This study design is a case-control study. In our country, individuals under the age of 19 years are evaluated and treated in the departments of pediatrics and department of child and adolescent psychiatry according the definition of World Health Organization (WHO) .
All patients between the ages of 6–18 years, who were followed up in our hospital, were included in this study. Some patients were excluded from the study because their parents could not fill the scales completely or could not complete the evaluation process (n = 7). As a result, a total of 20 children and adolescents with hemophilia A or B aged were included. All of the patients were boys. All of the children and adolescents who were enrolled in this study had been managed their treatment at the hemophilia centre of children hospital. Additionally, 20 male subjects with typical developing controls (TD) were recruited as the control group. TD group was selected randomly from pediatric outpatient clinics who do not have any chronic illness. Inclusion criteria for both hemophilia group and TDs were (i) age between 6 and 16 years, (ii) intelligence quotient (IQ) score higher than 70, (iii) children living in their own family house and attending a normal primary school, (iv) no history of head injury with loss of consciousness, and (v) no neurological or other serious medical diseases.
Instruments for assessment to children and adolescents
Firstly, to determine psychiatric disorders of children and adolescents, a semi-structured interview: Kiddie-Schedule for Affective Disorders and Schizophrenia, present and life time version (K-SADS-PL) was applied to parents by a specialist of child psychiatry. It screens child and adolescent pschiatric disorders and symptoms. At the end of the interview, it is determined whether there is any disorder according to the diagnostic criteria .
Child Depression Inventory (CDI) was used. It includes 27 items and each item scores as 0,1, or 2, and cutoff score of inventory for depression is 19 .
The Spielberger State-Trait Anxiety Inventory (STAI) consists of two questionnaires of 20 items each. The first questionnaire measures state anxiety (how one feels at the moment) and the second, trait anxiety (how one generally feels). Each item scores between 1 to 4, and the greater scores show the greater anxiety .
KINDLRQuestionnaire for Measuring Health-Related Quality of Life in children and adolescents was used. It could be completed both by children/adolescents and by their parents. Three versions of the KINDLR questionnaire are available as self-report measures for different age groups: Kiddy-KINDLR for children aged 4 to 7, Kid- KINDLR for children aged 8 to 12, and Kiddo-KINDLR for adolescents aged 13 to 16. The KINDLR questionnaire consists of 24 likert-scaled items associated with six dimensions: physical well-being, emotional well-being, self-esteem, family, friends, and everyday functioning (school or nursery school/kindergarten). All versions of the KINDLR contain an additional sub-scale entitled “Disease”, whose items can be completed in case of prolonged illness or hospitalisation .
Instruments for assessment to mothers
Beck Depression Inventory (BDI) was used. It is a 21-item self-report questionnaire in which each item consists of four statements indicating different levels of severity of a particular symptom experienced over the past week. Participants rate each item on 4-point scale ranging from 0 (absent) to 3 (severe), and total scores range from 0 to 63 .
Beck Anxiety Inventory (BAI) is a 21-item self-report measure that assesses level of anxiety and evaluates physical, emotional, and cognitive aspects of anxiety as well as fear of losing control. Total scores range from 0 to 63 and high scores indicate more severe anxiety symptoms .
The Burden Interview is a 22-item instrument to assess carers’ burden associated to the patient’s functional and behavioral disability and to the situation at home. The 22 items reflect respondent’s areas of concern such as health, social and personal life, financial situation, emotional well-being, and interpersonal relationships. The greater the total score, the greater the burden .
The last scale for mothers was KINDL parent form. There are two parents’ versions: Kiddy-KINDLR (4–7 years) and parents’ version of the KINDLR for 8 to 16 years old children and teenagers .
The interviews were performed during the usual visits of the patients to the hemophilia clinic. Firstly, the patients and their parents were interviewed together, and sociodemographic data was collected. Then, while children and adolescents were completing their scales, a child and adolescent psychiatry specialist applied to parents with the K-SADS-PL. After the KSADS-PL, the mothers completed their scales.
The Burden Interviews are especially related to caregiving for disabled persons so we did not give for control groups’ parents different from the patient group.
This investigation was approved by the Ethical Committee of Erciyes University (2016\501). Written consent was obtained from parents, and verbal assent was requested from children and adolescents to participate.
In the two-way paired t test, it was calculated to include 16 people that is enough to determine the differences between the scales at the level of 0.05 significance (α) with 80% power. In our study, it was planned to include 20 patients in the appropriate age range with follow-up in our department. Calculation was made with G * Power 188.8.131.52. package program. We used Shapiro-Wilk test to analyze homogeneity of variables. Among-group differences on demographic variables were analyzed using independent samples t tests. Mann-Whitney U test were used for non-homogenous variables. Associations between categorical variables were examined using chi-square analysis. Spearman and Pearson correlation tests were used for assesment of correlation. Logistic and multiple regression analyzes were also used. Data analysis was performed using SPSS 24.0, and p values < 0.05 were considered statistically significant.