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 .
| 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 |
† 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).
| 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.
| 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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