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ranging from compression stockings to intravenous immunoglobulin&#44; generating a mild to moderate response&#44; with unpleasant results in severe cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;4</span></a> The current article describes the authors&#8217; experience with intralesional corticotherapy in patient who present with the elephantiasic form&#44; noting a satisfactory and encouraging clinical response during the follow-up of over 11 months&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 47-year-old female patient has had Graves&#8217; disease since 2005&#46; Four years ago&#44; she presented with elephantiasic-like myxedema on the lower right limb&#44; with a similar condition&#44; to a lesser extent&#44; on the lower left limb one year ago&#46; The patient developed bilateral exophthalmos and severe retro-orbital impairment&#46; She was subjected to orbital decompression and iodine therapy in 2012&#44; developing hypothyroidism while maintaining clinical stability with levothyroxine use&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">On clinical examination&#44; she presented with non-depressible edema&#44; associated with nodules and yellowish-brown plaques that formed an elephantiasiform pattern on the lower right limb&#44; on the ankle and foot regions&#44; in addition to hyperpigmentation and fissures with hypertrichosis on the dorsum of the foot and phalanges&#44; and to a lesser extent&#44; non-depressible edema in the lower left limb &#40;LLL&#41; &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1 and 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The histopathological examination showed hyperortokeratosis in the epidermis&#44; an intense deposit of mucin between collagen bundles in the reticular dermis&#44; viewed in red on alcian blue coloration&#44; that was compatible with myxedema cutaneous &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">In light of confirmation of the clinical condition and the patient&#39;s desire to improve her appearance&#44; a therapeutic program was established with the use of triamcinolone acetate 20<span class="elsevierStyleHsp" style=""></span>mg&#47;mL without dilution&#44; applied over 50 points&#44; 0&#46;1<span class="elsevierStyleHsp" style=""></span>mL per point deposited through a 26G 1&#47;2 needle into the reticular dermis&#44; with the distance between two application points standardized at 1&#46;0<span class="elsevierStyleHsp" style=""></span>cm&#46; The following areas were treated&#58; dorsum and lateral region of the right foot&#44; proximal phalanx of the right first toe&#44; and the left ankle&#46; The initial frequency of the procedure was monthly&#46; After four months and based on a very satisfactory clinical response&#44; the interval between applications was increased to bimonthly&#44; with a reduction of the compound that was administered by 50&#37;&#44; maintaining a satisfactory clinical response&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">A clear decrease of non-depressible edema and nodules was observed&#44; improving the coloration of the affected skin&#44; allowing the patient to wear shoes that she was previously unable to due to her condition &#40;<a class="elsevierStyleCrossRefs" href="#fig0020">Figs&#46; 4 and 5</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">Graves&#8217; disease is triggered by the emergence of antibodies against TSH receptors&#46; Exophthalmia&#44; acropathia&#44; and pretibial myxedema are associated conditions and represent late manifestations&#44; affecting 15&#8211;50 per 100&#44;000 people per year&#44; primarily women&#46; Exophthalmia is usually present in patients with myxedema&#44; in approximately 15&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The etiology of myxedema is unknown&#44; but it is speculated that stimulation of anti-TSH receptor leads to the proliferation of fibroblasts&#44; causing an increase in hyaluronic acid and glycosaminoglycans and the consequent accumulation of fluids and compression of the small local lymphatics&#46; Other proposed causal factors are venous stasis and local trauma&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Clinically&#44; the lesions are light-colored&#44; but may also appear as yellowish-brown to reddish-brown&#46; Hyperpigmentation&#44; hyperkeratosis&#44; fissures&#44; hiperhidrosis and hypertrichosis are also present&#46; Lesions may have a dense aspect&#44; with the prominence of hair follicles&#44; generating the <span class="elsevierStyleItalic">peau d&#8217;orange</span> sign&#46; This dermopathy may be classified into four types&#58; non-depressible edema that is accompanied by changes in color&#44; plaques&#44; nodules&#44; and elephantiasiform&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;5</span></a> The most commonly affected location is the anterolateral region of the lower limb extremities&#44; possibly extending to the posterior face and feet&#46; The condition may persist for months to years&#44; and there are rare cases of spontaneous regression&#44; especially in localized conditions&#46; Elephantiasic forms are rare&#44; typically progressive and refractory to treatments&#44; leading to severe functional and emotional damage&#44;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> as presented in this report&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Treatment with topical medium- to high-potency corticosteroids under occlusion has been described&#44; generating a favorable clinical response but with a higher failure rate with regard to long-term remission&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;4</span></a> Trials with intralesional triamcinolone have reported therapeutic success and a higher percentage of complete remission in three to four years&#44; including dramatic responses&#44; without the recurrence of lesions&#44; as observed by Kumaran et al&#46; in 2015&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Several studies have combined medications&#44; such as pentoxifylline and topical or oral corticosteroid&#44; with less effective results in the cases of severe forms &#40;elephantiasiform and diffuse&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In 2015&#44; Lan et al&#46; compared the use of intralesional triamcinolone acetate in patients with several clinical forms of localized myxedema&#44; reporting satisfactory responses in all presentations&#44; corroborating the therapeutic initiative and the technique performed in the patient&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The injection technique with corticosteroids&#44; due to their anti-inflammatory and immunosuppressive properties&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> generates a significant and permanent reduction in pretibial dermal infiltrates in these patients&#44; without causing degeneration&#44; atrophy&#44; or hyperpigmentation after application&#46; In addition&#44; it is possible to maintain these results&#44;as in the case of the patient after 11 months&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Thus&#44; the present patient&#39;s therapeutic success using only intralesional corticosteroid without other adjuvant treatments was associated with a rapid clinical response and the absence of systemic side effects&#44; producing a positive emotional and social impact and encouraging the use of this technique in other patients with similar cases&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Financial support</span><p id="par0090" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Authors&#8217; contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Marina Ferreira&#58; Composition of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Luciana Helena Zacaron&#58; Approval of the final version of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Annair Freitas do Valle&#58; Approval of the final version of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">Aloisio Carlos Couri Gamonal&#58; approval of the final version of the manuscript&#59; conception and planning of the study&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0095" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Case Report
Successful therapeutic approach in a patient with elephantiasic pretibial myxedema
Marina Ferreiraa,
Corresponding author
marinaferreirajf@yahoo.com.br

Corresponding author.
, Luciana Helena Zacarona, Annair Freitas do Vallea, Aloisio Carlos Couri Gamonalb
a University Hospital, Universidade Federal de Juiz de Fora, Juiz de Fora, MG, Brazil
b Graduate and Post-Graduate Program in Dermatology, Faculdade Suprema, Juiz de Fora, MG, Brazil
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ranging from compression stockings to intravenous immunoglobulin&#44; generating a mild to moderate response&#44; with unpleasant results in severe cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;4</span></a> The current article describes the authors&#8217; experience with intralesional corticotherapy in patient who present with the elephantiasic form&#44; noting a satisfactory and encouraging clinical response during the follow-up of over 11 months&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 47-year-old female patient has had Graves&#8217; disease since 2005&#46; Four years ago&#44; she presented with elephantiasic-like myxedema on the lower right limb&#44; with a similar condition&#44; to a lesser extent&#44; on the lower left limb one year ago&#46; The patient developed bilateral exophthalmos and severe retro-orbital impairment&#46; She was subjected to orbital decompression and iodine therapy in 2012&#44; developing hypothyroidism while maintaining clinical stability with levothyroxine use&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">On clinical examination&#44; she presented with non-depressible edema&#44; associated with nodules and yellowish-brown plaques that formed an elephantiasiform pattern on the lower right limb&#44; on the ankle and foot regions&#44; in addition to hyperpigmentation and fissures with hypertrichosis on the dorsum of the foot and phalanges&#44; and to a lesser extent&#44; non-depressible edema in the lower left limb &#40;LLL&#41; &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1 and 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The histopathological examination showed hyperortokeratosis in the epidermis&#44; an intense deposit of mucin between collagen bundles in the reticular dermis&#44; viewed in red on alcian blue coloration&#44; that was compatible with myxedema cutaneous &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">In light of confirmation of the clinical condition and the patient&#39;s desire to improve her appearance&#44; a therapeutic program was established with the use of triamcinolone acetate 20<span class="elsevierStyleHsp" style=""></span>mg&#47;mL without dilution&#44; applied over 50 points&#44; 0&#46;1<span class="elsevierStyleHsp" style=""></span>mL per point deposited through a 26G 1&#47;2 needle into the reticular dermis&#44; with the distance between two application points standardized at 1&#46;0<span class="elsevierStyleHsp" style=""></span>cm&#46; The following areas were treated&#58; dorsum and lateral region of the right foot&#44; proximal phalanx of the right first toe&#44; and the left ankle&#46; The initial frequency of the procedure was monthly&#46; After four months and based on a very satisfactory clinical response&#44; the interval between applications was increased to bimonthly&#44; with a reduction of the compound that was administered by 50&#37;&#44; maintaining a satisfactory clinical response&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">A clear decrease of non-depressible edema and nodules was observed&#44; improving the coloration of the affected skin&#44; allowing the patient to wear shoes that she was previously unable to due to her condition &#40;<a class="elsevierStyleCrossRefs" href="#fig0020">Figs&#46; 4 and 5</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">Graves&#8217; disease is triggered by the emergence of antibodies against TSH receptors&#46; Exophthalmia&#44; acropathia&#44; and pretibial myxedema are associated conditions and represent late manifestations&#44; affecting 15&#8211;50 per 100&#44;000 people per year&#44; primarily women&#46; Exophthalmia is usually present in patients with myxedema&#44; in approximately 15&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The etiology of myxedema is unknown&#44; but it is speculated that stimulation of anti-TSH receptor leads to the proliferation of fibroblasts&#44; causing an increase in hyaluronic acid and glycosaminoglycans and the consequent accumulation of fluids and compression of the small local lymphatics&#46; Other proposed causal factors are venous stasis and local trauma&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Clinically&#44; the lesions are light-colored&#44; but may also appear as yellowish-brown to reddish-brown&#46; Hyperpigmentation&#44; hyperkeratosis&#44; fissures&#44; hiperhidrosis and hypertrichosis are also present&#46; Lesions may have a dense aspect&#44; with the prominence of hair follicles&#44; generating the <span class="elsevierStyleItalic">peau d&#8217;orange</span> sign&#46; This dermopathy may be classified into four types&#58; non-depressible edema that is accompanied by changes in color&#44; plaques&#44; nodules&#44; and elephantiasiform&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;5</span></a> The most commonly affected location is the anterolateral region of the lower limb extremities&#44; possibly extending to the posterior face and feet&#46; The condition may persist for months to years&#44; and there are rare cases of spontaneous regression&#44; especially in localized conditions&#46; Elephantiasic forms are rare&#44; typically progressive and refractory to treatments&#44; leading to severe functional and emotional damage&#44;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> as presented in this report&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Treatment with topical medium- to high-potency corticosteroids under occlusion has been described&#44; generating a favorable clinical response but with a higher failure rate with regard to long-term remission&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;4</span></a> Trials with intralesional triamcinolone have reported therapeutic success and a higher percentage of complete remission in three to four years&#44; including dramatic responses&#44; without the recurrence of lesions&#44; as observed by Kumaran et al&#46; in 2015&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Several studies have combined medications&#44; such as pentoxifylline and topical or oral corticosteroid&#44; with less effective results in the cases of severe forms &#40;elephantiasiform and diffuse&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In 2015&#44; Lan et al&#46; compared the use of intralesional triamcinolone acetate in patients with several clinical forms of localized myxedema&#44; reporting satisfactory responses in all presentations&#44; corroborating the therapeutic initiative and the technique performed in the patient&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">The injection technique with corticosteroids&#44; due to their anti-inflammatory and immunosuppressive properties&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> generates a significant and permanent reduction in pretibial dermal infiltrates in these patients&#44; without causing degeneration&#44; atrophy&#44; or hyperpigmentation after application&#46; In addition&#44; it is possible to maintain these results&#44;as in the case of the patient after 11 months&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Thus&#44; the present patient&#39;s therapeutic success using only intralesional corticosteroid without other adjuvant treatments was associated with a rapid clinical response and the absence of systemic side effects&#44; producing a positive emotional and social impact and encouraging the use of this technique in other patients with similar cases&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Financial support</span><p id="par0090" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Authors&#8217; contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Marina Ferreira&#58; Composition of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Luciana Helena Zacaron&#58; Approval of the final version of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Annair Freitas do Valle&#58; Approval of the final version of the manuscript&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct in the studied cases&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">Aloisio Carlos Couri Gamonal&#58; approval of the final version of the manuscript&#59; conception and planning of the study&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0095" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Localized pretibial myxedema is a dermopathy whose treatment is a challenge in dermatology&#44; occurring in 0&#46;5&#8211;4&#37; of patients with Graves&#8217; disease&#46; This autoimmune thyroid condition stimulates the production of hyaluronic acid and glycosaminoglycans that are deposited particularly in the pretibial region&#46; Clinically&#44; it presents as a localized&#44; circumscribed&#44; and non-depressible infiltrate in plaques&#46; Several treatment modalities have been proposed&#44; and their results vary&#44; with worse response observed in severe cases&#46; This report presents the case of a patient with elephantiasic pretibial myxedema who was subjected to intralesional corticosteroid applications&#44; resulting in an excellent and encouraging therapeutic response that was maintained&#46;</p></span>"
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Article information
ISSN: 03650596
Original language: English
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2023 September 90 76 166
2023 August 65 40 105
2023 July 56 27 83
2023 June 59 40 99
2023 May 53 23 76
2023 April 52 24 76
2023 March 106 48 154
2023 February 56 23 79
2023 January 67 45 112
2022 December 67 29 96
2022 November 74 57 131
2022 October 98 52 150
2022 September 57 70 127
2022 August 42 57 99
2022 July 39 60 99
2022 June 41 67 108
2022 May 41 65 106
2022 April 42 53 95
2022 March 47 68 115
2022 February 36 24 60
2022 January 59 86 145
2021 December 30 60 90
2021 November 44 46 90
2021 October 57 67 124
2021 September 36 55 91
2021 August 34 49 83
2021 July 40 52 92
2021 June 36 54 90
2021 May 55 94 149
2021 April 169 272 441
2021 March 46 61 107
2021 February 18 25 43
2021 January 20 19 39
2020 December 26 23 49
2020 November 29 20 49
2020 October 54 17 71
2020 September 40 15 55
2020 August 34 11 45
2020 July 69 16 85
2020 June 15 11 26
2020 May 8 11 19
2020 April 0 2 2
2020 March 0 2 2
2020 February 0 5 5
2020 January 0 0 0
2019 December 0 6 6
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