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[PMID]: 29188284
[Au] Autor:Diamond EL; Subbiah V; Lockhart AC; Blay JY; Puzanov I; Chau I; Raje NS; Wolf J; Erinjeri JP; Torrisi J; Lacouture M; Elez E; Martínez-Valle F; Durham B; Arcila ME; Ulaner G; Abdel-Wahab O; Pitcher B; Makrutzki M; Riehl T; Baselga J; Hyman DM
[Ad] Address:Memorial Sloan Kettering Cancer Center, New York, New York.
[Ti] Title:Vemurafenib for BRAF V600-Mutant Erdheim-Chester Disease and Langerhans Cell Histiocytosis: Analysis of Data From the Histology-Independent, Phase 2, Open-label VE-BASKET Study.
[So] Source:JAMA Oncol;4(3):384-388, 2018 Mar 01.
[Is] ISSN:2374-2445
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Importance: The histiocytic neoplasms Erdheim-Chester disease (ECD) and Langerhans cell histiocytosis (LCH) are highly enriched for BRAF V600 mutations and have been previously shown to be responsive to treatment with vemurafenib, an inhibitor of the BRAF V600 kinase. However, the long-term efficacy and safety of prolonged vemurafenib use in these patients are not defined. Here we analyze the final efficacy and safety data for vemurafenib in patients with ECD and LCH enrolled in the VE-BASKET study. Objective: To determine the efficacy and safety of vemurafenib in adults with ECD or LCH enrolled in the VE-BASKET study. Design, Setting, and Participants: The VE-BASKET study was an open-label, nonrandomized, multicohort study for patients with nonmelanoma cancers harboring the BRAF V600 mutation. Patients with BRAF V600-mutant ECD or LCH were enrolled in an "other solid tumor" cohort of the VE-BASKET study, and they were enrolled in the present study. Interventions: Patients received vemurafenib, 960 mg, twice daily continuously until disease progression, study withdrawal, or occurrence of intolerable adverse effects. Main Outcomes and Measures: The primary end point was confirmed objective response rate (ORR) by Response Evaluation Criteria in Solid Tumors (RECIST, version 1.1). Secondary end points included progression-free survival (PFS), overall survival (OS), metabolic response by modified positron-emission tomography (PET) Response Criteria in Solid Tumors (PERCIST) using 18F-fluorodeoxyglucose (FDG)-PET/computed tomography (CT), and safety. Results: A total of 26 patients from the VE-BASKET trial (22 with ECD, 4 with LCH) were included in the present study (14 women and 12 men; median age, 61 years; age range, 51-74 years). The confirmed ORR was 61.5% (95% CI, 40.6%-79.8%) in the overall cohort and 54.5% (95% CI, 32.2%-75.6%) in patients with ECD. All evaluable patients achieved stable disease or better. The median PFS and OS had not been reached in the overall cohort at study closure despite a median follow-up of 28.8 months; 2-year PFS was 86% (95% CI, 72%-100%), and 2-year OS was 96% (95% CI, 87%-100%). All 15 patients evaluated by FDG-PET/CT achieved a metabolic response, including 12 patients (80%) with a complete metabolic response. The most common adverse events (AEs) in the overall cohort included arthralgia, maculopapular rash, fatigue, alopecia, prolonged QT interval, skin papilloma, and hyperkeratosis. Hypertension and dermatologic AEs occurred at higher rates than those reported in metastatic melanoma. Conclusions and Relevance: In this study, vemurafenib had prolonged efficacy in patients with BRAF V600-mutant ECD and LCH and warrants consideration as a new standard of care for these patients.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180311
[Lr] Last revision date:180311
[St] Status:In-Data-Review
[do] DOI:10.1001/jamaoncol.2017.5029

  2 / 19066 MEDLINE  
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[PMID]: 29524253
[Au] Autor:Murad A; Bergfeld W
[Ad] Address:Department of Dermatology, Mater Misericordiae University Hospital, Dublin, Ireland.
[Ti] Title:5 alpha-reductase inhibitor treatment for frontal fibrosing alopecia: An evidence-based treatment update.
[So] Source:J Eur Acad Dermatol Venereol;, 2018 Mar 10.
[Is] ISSN:1468-3083
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Treatment for frontal fibrosing alopecia (FFA) is challenging and its treatment regimen often mirrors other lymphocytic-predominant cicatricial alopecia. 5 alpha-reductase inhibitor (5ARI) has been reported with some treatment success in severe cases of FFA. OBJECTIVE: To carry out evidence-based analysis of articles published on treatment efficacy and safety of 5 alpha-reductase inhibitor for the treatment of FFA. METHODS: Articles published on the use of 5ARI to treat FFA between 2005 to 2017 were reviewed, analysed and graded according to the American College of Physicians outcome study grading system. RESULTS: There were two studies with moderate-level of evidence that described the efficacy of 5ARI for treatment of FFA. 5ARI was commonly used as adjunctive therapy with positive results in recalcitrant disease. Mild to moderate hair regrowth was reported in one grade 2 and three lower grade (one grade 3 and two grade 4) studies. There is limited evidence on the safety aspects of this medication in most studies that were analysed. LIMITATIONS: Database studies might not fully account for confounders and is subjected to variations in methodology and data collection. CONCLUSION: This review demonstrated that FFA patients treated with 5ARI could achieve either disease stability or reduction in the rate of progression in selected cases. A well designed randomised, double-blind, controlled study would strengthen the role of 5ARI as part of treatment armamentarium for FFA. This article is protected by copyright. All rights reserved.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1803
[Cu] Class update date: 180310
[Lr] Last revision date:180310
[St] Status:Publisher
[do] DOI:10.1111/jdv.14930

  3 / 19066 MEDLINE  
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[PMID]: 29520806
[Au] Autor:Bassir SH; Chase I; Paster BJ; Gordon LB; Kleinman ME; Kieran MW; Kim DM; Sonis A
[Ad] Address:Division of Periodontology, Department of Oral Medicine, Infection, and Immunity, Harvard School of Dental Medicine, Boston, Massachusetts, USA.
[Ti] Title:Microbiome at sites of gingival recession in children with Hutchinson-Gilford progeria syndrome.
[So] Source:J Periodontol;, 2018 Feb 19.
[Is] ISSN:1943-3670
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:BACKGROUND: Hutchinson-Gilford progeria syndrome (HGPS) is a rare premature aging disorder with significant oral and dental abnormalities. Clinical symptoms include various features of accelerated aging such as alopecia, loss of subcutaneous fat, bone abnormalities, and premature cardiovascular disease. In addition, children with HGPS have been observed to suffer from generalized gingival recession. Whether periodontal manifestations associated with this syndrome are the results of changes in the oral flora is unknown. The present study aimed to identify the microbial composition of subgingival sites with gingival recession in children with HGPS. METHODS: Nine children with HGPS were enrolled in this study. Plaque samples were collected from teeth with gingival recession. DNA samples were analyzed using the Human Oral Microbe Identification Microarray (HOMIM). Microbial profiles from HGPS children were compared with microbial profiles of controls from healthy individuals (n = 9) and subjects with periodontal disease (n = 9). RESULTS: Comparison of microbial compositions of HGPS samples with periodontal health samples demonstrated significant differences for 2 bacterial taxa; Porphyromonas catoniae and Prevotella oulora were present in children with HGPS, but not normal controls. There were statistically significant differences of 20 bacterial taxa between HGPS and periodontal disease groups. CONCLUSION: Typical periodontal pathogeneses were not present at sites with gingival recession in HGPS children. The microbial compositions of sites of gingival recession and attachment loss in HGPS were generally more similar to those of periodontal health than periodontal disease. Species other than typical periodontal pathogens may be involved in this recession. This article is protected by copyright. All rights reserved.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1803
[Cu] Class update date: 180309
[Lr] Last revision date:180309
[St] Status:Publisher
[do] DOI:10.1002/JPER.17-0351

  4 / 19066 MEDLINE  
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[PMID]: 29482481
[Au] Autor:Kapoor R; Shome D
[Ad] Address:a Department of Cosmetic Dermatology & Facial Plastic Surgery, The Esthetic Clinics , Mumbai , India.
[Ti] Title:Intradermal injections of a hair growth factor formulation for enhancement of human hair regrowth - safety and efficacy evaluation in a first-in-man pilot clinical study.
[So] Source:J Cosmet Laser Ther;:1-11, 2018 Feb 26.
[Is] ISSN:1476-4180
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:BACKGROUND: Research has shown the efficacy of hair growth factors in hair regrowth. We describe the intradermal injections of a recombinant, bioengineered hair formulation, containing growth factors, into the scalp skin, for enhancement of hair regrowth and evaluate its efficacy. OBJECTIVES: The objective of this study was to assess the efficacy and safety of the hair growth factor formulation in reducing hair loss and enhancing hair growth. MATERIALS AND METHODS: This was an open-label, prospective, single-arm interventional pilot study in which 1000 patients were given intradermal injections of a hair formulation into the scalp skin. The formulation contains vascular endothelial growth factor, basic fibroblast growth factor, insulin-like growth factor, keratinocyte growth factor, thymosin ß4, and copper tripeptide-1 suspended in a sterile injectable vehicle. Intradermal injections of this hair formulation were injected into the scalp once every 3 weeks for a total of eight such sessions. Hair pull test was performed before every session. Videomicroscopic and global images were taken at baseline, fourth session, eighth session, and 2 months after the completion of the eight sessions. Relevant safety assessments through physical examination, questionnaires, and appropriate laboratory examination were conducted throughout the study. RESULTS: Significant reduction in hair fall was seen in 83% of the patients on hair pull test. Videomicroscopic image evaluation showed that most patients had a decrease in the number of vellus hairs, increase in number of terminal hairs, and increase in shaft diameter. Seventy-five percent of the patients believed that the hair injections were aiding the treatment of their hair loss, and it was also beneficial in post-hair transplant patients. At 1 year, a statistically significant increase in total hair count (P = 0.002) continued to be seen. Treatment was well tolerated. CONCLUSIONS: Intradermal injections of this hair formulation may be a promising option for treating male as well as female patterns of hair loss.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180308
[Lr] Last revision date:180308
[St] Status:Publisher
[do] DOI:10.1080/14764172.2018.1439965

  5 / 19066 MEDLINE  
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[PMID]: 29461879
[Au] Autor:Chang CJ; Yu DY; Chang SY; Hsiao YC
[Ad] Address:a Department of Plastic Surgery , Taipei Medical University Hospital , Taipei , Taiwan , Republic of China.
[Ti] Title:Comparing the effectiveness of laser vs. conventional endoforehead lifting.
[So] Source:J Cosmet Laser Ther;20(2):91-95, 2018 Apr.
[Is] ISSN:1476-4180
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:The objective of this study was to compare the efficacy and safety of laser versus conventional endoforehead lifting. Over a period of 12 years (January 2000-January 2012), a total of 110 patients with hyperactive muscles over the frontal region have been collected for a retrospective study. The SurgiLase 150XJ CO laser system, in conjunction with the flexible FIBERLASE, was used. The endoscope was 4 mm in diameter with an angle of 30°. The primary efficacy measurement was the assessment of the final outcome for using laser vs. conventional methods. Both groups were observed at three weeks, six weeks and six months after surgery. The most common complication in early convalescence (three weeks) was swelling. This was followed by local paraesthesia, ecchymosis, localized hematomas and scar with alopecia. All these problems disappeared completely after the 6-month study period. Based on a chi-square analysis, there were clinically and statistically significant differences favouring the laser endoforehead surgery in the operative time, early and late complications. All patients achieved significant improvement after both laser and conventional endoforehead surgery in the final outcome. However, the early and late complications indicated a greater difference in the laser group.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180308
[Lr] Last revision date:180308
[St] Status:In-Process
[do] DOI:10.1080/14764172.2016.1256487

  6 / 19066 MEDLINE  
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[PMID]: 29398487
[Au] Autor:Majtan T; Jones W; Krijt J; Park I; Kruger WD; Kozich V; Bassnett S; Bublil EM; Kraus JP
[Ad] Address:Department of Pediatrics, University of Colorado School of Medicine, Aurora, CO 80045, USA. Electronic address: tomas.majtan@ucdenver.edu.
[Ti] Title:Enzyme Replacement Therapy Ameliorates Multiple Symptoms of Murine Homocystinuria.
[So] Source:Mol Ther;26(3):834-844, 2018 Mar 07.
[Is] ISSN:1525-0024
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Classical homocystinuria (HCU) is the most common inherited disorder of sulfur amino acid metabolism caused by deficiency in cystathionine beta-synthase (CBS) activity and characterized by severe elevation of homocysteine in blood and tissues. Treatment with dietary methionine restriction is not optimal, and poor compliance leads to serious complications. We developed an enzyme replacement therapy (ERT) and studied its efficacy in a severe form of HCU in mouse (the I278T model). Treatment was initiated before or after the onset of clinical symptoms in an effort to prevent or reverse the phenotype. ERT substantially reduced and sustained plasma homocysteine concentration at around 100 µM and normalized plasma cysteine for up to 9 months of treatment. Biochemical balance was also restored in the liver, kidney, and brain. Furthermore, ERT corrected liver glucose and lipid metabolism. The treatment prevented or reversed facial alopecia, fragile and lean phenotype, and low bone mass. In addition, structurally defective ciliary zonules in the eyes of I278T mice contained low density and/or broken fibers, while administration of ERT from birth partially rescued the ocular phenotype. In conclusion, ERT maintained an improved metabolic pattern and ameliorated many of the clinical complications in the I278T mouse model of HCU.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1802
[Cu] Class update date: 180308
[Lr] Last revision date:180308
[St] Status:In-Data-Review

  7 / 19066 MEDLINE  
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[PMID]: 29341265
[Au] Autor:Sundberg JP; Hordinsky MK; Bergfeld W; Lenzy YM; McMichael AJ; Christiano AM; McGregor T; Stenn KS; Sivamani RK; Pratt CH; King LE
[Ad] Address:The Jackson Laboratory, Bar Harbor, ME, USA.
[Ti] Title:Cicatricial Alopecia Research Foundation meeting, May 2016: Progress towards the diagnosis, treatment and cure of primary cicatricial alopecias.
[So] Source:Exp Dermatol;27(3):302-310, 2018 Mar.
[Is] ISSN:1600-0625
[Cp] Country of publication:Denmark
[La] Language:eng
[Ab] Abstract:Primary cicatricial alopecias (PCAs) are a group of skin diseases in which there is progressive and permanent destruction of hair follicles followed by replacement with fibrous tissue. Unfortunately, by the time patients seek clinical evaluation of their hair loss, the skin is already inflamed and/or scarred, so there is little hope for a return to their normal hair growth pattern. Clinical and basic science investigations are now focusing on three forms of human PCA: lichen planopilaris (LPP), frontal fibrosing alopecia (FFA) and central centrifugal cicatricial alopecia (CCCA). Transcriptome, lipidome and other new technologies are providing new insight into the pathogenesis of some of these diseases that are being validated and further investigated using spontaneous and genetically engineered mouse models.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1801
[Cu] Class update date: 180309
[Lr] Last revision date:180309
[St] Status:In-Data-Review
[do] DOI:10.1111/exd.13495

  8 / 19066 MEDLINE  
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[PMID]: 29278862
[Au] Autor:Heppt MV; Letulé V; Laniauskaite I; Reinholz M; Tietze JK; Wolff H; Ruzicka T; Sattler EC
[Ad] Address:Department of Dermatology and Allergy, Ludwig-Maximilian-University Munich, Munich, Germany.
[Ti] Title:Frontal Fibrosing Alopecia: A Retrospective Analysis of 72 Patients from a German Academic Center.
[So] Source:Facial Plast Surg;34(1):88-94, 2018 Feb.
[Is] ISSN:1098-8793
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Frontal fibrosing alopecia (FFA) describes the scarring, band-like recession of the frontotemporal hairline. Treatment is difficult, and currently, no evidence-based therapy exists. The purpose of this study is to report clinical features and treatment responses in a large cohort of patients with FFA. The authors analyzed a series of 72 patients with a clinical or histologic diagnosis of FFA. A total of 70 patients were female (97.2%), and 2 were male (2.8%). In females, the first onset of FFA was postmenopausal in 81.4% ( = 57). Documented eyebrow loss was present in 61.1% ( = 44), whereas involvement of eyelashes and body hair was reported in only 4.2% ( = 3) and 5.6% ( = 4), respectively. Clinical symptoms were itching (40.3%, = 29) and trichodynia (4.2%, = 3) in the alopecic area. Virtually all patients were treated with topical high-potency steroids. Additional treatments were topical tacrolimus, systemic retinoids, and hydroxychloroquine. A total of 48 patients (66.7%) received a combination of high-potency steroids with topical pimecrolimus. In this subgroup, subjective improvement or disease stabilization was reported by 64.6% ( = 31), and the hairline was stabilized on average after 9 to 12 months of therapy. The combination therapy of topical high-potency steroids with pimecrolimus may be an effective and steroid-saving treatment for FFA.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180309
[Lr] Last revision date:180309
[St] Status:In-Process
[do] DOI:10.1055/s-0037-1615281

  9 / 19066 MEDLINE  
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[PMID]: 29264764
[Au] Autor:Quintard B; Giorgiadis M; Feirrera X; Lefaux B; Schohn C; Lemberger K
[Ad] Address:Parc Zoologique et botanique de Mulhouse, 51 rue du jardin Zoologique, 68100, Mulhouse, France. benoit.quintard@mulhouse-alsace.fr.
[Ti] Title:Evidence for the possible occurrence of Grave's disease in a blue-eyed black lemur (Eulemur flavifrons).
[So] Source:Primates;59(2):123-126, 2018 Mar.
[Is] ISSN:1610-7365
[Cp] Country of publication:Japan
[La] Language:eng
[Ab] Abstract:The blue-eyed black lemur (Eulemur flavifrons) is classified by the International Union for Conservation of Nature (IUCN) as critically endangered. A 23-year-old male housed at Mulhouse Zoo presented with lethargy, polyphagia, alopecia, and chronic weight loss. Clinical examination suggested an endocrine pathology such as hyperthyroidism. Secondary examinations included cervical ultrasound, thyroid biopsy, and scintigraphy. The latter revealed elevated thyroid activity. Blood analysis was performed to measure the level of anti-receptor thyroid-stimulating hormone antibodies, which allowed us to test the autoimmune hypothesis. The high level of antibodies together with levels of thyroid-stimulating hormone and the scintigraphy images led to the diagnosis of Grave's disease. Carbimazole treatment followed by thyroidectomy resulted in a quick weight gain and general improvement in health status. The following breeding season, the treated individual sired an offspring. To the authors' knowledge, this is the first report of likely Grave's disease in a non-human primate.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180309
[Lr] Last revision date:180309
[St] Status:In-Process
[do] DOI:10.1007/s10329-017-0644-5

  10 / 19066 MEDLINE  
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[PMID]: 29517680
[Au] Autor:Chen M; Zhou W; Zhang Z; Zou Y; Li C
[Ad] Address:Department of Endocrinology, the First Affiliated Hospital, College of Medicine, Zhejiang University.
[Ti] Title:An ovarian Leydig cell tumor of ultrasound negative in a postmenopausal woman with hirsutism and hyperandrogenism: A case report.
[So] Source:Medicine (Baltimore);97(10):e0093, 2018 Mar.
[Is] ISSN:1536-5964
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:RATIONALE: The incidence of severe hyperandrogenism associated with masculinity in women is very low. While rare and difficult to diagnose, androgen secreting tumors should be suspected in women with hyperandrogenism and hirsutism, especially in the postmenopausal population. Herein we present one case of ovarian Leydig cell tumor (LCT) with markedly elevated serum testosterone levels and frank hirsutism. PATIENT CONCERNS: A 60-year-old woman, presented with increased hair growth and androgenic alopecia and the hormonal laboratory examination showed that she had elevated serum testosterone level and normal dehydroepiandrosterone sulfate (DHEAS), androstenedione, 17- hydroxyprogesterone, cortisol and thyroid stimulating hormone (TSH). DIAGNOSES: The diagnosis of possible testosterone secreting tumor was performed when pelvic computed tomography (CT) and magnetic resonance image (MRI) showed a right adnexal mass of 15mm×16mm indicative of sex cord- stromal tumors. INTERVENTIONS: The patient received laparoscopic total abdominal hysterectomy and bilateral salpingo-oophorectomy. OUTCOMES: After operation, testosterone got back to the normal level and clinical symptoms subsided. LESSONS: It is common that postmenopausal androgen excess is a state of relative or absolute androgen excess originating from the adrenal gland and/or ovaries. In either case, doctors need to assess such patients and exclude relatively rare potential causes of tumors. Any woman who has hirsutism or frank evidence of markedly increased testosterone should exclude this kind of possibility of androgen producing tumors. It is possible to determine the origin of androgen hypersecretion with the severity of symptoms, the extent of androgen excess, and the relevant imaging studies. Since LCT are rare ovarian sex-cord stromal tumors, it can be beneficial for diagnosis with careful research of patient history of the defeminization followed by virilization, and a CT and MRI image.
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1803
[Cu] Class update date: 180308
[Lr] Last revision date:180308
[St] Status:In-Process
[do] DOI:10.1097/MD.0000000000010093


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