Lack of response to intravenous sodium thiosulfate in three cases of extensive connective tissue disease-associated calcinosis cutis

Song, Philip; Fett, Nicole; Lin, Jianguo; Merola, Joseph F.; Costner, Melissa; Vleugels, Ruth Ann

doi:10.1111/bjd.15783

Cited by 26 publications

(15 citation statements)

References 8 publications

(10 reference statements)

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“…Historically, treatment of calcinosis in JDM has posed a challenge, and spontaneous resolution is rare. Intravenous STS has proven useful when used in cases of calciphylaxis but offers less clinical resolution in connective tissue‐associated CC . The use of topical STS in cases of CC has been reported as efficacious but reports of its use in JDM with CC are infrequent .…”

Section: Discussionmentioning

confidence: 99%

Linear amyopathic dermatomyositis with calcinosis cutis responsive to topical sodium thiosulfate

Topham

Haynes

Frigerio

et al. 2019

Pediatric Dermatology

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A case of a 15-year-old male patient with a 3-year history of linear, segmental amyopathic dermatomyositis with calcinosis cutis is presented.The calcinosis was recalcitrant to treatment with topical steroids and hydroxychloroquine. Topical 10% sodium thiosulfate use for 8 weeks resulted in improvement. The use of topical sodium thiosulfate for patients in whom surgical extraction is not an option is detailed. | e103Pediatric Dermatology BRIEF REPORT | D ISCUSS I ONLinear lesions in children can invoke a relatively broad differential, with common aetiologies including lichen striatus, lichen planus, psoriasis, contact dermatitis, and morphea. The lesions presented in this case proved to be particularly challenging diagnostically given the relatively infrequent incidence of linear variant juvenile dermatomyositis (JDM). Indeed, only a handful of cases have been reported to date. 1,2 Furthermore, although certain features of this case, such as the presence of photosensitivity and possible Gottron's papules, were suggestive of JDM, the patient's unremarkable laboratory studies, as well as absence of nail fold involvement or myalgias, complicated the clinical picture. Ultimately, it was the histologic presence of calcinosis that suggested JDM, as calcinosis is common in JDM. 3 The association of calcinosis with the linear variant of JDM is less well known, with only a few reports to date. 2,4,5 We suspect that the patient suffered from localized rather than systemic JDM, which would complement the inconsistencies in his presentation. Other possible diagnoses were linear lichen striatus and morphea. Lack of prior case reports describing CC with lichen striatus made this explanation less favorable, while the absence of sclerosis pointed away from the diagnosis of linear morphea.Historically, treatment of calcinosis in JDM has posed a challenge, and spontaneous resolution is rare. Intravenous STS has proven useful when used in cases of calciphylaxis but offers less clinical resolution in connective tissue-associated CC. 6 The use of topical STS in cases of CC has been reported as efficacious 7,8 but reports of its use in JDM with CC are infrequent. [8][9][10][11] The mechanism of calcinosis destruction with STS involves dissolution of calcium depositions, calcium chelation, as well as prevention of further calcium precipitation. 7,12 STS also acts as a potent vasodilator as well as an antioxidant, leading to quick resolution of CC-associated pain. 9,12 In conclusion, we present a case of linear, segmental JDM with dystrophic CC that was clinically responsive to topical STS. JDM is frequently associated with CC, and therefore, an effective therapy for the calcinosis is needed. We propose topical STS as an effective, alternative therapy for patients in whom surgical extraction is not an option.

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Section: Discussionmentioning

confidence: 99%

Linear amyopathic dermatomyositis with calcinosis cutis responsive to topical sodium thiosulfate

Topham

Haynes

Frigerio

et al. 2019

Pediatric Dermatology

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“…Intravenous administration of sodium thiosulfate may subject patients to potential adverse effects, including fatigue, hypotension, gastrointestinal upset, and anion gap metabolic acidosis. 10,11,18,19 Although it may have fewer associated systemic adverse effects, topical sodium thiosulfate may not penetrate the skin adequately enough to dissolve deep calcifications, and patients must strictly adhere to the application directions. 13,20 Intralesional injection of sodium thiosulfate has the advantages of localized administration and reduction of unwanted systemic adverse effects while maintaining accessibility to deep lesions.…”

Section: Introductionmentioning

confidence: 99%

Efficacy of intralesional sodium thiosulfate for the treatment of dystrophic calcinosis cutis: A double-blind, placebo-controlled pilot study

et al. 2020

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Background: Intralesional injection of sodium thiosulfate has emerged as a promising therapy for calcinosis cutis, but to our knowledge there are no randomized controlled trials evaluating its efficacy as a treatment. Objective: Conduct a prospective, double-blinded investigation of intralesional sodium thiosulfate versus normal saline in the treatment of dystrophic calcinosis cutis. Methods: This prospective pilot study injected normal saline or sodium thiosulfate at 0.1 mL/cm 2 into lesions at baseline and at 1-and 2-month follow-up. Subjects were followed for a total of 12 weeks. An inperson Physician Global Assessment score was assigned by the injecting physician at each visit and was repeated by an independent observer. Results: Of 4 subjects who completed the study, only 1 experienced improvement in the size and Physician Global Assessment score of the lesion. By 3-month follow-up, there was no difference between the average size of the treatment and control lesions (P = .39). Limitations: This was a small single-center study with limited demographic diversity and a short follow-up period. Only dystrophic calcinosis cutis subjects were included, and subjects received only 3 monthly injections of sodium thiosulfate. Conclusions: With only 1 positive response, our results highlight the need for further study of sodium thiosulfate treatment for dystrophic calcinosis.

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“…The efficacy of this inorganic salt for calcium‐mediated disorders may be multifactorial, possibly including its anti‐inflammatory and antioxidant properties, and its ability to chelate calcium into calcium thiosulfate salts, thereby increasing the solubility of calcium . However, in one report, three patients with extensive and longstanding CC associated with ACTD who were treated with intravenous sodium thiosulfate had no improvement …”

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confidence: 99%

Topical sodium thiosulfate for calcinosis cutis associated with autoimmune connective tissue diseases: the Mayo Clinic experience, 2012–2017

Ernste

Davis

et al. 2018

Clin Exp Dermatol

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Summary In this case series, we retrospectively identified all patients treated with topical sodium thiosulfate (TST) for calcinosis cutis (CC) associated with underlying autoimmune connective tissue diseases at Mayo Clinic (Rochester, MN, USA) during the period 1 January 2012 to 27 June 2017. Of 28 patients identified (mean age 57.0 years; 96% female), 19 (68%) had clinical improvement of their CC with TST, 7 (25%) had no response and 2 (7%) had unknown response. There were adverse events in three patients: two had skin irritation and the third, who had a zinc allergy, experienced pain with application. Overall, our findings support those of previous case reports that TST appears to be a relatively well‐tolerated adjuvant treatment for CC, although future studies with a control group are warranted to assess the true efficacy of TST for the indication of CC.

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Lack of response to intravenous sodium thiosulfate in three cases of extensive connective tissue disease-associated calcinosis cutis

Cited by 26 publications

References 8 publications

Linear amyopathic dermatomyositis with calcinosis cutis responsive to topical sodium thiosulfate

Linear amyopathic dermatomyositis with calcinosis cutis responsive to topical sodium thiosulfate

Efficacy of intralesional sodium thiosulfate for the treatment of dystrophic calcinosis cutis: A double-blind, placebo-controlled pilot study

Topical sodium thiosulfate for calcinosis cutis associated with autoimmune connective tissue diseases: the Mayo Clinic experience, 2012–2017

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