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 – D1.10
Deli e able Repo
WP1– De elopmen o a Co e Ou come Se (COS) o AYAs wi h cance and
da a collec ion
D1.10
S udy 2 Ini ia ion Package
Due da e o deli e able: 31/03/2024
Ac ual submission da e: 31/03/2024
P ojec :
STRONG-AYA
Lead Con ibu o
Olga Husson (NKI)
Email
[email p o ec ed]
O he Con ibu o s
Wine e an de G aa ; Renske Fles; Simone Hanebaum; Sil ie Janssen
Emails
w. d.g [email p o ec ed]; [email p o ec ed]; [email p o ec ed]; [email p o ec ed]
Due da e
31 03 2024
Deli e y da e
30 03 2024
Deli e able ype
R
Dissemina ion le el
PU
Desc ip ion o Wo k
Ve sion
Da e
V1.0
30/03/2024
STRONG-AYA – No. 101057482 – D1.10
Desc ip ion:
S udy 2 ini ia ion package (be o e en olmen o he i s s udy pa icipan ) including:
-Regis a ion numbe o he clinical s udy in a egis y mee ing WHO Regis y c i e ia
-Final e sion o s udy p o ocol as app o ed by he egula o (s) / e hics commi ee(s)
-Regula o y and e hics (i applicable, ins i u ional) app o als equi ed o he en olmen
o he i s s udy pa icipan
Publishable summa y (max ½ page)
This epo p o ides an o e iew o he e hics app o al o he da a collec ion phase o STRONG AYA. The
a ached documen s include he s udy p o ocol iled wi h he Ins i u ional Re iew Boa d a he Ne he lands
Cance Ins i u e – An oni an Leeuwenhoek Hospi al.
STRONG-AYA – No. 101057482 – D1.10
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
5 STUDY 2 INITIATION PACKAGE .......................................................................................................................... 9
6 RESULTS ......................................................................................................................................................... 10
7 REPOSITORY FOR PRIMARY DATA (ANNEX) ..................................................................................................... 11
STRONG-AYA – No. 101057482 – D1.10
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 – D1.10
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 – D1.10
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 ies
1
. 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 diagnosis
2
, 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 2007
4
.
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
neoplasms
5
. 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
needs
6
. 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 ch
7
.
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
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 – D1.10
se ices
8
,
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 y
11
.
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 age
12
. 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 ol
13
.
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 AYA
14
. 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).
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:
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 – D1.10
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 .
5 S udy 2 ini ia ion package
The Ne he lands Cance Ins i u e (NKI) is he Coo dina o o STRONG AYA. As such i leads he STRONG AYA
p ojec and se s he p eceden and scope o he nex phase o he p ojec , namely da a collec ion o he COS.
The e ha e been delays in de eloping he COS in o de o unde ake he Delphi consensus p ocess and o
decide on he app op ia e me ics. As such, a p esen ime he NKI is he o he cen e o ha e secu ed e hics
app o al o he nex phase o he p ojec . The e a e se e al impo an con ex ual ac o s o conside a
p esen ega ding he e hics applica ion o he STRONG AYA p ojec .
1) Consen o he use o ou inely collec ed da a in esea ch a he NKI, as a comp ehensi e cance
cen e, is collec ed a pa ien egis a ion, and hus no u he pa ien consen is equi ed o use da a
ha is ou inely collec ed in clinical p ac ice and hus is a ailable by da a desk eques om he
elec onic heal h eco d
P o ocol e sion 1.0
4
S éphane Lejeune
Eu opean O ganisa ion o Resea ch and T ea men o
Cance EORTC BE
Ma ia Aguado
Eu opean O ganisa ion o Resea ch and T ea men o
Cance EORTC BE
No be Couespel
Eu opean Cance O ganisa ion
ECO
BE
No a Lo enzo
Eu opean Cance O ganisa ion
ECO
BE
Mi iam Rome o
Eu opean Cance O ganisa ion
ECO
BE
Ka alin
Riz ine
Lehoczky Funda ia You h Cance Eu ope YCE RO
U ska Kosi
Funda ia You h Cance Eu ope
YCE
RO
Tessa an de E e
FFUND B.V.
FFUND
NL
Ma ieke Dekke
FFUND B.V.
FFUND
NL
P o ocol e sion 1.0
5
Summa y
Backg ound
STRONG-AYA is 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 (AYA) wi h cance , de ined as
indi iduals aged 15-39 yea s a cance diagnosis. 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-adjus ed and high-quali y heal hca e. AYA-ca e and esea ch will bene i
om he collec ion, secu e access, and in eg a i e analysis o a la ge olume and highly inclusi e pool
o pa ien -cen 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. Wi hin STRONG-AYA we will se up a alue-based heal hca e esea ch
ecosys em o de elop da a-d i en, in e ac i e policy and isualiza ion ools ha b ing, in co-c ea ion
wi h all s akeholde s including pa ien s, no el insigh s in o AYA heal hca e.
Objec i es
The o e all p ojec objec i es include:
(1) The de elopmen o an age-speci ic Co e Ou come Se (COS) o AYAs wi h cance (in p ocess,
app o ed by Sou hamp on E hics Commi ee/IRB 78653. Expec ed d a COS: Decembe 2023);
2) The Implemen a ion o he COS in 5 na ional heal hca e sys ems (F ance, I aly, Ne he lands, Uni ed
Kingdom, Poland) and es ablishmen o na ional in as uc u es o he collec ion and managemen o
ou come da a;
3) The enac men o a pan-Eu opean STRONG-AYA ecosys em, including a p i acy-p ese ing
ede a ed lea ning in as uc u e;
4) The dissemina ion o he COS and i s insigh s gleaned om he de elopmen o bespoke analy ical
ools o a wide ange o local and pan-Eu opean s akeholde s o use eal wo ld insigh o de elop da a
d i en, alue-based ca e and esea ch ac oss he con inen .
This IRB p o ocol conce ns i sel wi h objec i e 2 and is in ended o ope a e as an umb ella o ‘mas e ’
p o ocol o his objec i e a he pan-Eu opean le el.
P ocedu e and aims
The p ima y aim o he STRONG-AYA conso ium is o de elop ou come p edic ion models
(p e alence/incidence o , isk ac o s o and mechanisms o poo ou comes) o AYAs wi h cance
h ough he c ea ion o na ional and pan-Eu opean ede a ed lea ning in as uc u e ecosys ems.
Once he d a COS has been inalised (Decembe 2023), he COS will be implemen ed as a esea ch
ins umen o gain eal-wo ld insigh om AYA da a ( e ospec i e and p ospec i e clinical and egis y
da a, whe e ele an ) and in o clinical p ac ice (p ospec i e) by building a Eu opean heal hca e
esea ch ecosys em o AYA wi h cance . Mo e speci ically, we aim o de elop ou come p edic ion
models (p e alence/incidence o , isk ac o s o and mechanisms o poo ou comes) o AYA wi h
cance h ough he me hod o Fede a ed Lea ning (FL). These models mean ha analysis is based on
da a om pa ien s aged 15-39 yea s a p ima y cance diagnosis ac oss mul iple in e na ional cen es,
P o ocol e sion 1.0
6
wi hou any indi idual le el pa ien da a lea ing he ins i u ion i o igina es om, he e o e p ese ing
pa ien da a p i acy.
Conclusion
We hypo hesise ha obus and gene alisable FL can be de eloped ac oss cen es, and ha his
me hod will p o ide s a is ical models ha explain he da a, and demons a e clinically use ul isk
ac o s o good and bad cance ou comes. By linking mul iple in e na ional cen es, he FL models can
be de eloped using he la ges a ailable coho o AYA cance pa ien s o u he de elop esea ch in o
AYA cance and o be e in o m he ca e o his unde se ed cance popula ion. This has p e iously
and ecen ly been success ul done o lung cance pa ien s (IRBd21-026_29-01-2021) and anal cance
pa ien s (IRBd21-166_06-08-2021).
P o ocol e sion 1.0
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1 BACKGROUND
In any heal hca e sys em i is essen ial ha pa ien s ha e access o high-quali y ca e and o deli e
angible imp o emen s in ou comes encompassing su i al, heal h- ela ed quali y o li e (HRQoL), he
abili y o a end school o wo k, ge and aise child en and ac i ely pa icipa e in socie y [1]. This is
pa icula ly impo an o adolescen s and young adul s (AYA) wi h cance , de ined as 15 o 39 yea s
a ini ial cance diagnosis [2]. Thei 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]. Ad ances
in cance ea men ha e led o inc eased su i al a es and in AYA’s e en o e 80% will su i e long-
e m (>5 yea s) [4], Howe e , despi e he ela i ely a ou able cance p ognosis, AYA cance pa ien s
a e a isk o ea men ela ed de elopmen o medical e ec s (e.g., ca dio ascula disease o
second malignancies), in e ili y o psychosocial e ec s (e.g., di icul y in oman ic ela ionships,
inancial oxici y due o unemploymen wi hou a p io ca ee job), and ha e an inc eased isk o
la e mo ali y [5, 6].
AYAs wi h cance a e dis inc om he paedia ic (<15 yea s) and he olde adul (>40 yea s) cance
popula ions illus a ed by he ollowing indings [2, 7]:
- Thei unique spec um o cance ypes (epidemiology), wi h bo h paedia ic- ype and adul -
ype umou s [8]. The AYA g oup aces an o e lap wi h paedia ic umou s (b ain umou s,
sa coma, leukaemia and lymphoma), bu also wi h umou s common a olde age (b eas and
inc easingly colo ec al cance ), and diagnoses which ypically peak a AYA age ( es icula
cance , hy oid cance and melanoma). Hence, bo h paedia ic oncologis s’ inpu and adul
oncologis s’ compe ences a e needed o deli e op imal ca e and ea men o AYAs.
- Di e ences in umou genomic, biology and clinical beha iou in AYA umou s compa ed o
child en and olde adul s [8, 9]. Age-speci ic molecula ea u es a e inc easingly seen as
he apeu ically ele an . The biology o he hos may also di e acco ding o age, wi h dis inc
pha macology and po en ial impac on he apy e icacy and oxici y p o iles. Jus ansla ing
he clinical managemen o child en o olde adul s in o he s anda ds o he ea men o
AYAs hus seems o be unjus i ied.
- A lack o awa eness ha cance may occu in his age g oup (among gene al popula ion and
heal hca e p o essionals (HCPs)),wi h o en esul s in diagnos ic delays and/o di icul access
o specialised ca e, po en ially esul ing in poo su i al ou comes [10]; Diagnos ic delay also
has a nega i e impac on pa ien s’ us in he heal hca e sys em and psychosocial ou comes
such as anxie y.
- A lack o AYA cance pa ien s’ pa icipa ion in clinical p o ocols ( epo ed a e o en e ing
clinical ials anged om 5% o 34% in published se ies), limi ing he e idence base o
ea men s [11];
- Adolescence and young adul hood a e complex phases o li e due o he many physical,
emo ional, cogni i e, and social ansi ions [12]. Impo an de elopmen al asks need o be
achie ed, such as o ming one’s own iden i y and a heal hy body image, es ablishing
au onomy, esponsibili y and independence, inishing educa ion and s a ing a ca ee , s a ing
a oman ic ela ionship and ha ing and aising young child en [12]. A cance diagnosis
challenges AYAs’ abili ies o achie e hese de elopmen al miles ones [13]. The way in which
AYA wi h cance adjus o hei cance expe ience migh ha e li e-long implica ions o he
quali y o hei su i al [14]. Li e-long consequences o ea men a e o en no pa o he
P o ocol e sion 1.0
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p ima y ea men discussions whe e he s ong ocus is on op imal su i al ou comes.
Examples o hemes ha AYA cance pa ien s would p e e o be discussed al eady e y ea ly
a e diagnosis a e e ili y, educa ion and wo k, oman ic ela ionships and sexuali y, aising
young child en and inancial consequences o disease and ea men . All opics ha a e no
pa o s anda d clinical consul a ions, due o lack o awa eness, ou ine (as AYA cance
pa ien s a e a e), ime cons ain s and lack o knowledge and suppo i e s a o discuss and
ac on hese issues.
- His o ically, he e was a lack o imp o emen in su i al a es as compa ed o o he age
g oups. Fo some umou ypes, such as b ain umou s and sa comas, su i al in AYA is s ill
poo e han in child en wi h he same disease [15] which is umou biology, pa ien and
ea men ela ed.
A MISSED CHANCE AND UNMET NEED:
The unique cha ac e is ics equi e access o oncology se ices which p o ide expe cance ca e and
conside AYAs’ age-speci ic and complex psychosocial and physical needs ega ding (1) physical
changes, de elopmen o sel -image, iden i y, ela ionships, sexuali y and independence; (2) age-
app op ia e in o ma ion and communica ion, sha ed decision-making, compliance and ea men
adhe ence; (3) p i acy and pee -suppo ; (4) peculia beha iou s o his age and isk- aking (including
alcohol/subs ance abuse). Howe e , in many pa s o he wo ld, AYAs wi h cance ace inequi ies o
ca e as hey a e poo ly se ed by he adi ional dicho omy o he in eg a ed paedia ic
(“pa ien / amily-cen ed”) ca e se ices e sus dispe sed (“disease-cen ed”) adul oncology se ices
[11, 16, 17]. Up o hal o AYA 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 he socie y [18].
Cance a AYA age is a e, al hough 4-6 imes mo e equen han paedia ic cance , bu he 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 HRQoL due o he impac o he
disease du ing o ma i e yea s and long- e m complica ions o disabili ies [19]. The AYA Wo king
G oup (WG) o he Eu opean Socie y o Medical Oncology (ESMO) and he Eu opean Socie y o
Paedia ic Oncology (SIOP Eu ope) concluded in 2021 ha inding apid solu ions o ‘speak he same
language’ among heal hca e p o essionals (HCPs) is essen ial o u he imp o e heal h ou comes o
AYAs [11]. The WG epo ed ha s anda d clinical ial endpoin s, such as i e-yea o e all su i al,
p og ession- ee su i al, and cance -speci ic su i al, o en do no add ess he speci ic needs o he
AYA popula ion. The WG ound a widesp ead geog aphic a ia ion, na ionally and in e na ionally, in
AYA ca e p og ams wi h an uns uc u ed, o en me ely philan h opic, unding [11]. When ESMO and
SIOPE membe s we e asked i hei pa ien s had access o specialized se ices o AYA wi h cance , o
i such se ices we e in de elopmen , only 33% con i med ha hey had, namely: 13% in Eas e n and
Sou h-Eas e n Eu ope, 45% in Wes e n Eu ope and 60% in No he n Eu ope [20]. In addi ion,
subs an ial inequali ies in suppo by specialised HCPs, such as psychologis s, social wo ke s,
physio he apis s, die icians and AYA-dedica ed nu ses we e ound be ween AYA ca e p og ams [20].
The WG 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 ol [11]. In 2016, he NCI upda ed he AYA cance p og ess e iew g oup epo
and examined scien i ic gaps and oppo uni ies o u u e AYA oncology [22]. I was concluded ha
P o ocol e sion 1.0
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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 AYA [22].
STRONG-AYA seeks o ake up his call om he ESMO-SIOPE WG and NCI o es ablish a co e ou come
se (COS), and o use eal-wo ld da a, pooled in a ede a ed lea ning in as uc u e, o
comp ehensi ely assess pa ien s’ age-speci ic needs, sc een o physical and psychosocial p oblems
and p o ide mul idisciplina y, holis ic, age-speci ic hospi al and communi y suppo . By e alua ing AYA
cance pa ien s’ heal hca e expe iences and ou comes we belie e hei ca e and ou comes can be
imp o ed and unjus i ied a ia ions in quali y o ca e educed. We belie e ha his can be done by
maximizing he po en ial o mul i s akeholde s’ da a. Al hough an e e inc easing amoun o da a is
a ailable, he collec ion, access, p ocessing, and use o hese da a a e s ill e y agmen ed wi hin and
especially ac oss na ional heal h sys ems. 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 de elopmen and
implemen a ion o a COS, a consensus-based ag eed minimum se o ou comes ha should be
measu ed and epo ed, wi h pa ien -cen ed ou comes o alue o AYAs wi h cance , ep esen s an
oppo uni y o enable da a-d i en heal hca e inno a ion and se e as a basis o add ess clinically
ele an ques ions o his ulne able pa ien g oup.
The c ea ion o STRONG-AYA as a new dedica ed, in e disciplina y, mul i-s akeholde Eu opean
ne wo k, consis ing o AYAs wi h cance , HCPs wo king in his ield, esea che s and policy-make s,
builds on p e ious ini ia i es, such as he Eu opean Ne wo k o Cance Resea ch in Child en and
Adolescen s p og am (ENCCA), a speci ic Eu opean Ne wo k o Teenage s and Young Adul s wi h
Cance (ENTYAC; EU FP7 2011-14).
STRONG-AYA: We ha e uni ed a ne wo k o p o essionals (HCPs and scien is s) and pa ien s, some
o which a e al eady collabo a ing wi hin ENTYAC and he ESMO/SIOPE AYA WG, who collec i ely
exp essed hei wish o join o ces o wo k on he speci ic challenges o AYA wi h cance . The ime is
igh o make a signi ican in es men o sus ainably align esea ch and heal hca e o in o m indi idual
decision-making and heal hca e policy. By c ea ing i e na ional ecosys ems and an o e all Eu opean
AYA heal hca e esea ch ecosys em, concen a ing on he dynamic gene a ion and use o ou come
da a by all impo an ac o s wi hin heal hca e and esea ch, we will wo k owa ds he mos e ec i e,
a o dable and sus ainable alue-based ca e o AYA wi h cance o maximize hei heal h ou comes.
Wi hin STRONG-AYA we will make use o ede a ed lea ning o implemen he COS and c ea e he
na ional and pan-Eu opean ecosys em. Con en ional da a analysis equi es sha ing and
cen aliza ion o da a. Bu , combining da a o igina ing om mul iple sou ces is di icul because o
e hical, adminis a i e, legal, and poli ical ba ie s associa ed wi h da a sha ing [23]. In o de o a oid
hese issues, we will use a hyb id model o cen alised and ede a ed analysis o lea ning echniques.
Cen alised app oaches will mainly be applied in he local ecosys ems, while ede a ed app oaches
a e speci ically ele an o lea ning be ween ecosys ems a a pan-Eu opean and he na ional le el.
Fede a ed analysis/lea ning is an A i icial In elligence echnique – i.e. machine lea ning – ha
e o mula es con en ional da a analysis algo i hms so ha da a cen aliza ion becomes unnecessa y
and consequen ly da a ans e and associa ed ag eemen s a e no needed [24]. Fede a ed
analysis/lea ning is de ined as analysing o lea ning om da a wi hou da a lea ing he sou ce:
algo i hms i e a i ely analyse sepa a e da a sou ces (si es) and only agg ega ed s a is ics a e
P o ocol e sion 1.0
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p oduced as ou pu s wi h he ou side wo ld [24]. In o he wo ds: esea ch ques ions and answe s a e
sha ed be ween he da abases ins ead o he da a. Fede a ed analysis/lea ning is a sus ainable
app oach o AYA da a as i o e s a p i acy-p ese ing solu ion o combine dispe sed da a o yield
o de s o magni ude mo e da a han adi ional app oaches [24]. The ede a ed analysis/lea ning
me hodology equi es and o ced he de elopmen o da a wi h seman ic in e ope abili y: FAIR da a
[25]. An ad an age o ede a ed analysis/lea ning is ha i makes implemen a ion o models in daily
clinical p ac ice possible (e.g. clinical decision suppo sys ems o diagnos ic decision-making),
he eby acili a ing a apid lea ning heal hca e p ac ice [26].
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2 STUDY OBJECTIVES
The p ima y aim o he STRONG-AYA conso ium is o de elop ou come p edic ion models
(p e alence/incidence o , isk ac o s o and mechanisms o poo ou comes) o AYAs wi h cance
h ough ede a ed lea ning (FL). These models will in o m u he AYA cance esea ch and es ablish
bes p ac ice in ca e. The FL me hod will desc ibe and model he ela ionships be ween he key
elemen s o da a om pa ien s aged 15-39 yea s a p ima y cance diagnosis ac oss mul iple
in e na ional cen es unde hei local, alid e hical app o al. The FL me hod enables he esea ch
eam o achie e his wi hou any indi idual le el pa ien da a lea ing he ins i u ion i o igina es
om, he e o e p ese ing pa ien da a p i acy. The scope o his p o ocol is, join ly, he collec ion o
da a de e mined by he COS de elopmen and he building and implemen a ion o he STRONG AYA
FL in as uc u e a he NKI. A ime o IRB submission (July 2023) we cu en ly in end o apply o only
collec da a as con ined by he TADP. I o when da a beyond he TADP is desi ed o STRONG AYA, a
new p o ocol o amendmen (in acco dance o IRB guidelines) will be de eloped.
The ollowing is equi ed o each he a o emen ioned objec i es:
1. Es ablish a COS o ou comes and a iables o da a collec ion and measu emen
o This is in p og ess and he d a COS is expec ed o be comple ed by he end o 2023.
The COS is being de eloped ia a sys emic li e a u e e iew, quali a i e in e iews
wi h pa ien s, HCPs, and ca egi e s, and a consensus-building Delphi p ocess. This
p ocess is de ined by he p o ocol pape and e hics app o al (78653) o he pa o
he STRONG-AYA s udy de ining he COS based p ima ily a he Uni e si y o
Sou hamp on. This p o ocol ou lined he scope and need o a COS; assembled a
wo king g oup; de eloped a s udy p o ocol encompassing a sys emic li e a u e
e iew, quali a i e in e iews and a Delphi s udy in alignmen wi h he EU CAYAS
NET s udy (h ps://ec.eu opa.eu/in o/ unding-
ende s/oppo uni ies/po al/sc een/how- o-pa icipa e/o g-
de ails/997929890/p ojec /101056918/p og am/43332642/de ails) o build mul i-
s akeholde consensus; as well as me ics and case-mix a iables. Re inemen o he
d a COS will ake place a e 2023.
2. Es ablish na ional ecosys ems in each o he i e pa icipa ing coun ies (Ne he lands, Uni ed
Kingdom, F ance, I aly, and Poland), consis ing o
a) An in e disciplina y, mul i-s akeholde AYA cance ne wo k o expe s o acili a e
collabo a ion, dissemina ion and communica ion, as well as he day- o-day collec ion o
AYA pa ien da a ia ou ine clinical ca e in elec onic heal h eco ds (EHRs), alida ed
pa ien - epo ed ou come (PRO) ques ionnai es, e c.
b) A echnical in as uc u e and ools o da a managemen ha use ede a ed lea ning in
line wi h FAIR (Findable, Accessible, In e ope able, Reusable) da a p inciples (In p og ess,
cu en ly de ined by he Da a Managemen Plan, echnical bluep in , and de eloping
ope a ional plans o he a ious ecosys ems). I is en isioned ha his echnical
in as uc u e will include in eg a ed web-po als wi h a ious and app op ia e le els o
access and pe missions o use by a ious s akeholde s including HCPs, pa ien s, and
policymake s
P o ocol e sion 1.0
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c) A go e nance model and legal and amewo k o ensu e compliance wi h Eu opean and
each coun y’s legal and policy s ipula ions ega ding he collec ion, use and sha ing o
heal h ou comes da a (e.g. obus da a p o ec ion and Gene al Da a P o ec ion
Regula ion (GDPR) compliance
d) Secu e and locally con olled da a wa ehouses o local da a esou ces ha indi idual
pa ne s a e able o access solely by hemsel es as independen ins i u ions, such as:
esea ch da abases,
clinical ials case epo s,
cance egis ies,
hospi al-based elec onic heal h eco ds,
eposi o ies o pa ien (o sel ) epo ed ou comes, and
o he such da abases ha he Pa ne deems ele an o he STRONG-AYA
e) Sus ainabili y model plan o ensu e long- e m sus ainabili y o he ecosys ems o suppo
Eu opean/na ional/ egional/local cance policy o ganisa ions o build app op ia e AYA
cance se ices
3. In pa allel, o es ablish a pan-Eu opean umb ella ecosys em o manage consis ency and
in e ope abili y ac oss na ional ecosys ems, o se e as a Eu opean ga eway, and o suppo
he c ea ion o ecosys ems in o he Eu opean coun ies.
4. To conduc he ollowing ac i i ies when he na ional ecosys ems become ope a ional:
a) Re ospec i e da a (mainly clinical) will be collec ed om na ional and egional egis ies
o p e iously conduc ed e hically app o ed popula ion-based s udies, his means da a
ha e o be s anda dized and mapped on a common da a model o in eg a e hem in he
in as uc u e o be used o analysis and ou come epo s.
b) P ospec i e COS da a (clinical and pa ien - epo ed) will be newly collec ed in
pa icipa ing clinical cen es among newly diagnosed AYA wi h cance ;
c) Re ospec i e COS da a (clinical and pa ien - epo ed) will be collec ed in pa icipa ing
clinical cen es (su i o s);
We hypo hesise ha obus and gene alisable ede a ed models can be de eloped ac oss coun ies,
and ha hese models will demons a e clinically use ul p e alence/incidence a es, isk ac o s and
mechanisms o poo ou comes as de ined in he COS. By linking mul iple in e na ional cen es, he
ede a ed models can be de eloped using he la ges a ailable coho o AYA cance pa ien s.
P o ocol e sion 1.0
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P ojec imelines:
Task
Q4
'22
Q1
'23
Q2
'23
Q3
'23
Q4
'23
Q1
'24
Q2
'24
Q3
'24
Q4
'24
Q1
'25
Q2
'25
Q3
'25
Q4
'25
Q1
'26
Q2
'26
Q3
'26
Q4
'26
Q1
'27
Q2
'27
Q3
'27
Q4
'27
WP1: De elopmen COS and da a collec ion
COS de elopmen : Con ene s akeholde s,
le e age li e a u e and build consensus ia he
Delphi panel
Digi al echnology equi emen s and design o
epo s (quali a i e esea ch in o ming echnical
unc ionali y o STRONG AYA in as uc u e)
Implemen a ion o COS and da a collec ion a
pa icipa ing cen es
COS e ision and e inemen
S a i ica ion and ou come p edic ion
WP2: Go e nance, da a secu i y
and e hics
Ecosys ems go e nance model and business
a chi ec u e
Pa ien
-
cen ed
and pa ien
-
in o med e hical
guidance ( o me ly S akeholde alue)
Go e nance, da a p o ec ion and e hics
WP3:
In as uc
u e and
A chi ec u e bluep in de elopmen
Pa ien ool de elopmen
P o ocol e sion 1.0
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P o ocol e sion 1.0
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5 Da a lows and ede a ed lea ning in as uc u e
Following a simila a ionale o he a omCAT2 s udy, sha ing da a insigh and coope a ion in mul i-
cen e s udies is in aluable o he de elopmen and alida ion o p ognos ic models in a e cance s,
which by de ini ion encompass all AYA cance s. Fede a ed lea ning in as uc u es and he da a low
p inciples behind i ensu e da a insigh s can be sha ed whils p ese ing pa ien p i acy and local
au onomy o e da a. Fu he mo e, he agile na u e o ede a ed lea ning allow insigh s o be d awn
whils wo king wi h, no agains , he di e se local con ex s and si ua ions a ound da a collec ion a
each pa icipa ing cen e.
Da a lows:
Da a lows wi hin he STRONG-AYA conso ium, and a he NKI, will ollow he ollowing p inciples:
Each Pa ne con ols and ope a es i s own na ional o local ecosys em. This is expec ed o consis o ,
bu is no exclusi ely limi ed o, local da a esou ces ha he Pa ne is able o access solely by
hemsel es as independen ins i u ions, such as:
• esea ch da abases,
• clinical ials case epo s,
• cance egis ies,
• hospi al-based elec onic heal h eco ds,
• eposi o ies o pa ien (o sel ) epo ed ou comes, and
• o he such da abases ha he Pa ne deems ele an o he STRONG-AYA as pe he COS
These “g ass oo s” da a sou ces a e allowed o be ei he e ospec i e, o p ospec i e, o a mix u e
o bo h. Since each Pa ne owns, con ols and ope a es in hei own space, hey will au onomously
decide when and which Da a o make accessible o Fede a ed Lea ning h ough he STRONG-AYA
conso ium. The e is no in ellec ual, echnical o conso ium-de i ed manda o y equi emen on
e e y Pa ne o make all Da a comple ely accessible a exac ly he same ime; no ing, as men ioned
abo e, ha he da a collec ion pa adigm mus allow o dynamism, adap abili y and s ep-by-s ep
accumula ion (and ma u a ion) o he Da a o e ime.
Each Pa ne will be esponsible o eques ing indi idual-le el subjec da a om p ima y da a
sou ces wi hin hei own a ea o con ol (as abo e, he elec onic heal h eco ds, cance egis ies,
e c. e c.) using local go e nance mechanisms and echnical ins umen s ha a e easible o he
Pa ne . These mechanisms and ins umen s may, o illus a ion pu poses only, include Da a
T ans e Ag eemen s be ween hemsel es and na ional cance egis ies, and cus omized da a
ex ac ion ools om elec onic heal h eco ds hos ed in hei local hospi al (see, o a pu ely
illus a i e example, Figu e 1 below). Exis ing go e nance and ins umen s should be e-used
whe e e possible, wi h a iew o g ow and sus ainably each a be e quali y o da a collec ion o e
ime.
P o ocol e sion 1.0
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Figu e 1: da a lows o STRONG AYA
Each Pa ne s o es pseudonymized da a hey ha e ex ac ed om hei local sou ces in o a secu ed
compu a ion de ice which is wi hin hei ins i u ional da a secu i y i ewall. This de ice esides on a
sub-ne wo k ha can be insula ed om o he mission-c i ical and ope a ional sys ems used by he
ins i u ion. The Pa ne needs o ensu e su icien secu i y o he p i a e da a in hei own
collec ion, using he ad ice o hei local da a p o ec ion o ice s and in o ma ion secu i y eams. I a
Pa ne uses pseudonym keys o link in o ma ion o he same human subjec om mul iple da a
sou ces, such key iles mus be s o ed on a sepa a e de ice om he Da a i sel .
T adi ionally, machine lea ning models a e ained on da a ha a e collec ed cen ally om mul iple
sou ces. Howe e , he e hical, legal and socie al implica ions ega ding sha ing o da a ou side he
sou ce o ganiza ion has led o a pa adigm shi in how machine lea ning models a e ained. Ins ead
o sha ing da a, algo i hms a e sen o each o he da a sou ces, whe e hey a e ained locally. The
locally ained algo i hms a e agg ega ed o de elop a global model in a manne as i he algo i hm is
ained by adi ional app oaches. The p i acy and con iden iali y o da a is p o ec ed a he sou ce
and only necessa y analysis is sha ed.
P o ocol e sion 1.0
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Fede a ed lea ning
Figu e 2: P inciple pa s o ede a ed lea ning
STRONG-AYA will use he Pe sonal Heal h T ain (PHT) pa adigm o c ea e i s ede a ed lea ning
in as uc u e. The Pe sonal Heal h T ain (PHT) is a p i acy-by-design in as uc u e o pe o ming
ede a ed/dis ibu ed machine lea ning. The da a p o ide s (hospi als) can pa icipa e in mul icen e
da a analysis wi hou exposing sensi i e da a ou side he o ganiza ion. The p inciple o PHT can co-
exis wi h o he p i acy enhancing measu es such as enc yp ed and pe u bed da a, bu he c ucial
poin he e is ha he PHT pa adigm ne e elies solely on he unb eakabili y o enc yp ion no he
i e e sibili y o pe u ba ion o sa egua d pa ien p i acy. PHT ensu es ha he machine lea ning
p ocess is comple ed in a da a agnos ic manne . The da a s a ion p o ides an execu ion pla o m o
he algo i hm and p o ides secu e access o he da a eposi o y. In he PHT in as uc u e, he
me apho ‘ ain’ e e s o he Docke con aine s con aining he da a que y and he algo i hm. These
con aine s p o ide an isola ed and secu e en i onmen o he que y and algo i hm. ‘T acks’ a e he
secu ed communica ion channels connec ing he cen al message b oke , he esea che and he
da a s a ions. T ains a e sen o he da a s a ion h ough he acks and only au hen ica ed use s a e
allowed o igge a ede a ed lea ning. The open sou ce implemen a ion o PHT, Van age
(h ps:// an age6.ai/) will be used as he so wa e solu ion o pe o ming p i acy p ese ing
ede a ed machine lea ning on he da ase s.
P o ocol e sion 1.0
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Figu e 3: PHT que y jou ney
The PHT was de eloped by expe s a he IKNL and he Uni e si y o Maas ich and has been applied
in nume ous oncological use-cases in he Ne he lands and in e na ionally. Selec ed p ojec s include
PROSPECT, a p os a e cance s udy, o which NKI-A L is a collabo a o ; a e cance egis y
de elopmen s such as RARECANne Asia and BLUEBERRY (IKNL), he a omCAT anal cance s udy (A L-
NKI).
The speci ic compu ing esou ces equi ed o STRONG-AYA a e ou lined in he echnical bluep in
a ached which is he e ol ing s anda d ope a ing p ocedu e de ining he in eg i y and sa e y o he
sys em alongside he Da a Managemen Plan.
In he p oposed ede a ed s uc u e, he exchange o s a is ical summa ies and model coe icien s
(e.g., odds a io) a e as close as possible o anonymiza ion wi hou becoming un i o he pu pose o
ac ionable insigh s and es able hypo heses o imp o e he ca e o AYAs wi h cance . In so a as he
same such s a is ics and esul s a e p in ed in public epo and a icles, hen he in o ma ion
exchanged in ede a ed lea ning is NOT any mo e iden i iable han a icles and epo s. Sa e y and
in eg i y o he local da a spaces is he esponsibili y o he local PI and his/he IT eam. Sa e y and
in eg i y o he ede a ion c oss-ins i u ional communica ions ne wo ks will be he esponsibili y o
Medical Da a Wo ks, who ha e been subcon ac ed o hos he STRONG-AYA cen al se e . The
design o he so wa e sa egua ds and he algo i hmic in eg i y is a join unde aking o UNIMAAS
and IKNL.
P o ocol e sion 1.0
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6 Da a analysis
Sample size:
As p e iously men ioned, since he main aim o his s udy is o implemen he COS, no o mal sample
size calcula ion will be done and will be dependen on he speci ic sub-ques ion on analysis
unde aken by esea che s using he STRONG AYA ecosys em
F om all AYA wi h cance diagnosed in he STRONG-AYA cen es, we expec ha he p ospec i e pa
o STRONG-AYA will be a ailable o a ound 4000 pa ien s each yea (please see he Table below) ha
will be ollowed longi udinally o e ime (each yea ). This numbe will only g ow when STRONG-AYA
will open in o he coun ies. When pa ien inclusion o he p ospec i e pa o he da a collec ion is
no possible di ec ly a e diagnosis, i is also possible o include pa ien s o su i o s la e in hei
pa ien jou ney ( e ospec i e clinical p ac ice). We will aim o a esponse a e o 25%.
AYA Cance pa ien s pe ins i u ion based on his o ical da a
Sho Name
His o ical
numbe s
( e ospec i e)
New cases expec ed pe yea (p ospec i e)
IRCCS
2000
400 (p ima y ocus on sa comas; b eas , es icula and
melanoma)
UL
6000
600 (all umou ypes)
MU
2000
355 (p ima y ocus on B eas ,
Gynae
cology
, Sa coma, Ge m cell,
CNS)
MSCN
RIO
600
150 (p ima y ocus es icula cance s, sa comas)
I
GR
200
0
800 (all umou ypes)
CLB
2000
150 (all umou ypes)
NKI
4000
500 (all umou ypes)
YCE
12775
>1000 (all umou ypes)
Wi h ega d o e ospec i e egis ies/popula ion-based s udy da a: As an impo an s ep owa ds
he de elopmen o u u e pe sonalised p edic ion models, STRONG-AYA will also u ilise da a om
he exis ing da abases a ailable wi hin i s conso ium (e.g. in The Ne he lands: clinical da a om he
Ne he lands Cance Regis y and pa ien - epo ed ou comes ia he PROFILES egis y o o e 4000
AYA who we e diagnosed wi h cance 5-20 yea s ago, as well as he SURVAYA da ase (IRBd18-122);
in UK: clinical da a om he Na ional Cance Regis y).
S a is ical analysis:
A ull da a analysis plan will be de eloped p io o da a analysis ini ia ion and will be p e- egis e ed in
a ele an public eposi o y (e.g. Open Science Founda ion, h ps://os .io). As he esea ch ques ions
applied o STRONG AYA will be highly a iable, his documen will be dynamic and e e -e ol ing.
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Desc ip i e da a analysis: p e alence/incidence poo ou comes
Summa y s a is ics will be exchanged be ween cen es in o de o calcula e p e alence/incidence
a es o poo ou comes and explo e coho di e ences p io o modelling. Ca ego ical a iables will
be compa ed using chi-squa e es , and nume ical a iables will be compa ed using a one-way
ANOVA es . All es s will be ca ied ou using summa y s a is ics (numbe o pa ien s, mean and
s anda d de ia ion alues) a he han indi idual pa ien da a. Es ima ed su i al / ecu ence a es
and po en ial ollow-up imes will be calcula ed by each cen e indi idually, using he Kaplan-Meie
es ima o . The median ollow-up ime will be calcula ed based on he in e se Kaplan-Meie
es ima o . A de ailed app oach o handling missing alues, on a wi hin- and be ween-cen e basis,
will be speci ied in he da a analysis plan.
Risk ac o s and mechanisms
Al eady-es ablished p edic i e and p ognos ic ac o s o he ou comes in ques ion will be iden i ied
h ough a sys ema ic e iew o he li e a u e. This app oach iden i ied he ini ial lis o ele an da a
o be collec ed; his was subsequen ly p io i ised in a Delphi s udy, and addi ional ac o s we e
added. A inal plan o ac o selec ion and model e inemen will be a ailable in he da a analysis
plan.
(mul i a ia e) Linea and logis ic eg ession analyses o con inuous and dicho omous ou comes
espec i ely, and Cox eg ession o ime-dependen ou comes will be used. Mode a ion and
media ion e ec s will be explo ed, depending on esea ch ques ion. Mul i a ia e analyses will be
adjus ed o iden i ied case-mix ac o s.
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7 ETHICAL CONSIDERATIONS
This s udy p o ocol assumes s anda d o ca e is p o ided o AYA cance pa ien s, he e is no clinical
in e en ion pe o med by o wi hin his p o ocol. The e o e, no in o med pa ien consen is needed
o collec he da a desc ibed in his p o ocol.
Each ins i u ion will acqui e sepa a e local app o als o accessing and collec ing pa ien da a o
esea ch; he local coo dina ing in es iga o will be esponsible o p o iding a copy o he le e
con i ming ha use o da a o esea ch is app o ed (e.g. om Ins i u ion Re iew Boa d, IRB),
including app o al e e ence numbe .
As no indi idual pa ien da a will be exchanged be ween ins i u ions, no da a ans e ag eemen s o
addi ional pa ien consen will be needed. Howe e , in cau ion o mi iga e p i acy isks, a Join
con olle ship ag eemen will be d a ed be ween all pa ne s o he STRONG AYA conso ium p io
o da a collec ion. Local in o ma ion go e nance (IG) and da a p o ec ion e iew o he dis ibu ed
lea ning in as uc u e may be needed, and should be ob ained whe e e app op ia e. The s udy
coo dina o s may be able o assis wi h suppo ing ma e ial.
A pan-Eu opean ROPA and DPIA has been conduc ed and ci cula ed o all STRONG AYA conso ium
membe s o e iew and sign. I is expec ed o be a dynamic documen ha e ol es wi h he p ojec
o e ime o ensu e bes p ac ice ega ding da a secu i y.
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8 ORGANISATION AND POLICIES
S udy o e sigh and managemen
De ails o he conso ium engagemen and p ojec managemen a e desc ibed in de ail in a
collabo a i e esea ch ag eemen and he conso ium ag eemen . In b ie , he conso ium consis s o
he senio in es iga o s, co-in es iga o s, and local ins i u ional pa icipan s. Conso ium
membe ship will be inclusi e, wi h all indi iduals con ibu ing (e.g. o local ins i u ional wo k)
eligible, bu each cen e will be asked o name a single cen e lead who will ake inal esponsibili y
o cen e con ibu ion.
Day- o-day managemen will be handled by he s udy coo dina o , suppo ed by he conso ium
leade ship eam (senio in es iga o s and co-in es iga o s). This g oup will mee egula ly o ensu e
smoo h s udy conduc and communica ion. The s udy coo dina o mee s wi h he in ol ed pa ies o
each wo k package mon hly o s ay on op o ac i i ies and conce ns and epo s weekly o he lead
PIs. A de ailed e e ence ega ding he p ojec ’s decision making s uc u es, communica ions
guidelines, quali y assu ance o deli e ables, con lic esolu ion and isk managemen plans, as well
as o he policies and p ocedu es, ha e been inco po a ed in o he P ojec Handbook. Addi ional
online mee ings o he ull conso ium will p o ide s udy o e sigh .
As pa ien -cen ed esea ch is a enan o he p ojec ’s objec i es and e hos, a Pa ien Ad iso y
Boa d is cu en ly being ec ui ed o p o ide pa ien ad ice on STRONG AYA’s ac i i ies. This is
u he complimen ed by an E hics and Regula o y Ad iso y Boa d and a Scien i ic Ad iso y Boa d o
ensu e ha STRONG AYA does no exis in an in ellec ual silo and is accoun able o expe s in he
ele an a eas.
Publica ion policy
STRONG AYA’s au ho ship policy ollows he accep ed p inciples o es ablishing au ho ship
acco ding o he In e na ional Commi ee o Medical Jou nal Edi o s which de ines he ou ollowing
c i e ia o claiming au ho ship:
Subs an ial con ibu ions o he concep ion o design o he wo k; o he acquisi ion,
analysis, o in e p e a ion o da a o he wo k;
D a ing he wo k o e ising i c i ically o impo an in ellec ual con en ;
Final app o al o he e sion o be published;
Ag eemen o be accoun able o all aspec s o he wo k in ensu ing ha ques ions ela ed o
he accu acy o in eg i y o any pa o he wo k a e app op ia ely in es iga ed and esol ed.
The con ibu ions o au ho s should be iden i iable, and au ho s should ha e con idence in he
con ibu ion o co-au ho s. Au ho ship posi ions and he co esponding au ho should be decided
be o e he wo k is comple ed and wi h consensus among he pa ies. I con lic a ises, he ma e will
ollow he in e nal con lic esolu ion pa hway. All pa ies lis ed as au ho s should mee all ou o
he c i e ia ou lined in he ICMJE guidelines. The co esponding au ho should make su e ha hey
a e a ailable h oughou he submissions p ocess o espond o edi o ial que ies and e iewe ’s
commen s in a imely manne . The au ho ship c i e ia is no in ended o use o disquali y STRONG-
AYA colleagues om au ho ship who o he wise mee au ho ship c i e ia by denying hem he
oppo uni y o mee he second and hi d c i e ia. In addi ion o lis ing au ho s, all scien i ic
publica ions s emming om STRONG AYA will acknowledge he conso ium ia he use o an
P o ocol e sion 1.0
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‘ac onym’ o s a emen ollowing he au ho ship lis “...on behal o he STRONG-AYA conso ium”.
A ull lis o STRONG-AYA con ibu o s, by ins i u ion, will be lis ed in he acknowledgemen s
o publica ions. This lis will be e iewed be o e publica ion submission o ensu e i is up o da e.
Au ho s a e expec ed o acknowledge non-academic suppo as well whe e possible, including bu
no limi ed o hose who ha e p o ided ma e ials o me hods, o ganiza ional suppo , echnical
suppo , clinical cases, pa ien s and hei ela i es, unding bodies, e c. All success ul publica ions
should be epo ed o he P ojec Coo dina o o inclusion in he dissemina ion acke . Fo he
p ima y s udy publica ions, we plan o name he senio in es iga o s and co-in es iga o s, and one
local lead pe ins i u ion as co-au ho s. We will apply o a conso ium “g oup” au ho ship, and
wi hin his g oup au ho ship, we will add he names o each addi ional pe son ha has con ibu ed in
a meaning ul way o his p ojec , including all ele an conso ium membe s.
In as uc u e use ag eemen
Medical Da a Wo k BV (MDW, h ps://medicalda awo ks.nl/) implemen s a p i acy p ese ing
dis ibu ed in as uc u e ha in es iga o s in STRONG-AYA will use. The e o e, an In as uc u e
Use Ag eemen (soon o ollow) is equi ed as a con ac ual ag eemen be ween each ins i u ion
and MDW, and he same ag eemen is equi ed be ween he p incipal in es iga o s and MDW.
MDW will no be conside ed as a “p ocesso ” o clinical da a acco ding o he de ini ion in he EU
Gene al Da a P o ec ion Regula ion, bu is solely he p o ide o he in as uc u e. As he
in as uc u e p o ide , MDW will en o ce he legal use o algo i hms and da a s a ions, and his
ag eemen shall de ine he e ms and condi ions o he use o he in as uc u e.
A empla e In as uc u e Use Ag eemen ( om MAASTRO) will be p o ided o all pa ne s in he
p ojec .
P o ocol e sion 1.0
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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 .
Pseudonymisa ion: he p ocessing o pe sonal da a in such a manne ha he pe sonal da a
can no longe be a ibu ed o a speci ic pe son, including pa ien s, wi hou he use o
addi ional in o ma ion, p o ided ha such addi ional in o ma ion is kep sepa a ely and is
subjec o echnical and o ganisa ional measu es o ensu e ha he pe sonal da a a e no
a ibu ed o an iden i ied o iden i iable na u al pe son in he con ex o he STRONG AYA
p ojec .
P o ocol e sion 1.0
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Abb e ia ions
Ac onym/Abb e ia ion
Meaning
DMP
Da a Managemen Plan
FAIR
Da a
p inciples o Findabili y, Accessibili y,
In e ope abili y, and Reusabili y
HCP
Heal h Ca e P o ide
PRO
Pa ien Repo ed Ou come
COS
Co e Ou come Se
PROM
Pa ien Repo ed Ou come Measu e
ROPA
Reco ds o P ocessing Ac i i ies
DPIA
Da a P o ec ion
Impac Assessmen
GDPR
Gene al Da a P o ec ion Regula ion
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 )
P o ocol e sion 1.0
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1. In oduc ion
1.1. Backg ound: adolescen s and young adul s wi h cance
STRONG-AYA is 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 (AYA) wi h cance ,
de ined as indi iduals aged 15-39 yea s a cance diagnosis. 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) alongside dec eased
quali y o li e due o cance and i s ea men . Unlike dedica ed heal hca e and ials o pedia 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 e y impo an social and economic demog aphic g oup, , need access o age-
adjus ed and high-quali y heal hca e. AYA ca e and esea ch will bene i om collec ion and pooling
o pa ien -cen e ed da a and collabo a ion among all s akeholde s. 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 he
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, pe haps because 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 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
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.
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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.
Despi e he inc easing awa eness and a g owing body o scien i ic li e a u e, hese unique
issues a e no ye 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 .
1.2. P ojec aims
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
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.
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.
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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.
AYAs, as a unique socio-economic g oup, need access o age-adjus ed and high-quali y
heal hca e. AYA ca e and esea ch will 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. Ou conso ium includes clinical and scien i ic leade s in AYA-ca e, da a science and
egis ies, The Eu opean Cance O ganisa ion (ECO), You h Cance Eu ope (YCE) and he Eu opean
O ganisa ion o Resea ch and T ea men o Cance (EORTC), building on p e ious ini ia i es and EU
g an s, and me ging expe ise wi h inno a ion. Wi hin STRONG-AYA, we will se up a alue-based
heal hca e esea ch ecosys em o de elop da a-d i en, in e ac i e policy and isualiza ion ools ha
b ing, in co-c ea ion wi h all s akeholde s including pa ien s, no el insigh s in o AYA heal hca e. This
will equi e he es ablishmen o symbio ic, independen , no - o -p o i sys ems o wo king based a
he local le el ac oss he i e heal h se ices cu en ly pa icipa ing in STRONG AYA, as well as a pan-
Eu opean ecosys em o manage he connec ions be ween hem.
The ecosys em business a chi ec u e deli e able will co e he ollowing aspec s o he
o ganisa ional and design aspec s o he STRONG AYA ecosys ems:
The STRONG AYA ision
Go e nance key p inciples ha guide he es ablishmen and managemen o he STRONG AYA
ecosys ems, including p esen and u u e pa icipa ing ecosys ems
The legal s uc u e unde pinning he o ma ion o he STRONG AYA ne wo k a bo h na ional
and pan-Eu opean le els
A synopsis o he da a access and lows in he ne wo k
The o ganisa ional aspec s and esponsibili ies o he ecosys ems
A cu so y ske ch o how STRONG AYA ecosys ems may be sus ained beyond he unding pe iod
Gi en he in e connec ed na u e o he STRONG AYA’s wo k packages, his deli e able pulls on
wo k unde aken ac oss se e al o i s wo k lows: WP2, WP3, and WP4. I is he encapsula ion o he
wo k unde aken hus a and he e o e should be iewed as a li ing documen ha will e ol e o e
ime.
2. Me hods and ma e ials
The WP2 leads, he EORTC, along wi h inpu om colleagues a he B igh lands Ins i u e o Sma
Socie y a Maas ich Uni e si y (BISS) and he Uni e si y o Leeds (UoL) designed a ques ionnai e o
assess he local legal p ocesses and go e nance con ex s wi hin each pa ne ins i u ion o es ablish
an ini ial comp ehensi e unde s anding o wha he STRONG AYA go e nance and legal s uc u es
should en ail. This is necessa y o mee he objec i es o he p ojec in e ms o da a deli e y,
echnology, and impac whils main aining compliance.
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The wo k package collabo a i ely de eloped ques ionnai es o ga he in o ma ion on he local
ins i u ional and na ional egula ions, p ocedu es and p ocesses a ound da a secu i y and lows. This
was hen expanded upon using in dep h de ailed on-one-one mee ings be ween WPLs and each
indi idual da a-con ibu ing pa ne :
The Ne he lands Cance Ins i u e (NKI) based a he An oni an Leeuwenhoek Hospi al in
Ams e dam
The Ma ie-Sklodowska-Cu ie Na ional Ins i u e o Oncology (MSCNRIO) in Wa saw
The Ins i u o Nazionale dei Tumo i (INT) in Milan
Ins i u Gus a e Roussy in Pa is
Cen e de Lu e Con e le Cance Leon Be a d in Lyon
The Ch is ie NHS Founda ion T us (a hospi al us a ilia ed wi h he Uni e si y o
Manches e ) in Manches e
The Leeds Teaching Hospi al NHS T us (a hospi al us a ilia ed wi h he Uni e si y o
Leeds) in Leeds
This es ablishes:
Wha a e he common elemen s o AYA heal hca e da a collec ed and easibly collec ed by
p ojec pa ne s (WP1)
Consensus and p elimina y Tie 1 lis o minimum COS, o lay he ounda ion o u u e
Delphi-s yle consensus on upg aded ex ended COS (WP1)
Exis ing legal, go e nance and da a managemen p ocedu es ha a e al eady in place a
pa ne ins i u ions (WP2, WP3)
he deg ee o unde s anding o ede a ed esea ch (da a accessing) ecosys ems, and how
hese can i wi hin he egula o y in e p e a ion o he pa icipa ing pa ne s (WP2, WP3)
he ex en o which he exis ing esea ch and ope a ional in as uc u es can be in eg a ed
wi h he STRONG AYA ision (WP3)
le el o deg ee o da a p epa edness (and hence da a p epa a ion needed) in o de o
es ablish he heal hca e o AYA pe sons needed o his p ojec (WP3)
In addi ion o his, The Uni e si y o Leeds, wi h inpu om conso ium membe s led in he
concep ualisa ion o he ecosys em’s o ganisa ional design and conduc ed a li e a u e e iew o
unde s and he wide con ex o heal hca e esea ch ecosys ems and wha hey en ail. The
Maas ich Uni e si y Clinical Da a Science lab (UNIMAAS) led on iden i ying he design p inciples and
echnology bluep in s equi ed o implemen ing a STRONG AYA echnical in as uc u e. UNIMAAS
and UoL a e wo king in close alignmen wi h he EORTC o de elop he Reco d o P ocessing Ac i i y
(ROPA) and Da a P o ec ion Impac Assessmen (DPIA) o he conso ium as a whole. Th ough
consul a ion wi h WPLs and use o expe ise, ECO has begun mapping s akeholde s o ano he
deli e able (D4.1) which will in o m he e olu ion o he ecosys em as i ma u es. This deli e able
also pulls on concep s a icula ed in he echnical bluep in c ea ed o WP3 in he con ex o D3.1
(Da a Managemen Plan) and D3.3 (de elopmen o a chi ec u al bluep in ). The legal p inciples
unde pinning he ecosys em a chi ec u e a e b oadly a icula ed below and will be discussed in
u he de ail in D2.2/D2.3 (STRONG AYA in o ma ion go e nance and e hics amewo k/e hics
guidelines). The discussion o sus ainabili y is in i s in ancy and ep esen s some ini ial hough s ha
will be u he de eloped including wi hin deli e able D4.12 ( o be p o ided in Sep embe 2025
(M36)).
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3. STRONG AYA Mission
STRONG AYA’s mission is o imp o e heal hca e se ices, esea ch and ou comes o Adolescen s and
Young Adul s (AYA) wi h cance . As ou lined in dep h abo e, AYAs wi h cance 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 du ing a whole li e o cance su i o ship. In addi ion, he e a e se e al challenges ha
AYAs p esen o adi ional esea ch and ca e. AYAs a e a e y complex g oup clinically, sociologically
and demog aphically which makes using s anda dised p o ocols e y di icul . No only a e hey
liminal, exis ing sociologically be ween childhood and ma u e adul hood, bu hey a e also
expe iencing apid social, pe sonal and economic change. The model o ca e and ea men p o ocols
used o AYAs will o en depend on hei local con ex and whe he hey a e ea ed in paedia ic,
adul o in eg a ed/comp ehensi e heal hca e se ings. A numbe o challenges limi e o s o
imp o e ca e and es ablish in e en ions o imp o e AYA quali y o li e. The liminali y o AYAs means
he e is a dea h o AYA dedica ed ials, hei g ea e mobili y makes i di icul o enable a el o
ial access and hey a e less likely o ha e inancial s abili y ha enables lea ing wo k o ial
pa icipa ion. AYAs wi h cance hen equi e a new app oach ha can allow esea che s and
clinicians o access eal wo ld insigh s om a la ge popula ion and has he agili y no only o adap o
a he e ogenous s udy popula ion bu also o a ying heal hca e sys ems. Ou ede a ed lea ning
ecosys ems will allow us o do bo h.
To achie e ou mission and o ec i y he dispa i y in AYA ca e and esea ch and o imp o e AYA
ou comes ac oss Eu ope, STRONG AYA is guided by he ollowing p inciples:
1) Heal hca e should be alue-based
STRONG AYA seeks o use da a-d i en insigh s as a s a ing le el o e idence o d i e apid
de elopmen s in esea ch, ca e and policy. AYA cance is conside ed ‘ a e’ in oncology wi h an
incidence a e o less han 6 pe 100,000 pe sons pe yea . This means ha clinical expe ience is
dilu ed and ( o example) AYA cance is mo e likely o be suspec ed la e and diagnose a
ad anced s age. The e a e speci ic di icul ies in conduc ing clinical ials o AYAs as well.15 Fo
he s udies ha do exis , numbe s o subjec s a e una oidably small, making s a is ically
signi ican indings di icul and he e o e i is unclea wha a e he po en ially ac ionable insigh s
o imp o e heal hca e. By b oadening he esea ch using no el p i acy-sa e app oaches owa ds
da a sha ing and da a analy ics, STRONG AYA will b ing no el insigh s in o AYA heal hca e,
encou age e hical e-use o sca ce AYA esea ch da a , p o ide eal-wo ld e idence o all
impo an AYA oncology decision-make s, and hus, inc easing he bene i o pa ien s whils
making ca e mo e e icien .
2) Access o and expe ience o heal hca e should be consis en and anspa en ac oss Eu ope
As AYAs a e an unde - esea ched demog aphic and s addle he adi ional di isions be ween
paedia ic and adul oncology, he ype o o app oach o ca e an AYA pe son expe iences a ies
signi ican ly be ween paedia ic and adul oncological ins i u ions, be ween egions, and
be ween coun ies. By es ablishing a Co e Ou come Se o AYA ca e and using da a-d i en
insigh s ga he ed ia ou pan-Eu opean ecosys em, STRONG AYA will sha e insigh s wi h policy-
15 See o example, Fe a i e al (2021), 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, 6(2), 100096. h ps://doi.o g/10.1016/j.esmoop.2021.100096; and
Fe n e al ((2014). A ailable, accessible, awa e, app op ia e, and accep able: a s a egy o imp o e pa icipa ion
o eenage s and young adul s in cance ials. The Lance . Oncology, 15(8), e341–e350.
h ps://doi.o g/10.1016/S1470-2045(14)70113-5.
P o ocol e sion 1.0
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make s o add ess and educe inequali ies ac oss he en i e disease pa hway o di e en cance s
in AYAs in di e en egions in Eu ope wi h an aim owa ds es ablishing a Eu opean-wide
s anda d o ca e. STRONG AYA will enable AYA heal hca e quali y ou comes moni o ing by
acking access o AYA nu se specialis s, e ili y consul an s, and social wo ke s o discussion
abou wo k and es ablish benchma king be ween he di e en coun ies on key pe o mance
indica o s.
3) Co-c ea ion wi h s akeholde s, especially pa ien s
Clinical ials and medical esea ch his o ically ha e been d i en by he ques ions and conce ns
clinicians and esea che s wish o in es iga e. Howe e , i is pa ien s who a e di ec ly impac ed
by he ou comes o esea ch. Thei ques ions and conce ns should be placed on equal oo ing
wi h clinical esea ch a eas o in e es . By in ol ing pa ien s in he p ocess o co-c ea ion,
STRONG AYA will c ea e a pla o m ha se es pa ien s and empowe s hem. STRONG AYA will
equip AYAs wi h cance wi h he da a o be e op imise hei heal hca e, and suppo sha ed
and pe sonalised decision-making be ween AYA and heal h ca e p o ide s. This also ex ends o
he con ol o , access o, and use o AYA’s da a in he con ex o esea ch and heal hca e.
4) Con ol o e da a
The key enabling echnologies in STRONG AYA will e ol e a ound pa ien s and da a-ga he ing
ins i u ions (such as clinical egis ies and ea ing hospi als) being able o e ain he ask o
con ol o e hei da a. Asking pa ien s and ins i u ions o hand o e hei da a o a cen alized
Eu opean eposi o y signi ican ly inc eases he “ us dis ance” be ween he pe son gi ing he
da a and he s ewa d gua ding he da a, in e ec e oding he ex en o di ec con olle ship held
by he da a con ibu o .
STRONG AYA implemen s a ede a ed pan-Eu opean da a access in as uc u e while
allowing da a con ibu o s, such as ea ing ins i u ions and pa ien o ganiza ions, o e ain di ec
con olle ship o e hei own da a. This keeps he da a p i a e and p o ec ed agains being used
by anyone ou side o he con olling ins i u ion. Fede a ed in eg a ion o mul i-ins i u ional da a
e ains local go e nance and con ol o e da a.
The inno a ion we implemen wi hin STRONG AYA is o de i e da a-d i en heal hca e
insigh s om mul iple se s o p i a e and geog aphically agmen ed da ase s; pa ne s
UNIMAAS and IKNL in he conso ium a e specialis expe s in his ques ion o “ ede a ed
lea ning” which nume ous success ul p ojec s on hei ack eco d.
5) FAIR and Fede a ed da a: sha ing insigh wi h compliance
STRONG AYA ollows he FAIR da a managemen p inciples which means ha da a emains
indable, accessible, in e ope able, and eusable e en while i s ays ully p i a e unde he di ec
con ol o dispa a e ins i u ions. The FAIR p inciples will acco dingly be applied a pa ne ,
na ional and pan-Eu opean le els o da a, and subsequen ly digi al a e ac s (e.g., so wa e,
publica ions, epo s) gene a ed om he p ojec wo k. STRONG AYA will u ilize ede a ed
analy ics, i.e. he pe o mance o s a is ical analyses and summa y isualisa ions o da a bu
wi hou ansmi ing any indi idual pa ien -le el da a ou side o he da a-owning ins i u ion.
STRONG-AYA also suppo s ede a ed lea ning such ha p ognos ic and p edic i e (s a is ical)
models can be de eloped wi hou exchanging indi idual pa ien -le el da a. P i acy p o ec ion o
ede a ed analy ics and ede a ed lea ning is based on he un easibili y o e e se-enginee ing a
pa icula indi idual subjec ’s iden i y om summa ies, s a is ical models and o he agg ega ed
P o ocol e sion 1.0
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esul s (which a e ypically p esen ed o epo s and publica ions). This means ha STRONG AYA
pa ne s and s akeholde s will be e hically, echnically and p agma ically empowe ed o sha e
insigh s bu no he ac ual da a. This allows STRONG AYA o gene a e new insigh s om
agg ega ed da a o achie e i s aims, in a way ha complies wi h na ional and Eu opean
s anda ds o p i acy, e hics and da a secu i y.
6) Sus ainable scalabili y: o e new oppo uni ies wi hou lea ing anyone behind
The de elopmen o a COS o AYAs wi h cance can enable STRONG AYA o o e new
oppo uni ies o he collec ion o p ospec i e da a ha some o ou pa icipa ing cen es ha e
ne e collec ed be o e suppo ing new esea ch and ca e inno a ion. Fede a ed lea ning ma ks a
new on ie in using da a o sol e mode n heal h challenges. Fede a ed lea ning is an agile and
adap able model ha allows us o lexibly inco po a e exis ing sys ems and p ac ices in o a la ge
ecosys em o knowledge exchange. A guiding p inciple o he STRONG AYA ecosys ems is o
accommoda e local cen es and hei con ex s whils connec ing hem ac oss Eu ope.
Ou ecosys ems a e ways o wo king ha aim:
To gene a e a s onge cul u e o success ul and sus ainable wo king oge he abou AYA
wi h cance
To in eg a e ‘ adi ional’ clinical, epidemiological and da a science p ocesses wi h pa ien -
cen ed da a, pa ien s and hei g oups, policy make s and o he s
To speed up key analyses o da a abou AYA wi h cance
To imp o e se ices, expe iences, and ou comes
Ou ecosys ems include:
Sophis ica ed websi es
A se o local da abases, se up in a e y speci ic way
Po als h ough which s akeholde s will en e and access in o ma ion
Tools ha we design and manage access o, ha can op imise he knowledge gained om
ou local da a
Expe ise equi ed o ope a e he ecosys em a bo h he Eu opean (expe guidance) and
local (clinical suppo , da a managemen , and echnical/IT suppo )
The COS and he ede a ed in as uc u e bo h allow STRONG AYA o scale and he e o e inc ease i s
sus ainable po en ial.
4. Go e nance P inciples, Da a Secu i y and E hics
4.1 Gene al Go e nance - GDPR
The in-dep h de elopmen o he go e nance p inciples o STRONG AYA will be comple ed in he i s
yea o he p ojec . An in en o y o ele an exis ing e hical and go e nance codes and guidelines
al eady p esen in he a ious pa ne ins i u ions will esul in he de elopmen o he p ojec ’s
e hical and legal guidelines and amewo k and will align p inciples om he di e en STRONG AYA
pa ne s as well as ele an EU go e nance and e hics p inciples, such as he ‘Eu opean E hical
P inciples o Digi al Heal h’ (2022) and key p inciples o da a p o ec ion as laid ou in he GDPR.
These p inciples include (bu a e no limi ed o):
Law ulness, ai ness and anspa ency: [da a shall be] p ocessed law ully, ai ly and in a
anspa en manne in ela ion o he da a subjec );
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Pu pose limi a ion: [da a shall be] collec ed o speci ied, explici and legi ima e pu poses
and no u he p ocessed in a manne ha is incompa ible wi h hose pu poses; u he
p ocessing o a chi ing pu poses in he public in e es , scien i ic o his o ical esea ch
pu poses o s a is ical pu poses shall, in acco dance wi h A icle 89(1), no be conside ed o
be incompa ible wi h he ini ial pu poses;
Da a minimisa ion: [da a shall be] adequa e, ele an and limi ed o wha is necessa y in
ela ion o he pu poses o which hey a e p ocessed;
Accu acy: [da a shall be] accu a e and, whe e necessa y, kep up o da e; e e y easonable
s ep mus be aken o ensu e ha pe sonal da a ha a e inaccu a e, ha ing ega d o he
pu poses o which hey a e p ocessed, a e e ased o ec i ied wi hou delay;
S o age limi a ion: [da a shall be] kep in a o m which pe mi s iden i ica ion o da a subjec s
o no longe han is necessa y o he pu poses o which he pe sonal da a a e p ocessed;
pe sonal da a may be s o ed o longe pe iods inso a as he pe sonal da a will be p ocessed
solely o a chi ing pu poses in he public in e es , scien i ic o his o ical esea ch pu poses
o s a is ical pu poses in acco dance wi h A icle 89(1) subjec o implemen a ion o he
app op ia e echnical and o ganisa ional measu es equi ed by his Regula ion in o de o
sa egua d he igh s and eedoms o he da a subjec ;
In eg i y and con iden iali y: [da a shall be] p ocessed in a manne ha ensu es app op ia e
secu i y o he pe sonal da a, including p o ec ion agains unau ho ised o unlaw ul
p ocessing and agains acciden al loss, des uc ion o damage, using app op ia e echnical o
o ganisa ional measu es;
Accoun abili y: The con olle shall be esponsible o , and be able o demons a e
compliance wi h, pa ag aph 1 [ he p e ious 6 p inciples]).
Whils i is p ema u e a he ime o w i ing (Sp ing 2023) o ully elabo a e on each o hese
p inciples, we can expand upon some p ac ical go e nance p inciples a ound da a access and use in
he ecosys ems.
The STRONG AYA go e nance p inciples chie ly conce n how he STRONG AYA conso ium de ines
wha i will do wi h da a and wha i canno do wi h da a e hically and legally. They add ess his a
wo geog aphical le els: he local (usually na ional) ecosys ems and a single pan-Eu opean
ecosys em.
4.2. Da a p o ec ion p inciples and go e nance
1) Local da a owne ship and s ewa dship
A he pan-Eu opean le el, a undamen al p inciple is ha STRONG AYA will no ake o dis ibu e
any conso ium membe ’s da a no ac as i s s ewa d. Each ins i u ion is he s ewa d o i s own
da a and he e o e mus use exis ing sa egua ds and comply wi h local egula ions and
p ocedu es o ensu e he secu i y o da a held on hei own machines.
2) Pseudonymisa ion and compliance e iew
The da a ha STRONG AYA pa ne s will make p i a ely accessible in a ede a ed way will be
pseudonymised, and he COS a iables will be e iewed by da a secu i y expe s o ensu e ha
he desi ed COS a iables a e a he absolu e minimum necessa y o ge he ac ionable insigh s
ha he conso ium wan s o achie e and no supe luous o ha o e all objec i e, acco ding o
da a minimiza ion p inciple (a .. 5.1.c o he GDPR).
E hical go e nance o STRONG AYA da a p o ec ion ac i i ies will ake place h ough an in e play o
na ional go e nance p ocedu es ( ela ed o he i e coun ies in ol ed in his p ojec ), local
(ins i u ional) go e nance p ocedu es and p ojec -le el go e nance cen alized in he S ong AYA
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Se es mul iple s akeholde s
enables a i ual desk op linkage, ha ing access o pa s o local IT sys ems
The highes laye will be public – anyone inding he websi e can see his laye – ou pu pose,
ou achie emen s, ou openly published epo s, summa ies o se ices we o e o o he s
The nex laye migh be accessible o all S ong-AYA membe s, upon login. I migh include
in e nal policy d a s, o example, and ‘Apps’
Ano he laye migh be only a ailable h ough a speci ic new eques – o example an indus y
pa ne , esea che , o policy-make wan ing o commission a new analysis
Fu he laye s may include p og essi ely mo e es ic ed in o ma ion, as a as we wan , and
we a e com o able wi hin ou in o ma ion secu i y ules and sys ems
2. a se o da abases a he local le el (Da a lake-houses) ha :
will s o e indi idual-pa ien da a in a p i a e box, o ‘sa e space’ wi hin ou local sys ems
will exis only acco ding o he local egula ions ha we ou inely use al eady, o ha we
de elop locally du ing S ong-AYA
will es ic access o local eam membe s acco ding o hei pe missions (unless [e.g. in one
na ion] pe missions a e gi en o ha e been gi en o sha e a a local le el)
3. Po als h ough which s akeholde s will all be able o:
En e in o ma ion and da a ( ha hey ‘own’ e hically and legally and a e pe mi ed o en e )
View in o ma ion and da a o moni o , e alua e and compa e ou comes (a a le el o de ail
ha i s wi h hei pe missions)
See ele an da a summa ies whe he o clinical, esea ch, se ice use o policy pu poses,
acco ding o hei pe missions
Re-use al eady exis ing ins i u ional in as uc u e and da a wa ehouses o he maximum
ex en possible
4. Tools ha can op imise he knowledge gained by us and o he s om ou wo k
By unning p ede ined analyses au oma ically (‘in eal ime’, ‘on demand’, ‘on he ly’) upon
ou da a
By building AYA-speci ic aspec s, in o ( o example):
U ilising exis ing epidemiological ques ions (Eu oCa e)
U ilising exis ing pa ien -d i en ques ions ( hei websi es)
U ilising exis ing policy ques ions (ECO)
The STRONG AYA ecosys ems a e also made up o indi iduals wi h expe ise and/o oles. Some o
hese oles may be held by he same o mul iple membe s wi hin o wi hou he STRONG AYA
conso ium a a gi en ime. These include:
1. Da a p o ide s
S o e
De elope
Agg ega o
Ha monize
Publishe
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Regis e
Da a main aine
2. Policies, laws and ules pa ies
Policy make s
S anda dised and egula ion pa ies
3. Keys one ac o s
Founde
4. Se ice p o ide s
Da a-as-a-se ice p o ide s
Analy ics-as-a-se ice-p o ide s
Da a se ice de elope
5. Reuse
Open da a-d i en o ganiza ion
Applica ion de elope
In e p e e
6. Da a in e media ies
Da a b oke s
Enable s
In eg a o s
Da a ex ac o and ans o me
Da a analyze
Da a isualize
7. Da a use /da a consume
Da a cu a o
In as uc u e p o ide
Da a sponso
Da a consul an
F om a p ac ical ope a ion pe spec i e, he indi idual capabili ies and expe s in STRONG AYA a e
bi u ca ed a he Eu opean and local/na ional le el. A he Eu opean le el, expe s guide he building
and main enance o he ecosys ems, including i s echnical equi emen s (UNIMAAS, UOL), i s legal
and e hical amewo ks (EORTC) and wha da a he ecosys ems will be collec ing and analysing
(SOUTHAMPTON). A local le el, ope a ional expe s a e needed o ensu e he ecosys ems unc ion.
These include da a collec o s in he o m o esea che s, clinicians, and clinical suppo s a such as
esea ch nu ses; Local legal and e hical expe s o ensu e compliance wi h local egula ions such as
DPOs and in e nal e hics e iew boa ds; da a manage s o manage he da a lake houses and ensu e
he da a is FAIR and accu a e; and inally da a scien is s o IT echnicians o main ain he
unc ionali y o he echnical in as uc u e.
Ou cul u e should build a ne wo k be ween us, which can imp o e ou pa ien s’ cance ou comes.
This will include:
Wo king his way ou inely, and inding i use ul (e en ewa ding, o enjoyable)
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Ways o gene a e co-ope a ion and connec ions, in e -dependence and c ea i i y, which
b ing da a science, clinical ca e, clinical esea ch, epidemiology and se ice use s close
oge he
Ways o hold da a ha ensu e i is clean, in eg a es, can be packaged, analysed, lea ned
om and applied
Be e use and e-use o ou da a o gene a e in o ma ion and expe ise wi hin and ou -wi h
S ong-AYA, a he han only mo ing knowledge o wa d in a adi ional, linea manne
P o ide – o- p ocesso - o- analys – o – knowledge – o – policy; We will seek o
e e se, change o e-o de ha sequence
Ways o wo k oge he , soon and o decades o come, when using ou sui able in o ma ion,
da a and analy ical esul s app op ia ely o he da a and he use
7. Sus ainabili y – ini ial hough s
The sus ainabili y o he STRONG AYA p ojec is embedded in i s way o wo king. Unlike adi ional
clinical ials, STRONG AYA is undamen ally abou he de elopmen o new p ocesses and ne wo ks
o enable scien i ic disco e y. Whils sus ainabili y ac o s o he p ojec will be explo ed in g ea e
dep h in deli e ables p oduced by WP4, i is use ul he e o ske ch ou some ini ial hough s on how
sus ainabili y is can be buil in o he STRONG AYA ecosys ems. Some a eas o conside a ion a e mo e
ma u e a his poin han o he s, and his epo will be upda ed o e lec e ol ing sus ainabili y
conce ns as he p ojec ma u es.
The i s conside a ion is he so wa e in as uc u e sus ainabili y. By pa ne ing wi h he IKNL, he
de elope o he an age6 so wa e ha ope a es he STRONG AYA echnical in as uc u e, we ha e
a di ec link o he echnical ope a ion and ma u a ion o he so wa e o e ime wi hin he
conso ium i sel . Mo eo e , he ede a ed analy ics and ede a ed lea ning codes ha will be buil
o ope a e STRONG AYA will be buil on op o he IKNL in as uc u e will be ully open sou ce code
and ully open access, gi en by UNIMAAS in a public so wa e eposi o y.
The agili y and adap a ion o e ed by he ede a ed lea ning in as uc u e and he app oach o ways
o wo king in STRONG AYA mean ha scaling STRONG AYA echnologically is s aigh o wa d and can
accoun o he di e si y o EHRs and da a p ocesses o new membe ins i u ions.
STRONG AYA is imagined o ope a e as a no o p o i business, which means sus ainabili y cos s will
be cen ed a ound expanding access o i s analy ics and new da a lake-houses and main aining he
da a manage s equi ed o ensu e he echnical in as uc u e con inues o ope a e a e he end o
he p ojec . Gi en he wide ange o s akeholde s, his could in ol e a subsc ip ion se ice o
esea ch cen es, egula o y bodies, NGOs, go e nmen s o policy make s who use STRONG AYA
insigh s egula ly, o paymen by analysis o by iew o mo e bespoke o ad-hoc use o he STRONG
AYA analy ics.
A g ea e challenge will be nego ia ing STRONG AYA’s sus ainabili y om a go e nance pe spec i e;
bo h wi h onboa ding new pa icipan s and o go e ning he p ojec a he end o he unding
pe iod. While i is oo ea ly ye o conside he o ganiza ional go e nance s uc u e a e unding, he
EORTC’s expansi e Eu opean knowledge and es ablishmen o bes p ac ices om he onse o he
p ojec , coupled wi h he success ul connec ion o STRONG AYA’s al eady di e se conso ium will
p o ide pe suasi e use-cases ha will, i is hoped, assuage da a secu i y conce ns and con ibu e
posi i ely o he Eu opean Digi al Heal h Space agenda. Fu he mo e, o he membe s o he STRONG
AYA conso ium a e in ol ed in o he Eu opean p ojec s ackling simila conce ns, such as he JANE
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p ojec led by he Ins i u o Nazionale dei Tumo i, o e ing STRONG AYA expe ise and he
oppo uni y o syne gise wi h o he Eu opean p ojec s.
8. Conclusion/ nex s eps
This documen summa ises key componen s o he ecosys em business a chi ec u e o STRONG
AYA. The de elopmen o he ecosys ems’ in as uc u e is ongoing and as such, his documen
ep esen s he alignmen achie ed hus a on he go e nance and echnical aspec s o he
ecosys em and he conso ium’s p og ess o da e. I should he e o e be ea ed as a ‘li ing
documen ’, cap u ing he wo k done o he poin o submission and concep s ag eed upon amongs
he conso ium hus a . I is an icipa ed ha hese s uc u es will e ine and e ol e o e he cou se
o he p ojec .
The STRONG AYA ecosys em business a chi ec u e ou lines he ini ial design o he na ional and pan-
Eu opean ecosys ems and he p inciples ha unde pin hem. I complemen s he echnical
in as uc u e o he STRONG AYA ne wo k by se ing ou he ecosys em’s o ganisa ional s uc u e.
The documen summa ises he mission and objec i es o he p ojec and go e nance p inciples; he
legal s uc u e and conside a ions equi ed o implemen he ecosys ems, an ab idgemen o he
da a managemen and echnical bluep in , he o ganisa ional s uc u e o he ecosys ems and b ie ly
ouches upon a eas o u he conside a ion o he sus ainabili y o he ecosys ems beyond he
unding pe iod (sus ainabili y planning is pa o o he deli e ables o he p ojec ).
I is supplemen a y o o he deli e ables o he p ojec . The managemen o da a is mo e
exhaus i ely de ined in he i s Da a Managemen Plan (D3.1, al eady submi ed a ime o w i ing in
M6) wi h a inal e sion due a he end o he p ojec . The echnical equi emen s a e discussed in
u he dep h in he e ol ing echnical blue p in (D3.3 – due M12). The go e nance and e hics
amewo k is being de eloped in D2.2 (expec ed M12). The ope a ional plans o he ecosys ems,
bo h local/na ional and pan-Eu opean, a e deli e ables 4.4, 4.6, 4.7 (due M9 and M12). Sus ainabili y
planning is he ocus o D4.12 (M36), and D4.15 (54). will con inue h oughou he li e o he p ojec .
