Cochlear implant in patients with autistic spectrum disorder–-a systematic review

Tavares, Flávia da Silva; Azevedo, Yaná Jinkings; Fernandes, Luísa da Matta Machado; Takeuti, Alice; Pereira, Larissa Vilela; Ledesma, Alelluia Lima Losno; Bahmad Jr, Fayez

doi:10.1016/j.bjorl.2020.11.020

Flávia da Silva Tavares

Universidade de Brasília (UnB), Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0002-9955-6388

Yaná Jinkings Azevedo

Universidade de Brasília (UnB), Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0001-6671-0679

Luísa da Matta Machado Fernandes

Instituto René Rachou – Fiocruz Minas, FESF-TECH Bahia e Grupo de pesquisa em Políticas de Saúde e Proteção Social, Belo Horizonte, MG, Brazil

http://orcid.org/0000-0002-5440-7928

Alice Takeuti

Universidade de Brasília (UnB), Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0001-8817-9495

Larissa Vilela Pereira

Universidade de Brasília (UnB), Faculdade de Ciências de Saúde, Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0003-3284-5641

Alelluia Lima Losno Ledesma

Universidade de Brasília (UnB), Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0003-4564-622X

Fayez Bahmad Jr

Universidade de Brasília (UnB), Faculdade de Ciências de Saúde, Programa de Pós-Graduação em Ciências da Saúde, Brasília, DF, Brazil

http://orcid.org/0000-0002-1575-7589

*Corresponding author. E-mail:fayezbjr@gmail.com (F. Bahmad Jr).

Peer Review under the responsibility of Associação Brasileira de Otorrinolaringologia e Cirurgia Cérvico-Facial.

Conflicts of interest

The authors declare no conflicts of interest.

	Title	Author	Location	Year of publication	Study design	Total N
1	Measuring progress in children with autism spectrum disorder who have cochlear implants	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	Michigan–-USA	2004	Cohort studies	7
2	Cochlear implant candidacy in children with autism	Hayman and Franck²⁰20 Hayman CD, Franck KH. Cochlear implant candidacy in children with Autism. Semin Hear. 2005;26:217–25.	Philadelphia, Pennsylvania–-USA	2005	Case reports	3
3	Children with cochlear implants and autism–-challenges and outcomes: the experience of the National Cochlear implant program, Ireland	Robertson¹¹11 Robertson J. Children with Cochlear implants and Autism–-challenges and outcomes: the experience of the national Cochlear implant programme, Ireland. Cochlear Implants Int. 2013;14:11–5.	Dublin–-Ireland	2013	Case reports	10
4	Cochlear implantation in children with autism spectrum disorder	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	Miami–-USA	2015	Cohort studies	15
5	Receptive speech in early implanted children later diagnosed with autism	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.	Belgrade–-Serbia	2016	Cohort studies	14
6	Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	Manchester, UK	2016	Prevalence studies	22
7	Cochlear implantation in autistic children with profound sensorineural hearing loss	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	Warsaw–-Polônia	2018	Prevalence studies	6

	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.
Were the two groups similar and recruited from the same population?	Y	Y	Y
Were the exposures measured similarly to assign people to both exposed and unexposed groups?	U	Y	Y
Was the exposure measured in a valid and reliable way?	Y	Y	Y
Were confounding factors identified?	N	Y	Y
Were strategies to deal with confounding factors stated?	NA	Y	U
Were the groups/participants free of the outcome at the start of the study (or at the moment of exposure)?	U	Y	Y
Were the outcomes measured in a valid and reliable way?	Y	Y	Y
Was the follow-up time reported and sufficient to be long enough for outcomes to occur?	Y	Y	Y
Was follow up complete, and if not, were the reasons to loss to follow up described and explored?	Y	Y	Y
Were strategies to address incomplete follow up utilized?	U	Y	U
Was an appropriate statistical analysis used?	U	Y	Y
Bias risk (%)	50%	100%	81.81%

	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.
Was the sample frame appropriate to address the target population?	Y	Y
Were study participants sampled in an appropriate way?	Y	Y
Was the sample size adequate?	U	U
Were the study subjects and the setting described in detail?	U	U
Was the data analysis conducted with sufficient coverage of the identified sample?	U	U
Were valid methods used for the identification of the condition?	Y	Y
Was the condition measured in a standard, reliable way for all participants?	Y	Y
Was there an appropriate statistical analysis?	U	U
Was the response rate adequate, and if not, was the low response rate managed appropriately?	Y	Y
Bias risk (%)	55%	55%

	Hayman and Franck²⁰20 Hayman CD, Franck KH. Cochlear implant candidacy in children with Autism. Semin Hear. 2005;26:217–25.	Robertson¹¹11 Robertson J. Children with Cochlear implants and Autism–-challenges and outcomes: the experience of the national Cochlear implant programme, Ireland. Cochlear Implants Int. 2013;14:11–5.
Were patient’s demographic characteristics clearly described?	Y	Y
Was the patient’s history clearly described and presented as a timeline?	Y	Y
Was the current clinical condition of the patient on presentation clearly, described?	Y	Y
Were diagnostic tests or assessment methods and the results clearly, described?	Y	Y
Was the intervention(s) or treatment procedure(s) clearly described?	Y	Y
Was the post-intervention clinical condition clearly described?	Y	Y
Were adverse events (harms) or unanticipated events identified and described?	Y	Y
Does the case report provide takeaway lessons?	Y	Y
Bias risk (%)	100%	100%

	Author	Sex	Age at implant	Associated disabilities	ADS type	Year of implantation	Test	Results
1	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	F	9-years	U	PDD-NOS–-3-years before – preimplant, 6-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated an increase in their comprehension of spoken words after implantation
2	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	4-years	U	PDD-NOS–-Pervasive developmental disorder, 3-years post-implant 7-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated an increase in their comprehension of spoken words after implantation. Showed improvement in the auditory comprehension test score, going from a standard below 40 in the preoperative period to 72 stitches 5 years after surgery Demonstrated strong improvements in the Expressive Vocabulary Test, going from 54 points in the 24a month after implant until 81 60 months after implant Achieved a score of 100% correct on both words and sentences two years after implantation
3	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	7-years	U	Autism, 2 years before – preimplant, 5-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Communication presented with other people through sign language and gestures
4	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	3-years	U	Autism, 1-year post- implant–-4-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated an increase in their comprehension of spoken words after implantation
5	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	8-years	U	Autism, 3-years before–-preimplant, 5-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated an increase in their comprehension of spoken words after implantation
6	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	3-years	U	Autism, 2 years post- implant–-5-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated no comprehension of spoken words preoperatively or at the 12 months post-activation interval.
7	Donaldson et al.¹⁹19 Donaldson AI, Heavner KS, Zwolan TA. Measuring progress in children with autism spectrum disorder who have cochlear implants. Arch Otolaryngol Head Neck Surg. 2004;130:666–71.	M	3-years	U	Autism, 1-year before–-preimplant, 2-years-old	Between 1998 and 2003	MacArthur Communication Development Inventory, EVT, PPVT-III, MAIS/IT-MAIS, GASP-W, GASP-S, Parental ratings	Demonstrated an increase in their comprehension of spoken words after implantation
8	Hayman and Franck20	M	11.3 years	None	Autism, preimplant	U	Early Speech Perception Test, MLNT	Have a total communication class with other children with cochlear implants. He is reading on almost a 3rd-grade level, and educators believe it will continue to improve.
9	Robertson 11	U	2.1 years	None	Autism, 8 months post-implant–-2,9- year-old	2005	U	PECS (Picture Exchange Communication System)
10	Robertson 11	U	2.6 years	None	Autism, 7-years post- implant–-9-years-old	2005	U	ISL (Irish Sign Language)
11	Robertson 11	U	2-years	None	Autism, 2.1-years post-implant–-4.1- years-old	2007	U	Spoken language Understands common phrases. Speech intelligible to familiar listeners
12	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	4 (1) and 10 (2) years	None	Autism not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation. ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Identification/recognition of simple phrases (2 words) and commands’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Simple Phrases and Commands (Where is X, let us go, etc.)’’
13	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	1.5 (1) and 6 (2) years	None	AutismNot informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation. ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Simple Phrases and Commands (Where is X, let’s go, etc.)’’
14	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	3-years	None	AutismNot informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation. ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘Some vocalization (consonants, vowels, nasal sounds)’’ for ‘‘Simple Phrases and Commands (Where is X, let’s go, etc.)’’
15	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	4 (1) and 15 (2) years	None	Autism not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation. ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘Identification/recognition of words’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Able to produce sentences’’.
16	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	1.67 years	None	PDD-NOS (Pervasive developmental disorder–-not otherwise specified) not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘No produce sentences’’
17	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	4-years	None	PDD-NOS (Pervasive developmental disorder–-not otherwise specified) not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘Identification/ recognition of words’’ for ‘‘Identification/ recognition of simple phrases (2-words) and commands’’. Speech Expression: unchanged. Kept ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
18	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	2-years	None	PDD-NOS (Pervasive developmental disorder–-not otherwise specified) not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Identification/ recognition of simple phrases (2-words) and commands’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Simple Phrases and Commands (Where is X, let’s go, etc.)’’
19	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	1.5 (1) and 2 (2) years	None	Autism not informed if pre- or post-implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39questions evaluating the subjective impression of CI benefits	Speech Perception: Understands conversations; Speech Expression: Able to produce sentences
20	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.	M	0.9-year	U	Autism not informed if pre- or post-implant	Between 2008 and 2009	Categories of Auditory Performance (CAP); Speech Intelligibility Rating (SIR)	Auditory processing developed slowly. Could identify environmental sounds or discriminate speech sounds, with very little progress up to six years old, despite extensive speech and language therapy.
21	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.	M	1-year	U	Autism not informed if pre- or post-implant	Between 2008 and 2009	Categories of Auditory Performance (CAP); Speech Intelligibility Rating (SIR)	Auditory processing developed slowly. Could identify environmental sounds or discriminate speech sounds, with very little progress up to six years old, despite extensive speech and language therapy.
22	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.	F	0.8-year	U	Autism not informed if pre- or post-implant	Between 2008 and 2009	Categories of Auditory Performance (CAP); Speech Intelligibility Rating (SIR)	Maintained the same pre- and post-IC characteristics Auditory processing developed slowly. Could identify environmental sounds or discriminate speech sounds, no progress up to six years old, despite extensive speech and language therapy.
23	Mikic et al.²²22 Mikic B, Jotic A, Miric D, Nikolic M, Jankovic N, Arsovic N. Receptive speech in early implanted children later diagnosed with autism. Eur Ann Otorhinolaryngol Head Neck Dis. 2016;133:36–9.	M	1-year	U	Autism not informed if pre- or post-implant	Between 2008 and 2009	Categories of Auditory Performance (CAP); Speech Intelligibility Rating (SIR)	Auditory processing developed slowly. Could identify environmental sounds or discriminate speech sounds, with very little progress up to six years old, despite extensive speech and language therapy.
24	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	F	2.10 years	None	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Non-user speech perception, No vocalization, Non-user the CI, Sign Communication
25	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1.10 years	None	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words Some vocalization (consonants, vowels, nasal sounds) User the CI Some vocalization and PECS (Picture Exchange Communication System) Communication
26	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	3.8 years	None	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Understands conversations, Able to produce sentences, User the CI, Oral Communication
27	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	2.4 years	None	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words Words only User the CI Words Communication
28	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1-year	None	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of simple phrases (two words) and commands, Words only, User the CI, Word, sing and PECS (Picture Exchange Communication System) Communication
29	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1.10 years	None	PDD-NOS (Pervasive developmental disorder–-not	Between 1989 and 2015	Speech perception and expression categories (adapted from otherwise specified), post-implant	Non-user speech perception, No vocalization, Non-user the CI, None Communication Yeargin-Alisopp et al. 2003)
30	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	1.9 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: No, Ling’s 6 sounds teste: No, Reaction to spoken name: No, Response to spoken request: No, Response to name: No, Response to environmental sounds: No, Behavior changes: None, Better family interactions: Yes
31	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	2.5 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: Yes (only to drum), Ling’s 6 sounds teste: No, Reaction to spoken name: No, Response to spoken request: No, Response to name: No, Response to environmental sounds: No, Behavior changes: None, Better family interactions: Yes
32	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	1.9 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: Yes (only to drum), Ling’s 6 sounds teste: No, Reaction to spoken name: No, Response to spoken request: No, Response to name: No, Response to environmental sounds: No, Behavior changes: Reduced anxiety, Better family interactions: Yes
33	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	1.9 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: Yes (only to flute and drum), Ling’s 6 sounds teste: No, Reaction to spoken name: No, Response to spoken request: No, Response to name: Yes, Response to environmental sounds: Yes, Behavior changes: Reduced anxiety, Better family interactions: Yes
34	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	1.3 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: Yes, Ling’s 6 sounds teste: Yes, Reaction to spoken name: Yes, Response to spoken request: Yes, Response to name: Yes, Response to environmental sounds: Yes, Behavior changes: Reduced anxiety, Better family interactions: Yes
35	Lachowska et al.²⁴24 Lachowska M, Pastuszka A, Łukaszewicz-Moszynsk´ a Z, Mikołajewska L, Niemczyk K. Cochlear implantation in autistic children with profound sensorineural hearing loss. Braz J Otorhinolaryngol. 2018;84:15–9.	M	2.2 years	None	Autism post- implant	U	Medical history, Reaction to music and sound, Ling’s six sounds test, Onomatopoeic word test, Reaction to spoken child’s name, Response to requests, the questionnaire given to parents, Sound processor fitting sessions and data	Reaction to music: Yes, Ling’s 6 sounds teste: Yes, Reaction to spoken name: Yes, Response to spoken request: Yes, Response to name: Yes, Response to environmental sounds: Yes, Behavior changes: Reduced anxiety, Better family interactions: Yes

	Author	Sex	Age at implant	Associated disabilities	ADS type	Year of implan- tation	Test	Results
1	Hayman and Franck20	U	1 year	Cortical blindness Globally delayed	PDD-NOS 2.5-years post-implant–-3- year-old	U	U	Socially and emotionally responsive, demonstrates better eye contact and orienting, improved listening response, and improved nonverbal communication such as indicating choices, show of affection, and social preferences, receptive language has improved markedly
2	Robertson 11	U	3.2 years	Developmental delay/mild intellectual disability	Autism 10-months post- implant–-3.10 year-old	2004	U	ISL (Irish Sign Language)/spoken language. Understands common phrases. Speech unintelligible
3	Robertson11	U	2,4 years	Verbal dyspraxia	Autism 1.2-year post-implant–-3.6- year-old)	2005	U	ISL (Irish Sign Language)/spoken language. Understands common phrases. Speech unintelligible
4	Robertson 11	U	4.4 years	Cerebral Palsy/intellectual disability	Autism 1.1-year post-implant–-5.5- year-old)	2005	U	PECS (Picture Exchange Communication System)/LAMH (Language Alternative for the Mentally Handicapped)
5	Robertson 11	U	3.10 years	Epilepsy, left hemiparesis	Autism 1.0-year post- implant–-4.10- year-old)	2007	U	ISL (Irish Sign Language)/spoken language. Understands common phrases. Speech unintelligible
6	Robertson11	U	2.3 years	Intellectual dis- ability/visual diffs/multiple medical problems	Autism, 10 months post-implant–-3.1- years-old	2009	U	PECS (Picture Exchange Communication System)/LAMH (Language Alternative for the Mentally Handicapped)
7	Robertson 11	U	13.1 years	Intellectual disability	Autism, 7.5-years before–-pre- implant, 5.6-years-old	2010	U	PECS (Picture Exchange Communication System)/LAMH (Language Alternative for the Mentally Handicapped)
8	Robertson 11	U	7-years	Intellectual disability	Autism, 4-years before–-pre- implant, 3-years-old	2011	U	PECS (Picture Exchange Communication System)/LAMH (Language Alternative for the Mentally Handicapped)
9	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	1.75 years	Rumination, GERD (Gastroe- sophageal reflux disease), Strabismus	Autism not informed if pre or post- implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: unchanged. Kept Awareness, detection, or localization of sound Speech Expression: unchanged. Kept ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
10	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	5.5 years	Gross motor delay, Strabismus	Autism not informed if pre or post- implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for Ïdentification/ recognition of words’’. Speech Expression: unchanged. Kept ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
11	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	4.5 years	Prematurity, encephalopa- thy, gross motor delay	Autism Not informed if pre or post- implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Awareness, detection or localization of sound’’. Speech Expression: unchanged. Kept ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
12	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	3.5 years	polymicrogyria, gliosis, develop- mental	Autism Not informed if pre or post- implant delay	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Awareness, detection or localization of sound’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
13	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	2-years	ADHD (Attention Deficit Hyperactiv- ity Disorder)	PDD-NOS 2.5-years post- implant–-3- year-old	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘Awareness, detection or localization of sound’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Able to produce sentences’’
14	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	4-years	Ushers Syndrome	Autism Not informed if pre or post- implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Awareness, detection or localization of sound’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Some vocalization (consonants, vowels, nasal sounds)’’
15	Eshraghi et al.²¹21 Eshraghi AA, Nazarian R, Telischi FF, Martinez D, Hodges A, Velandia S, et al. Cochlear implantation in children with Autism spectrum disorder. Otol Neurotol. 2015;36:121–8.	U	3 (1) and 12 (2) years	Meningitis at 18-months	Autism Not informed if pre or post- implant	Between 1992 and 2011	Speech perception and speech expression evaluation ABR, ESP test, MLNT, PBK test Parental survey: 39 questions evaluating the subjective impression of CI benefits	Speech Perception: changed of ‘‘No awareness of environment’’ for ‘‘Understands conversations’’. Speech Expression: changed of ‘‘No vocalization’’ for ‘‘Able to produce sentences’’
16	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	F	1.8 years	Learning disabilities	Autistic spectrum, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Non-user speech perception, Some vocalization (consonants, vowels, nasal sounds). Non-user the CI. Some vocalization Communication
17	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	3.2 years	Prematurity Learning disabilities	Autistic spectrum, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words. Some vocalization (consonants, vowels, nasal sounds) User the CI, PECS (Picture Exchange Communication System) Communication
18	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1.8 years	Meningitis Learning disabilities	Autistic spectrum, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of simple phrases (two words) and commands; Some vocalization (consonants, vowels, nasal sounds). User the CI. Sign and PECS (Picture Exchange Communication System) Communication
19	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	3.9 years	Prematurity	Autistic spectrum, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Non-user speech perception, No vocalization, Non-user the CI, Sign and PECS (Picture Exchange Communication System) Communication
20	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1.10 years	Communication disabilities	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words. Words only. User the CISome words, signs, and PECS (Picture Exchange Communication System) Communication
21	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	5-years	Meningitis, ADHD (Attention- Deficit Hyperkinetic Disorder) Communication disabilities	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Understands conversations, Able to produce sentences, User the CI, Oral Communication
22	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	6.11 years	Prematurity, ADHD (Attention- Hyperkinetic Disorder)	Autistic spectrum, post-implant Deficit	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Understands conversations, Able to produce sentences, User the CI–-unilateral, Oral and sign Communication
23	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	3-years	Smith-Lemli- Opitz Syndrome Global developmental delay	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of simple phrases (two words) and commands, Simple phrases and commands, User the CI–-unilateral, Phrases and sign Communication
24	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	3.1 years	Communication disabilities	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words. Some vocalization (consonants, vowels, nasal sounds) User the CI Some words and sing Communication
25	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	5.2 years	ADHD (Attention- Deficit Hyperkinetic Disorder) Learning disabilities	Autistic spectrum, post-implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of simple phrases (two words) and commands, Words only, User the CI, Word and sing Communication
26	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	1.3 years	Waardenberg syndrome, Learning disabilities	PDD-NOS post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Non-user speech perception, No vocalization, Non-user the CI, None Communication
27	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	4.6 years	Learning disabilities	PDD-NOS, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	‘‘Non-user speech perception, No vocalization; Non-user the CI, Sign and PECS (Picture Exchange Communication System) Communication
28	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	2.8 years	Prematurity Global develop- mental delay	PDD-NOS, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Awareness, detection of localization of sound, No vocalization, User the CI, Sign Communication
29	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	8.2 years	CMV (Cytomegalovirus infection)	Asperger’s disorder, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Understands conversations, Able to produce sentences, User the CI, Oral and sign Communication
30	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	2.8 years	Waardenberg syndrome, ADHD (Attention- Deficit Hyperkinetic Disorder)	Asperger’s disorder, post- implant 2003)	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al.	‘‘Understands conversations, Able to produce sentences, User the CI, Oral Communication’’
31	Valero et al.²³23 Valero MR, Sadadcharam M, Henderson L, Freeman SR, Lloyd S, Green KM, et al. Compliance with cochlear implantation in children subsequently diagnosed with autism spectrum disorder. Cochlear Implants Int. 2016;17:200–6.	M	2.10 years	Prematurity, Learning and commu- nication difficulties	Autistic Spectrum, post- implant	Between 1989 and 2015	Speech perception and expression categories (adapted from Yeargin-Alisopp et al. 2003)	Identification/recognition of words. Some vocalization (consonants, vowels, nasal sounds). User the CI. Sing and PECS (Picture Exchange Communication System) Communication

Outcome	No of studies (No of patients)	Study design	Factors that may decrease the certainty of the evidence					The effect per 1.000 patients tested	Test accuracy CoE
Outcome	No of studies (No of patients)	Study design	Risk of bias	Indirect evidence	Inconsistency Imprecision		Publication bias	The effect per 1.000 patients tested	Test accuracy CoE
True positives (patients with [condition of interest])	6 studies 95 patients	Cross-sectional (cohort type accuracy study)	Serious	Not serious	Not serious	Serious	Strong association	0 to 0	⊕⊕⊕◯ Moderate
False negatives (patients incorrectly classified as not having [the condition of interest])								0 to 0
True negatives (patients condition of interest])	1 study 7 patients	Cross-sectional (cohort type without [the accuracy study)	Serious	Not serious	Serious	Not serious	All potential confounding factors would reduce the demonstrated effect	0 to 0	⊕⊕⊕◯ Moderate
False positives (patients with [the condition of interest] incorrectly classified)								1000 to 1000

[1] *Corresponding author. E-mail:fayezbjr@gmail.com (F. Bahmad Jr).

Brasil

Brasil

Cochlear implant in patients with autistic spectrum disorder–-a systematic review

Abstract

Introduction:

Objective:

Methods:

Results:

Conclusion: