Frequency of social burden and underage children in neuro-oncological patients

We followed the ethical principles outlined in the 1964 Helsinki Declaration and its later amendments, and all procedures involving human participants in this study were approved by the institutional and local ethics committee (study ID: 2022–2809-Daten, ethics committee of the University Hospital, Jena, Germany). Data from this study will be made available upon reasonable request.

For this study, we retrospectively analysed the data of patients who met the following inclusion criteria:

The exclusion criteria for this study were: (1) Main diagnosis of a spinal tumour or other neuro-oncological tumours than intra-cerebral tumours and (2) incomplete psycho-oncological, palliative care, or quality-of-life screening.

The manuscript has been prepared in accordance with the Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) guidelines, as well as the Equator Network’s recommendations for scientific manuscript preparation (von Elm et al. 2007).

Treatment for brain tumour patients at our neuro-oncological centre is comprehensive and collaborative. We follow a multimodal, interdisciplinary approach that begins with a detailed consultation and evaluation of the patient’s wishes. Our neuro-oncological tumour board, which includes senior specialists in neurosurgery, neuroradiology, neuropathology, neurology, radiotherapy, and haemato-oncology, provides interdisciplinary advice and recommendations based on the guidelines of the relevant European specialist societies (Goldbrunner et al. 2020; Hoang-Xuan et al. 2023; Le Rhun et al. 2021; Soffietti et al. 2017; Weller et al. 2021).

At our centre, patients with benign brain tumours undergo regular clinical and radiological check-ups using MRI every 4 months. For malignant brain tumours, patients receive their first check-up 6 weeks after surgery or radiotherapy completion, and are subsequently monitored every 3 months using both clinical and radiological assessments. In case of special circumstances, control intervals are shortened or additional examinations such as FET-PET or MR perfusion examinations are carried out.

We offered all our inpatients and outpatients to participate in a screening for psycho-oncological issues, palliative medicine needs, and quality-of-life assessment as part of their routine care. An evaluation is conducted at every inpatient and outpatient visit. We use the distress thermometer to screen for psycho-oncological stress and the EORTC-qlq-C30 and -BN20 questionnaires to assess the patient’s quality of life (Rapp et al. 2018; Taphoorn et al. 2010; Schwarz and Hinz 2001). Additionally, we used the NCCN distress thermometer and the core data set of the German Society for Palliative Medicine (DPG) (Bausewein et al. 2005; Stiel et al. 2010; Radbruch et al. 2000a, b; Radbruch et al. 2000a, b).

We retrospectively collected demographic data, such as patients’ diagnoses, age, and gender, as well as information on quality of life, palliative care, and psycho-oncological burden from their charts and questionnaires. For further analysis, quality of life and the global health status was dichotomized into favourable (scores 5–7 on the 7-point Likert scale of the EORTC-qlq-C30 questionnaire) and unfavourable (scores 1–4). The follow-up period lasted until 31 January 2022.

In order to define benign and malignant diseases, we used the World Health Organization (WHO) classification for tumours of the central nervous system (Louis et al. 2021). Based on this definition, we classified grade 3 or 4 tumours according to the WHO classification as being malignant, and WHO grade 1 or 2 tumours as benign (Louis et al. 2021). Cerebral metastases and lymphomas were considered as potentially life-threatening diseases. For some patients with small meningiomas or schwannomas and a watch-and-wait strategy, the diagnosis was made on the basis of neuro-radiological imaging. The criteria of the relevant guidelines and the interdisciplinary assessment in the neuro-oncological tumour board were used as a basis for the classification (Goldbrunner et al. 2016, 2020).

To facilitate further analysis, we divided children into two groups based on their age: minors and adults. We determined the age of majority based on the United Nations (UN) Convention on the Rights of the Child from 1989 and the age limits for majority in most states (United Nations 1989). Specifically, we defined majority as starting from 18 years of age. Upon their initial visit to our neuro-oncological centre, we enquired about the children’s age, and subsequently computed the age at the time of diagnosis.

For continuous data, mean ± standard error of mean was used and for ordinal values, median values and minimum–maximum ranges. Normal distribution of data was assessed using the Shapiro–Wilk test of normality, and were found to be mostly non-normally distributed. To test for a significant difference between two independent groups with non-normally distributed data, the Mann–Whitney U test, a nonparametric test, was employed (Wilcoxon 1947, 1946, 1945; Mann and Whitney 1947). For categorical data, median and 25%–75% percentiles were presented. We chose a significance level α of 5% (0.05) for the present study and performed in total ten statistical evaluations (κ = 11). To adjust for multiple comparisons, Šidák’s correction was applied (α_adjusted = 1–(1–α)^1/κ), and a significance level of < 0.0046 was used (Šidák 1967). P values between 0.05 and 0.0046 were considered indicative of a tendency towards correlation.

From February 2021 to November 2022, our tertiary neuro-oncology centre treated 881 brain tumour patients who met the inclusion criteria, both as inpatients and outpatients, in a total of 3106 consultations. Of these patients, 540 were female (61.3%) and had a mean age of 61 ± 0.5 years (with a range of 16–88 years). The cohort consisted of 339 patients with meningiomas, 220 with gliomas, and 196 with pituitary tumours, among others (Table 1). A total of 228 patients suffered from malignant or potentially life-threatening intracranial tumours; in this cohort, mean age was 60 ± 1 years (with a range of 16–87 years) and 104 patients (46.6%) were female. The mean follow-up was 48.77 ± 1.09 month (range: 0–476 months).

The median distress score was 5 out of 10 for both the entire cohort (25th–75th percentile range 2–7) and for patients with malignancies (25th–75th percentile range 3–7) at the time of initial presentation in our tumour centre. More than three quarters of all patients reported feeling uncertain about their future, and nearly 60% of them reported a worsened outlook on the future (for exact numbers, see Table 2). Among patients with malignancies, 85% felt uncertain about their future, and 50% reported a worsened outlook on the future. The overall health condition and quality of life were rated as unfavourable in 23% and 24% of all patients and 27% and 40% of patients with malignant tumours, respectively.

Out of the total of 675 patients with comprehensive data, 138 individuals (20.4%) disclosed that they resided alone without any family members or companions and 48 out of 251 patients with malignant tumours lived alone. Additionally, 617 patients (which accounts for 80% of all patients, with information available for 773 patients) were in committed relationships, including 229 out of 280 patients with malignancies (81.7%) who had a complete set of information.

Based on responses to the EORTC-qlq-C30 and -BN20 questionnaires, over half of all patients and more than 65% of those with life-threatening tumours indicated that their illness or treatment disrupted their social activities and family life upon first presentation at our tumour centre (for exact numbers, see Table 2). Almost 30% of patients had moderate or severe complaints, as detailed in Table 2. About 27% of all patients (and 31% of patients with malignancies) expressed moderate or major concern that their family life could be disrupted. Approximately 15% of patients (17% of patients with malignancies) faced moderate or major financial difficulties due to their illness.

Out of 881 patients, 61 (6.9%) declined to provide information about their children, as well as 16 (7%) out of 228 patients with malignant diseases. A total of 162 patients of the entire cohort (19.6%) reported having no children. On average, each patient had 1.5 ± 0.04 children, with a range of 0–7 children. At the time of diagnosis, the total cohort had a total of 1083 children. At the time of their presentation to our clinic, patients’ children had a mean age of 33 ± 0.5 years, while at the time of diagnosis of intracranial neuro-oncological disease, their children’s mean age was 30 ± 0.5 years (Fig. 1). At the time of the parent’s tumour diagnosis, 231 patients (28.2%) had underage children.

Among the patients with malignancies, 212 provided complete information about their children. Of these, 177 patients (83.5%) had a total of 318 children at the time of tumour diagnosis, with a mean age of 33 ± 0.9 years (35 ± 0.8 years at the time of presentation to our clinic). Thirty-eight patients with malignancies (17.9%) had a total of 56 underage children at the time of tumour diagnosis, and currently have 53 underage children.

This study aimed to investigate whether underage children with a malignant disease experience a particular psycho-oncological burden. To achieve this, we compared the psycho-oncological burden of patients with malignant disease and underage children to that of patients with malignant disease but no underage children at the time of presentation in our tumour centre (Fig. 2). We found that patients with minor children had more financial worries (median 2 vs. 1, range 1–4; p < 0.0001) but less interference of their disease with social activities (median 2 for both groups, p < 0.0001), less psycho-oncological distress (median 4 vs. 5, range 1–10, p < 0.003), fewer setbacks (median 2 for both groups, range 1–4, p < 0.0001), and better future prospects (median 1 vs. 2, range 1–4, p < 0.0001). They were less limited in doing either work or other daily activities (median 2 for both groups, range 1–4, p < 0.0001) and in pursuing hobbies or other leisure time activities (median 2 for both groups, range 1–4, p < 0.0001). Their general health status and quality of life (median 5 vs. 4, range 1–7; p < 0.0001 for both parameters) were significantly better in week prior to the appointment. Patients with minor children also reported a better family life (median 2 for both groups, p < 0.003), but they had the same level of fear of disruption of family life as patients without minors (median 2 for both groups, p < 0.5).

We acknowledge several limitations of our present analysis. Firstly, all the data were derived from a retrospective, single centre study. Secondly, sample size calculation was lacking, but as a retrospective cohort was identified, post-hoc sample size calculation would be unusual. Future prospective studies should include a sample size calculation to draw more confirmative conclusions. Since the aim of the analysis was to determine the frequency of social and family stress on neuro-oncological patients, we have not performed a multivariate analysis, created a clinical prediction model for quality of life, nor have we assessed the burden carried by underage children. As our aim was to evaluate the impact of brain tumours on social functioning and distress, we used frequencies of each single item of the EORTC-qlq-C30 questionnaire and not the sum scores or subscore for further analysis (Aaronson et al. 1993). It should also be noted that we did not gather data on additional living conditions, e.g. single-parent households or individuals living alone without children. Additionally, the age of children might correlate with cancer patient's burden, e.g. that patients with younger children might suffer from higher distress; we have not accessed the impact of children's age on the distress of neuro-oncological parenting patients. These unexamined parameters could potentially influence the results as well. The potential younger age of patients with underage children might also influence the results, e.g. difference of age as reason for all the differences reported. Moreover, the present analysis compromised a very heterogeneous patient collective. The estimation of burden of parents with malignant diseases and minors based on 38 patients out of an entire cohort of 881 brain tumour patients and 228 with malignancies. However, it was the goal of our research to also determine the frequency of underaged children and social impairment caused by brain tumours. We did not analyse if and how different diagnoses and treatment protocols may affect the assessed parameters such as quality of life, social activities, or family life. Likely, the different disease stages, such as following first-line treatment, during follow-up, or end-of-life care, may differently affect psycho-oncological and social burden. The data arose from a tertiary neuro-oncological centre in Central/Eastern Germany. The geographical area from which we drew our patients is partially rural, resulting in considerable distances between patients' homes and the neuro-oncological center. The region was part of the former German Democratic Republic and a former communist country with a sometimes-lower socio-economic status than other regions of Europe. Thus, findings in this population cohort are not necessarily transferable to other regions in Europe and worldwide. Again, regional, cultural factors, and family ties may play an important role in quality of life, family, and social life. Fifthly, in this study, we pooled the quality of life at different points in the course of the disease, as opposed to tracking quality of life, social and family life over time in individuals, as done in other studies (Ernst et al. 2012). Certainly, the timing point of the illness will have an impact on social and family life, as well as quality of life. Sixth, we examined the social and family burden of cancer patients, particularly those with minor children. We did not differentiate whether the parent was the mother or the father. However, affected mothers and fathers may have different levels of burden. Finally, while our study focused on evaluating the social life, family functioning, and quality of life of neuro-oncological patients with and without dependent children, we did not assess the distress experienced by the children themselves. To gain a more comprehensive understanding of the impact of neuro-oncological diseases on families, future studies should examine the burden of the disease on both the patients and their children. Such studies could shed light on the unique challenges faced by families affected by neuro-oncological diseases and help inform the development of more effective support and care strategies.