The development of literacy skills in

children with Down syndrome:

Implications for intervention
Margaret Snowling, Hannah Nash and Lisa Henderson
The cognitive prole o!served in Down syndrome is typically "neven with
stronger vis"al than ver!al skills, receptive voca!"lary stronger than e#pressive
lang"age and grammatical skills, and often strengths in reading a!ilities$
However, there is considera!le variation across the pop"lation of children with
Down syndrome$ %e !egin !y o"tlining some of the methodological iss"es that
s"rro"nd research on literacy development in Down syndrome !efore s"rveying
what is known a!o"t literacy and literacy&related skills$ %e proceed to review
interventions to promote reading in school&age children with Down syndrome
and concl"de with directions for f"t"re research$
doi:'($)'(*+reviews+,(--
Methodological and conte#t"al iss"es in Down
syndrome research
In order to interpret research ndings it is critical to compare performance of
individ"als who have Down syndrome to that of a comparison gro"p$ There is
de!ate s"rro"nding the selection of appropriate comparison gro"ps in research
with special pop"lations$ The aim of matching gro"ps is to r"le o"t potential
e#planations of gro"p di.erences$ /hoosing what to match gro"ps on is driven
!y the partic"lar research 0"estions !eing asked and this iss"e is pertinent to
the st"dy of reading given that it is a componential skill$ Many st"dies have
incl"ded typically developing children matched for chronological age, for non&
ver!al mental age or on a meas"re of lang"age or reading a!ility$ Individ"als
with Down syndrome have also !een compared to individ"als with learning
di1c"lties of an "nknown origin and to individ"als who have learning di1c"lties
of a di.erent aetiology 2e$g$, specic lang"age impairment3$ The partic"lar
meas"res of lang"age, reading or non&ver!al a!ility "sed for matching can
a.ect the concl"sion drawn$ There are also !ehavio"ral aspects of the Down
syndrome phenotype other than non&ver!al a!ility and lang"age a!ility 2s"ch as
motivational style3 that may a.ect their performance on tasks, incl"ding
attainment tests, and need to !e taken into acco"nt$
In terms of ed"cation, there is strong evidence to s"ggest that the relatively
recent policy of ed"cating children with Down syndrome in mainstream schools
has had a positive e.ect on lang"age skills and academic attainments$ This
means that the ndings of st"dies cond"cted a n"m!er of years ago need to !e
interpreted with ca"tion$
4eading development in Down syndrome
Home and school in5"ences on literacy development
in Down syndrome
6 wide range of factors !oth intrinsic and e#trinsic to the child are known to
in5"ence literacy development
7'8
$ /hildren and yo"ng people with Down
syndrome attending mainstream schools o"tperform their peers in special
schools in reading and lang"age, emphasising the role of environmental
factors
7,,)8
$ Home environment is also likely to !e a critical factor and there is at
least anecdotal evidence that the introd"ction of reading in the pre&school years
to children with Down syndrome is !enecial$
6 dissertation !y 4icci s"ggests that home literacy environment is a predictor of
the interest children with Down syndrome have in reading
7*8
$ Moreover, parental
!eliefs a!o"t reading, incl"ding their propensity to ask 0"estions d"ring shared
reading, predicted children9s receptive voca!"lary and comprehension skills$
However, Trenholm and Mirenda who s"rveyed the carers of ,,* /anadian
adolescents and ad"lts with Down syndrome reported that, altho"gh there was a
wide range of reading materials in their homes, many parents e#pressed
concerns a!o"t the availa!ility of good literacy programmes
7:8
$ 6 nota!le
o!servation was that few parents reported asking any higher&level 0"estions
d"ring reading with their o.spring s"ggesting, perhaps, that comprehension
aspects may !e relatively neglected$
;honological awareness
It is well&esta!lished that phonological awareness, the a!ility to re5ect on the
so"nd str"ct"re of speech, is a strong predictor of individ"al di.erences in
reading skills in typically developing children$ ;honological awareness has !een
assessed in Down syndrome at the level of the sylla!le, onset and rime and
phoneme$
6n early st"dy !y /oss" and colleag"es s"ggested that children with Down
syndrome learn to read in the a!sence of phonological awareness
7-8
$ S"!se0"ent
st"dies have shown that children with Down syndrome have meas"ra!le levels
of phonological awareness
7<8
and that altho"gh phonological skills are weak, they
are nevertheless associated with variations in these children9s reading skills
7=&',8
$
It has !een s"ggested that compared to typically developing children, the
development of phonological awareness follows a di.erent path in children with
Down syndrome$ >om!ert fo"nd that a gro"p of ?rench children with Down
syndrome were poorer on tasks of rhyme oddity, rime @"dgement and phoneme
synthesis than on tasks tapping more e#plicit awareness of phonemes, s"ch as
phoneme co"nting, phoneme spelling and phoneme deletion, in contrast to the
ndings from typical development
7'(8
$ Snowling et al$ reported that children with
Down syndrome co"ld identify initial so"nds in words !"t fo"nd identifying rimes
di1c"lt
7',8
$ The o!servation of a specic decit in rhyme processing has !een
replicated !y a n"m!er of investigators
7'),'*,':8
$ There is also a s"ggestion from
some st"dies that letter so"nd knowledge is not related to reading or
phonological awareness skills as strongly in Down syndrome as in typical
development 2see 4A? ''3$
Together the ndings s"ggest that phonological awareness in Down syndrome is
only weakly associated with learning to read and is also poorer than e#pected
!ased on receptive voca!"lary$ %hen non&ver!al mental age, rather than
receptive voca!"lary knowledge, is "sed to match gro"ps, ndings s"ggest that
children with Down syndrome perform worse than controls on tests of rhyme
and initial phoneme awareness
7'-8
$ Th"s, phonological awareness may !e o"t of
line with general cognitive a!ility too$
However, there is a note of ca"tion$ In the case of ?ranBoise, the single case
st"dy 2with a relatively high IC3 reported !y 4ondal, performance on rhyme
detection and prod"ction tasks was at ceiling and her performance on some
phoneme tasks was also good
7'<8
$ Similarly, DS, an =&year&old child with Down
syndrome who had !een ta"ght to read at an early age, showed well developed
phoneme level skills and no sign of a rhyme decit
7'=8
$ She also performed at an
age appropriate level on tests of rapid a"tomatised naming and speech rate,
attesting to the integrity of speech o"tp"t processes, and "n"s"ally, her ver!al
as well as non&ver!al memory skills were well&developed$
4eading skills
4eading skills are often an area of relative strength for individ"als with Down
syndrome$ Most children with Down syndrome ac0"ire literacy skills, altho"gh a
great deal of varia!ility e#ists in the level of achievement o!tained
7E,'E&,:8
$ ?actors
that are associated with reading skills in Down syndrome incl"de cognitive
a!ility
7,)8
, e#pressive and receptive lang"age skills
7'-,,),,-,,<8
, and phonological
awareness
7',,,'8
$ It needs to !e noted, however, that there is variation in the tasks
"sed and inconsistency across st"dies, many of which have incl"ded children
from a variety of school !ackgro"nds$
6 n"m!er of st"dies s"ggest that word identication skills develop relatively well
in Down syndrome, perhaps s"ggesting a 9logographic9 approach
7,=8
, with
decoding a!ilities lagging !ehind
7,E8
$ In line with this Dennedy and ?lynn, and
Ferr"ci, Menghni and Ficari reported nonword reading decits in Down
syndrome which were o"t of keeping with levels of word identication
7'',)(8
$
However, ?owler et al$ fo"nd a signicant relationship !etween nonword reading
and word reading
7E8
, as did Day&4aining Gird et al$
7,'8
$ ?"rthermore, the e#ceptional
reader DS was a competent nonword reader
7'=8
$ In fact, she read nonwords more
5"ently than age&matched typically developing readers 2gaining a standard
score of ',,3, and she had no di1c"lty reading those witho"t orthographic
neigh!o"rs, s"ggesting she was relying on grapheme&phoneme
correspondences 2see 4A? '(3$
Longit"dinal st"dies
/ross&sectional st"dies of reading are limited in their a!ility to el"cidate the
process of reading development$ Longit"dinal st"dies allow the investigation of
growth in reading and phonological skills over time and have greater potential
for e#ploring ca"sal relationships$ Gyrne et al$ followed a gro"p of ,* children
with Down syndrome who attended HD mainstream schools over two years and
compared their progress with that of a gro"p of average readers and a gro"p of
slow readers from the same classrooms
7)'8
$ The gro"p of typically developing
average readers performed at higher levels on all lang"age, literacy and
memory meas"res and also progressed signicantly more in all areas over the
two years than the children with Down syndrome did$ The children with Down
syndrome made steady progress in reading acc"racy !"t their progress on
meas"res of reading comprehension, lang"age, spelling and memory was more
limited$
Day&4aining Gird, /leave and Mc/onnell followed the development of reading
skills in a small gro"p of ', children with Down syndrome over fo"r and a half
years
7,'8
$ They fo"nd improvements in word identication skills over time and
there was an advantage of word reading over nonword reading$ They also
monitored the development of phonological awareness$ There was no
improvement in segmentation skills over the years !"t spontaneo"s rhyming
skills improved steadily$
Similar ndings have !een reported !y H"lme, >oetI, Grigstocke, Nash and
Snowling, who followed :: children with Down syndrome aged : to '- years
2mean age EJ''3 over a period of two years, assessing them at three points in
time
7'*8
$ Their progress was compared to that of a comparison gro"p of -'
typically developing children matched for reading a!ility !"t of higher ver!al
and non&ver!al a!ility$ Despite starting o"t with similar levels of word
recognition, the children with Down syndrome made signicantly less progress
in reading acc"racy over time than the typically developing gro"p and in
partic"lar, their nonword reading skills lagged !ehind$ 6mong typically
developing children, the predicted relationship !etween phonological awareness
and reading development was fo"nd$ However, among children with Down
syndrome it was a meas"re of receptive voca!"lary that predicted reading
rather than phonological awareness$ Similarly, Gyrne et al$ fo"nd that reading in
their Down syndrome gro"p was associated with "nderstanding of grammar and
a"ditory memory
7)'8
$
/orrelates of reading in Down syndrome
Laws, Nye, Lom!ard and Griscoe reported preliminary ndings from a st"dy
comparing the reading and lang"age skills of children with Down syndrome with
those of children with specic lang"age impairment and children with specic
reading di1c"lties
7),8
$ The gro"ps were of similar age 2=&E years3 !"t varied in IC
and lang"age skill$ There was considera!le overlap !etween the reading scores
of the children with Down syndrome and those with specic reading di1c"lties$
However, levels of letter knowledge and of phonological awareness were
signicantly lower in the Down syndrome gro"p than in matched s"!gro"ps of
children with specic lang"age impairment and specic reading di1c"lties,
especially in rhyme awareness where few of the children with Down syndrome
scored a!ove chance$
Kne pla"si!le reason that children with Down syndrome are !etter at reading
than predicted !y their oral lang"age skills is that they have relatively good
vis"al skills 2e$g$ 4A? E3$ ?idler, Most and >"i!erson e#plored the relationship
!etween reading skills and vis"al perception in ,E children and adolescents with
Down syndrome and ,( with learning di1c"lties of mi#ed origin
7))8
$ The two
gro"ps were matched for age and non&ver!al mental age, and s"!se0"ently
fo"nd to !e e0"ated in reading skill$ ;articipants were administered tests of
vis"al memory, g"re&gro"nd discrimination, spatial relationships, vis"al clos"re
and vis"al discrimination$ There was a signicant correlation !etween a
composite score on the percept"al tests and word identication in the gro"p
with Down syndrome 2rL$--3 !"t not in the gro"p with mi#ed aetiologies rL$,)3$
%hen age was controlled, vis"al processing skills acco"nted for )*M of the
variance in word identication in the gro"p with Down syndrome$
The high incidence of mild to moderate hearing loss associated with Down
syndrome might in principle a.ect the development of phonological awareness
and reading skills$ However, altho"gh one st"dy fo"nd that a gro"p of children
with Down syndrome who co"ld read di.ered in their hearing thresholds from a
gro"p of children with Down syndrome who were not a!le to read
7,'8
, other
st"dies fo"nd no association with either phonological awareness or reading
levels
7'*,)*8
$
4eading comprehension
4eading comprehension in Down syndrome has attracted m"ch less research
interest than decoding a!ilityJ however it presents a signicant area of di1c"lty
for individ"als with Down syndrome
7,',)(,)',):8
$ Go"drea" showed that reading
comprehension and decoding skills were less well developed in a gro"p of yo"ng
people with Down syndrome compared with a yo"nger gro"p matched on non&
ver!al mental age
7'-8
$ However, !oth word identication and passage
comprehension were predicted !y meas"res of receptive voca!"lary, sentence
memory and MLH 2Mean length of "tterance3$ Similarly, ?owler et al$, and Laws
and >"nn reported relationships !etween lang"age and reading comprehension
in children with Down syndrome as in typical development
7E,,'8
$ In the case of the
e#ceptional reader descri!ed !y >roen et al$, comprehension for literal facts was
within the normal range !"t she had signicant di1c"lty answering
comprehension 0"estions that re0"ired her to make knowledge&!ased
inferencesJ in general, her reading comprehension was in line with wider
lang"age comprehension skills
7'=8
$ In s"mmary, reading comprehension appears
to lag !ehind acc"racy in Down syndrome !eca"se it is limited !y lang"age
skills$
Literacy interventions for children with Down
syndrome
;honological awareness training
In a small scale st"dy, Dennedy and ?lynn eval"ated whether phonological
awareness training wo"ld improve grapheme&phoneme skill and lead to clearer
speech prod"ction in a gro"p of three children with Down syndrome
7''8
2"sing an
intervention programme devised !y >illon
7)-8
3$ ?ollowing eight one&ho"r
intervention sessions the children gained higher scores on alliteration matching
and spelling tasks, tho"gh no statistics were reported to ascertain whether
these increases were statistically signicant$ The spelling data s"ggested that
over the co"rse of the intervention, participants moved from an awareness of
initial so"nds to an awareness of nal so"nds$ The increase in phonological
awareness did not generalise to the phoneme segmentation task, s"ggesting
that children with Down syndrome need to !e specically ta"ght the
phonological awareness skills needed for 5"ent decoding$ This st"dy fo"nd no
positive e.ect of training phonological awareness on speech prod"ction$
Fan Gysterveldt, >illon and Moran eval"ated the e.ects of a si# week
phonological awareness training programme on seven *&year&old children with
Down syndrome
7)<8
$ The intervention involved training parents to !ring their
children9s attention to letters and so"nds in words, and to initial phonemes
d"ring daily shared reading activities$ There were improvements in phonological
awareness and letter knowledge, and knowledge of phonemes depended "pon
whether or not the child knew the partic"lar letter that represented the
phoneme$ The ndings of this st"dy s"ggest that phonemic awareness may !e a
conse0"ence of letter learning in Down syndrome 2however, H"lme et al$, did
not replicate this item&specic e.ect
7'*8
3$
4eading intervention
Like other children with intellect"al disa!ility, children with Down syndrome are
often ta"ght to read "sing a 9sight word9 or 9Look and Say9 approach, in which
they learn to associate whole printed words with their spoken forms$ 6n o!vio"s
limitation of the sight word approach is that it does not e0"ip the child with
strategies that ena!le him or her to read "ntrained words$ 6n alternative
approach to reading instr"ction is 9word analysis9 or 9phonics9$ In an earlier
review of research on reading instr"ction for children with moderate mental
retardation, /onners s"ggested that a word&analysis approach is feasi!le and
appropriate for this pop"lation
7)=8
$ In line with this view, ?arrell and Alkins
reported ndings from a gro"p of children with Down syndrome who co"ld "se
9the alpha!etic principle9 in reading and writing and who attended to the forms
and so"nds of words
7)E8
$
6 direct comparison of two instr"ctional techni0"es for teaching oral reading
skills to children with Down syndrome was "ndertaken !y /"pples and Iacono
7*(8
$
In this st"dy, fo"r children were ta"ght "sing a whole&word 2look and say3
approach and three children were ta"ght with a word&analysis approach in which
children were ta"ght to read monosylla!ic words !y com!ining the phonological
"nits of onset and rime$ 6fter - weekly sessions lasting appro#imately *:
min"tes, * o"t of the seven children 2, from each intervention gro"p3 showed
meas"ra!le improvement in reading the trained words$ Knly two children from
the word&analysis gro"p were a!le generalise their skills and showed an
improvement in reading a set of "ntrained words 2note tho"gh that these words
had the same rime "nit as the trained words3$
Gaylis cond"cted a small&scale training st"dy "sing a programme designed to
develop the emergent literacy skills of '( children with Down syndrome who
possessed some letter so"nd awareness !"t did not "se this knowledge to
decode "nfamiliar words in their reading
7*'8
$ The programme was an adaptation
of the 9reading and phonology9 approach developed !y Hatcher, H"lme and
Allis
7*,8
$ /hildren were seen individ"ally for two one&ho"r sessions a week 2for E
weeks3 in which they received training in phonological awareness at the onset&
rime level incorporated into a programme of te#t reading enco"raging letter
knowledge, sylla!le segmentation skills, comprehension, spelling and writing$
The onset&rime level of phonological awareness was chosen !eca"se of the
ina!ility of the children to !lend at the phoneme level and !eca"se it was
considered this wo"ld red"ce the memory load placed on the children !y
presenting 9ch"nks9 of words rather than individ"al phonemes$
Aach of the ten children in the st"dy acted as their own control and performance
on tests of reading and phonological awareness was assessed twice !efore the
intervention !egan to provide !aseline meas"res 2t',t,3, the week following the
last teaching session 2t)3 and three months after the end of the teaching
sessions 2t*3$ 6ll ten children made gains in letter recognition and word
recognition and in a meas"re of !ook reading performance, tho"gh progress was
0"ite varia!le across the gro"p$ >ains in phonological awareness were m"ch
less consistent, with some children showing gains and others showing
deteriorations in performance$ ?or - of the '( children gains were larger than to
!e e#pected when compared with the progress made !y a larger cohort from
which the children were drawn$
6long similar lines, >oetI, H"lme, Grigstocke, /arroll, Nasir and
Snowling
7*)8
delivered a modication of the Hatcher et al$ approach
7*,8
, training
phonological awareness in the conte#t of learning letters so"nds and working
with words in !ooks
7**8
$ In this st"dy, phonological awareness training was
targeted at the phoneme levelJ the programme also incorporated sight word
learning activities and speech prod"ction e#ercises that foc"sed the child9s
attention on artic"lating so"nds in words and perceiving so"nd contrasts as well
as decoding strategies$
Aight children with Down syndrome received the intervention for a period of =
weeks 2>ro"p 'J delivered on a daily !asis in a *(&min"te one to one session3
!efore intervention for a f"rther - children in a 9waiting list9 control gro"p 2>ro"p
,3 commenced$
>ro"p ' made signicantly more progress in letter and word recognition d"ring
the period when they received intervention compared to the waiting control
gro"pJ e.ect siIes were large for %ord 4eading 2/ohen9s d L ($=(3 and
moderate for nonword reading 2/ohen9s d L ($*3$ The waiting gro"p started to
make progress once their intervention !egan and overall, children progressed
more in word reading skills over the d"ration of the intervention than they did
when they did not receive this intervention programme$ ;rogress in phonological
awareness was modestJ altho"gh there was signicant progress in alliteration
matching d"ring the intervention, the ma@ority of the children remained at
chance on tests of e#plicit phoneme awareness$ This nding contrasts with that
from case st"dies reported !y /ologon, /"pples and %yver who compared the
e.ects of instr"ction in phonological awareness 2thro"gh oral reading3 and
reading comprehension 2thro"gh silent reading3 in a sample of ': children with
Down syndrome aged !etween ,J'' and '(J= years
7*:8
$ The phonological
awareness intervention trained decoding skills at the onset&rime level and
individ"al phoneme level com!ined with pict"re matching and sentence
completion tasks$ The reading comprehension or 9silent reading9 intervention
trained word and pict"re matching, sentence and pict"re matching, action
sentence tasks and sentence completion tasks$ ;articipants were seen
individ"ally in a weekly session for '( weeks$ They were assessed pre&
intervention, immediately following completion of the intervention and for
maintenance testing si# months later$
6ltho"gh there was considera!le variation in progress across children, all
demonstrated signicant gains following the intervention$ ;articipants in the
phonological awareness intervention condition showed signicant improvement
on meas"res of phonological awareness, phonic decoding, letter&so"nd
knowledge and reading comprehension$ ;articipants in the silent reading
intervention condition showed signicant improvement on meas"res of reading
comprehension, reading a!ility, phonological awareness and letter&so"nd
knowledge$
?inally, a n"m!er of other small scale or pilot st"dies are in progress for children
with Down syndrome$ These incl"de an intervention to promote literacy
stim"lation d"ring @oint !ook reading which has positive e.ects
7*-8
and an
intervention programme to promote reading comprehension strategies
7*<8
$
/oncl"sions and f"t"re directions
4esearch on literacy development in Down syndrome has !"rgeoned in recent
years, tho"gh st"dy remains foc"sed on word level decoding a!ilities and
phonological skills with a relative neglect of spelling and reading comprehension
processes$ Goth areas of en0"iry co"ld o.er important insights into the
relationships !etween speech and lang"age skills, specic forms of ling"istic
representation and written lang"age a!ilities$ In addition, the ma@ority of
research remains small in scale, often involving children and yo"ng people
widely ranging in age and there is a dearth of longit"dinal st"dies$ Moreover,
relatively few comparisons have !een made !etween the literacy skills of
children with Down syndrome and those with learning di1c"lties of mi#ed
aetiology$
The pict"re that emerges is that of considera!le variation in the reading
attainments of children with Down syndrome$ The e#tent to which this is the
o"tcome of constit"tional vers"s environmental factors is "ncertain$ It appears
that ver!al rather than non&ver!al mental age is a predictor of individ"al
di.erences in word recognition in Down syndrome !"t nonetheless, word&level
reading skills are generally in advance of what might !e predicted given
receptive voca!"lary knowledge$ In terms of component reading skills, levels of
phonological awareness and decoding a!ility are generally lower than levels of
word identication 2!"t they are related and there are some e#ceptions3 and the
ma@ority of children with Down syndrome have poorer reading comprehension
than reading acc"racy, pro!a!ly d"e to lang"age limitations$ 6 consistent
nding across st"dies is of di1c"lties in rhyme recognition and awareness in this
pop"lation s"ggestive of atypical development of phonological skills$
%ith these ndings as a !ack&drop, a n"m!er of small&scale intervention st"dies
have eval"ated the e.ects of programmes targeting phonological awareness
and reading skills$ Most of these st"dies report very short&term interventions,
few have incl"ded control gro"ps and none have "sed randomised trials$ The
pict"re emerging to date is that word recognition skills are more amena!le to
training than phonological awareness skills or decoding a!ilities !"t more
research is re0"ired to clinch this iss"e$
?inally it sho"ld !e emphasised that, @"st as in the general pop"lation, there is
variance in a!ility in Down syndrome res"lting from genetic di.erences and
di.erences in the environments thro"gh which genes act$ Individ"als with Down
syndrome inherit a f"ll set of chromosomes from their parents, as typically
developing children do, along with the e#tra chromosome material$ They also
e#perience a wide range of environments at home and in school that will
contri!"te to their literacy o"tcomes$ There is an "rgent need for longit"dinal
st"dies that follow the development of literacy in Down syndrome from !efore
the start of formal schooling and that map relations !etween their general
cognitive a!ilities, lang"age and reading skills$ S"ch st"dies sho"ld also
investigate environmental in5"ences on literacy$ ?indings from these st"dies will
inform individ"al di.erence in responsiveness to intervention in Down
syndrome$
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6cknowledgements
%ork towards this review was f"nded !y a Gritish 6cademy 4eadership to MS, a
grant for the Health ?o"ndation and AS4/ st"dentships to HN and LH$ Thanks to
/harles H"lme and Dim Manderson for assistance at vario"s stages$
Margaret N Snowling, Hannah M Nash and Lisa M Henderson are at the Hniversity
of Pork, HD$

;aper prepared from presentations and disc"ssions at the Down Syndrome
4esearch Directions Symposi"m ,((<, ;ortsmo"th, HD$ The symposi"m was
hosted !y Down Syndrome Ad"cation International in association with the 6nna
and Nohn N Sie ?o"ndation, Denver$ Ma@or sponsors also incl"ded the Down
Syndrome ?o"ndation of Krange /o"nty, /alifornia and the National Down
Syndrome Society of the HS6$ Information a!o"t the symposi"m can !e fo"nd
at http:++www$dseinternational$org+research&directions+

4eceived: '< Nan"ary ,((=J 6ccepted: ,* Nan"ary ,((=J ;"!lished online: , N"ly
,((=$

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