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| ORIGINAL ARTICLE |
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| Year : 2022 | Volume
: 9
| Issue : 4 | Page : 291-298 |
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Psychological distress and related factors among parents having children with cleft lip and palate disorder: evidence from Sri Lanka
Aruni M K. Senavirathne, Prasanna Jayasekara, Nethma Kalani Jayasekara
Research and Surveillance Unit, Institute of Oral Health, Ministry of Health, Maharagama, Sri Lanka
| Date of Submission | 22-Apr-2022 |
| Date of Decision | 17-Oct-2022 |
| Date of Acceptance | 17-Oct-2022 |
| Date of Web Publication | 12-Feb-2023 |
Correspondence Address:
Prasanna Jayasekara
Research and Surveillance Unit, Institute of Oral Health, Maharagama 10280
Sri Lanka
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jdrr.jdrr_57_22
Background: Raising a child with a cleft lip and or palate disorder (CLPD) brings many challenges to the parents. Having children with CLPD can be a source of parental psychological distress (PPD). Stress experienced by parents may affect the development of a child. This study was designed to assess the prevalence and factors associated with PPD among parents having children with cleft lip and palate attending a major dental hospital in Sri Lanka. Materials and Methods: Cross-sectional study was conducted in cleft lip and palate clinics of Dental Hospital, Peradeniya, Sri Lanka. PPD was assessed using the General Health Questionnaire-30. Site of cleft and associated disabilities were recorded on data recording form using clinical records of the children. Chi-square and multivariate logistic regression analysis were used to analyze the data. Results: Out of 384 parents who accompanied their children to the clinic, 62.5% were mothers. The most common cleft site of the patients was cleft involving lip and palate (32.3%) and the least common site was isolated cleft of the soft palate (8.3%). The most common disability reported was speech problems (41.4%). The prevalence of PPD was 34%. Chi-square statistics showed that PPD was significantly associated with age of the child (P < 0.000), family income (P = 0.011), family structure (P < 0.000), previous knowledge on CLPD (P = 0.045), site of the cleft (P < 0.000), and disabilities associated with particular cleft (P < 0.000). Multivariate logistic regression analysis demonstrated that age of the child, family structure, cleft of the hard and soft palate, feeding difficulties, and speech problems were significant predictors for PPD. Conclusion: PPD is an important factor to consider in managing children having CLPD. This finding provides valuable information for the provision of multidisciplinary approach to manage PPD.
Keywords: Cleft lip, cleft palate, parental psychological distress
How to cite this article:
K. Senavirathne AM, Jayasekara P, Jayasekara NK. Psychological distress and related factors among parents having children with cleft lip and palate disorder: evidence from Sri Lanka. J Dent Res Rev 2022;9:291-8 |
How to cite this URL:
K. Senavirathne AM, Jayasekara P, Jayasekara NK. Psychological distress and related factors among parents having children with cleft lip and palate disorder: evidence from Sri Lanka. J Dent Res Rev [serial online] 2022 [cited 2023 Apr 1];9:291-8. Available from: https://www.jdrr.org/text.asp?2022/9/4/291/369588 |
| Introduction | |  |
The condition of cleft lip and or palate disorder (CLPD) occurs when a facial structure not properly closing together during the developmental phase. This condition includes cleft lip, cleft palate, isolated cleft palate, and both together. Feeding problems, speech problems, hearing problems, frequent ear infections, and oral health problems are associated with disabilities of this disorder. Usually, CLPD is associated with other congenital diseases.[1]
According to a systematic review done by Salari et al., the global prevalence of cleft palate, cleft lip and cleft lip and palate were 0.33, 0.3 and 0.45 respectively for every 1000 live births.[2] This rate varies considerably across different regions of the world. The prevalence of orofacial clefts in the Asian region was 1.57 for every 1000 live births.[3]
The epidemiological data on cleft lip and palate in Sri Lanka are limited. A prospective study conducted in the Central Province of Sri Lanka reported that the incidence of cleft lip with or without cleft palate was 0.83/1000 births, and for isolated cleft palate, the incidence was 0.19/1000 births.[4] A higher incidence of 2.2/1000 live births was reported in Anuradhapura of Sri Lanka in 2008.[5]
Psychological distress is one of the most common diseases in the society. Having children with deformities like CLPD can affect parent's emotional balance. Depression and anxiety of parents have been reported to be associated with CLPD of their children.[6] Ultimately, it would lead to change the social role of the parent. Parents of infants with CLPD generally are distressed as a result of lower infant weight gain. This may result from difficulties in direct breastfeeding and higher risk for upper respiratory infection of the child.[7]
In Sri Lanka, the psychological condition of parents having children with CLPD had not been given adequate attention and there was no systematic documentation related this condition. Sri Lanka is having self-sustaining multidisciplinary team approach for the treatment of cleft lip and palate. When a child presented to a government hospital with the CLPD at the beginning of their life, that child is followed up to 18 years until they have undergone plastic surgeries for esthetic correction.[8]
Although services are available for the psychological development of those affected children, there is limited attention paid for the psychological development of their parents, who have undergone different kinds of stressors at the same time.
The current study was conducted at Dental Hospital, Peradeniya (DHP), a tertiary care dental hospital in Sri Lanka, which acts as a referral center for all oral health needs. As the DHP is having well-planned treatment sequelae and offers surgical expertise to repair CLPD and to rehabilitate children with CLPD, patients are referred to this hospital from all over the country. Therefore, information gathered through this study will help to build a rational idea about the psychological condition of parents in Sri Lanka who are having children with CLPD.
This study was conducted aiming to assess the prevalence and associated factors of parental psychological distress (PPD) of parents having children with CLPD who were attending cleft lip and palate clinics, DHP.
| Materials and Methods | | |
The data for the present paper were obtained from a hospital-based cross-sectional study. Data were collected from January to April 2017. Ethical approval for this study was obtained from Ethical Review Committee, Faculty of Medicine, University of Colombo. Written informed consent was obtained from the parents, by providing information sheets and consent forms. Essential ethical details were provided to them including their right to confidentiality and their right to withdraw.
This study was conducted among parents having children with CLPD, undergoing treatment at cleft lip and palate clinics conducted by the oral and maxillofacial unit at DHP, located in the Central province of Sri Lanka. Only one parent of the affected child was included in the study.
Calculation of the sample size was done using the formula for estimating a population proportion with absolute precision. As there is no previous study on the prevalence of psychological distress among parents having children with CLPD in Sri Lanka, the prevalence was taken as 50%. The final sample size for this study was 384.
Nonprobability consecutive sampling method was used in this study due to low prevalence of the disease. All eligible parents having children with CLPD, who were participating in the cleft lip and palate clinics were recruited. Parents having children with births defects other than CLPD were excluded from the study.
Data collection took place at the hospital premises using a pretested self-administered questionnaire and a data recording form. Self-administered questionnaire consisted of two parts. The first part included questions on sociodemographic data (gender, ethnicity, and education level of the parents, parent living with spouse, family income, and family structure) and questions relevant to previous knowledge of parents about CLPD. The second part included 30-item General Health Questionnaire (GHQ-30) to assess the PPD. Clinical records of the children with CLPD were used to complete data recording form to assess age of the child, site of cleft, and associated disabilities.
Several measures were taken at the designing stage of the questionnaire, at the time of data collection, data entry, and analysis stage to ensure the validity and reliability of data. The first part of the questionnaire was validated for the judgmental validity in terms of face validity, content validity and consensual validity by the relevant experts in the field. Pretesting of the questionnaire was carried out among 10 parents having children with CLPD attending Lady Ridgeway Hospital for Children, Colombo, to assure that all the questions were clear and understandable to the participants. The average time taken for answering the questionnaire was 30 min.
The second part, GHQ-30 was already validated to Sri Lankan setup.[9] Psychological distress was calculated using marks obtain by parents from the GHQ-30. The responses for the 30 question items were rated on a four-point Likert scale. The Likert scale was transformed into GHQ score (0, 0, 1, 1). This produced a total score ranging from 0 to 30. As suggested by Abeysena et al., the cutoff score of 6 was taken as to decide the presence or absence of PPD.[9]
The data were analyzed by the Statistical Package for Social Sciences (SPSS, version 21, IBM Corp, Armonk, New York, NY, USA) . The relationship between the presence of PPD and the associated categorical variables was explored using the Chi-square test. The level of significance was set as P < 0.05. All variables with P < 0.05 were used for the multivariate logistic regression analysis which was performed using backward logistic regression methods which gradually removes the effect of confounding factors to identify the most important variables responsible for PPD.
| Results | | |
The current study population included 384 children who were undergoing treatment for CLPD, and whose parents responded to self-administered questionnaire. The mean age of children having CLPD in the sample was 58.8 months (standard deviation ± 43 months). Out of the parents included in the study, mothers accompanied 62.8% of their children. The majority of the participants were Sinhalese (78.9%), educated up to grade 11 (66.1%), had monthly family income <Rs. 25,000/(53.9%) and living with extended families (51.3%). Out of the affected children, the most common cleft site was cleft involving lip and palate (32.3%) and least common site was isolated cleft of the soft palate (8.3%). The most common disability reported was speech problems (41.4%).
The prevalence of psychological distress was 34.4%. As shown in [Table 1], PPD was higher among fathers than mothers but the difference is not significant. The PPD was significantly associated with age of the child (P < 0.000), family income (P = 0.011), family structure (P < 0.000), previous knowledge on CLPD (P = 0.045), site of the cleft (P < 0.000), and disabilities associated with particular cleft (P < 0.000). | Table 1: Factors associated with the parental psychological distress of the study population
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All variables associated with PPD having P < 0.05 were included in the multiple logistic regression analysis. The Omnibus Tests of Model Coefficients demonstrated the model has a Chi-square value of 222.074 and a P < 0.000 which indicates that the model is a significant predictor of PPD. Nagelkerke R-Square 0.607 revealed that 60.7% variance in the PPD was predicted by the predictor variables.
[Table 2] shows the adjusted associations between sociodemographic factors and other health-related factors with PPD. Controlling for confounding factors, PPD was significantly higher for parents having CLPD children with aged less 2 years (adjusted odds ratio [AOR], 16.327; 95% confidence interval [CI], 7.158–37.238). Parents who live with their extended families were significantly less likely to have PPD than living as nuclear families (AOR, 0.344; 95% CI, 0.182–0.649). Out of the sites of the cleft of the children, only cleft in the hard and soft palate was found to have a significant impact on PPD (AOR, 9.984; 95% CI, 2.313–43.107). Among the associated disabilities, feeding difficulties associated with cleft (AOR 2.758; 95% CI, 1.056–7.206) and speech problems associated with cleft (AOR 0.435; 95% CI, 0.196–0.967) were significant predictors for PPD in the current study. | Table 2: Multivariate analyses: Selected factors associated with parental psychological distress
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| Discussion | | |
PPD of parents having children with CLPD has given much less attention in Sri Lankan health literature. The current study conducted among hospital-based sample of parents identified that the prevalence of PPD among the study population was 34%. The crude analysis of this study demonstrated that the PPD was significantly associated with age of the child, family income, family structure, previous knowledge on CLPD, site of the cleft and disabilities associated with a particular cleft. However, multiple logistic regression analysis identified that out of all these variables, age of the child, family structure, cleft in the hard and soft palate, feeding difficulties associated with cleft and speech problems associated with cleft were significant predictors for PPD in the current study. These results may provide valuable information for multidisciplinary cleft care providers to manage the PPD.
Previous research in the field of CLPD has documented parents' emotional and social impact on their child's condition.[10] Children with CLPD face social marginalization and stigmatizing reactions from others. This may be particularly distressing for mothers and may evoke anxiety for their child's future. Managing parental distress has been highlighted in the health literature to reduce the risk for psychological problems of parents with children having cleft lip and palate. Improving mother–child interactions may affect the timing of treatment for CLPD.[11]
Overall, the present study demonstrates the importance of concept of family. Those living with the spouse and living with extended family had lower scores of PPD. The extended family could provide support with household work and childcare and lessen the burden on the parents. Fathers were more distressed than mothers regarding the condition of their children. Although fathers play a key role in supporting their families through the treatment process of the affected children, they are underrepresented in the research literature.[12] Initially, fathers may not show their own concerns. However, they may provide a strong moral support to their family following the diagnosis of CLPD condition of the infant. Efforts should therefore be made to involve fathers in clinical care wherever possible. The findings of the study done by Stock et al. suggested that psychosocial impact of CLPD on affected individuals could be improved by involving all members of the family.[13] The impact on the wider family is hardly investigated. The role of grandparents in providing care of their grandchildren with CLPD was investigated by Guest et al. and highlighted the importance of incorporating them in proving the moral support to the parents of the children.[14]
The present study reported that the age of patients was an important predictor for the psychological distress of the parents. The effect of age of the child on the psychological conditions of caregivers of patients with CLPD has also been confirmed by studies carried out by Aslan et al. in 2018[15] and Yuan et al. in 2022.[16] When a child is born with a cleft, parents are initially a state of shock and distress. However, after finding the possible causes for the condition and the best treatments options available for the condition, they overcome the initial psychological trauma. There was a significantly lower level of distress of parents when the child was more than 2 years of age in this study. It is mainly because as the patient grows up and the treatment progresses, the parents can take care of their children more easily. However, one problem that we cannot ignore is the child's psychological and developmental problems. Many parents of children with CLPD pay too much emphasis on improving the appearance and function of their children. However, the interpersonal communication and personality development of these children are ignored.[16] Although the burden of medical care for parents is reduced as children with CLPD grow up, parents need to be assisted with relevant professional services to promote the healthy growth of affected children psychologically and socially.
Feeding difficulties often experienced by infants with CLPD increases the psychological distress of parents. This relationship was clearly demonstrated in the present study as well as many other studies found in the health literature.[17],[18] This may be because the feeding time was significantly longer and exhausting both for baby and mother. The oronasal communication affects the functions performed by the oral and nasal cavities in individuals with CLPD. This reduces the ability to create negative pressure necessary for suckling. Further, these children have the risk of nasal regurgitation of food and excessive air intake resulting frequent burping and choking. Therefore, the clinician as well the parents need to pay their immediate attention on the feeding of the infants with CLPD in the newborn period.[19] The main priority during the first few months of life for all infants is to ensure adequate nutrition. The infant born with a cleft has similar nutritional requirements as other infants born without a cleft.[20] Mothers should be educated about the proper feeding techniques. The management of CLPD cases is lengthy and needs to include a multidisciplinary team approach.[21]
The current study identified speech problems associated with cleft as a significant predictor for PPD. The reason may be that the lack of speech and communication abilities of children with CLPD may increase the distress of the parents. Some children exhibit "cleft palate speech" characterized by atypical consonant productions, abnormal nasal resonance, and abnormal nasal airflow even with early cleft repair. These abnormalities can be corrected through speech therapy.[22] The speech therapist plays a major role in the cleft care team and needs to work closely with the surgeon and other team members to ensure that timely assessments and appropriate management. Improving this communication disorders in individuals with CLPD facilitates diminishing the distress of the parents.
Cleft care is most successful when services are not only comprehensive but also multidisciplinary in nature. Thus, it is important for each member of the cleft care team to understand the fundamental principles of care in the area of expertise of other members of the team.
GHQ-30 used in this study detects the current psychological distress level of the parents, which can be affected by temporary events. Qualitative research on the field can be conducted to overcome this limitation. Since this study was conducted only in one hospital, the findings of this study cannot be generalized to all the parents having children with CLPD in Sri Lanka.
| Conclusion | | |
The current study identified that psychological distress was present among one-third of the parents of children having CLPD. The age of the child, family structure, cleft in the hard and soft palate, feeding difficulties associated with cleft and speech problems associated with cleft were significant predictors for PPD.
This study highlights the importance of screening the parents of children having CLPD for factors affecting PPD in health-care settings, and initiating referrals for early intervention when these parental risk factors are present. It provides valuable information for multidisciplinary cleft care providers in terms of managing the PPD.
Ethical statement
Ethical approval for this study was obtained from Ethical Review Committee, Faculty of Medicine, University of Colombo (Ethical approval number EC - 16 - 135).
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Study instruments
Study reference No ......................................................................................
Questionnaire to assess psychological distress and related factors among parents having children with cleft lip and palate disorder
Part I – General information
Please tick (√) mark in the appropriate box
Part II – General Health Questionnaire (GHQ 30)
We would like to know if you have had any medical complaints and how your health has been in general over the past few weeks. Please answer ALL the questions on the following pages simply by underlining the answer which you think most relevant to you. Remember that we want to know about present and recent complaints, NOT you had in the past
Please read this carefully, answer ALL questions
Have you recently
Data recording form - details from clinical records
To be filled by the investigator
| References | | |
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| 2. | Salari N, Darvishi N, Heydari M, Bokaee S, Darvishi F, Mohammadi M. Global prevalence of cleft palate, cleft lip and cleft palate and lip: A comprehensive systematic review and meta-analysis. J Stomatol Oral Maxillofac Surg 2022;123:110-20. |
| 3. | Panamonta V, Pradubwong S, Panamonta M, Chowchuen B. Global birth prevalence of orofacial clefts: A systematic review. J Med Assoc Thai 2015;98 Suppl 7:S11-21. |
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| 7. | Tsuchiya S, Tsuchiya M, Momma H, Koseki T, Igarashi K, Nagatomi R, et al. Association of cleft lip and palate on mother-to-infant bonding: A cross-sectional study in the Japan Environment and Children's Study (JECS). BMC Pediatr 2019;19:505. |
| 8. | Lambadusuriya SP, Mars M, Ward CM. Sri Lankan cleft lip and palate project: A preliminary report. J R Soc Med 1988;81:705-9. |
| 9. | Abeysena C, Peiris U, Jayawardana P, Rodrigo A. Validation of the Sinhala version of 30-item General Health Questionnaires. International Journal of Collaborative Research on Internal Medicine and Public Health 2012;4:1373-81. |
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| 14. | Guest E, Costa B, McCarthy G, Cunniffe C, Stock NM. The experiences and support needs of grandparents of children born with cleft lip and/or palate. Cleft Palate Craniofac J 2019;56:1181-6. |
| 15. | Aslan BI, Gülşen A, Tirank ŞB, Findikçioğlu K, Uzuner FD, Tutar H, et al. Family functions and life quality of parents of children with cleft lip and palate. J Craniofac Surg 2018;29:1614-8. |
| 16. | Yuan L, Gao Y, Pan B, Wang J, Wang Y, Gong C, et al. Resilience and related factors: A comparison of fathers and mothers of patients with cleft lip and/or palate in China. Front Psychiatry 2021;12:791555. |
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[Table 1], [Table 2]
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