A Pilot Study to Evaluate the Hypoglossal Nerve Stimulator in Adolescents With Down Syndrome and Obstructive Sleep Apnea

NCT02344108 | Completed Results DMC FDA Device

• 2 other identifiers: 14-146H2019P001786
• Study Type: interventional
• Target: 42
• Locations: 1 country
• Sites: 5

Brief Summary

Obstructive sleep apnea (OSA) affects up to 1% of the general pediatric population and is associated with adverse behavior and quality of life, as well as long term cardiopulmonary system complications. Trisomy 21 (Down Syndrome) is the most common chromosomal disorder, with a incidence of approximately 1 per 660-800 births. Patients with Down Syndrome have a higher incidence of OSA than the general pediatric population, with rates of 30-60%, resulting in increased morbidity and decreased quality of life for affected individuals. In children, adenotonsillectomy (T\&A) is often a contributing factor to OSA, and adenotonsillectomy is a first line treatment. Children with Down Syndrome often undergo T\&A for obstructive sleep apnea, however 30-50% will have persistent obstructive sleep patterns requiring continuous positive pressure airway support (CPAP) or tracheotomy. Persistent obstruction is attributed to anatomic and physiologic differences in this population, including reduced muscular tone, macroglossia, maxillary hypoplasia, and lingual tonsil hypertrophy. This pilot study is designed to determine if the Inspire® Upper Airway Simulation System, Model 3024 IPG, and any subsequent iteration thereof that are approved under P130008 for the treatment of obstructive sleep apnea, which has already been approved for use in adults with OSA, can be safely implanted and used in adolescents and young adults with Down Syndrome.

Conditions

Sleep Apnea, Obstructive; Down Syndrome

Outcome Measures

Primary Outcomes (3)

• Serious Adverse Events

  In this study, adverse events were defined as "serious" if they resulted in: (1) death; (2) a life-threatening experience; (3) in-patient hospitalization or prolongation of hospital stay; (4) a persistent or significant disability/incapacity; (5) congenital anomaly/birth defect; or (6) events that jeopardized the health of the subject or required surgical intervention.

  1 year

• Non-Serious Adverse Events

  Non-serious adverse events include all other adverse events recorded during the study which were determined to be related or possibly related to the device, surgery, or research. It does not include adverse events that were determined to be serious, as previously defined. Safety events that were determined to be unrelated to the study were not included in this analysis.

  1 year

• Unanticipated Adverse Device Effects (UADE)

  Unanticipated adverse device effects (UADEs) are defined as adverse events which were determined to be serious, unexpected and related or possibly related to the investigational device.

  1 year

Secondary Outcomes (13)

• Apnea-hypopnea Index (AHI)

  1 year

• Obstructive Apnea-hypopnea Index

  1 year

• Central Apnea Index

  1 year

• Hypopnea Percentage

  1 year

• Oxygenation Percentage of Time SpO2 < 90%

  1 year

Study Arms (1)

Inspire® Upper Airway Simulation System

EXPERIMENTAL

Subjects meeting inclusion criteria, including sleep study and drug induced sleep endoscopy criteria, will undergo surgical placement of a hypoglossal nerve simulator (Inspire® Upper Airway Simulation System Model 3028 IPG). The simulator will be activated one month after surgery and subjects will undergo repeat sleep study evaluation and device titration at one, two, six and twelve months after implantation. Subjects will be followed for one year to determine safety and efficacy of the device.

Device: Inspire® Upper Airway Simulation System (Model 3028 IPG )

Interventions

Inspire® Upper Airway Simulation System (Model 3028 IPG ) DEVICE

Subjects will be implanted with a hypoglossal nerve stimulator. Safety and efficacy of implantation will be evaluated.

Also known as: hypoglossal nerve stimulator

Inspire® Upper Airway Simulation System

Eligibility Criteria

• Age: 10 Years - 21 Years
• Sex: all
• Healthy Volunteers: No
• Age Groups: Child (0-17), Adult (18-64)

You may qualify if:

• Only children and young adults with Down Syndrome age 10-21 years with prior adenotonsillectomy will be considered for the study.
• Subjects must have BMI \<95th percentile for age
• All subjects must have moderate to severe OSA (AHI \>10, AHI \<50, no more than 25% AHI attributable to central events) based on prior in-lab polysomnography performed after adenotonsillectomy.
• Subjects must have either tracheotomy or be ineffectively treated with CPAP due to noncompliance, discomfort, un-desirable side effects, persistent symptoms despite compliance use, or refusal to use the device.
• Children and their parents must be willing to have stimulation hardware permanently implanted, and be willing to participate in follow-up visits, postoperative polysomnography, and questionnaire completion.
• Children's parents must complete a questionnaire confirming that their child is capable of communicating feelings of pain or discomfort. They must also confirm they are able to assess their child for adverse effects related to device implantation.
• Children and their parents must be proficient in English for this pilot study in order to ensure full disclosure during the consent process, as well as have the ability to communicate with all staff, at all times, regarding any questions about participation or concerns about this device.
• In order to participate, subjects will require written consent from both parents. All study subjects must provide written assent as well.

You may not qualify if:

• Subjects will be excluded if they meet the following criteria: BMI \>95th percentile for age, apnea hypopnea index (AHI) \<10 or \>50 on in-lab polysomnography (PSG), central or mixed apneas accounting for \>25% of the total AHI, any anatomic finding on physical exam or drug induced sleep endoscopy (DISE) that would compromise the performance of stimulation (e.g. concentric soft palate collapse), other medical conditions resulting in medical instability (e.g. congestive heart failure, recent open heart surgery, immunosuppression, or chronic lung disease or aspiration), presence of another medical condition requiring future magnetic resonance imaging (MRI), history of cholesteatoma, or patients with another implantable device which could interact unintentionally with the Inspire system.
• Subjects in whom general anesthesia for a surgical procedure is contraindicated due to other medical illnesses or conditions will be excluded.
• Subjects with a life expectancy \< 12 months will be excluded.
• Subjects who are unable to communicate pain or discomfort to their caretaker/parent, based on parental or investigator assessment, will be excluded.
• Subjects with a history of bleeding or clotting disorders and those on blood thinning or NSAID medications will be excluded from participation.
• Subjects taking muscle relaxant medication will be excluded from participation.
• Female subjects who are pregnant or plan to become pregnant during the study period will be excluded. All female subjects will undergo urine beta-HCG testing on the day of procedures requiring general anesthesia (DISE, implantation, and any other unanticipated surgical procedures related to implantation). Subjects who are positive will not undergo surgical implantation or procedures under general anesthesia.
• Subjects deemed unfit for participation by investigators or any other reason will be excluded.

Sponsors & Collaborators

• Christopher Hartnick, M.D.: lead
• Emory University: collaborator
• Children's Hospital Medical Center, Cincinnati: collaborator
• Inspire Medical Systems, Inc.: collaborator
• University of Pittsburgh: collaborator
• Children's Hospital of The King's Daughters: collaborator

Study Sites (5)

Children's Healthcare of Atlanta - Egleston Hospital

Atlanta, Georgia, 30329, United States

Location

Massachusetts Eye and Ear

Boston, Massachusetts, 02115, United States

Location

Cincinnati Children's Hospital

Cincinnati, Ohio, 45229, United States

Location

Children's Hospital of Pittsburgh

Pittsburgh, Pennsylvania, 15224, United States

Location

Children's Hospital of The King's Daughters

Norfolk, Virginia, 23507, United States

Location

Related Publications (6)

• Yu PK, Stenerson M, Ishman SL, Shott SR, Raol N, Soose RJ, Tobey A, Baldassari C, Dedhia RC, Pulsifer MB, Grieco JA, Abbeduto LJ, Kinane TB, Keamy DG Jr, Skotko BG, Hartnick CJ. Evaluation of Upper Airway Stimulation for Adolescents With Down Syndrome and Obstructive Sleep Apnea. JAMA Otolaryngol Head Neck Surg. 2022 Jun 1;148(6):522-528. doi: 10.1001/jamaoto.2022.0455.

  PMID: 35446411 DERIVED

• Stenerson ME, Yu PK, Kinane TB, Skotko BG, Hartnick CJ. Long-term stability of hypoglossal nerve stimulation for the treatment of obstructive sleep apnea in children with Down syndrome. Int J Pediatr Otorhinolaryngol. 2021 Oct;149:110868. doi: 10.1016/j.ijporl.2021.110868. Epub 2021 Aug 5.

  PMID: 34371294 DERIVED

• Jayawardena ADL, Randolph GW, Hartnick CJ. Pediatric Modifications to Hypoglossal Nerve Stimulation for Obstructive Sleep Apnea: How I Do It. Laryngoscope. 2021 Feb;131(2):423-424. doi: 10.1002/lary.28661. Epub 2020 Apr 9. No abstract available.

  PMID: 32271464 DERIVED

• Caloway CL, Diercks GR, Keamy D, de Guzman V, Soose R, Raol N, Shott SR, Ishman SL, Hartnick CJ. Update on hypoglossal nerve stimulation in children with down syndrome and obstructive sleep apnea. Laryngoscope. 2020 Apr;130(4):E263-E267. doi: 10.1002/lary.28138. Epub 2019 Jun 20.

  PMID: 31219619 DERIVED

• Diercks GR, Wentland C, Keamy D, Kinane TB, Skotko B, de Guzman V, Grealish E, Dobrowski J, Soose R, Hartnick CJ. Hypoglossal Nerve Stimulation in Adolescents With Down Syndrome and Obstructive Sleep Apnea. JAMA Otolaryngol Head Neck Surg. 2018 Jan 1;144(1):37-42. doi: 10.1001/jamaoto.2017.1871.

  PMID: 29098288 DERIVED

• Diercks GR, Keamy D, Kinane TB, Skotko B, Schwartz A, Grealish E, Dobrowski J, Soose R, Hartnick CJ. Hypoglossal Nerve Stimulator Implantation in an Adolescent With Down Syndrome and Sleep Apnea. Pediatrics. 2016 May;137(5):e20153663. doi: 10.1542/peds.2015-3663.

  PMID: 27244805 DERIVED

MeSH Terms

Conditions

Sleep Apnea, Obstructive; Down Syndrome

Condition Hierarchy (Ancestors)

Sleep Apnea Syndromes; Apnea; Respiration Disorders; Respiratory Tract Diseases; Sleep Disorders, Intrinsic; Dyssomnias; Sleep Wake Disorders; Nervous System Diseases; Intellectual Disability; Neurobehavioral Manifestations; Neurologic Manifestations; Abnormalities, Multiple; Congenital Abnormalities; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Chromosome Disorders; Genetic Diseases, Inborn

Limitations and Caveats

Analysis was limited by the lack of a control group, although spontaneous resolution of OSA would be less likely to occur in an adolescent population with severe disease. Not all of the 12-month PSGs were full-night studies at one voltage. Some of the patients were young adults over 18 years old, and there are different scoring criteria for adolescents compared to young adults. All patients were assessed using the same criteria in order to maintain consistency across the study.

Results Point of Contact

• Title: Matthew E Stenerson, MS, Clinical Research Project Manager
• Organization: Mass General Brigham

matthew_stenerson@meei.harvard.edu

7155721100

Study Officials

• Christopher J Hartnick, MD

  Massachusetts Eye and Ear

  PRINCIPAL INVESTIGATOR

Publication Agreements

• PI is Sponsor Employee: No
• Restriction Type: GT60
• Restrictive Agreement: Yes

Study Design

• Study Type: interventional
• Phase: not applicable
• Allocation: NA
• Masking: NONE
• Purpose: TREATMENT
• Intervention Model: SINGLE GROUP
• Sponsor Type: OTHER
• Responsible Party: SPONSOR INVESTIGATOR
• PI Title: Principal Investigator

Study Record Dates

• First Submitted: January 13, 2015
• First Posted: January 22, 2015
• Study Start: February 1, 2015
• Primary Completion: July 23, 2021
• Study Completion: September 2, 2021
• Last Updated: April 12, 2022
• Results First Posted: February 11, 2022

Record last verified: 2022-04

Data Sharing

• IPD Sharing: Will not share

Locations

US (5)