Evans Syndrome After Unrelated Cord Blood Transplantation for Disseminated Langerhans Cell Histiocytosis in a Child

Chen, Rong-Long; Wu, Pei-Ling; Hsu, Yung‐Hsiang; Kuo, Pao‐Lin

doi:10.1097/mph.0b013e3180556467

Cited by 10 publications

(5 citation statements)

References 11 publications

(12 reference statements)

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“…Most were organ-specific ADs such as cytopenias followed by ADs of the thyroid and a few cases of miscellaneous multisystemic AD. The diagnoses of ADs in our cohort are comparable to those after CBT reported in the literature [6][7][8][9][10][11][12] and to ADs after allogeneic HSCT. 1,14 AIHA, ITP, and, more rarely, autoimmune neutropenia have been reported after allogeneic, noncord-blood-derived SCT with an incidence of 3%-4.5% in 3 single-center studies [14][15][16] The published cases have been reviewed recently.…”

Section: Discussionsupporting

confidence: 75%

“…Reports of ADs occurring after CBT are scarce and based on single case reports and small series, the largest including 10 patients. 6 The most prevalent ADs reported are AIHA, [6][7] Evans syndrome, 8 ITP, 6,9 thyroiditis, 10 and bullous dermatoses (pemphigus and pemphigoid). [11][12] This study was undertaken to characterize the nature, incidence, and risk factors for ADs developing after CBT in a large patient population.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

New autoimmune diseases after cord blood transplantation: a retrospective study of EUROCORD and the Autoimmune Disease Working Party of the European Group for Blood and Marrow Transplantation

Daikeler

Labopin

Ruggeri

et al. 2013

Blood

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Key Points• Autoimmune diseases do occur after CBT in approximately 5% of patients.• Of these, AIHA or ITP were observed the most often and were treated with prednisone, CSA, and RTX.To describe the incidence, risk factors, and treatment of autoimmune diseases (ADs) occurring after cord blood transplantation (CBT), we analyzed both CBT recipients reported to EUROCORD who had developed at least 1 new AD and those who had not. Fifty-two of 726 reported patients developed at least 1 AD within 212 days (range, 27-4267) after CBT. Cumulative incidence of ADs after CBT was 5.0% ؎ 1% at 1 year and 6.6% ؎ 1% at 5 years. Patients developing ADs were younger and had more nonmalignant diseases (P < .001). ADs target hematopoietic (autoimmune hemolytic anemia, n ‫؍‬ 20; Evans syndrome, n ‫؍‬ 9; autoimmune thrombocytopenia, n ‫؍‬ 11; and immune neutropenia, n ‫؍‬ 1) and other tissues (thyroiditis, n ‫؍‬ 3; psoriasis, n ‫؍‬ 2; Graves disease, n ‫؍‬ 1; membranous glomerulonephritis, n ‫؍‬ 2; rheumatoid arthritis, n ‫؍‬ 1; ulcerative colitis, n ‫؍‬ 1; and systemic lupus erythematosus, n ‫؍‬ 1). Four patients developed 2 ADs (3 cases of immune thrombocytopenia followed by autoimmune hemolytic anemia and 1 Evans syndrome with rheumatoid arthritis). By multivariate analysis, the main risk factor for developing an AD was nonmalignant disease as an indication for CBT (P ‫؍‬ .0001). Hematologic ADs were most often treated with steroids, rituximab, and cyclosporine. With a median follow-up of 26 months (range, 2-91), 6 of 52 patients died as a consequence of ADs. We conclude that CBT may be followed by potentially life-threatening, mainly hematologic ADs. (Blood. 2013;121(6):1059-1064)

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Section: Discussionsupporting

confidence: 75%

Section: Introductionmentioning

confidence: 99%

New autoimmune diseases after cord blood transplantation: a retrospective study of EUROCORD and the Autoimmune Disease Working Party of the European Group for Blood and Marrow Transplantation

Daikeler

Labopin

Ruggeri

et al. 2013

Blood

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“…Until now, four LCH patients who received CBT have been reported, and their characteristics and clinical course are shown in Table 1. All four, in addition to hematological dysfunction, were heavily pretreated prior to SCT, and were alive without recurrence of LCH at the time of reporting (11, 12, 17, 20, 21). They were all diagnosed with LCH under one yr old, and cord blood units containing a large number of nucleated cells could be infused, which achieved engraftment and complete donor chimerism.…”

Section: Discussionmentioning

confidence: 99%

Successful treatment of refractory Langerhans cell histiocytosis with pulmonary aspergillosis by reduced-intensity conditioning cord blood transplantation

Hatakeyama

Hori

Yamamoto

et al. 2010

Pediatric Transplantation

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The prognosis of multisystem LCH in children with risk organ involvement is extremely poor when they fail to respond to conventional chemotherapy. In such patients, allogeneic SCT may produce complete and sustained remission; however, high-dose myeloablative regimens are frequently associated with treatment-related morbidity and mortality. More recently, allogeneic SCT following an RIC regimen has been performed as an alternative salvage approach. We describe a nine-month-old boy with refractory multisystem LCH with pulmonary aspergillosis who was successfully treated with reduced-intensity cord blood transplantation.

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“…4,5 These disorders have been recently described among pediatric and adult patients undergoing CB transplantation (CBT), in several case reports or a cohort of patients with malignant and non-malignant diseases. [5][6][7] Studies of AIHA occurring after CBT are uncommon and based on case reports or small series, probably associated with graft-versus-host disease (GvHD). 8 These hematologic autoimmune disorders may be related to the use of unrelated donors and development of GvHD, possibly reflecting a dysregulation of the immune system.…”

Section: Introductionmentioning

confidence: 99%

Autoimmune Hemolytic Anemia After Cord Blood Transplantation: A Retrospective Single-Center Experience

Yuan

Liang

Dong

et al. 2023

JIR

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Objective To describe the incidence, possible risk factors, and treatment options of autoimmune hemolytic anemia (AIHA) occurring after cord blood transplantation (CBT). Methods We retrospectively analyzed the patients who underwent CBT at Peking University First Hospital between January 2004 and July 2022. Results We totally identified thirty-six patients who received CBT. Median age was 27.5 years (range, 1.6–52). With a median 6 (range 0.6–10.0) years survivor follow-up, six patients developed AIHA (2 Evans syndrome included) at a median of 168 (range, 122–264) days post-CBT for 8% cumulative incidence density 3 years. Its mortality was 50% and mainly associated with concomitant infections (CMV reactivation rate nearly 100%). The possible risk factors for developing AIHA are CMV reactivation, GvHD and HLA mismatch. Conclusion AIHA is a clinically significant common complication in recipients post-CBT. Corticosteroids combined with intravenous immunoglobulin (IvIg) is recommended for the treatment of warm antibody AIHA after CBT.

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Evans Syndrome After Unrelated Cord Blood Transplantation for Disseminated Langerhans Cell Histiocytosis in a Child

Cited by 10 publications

References 11 publications

New autoimmune diseases after cord blood transplantation: a retrospective study of EUROCORD and the Autoimmune Disease Working Party of the European Group for Blood and Marrow Transplantation

New autoimmune diseases after cord blood transplantation: a retrospective study of EUROCORD and the Autoimmune Disease Working Party of the European Group for Blood and Marrow Transplantation

Successful treatment of refractory Langerhans cell histiocytosis with pulmonary aspergillosis by reduced-intensity conditioning cord blood transplantation

Autoimmune Hemolytic Anemia After Cord Blood Transplantation: A Retrospective Single-Center Experience

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