The main hypothesis of this research is that the longer the time lapse before the first surgical intervention, the more likely are parental perceptions and feelings to upset the parent–child relationship and affect the harmonious development of the child.
There are also two secondary hypotheses:
1) that the parents for whom it has been possible to give a prenatal diagnosis are better prepared to accept the waiting time
2) that with time, the negative feelings of parents in the later surgery group (3 to 6 months after birth) tend to decrease and to come into line with those of parents whose children have had an early intervention, and also that the child's distress tends to decrease.
Main evaluation criteria
The main criteria used in this research to assess the psychological state of the infants and parental stress, as well as any possible parental psychopathology, are provided by the following measures:
1) for the children, the Alarme Détresse BéBé (ADBB) scale which measures relational withdrawal in infants. The measure is applied by a specially trained mental health professional [16
2) the Parenting Stress Index (PSI) which enables screening for parental attitudes that could be risk factors for the development of emotional and developmental disturbances in a young child [17
Secondary evaluation criteria
The secondary evaluation criteria are provided by the other questionnaires selected for this research, which are:
1) the Indice de Détresse Psychologique - Enquête Santé Québec health survey (IDPESQ) [18
] and the Edinburgh Post-partum Depression Scale (EPDS) [19
] which measure the psychological state of each parent
2) the Impact On Family Scale (IOFS) [21
] which assesses the family, social and financial impact of the malformation
3) Spanier's Dyadic Adjustment scale in the short version, which explores the parental couple [22
4) specifically developed questionnaires collecting information on the perception of the malformation and the care team, and on the parents' present relationship with their child. These questionnaires were designed to adapt to the moment of diagnosis (antenatal or at birth).
According to the type of CLP and the healthcare facility involved, the time lapse between diagnosis and the first surgery varies from birth to six months. Two evaluation times are planned:
· T0, when the infant is 4 months
· T1, when the child is 12 months, i.e. 6 months or more after the first surgical intervention
The choice of positioning T0 at 4 months is based on the following arguments:
· Because of the variability of the protocols used by the surgical teams in each of the centres, it is not possible to define T0 and T1 that correspond to a specific examination for all the centres;
· At this age, the children for whom the surgery occurs early have already had their operation. They can therefore be compared to children whose operation is to occur subsequently (up to 6 months), the two groups comprising roughly equivalent numbers. In a more detailed analysis, it will be possible to compare the children receiving surgery at birth with those receiving surgery at three months or more.
The choice of positioning T1 at 12 months is justified by the following argument:
· When the child is one year old, this is sufficiently distant from both birth and the first surgical intervention (by at least six months) to enable a repeat of examinations and questionnaires used in the first stage of the study. This will enable comparison of psychological perceptions of the parents and the relational development of the child between T0 and T1, as well as exploration of the question of the timing of surgery.
Only children with CLP are included, either isolated or familial, syndromic or nonsyndromic. Parents are included following informed consent for themselves and their child.
Two subgroups of children are also formed with respect to the time of diagnosis:
Sub-group 1: parents having received an antenatal diagnosis
Sub-group 2: parents having discovered the diagnosis at childbirth.
For both subgroups it is in the course of the post-natal consultation that the surgical team approaches parents to enter the study, after providing all required information, and without interfering in any way with the treatment protocol.
Non-inclusion criteria are:
children with CP alone
children born before 36 weeks amenorrhoea
children whose birth weight was under 2.5 kg
children placed in foster homes
parents under legal guardianship
parents insufficiently conversant with French and/or illiterate
Criteria for removal from the study
refusal to participate by one of the child's parents in the course of the study follow- up
the occurrence of a complication in the course of treatment and/or a serious illness requiring major specific treatment
an unexpected complication in connection with the surgical intervention
serious illness or death of one of the parents
the parents moving house outside the regions involved in the research
Four regional centres in France are taking part in this research:
· The Strasbourg CHU Competence Centre, Pr. C. Bruant-Rodier and Dr. I. Kaufmann
· The Centre Référent des Malformations Crânio-maxillo-faciales rares, Lille CHU (Reference Centre), Pr. Pellerin
· The Centre Référent des malformations rares de la face et de la cavité buccale, APHP Paris (Reference Centre) Pr. Vazquez
· The Nancy CHU Competence Centre, Pr. Simon
These facilities were selected according to their varying lengths of waiting period between birth and the first surgical intervention (Lille: "early intervention", immediately after birth; Strasbourg: "early intervention deferred 3 months"; Hôpital Armand Trousseau (Paris) and Nancy: "Later intervention, towards 6 months". The facilities in Paris and Lille are the only Reference Centres in France, and they are distinct in particular on account of the timing of the first surgical intervention.. Their teams have international renown in the field. By way of the inclusion of a psychologist on the staff of both centres, they have long been organised to take account of the psychological impact of these malformations on parents and children. The choice of the centres in Nancy and Strasbourg enables the inclusion of two Competence Centres. These facilities have a similar organisation with a smaller staff, and do not have a psychologist permanently available.
The documents collected directly in the observation file for each child concern the following:
· Tests and examinations performed at T0 and T1
· Brief analysis of the individual situation of the persons present performed by the psychologist present at times T0 and T1
· Information sheet drawn up by the surgeon giving the precise type of CLP, the time when diagnosis occurred, family history, the pregnancy, care provision to the child, and also the surgeon's impression of the relationship established with the parents
· Any undesirable event