This study reveals both the value of QoL assessment in EP Children and the limited extent to which it is used, and explains the reasons. It highlights the physicians’ perspective by revealing different strategies and different concerns among those questioned. It completes the findings of Baars on the perspective of general paediatricians [11].
The limited participation in the survey may be explained by the length of the questionnaire and by the two rounds of questions. However, other reasons may be suggested. Quality of life could appear to come into conflict with personal convictions in the highly sensitive field of management of EP Children. Some physicians justified their declining to take part by the complexity of the subject and their lack of knowledge of it. Physicians’ lack of interest compared with nursing staff or social workers has already been observed [7]. The main limit of this observational study is based on the high number of studied variables, which does not allow to justify the number of subjects to be studied. However, the participation rate is comparable to that reported in similar surveys [11, 14].
Interest in the subject, knowledge and practice varied according to age and specialty. Younger physicians gave more importance to QoL and appeared to put it into practice, as has already been described in a study comparing the perspectives of students and older physicians [15]. Similarly, the longer a child is followed by a professional, the better the latter can judge how the impairment is experienced and the impact it has. However, measurement of QoL is still little used and was reported by only two paediatric neurologists, and was certainly related to their experience of long-term follow-up. While the majority of physicians appeared interested in the topic, many had an indirect approach to QoL, as in the survey of Baars, where 61% of paediatricians assessed QoL informally [11]. The explanation given was lack of knowledge of the instruments available and the feeling that intuition is more appropriate than standardized measures, as has been reported in other disciplines [7, 9].
Even if they do not use a standardized approach, the physicians believe that the domains to be evaluated are those that are classically described in chronic diseases [11]. They consider the consequences of the symptoms, notably neurodevelopmental problems and the impact on those close to the child [1, 16–19]. The question of the autonomy appears to be an essential criterion, as described by Tanaka, whatever the age of the physicians questioned [15]. As in other disorders where the patient’s perspective is the most important, the physicians take into account the child’s perception and also that of the parents [1, 20]. The factors that finally govern the QoL of extremely preterm children appear to be close to those classically described in paediatrics [11, 21].
Physicians thus do not measure QoL but give responses that are socially “expected” of them: it is impossible for them to say that they are not interested in the patient’s QoL, the best viewpoint is that of the patient… that is, they give responses that are in contradiction with their practical experience. It is unlikely that this is related to total disinterest in the subject, but rather indicates following a conventional line of thought. The giving of expected responses is a mechanism identified in opinion surveys, and described as “selective reporting” [22].
Lastly, and this completes the study of Baars et al. [11], physicians describe an “ideal questionnaire” that would be likely to produce a QoL assessment that is relevant to their daily practice. This could direct the development of new tools, adapted to the population concerned. Obtaining the opinion of physicians on the best means of evaluating their patients’ QoL, and finding the most appropriate format of questionnaire, could encourage the practice of assessment [7, 11].
The main reasons for not assessing QoL in routine practice, reported in the results, concur with those described in experts in other disciplines: principally a doubt as to the possibility of measuring subjective data, the impossibility of relating statistical data to an individual situation, the importance of not assimilating QoL and state of health, and more particularly QoL and impairment, the feeling of the lack of a reliable instrument, and lack of time [7–9]. Beyond these reasons, the physicians questioned added the fear that the population evaluated would be stigmatized.
Three principal expectations with regard to standardized QoL assessment were expressed.
Firstly, assessment of patients’ QoL gives better understanding of their needs, and so of the measures that need to be implemented to respond to them, as has already been documented by Greenhalgh [7]. Data on QoL would shed light on public decisions through better appreciation of the social and economic impact of extremely preterm birth. Moreover, the idea of “burden” appears in the literature [16, 18, 23] in relation to the heavy demands of medical and rehabilitation management, the inadequacy of support and assistance, the occupational impact and the impact on siblings. This raises questions on the intensive measures that are implemented in the perinatal period and are then reduced, both in human terms (limited number of places in medical and social facilities, recent and limited development of follow-up networks to support and assist the children and their families, difficulties in the provision of schooling) and in economic terms (non-reimbursement of some rehabilitation treatments, low benefits).
Secondly, QoL data may be used as a management instrument in the relationship with the patient. They can improve the care relationship and information on outcome, as already described by Stahlmann in relation to prematurity [17] or by Greenhalgh from a more general standpoint [7]. This information transmitted in the perinatal period is marked by incertitude as to the prognosis. The parents need more concrete information than the conventional data on morbidity, described as a percentage of sequelae or of mortality. The parents may wish to know more about the children’s feelings and experience, as has been described in other settings [19, 24]. While information on the QoL of extremely preterm children appears to be useful to the parents, it is also useful to the physicians. Just as in chronic diseases where the patients’ daily life is affected, in extreme prematurity it is important to improve mutual knowledge between providers and recipients of care [21, 25]. This is what seems to be sought by the physicians questioned in our study, and was already suggested by the paediatricians in the survey of Baars [11] and by Barlesi in a survey among physicians in a thoracic oncology network [9].
While the above two types of expectation are classically described in the literature, the third expectation that became apparent in the results is more original. Given the specific nature of the problem raised by extreme prematurity, in particular the decisional dilemma of the limits of viability [23], the experts could wish for a tool to rationalize individual decision-making, notably to reflect on the “burden-benefit” balance of neonatal management. A very large majority of the physicians questioned considered that such data could rekindle the ethical debate on neonatal resuscitation practices. The potential decisional impact of such information was mentioned by the majority of physicians questioned, notably the possibility of therapeutic abstention, which appears innovative in France in research on the subject. In a survey among 318 neonatologists in Pacific Rim countries, Martinez studied the factors that influenced neonatal resuscitation decisions at the limits of viability. Perception of poor quality of life, at the same level as the presence of severe congenital deformities, the parents’ wish not to resuscitate their child, or a high probability of neonatal mortality, was identified as a decisional factor by over 70% of the neonatologists questioned. It was not stated, however, whether consideration of QoL had an objective or subjective basis [26]. But in our survey this opinion was not shared by those who had expressed reservations as to the standardized use of QoL. While QoL is used as a theoretical notion to argue decisions on limitation or cessation of treatments, by projecting the patient’s future, it is never based on objective results, as has already been described [12]. One of the principal obstacles to the use of QoL as a decision-making tool is that it may be seen as a judgement, given the perinatal context, on lives that are or are not worth living, and so become assimilated to a tool of sacrifice. However, although these results are debatable (interpretation of the results, differences in methodology), nothing in the literature on extreme prematurity truly describes the QoL of lives that would not be worth living [1, 27].
Quality of life assessment should be understood as an engagement to act. In the field of extreme prematurity, where “the child’s best interest” is one of the foundations of management [27, 28], the finality of action cannot be limited to a short-term result, reflected in a mortality rate or a survival rate, without being tempered by more qualititative elements.
Quality of life may be approached from an ethical viewpoint if concern with QoL means caring for the patient attentively and benevolently, improving their well-being by responding to their needs, and developing means to alleviate dependence. In the “child’s best interest”, physicians must question themselves as to the appropriateness of their acts and the outcome of their patients [29]. Addressing the QoL of extremely preterm children thus appears as a question not only of deontology but also of professional responsibility, with the need to integrate QoL data into care, clinical and economic strategies, an issue which has already been raised in other disciplines [30, 31].
