Characteristics of Healthcare Settings where Adolescents and Young Adults (AYAs) Receive Care for Acute Lymphoblastic Leukemia (ALL)

Abstract

Background:

Individuals diagnosed with cancer between 15 and 39y (AYA: adolescent and young adult) face unique vulnerability. Detail is lacking about care delivery for these patients, especially those with acute lymphoblastic leukemia (ALL). We address these knowledge gaps by describing AYA ALL care delivery details at NCI Community Oncology Research Program (NCORP) (sub)affiliates by model of care.

Methods:

Participating institutions treated at least one AYA with ALL from 2012–2016. Study-specific criteria were used to determine the number of unique clinical facilities (CF) per NCORP, and their model of care (adult/internal medicine (IM), pediatric, mixed [both]). Surveys completed by NCORPs for each CF by model of care captured size, resources, services, and communication.

Results:

Among 84 participating CFs (adult/IM=47, pediatric=15, mixed=24), 34% treated 5–10 AYAs with ALL annually; adult/IM CFs more often treated <5 (adult/IM: 60%, pediatric: 40%, mixed: 29%). Referral decisions were commonly driven by an age/diagnosis combination (58%), with frequent ALL-specific age minimums (87%) or maximums (80%). Medical, navigational, and social work services were similar across models while psychology was available at more pediatric CFs (pediatric: 80%, adult/IM: 40%, mixed: 46–54%). More pediatric or mixed CFs reported oncologists interacting with pediatric/adult counterparts, via tumor boards (pediatric: 93%, adult/IM: 26%, mixed: 96%) or initiating contact (pediatric: 100%, adult/IM: 77%, mixed 96%); more pediatric CFs reported an affiliated counterpart (pediatric: 53%, adult: 19%). Most CFs reported no AYA-specific resources (79%) or meetings (83–98%).

Conclusions:

System-level aspects of AYA ALL care delivery have not been examined previously. At NCORPs, these characteristics differ by models of care. Additional work is ongoing to investigate the impact of these facility-level factors on guideline-concordant care in this population. Together, these findings can inform a system-level intervention for diverse practice settings.

INTRODUCTION

Survival rates for individuals diagnosed with acute lymphoblastic leukemia (ALL) between 15 and 39y (AYA: adolescents and young adults) remain inferior to those in children (1–14y) and have not seen the extent of survival improvements evidenced by children.^1–6 These observations led the National Cancer Institute (NCI) to prioritize addressing the unique vulnerability in AYAs.^7–10

AYAs with ALL face survival differences based on where they receive care.⁶ Treatment regimens are one aspect of care that differ by site; superior AYA ALL survival has been observed with pediatric-inspired rather than adult-style therapy.¹¹ However, numerous aspects of care delivery have not been examined at the facility level. The majority of AYAs with ALL are treated at community cancer facilities rather than at specialized pediatric oncology or academic facilities; the proportion of AYAs at community centers increases with age^6,12,13 (1–14y: 30%, 15–21y: 49%, 22–39y: 88%),⁶ emblematic of the unique AYA interface with the U.S. healthcare system.¹⁴ Thus, it is crucial to study AYA care delivery in the context of community oncology.

Among AYAs with ALL, numerous patient-level explanations for inferior outcomes have been suggested (clinical, biological, behavioral), but minimal attention has been given to cancer care delivery.^11,15–21 When examining care delivery among AYAs, it is key to do so along the lines of model of care, as AYAs are treated in a variety of settings. These range from providing care in an adult/internal medicine model (IM: adult services/specialists only), a pediatric model (pediatric services/specialists only), or a mixed model (pediatric services/specialists embedded within a general hospital). These models differ along the lines of care organization²² and practice differences (comprehensiveness, family centered-ness, biopsychosocial approaches to care).^22–24 Robust evaluation of healthcare delivery uses the Donabedian Framework, requiring attention to domains within the structure (scaffolding of the system) and process (provision of care) of healthcare delivery.^25–28 Many inherent differences between how care is delivered in IM and pediatric-oriented practice models²⁹ are structural facets of individualistic (adult) vs. paternalistic (pediatric) models of care.²⁹

The NCI Community Oncology Research Program (NCORP) network, which provides opportunities to enroll onto research studies while remaining in the community close to support networks, is optimal for AYA-focused facility-level research.³⁰ NCORP research bases (currently seven) develop/coordinate cancer research for NCORP sites (currently 32 community, 14 minority-underserved [MU_NCORP: patient population comprised of at least 30% racial/ethnic minorities or rural residents]).³⁰ Each NCORP encompasses a varied number of healthcare practices (currently termed affiliates or sub-affiliates). [Supplemental-Fig.1A]³⁰ Although NCORP has periodically surveyed its network to identify research capacity, none of the Landscape surveys (v1.0 [2015],³¹ v2.0 [2017],³² or v3.0 [2022]) captured disease-specific or age-specific data.

Our aim was to describe aspects of the structure and process of care delivery for AYAs with ALL in the NCORP network, as a surrogate for real-world care for AYA ALL. This study operationalizes structure by first separating models of care as predominantly pediatric vs. adult/IM vs. mixed, then examining domains within structure and process according to these three models. The research presented here is one aspect of a comprehensive investigation of NCORP care delivery in AYA ALL.

METHODS

Eligible institutions were sub(affiliates) within an NCORP between 12/2017 and 04/2022 and treated at least one AYA (15–39y) with B- or T-cell ALL between 2012–2016, regardless of clinical trial participation.

Among NCORPs and (sub)affiliates that activated the study, the study team clarified the final subset of participating (sub)affiliates/NCORPs; this was accomplished via 23 months of outreach and iterative data submission to confirm eligibility (treating AYAs with ALL). Each (sub)affiliate registered via their preferred research base, whether pediatric (Children’s Oncology Group [COG]) or adult (SWOG Cancer Network [SWOG], ECOG-ACRIN Cancer Research Group [ECOG-ACRIN], Alliance for Clinical Trials in Oncology [Alliance]).³³ [Supplemental-Fig.1B]³³ COG led this study with Study Champions from Alliance, SWOG, and ECOG-ACRIN. (Sub)affiliates were registered according to their identification code for the Cancer Therapy Evaluation Program (CTEP), which serves as their identifier for the NCI and NCORP.³⁴ However, this code represents the institutional research and regulatory structures rather than the clinical structure and services for the delivery of care. Thus, institutions completed a preliminary (pre-) questionnaire to determine how many unique clinical facilities (CFs) were represented by their CTEP ID. [Supplemental-Fig.1C]

For the purposes of the study, the pre-questionnaire defined a CF as an independent clinical structure that could (but was not required to) be a distinct (sub)affiliate. Study criteria defining a CF included separate requirements/procedures for admitting privileges and requirement for hospital transfer documentation to move patients between facilities. Supporting characteristics included separate labs, buildings (regardless of tunnels or bridges), and/or admitting procedures. These criteria were captured with a series of questions to help determine the number of CFs.

For each CF, the site answered a series of questions to determine the model of care (pediatric, adult/IM, mixed [pediatric services embedded within a general healthcare facility]): who treated cancer (pediatric oncologists; medical oncologists; both), and what inpatient/outpatient oncology services were available (pediatric; medical/adult; both). Any inconsistencies between responses to questions and CF quantity/classification were discussed directly between the Study Chair/Co-Chair and the NCORP and/or (sub)affiliate. If CTEP IDs were shared between CFs that operated separately, each CF completed a questionnaire. One CF could have more than one CTEP ID. If CFs operated separately (regardless of a shared academic affiliation or health system), they completed separate questionnaires.

Site questionnaires (SiteQQs) were developed based on domains identified in the literature along with NCORP stakeholder input, then tailored into versions for each care model (pediatric, adult/IM, mixed).³⁵ SiteQQs captured CF structure (facility size,^36,37 practice size, training programs,^6,13,37,38 affiliations, electronic medical record [EMR]) and process (AYA referral process, age minimum or maximum, resources, physician-staff communication across pediatric/adult oncology,^39,40 clinical pathways for determining cancer care). Participating institutions completed a SiteQQ for each CF. To optimize data accuracy in each domain, a team approach to SiteQQ responses was encouraged; one senior individual within the NCORP or (sub)affiliate with specific responsibility or knowledge was asked to review the completed questionnaire (examples: NCORP Principal Investigator (PI), Administrator, or COG PI or CCDR Responsible Individual, etc.). Facility-reported NCI designations were cross-referenced with publicly available data. Questionnaires (pre-, SiteQQ) are available on request.

In order to describe the structure and process of care delivery for AYAs with ALL in the NCORP network, we describe continuous variables using medians (interquartile range [IQR]) and categorical variables by presenting the number and percentage of CFs per category. Kruskal Wallis and Fisher’s Exact tests were used to compare differences by care model. Due to the descriptive nature of this work, we did not adjust for multiple testing, and therefore statistical significance should be interpreted with care. This study was approved by the Pediatric Central Institutional Review Board (Rockville, MD), and local institutional review boards in accordance with institutional policies. R version 4.2.2 was use for analyses.

RESULTS

This study was activated by 33 (of 46) NCORPs on behalf of their associated 481 affiliates and sub-affiliates. After study outreach procedures and iterative data submission, 31 NCORPs confirmed they treated AYAs with ALL and would participate. Pre-questionnaires confirmed 86 unique CFs were represented. SiteQQs were submitted between study activation (12/2017) and 03/2020. [Supplemental-Fig.1C] General structure- and process-related characteristics of participating CFs are presented in Table 1, with AYA-specific process-related characteristics in Table 2.

Table 1:

Characteristics of Healthcare Facilities treating Adolescents and Young Adults with Acute Lymphoblastic Leukemia^*

	All Clinical Facilities (n=86)	Pediatric Model (n=15)	Adult/ Internal Medicine Model (n=47)	Mixed Model* (n=24)	P-value
STRUCTURE-RELATED CHARACTERISTICS
Total Inpatient Beds Median (Q1, Q3)
	248 (128, 456)	154 (108, 195)	259 (120, 483)	377.5 (252.5, 640.5)	0.003
Number of Credentialed Oncologists Median (Q1, Q3)
Medical Oncology	8 (5, 12)	NA	7 (5, 11)	10 (6.5, 15.5)	0.17
Pediatric Oncology	4 (3, 7)	7 (4, 9)	NA	4 (2.5, 5.5)	0.004
Residents are routinely involved in the care of patients N(%)
Medicine only	17 (19.77%)	0 (0%)	13 (27.66%)	4 (16.67%)	<0.001
Pediatrics only	13 (15.12%)	10 (73.33%)	0 (0%)	2 (8.33%)
Medicine and pediatrics	7 (8.14%)	0 (0%)	0 (0%)	7 (29.17%)
No residents	49 (56.98%)	4 (26.67%)	34 (72.34%)	11 (45.83%)
We have a Fellowship Program in ______ Hematology-Oncology N(%)
Medical/adult only	11 (12.79%)	0 (0%)	7 (14.89%)	4 (16.67%)	<0.001
Pediatric only	6 (6.98%)	6 (40.00%)	0 (0%)	0 (0%)
Medical and Pediatric	2 (2.33%)	0 (0%)	0 (0%)	2 (8.33%)
None	67 (77.91%)	9 (60.00%)	40 (85.11%)	18 (75.00%)
AYA	0 (0%)	0 (0%)	0 (0%)	0 (0%)
Facility Status**
University-affiliated	21 (24.42%)	4 (26.67%)	9 (19.15%)	8 (33.33%)	0.38
NCI-designated CCC	19 (22.09%)	3 (20.00%)	9 (19.15%)	7 (29.17%)	0.60
MU_NCORP	16 (18.60%)	7 (46.67%)	5 (10.64%)	4 (16.67%)	0.01
Other NCORP	79 (91.86%)	13 (86.67%)	43 (91.49%)	23 (95.83%)	0.58
Number of Other Facilities You are Affiliated With
1 pediatric facility	9 (10.47%)	NA	9 (19.15%)	NA	*
>1 pediatric facility	0 (0%)	NA	0 (0%)	NA
1 adult facility	6 (6.98%)	6 (40.00%)	NA	NA
>1 adult facility	2 (2.33%)	2 (13.33%)	NA	NA
Electronic Medical Record (EMR)
Facility has an EMR	84 (97.67%)	13 (86.67%)	47 (100%)	24 (100%)	0.03
EMR has alerts for non-lab items	37 (43.02%)	6 (40.00%)	17 (36.17%)	14 (58.33%)	0.11
PROCESS-RELATED CHARACTERISTIC
What Drives Referrals?
Age only	15 (17.44%)	3 (20.00%)	9 (19.15%)	3 (12.50%)	0.95
Insurance	1 (1.16%)	0 (0%)	1 (2.13%)	0 (0%)
Age + Diagnosis	50 (58.14%)	10 (66.67%)	24 (51.06%)	16 (66.67%)
Referrals Administrative Team	1 (1.16%)	0 (0%)	1 (2.13%)	0 (0%)
Referring Physician Preference	6 (6.98%)	0 (0%)	3 (6.38%)	3 (12.50%)
None of the Above	9 (10.47%)	2 (13.33%)	5 (10.64%)	2 (8.33%)
I donť know how this decision is made	4 (4.65%)	0 (0%)	4 (8.51%)	0 (0%)
Age Minimum/Maximum Policies
Age Minimum - ALL patients	41 (47.67%)	NA	41 (87.23%)	NA	*
Median (Q1, Q3) - years	18 (18, 18)	NA	18 (18, 18)	NA
Age Minimum - Other Cancers	42 (48.84%)	NA	42 (89.36%)	NA
Median (Q1, Q3) - years	18 (18, 18)	NA	18 (18, 18)	NA
Age Maximum - ALL patients	12 (13.95%)	12 (80.00%)	NA	NA
Median (Q1, Q3) - years	21.5 (21, 27.5)	21.5 (21, 27.5)	NA	NA
Age Maximum - Other Cancers	9 (10.47%)	9 (60.00%)	NA	NA
Median (Q1, Q3) - years	21 (21, 25)	21 (21, 25)	NA	NA
Oncologists Attend Each Other's (Medicine/Pediatrics) Tumor Boards (Frequency)
Never	36 (41.86%)	1 (6.67%)	34 (72.34%)	1 (4.17%)	<0.001
On a per-patient basis	41 (47.67%)	12 (80.00%)	11 (23.40%)	18 (75.00%)
Every 3–12 months	2 (2.32%)	0 (0%)	0 (0%)	2 (8.33%)
Every 2 months or more frequently	6 (6.97%)	2 (13.33%)	1 (2.13%)	3 (12.50%)
N/A	1 (1.16%)	0 (0%)	1 (2.13%)	0 (0%)
Oncologists Contact Each Other (across Medicine/Pediatrics) Outside of Tumor Boards (Frequency)
Never	11 (12.79%)	0 (0%)	10 (21.28%)	1 (4.17%)	0.01
On a Per-patient basis	70 (81.40%)	13 (86.67%)	34 (72.34%)	23 (95.83%)
Every 3–12 months	2 (2.32%)	2 (13.33%)	0 (0%)	0 (0%)
Every 2 months or more	2 (2.32%)	0 (0%)	2 (4.26%)	0 (0%)
Daily	0 (0%)	0 (0%)	0 (0%)	0 (0%)
N/A	1 (1.16%)	0 (0%)	1 (2.13%)	0 (0%)
Clinical Pathways in Place***
General Clinical Pathway: Adults	39 (45.35%)	0 (0%)	38 (80.85%)	1 (4.17%)	<0.001
General Clinical Pathway: Children	19 (22.09%)	11 (73.33%)	1 (2.13%)	7 (29.17%)
General Clinical Pathway: Adult + Children	18 (20.93%)	4 (26.67%)	3 (6.38%)	11 (45.83%)
Clinical Pathway: AYA ALL	52 (60.47%)	13 (86.67%)	26 (55.32%)	13 (54.17%)	0.07
Clinical Pathway: AYA, non-ALL	48 (55.81%)	12 (80.00%)	25 (53.19%)	11 (45.83%)	0.09

^*Because the survey questions differed by site type (Pediatric, Adult, Mixed), NA's are included for questions not asked of that specific site type. Formal hypothesis testing was not conducted for questions that were only answered by one facility model type.

^**CCC: Comprehensive Cancer Center; MU_NCORP: Minority-Underserved NCORP;

^***Question stem reads: Does your facility have a clear algorithm (or "clinical pathway") for determining cancer therapy for...

Table 2:

AYA-specific Process-related Characteristics of Healthcare Facilities treating Adolescents and Young Adults with Acute Lymphoblastic Leukemia

	All Clinical Facilities (n=86)	Pediatric Model (n=15)	Adult/ Internal Medicine Model (n=47)	Mixed Model (n=24)	P-Value
AYA-Specific Resources*
AYA Coordinator	6 (6.98%)	2 (13.33%)	2 (4.26%)	2 (8.33%)	0.31
AYA Navigator	5 (5.81%)	1 (6.67%)	3 (6.38%)	1 (4.17%)	1.0
AYA Social Worker	10 (11.63%)	2 (13.33%)	3 (6.38%)	5 (20.83%)	0.17
AYA Psychology	8 (9.30%)	2 (13.33%)	2 (4.26%)	4 (16.67%)	0.17
AYA Oncologist	12 (13.95%)	2 (13.33%)	3 (6.38%)	7 (29.17%)	0.04
No AYA-specific resources	68 (79.07%)	12 (80.00%)	41 (87.23%)	15 (62.50%)	0.06
AYA-specific Tumor Board (Frequency)
Never	81 (94.19%)	14 (93.33%)	46 (97.87%	21 (87.50%)	0.11
Every 3–12 mos	2 (2.32%)	1 (6.67%)	0 (0%)	1 (4.17%)
Every 2 mos or more	3 (3.49%)	0 (0%)	1 (2.13%)	2 (8.33%)
AYA-specific Journal Club (Frequency)
Never	84 (97.67%)	14 (93.33%)	46 (97.87%)	24 (100%)	0.40
Every 3–12 mos	2 (2.32%)	1 (6.67%)	1 (2.13%)	0 (0%)
Every 2 mos or more	0 (0%)	0 (0%)	0 (0%)	0 (0%)
AYA-specific Research Meeting (Frequency)
Never	77 (89.53%)	13 (86.67%)	42 (89.36%)	22 (91.67%)	0.67
Every 3–12 mos	2 (2.32%)	1 (6.67%)	1 (2.13%)	0 (0%)
Every 2 mos or more	7 (8.14%)	1 (6.67%)	4 (8.51%)	2 (8.33%)
AYA Program Meeting (Frequency)
Never	71 (82.56%)	11 (73.33%)	42 (89.36%)	18 (75.00%)	0.13
Every 3–12 mos	5 (5.81%)	2 (13.33%)	1 (2.13%)	2 (8.33%)
Every 2 mos or more	10 (11.63%)	2 (13.33%)	4 (8.51%)	4 (16.67%)

^*Question reads: Do you have staff at your facility who have been specifically designated to provide services for AYAs (these individuals may also care for other age groups)? Check all that apply.

Structure: Facility Characteristics

Facility Model:

Among participating CFs, 47 (55%) were classified as an adult/IM model, 15 (17%) as pediatric, and 24 (28%) as mixed.

Size:

Mixed CFs had the most inpatient beds (median=378; IQR, 253–641), followed by adult/IM (median=259; IQR, 120–483) and pediatric (median=154; IQR, 108–195) CFs. The median number of credentialed oncologists at pediatric and adult/IM CFs was in the same range, while mixed model CFs had fewer pediatric and more medical oncologists. Each year, adult/IM CFs more commonly saw <5 AYA ALL patients (adult/IM=60%, pediatric=40%, mixed=29%) and less commonly saw >10 AYA ALL (adult/IM=4%, pediatric=20%, mixed=29%). [Fig.1]

Figure 1.

Annual Volume of Adolescent and Young Adult (AYA) Patients with Acute Lymphoblastic Leukemia (ALL) according to model of care (pediatric, adult/internal medicine, mixed).

Training Programs:

More adult/IM (72%) than mixed (46%) or pediatric (27%) CFs reported no residents being involved in routine patient care. There were no hematology-oncology fellows at the majority of CFs (adult/IM=75%, pediatric=60%, mixed=85%. No CFs had a dedicated AYA Oncology fellowship.

Affiliations:

The minority of CFs were affiliated with a university (adult/IM:=19%, pediatric=27%, mixed: 33%), part of an NCI-designated Comprehensive Cancer Center (CCC) (adult/IM=19%, pediatric=20%, mixed=29%) and/or part of a minority-underserved NCORP (MU_NCORP) (adult/IM=11%, pediatric=47%, mixed=17%). Only 19% of adult/IM CFs but 53% of pediatric CFs (19%) reported an affiliation with a counterpart (pediatric or adult/IM) facility.

Structure: EMR

Most pediatric and all other CFs had an EMR system in place. Nearly half of the EMRs could provide alerts unrelated to lab values.

Process: Facility-level Policies

Referral Process:

The most common process to determine which oncology team (medical/pediatric) would accept a new patient was a combination of age and diagnosis (adult/IM=51%, pediatric=67%, mixed=67%) or age alone (adult/IM=19%, pediatric=20%, mixed=13%).

Age Minimum/Maximum Policies:

The majority of pediatric CFs had an age maximum (median=21y) for ALL (80%), and other cancers (60%). The overwhelming majority of adult/IM CFs had an age minimum (median=18y) for ALL (87%) and other cancers (89%).

Process: Resources

General Medical Resources:

Advanced practice providers (APP: nurse practitioner or physician’s assistant) were more common in the outpatient setting among adult/IM (inpatient=45%, outpatient=79%) and pediatric (inpatient=60%, outpatient=67%) CFs; mixed CFs reported APPs more commonly for adult (inpatient=50%, outpatient=83%) than pediatric (inpatient=25%, outpatient=54%) oncology. The majority of CFs reported a pharmacist on their oncology service. [Fig.2A]

Figure 2.

These panels depict the size, services and resources available by clinical facility model of care. (A) General Medical Services; (B) General Navigational Facilities; (C) General Psychosocial Services; (D) Other Services available by clinical facility model of care.

General Navigational Resources:

While the majority of CFs reported a nurse coordinator or navigator for their oncology service, a minority reported a clinical (non-nurse) or lay navigator for their oncology service. [Fig.2B]

General Psychosocial Resources:

Most CFs reported a social worker on their oncology service (adult/IM=89%, pediatric=93%; mixed/adult service=83%, mixed/pediatric service=88%). While the majority of pediatric CFs (80%) reported a psychologist for their service, only 54% of mixed/pediatric services, and a minority of adult (40%) or mixed/adult (46%) CF services included a psychologist. [Fig.2C]

Other General Resources:

The SiteQQ also provided CFs the opportunity to state ‘none of the above’ were part of their oncology team (some adult/IM and mixed CFs reported this, but no pediatric CFs), or voluntarily free-text other/additional individuals on their team. Additional medical personnel (13%) and genetic counseling (9%) were reported only by adult/IM CFs while additional psychosocial providers were reported more often in pediatric (57%) and mixed (38%) CFs than adult/IM CFs (11%). [Fig.2D, Supplemental-Table 1]

AYA-specific Resources:

Most CFs reported no AYA-specific resources, while a minority of CFs did report some: AYA Social Work (adult/IM=6%, pediatric=13%, mixed=21%), AYA Oncologist (adult/IM=6%, pediatric=13%, mixed=29%), AYA Psychology (adult/IM=4%, pediatric=13%, mixed=17%), AYA Navigator (adult/IM=6%, pediatric=7%, mixed=4%) and/or AYA Coordinator (adult/IM=4%, pediatric=13%, mixed=8%).

Process: Physician-Staff Communication

Contact across Services:

CFs reported the majority of oncologists informally contacted their counterparts in adult/pediatric oncology on a per-patient basis (adult/IM=72%, pediatric=87%, mixed=96%). However, 22% of adult/IM and 4% of mixed CFs reported oncologists never contacted their counterparts; no pediatric CFs lack contact.

General Tumor Boards:

Oncologists at pediatric and mixed CFs attended their counterpart’s tumor boards (medical/pediatric oncology) more often than oncologists at adult/IM CFs (never=72%), whether it was on a per-patient basis (pediatric=80%, mixed=75%) or at least every 2 months (pediatric=13%; mixed=13%).

AYA-Specific Tumor Boards and Meetings:

The majority of CFs reported having no AYA-specific meetings or tumor boards. However, the most commonly reported AYA-specific meetings were a program meeting or AYA-specific research meeting. One pediatric, one adult/IM, and three mixed CFs reported an AYA Tumor Board that met at least once a year.

Process: Clinical Pathways

General Clinical Pathways:

The majority of adult/IM and pediatric CFs reported having general clinical pathways that determined cancer care (adult/IM=81%, pediatric=73%), while 45% of mixed CFs reported having one for both adults and children (only adults=1%, only children=29%).

AYA-specific Clinical Pathways:

Most pediatric CFs and half of adult/IM and mixed CFs reported having clinical pathways for the treatment of AYAs with both ALL (adult/IM=55%, pediatric=87%, mixed=54%) and other malignancies (adult/IM=53%, pediatric=80%, mixed=46%).

DISCUSSION

Here we describe the characteristics of NCORP CFs treating AYAs with ALL according to model of care (pediatric, adult/IM, mixed). Notably, 61% of adult/IM CFs treated very few AYAs with ALL (<5/yr), and roughly a third of all types of CFs treated only 5–10 AYAs with ALL each year, while only a fraction of CFs treated a larger volume (>25/yr).

The small volume of AYAs treated across all models causes pause when one considers that sites who did not activate or participate in the study may also be treating small volumes of AYA ALL, thus amplifying the noted differences. In the setting of low AYA ALL volume and minimal regular communication across pediatric/adult oncology, it is reassuring that at least half of CFs had AYA ALL clinical pathways; nevertheless, more detail regarding pathways and related outcomes would be warranted (as would more common use of pathways).

While size (reflected by number of beds and oncologists) and having an academic component (training programs, university affiliation, CCC) varied along models of care, the models were more similar in terms of how referrals were directed, and restrictions according to age. The models of care had relatively similar availability for general medical, navigational, and social work services. However, psychology was more commonly available at pediatric CFs; this support is crucial for AYAs, which are a developmentally unique group of patients who face enhanced risk of psychological distress, especially those not engaging with professional mental health services.^41,42 Furthermore, ‘other’ services were reported by half of pediatric CFs and a quarter of mixed CFs for their pediatric service; most of these were psychosocial services not otherwise included in the questionnaire.

Physician/staff collaboration across oncology services is cited as key to use of pediatric (vs. adult) therapy, along with clinical trial enrollment, which, in turn, is associated with survival.^40,43 While ALL is the most common malignancy in pediatric oncology, it is much rarer in adult oncology,^44,45 with distinct toxicities among children and AYAs.^46,47 These observations would suggest a role for ongoing communication between clinicians caring for AYAs with ALL. Among these facilities, the majority of adult/IM CFs reported they never attended their pediatric affiliates’ tumor boards, and some reported never contacting pediatric oncologists. However, the potential for this may be hampered by the fact that only a small proportion of adult/IM and pediatric CFs reported an affiliation with an adult/pediatric counterpart and the majority of CFs across all models report no AYA-specific resources or meetings. With this lack of formal affiliation or infrastructure, praise may instead be warranted for attempting contact, with the majority of any formal or informal contact between pediatric and adult oncologists reported to be on a per-patient basis.

Previous research has shown that AYA outcomes are superior for patients treated at NCI-designated comprehensive cancer centers,^6,12,13 (including AYAs with ALL) supporting the notion that elements of healthcare delivery (structure and/or process) are associated with AYA ALL outcomes. However, there is a dearth of data regarding care delivery in AYA ALL and even less information about which domains are associated with outcomes. The services and characteristics examined here have not previously been measured in the context of AYAs (including ALL). An association between these characteristics and survival outcomes is often presented as fact in opinion pieces or clinical discussion, but to our knowledge has not been described in detail nor investigated in this context. Given the numerous domains within structure and process relevant to AYA care delivery, it will be crucial to understand the impact of these domains on clinical outcomes among AYAs to identify targets for systems-level interventions to optimize AYA ALL care delivery. Before examining outcomes, it is first necessary to delineate how these domains differ across models of care in the AYA population, to avoid making presumptions based on a subset of facilities.

This study must be considered in the context of its limitations. The number of NCORP (sub)affiliates activating the study (481) was larger than the number that participated (86); the study team worked closely with activated NCORPs to refine the list of participating (sub)affiliates based on their treatment of any AYA ALL. The remainder of limitations are due to use of facility-level data. This includes the perception by some that they treated few or “no” patients but were eligible on further review. To optimize the most accurate data possible, at least one senior individual (whose NCORP role was captured) reviewed and signed off on the SiteQQ. Nevertheless, data accuracy regarding national designations was suboptimal. For example, some sites reported they were CCCs, but the designation was not confirmed when compared with publicly available data. Although it is conceivable that this question was initially answered by staff in the affirmative with the belief that NCORP and/or NCTN designations were included in the CCC designation, a review by senior personnel should have noticed this discrepancy. Despite these limitations, there are no secondary administrative datasets that include the vast majority of details collected here; thus, with the safeguards described, this represents the best available option.

Conclusions

In summary, we describe how system-level characteristics of CFs differ along the lines of models of care. The small volume of AYAs treated across models of care prompts us to consider ALL treatment itself, which is fraught with nuance (how to measure and respond to treatment response [minimal residual disease]), and risks ranging from induction death to those needing attentive management of serious complications, such as pancreatitis, infection, or thrombosis.⁴⁸ Diseases and populations seen in small volume who require nuanced treatment beg development of a mechanism to guide clinicians treating those patients to provide care that optimizes survival, such as clinical practice guidelines. While such a mechanism is in development by national organizations, it is important to examine the system-level factors identified here (by model of care) and their impact on clinical care and outcomes so that such a mechanism can be implemented effectively across diverse practice settings.

This work was accomplished in the context of a larger study that evaluates facility-level factors and delivery of guideline-concordant care. Providing care consistent with quality indicators can be intervened upon at a systems (rather than clinical) level to improve survival.⁴⁹ While clinical and biological advances continue in parallel, this avenue provides the potential to impact a large amount of AYAs with ALL.

Supplementary Material

Supplemental Figures

Supplemental Figure 1. The structure of two programs of the National Cancer Institute are outlined here including (A) National Community Oncology Research Program, and (B) National Clinical Trials Network and Network-affiliated Cooperative Groups. The cooperative groups are both adult (SWOG, Alliance, ECOG-ACRIN, NRG Oncology, Canadian Network Group) and pediatric (COG). (C) Determination of Participating Institutions and Unique Clinical Facilities, using the structure outlined in (A) and (B).

CONTEXT SUMMARY.

Key Objective:

To describe aspects of care delivery for AYAs with ALL along the lines of models of care (adult, pediatric, mixed), using the NCORP as a surrogate for real-world care.

Knowledge generated:

Across care models, clinical facilities (CF) treat low volumes of AYA ALL (most adult CFs treat <5/yr). Similar approaches were reported across care models for age limits, referrals, and availability of navigational and social work services, with psychology more common at pediatric CFs. A small proportion of adult and pediatric CFs were affiliated with an adult/pediatric counterpart, and most CFs had no AYA-specific services or meetings; most adult CFs never attended pediatric affiliates’ tumor boards, and some never contact pediatric oncologists.

Relevance:

The characteristics examined have not been measured in the context of AYAs, nor by model of care. Understanding the impact of these factors on clinical outcomes (such as guideline-concordant care) can inform a system-level intervention for diverse practice settings; this work is ongoing.