Secondary objectives
The protocole design took account of the recommendations set out by the workshop of January 2006, entitled "Prioritizing a Research Agenda for Orofacial Clefts" [
8] conducted by The National Center on Birth Defects and Developmental Disabilities and the Center for Disease Control and Prevention. The aims of the meeting were to review existing research on orofacial clefts, and to identify gaps in knowledge that need additional public health research. Of the 18 priorities defined in order of importance by this commission comprising 45 experts, those relating to the present project are 5 in number:
Priority n°1: Characterisation of phenotypes so as to define aetiologically homogenous categories of CLP A classification established by way of agreement among surgeons of the two tertiary referral centres is used for each of the medical files entered into the database [
24,
25]. The grouping of the different forms of CLP into more homogenous categories improves not only the chances of identifying risk factors, but also the prognosis for each form.
Priority n°2: Early screening for retarded cognitive development among children with CLP, and determination of the instruments liable to detect it. There is a need to evaluate the timing of surgery so as to ascertain whether early intervention would optimise the child's development. Beyond the immediate research objectives, and in the longer term, the formation of a cohort of patients with CLP should enable follow-up of the outcome of these young patients and their parents, in particular in periods generally considered sensitive, such as entering infant or primary school, or reaching adolescence.
Priority n°3: Improvement of the quality-of-life of children with CLP and their families. According to the commission, it is essential to get to know the factors that can influence quality of life, among which: support from a pluri-disciplinary team, mental balance, type and timing of surgical acts, compliance with treatment protocols, and the experience of individuals involved in the care provision.The validation of international instruments specific to children with CLP could simplify and improve the assessment of quality of life among these children. A distinction needs to be made between the perceptions of this quality of life as seen and reported by parents, and the quality of life of the children themselves, or again as perceived by the healthcare team, so as to obtain a clearer picture of the situation. This study, for the first time in this particular area, implements a specific measure assessing the parent–child relationship and integrating the state of relational withdrawal of the infant (the ADBB scale). Indeed, in the literature the parent–child relationship has always been studied by questioning the parents. All the authors who have broached this subject recognise the difficulties and the limits of these instruments which can only give one aspect of reality. The ADBB scale (developed by Antoine Guedeney, co-investigator in the present project) focuses on the child. The scale has been used in numerous international studies, but never among children with CLP [
16].The PSI questionnaire has already been used for other studies on parents of children with CLP [
5,
26,
27]. The results obtained will enable us to make comparisons with those derived fromother studies. For the present study, the IOFS (Impact On Family Scale) has been translated and validated in French. It enables the evaluation of parental quality of life, and comparison of results with those obtained by Krammer in 2007 whose study concerned families of children aged between 6 and 24 months born with orofacial clefts [
28].
Priority n°4: The effects of the moment at which the diagnosis is received - before or after birth.Few studies have explored this theme. Better understanding of the factors liable to influence the perception of the diagnosis is required (parental stress, family support, the importance of the choice of neonatal care procedures). It is likewise important to know the manner in which parents were informed, and whether or not psychological back-up was offered. One of the secondary hypotheses of the present study is that the parents for whom a prenatal diagnosis was possible are better prepared to tolerate the waiting period until surgery.
Priority n°5: Care and treatment protocols for CLP.It is important to analyse the care strategies implemented for children with CLP, and also the chronology and frequencies so as to gain a better understanding of possible long-term consequences. The medical files stored in each of the participating centres give an account of the history and the chronology of the medical acts performed. Thus the protocols can be compared, in particular between the two Reference Centres in France (Lille and Paris) and the two Competence Centres (Nancy and Strasbourg).
Finally, and above all, if the results of the study point to the need for psychological support for parents of children with CLP, regardless of the timing of surgery, a list of correspondents could be made available by a reference psychiatrist belonging to the healthcare team (a recommendation of the Cleft Palate Cranio-facial Association quoted by Collet and Spetz in 2007) [
29].
Current state of the study
The study started in March 2010 and should be completed in the course of the second semester 2012. More than one hundred families of the 150 required have been included, among whom 18 have already been assessed at T0 and T1. The preliminary results cannot for the moment be reliably interpreted. However, concerning inclusions, it can be noted that certain parents refused to participate in this study. These refusals are mainly attributable to firstly the distance between home and the healthcare centre or the evaluation premises, and secondly to the repeated visits required for the care of the child, and also certainly to the reluctance of parents to confide their feelings and difficulties since the discovery of the malformation. As the study cannot intrude on the private lives of these families, it is impossible to press for agreement, especially in cases where the refusal is from one parent only. As for the evaluations, as mentioned earlier, in the literature the parent–child relationship is always studied by interviewing the parents. The choice and the relevance of the questionnaires used can be questioned. Indeed, self-administered measures are subject to caution, as parental responses can lack objectivity. The contribution of the ADBB scale for children in this age group in this instance enables the study of the parent- child relationship in a symmetrical manner, and also more objectively. The behaviour of an infant and any signs of withdrawal are unlikely to be dissimulated, which may not be the case with the parents.