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      Clinical and histopathological characteristics of atrophic pigmented dermatofibrosarcoma protuberans: A retrospective study of 14 cases

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          Abstract

          Background

          Dermatofibrosarcoma protuberans (DFSP) invades the dermis and subcutaneous tissue. DFSP with both atrophic and pigmentary (AP-DFSP) features is extremely rare and the clinical characteristics remain unknown. Here we aim to characterize the clinical, histopathologic and prognostic features of AP-DFSP.

          Methods

          Fourteen cases of patients with AP-DFSP were collected from our institution and published online, including four unreported cases and ten published cases. The clinical appearance, immunohistochemical markers, treatment, and prognosis were analyzed to obtain the clinical and histological features.

          Results

          There were six males and eight females with a mean age of 25 years old. The vast majority of lesions appeared in the trunk (10/14, 71.4 %) and limbs (3/14, 21.4 %), whereas a minority involved the infraorbital area (1/14, 7.2 %). The most typical manifestation was a depressed plaque-like lesion with fuchsia and bluish color. Histologically, AP-DFSP harbored both atrophic and pigmented features, presenting with a thinner dermis and intradermal melanin granules. Immunohistochemically, CD34 and vimentin were positive while S100 was negative in tumor tissues. The Ki67 index was less than 10 %. Thirteen of fourteen patients had complete excision surgery and follow-ups showed no local recurrence or distant metastasis.

          Conclusion

          Compared to DFSP, AP-DFSP shows more benign clinical and histological features with a good prognosis. Surgical intervention leads to a significant reduction in tumor burden and dramatically increases the likelihood of complete remission.

          Highlights

          • •

            Atrophic pigmented dermatofibrosarcoma protuberans (AP-DFSP) is extremely rare and harbors both atrophic and pigmentary features.

          • •

            AP-DFSP appears predominantly in young people with a mean age around 25 years old.

          • •

            Compared to classical DFSP, AP-DFSP has a much more indolent feature and a good prognosis after surgical excision.

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          Most cited references55

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          Vimentin in cancer and its potential as a molecular target for cancer therapy.

          Arun Satelli, Shulin Li (2011)
          Vimentin, a major constituent of the intermediate filament family of proteins, is ubiquitously expressed in normal mesenchymal cells and is known to maintain cellular integrity and provide resistance against stress. Vimentin is overexpressed in various epithelial cancers, including prostate cancer, gastrointestinal tumors, tumors of the central nervous system, breast cancer, malignant melanoma, and lung cancer. Vimentin's overexpression in cancer correlates well with accelerated tumor growth, invasion, and poor prognosis; however, the role of vimentin in cancer progression remains obscure. In recent years, vimentin has been recognized as a marker for epithelial-mesenchymal transition (EMT). Although EMT is associated with several tumorigenic events, vimentin's role in the underlying events mediating these processes remains unknown. By virtue of its overexpression in cancer and its association with tumor growth and metastasis, vimentin serves as an attractive potential target for cancer therapy; however, more research would be crucial to evaluate its specific role in cancer. Our recent discovery of a vimentin-binding mini-peptide has generated further impetus for vimentin-targeted tumor-specific therapy. Furthermore, research directed toward elucidating the role of vimentin in various signaling pathways would reveal new approaches for the development of therapeutic agents. This review summarizes the expression and functions of vimentin in various types of cancer and suggests some directions toward future cancer therapy utilizing vimentin as a potential molecular target.
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            Vimentin coordinates fibroblast proliferation and keratinocyte differentiation in wound healing via TGF-β-Slug signaling.

            Fang-Fang Cheng, Yue Shen, Ponnuswamy Mohanasundaram … (2016)
            Vimentin has been shown to be involved in wound healing, but its functional contribution to this process is poorly understood. Here we describe a previously unrecognized function of vimentin in coordinating fibroblast proliferation and keratinocyte differentiation during wound healing. Loss of vimentin led to a severe deficiency in fibroblast growth, which in turn inhibited the activation of two major initiators of epithelial-mesenchymal transition (EMT), TGF-β1 signaling and the Zinc finger transcriptional repressor protein Slug, in vimentin-deficient (VIM(-/-)) wounds. Correspondingly, VIM(-/-) wounds exhibited loss of EMT-like keratinocyte activation, limited keratinization, and slow reepithelialization. Furthermore, the fibroblast deficiency abolished collagen accumulation in the VIM(-/-) wounds. Vimentin reconstitution in VIM(-/-) fibroblasts restored both their proliferation and TGF-β1 production. Similarly, restoring paracrine TGF-β-Slug-EMT signaling reactivated the transdifferentiation of keratinocytes, reviving their migratory properties, a critical feature for efficient healing. Our results demonstrate that vimentin orchestrates the healing by controlling fibroblast proliferation, TGF-β1-Slug signaling, collagen accumulation, and EMT processing, all of which in turn govern the required keratinocyte activation.
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              Imatinib mesylate in advanced dermatofibrosarcoma protuberans: pooled analysis of two phase II clinical trials.

              Maria Debiec-Rychter, , Peter Hohenberger … (2010)
              Dermatofibrosarcoma protuberans (DFSP) is a dermal sarcoma typically carrying a translocation between chromosomes 17 and 22 that generates functional platelet-derived growth factor B (PDGFB). Two distinct phase II trials of imatinib (400 to 800 mg daily) in patients with locally advanced or metastatic DFSP were conducted and closed prematurely, one in Europe (European Organisation for Research and Treatment of Cancer [EORTC]) with 14-week progression-free rate as the primary end point and the other in North America (Southwest Oncology Group [SWOG]) with confirmed objective response rate as the primary end point. In the EORTC trial, confirmation of PDGFB rearrangement was required, and surgery was undertaken after 14 weeks if feasible. The SWOG study confirmed t(17;22) after enrollment. Sixteen and eight patients were enrolled onto the EORTC and SWOG trials, respectively. Tumor size ranged from 1.2 to 49 cm. DFSP was located on head/neck, trunk, and limb in seven, 11, and six patients, respectively, and was classic, pigmented, and fibrosarcomatous DFSP in 13, one, and nine patients, respectively. Metastases were present in seven patients (lung involvement was present six patients). Eleven patients (4%) had partial response as best response, and four patients had progressive disease as best response. Median time to progression (TTP) was 1.7 years. Imatinib was stopped in 11 patients because of progression, one patient because of toxicity, and two patients after complete resection of disease. Median overall survival (OS) time has not been reached; 1-year OS rate was 87.5%. Imatinib is active in DFSP harboring t(17;22) including fibrosarcomatous DFSP, with objective response rate approaching 50%. Response rates and TTP did not differ between patients taking 400 mg daily versus 400 mg twice a day.
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                Author and article information

                Contributors
                Songmei Geng
                Yale Liu
                Journal
                Journal ID (nlm-ta): Heliyon
                Journal ID (iso-abbrev): Heliyon
                Title: Heliyon
                Publisher: Elsevier
                ISSN (Electronic): 2405-8440
                Publication date PMC-release: 11 October 2024
                Publication date Collection: 30 November 2024
                Publication date (Electronic): 11 October 2024
                Volume: 10
                Issue: 22
                Electronic Location Identifier: e39271
                Affiliations
                [a ]Department of Dermatology, The Second Affiliated Hospital of Xi'an Jiaotong University, Shaanxi, China
                [b ]Department of Molecular and Cell Biology, University of California, Berkeley, Berkeley, CA, United States
                Author notes
                [* ]Corresponding author. Department of Dermatology, the Second Affiliated Hospital of Xi'an Jiaotong University, 157 West 5th Road, Xi'an, 710004, China. liuyale0703@ 123456xjtu.edu.cn
                [** ]Corresponding author. Department of Dermatology, the Second Affiliated Hospital of Xi'an Jiaotong University, 157 West 5th Road, Xi'an, 710004, China. gsm312@ 123456yahoo.com
                [1]

                These two authors contribute equally to this work.

                Article
                Publisher Item ID: S2405-8440(24)15302-9 Publisher ID: e39271
                DOI: 10.1016/j.heliyon.2024.e39271
                PMC ID: 11616497
                PubMed ID: 39634408
                SO-VID: 741b08f5-281e-40f4-b50d-c4eeddc505b7
                Copyright © © 2024 The Authors
                License:

                This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/).

                History
                Date received : 9 July 2024
                Date revision received : 8 October 2024
                Date accepted : 10 October 2024
                Categories
                Subject: Research Article

                Keywords: atrophic pigmented dermatofibrosarcoma protuberans,indolent character,good prognosis
                Data availability:
                Keywords: atrophic pigmented dermatofibrosarcoma protuberans, indolent character, good prognosis

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