Patient-centred oral cleft research

The perspective on the treatment of oral clefts (OCs) and related congenital craniofacial syndromes is shifting and a better understanding of what matters to our patients and a continuing dialogue are necessary throughout the long treatment trajectory from birth until adulthood. It is most important that patients and their parents understand the information provided, so that they can take part in the decision making.¹

However, research on patients with OCs is often focused on treatment and in patients with a complete unilateral lip, alveolar, palate cleft, this means that the patient undergoes lip, soft palate, hard palate and alveolar surgical closure mixed with ENT therapy, speech therapy, orthodontics and possibly psychosocial counselling over the years, comparing many different protocols.² When a new surgical technique is used within the same treatment protocol, one can only evaluate this well at the end of growth, after the treatment has been completed, so in (early) adulthood. This means that when researchers report on long or short term treatment methodological rigorousness is necessary.³ And whether research is on treatment outcome from a clinician or a patient perspective both should follow a strict methodological set up and report. The equator-network that aims to enhance the quality and transparency of health research should be the first stop for any researcher.⁴

Also in research on OCs the presentation of a fixed set of background data including ethnicity, gender, the same and correct phenotype, year of birth, age at outcome and consecutiveness of cases is needed. Other influencing factors, such as number of surgeons, surgical technique, number, timing, revisions and complications of surgeries, number of orthodontists, reporting time span and whether a standardized protocol was used, should be included in the report.⁵

Although the reporting on correct (sub) phenotypes of OCs is clearly needed,⁶ research is still often hampered by incomplete or unclear reporting.⁷ Clinicians should be able to use an easy registration form⁸ that records a precise anatomical description at birth, that could then be converted to different classification systems, such as the LAHSHAL⁹ and the Dutch Association for Cleft Palate and Craniofacial Anomalies (NVSCA) registration¹⁰ but also other commonly used systems such as the ICD.

Epidemiological research has been reported on in the past but it remains valuable. When researchers report on their consecutive cases over a certain time span readers can get an understanding whether the patient group described fits within the local prevalence or whether it is deviating.

Bias or potential confounders may then be diminished by a well composed study. This means that although Randomized Controlled Trials (RCTs) are the best evidence we can strife for there is still an important role for well reported case series, case control studies, cohort and cross sectional studies since not every part of OC treatment can be analysed through an RCT. A well designed multi-centre study could then provide even stronger evidence especially when using the same treatment protocol.¹¹

When reporting outcomes, current three-dimensional (3D) imaging and processing can lead to new insights in aetiology, diagnosis, treatment and outcomes of craniofacial malformations. The specialization of labs in 3D imaging has led to more precise and better evaluation with these 3D tools.¹² This has facilitated the recognition of facial areas involved in distinct syndromes, the follow-up over time of faces or dentition, the planning of surgical procedures and the improved outcomes of cranial surgery in infants.^{3, 13, 14} However, to compare or translate these new outcome tools to 2D tools in older reports, researchers should also provide raw data in older 2D outcome measures besides the 3D outcome measures, for instance in data repositories or appendices. However, the amount of data to compare becomes more complex which may make it more challenging to summarize and draw conclusions. Artificial intelligence (AI) might help to analyse these ‘big data’ by recognizing patterns potentially easier, but it remains crucial to solely include complete and methodologically sound data to prevent the so called ‘dreaming’ of the AI machine.¹⁵

Craniofacial research should aim to combine doctor driven data with patient centred data and the International Consortium for Health Outcomes Measurement (ICHOM) supports that aim by providing structured tools specific to the malformation or disease.¹⁶ Clinicians need an objective tool tailored to patients with a craniofacial condition to listen, communicate and provide a dialogue with them being OC specific Patient Reported Outcome (PRO) and Experience (PRE) measures.¹⁷ Hopefully these measures not only improve future outcomes for high income countries but also for low or middle income countries or countries where OC treatment or research is less well funded or less accessible. At the end of treatment, at nearly 22 years of age, these measures should result in added value for the patient or to Value-based care and to a better Oral Health related Quality of Life.

This special issue is hopefully a good example of this vision presenting meaningful data for craniofacial patients.

All authors contributed equally to this editorial.

None of the authors have a conflict of interest.

This manuscript was written in the light of the Helsinki declaration. However, no patient or animal data were used.