A new frontier of pediatric oncology rehabilitation: Establishment of a dedicated working-group within the Italian Association of Pediatric Hematology and Oncology

Abstract

PURPOSE:

In Italy, most children and adolescents affected by oncological diseases are treated in one of the centers within the Italian Association of Pediatric Hematology and Oncology (AIEOP). AIEOP relies on groups of experts; each of them develops interventions in their specific field of expertise to improve the diagnostic, therapeutic, and assistance pathway of patients and families. Although rehabilitation is an important field in pediatric oncology, until recently there was no working group dedicated to it. This study intends to show the steps that led to the creation of the rehabilitation working group in AIEOP.

METHODS:

First of all, a survey to identify the various rehabilitation approaches applied throughout Italian centers was sent via email to 39 AIEOP centers that provide rehabilitation.

RESULTS:

Answers received from 31 centers showed a lack of homogeneity in rehabilitation methods in terms of type of treatment, number of patients, and outcome measurement.

CONCLUSIONS:

Some results of the survey were chosen as basic criteria of clinical best practice on which to build the first rehabilitation working-group at the national level to be proposed to the AIEOP commission.

Keywords

Rehabilitation pediatric oncology network physical activity exercise

1. Introduction

In Italy, every year approximately 1380 children and 780 adolescents are diagnosed with oncological diseases [1]. Today, over 70% of children and adolescents with cancer are successfully treated. This result is produced by adequate facilities and by the various healthcare professionals who work together to ensure early diagnosis and the best treatment available. A growing consensus confirms the efficacy of networks and of national and international cooperation of healthcare professionals involved in the care of patients affected by complex diseases, ensuring high expertise on all diseases thanks to sub-groups dedicated to specific fields. This working methodology allows a sharing of evidence and clinical practice, cooperation in multicenter research projects, and represents the groundwork to define common standards of care [2, 3]. In Italy, treatment is offered by the Italian Association of Pediatric Hematology and Oncology (AIEOP), a network of 48 pediatric oncological centers dedicated to achieving constant improvements in the quality of care for pediatric cancer patients. AIEOP aims at providing uniformity in terms of structural, medical and assistance resources.

Children suffering from oncological diseases often have multiple rehabilitation needs resulting from chemotherapy and radiotherapy regimens, Hematopoietic Stem Cell Transplantation (HSCT), surgical and neurosurgical outcomes, complications and late effects affecting the cardiorespiratory system, motor performance, neurocognitive abilities, autonomy, and quality of life [4, 5]. In the past, children were advised to rest as much as possible. Currently, too much immobility may result in a further decrease of physical fitness and physical functioning [6]. These side effects might be prevented or reduced by introducing a physical exercise-training program during or after childhood cancer treatment [7]. Rehabilitation programs improve physical morbidity both in children with leukemia and lymphoma [8] and in the pediatric HSCT population and facilitate reintegration into everyday life [9]. In 2006, the Health Organization of Cancer Units for Rehabilitation Activities (HOCURA) project was developed and focused on oncology rehabilitation, starting from diagnosis up to palliative care. The project resulted in the publication of the “Oncological Rehabilitation White Book” [10], which defined some preliminary guidelines and had a chapter dedicated to pediatric oncological rehabilitation.

This document underlined the need to define recommended activities shared among the center’s network in order to offer equal rehabilitation care access to all patients. However, to date in Italy there is no collaborative network between Physiotherapists (PTs) and Development Neuro and Psychomotor Therapists (DNPTs) who handle the rehabilitation of children and adolescents affected by oncological diseases at affiliated AIEOP hospitals. Due to this lack of connection among therapists working in various centers, some important information is still missing. For example, the total number of pediatric patients who undergo rehabilitative treatment after oncological hospitalization remains unknown. Moreover, throughout Italy, a wide heterogeneity of treatment modalities is detectable with a very low percentage of evidence-based clinical behavior. Thus, the aim of this study was to describe current practices and identify limitations related to pediatric rehabilitation within the AIEOP centers in order to form a working group to improve and standardize services across sites.

2. Methods

In February 2015, an online multicenter national survey was created to investigate the state of the art of pediatric oncological rehabilitation in Italy. The survey was developed by 2 DNPTs and supervised by 1 psychologist, 1 child neuropsychiatrist, and 1 oncologist. Questionnaire items were selected starting from both clinical experience and literature. Informed consent to participate was obtained by telephone. Before being sent, the questionnaire was tested by DNPTs working at the Regina Margherita Children’s Hospital in Turin. The questionnaire consisted of 36 questions subdivided into 5 areas: 1) information about the rehabilitation professionals who take care of these children; 2) characteristics of each center; 3) modalities of the rehabilitative intervention; 4) outcomes and measures of the rehabilitative intervention; and 5) opinions about the survey and the project. There were 6 open and 30 closed questions. The closed questions were subdivided into ones that allowed only one answer ( $n=$ 22) and those that allowed multiple replies ( $n=$ 8). The rehabilitation services of the 52 centers that were members of AIEOP in 2015 were contacted, in addition to the staff at the “Eugenio Medea” Institute where patients are often treated after hospitalization. The centers that agreed to be contacted provided an email address of healthcare professionals who were then sent the survey. Data were collected from March 2015 up to the end of April 2015.

3. Data analysis

According to the qualitative nature of the data, descriptive analysis and frequencies were performed and illustrated in tables. The limited amount of available data prevented us from performing statistical tests.

4. Results

The questionnaire was sent to the 39 centers which provide rehabilitation for the population of interest, and was completed by 31 centers (participation rate 74%). Thirteen AIEOP centers were excluded because of the absence of a rehabilitation service ( $n=$ 11) or because therapists’ e-mail contacts could not be obtained ( $n=$ 2).

4.1 The participant

4.1.1 Qualification of the therapist(s) who completed the survey

In 29/31 centers a PT completed the survey, while in 2 a DNPT did so.

4.1.2 Participants’ experience

Therapists’ work experience varied from 8 to 38 years (24.7 years on average).

4.2 Center information

4.2.1 What is the reference department of the Rehabilitation Service in your center?

In 17/31 centers, the Rehabilitation Service is part of the Rehabilitation and Functional Recovery Department, in 6 it is part of the Child Neuropsychiatric Department, in 3 it is part of the Orthopedic Department, and in the remaining 5 centers it is in other departments.

4.2.2 Does your center have a Pediatric Oncology Department? Is there a Stem Cell Transplantation Unit? Is there a rehabilitation room?

In 23/31 centers there is a Pediatric Oncology Department, while in 6 there are dedicated rooms in other departments for these patients. In two centers, pediatric oncological patients are only treated as outpatients. In 21/31 centers there is a Stem Cell Transplantation Unit (SCTU). None of the centers with an Oncology Department or with a SCTU have a room dedicated to rehabilitation, although in one hospital a room will be built.

4.3 Organization of the rehabilitation of children and adolescents affected by cancer

4.3.1 Who makes decisions regarding rehabilitative assessment/treatment?

Referral for rehabilitation comes from various specialized physicians, even in the same center: an oncol- ogist was cited by 21 centers, a physiatrist by 19, an orthopedic pediatrician by 6, a child neuropsychiatrist by 4, a neurosurgeon and a pneumologist by 3 centers. Participants could choose more than one answer.

4.3.2 When is rehabilitation usually indicated during oncological treatment in your center?

The phase is highly variable: in 26/31 centers, a request for rehabilitative evaluation is made when specific problems appear and in 5/31 centers, it is made both before and after starting cancer treatment.

4.3.3 What are the main oncological illnesses you treat patients for? (kind of oncological illness, age ranges and kind of impairments)?

29/31 centers reported leukemia and lymphoma, 23 reported brain tumors, and 8 reported bone tumors. There is a high heterogeneity in age ranges: babies aged under 1 year are treated by 2/31 centers, pre-school aged children by 22, school-age children by 21, and preadolescents and adolescents by 15 centers. Table 1 represents the most common rehabilitation issues in pediatric oncology.

Table 1
Most common rehabilitation issues in pediatric oncology

Rehabilitation issues	Number of centres
	who deal with
	these issues
Respiratory issues	13
Restless Legs Syndrome	13
Sensory and motor alterations related to	9
Central Nervous System impairments
Muscle weakness	8
Orthopedic issues	6
Neuropathic disorders	3

Table 2

Centers in which rehabilitation treatment was performed considering the number of treated patients and the type of therapies

Number of children and adolescents	Patients receiving	Patients undergoing	Patients receiving
who receive rehabilitation	chemo/radiotherapy	HSCT	palliative therapies
Fewer than 5	1	2	2
Between 5 to 10	11	/	/
Between 10 to 20	14	12	17
Between 20 to 40	2	/	/
More than 50	2	7	3
No data or clinical records missing	2	/	9

4.3.4 In your center, how is the rehabilitation treatment organized (kind of intervention for areas of rehabilitation and how many therapists are involved)?

In 19/31 centers, children and adolescents are treated in the four following rehabilitation areas: neuro- psychomotor, neuromotor, orthopedic pediatrician, and respiratory areas. In the remaining 12/31 centers, treatment only includes some of these rehabilitative areas. In 24/31 centers, a single therapist deals with both orthopedic pediatrician, neuro-psychomotor, neuromotor and respiratory issues and in 18/24 centers, there is a shift rotation of therapists. In 6 centers, each therapist only deals with one kind of rehabilitative issue and if the patient has diverse rehabilitative problems, treatment is performed by various therapists.

4.3.5 In your center, how is the rehabilitation during different phases of the oncological treatments organized (the moment when rehabilitation is provided and the numbers of patients per year)?

In 20/31 centers, rehabilitative intervention is performed during active treatment and only during hospitalization. Following discharge, it continues as community rehabilitation. In 9/20 centers, after discharge, patients are further monitored at the hospital center with periodic evaluations, while in the remaining 11 there are no follow-ups for rehabilitation. In 5/31 centers, patients receive rehabilitation during active therapies both during hospitalization and after discharge until patients achieve a good functional level. Furthermore, in 4/31 centers, children/adolescents continue rehabilitation in the community and go to the hospital for periodic check-ups. In 2/31 centers, rehabilitation treatment is an out-patient service.

4.3.6 If you have a Stem Cell Unit, how is rehabilitation treatment organized (the time when rehabilitation usually starts, rehabilitative issues, numbers of patients per year)?

In the 21/31 centers with a SCU, rehabilitative assessments and treatment is provided if specific disorders appear. The main rehabilitative issue of these patients is muscular weakness (reported by 20/21 centers), followed by neurological problems (13/21 centers), respiratory issues (identified by 12/21 centers), and finally by graft-versus-host disease (GVHD) reported by 8/21 centers.

4.3.7 If there is a Palliative Care Unit, how is rehabilitation treatment organized (the time when rehabilitation usually starts, rehabilitative issues, numbers of patients per year)?

In 17/22 centers where rehabilitation is provided for patients undergoing palliative care, patients only receive rehabilitation interventions if there are specific disorders.

Table 2 represents the number of centers in which rehabilitation treatment was performed considering the number of treated patients and the type of therapies to which these patients are subjected (conventional antineoplastic therapies, HSCT, or palliative care).

4.4 Rehabilitation outcomes and measures

4.4.1 How is rehabilitative assessment managed in your center (what are the outcome measures and assessment tools)?

Functional abilities and pain were cited by 29/31 centers, muscular strength by 28, fatigue related to physical exercise by 27, range of joint movement by 24, quality of life by 20, and cutaneous tropism by 17 centers. Moreover, 5 centers cited other variables such as perception, visual function, ability to play, relational aspects, compliance to treatment, neurological aspects, pulmonary function, state of wounds, and lymphedema.

Twenty-two of the 31 centers declared they did not use standardized evaluation tools; in the remaining 9 centers, 7 use a standardized scale to assess pain (5 of those use the Visual Analogue Scale (VAS)), 6 use the Manual Muscular Test (MMT) and goniometric evaluation is performed by 4 centers. Four centers evaluate functional abilities, while 2 of these use the Gross Motor Function Measure Scale (GMFM) (Table 3).

Table 3
Use of standardized evaluation scales to evaluate various rehabilitative variables (these answers were given by the 9/31 centres who reported the use of standardized assessment tools)

Rehabilitation outcomes using	Number of centres who
standardized measures	used standardized
	measures
Quality of life	1
Cognitive abilities	1
Anthropometric variables	1
Functional motor abilities (GMFM)	4
Goniometric joint range of motion	4
Muscular assessment (MRC)	6
Pain Assessment (VAS)	7

Legend: GMFM (Gross Motor Function Measure); MRC (Medical Research Council Scale for Testing Muscle Strength); VAS (Visual Analogue Scale).

4.4.2 In your center, how is rehabilitative treatment organized (frequency and specific equipment)?

During hospitalization, rehabilitative sessions are generally performed daily. Fourteen of the 31 centers have specific equipment for rehabilitation treatment, exercise bicycles are used by 13, Wii Fit (an active video game developed by Nintendo for the Wii console) and treadmills by 7, and Kinesthetic (equipment for the passive mobilization of various parts of the body) by 4.

4.5 Comments on the survey

4.5.1 Did you find the questions interesting?

All participants but one felt the survey investigated significant topics.

4.5.2 What topics would you like to discuss further?

Only 12 therapists expressed their opinion, 8 cited standardized evaluation tools and rehabilitative protocols and 6 reported exercises for specific rehabilitative issues.

4.5.3 Do you think that onco-hematological pediatric rehabilitation would benefit from the creation of a network and would you be willing to take part?

All the PTs and DNPTs agreed that the creation of an Italian rehabilitative oncologic pediatric network would be useful. However, only 12/31 centers stated that they would be willing to be involved in the project.

5. Discussion

The aim of this study was to describe current practices and identify limitations related to pediatric rehabilitation within the AIEOP centers in order to form a working group to improve/develop uniform services across sites. In general, results underlined heterogeneity in the management of different pediatric oncological rehabilitation issues across Italy. Referring to participants, some of the therapists who completed the survey were DNPTs, i.e., Italian healthcare professionals who deal with the rehabilitation of neuropsychiatric illnesses in childhood in the areas of neuro-motricity, psycho-motricity, neuropsychology, and developmental psychopathology [11]. Considering professionals’ expertise, therapists who work with this type of population have some years of experience. On one hand, expertise of professionals ensures a high quality of rehabilitative treatment, but on the other hand it impedes the spread of experience to young colleagues, reducing the opportunities to involve them in future clinical studies. A more heterogeneous and extended working group could increase experiences and opinions as well as facilitate the definition of innovative approaches. Compared to the structural and logistic information of the hospital centers, our results showed a certain lack of homogeneity about who refers patients to Rehabilitation Services, as referrals are made by physicians with different subspecialties. Gohar et al. [12] suggest the implementation of standardized measures to evaluate musculoskeletal and neuromuscular side effects and referral guidelines. Since most centers have both a Pediatric Oncology Service and a SCU, at present, no center has a room dedicated to rehabilitation in these departments. The lack of dedicated rehabilitation rooms means patients cannot perform rehabilitation outside of their hospital room, even when they are not isolated. This absence may adversely affect rehabilitative intervention; as recognized by the National Institute for Health and Clinical Excellence [13], rehabilitation equipment can be an important part of therapy in children affected by cancer and environments may need to be adapted to allow installation of equipment. Furthermore, since the characteristics of how the room where rehabilitation treatment is set up can support various kinds of games and movement, it is very important to have a specifically dedicated place. For example, a large space in the middle of the room can support free movement and environmental exploration, while a corner set up with carpets and pillows can induce more static games. In most centers, rehabilitation referral is made when specific clinical issues emerge. Unfortunately, this means that a preventive rehabilitation treatment is not guaranteed in these centers. Many studies report decreased physical fitness (aerobic capacity and muscle strength) in patients and survivors of childhood cancer [14, 15, 16]. Reduced daily energy expenditure and lower levels of physical activity have been described as the most important causes of this reduced state of physical fitness in children with cancer [17]. Physical activity can prevent or diminish the negative effects of a sedentary lifestyle, such as obesity, poor skeletal health, fatigue, and poor mental health, thereby increasing the person’s quality of life [7]. As suggested by Gohar et al. [12], this consideration supports the need to increase the awareness of physicians about the benefits of early integration of a rehabilitative program into the medical treatment of these children.

Considering the type of patients treated, results underlined differences between the centers participating in the survey. Very few centers experienced the rehabilitation of children affected by bone tumors, of infants aged less than 1 year, and of subjects affected by neuropathic disorders. Specific rehabilitative guidelines would help to ensure that each patient receives suitable and effective rehabilitative care. In most centers, there are no therapists who are specialized in one of the intervention areas, and only a minority of the centers have a therapist who is completely dedicated to the treatment of children affected by oncological diseases. This structural organization makes it even more essential to be part of a rehabilitative network in order to have contact with colleagues who deal with the same kind of patients in other centers. The timing and duration of the rehabilitative intervention appears homogeneous, and almost all centers treat children/adolescents in hospitals for the duration of the active treatment and refer them to near-home rehabilitation services after the suspension of the treatment. Patients are not always monitored but have periodic hospital visits. In order to improve the continuity of a patient’s rehabilitation plan when referred from one hospital to another, or to the near-home rehabilitation services, it would be necessary to create a unique tool to share the most important rehabilitative information about the patient. Finally, consensus of common assessment tools among the different centers is crucial as it allows the creation of a common language among therapists necessary for data sharing and for the promotion of scientific research. This issue has been already reported by some authors [8, 18] who indicate the need to have measurement instruments for which reliability and validity has been established for this patient population. Considering the opinion of the therapists involved, their priority is the sharing with colleagues of standardized rehabilitation protocols and assessment tools. Another mentioned issue was the burden of the psychological aspects related to the disease. For all centers, it would be useful to create a collaborative rehabilitative network in which other professionals such as psycho-oncologists and oncologists are present.

6. Conclusion

Our research allowed us to define how the rehabilitation of children and adolescents with oncological diseases in Italy is currently structured. The resulting data indicate the presence of common pathways among participating centers, especially for general organization of treatment during active therapies, for principles of treatment, and for equipment used during rehabilitative treatment. We found a lack of homogeneity of experience in terms of specific rehabilitative needs, patients’ ages, evaluation tools, and exercise programs used. These data underline the need to develop rehabilitation standards of care and serve as a starting point for professional exchanges and a sharing of intervention strategies. This survey and the shortcomings highlighted were submitted to the AIEOP Committee with the proposal to create a national rehabilitation network with the main aim of filling these gaps. The proposal was launched and accepted, leading to the creation of a new Italian rehabilitation oncology network in 2016. Future principal objectives of the network will be the collection of epidemiological and clinical data, and the creation of a database in order to produce clinical studies on representative patient samples.

Footnotes

Acknowledgments

The authors thank all the therapists of the Italian Pediatric Oncology Rehabilitation Group who participated in the survey. Research was supported by Fondazione Umberto Veronesi.

Conflict of interest

Authors have full control of all primary data and agree to allow the journal to review their data if requested.

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