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      Hyperpigmentation following Treatment of Frontal Fibrosing Alopecia

      case-report

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          Abstract

          Introduction: Frontal fibrosing alopecia (FFA) is a scarring alopecia characterized by progressive recession of the frontotemporal hairline. Current treatment is aimed at stopping progression, and the combination of dutasteride and pimecrolimus is the most effective therapy. Side effects associated with dutasteride are erectile dysfunction as well as breast tenderness and enlargement, while pimecrolimus produces a burning sensation. Case Report: We present a 57-year-old postmenopausal female with a 3-year history of a scarring alopecic plaque in her frontotemporal region. Biopsy confirmed the diagnosis of FFA, and she was started on dutasteride 0.5 mg p.o. q.d., and later, topical pimecrolimus 1% b.i.d. was added. Eight months after initiating treatment, she showed hyperpigmentation on her metacarpophalangeal and interphalangeal joints, as well as on the cheeks and on the chin; dutasteride and pimecrolimus were discontinued. After 5 months of follow-up, her hyperpigmentation improved by 80% only by using photoprotection. Conclusion: Because of the variable clinical course of FFA, treatment is focused on halting its progression. Several therapeutic agents have been evaluated and the combination of dutasteride and pimecrolimus has shown a high response rate. There is no reported evidence of hyperpigmentation associated with this combination.

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          Most cited references17

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          Frontal fibrosing alopecia in postmenopausal women.

          Frontal fibrosing alopecia is a variety of cicatricial alopecia characterized by a band of frontal/frontoparietal hair recession and marked decrease or a complete loss of the eyebrows, typically observed in women who are postmenopausal. The purpose of this study was to report clinical and histopathologic findings and results of treatment in a group of women affected by the disease. A total of 14 women with alopecia of the frontal hairline were evaluated from June 2000 through July 2003 in our outpatient consultation for hair disorders. Clinical examination revealed a band of symmetric recession of the frontoparietal hairline extending to the preauricular areas associated with loss of follicular orifices, mild skin atrophy, and perifollicular erythema at the scalp margin. In all, 9 patients also had partial or total loss of the eyebrows. The histologic features of the scalp specimens were similar in all our patients with a reduction of the number of hair follicles, and a high number of intermediate and velluslike follicles. Intemediate and velluslike follicles were more commonly affected than terminal follicles by the lymphocytic inflammatory infiltrate and perifollicular fibrosis. Frontal fibrosing alopecia is a cicatricial alopecia that follows destruction of hair follicles by an inflammatory lymphocytic infiltrate that is localized around the upper portion of the hair follicle. It differs from lichen planopilaris because the lymphocytic infiltrate and fibrosis affect selectively the intermediate and the velluslike follicles of the frontal margin and eyebrows. The reason for this selective involvement is still unknown. Frontal fibrosing alopecia may represent a variety of lichen planopilaris with selective involvement of certain androgen-dependent areas. The affected follicles may have typical biologic markers that could explain the clinical and histologic features found in the disease. It is interesting to note that some of the patients treated with finasteride (2.5 mg/d) showed an arrest in the progression of the disease. Even if there is no proof for a hormonal basis of the disease, the effectiveness of finasteride in some patients may indicate that androgens might be partially responsible of the pathogenesis of the disease.
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            Treatment of frontal fibrosing alopecia and lichen planopilaris: a systematic review.

            Frontal fibrosing alopecia (FFA) is a primary lymphocytic cicatricial alopecia with characteristic clinical pattern of progressive frontotemporal hairline recession, perifollicular erythema and hyperkeratosis and symptoms of itch and burning, occurring mainly in post-menopausal women. FFA is considered a subtype of lichen planopilaris (LPP), based on their identical histopathology. Currently, no evidence-based treatment is available for FFA. Our aim was to determine the effectiveness of available treatment options for FFA, and to identify promising treatment options for future studies. For this, literature search was conducted to find all primary studies on the treatment of FFA and LPP. From the primary studies, data were subtracted and analysed. No randomized controlled trials were found, and one controlled trial. Treatment of 114 patients is described in the literature. They received 10 different regimes, of which oral 5-alpha-reductase inhibitors were provided most often, resulting in good clinical response in 45% of them. Hydroxychloroquine resulted in good clinical response in 30% of the 29 treated patients. Topical corticosteroid preparations are ineffective in FFA. The remaining treatments were all reported in less than 10 patients. For the treatment of LPP, topical corticosteroid preparations are the first line of treatment, followed by oral cyclosporine and systemic corticosteroids, although they are characterized by a high relapse rate. Summarizing, there is currently no effective treatment of FFA, the most effective being oral 5-alpha-reductase inhibitors that possibly affect the accompanying androgenetic alopecia. We argue that oral cyclosporine A might be a good candidate for future studies on the treatment of FFA.
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              Frontal fibrosing alopecia versus lichen planopilaris: a clinicopathological study.

              Frontal fibrosing alopecia (FFA) is an acquired scarring alopecia currently considered a clinical variant of lichen planopilaris (LPP). Our purpose was to examine the clinicopathological features of FFA. In addition, we investigated the similarities and differences between FFA and LPP. Biopsies from the scalp lesions of eight patients with FFA and eight patients with LPP were microscopically analyzed. Two cases of FFA and four cases of LPP were studied using direct immunofluorescence. In spite of the completely different clinical characteristics of FFA and LPP patients, the histopathological findings for the two entities were similar. Common microscopic findings for both FFA and LPP included an inflammatory lymphocytic infiltrate involving the isthmus and infundibulum of the hair follicles, the presence of apoptotic cells in the external root sheath, and a concentric fibrosis surrounding the hair follicles that resulted in their destruction with subsequent scarring alopecia. Biopsies taken from FFA patients showed less follicular inflammation and more apoptotic cells than those from LPP patients. In some cases of LPP, the inflammatory infiltrate involved the interfollicular epidermis, a finding never present in our FFA cases. Direct immunofluorescence was negative in the two cases of FFA studied and showed deposits of immunoglobulins and/or complement in two of the four LPP cases examined. The characteristic findings for FFA were more prominent apoptosis and less inflammation than found in LPP, along with spared interfollicular epidermis. FFA cases showed a rather characteristic histopathological pattern, although we could not find any clear-cut histological differences between FFA and LPP.
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                Author and article information

                Journal
                CDE
                CDE
                10.1159/issn.1662-6567
                Case Reports in Dermatology
                S. Karger AG
                1662-6567
                2013
                September – December 2013
                23 November 2013
                : 5
                : 3
                : 357-362
                Affiliations
                Department of Dermatology, Hospital Universitario ‘Dr. José Eleuterio González', Universidad Autónoma de Nuevo León, Monterrey, Mexico
                Author notes
                *Irma Margarita Pérez-Rodríguez, MD, Department of Dermatology, Hospital Universitario ‘Dr. José Eleuterio González', Avenida Madero y Gonzalitos s/n, Col. Mitras Centro, Monterrey 64460 (Mexico), E-Mail dra.irma.perez@gmail.com
                Article
                357022 PMC3861864 Case Rep Dermatol 2013;5:357-362
                10.1159/000357022
                PMC3861864
                24348386
                6e6a739d-58e1-4b0d-b417-778be1b23540
                © 2014 S. Karger AG, Basel

                Open Access License: This is an Open Access article licensed under the terms of the Creative Commons Attribution-NonCommercial 3.0 Unported license (CC BY-NC) ( http://www.karger.com/OA-license), applicable to the online version of the article only. Distribution permitted for non-commercial purposes only. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                History
                Page count
                Figures: 3, Pages: 6
                Categories
                Published: November 2013

                Oncology & Radiotherapy,Pathology,Surgery,Dermatology,Pharmacology & Pharmaceutical medicine
                Scarring alopecia,Hyperpigmentation,Frontal fibrosing alopecia

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