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Body Focused Repetitive Behavior Disorders: Significance of Family History

Sarah A. Redden, Eric W. Leppink, Jon E. Grant

PII: S0010-440X(15)30317-5

DOI: doi:10.1016/j.comppsych.2016.02.003

Reference: YCOMP 51629

To appear in: Comprehensive Psychiatry

Please cite this article as: Redden Sarah A., Leppink Eric W., Grant Jon E., BodyFocused Repetitive Behavior Disorders: Significance of Family History, ComprehensivePsychiatry (2016), doi:10.1016/j.comppsych.2016.02.003

This is a PDF file of an unedited manuscript that has been accepted for publication.As a service to our customers we are providing this early version of the manuscript.The manuscript will undergo copyediting, typesetting, and review of the resulting proof

http://dx.doi.org/10.1016/j.comppsych.2016.02.003http://dx.doi.org/10.1016/j.comppsych.2016.02.003http://dx.doi.org/10.1016/j.comppsych.2016.02.003http://dx.doi.org/10.1016/j.comppsych.2016.02.003

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Body Focused Repetitive Behavior Disorders: Significance of Family History

Sarah A. Redden, BA

Eric W. Leppink, BA

Jon E. Grant, JD, MD.

Department of Psychiatry & Behavioral Neuroscience, University of Chicago,

Pritzker School of Medicine, Chicago, IL, USA

Address correspondence to:

Jon E. Grant, J.D., M.D., M.P.H.

Department of Psychiatry & Behavioral Neuroscience

University of Chicago, Pritzker School of Medicine

5841 S. Maryland Avenue, MC-3077, Chicago, IL 60637

Telephone Number: 773-834-1325; Fax: 773-834-6761;

Email: jongrant@uchicago.edu

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Running title: Family history in trichotillomania and skin picking

Abstract

Background:The significance of family history in body-focused repetitive behavior disorders

(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

substance use disorder (SUD).

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

number of clinical and cognitive measures.

Results:77 (29.1%) participants had a first-degree family member with a BFRB and 59 (22.2%)

had a first-degree family member with a SUD. In terms of clinical severity, the amount of time

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

with a positive family history of a SUD. There were no significant cognitive differences based on

family history.

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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

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

skin picking disorder. Therefore, understanding the clinical and neurocognitive aspects of

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

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

clinical presentation than those without, and whether analysis of different familieshistories has

any clinical relevance.

2. Materials and methods

2.1 Subjects

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implementation of any treatments. Data were collected from September 2006 through January

2015.

2.2 Assessments

Participants were asked about the presence of lifetime BFRBs and SUDs (which included

alcohol and drug use disorders, but not nicotine) in all first-degree relatives. As some picking

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

definition of skin picking disorder or trichotillomania in a family member. Substance use

disorders were defined as the chronic use of drugs or alcohol resulting in either noticeable social

and occupational dysfunction or the need for a twelve-step program or formal treatment. All

information about relatives came from the proband. No direct evaluations of the first-degree

relatives were performed.

Current and lifetime psychiatric comorbidity was assessed using the Structured Clinical

Interview for DSM-IV (SCID) disorders23

and valid and reliable SCID-compatible modules for

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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 =

among the most extremely ill. The scale was used to assess only the severity of theBFRB

symptoms.

Sheehan Disability Scale (SDS).26The SDS is a valid and reliable, three-item, self-report

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.

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,

friendships, self-esteem, and standard of living.

Current depressive and anxiety symptoms were assessed using the 17-itemHamilton

Depression Rating Scale28

and theHamilton Anxiety Rating Scale,29

respectively.

2.3 Cogniti ve Testing

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2.4 Data Analysis

Based on the family history, participants were categorized into the following groups:

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

variables. Potential differences between the groups were explored using analysis of variance

(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

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

Bonferroni adjusted p

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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.

In terms of clinical measures, nothing was significant based on the Bonferroni

correction; however, those with a first-degree relative with a SUD reported more time each day

pulling or picking and greater depression symptomatology. There were also no statistically

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

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

without a family history of BFRBs. No significant cognitive differences were found based on

family history.

4. Discussion

The study confirms previous research showing that BFRBs are familial,13,14,19,20

as

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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

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

motor area involved in conditioned responses and response suppression).36,37In these

individuals, picking and pulling may function like an addiction and, although it awaits future

research, may even have a shared neurocircuitry with addictions.

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

individuals could mean that some sort of impulsivity is familial and gives rise to ADHD, more

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

controls. Those with trichotillomania demonstrated impaired performance versus controls, with

first-degree relatives occupying an intermediate position, thereby suggestive of some familial

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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

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

treatment remains uncertain as this time.

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

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

BFRBs remains unanswered. Additionally, whether this family history may be associated with

treatment response has also not been examined.

5. Conclusions

This study is the first to compare clinical measures based on two family history analyses

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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

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

DegreeRelativewith BFRB

(N=77)

Without 1st

DegreeRelativewith BFRB

(N=188)

Statistic p-value

With 1st

DegreeRelative withSUD

(N=59)

Without 1st

DegreeRelativewith SUD

(N=206)

Statistic p-value

DEMOGRAPHICS

Age (years) 34.4(11.4) 32.7 (11.2) 6656.5 .304 33.9 (11.4) 33.0 (11.2) 5782.5 .339

Age at Onset (years) 12.3 (6.8) 13.0 (7.9) 7034.0 .718 13.4 (9.3) 12.7 (7.0) 6069.0 .988

Sex, N (%),Female 70 (90.9) 167 (88.8) 1.269* .530 54 (91.5) 183 (88.8) .816* .665

Race, N(%),

Caucasian70 (90.9) 161 (85.6) 8.752* .119 48 (81.4) 183 (88.8) 9.649* .086

CLINICALTime spent in past

week pulling or

picking(minutes/day)

97.6(84.3) 86.6 (68.7) 4450.0 .606 117.9 (90.7) 81.7 (65.8) 3129.0 .022

Clinical Global

Impression-Severity4.44 (0.8) 4.40 (0.7) 7184.0 .914 4.5 (0.8) 4.4 (0.7) 5493.0 .202

Sheehan Disability

Scale10.8 (6.7) 10.8 (6.5) .003

+.955 10.7 (6.9) 10.9 (6.4) .027

+.870

Quality of Life

Inventory (T-score)43.5 (10.1) 43.0 (12.6) 7042.0 .963 42.0 (12.2) 43.5 (11.8) 5498.0 .339

Hamilton DepressionRating Scale

4.2 (3.5) 4.4 (3.6) 6984.0 .652 5.2 (3.6) 4.1 (3.5) 4982.0 .034

Hamilton Anxiety

Rating Scale4.3 (3.6) 4.3 (3.4) 7220.0 .975 5.1 (3.8) 4.1 (3.4) 5142.5 .070

History of BFRB

Treatment, N (%)37 (48.1) 74 (39.4) 1.695* .193 24 (40.7) 87 (42.2) .046* .831

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

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 SUDWith 1st

Degree

Relative

with BFRB(N=77)

Without 1st

Degree

Relative

with BFRB(N=188)

Statistic p-value

With 1st

Degree

Relative with

SUD(N=59)

Without 1st

Degree

Relative

with SUD(N=206)

Statistic p-value

COMORBIDITIES

Major Depressive

Disorder, N (%)28 (36.4) 72 (38.3) .087* .768 24 (40.7) 76 (36.9) .280* .597

Any Anxiety

Disorder, N (%)15 (19.5) 37 (19.7) .001* .970 11 (18.6) 41 (19.9) .046* .830

Obsessive

Compulsive

Disorder, N (%)

6 (7.8) 9 (4.8) .924* .336 4 (6.7) 11 (5.3) .1788* .673

Attention Deficit

Hyperactivity

Disorder, N (%)

8 (10.4) 17 (9.0) .116* .733 12 (20.3) 13 (6.3) 10.564* .001

Any Lifetime

Disorder, N (%)42 (54.5) 100 (53.2) .040* .841 37 (62.7) 105 (51.0) 2.542* .111

COGNITIVE VARIABLES

Stop Signal ReactionTime

a 186.2 (65.7) 191.7 (68.7) 2711.5 .729 202.3 (86.9) 186.3 (60.5) 2279.0 .795

Intra-

Dimensional/Extra-

Dimensional Set-

Shift Task: Extra-

Dimensional Shiftb

9.9 (10.5) 8.6 (9.8) 2745.5 .694 9.5 (10.1) 8.8 (10.0) 2318.5 .322

Intra-Dimensional

/Extra-Dimensional24.3 (20.1) 21.5 (21.9) 2604.0 .372 22.3 (19.0) 22.3 (22.2) 2367.0 .421

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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 subjectsa.Sample sizes differ for this variable: with 1stdegree relative with BFRB: n=117; without 1stdegree relative with BFRB: n=48; with 1 stdegree relative withSUD: n=126; without 1

stdegree relative with SUD: n=39.

b. Sample sizes differ for these variables: with 1stdegree relative with BFRB: n=119; without 1

stdegree relative with BFRB: n=48; with 1

stdegree relative with

SUD: n=126; without 1stdegree relative with SUD: n=41.