Abstract
Analyze contributing factors toward satisfaction after cleft lip and palate treatments in Senyum Bali Foundation. Qualitative case study, an interview with a semi-structured question from February to April 2023 by purposive sampling informants including parents, foundation staff, and plastic surgeon. Unsatisfying nose and teeth appearance due imperfect effects after surgery and requirement of more procedures. Mini model theory study that includes the appearance of the face and lips; speech and hearing; the functions of feeding, masticatory, breathing; psychosocial, quality of life, treatment cost-effectiveness, funding, health services, physician, implementing cultural contexts, surgical outcomes, and parents’ feedback.
Background
Cleft lip and palate is the second most common congenital disorder and the most commonly detected craniofacial malformation, with a prevalence that varies from 1 to 2.2 per 1000 births worldwide. This condition is closely related to the failure of the nose and maxillofacial development during embryogenesis in the sixth to eighth week of embryonic life. A few factors are involved in the etiology of this malformation: genetics and teratogenic exposure during the second to the third month of the pregnancy, with genetics being the more common etiology. In Indonesia alone, reports from the Indonesian Basic Health Research from 2013–2018 have shown that there has been an increase in the incidence of cleft lip (from 0.08% to 0.12%) in kids aged 24 to 59 months caused by genetic or environmental factors, such as medications consumed during pregnancy (amoxicillin, phenytoin, and oxprenolol), nutritional deficiencies, hypoxia, radiation, viral infection, teratogenic exposure, smoking, and an excess or deficiency of specific vitamins. 1
Cases of cleft lip and palate in developed countries are often identified before birth with ultrasonography. This allows medical professionals to educate the parents about the condition and the available procedures, which lead to an immediate response after the child's birth. 2 However, in developing countries, children with cleft lip and palate receive different treatments as some cases often receive delayed treatment, and those left untreated often suffer from malnutrition, dental problems, ear infections, speech problems, and extreme social stigma that can lead to child abandonment. Children with cleft lip and palate often need long-term treatment to counter the effects of years-long disruption in physiological processes and an unfavorable facial appearance that can equally affect their mental and social functioning. 1 The psychological impacts of this condition are often not limited to the patient. Still, they are also felt by the parents, over their child's condition and the stigma surrounding it. This will likely lead to a reluctance to socialize among the parents. As the only way to treat cleft lip and palate, a surgical procedure is also a long and arduous process that can affect the parents psychologically, socially, and economically.3,4
The Senyum Bali (Bali Smile) Foundation is an independent non-profit foundation that provides free medical treatments for cleft lip and is partnered with many hospitals in Bali. This foundation also works with a multidisciplinary team, such as speech therapists and dentists, to provide comprehensive treatment. Since its establishment 18 years ago, The Senyum Bali Foundation has never researched parental satisfaction whose children have benefitted from their services. 5
Hopefully, the child will experience improvements functionally, aesthetically, psychologically, and socially after the surgical procedure, which will affect the parents’ satisfaction. Research about parents’ satisfaction can illuminate the quality and cost-effectiveness of the surgical procedures and services provided thus far and what can be improved. Therefore, this research aims to assess the factors that affect the parental satisfaction of children suffering from cleft lip and palate in the Senyum Bali Foundation and whether the services provided are cost-effective.
Ethics Statement and Consent Statement
I certify that there is no actual or potential conflict of interest in relation to this article. This research has a Description of Ethical Approval from Ethical Committee of Health Research Wangaya Hospital Denpasar, Bali No. 070/5598/RSUDW. Inform consent is included in the title page from all the patient included in this research.
Methods and Materials
This study is a qualitative case study conducted from February to April 2023 at the Senyum Bali Foundation with a total of 10 subjects that were obtained using the purposive sampling method. The inclusion criteria for the subjects are (1) parents whose children suffered from cleft lip and/or palate and have undergone >1 surgical procedure facilitated by the Senyum Bali Foundation, (2) parents whose children suffered from cleft lip and/or palate and have undergone a surgical procedure at age >1 to 10 years old facilitated by the Senyum Bali Foundation, (3) parents whose children suffered from cleft lip and/or palate and have undergone a surgical procedure in the Denpasar area near the Senyum Bali Foundation, (4) parents whose children suffered from cleft lip and/or palate and have undergone a surgical procedure facilitated by the Senyum Bali Foundation and are willing to participate in the study, (5) Plastic surgeons who performed a surgical procedure for cleft lip and/or palate for the Senyum Bali Foundation, and (6) Staff of the Senyum Bali Foundation who are directly involved in providing services for patients with cleft lip and/or palate and are willing to participate in this study. The exclusion criteria include (1) parents of children with cleft lip and palate who have not received treatment, (2) staff of the Senyum Bali Foundation that was not involved directly in providing services to children with cleft lip and palate, and (3) plastic surgeons who did not treat cleft lip and palate in the Senyum Bali Foundation. These subjects include five parents of a child with cleft lip and palate that has undergone surgical treatment, one plastic surgeon, and four staff who work in The Senyum Bali Foundation that, was selected after an initial survey. A face-to-face in-depth interview with a semi-structured question was conducted to obtain information from all the chosen subjects. Additional official documents from the foundation, personal papers, and photos were collected when necessary. All the received information from the interview and observations were then extracted and sent back to the subjects to confirm whether the data transcription was accurate. Theme identification (coding) and categorization were then made to the data transcription, followed by data interpretation. Data remained in Bahasa Indonesia during the analysis process, and illustrative quotes were eventually translated into English.
Results and Discussions
The Appearance of the Face
The patients report facial appearance due to cleft lip/palate reconstruction.
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Appearance on the human face represents the initial description and identification of a person, and disturbances in facial structure have a high impact not only on the anatomy, physiology, and function of the facial region but also on the acceptance of the individuals in society.
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The facial appearance of sufferers of cleft lip and palate who have undergone repair surgery will give a better facial appearance that is neater and consequently allow the parents to feel satisfied, happy, and grateful because the patients also have their confidence improved. “I feel thrilled that after seeing the development of the second surgical procedure, my child is now happier and more confident, especially with his peers. So yes, this foundation helped to create a new smile for my child. I am truly grateful to this foundation for helping, meaning I could see the real smile from my child.” (MS, Parent of patient 5)
The Appearance of the Lip
The lip appearance on cleft lip reconstruction to achieve impeccable symmetry has become one of the most important goals of the plastic surgeon.
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The appearance of the lips of patients with cleft lip and palate who have undergone a surgical repair procedure will close the last gap so that the appearance of the patient's lips after a good repair will provide satisfaction for the sufferer's parents. “(I am) content, (the lip is) now able to close. Before, there was a gap.” (NM, Parent of patient 1)
The Appearance of the Nose
As well as the appearance of the lips, the nose appearance in cleft lip and palate repair to achieve symmetry is also one of the most important goals for surgeons in carrying out repair procedures.
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A study in Amsterdam said that patients who were very satisfied with the nose appearance were also delighted with the overall appearance of their faces.
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However, the appearance of the nose of people with cleft lip and palate who have undergone surgical repair procedures does not all give ideal results. The variation of each influences this condition, and the appearance of an asymmetrical nose gives a poor impression to the parents of sufferers. Several further surgical procedures are needed to improve the appearance of the patient's nose to make it look more proportionate. “Yes, often noses require further revision because children are still growing and developing, so the nose will undergo final repair after their faces have stopped growing. After seventeen years old (the nose) can be repaired again, and another change will be made. The change keeps happening as long as they grow, although the surgery was symmetrical already.” (SA, Plastic surgeon)
The Appearance of the Teeth
The teeth appearance in patients with cleft lip and palate is also often affected, characterized by a lack of alignment between the upper and lower incisors caused by abnormalities in the failure of the oral cavity to form. In patients with cleft lip and palate, dental problems can refer to several things, such as changes in the number of teeth, shape, and time of eruption.
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The poor appearance of the teeth of patients with cleft lip and palate after surgical repair procedures has dissatisfied the parents. Hence they hope for further treatment to improve the appearance of the patient's teeth. The patient's parents judge the poor teeth appearance to give a messy appearance. To enhance the appearance of the teeth, further actions are needed, such as jaw graft surgery and dental treatments to provide better results, such as orthodontic and prosthodontic treatment procedures performed by dentists. “For me, how the teeth look is a little bit lacking. I often find my child looking into the mirror asking, ‘Mom, why don’t I have any teeth?’” (NM, Parent of patient 1)
“Yes, that also requires more time because, in cases of cleft lip, the teeth are messed up, the growth is irregular, it also needs process, it doesn't grow in an orderly manner, and needs to be treated by a dentist, later it can be arranged. It requires orthodontic treatment. Well, probably, children in the foundation might certainly receive dental care. I am not sure if the foundation supports the care, but if it is not, then the teeth will not be well maintained because mostly, the parents who could not afford (the treatment) were the ones who sought the foundation.” (SA, plastic surgeon).
Speech Ability
The ability to speak is a form of expressing one's thoughts and feelings by articulating them.
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One out of 10 children with cleft lip and palate experienced trouble with speech.
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The procedure done on these children has a good outcome and has improved the child's speaking abilities. Thus, the parents felt delighted with the results. “Yes, the voice is good, normal like other children. There is no leak from the voice.” (NS, Parent of patient 4)
Hearing Ability
Children born with cleft palates often face problems in their middle ear, resulting in hearing ability. This becomes problematic since the palate muscles hold an essential role in the air entering the middle ear, which is involved in the hearing process and allows fluid drainage from the middle ear. However, after the procedure, there were no complaints about hearing abilities. “There are no disturbances, Doc, now that the canals from the ears and nose have been closed. It's getting better. In the past, if (the ears) were not cleaned, it became filthy.” (NS, Parent of patient 4)
Feeding Function
Infants with cleft lip and palate have difficulties while feeding. It takes the coordination of muscles in the mouth. This can be minimalized by holding the infant at a 45-degree angle, which could minimize the amount of liquid coming out of the infant's nose.
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However, this problem was said to be no longer present after the procedure. “During the first surgery, because the palate wasn't closed yet, the food came out of the nose, making it uncomfortable. After the palate surgery, the second operation (the palate) was already closed, so there were no complaints. With the second operation, the condition is more perfect than before.” (MS, Parent of patient 5)
Masticatory Function
Patients with cleft lip and palates were often found with abnormalities in their teeth shape caused by orofacial malformation, which affect the masticating ability of the patients. Those patients show poor masticatory function due to the malocclusion of their teeth’ vertical, anteroposterior, and transversal correlation.
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“None, the chewing is normal, (my child) could eat meat normally.” (NS, Parent of patient 4)
Breathing Function
Infants with cleft palate tend to experience nasal regurgitation while suckling since the palate abnormalities allow the breast milk or the food to come out from both the mouth and the nose, resulting in breathing disturbance. This might lead to choking and aspiration, where the milk and food would enter the airway through the nasal cavity.
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“During the first surgery, because the palate wasn't closed yet, the food came out of the nose, making it uncomfortable. After the palate surgery, the second operation (the palate) was already closed, so there were no complaints. With the second operation, the condition is more perfect than before.” (MS, Parent of patient 5)
Social Function
Social function can be defined by an individual's interaction with their surroundings and ability to fulfill their roles in their environment, such as working, participating in social activities, and maintaining relationships with others.
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Children with cleft lip and palate are believed to have a high risk of various disruptions in social functioning, such as behavioral problems, low self-esteem, and difficulties building a parent-child relationship.
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However, after the procedure, the children have newfound confidence, allowing them to interact with other children freely.
Yes, he (the child) has become more confident and easy to get along with. He invites me immediately to play with anyone he has just met. He is mentally strong and usually plays with his other kids his age even though he doesn't know them. (NS, Parent of patient 4)
Psychosocial Support
Patients who receive validation from people around them and their environment feel that they are as valuable as other people. Therefore, psychosocial support significantly impacts the patients to feel accepted in their environment and avoid the bad stigma that may arise in society.
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“Yes, it's comforting. As a parent, with the birth of a child like this, I didn’t immediately accept it, so I was told to be patient, to take good care of the child, and not to treat him any differently than the other kids. Generally, I have to be supportive.” (RY, Parents of Patient 3).
“Yes, of course, mental support, explaining and reassuring the parents, we will take action, we will try to do our best, we also explain the risks and to calm them, of course, we will also give them information about who the anesthesiologist is…. Like, okay, we have done this often, but some complications will occur, something we often go through, like how every path has its risks, but if we have somewhere to go, we have to go through it anyways. Yes, of course, they are happier after we explained because, before that, they had concerns, right? But by explaining the process, it will often calm them down.” (SA, Plastic Surgeon)
Perceived Quality of Life
Having a cleft lip and palate child can act as mental baggage for parents, causing sadness, anxiety, shame, guilt, low self-esteem, and even hatred that will affect the family dynamic. Parents will have to overcome their heightened emotional state and restructure their life to accommodate the needs of their children who need special care. However, after the operation, the parents were happy with how their children are now as normal as any other children, and they expressed their joy and satisfaction.
In the past, at first, I was afraid of how to give milk/water because he (the child) was vomiting, and I was emotional. But after we went through with it, it is better now. (NM, Parent of patient 1)
Treatment Factors
Managing cleft lip and palate requires a multidisciplinary team collaborating and committing professionals to care for the patients long-term.
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“Yes, (we) had another examination at the hospital after the surgery.” (RY, Parent of patient 3)
Cost-Effectiveness
Managing cleft lip and palate cases in low- and middle-income countries is still relatively neglected. Children suffering from the condition who do not receive any treatment are often experiencing malnutrition, poor teeth appearance and non-functioning teeth, ear infections, speech delay, and social stigma, which results in neglect and homicide to the babies.
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This economic evaluation helps clinicians and stakeholders make decisions when resources are scarce. Although the surgery requires extensive cost, especially for low-income people, this procedure is still worth doing because the abnormalities lie in the anatomic malformation. The surgical cost for repairing cleft lip and palate at the Senyum Bali Foundation is 7–10 million rupiah, while the package cost for gum graft surgery is 10–15 million rupiah for one patient. “I believe it is very effective because it has greatly improved their quality of life…. Just imagine that a child with a disability is undergoing curative surgery, his appearance has changed, and his function has returned almost to normal. It's very effective, right? Yes, there is no other option other than surgery, because the anatomical abnormality must be corrected, corrected by surgery, there is no other way, it's an anatomical abnormality” (SA, Plastic Surgeon).
Adequate Funding of Healthcare
The foundation must have adequate funding to facilitate the patient's surgical procedure. With a thorough and transparent financial report, donators feel more at ease to keep donating to the foundation. “As far as I know, what is certain for cleft lip is Smile Train Indonesia. It is indeed the biggest source of funding, so with the others, there are usually hotels or villas; they usually email Mrs. E if there is an event and ask to pay for one patient, usually for full coverage. There are many sources, from donations to patients, such as logistics and transportation like airplane tickets. Smile Train Indonesia usually covers the operation's cost, so we look for other donors. For the types of donations, there are regular donations every month, even though it's not much, via transfer usually, some come directly, and some - even though we don't know where they come from - would suddenly ask for one patient to pay for, then there's also the shop. Having two shops, that's also our source of funding, I don't know where the funds are allocated, but at least it's definitely to help finance the ones here. That might be Mrs. M's (founder of the Senyum Bali Foundation) aim when opening a shop like that. Then usually, if it's not in the form of money, usually in the form of used clothes, we resell them at our charity shop. (SU, Foundation Staff 1).”
Effective Delivery of Health Services
To effectively deliver healthcare services, adequate and accessible facilities are necessary. Especially for families in the East of Indonesia, several efforts are needed for optimal care. The foundation provides transportation to and from healthcare facilities, three meals a day, accompaniment during consultations, the surgery, and provisions such as nutritional supplements when needed. “We have two types of patients, patients who live here (in Smile House) from outside Bali and those who come from Bali. We accompanied them every time they went to the hospital, were taken home, have their three meals in the hospital covered. We also get donations in the form of milk or clothes that are still fit to wear; we usually support them too. We usually give them soap and toothbrushes too. It just so happens that we also get such a donation. Yes, we follow up until it's finished, but if they need to be facilitated/look for information about speech therapy, for example, something they need. Still, we don't have a good therapist available yet; we’ll tell them how and where to get them.”(SU, Foundation Staff 1).
Good Quality Physician
Providing the best care and seeking full service for patients is a form of the quality of a good doctor. In addition, it also provides comfort for the patient's parents because the results of the improvement in their child are as expected. “Yes, for sure, I always try to give my best. The existing conditions are never the same; even though the clefts look the same, one side or two sides are never the same, and one patient with another, we always try to give the best. it's the best.” (SA, Plastic Surgeon).
Implementing Cultural Contexts of Health Approach
One of the efforts to obtain health services requires several appeals, such as an approach through consideration of cultural background. In providing education and counseling in certain areas, especially in Eastern Indonesia, the Foundation approaches the Church through Pastors who convey health issues about a cleft lip and palate disorder as a condition that can be corrected, especially for sufferers who come from underprivileged families. They will be assisted in repairing operations to increase the quality of life, and sufferers can live like other ordinary people. Therefore, the success of health services can occur by paying attention to the background of the community and taking an approach in a cultural context. “Initially, it was very tough, but after running for several years, we provide transparent reports. For example, suppose donors donate to us for patient A from the initial cost to the penny. In that case, we report it, so if there are donors, they know where the funds were taken, and with that, surely, they will come for the second time … we are also working with the church because they are more willing to listen to the priests compared to doctors … we consider cultural aspects.” (SD, Foundation Staff 4).
Surgical Outcomes
Surgical procedure is generally done at a young age in cleft lip and palate cases to improve the patient's and their family's quality of life. The parents have expressed that favorable surgical outcomes lead to more significant overall improvements. Even though appearance-wise, it's still asymmetrical and often needs several revisions, results that show visible improvements bring joy to the parents.
“First of all is the results or how after the operation is finished, and maybe also the hospital services that I think will be their consideration. Yes, it depends on the aspects we are treating, right? There is a cleft lip and palate. For the lips, of course, the appearance is symmetrical; the appearance when silent and when speaking must be good, the stitches must be good, we try to make the scar as smooth as possible even though healing is difficult for us predictions too, with the nose, if the cleft lip, the lips and nose will have abnormalities, we try to be as symmetrical as possible, but usually, it's tough, yes, the maximum effort we make is symmetrical, yes, close to normal, yes, but not normal. For the most part, yes, there has been an immediate change that can be seen. They are happy, for sure.” (SA, Plastic Surgeon)
Parents’ Feedback
Listening to patient feedback helps build reciprocal relationships between service providers and patients, which is needed to improve patient care outcomes, patient loyalty, and satisfaction. Doctors always ask the patient's parents to make sure the patient's parents are satisfied with the results of the improvements made to the patient. Good results will satisfy the patient's parents, which can be expressed expressively through various positive responses.
“Many are expressive, especially those with cleft lips who say the results are good. Yes, it is with expressions like that that parents express their satisfaction. Many also come back here, even though they have only had one operation. good, they are satisfied, they often visit here, always in contact, even though patients are far away we still often exchange messages, so we feel that is an expression of parental satisfaction.” (ES, Foundation Staff 3).
Conclusion
Our study has concluded twenty latent variables from applying the triangulation theory and literature, which we assessed as the factors influencing parental satisfaction with cleft lip and palate, including the appearance of the face, the appearance of the lip, the nose, teeth, speech ability, hearing ability, feeding function, masticatory function, breathing function, social function, psychosocial support, perceived quality of life, treatment factors, cost-effectiveness, adequate funding of healthcare, effective delivery of health services, good quality of physician, implementing cultural contexts of health approach, surgical outcomes, dan parents’ feedback.
Footnotes
Acknowledgment
We want to thank Dewi Sri Surya Wuisan, Ir, MM, Ph.D., the Hospital Administration Program head, for her encouragement.
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
Ethical Statement
Written informed consent was obtained from all subjects for their anonymized information to be published in this article. This research has an Ethical Approval from the University's Ethical Committee of Health Research.
