Literature DB >> 32176143

Contextualizing the Modern Epidemiology of Neurofibromatosis Type 2 in an Era of Heightened Detection of Sporadic Vestibular Schwannoma.

Eric M Dowling1, John P Marinelli2, Christine M Lohse3, Matthew L Carlson1,4.   

Abstract

OBJECTIVE: Closely paralleling previous radiologic studies, recent population-based prevalence data suggest sporadic vestibular schwannoma (VS) affects over one in 2,000 adults and up to one in 500 in those aged 70 years or older. Attributable to increased utilization of magnetic resonance imaging and screening protocols for asymmetrical sensorineural hearing loss, the increasing detection rate of sporadic VS fundamentally changes the perception of VS as a whole. The primary objective of the current study was to contextualize modern epidemiological trends in neurofibromatosis type 2 (NF2) in light of these recent advancements in the understanding of sporadic VS. STUDY
DESIGN: Population-based study.
SETTING: Olmsted County, Minnesota. Population size on January 1, 2017: 159,689 people. PATIENTS: All patients with NF2 diagnosed between Jan 1, 1966 and Dec 31, 2016, identified using the Rochester Epidemiology Project. MAIN OUTCOME MEASURES: Incidence, prevalence.
RESULTS: Six incident cases were identified over the past 50 years. From 1966 to 2016, the age- and sex-adjusted incidence rate was 0.10 per 100,000 person-years. The incidence rate remained at 0.12 over the most recent decade. Five cases met disease prevalence criteria, and the age- and sex-adjusted prevalence of NF2 on Jan 1, 2017 was 3.1 per 100,000 persons. All prevalent cases were women, and the resultant prevalence among women only was 6.0 per 100,000 persons.
CONCLUSIONS: The modern age- and sex-adjusted incidence rate and prevalence of NF2 is 0.10 per 100,000 person-years and 3.1 per 100,000 persons, respectively. In contrast to trends in incidence rates of sporadic VS over the last half-century, this study demonstrates that the incidence of NF2 has remained relatively stable since 1966. This divergence in epidemiological trends is likely attributed to the nature of NF2, with early clinical manifestations resulting in diagnosis regardless of modern advances, whereas the increased incidence of sporadic VS is heavily influenced by improved detection in a greater population of patients with minimally symptomatic or asymptomatic tumors.

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Year:  2020        PMID: 32176143      PMCID: PMC8055299          DOI: 10.1097/MAO.0000000000002557

Source DB:  PubMed          Journal:  Otol Neurotol        ISSN: 1531-7129            Impact factor:   2.311


  27 in total

1.  Incidence of vestibular schwannomas in the United States.

Authors:  Varun R Kshettry; Jason K Hsieh; Quinn T Ostrom; Carol Kruchko; Jill S Barnholtz-Sloan
Journal:  J Neurooncol       Date:  2015-05-30       Impact factor: 4.130

2.  Use of a medical records linkage system to enumerate a dynamic population over time: the Rochester epidemiology project.

Authors:  Jennifer L St Sauver; Brandon R Grossardt; Barbara P Yawn; L Joseph Melton; Walter A Rocca
Journal:  Am J Epidemiol       Date:  2011-03-23       Impact factor: 4.897

3.  Occult schwannomas of the vestibular nerve.

Authors:  T J Stewart; J Liland; H F Schuknecht
Journal:  Arch Otolaryngol       Date:  1975-02

4.  Incidence of vestibular schwannoma and neurofibromatosis 2 in the North West of England over a 10-year period: higher incidence than previously thought.

Authors:  D Gareth R Evans; Anthony Moran; Andrew King; S Saeed; Nihal Gurusinghe; Richard Ramsden
Journal:  Otol Neurotol       Date:  2005-01       Impact factor: 2.311

5.  Real Incidence of Vestibular Schwannoma? Estimations From a National Registry.

Authors:  Maarten Kleijwegt; Vincent Ho; Otto Visser; Willem Godefroy; Andel van der Mey
Journal:  Otol Neurotol       Date:  2016-10       Impact factor: 2.311

6.  Birth incidence and prevalence of tumor-prone syndromes: estimates from a UK family genetic register service.

Authors:  D G Evans; E Howard; C Giblin; T Clancy; H Spencer; S M Huson; F Lalloo
Journal:  Am J Med Genet A       Date:  2010-02       Impact factor: 2.802

7.  A novel moesin-, ezrin-, radixin-like gene is a candidate for the neurofibromatosis 2 tumor suppressor.

Authors:  J A Trofatter; M M MacCollin; J L Rutter; J R Murrell; M P Duyao; D M Parry; R Eldridge; N Kley; A G Menon; K Pulaski
Journal:  Cell       Date:  1993-03-12       Impact factor: 41.582

8.  Alteration in a new gene encoding a putative membrane-organizing protein causes neuro-fibromatosis type 2.

Authors:  G A Rouleau; P Merel; M Lutchman; M Sanson; J Zucman; C Marineau; K Hoang-Xuan; S Demczuk; C Desmaze; B Plougastel
Journal:  Nature       Date:  1993-06-10       Impact factor: 49.962

9.  Central neurofibromatosis with bilateral acoustic neuroma: genetic, clinical and biochemical distinctions from peripheral neurofibromatosis.

Authors:  W R Kanter; R Eldridge; R Fabricant; J C Allen; T Koerber
Journal:  Neurology       Date:  1980-08       Impact factor: 9.910

10.  Empirical development of improved diagnostic criteria for neurofibromatosis 2.

Authors:  Michael E Baser; Jan M Friedman; Harry Joe; Andrew Shenton; Andrew J Wallace; Richard T Ramsden; D Gareth R Evans
Journal:  Genet Med       Date:  2011-06       Impact factor: 8.822

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  1 in total

1.  Stereotactic radiosurgery for vestibular schwannomas in neurofibromatosis type 2 patients: a systematic review and meta-analysis.

Authors:  Umberto Tosi; Omri Maayan; Anjile An; Miguel E Tusa Lavieri; Sergio W Guadix; Antonio P DeRosa; Paul J Christos; Susan Pannullo; Philip E Stieg; Andrew Brandmaier; Jonathan P S Knisely; Rohan Ramakrishna
Journal:  J Neurooncol       Date:  2022-01-18       Impact factor: 4.130

  1 in total

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