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The aetiology of anorexia nervosa is multifactorial and consists of a complex combination of genetic and environmental factors. Population-based twin studies estimate a heritability between 28 and 74%. 1–4 Anorexia nervosa is often associated with comorbid psychiatric disorders, such as depression, anxiety, obsessive–compulsive, personality and substance use disorders. 1,5,6 Familial and population-based studies report co-occurrence of anorexia nervosa and autism spectrum disorders (ASD) in families 7 and an overrepresentation of ASD in individuals with eating disorders. 8 Finally, in recent years a number of clinical and neuropsychological studies support the existence of shared traits between individuals with anorexia nervosa and ASD, including problems with central coherence, set-shifting and theory of mind in both disorders. 9–14 These traits could present shared endophenotypes of anorexia nervosa and ASD, which characterise individuals or specific subgroups of individuals with anorexia nervosa; subgroups that may represent those who have a prolonged or chronic course of illness. 5,14,15 Moreover, some authors have hypothesised that spectrum traits may manifest as anorexia nervosa in females and as ASD in males. 10 Tailoring treatment for those subgroups of individuals with anorexia nervosa who either have comorbid ASD or subthreshold autism spectrum symptoms and traits might alter the course of anorexia nervosa in a favourable direction. 16 It is therefore important to identify and to describe the co-occurrence of ASD in individuals with anorexia nervosa and in their families and vice versa. Clinical and population-based studies describing the coexistence of anorexia nervosa and ASD in families or on the psychological traits of social cognition are, however, limited by small samples of predominantly females (Ra ˚stam 2003, n = 51; Wentz 2005, n = 30). 6,8,17 Confirmatory evidence in large population-based samples does not yet exist. In Denmark we have access to data from public national registers with information on all people admitted to hospital for psychiatric disorders since 1969 onwards. 18 The aim of this study was to investigate the co-occurrence of anorexia nervosa and ASD in probands and in their first- and second-degree relatives in a nationwide study. We hypothesised (a) that ASD would be overrepresented in probands with anorexia nervosa and vice versa (comorbidity) and (b) in their families (cross-diagnostic genetic liability) and (c) that we could identify a dose–response effect of the presence of ASD in these relatives according to the degree of shared genetic material as a measure for a common genetic aetiology for anorexia nervosa and ASD. Finally, we repeated all analyses for comorbid and familial major depression and any psychiatric disorder, to determine whether any observed associations were specific to anorexia nervosa and to ASD. Method Study population We identified all individuals born in Denmark from 1 May 1981 to 31 December 2008 in the Danish Civil Registration System. 19 The register includes the personal identification number, gender, date of birth, continuously updated information on vital status and parents’ identification numbers. The identification number is used as a personal identifier in all national registers, enabling unique linkage between registers. We excluded those lacking a link to the mother and to information on parental ages at date of the child’s birth (n = 11 536 (0.7%) individuals, mostly lacking information on paternal age). 401 Autism spectrum disorder in individuals with anorexia nervosa and in their first- and second-degree relatives: Danish nationwide register-based cohort-study Susanne V. Koch, Janne T. Larsen, Svend E. Mouridsen, Mette Bentz, Liselotte Petersen, Cynthia Bulik, Preben B. Mortensen and Kerstin J. Plessen Background Clinical and population-based studies report increased prevalence of autism spectrum disorders (ASD) in individuals with anorexia nervosa and in their relatives. No nationwide study has yet been published on co-occurrence of these disorders. Aims To investigate comorbidity of ASD in individuals with anorexia nervosa, and aggregation of ASD and anorexia nervosa in their relatives. Method In Danish registers we identified all individuals born in 1981–2008, their parents, and full and half siblings, and linked them to data on hospital admissions for psychiatric disorders. Results Risk of comorbidity of ASD in probands with anorexia nervosa and aggregation of ASD in families of anorexia nervosa probands were increased. However, the risk of comorbid and familial ASD did not differ significantly from comorbid and familial major depression or any psychiatric disorder in anorexia nervosa probands. Conclusions We confirm aggregation of ASD in probands with anorexia nervosa and in their relatives; however, the relationship between anorexia nervosa and ASD appears to be non- specific. Declaration of interest C.B. is a consultant for Shire Pharmaceuticals. Copyright and usage B The Royal College of Psychiatrists 2015. The British Journal of Psychiatry (2015) 206, 401–407. doi: 10.1192/bjp.bp.114.153221

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  • The aetiology of anorexia nervosa is multifactorial and consists ofa complex combination of genetic and environmental factors.Population-based twin studies estimate a heritability between28 and 74%.14 Anorexia nervosa is often associated withcomorbid psychiatric disorders, such as depression, anxiety,obsessivecompulsive, personality and substance use disorders.1,5,6

    Familial and population-based studies report co-occurrence ofanorexia nervosa and autism spectrum disorders (ASD) infamilies7 and an overrepresentation of ASD in individuals witheating disorders.8 Finally, in recent years a number of clinicaland neuropsychological studies support the existence of sharedtraits between individuals with anorexia nervosa and ASD,including problems with central coherence, set-shifting and theoryof mind in both disorders.914 These traits could present sharedendophenotypes of anorexia nervosa and ASD, which characteriseindividuals or specific subgroups of individuals with anorexianervosa; subgroups that may represent those who have aprolonged or chronic course of illness.5,14,15 Moreover, someauthors have hypothesised that spectrum traits may manifest asanorexia nervosa in females and as ASD in males.10 Tailoringtreatment for those subgroups of individuals with anorexianervosa who either have comorbid ASD or subthreshold autismspectrum symptoms and traits might alter the course of anorexianervosa in a favourable direction.16 It is therefore important toidentify and to describe the co-occurrence of ASD in individualswith anorexia nervosa and in their families and vice versa.

    Clinical and population-based studies describing the coexistenceof anorexia nervosa and ASD in families or on the psychologicaltraits of social cognition are, however, limited by small samplesof predominantly females (Rastam 2003, n=51; Wentz 2005,

    n= 30).6,8,17 Confirmatory evidence in large population-basedsamples does not yet exist. In Denmark we have access to data frompublic national registers with information on all people admitted tohospital for psychiatric disorders since 1969 onwards.18 The aim ofthis study was to investigate the co-occurrence of anorexia nervosaand ASD in probands and in their first- and second-degree relativesin a nationwide study. We hypothesised (a) that ASD would beoverrepresented in probands with anorexia nervosa and vice versa(comorbidity) and (b) in their families (cross-diagnostic geneticliability) and (c) that we could identify a doseresponse effect ofthe presence of ASD in these relatives according to the degree ofshared genetic material as a measure for a common geneticaetiology for anorexia nervosa and ASD. Finally, we repeated allanalyses for comorbid and familial major depression and anypsychiatric disorder, to determine whether any observed associationswere specific to anorexia nervosa and to ASD.

    Method

    Study population

    We identified all individuals born in Denmark from 1 May 1981 to31 December 2008 in the Danish Civil Registration System.19 Theregister includes the personal identification number, gender, dateof birth, continuously updated information on vital status andparents identification numbers. The identification number is usedas a personal identifier in all national registers, enabling uniquelinkage between registers. We excluded those lacking a link tothe mother and to information on parental ages at date of thechilds birth (n= 11 536 (0.7%) individuals, mostly lackinginformation on paternal age).

    401

    Autism spectrum disorder in individualswith anorexia nervosa and in their first- andsecond-degree relatives: Danish nationwideregister-based cohort-studySusanne V. Koch, Janne T. Larsen, Svend E. Mouridsen, Mette Bentz, Liselotte Petersen,Cynthia Bulik, Preben B. Mortensen and Kerstin J. Plessen

    BackgroundClinical and population-based studies report increasedprevalence of autism spectrum disorders (ASD) in individualswith anorexia nervosa and in their relatives. No nationwidestudy has yet been published on co-occurrence of thesedisorders.

    AimsTo investigate comorbidity of ASD in individuals with anorexianervosa, and aggregation of ASD and anorexia nervosa intheir relatives.

    MethodIn Danish registers we identified all individuals born in19812008, their parents, and full and half siblings, and linkedthem to data on hospital admissions for psychiatric disorders.

    ResultsRisk of comorbidity of ASD in probands with anorexia

    nervosa and aggregation of ASD in families of anorexianervosa probands were increased. However, the risk ofcomorbid and familial ASD did not differ significantly fromcomorbid and familial major depression or any psychiatricdisorder in anorexia nervosa probands.

    ConclusionsWe confirm aggregation of ASD in probands with anorexianervosa and in their relatives; however, the relationshipbetween anorexia nervosa and ASD appears to be non-specific.

    Declaration of interestC.B. is a consultant for Shire Pharmaceuticals.

    Copyright and usageB The Royal College of Psychiatrists 2015.

    The British Journal of Psychiatry (2015)206, 401407. doi: 10.1192/bjp.bp.114.153221

  • Assessment of anorexia nervosa,ASD and any mental disorder

    Individuals in the study population, their parents, and their fulland half siblings were linked with the Danish Psychiatric CentralResearch Register,18 which registers all admissions to Danishpsychiatric in-patient facilities since 1 April 1969, and with theDanish National Patient Register,20 which registers alladmissions to somatic in-patient facilities since 1 January 1977.Since 1995 and onwards, the National Patient Register has alsoincluded psychiatric contacts in general hospitals, and both theNational Patient Register and the Psychiatric Central ResearchRegister have included information on out-patient contacts aswell. Until 31 December 1993, the diagnostic system usedwas ICD-8,21 and since 1 January 1994, the diagnostic systemICD-1022 has been used.

    Given our aim to study comorbidity between anorexia nervosaand ASD in an individual, we included all members of thepopulation classified as having anorexia nervosa or atypical anorexia(that is ICD-8 code 306.5x or ICD-10 code F50.0-F50.1) or infantileautism, atypical autism or Asperger syndrome (ICD-8 code299.00-299.01 or ICD-10 code F84.0-F84.1, F84.5) if they had arecord in the National Patient Register or Psychiatric CentralResearch Register of in- or out-patient care for that disorder. Casesof anorexia nervosa were not counted before the age of 9 years,because those diagnoses represented childhood eating disordersand not fully developed anorexia nervosa.23

    In a second step, to study familial aggregation of the twodisorders, we included index probands and their parents and siblingsdiagnosed with eating disorder (ICD-8 code 306.5 or ICD-10 codeF50.0-F50.1, F50.8), ASD (ICD-8 code 299.00-299.01 or ICD-10code F84.0-F84.1, F84.5, F84.8-F84.9), major depression (ICD-8code 296.09, 298.09 or ICD-10 code F32, F33) or any psychiatricdisorder (ICD-8 code 290-315 or ICD-10 code F00-F99), if theyhad a record of in- or out-patient care for that disorder. The dateof onset was defined as the first day of the first contact (in- orout-patient) leading to a diagnosis of that particular disorder.

    Study design

    A total of 1 724 189 people born in Denmark were followed upfrom date of birth until the first diagnosis with the disorder ofinterest, death, emigration from Denmark or 31 December 2012,whichever came first. The Danish Data Protection Agency hasapproved the study.

    Statistical analysis

    We estimated hazard ratios (HR; using Cox regression in Stata 12for Windows) for both anorexia nervosa and ASD for probands,who had anorexia nervosa or ASD, respectively, or were exposedto a first- or second degree relative with one or the other ofthe disorders, to detect co-occurrence of anorexia nervosa andASD independent of the sequence of the disorders.24 Moreover,we compared HRs for anorexia nervosa and ASD, respectively,in probands of families with major depression or any psychiatricdisorder to distinguish between a specific effect of co-occurrenceof the two disorders or a general psychiatric vulnerability. Analyseswere adjusted for maternal and paternal age, for calendar time(to adjust for birth-cohort effect) as a time-dependent variableand for gender by means of separate underlying hazardfunctions. Calendar time was categorised as 19811984, 19851989, 19901992, 19931994, 19951999, 20002004, 20052010and 20112012 taking into account the above mentioned changesin registration practice over time. Maternal age was categorised asless than 20 and 20 or older, whereas paternal age was categorisedas less than 20, 2039 and 40 or older according to how parental

    age acts as a risk factor for psychiatric illness in the offspring.25

    Familial psychiatric history was coded as time-dependent variablesfor probands, first-degree relatives (mothers, fathers and fullsiblings) and second-degree relatives (maternal and paternal halfsiblings) separately. Individuals without second-degree relativesand those without registry-identified fathers were analysedseparately. Furthermore, we performed supplementary analyses ofgender and age at outcome-diagnosis stratified by age 516 and516 years of age (results not shown). We used 2-tailed chi-squaretest for descriptive statistics.

    Results

    In the study population of 1 724 189 individuals, we identified5006 individuals (358 males, 4648 females; male:female ratio1:13.0) who had a diagnosis of anorexia nervosa, and 12 606individuals (10 012 males, 2594 females; male:female ratio 3.9:1)with a diagnosis of ASD (Table 1). In total, 45.8% of the femalesand 58.4% of the males were diagnosed with anorexia nervosa forthe first time before the age of 16 years (P50.001). More femaleswere given a first diagnosis of infantile or atypical autism (64.4%v. 59.1%, P=0.009) and fewer a diagnosis of Asperger syndrome(35.6% v. 40.9%, P50.001) before the age of 16 years comparedwith the males.

    Probands

    Risk of ASD in probands with anorexia nervosa

    Probands with a first diagnosis of anorexia nervosa, had a highlyelevated risk of receiving a second diagnosis of ASD (HR=15.08,95% CI 12.2318.58) (Table 2). This risk for males (HR= 22.04,95% CI 15.3931.58) was higher than for females (HR= 12.82,95% CI 9.9416.53). Males with anorexia nervosa had a higherrisk of a late autism spectrum diagnosis (516 years of age),whereas females with anorexia nervosa were equally diagnosedwith ASD before and after the age of 16 years (results not shown).However, we found an even higher risk for being diagnosed withASD in probands, whose first diagnosis was major depression(HR= 34.76, 95% CI 31.5138.36).

    Risk of anorexia nervosa in probands with ASD

    Likewise, probands, who had ASD as their first diagnosis, had anelevated risk of having a second diagnosis of anorexia nervosa(HR= 5.39, 95% CI 4.376.64) (Table 2) with no differences withrespect to gender or age at anorexia nervosa diagnosis (results notshown). The risk of developing anorexia nervosa, however, waseven higher after an initial diagnosis of major depression(HR= 17.67, 95% CI 16.1219.36).

    Family history

    Risk of ASD with a family history of anorexia nervosa

    A family history of anorexia nervosa was associated with anelevated risk of receiving a diagnosis of ASD (Table 3). The HRfor being diagnosed with ASD if a first-degree relative hadanorexia nervosa was 1.80 (95% CI 1.432.28) and the HR was1.44 (95% CI 0.942.21), if a second-degree relative had anorexianervosa. However, these HRs were of the same magnitude asthose seen in families with a history of major depression (HRfor first-degree relatives 1.78 (95% CI 1.651.92) and HR forsecond-degree relatives 1.37 (95% CI 1.111.69)) and those infamilies with any psychiatric disorder (Table 3). We thus didnot find a doseresponse effect according to the degree of sharedgenetic material among relatives.

    402

    Koch et al

  • Autism spectrum disorder and anorexia nervosa

    Risk of anorexia nervosa with a family history of ASD

    A family history of ASD also increased the risk of being diagnosedwith anorexia nervosa (HR= 1.45 (95% CI 1.091.94) for having afirst-degree relative with ASD and HR= 1.40 (95% CI 0.892.20)for having a second-degree relative with ASD) (Table 4). Similar tothe outcome of ASD, we did not observe a doseresponse effectaccording to the degree of shared genetic material among relatives.Furthermore, a family history of ASD yielded a significantly lowerrisk for developing anorexia nervosa compared with families witha history of anorexia nervosa (Table 4). Moreover, we observed anequally elevated risk for developing anorexia nervosa amongprobands, who had a family member with major depression(HR for first-degree relatives 1.34 (95% CI 1.191.51) and HRfor second-degree relatives 1.02 (95% CI 0.731.43)). Familieswith a history of any psychiatric disorder showed a similarpattern.

    Familial aggregation of anorexia nervosa

    A family history of anorexia nervosa was a risk factor fordeveloping anorexia nervosa with an HR of 3.92 (95% CI 3.124.93) if a first-degree relative had the disorder and an HR of 1.83(95% CI, 1.013.32) if a second-degree relative had the disorder(Table 4). This represents a dose response effect according tothe degree of shared genetic material and underscores the roleof genetic factors in the pathogenesis of anorexia nervosa. Asubanalysis showed that especially anorexia nervosa in mothersand sisters contributed to the increased risk (both genders);however, we identified only a few cases of anorexia nervosa amongfathers and brothers, which may have influenced the statisticalpower of the estimates (results not shown). Thus, we neither coulddetect any differential influences of a family history of anorexianervosa on the proband according to gender; nor did probandswith early diagnosis of anorexia nervosa seem to have a morepronounced aggregation of anorexia nervosa in the family.

    Familial aggregation of ASD

    Moreover, the risk for ASD was highly elevated when exposed toASD in a first- (HR= 7.79, 95% CI 7.158.50) and a second-degree relative (HR= 2.97, 95% CI 2.473.57) (Table 3). Femaleswere at highest risk (females HR= 10.24 (95 % CI 8.7112.04)v. males HR= 7.09 (95% CI 6.407.85)). ASD was most frequentlypresent in fathers and brothers (results not shown), but ASDamong the female members of the family also contributed tothe results.

    Unspecific diagnoses

    In additional analyses, we included a broader phenotype (theunspecific diagnoses of the two categories in ICD-10, such as codeF50.8 (eating disorders NOS) and code F84.8+.9 (ASD NOS),respectively) to explore the specificity of the findings, which didnot alter the main results.

    Other effects

    Additional analyses could not confirm any specific effect of any ofthe measured psychiatric disorders in half-siblings (divided intomaternal or paternal half-siblings), nor gender effects of full- orhalf-siblings. To exclude the possibility that unknown fatherscould have influenced the analyses, we repeated the analysesrestricted to families where all fathers were known, which alsodid not change the main findings.

    403

    Table

    1Individuals

    born

    inDenmark,1May1981to

    31December2008,whore

    ceivedeitherafirstdiagnosis

    ofanore

    xia

    nervosaorautism

    spectrum

    disord

    ers

    (ASD)bygenderandageatdiagnosis

    Ageatdiagnosis

    Ageatdiagnosis

    Ageatdiagnosis

    Diagnosis

    All

    516years

    516years

    Allfemales

    516years

    516years

    Allmales

    516years

    516years

    Anorexianervosa

    500

    6(100

    .0)

    233

    6(100

    .0)

    267

    0(100

    .0)

    464

    8(100

    .0)

    212

    7(10

    0.0)

    252

    1(100

    .0)

    358(100

    .0)

    209(100

    .0)

    149(100

    .0)

    ASD

    1260

    6(100

    .0)

    1085

    1(100

    .0)

    175

    5(100

    .0)

    259

    4(100

    .0)

    210

    3(100

    .0)

    491(100

    .0)

    1001

    2(100

    .0)

    874

    8(100

    .0)

    126

    4(100

    .0)

    Infantile/atypical

    autism

    712

    6(56.5)

    652

    4(60.1)

    602(34.3)

    150

    7(58.1)

    135

    4(64.4)

    153(31.2)

    561

    9(56.1)

    517

    0(59.1)

    449(35.5)

    Asp

    ergersyndrome

    548

    0(43.5)

    432

    7(39.9)

    115

    3(65.7)

    108

    7(41.9)

    749(35.6)

    338(68.8)

    439

    3(43.9)

    357

    8(40.9)

    815(64.5)

    Table

    2Hazard

    ratios(H

    Rs)forinfantile

    andatypicalautism,Asperg

    ersyndro

    me,andanore

    xia

    nervosaandatypicalanore

    xia

    giventh

    epro

    bandhasanore

    xia

    nervosa,autism

    spectrum

    disord

    ers

    (ASD)ormajordepre

    ssiona

    Probands

    history

    Allprobands

    Female

    probands

    Male

    probands

    ofpsych

    iatric

    disorder

    HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )

    HRforASD

    Nega

    tivehistory

    ofan

    orexianervosa

    1.00

    (Ref)

    170

    609

    6(99.7)

    1251

    2(99.3)

    1.00

    (Ref)

    83172

    8(99.4)

    253

    0(97.5)

    1.00

    (Ref)

    87436

    8(100

    .0)

    998

    2(99.7)

    Positiv

    ehistory

    ofan

    orexianervosa

    15.08(12.2318.58)

    548

    7(0.3)

    94(0.8)

    12.82(9.9416.53)

    508

    6(0.6)

    64(2.5)

    22.04(15.3931.58)

    401(0.1)

    30(0.3)

    Nega

    tivehistory

    ofdepression

    1.00

    (Ref)

    169

    092

    1(98.8)

    1210

    0(96.0)

    1.00

    (Ref)

    82227

    1(98.3)

    239

    0(92.1)

    1.00

    (Ref)

    86865

    0(99.3)

    971

    0(97.0)

    Positiv

    ehistory

    ofdepression

    34.76(31.5138.36)

    2066

    2(1.2)

    506(4.0)

    32.83(27.8738.67)

    1454

    3(1.7)

    204(7.9)

    35.64(31.5240.30)

    611

    9(0.7)

    302(3.0)

    HRforan

    orexianervosa

    Nega

    tivehistory

    ofASD

    1.00

    (Ref)

    170

    138

    0(99.0)

    491

    5(98.2)

    1.00

    (Ref)

    83103

    7(99.6)

    458

    0(98.5)

    1.00

    (Ref)

    87034

    3(98.4)

    335(93.6)

    Positiv

    ehistory

    ofASD

    5.39(4.376.64)

    1780

    3(1.0)

    91(1.8)

    5.22(4.116.64)

    372

    3(0.5)

    68(1.5)

    5.75(3.768.82)

    1408

    0(1.6)

    23(6.4)

    Nega

    tivehistory

    ofdepression

    1.00

    (Ref)

    169

    883

    8(98.8)

    441

    5(88.2)

    1.00

    (Ref)

    82093

    9(98.3)

    409

    6(88.1)

    1.00

    (Ref)

    87789

    9(99.3)

    319(89.1)

    Positiv

    ehistory

    ofdepression

    17.67(16.1219.36)

    2034

    5(1.2)

    591(11.8)

    16.84(15.3218.51)

    1382

    1(1.7)

    552(11.9)

    46.10(32.4365.54)

    652

    4(0.7)

    39(10.9)

    a.Of172

    418

    9individualsborn

    inDenmark1May

    1981

    to31

    Dece

    mber20

    08,5

    006(464

    8women,3

    58men)rece

    ivedadiagn

    osisofan

    orexianervosa,leav

    ing171

    918

    3co

    ntrols(834

    844women,8

    8442

    3men)a

    nd12

    606(259

    4women,10

    012men)rece

    ivedadiagn

    osis

    ofinfantileoratyp

    ical

    autism

    orAsp

    ergersyndrome,leaving171

    158

    3co

    ntrols

    (836

    814women,87

    476

    9men).Bold

    indicatesign

    ifica

    ntresu

    lts.

  • Koch et al

    404

    Table

    3Hazard

    ratios(H

    Rs)forinfantile

    andatypicalautism

    andAsperg

    ersyndro

    megiventh

    ere

    lativeshaveanore

    xia

    nervosa,autism

    spectrum

    disord

    ers

    (ASD),majordepre

    ssionorany

    psychiatric

    disord

    era

    Family

    history

    ofpsych

    iatric

    Allprobands

    Female

    probands

    Male

    probands

    disorder/typeofrelative

    HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n

    (%)Case

    group,n(%

    )

    History

    ofan

    orexianervosa

    First-degree,nega

    tive

    1.00

    (Ref)

    170

    149

    7(99.4)

    1253

    5(99.4)

    1.00

    (Ref)

    83187

    2(99.4)

    257

    8(99.4)

    1.00

    (Ref)

    86962

    5(99.4)

    995

    7(99.5)

    First-degree,positiv

    e1.80(1.432.28)

    1008

    6(0.6)

    71(0.6)

    1.84(1.133.01)

    494

    2(0.6)

    16(0.6)

    1.79(1.372.34)

    514

    4(0.6)

    55(0.6)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    46521

    5(27.2)

    425

    2(33.7)

    1.00

    (Ref)

    22802

    3(27.3)

    837(32.3)

    1.00

    (Ref)

    23719

    2(27.1)

    341

    5(34.1)

    Seco

    nd-degree,positiv

    e1.44

    (0.94

    2.21

    )286

    5(0.2)

    21(0.2)

    2.54(1.275.11)

    138

    9(0.2)

    8(0.3)

    1.14

    (0.66

    1.96

    )147

    6(0.2)

    13(0.1)

    History

    ofASD

    First-degree,nega

    tive

    1.00

    (Ref)

    169

    385

    6(99.0)

    1206

    2(95.7)

    1.00

    (Ref)

    82794

    5(98.9)

    243

    5(93.9)

    1.00

    (Ref)

    86591

    1(99.0)

    962

    7(96.2)

    First-degree,positiv

    e7.79(7.158.50)

    1772

    7(1.0)

    544(4.3)

    10.24(8.7112.04)

    886

    9(1.1)

    159(6.1)

    7.09(6.407.85)

    885

    8(1.0)

    385(3.9)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    45905

    4(26.8)

    415

    6(33.0)

    1.00

    (Ref)

    22490

    1(26.9)

    815(31.4)

    1.00

    (Ref)

    23415

    3(26.8)

    334

    1(33.4)

    Seco

    nd-degree,positiv

    e2.97(2.473.57)

    902

    6(0.5)

    117(0.9)

    3.62(2.515.21)

    451

    1(0.5)

    30(1.2)

    2.81(2.273.47)

    451

    5(0.5)

    87(0.9)

    History

    ofdepression

    First-degree,nega

    tive

    1.00

    (Ref)

    159

    854

    1(93.4)

    1185

    2(94.0)

    1.00

    (Ref)

    78109

    2(93.3)

    241

    4(93.1)

    1.00

    (Ref)

    81744

    9(93.5)

    943

    8(94.3)

    First-degree,positiv

    e1.78(1.651.92)

    11304

    2(6.6)

    754(6.0)

    1.90(1.642.22)

    5572

    2(6.7)

    180(6.9)

    1.74(1.601.90)

    5732

    0(6.6)

    574(5.7)

    Seco

    nd-degree,nega

    tive

    1.00

    44920

    9(26.3)

    418

    2(33.2)

    1.00

    22020

    7(26.3)

    820(31.6)

    1.00

    (Ref)

    22900

    2(26.2)

    336

    2(33.6)

    Seco

    nd-degree,positiv

    e1.37(1.111.69)

    1887

    1(1.1)

    91(0.7)

    1.68(1.132.51)

    920

    5(1.1)

    25(1.0)

    1.28(1.011.64)

    966

    6(1.1)

    66(0.7)

    History

    ofan

    ypsych

    iatricdisorder

    First-degree,nega

    tive

    1.00

    (Ref)

    130

    796

    5(76.4)

    931

    1(73.9)

    1.00

    (Ref)

    63823

    0(76.3)

    187

    7(72.4)

    1.00

    (Ref)

    66973

    5(76.6)

    743

    4(74.3)

    First-degree,positiv

    e2.02(1.942.10)

    40361

    8(23.6)

    329

    5(26.1)

    2.02(1.852.20)

    19858

    4(23.7)

    717(27.6)

    2.02(1.932.11)

    20503

    4(23.4)

    257

    8(25.8)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    36181

    7(21.1)

    352

    1(27.9)

    1.00

    (Ref)

    17724

    7(21.2)

    671(25.9)

    1.00

    (Ref)

    18457

    0(21.1)

    285

    0(28.5)

    Seco

    nd-degree,positiv

    e1.46(1.341.58)

    10626

    3(6.2)

    752(6.0)

    1.62(1.371.91)

    5216

    5(6.2)

    174(6.7)

    1.42(1.291.55)

    5409

    8(6.2)

    578(5.8)

    a.Of172

    418

    9people

    born

    inDenmark1May

    1981

    to31

    Dece

    mber20

    08,12

    606(259

    4women,10

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    ivedadiagn

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    autism

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    ergersyndrome,leaving171

    158

    3co

    ntrols

    (836

    814women,87

    476

    9men).Bold

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    Table

    4Hazard

    ratios(H

    Rs)foranore

    xia

    nervosaandatypicalanore

    xia

    giventh

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    lativeshaveanore

    xia

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    disord

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    disord

    era

    Family

    history

    ofpsych

    iatric

    Allprobands

    Female

    probands

    Male

    probands

    disorder/typeofrelative

    HR(95%

    CI)

    Controlgroup,n(%

    )Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n(%

    )Case

    group,n(%

    )HR(95%

    CI)

    Controlgroup,n(%

    )Case

    group,n(%

    )

    History

    ofan

    orexianervosa

    First-degree,nega

    tive

    1.00

    (Ref)

    170

    911

    3(99.4)

    493

    1(98.5)

    1.00

    (Ref)

    82991

    6(99.4)

    457

    8(98.5)

    1.00

    (Ref)

    87919

    7(99.4)

    353(98.6)

    First-degree,positiv

    e3.92(3.124.93)

    1007

    0(0.6)

    75(1.5)

    3.92(3.104.97)

    484

    4(0.6)

    70(1.5)

    3.84(1.599.30)

    522

    6(0.6)

    5(1.4)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    46800

    3(27.2)

    145

    4(29.0)

    1.00

    (Ref)

    22751

    0(27.3)

    135

    2(29.1)

    1.00

    (Ref)

    24049

    3(27.2)

    102(28.5)

    Seco

    nd-degree,positiv

    e1.83(1.013.32)

    288

    5(0.2)

    11(0.2)

    1.79

    (0.96

    3.33

    )138

    5(0.2)

    10(0.2)

    2.44

    (0.34

    17.51)

    150

    0(0.2)

    1(0.3)

    History

    ofASD

    First-degree,nega

    tive

    1.00

    (Ref)

    170

    050

    8(98.9)

    495

    9(99.1)

    1.00

    (Ref)

    82572

    6(98.9)

    460

    3(99.0)

    1.00

    (Ref)

    87478

    2(98.9)

    356(99.4)

    First-degree,positiv

    e1.45(1.091.94)

    1867

    5(1.1)

    47(0.9)

    1.51(1.132.03)

    903

    4(1.1)

    45(1.0)

    0.79

    (0.20

    3.16

    )964

    1(1.1)

    2(0.6)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    46166

    3(26.9)

    144

    6(28.9)

    1.00

    (Ref)

    22436

    5(26.9)

    134

    5(29.0)

    1.00

    (Ref)

    23729

    8(26.8)

    101(28.2)

    Seco

    nd-degree,positiv

    e1.40

    (0.89

    2.20

    )922

    5(0.5)

    19(0.4)

    1.34

    (0.83

    2.17

    )453

    0(0.6)

    17(0.4)

    2.17

    (0.53

    8.82

    )469

    5(0.5)

    2(0.6)

    History

    ofdepression

    First-degree,nega

    tive

    1.00

    (Ref)

    160

    530

    2(93.4)

    472

    0(94.3)

    1.00

    (Ref)

    77922

    2(93.4)

    439

    2(94.5)

    1.00

    (Ref)

    82608

    0(93.4)

    328(91.6)

    First-degree,positiv

    e1.34(1.191.51)

    11388

    1(6.6)

    286(5.7)

    1.28(1.131.46)

    5553

    8(6.7)

    256(5.5)

    2.11(1.453.07)

    5834

    3(6.6)

    30(8.4)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    45186

    2(26.3)

    143

    1(28.6)

    1.00

    (Ref)

    21970

    4(26.3)

    132

    8(28.6)

    1.00

    (Ref)

    23215

    8(26.3)

    103(28.8)

    Seco

    nd-degree,positiv

    e1.02

    (0.73

    1.43

    )19

    026(1.1)

    34(0.7)

    1.10

    (0.78

    1.54

    )991

    9(1.1)

    34(0.7)

    N/A

    983

    5(1.1)

    0(0.0)

    History

    ofan

    ypsych

    iatric

    disorder

    First-degree,nega

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    1.00

    (Ref)

    131

    224

    8(76.3)

    388

    8(77.7)

    1.00

    (Ref)

    63664

    4(76.3)

    362

    8(78.1)

    1.00

    (Ref)

    67560

    4(76.4)

    260(72.6)

    First-degree,positiv

    e1.29(1.211.38)

    40693

    5(23.7)

    111

    8(22.3)

    1.26(1.171.35)

    19811

    6(23.7)

    102

    0(21.9)

    1.74(1.382.20)

    20881

    9(23.6)

    98(27.4)

    Seco

    nd-degree,nega

    tive

    1.00

    (Ref)

    36372

    3(21.2)

    122

    5(24.5)

    1.00

    (Ref)

    17681

    6(21.2)

    113

    4(24.4)

    1.00

    (Ref)

    18690

    7(21.1)

    91(27.4)

    Seco

    nd-degree,positiv

    e1.06

    (0.92

    1.22

    )10

    716

    5(6.2)

    240(4.8)

    1.09

    (0.94

    1.25

    )52

    079(6.2)

    228(4.9)

    0.73

    (0.40

    1.33

    )55

    086(6.2)

    12(3.4)

    a.Of172

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    6(464

    8women,35

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    ing171

    918

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    ifica

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  • Autism spectrum disorder and anorexia nervosa

    Discussion

    Risk of anorexia nervosa and co-occurring ASD

    We studied comorbidity and familial aggregation of anorexia nervosaand of ASD in a nationwide sample. This study corroborates earlierfindings and confirms that individuals with anorexia nervosa havea higher risk of a co-occurring ASD, and that a family history ofanorexia nervosa increases the risk for developing ASD.7,8

    Similarly, individuals with ASD had an increased risk for a laterdiagnosis of anorexia nervosa and a family-member with ASDincreased the risk of developing anorexia nervosa. However, therisk of developing anorexia nervosa was also increased inindividuals with a diagnosis of major depression or anypsychiatric disorder. Moreover, the risk was also increased ifprobands had family members with any of these psychiatricdisorders. Furthermore, the study could not confirm a doseresponse effect of ASD in relatives of individuals with anorexianervosa according to the degree of shared genetic material. Hence,our results did not support the hypothesis that ASD acts as aspecific genetic or familial risk factor for developing anorexianervosa, because the risk for developing anorexia nervosa wasincreased both by the presence of ASD in probands (comorbidity)or ASD in a family-member (familial aggregation) but also thepresence of major depression or other psychiatric disordersincreased this risk. Earlier observations of an augmented risk forco-occurring anorexia nervosa and ASD in families, thus, mayexpress a non-specific genetic or familial vulnerability, whichmay also act as a risk factor for developing other psychiatricdisorders.

    Familiality

    This study replicates earlier findings concerning familialaggregation26 and heredity of anorexia nervosa,14 documentinga four times higher risk for developing anorexia nervosa inindividuals with a first-degree relative with anorexia nervosaand a two times higher risk of the disorder with a second-degreerelative. As expected, the doseresponse effect in relatives withASD is even stronger, which further confirms the heritability ofASD.27 These results support the good quality of the data.

    ASD as a non-specific risk factor for anorexia nervosa

    Several studies have demonstrated familial aggregation of majorpsychiatric disorders,28 including anorexia nervosa,26,29 in across-diagnostic perspective. Thus, familial aggregation of twoor more disorders has been demonstrated for several psychiatricdisorders, such as schizophrenia and bipolar disorder,30 Tourettesyndrome and obsessivecompulsive disorder31 and anorexianervosa and depression.1,32 Our findings, demonstrating thatASD is a non-specific familial risk factor for anorexia nervosaand vice versa, thus are in accordance with earlier findingssuggesting that psychiatric disorders in general predispose to thedevelopment of different psychiatric disorders in the offspring.33

    However, the specific co-occurrence of anorexia nervosa andASD in families, has to our knowledge not been studied previouslyin a large population-based setting. Our results indicate that asimple Mendelian, genetic pathway does not explain the cross-diagnostic familial aggregation, and interactions must be morecomplex.34 The interaction between genetic and environmentalfactors may act through pleiotropy (i.e. the same genes may giverise to different disorders). Thus, a genome-wide association studyhas revealed pleiotropy in a number of psychiatric disorders,35 andsuggests that pleiotropy act through identical genes that may bemodulated by environmental factors to be expressed as various

    endophenotypes,36 which again might predispose for differentpsychiatric disorders.

    Comparison with findings from neuropsychologicalstudies

    Neuropsychological studies, predominantly performed in adults,have reported the presence of social cognitive characteristics thatare pathognomonic of ASD in individuals with anorexia nervosaand their relatives.6,814,17 These studies suggest that at least asubgroup of individuals with anorexia nervosa demonstrate socialcognitive difficulties that may interfere with their ability to engagein family-based treatment, which is considered the treatment ofchoice for adolescents with anorexia nervosa.37,38 Most of thesestudies, however, find that the social cognitive deficits in patientswith anorexia nervosa are less severe than in patients with ASD,reporting intermediate measures of social cognitive performancebetween patients with ASD and healthy controls.11,13 None ofthese studies has included other comparison groups such aspatients and families with major depression or other psychiatricdisorders. Thus, existing data cannot conclude whether the socialcognitive deficits observed were specific to anorexia nervosa orwould also be observed in individuals with other psychiatricdisorders and in their family members. Although our results donot contradict the clinical observations; they argue for theinclusion of other pathological groups for comparison anddetermination of specificity in a cross-diagnostic perspective.

    Other explanations for differences between our epidemiologicalfindings and those of clinical studies of social cognition could bethat these studies often consisted of small or selected samples andoften included a combination of individuals with different eatingdisorders, i.e. also including those with bulimia nervosa, andindividuals at different stages of the diseases.8,17 Finally, studiesmay not have accounted fully for the fact that social cognitivecharacteristics can be influenced by underweight or other neuro-biological long-term effects of anorexia nervosa; thus, time tofollow-up might have influenced the findings, because theknowledge about the impact of the duration of starvation onneuropsychological functions is limited.8,39 Moreover, manystudies differ in their definition of full recovery of anorexianervosa.40

    Gender and age at onset in relation to co-occurringanorexia nervosa and ASD

    Males were more frequently diagnosed with anorexia nervosabefore the age of 16 years compared with females, but they didnot more frequently have relatives with ASD or anorexia nervosa.The data presented here cannot explain this finding. This couldsimply point to the fact that males may be admitted to hospitaltreatment at younger ages than females. Two Nordic clinicalstudies have reported that males referred to hospital treatmentwere both diagnosed with anorexia nervosa at younger ages, hada shorter course of illness and had an overall more favourableprognosis than females.41,42

    Males with anorexia nervosa also had a higher risk of having asecond diagnosis of ASD, but the pattern of anorexia nervosa andASD among their relatives did not indicate that genetic factorswere the explanation for this. It might be that males withunrecognised ASD first receive a diagnosis when they have contactwith the hospital system because of their eating problems. This issupported by the fact that the risk for developing ASD in themale group with anorexia nervosa was as high in the group of over16-year-olds as in the younger age group, which is in contrast to

    405

  • Koch et al

    the female group with anorexia nervosa, who more often werediagnosed with ASD before the age of 16 years.

    Overall, this study cannot conclude that groups defined byclinical characteristics such as gender or early-diagnosis anorexianervosa have a specifically increased risk for having ASD in thefamily, or vice versa. We are unable to determine from this studywhether anorexia nervosa and ASD are alternative gender-specificexpressions of the same genetic factor.

    Strengths and limitations

    We have included data from the total Danish population, whichaffords considerable statistical power, generalisability and aminimum of selection and recall bias. Hospital treatment is freeof charge in Denmark, which makes it accessible for all socio-economic groups. The Danish Civil Registration System doesnot allow people to be lost to follow-up and data are collectedprospectively for public administrative purposes and not for thisspecific study. Further strengths are the large number of cases,in particular in males, and exposed family members and theunique linkage to family members across two generations.

    Our study also has limitations. One is that we were onlyconsidering categorical diagnoses rather than more richlycharacterised dimensional characteristics, which may be moresuitable for describing cross-diagnostic traits or shared endo-phenotypes or subphenotypes. Hence, we were unable to includedata on subthreshold anorectic or autistic symptoms, or onpsychological traits concerning social cognitive deficits that mayinfluence the development of anorexia nervosa and ASD, or onpatients with an intermediate phenotype between anorexianervosa and ASD. These kinds of data are not available from theregisters where only patients who reach the full diagnostic criteria(for example infantile autism) are registered. For anorexia nervosaand bulimia nervosa it is furthermore assumed that more than50% of individuals with mild to moderate disorders are neveradmitted for hospital care,43 and therefore cannot be identifiedvia the registers.

    Eating disorder diagnoses in the Danish Registries are not yetsystematically validated. Register diagnoses of schizophrenia,44

    childhood autism45 and depression46 have, however, been validatedand have shown good validity, and thus, diagnoses in the registriesare deemed of good quality. One study has suggested thoughthat comorbid diagnoses have less good coverage compared withthe primary diagnoses,47 potentially leading to a number ofmissing comorbid diagnoses, which may have contributed tounderestimation of the co-occurrence of anorexia nervosa and ASD.

    Even though a small percentage of fathers were not identifiedat the childs birth and therefore hindered some linkages to familymembers, analysis restricted to probands, whose fathers wereknown, did not change the results; thus, this problem had a minorimpact on data quality. We decided to also include ICD-8diagnoses, to allow a larger population through the inclusion of olderbirth cohorts and mental health diagnoses of parents, even thoughthis system generated more incomplete and imprecise information,compared with ICD-10 diagnoses. Thus, inferior recognition ofpsychiatric disorders in parents may have contributed to lowerstatistical power in some of the subgroup analyses.

    Summary

    Our results suggest that an individual who has already developedanorexia nervosa is also at increased risk for being diagnosed withASD or vice versa. Further, we observe an aggregation of anorexianervosa and ASD in families. The risk for developing anorexianervosa, however, is equally increased if the individual hasdepression or any other psychiatric disorder, or if these disorders

    are present in the family. The present data, thus, do not support aspecific genetic relationship between anorexia nervosa and ASD;however, having any psychiatric disorder or having relatives withany psychiatric disorder is a non-specific risk factor for developinganorexia nervosa. Thus, our findings are in line with recent studiesthat point to overlapping genetic liabilities for several neuro-psychiatric disorders. The risk of developing anorexia nervosashowed a doseresponse effect according to having first- andsecond-degree relatives with anorexia nervosa and supports theheritability of anorexia nervosa; likewise the heritability of ASDis confirmed. Having family members with anorexia nervosa orASD did not seem to impose a specific risk on males to developanorexia nervosa compared with females or in relation to earlyonset of anorexia nervosa.

    Implications

    Our results indicate that the presences of a psychiatric disordermay act as a non-specific risk factor for the development of otherpsychiatric disorder in the offspring, which further extends ourknowledge concerning the risk of mental health problems inchildren of parents with psychiatric disorders. Earlier literaturehas focused mostly on children of parents with affective disordersor psychosis,28 but children with parents with anorexia nervosaare also at elevated risk.32 Moreover, this study emphasises theimportance of dimensional and cross-diagnostic approaches toadvance our understanding of the characteristics of socialcognitive functioning in individuals with anorexia nervosa,especially when distinguishing whether these represent state ortrait characteristics and whether these putative endophenotypesare of prognostic value. Furthermore, it is important to describeco-occurrence and sequential comorbidity with other psychiatricsymptoms or disorders in anorexia nervosa illness trajectory,and to be aware of the presentation of early signs andmanifestations of the disorder that may differ somewhat betweenthe genders. Similarly, future studies should further target thepossible aetiology and impact of shared cross-diagnostic geneticliabilities, preferably in longitudinal studies. A more thoroughassessment of neuropsychological characteristics and recognitionof co-occurrence of psychiatric disorders will hopefully lead toindividually tailored treatment-courses for individuals withanorexia nervosa, which in turn may lead to a shorter course ofthe illness and improved outcome.

    Susanne V. Koch, MD, PhD, Mental Health Centre for Child and AdolescentPsychiatry, Copenhagen Region and Institute of Clinical Medicine, Faculty of HealthSciences, University of Copenhagen, Copenhagen, Denmark; Janne T. Larsen, MSc,National Center for Register-based Research, School of Business and Social Sciences,University of Aarhus, Aarhus, and The Lundbeck Foundation Initiative for IntegrativePsychiatric Research, iPsych, Department of Biomedicine, Faculty of Health, AarhusUniversity, Denmark; Svend E. Mouridsen, MScPsych, Mette Bentz, MScPsych,Mental Health Centre for Child and Adolescent Psychiatry, Copenhagen Region,Denmark; Liselotte Petersen, MSc, PhD, National Center for Register-basedResearch, School of Business and Social Sciences, University of Aarhus, Aarhus,iPsych, Department of Biomedicine, Faculty of Health, Aarhus University, and TheLundbeck Foundation Initiative for Integrative Psychiatric Research, Aarhus, Denmark;Cynthia Bulik, PhD, FAED, Department of Psychiatry, University of North Carolina,Chapel Hill, North Carolina, USA, and Department of Medical Epidemiology andBiostatistics, Karolinska Institutet, Stockholm, Sweden; Preben B. Mortensen, MD,DMSci, National Center for Register-based Research, School of Business and SocialSciences, University of Aarhus, Aarhus, and The Lundbeck Foundation Initiative forIntegrative Psychiatric Research, iPsych, Department of Biomedicine, Faculty ofHealth, Aarhus University, Denmark; Kerstin J. Plessen, MD, PhD, Mental HealthCentre for Child and Adolescent Psychiatry, Copenhagen Region and Institute ofClinical Medicine, Faculty of Health Sciences, University of Copenhagen, Copenhagen,Denmark

    Correspondence: Susanne V. Koch, Mental Health Centre for Child andAdolescent Psychiatry, University of Copenhagen, Lers Parkalle 107, lth, DK2100, Copenhagen, Denmark. Email: [email protected]

    First received 23 Jun 2014, accepted 21 Aug 2014

    406

  • Autism spectrum disorder and anorexia nervosa

    Funding

    Mental Health Services, Capital Region, Copenhagen, Denmark.

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  • 10.1192/bjp.bp.114.153221Access the most recent version at DOI: 2015, 206:401-407.BJP

    Preben B. Mortensen and Kerstin J. PlessenSusanne V. Koch, Janne T. Larsen, Svend E. Mouridsen, Mette Bentz, Liselotte Petersen, Cynthia Bulik,register-based cohort-study

    nationwideand in their first- and second-degree relatives: Danish Autism spectrum disorder in individuals with anorexia nervosa

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