Effect of Cleft Types on Outcome of Unilateral Cleft Lip Repair.

INTRODUCTION: It is generally assumed that the major phenotypes of unilateral cleft lip (UCL) represent its extent of severity. There are a few studies on this, but objectivity has been lacking. It was the aim of this study to assess the effect of different phenotypes of UCL on the outcome of surgical repair.
METHODOLOGY: This study was a case series of the effect of phenotypes of cleft on the outcome of repair of UCL. Fifteen subjects each were in three UCL phenotype groups. Surgical outcome was assessed quantitatively with anthropometric measurements recorded from a full frontal face photograph of subjects. The analysis was done using Student's t-test and one-way ANOVA at P = 0.05.
RESULTS: The mean values of the preoperative horizontal and vertical lip height, and nasal width on the cleft side in the cleft lip (CL) group were closest to those of the noncleft side and control. The postoperative mean values of the horizontal and vertical lip height, and nasal width on the cleft side in the CL group were closest to those of the noncleft side and control, while those of the CL, alveolus and palate group were farthest from those of the noncleft side and the control. DISCUSSION: The different phenotypes of UCL have different degrees of tissue distortion and deficiencies. CL group had its measurements closest to that of the noncleft sides and control, suggesting that it has the least distortion.
CONCLUSION: The comparison between the three groups did not reveal any difference, suggesting that the skill of the surgeon and the selection of a well proven technique are more important factors in the outcome of repair of unilateral cleft lip. Copyright:

INTRODUCTION

The facial muscles are divided into three muscular rings. The first ring is formed by the nasolabial group, second by the bilabial group and the third by the labiomental ring.[12] All the three rings are disrupted when cleft of the lip with/without palate occurs.[2] The resultant disturbance in muscular function within these anatomic muscular units secondarily produces distortions in the growth of surrounding skeletal and cartilaginous structures that theoretically escalate over time. Accurate reconstruction of these different muscular layers of the lip is important for normal lip function, and also to stop further distortion of underlying hard tissue structures.[2]

The severity of unilateral cleft lip (UCL) has been correlated with the extent of facial tissue distortion.[3] Even though no objective index is available, as at the time of this study, it is generally assumed that the major phenotypes of UCL (cleft lip [CL] only, cleft lip and alveolus [CLA], and CL alveolus and palate [CLAP]) represent the extent of severity in that order.[4] And so the degree of tissue distortion might affect the degree to which the tissue can be apposed in UCL repair, ultimately affecting the outcome of surgical repair.

The ideal operation for the repair of a UCL should result in a symmetrical upper lip and nose, with the equal philtral column length on either side.[5] In addition, the Cupid's bow should be of adequate proportions. These criteria should be obtainable in a single operation without multiple minor revisional procedures.[5] The range of outcome of the treatment of CL and palate can, however, be considerable due to many variables including types/severity of cleft, variation in the sequence, timing and technique of treatment, the organization and delivery of care, as well as in the skills and experience in the individual surgeons.[67] On this premise, evaluation of treatment outcome is essential to allow for identification and implementation of the highest possible standards of care, as it gives a foreknowledge of area of cleft repair that are likely to necessitate the use of secondary techniques and adjunct flaps to achieve an ideal outcome. It has been reported that anthropometry and clinical examination best evaluate the morphology of repaired CL and nose[8] because anthropometry can show quantitatively the degree of deformity present in clefts

Justification

Studies that have quantitatively determined the extent of the effect of various types of UCL on the surgical outcome of repair of UCL are few,[91011] and to the best of our knowledge, none at all in Nigeria and the sub-Saharan Africa, so this study aimed to determine if there is association between types of UCL and outcome of surgical repair.

Study hypothesis

The study was based on the hypothesis that the type of UCL has effect on the surgical outcome of repair of UCL.

METHODOLOGY

This study was a case series of effect of the three types of UCLs (CL only, CLA, and CLAP) on treatment outcome of surgical repair of UCL. It was carried out between January 1, 2013 and July 31, 2014. The study was approved by the Health Research and Ethics Committee on October 9, 2012 (IRB number 34,512).

Sample size for this study was determined by using the formula for cohort and case–control study as proposed by Schlesselman.[12] The attrition rate was taken as 10% to cater for dropouts, so the sample size was 27.5, approximated to 28, nevertheless, 45 were used. This was statistically stratified into three to make each group (CL only, CLA and CLAP) contain 15 participants.

The participants were recruited from the cleft clinic of a University Hospital.

The aim was to evaluate the effect of types of UCL on treatment outcome, following repair of UCL.

The objectives are

To evaluate the surgical outcome of repair of CL only

To evaluate the surgical outcome of repair of CLA

To evaluate the surgical outcome of repair of CLAP

To compare the surgical outcome of repair of CL only, CLA, and CLAP.

Inclusion criteria

Participants with complete UCL with or without alveolus and palate

Participants who were 3 months old and above.

Exclusion criteria

Individuals with bilateral CL with or without palate

Individuals with syndromic cleft of the lip and/or palate

Individuals who were <3 months old

Individuals who required CL revision following primary CL repair

Individuals who were not fit for general anesthesia.

Written informed consents were obtained from parents/guardian of all participants before enrolment in the study [Appendix I]. Prior to this, detailed information and explanations of the study were given to the parents or guardians. Every one of them was given an opportunity to ask questions concerning the study, and appropriate clarifications were given before the commencement of the study. Opportunity to withdraw at any stage of the study was also made known without victimization or denial of treatment.

Data collection

The following data were recorded preoperatively on a pro forma: age, sex, weight, height, and type of clefts.

Preoperative evaluation

Routine preoperative blood investigations including hemoglobin estimation, electrolyte, urea and creatinine level, and electrocardiography was done for each subject. Echocardiography was also done when indicated. Participants were referred to the pediatrician for clinical evaluation, to rule out cardiovascular congenital anomalies, upper respiratory tract infection, ear infection, and other congenital anomalies that may be of clinical significance. Participants were at least 3 months old, 4.5 kg (10 pounds) with a minimum hemoglobin concentration of 10 g/dL.

Standardized preoperative photographs were taken for all participants before the surgeries were performed from which preoperative anthropometry was done as explained below in postoperative anthropometry.

Operative procedure

All participants had their surgical repair done under general anesthesia with endotracheal intubation. Two oral and maxillofacial surgeons that were skilled in Millard-Rotation advancement technique of CL repair with the same number of years of experience performed the operation.

Surgical outcome was done by using anthropometry as stated by Cutting and Dayan,[13] because it employed two-dimensional (2D) (photography) anthropometry where image acquisition is relatively simple, equipment are cheap and a very high level of technical expertise is not needed. It also correlates very well with clinical assessment. Anthropometric measurements were recorded from a 2D full frontal face photograph of subjects taken with a digital camera (Canon PowerShot SX500 IS, 30x optical zoom, 16MP, 24–720 mm wide angle). For standardization, each photograph was taken with the camera placed at 45 cm from each participant and the interpupillary plane parallel to the floor (the subjects were positioned in a way that the lens of the camera is perpendicular to the interpupillary line). The photographs were then imported into Adobe Photoshop software (Adobe Systems, Mountain view, California, 2009), for analysis.

These anthropometric measurements were recorded [Figure 1a and b]:

Figure 1

(a) Preoperative anthropometry. (b) Postoperative anthropometry reference point

Preoperatively;

Vertical lip height on the noncleft side: measured from the alar base to the peak of the Cupid's bow on the same side

Vertical lip height on the cleft side: measured from the alar base on the cleft side to a point where the white roll just starts to disappear

Horizontal lip height on the noncleft side: measured from the peak of the Cupid's bow on the noncleft side to the ipsilateral commissure of the mouth

Horizontal lip height on the cleft side: measured from where the white roll starts to fade out to the ipsilateral commissure

Nasal width: measured from the alar base to the mid-point of the columella for both sides

Total nasal width: measured from the alar base on cleft side to alar base on noncleft side.

Postoperative measurement; this was done 3 months after the surgery.

Vertical lip height: measured from the alar base to the peak of the Cupid's bow, for both cleft and noncleft sides

Horizontal lip height: measured from the peak of the Cupid's bow to the commissure for both cleft and noncleft sides

Nasal width: measured from the alar base to the mid-point of the columella for both cleft and noncleft sides

Total nasal width: measured from the alar base on cleft side to alar base on noncleft side

Philtral height: measured from the peak of the Cupid's bow to the mid-point of the columella for both cleft and noncleft sides

Cupid's bow width: measured from the peak of the Cupid's bow on one side to the peak on the other side.

Confounders

To minimize confounders as much as possible, the following steps were taken;

The camera has a stand that was permanently located at the distance, 45 cm, as stated above

Several photographs were taken of the participants out of which the best was chosen by the lead author and imported into the Adobe software, stated above

All participants with microform or incomplete CL phenotype were excluded from the study.

Age-matched controls were recruited from the outpatient immunization clinic of Community Health. Vertical lip height, horizontal lip height, nasal width, total nasal width, philtral height, and Cupid's bow width were measured as stated for postoperative measurement above.

Data analysis was performed using Statistical Package for Social Sciences (SPSS) for Windows (version 23.0, SPSS Inc., Chicago, IL, USA). Means of each variable measured preoperatively and postoperatively were generated for each group, and a comparison of these, cleft side with noncleft side and the control, was done, using Student's t-test. In addition, the mean values of the measured variables in each group were compared within the three groups using one-way ANOVA. Statistical significance was P ≤ 0.05.

RESULTS

A total of 45 participants requiring repair of UCL were enrolled for the study and analyzed. Fifteen participants each were in CL, CLA, and CL, alveolus and palate groups. Of the 45, 25 (55.6%) were males, and 20 (44.4%) were females, in the ratio of 1.3:1.

The preoperative mean values of the horizontal lip height, vertical lip height, and nasal width on the cleft side in the CL group were closest to those of the noncleft side and control. The comparisons did not show any statistically significant difference for horizontal lip height (cleft side and noncleft side and the control) and nasal width (cleft side and the control) in the CL group, as shown in Table 1.

Table 1

Preoperative anthropometry measurements of cleft side and noncleft side and control in the three groups

Variables	CL group		CLA group		CLAP		P
	Mean	SD	Mean	SD	Mean	SD	CL	CLA	CLAP
Horizontal lip height
Cleft side	15.80	5.20	15.21	2.23	14.67	2.78
Noncleft side	17.55	3.81	18.11	3.37	16.93	3.50	0.084	0.001*	0.013*
Control	18.04	2.72	19.10	3.48	18.24	2.98	0.182	0.003*	0.008*
Vertical lip height
Cleft side	13.97	6.47	12.16	3.54	11.99	2.04
Noncleft side	16.20	6.81	14.84	4.60	15.36	3.28	0.007*	0.001*	*0.001
Control	15.39	1.80	16.25	2.82	15.49	1.68	0.387	0.003*	0.001*
Nasal width
Cleft side	22.45	5.59	22.40	3.79	24.31	3.14	0.001*	0.001*	0.001*
Noncleft	17.47	4.81	15.83	3.17	16.45	2.28
Total nasal width of subject	38.99	10.46	38.79	6.47	40.09	5.35	0.011*	0.010*	0.001*
Total nasal width of control	31.73	2.81	31.45	5.93	31.14	1.70

*Significant at P≤0.05. SD: Standard deviation, CL: Cleft lip, CLA: Cleft lip and alveolus, CLAP: Cleft lip, alveolus and palate

Comparison of postoperative anthropometry measurement of cleft side with noncleft side and the control in the three groups

The mean values of the horizontal lip height, vertical lip height and nasal width on the cleft side in the CL group were closest to those of the noncleft side and control while those of the CL, alveolus and palate group were farthest from those of the noncleft side and the control [Table 2].

Table 2

Postoperative anthropometry measurements of cleft side and noncleft side and control

Variables	CL group		CLA group		CLAP		P
	Mean	SD	Mean	SD	Mean	SD	CL	CLA	CLAP
Horizontal lip height
Cleft side	17.60	2.86	19.19	4.11	17.36	3.22
Noncleft side	18.55	3.06	20.50	3.85	19.38	3.00	0.082	0.034*	0.010*
Control	18.04	2.72	19.10	3.48	18.24	2.98	0.651	0.947	0.412
Vertical lip height
Cleft side	16.98	3.17	16.48	4.21	16.76	3.03
Noncleft side	18.77	3.88	18.37	3.61	18.80	2.60	0.038*	0.002*	0.001*
Control	15.39	1.80	16.25	2.82	15.49	1.68	0.138	0.838	0.180
Nasal width
cleft side	17.89	2.84	20.06	4.19	18.79	3.15	0.527	0.003*	0.048*
Noncleft	17.52	2.48	17.00	2.71	17.42	2.20
Total nasal width of subject	35.41	4.85	37.06	6.18	35.87	4.90
Total nasal width of control	31.73	2.81	31.45	5.93	31.14	1.70	0.009*	0.012*	0.004*
Philtral height
Cleft side	12.59	2.14	12.70	2.00	12.54	1.66
With noncleft side	13.90	2.78	14.06	2.66	14.33	2.51	0.088	0.013*	0.001*
With control	12.07	1.03	12.43	2.25	11.88	1.86	0.314	0.718	0.295
Cupid’s bow width
Cleft subjects	12.02	2.66	12.08	1.95	11.76	2.34
Control	10.08	1.25	10.27	1.39	9.37	1.17	0.029*	0.015*	0.007*

*Significant at P≤0.05. SD: Standard deviation, CL: Cleft lip, CLA: Cleft lip and alveolus, CLAP: Cleft lip, alveolus and palate

Cleft lip only

The comparison showed that, there was no difference between the horizontal lip lengths of the cleft side, noncleft side and the control; no difference between the vertical lip heights of the cleft side and the control; no difference between the nasal widths of the cleft side and the noncleft side, and also, no difference between the philtral heights of the cleft side, noncleft side and the control.

Cleft lip and alveolus

The comparison showed that, for both horizontal and vertical lip heights, there was no difference between those of the cleft side and the control and also no difference between the philtral heights of the cleft side and those of the control.

Cleft lip, alveolus, and palate

The comparison was essentially like that of CLAP. It showed that, for both horizontal and vertical lip heights, there was no difference between those of the cleft side and the control and also no difference between the philtral heights of the cleft side and those of the control.

Comparison of postoperative anthropometry variables of repaired cleft in cleft lip only, cleft lip and alveolus and cleft lip, alveolus and palate groups

The CL only group had the greatest increase in the postoperative vertical lip height and greatest decrease in nasal width, while the CLA group had the greatest increase in vertical lip height and the philtral height. The CL, alveolus and palate group had the greatest reduction in the cupid bow width. However, the comparison did not show any significant difference between the three groups (P > 0.005) [Table 3].

Table 3

Postoperative anthropometry variables of repaired cleft side in cleft lip, cleft lip and alveolus and cleft lip and alveolus groups

Variables	CL group		CLA group		CLAP		df	P
	Mean	SD	Mean	SD	Mean	SD
Horizontal lip height	17.60	2.86	19.19	4.11	17.36	3.22	2	0.294
Vertical lip height	16.98	3.17	16.48	4.21	16.77	3.03	2	0.926
Nasal width	17.89	2.84	20.09	4.19	18.79	3.15	2	0.225
Total nasal width subject	35.41	4.85	37.06	6.18	35.87	4.90	2	0.687
Philtral height	12.59	2.14	12.70	2.00	12.54	1.66	2	0.974
Cupid’s bow width	12.02	2.66	12.08	1.95	11.76	2.34	2	0.021

*Significant at P is ≤0.05. SD: Standard deviation, CL: Cleft lip, CLA: Cleft lip and alveolus, CLAP: Cleft lip, alveolus and palate

DISCUSSION

The impact of different types of cleft phenotypes on various treatment outcomes in UCLP patients has been assessed to some degree,[7] but objectivity has been lacking. Preoperative measurements and comparisons of the three types of UCL showed there was indeed varying degrees of tissue distortion and relative hypoplasia in all the 3 cleft types, with CLAP exhibiting greatest distortion and relative hypoplasia. This result echoed that of Chou et al.[14] and in agreement with Suzuki et al.;[3] Bishara[9] and Carroll and Mossey.[15] Carroll and mosey[15] observed that the severity of CL is greatest with CLAP subtype. Literature seems to agree on this observation as we did not find a contrary opinion in literature. The reason for the least distortion and relative hypoplasia in CL only group is most likely to be because the cleft width is relatively small and so the displacement of the tissue is minimal as stated in Delaire and Precious.[1] It is also important to note that the cleft side of the CL only group is closest to its controls, further buttressing the fact that it has least tissue distortion.

Interestingly, the vertical lip heights in all the three groups were similar to those of the control and not to the noncleft side. This might be because the cleft is placed vertically in the lip tissue and so the hypoplasia effect is more pronounced vertically rather than horizontally. However, symmetry of cleft side with the control but not with the noncleft side has equally been reported in other studies.[161718] It is consequently, now being speculated that the noncleft side may not be an appropriate control for the cleft side.[1617]

Postoperatively, the distinction between the various CL types is still conspicuous. Most of the measurements in the CL only group were similar to those of the noncleft side and the control while the other groups were similar with only those of the control. This finding is similar to that of Hoh and Sulaiman[10] which reported different outcomes of surgical repair based on initial differing preoperative severities in UCL, and is completely in agreement with Campbell et al.[11] and Gundlach et al.[19] Abdurrazaq et al.[20] also alluded to the effect of cleft severity based on the different cleft phenotypes, on the outcome of surgical repair, especially that of the nose, even when primary rhinoplasty was done. The impact of surgery on the different types of UCL was however remarkable, as previously reported by Adetayo et al.[16] as the lip lengths, philtral heights were similar with those of the control, even in seemingly most severe CLAP group, which could mean that the surgeons were able to compensate for this differing severities because of their skills and experience.

CONCLUSION

The different phenotypes of UCL have different degree of tissue distortion and relative deficiencies which could be termed “severities” as shown by the preoperative measurements in Table 1, where minimum distortion was seen with CL only subtype. This group also has its measurements similar to those of the nonleft side and control, suggesting it has the least deviation from the normal. However, the comparison between the three groups did not reveal any difference, suggesting, again, that the outcome may not be entirely dependent on the UCL phenotypes. The skill of the surgeon and the selection of a well-proven technique can be considered as the more important factors in the outcome of repair of UCL. However, larger number of patients in each group might give a more reflective difference in outcome.

Limitation of the study

Low number of participants employed and nonuse of a standardized tool to measure the unilateral cleft severity might contribute to why a difference was not seen with all the parameters considered.

Future research

3D anthropometry measurement needs to be conducted to give a more accurate 3D evaluation of surgical outcome.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.