Chronic Cutaneous Graft-versus-Host Disease Manifesting as Calcinosis Cutis Universalis on a Background of Widespread Sclerodermatoid Changes

Man, Jeremy; Kalisiak, Michal S.; Birchall, I. W. J.; Salopek, Thomas G.

doi:10.2310/7750.2010.09034

Cited by 9 publications

(6 citation statements)

References 17 publications

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“…Sclerotic-type chronic skin graft-versus-host disease shares many clinical features with SS, suggesting a common pathway leading to calcinosis. The 7 cases described herein are similar to 2 previously reported cases in the literature, including a long latency period (12 and 14 years) and antecedent ScGVHD . The lower extremities are a common site of skin sclerosis in ScGVHD and were the most common location for calcinosis in these patients.…”

Section: Discussionsupporting

confidence: 83%

“…Systemic sclerosis and sclerotic-type chronic skin graft-versus-host disease (ScGVHD) share several clinical and histologic features; however, high-titer antibodies specifically associated with SS are not typically found in ScGVHD, and the fibrotic features in ScGVHD more closely resemble morphea and eosinophilic fasciitis . Whereas calcinosis is well described in SS, there are only 2 reports of calcinosis in the setting of chronic graft-versus-host disease (cGVHD), to our knowledge . We sought to identify patients with cGVHD with calcinosis to explore predisposing factors and clinical features.…”

mentioning

confidence: 99%

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Calcinosis Cutis in the Setting of Chronic Skin Graft-Versus-Host Disease

et al. 2020

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cluded. One patient who did not adhere to topical corticosteroid therapy and 3 patients with less than 5 years of follow-up (or loss to follow-up) were excluded. In the remaining 11 patients, mean age at diagnosis of primary lesion was 62.9 years (range, 26-88 years). Initial management of vSCC or dVIN included surgical excision alone in 4 patients, surgical excision with node biopsy in 2 patients, surgical excision with node dissection in 4 patients, and surgical excision with adjuvant laser therapy in 1 patient. Of 11 patients, 8 (73%) remained free of recurrence with a mean follow-up of 10.5 years (range, 5.1-16.5 years) (Table ). Two patients (18%) had recurrence of their vSCC, 1 of whom developed multiple recurrences of vSCC, and 1 patient (9%) had recurrence of their dVIN.Discussion | In the study cohort, patients with vLS who adhered to topical corticosteroid therapy had a vSCC or VIN recurrence rate of 27% compared with reported 5-year recurrence rates of 44% to 47%. 4,5 Only 1 patient with a recurrence of vSCC or dVIN had a subsequent recurrence compared with the 5-year subsequent recurrence rate of 80% for untreated vLS. 5 It has been hypothesized that chronic inflammation contributes to the increased risk of developing vSCC; thus, the antiinflammatory effect of topical corticosteroid may confer a protective benefit. 6 Topical corticosteroid treatment is inexpensive and safe 3 and could result in considerable cost savings and reductions in morbidity and mortality.A limitation of the present study is that it is a singlecenter retrospective medical record review with a small sample size; there was no control group (patients not treated with a topical corticosteroid) for comparison. A strength of the study is that all included patients were part of a large cohort with vLS who have been followed up prospectively since 2008. Larger prospective studies including a control group for comparison would strengthen the evidence of the findings in this report.Topical corticosteroid therapy may represent an important management strategy to reduce the recurrence rate of vSCC and dVIN in patients with vLS. These findings suggest the need for a large prospective trial. However, in the meantime, we encourage dermatologists to play an active role in assisting colleagues who specialize in gynecology-oncology to use a topical corticosteroid when treating patients with vLS.

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Section: Discussionsupporting

confidence: 83%

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confidence: 99%

Calcinosis Cutis in the Setting of Chronic Skin Graft-Versus-Host Disease

et al. 2020

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“…Calcinosis cutis associated with sclerodermoid GVHD remains an interesting yet rarely discussed topic in the literature (Table 1) [9,10]. In the two previously published cases and in our patient, the patient age was greater than 50 years, serum calcium was normal, the time interval between the initial diagnosis of sclerodermoid GVHD and the development of calcinosis cutis was greater than 10 years (average 11.5 years), and the aetiology of calcification was thought to be dystrophic, related to chronic sclerodermoid change.…”

Section: Discussionmentioning

confidence: 99%

Calcinosis Cutis Associated with Chronic Sclerodermoid Graft versus Host Disease: A Case and Review of the Literature

Deen

Byrom

Robertson

2020

Case Reports in Dermatological Medicine

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We present a rare case of calcinosis cutis associated with chronic sclerodermoid graft versus host disease in a 59-year-old male, 13 years following allogenic bone marrow transplantation. e etiology of calcification was thought to be dystrophic. Further research is needed to understand the link between calcinosis cutis and chronic sclerodermoid graft versus host disease to assist with selecting appropriate management for these patients.

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“…Additional signs and symptoms of evolving sclerotic involvement include widespread calcinosis, 91 edema of the affected extremity, muscle weakness, pain, and cramping. 92 It may be clinically difficult to differentiate edema associated with sclerotic skin involvement from drug-induced causes (eg, sirolimus, imatinib, or gabapentin), fluid overload, and deep venous thrombosis in patients with indwelling lines.…”

Section: Sclerotic Manifestations Of Chronic Gvhdmentioning

confidence: 98%