A population-based study of follow-up care for Hodgkin lymphoma survivors

Cancer ◽  
2010 ◽  
Vol 116 (14) ◽  
pp. 3417-3425 ◽  
Author(s):  
David C. Hodgson ◽  
Eva Grunfeld ◽  
Nadia Gunraj ◽  
Lisa Del Giudice
2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Christina Bergqvist ◽  
François Hemery ◽  
Arnaud Jannic ◽  
Salah Ferkal ◽  
Pierre Wolkenstein

AbstractNeurofibromatosis 1 (NF1) is an inherited, autosomal-dominant, tumor predisposition syndrome with a birth incidence as high as 1:2000. A patient with NF1 is four to five times more likely to develop a malignancy as compared to the general population. The number of epidemiologic studies on lymphoproliferative malignancies in patients with NF1 is limited. The aim of this study was to determine the incidence rate of lymphoproliferative malignancies (lymphoma and leukemia) in NF1 patients followed in our referral center for neurofibromatoses. We used the Informatics for Integrated Biology and the Bedside (i2b2) platform to extract information from the hospital’s electronic health records. We performed a keyword search on clinical notes generated between Jan/01/2014 and May/11/2020 for patients aged 18 years or older. A total of 1507 patients with confirmed NF1 patients aged 18 years and above were identified (mean age 39.2 years; 57% women). The total number of person-years in follow-up was 57,736 (men, 24,327 years; women, 33,409 years). Mean length of follow-up was 38.3 years (median, 36 years). A total of 13 patients had a medical history of either lymphoma or leukemia, yielding an overall incidence rate of 22.5 per 100,000 (0.000225, 95% confidence interval (CI) 0.000223–0.000227). This incidence is similar to that of the general population in France (standardized incidence ratio 1.07, 95% CI 0.60–1.79). Four patients had a medical history leukemia and 9 patients had a medical history of lymphoma of which 7 had non-Hodgkin lymphoma, and 2 had Hodgkin lymphoma. Our results show that adults with NF1 do not have an increased tendency to develop lymphoproliferative malignancies, in contrast to the general increased risk of malignancy. While our results are consistent with the recent population-based study in Finland, they are in contrast with the larger population-based study in England whereby NF1 individuals were found to be 3 times more likely to develop both non-Hodgkin lymphoma and lymphocytic leukemia. Large-scale epidemiological studies based on nationwide data sets are thus needed to confirm our findings.


2021 ◽  
Vol 151 ◽  
pp. 106586
Author(s):  
Meghan J. Walker ◽  
Olivia Meggetto ◽  
Julia Gao ◽  
Gabriela Espino-Hernández ◽  
Nathaniel Jembere ◽  
...  

Blood ◽  
2020 ◽  
Vol 136 (Supplement 1) ◽  
pp. 16-17
Author(s):  
Ilja Kalashnikov ◽  
Nea Malila ◽  
Sirkku Jyrkkio ◽  
Sirpa Leppa

Background Nodular lymphocyte predominant Hodgkin lymphoma (NLPHL) is a rare subtype of Hodgkin lymphoma (HL) with typically indolent clinical course. Although NLPHL is associated with favorable prognosis, late relapses, and a tendency to undergo transformation to aggressive lymphoma is seen in a substantial proportion of cases. Population-based long-term data on outcomes is limited. Aims We aimed to assess outcomes of the patients diagnosed with NLPHL in Finland. Methods We retrieved NLPHL patients diagnosed between 1995-2016 from the Finnish Cancer Registry (FCR) with complete follow-up to end of 2018. The Registry has an excellent coverage and provides accurate population-based nationwide data for all histologically verified cancers in Finland. Gender and events such as histologically verified progression and transformation were obtained from the FCR. Date of death or emigration was verified from the Population Register and underlying cause of death (COD) from Statistics Finland. COD was categorized into three groups: lymphoma, solid cancer, or other cause. Patients were categorized into diagnostic calendar periods (1995-2002, 2003-2010, 2011-2016), gender, according to relapse, transformation, and patient status (alive, dead, emigrated) by end of 2018. Standard descriptive statistical analyses were performed. Progression free survival (PFS) and all-cause overall survival (OS) was estimated via Kaplan-Meier method. Analysis of risk factors for OS and transformation was performed using the Cox proportional hazard model. Risk factors included diagnostic calendar period, age at diagnosis as a continuous variable (year), gender, relapse, and transformation. Multivariate analysis was performed including only factors with a p value < 0.05 from the univariate model. Two-tailed p-values < 0.05 were considered statistically significant. All statistical analyses were performed using R, version 4.0.2. Results We retrieved 414 patients with newly diagnosed NLPHL. Majority of the patients were males (n=320; 77%). Median age was 47 years (range 5 - 87), females being significantly older than males at diagnosis (59 vs 44 years; p < 0.001). We did not observe any statistically significant differences in age at diagnosis, gender distribution, progression, or transformation rates between the calendar periods. Five- and 10-year OS for the entire patient cohort were 89% and 84%, and 5- and 10-year PFS 77% and 64%, respectively (Fig. 1, 2). Eighty-six (21%) patients had at least one histologically verified progression (range 1 - 4). During the 11.1 years (range 0.7 - 23.9) follow-up, 81 (20%) patients died. The underlying COD was any lymphoma in 34 (42%), solid cancer in 14 (17%) and other cause in 33 (41%) patients. Gender, age at diagnosis, and transformation were included in the multivariate model with respect to OS. Age at diagnosis (HR 1.07; 95% CI 1.05 - 1.09), and transformation (HR 2.6; 95% CI 1.5 - 4.7), but not gender, remained independent. We found histologically verified transformation to large B cell lymphoma in 25 (7.4%) patients with ≥ 5-year follow-up from NLHPL diagnosis. Transformation was the first event in 15 (60%) patients, and 10 (40%) patients had one or more NLPHL progressions prior to transformation. The median age at the time of transformation was 57 years (range 20 - 87) and 17 (68%) of patients were males. The transformation free proportion over 5 years of follow-up was 96% and 93% over 10 years (Fig. 3). In multivariate analysis, gender, and age at diagnosis were not significantly associated with the risk of transformation. The 5-year OS after transformation was 50% which was significantly lower compared to the nontransformed patients with 5-year OS of 90%. Fourteen (56%) of the transformed patients died during follow-up, 13 from lymphoma and 1 from other cause. Conclusion In this large nationwide population-based study, females were 15 years older than males at the time of diagnosis. Twenty-five patients (7.4%) developed transformation to large B cell lymphoma. Age and transformation were independent risk factors for poor survival, whereas progression of NLPHL and gender were not associated with adverse outcome. We did not observe any survival difference between diagnostic calendar periods. Lymphoma was the main cause of death. Figure Disclosures Malila: Cancer Society of Finland: Current Employment. Jyrkkio:Hospital District of Southwest Finland: Current Employment.


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