Abstract
Objective
To assess the quality of life of parents of patients with cleft lip and/or palate (CL/P) undergoing cheiloplasty or palatoplasty in the pre and/or postoperative periods.
Design
This is a mixed method study. The sample consisted of parents/guardians of non-syndromic patients with CL/P undergoing treatment in the University Hospital of Western Paraná in Brazil. Data were collected from February 2022 to December 2023 and included demographic information, the Impact on Family Scale (33 items) to assess quality of life, and semi-structured interviews, followed by content analysis.
Results
Caregivers’ (N = 116) quality of life was generally in the middle to high range and similar across pre and postoperative time points. The most frequent topics identified in interviews were financial difficulties, time required for childcare, esthetic concerns, feeding, phonation, and integration of children into social environments with improvements reported after surgeries.
Conclusion
Quality of life of caregivers was similar across pre and postoperative periods and content analysis highlighted improvements in psychosocial and functional aspects. Early follow-up with specialized services is recommended to improve the quality of life of caregivers of children with CL/P.
Introduction
Cleft lip and/or palate (CL/P) are considered congenital diagnoses of craniofacial development that can occur either in isolation or in association with other medical concerns. Their prevalence varies by region, ranging from 1:700 to 1:2500 worldwide. 1 In Brazil, the prevalence is approximately 1:1700 live births, with the highest incidence found in the Southern region, around 1:1215 live births.2,3 The etiology is not yet well-established; however, multiple factors may be involved, including genetic susceptibility and exposure to environmental factors such as alcohol, smoking, nutritional deficiency, and medications (anticonvulsants, corticosteroids, and benzodiazepines). 4
Multiprofessional cleft treatments aim to establish quality of life, defined as individuals’ perception of their experience within the context of culture and value systems and in relation to their goals, expectations, standards, and concerns.5,6 Quality of life encompasses physical, psychological, and social well-being. Parents’ and patients’ perceptions of CL/P support the enhancement of multiprofessional care. 7
The birth of a child with CL/P can affect parents or caregivers and their quality of life in preoperative and postoperative periods. 8 Health-related quality of life (HRQoL) measures are used to assess quality of life, including subscales such as financial and social impact and interpersonal relationships. Children with orofacial clefts may face psychosocial difficulties due to differences in their speech and facial appearance, which can negatively influence their quality of life and family dynamics. 9
Aiming at the reduction of functional concerns and improvement in the patient's esthetic and psychological aspects, cleft treatment should be carried out by a multidisciplinary team, including dental professionals (pediatric dentistry, orthodontics, general dentistry, oral and maxillofacial surgery, endodontics, and prosthodontics), speech therapy, medical professionals (pediatrics, neurosurgery, craniofacial surgery, plastic surgery, and otolaryngology), nutrition, psychology, physiotherapy, social work, and nursing. The treatment begins in the neonatal period with instructions for parents on feeding and hygiene care. Surgical procedures involve the areas of pediatric, plastic, and maxillofacial surgery and start with cheiloplasty around 3 to 5 months, followed by palatoplasty from 12 to 18 months, subsequent alveolar bone graft between 8 and 12 years, followed by orthognathic surgery from 15 years onwards, and finally, rhinoplasties are performed.4,10,11
Surgical procedures are related to the recovery and maintenance of patients’ health, which can impact domains covered by quality of life, such as functional, social, psychological, and financial impacts. Therefore, this study aimed to assess the impact of cheiloplasty and palatoplasty surgeries on the quality of life of caregivers of patients with CL/P undergoing treatment at University Hospital of Western Paraná.
Methods
A mixed-method approach (quantitative and qualitative) was adopted to address the research question: What is the impact of cheiloplasty and palatoplasty surgeries on the quality of life of caregivers of patients with CL/P undergoing treatment at University Hospital of Western Paraná? The research was approved by the ethics and research committee under number 5,245,703.
All research participants were informed about the procedures, advantages, and disadvantages and were invited to participate in the study. Those who agreed to participate signed the Informed Consent Form. Data collection took place from February 2022 to December 2023 with caregivers of patients undergoing cheiloplasty (Q) or palatoplasty (P). Data was collected between 1 month to the day before the surgical procedure (0) and/or from 3 to 6 months postoperatively.
Participants provided sociodemographic data of the child (age, gender, diagnosis, and age at surgery) and of themselves (age, gender, education level, number of children, marital status, current family situation, and monthly income); completed a quality of life questionnaire; and were interviewed with 3 semi-structured questions for pre and post-surgical procedures.
Quantitative Study
The objective of the quantitative step was to describe the cross-sectional HRQoL at each time point. The questionnaire assessing HRQoL was the Impact on Family Scale12,13 (translated into Portuguese), consisting of 33 questions with 5 subscales: Financial Impacts (4 items), Social Relationships (15 items), Personal Impacts (5 items), Coping Strategies (3 items), and, if applicable, Impact on Siblings (6 items). Parents were asked to rate each item on a scale: 1—absolutely true, 2—true in most aspects, 3—not true in most aspects, or 4—absolutely not true. Subscale scores were calculated based on the average of each subscales’ items. In the version used in this study, except for the Coping Strategies subscale, lower scores reflect a greater negative impact on HRQoL and higher scores indicate better HRQoL. On the Coping Strategies subscale, lower scores reflect higher HRQoL. The quantitative data were tabulated in a Microsoft Excel® spreadsheet for subsequent descriptive statistics analysis.
Qualitative Study
The qualitative study aimed to conduct a convergent analysis with the quantitative study. 14 Face-to-face semi-structured interviews lasted on average 35 minutes (range 25 to 45 minutes) and were conducted by a member of the oral and maxillofacial surgery and traumatology team of the service.
Preoperative questions included: (1) What are the biggest challenges you and your family are currently facing; (2) What is your biggest expectation for the surgery? (3) What aspects of our child's life will improve after surgery?
Postoperative interview questions were: (1) What was the biggest expectation for the surgery? (2) Was that expectation met? (3) What aspects of care can be improved?
The data were transcribed into a Microsoft Word® document, and divided by pre or postoperative time point and by questions. Bardin’s Content Analysis (2011) 15 was used and consists of 3 phases: (i) pre-analysis; (ii) exploration of material, categorization, or coding; (iii) treatment of results, inferences, and interpretation. Initially, the data were identified according to the group: cheiloplasty (Q) or palatoplasty (P) and the time point: pre-surgical (0) and post-surgical (1). In the first phase of Content Analysis, a floating reading was performed to identify possible patterns among the responses. In the second phase, the text units were identified and subsequently regrouped, based on the most frequently mentioned categories. In the third phase, the results were critically analyzed.
Results
This study included an unpaired sample of parents or guardians of 116 children. Out of the total number of children, 61 underwent cheiloplasty surgery, with 31 parents addressed in the preoperative stage and 30 in the postoperative stage. Additionally, 55 children underwent palatoplasty surgery, with 32 parents addressed in the preoperative stage and 23 in the postoperative stage.
Quantitative Analysis
The mean patient age in the preoperative period for cheiloplasty was 7.42 months, ranging from 4 to 24 months, and patients had a mean age of 23.35 months before palatoplasty, ranging from 7 to 54 months. Across time points males predominated, accounting for 66.37% of patients, resulting in a ratio of 2 boys to every 1 girl with cleft lip and palate. Regarding the type of cleft, 12.06% had cleft lip, 75% had cleft lip and palate, and 12.93% had cleft palate. The mean age of parents or guardians was 29 years, ranging from 18 to 53 years, and89% were female. The mean monthly income was R$ 2976.81, ranging from R$0.00 to R$16,000.00, with the Brazilian minimum wage during the study period being R$1212.00. Complete descriptive statistics at each time point are shown in Table 1.
Description of Sociodemographic Data.
The results of the Impact on Family subscales are shown in Table 2. Scores were similar across the pre and postoperative periods for cheiloplasty and palatoplasty. Scores were generally in the mid-range with highest HRQoL consistently reported for positive Social Relationships and Impact on Siblings. Positive Coping Strategies were endorsed, with lower scores reflecting higher HRQoL. The lowest relative area of HRQoL across time points was for Financial Impacts.
Mean of Subscales.
Qualitative Analysis
The most frequently identified topics in parents’ interviews were financial difficulties and time required for childcare, esthetic concerns, feeding, phonation, and integration of children into social environments, as described below across time points.
Cheiloplasty
Preoperative Period In the cheiloplasty group, parents or guardians reported many preoperative challenges, including difficulties with feeding, concerns about esthetics, and the need to travel for treatment. They highlighted issues such as “consistency of treatment, expenses, time, absence from work, and physical and emotional exhaustion.” Additionally, the time dedicated to caring for a child with a cleft was described as requiring “care, time spent caring, more comprehensive than usual, affecting our daily life.” While some reported no difficulties, stating “everyone accepted it well, even during pregnancy,” others mentioned facing “psychological difficulty; I only discovered the condition at birth,” which appeared to reflect more difficult adjustments without the benefit of a prenatal diagnosis. Parents also reported effects on quality of life related to sibling relationships. One parent noted “jealousy in our other daughter and feeling neglected because we give more attention to him.” Financial impacts were also frequently mentioned: “I will have to stay away from my job for a few days” and “I had to leave work to take care of him.”
Feeding difficulties were a common preoperative concern among parents or guardians, referenced in many reports, such as: “breastfeeding was supposed to happen, but he couldn't suck properly,” “buying milk, because he couldn't breastfeed,” “we struggled until we found the right bottle to help him gain weight,” and “feeding with the spoon bottle—he takes it initially, but then he gets stressed and refuses it.” Beyond difficulties identifying the best bottle and expenses related to milk formulas, one family noted “he's only on the feeding tube, the speech therapist has started introducing food.”
When asked about preoperative expectations, most responses included hoping for a successful surgical procedure and for the children to recover well postoperatively, such as: “that he gets well, recovers soon, and doesn't get so stressed out… that he can develop because there are things he can't do due to the cleft.” There were also expectations about appearance: “good result to the point that no one notices so that he doesn't suffer from bullying,” “to see her even more beautiful than she already is” and “to see his smile closed.”
Regarding aspects expected to improve with surgical intervention, most respondents mentioned esthetic enhancement, reducing society's prejudice, for example, “he will look the same as his siblings” and “improving appearance to avoid bullying.” There were also expectations regarding relationships between siblings: “that his relationship with his older brother improves, because he is a twin, and I feel that his relationship with the other siblings is better.” Additionally, feeding appeared as an aspect that would improve postoperatively: “he uses tape every day and breastfeeds better, [surgery] will help to close his mouth to breastfeed better” and “improve feeding, because there are things he doesn't want to eat and may improve with surgery.”
Postoperative Period When parents or guardians were approached in the postoperative period about their expectations for the surgical procedure and whether these were met, responses often included esthetics, including concerns about ongoing visible differences: “if the scar would be perfect, without signs of healing.” Concern about prejudice continued to be identified “I am afraid he will suffer bullying, whether we like it or not, people look at him differently, I always tell my husband, don't even worry about it, he's perfect just the way he is.” Other parents stated: “if only the mark could be removed,” “maybe a rhinoplasty to improve appearance,” and “the nose was a bit down on one side, but I know it will be corrected later.”
Regarding areas for improvement, the responses were mainly related to functional aspects of lip movement, feeding, and speech. Ongoing concerns were noted, such as “there are movements he wants to make with his lip and he can't, for example, blowing a kiss, he tries to close his mouth to make the noise, but he can't.” Other parents noted some improvements in feeding and speech: “improved for feeding, drinking water” and “speech, pronouncing the right words and being able to eat better…I still fear giving him some foods: fruits, cookies, because what if he chokes!?” Ongoing areas of concern were often expected to be addressed through palatoplasty, such as: “I can't wait for him to have the palate done and not have [food] come out through his nose so that he can eat without me having to watch over him…every time he eats, I need to be attentive; there were weeks when he choked twice and turned purple.”
Analyzing the responses of parents and guardians, preoperative expectations were primarily focused on feeding and how esthetics would impact their children in social relationships. In the postoperative period, most parents or guardians expressed relief regarding societal prejudice. However, in cases of the cleft palate, parents still awaited palatoplasty to see additional improvements.
Palatoplasty
Preoperative Period The primary preoperative difficulties reported by parents and guardians in the group undergoing palatoplasty surgery were related to financial concerns and the time required for treatment and childcare. For example, parents reported “financial [stressors] and lack of time, balancing work with appointments,” “time to take care of my son,” “taking care of my son, so he does not get hurt, fall, hit his face,” and “my husband's unemployment.” Difficulties related to feeding were also observed, such as: “fear of choking and food goes into the nose.” Concerns about speech were observed in many cases: “I'm afraid about speech, which will interfere with the learning process and socialization.” Some parents noted strengths despite speech issues, such as “he doesn't say some words, but he socializes well” and “the family doesn't understand [his speech], but I do and his older brother also understands him well…or we understand him through gestures.”
Among the greatest expectations for the surgical procedure, parents identified integration of children into the social environment, often through improved speech. For example, parents hoped for “adaptation in the school environment with people who don't know him,” “that he can express himself better and not get so nervous because they don't understand him,” “improved speech, interaction with other people and communication,” and “he will be able to communicate, not only with me, but with other people too.” Greater comfort with safe feeding was also expressed: “for her to be freer because people are afraid, she will choke, so they don't take her out,” and “that he can eat everything and stop food from coming out of his nose.” Furthermore, they hoped for improvement in breathing: “to breathe better.” Parents also expressed an expectation of improvement in time spent with the child: “it won't be so rushed, my husband works overnight, it's tiring for him to leave work and come to accompany us.”
Postoperative Period The greatest postoperative expectations were regarding speech and feeding: “that she develops speech and that she could eat better, without worries,” “that she speaks and that she can eat, because before everything was mashed,” and “that she can speak without nasal [air] escape.” Most reports were positive about some surgical expectations being met: “regarding speech yes, regarding food not yet, [it's] less [than before surgery, but] it still comes back,” “feeding [expectations were met], but speech still not yet, because he was born with a tied tongue,” “feeding is excellent, infections and sinusitis have decreased a lot, but in speech there's still difficulty,” and “speech isn't very good, he doesn't speak much, but little by little… feeding, he eats better, more calmly.”
Regarding aspects that could improve, social issues and speech were frequently identified: “speech and interactions with other people could improve” and “in speech he still cannot pronounce [words] well, he calls me ‘gaga’, his sister ‘dada’, then mommy is ‘mama.'” esthetic concerns were also reported: “people still look [at child] differently” and “only with plastic surgery later, because of school.”
While feeding was a frequent concern alleviated in the post-operative period, parents’ main ongoing concerns were related to the social environment, both in family settings and future school interactions. Speech was noted as a key area related to both communication and social interactions.
Discussion
Cl/P are congenital medical concerns that affect craniofacial structures and influence patients functionally, psychologically, and esthetically, potentially impacting the quality of life of patients and their families. 6 This paper aimed to describe caregiver quality of life in the pre and postoperative periods of early cleft care in a Brazilian sample. Regarding the characteristics of patients in the study sample, the ratio of 1 girl to every 2 boys was obtained, consistent with the literature. 4 However, combined clefts (CLP) were the most prevalent, contrasting with other studies 3 conducted in Brazil where the proportion of clefts was similar among the 3 types of cleft. In the quantitative analysis, the means of quality of life in the pre and postoperative periods of patients undergoing cheiloplasty and palatoplasty surgeries were similar across time points and in the middle to high range of HRQoL.
Parents or guardians of patients with CL/P can be affected by many factors from birth. 16 In this study, some parents were aware of the cleft during prenatal care, while in others, it was only detected at birth. Late recognition increases the social impact on families.5,9,17 Following diagnosis, families must prepare for surgery and the perioperative period can positively or negatively influence the perception of quality of life. Many expectations are generated during the preparatory phase of the surgical procedure regarding form or esthetics, function impacting feeding and phonation, potential surgical complications, and increased demand for care for the child in the postoperative period.18–20 Cleft care providers can help adaptively inform parental expectations and support caregiver adjustment.
The qualitative data regarding expectations and postoperative outcomes align with other studies,21,22 which also identified esthetic improvement resulting from the surgical procedure, along with frequent concerns about bullying, especially in the school environment. Additionally, participants in this study identified a reduction in concerns about feeding, particularly in the palatoplasty postoperative period, as well as improvements in speech development and communication, which has been associated with improved psychosocial adjustment. During the qualitative analysis, functional improvements were identified as the main factor affecting quality of life, with a decrease observed in the preoperative period and improvement in the postoperative period.
Chronic conditions can promote positive adjustment in parents, as well as resilience development for daily coping and surgical procedures.23,24 While surgical treatment can assist in improved social inclusion for individuals affected by CL/P 25 it is important for cleft providers to understand the psychological and social issues involved with the patient and the family, as well as the individual risk factors that each family faces.26,27
Limitations of this study include the use of cross-sectional data collection that did not allow for longitudinal analysis across time points. Another limitation of the study is the postoperative period established for data collection, which was 3 to 6 months. Extending the postoperative data collection period, particularly for palatoplasty, could provide a more accurate assessment of quality of life by allowing for complete healing and the child's adaptation to postoperative functional needs, especially those related to speech. Additionally, each type of cleft could have been analyzed quantitatively in isolation. Our qualitative analysis showed that when parents are aware of the condition before birth, there was a higher quality of life from the preoperative period. Therefore, gathering information on the timing of diagnosis should be included in future studies. Use of existing standardized measures developed for CL/P populations as well as the general population is also recommended to allow for clinical interpretation and comparison across studies.
Conclusion
The objective of the research was to assess the impact on quality of life of Brazilian parents and/or guardians of patients of children with CL/P undergoing cheiloplasty and palatoplasty surgeries in the pre and postoperative periods. The qualitative analysis highlighted improvements in psychosocial and functional aspects and middle to high levels of quality of life were maintained across subscales in the preoperative and postoperative periods. Early multidisciplinary cleft care is recommended to support patients with CL/P and their caregivers starting at the time of diagnosis.
Footnotes
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Ethical Approval
This case report was approved by the ethics committee of the Western Parana State University under number 5,245,703.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
