Autologous hematopoietic stem cell transplantation for refractory antiphospholipid syndrome causing myocardial necrosis

Hashimoto, Naoaki; Iwasaki, Tsuyoshi; Sekiguchi, Mutsuo; Takatsuka, Hiroyuki; Okamoto, Takahiro; Hashimoto, Takashi; Sano, Hajime

doi:10.1038/sj.bmt.1704432

Cited by 17 publications

(5 citation statements)

References 18 publications

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“…One of the patients with refractory disease (patient 10) died of septic shock after receiving a conditioning regimen for autologous hematopoietic stem cell transplantation (HSCT) attempted at another institution. Most published data on HSCT in APS are derived from subsets of patients with SLE (). In an open‐label trial of 22 patients with SLE and APS, 18 patients receiving long‐term anticoagulation therapy prior to autologous HSCT were able to discontinue long‐term anticoagulation therapy, with 78% of the patients remaining free of thrombosis at a median followup of 15 months ().…”

Section: Discussionmentioning

confidence: 99%

Primary Antiphospholipid Syndrome–Associated Diffuse Alveolar Hemorrhage

Cartin‐Ceba

Peikert

Ashrani

et al. 2014

Arthritis Care & Research

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Objective. Diffuse alveolar hemorrhage (DAH) is an uncommon complication of primary antiphospholipid syndrome (APS). We aimed to describe the clinical characteristics, treatment, and outcomes of primary APS-associated DAH in a single center. Methods. We conducted a retrospective review of all adults with primary APS-associated DAH evaluated at Mayo Clinic over a 15-year period. DAH was defined as bilateral pulmonary infiltrates and bronchoalveolar lavage (BAL) fluid documenting progressively bloody returns and/or the presence of >20% hemosiderin-laden macrophages. Patients with other causes of DAH were excluded. Results. Eighteen patients were identified (median age 43 years). Capillaritis was present in surgical lung biopsy samples of 3 patients. BAL differential cell counts revealed predominantly neutrophils. All patients were treated initially with glucocorticoids. Cyclophosphamide (CYC) was used in 8 patients; complete remission was achieved in 3 patients treated with CYC alone and in 1 patient receiving combination therapy with rituximab (RTX). RTX was used in 9 patients; 2 patients achieved remission with RTX alone, whereas 3 patients required combination therapy with CYC or mycophenolate mofetil (MMF). No patient achieved complete remission while receiving single therapy with MMF, azathioprine, or plasma exchange. Intravenous gamma globulin therapy was administered in 5 patients; no patient achieved control of the disease. Six patients died, all because of complications related to uncontrolled DAH or its therapy. Conclusion. We present the largest case series of primary APS-associated DAH reported in the literature. DAH carries a very poor prognosis and therapeutic options are limited. Immunosuppression with either CYC or RTX is associated with the highest likelihood of remission induction and should be considered early.

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Section: Discussionmentioning

confidence: 99%

Primary Antiphospholipid Syndrome–Associated Diffuse Alveolar Hemorrhage

Cartin‐Ceba

Peikert

Ashrani

et al. 2014

Arthritis Care & Research

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“…The study selection process is reported in Figure 1. The use of allogenic/auto-HCT as rescue therapy for SLE/SAPS was assessed using data from 2 prospective [15,36], 10 retrospective studies [16][17][18][19][20][21][22][23]34]and 13 case reports [24][25][26][27][28][29][30][31][32][33]35,[37][38][39], for a total of 279 SLE patients. Of those, 54 patients also fulfilled the criteria of APS.…”

Section: Search Results and Characteristics Of Included Studiesmentioning

confidence: 99%