, Email xielp@ 123456zju.edu.cn
                Article
                tcrm-11-885
                10.2147/TCRM.S80431
                4455842
                26064056
                © 2015 Rao et al. This work is published by Dove Medical Press Limited, and licensed under Creative Commons Attribution – Non Commercial (unported, v3.0) License

                The full terms of the License are available at http://creativecommons.org/licenses/by-nc/3.0/. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed.

                Categories
                Case Series

                Medicine

                metastasis, paget’s disease, case report, follow-up, dissection

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