A two-centre study on facial morphology in patients with complete bilateral cleft lip and palate at nine years of age

Abstract

The aim of this study was to compare craniofacial morphology and soft tissue profiles in patients with complete bilateral cleft lip and palate at 9 years of age, treated in two European cleft centres with delayed hard palate closure but different treatment protocols. The cephalometric data of 83 consecutively treated patients were compared (Gothenburg, N = 44; Nijmegen, N = 39). In total, 18 hard tissue and 10 soft tissue landmarks were digitized by one operator. To determine the intra-observer reliability 20 cephalograms were digitized twice with a monthly interval. Paired t -test, Pearson correlation coefficients and multiple regression models were applied for statistical analysis. Hard and soft tissue data were superimposed using the Generalized Procrustes Analysis. In Nijmegen, the maxilla was protrusive for hard and soft tissue values ( P = 0.001, P = 0.030, respectively) and the maxillary incisors were retroclined ( P < 0.001), influencing the nasolabial angle, which was increased in comparison with Gothenburg ( P = 0.004). In conclusion, both centres showed a favourable craniofacial form at 9–10 years of age, although there were significant differences in the maxillary prominence, the incisor inclination and soft tissue cephalometric values. Follow-up of these patients until facial growth has ceased, may elucidate components for outcome improvement.

A favourable facial morphology is one of the key aspects of treatment outcome in patients with cleft lip and palate. Many studies have addressed this topic for patients with unilateral cleft lip and palate, but relatively few cephalometric studies on patients with complete bilateral cleft lip and palate (CBCLP) have been published. This is probably due to the low incidence of CBCLP compared with the rest of the cleft population . For patients with unilateral cleft lip and palate (UCLP), cephalometric studies revealed a pattern of consistency in the results at age 12 and 17 years, with ratings at the age of 9 years . Therefore, age 9–10 years was indicated as the target age for the assessment of facial growth in the record guideline specifications of the Eurocleft project . For CBCLP only 10 studies could be identified that report cephalometric data at the target age. Table 1 gives an overview of these studies .

Table 1
Overview of studies that show cephalometric data of patients with CBCLP at 9–10 years.
Authors Year Country Design Consecutive Target group N target Comparison N comp Variables studied
K rogman et al. 1982 USA CS retro ? BCLP centre’s protocol 22 UCLP 45 Lateral cephalogram hard tissue
CPO 50
P eat 1982 New Zealand CCT ? BCLP infant orthopaedics 14 BCLP no infant orthopaedics 12 Lateral cephalogram hard tissue
V argervik 1983 USA CS retro no CBCLP no premaxillary setback 17 CBCLP early premaxillary setback 12 Lateral cephalogram hard tissue
S emb 1991 Norway CS retro ? CBCLP centre’s protocol 57 CUCLP 5–18 years centre’s protocol 45–60 Frontal and lateral hard and soft tissue
H eidbüchel at al. 1994 Netherlands CS retro no BCLP centre’s protocol 16 CBCLP Oslo 90 Lateral cephalogram hard and soft tissue
G aukroger at al. 2002 UK; Norway CS retro ? BCLP centre’s protocol 13 CBCLP Oslo 26 Lateral cephalogram hard and soft tissue
S ilvera et al. 2003 Japan CS retro no BCLP two stage palatoplasty, Hotz plate 10 BCLP different treatment protocols 11 Frontal and lateral hard and soft tissues
L isson et al. , 2005 Germany no BCLP centre’s protocol 11 UCLP 12 Lateral cephalogram hard tissue
Non-cleft controls 20
G noinski and R utz 2009 Switzerland CS retro yes BCLP centre’s protocol 29 BCLP 5, 15, 19 years centre’s protocol 29 Lateral cephalogram hard and soft tissue
H olst et al. 2009 Germany CS retro ? BCLP centre’s protocol 21 UCLP 52 Lateral cephalogram hard tissue
Non-cleft controls 53

CS: case series; CCT: controlled clinical trial not randomized; retro: retrospective study; CUCLP: complete unilateral cleft lip and palate; CPO: cleft palate only.

L isson et al. is a double publication of L isson et al. , therefore only one has been included.

Only one study presented in the literature on CBCLP at the age of 9–10 years had a sample size of over 50 patients: the largest sample at this age is the Oslo sample with 57 patients . In the other studies the sample size ranges from 11 to 29 . Some of the studies in Table 1 are longitudinal observational studies, following their sample from 1 month until adulthood, and presenting data at 9 years of age . Certain studies compared their group with normative data for a non-cleft population , or with the protocols of other centres . In other studies, different treatment approaches within the same cleft centre were compared . CBCLP patients’ skeletal development was compared with UCLP patients in other studies .

From the published literature, the authors conclude that knowledge of facial morphology in patients with CBCLP is limited and potentially biased, as sample sizes are small and in all but one study, cases were not reported consecutively. Only two intercentre studies have been performed in which the outcome of CBCLP patients was compared . Delayed hard palate closure was not considered in these studies.

This study aims to compare the craniofacial morphology of patients with CBCLP at 9 years of age, consecutively treated in two European cleft centres with different protocols but with similar timing of delayed hard palatal closure. It examines the hypothesis that treatment outcome for craniofacial morphology at the two centres is the same.

Material and methods

Two cleft centres participated in this study: Gothenburg (Sweden, centre A) and Nijmegen (The Netherlands, centre B). Table 2 shows the treatment protocols used at the two centres. Lateral cephalograms of 83 consecutively treated patients with CBCLP from these two centres who were about 9 years of age were evaluated (Gothenburg N A = 44, 33 males, 11 females; Nijmegen N B = 39, 34 males, 5 females).

Table 2
Treatment protocols (primary procedures for lip, alveolus, and palate) for patients with a complete bilateral cleft lip and palate from birth until 10 years of age of the cleft palate centres in this study. Gothenburg: Centre A, Nijmegen: Centre B.
Age Centre A Centre B
Birth Infant orthopaedics, duration 1.5 years Infant orthopaedics with extra-oral strapping
Nose plugs Mean duration 9.2 months
Duration 2.5 years
3 months Bilateral lip adhesion
Mean age 3.3 months
6 months Soft palate closure One–stage lip closure (modified Manchester)
(centre’s own technique) Mean age 7.2 months
Mean age 8.5 months
12 months Modified Von Langenbeck
soft palate closure
Mean age 13.8 months
18 months Definitive bilateral lip and nose repair
Mean age 18 months
(centre’s own technique)
4 years Von Langenbeck hard palate closure (before 1975)
Mean age 3.8 years
9 years One side alveolar bone grafting (tibia) Hard palate closure and bilateral alveolar bone grafting (chin) (after 1975) and osteotomy of the premaxilla
Mean age 8.0 years Mean age 9.9 years
Hard palate closure with alveolar bone grafting of second side.
Mean age 8.5 years

The inclusion criteria were: CBCLP with a diagnosis confirmed by the preoperative written records, neonatal pictures of the face and/or casts taken preoperatively; patients with Simonart’s band(s) were included only if no hard tissue union was present (the side of the Simonart’s band was noted); Caucasian ethnic background; no associated congenital malformations, syndromes or mental retardation; treatment from birth onwards in the same centre; age 8–10 years; born before 1996.

Lateral cephalograms were available, taken in centric occlusion and oriented to the Frankfurt horizontal plane. The cephalograms from both centres were scanned on a 12-bit scanner (R2 ImageChecker M5000 DM, R2 Technology, Inc., Sunnyvale, CA, USA) at 150 dpi. All cephalograms were digitized with a commercially available software programme for cephalometric analysis (Viewbox 3/dHAL Software, Kifissia, Greece). The cephalometric reference points (18 hard and 10 soft tissue landmarks) used in the study are shown in Fig. 1 . From these landmarks, 21 cephalometric variables were calculated. To avoid errors due to magnification differences between the two centres only angular measurements and one ratio were used. All cephalograms were digitized by one operator (TB). To determine the measurement error, 20 randomly selected cephalograms were digitized twice by the same operator with a time interval of 1 month.

Fig. 1
Reference points on the profile cephalometric radiographs.

Skeletal reference points:
S Sella. The geometric centre of the Sella turcica.
N Nasion. The most anterior point at the frontonasial suture.
ANS Anterior nasal spine.
A Point A. The deepest point on the anterior contour of the upper alveolar process.
As Apex superius. The apex of the root of the upper central incisor.
Ls Incision superius. The incisal edge of the most prominent upper incisor.
Li Incision inferius. The incisal edge of the most prominent lower incisor.
Ai Apex inferius. The apex of the root of lower central incisor.
B Point B. The deepest point of the anterior contour of the lower alveolar process.
Pg Pogonion. The most anterior point of the mandibular symphysis.
Gn Gnathion. The most anterior inferior point of the bony chin.
Me Menton. The most inferior point of the mandibular symphysis.
Go Gonion point. Most posterior inferior point on the angle of the mandible.
Mtp Mandibular Tangent Posterior. The most posterior inferior point on the outline of the mandibular body.
R Ramus point. The most posterior-inferior point of the mandibular ramus.
Ar Articulare. The constructed point at the intersection of the images of the posterior margin of the ramus and the outer margin of the cranial base.
Ba Basion. The lowest point on the anterior margin of the foramen magnum in the median plane.
Pm Pterygo-maxillare. The intersection of the nasal floor and the apex of the pterygomaxillary fissure .
Soft tissue reference points:
n Soft tissue nasion. The deepest point on the frontonasal curvature.
an Anterior nasalis. The most prominent point on the nose tip.
sn Soft tissue subnasale. The point of intersection between the base of the nose and upper lip of soft tissue.
ss Soft tissue subspinale. The point of greatest concavity in the midline of the upper lip.
ls Labrale superius. The most prominent point of the upper lip.
li Labrale inferius. The most prominent point of the lower lip.
sm Soft tissue supramentale. The point of the greatest concavity in the midline of the lower lip.
pg Soft tissue pogonion. The most prominent point on the soft tissue of the chin.
gn The most anterior inferior point of the soft tissue chin.
me Soft tissue menton. The lowest point on the lower border of the mandible.
Reference lines:
SN Nasion-Sella line.
NL Nasal line. The line through pterygo-maxillare (Pm) and anterior nasal spine (Ans).
ILs Axis of upper incisors.
ILi Axis of lower incisors.
ML Mandibular line. The tangent of the lower border of the mandible through menton (Me) and Mandibular Tangent Posterior (Mtp).
RL Ramus line. The line through articulare(Ar) and ramus point (R).
E-line Aesthetic line. The line through soft tissue anterior nasalis (an) and soft tissue pogonion (pg).
Hard tissue angles:
SNA
SNB
ANB
SNPg
ILS-NL
ILS-SN
ILI-ML
SN-NL
SN-ML
NL-ML
RL-ML = Gonial angle
N-S-Ba
S-N-Ba
S-Ba-N
Soft tissue angles:
S-n-an
S-n-ss
S-n-sm
S-n-pg
n-an-pg
an-sn-ls = Nasolabial angle
n-sn-pg
Ratios:
Posterior facial height/anterior facial height = (Post-AnteFH) SGo/NMe.
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Feb 5, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on A two-centre study on facial morphology in patients with complete bilateral cleft lip and palate at nine years of age
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