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      A Systematic Review and Meta-Analysis of Mapping Biopsy for Primary Extramammary Paget’s Disease in Reducing Recurrence Following Surgical Excision

      research-article
      * , , , MD * , , , MD * , , , PhD , , MSc * , , § , , PhD * , , § , , PhD, MD * , , § , , MBBS, MRCP, MMed , , MBBS, MRCP, MMED , , MBBS, MRCP, MMed, FAMS, PhD , , , MB BChir, PhD, MRCP, FAMS , # , , MBBS, MRCP, MMED , ** , , MBBS, MRCP, MMed , ** , , MBBS, FRCR †† , , MBBS, FRCR ** , †† , , MBBS, FRCR, FAMS ** , ‡‡ , , MBBS, FRCR, FAMS ‡‡ , , MBBS, FRCR, MMed, MCI ‡‡ , , BMEDSc, MBBS, FRCPath, FRCAPA §§ , ‖‖ , , MBBS, PGDCP, FRCPath, MCS, FAMS §§ , , MBBS, MRCS, MMed, MCI, FAMS ¶¶ , , MB, BCh, BAO, LRCP&SI, MSurg * , , , MBBS, MMed, FRCS * , , ** , ## , , MB, BCh, BAO, MMed, FRCS * , , ** , , MBBS, MD, FRACS, FACS * , , , MBBS, MMed, FRCS * , , ** , ## , , MBBS, MMed, FRCS, PhD * , , § , # , ** , ## ,
      Annals of Surgery Open
      Wolters Kluwer Health, Inc.
      extra-mammary paget's disease, mapping biopsy, recurrence

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          Abstract

          Objective:

          To examine the association between the performance of mapping biopsies and surgical outcomes postexcision of extramammary Paget’s disease (EMPD).

          Background:

          Primary EMPD is a rare entity associated with poorly defined surgical margins and difficult-to-access sites of lesions. Surgical resection with clear margins remains the preferred management method. The use of mapping biopsies might be beneficial, particularly in lowering disease recurrence.

          Methods:

          Available literature was reviewed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses methodology before a fixed-effect meta-analysis was performed to identify the presence of a correlation between performing mapping biopsies and positive margins on permanent sections as well as disease-free survival. Additional study results not included in the quantitative assessment were qualitatively assessed and reported.

          Results:

          A total of 12 studies were shortlisted for final analysis. 294 patients who underwent mapping biopsies and 48 patients who did not undergo mapping biopsies were included in the assessment. Forest plot analysis revealed a pooled rate ratio of 0.50 (95% CI, 0.32–0.77) in the prevalence of positive margins in patients with mapping biopsies performed as compared to patients without. The pooled rate ratio of the prevalence of disease-free survival in patients with mapping biopsies performed as compared to patients without was 1.38 (95% CI, 1.03–1.84). Qualitative assessment of the remaining selected studies revealed equivocal results.

          Conclusions:

          Mapping biopsies are able to improve EMPD surgical excision outcomes but given the rarity of the disease and heterogeneity of mapping biopsy procedures, further confirmation with randomized controlled trials or a larger patient pool is necessary.

          Abstract

          Mini abstract

          In this systematic review and meta-analysis of 12 studies containing 342 patients who underwent and did not undergo mapping biopsies before surgical excision for primary extramammary Paget’s disease, we found that mapping biopsies are able to improve postexcision outcomes, taking certain caveats into consideration.

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          Most cited references27

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          The PRISMA 2020 statement: an updated guideline for reporting systematic reviews

          The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement, published in 2009, was designed to help systematic reviewers transparently report why the review was done, what the authors did, and what they found. Over the past decade, advances in systematic review methodology and terminology have necessitated an update to the guideline. The PRISMA 2020 statement replaces the 2009 statement and includes new reporting guidance that reflects advances in methods to identify, select, appraise, and synthesise studies. The structure and presentation of the items have been modified to facilitate implementation. In this article, we present the PRISMA 2020 27-item checklist, an expanded checklist that details reporting recommendations for each item, the PRISMA 2020 abstract checklist, and the revised flow diagrams for original and updated reviews.
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            Extramammary Paget's disease: treatment, prognostic factors and outcome in 76 patients.

            Extramammary Paget's disease (EMPD) is a rare cutaneous carcinoma usually presenting as a genital erythematous lesion in the elderly. Although most EMPD tumours are in situ, invasive EMPD has a poor prognosis. To evaluate the clinical and pathological features of EMPD and determine prognostic factors for survival. The medical records of 76 patients with EMPD were retrospectively reviewed. Of the 66 patients who underwent curative surgical excision, five (8%) developed local recurrence, but surgical margin ( 2 cm) was not correlated with local recurrence. Thirteen of the 76 patients (17%) developed systemic metastases and 10 of these died of disease. On univariate analysis, the presence of nodules in the primary tumour, clinical lymph node swelling, elevated serum carcinoembryonic antigen (CEA) levels, tumour invasion level and lymph node metastasis were significant prognostic factors. On multivariate analysis, invasion level and elevated serum CEA were the only factors that were significantly associated with reduced survival. Invasion level and lymph node metastasis are important prognostic factors in EMPD. In patients with in situ tumour, local tumour control is the major aim of treatment; however, wide surgical margins are not associated with a lower risk of local recurrence.
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              • Article: not found
              Is Open Access

              Paget disease of the vulva.

              In this review, we provide an overview of the clinical aspects, histopathology, molecular genetics, and treatment options for Vulvar Paget's Disease (VPD), a rare skin disease, most commonly found in postmenopausal Caucasian women. The underlying cause of VPD remains not well understood. VPD is rarely associated with an underlying urogenital, gastrointestinal or vulvar carcinoma. In approximately 25% of the cases, VPD is invasive; in these cases, the prognosis is worse than in non-invasive cases. Recurrence rates in invasive VPD are high: 33% in cases with clear margins, and even higher when surgical margins are not clear, regardless of invasion. Historically, surgical excision has been the treatment of choice. Recent studies show that imiquimod cream may be an effective and safe alternative.
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                Author and article information

                Journal
                Ann Surg Open
                Ann Surg Open
                AS9
                Annals of Surgery Open
                Wolters Kluwer Health, Inc. (Two Commerce Square, 2001 Market Street, Philadelphia, PA 19103 )
                2691-3593
                December 2023
                02 November 2023
                : 4
                : 4
                : e339
                Affiliations
                From the [* ]Division of Surgery and Surgical Oncology, Department of Sarcoma, Peritoneal and Rare Tumours (SPRinT), National Cancer Centre Singapore, Singapore
                []Division of Surgery and Surgical Oncology, Department of Sarcoma, Peritoneal and Rare Tumours (SPRinT), Singapore General Hospital, Singapore
                []Division of Clinical Trials and Epidemiological Sciences, National Cancer Centre Singapore, Singapore
                [§ ]Laboratory of Applied Human Genetics, Division of Medical Sciences, National Cancer Centre Singapore, Singapore
                []Division of Medical Oncology, National Cancer Centre Singapore, Singapore
                []Cancer Discovery Hub, National Cancer Centre Singapore, Singapore
                [# ]Institute of Molecular and Cell Biology, A*STAR Research Entities, Singapore
                [** ]SingHealth Duke-NUS Oncology Academic Clinical Program, Duke NUS Medical School, Singapore
                [†† ]Division of Radiation Oncology, National Cancer Centre Singapore, Singapore
                [‡‡ ]Division of Oncologic Imaging, National Cancer Centre Singapore, Singapore
                [§§ ]Department of Anatomical Pathology, Singapore General Hospital, Singapore
                [‖‖ ]Duke-NUS Medical School, Singapore
                [¶¶ ]Division of Surgery and Surgical Oncology, Department of Urology, Singapore General Hospital, Singapore
                [## ]SingHealth Duke-NUS Surgery Academic Clinical Program, Duke NUS Medical School, Singapore.
                Author notes
                Reprints: Chin-Ann Johnny Ong, MBBS, MMed, FRCS, PhD, Division of Surgery and Surgical Oncology, Department of Sarcoma, Peritoneal and Rare Tumours (SPRinT), National Cancer Centre Singapore, 30 Hospital Boulevard, Singapore 168583. Email: johnny.ong.c.a@ 123456singhealth.com.sg
                Article
                00009
                10.1097/AS9.0000000000000339
                10735084
                38144489
                177ae5f5-629e-4154-aba8-7c3173d20546
                Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc.

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.

                History
                : 8 February 2023
                : 11 August 2023
                Categories
                Meta-Analysis
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                extra-mammary paget's disease,mapping biopsy,recurrence

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