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World Journal of Clinical Cases World Journal of W J C C Clinical Cases Submit a Manuscript: http://www.f6publishing.com World J Clin Cases 2018 November 26; 6(14): 854-861 DOI: 10.12998/wjcc.v6.i14.854 ISSN 2307-8960 (online) CASE REPORT Intralesional and topical glucocorticoids for pretibial myxedema: A case report and review of literature Fan Zhang, Xin-Yue Lin, Jian Chen, Shi-Qiao Peng, Zhong-Yan Shan, Wei-Ping Teng, Xiao-Hui Yu Fan Zhang, Xin-Yue Lin, Jian Chen, Shi-Qiao Peng, Zhong- Manuscript source: Unsolicited manuscript Yan Shan, Wei-Ping Teng, Xiao-Hui Yu, Department of Endocrinology and Metabolism, Institute of Endocrinology, Corresponding author to: Xiao-Hui Yu, MD, PhD, Professor, Liaoning Provincial Key Laboratory of Endocrine Diseases, The Department of Endocrinology and Metabolism, Institute of First Affiliated Hospital of China Medical University, Shenyang Endocrinology, Liaoning Provincial Key Laboratory of Endocrine 110001, Liaoning Province, China Diseases, The First Affiliated Hospital of China Medical Uni­ versity, No. 155, Nanjing North Street, Heping District, Shenyang ORCID number: Fan Zhang (0000­0002­0176­2715); Xin­Yue 110001, Liaoning Province, China. [email protected] Lin (0000­0002­8168­7531); Jian Chen (0000­0003­3589­7236); Telephone: +86­24­83283135 Shi­Qiao Peng (0000­0002­2164­2825); Zhong­Yan Shan (0000­0002­2849­2380); Wei­Ping Teng (0000­0002­6445­6192); Received: August 8, 2018 Xiao­Hui Yu (0000­0003­4802­9673). Peer-review started: August 9, 2018 First decision: August 24, 2018 Author contributions: All the authors materially participated Revised: September 4, 2018 in this work, read and approved the final manuscript; Zhang F is Accepted: October 12, 2018 the first author for this study; Yu XH is the corresponding author Article in press: October 11, 2018 supervising this work; Zhang F and Lin XY managed the case Published online: November 26, 2018 and drafted the manuscript; Zhang F, Lin XY, Chen J and Peng SQ performed data analysis and interpretation based on literature review; Yu XH, Shan ZY and Teng WP reviewed the manuscript; Lin XY and Chen J prepared histopathological results; All authors participated sufficiently in the work and take public responsibility Abstract for its content. Pretibial myxedema (PTM), an uncommon manifestation of Graves’ disease (GD), is a local autoimmune reaction Informed consent statement: Written consent was obtained in the cutaneous tissue. The treatment of PTM is a from the patient. The patient consented to the publication of clinical challenge. We herein report on a patient with medical data (including figures from diagnostic imaging results PTM who achieved complete remission by multipoint and from histological examination results). subcutaneous injections of a long-acting glucocorticoid Conflict-of-interest statement: All authors declare no conflicts and topical glucocorticoid ointment application for a of interests. self-controlled study. A 53-year-old male presented with a history of GD for 3.5 years and a history of PTM CARE Checklist (2013) statement: This report follows the for 1.5 years. Physical examination revealed slight guidelines of the CARE Checklist (2013). exophthalmos, a diffusely enlarged thyroid gland, and PTM of both lower extremities. One milliliter of Open-Access: This article is an open­access article which was triamcinolone acetonide (40 mg) was mixed well with selected by an in­house editor and fully peer­reviewed by external 9 mL of 2% lidocaine in a 10 mL syringe. Multipoint reviewers. It is distributed in accordance with the Creative intralesional injections into the skin lesions of the Commons Attribution Non Commercial (CC BY­NC 4.0) license, right lower extremity were conducted with 0.5 mL of which permits others to distribute, remix, adapt, build upon this work non­commercially, and license their derivative works on the premixed solution. A halometasone ointment was different terms, provided the original work is properly cited and used once daily for PTM of the left lower extremity the use is non­commercial. See: http://creativecommons.org/ until the PTM had remitted completely. The patient’s licenses/by­nc/4.0/ PTM achieved complete remission in both legs after WJCC|www.wjgnet.com 854 November 26, 2018|Volume 6|Issue 14| Zhang F et al . Local glucocorticoid treatment for PTM an approximately 5-mo period of therpy that included triamcinolone injections once a week for 8 wk and then once a month for 2 mo for the right lower extremity and halometasone ointment application once daily for 8 wk and then once 3-5 d for 2 mo for the left lower extremity. The total dosage of triamcinolone acetonide for the right leg was 200 mg. Our experience with this patient suggests that multipoint subcutaneous injections of a long-acting glucocorticoid and topical glucocorticoid ointment application are safe, effective, and convenient treatments. However, the topical appli- cation of a glucocorticoid ointment is a more convenient treatment for patients with PTM. Key words: Intralesional injection; Pretibial myxedema; Figure 1 The patient’s lower extremities presented pretibial myxedema Glucocorticoid; Halometasone; Triamcinolone acetonide; before therapy. The bilateral pretibial and ankle skin was thickened with a hard texture, uneven surface, and non-pitting edema (yellow arrow). Case report © The Author(s) 2018. Published by Baishideng Publishing multipoint subcutaneous injections of a long­acting Group Inc. All rights reserved. glucocorticoid (triamcinolone) and topical application of a glucocorticoid ointment for the two legs. Core tip: Pretibial myxedema (PTM) is an extrathyroidal, dermatological manifestation of Graves’ disease. And PTM may be associated with autoimmunity. Local gluco- CASE REPORT corticoid therapy is the most common and effective Medical history option for PTM. However, any difference in efficacy A 53­year­old man visited our clinic for fatigue, irri­ between the external use of a glucocorticoid ointment tability, and weight loss for 3.5 years and PTM for 1.5 and intralesional glucocorticoid is unclarified. Our ex- years. Three and a half years before, he had felt weak perience with this patient suggests that multipoint subcutaneous injections of a long-acting glucocorticoid and had excessive sweating, palpitations, and a weight and topical glucocorticoid ointment application are loss of 10 kg. One month later, he saw a doctor at a safe, effective, and convenient treatments. However, local hospital. His thyroid function showed a decreased the topical application of a glucocorticoid ointment is a TSH level and an elevated free triiodothyronine (FT3) more convenient treatment for patients with PTM. level. His serum thyrotropin receptor antibody (TRAb) level was more than 40 IU/L (reference interval for TRAb: 0­1.75 IU/L). He was diagnosed with hyper­ Zhang F, Lin XY, Chen J, Peng SQ, Shan ZY, Teng WP, Yu thyroidism and prescribed methimazole 15 mg once XH. Intralesional and topical glucocorticoids for pretibial daily. However, the patient did not take the drug myxedema: A case report and review of literature. World J Clin regularly. One and a half years ago, swollen red areas Cases 2018; 6(14): 854­861 Available from: URL: http://www. with a diameter of about 5 cm and clear boundaries and wjgnet.com/2307­8960/full/v6/i14/854.htm DOI: http://dx.doi. a slightly tougher texture ­ but without itching ­ were org/10.12998/wjcc.v6.i14.854 found above both ankles. He was diagnosed at the local hospital as having PTM and prescribed halometasone 1 g twice a day and oral tripterygium. One week later, the myxedema had improved. He continued treatment for 2 INTRODUCTION wk but after discontinuing the halometasone ointment, Pretibial myxedema (PTM) is an extrathyroidal, der­ the myxedema relapsed and was accompanied by matological manifestation of Graves’ disease (GD). occasional acupuncture­like pain. Later, he gave up Recent research on the pathogenesis of PTM has treatment for 16 mo until he came to our hospital. shown that PTM may be associated with autoimmunity. The presence of thyroid­stimulating hormone (TSH) Physical examination receptors in fibroblasts of the skin may play a major The patient presented with eyelid edema, slight ex­ role in the pathogenesis of PTM. In general, PTM is self­ ophthalmos, and a diffusely enlarged thyroid gland limited and asymptomatic; however, advanced cases (grade Ⅱ-Ⅲ). The bilateral pretibial and ankle skin was may cause cosmetic or functional problems. Local thickened with a hard texture, uneven surface, and glucocorticoid therapy is the most common and effective non­pitting edema (Figure 1). option for PTM. However, any difference in efficacy between the external use of a glucocorticoid ointment Laboratory examination and diagnosis and intralesional glucocorticoid is unclarified. There­ The results of thyroid function showed a decreased fore, we report on a patient with PTM who underwent TSH level (0.0014 mIU/L; reference interval: 0.35­4.94 WJCC|www.wjgnet.com 855 November 26, 2018|Volume 6|Issue 14| Zhang F et al . Local glucocorticoid treatment for PTM A B C D Figure 2 Pathology of pretibial skin. A and B: The biopsy specimens showed hyperkeratosis of squamous epithelium and perivascular inflammatory cell infiltration under hematoxylin and eosin staining; C and D: Alcian blue staining showing mucin-like substances deposited between collagens and the widened gaps of the collagenous fibers (A: HE staining, × 20; B: HE staining, × 40; C: alcian blue staining, × 20; D: alcian blue staining, × 40). mIU/L), an increased FT3 level (7.27 pmol/L; reference dilute triamcinolone acetonide. The concentration of interval: 2.63­5.7 pmol/L), and a normal free thyroxine triamcinolone acetonide was 4 mg/mL. Ten injection (FT4) level (13.46 pmol/L; reference interval: 9.01­19.05 points were used along the upper border of the skin pmol/L). The titers of thyroglobulin antibody (TgAb), lesions and 0.5 mL of diluted triamcinolone was injected thyroid peroxidase antibody (TPOAb), and TRAb were at each point using a 1­mL syringe.
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