Lithuanian University of Health Sciences Faculty of Medicine Department of Pediatrics Rehabilitation

(1)

1 Lithuanian University of Health Sciences

Faculty of Medicine

Department of Pediatrics Rehabilitation

Title of Master’s Thesis:

The use of telehealth in early intervention for children with neurodevelopmental disabilities

Author: Basel Sijari

Supervisor:

PhD, assoc. Prof. Audrone Prasauskiene

(2)

2

SUMMARY

Author name: Basel Sijari

Research title: The use of telehealth in early intervention for children with neurodevelopmental disabilities.

Aim: To perform a systemic review of papers describing the use of telehealth in early intervention. Objectives:

1. Identify the extent and range of the existing literature on the use of telehealth for diagnosis and intervention for neurodevelopmental disorders.

2. Systematically review evidence on the impact of telemedicine on early diagnosis, outcomes, and care utilisation.

3. Identify gaps in the research and to delineate the future research needs.

Methodology: The systematic review was conducted in line with the Preferred Reporting Items for Systematic Reviews and Meta-analyses statement (PRISMA). The data sources include Medline (PubMed), along with reference lists of relevant reviews and involved articles. Study eligibility criteria were studies that assess the use of telehealth for implementation of an intervention, assessment and/or consultations in early intervention.

Results: Fourteen studies met the inclusion criteria and were included in the review. Analysis of the studies revealed that low-cost video conferencing is a reliable method for speech, language, and hearing impairments screening, and autism identification in children. Video conferencing appeared to be the effective mode of implementing behaviour, communication, speech and language targeted interventions for children with developmental language delays, learning disability, hearing loss, and autism spectrum disorders.

(4)

4

Santrauka

Autorius:: Basel Sijari

Darbo tema: Telemedicinos naudojimas raidos sutrikimus turinčių vaikų ankstyvojoje reabilitacijoje. Tikslas: Atlikti sisteminę straipsnių, kuriuose aprašomas telemedicinos naudojimas ankstyvojoje reabilitacijoje, apžvalgą.

Uždaviniai:

1. Nustatyti mokslinių tyrimų, analizavusių telemedicinos naudojimą raidos sutrikimus turinčių vaikų ankstyvojoje reabilitacijoje (AR), apimtis ir pobūdį.

2. Sistemiškai apžvelgti mokslinius įrodymus, susijusius su telemedicinos naudojimu raidos sutrikimų diagnostikai ir gydymui.

3. Nustatyti spragas moksliniuose įrodymuose ir įvertinti kokie papildomi tyrimai reikalingi.

Metodika: Sisteminė apžvalga atlikta vadovaujantis sisteminių apžvalgų ir meta-analizių rengimo

rekomendacijomis PRISMA. Atrinkti moksliniai straipsniai iš Medline (PubMed) duomenų bazės ir į šią apžvalgą įtrauktų straipsnių literatūros sąrašų. Įtraukimo į apžvalgą kriterijai – tyrimai, analizuojantys telemedicinos naudojimą konsultavimui, diagnostikai ir gydymui AR.

Rezultatai: Įtraukimo kriterijus atitiko 14 tyrimų, kurie buvo įtraukti į apžvalgą. Studijų analizė parodė, kad mažai kainuojančios video konferencijos yra patikimas metodas kalbos, kalbėjimo ir klausos sutrikimų rizikaą turinčių vaikų atrankai ir autizmo diagnostikai. Video konferencijos pasirodė esąs efektyvus metodas, taikant intervencijas, nukreiptas į elgesio, kalbos ir kalbėjimo sutrikimų šalinimą vaikams, turintiems kalbos raidos sutrikimą, protinę negalią, kurtumą ir autizmo spektro sutrikimus.

(5)

5

ACKNOWLEDGMENTS

I dedicate my thesis to my beloved mother, wife for their constant encouragement and unwavering faith in me, and I want to thank my father, Eng. Ammar Sijari for his enormous support, always inspiring me to follow my dreams. The journey would not have been possible without the help of my family, friends, professors and university staff.

(6)

6

CONFLICTS OF INTEREST

The author reports no conflicts of interest.

PERMISSION ISSUED BY THE ETHICS COMMITTEE

(7)

7

LIST OF ABBREVIATIONS

ADHD - Attention-Deficit Hyperactivity Disorder ASD - Autism Spectrum Disorders

ACO - Accountable Care Organization AVT - Auditory-Verbal Therapy ADI - Autism Diagnostic Interview

ADOS - Autism Diagnostic Observation Schedule CP - Cerebral Palsy

CME - Continuing Medical Education CNS - Central Nervous System

DLDs - Developmental Language Disorders

DPOAE - Distortion Product Otoacoustic Emissions EI - Early Intervention

FA - Functional Analysis

FCT - Functional Communication Training GDD - Global Developmental Delay IQ - Intelligence Quotient

LD - Learning Disabilities

NDD - Neuro Development Disorder NTP - National Toxicology Program PCBs - Poly-Chlorinated Biphenyls

(8)

8 RCT - Randomized Clinical Trial

U.S. - United States

(9)

9

CHAPTER 1: INTRODUCTION

Over the past century research has proven that early intervention may improve long-term outcomes for children with neurodevelopmental disorders. Implementation of evidence-based practice often needs a multidisciplinary team (e.g., speech-language pathologist, physiotherapist, occupational therapist, special educator, and psychologist). Therefore, as there is increasing the number of individuals diagnosed with ASD, so need for specialists trained to provide evidence-based practices increases.

Due to the less number of specialists, children with neurodevelopmental disorders are often evaluated and treated by less qualified personnel, particularly in rural areas where access to specialists is most limited. It is not always possible to routinely travel the distances it would require to have a possibility to connect trained specialists as well as parents of children with neurodevelopmental disorders who need services. Telepractice (also known as ‘telehealth’ and ‘telemedicine’) which involves the application of

communication technologies (e.g., computer-based video conferencing) enable specialists to consult and deliver services over far distances. Many works of literature support the use of telepractice to provide health care services to people in rural areas. For example, telepractice can be used to connect doctors in hospitals to home patients who need consultations. It is possible that telepractice may also be helpful in connecting specialists of early intervention to parents of children who have or are at risk of

neurodevelopmental disorders to provide screening tests, interventions, and supports. Although the use of telehealth in the assessment and treatment of children with neurodevelopmental disabilities was evaluated in a number of studies, only a few reviews of this research were published.

To better understand how telehealth used for the diagnosis and intervention of children with

(10)

10

CHAPTER 2: AIMS AND OBJECTIVES

Aim: To perform a systemic review of papers describing the use of telehealth in early intervention.

Objectives:

1. Identify the extent and range of the existing literature on the use of telehealth for diagnosis and intervention for neurodevelopmental disorders.

2. Systematically review evidence on the impact of telemedicine on early diagnosis, outcomes, and care utilization.

(11)

11

CHAPTER 3: LITERATURE REVIEW

3.1 Definition, prevalence, and etiology of neurodevelopmental disorders

Neurodevelopmental disorders (NDD) are the group of chronic diseases in which the development of the central nervous system (CNS) is disturbed. NDDs start in childhood and usually affect several or all areas of functioning. [1-2]

Some examples of NDD in children involve attention-deficit/hyperactivity disorder (ADHD), autism, learning disabilities (LD), cerebral palsy (CP), and weakness in vision and hearing. At birth, the brain is incompletely developed so symptoms of brain damage might be not visible. The symptoms of NDD would appear later as the child grows up and start using the function of affected area. Problems related to brain maturation and child development could be deficits in mental ability involving judgment, planning, problem solving and academic learning, communication disorders including language and speech, memory, behavior and other diseases, and cerebral palsy. Treatment is a combination of medications, multidisciplinary professional therapy, home programs, and school programs.

The prevalence of NDD varies between studies and regions, but it assumed that rates of NDD have

increased in the past ten years, especially in autism and ADHD. In the United States (U.S), the prevalence of NDD in children aged 3-17 years was approximately 15% in 2006–2008 [] . The highest prevalence 1 was found for ADHD and LD. Boys had the greatest prevalence compared with girls. Hispanic children had the lowest prevalence of NDD compared with non-Hispanic white and black children]2[. (Table1)

Table 1. The prevalence of neurodevelopmental disabilities in the United States 2006-2008 (Blumberg, 2011) [3]

Disability Prevalence

Any developmental disability 13.9 Attention-deficit/hyperactivity disorder 6.7 Autism 0.5 Blindness 0.1 Cerebral Palsy 0.4 Intellectual Disability 0.7 Learning Disability 7.0 Hearing Loss 0.5 Seizures 0.7

(12)

12 Although NDD are very prevalent, the etiology of such disorders is poorly understood. The most

described causes of NDD are genetic and metabolic conditions, pre- or postnatal insults due to hypoxia, alcohol or toxin exposure, nutritional deficiencies, infection, trauma, or metabolic changes, and

deprivation.

Poly-Chlorinated Biphenyls (PCBs), lead, and methylmercury are common environmental contaminants which have adverse effects on a child’s brain and nervous system. The National Toxicology Program (NTP) has approved that PCBs and lead have relations between these contaminants and NDD effects, including decreased intelligence quotient (IQ) and behavioral deficits like inattention and rushing behaviour. [4-5]Studies reported a relationship between adverse outcomes and PCB exposure during infancy and childhood. It approved that “PCBs have a role in neurobehavioral alterations which observed in newborns and young children of women with PCB”. [6] Methylmercury has supported that have developmental effects and neurotoxic on humans. Cases of such impacts were seen in Japan and Iraq during the war. Children who were exposed to methylmercury had severe disabilities such as mental retardation, deafness, CP, and blindness. [7]

Genetics play an important role in many NDD. These disorders result from a combination of genetic, environmental, biological, and psychosocial risk factors. A broad range of environmental risk factors may affect neurodevelopment, including (but not limited to) maternal use of alcohol, tobacco, or illicit drugs during pregnancy, lower socioeconomic status, and prenatal or childhood exposure to specific

environmental contaminants. Also, a wide variety of other environmental chemicals have been identified as potential concerns for childhood neurological development but have not studied for these effects as lead, mercury, and PCBs. [8-13]

3.2 Types of neurodevelopmental disorders

The following NDD are the most commonly identified for children: ADHD, autism spectrum disorders (ASD), global developmental delays (GDD), LD, CP, and hearing and vision impairments.

ADHD is a behavior disorder characterized by symptoms of inattention and hyperactivity, which occurs more severely and frequently than is typical for other individuals at the same stage of development. ADHD have effects on performance. Symptoms of ADHD tend to change over time. Children with ADHD frequently have other comorbid conditions: about half of them have LD, and about one of four children with ADHD has conduct disorders. [14-15]

(13)

13 skills such as organization, time planning, abstract reasoning, long or short-term memory and attention. Children with LD usually have average or above average intelligence, but there are differences in the way their brains process information. [16]

ASD is a range of conditions re-characterized by social-interaction difficulties, communication challenges and a tendency to engage in repetitive behaviours.

The term “spectrum disorders” refers to the fact that although people with ASD share some common symptoms, ASD affects different people in different ways, with some experiencing very mild symptoms and others experiencing severe symptoms. Children with ASD suffer from lack interest in people, have problems with the expression of feelings, and avoid people contact. [17]

GDD: is an umbrella term for conditions where there is a significant delay in two or more developmental categories: gross/fine motor, speech/language, cognition, activities of daily living and, social/personal. The term GDD is accepted to use for younger children (i.e., typically less than five years of age), while the term mental retardation is usually applied to older children when IQ testing is more valid and reliable. CP: is describing a group of permanent disorders that affect the development of the movement and

posture, and cause limitation of activity. Symptoms also can include disturbances of sensation, perception, cognition, and communication. CP is a consequence of immature brain damage before birth or during the first five years after birth. [18]

Hearing loss: is impairment in hearing, whether permanent or fluctuating, which affects a child’s

educational performance but not defined as deafness is a total or partial inability to hear due to disturbance of sound transmission in the outer, middle or inner ear. The conductive hearing happens when there is a problem with the ear canal, eardrum, or middle ear and its little bones (the malleus, incus, and stapes). The sensorineural hearing loss is due to problems of the inner ear, and also is known as a nerve-related hearing loss. Mixed describes the occurrence of conductive hearing loss that also has a sensorineural component. Speech and language delays refer to problems in communication and related areas such as oral motor function. [19]

(14)

14 3.3 Early intervention

Early Intervention (EI) is a support and educational system for very young children who have

developmental delays or disabilities due to biological, medical or environmental risk factors (e.g., poverty, child abuse, and neglect). The aims of EI are remedial or preventative and comprise the following: 1) promote child health and well-being, 2) enhance emerging competencies, 3) minimize developmental delays, 4) remediate existing or emerging disabilities, 5) prevent functional deterioration and, 6) promote adaptive parenting and overall family functioning. [20]

These goals are to provide individualized developmental, educational and therapeutic services for children in conjunction with mutually planned support for their families. EI can be in different places such as a hospital, rehabilitation center, or home. EI can start at any moment between birth and five years old. It is advised to interfere as soon as possible. It recognized that only through early identification and

appropriate programming infants can reach their full potential. EI help to decrease the impact of developmental problems on the family. The best time to do EI in infant lies between 40-44 weeks

postmenstrual age and 15 months postnatally. [21] The main advantage of EI is that the immature brain is plastic and able to change due to sensory or motor experience. There are a few disadvantages of EI as well. First, when an infant is born, it is not known what problem will develop later in his life. Second, a part of children at-risk may not have a developmental disorder later in their life; thereby EI is unnecessary for these children. A disadvantage of intervention which first starts when the disease has become

undeniable is that it begins relatively late from the plasticity of the brain. [21] 3.4 Telemedicine

Telemedicine: is defined as “using medical information exchanged from one site to another with

electronic communications to improve a patient’s clinical health status.”. Telemedicine and telehealth, as commonly used today, can be considered synonymous. [22]

The use of telemedicine had started since the civil war when the telegraphs were used for transmission of lists and to order supplies. The Advanced Research Projects Agency Network in 1969 was constructed by US Department of Defense which later evolved into the Internet. The invention of the mobile phones industry has improved access to both voice and video and provided possibilities for clinicians to collaborate more efficiently.

(15)

15 continuing medical education (CME), obtain free CME, and establish public groups to learn from other physicians. [23-24] Tele-consultation helps for disease management. [29-31] Benefits of such

consultations include: (1) improvement in the quality of health due to faster assessment provided by telephone consultations, (2) decrease in the cost of the healthcare services, (3) reduces school absences for the children, (4) less money spent by parents on travel, (5) less time away from employment for parents, and (6) decrease crowding in emergency departments. [29-31] Government forces, such as legislation mandating telemedicine reimbursement, are driving the further development of telemedicine. [30-36] Telemedicine has many usages in pediatrics. As technology strengthens the reduction in cost, telemedicine will enhance research, education, accessing to care, response to an emergency, and the delivery of general and specialty pediatrics in different settings. The barriers are payment, licensing across state borders, and liability. More research is needed to decide telemedicine’s best uses, quality improvement and patient safety implications, and cost-effectiveness in alternative payment systems such as accountable care organization.

3.5 Previous reviews of the use of telemedicine in early intervention

Although the use of telehealth in the assessment and treatment of children with NDD has been evaluated in many studies, only a few reviews of this research have been published. [37-38]

One systematic review explored results from studies regarding the use of telehealth in delivering services to individuals with ASD. The authors of this review concluded that telepractice is a promising service delivery approach in the treatment of individuals with ASD. However, the authors had noticed that research in this field was limited and indicated that more research is needed to prove the validity of the telehealth screening as well as the effectiveness of tele guided interventions, parent training, and support. [37]

The clinical review of Cason (2011) provided an update of studies related to telepractice in EI services for children throw two years. [38] The author has found that telepractice could be useful as an adjunct service delivery model for EI for children with different types of disabilities. However, the author did not perform a systematical search of studies and didn’t ascertain the methodological quality of included studies,

making it difficult to interpret research in telehealth application for EI. [38]

3.6 Systematic reviews

(16)

16 reviews in several points. Narrative reviews are mostly descriptive; do not involve a systematic search of the literature. Thus, narrative reviews while informative can often include an element of selection bias. The systematic reviews typically involve a detailed and comprehensive plan and searching strategy to identify, appraise, and synthesize all relevant studies on a particular topic.

An international group which included experienced authors and methodologists developed PRISMA (Preferred Reporting Items for Systematic reviews and Meta-Analyses): the PRISMA contains 27-item checklist (Table2) and a four-phase flow diagram.

Table 2. PRISMA Checklist Section/topic # Checklist item

TITLE

Title 1 _{Identify the report as a systematic review, meta-analysis, or both.} ABSTRACT

Structured summary

2 _{Provide a structured summary including, as applicable: background;} objectives; data sources; study eligibility criteria, participants, and interventions; study appraisal and synthesis methods; results; limitations; conclusions and implications of key findings; systematic review

registration number. INTRODUCTION

Rationale 3 _{Describe the rationale for the review in the context of what is already} known.

Objectives 4 _{Provide an explicit statement of questions being addressed with reference} to participants, interventions, comparisons, outcomes, and study design (PICOS).

METHODS Protocol and registration

5 _{Indicate if a review protocol exists, if and where it can be accessed (e.g.,} Web address), and, if available, provide registration information

including registration number. Eligibility

criteria

6 _{Specify study characteristics (e.g., PICOS, length of follow-up) and} report characteristics (e.g., years considered, language, publication status) used as criteria for eligibility, giving rationale.

Information sources

7 _{Describe all information sources (e.g., databases with dates of coverage,} contact with study authors to identify additional studies) in the search and date last searched.

Search 8 _{Present full electronic search strategy for at least one database, including} any limits used, such that it could be repeated.

Study selection

9 _{State the process for selecting studies (i.e., screening, eligibility, included} in systematic review, and, if applicable, included in the meta-analysis). Data

collection process

(17)

17 Data items 11 List and define all variables for which data were sought (e.g., PICOS,

funding sources) and any assumptions and simplifications made. Risk of bias

in individual studies

12 Describe methods used for assessing risk of bias of individual studies (including specification of whether this was done at the study or outcome level), and how this information is to be used in any data synthesis. Summary

measures

13 State the principal summary measures (e.g., risk ratio, difference in means).

Synthesis of results

14 Describe the methods of handling data and combining results of studies, if done, including measures of consistency (e.g., I2) for each meta-analysis.

Risk of bias across studies

15 Specify any assessment of risk of bias that may affect the cumulative evidence (e.g., publication bias, selective reporting within studies). Additional

analyses

16 Describe methods of additional analyses (e.g., sensitivity or subgroup analyses, meta-regression), if done, indicating which were pre-specified. RESULTS

Study selection

17 Give numbers of studies screened, assessed for eligibility, and included in the review, with reasons for exclusions at each stage, ideally with a flow diagram.

Study

characteristics

18 For each study, present characteristics for which data were extracted (e.g., study size, PICOS, follow-up period) and provide the citations. Risk of bias

within studies

19 Present data on risk of bias of each study and, if available, any outcome level assessment (see item 12).

Results of individual studies

20 For all outcomes considered (benefits or harms), present, for each study: (a) simple summary data for each intervention group (b) effect estimates and confidence intervals, ideally with a forest plot.

Synthesis of results

21 Present results of each meta-analysis done, including confidence intervals and measures of consistency.

Risk of bias across studies

22 Present results of any assessment of risk of bias across studies (see Item 15).

Additional analysis

23 Give results of additional analyses, if done (e.g., sensitivity or subgroup analyses, meta-regression [see Item 16]).

DISCUSSION Summary of evidence

24 Summarize the main findings including the strength of evidence for each main outcome; consider their relevance to key groups (e.g., healthcare providers, users, and policy makers).

Limitations _{25 Discuss limitations at study and outcome level (e.g., risk of bias), and at} review-level (e.g., incomplete retrieval of identified research, reporting bias).

Conclusions _{26 Provide a general interpretation of the results in the context of other} evidence, and implications for future research.

FUNDING

(18)

18

CHAPTER 4: RESEARCH METHODOLOGY AND METHODS

The Medline (PubMed) was systematically searched for titles and abstracts published from 2012. Search terms were: “telepractice,” “teleconference,” “skype” ©, “iChat” ©, “videoconferencing,” “distance education,” “telehealth,” “teletherapy,” “telerehabilitation”, “telemedicine” AND “” developmental delay”, “developmental disabilities”, “developmental education”, “early intervention”, “cerebral palsy”, “developmental coordination disorder”, “mental retardation”, “autism”, “rare diseases”, “autism”, ‘speech disorders”, “language diseases”, “language delay” in diverse combinations. The reference lists of relevant reviews and all included papers were checked for eligible articles missed from the original electronic searches. The search was updated on the Medline database on December 10, 20172.

Inclusions and exclusion criteria are provided in Table 3.

Table 3. Inclusion and exclusion criteria.

Inclusion Exclusion

Full-text articles Reviews

Within five years Older than five years

Human case studies Animal studies

Articles written in English Articles in other languages

Children studies Adult studies

Neurodevelopment diseases Other disorders

Early intervention studies Not early intervention studies

Original research Review articles, conference abstracts, editorials and commentaries

For all selected studies, the full text was retrieved and examined. Data on the telehealth were extracted using a standardized data extraction form. Data obtained included information about: (1) the study (author(s), year of publication, study location), (2) participants, aim of study, study design. (3) type of technology and service delivery via telehealth, (4) outcome measures, (5) results.

The information was analyzed using two approaches: (1) table in which a descriptive summary (author, year, country, the aim of the study, study design, participants, intervention, and results) was provided. (2) a narrative synthesis to summaries the data on effects of telehealth interventions.

Results of the data analysis organized along two themes: (1) telehealth interventions aimed to identify neurodevelopmental disabilities. (2) telehealth interventions designed to provide intervention or educational programs.

(19)

(20)

20

CHAPTER 5: RESULTS

The database search identified 420 titles in PubMed, narrowed down to 52. Twelve studies that met the requirements were analyzed, while the remaining 40 excluded (figure 1).

Articles were from USA were (10), and Australia (2). Two studies were randomized-controlled, and twelve studies were non-randomized pre/post cohort studies. Three studies analyzed the use of telehealth in the assessment and/or screening of children with NDD: ASD [41], developmental language disorder [40], and hearing [39]. Other studies investigated the effect of telehealth providing interventions: autism therapies [43,44,45,48], developmental language disorder (DLD) [42,47,49], and hearing loss [46,50]. Technology used: Most of the studies used widely available technology to implement telepractice, such as laptop computers with both internal or peripherally connected microphones and web cameras connected to the Internet via high-speed Internet connections. A video was transmitted at a rate of either 15 or 30 frames per second (fps). Skype and iChat video conferencing software programs were used as well. Skype and iChat both allow video and voice calling over a variety of different mobile internet devices and are currently available for free download.

Records identified through database searching (n = 420) S cre ening Inc luded Eligi bil it y Ide nti fic ati on

Additional records identified through other sources

(n =5)

Records after duplicates removed (n =30)

Records screened (n =395)

Records excluded (n = 343)

Full-text articles assessed for eligibility

(n = 52)

Full-text articles excluded, with reasons (n =38)

• Not pediatrics studies n=13

• Not neuro disabilities n = 9

• Not in the English language n= 6

• Validity and reliability n = 5

• Not early intervention n = 7

Studies included in the qualitative synthesis

(21)

21 Figure 1. Flowchart of the study design

5.1. Telehealth interventions aimed to identify neurodevelopmental disabilities

Three studies [39-41] explored Interventions aimed to provide screening and/or, assessment of speech, language, hearing impairments or autism (Table 4). Reese et al. (2015) examined the utility and validity of an ASD assessment protocol conducted via video conferencing. The protocol included: (1) a 20-minute observation of the child in an unstructured play setting with age-appropriate toys, b) coaching the parents to implement Autism Diagnostic Observation Schedule Autism (ADOS-2) activities, c) structured

interview using only the Autism Diagnostic Interview (ADI-R) algorithm items, and d) medical and family history. Results demonstrated excellent inter-rater agreement on diagnoses between clinicians in the VC setting and the interdisciplinary team [41]. Other two papers described the results of studies aimed to check the feasibility of VC for implementing speech and language [39,40] and/or hearing screening measures [40]. The methodology strong large study of Ciccia et al. (2011) has found that the reliability of VC based speech-language screening tool was high. VC based hearing screening tool appeared to be reliable (moderate reliability) as well. Moreover, both screening tolls were strongly supported by the families and the community [39]. Somewhat different results were found in a smaller study of Guiberson (2016). Although, VS related speech and language assessment measures were associated with

standardized language scores, only reported vocabulary had classification accuracy values that were desirable for screening for DLDs. Greatee screening efficacy was achieved when reported vocabulary was combined with a number of different words children produced during interactions with parents [40].

Table 4. Studies that investigated the use of telehealth for the screening of neurodevelopmental disabilities Author (year), country Participant characteris tics (number, age, diagnosis) Technology Service delivered Outcome assessment Results Ciccia et al. (2011), USA [39] N=411, (0-6 years) VC Speech, language, and hearing screening Comparison of pass/fail rates for all screening components between those

(22)

22 conducted via VC compared to those conducted at the clinics Parent satisfactions survey tympanometry screenings were 84%. Parent satisfaction rate was high (mean 4.5 from 5 point) Guibers on (2016), USA [40] N= 62 (2-2.11)

VC DLD screening The expressive language subtest of SPLS-4 in Spanish Only parent reported vocabulary had classification accuracy values that were desirable for screening for DLDs. Reese et al. (2015), USA [41] N= 17 (2.5-6) VC ASD ADOS ADI VC based protocols showed excellent accuracy in diagnosis (82%) with good specificity (78%) and excellent sensitivity (88%).

5.2. Telehealth interventions aimed to implement interventions

Nine studies [42-50] explored Interventions aimed to provide treatment (Table 5).

(23)

23 lowest for home telehealth, but both telehealth models were significantly less costly than in-home therapy [47].

Four studies evaluated the possibilities to deliver EI to children with speech and language impairments and/or hearing loss with promising results [46,47,48,50]. Parent who participated in couching

interventions improved in their knowledge of pediatric language-promoting activities and were more engaged in language enrichment activities with their child. [45,47] The data of the study of

Constantinescu et al. (2014) revealed that auditory verbal therapy (AVT) delivered via health had the same effect on language development as conventional AVT [46,50].

(24)

(25)

25 and messages providing information on local, low-cost community organizations specializing in early child development

(26)

26 G Constant inescu et al. (2012), Australia [50] N=17 (6 month-6.5 years, hearing loss)

VC AVT The Parent and

specialist reported survey Parents felt as comfortable as face-to-face when discussing problems with the therapist online, and were satisfied or as satisfied as face-to-face with their level and

their child’s level of

interaction/rappor t with the

(27)

27

CHAPTER 6: DISCUSSION OF RESULTS

A number of children with NDD are growing, so demand in EI is growing rapidly. Over the past century new researches in neurosciences has contributed to new approaches to EI for children with NDD. Early diagnosis, goal-directed, intensive and active interventions, individual, complex and family-centered healthcare have become mandatory components of EI. However, this approach requires one-on-one supervisions by highly trained professionals. Consequently, there is a significant gap between the intensive service requirements for children with NDD and the available resources to provide these services. Although telehealth might address this gap, the result of our review showed that the use of telehealth in EI isn’t well investigated. The studies that were conducted focused mostly on autism and speech and language delays and hearing loss. The use of telehealth in service delivery for parent and children with other kind of NDD, such as CP, ADHD, and GDD weren’t addressed in no one study. The studies that include these kinds of disabilities would be in great value for researchers and practitioners. All studies that investigated telehealth found that this service delivery approach is cheap, feasible and effective approach to implement screening as well as parent coaching to deliver EI interventions. The studies analyzed the validity of telehealth-based speech and language impairment, ASD, and hearing impairment screening. On study [refer] had statistical power to provide high level scientific evidence, others were underpowered []. However, the results of all these studies are promising. There is possibility that in future EI professionals will have cheap, feasible, reliable and valid tools for screening and

diagnosis of children with ASD and DLD. The main concern is that no one studies explored tools for identifying motor delays and/or CP. Future research is needed to address this gap.

The research exploring the use of telehealth to deliver interventions is limited as well. Similar to studies investigating telehealth-based screening tools, interventional studies focused only on ASD and DLD. Interventions aimed to help the families and children with other kind of disabilities are not investigated. Nevertheless, there is research evidence that telehealth-based interventions could achieve the same results as conventional practices.

Finally, telehealth as service delivery method is well supported by families, practitioners, and community. The studies revealed that parents feel as comfortable as face-to-face when discussing problems with the therapist online. This is very important issue, which should encourage researches to create new tools and therapies which could be delivered using remote technologies. Also, practitioners should introduce telehealth in their everyday practice.

(28)

28 included in the review. Also, quality appraisal of included studies wasn’t performed in line with PRISMA statement. The quality appraisal should be performed by at least two reviewers, who have special

(29)

29

CHAPTER 7: CONCLUSIONS

1- The use of telemedicine in EI is little investigated.

2- Current research data shows that telemedicine can be effective service delivery approach in EI for children with ASD, speech and language impairment and hearing loss.

(30)

30

CHAPTER 8: PRACTICAL RECOMMENDATIONS

Recommendations for the research: there is a small number of articles that describe the cost of telehealth and about early intervention, so there is a need for future studies to focus on these points.

Recommendations for the clinicians: use skype consultations via telehealth to improve the confidence of parents who have children with NDD.

(31)

31

CHAPTER 9:

REFERENCES

1- Boyle, C.A., S. Boulet, L.A. Schieve, R.A. Cohen, S.J. Blumberg, M. Yeargin-Allsopp, S. Visser, M. Kogan. Trends in the prevalence of developmental disabilities in US Children. 2011. Pediatrics. 127(6):1034-42.

2- Boyle CA, Decoufle P, Yeargin-Allsopp M. Prevalence and health impact of developmental disabilities in US children. 1994. Pediatrics. 93(3):399– 403

3- Coleen Boyle, Sheree Boulet, Laura Schieve, Robin Cohen, Stephen Blumberg, Marshalyn Yeargin-Allsopp, Susanna Visser, Michael Kogan. Trends in the Prevalence of Developmental Disabilities in US Children. June 2011. Pediatrics. Volume 127, pages 1034-1042

4- Stewart, P.W., D.M. Sargent, J. Reihman, B.B. Gump, E. Lonky, T. Darvill, H. Hicks, J. Pagano. Response inhibition during Differential Reinforcement of Low Rates (DRL) schedules may be sensitive to low-level polychlorinated biphenyl, methylmercury, and lead exposure in children. 2006. Environmental Health Perspectives. 114 (12):1923-9.

5- National Toxicology Program. NTP Monograph on Health Effects of Low-Level Lead. Research Triangle Park, NC: National Institute of Environmental Health Sciences, National Toxicology Program. 2012. http://ntp.niehs.nih.gov/go/36443.

6- Agency for Toxic Substances and Disease Registry (ATSDR). Toxicological Profile for Polychlorinated Biphenyls (PCBs). 2000. Atlanta, GA: U.S. Department of Health and Human Services. Public Health Service. http://www.atsdr.cdc.gov/toxprofiles/tp.asp?id=142&tid=26. 7- Amin Zaki, L., S. El hassani, M.A. Majeed, T.W. Clarkson, R.A. Doherty, M. Greenwood.

Intrauterine methylmercury poisoning in Iraq. 1974. Pediatrics 54 (5):587-95.

8- Aarnoudse Moens, C.S.H., N. Weisglas Kuperus, J.B. van Goudoever, J. Oosterlaan.

Meta-analysis of neurobehavioral outcomes in very preterm and/or very low birth weight children. 2009. Pediatrics. 124 (2):717-728.

9- Banerjee, T.D., F. Middleton, and S.V. Faraone. Environmental risk factors for attention-deficit hyperactivity disorder. 2007. Pediatric. 96 (9):1269-1274.

10- Bhutta, A.T., M.A. Cleves, P.H. Casey, M.M. Cradock, K.J.S. Anand. Cognitive and behavioral outcomes of school-aged children who were born preterm. 2002. JAMA the Journal of the American Medical Association. 288 (6):728-737.

(32)

32 12- R. E. Behrman and A. S. Butler. Preterm Birth: Causes, Consequences, and Prevention. 2007.

Institute of Medicine. Washington, DC: The National Academies Press.

13- Linnet, K.M., S. Dalsgaard, C. Obel, K. Wisborg, T.B. Henriksen, A. Rodriguez, A. Kotimaa, I. Moilanen, P.H. Thomsen, J. Olsen, et. al. Maternal lifestyle factors in pregnancy risk of attention deficit hyperactivity. 2003

14- Pastor, P.N., C.A. Reuben. 2008. Diagnosed attention deficit hyperactivity disorder and learning disability. 2004-2006. United States. Vital and Health Statistics 10 (237).

15- Larson, K., S.A. Russ, R.S. Kahn, N. Halfon. Patterns of comorbidity, functioning, and service use for U.S. children with ADHD. 2011. Pediatrics. 127 (3):462-470.

16- National Dissemination Center for Children with Disabilities. Disability Fact Sheet-No. 7: Learning Disabilities. 2010 April 6.

http://www.nichcy.org/InformationResources/Documents/NICHCY%20PUBS/fs7.pdf

17- Jessica Simacek, Adele F. Dimian, Jennifer J. McComas. Communication Intervention for Young Children with Severe Neurodevelopmental Disabilities Via Telehealth. 2017. Department of Educational Psychology. Doi:10.1007/s10803-016-3006-z.

18- Rosenbaum P, Paneth N, Leviton A, et al. A report: the definition and classification of cerebral palsy. April 2006. Developmental Medicine Child Neuro Support. 109: 8–14.

19- American Speech-Language-Hearing Association’s Position Statement on Telepractice http://www.asha.org/ docs/html/PS2005-00116.html

20- Shonkoﬀ JP, Meisels SJ, editors. Handbook of early childhood intervention. 2000. Cambridge. Cambridge University Press

21- De Graaf-Peters VB, Hadders Algra M. Ontogeny of the human central nervous system: what is happening when Early Human Developmental. 2006. 82:257-266.

22- American Telemedicine Association. What is telemedicine. Available at:

www.americantelemed.org/learn/what-is-telemedicine. Accessed March 31, 2014

23- Smith CE, Fontana-Chow K, Boateng BA, et al. Tele-education: linking educators with learners via distance technology. 2009. Pediatric Ann. 38(10):550–556. [PubMed: 19968192]

24- González Espada WJ, Hall-Barrow J, Hall RW, Burke BL, Smith CE. Achieving success connecting academic and practicing clinicians through telemedicine. 2009. Pediatrics. 123(3) Available at: www.pediatrics.org/cgi/content/full/123/3/e476

(33)

33 26- Hall-Barrow J, Hall RW, Burke BL Jr. Telemedicine and neonatal regionalization of care –

ensuring that the right baby gets to the right nursery. 2009. Pediatric Ann. 38(10):557–561. [PubMed:19968193]

27- Lim D, Oakley AM, Rademaker M. Better, sooner. Successful tele dermo scopy service. 2012. Australia’s Journal Dermatol. 53(1):22–25. [PubMed: 22309326]

28- Mahnke CB, Jordan CP, Bergvall E, Person DA, Pinsker JE. The Pacific Asynchronous Telehealth (PATH) system: A review of 1,000 pediatric teleconsultations. 2011. Telemedicine Journal

Educational Health. 17(1):35–39. [PubMed: 21214304]

29- Munoz RA, Burbano NH, Motoa MV, Santiago G, Klevemann M, Casilli J. Telemedicine in pediatric cardiac critical care. 2012. Telemedicine Journal Educational Health. 18(2):132–136. [PubMed: 22283363]

30- Izquierdo R, Morin PC, Bratt K, et al. School-centered telemedicine for children with type 1 diabetes mellitus. 2009. Journal Pediatric. 155(3):374–379. [PubMed: 19464030]

31- McConnochie K, Wood N, Herendeen N, ten Hoopen C, Denk L, Neuderfer J. Integrating telemedicine in urban pediatric primary care: provider perspectives and performance. 2010. Telemedicine Journal Educational Health. 16(3):280–288. [PubMed: 20406114]

32- McConnochie KM, Tan J, Wood NE, et al. Acute illness utilization patterns before and after telemedicine in childcare for inner-city children. A cohort studies. 2007. Telemedicine Journal Educational Health. 13(4):381–390. [PubMed: 17848106]

33- The Patient Protection and Affordable Care Act. 2010. Pub L No. 111-148Available at:

www.gpo.gov/fdsys/pkg/BILLS-111hr3590enr/pdf/BILLS-111hr3590enr.pdf. Accessed March 31,2014

34- Woolf SH. The meaning of translational research and why it matters. 2008. JAMA. 299(2):211– 213. [PubMed: 18182604]

35- Chiang MF, Gelman R, Martinez-Perez ME, et al. Image analysis for retinopathy of prematurity diagnosis. 2009. Journal AAPOS. 13(5):438–445. [PubMed: 19840720]

36- McConnochie KM, Wood NE, Herendeen NE, ten Hoopen CB, Roghmann KJ. Telemedicine in urban and suburban childcare and elementary schools lightens family burdens. 2010. Telemedicine Journal Educational Health. 16(5):533–542. [PubMed: 20575720]

(34)

34 38- Cason J. Telerehabilitation: An Adjunct Service Delivery Model for Early Intervention

Services. 2011. International Journal of Telerehabilitation. 3(1):19-30. doi:10.5195/ijt.2011.6071. 39- Ciccia, Angela Hein, Whitford, Bridgid, Krumm, Mark, McNeal, Kay. Improving the access of

young urban children to speech, language and hearing screening via telehealth. 2011. Journal of Telemedicine and Telecare. Doi:10.1258/jtt.2011.100810 e244.

40- Guiberson M. Telehealth Measures Screening for Developmental Language Disorders in Spanish-Speaking Toddlers. 2016. Telemedicine Journal and e-Health. 22(9):739-745.

Doi:10.1089/tmj.2015.0247.

41- R. Matthew Reese, T. Rene Jamison, Matt Braun, Maura Wendland, William Black, Megan Hadorn, Eve-Lynn Nelson, Carole Prather. Use of Interactive Television in Identifying Autism in Young Children. 2015 May. Methodology and Preliminary Data. 45(5): 1474–1482.

doi:10.1007/s10803-014-2269-5.

42- Jessica Simacek, Adele F. Dimian, Jennifer J. McComas. Communication Intervention for Young Children with Severe Neurodevelopmental Disabilities Via Telehealth. 2017. Department of Educational Psychology. Doi10.1007/s10803-016-3006-z.

43- Vismara, L. A., Young, G. S., Rogers, S. J. Telehealth for expanding the reach of early autism training to parents. 2012 Autism Research and Treatment. Doi:10.1155/2012/121878.

44- Ingersoll B, Berger NI Parent Engagement with a Telehealth-Based Parent-Mediated Intervention Program for Children with Autism Spectrum Disorders. 2015. Predictors of Program Use and Parent Outcomes Journal Medical Internet Research. 17(10): e227.

45- McDufe, A., Machalicek, W., Oakes, A., Haebig, E., Weismer, S. E., Abbeduto, L. Distance video-teleconferencing in early intervention. 2013. Topics in Early Childhood Special Education, 33, 172–185. Doi:10.1177/0271121413476348.

46- Constantinescu, Gabriella, Waite, Monique, Dornan, Dimity, Rushbrooke, Emma, Brown, Jackie, McGovern, Jane, Ryan, Michelle, Hill, Anne. A pilot study of telepractice delivery for teaching listening and spoken language to children with hearing loss. 2014. Journal of Telemedicine and Telecare. P 135-140. Doi:10.1177/1357633X14528443

(35)

35 48- Pearl, P.L., Sable, C., Evans, S., Knight, J., Cunningham, P. et al. International telemedicine

consultations for neurodevelopmental disabilities. 2014. Telemedicine and e-Health. 20(6), 559-562. Doi:10.1089/tmj.2013.0275

49- Lindgren S, Wacker D, Suess A, et al. Telehealth and Autism: Treating Challenging Behavior at Lower Cost. 2016. Pediatrics.137(S2): e20152851O

(36)

36

(37)