1. health and fitness
2. disorders

Epidemiologyof FASD in a Provincein Italy: Prevalence
of Childrenin a RandomSampleof
andCharacteristics
Schools
H EugeneHoyme'
wendyO. Kalberg,
Fiorcntino,
J. PhilllpGossage,
PhllipA. May,Daniela
Luigi
LyonsJones,MigueldelCampo'
Coriale,Kenneth
Giovanna
LutherK-Robinson,
andMauro
DavidBuckley,
LucaDeiana,
Tarani,l,lana Romeo,Piyadasaw.Koditlwakku,
Ceccanti
B&kgrourd: Accuratecstinatesof the lreval€n€ and claracl€rhtics of feral alcoùolsvndrÓme
IFAS) ard fetalalcohol spectrun disord{s (FASD) in a Wesre.nEùîopeanpoPulatìonarc lacking
and are ofparliculù ìnterestin setlingswberethcnsùal Pattemofalohol consunprionìs thoÙgh!!o
be daily drinking{ith mcàls.To addre$ úcs nsues,àrelid€miologt slùdv ofl'AS and other FASD
wds undertakenin ltalian schooh.
Merhods:Prinary schooìs{t:25) in 2 healthdishicrsofthe Lazio regionwererandohlv electcd
and reduited for the sludy. File hundred forry-rlree chiÌdren,50% ofthose enrolledi. îrst-gmde
clases. .eciv€d !.rental pefnission to paiiicipaie in a 2-riered.activ€caseasertalnnenr PreeÙng
prÒcess.
Dctailed€vduarion ofchildr€n selectedin ipr€linì'na.v erNringphase wascarriedoùr or
tnosewho weresnall for leishr. weight,and headcircumfcrene and/or rcfered bv teichús lor sùe
psted lea.ninE and behavioraìproblems. Detailcd evalùatìoú was €rried oul on cach child s;
(3) pre_
{l) physìcalerowih and dysmorpholosr.{21psycholosi.aldevelopmcntand b€hùvior,and
g.ÓÙp
A
ol ó7
intefliews.
for
FASD
via
nalemal
oth€r
.isk
lactors
rÒ
alcohol
and
naral cxposù.e
wasutili2edas a conpariso! group
childrer wiLhoùtFASD from rle sane classes
randonly selected
Results: UsiDs 2 d4oninatoB for pÉvalen* stimaiion, a consNative one and a s1nct samplebdsd estimale, the preval€n@of FAs in lhis proline of Ìtaly wò 3.7 to 7 4 p{ 1,000children wlen
ca$s of paúial FAS (PFAS) atrd a èse of almhoì_relaled neùrodcvelopmend denciÌs {ARND) wee
al 35per I,000oveEll o.
added1oFÀS cass, île nte of FASD was20.11040 5 p€r 1,000dd es!ìmared
belwÉn 2.3 and 4l% of dI clìldÉn. Tlìis exc€€dspreviosly pùblùhedestinatcsol bolh FAS and
FASD for the wesremworld Delailed dau re !É*ntcd rhar denonsttab rhe nlilitv of the guid€linesof
rhe revised hstilurc of Medi.irÈ dìagnostic c.i&na for FASD Child.er wì1h FASD aÉ signiiiontlv
more inpaired/afrcted (, < 0.05) Lldn nndonly Èlected coúparìsn children otr all measues or srowrh
deficiency, kcy facial featuÉs ol FASD, overall dlanorploloeJ $ora, lateuàee conpEhersion, nonverbal lQ, and b€havior. Maleml rcports ol cùrrent drìn*ing were sieDificantlv highe. Ibr úoth*s of
FASD childÉr thù @nparison moùes, bù1repofed nLes of oveiall drinking duing ptcgnancv were
not signllìcantly dif€renr. In conlrasl to expectations,daily d.inking among nothers of 1ìe Llmpariso.
groùp was nor coúnon. Howeve., dysúorpholoet soFs of tbe clildren were signìficantÌv corelattd
sith drinkire in ùe s@nd and ùnd tnmesters,dnlks pef cùÍen1 drìnking day, and curent drinks pe.
molrh. Fìmlly, children wjt! rhe lhysi@l fealures of FASD had lov* IQq ronverbal IQ was sienii:
icantly corelared wìth nead ciÈumfdene aod n€galively orela&d wirh ove.all drsnorlholoe/ smÉ,
$noolh phlltrum, and seveÉl othd facial and physì€l anonalis cha.acrerìsticof FAs.
coNlùsions: Using caefùl mcasùrcsofa$e.tainmenr in a Pnmai, shool sefting,theseresùlis
Drovidcrelalivelyhighestitratesof Lhepr€valenceofFASD and ralscthe queslio! of*hethcr FASD
is mo.e coúnor in the westernworld than previousl, estinarcd.
IGwords: Feral Alcohol Syrdrone {FÀS). Fetal Alcohol Spectrùm Dìsordc. (FAD),
Elidèmlology. Preral€ne, llaiy.
LaSapierca Rome
Fún J'hpUnì,erciryaf Ner Meri.a,Atbuqkrque, NewMexico (PAM,JPG.WOK,PTUK DB):Th.Unirùsiîj.fRotue
hal, I D F, GC, r,T, M A, LD, MC ) : StdnîÒrd Unifernty. StalÍÒ 1, calfomia ( H EI! ) : The stat. uhirùsìl! of New fark at Bu|lala, lJùflà|o, Ncf
rotlr (LKR)tThe UhíeeÍit, aJ Catifu ia, Sdn Dtuga, Cati'ot"ia ( KLJ ) : akd th. Oni*aitd t Panpeu Fabra Barcelanú Spaín ( MC )
A.ei,erljot lruhliaîion No@nber 1,2A05: acrcpled Altìl 26, 2006
(tilot projed flbenttuct # 5125?A-P1660.
Thk lajeú;u!
luhìed in part br the Ndtiùat l$înúe on akÒhÒl Abw and Al.ÒhÒlbn (NlAA,a)
pA s D crFAsD )-AA0 t 481t aú
78021t asl,r 'ah San Dieca Sîai Unive iq ) a! Fú aJ the tnt..ú4tionat cùsortiuh fù th. sttulr _al
oÌ
the
lazía
Resiú. a's?ssorutoalo sanita dela
resionat
deryrtneú
al
the
a
ihe
hultt
ù
wú
;ppùett
sa,.mùenl
i ^a 1as2sl . in rtat!.
ú smnífrcn
RèsioheLa.ìo (,4SL RMG ) , and a sant h! Slî 4C Oul""R.ptk1 rcqùests: phitip A. M dt, PhD, Cehtel on Àlcohohm, SubstdnceAbuv ahrl ,1ddicÌíons ( CAS A,4) , Th. Univercity oÌ New Merica 2654
yate Boute,a ,S.E. Albùqrcrque,NM 87106: Far:5A5 925-2313:E mail: pna!@,unh edu
Copt'tistu A 20A6bf the Rekor.h Saciery on AkohÒtam
DOI: 10,1tlt/j,r$ù02t7,2006.00183.x
,4/.tr, c/t Elr ner vol30. No9.2006:pp ì562-15?J
EPIDEMIOLOGYOF
FASDINA PFOVINCEIN I'IALY
-ra HE tPIDÉMIoLoGY oF feralaicoholsyndrome
STUDIESOF FASDPREVALENCE
ANDOTHER
ÉPIDEMIOLOGICAL
CHAMCTEFISTICS
l. (FAS), or ieral alcohol specrrumdrsorders(FASD)
from a populationolany kind, had nol beenresearched
Our review of the literaturc revealedno major epid€miobased perspectiveiD a Western European population.
plan
However,in 2003,a collaborative
betweenresearch- logic studiesof FAS or FASD prevíouslyurderlaken in
ers from the United Statesatrd ltaly was finalizedwith Italy or in Western Europe that utilìzed extensiveoufeach
officialsofthe U.S. National lrNtitute on Alcohol Abuse or otbermethodsof actlvecaseasc€rtainment.
Most studiesin the United Statesthat have atiempted to
and Alcoholism (NIAAA) and several governmental
the prevalence and other epidemiological charltaly
to
determine
the
nature
and
extent
of
define
agenciesin
FASD iD ltaly. While most population-basedepide- acteristicsof FASD have usedclinic- (Astl€yet al., 2004;
miology studies of FASD have been carried oùl in Sampson et al., 199'l) ot record-based systems(Chavez
populationswhere heavyepisodicddnking (e.g.,"bjnge et al., 1988;Egelandet al., 1995, 1998)witlìout active
drinking") is commoq in much of WesternEurope alco- recruiimeDt in defined populatioft. Sùch meihods are
hol consumplionis commonly believedto be modeúte, likely to underreport the extent and specificchaúcleristics
of the problem in any popùlation (Levershaand Marks,
daily, and fith meals.
1995).Without activecaseascertainment,
many children
with FAS and other FASD are neither det€ctcd (Clarreú
eta1.,2001;Egelandet al., 1998;Little et aì., 1990;Stratton
et ai., 1996)nor referredfor a diagnosis(seerevrewsrn
RÉLEVANTLITERATURE
ON FASDANDII'ATERNAL
Abel, 1995,1996,1998;Abeland Sokol, 1987,1991;May
DRINKINGIN ITALY
Strattonet al., 1996).Comparingstudand Gossage,2002;
Only a few cas€solchildren with FAS in Italy hav€ been ies of mainstream populations that utilize different
passivevs active)is pedloÙsiflaken literally.
d€sffibedin the publishedlileratùe (CaÌvaniet al., 1985a, methods(e.g-,
population-based,
1985b;Moretti and Mortali, 1982;Roccellaand Testa,
In
active case asce ainment studres
2003; scianafo et al., 1978:Scono et al., 1993).These of FASD casesare activ€ly sought for €xamination and
articlespresentdata oÍ 24 caseswhere the physicaland diagnosisthrcugh outreachin a defn€d population throug!
behavioral characteristicsare descdbedas similar to those ar organized network of training and coÍrmunlcauon
(Sratton et al-, 1996).AI previously published, active cas€
FASD childrenin U.S. studies.
In ltaly, where daily, moderatedrinking is believedto be ascertainment, population-based sludies of FAS, exc€pt
the pÌedominant pattern, some studies have shown no one, were carfied out in prcdominantly minority (usually
relationship between maiernal alcohol consumption, Americar Indian) and lor.socioeconomic-slatus (SES)
redùcedbirth weight,or pregnancyloss(De Nig s et al., communitiesin the United Statesand South Africa (Duiml98l; Parazzini€t al, 1994,1996;Primateslaet al., 1993). stra et al., 1993;May and Hynbaugh, 1982;May et al.,
Otl€r studies, however, have linked prenatal alcohol use 1983,2002;Quaid et a1.,1993).While most popì. ationand smokingwilh low birth weighl.NonsmokersiÍ Italy basedstudieshave usedaclive referml systems,in Solrlh
who drank l0 g (0.35oz) or more absolutealcohÒla day Africa in-school screenìngof firstgrade children has been
wereat thehighestriskfor havinglow-biith-weightinfants pursuedsuccessfùllyin sevenl waves(May el al., 2000,
(<2,500 g), and malernal alcohol consùmptìonof 20 g 2005;Viljoetretal.,2002,200t. This studyin Italy u.ilizes
(0.75oz or 1.5drinks) p€r day significantlyincreasedthe merhodssimilar ro thoseusedin SourhAfricr.
risk of pretermdelivery(Lazzaroniet al., 1993a).Bonati
Only one in-school study has been completediD ally
and Fellin (1991)found that more than one-thirdof4,966 population in the United States.Clarren et al. (2001) used
womendeliveringin Italian hospitalswefe daily drinkers, methods of passiveparental consent(all children were
and that nearly ali of thesewomen continùed drinfing inclùded unlessparents took specialm€asuresto wiihdraw
after recog tion ofpregnancy.The authorsdistinguished them), which yielded very high participation in I coùnty
beíreen women ìÀ'ho"drank betweenmeals" and those in Washitrgton State. In another Washington county,
who did not, with sliehtly lessthari l% falling into the active consent for children to pàrticipate was required,
fomer category.Overall,maternald nking wasnot asso- which yielded low participation (<25%). In the highciatedwith lower birth weight,but the autho$ concluded participationcounty, the rate of FAS was determjnedto
that birth weight is affected only by abùsivedrinking be 3.1 per 1,000,substantiallyhigher than estimat€sof
(Bonatiand F€llin, 1991),the smal proportionwbo drank FAS derivedùom passiveascertainment
methods.
betweenmeals.Primatestaet al. (1993)also reportedlow
Recent clinic- and registry-basedestimatesof the prevaratesofbinge drinkiîg (1.4%) amorg preprcgnantwomen lenceofFAS in the mainstr€amUnited Stat€spopulation
in Miìan. However,in this samestudy,9% ofthe women have varied between0.33 per 1,000births and 2.0 (Abel
rcportedrisky to very risky averageweeklyconsumption and Sokol, 1991;May and Gossage,2002jStrattonet al.,
of alcohol, with 29% continuing to drìnk daily dùring 1996). Furthermore, tle combined rate of FAS and
pregnancy,
ARND asimilar to FASD) has beeiì estimated from
clinicals!ùdiesat 9 per 1,000or approximaielyl% (SèÌÌp
soner a1.,r997).
Owìrgto a lack of studiesutilìzingactivecaseascertain
meni. and also becauseof recent advancemenlsrn the
clarificalionol the Instituteoî Medicine(IOM) categories
lbr FASD (Hoymeet al., 2005),we believerhar the overall
rate ofFASD Íray be higherin both th€ United Slatesand
suggest.
To develop
WeslernEuropethanculrentestjmates
prevalence
and
chrìracteris
úroreacculaaeestimalcsofthe
tics of FAS and FASD in Weslerr Eúope, specifically
in a setiing in which bjnec ddnking is thought to bc
uncoDmou, a l€am of U.S. aDd Itlllian investigators
carrìed out thìs active casc ascertainment,populaiion
basedstudy.
Thc datr orignlàrefrÒm ir school,î$t grade saDpleslÌoú
2 heaìthdlsrrictsofthe Lazìorcgionthat lje oùtsideof thelargemet
bt ! nùÌ,bef
ropolitanarcaofRÒúe.Thesrudyareàls characletized
ofsúall towùsand muicipaliti€s.soúe vilh sùbù.bane.oronrcs
(c.e..bedroon cÒúnúniriessomeNhatdelendenton Roúe) aúd
oùers that arc feìatilely to Òoúpletely self4lfncienl, rural, and
caseconlroldesign
The stùdyis a cros sectional.
obserlational.
with retrcsFctivecolleclioùof .ratemal exposurcinfou4lion.
sereselededfrom rhc ó8
Using. raldon numbertable.25schoÒls
îern
schoohin lhe2 dlstriclsNith nat gradecla$es.halianreleàrch
úe regiÒnal
schooiadùinisùatoBandeacìrot
menbeNapproached
ihe selecled
schoolsto explainthestùdyatd ganlFrmissiÒ!1opro
ceed.All pàrentsàtrd gúardiansof irsl g.adcclrildrenrvercthen
Tier ll
cotrtrctedlia lorúal schoolcomDunìcatioúchalnets,inclùdìlg
Thc totll nùmler Òl
parenroreanizalion
meetings
ln rheevenlnes.
schools
chiìdreneúolled as lìst grade$ nt the ràndoúty selectcd
was1.086.ConsenlfÒrmsNercsigncdud letùrùedby slighlÌyoler
-a
\ r l o l o f . h e r d ' e - r \A. f r e rh e q ' J . . e r o f{ r c c n i n c \ 1 \ ' o .
pcrepresent
lnd perticipated
plct€d,exacLly
halfofll[ chlldrcn,541,
with consèntlo particìpatc
lirsL
lier
ofscrccdrg.
The
cùìldreú
in the
lled
ol all childreaeúrÒ
in tlls studyarebelie!edto bercpresentativc
in nrst Bradeat ihc rundoúlt sehted schools.A1l .cscarchproce
d!rcs rvereàppróled by both ihe Ethics Comnitiee of the reglonal
lìealth disbicl in Italy (ASL RMG) and the Unive6ity of New
Mexico Heaìth SciencestlxMn ResearchRcvicw Connittee
(ap!ùul#01039).
Iiùial Datd Collection rier I Sùeeúitls
Dàtr collecdÒn
for L\ediasnoses
o@uredvia 2 tieA ofsùeening.
ln Tìer I. lreishr,wèigbl.and headcircumlircnccloccipitof,onÌdl
circuúfererce (OFC)I were measuredfo. eàc! chiid by the local
for growlh for eachmeuurc ivcfc
schoolphyslcjansPercenriles
U.S.NàLìóralCenrer
asìsnedbt s dy srarrsins receùlly.€vised
grorLh charLs(KùchùBkì er al., 2000).Chil
for HeallhStaristics
drd ar or beloù lhe l0ú percenlilcotr bcight,or v.ìsht, of OFC
wereadvarcedlo Tier TI of the studJ(seeFjg. l). I! additìot 1o
growth,bacherswereaskcdio refor lny child silh learmngór
behrlior problems.ReferElswe.enade Ònúe basisofa qùestìo,
hd led.nirg
nai.eúal in ludedltemsotrinattcntio!,hyperactivìly,
problenNdcrivedfron the QtreriorarioO$ervalilo per I ldùìlifi
cazionePercocedeìle Dift@1a di Ap!rcldnnerto (IPDA Tercnj
DisrupriveBehavior
€t a1..2002)and a tra.slatioDof dre PelhaDr
Disorder.atiì,gscàle(PelhameÌ al., 1992).Of thc 543childre!in th€
study.153mot oDeor bolb oi Lte abÒvecriLeriafor Tìer II ofthe
srudy. Oleràll, 31.5% of thc 158 childrcnwho enieredTi€r ll
(thc diagtroric lhase) of the study sÒlelybecauseof size and/
or OI."C.13.9%enieredby rctcrralsfof bolh poor erowrh and
deiercd because of econd.ry rcltrsah ol conselt or lrulilFle
!bsencesrekrlrì!g in noncomplcdÒnoflesdng.
Ps\,.hohsnd aúlRd ttiÒ&/ l/.4!rzJ. Pslchologicalard dc\cl
opúetrtal *alultioDs utilized a battcfy of tests Lhar itcludcd
nrcdsuresof perceptull rnd nÒùlerbal.easorl.e abilily, a langùrge
îìt t M
ideio lt ic ? race.lut? t
.Òmprchcnsionm€a$úe, a.d 2 m..s!rcs of behavior. The R.rc!
Coiorcd Prog,e$i!e Mrtriccs {CPMi Ravenet dl., 197ó)ìs. pcrccp
In Tier lI.: domajns ofdssessúentw€rc cxplorcd foreacb cliÌdl
'f!e
Lùal Lcstir$tfument lor asesi4 ooDvcrbal fe$onnrg ability.
(l) dysrorlhology. lhysical graivth. aùd developmcnli (2) psycho
CPM lerJor of rhc Raren ìs used!iù yoùns chlld'cn and rhe eld
lo-Èlcald€rcloDmenL(i elligenceand behavnr.)i 2rd il) marenril
rjsk factos. A raùdadìzed cuminatiÒd, by .l dy$norpholosists erly. Colphd witlì ù $ndÀrdizcd icst ofiaÌgùage ÀbiliLy.the RarcD
c.D pfolide a silgle tcsi ofinlelÌigeúce rhar is not cùhur!Ììy biased.
wÒlttug iogethcr ir 2 teanr. was cadcd out o\e, a period of
The Ruslionì Tcs! of Laúgroge Comprchcnsion(RuliÒni. 199.1)is
2 vccks, lbllowed iúùediatcly by tlìe ps'cholÒgìcaltesting.
and
D.r.lal
.tr lialim test of lirguistic undèsrandnlg dcveloPed!rd notn,cd on
Drvnoryhol.sj,
Phtsìùl
G'Òrrh
?h!\nal E:uú1
,rexl. Physi.al ascsmcnt loÌlowed Lhe rcliscd IOM crirerià the ÌÌ.li!n poriùlaLioùto Pro!ìdc !r lssesm€n! ofoùe\ ùtderstard
nrg ol llelian graùùar. The PaÈnrfièacher Desctiptivc Bchalior
(Hoy e et ùÌ., 2005), a diegnosticschrma lhat hds aho been pub
lislcd in llaly (Spàgnolo ci al.. 2005) and used ù othcr counlrLes: DúÒr'der(DBD) ratio scale(Ìel!.n ct al., 1992)Ànd the Pesonrl
DehaliÒ* CbecÌlist (PBCL l; SLÈissgúù er .1, 199E) nred\ure
souú Afrìcî. R!$ia, and Fdend (Aùlii RàNó et al.. 1005). wìth
IONI c,ilerìa, r chiid who displaysàlloflhe foLlowiìrgcha.acte,,st,cs behalior and pnrvided ILalian pafcnlll and leacherl]erù!1ioN of
.tcts crjicía for Ìhe dìagnosisofF S: 2 or i of Llteùrdinrl làcial lhechìldl beha\ior. ThcsetetLsprolided. battery lhativas briel. yet
culLuralÌylppropriaLe. Lo xsse$ the futrdionins of Lhechildre! on
rnoDralie! (sho pibcbral 1ì$urÈs.Lhir verùilioÀ bordef, aùdiù
snooLh phill,uD), fremial àùd/or postratll erowilr retardadot
srùùalintelligence. laDsuage.aDd bèhalior. The R.vcn. Peìhan,
rndPBCL have beenusedwidl schoolclrìÌdrenin other FASD stud
(< ll]rh t'ercentile).ald microcephaly(< l0th pcr@ntile) or otilcì
jcs ji other cóuntdes(Admlns et al.. 2001tSfonnard et rl.. 2005).
.'ldence of srructurnì brànl abrorùalitics. sì1h or Nil]1(tr'l connr
Chìldrer perlom ing foorlr ót úosl of dEse iesls (gcnc,alb I J Òr
ùation of malernal drinldrg. For pll1irlFAS (IFAS), a child ùust
ùorc fandàrd deliations below Lheúein) were citdidircs lbr a
erhibil?or mo.c tìcirlfeàLrLresand I or morc oldre lbllowrngc!àr
acrerisricsrp.enatal and/or poslnaLal grovth r.tardaljoD 1< loLh diùgnosisofFAS, PFAS. oTARND wÌrc! otler proLrlem\ol srosrlì.
LÒ alcohoi .rc prcsctt.
!.rcentlÌc), *,idence of .bnóúlal brú, st.uctúe ór grÒwtlt G.e., dlsiìorpholÒgy, ald matcùral exlorft
j0Òf Lherar
mi.ro*phàly < l01h perceDtile),or e!ìdetce ofchaEctcrisl,c b€ha N i r e l y c h i l d . e n w i L hl o . m o r e f e a t u r c s o fF A S D a n d
psldìolo8ica
ftrllhartery
of
I
t€sls(F,s. l).
don cortroh rcceired tle
riorxl or cognitile abroúlalities. sjtÍ or Niúoul elidencc ot matel
ttiat.ntul ú1tefikrs aùti trtatù,al Bonr Mass lútlet. Lll b\t2 ol
relaledneùroderclopnEital
naldrirkìÙg. forrdiaelosisofllcohol
rhc îuteirrdl nrlervieùs were caricd out al the schóÒls.TIE) were
djsorder (^RND), a child úusr lwe a soljd docùntntdLioù ofsig
nifi.ant prenatàlahohol cxposure.di\piày neurùlogi.cl or slructufal iniriated as sooÌ rs coùsenrforns were receìled.The questionnaire
corsisredof 175items, nary of which were dràwù lioù qùestion
bmin lbnoùralities (e.g.. microccphaly),or n,aDifer elideNe ofa
nùiLesùsd cÌsewh..e nì fAS epidenioiogy prcicc|s (M!) eL xl.,
cóúpler and chcracrerislic!attern Òfbeh.\Ìoralor cognitiveabnor
nalìlies in.osisrert rlith dclclolnenhl lelel lnd nol exllaiDcd by 2005; viljoeú er a1.,2002). Ìten* Nere reviewedby the biùatìoDal
rcscarchtelm a.d.hokn for scnsìririly and substaÌrile ànd cultùràl
gerelic predìsposilion, làmily bacl<eroùnd,or cnvironmenl alde
.elevarce.Tr.nsÌ!ted ftonì EDglishto ILàli.n, theyscrcchcckcd via
(Ho]tu er al.. :005). Diagnosis of FAS or PF-AS wrtbout ! con
back raDshtioù tèchnlqùes.DomaiN covcredby the inslruúeút are
nmcd history of rlcohÒl er!ó$uc nust bc lieùed as rentative.bùl
$ follovs: denogralhic and sÒciÒecoroùicireprodlctivc hisLory:
iOM dicria aUow diasDosisÒI Lhesecalegories{illro!! defitilc
nutdLìÒnaùd c.tirg paltern: drinking by quanlnl, frc.lùcncl. rnd
direcr reporîs ofcxpos!'! (StuLLoùeLà1.,1996).
SrariitÈSJ!/crr. Each childwls exrútued by tirning of the ,lcohol exposlrc bclbrc, duiDg. and af|èr rhe tu.lex
TheD) sh1orylloltigt,
I Òf2 Leaùsofdysnorpholo-qisLswórkìne bltudedfron anl knovl
lregúanc): and flnnll and hode en!ironúe!1. Body ma$ index
cde. ol thè chìld ard family. Inlctrarer relìabilìLr fo. rhe iead (BMI) scores {ere calcùlated \rirh 1lE lblbwing úielric fornùj.:
dy!ìoryhoiogìst\ of 11Èterns in simlld fudies Nas lbund to be ùeishr in kilogramsl(heighr in netcrt Low Dxteflral BMI bàs
0.821o 0.92 nr nrdcpendenta$elr,nènts ófket facìal lrc.surcnìcnts .ecently beerlinked ro.n ircreasedlikelihÒÒdfór IAS bidhs {May
(Nlay er dl., 2000:Viljoenet ai..2005).Thedùta Iìom eacirc$ld sere e t a l . . 2 0 0 5 K
i h r o l e e La 1 . , 2 0 0 4 ) .
The itrolbes ofalìstldeors werecontactedfor inlervie\ls togather
Écorded (h Efslish) br a nremberofLhÈreseirchte.D wofk,'sone
on one wirh Lhecx.ùiDins Fhysicians Eac[ of rhe over 40 feaùrcs iiioùration on nìàLern.l risk aùd lrotcctirc facrors nr this lopulà
crîùircd are fe.rlurrs linl'ed by rcsarch \rilh FASD. Basedo,r the tior ind lhc spccific exposure 1o àlcohol. If r ùothcr w.s noi
a\àilabÌc. coÌlateral inlìrúdLio! wàs disùetcly solghl whenever
shùdardized rssrssnen! a tolal d)sùofpÌrology sco& was celctr
hted lof eacl child In the scodúg sysren. some key lèarures of
fossible. Màteroal iùtelvieves ivùe blìnded rÒ all tufÒrúàtior on
thc children. All5l9 úorhers who corscDtcdio hare their childìeù
|'ASD are wclghcdlrore hclvìly than olhe.\. Stuallhe.d cicurìfcl
examincd,who we,e located,{ho conserred.and sho shorcd up ior
ence,shorLprlFbralissùfes. snooth philtNn. and lhnr verúilion
b Ò r d e r o f r h c ù ! ! c r l i p a r e a l l a s s ì s n e d a 3F.e a L Ì ú e s a $ l e ù cads c o r e aD ìnrerlie{ $ere inteNiewcd. Tnjs was done so as not 10singleoui
IÒr stlgn. ary pa.ticula.wontn in the colruùrities.
of 2 rrc low weìght fór age, nìdfàcial hypoprasia.ptosis. rnd ànL
EighLysir olthe 9:l mothcrs ofclildrcn ùiù ole or mÒrelèalúres
elerled úares. lvfost lèàturescaùy d weight ol I or 0. Thc highcst
torsible scoreis 3ó. Higher scorcsindicate nore fèàLuresconslstenl corsistcnt wrth FASD {91.5%) \r*e lnteNicNcd. Sixry three ofthe
67 (94%l modrc^ ofcbild.en uldnatelr îelaired as cortroh scrc
wilh F^SD (seeH.'me et al.,2005)
jntcrviewed(l-ig. 1). This p.pcr rlports pfjDlrily tre daLàfroln 18of
Ptulùni'dt! Cl6súto1ìÒh ol l/15 atd D.fcttud. ln Lle study, tlc
_hoÌding diagnosis pending rhe 2l (85.7%) molhen ofLhe 2z IASD clildrer (ihcrcwas I sel oi
tcrm "dcfcùed is ùs€d úeÉly às a
sàLheringoI additioral hfornation fÒ. d nnal diagrosis. Whcn a tlin, cÒúlared with thc 63 control molhers ibr wnÒú data ùer.
child is examinedby dlsnorpholoeisls in dìe firs1!ú ÒI fier ll of
Cale CÒrúa|r.e lot Fiù DtzpDrri. In rcrised IOM didgnÒric
rhc srudj, hcishe is asrigned. didgnosisof prolrableFAS, delired
as posibie FASD, or nÒLFASD. Probabl€ FAS, deÈred childLen. methodology, thè I separ.te data scts (a, d. and e àbÒ!e) àre
jndepeÀleúlly coucctcd and nuùutued by 3 sep...tc eroul]s of
and córliols.re rhùadDìùjstcEd LhebaLreryóf fturolsychological
par
in
case
confereùcer
FAS,
tesrs A lìnàldiàgnosisìs asignedlater
lrolèsnDals on ihe resealchteaìì, (1) chìld srowth. physic.l delel
rìal l-_AS.ARND, or noi FASD. Fourchildrcn left rhc stùdy as stitl o!ìneùr and dlsnorphologyi 12) tsycholoeical ard behdvior,ìl
behdlioral/leaning, and 52.ó% oi rhe children etrtcrcd solèly lor
iearirg and/of bchrvioi probleDs. Additiontrl ch,ldren *ere .lso
as cÒnlroh.
eúleÉd ituo Ticr lI scleenn,-q
ascsDcntsr a.d 1l) úatetual risk.trd protectì\edaLà.Dudne Îhedxta
phse. therejs ro shringoffndings rcros djsciplit$ Once
collecLiÒn
all dltalrecolltuLed and liled clcctronjcllÌyir a ùùrràlìad data bank,
rhen sùrNary 1ìodìngsare lJ.eparedon eachchild lia ! cdsecÒnlir
Lrrr. cl e,.n.r.tr5 i g
- 1 - r ' ' . A r . ' , c- o I ! r c n r ' . . l
erch of thc I subranliverdatadÒùains,.onrcs logelher over seletul
dats ro revieù and dì\cussTier II1ìndings on a crsè by c.sè b.ss.
A nù.ldiaerosis tbr edclrchild ard cortol {to @ nÎDr llut ratrdÒdnr
dolor ha\c on FASD) is asieftd. Reprlsrl.tilcs ol'
sclcctedcÒnLrols
eàchdisciplirc/datddomain p.esenldata tòr cacì chiì<lsLììlblnrdtdrs
io lhc rcasonthe child emeredT'cr lI of the studl. The dirgnosisis
dreDnade b!corsctrsus or, itne.essrry and vù! rarcl), b] vot..
S.k.tìor t)J E cantnl chil.hek. Controls artendnrg rhe snmc
nBt grade clascs $rre closen t, = 75) vla a f.ndon nùÌ,ber rtble
fon .lì 5.ll child.en IÒr whom th.rc were siéred cÒnsert foúns,
rceardh$ of the chìld\ sizc or a rèfùral for beharioml/Ìcrnring
p.obhns. They aie belielcd ro b! .epresenlaLive
ol Lie .lcragc of
modrl cbild eriollcd in these nst grade clNs. Cont.ol childrèn
ùldcNùnt ùe \creenìngand tcstnrs sìmrlhneÒusly sith dìc ildcx
ca5es.Earúines ìn all ofthe subsLatLivedomaì$ alld Prrents and
tcachcr vere blinded throùgholt the reserrchas lÒ lhe rerson lof
exrúiniùg or lesrìnglny of tÌe childreú. sùbjccls.ot coÌrroÌ5. The
CencmÌ publìc and rhe schools knc$ i1 rva\ a lrdy of devclop
Dcni. NineLeenof rhc rardo y sele.led comrols Ncrc oagitrlÌy
defcftd lpon dysúÒiphologiùl cxaùriùrLioÌ b{aùse of oúc or
*ith f_ASD.
irore phlsi.al feiturcs consisteDL
Endy ìr lhc study. I ÒfLhe 15 pickcd controls sere not rndùded
becru\e Òf Nilldrawn pcmission. ,nd laler 3 rsrc nol provided r
sccond p..missìon for lstchologìc!ì cfing TNo of the rardomll
schclcd chìÌdrer were foùnd to hale an FASD as a nral diaglosis
aùd reúoled ascortrols. but all óLherschosc! in lhc originrl colLról
seledion a.e inclùdedir the conlrol groL0 Òf ó7 (Fig. I).
illt hd! ol F.4SD ChìtLbùt. The data rcpoúed io tlris ÈI)er rep
fcsent only Lhe úoth.$ of children wìth dù IASD dìaerosis lnd
dodE6 of 6l connoh. There rerc 2l clÌildrcn d,agno\ed Nith .n
FASD. but bc.aùsc a sct of twins ras iicludcd. thcrc {'ere onl}- 2l
possibleùolher\ to i ervies. Up to 5 bìdogicdl nothe6 of f^SD
childLer arc noî hctded !n sÒúe anàlyss, bùQusc 2 rcfe umvril
lble dùe LÒadóltion or foslcf placentnl. I codld lol bc locat.d.
1 rcfusedrù i e.liew .epcatcdly, and I LenninaLedúe intcrvicrt
preùatnrcly. fourol lhe DÒLh€6ofFASD children denEd drirking
at all; ye1,all bur one of thcm Nho did denr dúúkine weie knosn
infonnalìy to rcÌixble collaleral i.fo rants (lcrchca, connnùnlly
nèùbeA, and social seÀìce worke,s) as drinkcs !Ddjor as having
rlcohol, olhersùbsLaneabusc.orcornorbidtryProbleús.
^il det. were enteredard processedby EPI lnlo (versionó) soft
warc oflhe U.S. CenLeBfor DìseascCoùîrol and Pfevenr,on(Deùú
el rl.. 1994).^nalrses lrindrily iúclùde testsof sigrillcanc! tbr bodr
dìsfete fchi square.Fishcis cxact te\ls, rnd Òddsntios (ORt fof
2x2 con,rari\ons ùiú 95% corfidcnce intervàls calcùlaredby thc
CÒrnlìeld rechdqùrl aùd contiNou! la.i.bles (/ rest! and Bhlock
dirùcrcc oiprofoùiDs tesLs)(Bldock, 1972)thai co!ìfaresuliecrs
rvùh corLrÒh. Zero oldd PclNón corelaLioús ùre prolldcd in
Tlbles 4 and 5. No adjùstnrcntswcrc nrade lor nùltiple cohParl
sons. Orc s.y andijsis ol lariance $as lscd to rùsl relalionsùìts
bctwccD 3 s.Òu])! h l..blc 2. aNl lor coúelati.1n signincaùccnr
Tùbles 4 ùnd 5. PaiNise fost lìoc analysesofbciNecn group dillt.
enc€sRere usedùriuznrg/ tesrs.
children in the ovcrall sample,lhe 22 childrcr diagrosed
controls.T1ìe
\rith an FASD, andihe óTrandoniy selected
chjldren
are
inclicatedin
diagnoses
of
the
FASD
exac!
Fig. l:4 childrenhadFAS, l7 hîd parlialFAS.and t \ùith
presertedin Table I arc sìlnilar
ARND. T1ìeI aggregalcs
in sex compositionand age.as 45 to 5l% of all groqrs
w€re ìÌale, nnd the mean agc fof Lhe3 groups was 80
montlìs(6.7yeam).Fùrlhermore,therewèsno appreciable
differencebclllccn total saDplemensurcsol growlh and
the coùrol group, indicalirg thal random selectionol'
controls produced r representalivesanlpLe.Owing lo
crite a, thc.c are signifìcantdiffer
adhefcnccto screening
cìrccsbetweenlhe childrcnwith FASD and controìsin lhc
followjngparàacters:heiglìt.weight,BMI pcrccnlile.and
(OFC).Iù thediasnosticprocess,
pool
headcìrcumference
gro\lth, sm. l headcircu!Ììference,
slìort palpebrallissures,
and/or fealùresof a hypoplasticmidfaceseùe to differen
tiate FASD subjects fioln non FASD children and
indjcate rjsk of mcntal deficiency. Irdjvidual lacial
feiìrul'eswele also found to be significantly diff€rent
bclweer groups.Pnip,rbrallissurelength,philtral l€ngth,
prosis.epicanthalîolds.antevertedrostrils,longphiltrun1,
smoothphiltnnn, and narrolvvcrnilion borderare all sig
the FASD group anclcontrols.
nificîntly clilfe.cn1betweer
The highesrORs for îaciai lèaturediffcrcnceswereupper
1ip l:cùtrùes:smooth philtrurn (OR:85.7) and n3rl.o\\,
vermìlion border (OR: lll.6). Other niror struclu.4l
anoDralicsllral difîerentìatethc 2 gfoups are as follo$'s:
raiÌroadtuackcar conigrmlion, camptodactÌy,and aìlcf
ation oî palmar cfeasesil] the children \\ith I-ASD.
Nonstatisricaliy significanl dìferences between FASD
subjectsànd conlrols w€re rìs foìlows: slrabisnus, heart
rì1urmùr,limited elbow sWination (p:0.06), olioodac
tyly, ànd gcneralclinìcalobserrationsofpoor 6ne motor
coordiíìa1ion.hypoplaslicmidface.and prognatbicchin.
The ncan total dys]lorphologyscor€rvrÌssigri ficàt1tlydifferent(/<0.001) lbr thc FASD group (12.5+ 3.9) and
controìs(3.3+ l-0), indicarirg, as prcdìclcd by tìre diag
nof;c process,substanlìallymore dysmorphicleatufesin
rhe FASD group. Orerall, 36.4% of t]1c childrcn diàg
nosed$'ith rn FASD had all 3 of the key îacial features
coúr]ììonlys€enwilh FAS (50% oî the F^S ànd 35.3%of
drePFAS children).
Devloltne t 0k! Rehúri.)i
Ir Tîblc 2. dcveloprÌentaland behalioral test lìùdings
are summarized,in addition 1(ra summaryofmxtefnal agc
and sclectedrìlaternaldrinkìng variablcs.The study chil
drenweredividedirlo 3 groups:2 F'ASDgroups,(l) thosc
tust prelinrinarily diagrosedasFAS and (2) lhoseìn whom
diagnosiswasiDiliallydeferredbut who laler convcrtedLo
à diagnosisof F'ASD, and (3) lhe controls.Of thosechìl
dreù wllh a preliDiDary diaglosls o[ FAS, 54.5% wcrc
ofdelìcienigrowthor sùall
for referredintolhe slud)'because
ln labte 1, the demographicand growlh paralnete|S
stud] children Àre pfesentedfor 3 categofies:all 543 hcad size.another3ó.4% of rhis gfoup were relèrgd lor
(95%Cr)b
22f
50.0
H6ight(cm)è,Med(sD)
Weisht(ks)",Mean(SD)
childen's sMl', À4ean(sD)
BMIpèrcenliè', Mean(sD)
occipilal circumference(cm)", r',!éan(So)
length(cm),lvean(SD)
Palp€brallissurc
Phi|tlumlenglh(cm),Mean(SD)
Short inner€nlhal distan@ {< 25%)
80.4(4.4)
(5_4)
121_5
16.9(2_8)
6 1 . 8( 3 1 . 4 )
52.0(1.5)
80.1(4.3)
116.2\5.2)
22_O14.4)
16.212.4)
52.2(33.9)
50.7(1.3)
2.4 \0.1)
1.5 (0_2)
14,2
55.2
79.9(3.4),
121.4\4.51.
25.5{4.5)
17.312.5)
69.6(23.1)
5 1 . 9( 1 . 1 )
2.5(0.1)
1.4(0.2)
Fine moior dysiunclion (7.)
Hypopldtc midlaco(%)
27.3
0,0
11.9
'Rd@d
22.7
6.0
rrack ea6 {%)
3.0
NS(0.670f
NS(0.80îd
NS(0.o7zd
0.001"
NS(0,1,|8f
NS(0-067'
NS(0.230f
13,6
14,9
36-4
9.0
0.002'
0.023'
68.2
0.001c
0.0
0.0
0.0
1.5
Umitedelbowsupinalion(qÉ)
0.0
13.6
0,0
0.0
3_0
NS(0.060f
clinódacryly (%)
31.8
26.9
NS(0.654f
22:7
7.5
0.05'
45.5
19.4
0.015"
0,0
4.5
0.0
9,0
NS(0.505f
Heart maliomalions (9.)
PalmarcÉas€ allóralion (%)
NS(0.564f
rchi-squaredlesl ol dala compaingchildrenwith FASDandconlrolsia Fishsasexacltest whenllrereafe celb wnhan expeted valueol <
inìhesludy,(2)Iorcompason,and(3)whendiasnosis
wasmade.
sexandused.(1)whenconsiderins
inclusion
ofchildr€n
'li,loasuremenls
atlimèof Tierlscreen;lhefelore,lhey
aÉ direclly@mparable
lo allolhergroups.
:
both sizeand behavioral/leamingproblems,and < 10% becauseofgroMh or OFC dofrciencyatrd al1the remaining
were rcferred for behavioravleaming problems only. In 81.8% werc rcfeffed by teache for learning/behavioral
the prcliminarily deferredgroùp, only I 8.2% were referred próblems. On language coÍìprehension (? = 0.009),
'1
IndiÒaìÒrsa
and Behavioral
Table2.Gen€raDevélopménlal
Lano!aoecomD.ehènsion(
51.8(19.7)4
5.3 (3.3)
10.4(3.4r
score
lótal dysmophorogy
À4ean
dri.ks percurèntweèki(sD) .
Meandrirks percureridrinkinqday'(SD)
3.5 (2.1)3
58.2(21.5f
1 1 . 7( 6 1 f
r6.2 (26.6)4
2.6(3.6)1
7 . 5$ . 4 4
1 . 5( 0 . 9 r
'R!stioni QùallaliveTest.
nonverbal IQ (p:0.005), and behavioral problems
O<0.001), lhe 3 groupspeform differentll. The children
prelinìinarjlydiaguosedas FAS had the worsi scoreson
ÌanglLageand nonverbal IQ, while the prelin'narily
def€ffed FASD children h.rd the most behavioralprob
lelns.Overall,the 2 FASD groupspeforded wolse than
conifols on àll standardlests.Post hoc analysisof inler
group diffefencesirdicates thal thc prcliminary FAS
groupsand the initially delerrcdgroupsdifferedon behav
ior problems,a fufther indic.ìlor thrr lhis later gfoup
includedmost of rhe beh.ìvior/leamjngproblem retèrrals
fionl teachers.Botb FASD grorps differed significantly
from contfols on all devclopìncntalmeas!ìreswith tlre
cxccptionol'behavior.Both the prelininary FAS childr€n
and lhe coDlrolsarc betlcr bchavcdthan lhc preliminarily
deferredchildrenwlro were latef diagrosedas havingan
FASD. Iotal dysmorphologyscoresfor eachgroupform a
spectrum(14.7+1.1, 10.,1+3.4,and 3.3+3.0) tìolr1
preiimiìary FAS lo contl01(p<0.001). Posthoc analyses
in scoresbetweeneach
alsoi dìcÀ!esignificaÍt differences
group.
In the second part of Tablc 2, the rnatcrnal agc of
the 3 groups displaysa conrinuum.The morhefs of the
prcliminary group wcrc the oldest (.tr= 32.4+ 5-2), lhe
prelimirary defeÍed interÍnediate(X: 31.1+ 3.2), ancl
the controls thc youngcst (,Y = 29.7+ 5.7) at delivery,
alrlìoughthe differenc€s\r!'ere
not stalisticallysignìfican!.
In gen€ral,lhc currcnl drinkìng reportedby the mothers
olihc clildrcù iD theseI groùpsalso exhibirsa spectrum
that mirrors the FASD v€rsus conlrol findings. The
nean number of drinks cùrrenily consurìed per week
(a! interview) by molhcn oI children dragnosedas
preliminaryFAS (16.2+ 26.6)€xce€ds
rhat ofthepreÌìm
inarily defered chiìdren(7.518.7) a,rd the controls
(1.5 + 2.0), ù1d the standard deviationsvary greatly in
the 3 groups, bejng the highest in the molhers ol lhe
children eventually diagnosed with an FASD- Binge
measures(mean drinks per currenr dfinking day) were
the hishesi for lhc prcìiirinary FAS group (2.6 a 3.6).
Post hoc t t€sts indicated that both FASD ìnalcrnal
gloups were signifìcanllydifferent than conrrol mothers
on both of thesevariables.Bulitis interestingto nole that
only 44% 1o50% ofthc molhersin any olìthese3 groups
reported drìnking at all dufirìs pregnancy (once they
knew lhey wcfe pregnanl). Il \ras the impressior ol the
inte iew€rsihat the veracityof ihe reporting olpremtal
dl-inkiÌlgwas questioùableîor sone wonleù. For exam
ple, ìn 19% of the inteNiews of the mothers of FASD
childr€n, the blinded i terviewerscheckeda box ihat
I n d r c a r c\ rd. f i c i o n l l b ó u r ' h| e| I r h t u l n p . s , r) e , l ^ n . e . .
Denogttrthir, Socioeonanh, a d Mate lal Dthlhtug
hì Table 3, socioeconomic
and drìnking indicatorsfor
rìothcrs of ciildrcn vr'ilh an FASD arc comparcdwith
thoseof controls.Demosnphic and socioeconomic
indi
cators for the 2 naternal groups\vereanalyzed,and the
srÌmmaryin Table 3 ìndicàiesvery lillle differeúce.M€àn
age, rur.ìl/urban residence,frequencl of church allend
aÍìce, rcligious attitude, and employnÌent weÌ€ not
or in
sigìfificantlydjfîerent in the highligited anaLyses
Meanagè (y) on dayof nredièw(sD)
ReligosityIndex [4ean(SD)
AmongìhÒsèemployed,holrs ol woii pefweek Mean(SD)
37.9 (5.3)
36.6(s.3)
NS(0.636f
29.4
20.6
NS(0727f
23.5
3 . 9( 1 . 7 )
s88
19.0
2.4\1 7)
59.7
0.0
3 0 . 3( 1 16 )
2.7
271 (9.s)
NS(0.422f'
NS(0.305r
ci)o
Vaue,OF (95o/.
cLif entdrinkel (or everdrinkers)(%)
rvleannlmber otdrinkslasìmonlh(c!rcnl dtnke6')(SD)
Percefidrinking3 mo beiorèindèr prèqnancy(everdrlnké6)
41.9173.7)
durng ndexpreonancy(everdrirkers)
Percenldrinking
F Gi alnesler ol pregnancywithindexch ld ('n)
SecondandlhirdtrimesterolpÉgnancywithindexchid (%)
1000
e.0(8e)
47.5
NS (0.ffi3)bOF
69.2
64.6
NS (0.754)bOR
50.0
37.5
333
, B
N S( 0 . 7 9 1 f O
N S ( 0 2 8 4 ) bO R
NS {O18lb OR
clr€nrsmÒker(orlhosewhoeve. snoked)(%)
NS (0.372)b.
25.0
Percenlsmoked3 nÒ bèlore ndexpfegnancy(ahÒngeversmokersl
Perc€riusedbbacco duringind€xprègnancy
{eversmoke6)
90.9
37.5
;
"Caiculallonsofchi-sqLarèbasedoddsralionotposslbletorlhsvarableasnsnota2x
ioonsumedacoho in 12 monthsprecèdingnlerview.
Thereúas a signiiìcantdiffer
othef valiablecomparisons.
,ìttainment.
lhe mothe.s of FASD
ence i educational
yet, overaÌl thcl arc
somewhat
bimodal;
clildfen being
lesseducatedlhan conúols.Aìso, Ìnothersof FASD chil
dfer rcporled signilìcantlyligher church attcndanceand
!o religìonasrcflectcdin thehighef
morcposiliveadherence
religiosily index scofes.Direct confirmatlon of drinkìng
$'asrot available1ìom inicrviewsfo.9 ofthe 2l nrothefs
of L-ASD chìldren.Four wereludged to be sùspeotand
inaccurateby blìnded iiterviewers,and 5 lverc n1issingusefuldatx w€reobtainedon the remajnrng
Neverrh€lcss,
l2 ofthe mo1he|sofFASD clildren, tLndîìl but I ofthese
molhers
reporlcdthat theywereculìent drinkers.Overaì1.
of chìldren\vìih FASD reporl dfìnkìns 42 d,nrks in the
morth beòre the interview (curreni drinking) conlpàred
with a significantly loiver average of 8 for conlrols
rcporiedfor the3 months
(tr:0.007). Drinking prevalencc
before the index pregnancydid not differ significantly
groups(91.7%vs 87.5%),ùof did reporreddriúk
be!ù,een
ins during presnancy (69.2% .!s 64.6%). Mothers of
F A S D ( r i d r e n \ r J r ( n r o r el l c l ) l o e p o í c o D , u r n i n g
alcohol i all trinesters.€speciallyir'ì tfimesters2 and l,
Snok
aÌrhoughnoneof the dillèrenc€sprovedsìg,rifìcant.
iig varìablesdid no! dìffer significantlybetw€eÌìmalernal
srbiectsand conirolsoverallor f-orany timcslcr.
Detel.phlentúla d 4,el1orphalag!Med! tesi|1RcLltionîo
Table 4 presentsPeanor correlation cocffcients for
devclopmeÍìtal,ìnddysmorphologylraits associaledwi!h
selecrecl
nlalcrnaldfinkirg lncasùres.Few of thc bivaiate
corrclalionsof drÌnking and specificpsychologicaland
developnentalneasùresarc significant.The ooe notable
exceptionis lhe Pelhaminaltentiorìscore,\\'hichis signifl
cantly associatedwith all drinking variablesexcept fof
currcnldrinks pcr drinkingda]. Howe\'€r,all ofthc drink
irìg neasures are signilìcantly associatedwìth total
dysnìorphologyscoreexceptfor dfinkìng dudtg the fircl
ùimester.Ddnks per monlh in secondand third trimesteN
and curcnt drì ks per d nking day are all positivelyasso
cjaLed0 = 0.25 0.27,/,<0.05) with high dysmofphology
scol€s.Thc highestcorfeìalionis betwe€ncuncnî d nks
score.
pèrmonth(f:0.32.1< 0.01)anddysnìorphology
border'
Naifow ve.mirllÒn
"Baircad'€ars"
Saabism!sc
Ep canthajolds'
Limled supnationol elbowsc
Pa ner creasoalleÉtÌons"
l f e p e s " n . e o l 5 È l ' a [ F q ù d r rI d n d i t è d b < é . c è F o L a- 0
Maternalanl ChildCharatteristí(sin Rahri.tl ro Childs
NontetbatlQ
Furlher data ìn Tablc 5 coÍe1ateselectcdÌnatemaland
IQ No e
clitd phlsicàlvariableswitlì the clild's noDverbaL
variables
a d
selected
lnate.nal
of the correlationsbetween
variables.
for
tlÌe
children's
HoNclcr,
IQ afc sìgnilìcant.
(r = 0.25),snoolh philtrùn (f - 0.29),
headcircrlllllcrcnc€
0.:,1), plosrs
ù raìlroad irask eù configuration (,:
0.22),and
(r': -0.24), limited supìnàiionof elbows(f:
iotal dysmoìphologyscore(, : 0.26)aresignificantlycol
r e r r c dN ' , h , h e c h i l J' , o n r c , . , .llQ . a l . e l | n e . r . r , i I
FASDdiagnosis.However,noDeoftlìesclrais, whentaken
jndivìd ally as zero ordcf correlttions,expl morc than
Table4. PearsonCorrealiÒnCoeflcenlslor Developmental-
Ònb
Lanquagecomprehens
o.23"
0.24
"FavenColoredMat cès.
0.31"
0.31"'
a.27'
0.25'
0.32"
5% lo 8% oîthe variancein a child's onverballQ. Larger 16 cascsof FASD are esliÌnatedto existamong thoscùot
This resultsin aù overaliestimatedFASD ral€ of
screened.
sa pleswould alloN ]nulliplecorreÌationstudies.
per
34.9
i,000 children,or 3.5%. We believethat tlis rute
is likely to err on the high side for thc rnofe severediag
noses(FAS and PFAS) if oneassu esthat th€ activcciìse
Table 6 preseìltsprevalence€stimateslbr vaîiouslevels ascertainmentmethods,bolh growlh and re{errals1ìom
ofFASD in the Italìan studypopulation.Overall.the raie teachers,may haverecrùiteda high proporlion ofthe chil
of FAS an.l partial FAS exc€ededoùr expectationsand dren who werccandìdàtesfor thesediagnoses.
also cun€nt estimatedrates fbr lhe United Staies.Esti
mat€dralesol F:ASDdirìgtosesare presentedin 2 waysir1
Table 6: ratescalcùlaiedon tlÌe basisol the samplecon
One of the greatestlimitationsto the prevalencecalcu
seùting!o partrcipale(r: 541) and also projectedto ihe
lotal nrunberofclildren in thefirst gradeclassroomsfronÌ lations of this study, aDd al1 active case ascertùinmeni
which the sanple rvasdruwD (,t:1,086). Thereforc.thc siudies,is theconsentrate-The ùct that consentto partic
fale of FAS overaìlis belween3.7 and 7.4 per 1.000chil ipatc was obtaircd for only s0% of the children ir the
dren. Parlial FAS is betwecn 15.7 aÍìd 3l.l per 1.000 randomly sclccledschoolsintroduccspolential bias for
children.The overall rangeof rates of FASD (nlcluding $'hichi! is d'îficult to accounl.This hasb€e a pÌoblemìn
thecaseofARND)ìs 20.3to40.5pcr1,000children.lfone U.S. stùdiesas well, as therejs frequentlya reluctanceol
excludcsthe casesIor which naterÍìal alcoholirtakc could guardiaDs.cspeciallybirth parents,to p.ovìde consentfor
not be confirmeddirectly,the observedr.ìtesofFASD are an eraminationofihei. child lbr FASD or other develop
slightlymorc thùn half (55%) of lhe aboveràtes:overall, ncùtal issues.In fact, becaNeofthe reluctar€eofparcnts
2.8105.5per 1,000for FAS;.ìnd7.4 and guàrdians,nìost active case ascertatnmel studles
11.Ito 22.0per 1,000ì
have avoidedin school studiesof FASD, relying iDslead
per
PF,A.S.
to 14.7
1,000
'Iabl€
6 assumethnt the on large oùtreachreferral networks wìlhin and between
The lower prevalenceratesin
2 methods of àctive rcouitment for the sù1plt to be publjs hcàlth and educatioDalsyslems.The one in school
(gro\rlh or OFC < l0% and/or rcferralfor learn studyin the United Statcs,in a countythat requiredaclivc
scr€ened
ing or behaviorÀÌproblems)capturcdall candidalesîor an consenlliom parenis,was only able !o recruil <25% of
FASD diagrosisamong the 541 childrcn who had con rhc children(Clarrerìe! a1.,2001).We have nttenìptedi
senledto parricipatc.Thc higher rates assumethc polar this study to correcrthis poteDrialskelvìDgol th€ data by
opposjtcrlhat the activ€recruìtmcntofcasesby ihe school scvcralmetlìods.Fiff, somcchildrcn,in addìtionto those
officralswasnot at àlìscleclive.and a child rot jn thc s1Ùdy refefr€d for physical growih alrd devclop enl, were
was oo 1'I1orc
or iesslikely io havcFASD as those5.13who refe ed tbr academicof behavioralproblems.Consen!
did paìticipale.The lruth may lie in the middle.Oî tlre 69 for rhese speciallyreîerred children was high irl most
ofteachers,admìnistra
Iandornly selectedcoDlrol/corÌparisonchildren who hàd schoolsbecauseof lhe persìslence
pormissioù1()participate,and wbo wcrc exaniied by blinded tors, aùd researchtearnmembers-Sccond,lo accountfor
dysmorphologyteams,2 were ùltifìaiely diagnosed\ìith possibleselectivity.we haveptovideda rangeofrate esti
an FASD (l FAS and I parlial FAS). Therefore,2.9%of m r r e . a r l . e r ' h nonn ed e f .r i r er J t € l. - i n r l l ) r. h e f o n u Ir o r
childrenÈom the consentjrgparlic of children found !o have an !-ASD in the randomly
the randomlyselÈcled
ipantpool bad an FASD. Projectinglhìsproportionro th€ selectcdcdsesliom dìe participantpool was used10 esli
543 childrer Dot parlìcipal;ngin the study,an additìonal male a sìngl€ mtc fof lhe sanple. Thercforc, we have
samplè'
enli€classÙ
samplè"
enlireclassb
provided high (sampleonly) rnd low (tolal enrollmert) clilúen with subslanlial prenatal alcohol exposure
growthand deveL
iì the \\orld. Their suppressed
rates of pre!.ìlencealong ivith a \jngie eslimate(35 per elsewhere
ol bolh probabi opmenl. depressediniell€clrÌàllunctionìng,xnd behavior
1.000)that ùa) combineîhe advanteges
lislic screeningand raDdomsÈlectionofcontfol subjccLs- problenrsarc similar to thoseiclenriîedand dcscrìbedjn
WhiLeit is diliìcult to compareilìc ratcsoflASD tound subpopulationsof ihe Unircd Shtcs and South Afiica.
Their height.weighi, ard BMI fèrcenlile weredeplessecl;
in this populaLion\iith other studiesin dèvelopedcour
the
most
shoft palp€búl nssuresand hypoplasticmìdl.lcìrl fea
rfies,rhe ratesofl-AS and PFAS wefeligh. Even
.
r
u
d
)
.
.
llre snooth philtrum and a
I
e
'
c
e
<
d
turcs \ìerecolnmoD,especiÀlly
c o r \ e r . - u \ . . , i n . r e o " a ' e .f r o r , l r .
prevalenceof hand delecls
The
narrow
vefnilior
bordcr.
fronì
the
United
Stalcs
esriìnalcsfrom clinic basedsludics
(Abel,1998;Sanpsonctal., 1997).
Forexalì]plc.
Sànrpson as also sìnilÀr 1(ralcolìol usiDgmrtcrnal populalions
Tbe intcllcclualperfornìancexÍìd pfob
er al. (1997)erimatedthe fateoI FAS and AIìND lo be studiedelsewh€fe.
l% in the U.S. pofulalìoÍì. While $'e uscd a differeúl. lematicbcha\riorof ltaliar cììildrenNith FASD lormed a
updrÌted, and Iìkely tnore sensilìvcdragnosilc scheme spcctr rn that was corfelatcd with Lhc level of curent
(Hoynreer al-, 2005)ì. Ilaly. ùe hav€fourd th.ri tbc raLc drirÌking feportÈdby lhe nroLlÌerard L1ìeselerity of lhc
oIFASD may be 2.0%to 4.1%(20.340.5pcri.000).Tlìe child s dysnorplìology.Iralìan childrcn with FASD lvere
F^S only Ool FASD) ratcsiìre,howevel.some\rúatclose rìgniiicantlyùore delicicntin verbàllQ and nonveÌbalIQ
to th€ 3.1per 1,000reporledby ChrreD et al. (2001)in the and more pfore to behaviorrl problemsthtìn controls.
irì sclìool!tudy in WashlngtonStni€ol the tjnited States. Onceagàin.the dala frorìì tÌis study fajscthe qùestior of
areproofofa sub
Yei, they are lnuch lo cì lhan the ln school samplcsof whetherhiglìlevelsol cufren1drìnking
SourhAliican childrenof46 ro 75 fr l blown FAS c.lses stantidlprenalaleffeclon rhe clildì behì1,ioror wlìether
pÈr 1,000(May et ., 2000: viÌtoen et al-. 100i). wheÌe tle postnaial environnent is mosl irîportîni. WheÍì
poor nutrjtioD.po!erly. blÍìgedrinkiDg,and olher faclors combhed \vitir lhe dlsnorphic feaîufesdocunìertedjn
childrcn dlagÍìosedrvith an FASD. Ihc prcnatrl cflèct is
colnbil]efor extrenelyhigh rulcs.
ria hous€hoìd
Our fìrdingsÍaisethe substantjaiqucslionas Lolvhelher evideÌìr,bul poslnalalbehaviofaiinffuences
popuÌatioù,
rnporlaoi.
In
this
Italian
afi: also
FASD prclalcnce is accufaLelyreporled or cstrmatedir conditions
but social
'ras
no
siglìificant
varjatior
by
SLS,
the United Staresof in îny Wesle.n Europeancounlry. tlÌcrc
floù
làmil]
to
tanrily
differed.
lhe rate of FAS basbeenestimaledrecentlyas 0.51()2.0 behavlor
ìn the sclìoolsofSouth Africa,lhc
(Sr.atlonet a1.,1996)of 0.5 to Li (May a,ìd (]ossage. As in p.cviÒussLudies
definitely led úe bLlÍìded
quilc
àrise
diagnosiic
dysmorphology
estimat€s
may
be
low,
as
lhcy
2002).Tbese
prinarily from passivecaseascertainmenlstudies.Fur reseafchlcam lo childf€nwbo had behavioraland lealn
lhÒnÌore. the eslimaiethat FASD nìdy alfecl l'70 ol tlle lng pfoblemsaÌìd more inportantly !o rÌotlrers who had
U.S. popuìrlior (SaNpsonet î1., 1997)or anir developed substrntialissuesofàlcolÌoluseînd comorbidjr),.but once
popllalioÍì may alsobe substînliall] low. ns lhìs in school again, :ln episodicpattern ol hcavy drinkìÙg sccns to
studt jn Italy prolides esdmatesof 2% to 4%. Marìy .nrcrge ro dillerentintethe mothers of FASD childf€D
aullìofs hîve suggeslcdlhal FAS and olher I'ASD are f-romcontrols,although c!ìdcncc ol e|i$dic d.ìnkìng is
lìigh rntesoî lessir1this llalian populationth.Ìr ir othef popuìatìoDsit
u'derreported,and studìeshavedocumeDted
llrc a.reragenùmlrer
undiagnoscdcnscsin sei,eralcounlfies (Clarrcn er al.. \Àhichwe halc workcd. Ncvcrthclcss,
200ì; Duimstfaet al.. 1993;Lclcrshaand lvlarks.1995; ofcurrenl ddrks pef \\eek reportedby Italian rlloilìersolto lhal rcporlcd
I-irtic c! al.- 1990;Kligne et al., 2001;Squafe.l99l). FASD children(16.2)is slfikiùglrsill1ìlaf
Therefore.our corclusion is rhat FAS aDd FASD afc by Sourh African women who have had IAS clildren
probÀblynore common in llìe $,estern,developedwofld (16.1aÌd 11.6)(May er al.,2005;Viljoeùer a1..2002).
ln
L|an currenLl] estinìated.In sùpporr of this corchìsion- generd.eveDthouglLabout thrce f-oufthsotÙÀterndj con
Clarfen et al. \rfole alle. lhcir ìn schoolsludy: none o1 tfols aúd 100% of the $omen who have ever coÍìsurned
r1ìelÈASl childfenhad beenideDtilieclitl ihe Washìnslorì alcoholreporteddrintìng in the past yeàr.dajìy drinking
Slale Regìslry.Jú ouf opinioù.tlone ofthesecasesoî F^S in this part ofÌt. y seenìsto be iesscomnlon lh.ìr we sus
would hî'"c beeninchded in.ìny passivesufleillîrce study pected.But to a greatefdegfeethan SourlìAiijcaD\\omer,
rcporllngthe prc\'al€rce
FAS ... . " ODlylhroughaddi Itrliîn sLrbjeclsreported fewer birg€s and dso seerned
discrLs
siudiesof FAS and otlìer Inofe chaìlcngìngto cngagenr liank .ìnd accur.ì1e
lioDal rclive ùse asceftainment
ofd.inkingdurirg thepfcnalalpcfiod.
È=ASDin tlìe United Stàrcsand wcstcrú Eùropecan the sior'ì
qucsiionofthe tÍùe prevaleÍìce
oîFASD be answer€d.
Whìlc somc sludiesof natemal driiking in ltah lìave
rlot linked materDatdrinking to màjor advcnc lctal out
(Lazzaroniet al., 1991,1993n,1993b),two other
corìres
li.tits ofChikúcn Iríîh F,,lSDii ltúb Rclutcdto Mate l.tl
rudies of prenalal diÌrking ìn Italy have reportedsub
Dri khs
st.ìnlialL)highcr lclcls of dnnkhg during pregùancytlìan
The childrenrÍì ltaly identifiedas haÌing an FASD ncet we Ìbund. l lralìaD hospilals,29% ol woNen rcportcd
the revisedIOM crirefia that we have ìrsed !o idenlify dfiiking daily lhroughout pregnancy and l% rvere
final case coúference in which aI diagnoses were com
pleted.In Italy, many peopÌehaveassistedininitiatingtheFoject. Luca Deiana, Luciana Chessa,M.D., Michele
Stagagno,M.D., and Agatino Battaglia,M.D., were all
instrumental ir hosting us aDd participatrng in tìe eafly
rraining and screeningof children. Maternal inteniewers
ì,ere outstandingin locating mothers and interviewing
them: Lucia Cupelli, Irene Di Stefàno, Marcella Scamp
oÍino, Anna Maria Galli, Federica Cereatti, and
FrancescaDe Rosa.We woùld also Iike to thank Stefano
Giacomelli,who coordinatedthe organizationofmate.nal
irrervìews and was very supportive dudng tlìe study ìn
many difîerenl ways. We also thank managers,school
physicìars, and psychologislsfrom ASL RMG' and
RMH** ftom whom we rcceived àssistarce: dotl.
P. Trccca,*dott. C. Carapeilese,'dot!. Di Giovanni,doii.
G- Ver-sace,**dott. V.De Carolis,** dott. N. Roma,**
dott. C.D'Anna,** dott. ssa L.Asci,*+ c.cironda,**
S- Gagliardi,** and A. Ponlecofli.** Thosewho assist€d
from tlÈ School Offic€ of l-azio Region* and Rome Prov
ince""were:dotL.ssaL. Signori.*dort.rta R. Mrs(acesj.*
ànd dott. ssa M.T. Silani." Finally, we thank dott.
F- Valeriani from SIFIP. FinàlÌy, in addiiion to 2 ol lhe
artlors of this paper @adela Fiorentino,and Giovanna
Coriale), the pyschologicaltestirg of the childrer was
carriedout witl assislanccfrom FnncescaDe Rosa and
Codnna Ceoldo.