Supporting Statement Part A
Documenting outcomes associated with persistent tic disorders (including Tourette syndrome) in children, adolescents, and young adults through surveillance
New
Rebecca Bitsko, PhD
Health Scientist
Centers for Disease Control and Prevention
Email: dvk2@cdc.gov
Phone: 404.498.3556
and
Lara Robinson, MPH, PhD
Team Lead
Centers for Disease Control and Prevention
Email: lpr0@cdc.gov
Phone: 404.498.3822
Section A.1. Circumstances Making the Collection of Information Necessary 4
Section A.2. Purpose and Use of Information Collection 5
Section A.3. Use of Improved Information Technology and Burden Reduction 6
Section A.4. Efforts to Identify Duplication and Use of Similar Information 6
Section A.5. Impact on Small Businesses or Other Small Entities 7
Section A.6. Consequences of Collecting the Information Less Frequently 7
Section A.7. Special Circumstances Relating to the Guidelines of 5 CFR 1320.5 7
Section A.9. Explanation of Any Payment or Gift to Respondents 8
Section A.10. Assurance of Confidentiality Provided to Respondents 8
Section A.11. Justification for Sensitive Questions 9
Section A.12. Estimates of Annualized Burden Hours and Costs 10
Section A.13. Estimates of Other Total Annual Cost Burden to Respondents or Record Keepers 11
Section A.14. Annualized Cost to the Government 12
Section A.15. Explanation for Program Changes or Adjustments 12
Section A.16. Plans for Tabulation and Publication and Project Time Schedule 12
Section A.17. Reason(s) Display of OMB Expiration Date Is Inappropriate 13
Section A.18. Exceptions to Certification for Paperwork Reduction Act Submissions 13
Attachment 1. Authorizing Legislation
Attachment 2. 60-day Federal Register Notice
Attachment 3-7. Surveys with questions, responses, and original source of questions
Attachment 3: Adult Survey and clinical assessment
Attachment 4: Parent Survey and clinical assessment
Attachment 5: Teen Survey and clinical assessment
Attachment 6: Child 9-11 Survey and clinical assessment
Attachment 7: Child 4-8 Clinical assessment (YGTSS only)
Attachment 8-12. Surveys in REDCap (note: skip patterns not reflected in REDCap attachments)
Attachment 8: Adult Survey in REDCap
Attachment 9: Parent Survey in REDCap
Attachment 10: Teen Survey in REDCap
Attachment 11: Child 9-11 Survey in REDCap
Attachment 12. Summary of content assessed for each respondent type.
Attachment 13. Appropriations letter
Attachment 14. Thank you note
Attachment 15. Research Determination
Attachment 16. Privacy Impact assessment
Attachment 17. Introductory Letter
Attachment 18. Participant Information Sheet for Parents/Children
Attachment 19. Participant Information Sheet for Adults
Attachment 20. Suicide resources
Attachment 21. References
Supporting Statement Part A. Justification
A. Justification
This Information Collection Request is submitted under the classification “New” request. The length of data collection requested for Office of Management and Budget (OMB) approval is 3 years. The National Center on Birth Defects and Developmental Disabilities (NCBDDD) at the Centers for Disease Control and Prevention (CDC) is making this request as authorized by Sections 301(a) and 317C of the Public Health Service Act, [42 U.S.C. Sections 241(a) and 247b-4, as amended] (Attachment 1).
A copy of the agency’s 60-day Federal Register Notice is attached (60-day Federal Register Notice Attachment 2).
Background
There are an estimated 1.4 million people in the U.S. affected by persistent tic disorders (PTD), including Tourette syndrome (TS).[1] To support people with these conditions, the impact of PTD/TS must be understood. Although some data on the impact of PTD/TS on social relationships and education are available, other potential outcomes associated with PTD/TS have not been well-documented, including costs associated with tic disorders, suicidality risk, health care transition, and how co-occurring disorders modify these outcomes. Limited data are available on how these outcomes may differ among sub-populations (e.g., by sex, race/ethnicity, age group, and geography [e.g., urban/rural]). Data on PTD/TS are particularly limited for adults.
A single source of population-based surveillance data exists for Tourette syndrome, the National Survey of Children’s Health (NSCH; OMB No. 0607-0990, exp. 04/30/2027), which is funded by the Health Resources and Services Administration (HRSA) and conducted by the U.S. Census. While this survey has been beneficial in describing some impacts associated with TS including bullying, parenting aggravation, healthcare needs, and school outcomes,[2-8] it does not include other persistent tic disorders that occur in approximately 1.6% of children, information on the priority outcomes of associated costs and suicidality risk, or information on key co-occurring disorders such as obsessive-compulsive disorder. NSCH is based on parent-report, and diagnosis of TS is not validated. Additionally, although the survey has a large sample size, only a small number of minority children and female children with TS are included in the NSCH data, which limits our ability to describe different outcomes by categories of race/ethnicity or among females.
In 2014, CDC funded a follow-up survey of the NSCH, the National Survey of the Diagnosis and Treatment of ADHD and Tourette syndrome (NS-DATA) to better understand characteristics of children with ADHD and TS. NS-DATA provided needed data on the diagnostic process, current treatment, and indicators of impairment but had a limited sample size that was not nationally representative and lacked a control group of children without ADHD or TS.[9-12]
CDC/NCBDDD funded a study that collected data in 2009-2011 on outcomes associated with TS focused on social outcomes (e.g., social problems, social deficits, self-concept) as well as the prevalence and impact of co-occurring conditions.[13] While not intended to document information on suicide, the measures of depression included in the study identified suicidal ideation as a potential important impact to study,[14] and therefore that is one of the priorities of the current project. Additionally, information from the 2009-2011 study is nearly 15 years old and contemporary information is needed.
Current population-based surveillance and research among children and adolescents with Tourette syndrome relies on data from the NSCH or administrative healthcare data. The scant data on outcomes among children, adolescents, and young adults with PTD/TS and absence of information on outcomes by other factors including age, sex, and type of tic disorder is insufficient to provide insight into the public health questions that remain for children, adolescents, and young adults with PTD/TS or to develop services and allocate resources designed to improve long-term health and wellbeing.
This data collection aims to document the impact of PTD/TS on outcomes including associated costs, prevalence of suicidality risk, transition to adult healthcare, and co-occurring conditions among children and adolescents aged 4–17 years, as well as among young adults aged 18–26 years with PTD/TS. Data will be collected primarily by electronic survey of parents of patients ages 4-17 years, child and adolescent patients aged 9-17 years, and young adult patients aged 18-26 years. In addition, there will be a clinical assessment including questions about current suicidal ideation and presence, type, and severity of tics; parents of children aged 4–17 years will participate in the clinical assessment alongside their child.
Patients and their parents will be recruited from 4 clinical sites. One site is The University of South Florida (USF) who will recruit from the Division of Pediatric Neuropsychiatry – Rothman Center, St. Petersburg, FL and the Division of Pediatric Neurology, Tampa, FL. The Tourette Association of America (TAA) will recruit from three sites that are designated as a TAA Center of Excellence. They have proposed the following three sites but have not finalized agreements and may select alternate sites: Mount Sinai West TAA Center of Excellence, New York, NY; Washington University TAA Center of Excellence, St. Louis, MO; University of Miami Miller School of Medicine TAA Center of Excellence, Miami, FL. Sites will identify eligible patients through usual clinic flow and outreach to patients who have previously been seen at the clinics for PTD/TS. The survey data will be linked with documentation of clinical criteria used to determine eligibility, data from the clinical assessment of current and past tic symptoms and severity and suicidality for individuals with tic disorders aged 9-26 years, as well as to additional clinical data from the medical record. These data will be used to inform where resources for families and healthcare providers (e.g., professional trainings) are most needed to support people with PTD/TS and their families and to address health inequities among the population.
Currently, Congress has appropriated $2 million per year to CDC to continue to educate physicians, educators, clinicians, allied professionals, and the general public about the disorder and to improve scientific knowledge on prevalence, risk factors, and co-occurring conditions of Tourette Syndrome. Project information and results will be disseminated to healthcare providers, patients, families, national patient and clinical organizations, the public, and other partners via emails, newsletters, websites, social media, publications, presentations, and other methods. Findings from this project will enable federal, state, and local governments and organizations to understand the needs of children, adolescents, and young adults with PTD/TS, allocate resources, and establish programs accordingly.
All survey data will be collected via electronic survey; respondents will be asked to complete the survey on their own but will be offered support if needed (e.g., reading of questions). The survey also includes skip patterns so that each respondent type (children, adolescents, parents, young adults) are only asked relevant questions (Attachments 3-11) and content (Attachment 12). Skip patterns are described in Attachments 3-7 but are not shown in the attachments showing the surveys coded in REDCap (Attachments 8-11).
As part of this new data collection, information available from electronic medical records (e.g., date of birth, health insurance status and type, co-occurring disorders, address, sex, race/ethnicity, treatment, Columbia Suicide Rating Scale) will be extracted and linked to the respondents’ survey information. These data will be evaluated for completeness and will inform whether ongoing collection of these data as part of the survey are needed, or if survey items can be eliminated.
We are also collecting individuals’ addresses to link to Census data to examine social vulnerability index, rurality, and distance to tic specialty clinics. Address data will be sent to CDC and the CDC program will work with CDC/ATSDR’s Geospatial Research, Analysis, and Services Program, or GRASP for the geocoding and linkage. Address data will be separated from the rest of the data, and once available, Census data (e.g., social vulnerability index, rurality, and distance to tic specialty clinics) will be linked to the main data sets for analysis.
Conversations with CDC program staff, partner organizations supporting families of children, adolescents, and young adults affected by PTD/TS, and healthcare providers with expertise in diagnosis and treatment of PTD/TS have identified the priority outcomes of PTD/TS, including costs, suicidality risk, health care transition, and the role of co-occurring disorders. These priorities were documented in a letter signed by over 100 members of Congress to the House Appropriations Subcommittee on Labor, Health, Human Services, and Education that specified needs related to documenting the impact of PTD/TS as well as better understanding priority needs (Attachment 13).
Most existing population-based national surveys collecting data on children, adolescents, and young adults do not inquire about PTD/TS (e.g., National Health and Nutrition Examination Survey (NHANES), OMB No. 0920-0950, exp. 04/30/2025, National Health Interview Survey (NHIS), OMB No. 0920-0214, exp. 12/31/2026). Only NSCH (OMB No. 0607-0990, exp. 04/30/2027) includes questions about Tourette syndrome in children but does not ask about other persistent tic disorders. Additionally, NSCH does not validate parent or caregiver responses. Thus far, these surveys have been used to broadly describe a limited selection of outcomes including healthcare needs and use, bullying, education, and parenting aggravation [2-8]. However, individuals with PTD/TS are a heterogeneous group, comprised of individuals with persistent motor tic disorder, persistent vocal tic disorder, and Tourette, and varying degrees of tic severity and tic-related impairment.
PTD/TS is a relatively rare condition, affecting approximately 2% of children and less than 1% of adults;[1] therefore, adding a question that would provide enough detail on PTD/TS type to these population-based surveys would not provide sufficient sample size to generate precise prevalence estimates for key impacts/outcomes among children with specific persistent tic disorders or TS.
Furthermore, data on outcomes of interest specific to those with PTD/TS, such as healthcare transition specific to care for PTD/TS, are not collected on existing surveys.
This data collection will not involve small businesses.
Each respondent will be asked to respond once. The project is designed to complement existing data, by including persistent tic disorders, including only individuals with clinically confirmed PTD/TS, and including clinical information including data from the electronic medical record. PTD/TS is not included in national surveys of adults or teens (e.g., NHIS, YRBS, NSDUH) and many of the questions incorporated on our surveys are from those data sources and will allow for new information to be collected on PTD/TS. These data are needed to inform the public health needs of and resource allocation for services for U.S. children, adolescents, and young adults with PTD/TS, a group that totals over 1.4 million.
There are no legal obstacles to reduce the burden.
This request fully complies with regulation 5 CFR 1320.5.
A copy of the agency’s 60-day Federal Register Notice is attached (60-day Federal Register Notice Attachment 2). The notice, as required by 5 CFR 1320.8 (d), was published on April 5, 2024 (Vol. 89, No. 67, pages 24007-24008). CDC received one public comment in response to this notice.
Respondents (one per parent/child pair) will receive a thank you note and $20 gift card upon completion of the survey as a token of appreciation (Attachment 14). Research suggests that providing tokens of appreciation to eligible participants increases response rates and prevents bias, making findings more generalizable to U.S. children, adolescents, and young adults with PTD/TS. Literature examining the benefit of tokens of appreciation for participation was summarized by Yu J, et al. in their paper “A quantitative review of research design effects on response rates to questionnaires”.[15] It reviewed 497 response rates found in 93 journal articles and found that response rates increased with monetary and non-monetary gifts to participants.
The proposed data collection will have no anticipated effect on the respondent’s privacy.
The research determination for this project has declared it “Not Research – Public Health Surveillance” and therefore exempt from IRB review (Attachment 15). However, this data collection effort is subject to the Privacy Act and will be managed in accordance with CDC’s System of Records Notice (SORN) #09-20-0136, Epidemiologic Studies and Surveillance of Disease Problems, Department of Health and Human Services/CDC/National Center for Infectious Diseases. A privacy impact assessment for this project was reviewed and approved by a CDC Senior Official for Privacy (See Privacy Act Attachment 16).
A brief clinical assessment (Yale Global Tic Severity Scale [YGTSS], Ask Suicide Screening Questions [ASQ]) and an electronic survey will be conducted at the clinic or during a telehealth visit (Attachments 3-11). Once all questions related to suicide risk are completed, respondents can complete the survey on their own, outside the clinic or telehealth appointment, if they request additional time to complete the survey. Data will be stored at the sites, linked with clinical data, and shared with CDC through secure data-transfer systems. Electronic data will be kept on password protected systems only accessible by Site and CDC project staff. The sites will conduct initial data cleaning and the CDC data manager will clean and remove Information in Identifiable Form from the survey data and create a dataset for use by project staff. A separate dataset will be saved with only the respondent ID and address information for use by GRASP. The dataset will not include Information in Identifiable Form. Project staff at funded sites may be given access to project data stored at CDC through secure data-transfer systems. Before obtaining the data, project staff must sign a Confidentiality and Data Use Acknowledgement form assuring they will not use these data in any way except for statistical reporting and analysis; they will not share the individual-level data with anyone not named on the data sharing and confidentiality form; they will not attempt to use the dataset to learn the identity of any person or establishment, and they will use reasonable measures to protect all individual-level data from eye observation, theft, or accidental loss or misplacement.
An introductory letter (Attachment 17) and participant information sheet (Attachments 18-19) will be provided to all adults and parents of children aged 4-17 years eligible to serve as respondents in the surveillance project. Children and adolescents aged 9-17 will also be provided the parent participant information sheet. The participant information sheet 1) informs potential respondents about the purpose and procedures of the project, 2) states that there are no known risks to the respondent and all personal information will be kept private to the extent allowed under federal laws, 3) states there is no benefit to completing the survey, but answers may help identify the impact of PTD/TS which can be used to inform future activities aimed at improving the health and wellbeing of individuals with PTD/TS. Further, respondents will be reminded that 1) their participation is voluntary and 2) they may choose not to answer a question at any time or may withdraw their survey from the project.
Because this work presents no more than minimal risk of harm to subjects and involves no procedures for which written consent is normally required outside of the research context, we will not request written documentation of informed consent/assent. However, the sites may require IRB in which case informed consent/assent may be incorporated into the project.
The survey asks questions about topics that may be considered sensitive: medical diagnoses and treatment, disabilities, special healthcare needs, use of mental health services, healthcare costs, and sexual orientation, income, employment, school absenteeism, and suicide. These topics are included in the survey because several reports indicate they are important issues for children, adolescents, or young adults with PTD/TS, and/or may be associated with priority outcomes. As mentioned, the participant information sheet (Attachment 18) states that participation is voluntary, nothing will happen if the person decides not to respond, that the respondent may skip any question they do not wish to answer, and that all information collected will be kept confidential. Additionally, the survey instrument also states that the respondent may skip any question he/she does not wish to answer (Attachments 3-11), and for surveys including questions on suicidality (i.e., adult and teen surveys), “This survey asks about mental health and emotional well-being. If you answer that you have had suicidal thoughts or behaviors, or purposely tried to hurt yourself, we may inform your doctor or other clinic staff. This would be to ensure your safety and provide you with support and care. By completing this survey, you accept and consent to this protocol. If you have concerns or need immediate help, please tell the clinic staff.” All respondents aged 9-26 years and parents serving as respondents will be provided information on the 988 Suicide and Crisis Lifeline (Attachment 20), and each site will have a protocol for immediately assessing any endorsement of assessments and survey items including self-harm and suicidality. There is also a statement in the introductory letter that reads: “None of your answers or your child’s answers will be linked to your name or your child’s name. Your child’s name will never be released as having PTD/TS. Your name will never be released as having completed the survey or having been asked to participate.” (Attachment 17). Additionally, most questions on this survey are derived from national surveys, including the National Survey of Children’s Health (OMB No. 0607-0990, exp. 04/30/2027), the National Health Interview Survey (NHIS OMB No. 0920-0214, exp. 12/31/2026), and NHIS-Teen (NHIS Teen OMB No. 0920-0214, exp. 12/31/2023) which has been found acceptable by parents, teens, and adults across the United States. Each site will create a protocol to implement immediate follow-up in cases where individuals endorse any items related to self-harm.
A question about race/ethnicity is included to assess differences in outcomes among individuals with PTD/TS by race/ethnicity. The program chose to include the mid-level of detail question on race/ethnicity with minimum categories only and examples. The question stem includes race and/or ethnicity and “select all that apply” and response options include each of the 7 minimum categories and detailed category examples that exactly match the 2024 SPD 15. This is included in the parent-report and adult self-report surveys (Attachments 3, 4, 8, 9). We selected the mid-level option because our sample size is relatively small and collecting additional details on race/ethnicity would pose additional burden that we would be unable to report at a greater level of detail due to the smaller number of respondents.
It is estimated that 2,100 respondents (1200 patients with PTD/TS and 900 parents of the underage patients) in total across all 3 years will complete the survey. The survey will be conducted 1 time only. Data will be collected at four sites, and we expect each site to recruit approximately 100 unique individuals with PTD/TS per year (total N=400/year), of which 75% will be children (n=300), and 25% young adults (n=100). Parents will complete a 25-minute survey and participate in a 20-minute clinical assessment (YGTSS) for all children ages 4–17 years (300 parents at 45 minutes for a total of 225 hours). Young children ages 4–8 years will participate in a clinical assessment of tics only (YGTSS; 60 children at 20 minutes for a total of 20 hours). Children ages 9–11 years will complete a 10-minute survey and 20-minute clinical assessment of tics and suicidality (YGTSS, ASQ; n=100 at 30 minutes for a total of 50 hours). Adolescents ages 12–17 years will also complete a 25-minute survey and a 20-minute clinical assessment of tics and suicidality (YGTSS, ASQ; n=140 at 45 minutes for a total of 105 hours). Young adults ages 18–26 years will complete a 40-minute self-report survey and a 20-minute clinical assessment of tics and suicidality (YGTSS, ASQ; n=100 at 1 hour for a total of 100 hours). The burden per response for each respondent type, in hours, was estimated based on piloting a pretest of the form with 5 individuals and incorporating estimates of time needed to complete previously validated measures from the literature. Given a 3-year length of approval, corresponding annual estimates are 700 respondents per year and 500 burden hours per year.
There are no costs to respondents other than their time.
EXAMPLE OF BURDEN TABLE:
A.12.A. Estimated Annualized Burden Hours
Type of Respondents |
Form Name |
No. of Respondents |
No. Responses per Respondent |
Average Burden per Response (in hours) |
Total Burden Hours |
Parents of children 4–17 years with a persistent tic disorder |
Parent |
300 |
1 |
45/60 |
225 |
Children 4–8 years with a persistent tic disorder |
Child 4–8 |
60 |
1 |
20/60 |
20 |
Children 9–11 years with a persistent tic disorder |
Child 9–11 |
100 |
1 |
30/60 |
50 |
Adolescents (teens) 12–17 years with a persistent tic disorder |
Teen |
140 |
1 |
45/60 |
105 |
Adults (18–26 years) with a persistent tic disorder |
Adult |
100 |
1 |
1 |
|
TOTAL |
|
700 |
— |
— |
500 |
Annualized burden costs are summarized in the table below. The hourly wage estimates are based on the U.S Bureau of Labor Statistics, May 2022 National Occupational Employment and Wage Estimates (available at http://www.bls.gov/oes/current/oes_nat.htm). The median hourly wage rate for all occupations ($22.26) was used for parent and adult respondents.
A.12.B. Estimated Annualized Burden Costs
Type of Respondents |
Form Name |
Total Burden Hours |
Average Hourly Wage Rate ($) |
Total Respondent Burden Costs ($) |
Parents of children 4–17 years with a persistent tic disorder |
Parent |
225 |
$22.26 |
$5,008.50 |
Children 4–8 years with a persistent tic disorder |
Child 4–8 |
20 |
$22.26 |
$445.20 |
Children 9–11 years with a persistent tic disorder |
Child 9–11 |
50 |
$22.26 |
$1,113.00 |
Adolescents (teens) 12–17 years with a persistent tic disorder |
Teen |
105 |
$22.26 |
$2,337.30 |
Adults (18–26 years) with a persistent tic disorder |
Adult |
100 |
$22.26 |
$2,226.00 |
TOTAL |
|
500 |
— |
$ 11,130 |
There are no costs to respondents associated with either capital and startup efforts or operation and maintenance of services for this project.
The average annualized cost to the Government to collect this information is $951,595 for this 3-year OMB approval period that is requested.
|
Total ($) |
|
Federal Government Personnel costs |
CDC Project Officer |
41,799 |
CDC Project Manager |
49,394 |
|
CDC Project Oversight |
19,758 |
|
CDC Collaborator |
33,440 |
|
CDC Collaborator |
8,360 |
|
CDC Collaborator |
19,758 |
|
ORISE |
ORISE fellow |
19,391 |
|
ORISE fellow |
9,695 |
Funded Sites |
Awardee #1 (Tourette Association of America – 3 sites) |
550,000 |
Awardee #2 (University of South Florida) |
200,000 |
|
Total |
|
$951,595 |
This is a new data collection.
The recruitment materials will be finalized and printed in the first month after OMB approval. Throughout the first to 36 months after OMB approval, recruitment materials, which include the participant information sheet and survey questionnaire, will be presented by the surveillance sites to eligible patients and their parents or caregivers. Each site will aim to recruit 100 patient respondents per year, for a total of 400 patient respondents per year (totaling 700 respondents per year, including parents of patients). Thank you letters and $20 gift cards will be given or sent to all patient respondents upon completion of the survey (one for adults and one for each pair of parent/child respondents). Sites will link, clean, and share data with CDC for further linkage to census data and cleaning. Finally, data analysis and dissemination of findings is expected to occur between 36 and 48 months after OMB approval, although some preliminary results may be disseminated sooner.
A.16.—Project Time Schedule |
||
|
||
Activity |
Timeframe |
|
Recruit Patients and Caregivers |
Prepare and print recruitment materials for distribution |
1 month after OMB approval |
Recruit patients/caregivers. Share participant information sheet with eligible patients and their caregivers. |
1–36 months after OMB approval |
|
Provide thank you letters and gift cards to respondents (one for each adult respondent, and one for each parent/child pair of respondents) |
1-36 months after OMB approval |
|
Analyze and Report Data from Surveillance Project
|
Sites link survey data and clinical data, conduct initial data cleaning (including separation of date of birth, address, and email address from main shared data), and share data with CDC |
3-36 months after OMB approval |
CDC links data with census tract data, finalize cleaning including deidentification of address data, and merge data sets across sites |
3-40 months after OMB approval |
|
Analyze data, draft reports, and present findings |
40-48 months after OMB approval |
|
NA
There are no exceptions to the certification.
| File Type | application/vnd.openxmlformats-officedocument.wordprocessingml.document |
| Author | Bitsko, Rebecca (Becky) (CDC/NCBDDD/DHDD) |
| File Modified | 0000-00-00 |
| File Created | 2025-07-02 |