FATHERS OF PATIENTS WITH SPORADIC NEUROFIBROMATOSIS 1, IN A CASE CONTROL STUDY WERE 1.5 YEARS OLDER THAN FATHERS OF THE CONTROL SUBJECTS AT THE BIRTH

Original Article
Paternal age and sporadic neurofibromatosis 1: A case-control study and consideration of the methodologic issues
Greta R. Bunin 1 *, Michael Needle 1, Vincent M. Riccardi 2
1Department of Pediatrics, Division of Oncology, Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine, Philadelphia
2The Neurofibromatosis Institute, La Crescenta, California

email: Greta R. Bunin (bunin@email.chop.edu)

*Correspondence to Greta R. Bunin, Children's Hospital of Philadelphia, Abramson Center, 34th St. & Civic Center Blvd., Philadelphia, PA 19104-4318

Funded by:
National Institutes of Health; Grant Number: HD28376
Texas Neurofibromatosis Foundation

Keywords
epidemiologic methods • germline mutation • maternal age


Abstract
Sporadic neurofibromatosis 1 (NF1) occurs in the absence of a family history of the disease and usually results from a new mutation in the germ cell of one of the parents, most commonly the father. Older paternal age may increase the risk for a new germinal NF1 mutation, but the results of studies to address this question conflict. We investigated paternal age in sporadic NF1 by using a case-control study design. Patients who were seen at two specialty NF clinics in Houston, Texas, born between 1970 and 1992 and living in the Houston area and surrounding counties, were studied. Birth certificates with information on the father were found for 89 cases. For each case, two birth certificates were chosen at random from the same year and county of birth. In this way, the control group of 178 individuals was formed. Fathers of patients with NF1 were 1.5 years older than fathers of control subjects at the birth of the child, but the difference was only of borderline statistical significance (P = 0.07). This paternal age difference was not changed by adjustment for socioeconomic status or maternal age. These and previous data are consistent with either a small paternal age effect in sporadic NF1 or a bias such as that resulting from the selection of cases and/or controls. Genet. Epidemiol. 14:507-516,1997. © 1997 Wiley-Liss, Inc.

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Received: 23 July 1996; Revised: 2 December 1996; Accepted: 2 January 1997
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