Role of Omega-DEK in Childhood Apraxia of Speech
试验速览
- 阶段
- 2 期
- 状态
- 尚未招募
- 发起方
- 入组人数
- 34
- 试验地点
- 3
- 主要终点
- Percent of Expanded Cohort Recruited
研究概览
简要总结
This is a 20-week study for children between 3 and 6 years old with confirmed childhood apraxia of speech (CAS). The study includes a 12-week open-label pilot feasibility study of an investigational drug (Omega-DEK) plus L-carnitine (Carnitor®), which is followed by an 8-week randomized, placebo-controlled discontinuation period among the same study participants.
详细描述
Verbal apraxia (VA) is a severe neurological motor planning speech disorder of unknown etiology. It is a devastating disorder that is insufficiently recognized by general pediatricians and often goes unaddressed and improperly treated during critical years of speech and language development. The high prevalence of this disorder excludes it as an "orphan" disease, although like autism, it may have met the definition over a decade ago. However, inadequate awareness of this condition among practitioners renders it a neglected disorder. Confusion around this condition is reflected by the vast number of terms used to define it, including Childhood Apraxia of Speech, Developmental Apraxia, Developmental Dyspraxia, Speech Apraxia, and Speech Dyspraxia to name a few.
Approximately half of children with autism spectrum disorders (ASD) have some degree of apraxia, although not all apraxic children are autistic. There is currently no recognized cure for VA, and it is thought to be a life-long condition. Standard treatment is extremely costly and involves intensive and frequent 1:1 speech therapy with a speech pathologist knowledgeable in VA. Typical response to therapy tends to be slow, and some children do not learn how to talk, thereby requiring alternate means of communication. Children with this disorder find it very difficult to correctly pronounce sounds, syllables, and words, despite intense effort. Intelligibility is poor, and some children remain completely speechless and require the use of augmentative communication devices, sign language and/or a picture exchange communication system.
Many children with VA present with a unique but homogeneous group of neurological symptoms that affect coordination, muscle tone and sensory issues in addition to expressive speech delay, suggesting a common underlying mechanism of disease. Vitamin E (vit E) deficiency causes a constellation of symptoms that overlap those of speech apraxia, limb dyspraxia, hypotonia and sensory integration dysfunction (including abnormalities in proprioception, vestibular sensation, and pain interpretation) that often occur in VA and ASD. Low bioavailability of vit E will create an environment within the cell membrane where vital polyunsaturated fatty acids (PUFAs) are vulnerable to lipid peroxidation and early destruction. This can lead to a functional PUFA deficiency and neurological sequelae that may be reversible through supplementation with PUFA/vit E. In addition, PUFA supplementation increases utilization of vit E in the body. These two supplements may have synergistic effects at higher doses.
An unexpected number of apraxic children have a carnitine deficiency, high antigliadin antibodies and carry a gluten-sensitivity major histocompatibility complex (HLA), suggesting abnormal fatty acid metabolism, increased oxidative stress and a potential link to gastrointestinal inflammation and gluten-sensitivity that creates a distinctive nutritional requirement in these children that may benefit from an investigational drug specifically formulated to targets unique deficiencies that contribute to VA. The researchers speculate that patients with gluten sensitivity or those carrying a celiac HLA with autism/VA may not have classic celiac disease, but perhaps a broader diagnosis of gluten-sensitivity associated with malabsorption and neurobehavioral consequences of nutritional deficiencies that needs consideration. The less sensitive celiac biomarker, antigliadin immunoglobulin G (IgG), frequently found in both ASD and VA may represent a biomarker that identifies an intervention-responder group. A recent double-blind randomized, placebo-controlled trial in irritable bowel syndrome concluded that a non-celiac gluten intolerance my exist, a concept in need of exploration in both ASD and VA.
In this study, children between 3 and 6 years old with confirmed childhood apraxia of speech (CAS) will take an investigational drug (Omega-DEK) plus L-carnitine (Carnitor®) for 12 weeks. This open-label period of the study is followed by an 8-week blinded trial where participants will be randomized to continue taking Omega-DEK or to take a placebo. Additionally, 10 participants will be enrolled in a cohort with expanded follow-up to examine the feasibility of conducting a 12-month open-label trial of Omega-DEK plus L-carnitine in participants aged ≥2 years who have a working diagnosis of CAS, intestinal lymphangiectasia, or fat malabsorption syndrome (in isolation or associated with other medical conditions such as cystic fibrosis, celiac disease, etc.).
研究设计
- 研究类型
- Interventional
- 分配方式
- Randomized
- 干预模型
- Parallel
- 主要目的
- Treatment
- 盲法
- Quadruple (Participant, Care Provider, Investigator, Outcomes Assessor)
入排标准
- 年龄范围
- 36 Months 至 6 Years(Child)
- 性别
- All
- 接受健康志愿者
- 否
入选标准
- •for Main Trial:
- •Confirmed diagnosis of childhood apraxia of speech/verbal apraxia by a qualified professional (SLP) based on established guidelines
- •Male and female, age 36 months - 6 years (inclusive)
- •Ability to comprehend and use Standard American English
排除标准
- •for Main Trial:
- •Children unable to tolerate oral supplementation
- •Known allergy to fish oil, palm kernel oil or other ingredients in investigational drug
- •Medical or genetic condition that in the opinion of the PI/Co-Is may affect participation and compromise results (including significant receptive language delay, moderate-severe cognitive delay, complex medical history, hearing loss, cerebral palsy, history of traumatic brain injury or severe anoxic event, Down's syndrome)
- •Known seizure disorder or history of febrile seizures
- •History of cardiac dysrhythmia or abnormal ECG at baseline
- •A prothrombin time test with an international normalised ratio (PT/INR) >1.2
- •Use of blood thinners, including chronic aspirin, chronic NSAIDS, warfarin etc.
- •A history of PUFA or vit E supplementation use within 3 months prior to enrollment in the study
- •On an elimination diet for < 3 months (gluten, casein, yeast free etc.) prior to enrollment, or planning to initiate a special diet during the study
- •Recent reintroduction of food items from elimination diet < 3 months
- •On any additional nutritional interventions/supplements < 3 months (i.e. high dose vitamins/minerals that exceed what would be found in a children's multivitamin supplement etc., probiotics)
- •Any new chronic medication < 3 months prior to enrollment (stable doses > 3 months allowed; medications for acute illness allowed including antipyretics, antibiotics, asthma medication)
- •Anticipated initiation of new chronic medication during study timeline including new attention-deficit/hyperactivity disorder (ADHD) medications, other behavior medications
- •Plans to try additional complementary interventions or diets during the study period
- •Planned surgery during or within 4 weeks after conclusion of trial
- •Inclusion Criteria for Expanded Cohort:
- •Working Diagnosis of CAS, or intestinal lymphangiectasia, or fat malabsorption syndrome (in isolation or associated with other medical conditions such as cystic fibrosis, celiac disease, etc.)
- •Male and female, aged ≥2 years
- •Ability to comprehend and use Standard American English
- •Exclusion Criteria for Expanded Cohort:
- •Subjects unable to tolerate oral supplementation
- •Known allergy to fish oil, palm kernel oil or other ingredients in investigational drug
- •History of cardiac dysrhythmia or abnormal ECG at baseline
- •PT-INR >1.
- •A clinical lab performed within a month of enrollment will be accepted to meet this exclusion criteria
- •Use of blood thinners, including chronic aspirin, chronic NSAIDS, warfarin etc. (Symptomatic use of NSAIDS for acute fever or pain permitted)
研究组 & 干预措施
Omega-DEK and L-carnitine for 12 Weeks Followed by Placebo for 8 Weeks
Children aged 3 to 6 years old with confirmed childhood apraxia of speech (CAS) receiving Omega-DEK and L-carnitine for the 12-week open-label part of the trial, who are then randomized to receive a placebo to match Omega-DEK for 8 weeks. L-carnitine is provided to participants only during the 12-week open-label portion of the trial.
干预措施: L-carnitine (Drug)
Expanded Cohort
Children aged 2 or older with a working diagnosis of CAS, intestinal lymphangiectasia, or a fat malabsorption syndrome (in isolation or associated with other medical conditions) receiving Omega-DEK and L-carnitine for 12 months.
干预措施: L-carnitine (Drug)
Omega-DEK and L-carnitine for 12 Weeks Followed by Omega-DEK for 8 Weeks
Children aged 3 to 6 years old with confirmed childhood apraxia of speech (CAS) receiving Omega-DEK and L-carnitine for the 12-week open-label part of the trial, who are then randomized to continue to receive Omega-DEK for 8 additional weeks. L-carnitine is provided to participants only during the 12-week open-label portion of the trial.
干预措施: L-carnitine (Drug)
Omega-DEK and L-carnitine for 12 Weeks Followed by Omega-DEK for 8 Weeks
Children aged 3 to 6 years old with confirmed childhood apraxia of speech (CAS) receiving Omega-DEK and L-carnitine for the 12-week open-label part of the trial, who are then randomized to continue to receive Omega-DEK for 8 additional weeks. L-carnitine is provided to participants only during the 12-week open-label portion of the trial.
干预措施: Omega-DEK (Drug)
Omega-DEK and L-carnitine for 12 Weeks Followed by Placebo for 8 Weeks
Children aged 3 to 6 years old with confirmed childhood apraxia of speech (CAS) receiving Omega-DEK and L-carnitine for the 12-week open-label part of the trial, who are then randomized to receive a placebo to match Omega-DEK for 8 weeks. L-carnitine is provided to participants only during the 12-week open-label portion of the trial.
干预措施: Omega-DEK (Drug)
Omega-DEK and L-carnitine for 12 Weeks Followed by Placebo for 8 Weeks
Children aged 3 to 6 years old with confirmed childhood apraxia of speech (CAS) receiving Omega-DEK and L-carnitine for the 12-week open-label part of the trial, who are then randomized to receive a placebo to match Omega-DEK for 8 weeks. L-carnitine is provided to participants only during the 12-week open-label portion of the trial.
干预措施: Placebo (Drug)
结局指标
主要结局
Percent of Expanded Cohort Recruited
时间窗: Up to Month 12
Feasibility of conducting a 12-month, open-label trial is measured by successful recruitment, which is defined as \<20% refusal of eligible children.
Percent of Expanded Cohort Retained
时间窗: Up to Month 12
Feasibility of conducting a 12-month, open-label trial is measured by successful retention, which is defined as \>85% of study participants completing the study.
Percent of Expanded Cohort Complying with Treatment Regimen
时间窗: Up to Month 12
Feasibility of conducting a 12-month, open-label trial is measured by successful compliance, which is defined as \>75% of participants with compliance with the treatment regimen.
Percent of Expanded Cohort With Complete Data
时间窗: Up to Month 12
Feasibility of conducting a 12-month, open-label trial is measured by collection of study data, which is defined as \>90% of participants providing outcome data.
Percent of Participants Retained in Study
时间窗: Up to Week 20
Feasibility of the intervention is assessed with the percentage of enrolled participants who complete the 20 week study. Successful retention is defined as \>85% of enrolled participants completing the study.
次要结局
- Percent of Participants With Complete Outcome Data(Up to Week 20)
- Change in Dynamic Evaluation of Motor Speech Skill (DEMSS) Score(Baseline, Week 12, Week 20)
- Mean Length of Utterances (MLU)(Baseline, Week 12, Week 20)
- Clinical Global Impression for Improvement Scale (CGI-I) Score(Weeks 1, 4, 8, 12 (during the open-label portion of the study), weekly during Weeks 13-22 (during the randomized portion of the study))
研究者
Claudia R. Morris
Professor
Emory University
