1 INTRODUCTION

Children, adolescents, and young adults with hematologic and/or oncologic conditions including cancer and sickle cell disease (SCD) have complex needs during treatment that include managing stress, anxiety, fatigue, and pain.^1-4 Pediatric patients and their families report pain as the most distressing aspect of their cancer treatments. If not adequately managed, pain can contribute to impairments in quality of life, mental health, and behavior.⁵ To address patients’ symptoms and provide psychosocial support, many cancer centers have integrated evidence-based interventions including massage therapy (MT) into their services. A recent review of cancer center websites found that MT was available in 84.3% of National Cancer Institute-designated comprehensive cancer centers and 80% of community hospitals.⁶

A few randomized controlled trials (RCTs) and systematic reviews have established the benefits of MT for pediatric patients with cancer. Specifically, a review of seven clinical trials (sample size range: 17–171) reported that MT contributed to decreased pain, stress, anxiety, and nausea.⁷ An RCT among 62 hospitalized children with cancer aged 10–18 years found that patients in the treatment group reported significant single-session reductions in pain intensity and reduced pain interference with walking.⁸ However, per the recent clinical practice guidelines for integrative medicine for pain management in oncology published by the Society for Integrative Oncology and American Society for Clinical Oncology, there is currently insufficient or inconclusive evidence to make integrative treatment recommendations for pediatric patients with cancer pain.⁹ Among adults, the strength of the recommendation was moderate for MT for general musculoskeletal pain, posttreatment pain in breast cancer, and pain during palliative or hospice care.⁹ Given the current state of the evidence for MT in pediatric cancer pain, more research is needed to investigate its clinical effectiveness.

Even fewer studies have examined MT among children and adolescents with SCD, despite these individuals often being hospitalized within the same medical oncology units as children and adolescents with cancer.¹⁰ A review of five RCTs found that MT can provide significant pain reduction; improve functional status; reduce depression, anxiety, and stress; and potentially reduce analgesic use and length of stay.¹¹ Given the significant pain burden,¹² severity of vaso-occlusive crises,¹³ barriers to accessing nonpharmacologic pain management,¹⁴ and structural racism¹⁵ affecting individuals with SCD, research is needed to understand the effectiveness of MT within this population as distinct from patients with other hematologic and/or oncologic conditions.

Many of the clinical trials referenced above have limitations, including (a) small sample size^{8, 16-18}; (b) a focus on one or two specific MT interventions for narrowly defined conditions; and (c) the inability to tailor individualized interventions to specific patients. The qualifications of individuals delivering MT have varied across studies, with few including a certified pediatric massage therapist (CPMT). Additionally, there remains a gap in understanding the delivery and characteristics (e.g., patients’ chief complaints, body regions addressed, techniques used, and pressure applied) of MT within a pediatric hematology and oncology population. No prior study has compared the impact of MT on symptoms between patients with SCD and patients with other hematologic/oncologic conditions. To address these gaps, this retrospective study sought to describe the clinical delivery of MT and compare differences in the effectiveness of MT on pain, stress, and anxiety between two patient groups: those with hematologic and oncologic conditions excluding SCD (HemOnc) and those with SCD alone (SCD).

2 METHODS

3 RESULTS

4 DISCUSSION

The purpose of this retrospective study was to describe the clinical delivery of MT and examine differences in MT's effectiveness on pre-to-post changes in acute pain intensity, stress, and anxiety between the HemOnc and SCD groups. Within a large pediatric academic medical center, MT services can be integrated within inpatient and outpatient care to not only enhance symptom management but also provide psychosocial support to patients and their families as they undergo treatment. Unlike care models that contract with MT services, the model presented in this study involves close collaboration with the medical team to provide coordinated care for symptom management and procedural support. The CPMT also provides valuable services consistent with the Standards for Psychosocial Care of Children with Cancer and Their Families.^{2, 23, 24} These services include: (i) conducting systematic assessments of patients’ and family members’ psychosocial healthcare needs; (ii) providing psychosocial support and interventions throughout the cancer journey; (iii) providing psychoeducation, information, and anticipatory guidance related to hospitalization; (iv) providing supportive interventions for painful procedures; and (v) facilitating communication and collaboration between patients, their families, and the medical team.²⁴ These psychosocial support interventions have been shown to contribute to clinically meaningful reductions in distress.²⁴

Similar to prior RCTs and observational studies of MT with this population, patients in the HemOnc group had a mean age between 10 and 14 years,^{4, 25, 26} and malignant hematology conditions were the most prevalent diagnoses.^{4, 8, 18, 25, 26} Female sex was slightly more common in both groups (52.3%), differing from prior RCTs and observational studies where participants were mostly male.^{4, 8, 18, 25, 26} Black/African American patients made up a higher proportion within our combined sample (43.2%) and HemOnc group (21.1%) than a prior observational study by Yun et al. (12.0%)⁴ and a prior RCT by Phipps et al. (15.2%).²⁶

Few MT studies have described specific MT session characteristics. MT interventions in this study lasted 32.2 ± 14.4 minutes on average, similar to a prior study by Post-White et al. (29.2 ± 11.4 minutes).¹⁸ Descriptions of MT techniques are essential for examining what constitutes safe and effective clinical practice in this population. Though MT practitioners may differ in their therapeutic approach, it is important for qualified CPMTs to conduct assessments to determine which MT techniques are appropriate for each patient. Systematically obtaining this important data also facilitates future research to compare the effectiveness of different MT techniques and to examine the optimal delivery of MT during patients’ hospital admissions.

Comparing changes in PROs with those from prior RCTs is challenging, given the heterogeneity in outcome measures.⁷ When comparing adjusted PROs reported among the current sample to three prior studies that utilized a pain NRS, patients in the HemOnc group reported higher mean pain intensity (adjusted mean: 4.46) than pediatric patients with cancer reporting 0–10 pain scale measures in Post-White et al. (all scores less than 2)¹⁸ and Batalha and Mota (median = 3),⁸ but lower than scores reported by children receiving palliative care and MT in a pilot study by Genik et al. (mean = 5.20 ± 2.68).²⁷

Patients in the SCD group reported significantly higher pretreatment pain intensity (adjusted mean = 6.95 vs. 4.46), stress (adjusted mean = 6.47 vs. 4.58), and anxiety (adjusted mean = 6.67 vs. 4.59) than patients in the HemOnc group. While acute cancer-related pain can arise from tumors impinging on the nerves, mucositis, and/or chemotherapy-induced peripheral neuropathy,²⁸ acute SCD pain typically arises from vaso-occlusive episodes, which are unpredictable and can occur with great frequency throughout childhood.²⁹ A recent longitudinal study of patients ages 10–18 with SCD and chronic pain found pain catastrophizing to be prevalent and predictive of future pain interference.³⁰ Additionally, pain in SCD is often undertreated due to factors including negative provider attitudes and structural racism.^{15, 31} Thus, a combination of physiological, psychosocial, and systems-level factors may have contributed to patients with SCD reporting higher pretreatment pain, stress, and anxiety in this study.

Despite these differences in pretreatment PROs, both the HemOnc and SCD groups reported statistically significant reductions in pain, stress, and anxiety, and the majority reported relief meeting clinically significant thresholds. The mean pain change within the SCD group (adjusted mean: −1.87) is significant, given the substantial pain burden within this population. This change exceeds the minimum clinically significant improvement reported in a prior study of patients aged 8–18 receiving pain treatment (0.9 units)³² and the average pain reduction associated with a day of pharmacologic treatment in the hospital (0.5 units).³³ Similar pain reduction was reported among 19 children with SCD receiving acupuncture (1.33 units).³⁴ Thus, our study supports the inclusion of patients with SCD within pediatric integrative oncology programs. Additionally, our data support MT's clinical effectiveness and inclusion as an evidence-based, nonpharmacologic pain modality in accordance with the 2018 Joint Commission guidelines for hospitals to promote and provide nonpharmacologic pain treatments.^{35, 36}

Importantly, no adverse events were reported or charted in the EHR over the course of this retrospective study among patients with a high prevalence of coagulation defects (46.1%), neutropenia (39.1%), and immune system disorders (26.7%). This high safety profile is consistent with a prior observational study of acupuncture among children and adolescents with cancer, which found no increase in acute or delayed adverse events in patients with and without thrombocytopenia or neutropenia.³⁷ Though a small number of adverse events have been reported in other adult medical populations,^{38, 39} it is clear that MT can be implemented safely for patients with a range of hematologic and/or oncologic conditions⁴⁰ including metastatic bone pain.⁴¹ Patients with hematologic and/or oncologic conditions may be at higher risk for adverse events, such as (i) bleeding (ranging from minor bruising to internal hemorrhage during periods of thrombocytopenia); (ii) fracture related to bone metastases; (iii) increased pain; (iv) infection of open wounds; (v) radiation dermatitis; and (vi) dislodgement of a thrombus in deep vein thrombosis.⁴⁰ However, when conditions such as these are present, CPMTs are trained to take appropriate safety precautions including: (a) reducing massage pressure when patients present with low blood counts; (b) not providing massage directly on tumors, bruises, areas with compromised skin integrity, or areas where invasive procedures were recently performed; and (c) ensuring that massage lotion does not come in contact with intravenous lines or radiated body sites. Thus, advanced training in pediatric MT should be considered an essential qualification for personnel providing MT within pediatric academic cancer care.

Limitations are common in all research, and limitations in the current study include the lack of PRO data collection within the entire sample; lack of relevant measures of other PROs including nausea, fatigue, and constipation where MT may have been effective; lack of a method for controlling for pain medications that may have been provided to patients immediately prior to MT; the use of observational data among a convenience sample without a control group as PROs are not reliably obtained from all pediatric patients; the potential for response bias (e.g., reporting a lower PRO score to please the CPMT) among patients providing post-session PROs; and the use of single-item NRS scores rather than more comprehensive instruments for pain, stress, and anxiety. Furthermore, this study was conducted at a tertiary pediatric academic cancer center where the inpatient and outpatient areas were co-located in the same building, so the generalizability of findings to other facilities may be limited. However, there are many strengths of this study that include the large sample size, diversity of sociodemographic and clinical populations, novel approach to using EHR data to measure the real-world effectiveness of MT, collection of PROs immediately before and after MT sessions, and the inclusion of patients reporting severe symptoms.

This retrospective study supports the short-term clinical effectiveness and safety of MT provided by a CPMT for addressing acute pain, stress, and anxiety among children, adolescents, and young adults with hematologic and oncologic conditions. Within a large pediatric academic medical center, MT services can be integrated within inpatient and outpatient care to enhance symptom management and psychosocial support. Future research is needed to optimize the delivery and effectiveness of MT, both individually and in collaboration with other integrative treatments (e.g., acupuncture and music therapy), for addressing these patients’ needs. Given the higher symptom burden reported by the SCD group, this study also supports the inclusion of individuals with SCD within integrative oncology services such as MT. Additionally, future studies could examine how best to optimize MT intervention delivery to meet the needs of children, adolescents, and young adults with hematologic and/or oncologic conditions throughout their treatment journey.

CONFLICT OF INTEREST STATEMENT

The authors declare no conflict of interest.

FUNDING INFORMATION

Elisabeth Severance Prentiss Foundation (Cleveland, OH).