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Frequency of Le Fort I Osteotomy After Repaired Cleft Lip and Palate orCleft PalatePhoebe M. Good, D.M.D., John B. Mulliken, M.D., Bonnie L. Padwa, D.M.D., M.D.

Objective: Diminished m axillary g rowth is a consequence of labiopalatal re-pair, and many patients with cieft iip and paiate require Le Fort I advancement.The goai of this study was to determine the frequency of maxillary hypopiasiaas measured by need for Le Fort I.Subjects: Retrospective cohort study of maies born before 1987 and femalesbefore 1989. Records of 173 patients with cieft lip and paiate and 34 with cieftpaiate were reviewed.Methods: Documented age, gender, cieft type, and need for Le Fort I. Pearsonchi-square and Fischer's exact analyses were performed to evaiuate the fre-quency of Le Fort I.Results: Of 217 patients with cieft iip and palate or cieft paiate, 40 were

syndrom ic; of the rem aining 177 patients, 69 had cleft lip, 78 had cleft iip andpaiate, and 30 had cieft paiate. Thirty-seven of 177 patients (20.9%) requiredLe Fort I, subcategorized by cleft type: 0/69 for cieft iip, 37/78 for cleft lip andpalate, and 0/35 for cleft paiate (p < .0001). Of the 37/78 (47.4%) cieft lip andpalate patients, the frequency of Le Fort I correlated with severity: 5/22 uniiat-erai incompiete cleft lip and paiate; 16/33 uniiaterai complete cleft lip and pai-ate; 1/2 biiaterai incompiete cieft lip and palate; 2/4 biiaterai asymmetric com-piete/incomplete c left lip and pa late; 13/17 bilateral complete c left lip and palate(p < .05).Conclusion: Overall frequency of Le Fort I was 20.9% in patients with cleftlip and paiate and cleft pa iate. Of those w ith cieft lip and paiate, 47.7% requiredmaxiilary advancement, but none with isoiated cleft iip or cieft palate requiredcorrection. Frequency of Le Fort I osteotomy correlated with the spectrum ofseverity of iabiopaiatai clefting.KEY WORDS: cleft l ip/palate. Le Fort I osteotomy, maxillary hypopiasia

Specialists treating children with cleft lip/palate have em-phasized the effect of labiopatatal repair on facial growth.There are several studies showing that patients with unrepairedcleft lip/palate have normal maxillary growth (Orti/,-Monas-terio et al.. 1966; Mars and Houston. 1990). There are also afew studies of patients with unoperated cleft lip/palate showing

Dr. Good is a dentai s tudent at Harvard School of Dental Medicine. Boston.Massachusetts . Dr. Mulliken is Professor of Surgery. Harvard Medical School.Department of Plastic and Oral Surgery. Children's Hospital, Boston. Massa-chusetts . Dr. Padwa is Associate Professor of Oral and Maxiilofacial Surgery,Harvard School of Dental Medicine. Depanment of Plastic and Oral Surgery,Children's Hospital. Boston. Massachu,setts .

Manuscript was presented orally al the annual meeting ofthe American As-socialion of Oral and Maxillotaciiil Surgeons. San Francisco. California. Oc-tober 1.2004.

Funtl ing was provided by the Research Train ing Grant NIH/NIDCRDE0726X and Office of Enrichment Programs. Harvard Medical School.

Submit ted May 2006; Accepted September 2006.Address correspondence to: Dr. Bonnie L. Padwa, Department of Plastic and

Oral Surgery. Cbildren's Hospital, 300 Longwood Avenue, Boston, MA 021 l. ' i .E-mail bonnie.padwalp'childrens.harvard.edu.

decreased m axillary growth (Innis, 1962; Atherton, 1967; Isiekwe and Sowemimo. 1984; Bisharaet al.. 1986). The validitof these studies has been questioned, because the sample sizewere too small for statistical significance (Will. 2000).

Diminished maxillary growth is commonly observed folowing cleft lip/palate closure, and most investigators believthat this is secondary to the labial or palatal repair, or bot(Ro ss. 1987a; Mars and Hou ston, 1990; Han et al., 1995; Capelozza Filho et al., 1996; Liao et al., 2002). Ross maintainethat there is an intrinsic deficiency in midfacial skeletal growtin unilateral complete cleft lip and palate (UCLP) that is accentuated by labiopalatal repair (Ross, 1987a). Several invetigators have tried to deteniiine whether it is labial or palatarepair that causes delicient tnaxillary growth. Capelozza Filhet al. (1996) studied 93 patients with repaired UCLP and founmaxillary retrusion in those who had labial repair only. Thospatients who had both labial and palatal repair did not diffsignificantly from the labial repair-on ly grou p. They concluded that deficient maxillary growth is a consequence of labiarather than palatal, closure. In contrast, other studies hav

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Good et al., FREQUENCY OF LE FORT I IN CLEFT LIP/PALATE 39 7

illary growth (Liao et al.. 2005), suggesting instead that palatalrepair is responsible {Ross, 1987a: Mars and Houston, 1990;Han et al.. 1995; Liao et al., 2002). Mars and Houston (1990)documented that patients with UCLP had normal maxillarygrowth following labial repair in early infancy, whereas max-illary hypopiasia was common following palatal closure before2 years. Han et al. (1995) compared longitudinal craniofacialgrowth among patients with unilateral complete cleft lip (CL),UCLP. and isolated cleft palale (CP). and found that Ihere wasless forward development of the posterior limit of the maxilla(perpendicular intersect of the palatal plane with a line fromthe inferior point of the pterygomaxillary fissure) in UCLP andCP patients as compared with CL patients. Liao et al. (2002)analyzed craniofacial morphology in patients with bilateralcomplete cleft lip and bilateral complete cleft lip and palate(BCLP), and concluded that palatal repair had a more adverseeffect on maxillary length than labial repair.

Whatever causes maxillary hypopiasia, it is accepted that asubgroup of patients with repaired labial and/or palatal cleftwiil need maxillary advancement. The frequency of Le Fort Iosteotomy in UCLP and BCLP patients is reported to be 22%to 27% (R oss. 1987b; Rosenstein et ai., 1991; Cohen et al..1995; DeLuke et al.. 1997). However, many of these studieshave limitations, such as small sample size, large age range,different elinical management protocols, failure to separatecleft types, combining of sexes, inadequate controls, and in-sufficient postoperative interval. Moreover, subjective criteriaoften have been used to determine the need for orthognathiccorrection (Rosenstein et al.. 1991). Clearly, further study in adefined population of patients is needed to better delineate therelationship between maxillary hypopiasia and closure of cleftlip and palate. Furthermore, it is important to examine if thecleft severity and/or the number of operative procedures cor-relate with abnormal maxillary growth.

The g oals of this study wer e: (11 to deter min e the freque ncyof maxiliary hypopiasia. as documented by the frequency ofLe Fort I correction in cleft patients treated by one surgeonusing the same prolocol; (2) to a.scertain the frequency of max-illary adva ncem ent for various categories of clefting (CL, CLP,and CP); and (3) to establish whether cleft severity correlateswith frequency of Le Fort I osteotomy.

M A T E R I A L S AN D M E T H O D S

This retrospective cohort study only included patients whohad completed facial growth: males bom before 1987 (18 yearsold) and females born before 1989 (16 years old). We reviewedrecord s ot 217 patients with various types of CL P (n = 170)and CP (n = 47) registered in the Cleft Program at Children'sHospital Boston. All patients had their primary repair(s) byon e surgeon (J.B.M.). Patients with CLP were subcategurizedas : uniiaterai incomplete cleft lip and palate (UICLP). unilat-eral complete cleft lip and paiate (UCCLP). bilateral incom-plete cleft lip and palate (BICLP). bilateral asymmetric com-plete/incomplete cleft lip and palate (BACLP). and bilateral

TABLE I Cleft Types in Study GroupsClefl Type

Cleft lipunilateralbilateral

Cleft lip and palateunilateralbilateral

Clefl palatesoftliiird and soft

M) . of Ptilieni.s69 (39.0%)

60 (51 incomplete, 9 complete)9 (4 incomplete. 1 asymm etric.

7 8 ( 4 4 . 1 % )55 (22 incomplete. 33 complete)23 (2 incomplete, 4 asymmetric.

3 0 ( 1 6 . 9 % )1713

4 complete)

17 co mplete)

syndromic CLP or CP (including Robin sequence) were ex-cluded. We documented age. gender, ethnicity, cleft type, needfor Le Fort I corre ction, and n umb er of total operativ e pro-cedures (labial, palatal, oronasal fistula closure, nasolabial re-vision, and pharyngeal Hap). Data were recorded from the pa-tient's hospital medic al recor ds, specifically the Cleft Prog ratiinotes and operative reports.

The patients were managed according to the protocol fol-lowed in the Cieft Program at Children's Hospitai Boston from1976 to 1989:1. Single-stage labial repair for unilateral (complete or incom-

plete) or bilateral incomplete CL (3 to 5 tnonths);2. A dentofacial orthopedic appliance for BCCLP to retract

the premaxilla. to align the dentoaiveolar segment.s, and tobring the labial elements closer together prior to synchro-nous nasoiabial repair (Mulliken. 1985). Some patients hada palatal device plus elastic traction to position the pre-maxilla. (The protocol changed in 1991 to use of the La-tham pin-retained appliance for UCCLP and BCCLP, butnone of these patients were included in this study.);

3. Labial adhesion for UCCLP and complete side in BACLP(I month). Definitive nasolabial repair (3 to 6 months);

4. Soft and hard palatal repair (8 to 10 months);5. Pharyngeal flap for velopharyngeal incompetence (5 to 6

years);6. Labionasal revision (if needed), either at 3 to 5 years or at

time of alveolar bone graft;7. Alveolar bone graft and closure of oronasal fistula (during

mixed dentition); and

CL C L P C P

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398 Cleft Palate-Craniofacial Journal. July 2007. Vol. 44 No. 4

FIGURE 2 Frequency of Le Fort I among CLP patients,

8. Le Fort I osteotomy and maxillary repositioning (after com -pletion of skeletal growth).

Descriptive statistics were calculated on all study variablesafter converting the qualitative data into quantitative valuesusing Microsoft Excel features. Pearson chi-square and Fi-scher's exact analyses were performed to evaluate the fre-quency of maxillary deficiency requiring Le Fort I correctionfor each cleft type. These same analyses also were performedto evaluate the association between all variables (sex, ethnicity,and total number of procedures) and Le Fort 1 osteotomy.

RESULTS

Of 217 patients, 40 were either syndromic CLP or CP orhad Robin sequence, leaving 177 patients for analysis ( i l lmales, average age 21.5 years; 66 females, average age 20.8years). This distribution of cleft types is shown in Table 1.

Of the 177 patients, 37 (20,9%) required a Le Fort 1 pro-cedure, subcategorized by cleft type: none for CL; 37/78(47,4%) for CL P and none for CP (Fig. 1), These differences

were statistically significant (j) < .0001). The frequency of LFort I in the various subcategories of the 37/78 CLP is showin Figure 2, Note the increased need for Le Fort I correctiowith the increasing extent of the cleft. The differences in frquency of Le Fort 1 osteotomy among uniiaterai and bilaterand incomplete and complete CLP were statistically signilica(p < ,05), Due to the small numbers, however, there were nsignificant differences when comparing bilaleral incompletbilateral asymmetric, and bilateral complete CLP,

We also examined other variables and their possible assciation with Le Fort I correction. P atients who had Le Fortosteotomy had significantly {p < .0001) more operative prcedures (mean, 6.4; range. 4 to 9) than those who did n(mean. 2.6; range, I to 8). There was no correlation betweesex and need for a Le Fort I procedure; 20.7% of males an21.2% of females had this procedure. Also, ethnicity was na predictor of need for Le Fort I; between the two most common ethnicities. 21.8% (34/156) of Caucasians and 25.0(3/12) of Hispanics had maxillary coiTection.

DISCUSS ION

The requirement for Le Fort I osteotomy for all types clefts treated by one surgeon using one protocol was 20,9%The need for maxillary correction correlated with the type cleft. None of the patients with a cleft of ihc primary pala(CL) or isolated cleft ofthe secondary palate (CP) required LFort 1 advancem ent (i.e.. did not have maxillary hypop iasiwhereas 37/78 (47,4%) of patients with CLP required osteoomy. Furthermore, the frequency of Le Fort I advancemecorrelated with anatomic severity, from unilateral to biiaterand from incomplete to comple te. Le Fort I osteotomy amonpatients with UCCLP (48,5%) was similar to that of BACL

PIGURK 3 A: Frontal and B: talcral pholoj;raphs (if t->i.'ar-ld ma n with rtpairctt It-ri DCCLI' dtinoiistralint; minor flallL-iii-d midface, C: LaU

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Good ei al, . FREQUENC Y OF LE FORT I IN CLEFT LIP/PALATE 39

B / C

KKil KK 4 Patient in Fin urt i two years afU-r orlhiiclnnlii- duninipensation followed by l.eFrt I osteolomv and nia\ill:ir> ad^anci-nit-nl. dt-rmal j-rafl tphiltral riclj-f. and nasal scptal restction. Ni)U' improved nasolahial and mirifiiiial prolik' (A: fronlal \k\\, H: laUrai viiw, C: iiphaloRram).

(50%), suggesting that the repaired complete side of the cleftis responsible for deficient maxillary growth.

The h igher rate of Le Fort I osteotom y in our CLP palienls(47.4%). compared with previous reports, requires explanation.The decision for maxillary adv anceme nt is based on subjectiveassessment, usually without standardized criteria, and there-fore, the decision tor orthognathic correction is likely to varyamong centers (Rosenstein et al., 1991). Although other stud-ios focused on patients with horizontal and vertical maxillaryhypopiasia. these studies may not have included adolescentswith repaired oral clefts who have a normal dental relationship,yet might benefit aesthetically from orthognathic conection.

The high er frequency of Le Fort I osteo tomy in our unit mayreflect our preference for operative correction for all patientwho have poor midfacial aesthetics despite their occlusai relationship (Fig s. 3 through 6 ); appro xima tely l()7r of this patient sample had maxillary advancement for this reason. Furthermore, other reported rates for Le Fort 1 osteotomy are diticult to interpret and compare, due to small sample size, failure to fully separate cleft types, and combining of patienttreated by different surgeons and protocols (Ross. l9H7b: Rosenstein et al., 1991 ; Cohe n et al.. 1995: DeL uke et al.. 1997Sehnitt et al.. 2004). For example. Ross (1987b) analyzed large sample (n ^ 100) of male patients with repaired U CC LP

FK it'KK 5 A: Krontal and K: lateral photog raphs f t7-vi;n-(ilil ;;irl nilli repaired IIACL I' (led aimpltlW riuhl inrampleU"). C: flinic al an d

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40 0 Clefl Palate-Craniofacial Journal. July 2(H)7. Vol. 44 No. 4

6 Improved appearance 8 months after ti-mm hiniaxilliirj advancement an d malar implants. (A: frontal view, B: lateral view, C: cephaiogram)

treated at 15 centers by various protoeols and calculated a LeFort I rate of 27%. Rosenstein et al. (1991) evaluated 36 pa-tients treated with primary bone grafting (25 with UCCLP and11 with BCCLP) and documented Le Fort I frequency of6/25 (24.0%) and 2/11 (18.2%), respectively. Cohen et al.(1995) ineluded patients with UICLP, UCCLP BICLP, BCCLP,but did not separate cleft types based on incomplete versuscomp lete forms. They do cumen ted Le Fort I correction in 10/38 (26.3%) patients with UCLP and 7/29 (24.9%) patients withBCLP. DeLuke et al. (1997) subcategorized patients and re-ported the frequency of Le Fort I as 2/5 (40.0%) in isolatedCP 5/15 (33.3%) in UCCLP and 2/8 (25.0%) in BCCLPSehnitt et al. (2004) reported that 7/22 (32%) of all UCLPpatients needed Le Fort I osteotomy. U nlike many previousreports, our study included a large cohort of patients and con-trolled for several important variables (i.e., surgeon, operativetechnique, and treatment protocol) that have been shown toinfluence outcomes (Ross. 1987a).

Ross (1987a) suggested that patients bom with UCCLP havean intrinsic deficiency in the midfacial skeleton that Is madeworse by operations. Our data supports this concept; maxillaryhypopiasia occurred in patients with CLP, but not in those withisolated CL or CP Patients who required Le Fort I osteotomyhad significantly more operations than did those who had ad-equate midfacial position. Several investigators have tried toindict labial or palatal repair as being responsible for maxillarygrowth aberrations (Ross, 1987a; Mars and Houston. 1990;Han et al.. 1995; Capetozza Filho et al., 1996; Liao et al.,2002). In our study, however, patients who had deficient max-illary growth had had both labial and palatal repair; this pre-cluded blaming either repair as the cause of maxillary hypo-piasia. We found that the severity of cleft type, as well asnumber and extent of operative procedures, predisposes pa-

The findings of this study aid in management and counselingof cleft patients. Members ofthe cleft lip/palate team can moreaccurately inform patients and their parent.s as to the likelyneed for Le Fort I osteotomy based on cleft type. Our protocolchanged in 1991 to the use of preoperative orthopedic manip-ulation for all infants with complete CLP. This separate cohortof patients will need a similar analysis of maxillary growthonce they have reached skeletal maturity. This would be anextension of the study from our unit showing that active infantorthopedics does not affect the dental arch of preadolescentchildren with repaired UCCLP (Chan et al., 2003).

CONCLUS ION

Once skeletal growth nears completion, patienis with re-paired CLP often exhibit a characteristic concave facial profile,which requires correction by Le Fort I osteotomy and m axil-lary advancement. The prevalence of maxillary bypoplasla, de-termined by frequency of Le Fort I osteotomy, was 20.9% forall types of patients with clefts (CL. CLP and CP), treated byone surgeon, following one protocol. For all types of CLP thefrequency of Le Fort I osteotomy was 47.4%. The severity ofthe cleft type correlated with deficient maxillary growth, asreflected by an increased frequency of Le Fort I osteotomy.Acknowledgments. We thank Dr, Catherine Hayes for assistance in statisticaanalyses.

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