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2013 ; 92
(2
): 69-81
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Severe aplastic anemia associated with eosinophilic fasciitis: report of 4 cases
and review of the literature
#MMPMID23429351
de Masson A
; Bouaziz JD
; de Latour RP
; Benhamou Y
; Moluçon-Chabrot C
; Bay JO
; Laquerrière A
; Picquenot JM
; Michonneau D
; Leguy-Seguin V
; Rybojad M
; Bonnotte B
; Jardin F
; Lévesque H
; Bagot M
; Socié G
Medicine (Baltimore)
2013[Mar]; 92
(2
): 69-81
PMID23429351
show ga
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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