Deaf and hard of hearing children are at risk of exclusion from community life and education, which may increase their risk of mental health conditions. This study explores the experience of deaf and hard of hearing children in the Gaza Strip, with particular focus on the factors that contribute to psychological wellbeing and distress. In-depth interviews were conducted with 17 deaf and hard of hearing children, 10 caregivers of deaf and hard of hearing children and eight teachers of deaf and hard of hearing children in mainstream and special schools, across the Gaza Strip. Further, three focus group discussions were held with deaf and hard of hearing adults and disability leaders, mental health specialists and other teachers of deaf and hard of hearing children. Data collection was completed in August 2020. Key themes identified in the analysis included lack of accessible communication, community exclusion, negative attitudes towards hearing impairment and deafness and the impact on deaf and hard of hearing children’s sense of self, and limited family knowledge on hearing impairment and deafness. Further findings focused on strategies to improve the inclusion of deaf and hard of hearing children and how to promote wellbeing. In conclusion, participants in this study believed that deaf and hard of hearing children in the Gaza Strip are at increased risk of mental health conditions. Changes are needed across community and government structures, including education systems, to promote the inclusion of deaf and hard of hearing children and to support their psychological wellbeing. Recommendations from the findings include increasing efforts to improve awareness and reduce stigma, providing better access to sign language for deaf and hard of hearing children, and offering training for teachers of deaf and hard of hearing children, especially in mainstream environments. One in five people experience hearing impairment, of whom more than 80% live in low- and middle-income countries (LMICs) . Whilst Deaf or hard of hearing children across the world can experience challenges in daily life, with regards to educational attainment, social inclusion and community participation –4. Deaf Little research exists on the experiences of deaf and hard of hearing children in LMICs. The vast majority of research comes from high-income settings and findings are not representative of LMICs, where there exist different conceptions and attitudes towards disability and hearing impairment, and often more limited availability of specialised support and services. Further, little evidence is available on the experiences of deaf and hard of hearing in conflict-affected settings, where, in general, people are at greater risk of mental health conditions . Deaf anThis study explores the experiences of deaf and hard of hearing children in the Gaza Strip, an area that has been affected by conflict and blockade, resulting in a deteriorating socioeconomic situation that has negatively impacted on the economy, education, food security, and access to basic services . There iOngoing conflict, economic hardship and restricted life prospects are associated with high rates of depression, anxiety and post-traumatic stress disorder for youth in the Gaza Strip –25. ThesGiven the dearth of evidence on deaf and hard of hearing children in the Gaza Strip, this study aimed to investigate the experiences of deaf and hard of hearing children in daily life and the factors that contribute to their inclusion, quality of life and mental health. This information can help contribute to the development of context appropriate policies and interventions.This study used qualitative research methods to explore the experiences of deaf and hard of hearing children in the Gaza Strip. In-depth interviews were conducted with 35 participants, including 17 primary school age deaf and hard of hearing children, 10 caregivers, and eight teachers of deaf and hard of hearing children in mainstream and special schools. Three focus group discussions were held with five deaf and hard of hearing adults and disability leaders, seven mental health and psychosocial support specialists and school counsellors, and 13 teachers of deaf and hard of hearing children in mainstream and special education settings.The research and analysis were guided by a steering committee of key stakeholders in the Gaza Strip. This group comprised: five deaf and hard of hearing people and representatives from organisations of deaf and hard of hearing people; two caregivers of deaf and hard of hearing children; four teachers of deaf and hard of hearing children; three mental health and psychosocial support specialists; and five representatives from government level institutions, including the Ministry of Education.The research was conducted across each region of the Gaza Strip. In the Gaza Strip, deaf children with severe to profound hearing impairment are typically taught in special schools. Hard of hearing children with mild to moderate hearing impairment are typically taught in mainstream schools. Mainstream schools are government run and free to attend. Special schools are not supported by government financing and are often managed by non-governmental organisations (NGOs). Families pay a fee for their child to attend a special school. Each mainstream and special school is required to have a school counsellor who is tasked with promoting mental wellbeing and supporting children experiencing distress. When a child presents with severe symptoms of psychological distress, they are referred by the school to counsellors at district level who provide more in-depth assessment and support. At this district level, there are mental health specialists trained to work with deaf and hard of hearing children.A summary of participant characteristics is presented in Participants for the in-depth interviews were purposively sampled through mainstream and special schools across four regions in the Gaza Strip  in order to maximise variation, as based on Patton’s maximum variation sampling strategy . ParticiParticipants for the three focus group discussions were recruited through local organisations of persons with disabilities (OPDs), disability NGOs, mental health associations and schools. The first group comprised of three deaf and hard of hearing adults and sign language users, one individual with a physical impairment and one individual with a visual impairment, who also works as a counsellor. These individuals were representatives from OPDs and NGOs supporting deaf and hard of hearing people. The second group comprised five school counsellors and two mental health professionals working with children in the community. The third included seven special school teachers and six mainstream school teachers. These teachers were not the same as those included in the in-depth interviews.Data collection was conducted in July 2020. Interviews were held in a trusted, central location, as requested by participants, many of whom expressed concern about being audio-recorded in their own home. As the research was conducted during the COVID-19 pandemic, government guidance on safety was followed.Semi structured, in-depth interviews with deaf and hard of hearing children, caregivers and teachers sought to explore the views, experiences, emotions and perspectives of deaf and hard of hearing children and caregivers in the Gaza Strip, both in school and the wider community. In addition, focus group discussions were held with individuals who have expertise in understanding the experiences and needs of deaf and hard of hearing children and their families. Through dynamic group discussion, these focus groups aimed to elicit multiple opinions on the experiences of deaf and hard of hearing children, as well as recommendations to improve quality of life and psychological wellbeing. Topic guides are available in The interview guides were developed by the research team, piloted with six participants  and adapted as needed thereafter. Questions explored the experiences of deaf and hard of hearing children, their families and their teachers, focusing on experiences in education, healthcare and community life. Specific questions were asked on communication, mental health, wellbeing and attitudes towards hearing impairment and deafness. Topic guides for the focus group discussions aimed to further understand these areas, explore the wider context for deaf people in the community, and appraise the factors contributing to wellbeing. Data was collected during the COVID-19 pandemic, however the study sought to understand experiences in more typical times, and we did not explicitly explore the experiences of deaf and hard of hearing children and families in the context of the pandemic.Interviews were conducted in Arabic with those using oral communication. Palestinian sign language interpretation was available for those using sign language for communication. Emotion cards were used to help children understand a question or to describe their feelings. Children were interviewed individually, unless the child or caregiver desired otherwise. There was occasionally need for a family member to sit in as interpreter, when a child communicated through a signed language developed with family at home. Each interview lasted between 30–60 minutes. Five pilot interviews were conducted, in which to practice interviewing techniques and refine the topic guides. The data from these interviews has been included in the analysis. All interviews were audio-recorded. Sign language users had their response reported orally by an interpreter. Audio-recordings were transcribed verbatim. Each was translated and transcribed directly into English, by an independent transcriber, and subsequently checked against the audio file by the interviewer. Each transcript was anonymised and stored on a secure server.Data was analysed using thematic analysis . In stepInterviews were conducted by a trained interviewer (RH), who received a three-day, one-to-one training with the lead author (NS), a qualitative researcher in global disability and mental health. The interviewer’s background was as an experienced audiologist at a major service unit for deaf and hard of hearing people in the Gaza Strip; Atfaluna Society for Deaf Children. The interviewer knew some local sign language, but not enough to conduct interviews without support from an interpreter. The interviews themselves were held at Atfaluna’s headquarters, at the request of participants. With Atfaluna known to many deaf and hard of hearing people and their families in the Gaza Strip, their responses may have been influenced by the dynamic. To mitigate this, we assured participants that their responses and data would be kept confidential and that their access to services would not be affected as a result of the interview. For many, Atfaluna offers a safe space for deaf and hard of hearing children and families, and in many instances, the interviewer reported the benefit of this association on building comfort and rapport.The analysis was led by NS, who is based in the United Kingdom. NS supported the data collection remotely through co-training, regular post-interview discussions with the interviewer and remote participation in workshops/advisory committees as travel to Gaza strip was not possible due to the COVID-19 pandemic. To limit potential biases in analysis led by an off-site foreign researcher, the interpretations were reviewed by the research team in the Gaza Strip with experience in supporting deaf and hard of hearing children in the region. The findings were further discussed with the steering committee in the Gaza Strip.To promote reliability, validity and rigour of this qualitative research, we adopted a number of methodological strategies, as recommended in the literature , 29: (1)Ethical approval for this study was obtained from the Research Ethics Committee at the London school of Hygiene & Tropical Medicine (19144) and the Palestinian Health Research Council (PHRC/HC/697/20).Informed written consent was received from all participants. Caregivers or guardians provided informed consent for children and further assent was sought from children using a simplified information sheet.Across the interviews, there was the consistent understanding from adult participants that deaf and hard of hearing children in the Gaza Strip were at increased risk of psychological distress and mental health concerns. Various associated factors were reported, including language deprivation, community attitudes and family support.Participants reported that deaf and hard of hearing children with milder levels of hearing impairment were less likely to experience challenges with communication and community participation, and they were often able to communicate orally. However, deaf and hard of hearing children with moderate to profound hearing impairment faced daily challenges with communication and engagement with others in the community as sign language use is limited across the region. Communication challenges restricted social inclusion and participation, negatively impacting on the psychological wellbeing of deaf and hard of hearing children.Although Palestinian sign language use is increasing across the Gaza Strip, it is still limited in healthcare, mainstream schools and community settings. Teachers, caregivers and deaf and hard of hearing adults told us how many deaf and hard of hearing children experience language deprivation, with families unable to teach them sign language or refusing to allow their child to learn.“I: Do they use sign language?P: No it is not allowed… once her uncle tried to use sign language and I prevented him from doing so. If I wanted her to learn and use sign language, I would give her sign language courses.”(Father of a child aged 10–12 with severe hearing impairment)We were told that many families in the Gaza Strip do not know Palestinian sign language and do not use it at home, even if their child uses the language at school and despite many of the children interviewed telling us that they prefer to communicate using sign language. Many of the families interviewed perceived sign language negatively and desired their child to use oral speech, even when the child had difficulty doing so. Some attributed sign language for their child’s poor oral skills and some did not understand it to be a language itself and their child’s natural form of communication. Limited sign language use and support resulted in examples of family and child having limited communication or regular misunderstanding and miscommunication.“If their cousins gather and talk, she will withdraw because she cannot hear and she cannot understand what they are saying, as they are talking fast. The same thing when my sisters and I gather. This thing bothers me and bothers her, but what we can do?”(Mother of a child aged 10–12 with moderate hearing impairment)The lack of engagement with sign language was not the case for all families, and there were positive examples of caregivers supporting the use of sign language by their child and seeking training for themselves. After learning themselves, there were examples told to us of family members training others, including hearing siblings, aunts, uncles and cousins. Support for sign language and accessible communication was often seen in families with multiple deaf and hard of hearing children, where siblings could communicate with and support one another. One child interviewed even taught their hearing friends at a mainstream school some simple signs, so they could better communicate and bond. However, despite wishing to support sign language use, some family members reported limited awareness on where to receive sign language training and communication support. Some children used home sign language as a result of limited exposure to Palestinian sign language and can only communicate with a few others as a result; often deaf and hard of hearing siblings from whom they learned to communicate.Some children interviewed had strong oral skills. They found it easier to participate in community life and were integrated into life at a mainstream school. That said, many of these children still expressed a preference for sign language, because oral communication was often tiring and difficult to maintain, especially in group situations and noisy environments.Many deaf and hard of hearing children in mainstream schools described difficulty in keeping up with teachers and peers, who often have limited awareness of deaf and hard of hearing children’s communication needs and how best to support them. We were told by many, including teachers, that difficulty with communication in mainstream schools significantly hampered the learning and education for many deaf and hard of hearing children. Limited communication also makes inclusion difficult with hearing peers in school and the community. This was reported to cause isolation and psychological distress. Some caregivers told us that they do not allow their deaf or hard of hearing child out into the community because of their difficulties communicating with others.“His nature is sociable, he tries to communicate with other kids and join them, but they cannot understand what he is saying, which makes him upset.”(Father of a child aged 6–9 with moderate hearing impairment)In contrast, the special school environment was more supportive of sign language and children and teachers in these settings reported very few instances of miscommunication or isolation of deaf children. A deaf adult told us how, as a child, he had to move back to a special school after joining a mainstream school, as communication in the mainstream environment was too challenging and teachers didn’t account for his deafness and communication needs. For example, he lipreads and teachers regularly faced away from him when talking. He expressed regret that he had to leave, as he wanted to learn in a mainstream environment.Language deprivation, challenges in communication and resulting social isolation were noted by many of the participants as the major influence on a child’s mental health and wellbeing.I: Are deaf children at risk of mental health issues?P: Yes, because they cannot keep up with the family talking. What’s causing a psychological problem is that a family will shorten up a long conversation into two words. This upsets a child and makes them prefer loneliness.Participants, including deaf adults, teachers and mental health specialists, discussed communication in the context of protecting and promoting mental health and wellbeing. Communication and the resulting social integration were described as the main source of a child’s confidence, self-esteem and wellbeing. Participants recommended that families and teachers be given sign language training to combat the language deprivation experienced by many deaf and hard of hearing children in the Gaza Strip and to promote their psychological wellbeing.“When the student has good communication in sign language with their family members, this comforts and relieves them very much. When the mother communicates in sign language with her child, the child becomes able to express and explain everything. The same thing happens when they communicate with their teacher, they feel that they are just as other classmates. When they go on a school journey and they communicate with teachers and other people, they break the ice and overcome the fear with outside. As a result, they become more confident… This gradually strengthens their personalities and their self-confidence.”Deaf adults told that us that communication was improving in the Gaza Strip, with Palestinian sign language more widely used than when they were children. However, improvements in the attitude towards and understanding of sign language was slow, and many families were still resistant to its use. Services and programmes to support use of sign-language, as well as speech and language therapy and occupational therapy, were often unavailable to caregivers, except through one or two specialist organisations. Foremost was Atfaluna Society for Deaf Children, which was often cited as the primary support for deaf children in the Gaza Strip, although not all participants were aware of the available support from this organisation and other specialist organisations supporting deaf and hard of hearing children. They did not have information on support available, where to seek support and how to access it. Deaf adults also told us that it was difficult to find qualified sign language interpreters in the region and sign language training was sparse. The services were not sufficient to meet the need.“Frankly, the Department of Education hasn’t given us sign language courses. I use simple, easy things for students. For example, go, come, and understand… I had four students with hearing disabilities and I couldn’t communicate well with them.”(Teacher in a mainstream school)Assistive technology was commonly discussed in the context of communication. Many caregivers described hearing aids as useful for children who use them and many wanted their child to use one, even if the improvements in hearing were marginal. However, some caregivers and children spoke of ill-functioning or ill-fitting hearing aids, negating any benefits for communication and limiting their ability to engage in community and classroom activities. Caregivers told us that paying for and maintaining the hearing aids is expensive, with some caregivers forgoing food to afford batteries. Repair services are also limited and there can be long waitlists if a hearing aid is damaged, especially if originally sourced from a country out of the Gaza Strip. Cochlear implants were perceived positively among the participants, with some having gone abroad to receive them, to countries such as Egypt and Australia. However, this was not available for all families of deaf and hard of hearing children. Many of those that had done so still relied on financial support from an NGO or international sponsor. Cochlear implantation is becoming more widely available in the Gaza Strip and many families note this as an appealing option to them. Deaf adults interviewed, however, were often disparaging of such assistive technologies and encouraged families to embrace their child’s hearing impairment and to cement a strong Deaf identity in them.Stigma and discrimination in the community also presented barriers to social inclusion and participation for deaf and hard of hearing children, and had a negative impact on psychological wellbeing. This was not the case for all deaf and hard of hearing children and adults, and some reported positive relationships with neighbours and hearing peers. However, we were told that many in the community still held discriminatory views and behaviours. Awareness of disability and deafness was said to be poor. Knowledge was often based on a medical or charity model of disability and deafness, with little understanding of rights-based perspectives in line with the United Nations Convention on the Rights of Persons with Disabilities (UNCRPD) . This liRespondents felt that it was common for the community to look on families and deaf and hard of hearing children with sadness and pity, and we were told that deaf people were seen to be unequal or were simply not acknowledged in society.“For example, if we are in a car, people will stare at him [her deaf son], and some of them ask, ‘Why is he like this?’. Someone said, ‘Poor boy’. I said, ‘Why? What’s wrong with him [her deaf son]? Thank God, they are a gift’.”(Mother of child aged 6–9 with severe hearing impairment)Participants reported that negative attitudes often proceed to more overt acts of discrimination. We were told that hearing people often use derogatory terms when talking about deaf people, including an Arabic slur which translates loosely to ‘dumb’ or ‘retarded’ (derogatory terms themselves across much of the world).“They whisper and point at me that I’m dumb [Arabic slur]”(Child aged 10–12 with severe hearing impairment)Discrimination was said to severely impact on deaf and hard of hearing children (and adults) in the Gaza Strip. Children and caregivers told us of the psychological distress caused when community members or other children made them feel different or marginalised. Children told us they often cried as a result. There were several examples of such instances. A father told us how he moved his family to another region of the Gaza Strip, as his son was facing such severe discrimination; others used to steal his hearing aids and run away. Similarly, a mother told us how her daughter was bullied constantly at school.“She was bullied at school by other children. They would remove her hearing aid and bully her. She would come home crying. I tried to speak to the teachers about this, but they didn’t help. No one understands her communication or possibly needs. I decided to move her to another school but the same thing happened. I eventually moved her to a special school, where things got better. She has since joined a mainstream government school after her communication improved.”(Mother of child aged 10–12 with moderate hearing impairment)There were a few examples reported in which bullying and discrimination dissipated once caregivers and teachers explained deafness to other children, other teachers and community members. Some caregivers reported speaking about deafness to teachers individually, as well as presenting to other caregivers and children at the school. Some teachers with experience and knowledge about deafness instructed other teachers at their school, with support from the school counsellor and headteacher. These efforts were reported to improve awareness of the experience of deaf and hard of hearing children and helped dissipate some of the stigma and discrimination seen. Caregivers are important advocates in these initiatives.“It was hard at the beginning. They mocked him every single day, which made him sad. He was crying all the time. I explained his case to the children and now they understand him and they started playing with him.”(Mother of child aged 6–9 in a mainstream school with moderate hearing impairment)Special schools offered a more inclusive and supportive environment, with no discrimination reported to us amongst teachers or other children. However, there were no reports of systematic governmental or organisational training and resources for special school teachers or others to instruct mainstream teachers on supporting the inclusion of deaf and hard of hearing children in mainstream schools.Further, few deaf and hard of hearing spaces and community structures were reported. Participants advocated for more deaf and hard of hearing friendly spaces, where deaf and hard of hearing children could learn about their hearing impairment, Deaf culture, sign language and be engaged with other deaf and hard of hearing people in a safe space. Participants also suggested that deaf and hard of hearing children need to be more included in community activities, to reduce isolation and improve psychological wellbeing.“For us as deaf people, not more than 3% are included in society. At the present time, the community should make more effort to give deaf people their own space, and to involve them in community activities, so that people know more about the skills and experiences of deaf people.”It was clear that many caregivers and families interviewed were proud of their deaf and hard of hearing children and expressed to us deep love, but some had not fully accepted their child’s hearing impairment and deafness. Caregivers were said to occasionally hide their deaf or hard of hearing child, so as not to affect their siblings’ future and experience in the community. Others did not want to see their child wearing a hearing aid and refused to let them learn sign language. A deaf adult told us of a story in which a mother refused for her deaf son to marry a deaf woman, as she feared their child would also be deaf; she wanted him to marry a hearing woman instead.Limited acceptance seemed to result from limited understanding and awareness of deafness and the rights of deaf and hard of hearing people, paired with the influence of negative community attitudes. Many of the caregivers interviewed had never received information or support on hearing impairment when their child was diagnosed, despite wishing for such. Caregivers called for structured training programmes with other caregivers, access to online resources and support from specialist organisations. Those that did receive such support reported positive benefits and typically presented with more positive attitudes to hearing impairment.For some other caregivers, internalised stigma among families appeared to result from the challenges they perceive a result of having a deaf or hard of hearing child.“It affects all my life system. It takes all of me, my efforts, my time, and it takes me from my other children.”(Mother of child aged 6–9 with severe hearing impairment)As a result of negative community and family attitudes, some children told us that they did not want to be deaf or hard of hearing, or to be different, and many were embarrassed and ashamed to use sign language in public or to be seen wearing a hearing aid. One father said his daughter feared she would never get married because of her hearing aid.“Once she said, ‘I know why they are treating me like that, it is because I wear a hearing aid.’ And one time I saw her crying because her sister got engaged and she said, ‘I will never get married because of my hearing aid’.”(Father of child aged 10–12 with severe hearing impairment)The discrimination in the community, and the internalised stigma among families and children, negatively impacts on deaf and hard of hearing children’s sense of identity and ultimately their mental health and wellbeing. Children were said to get upset and angry at being different to others. One mother told us of how she enrolled her daughter in psychosocial counselling, as she was very down and tired of being “different”.Participants believed community awareness and improving understanding of deafness and hearing impairment was of the utmost importance in addressing stigma, discrimination and improving engagement with Deaf culture and norms, such as sign language. Addressing stigma and discrimination was said to be vital in improving the psychological wellbeing of deaf and hard of hearing children. Some told us that this awareness is continually improving in the Gaza Strip, particularly among the younger generation, who are more knowledgeable and supportive of deaf and hard of hearing people, as a result of greater exposure to the internet and international campaigns.Children that engaged with deaf and hard of hearing peers and Deaf communities reported the positive impact on quality of life and psychological wellbeing. They enjoyed having deaf and hard of hearing friends and interacting with others with a shared experience. Many deaf and hard of hearing children in mainstream education said that they had few deaf and hard of hearing friends, although they would like to. Those that did have deaf and hard of hearing friends valued those relationships. “The first is to focus on things that help to integrate them into society, and the second is to spread culture about hearing disability among children so that they accept deaf colleagues or any other disability without making fun of them.”(Adult with a mobility impairment and representative from an organisation of persons with disabilities)Building a sense of self and building self-esteem was said to be key in promoting mental health and wellbeing among deaf and hard of hearing children. We were told that building a sense of identity and confidence in children starts with the family. Participants told us how important it was for families to learn about hearing impairment, deafness, deaf and hard of hearing rights, communication, sign language and how best to support their child. Many participants believed that deaf and hard of hearing children develop a strong sense of self and mental wellbeing when families are accepting and supportive of their children. Participants noted that caregivers must promote belief and expectation in their child.Further, we were told of the importance of caregiver involvement in their child’s education and life experience at school, especially in mainstream education. Doing so was considered to improve awareness and inclusion at schools, and caregivers expressed eagerness to work closely with teachers to ensure consistent communication and support.Examples of good practice were seen in some special schools, including awareness raising and psychosocial support. This included sessions with caregivers on how to communicate with and support their deaf or hard of hearing child. Further, we were recommended that deaf and hard of hearing role models visit schools, both to raise awareness amongst the students on deafness and inclusion and to help build the self-esteem and aspirations of deaf and hard of hearing children and their families.“We have a lot of attitudinal problems with adults and teenagers. We keep implementing awareness raising activities and psychosocial support sessions. These are very important to be included, especially as caregivers lack sign language, so they don’t communicate with their children. Teachers manage the whole communication process and guide the students. We also implement awareness raising activities with caregivers to guide them on how to communicate with and treat their children. We advise them to talk to their children, to understand them, and to identify and analyse their problems.”As presented, participants reported a number of factors that impact on deaf and hard of hearing children’s mental health and psychological wellbeing. Intervention and support is needed across a number of areas to promote wellbeing, including reducing stigma and improving communication. Deaf adults and teachers emphasised that the development of support for deaf and hard of hearing children and families at school and in the community is not going to happen without government support. According to the deaf adults we spoke with, inclusion at government level was viewed as talk with no action. We were told that this stemmed from limited understanding of hearing impairment and deafness at government level, which, in turn, contributed to the lack of inclusion and awareness in the community.“Even the government decision makers know nothing about us [deaf people]. What we are, what we feel and what we need. They don’t know us. There’s no awareness of what disability really is. They think we deaf people are less intelligent than others, they think our learning abilities are slow and hopeless… We, like all other disabilities, are no different and no less.”Deaf and hard of hearing children in the Gaza Strip were reported to be facing increased risk of mental health issues. Efforts are needed to build self-esteem and identity in deaf children, so that they may reach their potential and realise personal wellbeing.“When a child with disability is left behind because of their disability, this would complicate their mental health and cause them lots of psychosocial problems. We need to improve their skills, look into their talents and work on developing them. This would improve their self-trust and help them overcome their mental health and psychosocial problems.”(School counsellor at a mainstream school)For this to occur, families, teachers and the community need the understanding on how to support inclusion of deaf and hard of hearing children, which includes knowledge on communication and deaf culture.Participants in this study reported that deaf and hard of hearing children in the Gaza Strip are at increased risk of psychological distress, resulting from language deprivation, discrimination and exclusion from community participation. These findings are consistent with previous literature on the social inclusion and mental health of deaf and hard of hearing populations –13.Children in special school environments commonly used sign language and engaged with deaf and hard of hearing peers. Often, their caregivers were well-informed on the support available, on hearing impairment, on deafness and on deaf and hard of hearing rights, as a result of support and outreach from the special schools. These children appeared to be at lower risk of poor mental health. Children in mainstream environments were most likely to feel included if they used oral communication. These children talked of having hearing friends and generally positive experiences in the community. They tended to have milder hearing impairment and/or used a hearing aid or cochlear implant. They did not often report experiences of mental distress, likely related to fewer challenges in daily life and feeling part of the wider community. As is consistent with previous literature, our interviews indicated that the children most at risk of poor mental health were those experiencing language deprivation and social exclusion, especially in a mainstream schooling environment –33. OfteOur findings suggest that, in general, Deaf culture and awareness is limited in the Gaza Strip. Many deaf and hard of hearing children do not have a positive self-image and many deaf and hard of hearing children in the Gaza Strip experience prejudice and discrimination at some point in the community. This was not universal, with some describing positive experiences. Largely, prejudice and stigma appeared to stem from a lack of awareness on hearing impairment and deafness, and understanding of Palestinian sign language as a language. This lack of awareness and knowledge was viewed as a challenge at both community and government level, and was shown in school settings. Stigma is a common barrier experienced by youth with disabilities, but the current state of evidence on stigma-reduction activities for children with disabilities in LMICs is limited . Most stKey in our findings is the language deprivation experienced by some deaf and hard of hearing children in the Gaza Strip. Language deprivation in early life is a fundamental issue and has lifelong consequences . Beyond As well as improved access to Palestinian sign language, many deaf and hard of hearing children would benefit from improved access to oral-communication services and devices. Our findings reveal a need to improve access to assistive technology, including hearing aid and cochlear implant assessment, provision and maintenance. Many respondents in this study reported experiencing difficulties with poorly functioning assistive products, which hindered oral communication for those that could use and may prefer it. In order to provide early support and intervention, the government in the Gaza Strip may wish to provide systematic newborn hearing screening programmes. Regardless of a family’s decision on the use of hearing aids, cochlear implants and audiology therapies, we encourage health professionals to immediately refer deaf and hard of hearing children to Deaf associations, networks of parents of deaf and hard of hearing children and schools that cater for the needs of deaf and hard of hearing children when they first identify hearing loss.In addition to the above recommendations, training for families may be important to improve deaf and hard of hearing awareness and mitigate the risk of language deprivation. Evidence shows that early intervention with caregivers promotes better awareness on disability, rights and supporting the child . InterveWith the importance of education and the role that school plays in the inclusion and wellbeing of deaf and hard of hearing children, it is important that schools are be able to support deaf and hard of hearing children. Throughout the interviews, it was clear that special schools in the Gaza Strip were better equipped to provide this support to deaf and hard of hearing children, with Palestinian sign language being routinely used to communicate, aiding both education and social inclusion. With the promotion of inclusive education across the world, mainstream schools in the Gaza Strip need the resources and training to better support deaf and hard of hearing children in their classrooms, including sign language training and interpretation services . The relIn efforts to support deaf and hard of hearing children, there is need for government, funders and service providers to partner with deaf and hard of hearing communities and organisations of deaf and hard of hearing people in the Gaza Strip. Evidence has shown that organisations of persons with disabilities can produce positive outcomes for people with disabilities and these organisations are key in achieving equal rights for deaf and hard of hearing people and people with disabilities . In the There is also a need to develop mental health support that is targeted to the needs of deaf and hard of hearing children. Although important to address the risk factors of poor mental health, as identified in this study, it is equally important to ensure appropriate support for those that do experience distress. In a systematic review of mental health support for deaf and hard of hearing children, appropriate interventions included peer-support, caregiver-child interaction therapy and resilience training . Many inChildren and adolescents in the Gaza Strip are at increased risk of mental health conditions, regardless of disability . deaf anInteresting to note in this discussion is the limited information reported by participants on the direct impact of conflict and blockade on the psychological wellbeing of deaf and hard of hearing children. Few participants reported on these issues, instead focusing on the topics discussed in the results of this study. This may be because questions asked in the topic guides did not include conflict-specific questions. Questions asked more broadly on factors that influence the mental health of children in the Gaza Strip. There was opportunity for participants to mention conflict and blockade under these questions. It is important to note also that data collection occurred before the recent escalation of rocket fire in 2021 and there is need to assess the wellbeing of deaf and hard of hearing children  since this time, so that appropriate support can be provided.This research encountered challenges resulting from the COVID-19 pandemic. These affected the process of data collection and the involvement of the lead researcher in the Gaza Strip itself. Several checks were in place to strengthen the integrity of data and interpretations, including remote training and supervision, and discussion of the findings with a steering committee, including people with lived experience.It is also worth noting the greater number of female caregivers interviewed, compared to male. Fathers and other male caregivers were more difficult to recruit, as most were busy during working hours. Moreover, female caregivers in the Gaza Strip are most typically the primary caregiver for deaf and hard of hearing children and when we attempted to recruit to male caregivers, many told us that the mother or female caregiver would be able to give better information than they.Deaf and hard of hearing children in the Gaza Strip experience a number of stressors that are detrimental to their mental health and wellbeing, including language deprivation, inaccessible communication and discrimination. Families, civil society actors and education providers must be empowered with the knowledge, skills and resources to provide an inclusive and supportive environment to deaf and hard of hearing children.S1 Appendix(DOCX)Click here for additional data file.S2 Appendix(DOCX)Click here for additional data file.S3 Appendix(DOCX)Click here for additional data file. 6 Mar 2023PGPH-D-22-01952Communication, inclusion and psychological wellbeing among deaf and hard of hearing children: A qualitative study in the Gaza StripPLOS Global Public HealthDear Dr. Scherer,Thank you for submitting your manuscript to PLOS Global Public Health. After careful consideration, we feel that it has merit but does not fully meet PLOS Global Public Health’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.Your manuscript has been assessed by two expert reviewers, whose comments are appended below. The reviewers are generally positive about your study, but have made several requests for additional information on the methodology and discussion which will improve the reproducibility and clarify the implications of your study. Please ensure you respond to each point carefully in your response to reviewers document, and modify your manuscript accordingly.globalpubhealth@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pgph/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.Please submit your revised manuscript by Apr 20 2023 11:59PM. 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Please include a complete copy of PLOS’ questionnaire on inclusivity in global research in your revised manuscript. Our policy for research in this area aims to improve transparency in the reporting of research performed outside of researchers’ own country or community. The policy applies to researchers who have travelled to a different country to conduct research, research with Indigenous populations or their lands, and research on cultural artefacts. The questionnaire can also be requested at the journal’s discretion for any other submissions, even if these conditions are not met.  Please find more information on the policy and a link to download a blank copy of the questionnaire here: Additional Editor Comments (if provided):[Note: HTML markup is below. Please do not edit.]Reviewers' comments:Reviewer's Responses to Questions <font color="black"> Comments to the Authorpublication criteria? Is the manuscript technically sound, and do the data support the conclusions? The manuscript must describe methodologically and ethically rigorous research with conclusions that are appropriately drawn based on the data presented. </font> 1. Does this manuscript meet PLOS Global Public Health’s Reviewer #1: YesReviewer #2: Yes********** <font color="black"> 2. Has the statistical analysis been performed appropriately and rigorously? </font> Reviewer #1: N/AReviewer #2: N/A********** <font color="black"> 3. Have the authors made all data underlying the findings in their manuscript fully available (please refer to the Data Availability Statement at the start of the manuscript PDF file)?PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception. The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified. </font> The Reviewer #1: YesReviewer #2: No********** <font color="black"> 4. Is the manuscript presented in an intelligible fashion and written in standard English? </font> PLOS Global Public Health does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.Reviewer #1: YesReviewer #2: Yes********** <font color="black"> 5. Review Comments to the Author </font> Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. Reviewer #1: This is a good qualitative study on communication, inclusion, and psychological wellbeing among deaf and hard of hearing children. There is a good discussion on what sort of factors contribute to the psychological wellbeing and inclusion of Deaf and hard of hearing (DHH) children. The topic is interesting, and it seems the authors have spent some good time conceptualizing it. In addition, the general overview, and application of the study and its relevance in social contexts is missing.The following can help improve the manuscript.AbstractThe last bit of the abstract can be improved to better state the recommendations and implications of the findings.IntroductionThere is room for improvement in this section.1.Authors first must state the broad theme or topic of the study. After introducing the broad theme/topic, its academic and practical significance (if applicable) must be discussed. You should present a convincing response to the question, "Why should anyone care about this article?" Author has already presented some data regarding this, but it is not enough evidence for justification of the study.2. The author should indicates some gaps, inconsistencies in the literature that the current study has addressed and should explain the study’s main contribution .Materials and MethodsThe method used is appropriate for the type of study presented in the article.1. How the sample size (35) has been determined is not described, author should provide some scholarly reference here.2.It is mentioned that the interviews with the school students, caregiver and teacher were In-depth Interviews, and three Focus Group Discussions were held with DHH adults and disability leaders, mental health specialists.The author should explain explained why different interview techniques were used for different interviews.3.Much more could have been written about the validity and reliability of the study. Results and DiscussionThe results section gives an interesting description of a number of themes that have most likely had an important impact on the studies of the interviewed students.This section is well written.1."Communication, inclusion and psychological wellbeing among deaf and hard of hearing children: A qualitative study in the Gaza Strip" According to the title of your study, psychological wellbeing among deaf and hard of hearing children is a key factor of this study. But I did not find enough writing or analysis about ‘psychological wellbeing’ in the result section.ConclusionI suggest the authors club the conclusion, limitations of the study, and future prospectus and elaborate it under "Conclusions or concluding remarks" as a heading.Reviewer #2: Review of "Communication, inclusion and psychological wellbeing among deaf and hard of hearing children: A qualitative study in the Gaza strip"This is a very important study, giving voice to deaf and hard of hearing (DHH) children, adults and their advocates in the Gaza strip. By analyzing a sufficient sample of interviews and focus groups, and nicely presenting the findings, the authors point at the difficult situation of this population in terms of language deprivation, stigma, and lack of basic services. They indicate needs and suggest solutions, making this study valuable for policy makers and anyone aiming at improving the situation for this population. I would also like to commend the authors for including DHH people from Gaza strip in the steering committee of the research, thereby strengthening its rigor.I have several comments and suggestions to improve the manuscript.1. The introduction is well-written. Under 'Materials and Methods' (p. 4 line 84), please summarize the exact number of participants involved in the study at each category .2. Sentence 104 starting with 'Referral systems' needs further clarification.3. Appendix S1: Please provide the following information about the DHH children: Usage of hearing aids or cochlear implants, the age of fitting/implantation (if known), and communication mode .4. Data collection: Were the interviewers structured or half-structured? Please provide the full topic guides of the interviews and the focus groups, in the text or as an appendix.5. Data analyses: This section is relatively weak. Please provide a detailed explanation of the different stages of TA that you conducted according to Braun and Clarke, who, and how many people were involved at each stage. If more than one person was involved in determining codes/categorizations, at what stage were these analyses compared and discussed, and how were discussions solved?Results and Discussion1. The authors mentioned in the introduction the unique situation of children in conflict zones, such as suffering from multiple stressors and traumas. They also mentioned that the research was carried out during the COVID-19 pandemic. Yet, these topics were not discussed in the study itself. Were there any data stemming from the interviews which could inform us on the experience of coping with hearing loss in these contexts? Were participants asked about conflict/pandemic issues? If no data exists, a reflective notation may help the readers understand why certain topics were not discussed with participants or not raised by them.2. My overall impression of the study is that it advocates for the use of sign language in DHH children, their families, teachers, and community. While this is a very important recommendation, another direction exists as well, which is improving the oral-communication services and devices provided to DHH children and adults. These may include intensive speech therapies for children using hearing aids or cochlear implants (without which they can hardly benefit from them), auditory specialists , systematic newborn hearing screening, providing and informing on accessibility devices such as FM systems and lived transcriptions. These can be emphasized in more details both at the 'assistive technology and communication' section , and at the discussion and recommendations.3. Maybe related to that, the authors mention in the discussion the relatively positive situation of children in special schools, who use sign language, are engaged with DHH peers and seem to be at lower risk for poor mental health. This is of course important to note, yet, it is also important to consider in the discussion the word-wide dispute on schooling segregation and the role that special schools may at times play in segregating children from society and limiting their opportunities. It seems that in the current context at Gaza strip, special schools provide opportunities that lack in mainstream schools (which is well described by the authors), also because they are private. Yet, it is important to bring about a wider perspective on schooling options, based on experiences in other countries. Thus, adapting recommendations that strive at ideally providing DHH with all options, including full integration within mainstream schools (when done well).4. Results line 353: Please mention the (apparently) lack of systematic governmental or organizational training and resources for ambulant teachers, who can instruct mainstream teachers about the needs of DHH children and provide DHH children with personal educational and other assistances (thereby supporting their inclusion in mainstream schools).5. Line 405: When surveying detriment factors, emphasis needs to be given also to structural and material barriers , in addition to cultural and attitudinal ones.6. Discussion line 598: Mental health services for DHH people would benefit also from recruiting signing mental health professionals, DHH professionals, and sign language interpreters specialized in mental health.7. Finally, in your suggestions please provide an informative review of existing interventions designed especially for DHH children . These can include also interventions applied in high-income countries.Editing suggestions:1. Page 1 line 47: Please change to 'moderate to severe or profound (depending on your source)', instead of 'complete' hearing impairment.2. Line 58: The word 'are' is redundant.3. Line 88: 'and caregivers' is redundant.4. Line 124: The word 'to' seems incorrect here.********** <font color="black"> 6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.Do you want your identity to be public for this peer review? If you choose “no”, your identity will remain anonymous but your review may still be made public.Privacy Policy. </font> For information about this choice, including consent withdrawal, please see our Yes: Morshed AlamReviewer #1: Reviewer #2: No**********https://pacev2.apexcovantage.com/. PACE helps ensure that figures meet PLOS requirements. To use PACE, you must first register as a user. Registration is free. 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Please note that Supporting Information files do not need this step.While revising your submission, please upload your figure files to the Preflight Analysis and Conversion Engine (PACE) digital diagnostic tool,  31 Mar 2023AttachmentPLOS Global Public Health_Gaza_Response to Reviewers.docxSubmitted filename: Click here for additional data file. 21 Apr 2023Communication, inclusion and psychological wellbeing among deaf and hard of hearing children: A qualitative study in the Gaza StripPGPH-D-22-01952R1Dear Mr Scherer,We are pleased to inform you that your manuscript 'Communication, inclusion and psychological wellbeing among deaf and hard of hearing children: A qualitative study in the Gaza Strip' has been provisionally accepted for publication in PLOS Global Public Health.Before your manuscript can be formally accepted you will need to complete some formatting changes, which you will receive in a follow up email. 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For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified. </font> The Reviewer #1: YesReviewer #2: No********** <font color="black"> 5. Is the manuscript presented in an intelligible fashion and written in standard English? </font> PLOS Global Public Health does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.Reviewer #1: YesReviewer #2: Yes********** <font color="black"> 6. Review Comments to the Author </font> Please use the space provided to explain your answers to the questions above. 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VH  is the need of the hour. VH of parents of children with hearing assistive devices (HAD) reveals a literature gap, during the habilitation process of their children. To explore the vocal health of parents of children with hearing assistive devices.This cross-sectional study was conducted at Riphah International University from September to December 2021. Study recruited N=384 parents of Hearing Impaired children (HIC) using HAD for at least two years, of both genders and aged 2-9 years using convenience sampling. Voice-related quality of life (V-RQOL), and vocal health Index (VHI) -10 were used for data collection. Data was analyzed on SPSS Version 25. Descriptive statistics, Anova and t-test were utilized to see difference between means of groups. P<0.05 shows significant-results.Parents of children using hearing assistive devices had excellent V-RQOL score in 350(91.14%) parents. There was no significant difference in V=RQOL as regards type of hearing assistive device use (p=0.102), laterality of device use (p=0.918) and degree of hearing loss (p=0.143). However, type of hearing loss revealed significant difference (p=0.021). Also VHI score revealed significantly (p=0.008) lower means in parents of children with cochlear implants.Current study concludes that the parents raising hearing impaired children with hearing assistive devices, possess good vocal health as determined by VHI and V-RQOL scores with only a very small number of parents reporting vocal symptoms. Voice is essential for communication in daily life and it delivers required information including emotions, individuals’ traits and social status. Voice production is the end result of complicated fluid-structure-acoustic interactive sequences depending on structural properties of lungs, voice box and airway. The resultant outcome of this being voice acoustics and perception with imposition of variations due to vocal physiology and physical principles on the intensity, quality, frequency or pitch, prosody and overall modulation of voice. These characteristics ultimately convey meaning3Voice is used to pass on perceptual sensations with humans having an oblivious relationship with ability to interpret depictions of voice quality. However, this is not valid for some instrumental estimations of voice quality, hence self-assessment tools are used detect the different aspect related to vocal functioning which is not possible with instrumental estimations like acoustic assessments.5A study involving teachers of different classes and subjects reveal different level of vocal issues with a higher frequency of vocal abnormalities in primary teachers compared to instructors of optional subjects.9A local study noted that around 1.6 out of 1000 individuals suffer profound hearing impairment involving both ears.The advancement of hearing-related innovations has been working on step by step, for example, cochlear implants (CI) have compressed the impact of deafness by working with children with HI.The stressful conditions make the parents less responsive, more controlling and this ultimately leads to low quality and quantity of verbal communication.st December, 2021. Study recruited N=384 parents of hearing impaired children using convenience sampling from Pakistan Air Force Special Children’s School, Inayat Foundation, Alam School of Speech & Learning, Rangers Institute of Special Children and mainstream section of Hamza Foundation, Lahore. Sample included parents of HI children of both genders aged 2-9 years using hearing assistive devices for at least two years. Parents with poor health, known cases of asthma, nodules or polyps, on job with any kind of vocal stress and parents of children with any comorbid condition or any additional disability like cerebral palsy, seizures, visual problems, Downs syndrome etc., were excluded from the study. Sample size was calculated 384 on the basis of total population of hearing-impaired children 200,000 approximately by using 95% confidence level and 5% margin of error through Rao-soft online sample size calculator. Study was initiated following ethical approval of research from Research Ethics Committee of Riphah College of Rehab & Allied Health Sciences, Riphah International University vide registration No RCR-RE-MS-SLP/Spring 21/004 and informed consent of participants.This cross-sectional study was conducted at Riphah International University over a period of 16 weeks 1st September 2021 to 31Basic demographic sheet, voice-related quality of life (V-RQOL),100 -  x 100(Highest Possible Raw Score - # items)SPSS Version 25 was utilized for data analysis. Descriptive statistics were used with frequency and percentage calculated for categorical variables and chi-square used to see any difference between groups of child hearing characteristics, while Anova and t-test were utilized to see difference between means of groups. P<0.05 was considered significant.Study sample comprised mainly 344(89.6%) mothers of HI children using hearing assistive devices with most 166(43.2%) aged 30-39 years . CurrentStudy found marked difference in the VHI mean score when compared with V-RQOL score categories with lowest score (1.84±1.349) noted in the category of lowest V-RQOL i.e., Excellent (10-15) and difference was significant (p=0.000) when compared with other categories. Almost similar results were noted in the subscale of Functional, Emotional and Physical subscales (p<0.000) with lowest scores in the Excellent category of V-RQOL, .VHI-10 total scale and subscale mean scores revealed lower means in parents of children using cochlear implant compared to hearing aids and the difference was significant for total mean score (p=0.008), functional subscale (p=0.011) & physical subscale (p=0.016), however it was not significant for emotional subscale (p=0.104) . InteresStudy measured subjective outcomes of voice related quality of life by using V-RQOL measure and VHI, using affected parents’ perspectives regarding various aspects of their vocal loading, voice fatigue, vocal abuse or misuse of voice related quality of life. With a sample which comprised predominantly 344(89.6%) mothers & 40(10.4%) fathers of HI children using hearing assistive devices with majority 166(43.2%) aged 30-39 years, study revealed that parents of HI children using hearing assistive devices had excellent V-RQOL score in majority 350(91.14%) parents and only 6(1.6%) had poor V-RQOL score. Also, V-RQOL did not reveal significant association with degree of HL (p=0.143) and laterality of hearing assistive device use p=0.918), though there was significant association (p=0.021) with type of HL with mixed HL having more cases with poor V-RQOL. This is in compliance with literature where in a local study by Ishtiaq N et al revealed low level of stress in parents of HI children compared to parents of children with autism., though 19In present study type of hearing assistive device use did not reveal any significant difference (p=0.102) with V-RQOL, though there was no parent in cochlear implanted category having poor score. This is in conformity with a study by Wischmann S et al. in 2022, aimed to investigate long-term development of basic language in new generation of children with hearing-impairment (HI) using hearing aids (HA) for hearing loss of 30dB to 80dB and bilateral CI for hearing loss more than 80 dB, revealed normal social wellbeing and language skills, which favors the fact that parental vocal health and quality of life is not affected.22Morningstar M et al. in their study reported that Interaction between infant and parent can alter parental prosody, which is good for infants’ language development.25Current study found marked difference in the VHI mean score when compared with V-RQOL score categories with lowest score (1.84±1.349) noted in the category of lowest V-RQOL ie. Excellent (10-15) and difference was significant (p=0.000) when compared with other categories. Almost similar results were noted in the subscale of Functional, Emotional and Physical subscales (p<0.000) with lowest scores in the Excellent category of V-RQOL. A moderate to strong association between VHI and its domains with V-RQOL have been reported by Lu D et al. in a study involving teachers.26Voice related Quality of life (V-RQOL) was administered in English language which is not the mother tongue of the sample population which could induce biasness in scores.Current study concludes that the parents raising hearing impaired children with hearing assistive devices, possess good vocal health as determined by VHI and V-RQOL scores with only a very small number of parents reporting vocal symptoms.UQ: Did the data collection, analysis and interpretation and responsible for integrity of the research.NM: Did the supervision and critical revision of the articleGS: Did the conception of work and writing of the manuscript.


Cochlear implantation (CI) allows rehabilitation for patients with severe to profound hearing impairment. Although the use of a robotic assistant provides technical assistance to the surgeon, the assessment of the impact of its use on auditory outcomes remains uncertain. We aim to compare the hearing results of patients who underwent bilateral cochlear implantation; one side was performed with manual insertion and the other side with robot-assisted insertion. The electrode array intrascalar positioning and the surgery duration were also studied. This retrospective intra-individual study involved 10 patients who underwent bilateral cochlear implantation. The study included two infants and eight adults. The unique composition of this cohort enabled us to utilize each patient as their own control. Regarding speech disyllabic recognition, pure tone average, ECAP, ratio of array translocation, basilar membrane rupture, and percentage of translocated electrodes, there was no difference between manual and robot-assisted CI groups. This study is the first to compare intra-individual hearing performance after cochlear implantation, either manually or robot-assisted. The number of patients and the time delay between manual and robotic implantation may have led to a lack of power, but there was no apparent difference in hearing performance between manual and robotic implantation. Cochlear implantation allows rehabilitation for patients with severe to profound hearing impairments. It can be used for both adults and children with syndromic and non-syndromic deafness . It may ® , which provides autonomous, minimally invasive access to the inner ear by drilling a path through the temporal bone . The study conducted by Baron et al. on pediatric populations compared two groups according to the inter-CI interval: < 36 months for the “early group” versus ≥ 36 months for the “late group” . Using tThe length of hearing loss before the second cochlear implant was a factor negatively correlated to speech understanding . RegardiTo note, Zeitler et al. reported that speech perception continued to improve, whatever the inter-CI interval . This intra-individual study overcomes the variability caused by the etiology of the hearing loss and the type of implant used. However, it has some limitations. This study cannot exclude variability due to the duration of profound deafness; only one patient underwent synchronous implantation, which may have biased the results in favor of robotic implantation in the others. A large multi-center, prospective, single-blind, randomized trial will be needed to ensure that there is no effect of robot-assisted insertion on hearing outcomes. A prospective intra-individual study with simultaneous implantation  would be another option.This study is the first to compare intra-individual hearing performance after cochlear implantation, either manually or robot-assisted. The number of patients and the time delay between manual and robotic implantation may have led to a lack of power, but there was no apparent difference in hearing performance between manual and robotic implantation. There was no significant difference between the two methods in terms of operative time or the incidence of array translocation or basilar membrane rupture. No complications were observed. This confirms the overall safety and reliability of both manual and robotic approaches to cochlear implantation.


Sensorineural hearing loss (SNHL), caused by pathology in the cochlea, is the most common type of hearing loss in humans. It is generally irreversible with very few effective pharmacological treatments available to prevent the degenerative changes or minimise the impact. Part of this has been attributed to difficulty of translating “proof-of-concept” for novel treatments established in small animal models to human therapies. There is an increasing interest in the use of sheep as a large animal model. In this article, we review the small and large animal models used in pre-clinical hearing research such as mice, rats, chinchilla, guinea pig, rabbit, cat, monkey, dog, pig, and sheep to humans, and compare the physiology, inner ear anatomy, and some of their use as model systems for SNHL, including cochlear implantation surgeries. Sheep have similar cochlear anatomy, auditory threshold, neonatal auditory system development, adult and infant body size, and number of birth as humans. Based on these comparisons, we suggest that sheep are well-suited as a potential translational animal model that bridges the gap between rodent model research to the clinical use in humans. This is especially in areas looking at changes across the life-course or in specific areas of experimental investigation such as cochlear implantation and other surgical procedures, biomedical device development and age-related sensorineural hearing loss research. Combined use of small animals for research that require higher throughput and genetic modification and large animals for medical translation could greatly accelerate the overall translation of basic research in the field of auditory neuroscience from bench to clinic. Hearing loss affects approximately 1.5 billion people globally and imposes a significant burden on individuals and society, affecting communication, quality of life, and productivity . Risk faMacaca fuscata) compared to small laboratory animals. Monkeys  and sheep  have smaller litter size compared to rodents [The life span of laboratory animals is relatively short compared to humans Table . The lif rodents , 5. Smal rodents  make rodAhl2, mt-Tr, and Cdh23ahl have been identified in some specific strains of mice [There are various inbred and outbred strains available in rodents, and most of their genome are comprehensively researched. Inbred strains of rodents can provide better stability of genotype and minimal individual difference. Some of the hearing loss related gene locus variation, such as  of mice . The gen of mice . However of mice . The com of mice , enablin of mice . We expeThe adult body weight is an important factor when designing a suitable animal experiment with appropriate housing and space requirements, adjusting the amount of therapeutic compounds for systemic administration, and volume of tissues or blood that can be sampled. Human average adult body weight is approximately 62 kg  in the sMacaca fuscata, 28 Hz–37 kHz), and chinchilla (50 Hz–33 kHz) have a similar hearing range to humans [Macaca fuscata, 1 dB SPL at 4 kHz), dog (0 dB SPL at 2–8 kHz), rabbit (4 dB SPL at 2 kHz), and chinchilla (8 dB SPL at 500 Hz–4 kHz) have their greatest sensitivity in a similar frequency range to humans [Hearing in animals can be assessed using a variety of behavioural tasks or physiological measures for comparison with human performance. The estimation of auditory thresholds using the Auditory Brain Response (ABR) to tone bursts across frequency and Otoacoustic Emissions (OAE) are the most commonly used physiological assessment and which can be compared to human pure tone audiometry, or similar physiological measures. Here the hearing frequency range and sensitivity developed by pure tone audiometry in humans and ABR or behavioural testing in animals is compared Fig. , Table 2o humans , 44, 45.o humans –48. The o humans . Monkey o humans , 50. Otho humans , 48, 51 Frequency representation in the cochlea is organised spatially by the tonotopic map along the length of the cochlea such that higher frequencies are detected at the basal end, while more lower frequency sounds are detected towards the apical tip of the cochlea . In humaThe difference in hearing range and tonotopic representation within the cochlea may differentially influence animal’s response to the same environmental noise and development of noise-induced hearing loss models. In human occupational noise-induced hearing loss, the main auditory threshold shift is at 3–6 kHz  with progression to predominately higher frequencies followed by lower frequencies , 60. HowIn other types of SNHL independent of noise, the difference in the frequency range may matter less. For example, in humans, cisplatin-induced SNHL affects the hearing threshold at in high frequencies (> 4 kHz) the most with hair cell loss observed in high frequency region of the cochlea , 66. In When using animal models for human medical research, it is important to correlate approximate equivalent age and stage of development between species . SeveralAhl variation which is responsible for the early-onset ARHL occurring at 3 month after birth [Age-related hearing loss (ARHL) or presbycusis is observed in humans, small rodents, and non-human primates. In humans, the prevalence of ARHL increases non-linearly with age and exceeds 30% within the 60–69 year old populations . In rheser birth , 92. In er birth . The ARHer birth , 94, theCommon marmoset), cat, rabbit, guinea pig, chinchilla, rats, and mice (5–24 mm) are shorter compared to human (32 mm) [Common marmoset, 5.22 µl), mice (0.32 µl), and rats (1.04 µl) are substantially smaller compared to humans (29.22 µl) [Here we will compare the main laboratory animal species and humans, focusing on the geometry and some of the anatomical features of the cochlea. The cochlea of humans is approximately 6 mm in diameter and is embedded in the temporal bone. It is a fluid-filled structure and consists of 3 fluid filled compartments . The cochlea of mammals all shares the same basic spiral structure, however the length of the cochlea and the number of turns differs considerably. The human cochlea spirals 2.6 turns around the modiolus , whereas (32 mm) , 98–103.9.22 µl) , 104. In9.22 µl)   and outer hair cells (OHC) in the organ of Corti (OoC). The afferent innervation by the spiral ganglion neurons (SGNs) transmit sound from the hair cell to the brain . In SNHLSpiral ganglion neurons are the primary afferent neurons and the SGN cell bodies are located in the modiolus, specifically Rosenthal’s canal, of the cochlea. Humans have a higher SGN count than rats and mice and a lower SGN density than dog, guinea pig, chinchilla, rats and mice Table . The numRhesus macaque, 40–60 µm) have similar RWM thickness to humans [2) is larger than chinchilla (1.06 mm2) and guinea pig (1.18 mm2) and similar to sheep (approximately 3.79 mm2) and cats (approximately 1.77–3.14 mm2) [The round window (RW) is one of two membrane-bound openings on the bone surrounding the cochlea, separating the middle and inner ears. The RW is covered by an epithelial membrane, round window membrane (RWM), and is commonly used as access to the cochlea from the middle ear in cochlear implantation surgery , inner eo humans , 139. Al.14 mm2) –143  survival, as well as understanding post-CI pathophysiological responses in the cochlea such as inflammation and fibrosis, which is thought to negatively impact the CI outcomes. In this regard, reproducible surgical approaches for CI implantation have been well established in small  animal models , 154. FoThe size and geometry of the cochlea is critical for fitting human CI. If the study needs to take into account of the feasibility of CI surgery procedure, then the overall anatomy of the skull and temporal bone becomes a relevant factor to be considered for selecting the right large animal model. While the cochlear length in pig is long enough for a full length human CI, the temporal bone anatomy of pig head is not ideal for CI surgery . The lenWe have summarised data available to allow comparison of laboratory animal models for purpose of identifying advantages and disadvantages that large animal models may offer over the conventional small animal models used in auditory research. These advantages and disadvantages are summarized below.tau neurofibrillary accumulations are the hallmark signatures of Alzheimer’s disease (AD) [tau tangles in sheep is mostly in the temporal lobe and entorhinal cortex [tau pattern in humans [The first potential advantage is the longevity of larger animals used in research. An example of the importance of this advantage comes from models of human neurodegenerative disease. Age-dependent amyloid β deposits and ase (AD) . This siase (AD) . Therefoase (AD) . In contase (AD) –179. Thel cortex , which in humans –183.Aging is a major contributor to hearing loss in humans , and ARHIn addition, the longer gestation period of large animals may also be an advantage in studying congenital/early onset hearing loss. Some prenatal risk factors such as asphyxia, maternal infection, ototoxic drug exposure, and low for gestational birth weight –194 causSecond advantage that large animals have to offer is the size and the anatomical similarities of the peripheral auditory system between large animals and human as we have summarised in this review. Our comparison shows that in terms of the cochlear size anatomical features (such as RWM), and fluid volume inside the cochlea, large animals are more similar to human than small animals. These features make large animals more translatable models for development of devices or procedures for the inner ear. As mentioned, sheep animal model has an advantage as commercial and full-sized human CIs can be used without adjustment . More reahl/+ ahl) that confers resistance to hearing loss compared to CBA/CaJ strain mice, and have no detectable hearing loss even after their 24 months of ages [On the flip side to the advantage of the longevity, the longer gestation period and the smaller litter size can be the major disadvantage of large animals such as sheep and monkey, if research requires development of transgenic animal models to recreate certain SNHL pathologies and higher throughput research. Rodents have a shorter gestational period (22–23 days) and larger litter size (7–12) . Genetic of ages , 214. WhFinally, one key aspect for consideration is the ethics and cost associated with the maintenance of large or small animals for research purposes, including housing needs and resources required to maintain these animals. In common with use of any animals for research, full justification for their use is required and every available effort made to minimise numbers used and any suffering must be consistent with internationally accepted conventions stated in the ARRIVE guidelines . With reIn our experience in NZ, because we have a major primary industry centred around farming of sheep, we can acquire experimental subjects from large commercial populations on a regular basis. The cost of sheep for research of course depends on the local market at the time, currently ranging from NZ$250 to NZ$350 per ewe. This is compared to the cost of standard  rodents which range from NZ$60 to NZ$200 depending on the age. Additional costs for sheep work is the requirement to have a team of investigators capable of handling the animals, larger facilities required for surgeries and manipulation of the sheep, and larger volumes of reagents and medications. When live sheep work is undertaken in an urban laboratory setting the cost of can be around, 5–10 times higher (~ $3000) than the cost of performing equivalent experiment on rats (~ $300). However, our research farm laboratory facility and pasture are co-located which negates the problems of transporting animals long distances and also offers superior animal welfare standards and lower costs. In our experience, the use of large animals requires collaborative work with researchers with strong veterinary and animal husbandry backgrounds. Finally, every effort should made to share the use of valuable animals with other researchers who can use other tissues and samples. We hope this review will be helpful to those considering utility of large animal models and will support collaborative use of large animal models in the auditory research field.In this article, we compared some of the key features of the auditory system between different animal models. The shorter reproductive cycle and larger litter size makes small animals, such as mice, have an advantage over large animals for establishing the genetically modified hearing loss models. Large animals such as sheep have similar hearing range, adult and infant body size, and cochlear and round window membrane anatomy to humans and longer life span compared to other small animals. With similar cochlear geometry, perilymph volume (or ST volume), RWM thickness and diameter, and cochlear length to humans, sheep may be an ideal animal model for preclinical testing of new therapies, including diagnostic, drug delivery and prosthetic devices such as CI and other emerging technologies. Combined use of small animals for research that require higher throughput and genetic modification and large animals for medical translation will greatly accelerate the overall translation of basic research in the field of auditory neuroscience from bench to clinic.


OTOF, which encodes the calcium-binding protein otoferlin, are responsible for 1–8% of congenital, nonsyndromic hearing loss and are the leading cause of auditory neuropathy spectrum disorders. The natural history of otoferlin-related hearing loss, the relationship between OTOF genotype and hearing loss phenotype, and the outcomes of clinical practices in patients with this genetic disorder are incompletely understood because most analyses have reported on small numbers of cases with homogeneous OTOF genotypes. Here, we present the first systematic, quantitative literature review of otoferlin-related hearing loss, which analyzes patient-specific data from 422 individuals across 61 publications. While most patients display a typical phenotype of severe-to-profound hearing loss with prelingual onset, 10–15% of patients display atypical phenotypes, including mild-to-moderate, progressive, and temperature-sensitive hearing loss. Patients’ phenotypic presentations appear to depend on their specific genotypes. For example, non-truncating variants located in and immediately downstream of the C2E calcium-binding domain are more likely to produce atypical phenotypes. Additionally, the prevalence of certain sequence variants and their associated phenotypes varies between populations due to evolutionary founder effects. Our analyses also suggest otoacoustic emissions are less common in older patients and those with two truncating OTOF variants. Critically, our review has implications for the application and limitations of clinical practices, including newborn hearing screenings, hearing aid trials, cochlear implants, and upcoming gene therapy clinical trials. We conclude by discussing the limitations of available research and recommendations for future studies on this genetic cause of hearing loss.Congenital hearing loss affects one in 500 newborns. Sequence variations in  OTOF gene, which encodes the membrane-associated protein otoferlin, are responsible for 1–8% of congenital, nonsyndromic hearing loss cases and are the leading cause of auditory neuropathy spectrum disorders  with congenital or prelingual onset  based only on OAE detection  on February 14, 2022. An abstract scan requiring human data yielded 109 unique publications. After the original query date, two additional papers  calculated the air-conduction, pure-tone average hearing thresholds determined by behavioral or electrophysiological audiometry at 0.5, 1, 2, and 4 kHz between ears, or (2) used the average hearing threshold reported by the authors. When a range of hearing thresholds was reported, we used the mean of that range. If a range was unbounded at the upper end , we used the lower limit . We then assigned a hearing phenotype to each individual using a simplified version of the classifications established by Clark : 0–25 dB2 domain, C2de, according to Pangršič et al.  unless specifically described as non-truncating (n = 1). Single-residue, in-frame deletions and missense variants were classified as non-truncating (NT). In cases of multiple homozygous variants, the most severe variant was used to classify subjects. We defined the boundaries of functional regions within otoferlin according to Vona et al.  and, forč et al. . One NT Distortion product otoacoustic emissions (DPOAEs) and transient evoked otoacoustic emissions (TEOAEs) were all categorized as “OAEs.” OAE results were classified as “present,” “absent,” “reduced,” or, in cases where OAEs were lost over time, “present, then absent.” “Present” was used when (1) qualitatively reported by the authors or, if raw data were provided, (2) OAEs were present  at 50% or more of the frequencies tested in both ears. “Reduced” was used when (1) both ears had present OAEs in less than 50% of frequencies or (2) only one ear had present OAEs in at least 50% of frequencies. “Absent” was used when (1) described qualitatively by the authors or (2) if raw data indicated absent  OAE responses at all tested frequencies. The results of OAE-based NBHS were only included in NBHS analyses, not in other OAE analyses.We compared the results of NBHS that utilized ABRs, OAEs, and unspecified test methodology. Hearing aid outcomes were reported, predominantly in subjective terms, for 32 patients from 12 publications, which we summarized in Table Age data were used only if a specific age was indicated for the case. Age ranges were not used unless the entire range fit within a single bin. If hearing thresholds were measured at multiple ages, we used the youngest age. If OAEs were measured and present at multiple ages, we used the oldest age.OTOF variants  were homozygotes and 160 (40%) were compound heterozygotes. One hundred and sixty-five cases (41%) had two NT variants  or the regions flanking the final C2 domain, C2F . Twenty percent (n = 83) of all NT alleles (n = 410) were associated with atypical phenotypes, but in the C2E domain and the region immediately downstream (C2E-C2F), 57% (n = 35 of 61) of NT alleles were associated with atypical phenotypes. Figure We next investigated whether certain NT variants, or NT variants in certain regions of the gene, might be associated with particular phenotypes Fig. . A schemAs common allelic variants are not evenly distributed across different populations and geographic regions, we tabulated the number and locations of NT and T alleles reported in at least five individuals and two families from our dataset Table .Table 2GOf the 231 patients with OAE data, OAEs were present in 166 72%) and absent in 45  were failed and three 8.6%) were passed  and 24 (5.9%) NT alleles, respectively, in our dataset, are debated , 27 (6.6%), and 74 (18%) NT alleles, respectively, in our dataset, bind calcium and are functionally important  alleles in our dataset, lending support for its hypothesized functionality , and the proportion of those alleles associated with typical severe-to-profound phenotypes , suggest that conservation of this region is critical for otoferlin functionality  and 24 .9% NT al) and 24 .9% NT al) and 24 .9% NT alIn addition to the ten alleles associated with temperature-sensitive phenotypes described in the results section, one additional variant, p.I515T, was identified in two siblings with temperature-sensitive DFNB9 phenotypes  than in cases with atypical phenotypes , and none of the mild (n = 10) or temperature-sensitive (n = 13) cases had absent OAEs Fig. C. Additi2E domain might have a stable phenotype, whereas mild SNHL in a child with NT variants between C2E and C2F may be more likely to progress  of the variant and the region of the protein it affects. For instance, a child born with mild SNHL and NT variants in the Cess Fig. B.OTOF variants can disrupt temporal dynamics of vesicular exocytosis necessary for synchronous postsynaptic neural activity (measured by ABR) while still allowing enough dysregulated exocytosis for sound detection  cases with absent ABR at maximum stimulation displayed atypical phenotypes as measured by audiometric hearing thresholds, including seven (4.5%) with mild, eight 5.2%) with moderate, and eight (5.2%) with temperature-sensitive hearing loss , which may differ in clinical practices and OTOF allele frequencies. Consequently, foremost among the potential confounds of our analyses is the disproportionate influence of a small number of papers with large sample sizes derived from populations that may not be representative of the global population. For example, 55 of the 422 cases in our dataset came from 13 consanguineous Pakistani families described in Choi et al. , phenotypic bins , diagnostic criteria , inclusion/exclusion criteria, and genetic sequencing methodologies. For instance, some cases were described as having hearing thresholds “greater than” a certain value ; as we used the lower limit  to assign a phenotype to these cases, our analysis may overestimate the number of severe cases and underestimate the number of profound cases. Methodological variability is inevitable, but detailed reporting of methodological techniques and case-specific data (rather than group data only) ensures transparency, facilitates accurate interpretation, and enables subsequent quantitative reviews and meta-analyses.OTOF sequence variants affect the vestibular system. Otoferlin is expressed in vestibular hair cells and there are limited reports of patients with both OTOF variants and vestibular dysfunction, but no causal relationship has been established  reported to online databases is valuable. Through the collective effort of researchers and clinicians around the world, OTOF-related hearing loss will continue to be better understood and more effectively treated in the future.Moving forward, one open question is how, if at all, ed Varga . It is a