NIH Clinical Center Search the Studies: Study Number, Study Title

Protocol Details

Mapping the Genotype, Phenotype, and Natural History of Phelan McDermid Syndrome

This study is NOT currently recruiting participants.

Summary | Eligibility | Citations | Contacts

Summary

Number

15-M-0139

Sponsoring Institute

National Institute of Mental Health (NIMH)

Recruitment Detail

Type: No longer recruiting/follow-up only
Gender: Male & Female
Min Age: 1 Years
Max Age: N/A

Referral Letter Required

No

Population Exclusion(s)

None

Keywords

Biomarkers;
Phenotyping;
Brain Imaging;
Phelan McDermid Syndrome;
Neurodevelopmental;
Natural History

Recruitment Keyword(s)

None

Condition(s)

Phelan McDermid Syndrome;
22q13 Deletion Syndrome;
Pathogenic Deletions or Mutations of the SHANK3 gene

Investigational Drug(s)

None

Investigational Device(s)

None

Intervention(s)

None

Supporting Site

National Institute of Mental Health

Background:

- Phelan-McDermid Syndrome (PMS) is a rare disease caused by a change in a gene. People with Phelan-McDermid Syndrome often have intellectual disability or autism or other delays in development. They may also have sleep problems, seizures, kidney problems, and other symptoms. Researchers want to learn more about this disease.

Objective:

- To learn more about the medical, behavioral, and cognitive features of Phelan-McDermid Syndrome; to follow the natural history of the syndrome; to identify additional genetic factors that contribute to the symptoms.

Eligibility:

- People ages 18months 21 who have been diagnosed with Phelan-McDermid Syndrome

- First-degree relatives of people with Phelan-McDermid Syndrome.

Design:

- Participants will have 5 study visits over 3 years.

- At the start, 12-month, and 24-month visits, participants will visit the study center for several tests. They will have blood drawn. They will have cognitive testing. Their head size, weight, and height will be measured.

- Participants will have physical and neurological exams. They may be asked to identify objects, focus on certain information, solve problems, or play games.

- Parents will be asked questions about their medical and family history. They will get questions about the participant s current symptoms, functioning, and communication.

- Photos of the participant s feet, face, and hands will be taken.

- At the 6- and 18-month points, participants or parents will complete questionnaires at home.

- Researchers may ask to access the participant s or parent s medical records.

- If the participant has certain scans or heart tests due, researchers will ask for the results. They may add 15 minutes to a clinically indicated MRI for research.

- Relatives in this study may be asked to give blood once a year for 3 years.

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Eligibility

INCLUSION CRITERIA (PMS):

1. Individuals 18 months of age or older at time of enrollment with pathogenic deletions or mutations of the SHANK3 gene

2. Primary communicative language is English or Spanish.

INCLUSION CRITERIA (Parents and Siblings):

1. Individuals must be biological first-degree relatives whose primary communicative language is English or Spanish with capacity to provide consent or a legal guardian who may provide consent on their behalf.

INCLUSION CRITERIA (Controls):

1. Between the ages of 2-11 years

2. No underlying genetic diagnosis or other chronic medical condition associated with increased risk for ASD (e.g. traumatic brain injury, cerebral anoxia, intrauterine drug exposure)

3. Typical neurodevelopment for age (i.e., no established diagnosis or clinical suspicion for ASD or ID)

A subset of at least 5 (n=5) subjects and their participating relatives will be Spanish speaking to be included as part of the Administrative Diversity Supplement.

INCLUSION CRITERIA (Parent and Unaffected Siblings):

1. Individuals must be biological first-degree relatives who are English speaking with capacity to provide consent. A legal guardian may provide consent on their behalf and assent will be obtained when relevant.

EXCLUSION CRITERIA (PMS and Controls):

1. Has taken an investigational drug as part of another research study, within 30 days prior to study enrollment

2. For subjects involved in imaging biomarker assessment: contraindications to 3T MRI scanning, such as metal implants/non-compatible medical devices or medical conditions, including vagus nerve stimulator

3. For subjects involved in EEG/ ERP biomarker assessment: contraindications to EEG/ERP, such as uncooperative or destructive behaviors preventing lead placement or capture by ERP/VEP equipment. Under the age of 2 or over the age of 11 at the

time of enrollment.

4. Unwilling or unable to comply with study procedures and assessment

5. For healthy control subjects: Auditory or visual impairments that are uncorrected


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Citations:

Boeckers TM. The postsynaptic density. Cell Tissue Res. 2006 Nov;326(2):409-22. Epub 2006 Jul 25.

Betancur C, Buxbaum JD. SHANK3 haploinsufficiency: a "common" but underdiagnosed highly penetrant monogenic cause of autism spectrum disorders. Mol Autism. 2013 Jun 11;4(1):17. doi: 10.1186/2040-2392-4-17.

Cooper GM, Coe BP, Girirajan S, Rosenfeld JA, Vu TH, Baker C, Williams C, Stalker H, Hamid R, Hannig V, Abdel-Hamid H, Bader P, McCracken E, Niyazov D, Leppig K, Thiese H, Hummel M, Alexander N, Gorski J, Kussmann J, Shashi V, Johnson K, Rehder C, Ballif BC, Shaffer LG, Eichler EE. A copy number variation morbidity map of developmental delay. Nat Genet. 2011 Aug 14;43(9):838-46. doi: 10.1038/ng.909. Erratum in: Nat Genet. 2014 Sep;46(9):1040.

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Contacts:

Principal Investigator

Referral Contact

For more information:

Audrey E. Thurm, Ph.D.
National Institute of Mental Health (NIMH)



Margaret J. Pekar
National Institute of Mental Health (NIMH)
National Institutes of Health
Building 10
Room 1C250
10 Center Drive
Bethesda, Maryland 20892
(301) 435-7962
pekarm@mail.nih.gov

Office of Patient Recruitment
National Institutes of Health Clinical Center (CC)
Building 61, 10 Cloister Court
Bethesda, Maryland 20892
Toll Free: 1-800-411-1222
Local Phone: 301-451-4383
TTY: TTY Users Dial 7-1-1
ccopr@nih.gov

Clinical Trials Number:

NCT02461420

--Back to Top--

NIH Clinical Center Search the Studies: Study Number, Study Title

Protocol Details

Mapping the Genotype, Phenotype, and Natural History of Phelan McDermid Syndrome

This study is NOT currently recruiting participants.

Summary | Eligibility | Citations | Contacts

Summary

Number

15-M-0139

Sponsoring Institute

National Institute of Mental Health (NIMH)

Recruitment Detail

Type: No longer recruiting/follow-up only
Gender: Male & Female
Min Age: 1 Years
Max Age: N/A

Referral Letter Required

No

Population Exclusion(s)

None

Keywords

Biomarkers;
Phenotyping;
Brain Imaging;
Phelan McDermid Syndrome;
Neurodevelopmental;
Natural History

Recruitment Keyword(s)

None

Condition(s)

Phelan McDermid Syndrome;
22q13 Deletion Syndrome;
Pathogenic Deletions or Mutations of the SHANK3 gene

Investigational Drug(s)

None

Investigational Device(s)

None

Intervention(s)

None

Supporting Site

National Institute of Mental Health

Background:

- Phelan-McDermid Syndrome (PMS) is a rare disease caused by a change in a gene. People with Phelan-McDermid Syndrome often have intellectual disability or autism or other delays in development. They may also have sleep problems, seizures, kidney problems, and other symptoms. Researchers want to learn more about this disease.

Objective:

- To learn more about the medical, behavioral, and cognitive features of Phelan-McDermid Syndrome; to follow the natural history of the syndrome; to identify additional genetic factors that contribute to the symptoms.

Eligibility:

- People ages 18months 21 who have been diagnosed with Phelan-McDermid Syndrome

- First-degree relatives of people with Phelan-McDermid Syndrome.

Design:

- Participants will have 5 study visits over 3 years.

- At the start, 12-month, and 24-month visits, participants will visit the study center for several tests. They will have blood drawn. They will have cognitive testing. Their head size, weight, and height will be measured.

- Participants will have physical and neurological exams. They may be asked to identify objects, focus on certain information, solve problems, or play games.

- Parents will be asked questions about their medical and family history. They will get questions about the participant s current symptoms, functioning, and communication.

- Photos of the participant s feet, face, and hands will be taken.

- At the 6- and 18-month points, participants or parents will complete questionnaires at home.

- Researchers may ask to access the participant s or parent s medical records.

- If the participant has certain scans or heart tests due, researchers will ask for the results. They may add 15 minutes to a clinically indicated MRI for research.

- Relatives in this study may be asked to give blood once a year for 3 years.

--Back to Top--

Eligibility

INCLUSION CRITERIA (PMS):

1. Individuals 18 months of age or older at time of enrollment with pathogenic deletions or mutations of the SHANK3 gene

2. Primary communicative language is English or Spanish.

INCLUSION CRITERIA (Parents and Siblings):

1. Individuals must be biological first-degree relatives whose primary communicative language is English or Spanish with capacity to provide consent or a legal guardian who may provide consent on their behalf.

INCLUSION CRITERIA (Controls):

1. Between the ages of 2-11 years

2. No underlying genetic diagnosis or other chronic medical condition associated with increased risk for ASD (e.g. traumatic brain injury, cerebral anoxia, intrauterine drug exposure)

3. Typical neurodevelopment for age (i.e., no established diagnosis or clinical suspicion for ASD or ID)

A subset of at least 5 (n=5) subjects and their participating relatives will be Spanish speaking to be included as part of the Administrative Diversity Supplement.

INCLUSION CRITERIA (Parent and Unaffected Siblings):

1. Individuals must be biological first-degree relatives who are English speaking with capacity to provide consent. A legal guardian may provide consent on their behalf and assent will be obtained when relevant.

EXCLUSION CRITERIA (PMS and Controls):

1. Has taken an investigational drug as part of another research study, within 30 days prior to study enrollment

2. For subjects involved in imaging biomarker assessment: contraindications to 3T MRI scanning, such as metal implants/non-compatible medical devices or medical conditions, including vagus nerve stimulator

3. For subjects involved in EEG/ ERP biomarker assessment: contraindications to EEG/ERP, such as uncooperative or destructive behaviors preventing lead placement or capture by ERP/VEP equipment. Under the age of 2 or over the age of 11 at the

time of enrollment.

4. Unwilling or unable to comply with study procedures and assessment

5. For healthy control subjects: Auditory or visual impairments that are uncorrected


--Back to Top--

Citations:

Boeckers TM. The postsynaptic density. Cell Tissue Res. 2006 Nov;326(2):409-22. Epub 2006 Jul 25.

Betancur C, Buxbaum JD. SHANK3 haploinsufficiency: a "common" but underdiagnosed highly penetrant monogenic cause of autism spectrum disorders. Mol Autism. 2013 Jun 11;4(1):17. doi: 10.1186/2040-2392-4-17.

Cooper GM, Coe BP, Girirajan S, Rosenfeld JA, Vu TH, Baker C, Williams C, Stalker H, Hamid R, Hannig V, Abdel-Hamid H, Bader P, McCracken E, Niyazov D, Leppig K, Thiese H, Hummel M, Alexander N, Gorski J, Kussmann J, Shashi V, Johnson K, Rehder C, Ballif BC, Shaffer LG, Eichler EE. A copy number variation morbidity map of developmental delay. Nat Genet. 2011 Aug 14;43(9):838-46. doi: 10.1038/ng.909. Erratum in: Nat Genet. 2014 Sep;46(9):1040.

--Back to Top--

Contacts:

Principal Investigator

Referral Contact

For more information:

Audrey E. Thurm, Ph.D.
National Institute of Mental Health (NIMH)



Margaret J. Pekar
National Institute of Mental Health (NIMH)
National Institutes of Health
Building 10
Room 1C250
10 Center Drive
Bethesda, Maryland 20892
(301) 435-7962
pekarm@mail.nih.gov

Office of Patient Recruitment
National Institutes of Health Clinical Center (CC)
Building 61, 10 Cloister Court
Bethesda, Maryland 20892
Toll Free: 1-800-411-1222
Local Phone: 301-451-4383
TTY: TTY Users Dial 7-1-1
ccopr@nih.gov

Clinical Trials Number:

NCT02461420

--Back to Top--