1
A new, in e disciplina y, mul i-s akeholde Eu opean ne wo k o imp o e
heal hca e se ices, esea ch and ou comes o Adolescen s and Young Adul s
wi h cance .
STRONG-AYA – No. 101057482 – D1.2
Deli e able Repo
WP1 – Repo on he ini ial Co e Ou come Se
Deli e able 1.2
Repo on he ini ial Co e Ou come Se
Due da e o deli e able: 30/09/2023
Ac ual submission da e: 28/03/2024
P ojec : STRONG-AYA
Lead Con ibu o UOS- Ki s y Way, Anne-Sophie Da ling on, Nicole Collaço
Email A.Da ling on@so on.ac.uk
O he Con ibu o s NKI – Sil ie Janssen
Emails [email p o ec ed]l
Due da e
30 09 2023
Deli e y da e
28 03 2024
Deli e able ype
R
Dissemina ion le el PU
Desc ip ion o Wo k Ve sion Da e
V1.0 27/03/2024
STRONG-AYA – No. 101057482 – D1.2
Desc ip ion:
This epo con ains he ini ial Co e Ou come Se , including he me hodology
ha ook place o i s de elopmen . Resul s om he li e a u e e iew,
quali a i e in e iews, Delphi su ey ounds as well as he consensus mee ing
ha e been epo ed o show he p agma ic decision-making p ocesses behind
he de elopmen o he Ini ial COS
summa y (max ½ page)
The aim o Wo k Package 1 (WP1) o STRONG AYA is o de elop a Co e Ou come Se (COS) o adolescen s
and young adul s (AYAs) wi h cance . The e is speci ic scien i ic me hodology ha mus be ollowed o
de elop a COS [1-4], as well as iden i y he measu emen s ins umen s o measu e he i ems in he COS
[5]. The s ages o making decisions on he inal ou comes o be included in a COS a e h ee- old: (1) A
e iew o he li e a u e o disco e which ou comes a e impo an o AYAs wi h cance , (2) quali a i e
in e iews wi h s akeholde s (AYA pa ien s, hei ca egi e s, and heal hca e p o essionals) o hea hei
pe cep ions o wha ou comes a e impo an o AYAs wi h cance , (3) consensus me hodology, known as
a Delphi s udy.
Following a e iew o he li e a u e and quali a i e in e iews, a o al o 129 ou comes we e accumula ed
and conside ed impo an o AYAs wi h cance . These 129 ou comes we e hen ca ego ised in acco dance
wi h Dodd’s 38-i em i em axonomy o ou comes in medical esea ch [6], which o med he skele on o
he Delphi su ey. The Delphi su ey has a o al o h ee ounds, which aims o come o a consensus
be ween s akeholde g oups on which ou comes a e he u mos impo an o measu e o AYAs wi h
cance . Following he i s wo ounds o he Delphi su ey, a o al o 61 i ems me he inclusion c i e ia
(ou comes ha ecei ed ≥70% esponden s a ing i as a 7-9 on he Like -scale) and we e aken o wa d
o a consensus mee ing, which in ol ed 12 membe s o he STRONG AYA conso ium o ming an expe
panel o make p agma ic decisions on he inal ou comes o be included in he Ini ial COS.
The Ini ial COS consis s o 21 domains; he e a e 15 CORE domains, 3 ON TREATMENT domains and 3 OFF
TREATMENT domains. The de elopmen o he Co e Measu emen Se o supplemen he ini ial COS is
s ill being de eloped. Round 3 o he Delphi su ey is s ill ongoing; esul s om he hi d and inal Delphi
ound will be used o in o m he Final COS. In he mean ime, he Ini ial COS will be implemen ed
h oughou he heal hca e esea ch ecosys em in u u e wo k packages in he STRONG AYA p ojec
STRONG-AYA – No. 101057482 – D1.2
1 Table o Con en s
PUBLISHABLE SUMMARY (MAX ½ PAGE) .............................................................................................................. 3
1 TABLE OF CONTENTS ..................................................................................................................................... 4
2 DEFINITIONS ................................................................................................................................................. 5
3 ABBREVIATIONS............................................................................................................................................ 6
4 INTRODUCTION ............................................................................................................................................ 7
PROJECT BACKGROUND ...................................................................................................................................... 7
DELIVERABLE INTRODUCTION .............................................................................................................................. 9
5 DEVELOPING THE INITIAL CORE OUTCOME SET............................................................................................ 10
BACKGROUND ............................................................................................................................................................. 10
METHODS .................................................................................................................................................................. 10
Phase 1: Scope de ini ion .................................................................................................................................... 11
Phase 2: Es ablishing he need o a COS o AYAs ............................................................................................. 11
Phase 3: Composi ion o AYA Wo king G oup .................................................................................................... 11
Phase 4: S udy p o ocol de elopmen ................................................................................................................ 12
Phase 5: De e mine “wha ” o measu e – he ou comes in he COS .................................................................. 12
Phase 6: De e mine “how” o measu e he COS ................................................................................................. 21
De e mine “case-mix a iables” ......................................................................................................................... 23
RESULTS .................................................................................................................................................................... 23
The Li e a u e Re iew ......................................................................................................................................... 23
Quali a i e In e iews ........................................................................................................................................ 24
The Delphi su ey ............................................................................................................................................... 25
FUTURE WORK ............................................................................................................................................................ 30
6 CONCLUSION ............................................................................................................................................... 31
7 REPOSITORY FOR PRIMARY DATA (ANNEX) ................................................................................................. 31
STRONG-AYA – No. 101057482 – D1.2
2 De ini ions
STRONG AYA conso ium membe s a e e e ed o as ollowing wi hin his ex :
1. NKI-AVL – S ich ing he Nede lands Kanke Ins i uu – An oni an Leeuwenhoek Ziekenhuis (NL)
2. YCE – You h Cance Eu ope (RO)
3. INT – Fondazione IRCCS Ins i u o Nazionale dei Tumo i (IT)
4. FFUND – FFUND BV (NL)
5. CLB – Cen e de Lu e Con e le Cance Leon Be a d (FR)
6. ECO – Eu opean Cance O ganisa ion (BE)
7. UNIMAAS – Uni e si ei Maas ich (NL)
8. IKNL – S ich ing In eg aal Kanke cen um Nede land (NL)
9. EORTC – Eu opean O ganisa ion o Resea ch and T ea men o Cance AISBL (BE)
10. IGR – Ins i u Gus a e Roussy (FR)
11. MSCNRIO – Na odowy Ins y u Onkologii im. Ma ii Sklodowskiej-Cu ie – Pans wowy Ins y u
Badawczy (Ma ie Sklodowska-Cu ie Na ional Resea ch Ins i u e o Oncology) (PL)
12. UOM – Uni e si y o Manches e (UK)
13. UOL – Uni e si y o Leeds (UK)
14. LTHT – Leeds Teaching Hospi als Na ional Heal h Se ice T us (UL)
15. SOUTHAMPTON – Uni e si y o Sou hamp on (UK)
 G an Ag eemen (including i s annexes and amendmen s): he ag eemen signed be ween he
bene icia ies o he HORIZON Resea ch and Inno a ions Ac ions (he ea e e e ed o as Ho izon)
and he Eu opean Heal h and Digi al Execu i e Agency (he ea e e e ed o as HADEA) o he
unde aking o he STRONG AYA p ojec (G an Ag eemen no. 101057482).
 Bene icia y: Signa o ies o he G an Ag eemen
 Associa ed Pa ne : En i ies which pa icipa e in he ac ion bu wi hou he igh o cha ge cos s
o claim con ibu ions.
 P ojec : he sum o all ac i i ies ca ied ou in he amewo k o he G an Ag eemen .
 Conso ium: he STRONG AYA conso ium, including all he a o emen ioned pa ne s.
 Conso ium Ag eemen : The ag eemen made be ween STRONG AYA membe s o he
implemen a ion and execu ion o he ac ion ou lined in he G an Ag eemen . The ag eemen
shall no a ec he pa ies’ obliga ions o HADEA on behal o he Eu opean Union, and/o o one
ano he a ising om he G an Ag eemen .

STRONG-AYA – No. 101057482 – D1.2
3 Abb e ia ions
Ac onym/Abb e ia ion Meaning
HCP Heal h Ca e P o ide
PRO Pa ien Repo ed Ou come
PROM Pa ien Repo ed Ou come Measu e
COS Co e Ou come Se
WP Wo k Package
WPL Wo k Package Lead(s)
WP1 Wo k Package 1 (De elopmen Co e Ou come Se
AYA wi h cance & da a collec ion)
WP2 Wo k Package 2 (Go e nance, Da a Secu i y and
E hics)
WP3 Wo k Package 3 (In as uc u e and In e ope abili y)
WP4 Wo k Package 4 (Ope a ion o STRONG AYA
ecosys ems, s akeholde and pa ien in ol emen ,
dissemina ion, exploi a ion, communica ion)
WP5 Wo k Package 5 (Scien i ic coo dina ion and p ojec
managemen )
KPI Key Pe o mance Indica o
OA Open Access
PAB Pa ien Ad iso y Boa d
EC Eu opean Commission
HADEA Eu opean Heal h and Digi al Execu i e Agency
SC S ee ing Commi ee
MT Managemen Team
STRONG-AYA – No. 101057482 – D1.2
4 In oduc ion
P ojec backg ound
Cance a adolescen and young adul (AYA) age is a e, al hough 4-6 imes mo e equen han paedia ic
cance (i.e. p epubescen pe iod). Howe e , his a i y does no e lec he signi ican pe sonal and
socie al cos s o cance in his popula ion, as e lec ed in he po en ial yea s o li e los o sa ed, he
dec eased p oduc i i y and quali y-o -li e due o he impac o he disease du ing o ma i e yea s, and
he long- e m complica ions o disabili ies1. AYAs wi h cance o m a unique g oup; hey ace age-speci ic
issues (e.g. In e ili y, unemploymen , inancial p oblems) and dec eased quali y o li e due o cance and
i s ea men . Unlike dedica ed heal hca e and ials o paedia ic cance pa ien s, AYA-speci ic
heal hca e se ices a e sca ce and a y ac oss Eu ope. AYAs who a e a he co e o socie y and economy
need access o age-app op ia e and high-quali y heal hca e.
De ining AYAs wi h cance as 15 o 39 yea s a ini ial cance diagnosis2, hei annual cance incidence is
42.2/100.000, wi h 156.431 cases in Eu ope and 1.231.007 cases wo ldwide epo ed in 2018 ( oge he
6.8% o all cance s)3. Popula ion-based da a om 27 Eu opean coun ies suppo s ha AYAs ha e lowe
su i al han child en bu highe han adul s a ec ed by cance . Ad ances in cance ea men ha e led
o inc eased su i al a es o AYAs wi h cance , imp o ing by 82% o all cance s be ween 1990 and
20074.
Howe e , su i al imp o emen in AYA is mo e challenging han o child en and olde cance su i o s,
which migh be due o he ac ha AYA ha e he highes absolu e excess isk o second p ima y malignan
neoplasms5. AYA ace some dis inc challenges gi en ha hey do no belong o nei he paedia ic no
adul oncology g oups. Cha ac e is ic ea u es o his popula ion g oup include unique spec um o cance
ypes, di e en umou biology, unique complex psychological needs, dis inc la e sequelae, including
impai ed e ili y, and pallia i e ca e. These ai s imply ha clinical managemen , ea men , diagnosis,
psychological suppo will need o be designed and de eloped o AYA’s speci ic needs. Fo example, AYAs
diagnosed wi h b eas and p os a e ca cinomas ha e wo se su i al han olde pa ien s because o he
biological di e ences be ween hem, highligh ing he need o a ge sc eening me hodologies, ea men
and policies o hei needs6. AYAs wi h cance also ace signi ican psychological challenges, including
subs ance abuse, men al heal h issues, suicidal idea ions and inc eased emo ional bu den om cance
and cance - ela ed mo bidi y. Finally, ailo ing cance ca e o AYA’s needs is di icul and due o many
di e en complex ac o s including he low a e o pa icipa ion by AYAs in clinical ials and cance
esea ch7.
1 S oneham SJ. AYA su i o ship: The nex challenge. Cance 2020; 126: 2116-2119.
2 Adolescen and Young Adul Oncology Re iew G oup. Closing he gap: Resea ch and Ca e Impe a i es o Adolescen s and Young Adul s wi h Cance .
Na ional Ins i u e o Heal h, Na ional Cance Ins i u e, and Li es ong Young Aul Alliance: Be hesda, MD, USA, 2006.
3 T ama A, Bo a L, S elia o a-Fouche E. Cance Bu den in Adolescen s and Young Adul s: A Re iew o Epidemiological E idence. Cance J 2018; 24: 256-
266
4 T ama A, Bo a L, Foschi R e al. Su i al o Eu opean adolescen s and young adul s diagnosed wi h cance in 2000-07: popula ion-based da a om
EUROCARE-5. Lance Oncol 2016; 17: 896-906.
5 Keegan THM, Bleye A, Rosenbe g AS e al. Second P ima y Malignan Neoplasms and Su i al in Adolescen and Young Adul Cance Su i o s. JAMA
Oncol 2017; 3: 1554-1557.
6 S a k D, Bielack S, B ugie es L e al. Teenage s and young adul s wi h cance in Eu ope: om na ional p og ammes o a Eu opean in eg a ed coo dina ed
p ojec . Eu opean Jou nal o Cance Ca e, 25(3), 419–427.
7 Hayashi RJ. Adolescen and young adul cance su i o ship: The new on ie o in es iga ion. Cance 2019; 125: 1976-1978.
STRONG-AYA – No. 101057482 – D1.2
Despi e he inc easing awa eness and a g owing body o he scien i ic li e a u e, hese unique issues
emain o be ully ecognised and add essed by he Eu opean heal h sys ems and AYAs wi h cance a e
equen ly unde se ed. In pa , his may ha e esul ed om he adi ional dicho omy be ween he
in eg a ed paedia ic (“pa ien / amily-cen ed”) ca e se ices e sus dispe sed (“disease- cen e ed”)
adul oncology se ices8,9,10. Up o hal o AYAs wi h cance epo unme in o ma ional and se ice needs,
impac ing hei di ec (su i al a es) and indi ec (long e m e ec s and men al heal h) eco e y o
pa icipa e in socie y11.
Fu he mo e, aligned wi h his ba ie is he low a e o heal h ca e u ilisa ion by AYAs, especially p ima y
ca e, gi en hei challenges o sus ain heal h insu ance co e age12. Acco ding o a su ey conduc ed
h ough he ne wo ks o he AYA Wo king G oup o he Eu opean Socie y o Medical Oncology (ESMO)
and he Eu opean Socie y o Paedia ic Oncology (SIOP Eu ope), 67% o p ac i ione s do no ha e
access o specialised cen es o AYA wi h cance , 67% had no access o a specialis cance se ice o la e
e ec s managemen and 38% had no access o e ili y specialis s. Unde -p o ision and inequali y o AYA
cance ca e is common ac oss Eu ope and especially in he Eas e n and Sou he n-Eas . Fu he mo e, he
Wo king G oup also epo ed an absence o ou come measu es o moni o ing and e alua ing AYA cance
ca e p og ams and con ol13.
Among he ecommended u u e s eps, i has been iden i ied ha one o he mos impo an
con ibu ions o AYA esea ch would be o pool da a (e.g. pa ien - epo ed ou comes, clinical and
ea men da a) ac oss ins i u ions and coun ies and c ea e la ge coho s o esea che s o
Add ess he bu den o cance in AYA14. The e is a lack o da a s anda diza ion, da a in e ope abili y and
(p ospec i e) collec ion o ou comes o ele ance o AYAs wi h cance .
The STRONG-AYA p ojec aims o ackle he unde ep esen a ion o AYA’s expe iences and ou comes
when na iga ing he heal hca e sys em and in clinical ca e by de eloping na ional in as uc u es o
ou come da a managemen and clinical decision-making wi hin a pan-Eu opean ecosys em and
es ablishing communica ion eedback o AYAs wi h cance and he heal hca e sys ems. This will be key
o imp o ing heal hca e se ices, esea ch, ou comes and policies o AYAs and o ul ima ely be e cance
ca e o his pa ien g oup.
To his aim, STRONG-AYA b ings oge he an in e na ional mul i-disciplina y conso ium ac oss se en
Eu opean coun ies, led by he Ne he lands Cance Ins i u e (NKI) and composed o academic esea ch
o ganisa ions (Eu opean O ganisa ion o Resea ch and T ea men o Cance (EORTC), Uni e si y o
Sou hamp on, Uni e si y o Leeds, Uni e si y o Manches e , Maas ich Uni e si y, Ne he lands
Comp ehensi e Cance O ganisa ion (IKNL)), clinical pa ne s (I alian Na ional Tumou Ins i u e, Léon
Bé a d Cen e, Gus a e Roussy Ins i u e, Ma ia Sklodowska-Cu ie Na ional Resea ch Ins i u e o Oncology,
he Leeds Teaching Hospi als Na ional Heal h Se ice T us ), s akeholde and pa ien o ganisa ions (You h
Cance Eu ope, Eu opean Cance O ganisa ion) and a consul ing company (FFUND).
8 Fe a i A, S a k D, Pecca o i FA e al. Adolescen s and young adul s (AYA) wi h cance : a posi ion pape om he AYA Wo king G oup o he Eu opean
Socie y o Medical Oncology (ESMO) and he Eu opean Socie y o Paedia ic Oncology (SIOPE). ESMO Open 2021; 6: 100096.
9 Osbo n M, Johnson R, Thompson K e al. Models o ca e o adolescen and young adul cance p og ams. Pedia Blood Cance 2019; 66: e27991.
10 Fa dell JE, Pa e son P, Wake ield CE e al. A Na a i e Re iew o Models o Ca e o Adolescen s and Young Adul s wi h Cance : Ba ie s and
Recommenda ions. J Adolesc Young Adul Oncol 2018; 7: 148-152.
11 Keegan TH, Lich ensz ajn DY, Ka o I e al. Unme adolescen and young adul cance su i o s in o ma ion and se ice needs: a popula ion-based cance
egis y s udy. J Cance Su i 2012; 6: 239-250.
12 Hayashi RJ. Adolescen and young adul cance su i o ship: The new on ie o in es iga ion. Cance 2019; 125: 1976-1978.
13 Salous os E, S a k D, Michailidou K e al. Repo on ESMO/SIOPE Eu opean Landscape p ojec key esul s: Mapping he s a us and needs in AYA cance
ca e. La e-b eaking and de e ed publica ion abs ac s public heal h 2017; 28, 5: V643.
14 Smi h AW, Seibel NL, Lewis DR e al. Nex s eps o adolescen and young adul oncology wo kshop: An upda e on p og ess and ecommenda ions o
he u u e. Cance 2016; 122: 988-999.
STRONG-AYA – No. 101057482 – D1.2
Building on p e ious ini ia i es, a STRONG-AYA da a ecosys em will be se up o alue-based ca e,
esea ch and policy o AYA wi h cance by:
1. De eloping a Co e Ou come Se (COS) speci ically o AYAs wi h cance , ia a pa icipa i e
consensus p ocess de ining mos impo an aspec s o hose di ec ly a ec ed by AYA cance ,
including pa ien s and heal hca e p o essionals.
2. Implemen ing he COS ac oss se e al na ional Eu opean heal hca e sys ems. Da a will
be collec ed a local le el and will hen be included in a da a in eg a ion pla o m. An o e all
ecosys em amewo k o da a analy ics and ou pu will be c ea ed suppo ing ede a ed
analyses and he c ea ion o epo s ac oss clinical and pa ien - epo ed da a and making
na ional eposi o ies o (pa ien - epo ed) heal h da a, a ailable o indi idual pa ien s,
pa ien o ganisa ions, egula o y au ho i ies, as well as he pa ien s’ heal h ca e p o ide s o
in o m clinical decision-making. The i e esul ing na ional ecosys ems will be connec ed o
each o he in o he pan-Eu opean ecosys em using a ede a ed app oach also u ilizing he
o e all ecosys em amewo k.
3. Dissemina ing he COS o a wide ange o local as well as pan-Eu opean s akeholde s,
in pa icula by de eloping analy ical ools o p ocess and p esen pa ien ou come da a and
es ablish eedback loops ha in o m pa ien s and clinicians.
Implemen ing hese p ojec objec i es a e i e wo k packages wi hin he STRONG-AYA p ojec :
 WP1: De elopmen Co e Ou come Se AYA wi h cance and Da a Collec ion (Lead:
SOUTHAMPTON)
 WP2: Go e nance, Da a Secu i y and E hics (Lead: EORTC)
 WP3: In as uc u e and In e ope abili y (Lead: UNIMAAS)
 WP4: Ope a ion o STRONG-AYA ecosys ems, S akeholde and Pa ien in ol emen ,
Dissemina ion, Exploi a ion, Communica ion (Lead: E.C.O.)
 WP5: Scien i ic Coo dina ion and P ojec Managemen (Lead: NKI)
STRONG-AYA will enable AYA ca e and esea ch o bene i om collec ion and pooling o pa ien -cen e ed
da a and collabo a ion among all s akeholde s: pa ien s, heal hca e p o essionals, scien is s, and
policymake s. Mo e widely, he p ojec will le e age he ne wo k o in e es ed o ganisa ions and
ne wo ks es ablished unde STRONG-AYA o long- e m s eng hened p omo ion o he necessa y
implemen a ion o specialis AYA cance se ices ac oss Eu ope. This will ul ima ely b ing no el insigh s
in o AYA cance ca e, esea ch and policy, con ibu ing o he long- e m imp o emen o ou comes o
people wi h AYA cance .
Deli e able in oduc ion
WP1 (de eloping a Co e Ou come Se ): The o e all aim o WP1 is o de elop a Co e Ou come Se (COS)
o AYAs wi h cance . We aim o assess which ou comes, including pa ien - epo ed ou comes, clinical,
and ea men ela ed ou comes, a e mos impo an o measu e o AYAs wi h cance , om he poin o
iew o AYAs wi h cance , hei ca egi e s, heal hca e p o essionals, esea che s and policymake s. The
p ocess o de eloping a COS is ou lined in se e al guidance documen s, and ollows a clea p ocess [1-4].
This includes adhe ing o he 11 minimum s anda ds o Co e Ou come Se -STAnda ds o De elopmen
(COS-STAD) [2] and he 13 i ems conside ed essen ial documen a ion as desc ibed in he Co e Ou come
Se -STAnda dised P o ocol I ems (COS-STAP) [3].
STRONG-AYA – No. 101057482 – D1.2
in which ou comes we e ex ac ed and ca ego ized in o ele an domains. Ou comes we e coun ed
o iden i y weigh o domains.
The Delphi p ocess
The Delphi echnique is a mul is age p ocess, designed o ans o m opinion in o consensus by i e a i e
ounds o ques ionnai es, in e spe sed wi h con olled eedback [14]. Anonymi y o indi idual panel
membe s wi hin speci ic s akeholde g oups emo es inhe en bias, such as g oup con o mi y, as
pa icipan s do no in e ac di ec ly wi h each o he , so si ua ions whe e he g oup is domina ed by he
iews o ce ain indi iduals can be a oided [15]. Delphi s udies a e ypically 2-3 ounds, al hough aspec s
o i s me hodology can be in e p e ed in a a ie y o ways.
Delphi su ey de elopmen
The ou comes iden i ied as ele an o AYAs wi h cance ia he e iew o he li e a u e (s ep 1) and he
quali a i e in e iews (s ep 2) o med an ini ial lis , which would be aken o wa d o he i s ound o
he Delphi su ey. The ini ial lis o ou comes we e ca ego ized using Dodd’s 38-i em axonomy o
ou comes in medical esea ch [6], and o med he skele on o he Delphi su ey (Table 3). The axonomy
was adap ed, in e ms o o de o ou comes and speci ic wo ding, o adjus o he lay audience o his
Delphi su ey. The o de ing o domains in he axonomy was also adjus ed o imp o e he cohe ence o
he su ey, o he pu poses o he Delphi.
Table 3: The skele on o he Delphi su ey, using an adap ed e sion o Dodd’s 38-i em axonomy o
ou comes in medical esea ch [6]
Dodd’s 38-i em axonomy o ou comes in medical esea ch
Ou comes o disease
Mo ali y/su i al
Physiological/clinical
Ou comes ela ing o neoplasms
Ca diac ou comes
Ci cula ion (blood, ascula and lympha ic sys em) ou comes
Respi a o y sys em (ches and lungs) ou comes
S omach (gas oin es inal) and diges ion ou comes
Ne ous sys em ou comes
Kidney and u ina y sys em ou comes
Li e and panc eas (hepa obilia y) ou comes
Immune sys em ou comes
In ec ion ou comes
Ea , hea ing, and balance ou comes
Eyes and ision ou comes
Skin and subcu aneous issue ou comes
Musculoskele al and connec i e issue ou comes
Nu i ion and me abolism ou comes
Inju y and poisoning ou comes

STRONG-AYA – No. 101057482 – D1.2
Musculoskele al and connec i e issue ou comes
Ho mones (endoc ine sys em) ou comes
Rep oduc i e sys em ou comes
P egnancy and childbi h (be o e, du ing and a e childbi h) ou comes
Gene ic ou comes
Psychia ic ou comes
Gene al ou comes
Func ioning
Physical unc ioning
Social unc ioning
Role Func ioning
Emo ional and Psychological Func ioning / Wellbeing
Cogni i e Func ioning
Global Quali y o Li e
Pe cei ed Heal h S a us
Deli e y o Ca e
Pe sonal Ci cums ances
Resou ce Use
Economic
Hospi al
Need o u he in e en ion
Socie al and ca e bu den
Ad e se e en s / e ec s
D a e sions o he su ey i ems we e e alua ed by 8 membe s o he STRONG AYA conso ium, including
wo k package leads (n = 2), esea che s (n=2) and HCPs (n = 4). This was o ensu e all ou comes we e: (1)
ph ased in layman’s e ms o ensu e pa icipan s could accu a ely in e p e hem, and (2) ca ego ized
co ec ly acco ding o he axonomy. The leng h o he su ey was highligh ed as an o e a ching conce n;
he e o e, e o s we e made o elimina e any po en ial epe i i eness in ou come i ems and hus educe
he o e all numbe o i ems o be included.
The inal lis o ou comes, as well as he su ey ins uc ions, we e pilo es ed by h ee esea che s wi hin
he STRONG AYA conso ium, and eedback was p o ided. This was o es he usabili y o he su ey
pla o m, as well as he in e p e a ions o each ou come.
Rec ui men o he Delphi su ey
The Delphi p ocess was comp ised o h ee expe s akeholde panels:
(4) AYAs (aged 15-39 a diagnosis) and ca egi e s (including pa en s / gua dians, amily membe s,
pa ne s and ca egi e s).
(5) Heal hca e p o essionals who p o ide ca e o AYAs wi h cance .
(6) Resea che s and policymake s who a e ac i e in he ield o AYAs wi h cance .
STRONG-AYA – No. 101057482 – D1.2
The ec ui men a ge o he Delphi su ey was 100 pa icipan s pe s akeholde g oup (300 in o al).
This would allow o d op-ou a es mo ing in o subsequen Delphi ounds. Rec ui men ma e ials we e
de eloped, including a 1-Page ec ui men le e ou lining he s udy, email empla es, and social media
ad e s. Pa icipan s o each s akeholde g oup we e ec ui ed in he ollowing ways:
Table 4: Desc ip ion o he ec ui men s a egies o each s akeholde g oup in he Delphi su eys
(1) AYAs and Ca egi e s (2) Heal hca e
p o essionals
(3) Resea che s and
policymake s
AYAs and ca egi e s ha ook
pa in he quali a i e
in e iews we e asked i hey
would also like o ake pa in
he Delphi su ey.
Heal hca e p o essionals ha
ook pa in he quali a i e
in e iews we e asked i hey
would also like o ake pa in
he Delphi su ey.
Resea che s we e con ac ed ia
hei email add esses which
we e ound in ecen
publica ions in AYA jou nals (e.g.
Jou nal o Adolescen and Young
Adul Oncology).
You h Cance Eu ope a e a
cha i y made up o you h
cance o ganisa ions ac oss
Eu ope. A con ac a he
cha i y con ac ed po en ial
pa icipan s o in i e hem o
ake pa in he Delphi su ey.
They also ad e ised h ough
social media channels
(LinkedIn, Facebook).
The Eu opean Socie y o
Paedia ic Oncology (SIOPE) is
a pan-Eu opean o ganisa ion
ep esen ing all p o essionals
wo king in he ield o
childhood and AYA cance s.
They sha ed he ec ui men
le e wi h all membe s o he
Eu opean Ne wo k o
Teenage s and Young Adul s
wi h Cance (ENTYAC).
Resea che s p o ided con ac
de ails o o he esea che s who
may wish o ake pa .
The You h and Cance
Founda ion (S ich ing Jonge en
en Kanke ) is a pa ien
o ganisa ion o AYAs wi h
cance . They sha ed he social
media ad e s ia hei
Ins ag am and Facebook
s o ies.
The Eu opean Cance
O ganisa ion (ECO) a e
ne wo k o oncology
p o essionals and cance
pa ien s. They ad e ised he
Delphi su ey in hei mon hly
newsle e .
The Eu opean Cance
O ganisa ion (ECO) a e ne wo k
o oncology p o essionals and
cance pa ien s. They ad e ised
he Delphi su ey in hei
mon hly newsle e .
The Eu opean Cance
O ganisa ion (ECO) a e
ne wo k o oncology
p o essionals and cance
pa ien s. They ad e ised he
Delphi su ey in hei mon hly
newsle e .
Pa icipan s sha ed wi h hei
ne wo ks o HCPs a local
hospi als.
Pa icipan s di ec ed o he
AYAs and ca egi e s o ake
pa , h ough wo d o mou h
HCPs ale ed hei AYA pa ien
communi y, ia communi y
pa ien g oups and local
cha i ies.
STRONG-AYA – No. 101057482 – D1.2
o ad e isemen s in hei local
cance communi ies.
Su ey da a collec ion p ocedu e
The online su ey pla o m used o collec esponses was Qual ics (h ps://www.qual ics.com/uk/).
The pa icipan in o ma ion shee was p o ided a he s a o he su ey, as well as a checkbox o
pa icipan s o p o ide hei consen o aking pa . Pa icipan s we e eminded o hei igh o wi hd aw
om he s udy a any ime, p i acy and accessibili y in o ma ion, and how hei da a will be used.
Ini ial, gene ic demog aphic ques ions ( ele an o all s akeholde g oups) we e included a he s a o
he su ey o collec in o ma ion, such as: age, gende , e hnici y, coun y o esidence, educa ion, and
socioeconomic s a us. Speci ic demog aphic ques ions ele an o sepa a e s akeholde g oups we e hen
asked. Fo example, AYAs we e asked abou hei diagnosis ype, age a diagnosis, ea men s a us, and
clinic- ype. Ca egi e s we e asked he same ques ions, in ela ion o he AYAs hey ca e o . HCPs,
esea che s and policymake s we e asked ques ions ela ing o hei p o ession, yea s’ expe ience, and
hei expe ise wi hin hei p o ession.
Ou Delphi p ocess included h ee su ey ounds:
(1) Ini ial i em a ing, wi h all 129-ou comes
(2) I em e- a ing, wi h eedback om p e ious su ey ound
(3) I em anking, choosing a op 15 ou comes o on and o ea men *
* Round 3 o he Delphi su ey is cu en ly unde way a he ime o his epo
Round 1 o he Delphi Su ey
Following demog aphic ques ions, he su ey asked pa icipan s o a e he impo ance all 129 ou comes
on a 9-poin Like Scale, as pe ecommenda ions ou lined by he GRADE wo king g oup [16]. A a ing
scale o “no impo an ” ( a ing o 1–3), “impo an , bu no c i ical” ( a ing o 4–6), o “ex emely
impo an / c i ical” ( a ing 7–9) was p o ided. An op ion o “unable o answe ” was also p o ided. I was
explained o pa icipan s ha hey should no a e oo many ou comes as “ex emely impo an ”, as he
end-goal was o ha e only app oxima ely 10-15 ou comes ha a e kep o inclusion in he inal COS.
Pa icipan s we e eminded on each page o he su ey ha hey should a e he ou comes wi h emphasis
on he AYA (aged 15-39) demog aphic, o a oid esul ing in a COS ha is no speci ic o he AYA age-g oup.
Pa icipan s we e equi ed o p o ide a a ing o e e y i em in he su ey. A he end o he su ey was
an open- ex box o pa icipan s o p o ide any ou comes hey hough we e missing in he su ey, ha
we e impo an o measu e o AYAs wi h cance .
T ansla ions o he su ey we e p o ided o he ollowing languages: Du ch, F ench, I alian, Ge man,
Spanish and Polish. This was o inc ease he each and cul u al di e si y o he Delphi su ey.
Round 2 o he Delphi Su ey
STRONG-AYA – No. 101057482 – D1.2
The second su ey ound included he same ini ial demog aphic ques ions as ound 1. Pa icipan s
we e p o ided wi h a pe sonalised URL, he e o e hei esponses o he demog aphic ques ions we e
sa ed and ca ied o wa d om ound 1 and hey we e able o make changes (i necessa y).
The concep o a second Delphi su ey ound is o p o ide eedback o pa icipan s o he esul s om
ound 1, and allow hem o po en ially change hei a ing on he 9-poin Like scale, o begin a
con e gence o opinion (consensus) and come o an ag eemen on he mos impo an ou comes o
measu e o AYAs wi h cance . Feedback om ound 1 was p o ided as a pe cen age abo e each numbe
on he Like scale (i.e. a pe cen age o how many o he pa icipan s in hei s akeholde g oup a ed he
ou come as X le el o impo ance). Pa icipan s we e also p o ided wi h hei o iginal a ing om ound
1 as a p e- illed do on he Like scale. See Figu e 3.
All i ems lis ed in ound 1 o he Delphi su ey we e ca ied o wa d o ound 2. Pa icipan s we e also
asked o a e six addi ional ou comes ha pa icipan s had lis ed as impo an ye missing om ound 1
(using he open ex box).
Figu e 3: A snapsho o ound 2 o he Delphi su ey, e idencing he use o eedback (%) and p e iewing
pa icipan ’s ound 1 esponses.
Consensus mee ing
In o de o an Ini ial COS o be de eloped in ime o begin implemen a ion o da a collec ion h oughou
he heal hca e esea ch ecosys em o he STRONG AYA p ojec , a consensus mee ing was scheduled p io
o conduc ing Round 3 o he Delphi su ey. This was o allow decisions o be made ega ding he
ou comes o be included in he ini ial COS. This mee ing ook place on 18/01/2024 and a endees included
12 pa icipan s om he STRONG AYA conso ium, who o med an expe panel.
All ou comes ha ecei ed ≥70% esponden s a ing i as a 7-9 on he Like -scale, ac oss all s akeholde
g oups, we e p esen ed o he expe panel. Based on discussions ega ding he di icul y o dis inguishing
STRONG-AYA – No. 101057482 – D1.2
he mos impo an ou comes o AYAs wi h cance , i was decided ha he ini ial COS would iden i y
ou comes o bo h ON and OFF ea men phases. The de ini ions o ON and OFF ea men we e as
ollows:
On ea men = a cance pa ien ecei ing cu a i e ea men , including main enance ea men , who is
no belie ed o be disease- ee.
O ea men = a cance pa ien ea ed wi h cu a i e in en , who is disease- ee, o ecei ing li e-
ex ending o pallia i e ea men / ca e
An online poll ook place (using a polling pla o m, Ve ox) whe eby pa icipan s we e asked o selec he
mos impo an 15 ou comes o measu e o AYAs wi h cance o bo h ON and OFF ea men . The
numbe “15” was decided ollowing explo a ion o he li e a u e and a e aging he numbe o COS i ems
o cance COS published since 2020.
Resul s om his mee ing we e used o in o m he i s d a o he Ini ial COS. This d a o he Ini ial COS
was ci cula ed o he STRONG AYA conso ium, plus membe s o he public ad iso y boa d o eedback.
Feedback was p o ided om 7 esea che s and HCPs in he STRONG AYA conso ium and 4 pa ien
ad iso y boa d membe s.
Round 3 o he Delphi Su ey
Round 3 aims o pa icipan s o be mo e succinc wi h hei decision making. Any ou come ha ecei ed
≥70% esponden s a ing i as a 7-9 on he Like -scale was ca ied o wa d o ound 3 o he Delphi
p ocess, pe s akeholde g oup. Fo example., he AYA and pa en /ca egi e s akeholde panel we e only
p esen ed wi h he lis o ou comes ha ecei ed ≥70% esponden s a ing i as a 7-9 on he Like -scale
by hei s akeholde g oup (see Table 6) .
Simila o he consensus mee ing, pa icipan s we e asked o iden i y he op 15 ou comes o AYAs wi h
cance , whils bo h ON and OFF ea men . Addi ionally, hey we e asked o hen ank he op 15 ha
hey chose, in o de o “mos impo an ” and “leas impo an ”, o bo h ON and OFF ea men . I is
hoped ha his me hodology will elici g ea e consensus om s akeholde g oups, hus esul ing in he
Final COS.
Round 3 is cu en ly ongoing a he ime o his epo , and is due o close wi hin he upcoming weeks.
Resul s om his su ey ound will hen in o m he Final COS.
Phase 6: De e mine “how” o measu e he COS
A join ini ia i e be ween he COnsensus-based S anda ds o he selec ion o heal h Measu emen
INs umen s (COSMIN) ini ia i e [5] and he COMET ini ia i e [1] ha e de eloped a guideline on how o
selec measu emen ins umen s o ou comes included in a COS (Figu e 4).

STRONG-AYA – No. 101057482 – D1.2
Figu e 4: Flowcha o he selec ion o ou come measu emen ins umen s o COS [17]
S ep 4: Selec one ins umen o
each ou come in he COS
Use a consensus p ocedu e o
ag ee on he ins umen s o each
ou come included in he COS
E alua e he me hodological quali y
o he s udies on measu emen
p ope ies
E alua e he quali y o he
me hodological measu emen
p ope ies
Combine he e idence on he
me hodological quali y o he s udies
wi h he quali y o he measu emen
p ope ies
E alua e he easibili y o he
ins umen s
Pe o m a new li e a u e sea ch
Upda e he li e a u e sea ch
S ep 3: E alua e he quali y o
he ins umen s
No, no up- o-da e
No, o poo quali y
No,
quali y assessmen no
pe o med
S ep 1: Concep ual conside a ions
De ine cons uc o be measu ed
De ine a ge popula ion
S ep 2: Find all exis ing ins umen s
Is a good quali y, up- o-da e
sys ema ic e iew o ou come
measu emen s a ailable?
STRONG-AYA – No. 101057482 – D1.2
To de elop he Co e Measu emen Se o he Ini ial COS, he guidelines p oposed in Figu e 4 we e
ollowed, wi h he excep ion o upda ing / pe o ming a li e a u e sea ch in he absence o a good quali y,
up- o-da e e iew. The consensus p ocess (s ep 4) will be in he o m o a mee ing due o ake place on
26 h Ma ch 2024, whe eby membe s o he STRONG AYA conso ium om he Uni e si y o Sou hamp on
and NKI will mee o make p agma ic decisions on which a e he mos sui able measu emen ools, in
e ms o hei psychome ic p ope ies, accessibili y, ansla ion a ailabili y, and numbe o
i ems/subscales.
Once he Final COS has been de eloped, he scien i ic p ocess o measu emen ins umen selec ion
de ailed in Figu e 4 will be ollowed ex ensi ely.
De e mine “case-mix a iables”
As pa o he da a ex ac ion p ocess o he li e a u e e iew (see sec ion 5.5.1), case mix a iables
published in he li e a u e we e collec ed. These will hen be pooled and analysed, eady o
implemen a ion alongside he Ini ial COS in u u e wo k packages in he STRONG AYA p ojec . This p ocess
is ongoing a he ime o his epo .
Resul s
The Li e a u e Re iew
A o al o 14 e iewe s om he STRONG AYA conso ium conduc ed i le and abs ac sc eening, and ull
ex sc eening. 15 e iewe s conduc ed da a ex ac ion.
The ini ial sea ch iden i ied 37127 eco ds h ough he i e da abases (Figu e 5). A e duplica es we e
emo ed, he sea ch e u ned 19826 eco ds. Following sc eening 17301 eco ds on i le and abs ac s,
11727 eco ds we e excluded. This esul ed in 5574 eco ded being assessed o eligibili y, om which
2980 we e excluded. In o al, 2594 s udied we e included in he e iews; ou comes om hese eco ds
we e colla ed and in o med he de elopmen o he Delphi su ey. Impo an ly, e inemen o he
li e a u e sea ch is s ill ongoing a he ime o his epo . The numbe o a icles epo ed din Figu e 5
we e accu a e o de eloping an ini ial lis o ou comes o he Delphi su ey, howe e mo e a icles ha e
been excluded since his poin , and mo ing o wa ds o he w i e up o he Li e a u e Re iew.
STRONG-AYA – No. 101057482 – D1.2
Figu e 5: Flow cha o s udy sc eening and inclusion p ocess, acco ding o he PRISMA F amewo k.
Quali a i e In e iews
A o al o 56 in e iews we e conduc ed be ween Ap il 2023-June 2023; 27 AYAs, 4 pa en s / ca egi e s,
and 25 HCPs. Demog aphic in o ma ion and esul s ela ing o he p oduc ion i a digi al ool o he
STRONG AYA p ojec a e ou lined in Deli e able 1.11. A hema ic con en analysis esul ed in 22 ou come
domains ha we e pe cei ed as impo an o AYAs wi h cance . Table 5 summa ises he domains
iden i ied in he in e iews, including which s akeholde g oup iden i ied wha as impo an . These 22
domains, plus he indi idual ou comes wi hin each domain, we e hen o in o m he p oduc ion i he
Delphi su ey.
Iden i ica ion o s udies ia da abases and egis e s
Reco ds iden i ied h ough
da abase sea ching (n = 37127):
Medline ALL (n = 13088)
Embase (n = 12039)
Web o Science Co e Collec ion
(n = 11146)
Coch ane Cen al Regis e o
Con olled T ials (n = 654)
Google Schola (n = 200)
Duplica e eco ds emo ed be o e
sc eening (n = 19826):
Medline ALL (n = 119)
Embase (n = 10089)
Web o Science Co e Collec ion
(n = 9021)
Coch ane Cen al Regis e o
Con olled T ials (n = 420)
Google Schola (n = 177)
Reco ds sc eened on i le and
abs ac (n = 17301) Reco ds Excluded (n = 11727)
Full ex eco ds assessed o
eligibili y (n = 5574)
Reco ds excluded (n = 2980):
S udies included in e iew
(n = 2594):
Iden i ica ion Sc eening
Inclusion
STRONG-AYA – No. 101057482 – D1.2
Table 5: Domain esul s om he hema ic con en analysis o he quali a i e in e iews, pe
s akeholde g oup
Domain AYAs HCPs Pa en s /
ca egi e s
Fe ili y X X X
Rela ionships/sexual in imacy X X X
Changes o amily dynamics/ oles X X X
Men al heal h/psychological X X X
Iden i y X X
Social unc ioning (excl. oman ic
ela ionships) X X X
Connec ing o o he pee s wi h
cance o he same li e s age X
Educa ion X X X
Wo k (posi i e) X X
Wo k (nega i e) X X
Financial implica ions X X X
Body image (sel -es eem) X X
Su i al/mo ali y X X X
Loss o con ol X
Loss o independence X X X
Long e m e ec s o cance and
ea men /a ec s pos ea men X X
Li ing wi h and beyond cance X X X
Managing impac s on daily
ou ines/li es X
Beha iou X X
Heal hca e X X X
Physical X X
Cogni i e X
The Delphi su ey
Gene a ing he ini ial lis o ou comes