ArticleEuropean journal of neurology2016
Brain tumors in patients with myotonic dystrophy: a population-based study.
Article in European journal of neurology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers, 1 of them a synthesis that pooled it.
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Who cites it
10 citing papers in PubMed, 1 synthesis or guideline pooled it, 17 citations in OpenAlex.
- Cancer phenotype in myotonic dystrophy patients: Results from a meta-analysis.Muscle & nerve · 2018Pooled it
- Cancer Risk in Patients With Muscular Dystrophy and Myotonic Dystrophy: A Register-Based Cohort Study.Neurology · 2024Article
- Systematic thyroid screening in myotonic dystrophy: link between thyroid volume and insulin resistance.Orphanet journal of rare diseases · 2019Article
- Survival patterns and cancer determinants in families with myotonic dystrophy type 1.European journal of neurology · 2019Article
- Cancer Risk in Myotonic Dystrophy Type I: Evidence of a Role for Disease Severity.JNCI cancer spectrum · 2018Article
- Health economic evaluation of a serum-based blood test for brain tumour diagnosis: exploration of two clinical scenarios.BMJ open · 2018Article
- Risk of skin cancer among patients with myotonic dystrophy type 1 based on primary care physician data from the U.K. Clinical Practice Research Datalink.International journal of cancer · 2018Article
- Imaging of the thymus in myotonic dystrophy type 1.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2018Article
- Pigmentation phenotype, photosensitivity and skin neoplasms in patients with myotonic dystrophy.European journal of neurology · 2017Article
- Myotonic Dystrophies: State of the Art of New Therapeutic Developments for the CNS.Frontiers in cellular neuroscience · 2017Review
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Authors and funding
6 authors at 5 institutions in 3 countries.
Funding
Abstract
background and purposePatients with myotonic dystrophy (DM) are at high risk of brain cancer. This study describes the spectrum of brain neoplasms in DM patients.
methodsData from 1119 DM patients identified from the National Swedish Patient Register between 1987 and 2007 were linked to the National Cancer and the Cause of Death Registers. Standardized incidence ratios (SIRs) and cumulative incidence to quantify the relative and absolute risks of brain neoplasms were calculated and the Kaplan-Meier estimator was used for survival analysis. Patient follow-up started at birth or the age at the start of Swedish cancer registration (1 January 1958) and ended at the age of brain neoplasm diagnosis, death or on 31 December 2007.
resultsTwenty patients developed brain neoplasm during follow-up {median age 53, range 2-76 years, accounting for a five-fold excess risk of brain tumors during the patient lifetime [SIR = 5.4, 95% confidence interval (CI) 3.4-8.1, P = 1 × 10(-5) ]}. Astrocytoma was the most common histological subtype (n = 16, 80%), and almost all cases (n = 19) developed after age 20. No statistically significant differences in gender-specific risks (SIR in men 6.3 and in women 3.8, P-heterogeneity 0.46) were observed. After accounting for competing mortality related to DM, the cumulative incidence of brain neoplasms reached 2.9% (95% CI 1.8%-4.7%) by age 70. Five-year survival after brain tumor diagnosis was 52% (95%CI 29%-75%) overall (number at risk 8) and 34% (95% CI 26%-47%) for malignant neoplasms (number at risk 5).
conclusionDespite the high relative risk of DM-related brain tumors, the absolute risk is modest. Nonetheless, careful evaluation of DM patients with new central nervous system symptoms is warranted.
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