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Body Focused Repetitive Behavior Disorders: Significance of Family History
Sarah A. Redden, Eric W. Leppink, Jon E. Grant
PII:
DOI:
Reference:
S0010-440X(15)30317-5
doi: 10.1016/j.comppsych.2016.02.003
YCOMP 51629
To appear in:
Comprehensive Psychiatry
Please cite this article as: Redden Sarah A., Leppink Eric W., Grant Jon E., Body
Focused Repetitive Behavior Disorders: Signicance of Family History, Comprehensive
Psychiatry (2016), doi: 10.1016/j.comppsych.2016.02.003
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Body Focused Repetitive Behavior Disorders: Significance of Family History
Sarah A. Redden, BA
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Eric W. Leppink, BA
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Email: jongrant@uchicago.edu
Declaration of interest: This study was supported by internal funds. Ms. Redden reports no
conflicts of interest. Dr. Grant has received research grants from National Center for Responsible
Gaming, the American Foundation for Suicide Prevention, the Trichotillomania Learning Center,
and Forest and Psyadon Pharmaceuticals. He receives yearly compensation from Springer
Publishing for acting as Editor-in-Chief of the Journal of Gambling Studies and has received
royalties from Oxford University Press, American Psychiatric Publishing, Inc., Norton Press, and
McGraw Hill. Mr. Leppink reports no conflicts of interest.
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Running title: Family history in trichotillomania and skin picking
Abstract
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(BFRBs) (i.e. trichotillomania and skin picking) has received scant research attention. We sought
to understand the clinical and cognitive impact of having a first-degree relative with a BFRB or a
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Methods: 265 participants with BFRBs undertook clinical and neurocognitive evaluations.
Those with a first-degree relative with a BFRB or a SUD were compared to those without on a
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Results: 77 (29.1%) participants had a first-degree family member with a BFRB and 59 (22.2%)
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had a first-degree family member with a SUD. In terms of clinical severity, the amount of time
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spent picking or pulling per day in the past week was higher among those with a first-degree
relative with a SUD. There was a higher rate of ADHD and higher HAM-D scores among those
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family history.
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with a positive family history of a SUD. There were no significant cognitive differences based on
Conclusions: These results indicate that among those with BFRBs, having a first-degree family
member with a SUD may be associated with a unique clinical and cognitive presentation.
Whether family history also is associated with differential response to treatments awaits further
research.
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1. Introduction
pulling out of ones own hair, leading to hair loss and marked functional impairment.1,2 Skin
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picking disorder is characterized by the repetitive and compulsive picking of skin which causes
tissue damage.3 Trichotillomania and skin-picking appear to have substantial clinical and
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possibly even neurobiological similarities, and based on available evidence, have been described
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Although both trichotillomania and skin picking may seem like simple behaviors,
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research has demonstrated that BFRBs are complex, highly individualistic disorders.5 The
clinical utility of identifying potential subtypes of BFRBs, therefore, has been examined in the
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literature, including focused versus automatic behaviors,6 early versus late age at onset,7-9 and
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comorbidity patterns.10 One area that has received little attention is the possible importance of
family history.
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BFRBs have long been thought to have a familial basis.11-17 Several family studies have
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reported elevated rates of BFRBs in first-degree relatives of probands with a BFRB.14,18,19 The
suggestion that BFRBs may be familial seems consistent with existing twin studies which
suggest the disorders are heritable in addition to being familial.20,21
Although studies have examined the familial aspects of BFRBs, the family history issue
is perhaps more complex than initially thought. One family history study of trichotillomania that
included a control group found that the first-degree relatives of subjects with trichotillomania
were significantly more likely to have substance use disorders (21.6% alcohol and 14.7% drug
use disorders) than relatives of non-ill comparison subjects (7.7% alcohol use disorders and 2.2%
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drug use disorders).14 Similarly, a study of 34 patients with skin picking disorder found lifetime
alcoholism in 37.5% of first-degree relatives.22
Thus, the existing literature suggests that BFRBs run in families where we also see high
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rates of BFRBs and SUDs. Existing data, however, do not provide information as to what, if
anything, these types of familial associations may mean for the person with trichotillomania or
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skin picking disorder. Therefore, understanding the clinical and neurocognitive aspects of
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BFRBs and how these factors differ between individuals with different types of family histories
may be important in order to identify potential clinical and cognitive subtypes, improve
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neurobiological models, and optimize treatment. The purpose of this study is to investigate
whether adults with BFRBs with a first-degree relative with a BFRB or a SUD have a different
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clinical presentation than those without, and whether analysis of different families histories has
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2.1 Subjects
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implementation of any treatments. Data were collected from September 2006 through January
2015.
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2.2 Assessments
Participants were asked about the presence of lifetime BFRBs and SUDs (which included
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alcohol and drug use disorders, but not nicotine) in all first-degree relatives. As some picking
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and pulling behavior may occur in family members without rising to the level of a disorder, only
severe picking resulting in chronic lesions and pulling resulting in noticeable alopecia met the
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and occupational dysfunction or the need for a twelve-step program or formal treatment. All
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information about relatives came from the proband. No direct evaluations of the first-degree
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Current and lifetime psychiatric comorbidity was assessed using the Structured Clinical
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Interview for DSM-IV (SCID) disorders23 and valid and reliable SCID-compatible modules for
impulse control disorders using the Minnesota Impulse Disorders Interview (MIDI).24 The
diagnosis of ADHD was based on self-report history by the participant during a semi-structured
interview.
Each participant completed measures regarding the severity of their hair pulling or skin
picking. For purposes of this combined dataset, we extracted minutes per day pulling or picking
as this single measure could be combined across disorders whereas the disorder-specific scales
could not be. In addition, each participant was asked if they had previously received any
treatment for their picking or pulling behavior.
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In terms of formal measures, all participants completed the following at baseline:
Clinical Global Impression- Severity (CGI).25 The CGI is a valid and reliable, 7-item
scale used to assess symptom severity. It uses a Likert-scored scale with 1 = not ill at all to 7 =
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among the most extremely ill. The scale was used to assess only the severity of the BFRB
symptoms.
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Sheehan Disability Scale (SDS).26 The SDS is a valid and reliable, three-item, self-report
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scale assessing psychosocial functioning in three areas of life: work, social or leisure activities,
and home and family life. Scores on the SDS range from 0 to 30.
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Quality of Life Inventory (QoLI).27 The QoLI is a valid and reliable 16-item, self-report
positive psychology scale assessing areas of life such as health, love, work, recreation, home,
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Current depressive and anxiety symptoms were assessed using the 17-item Hamilton
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Depression Rating Scale28 and the Hamilton Anxiety Rating Scale,29 respectively.
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Stop-signal task (SST). The Stop-signal task from the CANTAB is a well-validated task
quantifying the ability to suppress impulsive responses.30 This task provides a sensitive estimate
of the time taken by the participants brain to stop a prepotent response, referred to as the Stopsignal reaction time (SSRT).
Intra-dimensional/Extra-dimensional Set Shift task (IDED).31 The IDED task in the
CANTAB includes aspects of rule learning and behavioral flexibility, and was derived from the
Wisconsin Card Sort Test.32 There are nine stages to the task, requiring different components of
set acquisition, reversal, and flexibility.
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Based on the family history, participants were categorized into the following groups:
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family history positive/negative for a BFRB and family history positive/negative for a SUD. We
used one-sample Kolmogorov-Smirnov Tests to test for normal distribution for all continuous
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variables. Potential differences between the groups were explored using analysis of variance
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(ANOVA) for normally distributed variables, while Mann-Whitney Tests were used to determine
significant differences in the variables that were not normally distributed. Chi-square tests were
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used for non-parametric tests as appropriate. A Bonferroni correction was conducted to correct
for multiple comparisons. With seven clinical variables, significance was defined as the
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Bonferroni adjusted p<.007 (Table 1) and with five comorbidities, it was defined as p<.01 (Table
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2). Because Bonferroni corrections are considered overly restrictive,33 we also highlight select
variables that are not statistically significant as we believe they have potential clinical
3. Results
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145 participants with trichotillomania and 120 with skin picking disorder took part in the
study (mean age SD= 33.2 11.3 years; n = 237 (89.4%) females). Of the 265 total
participants with a BFRB, 77 (29.1%) had at least one first-degree relative with trichotillomania
or skin picking, and 59 (22.3%) had at least one first-degree relative with a SUD. Specifically, of
the subjects with trichotillomania, 34 (23.4%) had a relative with a BFRB and 30 (20.7%) had a
relative with a SUD. In the subjects with skin picking disorder, 43 (35.8%) had a relative with a
BFRB and 29 (24.2%) had a relative with a SUD. There was little overlap in these family
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histories as only 11 (4.2%) had at least one first-degree relative with both a BRFB and SUD.
Clinical characteristics of those with and without a positive family history are contrasted in Table
1.
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pulling or picking and greater depression symptomatology. There were also no statistically
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significant clinical differences between adults with and without a family member with a BFRB.
Comorbid conditions and cognitive variables are presented in Table 2. Those with a
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family history positive for SUD reported significantly higher rates of co-occurring ADHD
(p=.001). There were no significant differences in terms of comorbidities between those with and
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without a family history of BFRBs. No significant cognitive differences were found based on
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family history.
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4. Discussion
The study confirms previous research showing that BFRBs are familial,13,14,19,20 as
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approximately one-third of our participants had a first-degree family member with a BFRB.
Given that almost one-fourth of the participants had a first degree family member with a SUD,
these data also suggest that substance addictions may run in families with BFRBs as well. The
high rate of SUDs in family members is in keeping with previous research14 and adds to the
speculation that at least in some individuals, BFRBs may be forms of addiction.34
Only in the case of SUDs did family history appear to have any clinical relevance. BFRB
participants with a family history of SUDs spent more time engaged in pulling and picking. One
possible explanation for this finding is that the picking and pulling are responses to the emotional
chaos of families with substance addictions.35 In these families, picking and pulling may function
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very early on as self-soothing mechanisms in response to the potentially chaotic environments.
Another possible, non-mutually exclusive, explanation is that a family history of SUDs may
reflect a shared genetic/biologic etiology for picking and pulling (for example neuroimaging
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evidence suggests a possible shared circuitry involving orbitofrontal cortices, anterior cingulate
cortices, and neuro regions such as the right inferior frontal gyrus and the pre-supplementary
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individuals, picking and pulling may function like an addiction and, although it awaits future
research, may even have a shared neurocircuitry with addictions.
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In terms of co-occurring disorders, those with a family history of SUDs had higher rates
of comorbid ADHD. In light of the other findings, the elevated rates of ADHD in these
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individuals could mean that some sort of impulsivity is familial and gives rise to ADHD, more
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picking and pulling, and substance addictions. Some support for this comes from a recent study
of response inhibition in adults with trichotillomania, their first-degree relatives, and healthy
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controls. Those with trichotillomania demonstrated impaired performance versus controls, with
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incorporate behavioral elements addressing this issue specifically, or at least impulsivity more
generally, as well as the picking and pulling. Finally, one recent pharmacological treatment study
found that those adults with trichotillomania and a positive family history for alcoholism
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responded to the opioid antagonist, naltrexone, whereas those without this family history did
not.39 Whether family history reflects viable subtypes within BFRBs that could improve
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Although BFRBs have long been known to run in families, we found no evidence that the
presence or absence of this familial link results in a different clinical presentation. One
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explanation could be that the BFRB family history simply did not differentiate the specific
clinical variables we examined. Whether it could be a meaningful variable for other aspects of
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BFRBs remains unanswered. Additionally, whether this family history may be associated with
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5. Conclusions
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This study is the first to compare clinical measures based on two family history analyses
and as such highlights how family history may be associated with unique clinical presentations
of BFRBs (for example more minutes spent picking or pulling per week and higher rates of
comorbid ADHD in those with a first degree relative with a SUD). This study, however, has
several limitations. First, BFRB probands were recruited because they had sought treatment or
responded to an advertisement for research. A community sample may provide very different
information. Second, ADHD was evaluated in a semi-structured interview with subjects
reporting if they had been previously diagnosed with ADHD. Diagnosis of ADHD may have
been more accurate if it was evaluated using a valid and reliable tool instead. Third, combining
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the data from those with trichotillomania and those with skin picking disorder could have
obscured the findings for either group alone. Finally, no interviews of family members were
conducted and so these results may under- or over-estimate the family rates of BFRBs and
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SUDs.
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Table 1: Demographic and Clinical Variables in 265 Body-Focused Repetitive Behaviors (BFRBs) Subjects With and Without
First-Degree Relatives with BFRBs or Substance Use Disorders (SUDs)
BFRB
SUD
With 1st
Without 1st
With 1st
Without 1st
Degree
Degree
Degree
Degree
Relative
Relative
Statistic
p-value Relative with
Relative
Statistic
p-value
with BFRB with BFRB
SUD
with SUD
(N=77)
(N=188)
(N=59)
(N=206)
DEMOGRAPHICS
34.4(11.4)
32.7 (11.2)
6656.5
.304
33.9 (11.4)
33.0 (11.2)
5782.5
.339
Age (years)
12.3 (6.8)
13.0 (7.9)
7034.0
.718
13.4 (9.3)
12.7 (7.0)
6069.0
.988
Age at Onset (years)
Sex, N (%), Female
70 (90.9)
167 (88.8)
1.269*
.530
54 (91.5)
183 (88.8)
.816*
.665
Race, N(%),
70 (90.9)
161 (85.6)
8.752*
.119
48 (81.4)
183 (88.8)
9.649*
.086
Caucasian
CLINICAL
Time spent in past
97.6(84.3)
86.6 (68.7)
4450.0
.606
117.9 (90.7)
81.7 (65.8)
3129.0
.022
week pulling or
picking(minutes/day)
Clinical Global
4.44 (0.8)
4.40 (0.7)
7184.0
.914
4.5 (0.8)
4.4 (0.7)
5493.0
.202
Impression-Severity
Sheehan Disability
10.8 (6.7)
10.8 (6.5)
.003+
.955
10.7 (6.9)
10.9 (6.4)
.027+
.870
Scale
Quality of Life
43.5 (10.1)
43.0 (12.6)
7042.0
.963
42.0 (12.2)
43.5 (11.8)
5498.0
.339
Inventory (T-score)
Hamilton Depression
4.2 (3.5)
4.4 (3.6)
6984.0
.652
5.2 (3.6)
4.1 (3.5)
4982.0
.034
Rating Scale
Hamilton Anxiety
4.3 (3.6)
4.3 (3.4)
7220.0
.975
5.1 (3.8)
4.1 (3.4)
5142.5
.070
Rating Scale
History of BFRB
37 (48.1)
74 (39.4)
1.695*
.193
24 (40.7)
87 (42.2)
.046*
.831
Treatment, N (%)
All items are Mean (SD) unless noted
Statistics are Mann-Whitney Test (U) unless noted, *Chi-Square Test, +ANOVA (F), P-Value Bonferroni Corrected is significant at
.007.
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BFRB: Body-Focused Repetitive Behaviors, SUD= Substance Use Disorder, N=Number of subjects, %=Percent of Subjects
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Table 2: Comorbidities and Neurocognitive Variables in Body-Focused Repetitive Behaviors (BFRB) Subjects With and
Without First-Degree Relatives with BFRBs or Substance Use Disorders (SUDs)
BFRB
SUD
st
st
st
With 1
Without 1
With 1
Without 1st
Degree
Degree
Degree
Degree
Relative
Relative
Statistic
p-value Relative with
Relative
Statistic
p-value
with BFRB with BFRB
SUD
with SUD
(N=77)
(N=188)
(N=59)
(N=206)
COMORBIDITIES
Major Depressive
28 (36.4)
72 (38.3)
.087*
.768
24 (40.7)
76 (36.9)
.280*
.597
Disorder, N (%)
Any Anxiety
15 (19.5)
37 (19.7)
.001*
.970
11 (18.6)
41 (19.9)
.046*
.830
Disorder, N (%)
Obsessive
6 (7.8)
9 (4.8)
.924*
.336
4 (6.7)
11 (5.3)
.1788*
.673
Compulsive
Disorder, N (%)
Attention Deficit
8 (10.4)
17 (9.0)
.116*
.733
12 (20.3)
13 (6.3)
10.564*
.001
Hyperactivity
Disorder, N (%)
Any Lifetime
42 (54.5)
100 (53.2)
.040*
.841
37 (62.7)
105 (51.0)
2.542*
.111
Disorder, N (%)
COGNITIVE VARIABLES
Stop Signal Reaction
186.2 (65.7) 191.7 (68.7)
2711.5
.729
202.3 (86.9) 186.3 (60.5)
2279.0
.795
Timea
IntraDimensional/Extra9.9 (10.5)
8.6 (9.8)
2745.5
.694
9.5 (10.1)
8.8 (10.0)
2318.5
.322
Dimensional SetShift Task: ExtraDimensional Shiftb
Intra-Dimensional
24.3 (20.1)
21.5 (21.9)
2604.0
.372
22.3 (19.0)
22.3 (22.2)
2367.0
.421
/Extra-Dimensional
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Set-Shift Task:
Total Errorsb
All items are Mean (SD) unless noted
Statistics are Mann-Whitney Test (U) unless noted, *Chi Square Test, P-value Bonferroni corrected is significant at .01
BFRB=Body-Focused Repetitive Behaviors, SUD= Substance Use Disorder, N=Number of subjects, %=Percent of subjects
a. Sample sizes differ for this variable: with 1st degree relative with BFRB: n=117; without 1st degree relative with BFRB: n=48; with 1st degree relative with
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