ORIGINAL ARTICLES
A Search for Susceptibility Loci for Anorexia Nervosa:
Methods and Sample Description
Walter H. Kaye, Lisa R. Lilenfeld, Wade H. Berrettini, Michael Strober,
Bernie Devlin, Kelly L. Klump, David Goldman, Cynthia M. Bulik,
Katherine A. Halmi, Manfred M. Fichter, Allan Kaplan, D. Blake Woodside,
Janet Treasure, Katherine H. Plotnicov, Christine Pollice, Radhika Rao, and
Claire W. McConaha
Background: Eating disorders have not traditionally
been viewed as heritable illnesses; however, recent family
and twin studies lend credence to the potential role of
genetic transmission. The Price Foundation funded an
international, multisite study to identify genetic factors
contributing to the pathogenesis of anorexia nervosa (AN)
by recruiting affective relative pairs. This article is an
overview of study methods and the clinical characteristics
of the sample.
Methods: All probands met modified DSM-IV criteria for
AN; all affected first, second, and third degree relatives
met DSM-IV criteria for AN, bulimia nervosa (BN), or
eating disorder not otherwise specified (NOS). Probands
and affected relatives were assessed diagnostically with
the Structured Interview for Anorexia and Bulimia. DNA
was collected from probands, affected relatives and a
subset of their biological parents.
Results: Assessments were obtained from 196 probands
and 237 affected relatives, over 98% of whom are of
Caucasian ancestry. Overall, there were 229 relative pairs
who were informative for linkage analysis. Of the pro-
band-relative pairs, 63% were AN-AN, 20% were AN-BN,
and 16% were AN-NOS. For family-based association
analyses, DNA has been collected from both biological
parents of 159 eating-disordered subjects. Few significant
differences in demographic characteristics were found
between proband and relative groups.
Conclusions: The present study represents the first large-
scale molecular genetic investigation of AN. Our success-
ful recruitment of over 500 subjects, consisting of affected
probands, affected relatives, and their biological parents,
will provide the basis to investigate genetic transmission
of eating disorders via a genome scan and assessment
of candidate genes. Biol Psychiatry 2000;47:794 – 803
© 2000 Society of Biological Psychiatry
Key Words: Anorexia nervosa, bulimia nervosa, eating
disorders, genetics, linkage analysis, affected relative pairs
Introduction
A
norexia nervosa (AN) is characterized by an obses-
sively morbid dread of body fat that drives, then
sustains, an unrelenting avoidance of normal body weight
(American Psychiatric Association 1994). It is widely
assumed to have a complex, multifactorial etiology, yet
psychological theories of pathogenesis have largely pre-
vailed in explanatory paradigms put forward in recent
decades. Likewise, social influences have been implicated
in its causation in the light of mainstream cultural attitudes
in industrialized countries favoring thinness as a beauty
ideal.
Although a role for causal psychosocial factors is not in
question, certain well-established observations regarding
AN presentation argue persuasively for its qualitative
distinction from mere extreme dieting, and against an
etiological focus on nonbiological determinants alone in
accounting for its development, progression, and morbid-
ity. For example, weight-loss efforts in the general female
population are frequent, yet AN is hardly a common
illness—its lifetime prevalence among females is esti-
mated to be 0.1%– 0.7% (Hoek 1998). Second, in the
majority of acutely ill patients, dietary restriction is highly
entrenched, and resistance to initial therapeutic interven-
tion is reflected in an unusually protracted time to achieve
full remission of illness (Strober et al 1997). Third, AN
appears to covary with other psychopathological risk
From The Price Foundation Collaborative Group: Eating Disorders Module,
Western Psychiatric Institute & Clinic, University of Pittsburgh School of
Medicine, Pittsburgh, Pennsylvania (WHK, BD, KLK, KHP, CP, RR, CWM);
Department of Psychology, Georgia State University, Atlanta (LRL); Center
for Neurobiology and Behavior, Department of Psychiatry, University of
Pennsylvania School of Medicine, Philadelphia (WHB); Neuropsychiatric
Institute and Hospital, School of Medicine, University of California at Los
Angeles, Los Angeles (MS); National Institute on Alcohol Abuse and Alco-
holism (NIAAA), National Institutes of Health (NIH), Bethesda, Maryland
(DG); Virginia Institute for Psychiatric and Behavioral Genetics, Department
of Psychiatry, Virginia Commonwealth University, Richmond (CMB); New
York Presbyterian Hospital-Westchester, Weill Medical College of Cornell
University, White Plains, New York (KAH); Klinik Roseneck, Hospital for
Behavioral Medicine, Munich, Germany (MMF); Department of Psychiatry,
The Toronto Hospital, Toronto, Canada (AK, DBW); and the Institute of
Psychiatry, Maudsley and Bethlem Royal Hospital, London, England (JT).
Address reprint requests to Walter H. Kaye, M.D., Western Psychiatric Institute and
Clinic, 3811 O’Hara Street E-724, Pittsburgh PA 15213.
Received December 14, 1998; revised May 5, 1999; revised August 16, 1999;
accepted August 30, 1999.
© 2000 Society of Biological Psychiatry 0006-3223/00/$20.00
PII S0006-3223(99)00240-1