Boyne Research Institute

Drogheda, Ireland

Boyne Research Institute

Drogheda, Ireland

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PubMed | University of Groningen, St Jude Childrens Research Hospital, Emma Childrens Hospital Academic Medical Center, Boyne Research Institute and 4 more.
Type: Journal Article | Journal: Cancer | Year: 2016

Fertility preservation care for children, adolescents, and young adults (CAYAs) with cancer is not uniform among practitioners. To ensure high-quality care, evidence-based clinical practice guidelines (CPGs) are essential. The authors identified existing CPGs for fertility preservation in CAYAs with cancer, evaluated their quality, and explored differences in recommendations.A systematic search in PubMed (January 2000-October 2014); guideline databases; and Web sites of oncology, pediatric, and fertility organizations was performed. Two reviewers evaluated the quality of the identified CPGs using the Appraisal of Guidelines for Research and Evaluation II Instrument (AGREE II). From high-quality CPGs, the authors evaluated concordant and discordant areas among the recommendations.A total of 25 CPGs regarding fertility preservation were identified. The average AGREE II domain scores (scale of 0%-100%) varied from 15% on applicability to 100% on clarity of presentation. The authors considered 8 CPGs (32%) to be of high quality, which was defined as scores 60% in any 4 domains. Large variations in the recommendations of the high-quality CPGs were observed, with 87.2% and 88.6%, respectively, of discordant guideline areas among the fertility preservation recommendations for female and male patients with cancer.Only approximately one-third of the identified CPGs were found to be of sufficient quality. Of these CPGs, the fertility preservation recommendations varied substantially, which can be a reflection of inadequate evidence for specific recommendations, thereby hindering the ability of providers to deliver high-quality care. CPGs including a transparent decision process for fertility preservation can help health care providers to deliver optimal and uniform care, thus improving the quality of life of CAYAs with cancer and cancer survivors. Cancer 2016;122:2216-23. 2016 The Authors. Cancer published by Wiley Periodicals, Inc. on behalf of American Cancer Society.


PubMed | Markusovszky Hospital, San Gerardo Hospital, French Institute of Health and Medical Research, Semmelweis University and 13 more.
Type: Journal Article | Journal: PloS one | Year: 2016

Childhood cancer survivors are at high risk of long-term adverse effects of cancer and its treatment, including cardiac events. The pan-European PanCareSurFup study determined the incidence and risk factors for cardiac events among childhood cancer survivors. The aim of this article is to describe the methodology of the cardiac cohort and nested case-control study within PanCareSurFup.Eight data providers in Europe participating in PanCareSurFup identified and validated symptomatic cardiac events in their cohorts of childhood cancer survivors. Data on symptomatic heart failure, ischemia, pericarditis, valvular disease and arrhythmia were collected and graded according to the Criteria for Adverse Events. Detailed treatment data, data on potential confounders, lifestyle related risk factors and general health problems were collected.The PanCareSurFup cardiac cohort consisted of 59,915 5-year childhood cancer survivors with malignancies diagnosed between 1940 and 2009 and classified according to the International Classification of Childhood Cancer 3. Different strategies were used to identify cardiac events such as record linkage to population/ hospital or regional based databases, and patient- and general practitioner-based questionnaires.The cardiac study of the European collaborative research project PanCareSurFup will provide the largest cohort of 5-year childhood cancer survivors with systematically ascertained and validated data on symptomatic cardiac events. The result of this study can provide information to minimize the burden of cardiac events in childhood cancer survivors by tailoring the follow-up of childhood cancer survivors at high risk of cardiac adverse events, transferring this knowledge into evidence-based clinical practice guidelines and providing a platform for future research studies in childhood cancer patients..


Green R.F.,Centers for Disease Control and Prevention | Byrne J.,Boyne Research Institute | Crider K.S.,Centers for Disease Control and Prevention | Gallagher M.,Centers for Disease Control and Prevention | And 2 more authors.
Frontiers in Genetics | Year: 2013

Periconceptional folic acid use can often prevent neural tube defects (NTDs). Variants of genes involved in folate metabolism in mothers and children have been associated with occurrence of NTDs. We identified Irish families with individuals affected by neural tube defects. In these families, we observed that neural tube defects and birth defects overall occurred at a higher rate in the maternal lineage compared with the paternal lineage. The goal of this study was to look for evidence for genetic effects that could explain the discrepancy in the occurrence of these birth defects in the maternal vs. paternal lineage. We genotyped blood samples from 322 individuals from NTD-affected Irish families, identified through their membership in spina bifida associations. We looked for differences in distribution in maternal vs. paternal lineages of five genetic polymorphisms: the DHFR 19 bp deletion, MTHFD1 1958G>A, MTHFR 1298A>C, MTHFR 677C>T, and SLC19A1 80A>G. In addition to looking at genotypes individually, we determined the number of genotypes associated with decreased folate metabolism in each relative ("risk genotypes") and compared the distribution of these genotypes in maternal vs. paternal relatives. Overall, maternal relatives had a higher number of genotypes associated with lower folate metabolism than paternal relatives (p = 0.017). We expected that relatives would share the same risk genotype as the individuals with NTDs and/or their mothers. However, we observed that maternal relatives had an over-abundance of any risk genotype, rather than one specific genotype. The observed genetic effects suggest an epigenetic mechanism in which decreased folate metabolism results in epigenetic alterations related to the increased rate of NTDs and other birth defects seen in the maternal lineage. Future studies on the etiology of NTDs and other birth defects could benefit from including multigenerational extended families, in order to explore potential epigenetic mechanisms. © 2013 Fisk Green, Byrne, Crider, Gallagher, Koontz and Berry.


Terenziani M.,Fondazione IRCCS Instituto Nazionale dei Tumori | Spinelli M.,Fondazione MBBM | Jankovic M.,Fondazione MBBM | Bardi E.,Markusovszky Hospital | And 5 more authors.
Pediatric Blood and Cancer | Year: 2014

Background: Fertility is impaired in many survivors of childhood cancer following treatment. Preservation of fertility after cancer has become a central survivorship concern. Nevertheless, several doctors, patients, and families do not discuss fertility and recommendations for fertility preservation in pediatrics are still lacking. Recommendations based on scientific evidence are needed and before their development we wanted to assess the practice patterns of fertility preservation in Europe. Procedures: On behalf of the PanCare network, we sent a questionnaire to pediatric onco-hematology institutions across Europe. The survey consisted of 21 questions assessing their usual practices around fertility preservation. Results: One hundred ninety-eight institutional representatives across Europe received the survey and 68 (response rate 34.3%) responded. Pre-treatment fertility counseling was offered by 64 institutions. Counseling was done by a pediatric onco-hematologist in 52% (33/64) and in 32% (20/64) by a team. The majority of institutions (53%) lacked recommendations for fertility preservation. All 64 centers offered sperm banking; eight offered testicular tissue cryopreservation for pre-pubertal males. For females, the possibility of preserving ovarian tissue was offered by 40 institutions. Conclusions: There is a high level of interest in fertility preservation among European centers responding to our survey. However, while most recommended sperm cryopreservation, many also recommended technologies whose efficacy has not been shown. There is an urgent need for evidence-based European recommendations for fertility preservation to help survivors deal with the stressful topic of fertility. © 2014 Wiley Periodicals, Inc.


Byrne J.,Boyne Research Institute
Irish Journal of Medical Science | Year: 2011

Background: Neural tube defects (NTDs) and birth defects overall are more likely to occur among maternal compared to paternal relatives in two generations (uncles/aunts and first cousins) of Irish families where an individual has been born with an NTD. Aims: The aim of this study was to determine if the matrilineal excess persisted into the third generation. Methods: First cousins were interviewed about their pregnancy outcomes and their offsprings' health. Results: Maternal first cousins once removed (FCOR) were more likely to have birth defects than paternal FCOR: 6.7 versus 3.5% (adjusted odds ratio 1.49, 95% CI 0.57, 3.89). No NTDs occurred. Folic acid supplementation significantly reduced the risk of birth defects (P = 0.04). Conclusions: This study demonstrates an excess of birth defects among maternal relatives in three consecutive generations of NTD families, and supports the hypothesis that an underlying mechanism links distant maternal relatives in at least some NTD families. © Royal Academy of Medicine in Ireland 2010.


Walsh P.M.,National Cancer Registry | Byrne J.,Boyne Research Institute | Capra M.,Our Ladys Hospital | Comber H.,National Cancer Registry
European Journal of Cancer | Year: 2011

Survival after childhood cancer varies across Europe, but national or regional studies have so far shown no survival differences related to socio-economic disparity. The relationship of childhood cancer survival to disparity has not been studied in Ireland. We assessed observed survival for Irish children (ages 0-14 years) diagnosed with cancer during the period 1994-2005, overall (for all cancers included in the 3rd edition of the International Classification of Childhood Cancer) and for three main diagnostic groups - leukaemias, lymphomas, and central nervous system tumours. Comparisons were made between two diagnosis periods (1994-1999 and 2000-2005), between four regions of residence, and between five area-based deprivation categories. Regional patterns of treatment were examined to help assess the impact of centralisation of services. There was only limited evidence of improvements in survival over time. No clear evidence was found of deprivation-related influences on childhood cancer survival in Ireland, overall or for the three main diagnostic groups examined, although a weak trend was apparent for lymphoid leukaemias. Regional variation in survival was likewise not clear-cut, with the possible exception of CNS tumours (significantly higher survival amongst patients resident in the Western region). The absence of clear trends or patterns for regional or deprivation-related variation in survival may reflect a high degree of coordination and uniformity of treatment (and perhaps diagnostic) services, and application of standard treatment protocols nationally. © 2011 Elsevier Ltd. All rights reserved.


Byrne J.,Boyne Research Institute
Irish Journal of Medical Science | Year: 2011

Background: Miscarriages occur to excess in sibships with neural tube defects (NTDs) and among maternal versus paternal relatives in NTD families. Folic acid prevents most NTDs. Its potential to prevent miscarriages has been controversial. Aim: We evaluated the relationship of maternal line and periconceptional folic acid with miscarriage. Methods: First cousins in Irish families with NTDs were interviewed about pregnancy outcomes and the health of their offspring. Results: Miscarriages were not more frequent among pregnancies of maternal versus paternal first cousins. Folic acid intake during early pregnancy significantly reduced the risk of miscarriage from 15.7 to 9.6%, for an adjusted odds ratio of 0.37 (95% confidence interval 0.19, 0.72, p = 0.005). Conclusions: Folic acid during pregnancy was associated with a reduction of approximately 60% in miscarriages. Miscarriages are common-one in every eight pregnancies in this study. If incorporated into pre-pregnancy counseling, these results could have significant public health impact. © Royal Academy of Medicine in Ireland 2010.


Byrne J.,Boyne Research Institute
Irish Journal of Medical Science | Year: 2010

Background: Maternal first cousins of an individual with a neural tube defect (NTD) are at increased risk for an NTD. It is not known if they are also at risk for other serious birth defects. Methods: We carried out an interview study of uncles and aunts and first cousins in Irish NTD families covering their pregnancy histories and the health of family members. Results: Maternal first cousins were more likely than paternal first cousins to have a birth defect (9.4% vs. 5.5%, p = 0.02; adjusted odds ratio: 1.72, 95% confidence interval: 1.04, 2.84). Conclusions: This study shows that two generations of distant relatives (uncles/aunts and first cousins) in NTD families have similar maternal excesses of NTDs and birth defects overall. Inheritance mechanisms favouring matrilineal transmission, currently unknown, may contribute to birth defect occurrence in these families. © 2009 Royal Academy of Medicine in Ireland.


Walsh P.M.,National Cancer Registry | Byrne J.,Boyne Research Institute | Kelly M.,National Cancer Registry | McDevitt J.,National Cancer Registry | Comber H.,National Cancer Registry
PLoS ONE | Year: 2014

We evaluated the relationship between breast cancer survival and deprivation using data from the Irish National Cancer Registry. Cause-specific survival was compared between five area-based socioeconomic deprivation strata using Cox regression. Patient and tumour characteristics and treatment were compared using modified Poisson regression with robust variance estimation. Based on 21356 patients diagnosed 1999-2008, age-standardized five-year survival averaged 80% in the least deprived and 75% in the most deprived stratum. Age-adjusted mortality risk was 33% higher in the most deprived group (hazard ratio 1.33, 95% CI 1.21-1.45, P<0.001). The most deprived groups were more likely to present with advanced stage, high grade or hormone receptor-negative cancer, symptomatically, or with significant comorbidity, and to be smokers or unmarried, and less likely to have breast-conserving surgery. Cox modelling suggested that the available data on patient, tumour and treatment factors could account for only about half of the survival disparity (adjusted hazard ratio 1.18, 95% CI 0.97-1.43, P = 0.093). Survival disparity did not diminish over time, compared with the period 1994-1998. Persistent survival disparities among Irish breast cancer patients suggest unequal use of or access to services and highlight the need for further research to understand and remove the behavioural or other barriers involved. © 2014 Walsh et al.


PubMed | Boyne Research Institute and National Cancer Registry
Type: Journal Article | Journal: PloS one | Year: 2014

We evaluated the relationship between breast cancer survival and deprivation using data from the Irish National Cancer Registry. Cause-specific survival was compared between five area-based socioeconomic deprivation strata using Cox regression. Patient and tumour characteristics and treatment were compared using modified Poisson regression with robust variance estimation. Based on 21356 patients diagnosed 1999-2008, age-standardized five-year survival averaged 80% in the least deprived and 75% in the most deprived stratum. Age-adjusted mortality risk was 33% higher in the most deprived group (hazard ratio 1.33, 95% CI 1.21-1.45, P<0.001). The most deprived groups were more likely to present with advanced stage, high grade or hormone receptor-negative cancer, symptomatically, or with significant comorbidity, and to be smokers or unmarried, and less likely to have breast-conserving surgery. Cox modelling suggested that the available data on patient, tumour and treatment factors could account for only about half of the survival disparity (adjusted hazard ratio 1.18, 95% CI 0.97-1.43, P=0.093). Survival disparity did not diminish over time, compared with the period 1994-1998. Persistent survival disparities among Irish breast cancer patients suggest unequal use of or access to services and highlight the need for further research to understand and remove the behavioural or other barriers involved.

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