Imatinib is effective for prevention and improvement of fibrotic fasciitis as a manifestation of chronic GVHD

Osumi, Tomoo; Miharu, Masashi; Tanaka, Ryuma; Du, Wei; Takahashi, Tsuyoshi; Shimada, Hideaki

doi:10.1038/bmt.2011.10

Cited by 12 publications

(11 citation statements)

References 7 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…However, because of lack of details, that is, clinical characteristics, mode of diagnosis, and lack of biopsy confirmation, these cases were excluded. 4,8,[24][25][26] Similar to the majority of patients reported in the literature, 3,4,8,14,22,[24][25][26] our patient had characteristic histopathologic findings of EF with thickening, edema, fibrosis, and inflammatory cells seen primarily in the fascia and subcutaneous fat (Fig. In addition, our patient had a typical "peau d'orange" appearance of his skin with a positive groove sign ( Fig.…”

Section: Discussionsupporting

confidence: 83%

“…This included 7 individual case reports 8 There were 3 additional case series with cGVHD and fibrotic features, in which EF was reported among a few cases within each series. This included 7 individual case reports 8 There were 3 additional case series with cGVHD and fibrotic features, in which EF was reported among a few cases within each series.…”

Section: Discussionmentioning

confidence: 99%

“…1) similar to what has been reported in 18 of the 31 prior case reports. 3,4,8,13,14,[22][23][24][25][26] Only 1 patient showed complete resolution of their EF manifestations, which included swelling and tenderness of the extremities, skin changes, and peripheral eosinophilia within 2 to 4 weeks of initiating prednisone therapy. 2) There were 2 cases in the literature reporting more extensive histopathologic findings, with involvement of the upper dermis and fibrinoid degeneration of the fascia and pronounced cuffing of the epimysium, along with atrophy and necrosis of the muscle fibers.…”

Section: Discussionmentioning

confidence: 99%

“…1,[7][8][9][10][11] We report an interesting case of cGVHD presenting as EF, which was refractory to glucocorticoids and cyclosporin A. There is an emerging body of literature featuring the effectiveness of imatinib in a variety of fibrotic skin conditions including cGVHD presenting as EF.…”

mentioning

confidence: 99%

See 3 more Smart Citations

Chronic Graft-Versus-Host Disease Presenting as Eosinophilic Fasciitis

Ganta

Chatterjee²,

Pohlman³

et al. 2015

JCR Journal of Clinical Rheumatology

View full text Add to dashboard Cite

Chronic graft-versus-host disease (cGVHD) is one of the main late complications of allogeneic hematopoietic stem cell transplant and a major contributor to the mortality and morbidity in surviving recipients. Skin is the most common involved organ in cGVHD and may mimic a wide spectrum of dermatological conditions in its clinical and histopathologic manifestations. Some of the commonly simulated diseases are scleroderma, morphea, and lichen sclerosus. Chronic GVHD simulating eosinophilic fasciitis (EF) is relatively rare, frequently presenting with skin induration, a typical "peau d'orange" appearance, peripheral blood eosinophilia, myalgia, arthralgia, and arthritis leading to joint contractures in severe cases.Diagnosis is based on clinical manifestations and histopathology. Treatment is challenging because most cases are refractory to first-line therapy of glucocorticoids and calcineurin inhibitors (CNIs), and there is no standard second-line therapy.We report a comprehensive review of literature on all reported cases of CGVHD presenting as EF. We also describe an additional interesting case of cGVHD presenting as EF that was resistant to traditional therapy of high-dose glucocorticoids and cyclosporin A, but showed complete resolution of skin manifestations after addition of imatinib.Chronic GVHD presenting as EF is a rare variant of sclerodermatous cGVHD. Diagnosis is difficult, and treatment of cGVHD mimicking EF remains a therapeutic challenge because of obscure pathogenesis and poor response to traditional immunosuppressive medications. Emerging insights into the pathogenesis of cGVHD have resulted in the development of novel targeted therapies, which may improve outcomes and should be attempted in this subset of the disease. Larger studies are warranted to substantiate these preliminary findings.

show abstract

Section: Discussionsupporting

confidence: 83%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

See 2 more Smart Citations

Chronic Graft-Versus-Host Disease Presenting as Eosinophilic Fasciitis

Ganta

Chatterjee²,

Pohlman³

et al. 2015

JCR Journal of Clinical Rheumatology

View full text Add to dashboard Cite

show abstract

“…Binding of agonistic autoantibodies to platelet-derived growth factor receptor can cause cutaneous fibrosis in patients with scleroderma or cGVHD, 47,48 although clinical trials testing the use of tyrosine kinase inhibitors to block the downstream signaling pathway mediated through platelet-derived growth factor receptor have not produced consistent results in patients with cGVHD. [49][50][51][52][53] The differences among these trials may be explained by variation in the spectrum of pathogenic autoantibodies or by antibody-independent mechanisms. Further studies will be needed to identify the mechanisms that account for antibody-mediated thymic and lymphoid tissue damage and cutaneous sclerosis in cGVHD.…”

Section: Discussionmentioning

confidence: 99%