Ci a ion: Rod íguez, A.A.; Amay a,
I.; López-Paz, J.F.; Ma ínez, O.;
Ga cía, M.; Salguei o, M.;
Al-Rashaida, M.; Luna, P.M.;
Pé ez-Nuñez, P.; Passi, N.; e al. The
Role o Associa ions in Reducing he
Emo ional and Financial Impac on
Pa en s Ca ing o Child en wi h
Duchenne Muscula Dys ophy: A
C oss-Cul u al S udy. In . J. En i on.
Res. Public Heal h 2022,19, 12334.
h ps://doi.o g/10.3390/ije ph191912334
Academic Edi o : Paul B. Tchounwou
Recei ed: 6 Sep embe 2022
Accep ed: 24 Sep embe 2022
Published: 28 Sep embe 2022
Publishe ’s No e: MDPI s ays neu al
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Licensee MDPI, Basel, Swi ze land.
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A ibu ion (CC BY) license (h ps://
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In e na ional Jou nal o
En i onmen al Resea ch
and Public Heal h
A icle
The Role o Associa ions in Reducing he Emo ional and
Financial Impac on Pa en s Ca ing o Child en wi h
Duchenne Muscula Dys ophy: A C oss-Cul u al S udy
Alicia Au o a Rod íguez 1,* , Imanol Amay a 1, Juan F ancisco López-Paz 1, Osca Ma ínez 1, Mai ane Ga cía1,
Mónika Salguei o 2, Mohammad Al-Rashaida 3, Paula Ma ía Luna 1, Paula Pé ez-Nuñez 1, Nicole Passi 1,
I une Ga cía1and Ja ie a O ega 4
1Neu o-e-Mo ion Resea ch Team, Facul y o Heal h Sciences, Uni e si y o Deus o, A . Uni e sidades, 24,
48007 Bilbao, Spain
2Depa men o Clinical and Heal h Psychology, and Resea ch Me hodology, Facul y o Psychology,
Uni e si y o he Basque Coun y, Tolosa Hi ibidea, 70, 20018 Donos ia, Spain
3Depa men o Educa ion, College o A s and Sciences, Dubai Campus, Abu Dhabi Uni e si y,
Abu Dhabi 59911, Uni ed A ab Emi a es
4Cen o In es igaciones de Psicología y Psicopedagogía [CIPP], Facul ad de Psicología y Psicopedagogía,
Pon i icia Uni e sidad Ca ólica A gen ina, Buenos Ai es 1107, A gen ina
*Co espondence: alicia [email p o ec ed]; Tel.: +34-944-139-000 (ex . 2577)
Abs ac :
Ca egi e s’ emo ions and inances a e a ec ed by he de e io a ion o unc ional capaci y o
pa ien s wi h Duchenne muscula dys ophy (DMD), bo h in Mexico and Spain. Pa ien associa ions
may educe his impac on ca egi e s. This s udy aims o s udy he ole o wo models o associa ions,
inspi ed by wo di e en cul u al models, in how he se ices hey p o ide can help dec ease he
emo ional and inancial impac on he ca egi e s o child en wi h DMD. The sample consis ed o
34 ca egi e s om Mexico and 40 om Spain ec ui ed om Spanish hospi als and a e disease
o ganiza ions in Spain and Mexico. The ins umen s used consis ed o a sociodemog aphic and
socioeconomic ques ionnai e, he Ca e Qol-7D, he PHQ-15, he Za i Ca egi e ’s Bu den Scale and
he SWLS. The esul s showed ha ca egi e s in Mexico a e in be e physical and psychological
heal h han ca egi e s in Spain. They also ecei e mo e subsidies han hose in Spain. Ca egi e s in
Mexico ha e a g ea e well-being and a e less a ec ed by he economic impac o he disease due
o he associa ions’ day- o-day wo k and he ac ha hey gene a e a ne wo k o heal h se ices
ha hey make a ailable o he pa ien ee o cha ge. These di e ences may also be a ibu able o
cul u al issues and o he ac ha Mexico has a deeply es ablished cul u e o suppo .
Keywo ds:
Duchenne muscula dys ophy; ca egi e s; economic cos s; psychological heal h; quali y
o li e; non-go e nmen al o ganiza ions; Mexico; Spain
1. In oduc ion
O e he yea s, he in e es in and awa eness o a e diseases (RDs) ha e g own in
bo h he s a e and he socie y. RDs a e ch onic, low-p e alence condi ions, cha ac e ized
by diagnos ic delay, unce ain clinical cou se and lack o cu a i e ea men s [
1
]. Some
c oss-cul u al s udies ha e a emp ed o analyze he economic and emo ional impac o
a ch onic illness on pa ien s’ li es [
2
,
3
] and on he ca egi e s o child en wi h RDs [
4
,
5
]
and how he social model o each coun y can in luence his impac . Howe e , o ou
knowledge, none o hese s udies conside ed he di e en cul u al bene i s p o ided by
associa ions om wo di e en coun ies o RDs pa ien s’ ca egi e s. The s uc u e and
aims o he se ices p o ided by he associa ions a e de e mined by public heal h policies.
When compa ing he public policies in La in Ame ica and Eu ope, public policies
ela ed o RDs we e de eloped a a ela i ely la e s age in La in Ame ica [
1
]. As a esul , RD
pa ien s a e only pa ially ca ed o . In policy e ms, hese diseases ha e been associa ed
In . J. En i on. Res. Public Heal h 2022,19, 12334. h ps://doi.o g/10.3390/ije ph191912334 h ps://www.mdpi.com/jou nal/ije ph
In . J. En i on. Res. Public Heal h 2022,19, 12334 2 o 15
wi h ca as ophic diseases, high-cos diseases, ch onic non-communicable diseases and he
disabled popula ion. Howe e , RDs ha e no been speci ically add essed by he au ho i ies.
Six coun ies, including Mexico, ha e in oduced legisla ion on RDs in La in Ame ica [
6
].
None heless, he na ional plans, p og ams and s a egies ha deal wi h he needs o his
g oup a e missing [
7
]. Consequen ly, pa ien associa ions a e he ones ha ha e aken
he esponsibili y o look ou o he RDs popula ion’s well-being [
6
]. Fu he mo e, RDs
ha e become mo e isible and ga he ed mo e in e es hanks o pa ien ede a ions and
o ganiza ions [6].
The de elopmen o policies aimed a sa egua ding he igh s o RDs in he EU has
ollowed a di e en cou se, s a ing ea lie and consolida ing be ween he yea s 2000
and 2004 [
8
]. In Spain, a na ional s a egy was p esen ed back in 2009 in o de o adap
heal hca e ac ions o he needs o pa ien s and o be e moni o hei implemen a ion [
9
].
The moni o ing showed ha heal hca e and social sys ems in Spain do no adequa ely mee
he needs o RD pa ien s, which is simila o he si ua ion in Mexico [10,11].
Due o he lack o public unds, he e is a dec ease in he income o pa ien s and hei
amilies [
12
–
14
]. I is es ima ed ha hey spend a la ge pa o hei income on medicines,
a el, home adap a ions, complemen a y he apies and unique ood p oduc s [
15
]. This
can lead o p oblems in he amily budge , which is u he agg a a ed i any o he amily
membe s ha e o educe hei wo king hou s o qui hei jobs due o illness [16].
In Mexico, non-go e nmen al o ganiza ions p o ide comp ehensi e and mul idisci-
plina y ca e suppo o RD pa ien s and hei amilies, he eby educing his economic
imbalance [
17
]. They o e guidance, dissemina e in o ma ion, aise awa eness, sensi ize
he popula ion and p o ide social educa ion abou RDs [18].
The si ua ion is simila in Spain [
19
]. Suppo o ganiza ions o people wi h RDs a e
essen ial o hei isibili y and a e conside ed a public heal h p io i y in Eu ope [
20
]. In
addi ion, EUROPLAN [
21
] leads ac ions o p omo e esea ch in his a ea and encou age
collec ing da a on hese RDs. The pu pose is o assess he ac ual si ua ion in Spain and
o he coun ies ega ding hese condi ions.
Suppo o ganiza ions o people wi h RDs led o he es ablishmen o a ne wo k,
which, among o he goals, aims o sp ead knowledge among he public o imp o e he
cu en unde s anding o hese diseases. They ha e also played a key ole in imp o ing
ca e and esea ch on RDs, as hey p oposed a join s a egy and a se ies o ac ions o ensu e
ha he needs o pa ien s and hei amilies we e me [22].
Some associa ions in Spain and Mexico gi e isibili y and suppo o a g oup o a e
diseases called neu omuscula diso de s (NMDs). One o hese is Duchenne muscula
dys ophy (DMD), an X-linked ecessi e he edi a y diso de , whe e p og essi e muscle
degene a ion leads o he loss o he abili y o walk independen ly by he age o 13 [
23
–
26
].
The p e alence o DMD is less han 10 cases pe 100,000 males and seems o be he same
be ween egions, and DMD in emales is e y a e (<1 pe million) and is limi ed o case
epo s o indi iduals wi h he Tu ne synd ome, a ansloca ion in ol ing DMD o hose
wi h bi-allelic DMD mu a ions [27].
DMD en ails a high deg ee o disabili y and dependency. Pa ien s wi h DMD need
cons an ca e and supe ision by in o mal ca egi e s–pa en s o o he i s -deg ee ela i es.
Gi en ha his ype o ca e equi es speci ic expe ise, akes a long ime and is associa ed
wi h addi ional inancial cos s, i s impac on he ca egi e ’s li e is no iceable [28].
Many ca egi e s o child en wi h DMD qui hei jobs o educe hei wo king hou s
o ca e o hem. Fo hose who con inue o wo k, he p oduc i i y declines due o equen
absences om wo k. In addi ion o he economic impac esul ing om job loss, he
a ec ed amilies bea subs an ial cos s associa ed wi h insu ance p emiums and medical
ca e co-paymen s [29,30].
Addi ionally, he ca egi e s’ emo ions, amily li e and social li e a e a ec ed due o he
de e io a ion o pa ien s’ unc ional capaci y [
31
]. The child’s disabili y causes changes in
he ca egi e ’s daily li e ac i i ies. Ca ing o a pe son wi h DMD is also e y s ess ul and
can impac he ca egi e ’s physical and psychological well-being [
30
]. S udies ha e shown
In . J. En i on. Res. Public Heal h 2022,19, 12334 3 o 15
ha ca egi e s su e om high demands, s ess, o e load, dis ess and lowe quali y o
li e (QoL) [32].
The ca egi e s o people wi h hese pa hologies su e om a signi ican bu den,
causing an impedimen o hei social li e [
33
]. Fu he mo e, ca ing o a dependen pe son
no only a ec s psychological well-being bu can also educe he ca egi e ’s physical
heal h [
34
]. In addi ion, hei pe cei ed sa is ac ion wi h li e, and hus, hei subjec i e
well-being, is also wo se han ha o pa en s o child en wi hou any illness [35].
Due o he impac o DMD on he ca egi e s’ li e, he associa ions wo k o help ca e-
gi e s o e come he challenges hey ace [
36
]. Howe e , he e a e no p e ious s udies ha
analyze he posi i e impac o associa ions on he li es o pa en s wi h
child en wi h DMD.
I is pa icula ly wo h analyzing wo models o heal h ca e o be e unde s and how
associa ions play an impo an ole. In Spain, heal h ca e consis s o public heal h ca e, so
pa ien s and ca egi e s do no ha e o bea hese cos s, and he associa ions ake ca e o
o he aspec s, such as social suppo . Howe e , his public heal hca e is no simul aneous
ca e, i.e., pa en s a e gene ally no scheduled o all heal h examina ions on he same day
and o en ha e o ake o e he cos s o p i a e specialis s because hey a e no sa is ied wi h
he ca e ecei ed. Con a iwise, in Mexico, al hough heal hca e is p i a e, he associa ions
co e he cos o ca e and p o ide he pa ien wi h a eam o heal h p o essionals who all
see he pa ien on he same day. The e o e, ca e is comp ehensi e. These c oss-cul u al
di e ences a e no only ela ed o he ype o ca e bu also o o he psychosocial a iables.
The ype o cul u e, adi ion o social s uc u e is also e lec ed in pa ien o ganiza ions [
37
].
So a , no s udies ha e analyzed collec i ely and compa a i ely he impac o di -
e en models o associa ions in e ms o he se ices p o ided on ca egi e s’ well-being.
Rega ding he las poin , he e ha e also been no s udies speci ically ocused on he ole
o associa ions in di e en coun ies in educing he impac o he disease on ca egi e
well-being in e ms o inancial cos s, QoL, li e sa is ac ion, bu den and soma ic symp oms.
The o iginali y o he p esen s udy esides in he analysis o wo di e en models o associ-
a ions, he di e ences be ween which a e based on cul u e and he heal h ca e sys em.
Because o all he abo e, his s udy aims o e alua e he ole o wo models o associa-
ions, inspi ed by wo di e en cul u al models, in educing he emo ional and inancial
impac on he ca egi e s o child en wi h DMD. This s udy is he i s o ou knowledge
o conduc a comp ehensi e analysis o he in e ac ion be ween he men ioned a iables.
Speci ically, his s udy desc ibes he inancial cos s a ising om illness, bu den, li e sa -
is ac ion, QoL and soma ic symp oma ology in ca egi e s o child en wi h DMD a a
c oss-cul u al le el in Mexico and Spain.
2. Ma e ials and Me hods
2.1. Pa icipan s
The sample consis ed o 74 ca egi e s o child en wi h DMD om Spain and Mexico.
Fo y ca egi e s om Spain we e ec ui ed om a ious DMD o ganiza ions (ASEM and
BENE), om he Hospi al de Basu o and he Hospi al Uni e si a io de C uces. Thi y- ou
ca egi e s om Mexico we e ec ui ed om he Coalición La inoame icana de Duchenne-
Becke . All pa icipan s we e membe s o DMD o ganiza ions.
The inclusion c i e ia we e he ollowing: (a) To be an in o mal ca egi e (pa en )
o a child diagnosed wi h DMD; (b) be o e 18 yea s o age; (c) o be willing o sign he
in o med consen documen be o e pa icipa ing in he s udy; and (d) o be a esiden in
Spain o Mexico and ha e Spanish as one o he p ima y languages o communica ion.
Exclusion c i e ia: (a) in o mal ca egi e o a child wi h any o he diagnosis no
seconda y o he diagnosis o DMD; (b) any o he psychological o psychia ic diagnosis
no seconda y o DMD; (c) uncompensa ed senso y de ici s ha p e en he adminis a ion
o he e alua ion p o ocol; and (d) illi e acy.
In . J. En i on. Res. Public Heal h 2022,19, 12334 4 o 15
2.2. Ins umen s
2.2.1. Measu es
Sociodemog aphic Da a
The i s ins umen was a 17-i em ad hoc ques ionnai e, which collec ed he pa ic-
ipan s’ sociodemog aphic da a (e.g., sex, age, academic le el, ype o employmen and
ma i al s a us).
Economic Da a
A 248-i em semi-s uc u ed ques ionnai e was used o assess he inancial cos s as-
socia ed wi h ca e. The ques ionnai e includes ques ions on assis i e echnology ha he
amily had o buy, he money spen on ac i i ies o imp o e he heal h o bo h he child and
he ca egi e , he ime spen engaging in hese ac i i ies and he loss o wo k p oduc i i y.
The subsidies paid by he s a e and he unding p o ided by he associa ions o co e he
expenses a e also assessed. Fo example, a numbe o ques ions comp ising his ques ion-
nai e ask abou he cos s o medical ca e, physio he apy, psychological ca e, pu chase o
wheelchai s, splin s, e c. This ques ionnai e was de eloped by Rod íguez e al. [
31
] and
was suppo ed by o he s udies conduc ed wi h people wi h NMD [38–42].
Soma ic Symp oms
The PHQ-15 [
43
] was used o assess he soma ic symp oms associa ed wi h ca e. This
ques ionnai e consis s o 15 i ems e e ing o 15 possible physical p oblems ha ca egi e s
may ha e had in he p e ious ou weeks. The o al PHQ-15 sco e anges om 0 o 30,
and sco es o >5, >10, >15 ep esen mild, mode a e and se e e le els o soma iza ion.
The possible esponse op ions o each one o he 15 i ems a e: “no bo he ed”, o absence
o a physical p oblem (0 poin s), “a li le”, o p esence o a p oblem (1 poin ), o “a lo ”,
o signi ican p esence o a p oblem (2 poin s) [
43
]. S udies, such as ha by Mon albán
e al., [
44
] ha e demons a ed high in e nal consis ency (C onbach’s alpha 0.78). The
Mexican e sion ob ained a C onbach’s Alpha o 0.77 [
45
]. The C onbach’s alpha coe icien
in his s udy was 0.88.
Ca egi e Bu den Scale
The Za i Ca egi e Bu den Scale [
46
] was used o assess he pa icipan s’ bu den
due o he ca ing expe ience. The scale measu es he wo kload o ca egi e s o dependen
people and de e mines he bu den o he ca egi e ’s expe iences as an o e all sco e. I is
a one-dimensional scale ha consis s o 22 i ems, measu ed on a ou -poin Like - ype
esponse scale. I has been shown o ha e a good in e nal consis ency in Spain (C onbach’s
alpha 0.91) [
47
] and Mexico (C onbach’s alpha 0.84) [
48
]. In he cu en s udy, C onbach’s
alpha coe icien was 0.90.
Sa is ac ion wi h Li e
The Sa is ac ion wi h Li e Scale (SWLS) is a unidimensional scale employed o assess
he ca egi e ’s sa is ac ion wi h li e [
49
]. The SWLS measu es he indi idual’s le el o
sa is ac ion wi h li e a ha momen . I consis s o i e ques ions, whe e each ques ion is
a ed on a 7-poin scale (1 = do no ag ee a all wi h he i em, 7 = s ongly ag ee wi h he
i em). The possible ange o his scale is om 1 o 7 pe ques ion. Howe e , he Spanish
e sion is a ed on a 5-poin scale. Li e sa is ac ion e e s o a subjec i e cogni i e p ocess
in which people judge hei o e all sa is ac ion wi h hei cu en si ua ion conce ning sel -
de ined s anda ds o expec a ions o wha hey would like hei li e o be [
50
]. C onbach’s
alpha was 0.89 [
51
]. In he Spanish e sion, he scale was shown o ha e high in e nal
consis ency, wi h C onbach’s alpha coe icien s anging om 0.79 o 0.89 [
52
]. The Mexican
e sion ob ained a C onbach’s alpha o 0.83 [
53
]. A C onbach’s alpha coe icien o 0.88
was ob ained in he s udy.
In . J. En i on. Res. Public Heal h 2022,19, 12334 5 o 15
Quali y o Li e
QoL was assessed using he Ca e Qol [
54
]. This ques ionnai e measu es ca e- ela ed
QoL and consis s o 7 i ems. The in aclass co ela ion coe icien s (ICCs) o he Ca e Qol7D
show alues be ween 0.55 and 0.94 [
54
]. The Ca e QoL includes wo componen s. The
i s componen is he Ca e Qol-VAS, which measu es well-being in e ms o happiness
using he isual analog scale (VAS), wi h he endpoin s being “comple ely unhappy”
and “comple ely happy”. The second componen is he Ca e QoL-7D, which measu es
subjec i e bu den h ough se en i ems ha e alua e he ollowing dimensions: ul illmen ,
ela ional p oblems, men al heal h, inancial p oblems, daily li e ac i i ies, ex e nal suppo
and physical heal h [
55
]. The u ili y a es o he Ca e Qol we e de eloped o calcula e a
Ca e Qol-7D u ili y sco e om he esponses o he se en dimensions, anging be ween 0
(“wo s imaginable ca egi ing si ua ion”) and 100 (“bes imaginable ca egi ing si ua ion”),
o which disc e e choice expe imen s we e used [
56
]. The Ca e Qol-7D had a C onbach’s
alpha o 0.641 [
57
] and a C onbach’s alpha o 0.62 in ano he s udy [
58
]. The p esen s udy
ob ained a C onbach’s alpha coe icien o 0.63.
2.3. P ocedu e
The sample was ec ui ed om di e en suppo associa ions o pa ien s wi h DMD
and wo hospi als in Bizkaia. Those in e es ed in pa icipa ing in he s udy we e in o med
abou he e alua ion p ocess. The da a we e collec ed using a sel -adminis e ed p o ocol
ia he “Qual ics” i ual pla o m, accessed h ough a pe sonal link. All ca egi e s ag eed
o pa icipa e in he s udy by gi ing consen be o e comple ing he su ey. They we e
p o ided wi h a elephone numbe o con ac he esea che i hey wished o ask any
ques ions while comple ing he su ey. The du a ion o he p o ocol was app oxima ely
one hou . The Responsible E hics Commission app o ed he esea ch (Re : ETK-39/18-19),
which was conduc ed in acco dance wi h he Decla a ion o Helsinki.
2.4. S a is ical Analysis
Desc ip i e s a is ics we e used o desc ibe he pa icipan s. The con inuous a iables
we e desc ibed by mean and s anda d de ia ion, and he ca ego ical a iables by equency
and pe cen age. Rega ding he socio-economic a iables, he mean sco es ob ained om
he s uc u ed i ems we e calcula ed. Simila ly, he mean sco es o he PHQ-15, Za i and
SWLS global sco es we e calcula ed. Fo he Ca e QoL-Ta i , he syn ax p o ided by he
au ho s o he ins umen was applied. The inancial cos s in Spain we e exp essed in EUR,
while in Mexico, hey we e desc ibed in MXN. All expenses we e hen con e ed o EUR
o acili a e compa ison. The Kolmogo o –Smi no es was used o es o no mali y
o he ou come a iables. Mos a iables did no ha e a no mal dis ibu ion because he
coe icien (K-S) was signi ican (p> 0.05). Da a we e analyzed using he Mann–Whi ney
U- es and Chi-squa e es . A p- alue below 0.05 was de ined as s a is ically signi ican .
IBM SPSS S a is ics 26.0 was used o all analyses.
3. Resul s
Fo y ca egi e s o child en wi h DMD om Spain and hi y- ou ca egi e s o
child en wi h DMD om Mexico pa icipa ed in he s udy. Table 1shows he da a ela ed
o he sampling dis ibu ion acco ding o he ca egi e ’s sex, ma i al s a us, academic
le el and employmen s a us. Rega ding he sociodemog aphic p o ile o bo h g oups o
ca egi e s, he e we e a mo e signi ican numbe o male ca egi e s in he Spanish sample
and a g ea e numbe o women in he Mexican sample (
χ2
(1) = 9.82, p= 0.02). As a as he
ma i al s a us was conce ned, ma ied people p edomina ed in he Spanish sample, while
in Mexico, no ha ing a pa ne o no being ma ied p e ailed (
χ2
(5) = 5.90, p= 0.023).
In e ms o employmen , mos Spanish ca egi e s we e employees, while he Mexican
ca egi e s we e engaged in housewo k (χ2(8) = 17.56, p= 0.025).
In . J. En i on. Res. Public Heal h 2022,19, 12334 6 o 15
Table 1. Dis ibu ion o sociodemog aphic and medical a iables.
Va iable Spain
(n= 40)
Mexico
(n= 34)
Gende Male 10 (25%) 0 (0%)
Female 30 (75%) 34 (100%)
Ma i al s a us
Ma ied 32 (80%) 17 (48.57%)
Li ing as a couple 3 (7.5%) 6 (17.64%)
Di o ced 2 (5%) 7 (20.58%)
Sepa a ed 3 (7.5%) 1 (2.94%)
Single 0 (0%) 1 (2.94%)
Widow/e 0 (0%) 2 (5.88%)
Occupa ion
Employee 16 (40%) 7 (20.58%)
Sel -employed 3 (7.5%) 2 (5.88%)
Unpaid wo k 2 (5%) 0 (0%)
Unemployed ( o heal h easons) 1 (2.5%) 1 (2.94%)
Unemployed ( o o he easons) 6 (15%) 2 (5.88%)
Re i ed 1 (2.5%) 3 (8.82%)
Housewo k 5 (12.5%) 17 (50%)
S uden 5 (12.5%) 1 (2.94%)
Disabled 1 (2.5%) 1 (2.94%)
The mean age o he Spanish ca egi e s was 46.15
±
8.14, and he mean age o he
Mexican ca egi e s was 41.18
±
10.14. The a e age yea s o educa ion o he ca egi e s
we e 14.09
±
7.02 (Spain) and 10.84
±
4.57 (Mexico); he a e age numbe o child en was
2.15
±
2.53 (Spain) and 2.53
±
0.86 (Mexico), and he a e age age o he child wi h NMD
was 14.58
±
5.38 (Spain) and 13.15
±
4.63 (Mexico). S a is ically signi ican di e ences
we e obse ed be ween Mexico and Spain in ca egi e age (U = 398.50, p= 0.002), yea s o
educa ion (U = 384, p= 0.027) and he numbe o child en (U = 448, p= 0.006).
Fi s , he ollowing mon hly expenses incu ed by ca egi e s in bo h Mexico and
Spain we e calcula ed: ees expended on heal h p o essionals o he child; cos s expended
on heal h p o essionals o he ca egi e s; annual expenses on assis i e echnology o
he child; and he amoun o money subsidized o each expendi u e. The expenses and
subsidies a e p esen ed in EUR o easy compa ison.
As household income la gely in luences he impac o household expenses, he annual
sala y in bo h coun ies was also assessed. In Spain, he a e age yea ly sala y o a amily
uni was 38,197.16
±
43,350.57. In Mexico, i was 8272.47
±
20,762.94. Gi en he sala y
di e ence be ween he wo coun ies, he pe cen age o household income spen on hese
expenses was assessed o see he eal impac on he amily. The ollowing o mula was
used o calcula e he pe cen age o amily income alloca ed o each paymen : (Annual
se ice cos —annual subsidies g an ed)/Annual household income. The pe cen ages a e
shown in Table 2.
Table 2. Pe cen age o annual income ela ed o ca egi e s’ heal h expenses (n= 74).
Spain
Annual Pe cen age (%)
M±SD
Mexico
Annual Pe cen age (%)
M±SD
Expenses associa ed wi h se ices p o ided by
heal h p o essionals o he child 4.61 ±9.61 5.89 ±13.73
Expenses ela ed o assis i e echnology 8.68 ±17.23 7.42 ±18.02
Expenses associa ed wi h se ices p o ided by
heal h p o essionals o he ca egi e 0.73 ±2.05 3.45 ±11.20
Desc ip i e analyses we e ca ied ou on he psychological a iables o bu den, li e
sa is ac ion, soma ic symp oms and QoL. An analysis was also ca ied ou o s udy any
In . J. En i on. Res. Public Heal h 2022,19, 12334 7 o 15
s a is ically signi ican di e ences in hese sco es be ween Mexico and Spain. Fo soma ic
symp oms, he sco es o Spain we e 12.42
±
7.22, a medium deg ee o se e i y, and
7.65 ±5.38
o Mexico, a mild deg ee o se e i y. These di e ences in soma ic symp oms we e ound o
be s a is ically signi ican (U = 244, p= 0.011). In e ms o ca egi e ’s bu den, he sco es we e
35.06
±
15.46 o Spain, co esponding o “mild o mode a e” se e i y, and 25.07
±
12.44 o
Mexico, co esponding o “mild o mode a e “o e load. The di e ences in ca egi e ’s bu den
sco es we e s a is ically signi ican (U = 287.5, p= 0.028). Rega ding li e sa is ac ion, ca egi e s
sco ed 14.35
±
5.66 (Spain) and 18.33
±
3.69 (Mexico). The Spanish adap a ion o his scale
has a 5-poin Like scale; he e o e, he minimum sco e ha could be ob ained was 5, and he
maximum sco e was 25. The o he in e na ional e sions ha e a 7-poin Like scale [
59
]. The
di e ences we e s a is ically signi ican in li e sa is ac ion (
U = 214,
p= 0.007). Fo QoL, sco es
o 43.59 ±26.58 (Spain) and 46.99 ±26.99 (Mexico) we e ob ained.
Rega ding he numbe o hou s pe week spen by ca egi e s on suppo ing hei child,
he a e age numbe o hou s in Spain was 44.62
±
37.75, whe eas he a e age numbe o
hou s in Mexico was 35.29 ±49.45.
The e we e s a is ically signi ican di e ences be ween Mexico and Spain ega ding
he expenses associa ed wi h heal h p o essionals who p o ided se ices o he child and
he ca egi e and ones associa ed wi h assis i e echnology. These di e ences we e ound
in expenses ela ed o assis i e echnology (U = 444, p= 0.010), in subsidies o heal h
p o essionals o p o ide se ices o he child (U = 440, p= 0.010), in subsidies o assis i e
echnology (U = 361, p= 0.000) and in subsidies o heal h p o essionals o p o ide se ices
o he ca egi e (U = 600, p= 0.027). All he o he analyses indica ed no s a is ically
signi ican di e ences (Table 3).
Table 3. Mann–Whi ney U- es analysis o clinical a iables and na ionali y (n= 74).
Clinical Va iables Sociodemog aphic
Va iables nMid-
Range A e age U pR
Expenses associa ed wi h heal h
p o essionals o he child
Spain
Mexico
40
34
40.20
34.32
1384.10
151.58 572 0.191 0.320
Expenses associa ed wi h assis i e
echnology
Spain
Mexico
40
34
43.40
30.56
44734.12
3487.66 444 0.010 0.300
Expenses associa ed wi h heal h
p o essionals o he ca egi e
Spain
Mexico
40
34
37.54
37.46
213.30
75.98 678.5 0.984 0.140
Subsidies o expenses associa ed wi h
heal h p o essionals o he child
Spain
Mexico
36
34
30.72
40.56
161.36
49.52 440 0.010 0.130
Subsidies o assis i e echnology Spain
Mexico
40
34
45.48
28.12
2884.75
249.00 361 0.000 0.380
Subsidies o expenses associa ed wi h
heal h p o essionals o he ca egi e
Spain
Mexico
40
34
35.50
39.85
0.00
22.05 600 0.027 -
Pe cen age o amily income spen on
heal h p o essionals o he child
Spain
Mexico
32
28
32.91
27.75
0.04
0.05 371 0.192 −0.05
Pe cen age o household income
spen on assis i e echnology
Spain
Mexico
35
27
33.63
28.74
0.08
0.07 398 0.281 0.122
Pe cen age o amily income spen on
heal h p o essionals o he ca egi e
Spain
Mexico
39
31
34.49
36.77
0.00
0.03 565 0.565 −0.173
Hou s spen on ca e Spain
Mexico
40
34
41.00
33.38
44.62
35.29 540 0.123 0.106
Soma ic symp oms Spain
Mexico
31
26
34.13
22.88
12.42
7.65 244 0.011 0.347
Sa is ac ion wi h li e Spain
Mexico
31
24
22.90
34.58
14.35
18.33 214 0.007 −0.379
Ca egi e ’s bu den Spain
Mexico
32
27
34.52
24.65
35.06
25.07 287.5 0.028 0.332
Quali y o li e Spain
Mexico
40
34
36.19
39.04
43.59
46.99 627.5 0.568 0.063
In . J. En i on. Res. Public Heal h 2022,19, 12334 8 o 15
4. Discussion
Pa ien associa ions play a p o ec i e ole in amilies, educing he disease’s psycholog-
ical and economic impac . They ha e been shown o posi i ely a ec esea ch de elopmen ,
go e nmen egula ions and clinical ials [
35
]. Fo his eason, his s udy examined he ole
o o ganiza ions in educing he emo ional and economic impac o illness on ca egi e s,
analyzing he inancial expenses incu ed by ca egi e s o people wi h DMD in Mexico
and Spain and he physical and psychological consequences o p o iding ca e. Conside -
ing hese a iables, he ole o associa ions in he ca e o pa en s o child en wi h DMD
was analyzed.
While he socioeconomic si ua ion in each o hese coun ies di e s in many aspec s,
i was ound ha he economic impac o DMD on hese amilies was high and signi i-
can in bo h coun ies. Heal h and social sys ems do no adequa ely add ess he needs
o pa ien wi h hese condi ions. Families in bo h coun ies spend a la ge pa o hei
income on heal h p o essionals o bo h he child and he ca egi e . Howe e , he DMD
pa ien suppo associa ions play an essen ial ole in educing hese amilies’ economic and
psychological impac .
To s a wi h, he highes annual expenses incu ed by hese amilies we e ela ed o
assis i e echnology o he ca e o hei child, ollowed by cos s associa ed wi h heal h
p o essionals o ea he child and, las ly, expenses ela ed o p o essionals p o iding
se ices o he ca egi e . No su p isingly, in bo h Spain and Mexico, he highes cos s
we e linked o pu chasing assis i e echnology o imp o e mobili y, as he a e age age
o he pa ien s in his s udy was be ween 13 and 14 yea s old. I is om his age g oup
onwa ds ha he deg ee o disabili y inc eases. Simila ly, ano he s udy ha ocused on
amily membe s o people wi h a e NMDs ound ha assis i e echnology usually in ol es
inc eased cos s o he amily [
12
]. These expendi u es on echnical aids we e signi ican ly
di e en be ween he wo coun ies. Mexican ca egi e s we e ound o spend less money
han Spanish ca egi e s on echnical aids, his being he mos p ominen expendi u e
obse ed in he p esen s udy. One eason o his di e ence is he undamen al wo k o
he associa ions in p o iding he pa ien wi h echnical aids, such as wheelchai s o o he
o hopedic aids.
On op o ha , hese da a become e en mo e subs an ial when conside ing he pe cen -
age o household income alloca ed o cos s associa ed wi h he disease. I should be no ed
ha he minimum income pe amily uni is di e en in Mexico and Spain. The e o e, i is
i al o conside each amily’s minimum income and he cos s and subsidies ecei ed. The
highes household income pe cen ages in Spain and Mexico we e also ound in assis i e
echnology cos s. Howe e , he a es we e also ema kable ega ding he cos s associa ed
wi h p o essionals suppo ing he child and he ca egi e . Fo Mexico, hese esul s we e
consis en wi h a p e ious s udy ha es ima ed ha he amilies o people wi h DMD spend
much o hei income on ca e [
60
]. In he case o Mexico, i is he associa ion ha educes
he impac .
In e ms o he numbe o hou s spen p o iding ca e pe week, i was highe in Mexico
and Spain han in p e ious s udies ocused on he ca egi e s o people wi h schizoph e-
nia [
61
], ca egi e s o olde people [
62
] and ca egi e s o people wi h demen ia [
63
].
Howe e , he e we e no signi ican di e ences be ween he wo coun ies.
Mo eo e , he e we e di e ences be ween Spain and Mexico in e ms o subsidies. The
esul s showed ha he subsidies di ec ed a heal h se ices a e highe in Spain han in
Mexico. This di e ence may be due o he ac ha , in Spain, he heal h sys em is public,
and he e o e, he e is a wide ange o public se ices a ailable o he pa ien , ee o
cha ge. Howe e , when analyzing he o e all impac o hese expenses and subsidies on
he ca egi e ’s li e, conside ing hei sala y, he e we e no signi ican di e ences. The same
phenomenon was obse ed when s udying he subsidies o echnical aids. A he i s
glance, echnical aids subsidies a e highe in Spain han in Mexico, bu when conside ing
all he inancial ac o s, he economic impac is highe in Spain. The e o e, when economic
In . J. En i on. Res. Public Heal h 2022,19, 12334 9 o 15
impac s udies a e conduc ed, i is ele an o conside he sala y o he pe son, he subsidies
ecei ed and he emo ional impac .
On he o he hand, i can be obse ed ha ca egi e s o child en wi h DMD ecei e
mo e subsidies o he ca egi e in Mexico han in Spain. I is impo an o no e ha he
unding sou ces a e di e en in he wo coun ies; in Mexico, hey come mainly om
pa ien suppo associa ions [
14
], while in Spain, hey o igina e om bo h he s a e and he
associa ions o people wi h DMD [64].
The in e en ions in Mexico a e mul idisciplina y and come om he associa ions
hemsel es. The associa ions p o ide pa ien s and amilies wi h he se ices o p o essionals
(neu ologis s, pulmonologis s, ca diologis s, e c.) necessa y o p o ide comp ehensi e
ca e o hei needs. As men ioned abo e, his c oss-cul u al analysis is based on wo
di e en models. In he Mexican case, each pa ien is ca ed o simul aneously by o he
p o essionals, al hough he pa ien o ganiza ions do no p o ide his clinical coo dina ion
in Spain. In Spain, he e is a e y agmen ed model, in which ca e is public and ee bu no
comp ehensi e, so ca e will no be simul aneous. In his case, he associa ions demand ha
pa ien s ecei e comple e and simul aneous ca e. The e o e, mul idisciplina y ea men
and ca e should be a posi i e elemen o conside when es ablishing he ac ion plans o
educe he impac o he disease in his g oup.
O ganiza ions s uggle e e y day o imp o e hei QoL and educe he impac o he
disease. They a e ac i ely in ol ed in und aising o esea ch in he hope o inding a cu e
o DMD. They also encou age he c ea ion o pa ien egis e s o acili a e he ec ui men
o pa icipan s o clinical ials [
65
]. Many associa ions p o ide he medical se ices ha
he child needs, which conside ably educes he cos s associa ed wi h he disease [66].
In addi ion o he economic cos s, he disease has a signi ican emo ional impac
on amilies. Ca egi e s in Spain had sco es consis en wi h medium-se e i y soma ic
symp oms and mild o mode a e bu den le els. In con as , ca egi e s in Mexico had
sco es consis en wi h mild symp oms and bu den le els. The p esence o soma iza ion
in ca egi e s is common in he li e a u e [
67
,
68
]. This is consis en wi h he ac ha
ca ing o a pe son wi h dependency no only a ec s psychological well-being bu can
also ha e a nega i e impac on he ca egi e ’s own heal h [
35
]. The esul s ob ained in
soma ic symp oma ology we e highe in he case o Spain and lowe in he case o Mexico
compa ed o o he s udies wi h pa ien s wi h anxie y and dep ession [
44
], wi h ca egi e s
o people wi h Alzheime ’s disease [
69
] and wi h ca egi e s o pa ien s wi h RDs [
70
].
The same occu ed in he o e load sco es. The esul s ob ained we e highe o he Spain
sample and lowe o he Mexico sample han he esul s obse ed in ca egi e s o pa ien s
wi h RD, speci ically wi h spinal muscula a ophy [
71
] and wi h Wol –Hi schho n [
72
]. In
he case o s udies ca ied ou speci ically in Mexico and wi h a no ma i e popula ion, we
ound ha he sco e ob ained in he p esen sample is highe in bo h cases [73].
The p esence o emo ional dis u bances in ca egi e s o dependen pe sons has been
widely s udied and co obo a ed by di e en s udies [
74
–
76
]. The pa ien ’s dependency in
hei daily li e causes p og essi e physical and men al de e io a ion in he ca egi e , which
b ings along dep essi e symp oms and pu s hei psychological heal h a isk [
77
]. Simila
esul s we e ound in he p esen s udy. O he au ho s ha e desc ibed ha ca egi e s o
child en wi h NMD ypically epo high le els o s ess ela ed o he pa ien ’s demands,
accep abili y, social isola ion, [
78
] and low QoL [
79
]. O he s udies ha e shown ha hese
ca egi e s su e om high le els o anxie y and dep ession because o hei ca egi ing
esponsibili y [79,80].
A educ ion in li e sa is ac ion can also be obse ed wi hin he physical and psycholog-
ical impac . I is no ed ha ca egi e s eel ha hey ha e no achie ed wha hey wan ed
in li e and ha hei li e ci cums ances a e no a o able. The mean li e sa is ac ion in he
s udy was lowe han in o he s udies conduc ed on a “no ma i e” popula ion [
81
–
83
].
Howe e , he mean li e sa is ac ion in Mexico was simila o ha ound in a p e ious s udy
wi h a Mexican popula ion [53].
