Eosinophilic fasciitis and aplastic anaemia

Narayanan, Mechu; Yin, Jun; Love, Elyse; Geary, C. G.; Holt, P. J.; Freemont, A. J.; Feinmann, R.

doi:10.1111/j.1365-2257.1988.tb01197.x

Cited by 9 publications

(13 citation statements)

References 3 publications

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“…We selected 19 patients from 15 different articles published between 1978 and 2009 in the English, French, German, and Portuguese literature. 2 , 13 , 22 , 24 , 30 , 38 , 57 , 58 , 73 , 77 , 89 , 95 , 111 , 117 , 128 Patients were selected if they displayed clinical features of EF and pancytopenia and if AA was confirmed by bone marrow examination. The diagnosis of EF was confirmed by a deep skin biopsy, including the fascia, in all but 2 cases.…”

Section: Methodsmentioning

confidence: 99%

“…EF is sometimes associated with hematologic diseases, particularly with aplastic anemia (AA) (n = 19), 2 , 13 , 15 , 22 , 24 , 30 , 33 , 38 , 57 , 58 , 73 , 77 , 89 , 95 , 111 , 117 , 128 but also with T-cell lymphoma (n = 5), 27 , 36 , 65 , 72 , 83 cutaneous T-cell lymphoma (n = 1), 25 Hodgkin disease (n = 3), 84 , 90 , 98 myeloproliferative syndromes (n = 3), 61 , 75 , 85 myelomonocytic leukemia (n = 2), 75 , 85 chronic lymphocytic leukemia (n = 2), 12 , 75 multiple myeloma (n = 1), 68 and myeloblastic leukemia (n = 1), 90 and, less commonly, with solid tumors such as breast cancer (n = 5), 12 , 90 , 109 , 127 choroidal melanoma (n = 1), 125 colorectal cancer (n = 1), 94 and prostate cancer (n = 1). 90 Diffuse EF has also been reported in association with autoimmune disorders, such as Hashimoto thyroiditis (n = 6), 2 , 5 , 13 , 59 , 114 systemic lupus erythematosus (n = 4), 6 , 43 , 45 , 74 Crohn disease (n = 1), 82 Graves disease (n = 1), 114 glomerulonephritis (n = 1), 63 rheumatoid arthritis (n = 1), 81 type 1 diabetes (n = 1), 46 and autoimmune cytopenias, including autoimmune hemolytic anemia (n = 2), 5 , 44 immune thrombocytopenic purpura (n = 2), 5 , 111 amegakaryocytic thrombocyt...…”

Section: Introductionmentioning

confidence: 99%

“…It is still uncertain whether AA associated with EF is an autoimmune disease and/or the initial manifestation of an evolving clonal myeloid disorder. Among the 19 reported patients with EF and associated AA, 2 , 13 , 15 , 22 , 24 , 30 , 33 , 38 , 57 , 58 , 73 , 77 , 89 , 95 , 111 , 117 , 128 8 died of complications from AA. Although most of these deaths occurred in patients receiving corticosteroids and/or antithymocyte globulin (ATG)-based regimens (without cyclosporine A [CsA]) in the 1980s, the current conventional immunosuppressive therapy of ATG and CsA was ineffective in 3 of 6 (50%) cases.…”

Section: Introductionmentioning

confidence: 99%

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Severe Aplastic Anemia Associated With Eosinophilic Fasciitis

Masson

Bouaziz²,

Latour

et al. 2013

Medicine

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Diffuse eosinophilic fasciitis (Shulman disease) is a rare sclerodermiform syndrome that, in most cases, resolves spontaneously or after corticosteroid therapy. It has been associated with hematologic disorders, such as aplastic anemia. The clinical features and long-term outcomes of patients with eosinophilic fasciitis and associated aplastic anemia have been poorly described. We report the cases of 4 patients with eosinophilic fasciitis and associated severe aplastic anemia. For 3 of these patients, aplastic anemia was refractory to conventional immunosuppressive therapy with antithymocyte globulin and cyclosporine. One of the patients received rituximab as a second-line therapy with significant efficacy for both the skin and hematologic symptoms. To our knowledge, this report is the first to describe rituximab used to treat eosinophilic fasciitis with associated aplastic anemia.In a literature review, we identified 19 additional cases of eosinophilic fasciitis and aplastic anemia. Compared to patients with isolated eosinophilic fasciitis, patients with eosinophilic fasciitis and associated aplastic anemia were more likely to be men (70%) and older (mean age, 56 yr; range, 18–71 yr). Corticosteroid-containing regimens improved skin symptoms in 5 (42%) of 12 cases but were ineffective in the treatment of associated aplastic anemia in all but 1 case. Aplastic anemia was profound in 13 cases (57%) and was the cause of death in 8 cases (35%). Only 5 patients (22%) achieved long-term remission (allogeneic hematopoietic stem cell transplantation: n = 2; cyclosporine-containing regimen: n = 2; high-dose corticosteroid-based regimen: n = 1).

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Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Severe Aplastic Anemia Associated With Eosinophilic Fasciitis

Masson

Bouaziz²,

Latour

et al. 2013

Medicine

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“…Pediatric disease, however, has also been documented 3 . EF has been frequently associated with hematological disorders such as aplastic anemia, hemolytic anemia, peripheral autoimmune thrombocytopenia, pernicious anemia, leukemias, and lymphomas 4,5 . Some autoimmune diseases such as Hashimoto's thyroiditis, morphea, systemic sclerosis, Sjogren's syndrome and antiphospholipid antibody syndrome have also been reported in patients with EF 6 .…”

Section: Discussionmentioning

confidence: 99%

Two Cases of Eosinophilic Fasciitis

et al. 2011

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“…Eosinophilic fasciitis has been frequently reported in association with hematological disorders, such as aplastic anemia, hemolytic anemia, peripheral autoimmune thrombocytopenia, pernicious anemia, leukemias, and lymphomas. 2,3 An association with malignancies such as mammary carcinoma and prostatic carcinoma has also been described. Some autoimmune diseases like Hashimoto's thyroiditis, morphea, systemic sclerosis, Sjögren's syndrome and antiphospholipid antibody syndrome 4 have also been described in patients with EF.…”

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confidence: 99%