AA Figueroa

BK Cha

DM Luke De

F Molina

G Dua

JC Posnick

JW Polley

LK Cheung

P Scolozzi

S Gürsoy

SH Baek

SM Kim

Y Jeblaoui

YW Park

English

Young-Wook Park

Kwang-Jun Kwon

Min-Keun Kim

Crossref

CASE REPORT Open AccessLong-term follow-up of early cleft maxillarydistractionYoung-Wook Park, Kwang-Jun Kwon and Min-Keun Kim*AbstractBackground: Most of cleft lip and palate patients have the esthetic and functional problems of midfacialdeficiencies due to innate developmental tendency and scar tissues from repeated operations. In these cases,maxillary protraction is required for the harmonious facial esthetics and functional occlusion.Case presentation: A 7-year old boy had been diagnosed as severe maxillary constriction due to unilateralcomplete cleft lip and palate. The author tried to correct the secondary deformity by early distraction osteogenesiswith the aim of avoiding marked psychological impact from peers of elementary school. From 1999 to 2006,repeated treatments, which consisted of Le Fort I osteotomy and face mask distraction, and complementarymaxillary protraction using miniplates were performed including orthodontics. But, final facial profile was notsatisfactory, which needs compromising surgery.Conclusions: The result of this study suggests that if early distraction treatment is performed before facial skeletalgrowth is completed, an orthognathic surgery or additional distraction may be needed later. Maxillofacial plasticand reconstructive surgeons should notify this point when they plan early distraction treatment for cleft maxillarydeformity.Keywords: Unilateral complete cleft lip and palate, Maxillary constriction, Early distraction treatmentBackgroundThe midfacial hypoplasia or maxillary constriction isa common secondary deformity in congenital cleft de-formity involving primary palate. The causes of themidfacial hypoplasia or maxillary constriction are in-nate growth impairment [1] and scar contracture en-gaged in hard palate during the palate repair [2].Despite of orthodontic treatment, up to 25 % of pa-tients with cleft lip and palate needs surgical inter-ventions to achieve balanced and harmonious facialappearance [3].Traditional approach to manage the cleft maxillarydeformity is orthognathic surgery, which sometimeshas difficulties to achieve the surgical goal due to theskeletal clefting and excessive soft tissue scarring.Moreover, Le Fort I advancement and miniplatefixation in adult patients with cleft lip and palate de-formity showed a mean skeletal relapse of 23 % eventhough autogenous iliac bone graft had been per-formed [4].After the pioneering study [5, 6], the maxillary dis-traction technique is considered as the valuablealternative to orthognathic surgery for patients withmaxillary constriction secondary to orofacial cleft [7,8]. Moreover, this technique can be applicable duringthe period of mixed dentition, which is appealing forwhom the wait for the skeletal maturity could bepsychologically unendurable.Now, the present author reports a long-term clinicalresult of early maxillary distraction, i.e., distraction dur-ing mixed dentition for a patient with unilateral cleft lipand palate. The rationale of the early distraction was notonly psychological relieve of the patient but also withthe purpose of guiding normal maxillomandibular rela-tion until skeletal maturation. The aim of this study wasto analyze the affecting factors for successful outcome incleft maxillary distraction treatment and to provide aparticular clinical experience which might influencesurgeon’s choice of treatment strategy: conventionalosteotomy versus distraction osteogenesis.* Correspondence: omfsmk@gwnu.ac.krDepartment of Oral and Maxillofacial Surgery, College of Dentistry,Gangneung-Wonju National University, 7 Jukheon-gil, Gangneung 210-702,South KoreaMaxillofacial Plastic andReconstructive Surgery© 2016 Park et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made.Park et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 DOI 10.1186/s40902-016-0069-xCase presentationThe patient was born in 1992 with a complete unilateralcleft lip and palate and first visited to the Department ofOral and Maxillofacial Surgery, Gangneung-WonjuNational University Dental Hospital in 1997. He hadundergone cheiloplasty 5 months after birth and palato-plasty at the age of 17 months in other hospital. He hadno accompanying anomalies and also no other spe-cific medical history. We diagnosed him as a severemaxillary constriction with anterior crossbite andintraoral nasolabial fistula (Fig. 1) and planned maxil-lary distraction for early correction of patient’s maxil-lomandibular relation.On Jul 28, 1999, a high-level transverse maxillaryLe Fort I osteotomy just below the infraorbital for-amen was performed to avoid injuring the uneruptedpermanent tooth buds. The pterygomaxillary junc-tion was separated, but maxillary down-fracturingwas not performed. No movement of the osteoto-mized segments or internal fixation was achieved in-traoperatively. The halo portion of the distractiondevice was placed after the closure of the surgicalwound. Seven days after the operation, face maskdistraction was applied with external elastic force of1000 g per side for 1 month to achieve the desiredmaxillary position. The amount of maxillary ad-vancement was 10 mm at A point. The direction ofmaxillary protraction was almost parallel to the pal-atal plane. After 6-month retention period, we fin-ished the early distraction treatment (Fig. 2). Weconcomitantly delivered a chin cup for the purposeof restriction of mandibular growth with full-timeorthopedic force of 400 g per side. After maxillaryprotraction, we applied a fan type active plate formaxillary expansion for 11 months for occlusalinterdigitation.On Apr 17, 2002, alveolar bone grafting was per-formed as the canine was erupting using cancellous iliaccrestal bone. As the mandible was growing, the positionof the maxilla was getting deficient for ideal maxillo-mandibular position. So, complementary protraction ofthe maxilla was planned using miniplate as a skeletalanchorage. On May 18, 2005, seven-holed curvedminiplates (M4 Rigid Fixation System, OsteoMed,USA) were fixed to the thick zygomatic buttress areawith three 6-mm screws under general anesthesia.The lower end of the plate was exposed to the oralcavity via attached gingiva between maxillary canineand the first premolar. Three weeks after the oper-ation, the orthopedic force of 400 g per side for6 months was applied at least longer than 12 h perday with the use of protaction head gear. The direc-tion of the orthopedic force was 30° downward to theocclusal plane (Fig. 3).After protraction treatment, the mandible showedan anterior and superior rotation with loss of anteriorfacial height, and upward inclination of the occlusalplane was detected (Table 1). Facial profile was notsatisfactory due to the hyperplastic mandible andprominent frontal bossing (Fig. 4). To compromisepatient’s profile, facial contouring surgery wasplanned. On Jan 9, 2015, reduction genioplasty, para-nasal augmentation, and corrective rhinoplasty wereperformed, and 8-month follow-up photograms arepresented in Fig. 5.DiscussionManagement of severe cleft maxillary constrictionpresents a challenge for maxillofacial plastic andreconstructive surgeons. Age, status of maxillary seg-ments, amount of required maxillary protraction,type of distraction device, vector control, and stabil-ity should be carefully considered when surgeonsplan cleft maxillary distraction treatments. In thispresenting case, the overall result was not satisfac-tory for ideal patient’s profile, and the causes arediscussed.Fig. 1 Pretreatment intraoral photograms at the time of initial diagnosis presenting severe maxillary constriction and anterior crossbite (a) andthe nasolabial fistula (b) taken on Feb 25, 1999Park et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 Page 2 of 6Cheung et al. concluded distraction osteogenesistends to be preferred to conventional osteotomy foryounger cleft lip and palate patients with severemaxillary deformities in a clinical study [9]. In cleftpatients with maxillary deformity, distraction osteo-genesis was commonly performed in their age of 6to 15 [10]. At initial diagnosis and treatment plan,our hypothesis was that early established normal oc-clusion would guide normal maxillomandibularrelation at the end stage of maxillofacial growth.But, in this study, established normal occlusal inter-digitation during mixed dentition had not main-tained during the period of mandibular growthspurt. Also, it was not clear to decide the amount ofmaxillary protraction considering individual growthpotential. As a result, patient profile was not im-proved, which needs compromising contouring sur-gery. Practically, it was not persuasive to restartFig. 2 Pre-distraction and post-distraction frontal facial views (a, b) and lateral cephalograms (c, d). On Jul 28, 1999, a high-level maxillary Le Fort Iosteotomy with separation of pterygomaxillary junction was performed. Seven days after the osteotomy, face mask distraction was performedwith external elastic force of 1000 g per side for 1 month to achieve the desired maxillary position and occlusion. The direction of the force wasalmost parallel to the occlusal planePark et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 Page 3 of 6preoperative orthodontic treatment for orthognathicsurgery after completing distraction treatment. So,we had chosen the compromising surgery andfinalize the tedious treatment.Cleft maxillary distraction would be more effectiveif the alveolar bone grafting was performed before-hand [11]. We performed the distraction treatmentbefore alveolar bone grafting. So, we connected thealveolar segments by resin splint before applying thedistraction force. Nonetheless, distraction forceseemed to push the segments to the alveolar gap,thereby decreasing the amount of maxillary protrac-tion. Also, we had used a face mask to transfer thedistraction force because the more effective RED(external regid fixation) system [12, 13] had not beenso popular that time especially to children at schoolage. In this present case, face mask distraction whichused the teeth as a support, showed limited effect forideal and suitable three-dimensional movement of themaxillary segment.After face mask distraction, as the mandible wasgrowing, we needed more maxillary space for idealocclusion and maxillomandibular relation. So, we pio-neerly applied miniplate as a skeletal anchorage formaxillary protraction [14, 15]. Seven-holed curvedminiplates successfully transferred the protractionforce to the maxilla. But, face mask protractionlacked exact vector control and finally dentoalveolarcompensation developed. Also, protraction face maskand miniplate anchorage seemed to be weak to over-come the tensile force from palatal scar in this par-ticular case.ConclusionsIn summary, the author presents a clinical outcomeof repeated treatments for secondary maxillary con-striction of unilateral cleft lip and palate. In theseFig. 3 We performed complementary maxillary protraction using miniplates as the skeletal anchorage. On May 18, 2005, seven-holed curvedminiplates were fixed to the zygomatic buttress. Three weeks after the operation, the orthopedic force of 400 g per side for 6 months was appliedusing a protraction head gear. The direction of the force was 30° downward to the occlusal plane. Pre-protraction (a), after miniplate fixation (b),and post-protraction (c) lateral cephalogramsTable 1 Cephalometric measurements after maxillary distractionand protraction treatmentMeasurement Unit Norma 2011-01-12SNA degree 82.48 75.3SNB degree 80.42 85.1ANB degree 2.05 −9.8Angle of convexity degree 2.36 −26.5Mandibular length mm 121.8 133.8Midfacial Length mm 93.6 89.2Mandibular plane degree 22.75 14.0Occlusal plane-SN degree 15.24 0.9Palatal plane angle degree 0.5 −10.4Gonial angle degree 130.0 121.2Lower anterior facial height mm 76.34 75.3Nasolabial angle degree 105.0 70.2Y-axis to FH degree 61.72 51aNormal measurements of Korean people(The council of the faculty of orthodontics. Textbook of orthodontics, 2nd ediSeoul: Daehannarae; 2006. p.186–187.)Park et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 Page 4 of 6growing patients, the appropriate degree of correctioncould not be predicted. And, there was no evidencethat corrected occlusion during mixed dentition couldguide normal maxillomandibular relation at the endstage of maxillofacial growth. Therefore, the result ofthis study suggests that if early distraction treatmentis performed before facial skeletal growth is com-pleted, an orthognathic surgery or additional distrac-tion may be needed later. Maxillofacial plastic andreconstructive surgeons should notify this point whenthey plan early distraction treatment for cleft maxil-lary deformity.ConsentWritten informed consent was obtained from the patientfor publication of this case report and any accompanyingimages.Fig. 4 Post-treatment (maxillary distraction and complementary protraction) oblique (a), frontal (b), and profile (c) views taking on Jul 21, 2014Fig. 5 On Jan 9, 2015, the patient underwent reduction genioplasty, paranasal augmentation, and corrective rhinoplasty using autogenous ribcartilage. Oblique (a), frontal (b), and profile (c) views 8 months after compromising contouring surgeryPark et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 Page 5 of 6Competing interestsThe authors declare that they have no competing interests.Author’s contributionsYW surgically treated all patients and wrote the manuscript. MK and KJcontributed significantly to the treatment of the patient. All authors read andapproved the final manuscript.AcknowledgementsI cordially thank Prof. Bong-Kuen Cha (Department of Orthodontics,Gangneung-Wonju National University Dental Hospital) for his orthodontictreatment of this patient.Received: 14 March 2016 Accepted: 28 April 2016References1. Kim SM, Kim JH, Kim JH, Park YW, Lee JH, Lee SK (2007) Abnormal growthpattern of human fetal maxilla with cleft lip and palate. J Korean Assoc OralMaxillofac Surg 33:238–462. Park YW, Min BI (1990) A clinical study on secondary cleft lip and/or palatedeformity. J Korean Assoc Oral Maxillofac Surg 16:101–113. De Luke DM, Marchand A, Robles EC, Fox P (1997) Facial growth and theneed for orthognathic surgery after cleft palate repair: literature review andreport of 28 cases. J Oral Maxillofac Surg 55:694–74. Posnick JC, Dagys AP (1994) Skeletal stability and relapse patterns after LeFort I maxillary osteotomy fixed with miniplates: the unilateral cleft lip andpalate deformity. Plast Reconstr Surg 94:924–325. Molina F, Ortiz Monasterio F, de la Paz AM, Barrera J (2011) Maxillarydistraction: aesthetic and functional benefits in cleft lip-palate andprognathic patients during mixed dentition. Plast Reconstr Surg 101:951–636. Polley JW, Figueroa AA (1998) Rigid external distraction: its application incleft maxillary deformities. Plast Reconstr Surg 102:1360–727. Scolozzi P (2008) Distraction osteogenesis in the management of severemaxillary hypoplasia in cleft lip and palate patients. J Craniofac Surg 19:1199–2148. Gürsoy S, Hukki J, Hurmerinta K (2010) Five-year follow-up of maxillarydistraction osteogenesis on the dentofacial structures of children with cleftlip and palate. J Oral Maxillofac Surg 68:744–509. Cheung LK, Chua HDP, Hägg MB (2006) Cleft maxillary distraction versusorthognathic surgery: clinical morbidities and surgical relapse. Plast ReconstrSurg 118:996–100810. Cheung LK, Chua HDP (2006) A meta-analysis of cleft maxillary osteotomyand distraction osteogenesis. Int J Oral Maxillofac Surg 35:14–2411. Jeblaoui Y, Morand B, Brix M, Lebeau J, Bettega G (2010) Maxillary distractioncomplications in cleft patients. Rev Stomatol Chir Maxillofac 111:e1–612. Figueroa AA, Polley JW, Friede H, Ko EW (2004) Long-term skeletal stabilityafter maxillary advancement with distraction osteogenesis using a rigidexternal distraction device in cleft maxillary deformities. Plast Reconstr Surg114:1382–9213. Dua G, Navin Kumar A, Roy ID, Roy SK (2014) Maxillary distraction osteogenesisin cleft lip and palate cases with midface hypoplasia using rigid externaldistractor: an alternative technique. J Craniofac Surg 25:746–5114. Baek SH, Kim KW, Choi JY (2010) New treatment modality for maxillaryhypoplasia in cleft patients. Angle Orthod 80:783–9115. Cha BK, Ngan PW (2011) Skeletal anchorage for orthopedic correction ofgrowing class III patients. Semin Orthod 17:124–37Submit your manuscript to a journal and beneﬁ t from:7 Convenient online submission7 Rigorous peer review7 Immediate publication on acceptance7 Open access: articles freely available online7 High visibility within the ﬁ eld7 Retaining the copyright to your article    Submit your next manuscript at 7 springeropen.comPark et al. Maxillofacial Plastic and Reconstructive Surgery  (2016) 38:20 Page 6 of 6

Maxillofacial Plastic and Reconstructive Surgery

Long-term follow-up of early cleft maxillary distraction

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Long-term follow-up of early cleft maxillary distraction

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