With 5-year survival rates among pediatric brain tumor patients approaching 75% [1] , physicians now face the challenge of enhancing psychosocial outcomes. Both children undergoing treatment for brain tumors and survivors require information regarding their diagnosis, treatment, follow-up care, and late effects [2] [3] . Continuous and honest communication is important to help these children build trusting relationships with medical professionals and participate in treatment and health management [4] [5] . The first step in facilitating this open communication is ensuring that medical professionals and parents provide children with accurate and appropriate information regarding their diagnosis [6] .

Although several studies in the past 30 years have discussed the degree of information that should be provided to children with life-threatening diseases [3] [6] - [9] , several factors must be considered for those with brain tumors. First, when children are diagnosed with a brain tumor, they might appear unable to comprehend detailed information due to symptoms such as headache, neurological dysfunction, and cognitive impairment [10] . Second, as several types of brain tumors are associated with high mortality [11] , information might be withheld out of concern for the child’s psychological well-being. In fact, a marked proportion of pediatric oncologists (14.4%) reported that they only revealed the diagnosis to children with a good prognosis [8] . Third, children with brain tumors showed more procedural and treatment anxiety than those with other types of cancer [12] . Children with brain tumors might receive limited information regarding their disease and therefore experience a higher degree of anxiety.

In a Japanese study conducted in parents of children with hematological and solid cancer in the 1990s, the majority of mothers (53/57) concealed the diagnosis to prevent their child from experiencing stress [13] . The remaining mothers (4/57) revealed the diagnosis to provide peace of mind (e.g. before stem cell transplantation [SCT]) [13] . Parents’ will must be considered when informing children of their diagnosis [8] [14] . Useful references for parents deciding to disclose or conceal their child’s diagnosis are the proportion of informed children, conditions for which a higher or lower level of information is provided, and whether informed children are more anxious or reassured than uninformed children. To provide further references for parents, we developed a study protocol based on the hypotheses that 1) children with brain tumors are provided information on their disease based on age and intellectual level; 2) a high proportion of information regarding diagnosis is provided to children treated at specific institutions, with a good prognosis, receiving specific treatment, and whose parents are without anxiety/depression; and 3) informed children are not more anxious, worried, or incommunicative than uninformed children.

Here, we clarified the degree of information provision to children with brain tumors regarding their diagnosis, factors relevant to this provision, and the relationship between this provision and psychosocial outcomes.

This study was conducted in combination with the development of the Japanese version of the PedsQL Brain Tumor Module [15] . We used the data to focus on how parents perceive the provision of information regarding diagnosis to their children.

Here, we report the degree of information provision to children with brain tumors in Japan and describe factors influencing this provision. Age and intellectual disability were the only factors relevant to the degree of information provision. Informed children did not appear more anxious, worried, or incommunicative than uninformed children. Quite the contrary, more-informed children showed a higher degree of treatment reassurance.

To our knowledge, this is the first report on the degree of information provision to children with brain tumors.

AA, P-value with analysis of covariance (ANCOVA) adjusted by age (“age-adjusted”); PedsQL, Pediatric Quality of Life Inventory; SD, standard deviation; Higher scores indicate higher quality of life (lower anxiety or more communication). Example items of each scale were “I get scared about having needle sticks” (Procedural Anxiety), “I get scared when I am waiting to see the doctor” (Treatment Anxiety), “I worry that my disease will come back or relapse” (Worry), and “It is hard for me to tell the doctors and nurses how I feel” (Communication). Missing data were excluded.

Parents of children aged 5 years or older provided at least a moderate level of information regarding their disease. The main reason for non-disclosure of disease by parents was concern of shocking children psychologically [14] . Our results might be used as a reference when deciding to disclose or conceal information to their children, particularly those concerned about their child being too young to cope with such information.

The majority of parents of children aged 4 years or younger provided a low level of information regarding their disease. Based on free descriptions, most information provided appeared to regard concrete and visible procedures, out of consideration for potential fear and anxiety the child might experience. Parents explained their intentions, particularly in cases where they might require their child’s cooperation or when their child was hurt. However, the majority of parents did not intend to explain the diagnosis to their child. Although more information, including disease name, was provided as age increased, there was no defined cutoff age at which certain information was deemed acceptable to disclose. Even for high-school students, approximately half did not know the name (“brain tumor”) of their disease. Parents often avoided providing accurate information about their child’s disease, instead explaining in an easy-to-understand manner (e.g. “a boil in the head”).

This unspoken policy of secrecy regarding disease is more common in Japan than the UK and USA [9] [27] . For example, in the USA, during the 1990s the majority (75%) of parents of young children with cancer (mean age, 5.45 years [SD 1.00]) reported that their children were aware of their diagnosis [7] . In contrast, in Japan, during the 1990s the majority of (64%) pediatric oncologists had never revealed the diagnosis to their patients [8] . Several studies have since clarified that the pattern of information provision differs between cultures [9] [28] - [30] . Similar to Japan, populations in several countries and areas such as Korea [31] , China [32] , Taiwan [33] , Iran [30] , and Italy [34] exhibited similar trends regarding the level of information on cancer diagnosis provided to “fragile” patients. The free descriptions from parents in this study demonstrate one way of talking to children about their disease in an appropriate manner.

AA, partial correlation adjusted by age of child; CA, correlation coefficient corrected for attenuation.

Our findings describe the current degree of information provision to children with brain tumors in Japan. In principle, children must be provided all information regarding their disease with consideration for their age and developmental level [5] [35] [36] . In addition, to encourage the provision of information to children with brain tumors, cultural background should be considered.

The age and intellectual disability of children were identified as factors relevant to the degree of information provision, findings consistent with a previous study showing that age was associated with disclosure of diagnosis to children with HIV/AIDS [37] . Other factors, such as child’s gender and socioeconomic status, were selected in the predictive model but were not significantly associated with disclosure, unlike the previous study [37] . In the present study, specific treatments (SCT and neurosurgery) increased information provision to children and were selected as considerable factors using the best-subset procedure. However, the influence of specific treatment on information provision was minor. Throughout the course of treatment, parents and physicians had consultations regarding information provision to their child. These findings indicate the consistent attitude of parents and physicians regarding the provision of information irrespective of tumor type and treatment course. Of course in clinical practice, the degree of information provision is decided case-by-case [8] . On age bracket stratification, children aged 12 years or older appeared to have been informed about their diagnosis more often on a case-by-case basis than younger children. There might also be instances of case-by-case decisions beyond the studied factors. For example, unstudied factors such as the requests of children might influence the degree of information provision.

Disclosure of a cancer diagnosis to children varies between hospitals [27] , and parents’ will regarding disclosure is affected by that of the physician [13] [14] . We therefore found it surprising that the recruited institutions (six hospitals and one association) were located in different regions yet had no effect on information provision. The recruited hospitals are considered the location where children received their treatment, even for children currently receiving no treatment, as most pediatric brain tumor survivors in Japan receive follow-up at the same hospital continuously, for an average of 15 years after diagnosis [38] . Further, the CCAJ is the largest organization supporting children with cancer and their families and provides various means of support to members (parents), including counseling on how to inform children of their disease [39] . These facts suggest that each recruiting institution conducted the best possible practice for information provision and open communication to children with brain tumors. We requested that this study be conducted with leading physicians of treatment for pediatric brain tumors. The findings of this study might therefore reflect a higher level of information provision to children than other institutions in Japan.

Here, we confirmed that informed children are not more anxious, worried, or incommunicative than uninformed children, a finding consistent with adult cancer patients in China [40] . Further, informed children were less anxious about treatment, hospitals, and physicians than uninformed children. The disclosure to children of information regarding their disease appears to contribute to their trust of medical and health care professionals. To our knowledge, this is the first empirical evidence of a relationship between information provision to children with cancer and psychosocial outcomes. This finding might help parents communicate their child’s diagnosis to provide them both peace of mind.

The reverse relationship between information provision to children and anxiety requires discussion, as less information might have been provided to children perceived as anxious. However, two results of the present study support a directional relationship between information provision and anxiety of children. First, this study measured trait anxiety, which is a stable characteristic of children [22] [23] , and confirmed that trait anxiety was not related to the degree of information provision. This finding therefore suggests that parents determining the level of information to provide to children based on their trait anxiety is unlikely. Second, we confirmed that the relationship between information provision to children and their self-reported anxiety was clearer than that between information provision to children and parent-reported anxiety of children. If parents were indeed determining the level of information they should provide to a child based on the child’s level of anxiety, then perceived anxiety of children should be more related to provision of information than child-reported anxiety; however, this was not the case. Therefore, a directional relationship between information provision and psychosocial outcomes of children might exist, rather than reverse relationship.

Although ethically speaking diagnosis information should be provided to children, obtaining empirical evidence on outcomes of such information provision is difficult, as controlled intervention studies are unethical. While a proportion of previous cross-sectional observational studies in adult cancer patients noted relationships between information provision to patients and their health outcomes [40] , the possibility of a reverse relationship (relationships between patients’ health outcomes to the degree of information provision) could not be discounted. We therefore consider the parent-child paired data to be a methodological strength of the present study that enables the acquisition of strong evidence regarding the effect of information provision on the psychosocial outcomes of children, despite this being a cross-sectional study.

Provision of information by parents might influence psychosocial outcomes of children pleiotropically. When parents provide information regarding diagnosis to their child, the psychosocial outcomes of the child might be improved due to the acquirement of new knowledge of themselves, confidence in the ability to manage their physical and psychosocial complaints, or relief by being informed. Furthermore, of note, the relationship between parents’ provision of information and children’s psychosocial outcomes is not necessarily mediated by children’s understanding or recall of such information. If, even after receiving information regarding their diagnosis, children do not understand (or forget) the information, their psychosocial outcomes may still be improved due to parents’ relief in having explained the situation to the child, heightened family functioning due to parents’ peace of mind, or parents and medical staff being more honest and open and therefore in a better position to care for children. While the present study somewhat clarified the pleiotropic influence of provision of information perceived by parents on the psychosocial outcomes of children, further research is required to clarify the process and mechanism by which this influence is enacted.

Several other limitations to the present study warrant mention. First, our ability to generalize the data is limited. While the response rate at hospitals was high, rates at the CCAJ are unknown, as several hundred families were notified of this study through the CCAJ, including those not eligible to participate. Parents might not have enrolled to prevent their children from any possible negative psychosocial outcomes due to participation in this study. Further, the findings of this study might reflect a higher level of information provision to children under the best possible practice than that in other institutions in Japan.

Second, our results should be interpreted with caution, as this was a cross-sectional study conducted at one time point. When children were informed of their diagnosis and how they experience current psychosocial outcomes after information provision remains unknown. We also measured the psychological distress of parents using the K10 score, which only indicates their current condition. Past mental conditions or medical history of parents could influence their desire to provide information to children regarding their disease.

Third, our results should also be interpreted with caution, as the degree of information was determined according to simple and subjective questions. Parents were only questioned regarding the diagnosis (disease name) and condition of their child, and information provided in the present study does not include the prognosis of the disease. In addition, what was considered “little information” by some parents may have been considered “moderate information” by others. However, these questions could be considered effective indicators of information provision in context, as statistically significant results were obtained even when the question had some degree of error and no parents selected the fourth response (instead providing other degrees of information). The question and response choices in the present study matched those of parents in a clinical setting in Japan [41] , where an important step is that children were provided information regarding disease condition (difference between the first and second response) and regarding disease name (difference between the second and third response.

Fourth, we did not conduct an a priori sample size calculation because the present study was conducted in parallel with a related study that has a predetermined sample size [15] . This study indicated that the influence of disease and treatment factors on information provision was minor, if present at all. A larger sample might clarify additional relationships between relevant factors and information provision. Specific treatments and the anxiety or depression of parents can be considered, among others.

In the future, a qualitative study should be conducted to explore types of practice implemented in leading hospitals and analyze how this can be applied to other hospitals. How information is provided (by parents, by physicians, by nurses, or by them role sharing) is also important. A more detailed survey should then be conducted to clarify the contents of “provided a moderate level of information,” which would be useful for parents and medical professionals when explaining the disease and treatment to children with brain tumors.

Here we report the degree of information provision to children with brain tumors in Japan in 2008. Parents of children with brain tumors provided information dependent on the child’s age and intellectual level. Children aged 5 years or older received information on a moderate or detailed level regarding their disease. Regarding ethnical/cultural considerations, children should not uniformly receive information regarding their diagnosis. The provision of information did not make children more anxious, worried, or incommunicative. This study provides evidence that information provision to children regarding their disease is the first step in fostering open communication with medical professionals.

This study was supported by a Grant-in-Aid for Pediatric Cancer Treatment and Research from the CCAJ 2008 and a Grant-in-Aid for Cancer Research from the Ministry of Health, Labour and Welfare of Japan (No. 18-14) 2008.