Dissertation (PhD)--Stellenbosch University, 2003. / ENGLISH ABSTRACT: Introduction: Fetal alcohol syndrome (FAS) consists of multi-system abnormalities and is
caused by the excessive intake of alcohol during pregnancy. The teratogenic effect of alcohol on
the human fetus has now been established beyond reasonable doubt and FAS is the most
important human teratogenic condition known today. The syndrome, first described by Lemoine
in1968 in the French literature and in the English literature by Jones and Smith in 1973, has since
been corroborated by numerous animal and human studies.
This study has grown out of several epidemiological, prenatal and infant studies in areas of the
Western Cape that are currently being undertaken by the Foundation for Alcohol Related
Research (FARR). Preliminary data from studies in Wellington have confirmed that a significant
proportion of school-entry children have FAS. The prevalence ofF AS in this community exceeds
that for Down syndrome by a factor of30 times. The frequency ofFAS in high-risk populations
of the Western Cape is the highest reported anywhere in the world. With this background, and the
paucity of FAS literature related to dentistry, the aim of this study was to determine the
craniofacial and oral manifestations ofF AS in a sample of school-going children in the Western
Cape.
Methodology: This study is a descriptive, case-control, cross-sectional study using a random
cluster sampling method. On the day of examination, children were weighed, and their height and
head circumference were measured. They then had photographs and radiographs taken, followed
by an oral examination. For each child, the following information was recorded on the data
capture sheet: date of birth, gender, head circumference, weight and height, enamel opacities,
dental fluorosis, plaque index, gingival bleeding index, dentition status, oral mucosal lesions and
dentofacial anomalies.
Results: The total sample of90 children with diagnosed FAS and 90 controls, were matched for
age, gender and social class. There were no significant age differences between the two groups
(p=0.3363) and the mean ages were 8.9 and 9.1 for the FAS and control groups respectively. Head circumference (HC) differed significantly between the two groups (p<O.OOO 1) and the three
photographic diagnostic measurements were all influenced by head circumference. The
prevalence of enamel opacities between FAS and controls was not significantly different and
averaged around 15% for both groups. The opacities were found largely in the maxillary central
incisor and lower first molar teeth. More than three quarters of both the cases and the controls
demonstrated the presence of plaque and almost two thirds demonstrated gingival bleeding on
probing. FAS patients had statistically significantly (p<O.OO 1) more dentofacial anomalies than
the controls. The mean dmft score for the FAS sample was slightly higher, though not
significantly different from that of the controls and the decayed component (d) made up the
largest part of the index in both groups. None of the FAS children had any missing or filled teeth,
and in the case of the controls these were also rarely found. Thirty nine children (21.67%) of the
total sample were caries-free.
Discussion: This study represents one of the largest sample sizes documenting the craniofacial
and oral and dental manifestations of the FAS to date. Forty two per cent of the FAS sample
manifested growth retardation and this was statistically significant (p<O.OOOl) when compared to
their controls. Analysis of the face using anthropometry supports many of the previous clinical
descriptions of the effects of neonatal alcohol exposure and offers some new perspectives on the
FAS facial phenotype. The characteristic dysmorphic facial features found included ptosis of the
upper eyelids, epicanthic folds, short upturned nose, thin vermillion border of the upper lip and a
smooth philtrum.
Overall the analysis of the caries data for this study in respect of differences between cases and
controls was found to be unremarkable. The lack of difference in the primary and permanent
dentitions between the cases and controls could have been anticipated in this population due to
the high prevalence of dental caries among children from the Western Cape. The FAS children
showed significantly lower dental ages when compared to the controls. Dental maturation has
previously been shown to be mildly, but consistently, delayed in children with delayed
development and therefore this is a not surprising finding for the FAS children in this study. Differences between skeletal age and chronological age were noted for both boys and girls, but as
a whole, in the present study groups (FAS and controls) showed little variation in skeletal
development.
Measurements related to the face height and mandibular size appear to be the most important in
distinguishing the FAS children from the controls. Most (5 out of8) of the discriminating linear
measurements studied lie in the front of the skull area. Most of the discriminating measurements
are vertical measurements and only two of the measurements are lines between soft tissue points.
When comparing the photographic analyses of the facial features versus the cephalometric
assessments; the four facial features most typical of aF AS child had a Positive Predictive Value
(PPV) of92% and a Negative Predictive Value (NPV) of90% and the eight linear measurements
from the cephalometric analyses had a PPV of 92% and a NPV of 95%. One can therefore
conclude that the external facial features are probably more reliable in discriminating between the
two groups than the cephalometric measures. For further analyses, other models where a single
angular measurement explains a combination of linear measurements need to be investigated.
This might further improve the discriminating abilities of the cephalometric measurements as a
whole.
Conclusions: This study has shown the importance of the oral and craniofacial features ofFAS.
FAS can no longer be viewed as just a rare and peculiar childhood disorder. Awareness and
recognition of children with FAS is important so that they can be correctly diagnosed and
referred appropriately. Prevention of the secondary disabilities and most importantly, the
prevention of FAS in subsequent programmes can be planned. The dentist who treats children
with FAS must recognise that such patients might be emotionally and mentally handicapped and
may make treatment difficult and there may be a need for the child to be treated with behaviour
modification and/or premedication before restorative treatment. The dentist should also be aware
of the need for an accurate medical history, and possible medical consultations, before treatment
can be undertaken safely. / AFRIKAANSE OPSOMMING: Fetale alkoholsindroom (FAS) bestaan uit multisisteem abnormaliteite en word veroorsaak deur
oormatige inname van alkohol tydens swangerskap. Die teratogeniese uitwerking van alkoholop
die menslike fetus word nie meer betwyfel nie en FAS is die belangrikste menslike teratogeniese
toestand tans bekend. Die sindroom, soos aanvanklik deur Jones en Smith in 1973 beskryf, is
sedertdien deur vele studies op mens en dier bevestig.
Hierdie studie het gegroei uit vele epidemiologiese-, prenatale- en kleuterstudies in dele van die
Weskaap wat tans onderneem word deur die Stigting vir Alkoholverwante Navorsing. Voorlopige
data van die studies in Wellington bevestig dat 'n betekenisvolle deel van skoolbeginners FAS
het. Die prevalensie van FAS in hierdie gemeenskap oortref dié van Down se sindroom met 'n
faktor van 30. Die frekwensie van FAS in die Weskaap is die hoogste wat in die wêreld
gerapporteer is. Met hierdie agtergrond, en die skaarste aan FAS literatuur wat op tandheelkunde
betrekking het, was die doel van hierdie studie om die kraniofasiale en mondmanifestasies van
fetale alkoholsindroom in 'n monster van skoolkinders in die Weskaap te ondersoek.
Metodologie: Hierdie studie was 'n beskrywende, gevallebeheerde deursneestudie waarin 'n
lukrake gebondelde monstermetode gebruik is. Op die dag van die ondersoek is die kinders
geweeg en hulle lengte en kopomtrek bepaal. Hierna is foto's en x-straalopnames geneem,
gevolg deur 'n mondondersoek. Die volgende inligting is vir elke kind aangeteken:
geboortedatum, geslag, kopomtrek, massa en lengte, glasuur-opasiteite, tandfluorose,
plaakindeks, gingivale bloedingsindeks, gebitstatus, mukosale letsels en dentofasiale anomalieë.
Resultate: Die totale monster, bestaande uit 90 kinders met gediagnoseerde fetale
alkoholsindroom en 90 bypassende kontroles, is vergelyk ten opsigte van ouderdom, geslag en
sosiale klas. Daar was geen betekenisvolle ouderdomsverskille tussen die twee groepe nie (p-
=0.3363). Kopomtrek het betekenisvol tussen die twee groepe verskil (p<0.0001), en die drie
fotografiese diagnostiese afmetings is almal beïnvloed deur kopomtrek. Die prevalensie van glasuur-opasiteite tussen die FAS- en kontrolegroep was nie betekenisvol nie
en het rondom 15% vir beide gewissel. Die opasiteite is hoofsaaklik gesien in maksillêre sentrale
snytande en mandibulêre eerste molare. Meer as driekwart van beide groepe het plaak getoon, en
byna tweederdes het gingivale bloeding met sondering gehad. Die gevallegroep het statisties
betekenisvol meer (p<O.OO1) dentofasiale anomalieë getoon. Die gemiddelde dmft telling vir die
FAS groep was effens hoër, alhoewel nie betekenisvol nie, as die kontrolegroep, en die "delayed"
(vertraagde erupsie) komponent (d) het die grootste deel van die indeks uitgemaak in beide
groepe. Geen van die FAS kinders het enige afwesige tande (m) of hers telde tande (f) gehad nie,
soos ook gevind in die kontrolegroep. Nege-en-dertig kinders (21.67%) van die totale monster
was kariesvry.
Bespreking: Hierdie studie verteenwoordig een van die grootste monstergroottes tot op datum
waarin ondersoek ingestel is na die kraniofasiale en mond- en tandmanifestasies van die fetale
alkoholsindroom. Twee-en-veertig persent van die FAS monster het vertraagde groei getoon en
dit was statisties-betekenisvol (p<O.OOOl)vergeleke met die kontrolegroep. Antropometriese
analise van die gesig steun die vele kliniese beskrywings van neonatale blootstelling aan alkohol,
en bied ook nuwe perspektiewe op die FAS gesigsfenotipe. Die kenmerkende dismorfiese
gesigseienskappe wat gevind word, sluit ptose van die boonste ooglede, epikantusvoue, kort
opgedraaide neus, dun vermiljoen rand van die bolip en 'n gladde filtrum in.
In die geheel was die analise van die karies data ten opsigte van verskille tussen gevalle en
kontroles onopvallend. Die afwesigheid van 'n verskil in die primêre en sekondêre gebitte in die
gevalle en kontroles kon in hierdie bevolking verwag gewees het as gevolg van die hoë voorkoms
van tandkaries onder kinders in die Weskaap. Die FAS kinders het betekenisvol-laer
gebitouderdomme gehad as die kontrolegroep. Gebitmaturasie is in geringe maar deurlopende
mate vertraag in kinders met vertraagde ontwikkelings, soos voorheen al getoon, en is daarom nie
verbasend vir die FAS kinders in hierdie studie nie. Verskille tussen skeletale ouderdom en
chronologiese ouderdom is gevind in beide seuns en dogters, maar in die geheel het dié huidige
groepe (FAS en kontroles) min variasie in skeletale ontwikkeling getoon. Dit wil voorkom of afmetings wat verband hou met die gesigshoogte en grootte van die
mandibula die belangrikste is om FAS kinders van die kontrolegroep te onderskei. Meeste (5 uit
8) van die diskriminerende lineêre afmetings wat bestudeer is, lê op die voorkant van die skedel.
Die meeste is vertikale afmetings, terwyl slegs twee lyne tussen sagte weefsel punte. Waneer die
fotografiese analises van die gesigseienskappe vergelyk word met die sefalometriese
waarnemings, word gevind dat die vier gesigseienskappe tipies van 'n FAS kind 'n Positiewe
Voorspelbare Waarde (PVW) van 92% en 'n Negatiewe Voorspelbare Waarde (NVW) van 90%
het, en die agt lineêre afmetings vanaf die sefalometriese analise 'n PVW van 92% en 'n NPV
van 95% het. Daar kan dus afgelei word dat die eksterne gesigseienskappe waarskynlik meer
betroubaar is om te onderskei tussen die twee groepe. Vir verdere analise behoort ander modelle
waar 'n enkel hoekige afmeting 'n kombinasie van lineêre afmetings verduidelik, ondersoek te
word. Dit mag die diskriminerende vermoëns van sefalometriese afmetings in die geheel verder
bevorder.
Gevolgtrekking: Hierdie studie het die belang van orale en kraniofasiale eienskappe van FAS
getoon. Die toestand kan nie langer as 'n seldsame en eienaardige aandoening van kinders
beskou word nie, en bewustheid en herkenning van fetale alkoholsindroom pasiënte is belangrik
sodat hulle korrek gediagnoseer en op gepaste wyse verwys kan word. Die tandarts wat FAS
pasiënte behandel, moet besef dat sulke pasiënte emosioneel en geestelik belemmer mag wees en
dus hantering en behandeling bemoeilik. Daar mag 'n behoefte ontstaan vir gedragsmodifikasie
enlofpremedikasie voor herstellende behandeling. Verder moet die tandarts bewus wees van die
behoefte aan 'n akkurate mediese geskiedenis, en moontlik konsultasie met 'n geneesheer, voor
behandeling veilig ingestel kan word.
| Identifer | oai:union.ndltd.org:netd.ac.za/oai:union.ndltd.org:sun/oai:scholar.sun.ac.za:10019.1/53425 |
| Date | 12 1900 |
| Creators | Naidoo, Sudeshi |
| Contributors | Dreyer, W., Viljoen, D., Stellenbosch University. Faculty of Medicine & Health Sciences . Dept. of Interdisciplinary Health Sciences. |
| Publisher | Stellenbosch : Stellenbosch University |
| Source Sets | South African National ETD Portal |
| Language | en_ZA |
| Detected Language | English |
| Type | Thesis |
| Format | 283 leaves : ill. |
| Rights | Stellenbosch University |
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