Cleft Lip and Palate Management by U.K. Consultant Oral and Maxillofacial Surgeons: A National Survey P.A. BRENNAN, F.D.S.R.C.S., F.R.C.S., F.R.C.S.I. L.V. MACEY-DARE, M.SC., F.D.S.R.C.S., M.ORTH.R.C.S. T.R. FLOOD, F.D.S.R.C.S., F.R.C.S. A.F. MARKUS, F.D.S.R.C.P.S., F.D.S.R.C.S. R. UPPAL, B.D.S. Objective: Following the report of the Clinical Standards Advisory Group (CSAG), a national survey of U.K. consultant oral and maxillofacial surgeons was performed to determine the current cleft lip and palate practice of this group prior to the implementation of proposed radical changes in the delivery of cleft services. The views of these surgeons regarding the proposed changes was also sought. Design: An anonymous postal questionnaire sent to all 266 consultants in the U.K., which requested information on the practice of cleft surgery during a defined 1-year period (March 1997 through February 1998). It included the numbers and types of procedures performed, the involvement of multidisciplinary care, research and audit activity, and questions regarding the implementation of CSAG. Results: One hundred ninety-one replies (72% response rate). Seventy-three surgeons were actively involved with mainly secondary cleft surgery. A varied number and range of procedures were undertaken, with most surgeons performing less than five of each procedure per year. Audit and research activity was 26%. The majority of both noncleft and cleft surgeons agreed with proposals made by CSAG (except for cleft osteotomy procedures). Conclusions: In the U.K. at present, there are many oral and maxillofacial surgeons performing mainly secondary cleft surgery; overall, the number of procedures performed by these surgeons per year is small. Intraspecialty referral is suggested to further improve patient outcome. KEY WORDS: cleft lip and palate, Clinical Standards Advisory Group, oral and maxillofacial surgeons

The management of the cleft lip and palate patient in many parts of the world is undertaken by a number of the surgical specialties including oral and maxillofacial surgery, plastic surgery, otorhinolaryngology, and even general surgery. Oral and maxillofacial surgeons are, by virtue of their considerable

training, uniquely qualified to manage both hard and soft tissue conditions affecting the orofacial region. In contrast, the majority of plastic surgeons are not trained in hard-tissue surgery. Therefore the collaboration of specialties in the management of conditions such as the cleft lip and palate deformity is paramount, with optimum patient care and improved outcome being the ultimate goal. In the U.K., treatments provided by the National Health Service have undergone extensive audit to facilitate these aims (Parberry and Banerjee, 1995). In 1998 the report of the Clinical Standards Advisory Group (CSAG) into the management of cleft lip and palate surgery in the U.K. was published. The report has been summarized by Williams and Markus (1998). It addressed past practice and proposed changes to the management of cleft lip and palate care in the U.K. The objectives were achieved by accurately establishing the incidence of the deformity, identifying minimum standards, and reviewing outcomes in other European centers and by both a process and outcome audit for 5- and 12-year-olds born with unilateral cleft lip and palate. It was also necessary to identify variations in standards of care among

Mr. Brennan is a Specialist Registrar and Mr. Uppal is a Senior House Officer in Oral and Maxillofacial Surgery, Queen Alexandra Hospital, Portsmouth, United Kingdom. Ms. Macey-Dare is a Senior Registrar in Orthodontics, Queen Alexandra Hospital, Portsmouth, United Kingdom. Mr. Flood is a Consultant Oral and Facial Surgeon, Salisbury Hospital, Salisbury, United Kingdom. Mr. Markus is a Consultant Oral and Maxillofacial Surgeon, Poole General Hospital, Poole, United Kingdom. Submitted August 1999; Accepted March 2000. This study was funded by Geistlich Biomaterials, Long Lane, Chester, United Kingdom. This paper was presented at the Annual meeting of the British Association of Oral and Maxillofacial Surgeons, held in Birmingham, United Kingdom, June 2–4, 1999. Reprint requests to: Mr. Peter Brennan, Specialist Registrar in Oral and Maxillofacial Surgery, c/o 11, Oxlease Close, Romsey, Hants SO51 7HA, United Kingdom. E-mail: [email protected]. 44

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FIGURE 2 Frequency of cleft lip revisions performed in the study year.

FIGURE 1 A: Frequency of unilateral alveolar grafts performed in the study year. B: Frequency of bilateral alveolar bone grafts performed in the study year.

centers in this country. This was achieved by visiting 17 representative units, chosen on an agreed plan, taking into account levels of organization, the numbers of cleft lip and palate patients treated, and geographical location. Probably of greatest interest are the outcomes and the inferred relationship between volume and outcome. There are currently 57 teams in the U.K. involved in cleft care, with 75 surgeons performing primary repairs. This includes 56 plastic surgeons, 15 oral and maxillofacial surgeons, 3 pediatric surgeons, and 1 otolaryngologist. Extensive epidemiological research has demonstrated that there are between 900 and 1000 new cleft cases per year in the U.K. The outcome audits published in CSAG showed that 70% of 12-year-olds had a skeletal class 3 relationship, as opposed to 5% in the noncleft population. Bone grafting of the alveolus between the ages of 9 and 11 was seriously deficient or a complete failure in 42% of 12-year-olds. Speech intelligibility was poor or nonexistent in 41% of 5-year-olds and 19% of 12year-olds. Interestingly 46% of 5-year-olds and 15% of 12year-olds requiring speech therapy were not receiving it. The findings of CSAG confirmed the previous Eurocleft results published by Shaw (1992).

No relationship between volume and outcome was demonstrable; however, the four surgeons with the best results were middle-volume operators. A summary was produced by the committee of CSAG defining what they considered to be the ideal pattern of clinical care from diagnosis to maturity to establish a pattern on which future protocols could be based. The CSAG report has generated many debates in the U.K. among the specialties involved with most agreeing, in general, with the recommendations. In light of the continuing debate surrounding the CSAG report, we decided to seek the views of all consultant oral and maxillofacial surgeons in the U.K. by means of a postal questionnaire. To our knowledge, there has not been a previous national survey undertaken in the U.K. to assess the management of patients with cleft lip and palate by oral and maxillofacial surgeons. National U.K. surveys have, however, been conducted among plastic surgeons by Pigott (1992) and orthodontists by Williams et al. (1996). The study by Pigott (1992) addressed the issues of primary lip and palate repair, while the latter survey looked at orthodontic services. The purpose of this survey was firstly to establish a database of current cleft practice by oral and maxillofacial surgeons in the U.K. prior to the implementation of CSAG, which could subsequently be used

FIGURE 3 Frequency of pharyngoplasty performed in the study year.

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Cleft Palate–Craniofacial Journal, January 2001, Vol. 38 No. 1

TABLE 1. Number of Consultants Routinely Performing the Procedures Shown in Patients Undergoing Alveolar Bone Grafting (ABG) Procedure Related to ABG

Assessed by orthodontist before surgery Presurgery palatal expansion Revise lip at time of surgery Extract teeth at time of surgery Routinely use healing plate Total number of consultants

FIGURE 4 Frequency of cleft rhinoplasty performed in the study year.

Number (%)

58 36 5 49 6 58

(100) (62) (9) (84) (10)

surgeons, orthodontists, and other members of the multidisciplinary team and 19 (26%) reported that were actively involved in research or audit of cleft management. Primary Surgery

to compare with the proposed services in the future. Secondly, we wished to seek the views of this group regarding the proposed reconfiguration of cleft services. METHOD An anonymous postal questionnaire was sent to all consultant oral and maxillofacial surgeons in the U.K., identified from the handbook of the British Association of Oral and Maxillofacial Surgeons. The questionnaire was divided into two parts. In the first part, questions concerning the practice of cleft lip and palate surgery were addressed, and these were subdivided into the headings of primary lip and palate repair, alveolar bone grafting, pharyngoplasty, cleft lip revision, cleft rhinoplasty, and osteotomy. The questionnaire inquired about the total numbers of each procedure performed during the year March 1997 to February 1998 and to specific issues regarding patient management. In the second part, opinions as to the reconfiguration of the subdivisions of cleft practice named above were sought. A reply prepaid envelope was enclosed, and the consultants were given 2 months to respond. Because this was an anonymous questionnaire, no reminders were sent. RESULTS Two hundred sixty-six designated questionnaires were sent, with 191 consultants replying (72% response rate) within the designated 2-month period. Of these, 73 (38%) stated that they were regularly involved in one or more aspects of cleft care, and 118 (62%) stated that they had no cleft practice. Out of the 118 replies, 40 also had views concerning the reconfiguration of services. The remaining 78 respondents did not comment.

Seven respondents stated that they undertook primary repair of cleft lip and palate. However, one of these gave no results. The remaining six undertook between 3 and 22 lip repairs in the study year. All the surgeons stated that they performed the Delaire repair. For the management of cleft palate, six surgeons performed between 2 and 18 repairs during the study year. Alveolar Bone Grafting Fifty-eight (79%) surgeons stated that they performed alveolar bone grafts; however, the numbers being undertaken per consultant per year varied widely (Figs. 1A and 1B). The management of patients prior to and during alveolar bone grafting also varied (Table 1), as did the procedure for harvesting the bone (Table 2). Secondary Surgical Procedures The numbers of consultants performing cleft lip revisions, pharyngoplasty, and cleft rhinoplasty were 13 (18%), 13 (18%), and 18 (25%), respectively. As can be seen from Figures 2, 3, and 4, the total numbers of these procedures performed during the year in question were small. Fifty-seven (78%) consultants reported that they undertook cleft osteotomies regularly, with the Le Fort 1 osteotomy being the most common maxillary procedure (Table 3). The numbers for both maxillary procedures and bimaxillary osteotomies are shown in Figures 5 and 6, and the clinical management of osteotomy patients is summarized in table 4.

TABLE 2. Site of Bone Harvest Used by Consultants in Alveolar Bone Grafting Site of Bone Harvest

Multidisciplinary Management Of the 73 consultants who stated that they undertook regular cleft work, 55 (75%) regularly attended joint clinic with plastic

Iliac crest (open procedure) Iliac crest (trephined) Tibia Total number of consultants

Number (%)

50 (86) 5 (9) 3 (5) (58)

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TABLE 3. Number of Consultants Routinely Performing Cleft Osteotomies in the Maxilla at the Level Shown Level of Maxillary Osteotomy

Le Fort 1 Le Fort 1 or 2 Le Fort 1, 2, or 3 Variable Total number of consultants

Number (%)

39 8 1 9 57

(68) (14) (2) (16)

Views Regarding Reconfiguration The 73 surgeons who stated that they regularly undertook cleft procedures all had views regarding the reconfiguration of services; these are shown in Table 5. Forty of the 118 surgeons who did not provide cleft services also expressed their views regarding the reconfiguration of services (Table 6). DISCUSSION The results of this U.K. study show that 38% of the oral and maxillofacial surgeons who responded are involved with one or more aspects of cleft care with the majority being involved in secondary surgery. In comparison, the postal questionnaire of U.K. plastic surgeons conducted by Pigott (1992) found that of the 101 respondents, 71 (70%), performed primary lip and palate repair. It would appear that the number of plastic surgeons undertaking these types of procedures is decreasing. Shaw et al. (1996b), reporting on a postal questionnaire carried out in 1992, stated that of the 76 surgeons who performed primary lip and palate repair, 64 were plastic surgeons, and only 8 were oral and maxillofacial surgeons. The CSAG report showed that the number of plastic surgeons performing primary procedures has fallen further to only 57 with the number of oral and maxillofacial surgeons increasing to 15. It was disappointing that less than half of these 15 surgeons responded to our survey. However, of those who did reply, two performed 10 or more primary lip repairs and 3 performed

FIGURE 6 Frequency of bimaxillary procedures performed in the study year.

10 or more palate repairs during the study year. In other studies, these would be classified as high-volume operators, which is encouraging since it has been a stated aim of the British Association of Plastic Surgeons (1994) that cleft surgery should not be carried out by the occasional operator. This has been further reinforced by the CSAG report. In one cohort of children studied by CSAG, 83 surgeons reported that they undertook primary lip repair. Of these, only seven surgeons (8%) performed 10 or more primary lip repairs over the defined 2-year period. Of the remaining 76 surgeons, 66 performed between only one and four cases of primary lip repair. In our opinion, the move to fewer, high-volume operators should be supported in all aspects of cleft care. This could possibly be achieved by a greater degree of intraspecialty referrals. With regard to alveolar bone grafting, 72% of those surgeons studied in the CSAG report performed one or less procedures per year, 26% between two and four procedures per year, and only 2% more than five procedures per year. Our study has shown that among the 58 oral and maxillofacial surgeons who stated that they performed unilateral alveolar bone grafting (Fig. 1A), 10% performed no procedures, 52% performed between one and four procedures, and 38% performed five or more procedures during the study year. The last group could again be classified as high-volume operators. The numbers for bilateral procedures are shown in Figure 1B. This shows that 11 (19%) performed four of more procedures during the study year. Overall, the oral and maxillofacial surgeons who responded to our survey would appear to be performing

TABLE 4. Number of Consultants Routinely Performing the Procedures Shown in Patients Undergoing Cleft Osteotomies Procedure Related to Cleft Osteotomy

FIGURE 5 Frequency of maxillary osteotomies performed in the study year.

Assessed with orthodontist prior to surgery Preoperative orthodontics Bone grafting of maxilla Lip revision at time of osteotomy

Number (%)

57 56 24 3

(100) (98) (42) (5)

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TABLE 5. Cleft Surgeons’ Views on Reconfigurations of Services

Cleft Service

Primary lip and palate repair Alveolar bone grafting Cleft lip revision Pharyngoplasty Cleft rhinoplasty Cleft osteotomy

TABLE 6. Noncleft Surgeons’ Views on Reconfigurations of Services

Number (%) (n 5 73)

60 37 53 55 49 36

(82) (51) (73) (75) (67) (49)

more procedures per consultant than the surgeons studied in the CSAG report. The number of consultants undertaking cleft lip revision, pharyngoplasty, and rhinoplasty were small, demonstrating the need for reconfiguration of services in these areas. Another area of interest is cleft osteotomy procedures, which are almost exclusively undertaken by the specialty of oral and maxillofacial surgery in the U.K. Of the 57 who stated that they undertook these procedures in our survey, the majority (72%) performed two or less bimaxillary procedures during the study year, and only 9% performed five or more procedures. All the consultants saw their osteotomy patients in a combined clinic, and all but one (98%) routinely used preoperative orthodontics. It might seem sensible to consider referring both alveolar bone grafts and cleft osteotomies to named high-volume operators; however, this could create problems regarding access to orthodontic treatment. The numbers question is clearly important and has previously been raised by Markus (1995). The report of the Royal College of Surgeons published by Shaw et al. (1996a) suggested that a minimum of 28 new patients each year would generate sufficient numbers of unilateral cleft lip and palate deformities for the purposes of creditable audit within 5 years. Our study has demonstrated that this target could easily be achieved by intraspecialty referral, but a well-designed audit relating to outcome would need to be in place beforehand. Since our survey showed that only 19 (26%) of the consultants performing cleft procedures were actively involved with research and audit at present, this is an obvious area for attention. Although this study has shown a clear difference in opinion regarding the reconfiguration of services for cleft patients between cleft and noncleft surgeons, the latter group were also overwhelmingly in favor of reconfiguration for all aspects of cleft care. The majority of cleft surgeons favored reconfiguration of all aspects of cleft surgery with the exception of cleft osteotomy surgery, which nonetheless received the support of

Cleft Service

Primary lip and palate repair Alveolar bone grafting Cleft lip revision Pharyngoplasty Cleft rhinoplasty Cleft osteotomy

Number (%) (n 5 40)

38 30 38 34 32 32

(95) (75) (95) (95) (80) (80)

49% of the surgeons responding in this group. The need for a major reconfiguration of cleft services in the U.K. is not in question; however, reorganization must take into account accessibility. Cleft services can be provided only in a properly equipped, truly multidisciplinary setting with a team of fully trained surgeons practicing cleft surgery on a regular basis. The justification for oral and maxillofacial surgeons continued involvement in all stages of cleft care remains strong as long as they accept the need to maintain high standards and improve outcomes in an area of surgery that will demand increasingly high standards by patients, purchasers, and our peers. Acknowledgments. We would like to thank all the surgeons who took the time to complete and return the questionnaire. We would also like to thank Geistlich Biomaterials for their financial support.

REFERENCES British Association of Plastic Surgeons. Plastic Surgery in the British Isles. London: British Association of Plastic Surgeons; 1994. Markus AF. Surgery by numbers. Br J Oral Maxillofac Surg. 1995;33:205– 206. Parberry AC, Banerjee AK. Quality assurance and quality management in the National Health Service. J R Soc Health. 1995;115:109–112. Pigott RW. Organisation of cleft lip and palate services—results of a questionnaire. Br J Plast Surg. 1992;45:385–387. Report of the Clinical Standards Advisory Group (CSAG). London: HMSO; 1998. Shaw WC. A six centre international study of treatment outcome in patients with clefts of the lip and palate. Part 5. General Discussion and conclusions. Cleft Palate Craniofac J 1992;29:413–418. Shaw WC, Williams AC, Sandy JR, Devlin HB. Minimum standards for the management of cleft lip and palate: efforts to close the audit. Ann R Coll Surg Engl. 1996a;78:110–114. Shaw WC, Williams AC, Sandy JR, Devlin HB. The surgical care of cleft lip and palate patients in England and Wales. Br J Plast Surg. 1996b;49:150– 155. Williams JL, Markus AF. Cleft care: life after CSAG. Br J Oral Maxillofac Surg. 1998;36:81–83. Williams AC, Sandy JR, Shaw WC, Devlin HB. Consultant orthodontic services for cleft patients in England and Wales. Br J Orthod. 1996;23:165– 169.