Psychological Distress and Sleep Disturbance Throughout Thyroid Nodule Screening, Diagnosis, and Treatment.

CONTEXT: Many controversies exist regarding screening and treatment of thyroid cancer (TC), especially papillary thyroid microcarcinoma (PTMC). OBJECTIVE: The aim of this study was to evaluate patients' psychological distress and sleep disturbance throughout thyroid nodule (TN) screening, diagnosis, and treatment. METHODS: A total of 2834 participants (1153 participants with TNs) were enrolled during the screening phase, and 1105 individuals with TNs (87 individuals with TC) were enrolled during the diagnosis phase. Of the 87 TC patients, 66 underwent immediate operation (OP), and 21 patients with PTMC opted for active surveillance (AS). Four validated scales were applied to quantify the outcome indicators at prescreening, postscreening, postdiagnosis, and posttreatment. RESULTS: Higher psychological distress and sleep disturbance were found postscreening than prescreening in subjects with TNs, but no differences in those without nodules. Compared with postscreening, higher scores of psychological distress and sleep disturbance were identified in patients with suspicious TC treated with fine needle aspiration (FNA) or with AS. Lower psychological distress and sleep disturbance were noted for patients with benign nodules than for TC patients. OP for TC, especially PTMC, did not alleviate psychological distress or sleep disturbance compared with the same parameters in patients who underwent AS. CONCLUSION: Based on the findings of impaired psychological health and sleep quality, screening for TNs in adults who show no symptoms should be performed with caution. Psychological distress and sleep disturbance should also be taken into consideration when FNA is performed for suspected TC or OP for papillary thyroid cancer, especially PTMC.

The quality of life in papillary thyroid microcarcinoma patients undergoing lobectomy or total thyroidectomy: A cross-sectional study.

OBJECTIVE: Papillary thyroid microcarcinoma (PTMC) has a good prognosis and a long survival time, surgery is the common treatment including total thyroidectomy (TT) and unilateral lobectomy (LT), but recent studies showed that TT does not show an advantage over LT for PTMC in preventing cancer recurrence and reducing mortality. Given this, the health-related quality of life (HRQoL) has become one of the important factors that physicians must consider when making treatment decisions. The aim of this study was to compare the HRQoL of patients between undergoing TT and LT. METHODS: From October 2019 to December 2019, 69 PTMC patients were enrolled in our study, including 34 in the LT group and 35 in the TT group, respectively. We used three questionnaires which included the 36-item short-form health survey (SF-36), thyroid cancer-specific quality of life (THYCA-QOL), and Fear of Progression Questionnaire-Short Form (FoP-Q-SF) for each patient to evaluate their scores of HRQoL. RESULTS: According to the SF-36, the scores of the domain for the role limitation due to physical problems, emotional problems, and social function (RP, RE, and SF) as well as Physical Component Summary (PCS) and Mental Component Summary (MCS) showed a significant negative linear association between the LT group and TT group: RP (coefficient [coef]: -33.953 [confidence interval (CI) -51.187 to -16.720], p < 0.001, RE (coef: -21.633 [CI -39.500 to -3.766], p = 0.018), SF (coef: -10.169 [CI -19.586 to -0.752], p = 0.035)and PCS (coef: -10.571 [CI -17.768 to -3.373], p = 0.005), MCS (coef: -10.694 [CI -19.465 to -1.923], p = 0.018). The THYCA-QOL showed that the scores of the TT group were higher than that of the LT group in the problem of scar (coef: 16.245 [CI 1.697 to 30.794], p = 0.029 according to the multivariate analysis), suggesting a higher level of complaint in the TT group. There was no statistically significant difference in the scores of FoP-Q-SF between the two groups. CONCLUSIONS: In patients with PTMC, LT offers an advantage over TT in terms of HRQoL, which supports the role of LT as an alternative strategy to TT.

Factors affecting online health community participation behavior in patients with thyroid cancer.

Globally, cancer patients obtain much of their disease information online. Online health communities allow patients to share questions and information about diseases. However, there have been few studies on the factors affecting online health community participation behavior in cancer patients. Online social networking is associated with mental health problems, and patients with thyroid cancer experience high levels of distress, anxiety and depression. The purpose of this study was to investigate factors associated with use of online health communities by patients with thyroid cancer to understand the characteristics of patients participating in such online communities. A questionnaire survey was completed by 114 thyroid cancer patients admitted for surgery at a general hospital in Seoul, Korea. General characteristics, clinical characteristics, attitude toward cancer, distress, and anxiety and depression scores of patients who joined an online health community (user group) and patients who did not (non-user group) were compared. The factors affecting online health community participation were education (p = 0.049), tumor size (p = 0.010), attitude toward cancer (p = 0.022), and anxiety and depression (p = 0.021). The average score of satisfaction with the online health community was 4.25 of 5. The user group had larger tumors, a high awareness of the risk of thyroid cancer, and high levels of anxiety and depression. Patients who actively used the online health community have relatively larger cancer size and had higher levels of mental stress. As such patients are often very anxious and depend heavily on the gathered information, the quality of this information is important. Healthcare professionals need to develop appropriate interventions for patients participating in the online health community.

Quality of Life in Patients with Papillary Thyroid Microcarcinoma According to Treatment: Total Thyroidectomy with or without Radioactive Iodine Ablation.

BACKGROUND: Recently, there has been some controversy regarding the role of radioactive iodine (RAI) ablation in the treatment of low-risk differentiated thyroid carcinoma (DTC), especially papillary thyroid microcarcinoma (PTMC). This study aimed to compare quality of life (QoL) parameters between patients with PTMC who underwent total thyroidectomy (TT) alone and those who underwent TT with RAI ablation. METHODS: In this cross-sectional study, patients with PTMC who underwent TT with/without RAI remnant ablation were prospectively enrolled between June 2016 and October 2017. All patients completed three questionnaires: the 12-item short-form health survey (SF-12), thyroid cancer-specific quality of life (THYCA-QoL) questionnaire, and fear of progression (FoP) questionnaire. RESULTS: The TT and TT with RAI groups comprised 107 and 182 patients, respectively. The TT with RAI group had significantly lower serum thyrotropin (TSH) levels than the TT group. However, after matching for TSH levels between the groups (n=100 in both groups), there were no significant differences in baseline characteristics. According to the SF-12, the score for general health was significantly lower in the TT with RAI group than in the TT group (P=0.047). The THYCA-QoL also showed a significant difference in the "felt chilly" score between groups (P=0.023). No significant differences in FoP scores were observed between the groups. CONCLUSION: Patients with PTMC who underwent TT with RAI ablation experienced more health-related problems than those managed with TT alone. These findings support the idea that RAI ablation should be carefully considered in patients with low-risk DTCs.

Longitudinal Assessment of Quality of Life According to Treatment Options in Low-Risk Papillary Thyroid Microcarcinoma Patients: Active Surveillance or Immediate Surgery (Interim Analysis of MAeSTro).

Background: In this ongoing multicenter prospective cohort study on active surveillance (AS) in low-risk papillary thyroid microcarcinoma (PTMC), we aimed to compare the quality of life (QoL) of participants based on their choice of treatment, that is, AS or immediate surgery (OP). Methods: QoL of 203 participants who chose AS and 192 participants who underwent OP was evaluated using a thyroid-specific QoL questionnaire at diagnosis and during follow-up (median 8 months). Results: The mean ages of the participants in the AS and OP groups were 47.3 ± 11.7 and 45.6 ± 10.5 years (p = 0.138), respectively, and the mean tumor sizes were 5.7 ± 1.6 and 6.5 ± 2.1 mm (p = 0.065), respectively. At baseline, significantly better psychological health (7.1 ± 1.3 vs. 6.8 ± 1.6, p = 0.023) and overall health (6.8 ± 1.2 vs. 6.5 ± 1.3, p = 0.018) were observed in the AS group than in the OP group. During follow-up, significantly better physical (7.9 ± 1.1 vs. 7.4 ± 1.2, p < 0.001), psychological (7.4 ± 1.3 vs. 6.9 ± 1.6, p = 0.004), and overall health (6.9 ± 1.0 vs. 6.5 ± 1.1, p = 0.002) were observed in the AS group than in the OP group, whereas spiritual health was comparable between the two groups. Compared with the AS group, the OP group experienced more fatigue, changes in voice and appearance, less satisfaction, and low fear of recurrence. The self-assessed financial burden was similar at baseline and follow-up in both groups. Conclusion: The QoL of PTMC patients is different according to the type of treatment. Better psychological health at baseline and physical and psychological health at follow-up were observed in the AS group than in the OP group. However, studies with longer follow-up periods are needed.

Quality of Life in Patients with Papillary Thyroid Microcarcinoma Managed by Active Surveillance or Lobectomy: A Cross-Sectional Study.

Background: Active surveillance (AS) is recommended as an alternative to immediate surgery in patients with papillary thyroid microcarcinoma (PTMC), but the impact of AS on quality of life has not been reported. The aim of this study was to compare quality-of-life parameters in patients with PTMC under AS versus those who underwent lobectomy (LB). Methods: In this cross-sectional study, patients with PTMC were prospectively enrolled between June 2016 and October 2017. All patients completed three questionnaires: the 12-item short-form health survey, thyroid cancer-specific quality of life, and fear of progression. The results were compared after adjusting for age, sex, and serum thyrotropin levels. Results: The AS group comprised 43 patients, and the LB group comprised 148 patients. According to the 12-item short-form health survey questionnaire, the score for role limitations due to emotional problems showed a significant negative association between the groups (coefficient [coef]: -7.71 [confidence interval (CI) -15.26 to -0.16], p = 0.045). The thyroid cancer-specific quality of life questionnaire also showed statistically significant differences between the groups with respect to three scores: neuromuscular problems (coef: 4.99 [CI 0.63-10.62], p = 0.020), throat/mouth problems (coef: 5.28 [CI 0.18-10.38], p = 0.043), and scar problems (coef: 9.34 [CI 4.38-14.29], p < 0.001), suggesting a higher level of complaint in the LB group than in the AS group. No significant differences in fear of progression scores were seen between the two groups. Conclusions: Patients with PTMC underwent LB experienced more health-related problems than those managed by AS. These findings support the role of AS as a reasonable management option for patients with PTMC.

Papillary Thyroid Cancer: The Good and Bad of the "Good Cancer".

BACKGROUND: Papillary thyroid cancer is often described as the "good cancer" because of its treatability and relatively favorable survival rates. This study sought to characterize the thoughts of papillary thyroid cancer patients as they relate to having the "good cancer." METHODS: This qualitative study included 31 papillary thyroid cancer patients enrolled in an ongoing randomized trial. Semi-structured interviews were conducted with participants at the preoperative visit and two weeks, six weeks, six months, and one year after thyroidectomy. Grounded theory was used, inductively coding the first 113 interview transcripts with NVivo 11. RESULTS: The concept of thyroid cancer as "good cancer" emerged unprompted from 94% (n = 29) of participants, mostly concentrated around the time of diagnosis. Patients encountered this perception from healthcare providers, Internet research, friends, and preconceived ideas about other cancers. While patients generally appreciated optimism, this perspective also generated negative feelings. It eased the diagnosis of cancer but created confusion when individual experiences varied from expectations. Despite initially feeling reassured, participants described feeling the "good cancer" characterization invalidated their fears of having cancer. Thyroid cancer patients expressed that they did not want to hear that it's "only thyroid cancer" and that it's "no big deal," because "cancer is cancer," and it is significant. CONCLUSIONS: Patients with papillary thyroid cancer commonly confront the perception that their malignancy is "good," but the favorable prognosis and treatability of the disease do not comprehensively represent their cancer fight. The "good cancer" perception is at the root of many mixed and confusing emotions. Clinicians emphasize optimistic outcomes, hoping to comfort, but they might inadvertently invalidate the impact thyroid cancer has on patients' lives.

Measurement and Variation in Estimation of Quality of Life Effects of Patients Undergoing Treatment for Papillary Thyroid Carcinoma.

BACKGROUND: Thyroid cancer incidence is increasing. The effect of diagnosis and treatment on health-related quality of life (HRQoL) is an essential variable in the absence of a change in life span for the majority of patients. HRQoL instruments, with data useful for between-disease comparisons, are being increasingly used for health policy and outcomes evaluation. Variation exits among the instruments based on the impact of a specific disease. We assessed which of four well-validated, preference-based surveys detect changes in health and clinical intervention in patients diagnosed with papillary thyroid cancer (PTC). METHODS: Four commonly used HRQoL questionnaires (Short Form-12v2® [SF6D], EuroQol-5D [EQ5D], and Health Utilities Index Mark 2 and 3 [HUI2, HUI3]) were administered to patients with the diagnosis of PTC at three perioperative time points during the first year of treatment. Clinicopathological and treatment course data were assessed for HRQoL impact including complications from surgery, re-operation for persistence/early recurrence, and adjuvant radioactive iodine treatment. We compared standard metrics, including ceiling effect, intraclass correlation coefficient, effect sizes, and quality-adjusted life-years between the four instruments. RESULTS: Of 117 patients, 27% had a preoperative diagnosis of anxiety or depression, 41% had regional lymph node metastases, three had distant metastases and 49% underwent adjuvant radioactive iodine treatment. The ceiling effect (i.e., proportion with a perfect score) was greatest with EQ5D and least with SF6D. Index scores ranged from 0.77 (SF6D) to 0.90 (EQ5D). All scores declined at two weeks postoperatively and returned to pretreatment levels at six months. The SF6D was the only instrument to exceed the conventional minimally important difference between all three time points. Quality-adjusted life-years were as follows: SF6D, 0.79; EQ5D, 0.90; HUI2, 0.88; and HUI3, 0.86. CONCLUSIONS: Our results reflect the general good health of PTC patients. The effect on quality of life is primarily related to emotional and social impacts of treatment. The results support the measurement of a similar underlying construct, although variation in detecting changes in health exists between the instruments. Of the instruments assessed, the SF6D is the most responsive to treatment effects and should be utilized in future economic analyses in this patient population.

Quality of life, body mass index, and physical activity among uterine cancer patients.

OBJECTIVE: The purpose of this study was to assess the independent and joint effects of body mass index (BMI) and physical activity (PA) on overall quality of life (QoL) in survivors of uterine cancer. METHODS: We conducted a survey among uterine cancer patients who received curative therapy at the University of Pennsylvania between 2006 and 2010. Surveys assessed the weight, height, PA (college alumnus survey), and QoL (Functional Assessment of Cancer Therapy-Gynecologic Oncology Group). RESULTS: The response rate to the survey was 43%. Among 213 patients, the mean (SD) BMI was 31.1 (8.9) kg/m, and 48% reported greater than or equal to 150 min·wk of PA. Higher BMI was independently associated with poorer overall QoL (P = 0.050), including physical (P = 0.002) and functional well-being (P = 0.008). Higher min·wk of PA was not independently associated with any QoL outcome. However, among patients who engaged in greater than or equal to 150 min·wk of PA, the negative association between BMI and overall QoL was attenuated (P = 0.558), whereas among patients who engaged in less than 150 min·wk of PA, the negative association between BMI and overall QoL persisted (P = 0.025). Among patients who engaged in greater than or equal to 150 min·wk of PA, the negative association between BMI and physical and functional well-being was attenuated (P = 0.765 and P = 0.284), whereas among patients who engaged in less than 150 min·wk of PA, the negative association between BMI and physical and functional well-being persisted (P < 0.001 and P = 0.010), respectively. CONCLUSIONS: Body mass index is associated with poorer QoL among uterine cancer patients. The findings from this cross-sectional study are consistent with the hypothesis that endometrial cancer survivors who are able to perform 150 min/wk of PA may be protected from the negative effects of BMI on QoL.

Quality of life and anxiety in adolescents with differentiated thyroid cancer.

CONTEXT: Adolescents with differentiated thyroid cancer (DTC) require lifelong monitoring with a high possibility of reoperation or radioactive iodine. Although adult DTC survivors have similar or slightly worse quality of life (QOL), this has not been evaluated in the pediatric population. OBJECTIVE: Our objective was to compare QOL and anxiety in adolescents with DTC to patients with acquired autoimmune hypothyroidism. DESIGN, SETTING, AND PATIENTS: In this cross-sectional pilot study, three validated questionnaires were administered to 16 adolescents with DTC and 16 controls for assessment of QOL and anxiety levels. These included teen and parent PedsQL, Multidimensional Anxiety Scale for Children, and Coddington Life Events Scales for Adolescents. The contribution of age, time since diagnosis, and biochemical variables were compared with the outcome measures. RESULTS: There were 16 DTC patients (seven males); 13 had papillary carcinoma, one had follicular carcinoma, and two had mixed type. At diagnosis, five DTC patients had lymph node involvement and two had lung metastases, although at time of assessment, only one DTC patient had lymph node involvement. DTC patients were older than control subjects (P=0.004) and had lower TSH levels than control subjects at time of assessment (P=0.013). QOL and anxiety levels did not differ between DTC patients compared with control subjects and with previously reported scores in a healthy cohort. QOL and anxiety level parameters were not influenced by age, time since diagnosis, or free T4 levels measured at the time of assessment. CONCLUSIONS: Adolescents with DTC have similar QOL and anxiety levels compared with autoimmune hypothyroidism patients and with a healthy normative population.

Prophylactic pancreatectomy for intraductal papillary mucinous neoplasm does not negatively impact quality of life: a preliminary study.

BACKGROUND: Uncertainties remain over whether prophylactic surgery or surveillance is the better management option for intraductal papillary mucinous neoplasm of the pancreas. The aim of this preliminary study was to determine if differences in anxiety and quality of life exist between patients who have surgery or undergo surveillance. METHODS: Recruited patients were given the Hospital Anxiety and Depression Scale, a general survey that evaluates anxiety, and the Functional Assessment of Cancer Therapy-Pancreas, a disease-specific survey that assesses quality of life. Questionnaires were scored by standardized algorithms and compared using Student's t test or Wilcoxon rank-sum test. RESULTS: Sixteen patients had surgery and 16 patients were undergoing surveillance. Mean age was 66.8 ± 19.9 years. Responses from both groups were remarkably similar. Surgery patients scored higher on the anxiety questionnaire than surveillance patients, although not statistically significant (p = 0.09). Surgery patients scored lower on the functional well-being domain of the quality-of-life instrument (p = 0.03), though there were no differences in overall quality of life. CONCLUSION: Prophylactic surgery does not reduce quality of life, and a protocol of surveillance does not appear to generate undue anxiety in this select patient group. Further investigation with more patients is required to validate these findings.

Health-related quality of life in long-term follow-up of patients with cured TNM Stage I or II differentiated thyroid carcinoma.

OBJECTIVE: The purpose of this study was to assess the impact of cured low-risk differentiated thyroid carcinoma (DTC) on health-related quality of life (HRQoL) after long-term follow-up. DESIGN: A cross-sectional study including an age- and gender-standardized sample of the general population. PATIENTS AND METHODS: HRQoL was assessed by the 15D in 341 DTC patients, whose initial treatment for stage I or II DTC was performed 12.4 years (range 5-20) earlier. The results were compared to those of an age- and gender-standardized sample of the general population (n= 6001). Determinants of HRQoL in DTC patients were assessed by the Tobit model. RESULTS: Mean total 15D scores did not differ between patients and control subjects. In single dimensions, DTC patients were significantly worse off with regards to sleeping, speech and distress (P= 0.001, 0.002 and 0.012, respectively), but better off with regards to discomfort and symptoms (P < 0.001). Within the patient group, the only significant independent predictor of HRQoL was age at the time of the initial treatment (P < 0.001). CONCLUSIONS: After long-term follow-up, overall HRQoL in DTC patients is comparable to that of the general population. DTC patients demonstrate an age-related decline in HRQoL, similar to that seen in the population in general.

Pain perception, blood pressure levels, and peripheral benzodiazepine receptors in patients followed for differentiated thyroid carcinoma: a longitudinal study in hypothyroidism and during hormone treatment.

BACKGROUND: Elevated blood pressure levels that are associated with hypalgesia and hypothyroidism have major influences on the cardiovascular system. The potential modulation of pain sensitivity by thyroid hormones is largely undetermined. Moreover, a few experimental studies show that peripheral benzodiazepine receptors (PBRs), which may be altered in hypothyroidism, seem to be related with pain perception. METHODS: Dental pain threshold and tolerance were evaluated in 19 patients followed for differentiated thyroid carcinoma (1) in severe short-term hypothyroidism (phase 1) and (2) during thyroid stimulating hormone-suppressive LT4 treatment (phase 2). PBR expression (cytofluorimetric evaluation) on peripheral blood mononuclear cells was also investigated in the 2 phases. RESULTS: Pain perception differed throughout the study, the dental pain threshold was higher in phase 1 (P<0.05) whereas pain tolerance was higher but not significantly (P=0.07). Although the systolic blood pressure was higher during hypothyroidism (P<0.01), no relationship was found between blood pressure changes and pain sensitivity variations. Moreover, the multiple regression analysis showed an independent association of the clinical phase with pain sensitivity (r=-2.61, P=0.029), while accounting for systolic blood pressure. The intensity of PBRs was significantly higher in the first phase of the study (P=0.047) whereas the ratio did not significantly differ. However, no relationship was observed between pain sensitivity and PBRs. DISCUSSION: In conclusion, in athyreotic patients, the pain sensitivity is related to the thyroid status and is independent of the increase in blood pressure induced by thyroid hormone deprivation. The PBRs do not seem to have major influence on pain sensitivity changes in hypothyroidism.

Health-related quality of life in thyroid cancer survivors.

OBJECTIVE: The study objective was to study the impact of the diagnosis, treatment, and follow-up of differentiated thyroid cancer (DTC) on the quality of life and related issues in an urban multi-ethnic Asian population. DESIGN: A self-administered questionnaire containing the Short Form 36 Health Survey (SF-36) and assessing sociodemographic, disease, and treatment-related status was mailed to patients with DTC. MAIN OUTCOMES: One hundred fifty-two (52.4%) of 290 patients answered the questionnaire. There was a statistically significant decrease in SF-36 scores between thyroid cancer survivors and the general population in all domains except for social functioning (SF). Physical functioning (PF) was worse in those survivors who were aged 50 years or older. Mental health (MH) scores were better in those who had more than 12 years of formal education. Being employed had a positive influence on role physical (RP) and role emotional (RE) scores. Being of Malay/Indian ethnicity strongly correlated with lower scores in bodily pain (BP), SF, RE and MH domains. CONCLUSION: Although most patients with well-differentiated thyroid cancer have near normal life expectancy, our study has shown that there is a significant decrease in their quality of life, especially in the elderly and poorer educated. Returning to work should be encouraged to improve the quality of life in DTC survivors.

Health-related quality of life, anxiety and depression in thyroid cancer patients under short-term hypothyroidism and TSH-suppressive levothyroxine treatment.

OBJECTIVE: Very few previous studies have compared the degree of health-related quality of life (HRQL), depression and anxiety of differentiated thyroid cancer patients (DTC) under short-term hypothyroid-ism and levothyroxine treatment. METHODS: Using patient-completed instruments, we examined the frequency of physical complaints, HRQL, anxiety and depression in 130 DTC patients hospitalized for radioiodine therapy or whole-body diagnostics (age 52 years, female 71%) under short-term hypothyroidism (4 weeks of levothyroxine withdrawal; DTC-H) and in 100 DTC out-patients under TSH-suppressive doses of levothyroxine subsequent to radioiodine therapy (DTC-L; age 49 years, female 81%). RESULTS: Compared with the German general population, DTC-H as well as DTC-L patients had significantly impaired HRQL. Notably, the decrease in HRQL was significantly higher in DTC-H than in DTC-L patients. Surprisingly, the prevalence of anxiety (44.6%) but not depression (17.7%) was much higher in the DTC-H patients than in the general population. In contrast to expectations, similar results for anxiety (44.0%) and depression (17.6%) were observed in the DTC-L patients. CONCLUSIONS: This mounting evidence suggests that a consistent pattern of HRQL impairment is experienced by patients with DTC. The high frequency of anxiety and the significantly reduced HRQL should be considered in the aftercare of DTC patients.

Aggressive management of recurrent ovarian cancer--the challenge of individualizing cancer therapy illustrated by a case report.

BACKGROUND: In clinical practice, treatment recommendations and the patient's wishes often diverge, facing the physician with difficult choices. CASE REPORT: The clinical course of a 36-year-old patient with 'platinum-refractory' ovarian cancer is reported. The patient experienced a symptomatic relapse 7 months after debulking surgery and completion of platinum-based first-line chemotherapy. As she had given birth to a son 22 months before diagnosis, she fought with outmost determination against her disease. Her husband supported her, and both asked for maximal therapy, including intensive care treatment for recurrent respiratory tract infections and total parenteral nutrition (TPN). For the patient, it was of major importance to stay with her family and make sure that her son would be able to remember his mother. Problems related to TPN and progression of disease affected her individual perception of quality of life to a much lower extent than expected and perceived by her caretakers. All professional health care providers were more than once very reluctant to continue treatment and only after extensive counseling gave in to the demand of the patient for further treatment, considering the effort futile - only to be surprised by treatment response and recovery. After 3 years of palliation, the tumor was resistant to all cytotoxic regimens and the patient died 2 months after withdrawal of chemotherapy. CONCLUSION: This case report illustrates that also in the age of evidence-based medicine individualized treatment beyond proven strategies can offer patient benefit. Taking the child's development into account makes it impossible to determine the cost-benefit ratio.