Explorative study on = the aftercare of pediatric brain tumor survivors: a parents=92 = perspective

Eline J. Aukema^{1, 4}, Bob F. Last^{1, 2},=20 A. Y. Netteke Schouten-van Meeteren³ and=20 Martha A. Grootenhuis¹

Received:=20 10 February 2010 Accepted:=20 23 August 2010 Published online:=20 10 September 2010

Introduction

The overall survival rate of children with a brain = tumor has=20 increased substantially due to multimodal treatment strategies [1].=20 As a consequence of the tumor and its multimodal treatment, survivors of = childhood brain tumors are at considerable risk for a variety of = long-term=20 sequelae. The current literature comprehensively outlines the multiple=20 neurocognitive difficulties [2=964],=20 problems in physical functioning [5,=20 6]=20 and psychosocial difficulties [7=9611]=20 experienced by survivors of childhood brain tumors and the impact on = family=20 functioning [12,=20 14].=20 There is growing evidence that not only survivors treated with = cranio(spinal)=20 radiotherapy, but also survivors of low grade brain tumors treated with = surgery=20 only, experience several long-term sequelae [15,=20 16].=20

The group of brain tumor survivors is growing and = results in an=20 increased demand for structured aftercare for which multidisciplinary = long-term=20 follow-up programs (e.g. [10,=20 17=9620])=20 are desirable. For example, Wallace et al. [20]=20 proposed a three-level follow-up model to organize the intensity and = frequency=20 of the follow-up care. Recently, Bowers et al. [21]=20 evaluated the use of these long-term follow-up programs in different = centers=20 from the Children=92s Oncology Group (COG) group members and found = considerable=20 variations in follow-up care in different centers despite these = guidelines. They=20 reported multiple barriers in the delivery of follow-up care, barriers = in=20 finance and insurance, but also more intrapersonal barriers knowing = patients=92=20 uncertainty about the need to follow-up care and the desire not to be = followed=20 in a pediatric aftercare program.

Variation in brain tumors and treatment modalities may = influence=20 the amount, interval and length of the follow-up care based on health = risks.=20 More knowledge about the necessary follow-up care for children who had = surgery=20 only as treatment for their brain tumor compared to those treated with = adjuvant=20 therapy is needed. The follow-up care can vary in the degree of care = provided.=20 This could be caused by either insufficient awareness of long-term = sequelae or=20 due to restrictions in multidisciplinary care for instance a separate=20 neurosurgery department. Research so far indicates that timely = intervention and=20 preventive programs improve different aspects of quality of life of = survivors of=20 childhood cancer. Recently, several evidence-based interventions became=20 available, including cognitive remediation [22=9624]=20 and emotional and social programs [25=9627].=20 By remediating cognitive functions and offering early interventions at = school=20 (e.g. extra tutoring, audio books, use of a laptop) new learning = facilities are=20 provided, improving future perspective and psychosocial functioning of = these=20 survivors.

Although the need for aftercare for sequelae of some = survivors=20 are obvious, especially for children treated with adjuvant therapy, = information=20 is lacking on whether this need is detected in time by professionals and = whether=20 existing aftercare is timely initiated. Furthermore because of the long = lasting=20 existence of the sequelae and the risk for these survivors to grow = further into=20 their deficits [16],=20 it is essential that parents and survivors are well informed about the = possible=20 sequelae. Thereby they should have knowledge about available aftercare = services.=20 Little is known about the parents=92 perspective on the follow-up = aftercare; such=20 as whether their own needs for aftercare for their child are met, = whether=20 parents have enough knowledge of and make use of aftercare services = available or=20 whether parents experience certain barriers in the care trajectory after = the=20 treatment.

With a survey we retrieved information from parents of = a diverse=20 group of brain tumor survivors in order to gain insight in the needs for = aftercare, the course of aftercare and needed improvements regarding the = provided aftercare.

The aims of this explorative study were to: (1) = describe the=20 need for aftercare for long-term sequelae perceived by parents and to = explore=20 the aftercare, (2) analyze differences in the need for aftercare in = children=20 treated with or without adjuvant therapy and (3) evaluate parents=92 = knowledge=20 about and use of aftercare services available in the Netherlands. =

Methods

Procedure

Parents of all children who had completed treatment = for a brain=20 tumor between 1990 and 2006 at the Emma Children=92s Hospital Academic = Medical=20 Center, Amsterdam, The Netherlands (N =3D 57), were = invited to participate=20 in the study. Children with neurofibromatosis were excluded because of = their=20 specific cluster of impairments.

The inclusion criteria were: (1) treated for a brain = tumor of=20 any histology diagnosed between the ages of 0 and 18, (2) current age of = the=20 survivor between 4 and 20 years, (3) complete remission or stable = residual=20 tumor, (4) at least 1 year after the end of treatment and (5) able = to=20 complete a questionnaire in Dutch. Parents of survivors who met the = inclusion=20 criteria in May 2007 were invited to this study, which was approved by = the local=20 medical ethics committee.

Parents were firstly contacted by phone and then = received an=20 introductory letter explaining the aim of the study and asking for their = participation. The letter was accompanied by questionnaires including a = survey=20 about the aftercare, as reported in this article, an informed consent = form, and=20 a stamped return envelope. Each family received one packet of = questionnaires by=20 mail to be completed at home. Non-responders were contacted by phone and = a=20 reminder letter was sent to increase response.

Measures

The survey with eight questions per domain of child = functioning=20 and parenting includes both pre-coded as open questions about the need = for=20 aftercare, the type of provided aftercare, timing and referral, needs = for=20 improvements and knowledge about and use of aftercare services. The = choices of=20 the specific domains of child functioning and parenting were based on = the known=20 long-term sequelae of childhood cancer survivors [2=9616]=20 and aftercare services available in the Netherlands. The questions were=20 formulated by several experienced oncologists and psycho-oncologists of = our=20 hospital.

Answering options with service examples were given as = well as=20 space to write down other options. Afterwards, answers were categorized = in the=20 categories described below.

First, the prevalence and type of long-term = sequelae in=20 five different domains and the need for aftercare were evaluated = among=20 the parents. The domains covered different types of long-term sequelae,=20 classified as follows: (1) neurocognitive problems (regarding = attention,=20 memory, processing speed, executive function, visual perceptual motor = skills and=20 fine motor skills); (2) physical problems (regarding energy level = [e.g.=20 headache, fatigue], hormone function [e.g. early puberty, growth = retardation],=20 motor function [e.g., fine and gross dexterity, balance, strength], = epilepsy,=20 visual and auditory function); (3) emotional problems (including=20 internalizing [fearfulness, worrying, difficulties in expressing = emotions],=20 externalizing [frustration, anger] illness experience [difficulties with = the=20 disease, feeling different] and self-esteem); (4) social problems = (regarding social isolation [e.g. contact making, maintain = relationships],=20 adult-centered, being bullied and quarreling with peers); and (5) = parenting=20 problems (including autonomy encouragement, differences in parenting = style,=20 overprotection and overindulgent behavior).

Based on these answers, a mean score of need for care = in the=20 different domains (0=965) was calculated per participant.

Second, the type of aftercare as provided was asked, = categorized=20 in specific answer options and examples of aftercare services available = in the=20 Netherlands for each domain. These were, for neurocognitive = problems:=20 educational support at a regular school, special needs school, = neurocognitive=20 test administration and/or remediation; for physical problems:=20 physiotherapy and/or occupational therapy, medication, specific diet, = motor=20 support equipment (e.g. motor aids, adjusted sport participation), = visual=20 support (e.g. eyeglasses or training to cope with visual deficits), = auditory=20 support and special education needs based on motor dysfunction; for = emotional=20 problems: therapy from a psychologist, other forms of supportive = care (e.g.=20 homeopathy, creative therapy), parental support and contact with fellow=20 patients; for social problems: therapy from a psychologist, = social skills=20 training, parental support and contact with fellow patients; and for=20 parenting problems: therapy from a psychologist or another = professional=20 giving parenting support and contact with fellow parents. We evaluated = the=20 aftercare in terms of the referrer and need for improvements regarding = (1)=20 timely response to the need for aftercare, (2) psycho-education and = support in=20 finding aftercare services, (3) proactive approach from school and (4) = proactive=20 screening of needs for aftercare. To explore who referred participants = to the=20 aftercare, we created the following categories: hospital staff = (doctors,=20 psychologists or other caregivers from the hospital), school = staff=20 (teachers or other staff members of the school) and parents = themselves=20 (whether or not in cooperation with the general practitioner).

Finally, the knowledge of the existence and use of = different=20 aftercare institutions and websites in The Netherlands were asked. The = knowledge=20 was categorized in websites with psycho-education about acquired brain = injury=20 and brain tumors, institutions for aftercare services (rehabilitation = centers,=20 hospitals), parents=92 association for children with cancer and family = camps, and=20 school services (websites with school aids, information and school = liaisons).=20

Individual patient data regarding gender, date of = birth and=20 medical data regarding date of primary diagnosis, histological type of = tumor,=20 location of tumor, type of treatment, secondary operations (including=20 progression and relapse) and duration of adjuvant treatment were = gathered from=20 the medical files.

Statistical analyses

The participants and non-participants were compared = with respect=20 to demographic and medical variables using Independent Samples = t-tests=20 and the chi square (χ ²) tests = (p < 0.05). Frequency=20 analyses were conducted to assess the prevalence and kind of = parent-reported=20 long-term sequelae, the need for aftercare and the amount of domains for = which=20 aftercare was needed (0=965) was calculated. Then, associations between = need for=20 aftercare and treatment (=91surgery only=92 and =91surgery and adjuvant = therapy =92)=20 were examined with χ ² tests at = p < 0.05 and with=20 Mann=96Whitney U-tests, differences in the amount of need for = care in=20 different domains (0=965) between these two groups were examined at=20 p < 0.05. Also, frequency analyses were used to = evaluate the provided=20 aftercare and the familiarity with websites and institutions. The = Statistical=20 Package for Social Sciences (SPSS) version 16 was used for all analyses. =

Results

Participants

Forty-two (74%) of the 57 parents participated in = the study=20 and returned their completed survey. Characteristics of the participants = and=20 non-participants are presented in Table 1.=20 Half of the participants were treated with surgery plus adjuvant = therapy.=20 Non-participants did not differ from participants in demographic and = medical=20 variables. The reasons given for not participating were lack of interest = or no=20 time to fill in the questionnaires.

Table 1 Characteristics of the = (non-)participants=20 (N =3D 57)

	Participants (n =3D 42) =	Non-participants (n =3D 15) =
M	SD	Range	n	M	SD	Range	n
Age at study (years)	14.7	3.8	5.7=9619.8	42	14.9	4.1	7.7=9619.8	15
Age at start treatment (years)	6.5	3.3	1.9=9616.5	42	8.1	4.4	2.2=9616.8	15
Time since start treatment (years)	8.1	3.9	1.6=9615.7	42	6.6	3.6	1.7=9614.4	15
Duration of adjuvant treatment (years)	1.3	1.7	0.2=968.0	22	1.4	1.1	0.4=962.8	4
	n	%			n	%
Gender
Male	19	45			10	67
Female	23	55			5	33
Tumor location
Infratentorial	26	62			10	67
Supratentorial	16	38			5	33
Tumor Type
High grade	19	45			3	20
Low grade	23	55			12	80
Treatment
Surgery only	20	48			11	73
Surgery plus adjuvant therapy	22	52			4	27
Other	=96	=96			=96	=96
More specific
Surgery only	20	48			11	73
Surgery and radiation therapy	1	2			=96	=96
Surgery and chemotherapy	5	12			=96	=96
Surgery, radiation therapy and = chemotherapy	16	38			4	27
More than one operation (including = progression,=20 relapses)	8	19			3	20
Age at diagnosis
≤3 years	5	12			1	7
>3 years	37	88			14	93
Respondent
Father	6	14
Mother	32	76			=96	=96
Both	4	10

Needs for and exploration of the aftercare in = different=20 domains of long-term sequelae

The need for aftercare and received aftercare and = needs for=20 improvements with the aftercare trajectory are presented in = Tables 2=20 and 3.=20

Table 2 Need for=20 aftercare per domain (N =3D 42)

	Neurocognitive	n (%)	Physical	n (%)	Emotional	n (%)	Social	n (%)	Parenting	n (%)
Prevalence problems		31 (74)		38 (90)		27 (64)		24 (57)		16 (38)
Specific problems^a
	Attention	15 (48)	Motor function	28 (74)	Internalizing	16 (59)	Social isolation	21 (88)	Autonomy encouragement	11 (69)
	Processing speed	14 (45)	Energy level	19 (50)	Illness experience	12 (44)	Being bullied	9 (38)	Overprotective	9 (56)
	Fine motor skills	13 (42)	Hormone function	14 (37)	Self-esteem	9 (33)	Adult centered	7 (29)	Overindulgent	6 (38)
	Memory	11 (36)	Visual problem	12 (32)	Externalizing	8 (30)	Quarrel with peers	6 (25)	Parental difference	4 (25)
	Executive functions	8 (26)	Epilepsy	6 (16)
	Visuoperceptual motor skills	7 (23)	Hearing loss	6 (16)
Need for aftercare
Yes		30 (97)		34 (89)		22 (81)		21 (88)		11 (69)
No		1 (3)		4 (11)		5 (19)		3 (13)		5 (31)
Received aftercare
Yes		28 (93)		31 (91)		19 (86)		16 (76)		8 (72)
No but was indicated		2 (7)		3 (9)		3 (14)		5 (24)		3 (27)
Type of aftercare^a
	Neurocognitive test assessment and/or = remediation	17 (61)	Physiotherapist/Occupational therapist	25 (81)	Psychologist	15 (79)	Psychologist	11 (69)	Parental support	7 (88)
	Special educational services:	23 (82)	Medication	15 (48)	Fellow patient contact	6 (32)	Social skills training	7 (44)	Fellow parent contact	1 (13)
	- educational support at a regular = school	17 (61)	Motor support equipment	12 (39)	Other emotional supportive care	6 (32)	Fellow patient contact	2 (12)
	-transition to special education school	13 (46)
			Visual support	12 (39)	Parental support	2 (11)	Parental support	1 (6)
			Specific diet	4 (13)
			Special motor needs school	3 (10)
			Auditory support	3 (10)

^aMore options were possible in one = patient=20

Table 3 Evaluation=20 of the received aftercare in different domains

	n (%)	n (%)	n (%)	n (%)	n (%)
Need for aftercare
Yes	30 (97)	34 (89)	22 (81)	22 (88)	11 (69)
Received aftercare
Yes	28 (93)	31 (91)	19 (86)	17 (77)	8 (73)
No but was indicated	2 (7)	3 (9)	3 (14)	5 (23)	3 (27)
Referrer^a
Hospital staff	10 (36)	29 (85)	8 (42)	2 (17)	1 (13)
Schoolteacher or staff	9 (32)	5 (15)	3 (16)	6 (35)	1 (13)
Parents themselves	10 (36)	4 (12)	8 (42)	9 (53)	4 (50)
Not specified	2 (7)	2 (6)	4 (21)	2 (12)	2 (25)
Need for improvements^a	17 (57)	11 (31)	11 (50)	12 (57)	7 (64)
More specific
Timely response to need for care	13 (43)	9 (27)	7 (32)	8 (36)	5 (45)
Psycho-education about possible long-term = sequelae=20 and available aftercare services and support in finding aftercare=20 service	11 (37)	6 (18)	9 (41)	5 (23)	5 (45)
Proactive approach from school	6 (20)	2 (6)	1 (5)	1 (5)	=96
Proactive screening of needs for = aftercare	7 (23)	3 (9)	4 (18)	6 (27)	3 (27)

^aMore options were possible in one = patient=20

Overall, most parents reported need for aftercare in = several=20 domains: 17% reported problems in two domains, 29% reported problems in = three=20 domains, 19% reported problems in four domains and 14% reported problems = in all=20 domains. Overall, the mean amount of domains for which aftercare was = needed=20 according to the parents was 2.8 (median =3D 3, = SD =3D 1.4).

Neurocognitive domain

Two thirds (N =3D 31; 74%) of the = parents reported=20 neurocognitive problems, most often consisting of problems with = attention,=20 processing speed and fine motor skills. Thirty of these survivors (97%) = needed=20 aftercare; 28 survivors received specialized aftercare, two survivors = did not=20 receive the care they needed. Aftercare was comprised of use of special=20 educational services knowing remedial teaching at a regular school or = transition=20 to a school for special education and neuropsychological testing with or = without=20 remediation.

Parents reported that the hospital staff, school and = parents=20 themselves equally acted as referral sources for specific aftercare. = Half of the=20 parents expressed their needs for improvements regarding a sooner = response to=20 the need for aftercare and more support and psycho-education in general. = In=20 particular, they reported that school should point out problems more = actively.=20

Physical domain

Survivors with physical problems = (N =3D 38, 90%) covered=20 motor dysfunctions, such as fine and gross dexterity and balance, being = tired=20 and having headaches, as well as early puberty and delayed growth. Most = of these=20 survivors (N =3D 34, 89%) needed professional = aftercare; 31 survivors=20 received specialized aftercare; three did not receive aftercare though = it was=20 needed.

Most survivors received aftercare from a = physiotherapist and/or=20 occupational therapist, and received medication (anti-epileptic drugs = and/or=20 hormones) and motor or visual support equipment.

In most cases, the hospital staff referred = participants to the=20 particular specialist or therapist. A third of the parents had = suggestions for=20 improvements regarding the timing and the need for more support and = information=20 about getting the needed aftercare.

Emotional domain

Twenty-seven parents (64%) reported emotional problems = in their=20 child, especially internalizing symptoms regarding illness experiences = and=20 self-esteem. The majority of these survivors needed aftercare = (N =3D 22,=20 81%); 19 survivors received aftercare; three did not receive the care = that was=20 needed. The aftercare given included treatment by a psychologist, other=20 emotional supportive care and fellow patient contact.

The hospital staff and parents initiated this type of = aftercare.=20 Needs for improvement were expressed by half of the parents. More = specifically,=20 parents asked for more support and psycho-education on the possible = emotional=20 long-term sequelae, asked for information on aftercare available and = asked for a=20 more timely response on emotional problems.

Social domain

Social problems were reported in 24 survivors (57%) = and involved=20 social isolation, being bullied and adult-centered behavior. The = majority of=20 these survivors (N =3D 21, 88%) needed aftercare; 16 = received specialized=20 aftercare, and five did not receive aftercare although the need existed=20 according to the parents.

Aftercare was primarily comprised of treatment by a = psychologist=20 or a social skills training. In almost half of the patients the care was = initiated by the parents themselves, followed by school and the hospital = staff.=20 Parents expressed their need for improvements regarding a proactive = screening=20 for social problems in general and thereby a sooner referral for = specific=20 aftercare. Parents also wanted to receive more information about = possible=20 difficulties that could arise in the child=92s social behavior. =

Parenting domain

A third of the parents (N =3D 16, = 38%) reported problems=20 with encouraging autonomy in their child and being overprotective and=20 overindulgent to the child. Eleven (69%) needed extra care, of which = eight=20 received aftercare and three did not get the needed aftercare. Mostly, = aftercare=20 was comprised of professional parental support as was initiated by = parents=20 themselves.

More than half of the parents reported that = improvements were=20 needed, in particular more information about possible parenting = difficulties and=20 better screening and, if necessary, referral for these difficulties. =

Associations and differences between need for = aftercare and=20 type of treatment

No significant associations were found between the = need for=20 aftercare in the different domains and treatment modalities.

The groups also did not differ in the mean amount of = domains for=20 which aftercare was needed (mean amount of domains in =91surgery only=92 = group =3D 2.7=20 and in =91surgery plus adjuvant therapy=92 group =3D 3.0 = domains; U =3D 189,=20 p =3D 0.4).

Knowledge about and use of relevant institutions = and=20 websites

The familiarity and use of relevant institutions and = websites=20 for support and interventions in case of long-term sequelae are reported = in=20 Table 4.=20

Table 4 Knowledge=20 about and use of relevant institutions and websites

	n	%	n	%
Websites with psycho-education about acquired = brain injury=20 and brain tumors	27	64	8	19
Institutions for aftercare services = (rehabilitation=20 centers, hospitals)	34	81	19	45
Parents=92 association for children with cancer = and family=20 camps	29	69	16	38
School services (websites with school aids, = information=20 and school liaisons)	13	31	4	10

More options were possible in one=20 patient

Parents were most familiar with the aftercare services = in the=20 hospital (81%), the parents=92 association of children with cancer and = camps for=20 survivors (69%) and websites with psycho-education about required brain = injury=20 and brain tumors (64%). They were least familiar with educational = services and=20 special educational aids (31%).

Despite the knowledge of relevant institutions and = websites,=20 only 45% of parents actually made use of institutional aftercare, = followed by=20 the parents=92 association of children with cancer and camps for = survivors (38%),=20 use of websites (19%) and finally school services (10%). =

Discussion

Despite the fact that childhood brain tumor survivors = are at=20 considerable risk for long-term sequelae for which aftercare is needed, = little=20 is known about the recognition of this need for aftercare and whether = the=20 aftercare meets the needs of the parents of this group of survivors. =

Our explorative study from a parents=92 perspective = highlights a=20 considerable need for aftercare, not only for physical and = neurocognitive=20 problems, but also a substantial need for social, emotional and = parenting=20 aftercare. These findings are comparable with the outcome of former = studies=20 [2=966].=20 This need for aftercare has been expressed by the parents regardless of = whether=20 survivors had received adjuvant therapy, which is in line with the = findings in=20 earlier studies [15,=20 16].=20

Remarkably, our survey reveals that despite increased = awareness=20 of long-term sequelae among professionals, parents reported considerable = delays=20 in identification of the sequelae, and that more timely reactions to the = needs=20 for aftercare are required in particular for psychosocial problems = resulting=20 from disease and treatment.

Furthermore, parental knowledge about aftercare = services is=20 lacking, indicating a necessity for a timely and repeated screening of = long-term=20 sequelae that should result in providing specialized aftercare and=20 psycho-education to all brain tumor survivors.

Evaluation of the aftercare

Although parents reported that most survivors received = specialized aftercare when needed, a substantial number of parents = expressed the=20 need for improvements regarding the aftercare trajectory. The need for = aftercare=20 was most unmet for parenting problems, and most met for neurocognitive = problems.=20 For the psychosocial domains and parenting domain, more than half of the = parents=20 reported that improvements were needed regarding the timing of the = response to=20 the need for aftercare, more psycho-education and support in finding the = aftercare services and a proactive approach of the needs.

More specific, a timely response to the need for = aftercare and=20 timely referral was highest for physical aftercare, medium for = emotional, social=20 and neurocognitive aftercare, and lowest for parenting aftercare. = Parents often=20 had to refer their child for psychosocial aftercare themselves and the = timing of=20 the referral by professionals was often perceived as too late. In = addition, more=20 psycho-education about psychosocial long-term sequelae and support in = finding=20 aftercare services and a proactive screening of needs for aftercare were = suggestions to improve the aftercare for psychosocial and parenting = problems. A=20 proactive approach from the school (liaison) could be helpful in = identifying=20 difficulties and could lead to a timely intervention, especially for = learning=20 difficulties. From this, we conclude that the aftercare providers = respond to=20 physical problems appropriately, while more awareness is needed for = aftercare=20 for psychosocial problems and parenting difficulties. This finding is = consistent=20 with literature which indicates that psychological aftercare is needed. = Eiser=20 [10]=20 stated that psychological care of survivors needs to be integrated in = the=20 routine assessment. Recklitis et al. [28]=20 also points to the need to provide routine psychological assessment in = addition=20 to screening for physical problems.

Failure to detect and thereby meet the needs for = aftercare=20 timely could be explained from the perspective of several barriers. = First of=20 all, specific knowledge about late sequelae from childhood brain tumors = must be=20 well communicated with the parents. Second, it could be the case that = doctors=20 did not respond adequately to the needs of parents. Poor = doctor=96patient=20 communication and contact can be viewed as an interpersonal barrier. It = is known=20 that the quality of the consult is determined by the quality of the=20 doctor=96patient relationship, which is predictive for compliance with = the=20 doctor=92s advice [29].=20

Moreover, intrapersonal barriers in parents could also = have led=20 to delayed referral and in delayed use of the existing and familiar = aftercare=20 services. Parents may have underreported the existing problems in their = child=20 for a while in order to maintain a high degree of optimism. This = behaviour,=20 which may be viewed as denial, could also be viewed as =91selective = information=20 processing=92 [30].=20 In addition, looking to the parental reports we consider that at least a = substantial part of delay in aftercare has been caused by a lack of = urgency of=20 the problems from a parent=92s perspective in our survivor group. The = fact that a=20 substantial part of the parents are familiar with aftercare services but = only a=20 few use these services seems indicative for this delay. Therefore, = parents could=20 underreport the existing problems through lack of energy to be = confronted with=20 new problems.

Overall, half of the parents showed a lack of = knowledge about=20 long-term aftercare services, which shows a barrier in the prevention of = future=20 problems in this survivor group at risk.

Interestingly, Oeffinger and Wallace [31]=20 described several potential tools in overcoming some of the barriers = mentioned=20 before by introducing a Passport for Care, a web-based program with=20 individualized summary of the patients history combined with the COG = long-term=20 follow-up guideline. Also, our results support recommendations from = Hewitt et=20 al. [32]=20 for a survivorship care plan which addresses psycho-education about = long-term=20 aftercare and psychosocial support services available. Online = psycho-education=20 and recording the consult might improve the accessibility of the = necessary=20 information [33].=20

Parenting problems

In our group of survivors, one third of the parents = reported=20 parenting problems, including difficulties with encouraging autonomy in = their=20 child and being overprotective. These problems seem to have remained = unnoticed=20 by professionals at school and the hospital staff. From this, we = conclude,=20 consistent with literature [12=9614],=20 that more attention is needed to estimate the burden and needs of = parents.=20 Coaching parents in coping with their children=92s disabilities might = reduce=20 parental stress and empower their parenting style in everyday life = [34].=20 It is known that parents confronted with their child=92s = life-threatening disease=20 or near-fatal accident may react with a long-lasting fear of losing = their child,=20 even if the immediate threat has disappeared or faded away. Parents=92 = perception=20 of the increased vulnerability of their child could lead to = overprotection of=20 the child [35],=20 which might hinder the child in achieving its developmental milestones = [36].=20

Relationship with treatment modality

Although our group of participants is rather small, we = did not=20 find any significant relationship between the need for aftercare in the=20 different domains and treatment modalities. More specifically, we did = not find a=20 relationship between the need for neurocognitive aftercare and adjuvant = therapy.=20 Learning difficulties have been related to adjuvant therapy earlier = (e.g.=20 [4]),=20 although learning difficulties have also been described in survivors = with=20 low-grade brain tumors treated with surgery only [15,=20 16].=20

Our data further indicate that survivors of low-grade = tumors=20 treated with surgery only have the same overall need for aftercare = compared with=20 those who had adjuvant therapy, which stresses the need for follow-up = care. Ris=20 et al. [15]=20 also recommended routine follow-up for children after treatment for = low-grade=20 tumors.

Clinical implications and future research

Our study makes it clear that parents ask for more = attention for=20 psychosocial long-term sequelae in childhood brain tumor survivors, = regardless=20 of treatment modalities, pointing to the need of a multidisciplinary = follow-up=20 screening approach for all brain tumor survivors. In addition, the = oncologist=20 and the (neuro) psychologist should screen which families are in need = for=20 specialized aftercare at particular moments of transition during the = development=20 of the child growing in adulthood. This is desirable as survivors run = the risk=20 of developing deficits and a substantial number of survivors require = special=20 education (31% compared to 3% in the normal Dutch population). Further = research=20 on evaluating the needs of children and their parents for care should = lead to=20 more satisfaction.

Collaboration with school liaisons and the school of = the=20 survivor is needed to detect difficulties in time. Moreover, supportive = actions=20 can be initiated from the hospital regarding adaptations in primary or = secondary=20 education or follow-up training. Patient Reported Outcome (PRO) = assessments and=20 internet-based screening tools could be helpful and should be studied on = their=20 efficacy in assisting care providers in retrieving the needs for = survivors and=20 their parents [37].=20

Early interventions and preventive psycho-educational = programs=20 can improve coping with the long-term sequelae and improve different = aspects of=20 quality of life. These interventions directed to the survivors and the = parents,=20 include information about programs for improvements for neurocognitive = deficits=20 [21=9623],=20 empowerment of the child [38]=20 or coaching parents in coping with changes of their child [34].=20 Additionally, schoolteachers should be informed as well.

Limitations

Several limitations of the present research should be = noted. A=20 major limitation is the use of our survey in which we evaluated the = aftercare=20 from a parents=92 perspective to retrieve the needs for and received = aftercare of=20 this group of survivors. A parents=92 view could add more insight in = their=20 perception of improvements of the needs for aftercare, while = underestimating or=20 overestimating the needs for aftercare could be influenced by the = subjective=20 parental disease experience and could have been prevented by adding more = informants [39].=20 Despite this limitation, our results are in line with previous studies = and=20 identify a high need for aftercare in several domains of child = functioning and=20 in parenting [1,=20 9,=20 15,=20 19,=20 34,=20 35].=20 Moreover, 82% of the participants needed special educational services = and a=20 substantial number of the children had to transit to special education. = This=20 underlines the problems these children have to cope with.

A second limitation is found in the fact we could only = tentatively indicate the difference in need for aftercare based on the=20 conditions surgery only or surgery and adjuvant therapy because of our=20 relatively small group in relation to the diversity of patient = variables. The=20 influence of radiation therapy could not be evaluated as a separate risk = factor.=20 Nevertheless, the high similar need for aftercare in all survivors = regardless of=20 the type of treatment indicates that attention for aftercare regarding=20 psychosocial problems and parenting difficulties are a necessity for all = brain=20 tumor survivors. Third, the parental evaluation of the received = aftercare may be=20 influenced by barriers in the health care system (for instance long = waiting=20 lists, long distance to special services) and preexisting psychosocial = problems=20 (for instance psychiatric problems in the family, language problems, = multiple=20 social problems). In our survey, we did not explore these potential = barriers,=20 nor did we explore intrapersonal barriers as mentioned before. More = research is=20 needed to explore these barriers in more detail. Fourth, because this is = a=20 single center study, representation is limited. Nevertheless, it should = be=20 mentioned that to our knowledge this is the first evaluation of the = needs for=20 aftercare from a parents=92 perspective.

Finally, although the survivors were treated in a = broad time=20 span (16 years), it should be stated that our center has a long = tradition=20 in providing psychosocial aftercare to children treated for cancer, so=20 evaluation of these data from survivors treated at different time points = makes=20 sense [40].=20

Conclusions

Despite extensive knowledge about the serious = long-term sequelae=20 brain tumor survivors could suffer from, this study reveals that = increased=20 awareness and a more timely reaction to the need for aftercare is = required to=20 meet the need for psychosocial aftercare and parental aftercare more = adequately.=20 Treatment centers should focus on a solid follow-up program by medical = and=20 psycho-social staff to perform a timely referral for aftercare in this = patient=20 group. Information programs for neurocognitive deficits and = psycho-education=20 about long-term sequelae, strategies aiming at empowerment of the = child=92s=20 autonomy and coping behavior are highly recommended. A well organized=20 co-operation with schools is a prerequisite to meet the needs of = survivors and=20 their parents more adequately.

Acknowledgement The authors express their gratitude to the parents of = survivors who=20 participated in this study. They also wish to thank Hannah Karst for her = help in=20 collecting the data. This study was supported by Dutch foundation KIKA = =91Kinderen=20 Kankervrij=92 (Children Cancerfree).

Conflict of interest = statement =20

None of the authors has a conflict of interest to = declare in=20 relation to this work.

Open=20 Access =20

This article is distributed under the terms of the = Creative=20 Commons Attribution Noncommercial License which permits any = noncommercial use,=20 distribution, and reproduction in any medium, provided the original = author(s)=20 and source are credited.

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Original Article