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 Table of Contents  
STATE OF THE ART
Year : 2020  |  Volume : 7  |  Issue : 2  |  Page : 75-79

Outcome measures in cleft lip and palate: An Indian perspective


1 Plastic Surgery, National Heart Institute, East of Kailash, New Delhi, India
2 Plastic Surgery, National Heart Institute, East of Kailash; Max Smart Superspeciality Hospital, Saket, New Delhi, India

Date of Submission07-Jul-2020
Date of Acceptance13-Jul-2020
Date of Web Publication31-Jul-2020

Correspondence Address:
Dr. Anshumali Misra
National Heart Institute, East of Kailash, New Delhi - 110 017
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_22_20

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  Abstract 


Outcome measurement is the definitive way to prove or disprove the usefulness of a procedure and its impact on a disease process. Cleft lip and palate is a disease with many facets and involvement of many subspecialties in its treatment. It is therefore necessary to have a comprehensive approach towards outcome measures for satisfactory treatment endpoint. Patient opinion even in case of children is very important and should actually form a very important component of overall outcome measures. Authors' experience in the Indian subcontinent shows that many children have been very happy with results that may be found wanting by some. In some cases that proved a boon for the child to grow in the world facing adversities. Authors' have proposed a simplified standard of points for outcome measures for uniformity of results.

Keywords: Cleft lip and palate, cleft lip, cleft palate, Indian context, outcome, patient perspective, result


How to cite this article:
Agrawal K, Misra A. Outcome measures in cleft lip and palate: An Indian perspective. J Cleft Lip Palate Craniofac Anomal 2020;7:75-9

How to cite this URL:
Agrawal K, Misra A. Outcome measures in cleft lip and palate: An Indian perspective. J Cleft Lip Palate Craniofac Anomal [serial online] 2020 [cited 2020 Oct 23];7:75-9. Available from: https://www.jclpca.org/text.asp?2020/7/2/75/291139




  Introduction Top


Since early days of medicine, there has always been a need for evidence-based practices which enable the physician to determine the best treatment strategies and replicate the results again and again. The importance of evidence-based medicine was stressed by Hippocrates who declared that the physician needs to “rely on actual evidence rather than on conclusion resulting solely from reasoning because arguments in the form of idle words are erroneous and can be easily refuted.”[1]

Outcome measures are one of the corner stones of evidence-based medicine. These help ascertain the best possible practices which are consistently reproducible or should be consistently reproducible every time one ventures to do so. With evolution in time, cleft care has become a specialty in its own right. From the days of yore, where it was dominated by the surgeons alone and was limited to just correction of the facial form, cleft care is now a comprehensive effort by surgeons, speech therapists, dentists and maxillofacial surgeons, otolaryngologists, and more recently nutritionists and social care workers to achieve aesthetic and functional correction to the maximum and help the child lead a socially productive life. There is flip side to this advancement as well. Incorporation of all these specialties along, multifactorial nature of the disease and varied morphology of every patient makes the standardization of outcome measures very difficult.

In a broad sense, the goal of treating congenital conditions such as clefts is as follows:

  1. Optimize function
  2. Good esthetic outcome
  3. Health-related quality of life.[2]


Cleft patients have a very long course of treatment from child hood till one is a young adult and sometimes beyond. The development during this time is physical, mental, and psychosocial. All these have different impact on the deformity and patient at various levels. Furthermore, these are dealt by different specialists at different times, each of whom may individually focus on work at hand competently but the question of interplay of various specialties is found to be wanting in most cases. It is, therefore, of paramount importance to assimilate information and ascertain definitive end points which may be taken as satisfactory or good result for the particular problem.

Outcome measurement in the pediatric age group and more specifically cleft patients is very challenging. This is because of a variety of reasons.

  1. Many a times, the problem which one is trying to correct is actually not present at the time of treatment, an example is palate repair affecting the speech. The repair is to be done before the speech is evolved and hence the results are available after a certain period of time. If one waits till speech is evolved the damage has already been done. There has been a raging debate between the proponents of early surgery for speech and delayed surgery to allow better growth of the facial bones and facial features, the definitive answer to which is still an enigma
  2. Esthetic correction of any deformity is a very subjective. Race, ethnicity, and geographical location all have a major impact on the perception of normal
  3. Perception of deformity changes as the child grows in age and has enhanced social interaction. The expectation from the outcome also changes because of this.[2]


Another very important aspect in outcome measures is the patient's view point. Children have been able to self-report from the age of 6 years[3] and it is their perspective that needs to be accounted for. Children have less developed understanding of health concepts but as was shown in the landmark EURO cleft study[4] there was a significant level of dissatisfaction in teenagers about certain features after surgery. Given an option most patients wanted surgery for improvement and so did their parents even if they were grossly happy with the outcome. This study also showed that parents tend to agree with teen-aged children when it came to perception and need for further improvement. This aspect of outcome measurement has also raised a different set of problems. The expectations in some cases appear to be unrealistic and motivated by virtual imagery. Furthermore, the questionnaire used for such evaluation may sometimes be unable to achieve adequate assessment of esthetic outcome as the objectivity of the answers are a misfit to the subjective nature of this assessment.[4]

The International Consortium for Health Outcomes Measurement (ICHOM) convened an international multidisciplinary working group to develop a standard set of outcome measures for comprehensive appraisal of cleft care. The resulting standard set for cleft lip and palate (CLP) in nonsyndromic cases was presented in 2016. [Table 1] summarizes the set of standards recommended and methods of evaluation.[5]
Table 1: Standard set of outcome measures for the comprehensive appraisal of cleft care

Click here to view


These set of recommendations are quite extensive and application at ground level may be difficult in all cases. For a more comprehensive approach, the outcome measures in CLP can be evaluated in the following heads.

Defining the goals of treatment

In a disease as complex as cleft this can be an enormous task. This involves an-interplay between the esthetic and functional goals to achieve a level satisfaction acceptable to the patient and care givers of the patient. The advantages and disadvantages of individual techniques also have a part to play. The use of one particular technique may be superior to others in a particular case. The goals of treatment have to be individualized but also kept within a reproducible and comparable framework for easy and consistent evaluation and comparison. It is often possible that these goals not adequately address the ongoing process, rather terminate with the step at hand. In a disease as complex as cleft, the person heading that particular step may take precedence but a widely consultative approach with all stake holders both on the patients side and in the treating team should be taken to avoid clash of ideas, dissatisfaction, and perception of poor results when that is not the case.

Functional outcomes in cleft lip and palate

Functional outcomes in CLP differ with the type of patient and the problem that is being dealt with. For example, in cleft lip a good functional outcome will be good lip seal and adequate muscle repair without notching. In cleft palate surgery, good functional result will entail no residual defect, no fistula after surgery, well-developed speech without further need of corrective surgery (as done in velopharyngeal incompetence) and minimal effect on facial growth. Similarly, in orthognathic surgery, the goals shall change to stable bony skeleton with near normal anthropometric measurement and dental occlusion as close to normal as possible [Table 1].

The problem of time frames makes it very challenging task to evaluate the functional results without bias as the post intervention changes do not happen simultaneously. Many standardized questionnaires such as the GOSLON yardstick have been used facial growth and occlusion defects.[6] Similar methods in native languages have been adopted for speech assessment. With the assessment being spread out to each subspecialty, it is now becoming more compartmentalized and therefore more consistent.

Esthetic outcomes in cleft lip and palate

The assessment of esthetic outcomes in CLP is a very daunting task. Many variables can affect this assessment. Starting from expectation of the patients and care givers, variability in documentation and diverging perspectives of the patient, clinician, and general public are all responsible for the difficulty in objectively assessing the esthetic outcomes of CLP. Many psychometric tests such as Rasch analysis have been used for esthetic outcome measurement of CLP patients.[7] It is of importance to note that eventual goal of treatment in all these cases is to improve patients own perception of their appearance and wellbeing. Defining esthetic parameters with this core idea is probably the most acceptable way [Table 1]. This method though has an inherent problem of the observer's bias. It is important therefore to use cephalometric-guided scales as a tool for objective assessment of any esthetic result.[8] Perceptive differences are present between cleft team members and the patients and care givers. Professionals were less satisfied with surgical and esthetic treatment outcomes and thought that there was more need for corrective surgery as against the lay people who were far more satisfied with the result and thought that further surgery may not be needed. This approach could avoid many surgeries which would not satisfy the patient.[9]

Other facets of outcome measures in cleft lip and palate

As described in ICHOM study, nutrition of the child ascertained by body weight, otologic health, psychosocial development, and burden of care have also been assessed as parameters for outcome measures. These have to be assessed by patient reported outcome (PRO) measurement data. Many such PRO instruments have been used with CLEFT-Q[10] being the most common. Here, the patient response can be monitored from 6 years to 22 years. This makes the tool applicable in the entire journey of the patient from childhood to adulthood.


  Documentation and Standardization Top


These two words are very important to facilitate outcome measurement. Any assessment is possible only if data are comparable. In case of CLP, this starts from following a standard classification, pictorial depiction of the deformity at each stage of treatment to corroborate with the classification used and use of same tools for speech recording. Ensuring that data collection is uniform is the first step toward the assessment of outcome within one unit which can then be compared to outcome of other units. Standard of care can be defined only when one particular results can be repeatedly and efficiently reciprocated in the hands of multiple persons with low degree of variability.


  Authors' Perspective Top


Authors have very long experience of taking care of children with CLP and have long association with their care giving parents. As surgeon and cleft care givers, we always consider the anatomical, functional, and psychological outcome as the measures of result of cleft surgery. However, we should also consider the patients perspective in assessing the final outcome as they have to lead their life, and not the surgeon or the researcher.

There are many children when they reach the professional institutions, get acclimatized to a set of deformities. Sometimes, they become their identity. Occasionally, they become the reasons for outperforming other children who do not have such abnormalities. The CLEFT-Q score or GOSLON index are not relevant to them. Scientifically, they need more surgery, more care, more support, however, these children are happy and adjusted. In spite of offering all the care and attention, having multiple sessions of friendly counseling, respectfully decline further treatment. These children perform well and have very standard of quality of life.

There are another set of children, especially in developing countries like ours, who are “burn out” cases because of repeated surgery, repeated visits to hospitals and ancillary care givers for dental and speech care. These visits and hospital admissions compromise their studies, extracurricular activities, and normal lifestyle. In spite of being normal otherwise, they are over protected and are cared for more than other children in family.

In developing countries, many of these families are already financially compromised. Repeated hospitalization and visits to health centers for care pose financial burden to the family. The expenses for the hospitalization, travel and loss of wages of the parents pose financial burden apart from nonproductive time to be spent. This factor should also be taken into consideration while assessing the outcome. In attempt to achieve parametric correction, are we posing excessive financial burden to the family.

Hence, we need to go beyond the boundaries of our scientific analysis and measure the outcomes what the children and their parents want. We should respect their desire and decision. Hence, we must involve the children and parents in decision-making after requisite primary surgeries are performed. Moreover, we must give due respect to their decision. Moreover, the outcome should be rated only and only on the basis of the quality of life these children lead at the end of the day. We do not refute the importance of previously discussed outcome parameters. However, these should be the pedestal to reach to the final goal of the patient satisfaction. The authors' recommendations for outcome measures are summarized in the [Table 2].
Table 2: Defining the goals of treatment

Click here to view



  Conclusion Top


Evaluating the outcomes in cleft patients is very complex because the methodology and target population both are very complex but evaluation of outcome is important to implement better and holistic clinical approach. Many differences in perception exist between professionals who are a part of the cleft team and laypeople. This discrepancy makes the health-care professionals responsible toward offering best possible care to the patient. This is possible only with standard care practices which can be discerned by evaluation of outcomes to decide the best procedures in a present scenario.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Mountokalakis TD. Hippocrates and the essence of evidence based medicine. Hosp Chron 2006;1:7-8.  Back to cited text no. 1
    
2.
Wong WY, Forrest CR, Goodacre TE, Klassen AF. Measuring outcomes in craniofacial and pediatric plastic surgery. Clin Plastic Surg 2013;40:305-12.  Back to cited text no. 2
    
3.
Riley AW. Evidence that school-age children can self-report on their health. Ambul Pediatr 2004;4:371-6.  Back to cited text no. 3
    
4.
Semb G, Brattström V, Mølsted K, Prahl-Andersen B, Zuurbier P, Rumsey N, et al. The Eurocleft study: Intercenter study of treatment outcome in patients with complete CLP. Part 4: Relationship among treatment outcome, patient/parent satisfaction, and the burden of care. Cleft Palate Craniofac J 2005;42:83-92.  Back to cited text no. 4
    
5.
Allori AC, Kelley T, Meara JG, Albert A, Bonanthaya K, Chapman K, et al. A standard set of outcome measures for the comprehensive appraisal of cleft care. Cleft Palate Craniofac J 2017;54:540-54.  Back to cited text no. 5
    
6.
Sinko K, Caacbay E, Jagsch R, Turhani D, Baumann A, Mars M. The GOSLON yardstick in patients with unilateral cleft lip and palate: Review of a Vienna sample. Cleft Palate Craniofac J 2008;45:87-92.  Back to cited text no. 6
    
7.
Boone WJ. Rasch analysis for instrument development: Why, when, and how? CBE Life Sci Educ Wint 2016;15:rm4.1-7.  Back to cited text no. 7
    
8.
Bearn DR, Sandy JR, Shaw WC. Cephalometric soft tissue profile in unilateral cleft lip and palate patients. Europen J Orthodont 2002b; 24:277-84.  Back to cited text no. 8
    
9.
Alhayek S, Alsalem M, Alotaibi Y, Omair A. Evaluation of facial appearance in patients with repaired cleft lip and palate: Comparing the assessment of laypeople and healthcare professionals. Maxillofac Plast Reconstr Surg 2019;41:5.  Back to cited text no. 9
    
10.
Wong Riff KW, Tsangaris E, Forrest CR, Goodacre T, Longmire NM, Allen G, et al. CLEFT-Q: Detecting differences in outcomes among 2434 patients with varying cleft types. Plast Reconstr Surg 2019;144:78e-88e.  Back to cited text no. 10
    



 
 
    Tables

  [Table 1], [Table 2]



 

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