Models for estimating and projecting global, regional and national prevalence and disease burden of asthma: protocol for a systematic review
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Bhuia MR, e al. BMJ Open 2017;7:e015441. doi:10.1136/bmjopen-2016-015441
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ABSTRACT
In oduc ion Models ha ha e so a been used o
es ima e and p ojec he p e alence and disease bu den
o as hma a e in mos cases inadequa ely desc ibed and
i ep oducible. We aim sys ema ically o desc ibe and
c i ique he exis ing models in ela ion o hei s eng hs,
limi a ions and ep oducibili y, and o de e mine he
app op ia e models o es ima ing and p ojec ing he
p e alence and disease bu den o as hma.
Me hods We will sea ch he ollowing elec onic
da abases o iden i y ele an li e a u e published om
1980 o 2017: Medline, Embase, WHO Lib a y and
In o ma ion Se ices and Web o Science Co e Collec ion.
We will iden i y addi ional s udies by sea ching he
e e ence lis o all he e ie ed pape s and con ac ing
expe s. We will include obse a ional s udies ha used
models o es ima ing and/o p ojec ing p e alence and
disease bu den o as hma ega ding human popula ion
o any age and sex. Two independen e iewe s will
assess he s udies o inclusion and ex ac da a om
included pape s. Da a i ems will include au ho s’ names,
publica ion yea , s udy aims, da a sou ce and ime pe iod,
s udy popula ion, as hma ou comes, s udy me hodology,
model ype, model se ings, s udy a iables, me hods o
model de i a ion, me hods o pa ame e es ima ion and/
o p ojec ion, model i in o ma ion, key indings and
iden i ied esea ch gaps. A de ailed c i ical na a i e
syn hesis o he models will be unde aken in ela ion o
hei s eng hs, limi a ions and ep oducibili y. A quali y
assessmen checklis and sco ing amewo k will be used
o de e mine he app op ia e models o es ima ing and
p ojec ing he p e alence anddiseasebu den o as hma.
E hics and dissemina ion We will no collec any p ima y
da a o his e iew, and hence he e is no need o o mal
Na ional Heal h Se ices Resea ch E hics Commi ee
app o al. We will p esen ou indings a scien i ic
con e ences and publish he indings in he pee - e iewed
scien i ic jou nal.
BACkgRound
As hma is now one o he mos common
long- e m condi ions in he wo ld, and i is
esponsible o subs an ial mo bidi y and in
some cases mo ali y.1 The o e all wo ldwide
end in he p e alence o as hma appea s o
ha e pla eaued in some pa s o he wo ld, while
i is s ill inc easing in some coun ies.2 As hma
has been anked as he 14 h mos impo an
cause o yea s li ed wi h disabili y (YLDs) in he
wo ld,3 and i accoun s o 1% o all disabili-
y-adjus ed li e yea s (DALYs) los globally.4
The socie al and heal hca e cos s
a ibu ed o as hma a e also high ac oss
di e en wo ld egions: o ins ance, ac oss
Eu ope, he cos o pe sis en as hma among
hose aged 15–64 yea s was es ima ed in 2010
alues a abou €19.3 billion5; in Asia-Paci ic
egion, he o al annual pe -pa ien soci-
e al cos s o as hma a ied om US$184
in Vie nam o US$1189 in Hong Kong
(2000 a es).6 Likewise, a na ional le els,
as hma imposes conside able economic
bu den o he heal hca e sys em besides
i s nega i e impac on he quali y o li e o
indi iduals and amilies.7 8 Fo example,
ecen es ima es ound ha as hma cos s
a leas £1.1 billion pe yea o UK and i s
membe na ions,9 while in he USA he o al
cos a ibu ed o as hma in 2007 was es i-
ma ed a abou US$56 billion.10
Al hough a ying es ima es o as hma p e -
alence and bu den a he na ional, egional
and global le els ha e been epo ed in
Models o es ima ing and p ojec ing
global, egional and na ional p e alence
and disease bu den o as hma: p o ocol
o a sys ema ic e iew
Mohammad Romel Bhuia,1 B igh I Nwa u,1,2 Ch is ophe J Wei ,3 Aziz Sheikh1
To ci e: BhuiaMR, Nwa uBI,
Wei CJ, e al. Models o
es ima ing and p ojec ing
global, egional and na ional
p e alence and disease bu den
o as hma: p o ocol o a
sys ema ic e iew. BMJ Open
2017;7:e015441. doi:10.1136/
bmjopen-2016-015441
►P epublica ion his o y
and addi ional ma e ial a e
a ailable. To iew hese iles
please isi he jou nal online
(h p:// dx. doi. o g/ 10.1136/
bmjopen-2016-015441).
Recei ed 4 Decembe 2016
Re ised 31 Janua y 2017
Accep ed 4 Ap il 2017
1As hma UK Cen e o Applied
Resea ch, Cen e o Medical
In o ma ics, Ushe Ins i u e o
Popula ion Heal h Sciences and
In o ma ics, The Uni e si y o
Edinbu gh, Edinbu gh, UK
2School o Heal h Sciences,
Uni e si y o Tampe e, Tampe e,
Finland
3Edinbu gh Clinical T ials Uni ,
Ushe Ins i u e o Popula ion
Heal h Sciences and In o ma ics,
The Uni e si y o Edinbu gh,
Edinbu gh, UK
Co espondence o
M . Mohammad Romel Bhuia;
Mohammad- Romel. Bhuia@ ed.
ac. uk
P o ocol
S eng hs and limi a ions o his s udy
►To he bes o ou knowledge, his is he i s
sys ema ic e iew o syn hesise and c i ique
exis ing models o es ima ing and/o p ojec ing he
p e alence and diseasebu den o as hma o scope
he ele an e idence base.
►The e is no geog aphical and language limi a ions.
►Comp ehensi e and highly sensi i e sea ch
s a egies, iden i ica ion o s udies om leading
medical and public heal h da abases, and
in ol emen o a panel o expe will ensu e quali y
o unde lying e idence base.
►Panel o expe s should be consul ed due o lack o
s anda d guidelines o e alua ing models.
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2Bhuia MR, e al. BMJ Open 2017;7:e015441. doi:10.1136/bmjopen-2016-015441
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he published li e a u e,3 11–16 almos all appea o ha e
majo limi a ions in e ms o inadequacy o he analy ical
app oach used and lack o ep oducibili y.17–19 The e is
he e o e a need o gene a ing alid and ep oducible
es ima es o disease p e alence and bu den o as hma o
in o m e idence-based policy delibe a ions. De eloping
anspa en p ocesses o gene a ing he na ional, egional
and global es ima es o p e alence and disease bu den o
as hma will hus enhance ep oducibili y ac oss se ings
and will allow eliable p ojec ion o u u e es ima es.
In heal hca e policy, a model can be de ined as a logical
ma hema ical amewo k o analy ical me hodology ha
in eg a es heo ies and da a o d aw in e ences ega ding
pa ame e s o in e es o clinicians and decision make s.20 21
Models a e widely used o es ima e disease bu den,9 22–24
end in p e alence25 26 and u u e p ojec ions27 o di e en
epidemiological cha ac e is ics o as hma. Al hough
cu en p e alence o as hma can be es ima ed wi hou
applying a model, many s udies15 17 28–31 es ima ed as hma
p e alence applying modelling echniques, pa icula ly
wi h he aim o adjus ing o ce ain popula ion cha -
ac e is ics, such as age, sex, ime, geog aphy and o he
con ex ual pa ame e s, which may a y ac oss s udies. Fo
example, he In e na ional S udy o As hma and Alle gies
in Childhood S ee ing Commi ee applied gene alised
linea mixed model o es ima e he global p e alence o
as hma in o de o adjus o wi hin-coun y and be ween-
coun y a ia ions.15 Adeloye e al17 applied a non-linea
model o es ima e egional (A ica) p e alence o as hma.
The Global Bu den o Disease s udies also de eloped some
compu e based disease modelling ools (such as DisMod,
DisMod-MR) o es ima e he p e alence and bu den o
a ious diseases.28 32 Howe e , exis ing models o es i-
ma ing and p ojec ing p e alence and disease bu den o
as hma a e in mos cases poo ly desc ibed, he eby limi ing
he oppo uni y o assess hei ep oducibili y. In o de o
gain a be e app ecia ion o he pe o mance o exis ing
models and hei capaci y o ep oducibili y in es ima ing
he bu den o as hma, a sys ema ic app aisal o he unde -
lying e idence base is equi ed.33
objec i es
The aims o his sys ema ic e iew a e o (1) sys ema ically
desc ibe and c i ique he exis ing models o es ima ing
and/o p ojec ing he global, egional and na ional p e -
alence and disease bu den o as hma in ela ion o hei
s eng hs, limi a ions and ep oducibili y, and (2) de e -
mine he app op ia e models o es ima ing and p ojec ing
he p e alence and disease bu den o as hma.
MeThodS
eligibili y c i e ia
Types o s udies
Any s udy ha de eloped models o es ima ing and/o
p ojec ing p e alence and disease bu den o as hma will be
included in his e iew. S udies ha es ima ed p e alence
and disease bu den wi hou modelling will be excluded.
Models ha es ima ed indi idual isk a he han popula ion
bene i s, such as decision analy ic models and indi idual
p ognos ic models, will be excluded. Mo eo e , s udies wi h
models ha simply desc ibe animals, clinical se ies and cell
lines will be excluded. Compa a i e in e en ion s udies
will also be excluded. Po en ial sou ces o e idence such
as o iginal esea ch a icles and e iew a icles, including
sys ema ic e iews, me a-analyses and me a-syn heses o
obse a ional s udies, will be included.
Pa icipan s
Eligible pa icipan s in his e iew will include human
popula ions o any age and ei he sex.
Yea s conside ed
We will include s udies om Janua y 1980 o Ap il 2017.
The s a da e has been se up om he ime when model-
ling echniques s a ed o be applied b oadly o s udy he
epidemic o non-communicable diseases.34
Se ing
Resea ch a icles om any coun y and any se ing
(u ban/ u al) will be included in his e iew.
Language
The e will be no language es ic ions and, whe e possible,
we will ansla e he li e a u e published in languages
o he han English.
In o ma ion sou ces
Da abase sea ches and o he sou ces o iden i y s udies
We will conduc sea ches o iden i y bo h published and
unpublished modelling s udies in he ollowing elec-
onic da abases: Medline, Embase, WHO Lib a y and
In o ma ion Se ices (lib a y ca alogue o books and
epo s) and Web o Science Co e Collec ion. The e e -
ence lis s o all he included pape s will be sea ched o
addi ional s udies. We will also con ac a panel o expe s
in an a emp o iden i y addi ional unpublished o in
p og ess s udies.
Sea ch s a egy
A comp ehensi e li e a u e sea ch will be unde aken o
iden i y bo h published and unpublished (g ey li e a u e)
p ima y s udies as well as e iews. The sea ch s a egy has
been de eloped o sea ching li e a u e in Medline and
Embase (see online supplemen a y appendix) in consul-
a ion wi h a senio medical lib a ian a The Uni e si y
o Edinbu gh, and his will be adap ed in sea ching
o he da abases. The sea ch e ms include he concep s
o ‘modelling’, ‘p e alence and disease bu den’ and
‘as hma’.
S udy eco ds
Da a managemen
The e ie ed eco ds om all da abases will be expo ed
o EndNo e Lib a y, which will be used h oughou he
e iew o s udy sc eening, deduplica ion and o e all
managemen o he e ie ed eco ds.
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Bhuia MR, e al. BMJ Open 2017;7:e015441. doi:10.1136/bmjopen-2016-015441
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Selec ion p ocess
Two e iewe s will independen ly check and sc een
he i les and abs ac s o iden i ied a icles agains he
inclusion c i e ia. Full- ex copies o po en ially ele an
s udies will be ob ained and assessed by wo independen
e iewe s on he basis o hei eligibili y o inclusion. Any
disc epancies will be esol ed by discussion, and disag ee-
men s will be a bi a ed by a hi d e iewe .
Da a ex ac ion
A da a ex ac ion o m will be used o ex ac ele an
da a om included s udies. We ha e de eloped a d a
da a ex ac ion o m. Du ing he e iew p ocess, his
d a will be e ined and he da a ex ac ion o m will be
upda ed acco dingly. The da a ex ac ion o m will be
p e-pilo ed p io o ull use in he e iew. Da a ex ac ion
will be pe o med independen ly by wo e iewe s.
da a i ems
In o ma ion ega ding di e en componen s o he
models will be eco ded o ge a comp ehensi e pic u e
o he models. The ollowing da a i ems will be ex ac ed
om each s udy: au ho s’ names; publica ion yea ; s udy
aims; da a sou ce and ime pe iod; s udy popula ion;
as hma ou comes (p e alence/disease bu den); s udy
me hodology; model ype; model se ings; model o mu-
la ion (s uc u e, speci ica ion, assump ions, me hods o
model de i a ion, me hods o pa ame e es ima ion and/
o p ojec ion, heo e ical basis o he models) s udy a i-
ables; a ailabili y o da a and codes; indings om he
models; model i in o ma ion; key indings o he s udy;
and iden i ied esea ch gaps. In o ma ion ega ding
he model a ailabili y, anspa ency, sensi i i y analysis,
model alida ion, add essing missing da a, policymake s’
in ol emen , dissemina ion and expe in ol emen ,
limi a ion discussed and ep oducibili y o he model will
also be ex ac ed. Desc ip i e ables will be used o abu-
la e hese i ems. The sys ema ic e iew will be epo ed
ollowing he guidelines o he P e e ed Repo ing I ems
o Sys ema ic e iew and Me a Analysis (PRISMA) check-
lis (see online supplemen a y ma e ial).35
ou comes and p io i isa ion
The ou comes ha a e o in e es include p e alence and
disease bu den o as hma. The e a e a ious measu es
a ailable o quan i y disease bu den. All he es ablished
measu es o disease bu den will be conside ed in his
e iew. P ima y and seconda y ou comes a e ca ego ised
as ollows.
P ima y ou comes
1. P e alence o as hma.
2. Di e en measu es o disease bu den o as hma.
The measu es a e: DALYs, YLDs, mo ali y,
heal hca e cos (cos o illness, d ug cos , hospi al
cos /hospi alisa ion cos ), li e expec ancy, p ima y
ca e, ambula o y ca e, eme gency isi , absen ees,
yea s li e los , po en ial yea s o li e los , heal hy
yea s o li e los , ac i e li e expec ancy, disabili y- ee
li e expec ancy, disabili y-adjus ed li e expec ancy,
heal hy li e expec ancy, and so on.
Seconda y ou comes
1. Incidence o as hma.
Risk o bias in indi idual s udies
To he bes o ou knowledge, he e is no exis ing quali y
app aisal ool o assess quali y o models. So we ha e
d awn on i s p inciples and adap ed ele an sec ions
om pe inen epo ing guidelines36 and o he guide-
lines o good p ac ice in modelling s udies20 21 37 38 o
de elop ou own model e alua ion amewo k. This will
in ol e independen assessmen o he s eng hs and
limi a ions o he models on he basis o model s uc u e,
speci ica ion, assump ions, sensi i i y analysis, model
alida ion, dealing wi h missing da a, heo e ical basis o
he models, inco po a ion o con ounding ac o s and
lag imes, and whe he po en ial me hodological limi a-
ions a e desc ibed. Rep oducibili y o he model will be
assessed on he basis o a ailabili y o he models, da a,
codes and me hods o pa ame e es ima ion.
To e alua e he models used in included s udies and
o iden i y he bes models, we ha e p epa ed a check-
lis o i ems and o mula ed a sco ing s a egy (see
online supplemen a y appendix) ha we will use o
hese pu poses. P io o use o he checklis , we plan o
consul wi h a panel o expe s in he ield o modelling
s udies o gain hei insigh s and c i icisms o he check-
lis ; we will hen in eg a e eedback colla ed in p epa ing
he inal e sion o he checklis o be used in ou s udy.
da a syn hesis
A abula summa y o he da a will be p esen ed o
summa ise o e all e idence. A de ailed c i ical na a i e
syn hesis o he models will be unde aken ega ding
hei s eng hs, limi a ions and ep oducibili y.
PRoToCol RegISTRATIon
A de ailed p o ocol o he sys ema ic e iew will be
egis e ed wi h he In e na ional P ospec i e Regis e
o Sys ema ic Re iews p io o commencing he e iew
acco ding o he PRISMA-P o ocols 2015 s a emen .39
ConCluSIonS
To he bes o ou knowledge no e iew has been unde -
aken ye o app aise he models o es ima ing and
p ojec ing he global, egional and na ional p e alence
and disease bu den o as hma. This sys ema ic e iew is
he e o e he i s s udy o syn hesise exis ing models o
es ima ing and p ojec ing p e alence and disease bu den
o as hma. The e iew will also map he app op ia e
models ha will subsequen ly be used o ob ain cu en
es ima es and p ojec u u e end o global, egional and
na ional p e alence and disease bu den o as hma.
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Open Access
eThICS And dISSeMInATIon
We will no collec any p ima y da a o his e iew, and
hence he e is no need o o mal Na ional Heal h Se ice
Resea ch E hics Commi ee e iew. This wo k is howe e
subjec o Ins i u ional Re iew Boa d o e sigh by The
Uni e si y o Edinbu gh’s Cen e o Popula ion Heal h
Sciences. Findings om he e iew will be p esen ed a
scien i ic con e ences and be published in he pee - e-
iewed scien i ic jou nal.
Acknowledgemen s We a e hank ul o Ma shall Dozie , Senio Liaison Lib a ian
o he College o Medicine and Ve e ina y Medicine, The Uni e si y o Edinbu gh,
o he suppo in de eloping he sea ch s a egies. We a ealso hank ul o D
Susannah McLean o p o iding necessa y sugges ions and esou ces ega ding
modelling s udies, D Niall Ande son o his sugges ions in de eloping sco ing
amewo k o app aise he models, And ew S odda o sugges ing sea ch e ms
ela ed o economic bu den o as hma, and he As hma UK Cen e o Applied
Resea ch o p o iding pla o m o de elop his p o ocol.
Con ibu o s AS concei ed he idea o his wo k. MRB d a ed he p o ocol unde
he supe ision o AS, BIN and CJW. The d a was e ised acco ding o se e al
ounds o c i ical commen s om AS, BIN and CJW. All he au ho s will be in ol ed
in he sys ema ic e iew p ocess.
Funding MRB is suppo ed by he College o Medicine & Ve e ina y Medicine, The
Uni e si y o Edinbu gh, and he Fa Ins i u e, UK. BIN and AS a e also suppo ed by
he Fa Ins i u e, UK. The Fa Ins i u e is unded by a conso ium o unde s led by
he Medical Resea ch Council (MRC).
Compe ing in e es s None decla ed.
P o enance and pee e iew No commissioned; ex e nally pee e iewed.
Open Access This is an Open Access a icle dis ibu ed in acco dance wi h he
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o he s o dis ibu e, emix, adap and build upon his wo k, o comme cial use,
p o ided he o iginal wo k is p ope ly ci ed. See: h p:// c ea i ecommons. o g/
licenses/ by/ 4. 0/
© A icle au ho (s) (o hei employe (s) unless o he wise s a ed in he ex o he
a icle) 2017. All igh s ese ed. No comme cial use is pe mi ed unless o he wise
exp essly g an ed.
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Open Access
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e iew
bu den o as hma: p o ocol o a sys ema ic
disease egional and na ional p e alence and
Models o es ima ing and p ojec ing global,
Sheikh
Mohammad Romel Bhuia, B igh I Nwa u, Ch is ophe J Wei and Aziz
doi: 10.1136/bmjopen-2016-015441
2017 7: BMJ Open
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