Persson, M., Sandy, JR, Waylen, A., Wills, AK, Al-Ghatam, R., Ireland, AJ, Hall, AJ, Hollingworth, W

14 Pages·2017·0.27 MB·English

ORIGINAL ARTICLE
M. Persson A cross-sectional survey of
J. R. Sandy
5-year-old children with
A. Waylen
A. K. Wills non-syndromic unilateral cleft lip
R. Al-Ghatam
and palate: the Cleft Care UK study.
A. J. Ireland
Part 1: background and
A. J. Hall
W. Hollingworth methodology
T. Jones
T. J. Peters
R. Preston
D. Sell
J. Smallridge
H. Worthington
A. R. Ness
Authors'affiliations: Persson M.,Sandy J.R.,WaylenA.,Wills A. K.,Al-Ghatam R.,IrelandA.
M.Persson,CentreforAppearance J., Hall A.J., Hollingworth W., Jones T.,PetersT. J., PrestonR.,SellD.,
Research,UniversityoftheWestof
Smallridge J., WorthingtonH., Ness A.R. Across-sectional surveyof
England,Bristol,UK
J.R.Sandy,A.Waylen,A.K.Wills, 5-year-old children with non-syndromicunilateral cleft lip and palate:
R.Al-Ghatam,A.J.Ireland,T.Jones, the CleftCare UKstudy. Part1: backgroundand methodology
A.R.Ness,SchoolofOralandDental OrthodCraniofac Res2015; 18(Suppl. 2):1–13. ©2015The Authors.
Sciences,UniversityofBristol,Bristol,UK
Orthodontics&CraniofacialResearchPublishedbyJohnWiley&SonsLtd
R.Al-Ghatam,Dental&Maxillofacial
Centre,RoyalMedicalServices,West
Riffa,KingdomofBahrain Structured Abstract
A.J.Hall,Children’sHearingCentre,
Objectives – We describe the methodology for a major study investigat-
UniversityHospitalsBristolNHSFounda-
ing the impactofreconfigured cleft carein theUnitedKingdom (UK)
tionTrust,Bristol,UK
A.J.Hall,CentreforChild&Adolescent 15 yearsafteraninitial survey, detailedin the ClinicalStandards Advisory
Health,SchoolofSocial&Community Group (CSAG) reportin 1998,had informedgovernmentrecommenda-
Medicine,UniversityofBristol,Bristol,
tionson centralization.
UK
W.Hollingworth,SchoolofSocial&Com- Setting and Sample Population – This is a UK multicentre cross-sec-
munityMedicine,UniversityofBristol, tionalstudy of 5-year-oldsborn withnon-syndromic unilateral cleft lip and
Bristol,UK
palate.Children bornbetween1April 2005and 31March2007were
T.Jones,MusgroveParkHospital,
seen incleft centreaudit clinics.
Taunton,UK
Materials and Methods – Consent was obtained for the collection of rou-
tineclinical measures(speech recordings, hearing, photographs,models,
Date:
Accepted30August2015 oral health,psychosocialfactors)and anthropometricmeasures (height,
DOI:10.1111/ocr.12104 weight, headcircumference). The methodologyforeachclinicalmeasure
©2015TheAuthors. followed thoseof the earliersurveyasclosely aspossible.
Orthodontics&CraniofacialResearch
PublishedbyJohnWiley&SonsLtd
ThisisanopenaccessarticleunderthetermsoftheCreativeCommonsAttributionLicense,whichpermitsuse,
distributionandreproductioninanymedium,providedtheoriginalworkisproperlycited.
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
T.J.Peters,SchoolofClinicalSciences, Results –Weidentified359 eligible children andrecruited 268 (74.7%)
UniversityofBristol,Bristol,UK tothe study. Elevenseparaterecords foreachchildwere collected at the
R.Preston,CleftLipandPalateAssocia-
audit clinics.In total,2666(90.4%) werecollected fromapotential 2948
tion,London,UK
D.Sell,SpeechandLanguageTherapy records. Theresponserates forthe self-reported questionnaires,
DepartmentandCentreforOutcomes completedathome,were52.6%fortheHealthandLifestyleQuestionnaire
andExperienceResearchinChildren’s
and52.2%fortheSatisfactionwithServiceQuestionnaire.
Health,IllnessandDisability(ORCHID),
Conclusions –Response ratesand measureswere similarto those
GreatOrmondStreetHospitalNHSFoun-
dationTrust,London,UK achievedin theprevious survey.There arepractical, administrativeand
J.Smallridge,SouthThames’CleftUnit, methodological challenges in repeating cross-sectional surveys15 years
Guy’sandStThomasHospital,London,
apart andproducing comparable data.
UK
J.Smallridge,CleftNetEastCleftNet-
work,Addenbrooke’sHospital,Cam- Keywords: cleft lip;cleft palate;cross-sectional studies
bridge,UK
H.Worthington,SchoolofDentistry,
UniversityofManchester,Manchester,
UK
A.R.Ness,NationalInstituteforHealth
Research(NIHR)BiomedicalResearch
UnitinNutrition,DietandLifestyleatthe
UniversityHospitalsBristolNHSFounda-
tionTrust,UniversityofBristol,Bristol,
UK
Correspondenceto:
ProfessorJ.R.Sandy
UniversityofBristolDentalSchool
LowerMaudlinStreet
BristolBS12LY
UK
E-mail:[email protected]
Introduction advice on access to availability of selected
National Health Services (NHS) specialized ser-
The organization of care for children born with a vices. A number of areas were examined includ-
cleft of the lip and/or palate in the United King- ing childhood leukaemia, schizophrenia and
dom (UK) underwent significant change over the women in normal labour. For cleft lip and palate,
last 15 years as the Clinical Standards Advisory the CSAG committee commissioned a research
Group (CSAG) report was published in 1998 (1) team to undertake studies of non-syndromic
and the onset of the CCUK study. The outcomes cases of unilateral cleft lip and palate (UCLP) in
of the CSAG study were widely reported, and children aged five and twelve years throughout
there have been considerable operational and ser- the UK. A cross-sectional survey of the process of
vice reconfigurations in this area of UK healthcare care assessed key outcomes including speech,
since those reports (2–6). The 57 centres operat- hearing, dento-alveolar and skeletal relations,
ing on children born with some form of oro-facial bone grafting, facial appearance and patient/par-
clefting in the UK in 1998 have been reduced to ent satisfaction. The CSAG survey team identified
11 centres or managed clinical networks. The 326 5-year-olds born with UCLP in the UK over a
implementation period has been prolonged, but 2-year period, and outcome records were col-
all cleft services are now in an ongoing process of lected for 239 of these children. This represented
centralization. The impact of these changes on 73% of those eligible. A full account of the
care and outcome is unclear (7). methodology is presented elsewhere (1). The
The CSAG was set up by the UK Health Minis- findings of the report were far-reaching and
ters in 1991 as an independent source of expert accepted for the following reasons:
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Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
1. The evidence in the CSAG report was com- focus the results and discussion of this study on
pelling and arose from a detailed and meticu- the comparability of measures and response
lous observational study. rates with the previous survey.
2. The recommendations of the CSAG report
were supported by all members of the CSAG
committee including those whose clinical Subjects and methods
practice was likely to be directly affected by
implementation. The original cross-sectional CSAG study did not
3. The recommendations were accepted by gov- require research ethics committee approval as it
ernment and supported by the relevant pro- was considered to be an audit. However,
fessional organizations. because of the changes in ethics procedures and
4. The proposed changes were in line with evi- a wish to incorporate collection of research data
dence from centres abroad, which were gen- into the present survey, ethical approval was
erally agreed to provide excellent services. obtained (REC reference number: 10/H0107/33,
5. Virtually all cleft care in the UK is provided South West 5 REC). Approval included consent
within the NHS. to link medical and other records in a number
6. Most importantly, there was, and still is, an of areas (such as education) and for additional
active and well-informed patients/parents/ measures (height, weight, head circumference)
professionals group (Cleft Lip and Palate and questions (psychosocial, health and lifestyle
Association, CLAPA) that accepted in princi- and economic) to be collected. There was also
ple the recommendations of the CSAG report. approval to approach families for further
research in future.
The key recommendation, unanimously sup-
ported by the CSAG committee, was that the
number of centres offering cleft services in the Studydesign
UK should be reduced from 57 to around 8–15.
Inevitably, not all regions could implement The original CSAG survey published in 1998 (1)
these changes at the same time but there were was cross-sectional and attempted to locate and
opportunities for the individual centralization study all 5-year-old children born between 1
processes to learn from each other and to April 1989 and 31 March 1991 with non-syn-
request direction from the Department of dromic UCLP. To compare a similar group post-
Health. centralization, we conducted a further cross-sec-
In the years after the CSAG report, there have tional study of 5-year-olds born with UCLP
been preliminary studies in secondary alveolar (known as Cleft Care UK) treated within a cen-
bone grafting and dento-alveolar relations that tralized or centralizing service. We adopted a
have reported improvements in outcomes (8, 9). protocol that was as similar as possible
These evaluations took place in the early stages (although extended in places to include addi-
of centralization and were either regional (rather tional items) to the original study. It was not felt
than national) or not comprehensive in scope. necessary to survey all children in the UK born
Given these shortcomings, a national examina- with all expressions of clefting as several multi-
tion of the impact of these changes to cleft ser- centre comparisons have provided evidence that
vices some 15 years after the centralization care for, and outcomes in, UCLP cases are repre-
process was both timely and relevant. In this sentative of the quality of care and outcomes in
study, we describe design issues, the conduct of a centre (3). The original CSAG survey also
the fieldwork and the data collection methods included 12-year-olds. At the time of the current
used in the study. We have included detailed study, however, this age group would not all
descriptions of the coding and analysis of these have been cared for in a centralized service so
data in the later studies in this supplement. We they were not resurveyed.
OrthodCraniofacRes2015;18(Suppl.2):1–13 | 3
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
Participanteligibility We did not exclude cases until they had been
discussed, on a case-by-case basis, with the cleft
We collected records of 5-year-old children from centre and the research team during the sched-
cleft centres in the UK born during a 2-year per- uled audit clinic. Cleft centres were asked to
iod. The original inclusion criteria comprised the provide clinical photographs of those children
following: born with UCLP and excluded from the study
(cid:129) because they had soft tissue Simonart’s bands of
Five-year-old children born with non-syn-
more than 5 mm in width.
dromic complete unilateral cleft of the lip and
palate, including any with soft tissue Simo-
nart’s bands of less than 5 mm. Studyclinics
(cid:129)
Children born between 1 April 2005 and 31
March 2007. Post-centralization, regular audit clinics were set
(cid:129)
The child was aged between 5 years 3 months up by most cleft centres, and defined age groups
and 5 years 9 months. If a child failed to (including children around the age of 5 years)
attend the initial scheduled research audit are now routinely reviewed. Measures of out-
clinic, they were invited to attend a subse- comes for appearance, dental arch relationship,
quent audit clinic up until the age of 6 years speech and hearing (function), oral health and
and 5 months. Some children were seen at psychosocial adaptation are collected at these
younger and older ages than originally stipu- audit clinics. To support the research study, cleft
lated (Fig. 1); we decided to include these centres agreed to organize designated audit clin-
children and to examine and adjust for age in ics and invite eligible 5-year-olds within the
analyses where appropriate. agreed age range. These clinics collected addi-
tional information to standard audit clinics (such
Exclusion criteria comprised the following:
as head circumference); hereafter, we refer to
(cid:129)
ChildrenbornwithUCLPwhosedevelopmental these clinics as audit clinics. The research team
delaywassufficienttopreventthemfromcoop- (comprising psychologists and dental academics)
eratingwithprocedures(suchasspeechrecord- liaised and worked with an identified key mem-
ings)thatwereneededfordatacollection. ber of each cleft centre to arrange dates and
(cid:129)
Refusal to participate in the study by either details of audit clinics and to collect data. Eligi-
parents or children. ble families were sent written information about
Fig.1. Histogram of the age dis-
tributionoftheCCUK(filledbars)
and CSAG children at presenta-
tiontothedatacollectionclinics.
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Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
the study from the cleft centre together with the researchers in a free-post labelled envelope.
their appointment details for the audit clinic. On If questionnaires were not returned within 7–
the day of the audit clinic, the parent(s) and 14 days, the research teams posted one remin-
their child were asked whether they would par- der with a further copy of the questionnaires
ticipate in the study and, if they agreed, consent together with a free-post labelled envelope.
was obtained from the parents and assent
obtained from the child. The research team Surgicaltreatment
sought consent and assent for the majority of
children, but if the researchers were unable to The type of primary lip repair, surgical compli-
attend the audit clinic, the clinical team at the cations, type of palate repair and whether antibi-
centre sought these agreements. If the parent otics were used at the time of lip and palate
refused to participate in the study, the data closure were recorded from medical notes. The
obtained during the clinical examinations were surgeons examined the child to assess whether
stored in the child’s medical files and not any oral fistulae were present and whether there
accessed by researchers. If the parents and child were any functional problems such as nasal
failed to attend their initial audit clinic, further regurgitation of liquids and food.
invitations were sent until no further audit clin- The surgeon also documented their subjective
ics were organized and therefore available. Audi- rating of the surgical outcome using a four-point
ology and speech assessments and recordings Likert scale ranging from poor to very good on
were carried out by the centre specialist team or each of the following: the scar quality of the lip,
by local specialist staff (i.e. audiologists and Cupid’s bow, lip length, frontal view and inferior
speech and language therapists). Dental exami- view of appearance/symmetry of the nose. The
nation and assessment were carried out by Bri- vermillion border was assessed on three parame-
tish Association for the Study of Community ters: notching and/or deficiency, was it balanced
Dentistry (BASCD)-calibrated (10) paediatric (i.e. equal on both sides), or was it or too full or
dentists, and psychological assessments were bulging. The functionality of the lip was assessed
completed by a psychologist. In those centres when smiling (symmetrical/equal length or
that did not have access to a paediatric dentist asymmetrical/shortened) and pouting (symmet-
or psychologist, the research team conducted rical or asymmetrical).
the assessment. Standardized photographic
views (extra-oral and intraoral) were taken by Assessmentofdentalarchrelationship
either the medical photographer or the
orthodontist at the cleft centre. Impressions for Alginate impressions of the child’s teeth were
dental study models were taken by the taken together with a wax squash-bite in centric
orthodontists, and they also completed details of occlusion and a record of the overjet. The
the child’s orthodontic history. The surgical impressions were placed in labelled plastic bags
forms were completed by surgeons using infor- and transported to an orthodontic laboratory by
mation from the medical notes. The surgeons the research team in a ‘cool bag’. For consis-
also assessed the appearance of the lip and nose. tency, a single laboratory technician handled all
Parents/guardians were given a number of ques- of the impressions and constructed all of the
tionnaires to be completed in the audit clinic models. The impressions were cast in white
and placed in a questionnaire box provided by plaster and study models constructed in a stan-
the research team. Alternatively, they could mail dardized format with the participants ID number
them back in a free-post labelled envelope pro- inscribed in the base. Some cleft centres pro-
vided to them. The parent/guardian was also vided existing study models, which were then
asked to complete the Health and Lifestyle duplicated in the laboratory and the original
Questionnaire and the Satisfaction Questionnaire returned to the centre. If impressions could not
in their home environment, to be returned to be obtained, then intraoral photographs of the
OrthodCraniofacRes2015;18(Suppl.2):1–13 | 5
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
teeth in occlusion were taken. These intraoral buccal occlusion, oral cleanliness and the
photographs included a frontal view, right and number of decayed, missing and filled teeth
left lateral views and, if possible, a palatal view. (dmft). The presence/absence of fistulae was
The interarch relationships of the 5-year-olds’ also noted but only if a surgical assessment
study models were used to indicate the effects of (where fistulae were also recorded) was not
surgery on dental arch relationships, and this conducted on the day of the audit.
was assessed with the well-established 5-Year-
Olds’ Index (11). Audiology
Facialaesthetics The history of audiological and otological inter-
ventions was derived from the medical notes
A two-dimensional assessment of the child’s face and through questioning the parents. This his-
was made from profile and frontal photographs tory included the management of middle ear
that were taken using published guidelines (12, effusion through watchful waiting, insertion of
13). Photographs were taken with a standardized grommets, the use of hearing aids (past and cur-
camera set-up (Camera Nikon D3s or other rent) and other medical otological procedures.
equivalent camera, a 105-mm macro lens and The audiological/otological assessment com-
lighting equipment for the camera). The child prised a full audiogram which tested air conduc-
was asked to sit on a chair positioned 0.5 m in tion (AC) and bone conduction (BC) as
front of a standardized black or white non-reflec- appropriate to assess hearing thresholds. The
tive wall mounted background. If necessary, the management, type and degree of hearing loss
child’s hair was arranged to show the entire ear. were recorded on the audit assessment day with
The following views were taken for each child middle ear function assessed by tympanometry
with the lens length fixed to give standardized and otoscopy.
magnification (the numbers in brackets are the
magnification settings on the camera): left lateral Somaticgrowth
face (1:8), right lateral face (1:8), ¾ left lateral
face (1:8), ¾ right lateral face (1:8), facial (1:8), An assessment of growth was made by measur-
facial smiling (1:4), whistling (1:4), worm’s eye ing the child’s height, weight and head circum-
view (1:4), lip and nose (1:3). ference. The height was measured in
The images were anonymized and cropped to centimetres to one decimal place using the
allow unbiased assessment of only the nose and Leicester Height Measure Scale (First Aid Ware-
lip area. They were rated independently by a house, 17 Chesford Grange, Woolston, Warring-
panel of assessors to determine the appearance ton, WA1 4RQ, UK). The child was asked to
of the lip, nose and profile of 5-year-olds. To remove their shoes and then positioned on the
evaluate the parents’ perception of their child’s height measure with their feet flat, the underside
appearance, the 20-item Satisfaction with of their heels in contact with the ground and the
Appearance Scale was used (14–16). backboard of the measuring device. The heels
were placed together, so that the medial malleoli
Oralhealth were touching (unless the child had ‘knock
knees’). The child was asked to stand straight
The child’s dental history was derived from par- with the shoulders relaxed and sloping forward
ental accounts and checked with hospital notes in a natural position. The hands and arms were
where available. Information included identifica- loose and relaxed with the palms facing medi-
tion of the usual care provider (general dental ally. The head was positioned with the Frankfort
practitioner or specialist paediatric dentist), the plane (the line between the lower orbit of
use of neonatal appliances and past dental the eye and the upper margin of the external
treatment. A clinical record was made of the auditory meatus) parallel to the floor. Weight
6 | OrthodCraniofacRes2015;18(Suppl.2):1–13
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
was measured in kilograms to one decimal place described in detail elsewhere (18). All recordings
on a calibrated Seca weight scale (model 899; were made by one of the centre-based SLTs,
Our Weigh Ltd, 10 Fore Street, St Mary Church, who had been trained in the CAPS-A. Recordings
Torquay, Devon, TQ1 4NE, UK). The child were made in a quiet room with the child facing
(lightly clothed and no shoes) was asked to step natural light if possible. A microphone was
on to the middle of the scale. placed on a stand, 23–30 cm away from the
Head circumference was measured in centime- child, at the level of their mouth and to one side.
tres to one decimal place, with the child sitting The face and upper neck were framed in the pic-
comfortably and relaxed with the Frankfort ture. The speech sample picture material was
plane parallel to the floor. The measurement presented beside the camera at the child’s eye
was taken with a reusable Lasso-O tape from level. Following data collection, the SLT checked,
Harlow Printing Ltd (Maxwell Street, South using the headphones, that a high-quality sam-
Shields, Tyne & Wear, NE33 4PU, UK) with the ple had been recorded.
tape being taut but not tight. The head was mea- The speech sample comprised the following:
sured at the widest horizontal circumference
1. A sample of 2 min of conversation, which was
above the eyebrows and ears.
encouraged with open-ended questions
through a progression of enquiries on a par-
Speechmethods
ticular topic.
2. Counting from one to twenty and from 60 to
The assessment of speech included a therapy
70.
history questionnaire which captured informa-
3. Saying (not singing) a nursery rhyme such as
tion about current and past experience of speech
Jack and Jill.
and language therapy services. Details were
4. Repetition of each of the 16 sentences after
gathered regarding waiting times for interven-
the therapist using the Great Ormond Street
tion, amount of contact time, location, focus of
Speech Assessment picture book.
therapy, therapists’ views of contributing factors
to outcomes and assessment of residual needs Assessment included nasality (hypernasality,
for speech and language therapy. Factors the hyponasality), nasal airflow (nasal emission,
speech and language therapist judged had con- nasal turbulence), cleft speech characteristics,
tributed to the outcome were identified. Data on non-cleft speech immaturities and intelligibility/
suspected and confirmed velopharyngeal insuffi- distinctiveness. This is described in detail in the
ciency and history of velopharyngeal surgery speech study of this series.
were also gathered. The history was taken in the
audit clinic by the speech and language therapist Psychosocialfactors
with information from parents, the medical
notes and local SLT notes. If needed, the thera- These data were collected using a standardized
pist subsequently sought further information questionnaire of 18 items that was originally
from local services. An estimate of residual developed by the Royal College of Surgeons of
needs for speech and language therapy was England Steering group (19) for the first CSAG
made. This estimate was based on the clinical study. Since the implementation of centraliza-
assessment made by the specialist therapist at tion, this questionnaire has been modified by
the time of the recording. The speech outcome the Psychology Specialist Interest Group (SIG).
measures used the Cleft Audit Protocol for The final version of questions used in the study
Speech-Augmented (CAPS-A) tool (17) developed to assess parental/guardian perceptions of the
in the UK for audit purposes in response to the impact of the cleft on their child was agreed by
CSAG report. For the assessment, speech audio– the Psychology SIG and the researchers before
video recordings were collected using the data collection began. The Strength and Difficul-
equipment, procedures and speech sample as ties Questionnaire with 35 items was used (20).
OrthodCraniofacRes2015;18(Suppl.2):1–13 | 7
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
Healthandlifestyle Respondents’ choices are assumed to reflect the
underlying value (utility) they place on service
Three questionnaires were used to collect basic attributes (29).
demographic data on the child’s temperament,
potential determinants or modifiers of psycho- Satisfactionwithservicequestionnaire
logical adjustment, and family costs of parental
preferences about the child’s cleft care. The An additional questionnaire was used to assess
basic socio-demographic section included ques- parental satisfaction with the cleft centre service
tions on ethnicity, parental age at the birth of delivery.
the child with a cleft, the highest level of paren-
tal education, most recent parental occupation,
number of people living in the home and rela- Results
tionship to the child as well as details of other Overallrecruitment
family members with cleft lip/palate and their
relationship to the child born with cleft lip and The cleft centres identified 359 eligible children
or palate. In the Health and Lifestyle Question- born in the period 1 April 2005 to 31 March
naire pack (140 items), established and validated 2007, and this figure was corroborated through
questionnaires were used to assess the individ- the national database CRANE (https://
ual characteristics of both the parent and child. www.crane-database.org.uk/). Eighty-five fami-
These included the Emotion, Activity and Socia- lies failed to attend the initial as well as the sub-
bility Scale (21), Parent–Child Relationships (22), sequent research audit clinics they were offered
Vulnerability and Over-protection (23), the War- and were not part of the study. Six families
wick–Edinburgh Mental Well-being Scale (23, declined to participate (2% of the 274
24), the Life Orientation Test – revised (25) and approached). Reasons included not wanting to
the 10-item Perceived Stress Scale (26). The participate, lack of time to complete the addi-
costs and discrete choices questionnaire mea- tional questionnaires or difficulties in re-arrang-
sured personal and indirect costs of the child’s ing audit clinics to suit them. We collected data
care incurred by the parent/guardian and was between 12 January 2011 and 12 December 2012
either sent out to families who attended early but continued to request missing data from the
audit clinics or included in the packs handed cleft centres until 30 April 2013. The study was
out at the audit clinic for the later recruits to the officially closed on the 15 May 2013 when we
study. The questionnaire was developed by the had recruited 268 (87 females, 181 males) of the
research team. A discrete choice experiment 359 eligible children 5-year-olds born with UCLP
(DCE), using best–worst scaling (27), was devel- (74.7%). Of these, 18% had a Simonart’s band.
oped to estimate the relative value of different
attributes of centralized cleft care services (28). Ageofchildren
The first stage of the DCE involved the identifi-
cation of attributes through a review of the liter- The cleft centres aimed to bring children to the
ature and semi-structured qualitative interviews audit clinics when they were aged between
with parents/guardians of children born with 5 years 3 months and 5 years 9 months. This
UCLP. The second stage entailed the assignment was not possible for all children as it required
of levels to the attributes identified, which was cleft centres to schedule special audit clinics that
explored with parents/guardians during the would take place when the children were at the
semi-structured interviews. The final stage will required age. We agreed that they should not
involve asking parents/guardians to choose run additional audit clinics to avoid increasing
between two or more hypothetical service con- the burden on staff and families. As a result, 20
figurations that have been created to contain children (7%) attended the audit clinics before
different combinations of attribute levels. the age of 5 years and 3 months. One hundred
8 | OrthodCraniofacRes2015;18(Suppl.2):1–13
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
and eighty-three children (68%) attended the would not cooperate or had missed the assess-
audit clinics within the optimal time span, but ment), it was not possible to obtain all records.
others missed their appointment and had to be In the event, 2666 data assessment records were
rescheduled. Thus, 65 children (24%) were older at least partially complete (90.4%).
than 5 years 9 months when they attended an
audit clinic. The mean age in the study was Self-completionquestionnaireresponserate
5 years and 7 months (range 4 years and
6 months to 7 years and 6 months). Two hundred and forty-six questionnaires
(Health and Lifestyle and Satisfaction with Ser-
Availabilityofclinicdata vice) were given to families in the audit clinic
for completion at home and postal return to the
Table 1 outlines the assessments made and the research team. Twelve families declined to
number of children who took part in each study. answer the questionnaires because of either lan-
The research teams attended 86 audit clinics guage difficulties or time constraints. Ten fami-
across the UK. At the audit clinics, it was possi- lies did not receive information or received the
ble to obtain 11 assessment records for each questionnaires during the audit clinic and as a
child. As there were 268 participants, a total of result did not participate in this part of the
2948 assessment records were, in theory, obtain- study. The response rate (those who returned
able. For a number of reasons (e.g. the child the questionnaire having completed at least a
part of it) was 52.6% (n = 141) for the Health
Table1. Numberofchildrenwithdataoneachassessment and Lifestyle Questionnaire and 52.2% (n = 140)
in the Cleft Care UK study and comparative information
for the Satisfaction with Service Questionnaire.
fromtheCSAGstudy(1)
Obtainedfrom ComparisonwiththepreviousCSAGsurvey
5-year-olds Obtainedfrom
whoattended 5-year-oldswho The 74.7% recruitment rate in CCUK is margin-
theCleftCare werepartofthe ally higher than the 73% recruitment rate
Specialty UKstudy CSAGstudy1998 achieved in the original 1998 CSAG survey.
Table 1 describes the response rates for CCUK
Audiology n n
and CSAG; the figures were broadly similar.
Speechandlanguage 227 200
Table 2 shows the age, sex, ethnic and socio-
forms
economic characteristics of the children in the
Speechrecordings 261 268
CCUK and CSAG surveys where data were avail-
Anthropometry 248 238
able. The CCUK children were assessed closer to
Facialaesthetics 242 –
the target age of 5 years – Fig. 1 shows that the
Oralhealth 252 200
CSAG children were older (difference in medi-
Dentalstudymodels 264 239
ans: +0.9 years) and had a wider spread of ages.
Orthodonticform 198 223
The sex and socio-economic distributions
Surgicaldetails 263 239
(Townsend index of deprivation) of the two
Psychology 243 297
cohorts were similar.
questionnaire
Strengthand 253 220
difficulties
Discussion
questionnaire
Healthandlifestyles 215 –
We set out to collect data from all 5-year-olds
(postal)questionnaire
born with non-syndromic UCLP in the UK over
Satisfactionwith 141 –
a 2-year period using comparable methods to
Service
the previous 1998 CSAG survey (1). This
OrthodCraniofacRes2015;18(Suppl.2):1–13 | 9
Perssonetal.Across-sectionalUKoutcomesforcleft–methodology
Table2. DescriptionofthestudysampleinCCUKandthe5-year-oldgroupfromtheoriginalCSAGstudy(1)
CCUK CSAG
N Median(IQR)unlessstated N Median(IQR)unlessstated p-Value‡
Male(n,%) 268 181(67.5%) 239 159(66.5%) 0.80
Age(years) 268 5.5(5.4–5.7) 239 6.4(5.9–6.9) <0.001
Whiteethnicity(n,%) 121 111(91.7%) – –
Deprivationscore(rankingoutof100)* 210 21.1(11.7,35.7) 94† 20.2(11.3,32.4) 0.58
*English Index of Multiple Deprivation based on postal codes: 2007 for CCUK and 2004 for CSAG (http://geoconvert.mimas.ac.uk/
help/faq.htm#walkthrough). Geocoding began in 2004 so this is the earliest comparable index available for CSAG. Measurement
range=0/100inpercentileswherelowerscoresindicatethemostdeprivedpostalareas.
†Postcodeswereonlypartiallyrecorded.
‡z-TestforproportionsandWilcoxonranksumtestofmedians.
included all those with Simonart’s bands, which further for treatment and to attend audit clinics.
comprised approximately 18% of the sample. This may in part have explained why more peo-
Five-year-old children were examined as they ple did not attend the centralized audit clinics.
were likely to have received all their care within
a centralized service. We recruited 268 partici- Auditvs.research
pants and obtained data on most clinical out-
comes for over 90% of those enrolled. The Cleft Care UK research team supported the
local cleft centres with obtaining consent and
Comparisonofresponseratesbetweensurveys assent and in data collection. There were delays
in obtaining research approval for this study that
Although the response rates for these two sur- we have highlighted elsewhere (30) and reduced
veys were similar, we had expected that post- response rates as a result of the need to obtain
centralization recruitment to CCUK would be consent. However, obtaining research permis-
higher. We thought that we would find it easier sions has had advantages. It has made it possi-
to recruit from fewer centres that had dedicated ble to approach participants with further
cleft teams working to defined job plans and research questions at a later date. For example,
running regular audit clinics. However, we did the DCE was not included in the original CCUK
manage to recruit individuals closer to the target protocol. It has also allowed us to develop
age in CCUK vs. the CSAG survey. The data in research capability and capacity within the cen-
the CSAG 1998 study were mainly collected by tralized service that will be available to support
the research team, whereas in Cleft Care UK, future research projects (31).
more of the data collection was undertaken by
the cleft centres. As CSAG was approved as an Theuseofdifferentclinicaloutcomes
audit project, information was collected from
the clinical records of children even if they did Speech is recognized as an important functional
not attend. In the CCUK study, however, infor- outcome measure of cleft care. Enabling children
mation was collected only for those children with cleft palate to have normal speech by
who attended the audit clinics and where their 5 years of age is a shared goal for both the fam-
parents gave informed consent. In the CSAG ily and the cleft team as it is important both
study, parents completed the Satisfaction with socially and educationally. Britton et al. (32), in
Service Questionnaire when they attended the fact, argue that speech outcomes represent a
audit clinic, whereas in CCUK, questionnaires cleft team’s multidisciplinary outcome, encom-
were completed at home and returned by post. passing timely and effective primary surgery,
Centralization does require families to travel well-coordinated follow-up, proactive hearing
10 | OrthodCraniofacRes2015;18(Suppl.2):1–13