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Results of Randomized Controlled Trial of Soft Palate First Versus

Hard Palate First Repair in Unilateral Complete Cleft Lip and Palate
Bruce Richard, M.B.B.S., M.S., F.R.C.S.(Plast), Joyce Russell, B.D.S., Ph.D., F.D.S.R.C.P.S., M.Orth., R.C.S.,
Siobhan McMahon, B.Sc., M.R.C.S.L.T., Ron Pigott, F.R.C.S., F.R.C.S.I.

Objective: To compare the outcomes for primary repair of unilateral cleft lip
and palate, operating on the soft palate first versus the hard palate first.
Design: Randomized controlled trial.
Setting: The Regional Cleft Service of West Nepal.
Patients: Forty-seven consecutive patients with nonsyndromic unilateral
cleft lip and palate, of whom 37 were assessed 4 to 6 years after completing
primary surgical repair.
Interventions: Primary repair of unilateral cleft lip and palate by two differing
sequences: (1) soft palate repair, with hard palate and lip repair 3 months later;
and (2) lip and hard palate repair, followed by the soft palate repair 3 months
later.
Main Outcome Measures: Analysis of dental study models, weight gain, and
speech recordings.
Results: Four to 7 years after completing the cleft closure, there was no
significant difference in facial growth between the two types of repair sequencing. Completing posterior repair first had no effect on anterior alveolar gap
width. It narrowed the hard palate gap by reducing the intercanine distance.
Anterior repair dramatically closed the anterior alveolar gap, and narrowed the
intercanine distance. Comparing anterior alveolar gap width with age at first
presentation demonstrated that there was no spontaneous narrowing of the
cleft in older children. Completing posterior closure first had a weight gain
advantage over anterior closure first. Improved oropharyngeal closure, and
thus swallowing, is the likely explanation.
Conclusion: Changing the sequencing of cleft closure has no demonstrable
difference in facial growth at 4 to 7 years after completion of the primary surgery.
KEY WORDS: cleft lip and palate, maxillary arch, randomized controlled trial,
surgery

Poor maxillary growth in unilateral cleft lip and palate is


multifactorial in etiology, and early palatal closure is likely to
be a contributing factor. It is recognized that normal downward

and forward development of the congenital cleft maxilla will


occur if the cleft is not repaired (Mars and Houston, 1990b;
McCance et al., 1990). However, it is not known whether the
deleterious effect of palatal surgery is due to interruption of
blood supply, scar contracture from relieving incisions, the effect of raising mucoperiosteal palatal flaps, or a surgeon-related factor. A national study in the U.K. found that up to 40%
of patients with repaired complete unilateral cleft lip and palate
(UCLP) require a maxillary advancement osteotomy in late
adolescence (Williams et al., 2001), and surgical scarring is
presumed responsible for some of this impaired maxillary
growth.
To ameliorate this scarring, Malek and Psaume (1983) proposed closing the soft palate first, with the expectation of narrowing the width of the hard palate defect. The intention here
is that its later repair would require little palatal dissection,
thus minimizing surgical scar formation and permitting uninhibited maxillary growth. Abyholm et al. (1981), however,

Dr. Richard was Plastic Surgeon to the Western Regional Hospital, Pokhara,
Nepal, and is currently Cleft Surgeon at Birmingham Childrens Hospital, Birmingham, U.K. Dr. Russell is Orthodontist for the Cleft service of Alder Hey
Childrens Hospital, Liverpool, U.K. Ms. McMahon is Senior Speech and Language Therapist for the Cleft service of Alder Hey Childrens Hospital, Liverpool, U.K. Dr. Pigott was Senior Plastic Surgeon at Frenchay Hospital, Bristol, U.K., and is now retired.
Portions of this article were presented at the winter scientific meeting of the
British Association of Plastic Surgeons, London, U.K., December 37, 1999;
the summer scientific meeting of the British Association of Plastic Surgeons,
Glasgow, U.K., June 2001; and at the annual scientific meeting of the Craniofacial Society of Great Britain and Ireland, Leeds, U.K., April 911, 2003.
Submitted April 2005; Accepted August 2005.
Address correspondence to: Dr. Bruce Richard, Department of Plastic Surgery, Birmingham Childrens Hospital, Birmingham, U.K. E-mail bruce.
richard@bch.nhs.uk.
329

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Cleft PalateCraniofacial Journal, May 2006, Vol. 43 No. 3

chose to close the hard palate with a single layer vomerine


flap at the same time as performing the primary lip repair, thus
obviating the need for palatal shelf dissection.
These two European centers (in Paris, France, and Oslo,
Norway) have received considerable attention in the latter part
of the 20th century, partly because of the diametrically opposed sequencing of repair of the hard and soft palate components of the cleft. In each center, large numbers of patients
underwent operations that followed a strict protocol, and in
both centers a high proportion of patients achieved good maxillary forward growth with a low requirement for maxillary
osteotomy.
The Norwegian center at the Rikshospitalet, Oslo, Norway,
used a sequence of initial repair of the hard palate with a
vomer flap and simultaneous lip repair, with continuity of repair between the hard palate and floor of the nose at the age
of 3 to 4 months. This was followed by soft palate repair, using
the von Langenbeck technique, at the age of 12 months. Although there was concern about the use of a vomerine flap to
close the hard palate (Delaire and Precious, 1985), many units
began using this sequence, but with variations of timing.
The opposite order of repair was practiced at the French
center of Hopital Robert Debre, Paris, France, by Malek and
Psaume (1983), who used a sequence of initial repair of the
soft palate only at 3 months, followed by the lip and hard
palate at 6 months. The Malek philosophy was adopted by the
Belgian center at the Brugmann Hospital, Brussels, Belgium,
and also in Nantes, France, where Delaire had an 11-month
gap between the staged closure of the cleft.
Representative groups of both Oslo and Paris patients have
been analyzed by independent assessors, albeit with different
methods of assessment. The Oslo method was assessed using
the GOSLON yardstick (Mars et al., 1987) showing less than
10% of patients likely to benefit from an osteotomy (groups 4
and 5) (Mars et al., 1992). Malek (2001) reported a 10% osteotomy rate on a personal series of 92 cases of UCLP. DeMey
et al. (1992a, 2002) also had a cohort of their patients assessed
by GOSLON, which showed that 6% required an osteotomy.
Both Oslo (Semb, 1991) and Paris (Ross, 1995) have had assessment of cephalometric data that showed excellent midfacial growth.
However, it was not known how the techniques would compare in the hands of one surgeon with a substantial caseload
and with no prior commitment to either sequence (i.e., equipoise). Therefore a randomized controlled trial was designed
to test the hypothesis that, in patients with UCLP, first closing
the soft palate and three months later the hard palate and lip
(posterior to anterior sequence, P-A) will not give better maxillary growth than first closing the lip and hard palate and three
months later the soft palate (anterior to posterior sequence, AP). This trial randomized cases to each sequence, and aimed
to assess outcome in terms of maxillary growth and speech.
This paper reports the results 4 years after the last patient
entered the trial.

METHODS
Participants
The first author was responsible for cleft care in the western
half of Nepal from 1990 to 1998, and with a caseload of more
than 80 new patients a year, he was able to randomize patients
with UCLP to each of the operation sequences. In Nepal, patients present at differing ages and come from two distinct
ethnic groups, based on their geographic origins and tribal
grouping (Sapkota, 1997). These are Indo-Aryan ethnicity
(64%) and Tibeto-Mongoloid ethnicity (36%). The trial was
carried out in compliance with World Health Organization regulations for research in developing countries (Council for International Organizations of Medical Sciences [CIOMS] and
the World Health Organization [WHO], 1993), and gained the
ethics approval of the local research committee and the National Health Research Council of Nepal. Proxy informed consent was obtained from the parents of each child using health
education materials, photographs, a printed explanation in Nepali, and written consent obtained by a research assistant.
Only children with nonsyndromic UCLP who were between
the ages of 3 and 60 months were considered eligible for the
trial. A Simonarts band (a soft tissue adhesion between the
medial and lateral margins of the cleft in the lip or nostril, or
between the clefted alveolar processes, concurrent with a complete skeletal cleft of the alveolus) was allowed as long as there
was no bony element (Semb and Shaw, 1991).
Patients were allocated to an operative sequence by stratified
block randomization. The stratification factors were age (3 to
18 months, 19 to 36 months, and 37 to 60 months) and ethnicity (Tibeto-Mongoloid or Indo-Aryan). Gender and side of
cleft were ignored in the expectation that these would be acceptably distributed with an adequate sample size. Patients
were allocated to the correct block randomization (three age
groups and two races) using six prepared bags of cards, where
half the cards had anterior first and half posterior first
written on the card as the operation sequence. The cards were
drawn by a third party. At least 19 patients were required for
statistical significance in each sequence group to discern a systematic difference of 0.75 of a GOSLON point at .05 significance level and 80% power at age 8 to 10 years (Shaw et al.,
1992). To allow for loss to follow-up, the intention was to
recruit 25 to each sequence group. A total of 47 patients were
entered (Tables 1 and 2).
Interventions
Surgical techniques were standardized as far as possible,
because the only variable to be tested by the hypothesis was
the sequence of procedures. The anterior operation consisted
of a lip repair by Millard rotation advancement (Millard Jr.,
1964, 1976), a nasal correction using the McComb procedure
(McComb, 1985), and a hard palate repair by single layer vomerine flap. The posterior operation consisted of a soft palate
repair with medial von Langenbeck incisions (Murison and

Richard et al., PRIMARY REPAIR OF UCLP: RESULTS OF AN RCT

TABLE 1 Trial Profile

TABLE 2 Patient Demographics: Preoperative Characteristics for


Patients Randomized to Either Posterior to Anterior (P-A) or Anterior to
Posterior (A-P) Sequencing of Cleft Repair

Registered or Eligible Patients


(n 5 47)
Boys
Girls
Tibeto-Mongoloid
Indo-Aryan

n
n
n
n

5
5
5
5

27
20
17
30

Randomization
(nonrandomized, n 5 0)
Anterior-Posterior Sequence
Posterior-Anterior Sequence
(n 5 24)
(n 5 23)
Boys
Girls
Tibeto-Mongoloid
Indo-Aryan

n
n
n
n

5
5
5
5

16
7
9
14

Boys
Girls
Tibeto-Mongoloid
Indo-Aryan

n
n
n
n

Follow-up Phase
(n 5 16)

Follow-up Phase
(n 5 19)

Boys
Girls
Tibeto-Mongoloid
Indo-Aryan

n
n
n
n

5
5
5
5

13
3
4
12

Boys
Girls
Tibeto-Mongoloid
Indo-Aryan

n
n
n
n

Withdrawn
(n 5 6)

Withdrawn
(n 5 4)

Deceased
Syndromic
Lost contact

n51
n50
n55

Deceased
Syndromic
Lost contact

331

5
5
5
5

11
13
8
16

5
5
5
5

9
10
7
12

Mean age
Indo-Aryan
Boys
P-L*
TR-TL
CR-CL
cr-cl

P-A

A-P

18.7 mo
61%
70%
9.9 mm
37.3 mm
30.2 mm
10.5 mm

18.8 mo
67%
46%
11.0 mm
38.4 mm
31.3 mm
10.2 mm

* P-L 5 anterior alveolar gap; TR-TL 5 intertuberosity distance; CR-CL 5 intercanine distance;
cr-cl 5 width of the cleft at the canine level.

of the cleft at the canine level cr-cl (ICCW-intercanine cleft


width; see Fig. 1). All model measurements were repeated 2
months later to assess intraobserver reliability.
Preoperative Weight
Children were weighed unclothed before each operation.
Orthodontic Examination

n50
n51
n53

Pigott, 1992). The two operations were undertaken 3 months


apart. Alveolar repair was deferred to the time of alveolar bone
graft (between 8 and 12 years).
Recording
Patient information was recorded on a standard form. Data
were entered into a computer database (Epi-Info, version 6.2;
Dean et al., 1996). Patients were followed at the Cleft Palate
Clinic, Western Regional Hospital, Pokhara, Nepal. Four years
after completing the trial, follow-up records were obtained in
Nepal by a visiting team from Alder Hey Childrens Hospital,
Liverpool, U.K.

Four years after completing entry of patients to the trial, a


team visited Nepal to collect audit data on the cohort of children. Orthodontic study models were obtained from alginate
impressions on the 37 patients attending. It is proposed to use
the GOSLON yardstick (Mars et al., 1987) to assess maxillary
growth when the entire sample has reached the 8- to 12-year
review. For now, using the Five-Year-Old Index (Atack et al.,
1997) for the 4- to 8-year-olds and the GOSLON Yardstick
for children older than 8 years would have split the groups
into statistically inadequate numbers. Consequently, incisor
overjet (measured clinically to the nearest millimeter using a
stainless steel ruler) was used as the measure of forward
growth for this report. It has been shown that overjet explains
87% of the variance of the GOSLON score and thus has a

Outcome Measures
Preoperative Maxillary Models
For both sequences, a quick-setting, addition-cured silicone
putty (Optosilt) maxillary arch impression was taken before
each of the two operations once the child was anesthetized.
Plaster of Paris models were made, numbered, and stored.
Models were measured randomly by an orthodontist (J.R.) using a Reflex Metrograph (Ross Instruments Ltd., Salisbury,
U.K.) three-dimensional measuring device (Scott, 1981). The
measurements were made to mucosal reference points considered stable by previous authors (Ball et al., 1995), and included
the anterior alveolar gap P-L, intertuberosity distance TR-TL,
intercanine distance CR-CL, and where unrepaired, the width

FIGURE 1 Mucosal reference points for maxillary arch model measurements. P-L 5 anterior alveolar gap, TR-TL 5 the intertuberosity distance,
CR-CL 5 the intercanine distance, and cr-cl 5 the width of the cleft at
the canine level.

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Cleft PalateCraniofacial Journal, May 2006, Vol. 43 No. 3

significant predictive value (Morris et al., 1994). Orthodontic


assessment and speech recordings also were obtained from the
local population to create a control group. For each of our
patients, two noncleft children, matched for age, sex, and ethnicity, had a dental examination and speech assessment. Thus,
50 Indo-Aryan and 24 Tibeto-Mongoloid children were examined for incisor overjet.
Speech and Velopharyngeal Function
A sample of spontaneous and rote speech (counting) was
obtained for each child and recorded using a JVC digital video
recorder (GR-DVL 9500, Victor Company of Japan, Limited,
Japan) and Sony Mini Disc recorder (MZ-R70, Sony Corporation, Japan). There is currently no standardized speech assessment available for Nepali speakers. However, sentence imitation frequently is used to obtain a speech sample for analysis, and a Nepali sentence set has been in use at the hospital
for some time. The sample contains a sentence for each phoneme occurring in Nepali, with each phoneme used initially,
medially, and, where these occur, finally in words. This sentence set, therefore, was used to obtain a further sample of
each childs speech for later analysis. In addition, each child
was assessed while counting and repeating the sentences using
the Kay Nasometer II (Kay Elemetrics, Lincoln Park, NJ) to
produce nasalance scores.
The speech samples for the cleft children and matched controls subsequently were analyzed to compare features of resonance and consonant production between the two groups.

TABLE 3 Reproducibility of Reflex Metrograph Measurements in


Millimeters (mm)
Mean
Difference
P-L preop*
P-L postop
TR-TL preop
TR-TL postop
CR-CL preop
CR-CL postop
cr-cl preop
cr-cl postop

0.24
0.17
20.39
20.29
1.71
0.00
0.89
0.58

95% Limits
23.15,
22.20,
24.13,
23.91,
22.99,
24.47,
21.54,
22.56,

23.64
22.54
23.35
23.33
26.41
24.47
23.32
23.72

Error
(Dahlberg)
1.22
0.85
1.37
1.31
2.07
1.60
1.07
1.18

* P-L 5 anterior alveolar gap; TR-TL 5 intertuberosity distance; CR-CL 5 intercanine distance;
cr-cl 5 width of the cleft at the canine level; preop 5 preoperative; postop 5 postoperative.

ratio was 20:10 for the Indo-Aryan, and 7:10 for the TibetoMongoloid racial groups. Four years after completing entry of
patients to the trial, a team visited Nepal to collect audit data
on the cohort of children. Of the original patients, 38 returned
for assessment. Their ages ranged from 4 to 11 years. One
child was excluded (it was now apparent that she had a syndromic cleft), leaving 37 patients for assessment. Of the nine
patients who failed to attend, one girl had died from snakebite
and the rest were in a war zone and the team could not safely
access them. Table 1 shows the trial profile.
Preoperatively, there was no difference in age, anterior alveolar gap size, or ethnicity between children randomized to
the P-A or A-P sequence of repair. The ratio of boys to girls
was as expected for UCLP, and although more boys than girls
were randomized to P-A than to A-P, this was not statistically
significant (Table 2).

Otological Examination
Changes in the Maxillary Arch
Patients were examined preoperatively and 6 weeks postoperatively. Any found to have a hearing loss or otitis media
remained under regular review and active treatment. At the 4year review, patients had a hearing assessment with tympanography, audiometry, and an otological examination.
Statistical Methods
Statistical comparisons were carried out using SPSS 10.0.7
for Windows (SPSS Inc., Chicago, IL). The demographics of
the patients entering the trial were compared using t tests (PL, TR-TL, CR-CL, cr-cl), Mann-Whitney U test (age), and chisquare tests (gender and ethnicity). Fistula rates were compared by means of chi-square. The reproducibility of the maxillary model measurements was assessed using the method of
Bland and Altman (1999) and using Dahlbergs formula
(1940). Changes in maxillary arch measurements, weight gain,
and clinical incisor overjets were compared using t tests.
RESULTS
A total of 47 patients were entered into the trial over 3.5
years. Of these, 23 were randomized to the P-A sequence of
operations and 24 patients to the A-P first. The boy to girl

After completing the first and second surgery to repair the


cleft on 47 patients, the maxillary arch models were analyzed.
Table 3 demonstrates the reproducibility of the measurements,
which is shown as the mean difference in measurements in the
first column, followed by the 95% limits of agreement using
the method of Bland and Altman (1999). The error of the
method according to Dahlbergs formula (1940) is shown also.
The reproducibility was satisfactory, even though there were
difficulties using some of the mucosal reference points due to
the presence of teeth in the older patients.
From Table 2, it can be seen that there was no difference in
age, anterior alveolar gap size, intertuberosity, and intercanine
or cleft width between UCLP children randomized to the A-P
or the P-A sequence. Nor were there any differences in these
measurements between the Indo-Aryan and the Tibeto-Mongoloid ethnic groups. This cohort of 47 patients first presented
at differing ages and thus gives us a cross-sectional view of
the anterior gap size. This was neither larger nor smaller in
the older children, regardless of children presenting with ages
ranging from 3 months to 56 months. Figure 2 shows the even
spread of anterior cleft gap size against the increasing age at
presentation (Pearson correlation coefficient r 5 .12).
Table 4 compares the changes in measurements for each

Richard et al., PRIMARY REPAIR OF UCLP: RESULTS OF AN RCT

333

TABLE 4 Compares the Changes in the Maxillary Arch Measurements


Between the First and Second Operations for Each Operative Sequence
Reductions in Maxillary Arch Measurements in Millimeters (mm)
PosteriorAnterior
Sequence

AnteriorPosterior
Sequence

Comparison of
P-A With A-P
p Value

0.3
0.6
1.7*
1.6**

7.0***
0.9
4.3**
N/A

p , .001***
NS
p , .001***
N/A

P-L
TR-TL
CR-CL
cr-cl

FIGURE 2 Anterior alveolar gap size as a function of age at presentation.

mucosal reference point on the study models (taken just before


each surgery) for the two sequences and demonstrates the dramatic effects that the surgery has on the maxillary arch form.
For the A-P sequence, the anterior gap (P-L) was reduced
by 7.0 mm, and the intercanine (CR-CL) distance narrowed
by 4.3 mm. The hard palate defect (ICCW) change could not
be measured in the A-P sequence because of the vomer flap.
The P-A sequence neither opened nor closed the anterior
cleft gap (P-L), but the width of the cleft defect at the level
of the canines (ICCW) narrowed by 1.6 mm and the intercanine distance (CR-CL) narrowed by 1.7 mm. The intertuberosity
(TR-TL) distance was not altered significantly in either sequence. As shown in Table 4, the differences in P-L and CRCL values were statistically significant between P-A and A-P
sequencing (p , .001). A summary of the above changes to the
maxillary arch form caused by the first operation can be seen in
Figure 3.
Weight Gain After the First Operation
A greater weight gain advantage was noted in the children
with the P-A sequence, as opposed to the A-P sequence. The
P-A children had a mean weight gain of 1.4 kg, whereas the
A-P sequence gained only 0.69 kg. These gains represented a
21% increase in weight for the P-A children against an 11%
gain for the A-P children (Table 5 and Fig. 4).

P-A 5 posterior-anterior; A-P 5 anterior-posterior; P-L 5 anterior alveolar gap; TR-TL 5


intertuberosity distance; CR-CL 5 intercanine distance; cr-cl 5 width of the cleft at the canine
level; NS 5 not significant; N/A 5 not applicable.
* p , .05; ** p , .01; *** p , .001.

Overjet Results
The mean overjet measurement for all patients was 21.3
mm (range, 27 to 4 mm; 95% confidence limit (CL), 22.1,
20.5), whereas the mean overjet for the age matched controls
was 1.9 mm (range, 21 to 5; 95% CL, 1.6, 2.1). There was
no significant difference in overjet between the P-A (21.6
mm; 95% CL, 22.9, 20.3) and the A-P (21.1 mm; 95% CL,
22.1, 20.1) sequences (Table 6 and Figure 5). There was a
significant difference between the two ethnicities for both control and patient groups, because the patients of Tibeto-Mongoloid origin had less maxillary protrusion.
Speech and Velopharyngeal Function
Of the 37 children who attended follow-up, 15 had undergone secondary velopharyngeal surgery during the intervening
period. These were evenly distributed between both operative
sequences, with 8 from the P-A sequence and 7 from the AP sequence group. Because it was no longer possible to compare the outcome in relation to the original surgery, these children were excluded from the subsequent speech analysis.
The speech of the 22 children who had not had secondary
velopharyngeal surgery, 9 from the P-A sequence and 13 from
the A-P sequence, was analyzed in terms of resonance and

FIGURE 3 Summary of changes in the maxillary arch form produced by the two different operative sequences.

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Cleft PalateCraniofacial Journal, May 2006, Vol. 43 No. 3

TABLE 5 Weight Gain After the First Operation, Relative to the


Sequence Used

Mean difference in weight, 3 months


after first operation
% increase in weight since the first
operation

PosteriorAnterior
(P-A)
Sequence

AnteriorPosterior
(A-P)
Sequence

Comparison
of P-A
With A-P
p Value

1.4 kg

0.69 kg

p , .06 NS*

21%

11%

p , .09 NS

* NS 5 nonsignificant.

speech sound production. Hypernasal resonance significant


enough to warrant further surgery was noted in 9 children, 5
from the P-A sequence and 4 from the A-P sequence. Nasalance scores obtained from children in the treated groups were
compared with those from the control group. The patient group
had higher nasalance scores than the controls and this difference was highly significant.
Audiological Assessment
At the 4- to 7-year audit, 33 patients attended for assessment
in the hospital, of which 29 had a clinical ear, nose, and throat
examination, as well as tympanography and/or audiometry. Of
27 patients tympanograms, only 4 had bilateral normal results
(type A), whereas the other 24 patients had a flat (type B)
result for either one or both ears. Audiometry on 18 patients
was normal in 6 (,26 dB), mild air conduction loss (26 to 40
dB) in 9, and moderate air conduction loss (41 to 55dB) in 3
patients. No patient had any sensorineural deafness. There was
no significant difference between the two operation sequences
in any of the above groups.
Surgical Complications
Patients were assessed for fistula at 6 weeks after the second
operation and of the 47 patients, 16 were noticed to have a
fistula. One patient failed to return and one patient was subsequently removed from the trial because it became apparent

FIGURE 4 Comparison of weight gain after the first operation with the
type of operation P-A or A-P.

that she was syndromic. Of the 16 fistulas, 4 closed spontaneously, 5 had a nonsymptomatic slit fistula at the junction of
the hard and soft palate, 6 required an operation (3 anterior
and 3 junction of hard and soft palate), and 1 was lost to longterm follow-up. The symptomatic fistula rate was 13%. Of the
16 fistulas noted, 10 were in the anterior-first group and 6 were
in the posterior-first group. This was not a significant difference in fistula rate.
DISCUSSION
There are very few randomized controlled trials in cleft care
and many unanswered issues, with clinical decisions often
based on tradition and local consensus rather than evidence.
In Europe, two main schools of thought regarding optimum
cleft closure have developed and have been championed by
various individuals. Most agree that skeletal and soft tissue
growth is influenced by both the congenital anomaly and its
surgical treatment. Ross (1987) claims that variations in the
timing of hard and soft palate repair within the first decade do
not influence facial growth in the anteroposterior or vertical
dimensions. Others claim that early palatal closure certainly

TABLE 6 Incisor Overjet Results for the Two Operative Sequences Compared Against Ethnicity, Gender, and a Control Group
Sample
Size
(n)

Mean Overjet
(mm)
Range in ( )

95% Confidence
Limits

All patients
All controls

37
74

21.3 (27 to 4)
1.9 (21 to 5)

22.1, 20.5
1.6, 2.1

p , .001

Boys, cleft
Girls, cleft

22
15

21.09 (27 to 3)
21.67 (25 to 4)

22.2, 0.0
22.9, 20.4

p , .47 NS*

Indo-Aryan, cleft
Tibeto-Mongoloid, cleft

25
12

20.44 (20.4 to 25)


23.25 (27 to 21)

21.3, 0.5
24.3, 22.1

p , .001

Indo-Aryan, control
Tibeto-Mongoloid, control

50
24

2.2 (1 to 5)
1.29 (21 to 4)

1.9, 2.4
0.7, 1.9

p , .008

Posterior-Anterior sequence
Anterior-Posterior sequence

17
20

21.6 (27 to 3)
21.1 (25 to 4)

* NS 5 nonsignificant.

22.9, 20.3
22.1, 20.1

Comparison
of Groups
(p Value)

p , .54 NS

Richard et al., PRIMARY REPAIR OF UCLP: RESULTS OF AN RCT

FIGURE 5 Comparison of dental index for each operative sequence controlled for ethnicity.

influences midfacial growth adversely (Mars and Houston,


1990a). Late palatal closure, though permissive of maxillary
growth, has poor speech results (Bardach et al., 1984).
The least damaging style and timing of palatal surgery has
yet to be determined. The Oslo team has popularized closure
of the lip and anterior hard palate with vomerine flap first,
followed by closure of the rest of the palate 3 months later
(Abyholm et al., 1981a; Semb, 1991). Their growth results as
measured by the GOSLON scale (Mars et al., 1987) compare
well with other centers in Europe, with a low requirement for
maxillary osteotomy (Shaw et al., 1992). However, De Mey et
al. (1992b) have published results of a longitudinal study suggesting that maxillary growth and speech were better in patients treated by the Malek technique, starting with closure of
the soft palate (Malek and Psaume, 1983, 1983), than in those
treated with closure of the lip first. Malek advocated closure
of the soft palate first, followed by closure of the lip and hard
palate after 3 months, suggesting that less dissection of the
hard palate area would be required.
With the advent of maxillary growth assessment by GOSLON, an independent review of results is possible for any
group of patients. This study was therefore designed to compare the effect on facial growth of closure of the anterior cleft
first versus closure of the posterior cleft first.
This study has four main findings a minimum of 4 years
after the primary surgery:
1. There was no difference in the width of the anterior cleft
in children presenting for surgery at different ages between 3
and 60 months. It has been claimed by some that the original
cleft spontaneously narrows in the first few months. Malek
believed this, stating the cause to be that the tongue falls back
into the nasopharynx and cannot exert any pressure on the
maxillary fragments (Malek and Psaume, 1983). Hill et al.

335

(2002) has shown that there is a nil change in average anterior


gap size from birth to 3 months, although for some individuals
in the sample the size diminished and for others it enlarged.
The prospective clinical trial in the Netherlands on the effects
of presurgical orthopedic treatment (PSOT) showed no significant reduction in anterior cleft gap until after the lip repair in
the non-PSOT group (Prahl et al., 2003). In the West, nearly
all clefts are closed anteriorly in the first few months of life.
This sample from Nepal allows us to look at a cross-section
of patients presenting at differing ages. The correlation between size of the anterior alveolar gap and age at presentation
indicated that there is no spontaneous narrowing of a cleft in
the first 5 years (Fig. 2).
2. The patients who underwent posterior palatoplasty first
(P-A) had a better weight gain than those who experienced
anterior palatoplasty and lip repair first (A-P). This was a supplemental finding from this randomized controlled trial. There
are several possible reasons for poor weight gain with a cleft.
The most commonly assumed difficulty is that of inability to
suck. This makes breast-feeding or emptying a noncompressible bottle difficult, because both require a negative pressure
(Bullock et al., 1990). The hard palate defect also makes compressing the nipple and thus emptying the lactiferous ducts
difficult. Babies with clefts have a normal weight at birth, but
can fail to thrive until the defect is closed. In Nepals western
hills, 42% of children are malnourished and fall below the
third percentile for weight (Nepal Family Health Survey,
1996). However, 60% of the children with clefts were malnourished by the same definition (unpublished data on a cohort
of 391 cleft children treated in Pokhara, Nepal). Children with
clefts are fed by a cup and spoon using expressed breast or
buffalo milk. Only a tiny minority of the rich could afford to
use formula milk and hygiene issues with milk preparation,
storage, and bottle cleaning make this method both a dangerous and an expensive way to feed the child with a cleft. Even
in the U.K., 49% of children with isolated cleft palate and 32%
of those with cleft lip and palate fail to thrive, whereas only
10% of those with a cleft lip alone will fail to thrive (Pandya
and Boorman, 2001). Supplemental feeding and feeding support by a cleft nurse is successful in achieving weight gain
while waiting for the cleft palate operation (Pandya and Boorman, 2001). This study has found that those children who had
the soft palate repair as the first operation had an average
weight gain of 1.4 kg in the next 3 months, whereas the anterior first group had half this amount of weight gain (0.69 kg;
see Fig. 4). Although this advantage was not quite statistically
significant (p , .06), it seems likely to be relevant for cleft
care. Closing the soft palate alone will not aid the production
of a negative intraoral pressure, but it might allow improved
swallowing. A cleft soft palate allows reflux of milk at the
back of the oropharynx to be regurgitated through the nose,
as often as milk is actually swallowed. In a recent randomized
controlled trial of presurgical orthopedics, the use of a presurgical orthopedic plate did not result in weight gain or easier
breast feeding, compared with patients who did not have a
plate (Prahl et al., 1996).

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Cleft PalateCraniofacial Journal, May 2006, Vol. 43 No. 3

The results of the present study suggest that closing the


posterior palate is a more important parameter than closing the
lip and anterior hard palate, in that a reconstituted posterior
palate allows competent swallowing, and thus provides a nutritional advantage. This might have clinical relevance for children with clefts who persist in failing to thrive and are at risk
of complications due to poor weight gain, especially in less
developed countries. A lesser operation might be a soft palate
repair in the first instance to improve weight gain. This tendency warrants further investigation.
3. With respect to maxillary arch changes, posterior repair
first had no effect on the anterior alveolar gap. It narrowed the
hard palate gap by reducing the intercanine distance. P-A repair reduced the intercanine distance by 1.7 mm, and reduced
the hard palate gap at the intercanine level by 1.6 mm. Thus,
the narrowing of the hard palate gap appeared to be at the
expense of maxillary plasticity and not a true narrowing by
elevating the shelves, or traction-stimulated growth of the
shelves (see Fig. 3).
Anterior repair dramatically closed the anterior alveolar gap
(mean, 7.0 mm), and narrowed the intercanine distance by 4.3
mm. Not only were these changes significant within each operative group between first and second models, but the differences in the change in anterior cleft gap and the change in
intercanine distance between the posterior- and anterior-first
groups were significant also. Neither operation significantly
changed the intertuberosity distance. The tendency was toward
a slight narrowing. This is contrary to the slight increase expected from growth (Aduss and Pruzansky, 1968). The malleability of the maxilla during surgery on the cleft is in its
anterior portion, whereas the posterior maxilla seems more resistant to these forces. However, these changes are the only
ones that occurred in the 3 months between operations. Those
who carry out soft palate first often wait a further 12 months
before closing the hard palate. The effect of a longer period
of soft palate closure on the intertuberosity width is not known.
It is possible that a lip and soft palate repair would combine
these changes and narrow the hard palatal cleft before any
surgery was carried out on the hard palate itself.
Aduss and Pruzansky (1968) demonstrated that nonsyndromal UCLP patients have a wider intertuberosity distance at
birth than noncleft controls do (33 versus 27 mm). This excess
width is similar to the hard palate defect width, and thus most
of the cleft gap is maxillary displacement rather than intrinsic
deficiency. Coupe and Subtelny (1960) published a study of
posteroanterior cephalograms of infants with UCLP and noncleft controls. This showed evidence of both laterally displaced
maxillae and intrinsic deficiency in the width of the clefted
hard palatal shelves. Cleft closure molds the maxillary arches
and narrows the width and depth excesses of the cleft palate
such that these laterally displaced shelves have narrowed to
within one standard deviation of the intertuberosity width of
noncleft children by the age of 5. The present results support
this view of maxillary molding. The malleability of the maxilla
during surgery on the cleft is in its anterior portion, whereas
the posterior maxilla seems more resistant to these forces. Bru-

sati (2003) has recently shown that 73% of those with a missing lateral incisor fall in his poor facial growth group, whereas
only 39% of such patients are in his good facial growth group.
This is reasonable evidence that there is also an effect of intrinsic deficiency in the long-term growth prospects of a child
with UCLP, especially considering that those with wide clefts
did not have a higher chance of poor facial growth.
There are other variables when comparing cohorts of patients having anterior-first versus posterior-first sequencing of
surgery. One is that many European surgeons have abandoned
Maleks original protocol of soft palate alone, and now combine lip and soft palate as the first operation around age 6
months, then completing the hard palate repair, often with a
gingivoperiosteoplasty at 24 to 36 months. Many surgeons using an Oslo-style technique do not have continuity of the
vomerine flap into the floor of nose and lip repair, and thus an
alveolar gap remains until the later bone graft. Abyholm used
a supraperiosteal dissection for his lip repairs and many surgeons, although using his sequence, do a subperiosteal dissection for the lip closure. All these many variables make comparisons between techniques difficult, but in this series the surgical techniques were uniform for all patients. Only the sequence of the repair was compared.
4. One of the major outcomes of cleft lip and palate repair
is midfacial growth, and 4 to 7 years after completing the cleft
closure in our patients there was no significant difference in
facial growth between the two types of repair sequencing as
assessed by incisor overjet. There is considerable debate as to
the factors determining a poor outcome. These include an intrinsic deficiency of the maxilla or of its growth potential, a
reduction in growth brought about by the surgical method of
cleft closure, the sequencing of the cleft closure, or the skill
of the person carrying out the closure.
The original protocol for this project was based on seeking
a difference in the GOSLON score of 0.75 at age 10, thus
requiring 19 patients for a two-group comparison. We achieved
these numbers in randomization, but too few children have
sufficient permanent teeth to allow for this analysis yet. These
interim results regarding facial growth as a result of the two
sequences of closure finds no significant difference. However,
the overjet was measured on an age-mixed cohort, including
patients in deciduous, mixed, and permanent dentitions, which
has increased the variability within this measure. The delay
between operations of only 3 months may be an issue, because
other surgeons leave a 6-, 9-, or 18-month gap between closure
of the soft palate and subsequent hard palate closure. We do
not know what effect a longer time interval would have on
hard palate narrowing and whether any narrowing would represent a change in palatal shelf position and/or relative narrowing of intertuberosity distance. Intrinsic deficiency aside,
we do not know whether it is the surgeon or the technique that
has the strongest effect on outcome measurements. Evidence
that it is the surgeon rather than the procedure that delivers
good speech results and facial growth comes from cohort studies of single operators using techniques that others have used
with inferior results (Choudhary et al., 2003; Ross, 1987).

Richard et al., PRIMARY REPAIR OF UCLP: RESULTS OF AN RCT

There is also evidence from comparisons of junior and senior


cleft surgeons; the repeat comparison of the same people a few
years later shows that their speech results depended more on
their innate ability than on their training and experience (Rintala and Haapanen, 1995). The conclusion from Rosss (1987)
study was that some surgeons cause less growth inhibition than
others. However, there is evidence that it is the procedure rather than the surgeon that gives good speech results and facial
growth. Schweckendiek and Doz (1978) showed good facial
growth with a late hard palate repair, implying that it is the
surgery itself that is the problem. There has been a trend toward von Langenbeck from Veau-type palate repairs in the
belief that the von Langenbeck dissection is less traumatic to
the maxillas growth potential (Choudhary et al., 2003). Pigott
had a working lifetime of three patient cohorts where he
changed from a Veau to von Langenbeck and then to medial
von Langenbeck dissections and has showed a strong trend of
improvement in anteroposterior facial growth (Pigott et al.,
2002). The trend was nonsignificant. This may represent inadequate sample size or it could be his personal learning curve.
There are several large studies (Ross, 1987; Bearn et al.,
2001; Sandy et al., 2001; Sell et al., 2001; Williams et al.,
2001; Shaw et al., 2001; Williams and Sandy, 2003) that have
failed to determine the cause of poor facial growth. Attributing
responsibility neither to the surgeon nor any technique, they
have pleaded for organizational change to allow for protocoldriven management.
So where do we go from here? In the U.K., there now exist
larger regional centers with fewer operators carrying larger
caseloads. We may be in a position to test surgical techniques
and protocols, and the surgeons who carry them out. We know
little about the parameters that determine the optimal outcome,
so every patient should be part of our endeavor to measure
outcome and test what issues are important for that outcome.
Acknowledgments. We thank Binod Koriala for his organizational and administrative skills in managing the cleft database in Pokhara, Rosie Sleator for local
speech and language input, Dr. Paul Davies and Dr. Gill Lancaster for their
statistical advice and to the British Cleft Lip and Palate Association and the
Craniofacial Society of Great Britain & Ireland for financial support.

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