Onset and outcome of pregnancy after autologous haematopoietic SCT (AHSCT) for autoimmune diseases: a retrospective study of the EBMT autoimmune diseases working party (ADWP)

Snarski, Emilian; Snowden, John A.; Oliveira, Maria Carolina; Simões, Belinda Pinto; Badoglio, Manuela; Carlson, Kristina; Burman, Joachim; Moore, John J.; Rovira, Montserrat; Clark, R E; Sáiz, Albert; Hadj‐Khelifa, S.; Tan, Jeffrey; Crescimanno, Grazia; Musso, Maurizio; Martin, Thierry; Farge, Dominique

doi:10.1038/bmt.2014.248

Cited by 46 publications

(35 citation statements)

References 11 publications

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“…A small study of patients who underwent low-intensity or intermediate-intensity regimens demonstrated recovery of the menstrual cycle in all patients who were aged ≤32 years, but recovery of menstruation after treatment was reported up to age 41 years 66 . Permanent infertility is a risk, but in a retrospective study of 324 women who underwent HSC transplantation for autoimmune disease, 15 pregnancies were reported, and no congenital, developmental or other disease was reported in the children 67 . Patients with MS who undergo AHSCT can experience disease-specific adverse effects, such as frequent urinary tract infection, the Uhtoff phenomenon, limb spasticity and reduced mobility, all of which can increase the risk of complications and require expert management 68 .…”

Section: [H2] Safety and Tolerabilitymentioning

confidence: 99%

Autologous haematopoietic stem cell transplantation for treatment of multiple sclerosis

et al. 2017

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Autologous haematopoietic stem cell transplantation (AHSCT) is a multistep procedure that enables destruction of the immune system and its reconstitution from haematopoietic stem cells. Originally developed for the treatment of haematological malignancies, the procedure has been adapted for the treatment of severe immune-mediated disorders. Results from ~20 years of research make a compelling case for selective use of AHSCT in patients with highly active multiple sclerosis (MS), and for controlled trials. Immunological studies support the notion that AHSCT causes qualitative immune resetting, and have provided insight into the mechanisms that might underlie the powerful treatment effects that last well beyond recovery of immune cell numbers. Indeed, studies have demonstrated that AHSCT can entirely suppress MS disease activity for 4–5 years in 70–80% of patients, a rate that is higher than those achieved with any other therapies for MS. Treatment-related mortality, which was 3.6% in studies before 2005, has decreased to 0.3% in studies since 2005. Current evidence indicates that the patients who are most likely to benefit from and tolerate AHSCT are young, ambulatory and have inflammatory MS activity. Clinical trials are required to rigorously test the efficacy, safety and cost-effectiveness of AHSCT against highly active MS drugs

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Section: [H2] Safety and Tolerabilitymentioning

confidence: 99%

Autologous haematopoietic stem cell transplantation for treatment of multiple sclerosis

et al. 2017

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“…6 Although our study involved a small pool of patients, the results provide an interesting observation that gives a new perspective to the previously reported 4.6% of patients with pregnancies after AHSCT treatment for autoimmune disease (who were a part of the European-wide European Blood and Marrow Transplantation study). 7 A significant number of responders in our study had already fulfilled their reproductive needs and did not wish to become pregnant again, with 73% of women having children before AHSCT. Therefore, the percentage of pregnancies in the entire population may not be as important as percentage of pregnancies among the women who declare pregnancy desire, and this should be explored in further studies.…”

Section: Key Resultsmentioning

confidence: 98%

“…This rate is probably one of the highest for any type of hematopoietic stem cell transplant patient group. 7 With all points considered, this study provides helpful guidance to practitioners counselling patients in making an informed decision before undergoing AHSCT for autoimmune diseases. Such an online questionnaire could be an easy tool for data gathering and follow-up of patients after treatment for rare diseases, especially in outpatient units as part of longterm follow-up.…”

Section: Discussionmentioning

confidence: 99%

“…Well-established risk factors of gonadal malfunction after hematopoietic stem cell transplant for traditional indications are age at transplant > 30 years, postpuberty status at transplant, male sex, use of radiotherapy, multiple doses of chemotherapy before hematopoietic stem cell transplant, inclusion of alkylating agents in the conditioning regimen before transplant, and type of transplant (allogeneic > autologous). 6 7 Studies that include larger populations can be difficult to perform due to the need to coordinate between many international treatment centers with few patients scattered among them and the requirement to monitor patients over an extended period after treatment completion.…”

Section: Introductionmentioning

confidence: 99%

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Maciejewska

Snarski²,

Wiktor-Jedrzejczak³

2016

Exp Clin Transplant

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Objectives: Autologous hematopoietic stem cell transplant has become a successful treatment option in aggressive autoimmune diseases. Young women who are consulted before this treatment are faced with an absence of data on the transplant's effect on their fertility. The aim of our study was to analyze information on menstruation recovery after auto logous hema topoietic stem cell transplant for autoimmune diseases. Materials and Methods: An anonymous online questionnaire was distributed among members of an international Web forum of patients who had autologous hematopoietic stem cell transplant for autoimmune disease. Data were collated and analyzed. Results: We obtained responses from 28 female patients to the questionnaire. The conditioning regimens used in this population were cyclophosphamide ± antibodies or BCNU (carmustine), etoposide, Ara-C (cytarabine), and melphalan chemotherapy. All patients who were transplanted at the age of 32 years or younger showed restored menstruation after an average (standard deviation) of 5.38 months (5.34 mo). In patients aged 33 to 41 years, menstruation was restored in 38%. We found that 73% of patients already had children before autologous hematopoietic stem cell transplant, and only 15% of responders declared desire for pregnancy after transplant. Conclusions: Our online analysis showed feasibility as a questionnaire for assessing fertility after autologous hematopoietic stem cell transplant. The results show that menstruation is restored in all patients of 32 years of age or younger, regardless of the conditioning regimen. Many of these women are likely to maintain fertility and may eventually become pregnant. However, a significant number of responders who underwent autologous hematopoietic stem cell transplant for autoimmune diseases already had children before transplant and indicated no desire for pregnancy after transplant; the data on pregnancy occurrence in this group should be interpreted in this context. Key words: Fertility, Online questionnaire, Multiple sclerosis, Stem cell therapy IntroductionAutologous hematopoietic stem cell transplant (AHSCT) in autoimmune diseases has recently gained international attention due to multiple publications of randomized trials showing advantages of this form of treatment over standard therapies in multiple sclerosis, 1,2 systemic sclerosis, 3,4 and Crohn disease. 5 With the growing numbers of patients with autoimmune diseases in which AHSCT can be considered as a therapy option, consulting physicians face the common question of the posttransplant effect on gonadal function and fertility. This question is particularly important in young women undergoing this procedure. Well-established risk factors of gonadal malfunction after hematopoietic stem cell transplant for traditional indications are age at transplant > 30 years, postpuberty status at transplant, male sex, use of radiotherapy, multiple doses of chemotherapy before hematopoietic stem cell transplant, inclusion of alkylating agents in the conditioning regimen...

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“…43 Late effects of HSCT and nontransplant treatments also need to be considered, including impacts on the endocrine, reproductive, and cardiovascular systems as well as secondary autoimmune diseases and neoplasia. 6,[44][45][46] In conjunction with both prospective and registry studies, mechanistic studies have been progressively produced. [47][48][49][50] The recent publication of guidelines for studies of immune reconstitution studies and biobanking provides the necessary framework for further development of mechanistic studies.…”

Section: Discussionmentioning

confidence: 99%

Evolution, trends, outcomes, and economics of hematopoietic stem cell transplantation in severe autoimmune diseases

et al. 2017

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Onset and outcome of pregnancy after autologous haematopoietic SCT (AHSCT) for autoimmune diseases: a retrospective study of the EBMT autoimmune diseases working party (ADWP)

Cited by 46 publications

References 11 publications

Autologous haematopoietic stem cell transplantation for treatment of multiple sclerosis

Autologous haematopoietic stem cell transplantation for treatment of multiple sclerosis

Untitled

Evolution, trends, outcomes, and economics of hematopoietic stem cell transplantation in severe autoimmune diseases

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