Development and validation of psychological status questionnaire for parents of infantile hemangiomas

Introduction

Infantile hemangioma (IH) is one of the most common benign tumors in infants, the incidence of which can reach 4–5% (1). It is mostly located on the face and neck (2,3), and has a specific natural course, including a stage of hyperplasia, regression, and extinction. Some IHs are self-limiting and require no specific treatment (4). However, professor Jinzhe Zhang, one of the founders of China’s first pediatric surgery department (5), repeatedly emphasized that the management of IH was prone to overtreatment, as most of the treatment rendered did not take the natural course of the disease into consideration (6). Many authors have labeled the overtreatment of IH as unnecessary (7-9). However, these authors have tended to place more focus on the diagnosis and treatment of the disease and ignore the influence of parental psychological factors.

As we know, overtreatment is a medical behavior or a medical process, which is caused by a variety of causes exceeding the actual need for diagnosis and treatment of the disease (10). As to the cause of overtreatment, besides the current medical process and ethical aspects, one of the most important contributing factors is the patient expectations or pressure/request, which cannot be ignored during the course of treatment (11,12). At the proliferative phase, IH typically grows rapidly, with the classical clinical manifestations of flushed skin on the face and around the head and neck. This has an enormous effect on the child’s appearance, even causing obvious facial deformity (13,14). The physical appearance of the child has a psychological impact on both children and parents (15,16). These complicated psychological states disrupt the clear communication between doctors and patients, thereby undermining the parents or caregiver’s understanding of IH and its treatment (17). It is therefore paramount to clearly understand the parents or caregiver’s psychological state in order to provide proper counseling for appropriate and effective management (18). Unfortunately, there were previously no standardized questionnaires about the psychological status for Chinese parents with IH children.

To address this, the aim of current study was to design and validate a Psychological Status Questionnaire for Infantile Hemangiomas’ parents (IH-PSQ). We hypothesized that IH impacts the affected the parent’s emotional and social functioning. This paper describes the development and validation of IH-PSQ instrument for measuring the psychological status of IHs’ parents during this early critical period.

We present the following article in accordance with the MDAR reporting checklist (available at https://dx.doi.org/10.21037/tp-21-554).

Methods

The questionnaire was developed according to standard questionnaire development guidelines and methodologies (19) according to the following steps: questionnaire creation, questionnaire validation, and analysis of influencing factors. A multidisciplinary team, composed of health care professionals such as 3 IH experts, 3 psychologists, and 2 IH nursing experts, collaborated with the IH patients and their families.

Statistical analysis

The data collected were entered and analyzed using SPSS 20.0. Statistical analyses included descriptive statistics, factor analyses, reliability analyses, product-moment correlations, independent-sample t-tests, One-way ANOVA and regression analyses.

Questionnaire creation

During the conceptual phase, a series interview was conducted with clinicians, patient-reported outcome experts, and IH parents to collect parents’ opinions and complaints as the initial interview questions. Semi-structured individual interviews of 10 IH children’s parents were then conducted. Participants were asked questions about psychological status for the disease. Answers were transcribed and a content analysis of the responses was carried out according to published methodology, in order to build an initial wording report. Based on this initial wording report, the working group drew up a list of items that were reformulated as simple, comprehensible questions. They evaluated the grammar, wording, and scaling of each item.

Questionnaire validation

During this phase, the initial questionnaire and the correlated demographic and clinical features questionnaire were administered to a random sample of parents of IH patients (n=350). After reading the informed consent form, the parents completed the questionnaire survey on the spot while waiting at the Clinical Research Center of Vascular Abnormalities in Jiangxi Province, China.

Then, the item analysis of the questionnaire was determined using extreme value comparisons (decision values), entries and total score correlation (including entries and total score correlation, correction entries, and total score), and homogeneity test (including the alpha value after deletion, commonality, and factor load). The validity evaluation of the initial IH-PSQ was mainly based on two aspects: exploratory factor analysis and criterion-related validity. A total of 50 parents of IH children were randomly selected to complete an SAS (Self-rating Anxiety Scale) and SDS (Self-rating Depression Scale) at the same time. Pearson correlation analysis was conducted between the initial IH-PSQ and the scores of SAS and SDS to verify the validity of the questionnaire; The reliability analysis of the initial IH-PSQ was mainly based on three aspects: including internal consistency reliability, split half reliability, and test-retest reliability. Complete data was available for 50 (randomly selected) participants tested at the 2-week interval, to verity the test-retest reliability.

Analysis of influencing factors

In order to exclude the possible interaction between factors, this study used multiple linear regression to analyze the influencing factors of the questionnaire. According to the character of the data, this study set the working conditions of the parents, home address, duration of illness, and the main caregivers as the dummy variables; the educational level of the parents, number of children, monthly income of the family, whether the child had been treated, single or multiple hemangioma, tumor size, and whether there were complications as independent variables, and the score of IH-PSQ as the dependent variable. Backward regression analysis was used to screen the influencing factors of the score of IH-PSQ.

All procedures performed in this study involving human participants were approved by the Ethics Committee of The First Affiliated Hospital of Gannan Medical University (No. LLSC-2021062503) and were in accordance with the Declaration of Helsinki (as revised in 2013). All participants voluntarily participated in the study and signed a written informed consent before taking part.

Results

Questionnaire creation

According to the results of the semi-structured phenomenological interview with the parents of IH children, feelings mentioned more than four times were selected as the initial item (total 35 items), which formed the item pool.

A total of 10 parents of children with IH read the items severally, and proposed amendments on the existence of ambiguity, inaccurate expression, and unrealistic entry. The multidisciplinary team evaluated and corrected the objectivity, accuracy, comprehensiveness, and the form of the questionnaire entries. The initial questionnaire was formed according to the results of this evaluation (Table 1). The answer option was based on the LIKERT 5-level system for quantification as follows: “never/not applicable” means 0 point, “rarely” means 1 point, “sometimes” means 2 points, “often” means 3 points, “very often” means 4 points, and “constantly” means 5 points. To prevent confusion with any changes in perception due to symptoms related to comorbidities, the majority of questions included the wording “After your child got IH”.

Questionnaire validation

Correlated demographic and clinical features

A total of 350 initial questionnaires were distributed, and the recovery rate was 100%. We excluded 27 questionnaires on account of the questionnaire being incomplete, leaving a total of 323 (92.29%) effective questionnaires.

Table 2 and Table 3 show the demographic data of the 323 participants and their children, which were collected from their parents through self-reported questionnaires.

Item analysis

The item analysis of the entire initial questionnaire is shown in the Table 4, and the criteria for each line are at the bottom of the table. Even if only one rule was violated, the item was deleted. The results showed that item 1, item 4, item 11, item 21, item 26, item 29, item 31, and item 35 did not meet the standard, leaving 27 items after their deletion.

Validity evaluation

Factor analysis was used to investigate the construct validity of the questionnaire. The software SPSS 18.0 (IBM Corp., Chicago, IL, USA) showed that Kaiser-Meyer-Olkin (KMO) value was 0.750, and the χ² value of Bartlett spherical test was 6,114.620 (P<0.05). There was a significant difference between the correlation coefficient matrix and the unit matrix, indicating that the questionnaire was suitable for factor analysis. The principal component analysis (PCA) method was used to select the orthogonal rotating shaft mode, and the factor with an eigenvalue of greater than 1 was selected.

The results of exploratory factor analysis found that 6 factors could be extracted with a cumulative contribution rate of 85.014%. Factor 6, however, contained only 2 items (item 3 & 12), which did not meet the requirements. Deleted items 3 and 12 could be extracted after 5 factors, with a cumulative contribution rate of 83.239%. All the entries on the corresponding load factor were above 0.4. However, item 13 in the first factor and load on the fifth factor was above 0.4 at the same time, and item 7 in the second factor and the fourth factor load was higher than 0.4 at the same time. By the same method, the second factor analysis, in turn, deleted items 13 and 7. The results showed that the load of all items only on the corresponding factor was above 0.4, which met the requirements of the questionnaire.

According to the contents, the first factor, which included 6 items, demonstrated the degree of depression of parents of children with IH. The second factor, which included 5 items, represented the anxiety level of the parents. The third factor, which included 4 items, represented the psychological balance of the parents. The fourth factor which included 5 items represented the parents’ level of fear for the disease. The fifth factor, including 4 items, represented the degree of stigma of the parents due to the disease. These results (Table 5) demonstrated a well-structured and more reasonable questionnaire that can better fit parents of children with IH.

Cronbach’s α of SAS and SDS were 0.845 and 0.812, respectively, and the correlation coefficients between SAS, SDS, and IH-PSQ were 0.874 and 0.754, respectively (P<0.01), as shown in Table 6.

Reliability analysis

Cronbach’s α of IH-PSQ was 0.957, while Cronbach’s α of 5 factors were 0.941, 0.914, 0.915, 0.849, and 0.839, respectively. The IH-PSQ split-half reliability was 0.971, and the split-half reliabilities of 5 factors were 0.957, 0.879, 0.899, 0.911, and 0.865, respectively (Table 7).

The correlation coefficient of test-retest reliability of IH-PSQ was 0.967, and the correlation coefficient of test-retest reliability of 5 factors was 0.987, 0.947, 0.977, 0.967, and 0.947, respectively (Table 8).

A final, validated version of IH-PSQ is provided in Table 9.

Analysis of influencing factors

According to the survey of the 323 parents of children with IH, the average score of IH-PSQ was 60.20±20.12.

The results of multiple linear regression showed that education level of parents, duration of illness, tumor size, the presence or absence of complications, family monthly income, single or multiple occurrences, and whether to receive treatment entered the regression equation, indicating that these factors will affect the score of IH-PSQ, as shown in Table 10.

Discussion

The key reason that catalyzed this study was the unfortunate phenomena of overtreatment among some children with IH. Overtreatment of infants and young children exists in many countries (20). The reasons for this phenomenon included 2 aspects: doctor’s reasoning, including the fear of accidents (12), lack of corresponding standards (20), and some moral factors (12); the other was parental psychological factors (11,12). However, it was the guardians who ultimately decided the treatment of IH children. It was of positive significance to explore the psychological state of parents of children with IH in the pursuit of better service of children with IH (18).

The tools of investigation were important in this study. At the beginning, some researchers used the quality-of-life questionnaire to assess IH families (16), and emphasized the necessity for a specific questionnaire (16,18). Next, 2 specialized questionnaires appeared: 1 was the IH-QoL (Infantile Hemangioma Quality-of-Life), which was verified by 220 respondents, containing 29 items, investigating the physical condition and social interaction of children with IH and the emotional and psychological functions of the parents of children with IH (21); The other was the Hemangioma Family Burden (HFB), which was verified by 75 respondents, containing 20 items, investigating family life, relationship and work, emotion/feeling, psychology, and disease management of the IH family (22). Their research had a positive effect on the better service of IH families. Researchers from another region of China used the IH-QoL to investigate local IH families and got similar results (23). However, we had noticed that the questionnaire mentioned had some regional limitations. We needed to go through a series of processes such as translation and re verification prior to its application. In addition, we required a questionnaire focusing on the psychological status of parents of children with IH, because it has a great impact on the direction of treatment. However, the two above mentioned questionnaires seemed to focus more on the whole IH family. Based on this, we finally decided to develop a regional questionnaire that met our requirements. We would like others to use it in other areas, subject to re-verification.

It was difficult to name this questionnaire. There was no other single questionnaire that involved all the dimensions mentioned in this questionnaire, namely anxiety, depression, psychological imbalance, stigma and fear of the disease, and the survey participants were parents of children with IH. These dimensions actually involved the emotional, psychological, and mental state of parents. After several rounds of team discussions, we finally chose the name of the Psychologic Status Questionnaire for Infantile Hemangiomas’ parents (IH-PSQ) because it was more inclusive.

As the location of the survey was the outpatient department of the hospital, the information we gathered was more comprehensive. The possible influencing factors of our questionnaire were more than those of the studies we had previously seen, but it was still not a comprehensive overview. The study showed that there were two aspects affecting the score of the questionnaire: parent-driven factors were monthly income and education level. That means that parents with lower education levels and lower family income are more likely to be affected by psychological conditions such as anxiety and depression; disease-related factors were time of illness, tumor size, presence or absence of complications, single or multiple hemangiomas, and whether they had already been treated. That means the longer the illness, the larger the tumor, the more tumors, complications, and no treatment can make the parents more susceptible to depression, anxiety and other psychological states. Clinicians should focus on those factors in order to better communicate with the parents of children with IH. Some of these factors have been previously mentioned, such as the educational level of parents (23) and the size of the tumor (22,23). Some of the reasons that were not mentioned may be related to the different survey tools and the number of survey-related factors. In addition, what we need to know is that the psychological adjustment ability and positive attitude of parents have a very positive significance to relieve stress and the quality of family life (21). This should be advocated clinically and will help many IH families.

There are two main limitations to the study. The sampling was mainly from the patients of the Clinical Research Center of Vascular Abnormalities in Jiangxi Province, China, which was convenient but had systematic errors. We should enlarge the sample size and include different regions and hospitals so as to reduce the systematic error and selection deviation. Also, our goal of reducing IH overtreatment was only the first step, and we did not identify a link between questionnaire scores and overtreatment. The link will become clearer with the increase of the clinical application of the questionnaire, and we will finally achieve the purpose of reducing overtreatment. What we know is that the parents’ psychological distress will be better because of the treatment. When we introduced the treatment of IH to the parents in detail, it was obvious that their psychological state was better, which fully indicated that the change of cognition of the disease could help the parents to relieve their psychological distress.

The verification process of the questionnaire involved 323 families with IH. A questionnaire with 5 dimensions was produced through a series of processes such as item analysis, validity analysis, and reliability analysis. The structure and reliability of the questionnaire was supported by strong evidence. The objective assessment of the psychological status of parents of children with IH will enhance communication between the parents and health care providers, thereby improving information transfer, creating a real opportunity for practitioners to gain better understanding of certain issues brought up by the patients or their families, and avoid overtreatment. Moreover, IH-PSQ may help parents of children with IH to understand their psychological status and seek help from psychiatrists to harmonize their daily life.

Conclusions

The IH-PSQ demonstrated its reliability and validity, and can therefore be used to better understand the multidimensional nature of the psychological status of parents of children with IH, including anxiety, depression, psychological imbalance, sense of shame, and disease fear. Using the verified IH-PSQ, we provided further insight into the psychological status of local parents of children with IH and highlighted potential factors for future focus in assisting families with affected children, which include parents’ own factors (including monthly income and education level) and disease-related factors (including duration of illness, tumor size, the presence or absence of complications, single or multiple occurrences, and whether treatment should be implemented).

Acknowledgments

The investigators would like to acknowledge the junior fellow apprentice who participated in the co-design process. Thanks are extended for the platform provided by the First Affiliated Hospital of Gannan Medical University and Jiangxi Provincial Clinical Medical Research Center for Vascular Abnormalities, and for the technical support provided by Gannan Medical University.

Funding: This project was supported by the Key Project of Natural Science Foundation of Jiangxi Province (No. 20192ACBL20005) and the Youth Project of Jiangxi Provincial Department of Education (No. GJJ201550). In addition, this study was supported by the Jiangxi Provincial Clinical Research Center for Vascular Anomalies and Jiangxi Provincial Key Laboratory of Traditional Chinese Medicine Prevention and Treatment of Hemangioma.

Footnote

Reporting Checklist: The authors have completed the MDAR reporting checklist. Available at https://dx.doi.org/10.21037/tp-21-554

Data Sharing Statement: Available at https://dx.doi.org/10.21037/tp-21-554

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://dx.doi.org/10.21037/tp-21-554). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study involving human participants were approved by the Ethics Committee of The First Affiliated Hospital of Gannan Medical University (No. LLSC-2021062503) and were in accordance with the Declaration of Helsinki (as revised in 2013). All participants voluntarily participated in the study and signed a written informed consent before taking part.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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(English Language Editor: J. Jones)