The delivery of surgical cleft care in the United Kingdom

A.C. WILLIAMS*, D. BEARN#, J.D. CLARK†,W.C. SHAW# and J.R. SANDY*
*Division of Child Dental Health, University of Bristol Dental School, Bristol, #Department of Orthodontics, University Dental Hospital Manchester, Manchester and †Department of Dental Health, Dental Hospital and School, Dundee, UK

Background and purpose: A national survey of cleft teams was undertaken as part of the Clinical Standards Advisory Group investigation of the current status of cleft care in the United Kingdom (UK). Methods: Fifty-seven cleft teams were identified, of which 90% responded to the survey. Main findings: Nine cleft teams had been established since 1992. Only one region, Northern Ireland, had a centralised cleft service and, despite 82% of teams having databases, only four were able to produce corroborated evidence of receiving at least 30 annual new referrals during 1995. There was a wide variation in the facilities provided by individual cleft teams - only six teams were able to provide all of the key facilities recommended by the Royal College of Surgeons Steering Group on cleft lip and palate. Facilities such as antenatal and neonatal counselling, protocols for record keeping and long-term treatment were similar for high and low volume teams. High volume teams were more likely to have established links with a full range of specialties including psychology, clinical genetics and paediatrics than low volume teams. Conclusion: A national survey of cleft services has demonstrated a need for reorganisation. This is now in process and once established will require continual monitoring and assessment.

In 1996, the Royal College of Surgeons (RCS) Steering Group on cleft lip and palate published Recommendations for Minimum Standards of Management of Cleft Care.¹ The Recommendations, were based on agreed “good practice” both in Europe and the United States, where it is believed that cleft care should be provided by multidisciplinary teams based in regional centres.² A child born with a cleft and their families should have access to a wide range of clinical specialties including paediatrics, clinical psychology and clinical genetics.

The Steering Group recommended that 30 new cases per year should be a minimum involvement for operators undertaking primary cleft surgery. Cleft surgery should be limited to surgeons with extended cleft lip and palate training. A survey of cleft surgeons operating during 1991-1992 found that cleft care in England and Wales was based on low volume operators treating an average of less than 10 new cases per year.³ Others have argued that cleft care should be based on smaller teams providing a more accessible service for patients. ^4,5,6

The Steering Group emphasised the need for cleft teams to participate in multicentre audit and research. Well-tried treatment protocols should be followed and new procedures only introduced as part of an ethically approved trial. A survey of consultant orthodontists in England and Wales undertaken by the RCS Steering Group showed less than half the cleft teams operating in 1991/2 had adopted a standardised record keeping protocol for their patients.⁷

In 1995, The Clinical Standards Advisory Group (CSAG) commissioned a national study of the current status of cleft care in the UK.⁸ As part of this study, a detailed questionnaire survey was undertaken of teams involved in cleft care during 1995. The aim of this survey was to gather information on the organisation of cleft care in the UK and compare current practice with the recommendations made by the RCS Steering Group.

A detailed questionnaire was constructed to gather information on the structure and organisation of cleft teams in the UK. Information was also requested on referral patterns to cleft teams and the organisation of cleft clinics. The use of databases, the collection of standardised records and participation in audit was evaluated.

A network of regional co-ordinators was established and comprised representatives from consultant orthodontists, plastic surgeons and oral and maxillofacial surgeons involved in cleft care in each Region. The regional co-ordinators were asked to identify cleft teams in their region to the Research team. A list of cleft teams in England and Wales was available which had been compiled from a previous survey.3 This was updated with the information gained from the regional co-ordinators and extended to include cleft teams in Scotland and Northern Ireland. The leader of each cleft team was sent a questionnaire by post. If there was no response after a period of 6 weeks a further questionnaire was sent to the cleft team leader with a follow up telephone contact if necessary.

Each cleft team leader was asked to provide a list of the names of children born with a complete unilateral cleft lip and palate (UCLP) during the period 1.4.89-31.3.91 and currently under the care of their team. The lists of names were corroborated by an independent search of the operating books and from clinic lists provided by orthodontists and speech and language therapists. These children made up the younger age cohort for the clinical outcome study undertaken as part of the CSAG study.⁸

A total of 59 cleft teams were identified and sent copies of the Cleft Team Questionnaire. Two teams were no longer involved in cleft care and, therefore, 57 teams were operating in 1995. Nine cleft teams had been established since 1991, two of these were led by plastic surgeons and the remaining seven, by oral and maxillofacial surgeons. There were seven cleft teams in Scotland, one in Northern Ireland, two in Wales and forty-seven in England. Only one Region, Northern Ireland, had a centralised service for cleft patients. In contrast the Northern and Yorkshire Region had a total of 10 separate teams caring for cleft patients.

Fifty-one cleft teams (90%) eventually returned completed copies of the questionnaire, although three failed to do so in time for inclusion within the CSAG report.9 Each team was contracted to provide services for an average of 4.2 purchasers (range 1-16). The number of maternity units referring patients to each team varied from 1-15 (n = 5). Cleft teams reported receiving an average of 23.5 new referrals during 1995 (range 3-65). When teams were requested to provide names of new referrals for corroboration, only half (n=19) of the teams which returned the questionnaire were able to provide this information (Table 1).

Table 1: The number of new referrals received by UK cleft teams during 1995 (self-reported data)

Number of referrals	Cleft teams	Cleft teams which provided lists of names during 1995 of new referrals
Age (yr)	Number (%)	Number
1-9	8 (16)	4
10-19	13 (25)	9
20-29	4 (8)	2
30-39	6 (12)	1
40-49	5 (10)	3
>49	2 (4)	0
No information	13 (25)	32
Total	51 (100)	51

Seventy-five surgeons from four surgical specialities -plastic, oral and maxillofacial, paediatric and ENT - were operating on clefts during 1995. The number of surgeons associated with each cleft team ranged from 1-4. The mean number of new referrals per surgeon during 1995 was 18 (range 3-65).

Forty-one teams provided data on the number of lip and palate repairs undertaken by individual surgeons during 1995. The mean number of lip repairs undertaken by the 48 surgeons in these teams was 9.1 (range 3-36). The mean number of palate repairs undertaken by 50 surgeons associated with the same teams was 13.2 (range 1-60).

Fifty-one cleft teams provided information on the number of surgeons undertaking secondary procedures for cleft patients. Seventy-five surgeons undertook alveolar bone grafts during 1995 and 66 surgeons were involved in orthognathic surgery for cleft patients during this time. One hundred and thirteen orthodontists were involved in cleft care during 1995, the number of orthodontists associated with each cleft team varied from 1 to 7.

Access to peripatetic services such as genetic counselling, psychology and paediatric dentistry varied. The majority of teams (59%) had access to a paediatrician and a geneticist (78%) but only 19 teams (37%) were linked to a named psychologist. Less than a quarter of cleft teams had links with all three specialities (Table 2). Advice from a named consultant in restorative dentistry was available to 35 cleft teams (69%) but only 22 teams (43%) had a direct link with a named consultant in paediatric dentistry. The patients under the care of 13 cleft teams (26%) had no access to either a consultant in restorative dentistry or a consultant in paediatric dentistry.

Table 2: The number of cleft teams in the UK with established formal links for peripatetic specialties including paediatricians, clinical geneticists and/or clinical psychologists

Specialities with a formal link to the cleft team	Cleft teams which returned the questionnaire n=51 freq (%)	High volume (>/=10 UCLP cases) cleft teams n=12 freq (%)	Low volume (<10 UCLP cases) cleft teams n=34 freq (%)
Paediatrician	30 (59)	10 (83)	13 (38)
Clinical psychologist	19 (37)	5 (42)	13 (38)
Clinical geneticist	40 (78)	9 (75)	27 (79)
Paediatrician + psychologist + geneticist	12 (24)	5 (42)	6 (18)

Table 3 shows that 46 (92%) of the 50 cleft teams which responded to this section of the questionnaire had a named member of the team available to give feeding advice to the parents of a baby born with a cleft. Thirty teams (60%) were able to ensure that the parents were visited within 24 hours of the birth. One team leader was only able to confirm that the parents would be seen within the first three days. Three cleft team leaders could provide no information about when a new baby would be first seen by their team. Antenatal diagnosis of a cleft is becoming more common and the majority of cleft teams (82%) had arrangements in place for counseling of parents once such a diagnosis is made.

Table 3: The provision of antenatal counseling, specialist feeding advice and/or parental counseling within 24 hours by UK cleft teams

Facilities provided	Cleft teams which returned the questionnaire n=50 freq (%)	High volume (>/=10 UCLP cases) cleft teams n=12 freq (%)	Low volume (<10 UCLP cases) cleft teams n=34 freq (%)
Antenatal counselling	30 (59)	11 (92)	25 (74)
Specialist feeding advice	46 (92)	10 (83)	30 (88)
Counselling for parents within 24hrs	30 (60)	6 (50)	19 (56)
Antenatal counselling + specialist feeding advice within 24hrs	22 (44)	4 (33)	14 (41)

Thirty-seven teams (73%) had agreed longitudinal treatment protocols for cleft patients - 16 teams (31%) were able to provide a copy of this protocol (Table 4). Forty-two cleft teams (82%) held databases of cleft patients and 40 (78%) were sending information on cleft births to either of the two national databases, CARE and SCALP. Sixteen teams were also participating in regional databases. Twenty-five cleft teams were involved in other inter-centre cleft audit initiatives.

Six key facilities which cleft teams should offer their patients were considered; (1) arrangements for antenatal counselling; (2) specialist feeding advice available within 24 hours; (3) access to a psychologist; (4) clinical genetics available on request; (5) the existence of a database and (6) written protocols in place for long-term care and record keeping. Only 6 of the 51 teams who returned the questionnaire were able to confirm that all of these facilities were available.

Table 4: The current use of data bases, long term treatment protocols and protocols for record keeping by cleft teams in the UK

Facilities provided	Cleft teams which returned the questionnaire n=50 freq (%)	High volume (>/=10 UCLP cases) cleft teams n=12 freq (%)	Low volume (<10 UCLP cases) cleft teams n=34 freq (%)
Data base	42 (82)	11 (92)	25 (74)
Protocol for long-term care - exists - provided	37 (73) 16 (31)	8 (67) 4 (33)	23 (68) 11 (32)
Protocol for record keeping - exists - provided	39 (76) 29 (57)	10 (83) 7 (58)	24 (71) 18 (53)
Data base + protocol for long-term care + protocol for record keeping	11 (22)	3 (25)	7 (21)

The absence of corroborated figures for the number of new referrals received by cleft teams during 1995 makes comparison of the facilities provided by ‘high’ versus ‘low’ volume teams, based on current referral rates, impossible. Independent data, however, was available from the CSAG study for the number of 5-year-olds currently under treatment, with 46 of the 51 teams having completed the questionnaire (Table 5).⁸ This was used as the basis for comparing facilities offered by cleft teams. Five teams had started operating after 1991 and no estimate of volume, therefore, could be made. These teams were then excluded from this part of the study.

Number of Equivalent Number % UCLP cases annual number of cleft identified of cleft referrals teams

Table 5: The number of children born with complete unilateral cleft lip and palate during the period 1.4.89-31.3.91 identified to the CSAG research team by cleft teams who completed the questionnaire

Number of UCLP cases identified	Equivalent annual number of cleft referrals	Number of cleft teams	%
0	0	7*	16
1-4	3-12	16	32
5-9	15-27	16	30
10-14	30-42	8	16
>14	>42	4	6
Total		51	100

For the comparison, a ‘high’volume cleft team was defined as receiving the equivalent of 30 or more new referrals annually. Approximately 2 000 children were born with clefts in the UK during the period 1.4.89-31.3.91 and, based on these figures, the incidence of complete UCLP would be approximately 16%.^9,10 Thus, a cleft team which receives 30 or more new referrals of all types of cleft per year would expect to receive approximately five cases of complete UCLP annually. Therefore, over the 2-year period of this study ‘high’ volume cleft teams should have produced 10 or more cases of UCLP.

Twelve cleft teams who returned the questionnaire were classified as ‘high’ volume for the purposes of this comparison and 34 were classified as ‘low’ volume. No differences were found in the proportion of high versus low volume cleft teams that have arrangements in place for neo-natal and antenatal counselling to parents (Table 3). The use of databases and protocols for long-term care and record keeping was also similar for both categories of cleft teams (Table 4). However, when formal links with peripatetic specialities were compared, children under the care of high volume teams were more likely to have access to the full range of clinical specialties (Table 2).

This national survey of cleft teams in the UK has shown that, despite the existence of recommendations for minimum standards of cleft care, only a small percentage of cleft teams are able to offer their patients a full range of basic facilities.

Cleft care in the UK continues to be based on low volume operators. Recommendations have been made for centralisation of cleft care in the UK but we noted an increase in the number of cleft teams operating in England and Wales since the last published survey.^1,3,11 Despite the finding that 82% of teams now have databases, few cleft teams were able to provide corroborated evidence of the number of new referrals received during 1995. There are no reliable figures for the total number of cleft births in the UK, but the OPCS recorded 630 cleft births in England and Wales during 1994, an incidence of 0.95 per 1000 live births. Given that there were 731,645 live births in England and Wales during 1995, approximately 700 babies should have been born with clefts. This would give an average of 12 new cases during 1995 for each of the 57 teams in the UK, substantially below the recommendation made by the RCS Steering Group. Surgical experience is further diluted; some cleft teams have up to four surgeons undertaking primary repairs. Secondary surgery for cleft patients is undertaken by an even greater number of surgeons. It is not known how many alveolar bone grafts are performed each year in the UK but the figure is unlikely to exceed 500 since less than half of reported cleft cases involve the alveolus. Seventy-five surgeons were reported to be undertaking alveolar bone grafts during 1995. This gives an average of less than seven bone grafts per surgeon per year, again much lower than the minimum of 20 cases originally recommended by the RCS Steering Group. This may explain the finding in the CSAG study that almost half of alveolar bone grafts were unsuccessful.⁸ A further cause for concern is the finding that 66 surgeons were involved in orthognathic surgery for cleft patients during 1995. There have been no multicentre audits, to date, on the outcome of orthognathic surgery for cleft patients and the number of surgeons involved precludes any individual surgeon from generating sufficient cases for meaningful comparison of results.

There also appears to have been little progress towards centralisation within the orthodontic speciality. An average of two consultant orthodontists are linked to each cleft team in the UK and, even if it is assumed that every cleft patient will require orthodontic treatment, each orthodontist will treat an average of less than 10 new cleft cases per year. The involvement of the orthodontist in the timing of alveolar bone grafting is important in the success of the procedure.¹² The poor results of alveolar grafting seen in the UK may reflect the lack of experience of some orthodontists in the preparation of cases for bone grafting.

Preventive dental care is a priority for cleft patients and it has been recommended that these children should have priority access to a consultant paediatric dentist. There are only one or two consultant paedodontists in post outside dental teaching hospitals in the UK so it is perhaps not surprising that less than half the cleft teams have established links with these specialists. A similar problem exists with the consultants in restorative dentistry but more cleft teams had managed to gain access to this service. The CSAG study found that a significant number of children were in need of dental care despite being registered with a general dental practitioner. It is unlikely that the number of consultants in paediatric dentistry will be expanded in the near future and cleft teams need to explore novel ways to establish links with this speciality.

The paediatrician is well-placed to assess the child’s progress as a whole and it is recommended that he/she should be involved with cleft care throughout the child’s life.¹³ It was encouraging to find that many cleft teams had established formal links with paediatricians. Clefting may be associated with a range of congenital syndromes, which need to be identified at an early stage. Most cleft teams in the UK have established a formal link with a clinical geneticist.

A child born with a cleft may be teased at school and have a poor self-image.^{14 ,15} Some parents find it difficult to accept their child and may still be distressed many years after the birth.¹⁶ The RCS Steering Group has recommended that all cleft patients and their parents should have access to psychological counselling but only one third of cleft teams were able to offer this to their patients. This is not a situation peculiar to the UK. A comprehensive national survey of cleft and craniofacial teams in the United States of America and Canada highlighted concerns on recording and testing for psychological problems in these patients. There were also issues on record collection and peripatetic services, which are similar to those reported here.¹⁷

Improvements in ultrasound scanning have resulted in antenatal diagnosis of clefts becoming more common. There are reports that termination of pregnancy may follow the detection of a cleft on a scan.¹⁸ Skilled specialist counselling is required following diagnosis. The majority of cleft teams have considered this issue and have the necessary arrangements in place. However, many children are born with an unpredicted cleft - a distressing experience for both parents and staff at the maternity unit. The need for immediate support and specialist feeding advice from a member of the cleft team has been recognised for some time. It is disappointing that one third of cleft teams were unable to provide such advice within 24 hours of the birth, as recommended.¹⁹ The finding that the leaders of three cleft teams had no idea when a member of the team would first be called to see a baby with a cleft was disturbing.

The postal survey of cleft teams undertaken by the RCS Steering Group in England and Wales in 1992 showed that ‘high volume’ cleft teams were more likely to collect standardised records and have active data bases than ‘low volume’ cleft teams.²⁰ This latter survey was based on self-reported data. In the CSAG survey we used the number of 5-year-olds identified independently from a number of sources to give an indication of the number of children being cared for by current cleft teams. On this basis, it was encouraging to find that ‘low volume’ teams have now adopted the use of databases and protocols for record keeping. The finding from the previous survey that there is a tendency for ‘high volume’ teams to be able to offer their patients access to a greater range of clinical specialties, compared with the low volume teams has been reinforced by the current survey. However, less than half of the ‘high volume’ teams surveyed were able to offer their patients access to a full range of clinical specialties and it has been shown that a threshold of thirty new cases per year is too low to support a full cleft team.²¹

Training for cleft surgeons involved in cleft care clearly needs to be reviewed. The size of a unit and the quality of it’s multi-disciplinary activity are of great importance in training specialists for the future. The surgery of clefts is complex and training, as distinct from service, is possible only where there is a high volume and a limited number of trainees geared to the numbers of vacancies for cleft surgeons. If standards are to be improved it is essential that young specialists undergo a properly structured training programme. Not one of the units in the United Kingdom received sufficient referrals for it to be designated as a training unit that could support two major cleft surgeons and one trainee.

In conclusion, this article which is based on a survey of 90% of cleft teams operating in the UK during 1995, has shown that there has been a dilution of cleft care since the survey by the RCS Steering Group in 1992. Progress has been made towards the establishment of databases and protocols for the collection of records but few cleft teams are able to offer their patients access to the full range of clinical specialties recommended by the Steering group.

The full findings of the CSAG study have been detailed elsewhere.^8,10,21-24 The report generated a flurry of debate, particularly on volume and outcomes. The evidence, however, still points to a need for centralisation to improve outcomes.^21,25 This has not been matched with appropriate evidence by opponents to this view.

Political changes and reorganisation of regional boundaries have made the original intentions of CSAG recommendations difficult to apply to the UK as a whole. The move to devolution of government has meant that Northern Ireland, Scotland and Wales have made their own arrangements and interpretation of the CSAG report and recommendations. In Northern Ireland, a single centre has been retained for dealing with, on average, less than forty new clefts each year.²⁶

In Scotland, a historical account of cleft care is of relevance before considering the CSAG changes. The Scottish Association for Cleft Lip and Palate (SCALP) was formed in 1989 by clinicians from different specialties involved with the care of patients with cleft lip and palate. The clinicians were grouped into six geographical teams based in; Aberdeen, Ayr, Dundee, Edinburgh, Forth Valley and Glasgow. Each team comprised four core members; a cleft surgeon, speech and language therapist, orthodontist and audiologist or ENT surgeon.

This arrangement was replaced in April 2000 by a Managed Clinical Network for Cleft Services in Scotland (CLEFTSiS) comprising all clinicians involved in cleft care. The objective of CLEFTSiS is to provide high quality care for patients with orofacial clefting (OFC) as close as possible to their home to minimise inconvenience and cost. The recommendations of the Scottish Needs Assessment (SNAP) report ²⁷ and, at least in regard to cleft surgery, the CSAG report on cleft lip and palate are being followed. The number of cleft surgeons has been reduced from six to four, working at three sites, Aberdeen, Edinburgh and Glasgow. With between 80 and 100 patients born each year with OFC, a further reduction of at least one cleft surgeon is planned. Other specialist care is provided as close as possible to the patient’s home by clinicians in the CLEFTSiS network, rather than through the ‘hub and spoke’ structure which is being implemented in England.

A lead Trust and a lead clinician from the same Trust (e.g. Tayside University Hospitals NHS Trust) are responsible to the National Services Division (NSD) of the Common Services Agency (CSA) in Scotland for co-ordinating the network. They are also responsible to the Clinical Standards Board for Scotland (CSBS) for standards of care which include the reporting of the results of the currently available routine outcome assessments of primary surgery, secondary bone grafting, speech and hearing as well as the findings of parent / patient satisfaction questionnaires. Members of the Network continue to be accountable to the Trusts holding their contract of employment but the lead clinician has a responsibility to ensure that the employing Trust is made aware of any performance issues relating to its employees about which the lead clinician becomes aware.

The CLEFTSiS management structure is made up of an Executive Group comprising, the Lead Clinician, Convenors of the specialty groups, the 0.8 WTE Administrator funded by the NSD, the Chairman of SCALP, and a lay representative from the Cleft Lip and Palate Association (CLAPA). There are six specialty groups - cleft surgery, speech and language therapy, orthodontics, audiology, audit, research and an ‘associated specialties’ group comprising paediatricians, geneticists, oral and maxillofacial surgeons, paediatric dentists, dental hygienists and paediatric radiographers.

In Wales, no finalised plan has evolved but based on population figures a single centre would be responsible for approximately 60 new cleft cases per year. Wales have continued to seek other solutions and these include a managed network between their existing centres or links with those designated centres close to the borders.

In England, the NHS Executive formed a Cleft Implementation Group (CIG) to provide advice and guidance to Regional Offices who were asked to bring their services into line with CSAG recommendations. A framework for this process was provided through a Health Services Circular (HSC 1998/238).

Regional Offices appointed Regional Leads to organise these changes in their Regions. Inevitably, with significant changes in the delivery of care there were views to be sought from parents, patients, professionals and Primary Care Groups/Trusts. CIG has broad professional representation and includes the Chief Executive of the CLAPA. The change of service in some Regions has not required consultation since the process of centralisation was already in hand at around the time of the CSAG study. In others, a full consultation with the Community Health Councils will probably be requested. It is possible, depending on patient flows and cross regional boundaries that a centre may not be appropriate in a region. Clearly this requires collaboration, co-operation and a change in culture very much in line with the new arrangements for commissioning specialised services. Cleft lip and palate care is well in advance of many specialised services with the national review and recommendations having already been established. The changes will require continual monitoring and assessment to ensure that an improved service and outcome has resulted from this reorganisation.

The authors are grateful to the Cleft Lip and Palate CSAG Committee led by Professor John Murray for helpful comment and discussion. We are particularly grateful to the CSAG Support Team, especially Dr John Newton and Dr Elizabeth West. The CSAG study was funded by the Department of Health. This study would not have been possible without the help and co-operation of the UK cleft teams to whom we extend our grateful thanks.

Correspondence: Professor J.R. Sandy, Division of Child Dental Health, University of Bristol Dental School, Lower Maudlin Street, Bristol BS1 2LY, UK