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Case Report

Case Report: DSM–5 misses an edge case in tic disorders nosology

[version 1; peer review: 2 approved]
PUBLISHED 03 Jun 2020
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This article is included in the Tics collection.

Abstract

A boy with multiple phonic tics, one lifetime motor tic, and no impairment or marked distress does not meet criteria for any DSM–5 tic disorder diagnosis. The next version of the Diagnostic and Statistical Manual should adjust the criteria for Tourette's Disorder and/or for "other specified tic disorder" and "unspecified tic disorder."

Keywords

Tourette syndrome, tic disorders, nosology, DSM-5, case report

Introduction

Over time, tic disorder nosology has changed1. The current research criteria changed modestly with the adoption of DSM–52. I describe a case of a child with motor and vocal tics that demonstrates a gap in DSM–5’s diagnostic criteria for tic disorders3.

Case report

At age 9, a right-handed, non-Hispanic white boy and his mother participated in a thorough research assessment as part of the New Tics study4. The study was approved by the Washington University in St. Louis Human Research Protection Office, IRB ID #201109157, and his mother gave informed consent. This visit included questionnaires, K-SADS parent and child interviews, history of illness, neurological exam, 30 minutes of observation of the child alone via video, and YGTSS rating. His mother was a reliable informant, an elementary school teacher well informed about tics, and his father was a physician. His mother dated his tic onset to 8 months ago, at age 8. The child had seasonal allergies but the phonic tics were present when he had no allergy symptoms, and the tics did not respond to cetirizine. He was taking extended-release mixed amphetamine salts (40 mg daily) for ADHD with good response. K-SADS diagnoses were specific phobia, past social anxiety disorder, past nocturnal enuresis, predominantly inattentive ADHD since age 6, and provisional tic disorder.

Neurological exam was normal except for a medium-loud snort occurring once during the exam. He had simple phonic tics (sniff or snort, cough, clear throat), one motor tic (biting lower lip softly, seen during video observation) and no complex tics. He bit his nails sometimes since early childhood, but this was not counted as a tic given the timing and its high prevalence among young children5. He also had one probable simple motor stereotypy (rarely shook his hands up and down near his chest before sports or social events since age 5 or younger; he said “I like doing that” and said it didn’t feel like his tics; seen only once in over an hour of observation). YGTSS scores were: motor tics 5, phonic tics 12, impairment 10.

He returned at 12 months after tic onset. The stimulant continued at the same dose, now without an antihistamine. The same tics continued within the past week, though not every day, but no tics were observed at the visit. YGTSS scores were: motor tics 4, phonic tics 6, impairment 0. By 24 months after tic onset, he was taking no medications. The lip biting had disappeared but the phonic tics continued. He reported that “they’re kind of annoying and I would like them to go away,” but he did not have marked distress and the tics did not affect self-esteem, family life, friendships or school functioning. No tics were observed during thorough history and a neurological examination, but sniffing, coughing and forceful nasal exhalations were observed by video when he was alone in the room. No motor tics were observed. YGTSS scores were: motor tics 0, phonic tics 9, impairment 0. Diagnostic Confidence Index score was 356.

Discussion

This boy has a fairly typical history for mild Tourette syndrome, except that he has only one motor tic. (Other clinicians may choose to count the nail biting or hand shaking as tics, but for the present discussion the main point is that some children will have a presentation with vocal tics and one motor tic.) The DSM–5 criteria for Tourette’s Disorder require “multiple” motor tics2. The criteria for Persistent (Chronic) Vocal Tic Disorder exclude patients who have experienced both motor and vocal tics. At the follow-up visits, the duration of ticcing excludes Provisional Tic Disorder, and the history and examination provided no evidence for causation by a substance or non-psychiatric illness. The residual categories, Other Specified Tic Disorder and Unspecified Tic Disorder, require “clinically significant distress or impairment in social, occupational, or other important areas of functioning.” This last criterion is shared with most DSM–5 disorders, but since DSM–IV–TR it has been omitted for Tourette’s Disorder7. Thus this boy does not meet DSM–5 criteria for any tic disorder. Woods and Thomsen addressed the situation in which a patient has vocal tics and exactly one motor tic, and concluded that “the requirement that multiple motor tics exist seems arbitrary and unnecessarily exclusive”8. A DSM–5 work group discussed the nosological issues in detail7. They retained the “multiple” motor tic requirement for Tourette’s Disorder, and did not propose reinstating the impairment or distress criterion in the “not otherwise specified” diagnosis. Roessner and colleagues provided critical feedback on the proposed criteria for tic disorders, but also assumed the impairment or distress criterion would be absent for all tic disorders9. This change appears to have been inadvertent.

Conclusions

There is no clinical import for this child, as his symptoms bother him only slightly. But this case demonstrates that the current DSM–5 criteria inadvertently provide no diagnosis in this case, which may occasionally affect research on tic disorders. The exclusion of one motor tic from both Tourette’s Disorder and Persistent Vocal Tic Disorder leaves a gap. The residual diagnostic categories no longer cover this gap since DSM–5 requires the “impairment or distress” criterion for them, though that requirement may have been accidental. I propose that future revisions omit it for all tic disorders. I also agree with Woods and Thomsen’s opinion that one motor tic and multiple phonic tics is best described as Tourette syndrome.

Data availability

All data underlying the results are available as part of the article and no additional source data are required.

Consent

Written informed consent for publication of their clinical details was obtained from the parent of the patient.

Comments on this article Comments (1)

Version 1
VERSION 1 PUBLISHED 03 Jun 2020
  • Author Response (F1000Research Advisory Board Member) 17 Nov 2023
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    17 Nov 2023
    Author Response F1000Research Advisory Board Member
    The following recent article substantially updates the available information on the relationship of nail-biting to Tourette syndrome:

    Hsueh C-W, Chen C-W: Prevalence of nail biting and its chronological relationship with tics ... Continue reading
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Black KJ. Case Report: DSM–5 misses an edge case in tic disorders nosology [version 1; peer review: 2 approved]. F1000Research 2020, 9:505 (https://doi.org/10.12688/f1000research.23991.1)
NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article.
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Open Peer Review

Current Reviewer Status: ?
Key to Reviewer Statuses VIEW
ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approvedFundamental flaws in the paper seriously undermine the findings and conclusions
Version 1
VERSION 1
PUBLISHED 03 Jun 2020
Views
11
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Reviewer Report 17 Jun 2020
Carol A. Mathews, Department of Psychiatry, Genetics Institute, University of Florida, Gainesville, FL, USA 
Approved
VIEWS 11
This case report discusses a 9 year old boy who presents with persistent, multiple vocal tics, and one motor tic.  Based on history and physical exam, he clearly meets criteria for multiple chronic tics, but has no demonstrated impairment. I ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Mathews CA. Reviewer Report For: Case Report: DSM–5 misses an edge case in tic disorders nosology [version 1; peer review: 2 approved]. F1000Research 2020, 9:505 (https://doi.org/10.5256/f1000research.26466.r64376)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response (F1000Research Advisory Board Member) 26 Jun 2020
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    26 Jun 2020
    Author Response F1000Research Advisory Board Member
    I appreciate Dr. Mathews's thoughtful input, including the 2 recent references, and agree with her comments about the likely nosological unity of the various DSM–5 chronic tic disorders including TS.
    Competing Interests: No competing interests were disclosed.
COMMENTS ON THIS REPORT
  • Author Response (F1000Research Advisory Board Member) 26 Jun 2020
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    26 Jun 2020
    Author Response F1000Research Advisory Board Member
    I appreciate Dr. Mathews's thoughtful input, including the 2 recent references, and agree with her comments about the likely nosological unity of the various DSM–5 chronic tic disorders including TS.
    Competing Interests: No competing interests were disclosed.
Views
10
Cite
Reviewer Report 11 Jun 2020
Andrea Cavanna, Department of Neuropsychiatry, BSMHFT, University of Birmingham, Birmingham, UK 
Approved
VIEWS 10
Kevin Black’s case report highlights a possible hole in the current classification scheme for tic disorders.
The author describes the clinical presentation of a 9-year-old boy who has a fairly typical history for mild Tourette syndrome, except that he ... Continue reading
CITE
CITE
HOW TO CITE THIS REPORT
Cavanna A. Reviewer Report For: Case Report: DSM–5 misses an edge case in tic disorders nosology [version 1; peer review: 2 approved]. F1000Research 2020, 9:505 (https://doi.org/10.5256/f1000research.26466.r64373)
NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article.
  • Author Response (F1000Research Advisory Board Member) 15 Jun 2020
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    15 Jun 2020
    Author Response F1000Research Advisory Board Member
    I thank Dr. Cavanna for the review. I agree with his comment about the artificial and historical nature of the distinction between motor and vocal tics.
    Competing Interests: No competing interests were disclosed.
COMMENTS ON THIS REPORT
  • Author Response (F1000Research Advisory Board Member) 15 Jun 2020
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    15 Jun 2020
    Author Response F1000Research Advisory Board Member
    I thank Dr. Cavanna for the review. I agree with his comment about the artificial and historical nature of the distinction between motor and vocal tics.
    Competing Interests: No competing interests were disclosed.

Comments on this article Comments (1)

Version 1
VERSION 1 PUBLISHED 03 Jun 2020
  • Author Response (F1000Research Advisory Board Member) 17 Nov 2023
    Kevin J Black, Departments of Psychiatry, Neurology, Radiology and Neuroscience, Washington University in St. Louis School of Medicine, St. Louis, 63110-1093, USA
    17 Nov 2023
    Author Response F1000Research Advisory Board Member
    The following recent article substantially updates the available information on the relationship of nail-biting to Tourette syndrome:

    Hsueh C-W, Chen C-W: Prevalence of nail biting and its chronological relationship with tics ... Continue reading
Alongside their report, reviewers assign a status to the article:
Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested
Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.
Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions
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