Abstract
The purpose of this review was to evaluate the effect of 1-stage versus 2-stage palate repair with delayed hard palate closure on facial growth in patients with cleft lip and palate. A literature survey from the PubMed database from January 1966 to December 2007 used the medical subject headings terms ‘facial growth’, ‘cleft lip and palate’, ‘cephalometry’, and ‘1-stage palate repair’ in combination with ‘2-stage palate repair’ or ‘delayed hard palate closure’. The Cleft Palate-Craniofacial Journal from 1964 to November 2007 was hand searched. Controlled studies written in English were selected. Two reviewers selected and extracted the data independently and assessed the quality of the studies. On the basis of this search only nine studies were included in this review. All studies were retrospective and non-randomized. Six studies were cross-sectional, and three were longitudinal. Heterogeneity and methodological deficiencies in the studies prevented conclusions regarding stage of palate repair and facial growth. Further well-designed controlled studies and long-term studies are needed, and researchers should focus on the variation in hard palate repair timing before 3 years of age and consider patient speech.
Hard palate repair has been associated with maxillary hypoplasia . Theoretically late hard palate repair should result in better growth, because possible interference with maxillary growth is postponed to a later age when less growth remains. The idea is supported by the good growth results following the S chweckendiek technique . This idea has led to the development of a clinical protocol of delayed hard palate closure. This protocol involves staged palate repair with closure of the soft palate in infancy and the use of a speech prosthesis until closure of the hard palate at the age of late primary or early mixed dentition.
Evidence for the benefit of this staged palate repair on maxillary growth has been scanty, though L ilja et al. provided some evidence that maxillary growth using the staged repair with hard palate closure at 9 years of age was superior, and S ilvera et al. found that patients who had staged repair with hard palate closure at 6 years of age had more favourable maxillary growth than did those who underwent 1-stage repair at 1.5 years of age. F riede et al. questioned whether it is necessary to delay closure until 9 years of age rather than 5 years, because similar and satisfactory maxillary growth was found in two samples in which patients underwent surgery at the two different ages. They speculated that by starting closure of the soft palate, the size of the remaining hard palate narrows spontaneously , facilitating a smaller, less invasive, later hard palate repair. This results in the closure of the residual defect without leaving any raw palatal bony surfaces open to secondary epithelialisation. R oss reported no maxillary growth difference between patients with repair of the palate in 1 or 2 stages. S mahel et al. found no difference between repair in 1 stage at 3 years and 2 stages with hard palate closure at 6 years. R ohrich et al. found no difference between repair in 1 stage at 11 months and in 2 stages with hard palate closure at 4 years; and S tein et al. found no difference between 1-stage repair at 2 years and 2-stage repair with hard palate closure at 7 years. H olland et al. found that 1-stage palate repair at 12 months provided better maxillary growth than did 2-stage palate repair with hard palate closure delayed until 7 years. They ascribed the relative maxillary hypoplasia to palatal scarring caused by the pin-retained speech prosthesis and/or the increased number of secondary surgical procedures for speech.
To evaluate the effect of 1-stage versus 2-stage palate repair with delayed hard palate closure on facial growth in patients with cleft lip and palate, a systematic review was undertaken. The authors aimed to answer the following questions in patients with cleft lip and palate. Does stage of palate repair have an influence on the growth of the cranial base? Does 2-stage palate repair lead to better maxillary forward growth than 1-stage repair? Does stage of palate repair have an influence on the growth of the mandible? Does 2-stage palate repair lead to better antero-posterior jaw relation than 1-stage repair?
A quality analysis of the methodological soundness of the studies in the review was performed.
Methods
To identify all studies that examined the relationship between stage of palate repair and facial growth, a literature survey was performed using the following criteria. The Medline database (EntrezPubMed, www.ncbi.nim.nih.gov ) from January 1966 to December 2007 was searched using the medical subject headings terms ‘facial growth’, ‘cleft lip and palate’, ‘cephalometry’, and ‘1-stage palate repair’ in combination with ‘2-stage palate repair’ or ‘delayed hard palate closure’. The Cleft Palate-Craniofacial Journal (formerly The Cleft Palate Journal ) from 1964 to November 2007 was hand searched. The reference lists of all relevant publications were reviewed to identify any publications, not already identified using the two search strategies.
Controlled studies published as full-length articles reporting quantitative data on the effect of stage of palate repair on facial growth were selected. No restrictions were placed on sample size, but case series without controls, case reports, abstracts, letters, and review articles were not considered. Only papers written in English were included. Two independent reviewers assessed all the articles regarding the inclusion and exclusion criteria. Inter-examiner conflicts were resolved by discussion of each article to reach a consensus.
Data were extracted on the following items: year of publication, design, sample size, material and age, methods and measurements, and outcomes (for details see Tables 1 and 2 ). To document the methodological soundness of each article, a quality evaluation using a checklist was performed (for details see Table 3 ). The data were extracted from each article by two independent evaluators without blinding. Inter-examiner conflicts were resolved by discussion of each article to reach a consensus.
Study number/author(s) | Design | Sample size | Cleft type and subtype | Population studied | Surgery | Age at assessment (years) | ||
---|---|---|---|---|---|---|---|---|
Sequence (mean age at surgery in months) | Technique of hare palate repair | No. of surgeon(s) | ||||||
1. Ross | Cross-sectional | 247 | CUCLP (247M) | (a1) | L (?) → SP + HP (<20) | ? | >1 | 15 |
Multicenter | 133 | CUCLP (133M) | (a2) | L (?) → SP (?) → HP (<11–108) | ? | >1 | 15 | |
2. M olsted et al. | Cross-sectional | 23 | CUCLP (14M; 9F) | Sweden | L (3) → SP (9) → HP (108) | (V)(P)MF | 6 | 8–10 |
Multicenter | 23 | CUCLP (16M; 7F) | Netherlands | L (6) → SP + HP (12) | ? | 10 | 8–10 | |
26 | CUCLP (16M; 10F) | Sweden | L (6) → SP + HP (<24) | ? | 9 | 8–10 | ||
23 | CUCLP (10M; 13F) | UK | L (5) → SP + HP (12) | PMF (Pushback) | 5 | 8–10 | ||
3. S mahel et al. | Cross-sectional | 24 | CUCLP (17M; 7F) | Czeck | L (7) → SP + HP (48) | PMF (Pushback) | 1 | 10.4 |
27 | CUCLP (27M) | Czeck | L + SP (6.9) → HP (73) | PMF (Pushback) | >1 | 10.3 | ||
4. R ohrich et al. | Cross-sectional | 21 | 15CUCLP, 6CBCLP (12M; 9F) | UK | L (3) → SP + HP (11) | PMF (Pushback) | 1 | 17 (14–21) |
23 | 16CUCLP, 7CBCLP (14M; 9F) | UK | L (3) → SP (11) → HP (49) | VMF | 1 | 18 (15–19) | ||
5. S ilva F ilho et al. | Cross-sectional | 53 | CUCLP (33M; 20F) | Brazil | L (9) → SP + HP (19) | PMF (vL) | 2 | 4–7 |
22 | CUCLP (12M; 10F) | Brazil | L + SP (5.5) → HP (20) | PMF (Malek) | 1 | 4–7 | ||
6. S ilvera et al. | Longitudinal | 11 | CBCLP (4M; 7F) | Japan | L (3–11) → SP + HP (18) | PMF (Pushback) | 4 | 6, 8, 10, 12 |
10 | CBCLP (5M; 5F) | Japan | L (5&8) → SP (18) → HP (72) | (V)(P)MF | 1 | 6, 8, 10, 12 | ||
7. C orbo et al. | Longitudinal | 11 | CUCLP (8M; 3F) | Belgium | L + SP + HP (3) → A (120) | PMF (Malek) | ? | 7, 12 |
10 | CUCLP (4M; 6F) | Belgium | SP (3) → L + HP (6) → A (120) | PMF (Malek) | ? | 7, 12 | ||
8. S tein et al. | Longitudinal | 22 | CUCLP (15M; 7F) | Germany | L (4) → SP + HP (23) → A (150) | PMF | >1 | 6, 10, 15, 18 |
Multicenter | 21 | CUCLP (14M; 7F) | Germany | L (4) → SP (23) → HP (86) → A (127) | PMF | >1 | 6, 10, 15, 18 | |
9. H olland et al | Cross-sectional | 41 | CUCLP (23M;18F) | USA | L (?) → SP + HP (12) | VMF | 4 | Adults |
41 | CUCLP (25M;16F) | USA | L (?) → SP (12) → HP (84) | PMF (Modified vL) | 4 * | Adults |
Variables | 1 | 2 | 3 | 4 | 5 | 6 | 7 | 8 | 9 |
---|---|---|---|---|---|---|---|---|---|
Cranial base | |||||||||
S-N (mm) | X | X | |||||||
Cranial base angle (N-S-Ba,°) | X | X | X | X | |||||
Maxilla | |||||||||
Pharynx (Ba-PMP, mm) | ↑ | ||||||||
Length (mm) | X PMP-ANS ↓PMP-A |
X PMP-A | X PMP-ANS | X PMP-ANS | |||||
Protrusion (°) | X SNA | X SNA | X SNA | X SNA X S-N-ANS |
X SNA ↑/X FH-NA(a) |
X SNA | X SNA | ↓SNA | |
Anterior height (mm) | ↓N-ANS | X N-ANS | X N⊥PP | ||||||
Posterior height (mm) | X R-PMP | X S-PMP | ↑S⊥PP | ||||||
SN-PP (°) | X | X | X | ||||||
Mandible | |||||||||
Length (mm) | X Co-Gn | X Go-Gn+Co-Go | X Go-Pog | ||||||
Protrusion (°) | X SNB | X S-N-Pog | X SNB | X SNB,S-N-Gn | X SNB | X SNB | X SNB | X SNB | |
Gonial angle (°) | X Ar-Go-Gn | X Co-Go-Gn | X Co-Go-Gn | X Ar-Go-Gn | |||||
SN-MP (°) | ↓ | X | X | X | X | X | |||
Jaw relation | |||||||||
ANS-N-Pog (°) | X | X | |||||||
ANB (°) | X | X | X | ↑/x (a) | X | X | ↓ |