Home  -  About us  -  Editorial board  -  Search  -  Ahead of print  -  Current issue  -  Archives  -  Instructions  -  Subscribe  -  Contacts  -  Advertise - Login 
 
 
     

 Table of Contents  
ORIGINAL ARTICLE - PROSPECTIVE STUDY
Year : 2017  |  Volume : 7  |  Issue : 1  |  Page : 57-63

Maxillary distraction osteogenesis in unilateral cleft lip and palate patients with rigid external distraction system


1 Department of Orthodontics, University of Alabama, Birmingham, Alabama, USA
2 Department of Oral and Maxillofacial Surgery, University of Alabama, Birmingham, Alabama, USA

Date of Web Publication21-Jun-2017

Correspondence Address:
Chung How Kau
Department of Orthodontics, University of Alabama, Birmingham, Alabama
USA
Login to access the Email id


DOI: 10.4103/ams.ams_174_16

PMID: 28713737

Rights and Permissions
  Abstract 

Aim: Distraction osteogenesis (DO) is a treatment option for patients with maxillary hypoplasia secondary to cleft lip and palate (CLP). Purpose: The aim of this study is to present a technique for maxillary DO using Le Fort I osteotomy with rigid external distraction (RED) system. Subjects and Methods: The patient presented in this paper was an Asian female with CLP aged 13 years and 6 months. She presented with severe midfacial deficiency with a Class III dental malocclusion with a negative overjet and concave facial profile. Cone-beam computed tomography images were recorded preoperatively and the operation performed involved a high Le Fort I osteotomy. The appliance fabricated was banded to upper first molars used for anchorage of the RED system. Distraction of the maxilla was initiated after 7-day latency period. Results: Postoperative cephalometric analysis showed maxillary advancement anteriorly and superiorly, the total distraction treatment period was 10 days. The maxillary advancement was 10.5 mm and the SNA angle increased from 67.5° to 77.9°. Furthermore, the ANB angle changed from −9.8° to 1.6° and the occlusion changed from Class III to Class I. The profile of the face changed from concave to convex and a much better esthetic result was achieved. Conclusion: The study suggests RED system to be a reliable alternative procedure for the treatment of midfacial hypoplasia with or without cleft. Furthermore, it minimizes the risk of the surgical procedure and shortens the operating time.

Keywords: Cleft, orthodontics, rigid external distraction, surgery


How to cite this article:
Alkhouri S, Waite PD, Davis MB, Lamani E, Kau CH. Maxillary distraction osteogenesis in unilateral cleft lip and palate patients with rigid external distraction system. Ann Maxillofac Surg 2017;7:57-63

How to cite this URL:
Alkhouri S, Waite PD, Davis MB, Lamani E, Kau CH. Maxillary distraction osteogenesis in unilateral cleft lip and palate patients with rigid external distraction system. Ann Maxillofac Surg [serial online] 2017 [cited 2021 Jul 27];7:57-63. Available from: https://www.amsjournal.com/text.asp?2017/7/1/57/208639




  Introduction Top


Cleft lip and palate (CLP) is a birth defect that happens when the fetus' tissue of the lip and/or the palate do not fuse between the 5th and 12th week of pregnancy.[1] Clefts affect approximately 1 in every 600 new born babies.[2] However, the prevalence varies across geographic areas and ethnicity. A nonsyndromic CLP incidence occurs approximately 1.30 per 1000 for Asians, 1.41 per 1000 among Japanese, and 1.21 per 1000 among Chinese population [3] whereas 1.12 per 1000 live birth in European and American communities.[4] CLP affects the face and oral cavity not only in terms of esthetic and functional problems but also psychological aspects.[5] Multidisciplinary treatment and wide range of supportive care are employed on CLP patient to restore this defect to the normal range as possible, during patient lifetime.

Maxillary hypoplasia in CLP deformities results from congenital reduction in midfacial growth and the effects of the surgical scar from CLP repair.[2] Turvey et al. suggested that this disproportionate jaw growth is the biologic consequence of prior surgical intervention for closure of the soft tissues and is not related to the congenital cleft deformity.[6]

Midface hypoplasia has the following characteristics: concave facial profile, inverted nasal tip, wide alar base, acute nasolabial angle, and excessive exposure of sclera. Intraoral findings are anterior and posterior crossbite, CLP, accentuated curve of Spee, Class III dental malocclusion, multiple missing teeth, oronasal communication, and residual cleft. Speech disturbances are also usually present due to velopharyngeal incompetency and oronasal communication.[7]

Midfacial hypoplasia is commonly treated by performing conventional Le Fort surgery to displace the maxilla anteriorly and stabilization afterward with rigid fixation. However, several limitations have been reported with this procedure.[8] Therefore, distraction osteogenesis (DO) has become an alternative technique to treat craniofacial dysplasia to overcome some of this limitations. DO is a surgical technique for the growth of new bone through the application of tensile stresses to a preexisting tissue lying between two bone ends. This technique of skeletal regeneration generally involves four stages: (1) creation of a full osteotomy or corticotomy, (2) latency period or callus formation between two bones ends, (3) distraction period or bone formation by callus stretching, (4) consolidation or new bone tissue maturation.[6],[9]

Polley and Figueroa introduced maxillary distraction osteotomy in 1997 using a rigid external distraction (RED) device.[10] RED system has provided a significant alternative to treat severe midface hypoplasia with minimally invasive procedure, especially those secondary to orofacial clefting with the absence of maxillary and alveolar bone and the presence of scaring.[1] The gradual advancement and lengthening of the soft tissue in distraction osteogeneses procedure offer lower relapse rates 5.56%–10%[1] and more advancement.

In this case report, we describe the treatment with the combination of DO using RED system and orthodontics for a girl with a midfacial hypoplasia associated with a nonsyndromic unilateral CLP.


  Subjects and Methods Top


A 13 years and 6-month-old Asian female visited the orthodontics department at UAB, with a chief complaint of feeding and speaking difficulties with a compromised esthetics and occlusion. The clinical and radiographic examination (all records obtained with patient consent) indicated that she had unilateral repaired CLP associated with midface hypoplasia. Her medical history included right-sided CLP grafting surgery at age of 2 years, left iliac bone graft to maxilla for reconstruction, and closure of the oronasal fistula at 7 years old. She had unknown familial history since the patient was adopted.

The pretreatment facial photographs presented asymmetrical face with inverted nasal tip toward the left side, a chin point that was deviated 2 mm toward the right-side, wide alar base, decrease the lower face height, short upper lip, scar on the right-side of the upper lip due to early closer to the cleft lip, everted upper and lower lip, concave facial profile, acute nasolabial angle, no incisal display in the smile frontal photograph, lower dental midline deviated to the right from the facial midline [Figure 1].
Figure 1: Frontal and lateral views of 13 years and 6 months Asian female

Click here to view


Functional examination presented with normal temporomandibular joint function, speech disturbances, feeding difficulties due to velopharyngeal incompetency, and oronasal communication.

Intraoral examination showed that the dental arch form was ovoid, asymmetrical, and small upper arch in the three dimensions, retroclined upper left incisor, supernumerary tooth, deficiency in the alveolar bone due to palatal cleft, Class III molars relationships, negative 10 mm overjet, 8 mm overbite, a transversal and sagittal deficiency maxillary arch [Figure 2]. The lateral cephalometric analysis [Figure 3] and [Figure 4] showed an SNA angle of 67.5°, an SNB angle of 77.3°, and ANB angle of −9.8°. The mandibular plane angle was 30.7°. The maxillary left incisor was palatally inclined at an angle of 92.9° to the SN plane. Interincisors angle was 147.6°; soft tissue convexity was 162.0°.
Figure 2: Models and intraoral photograph

Click here to view
Figure 3: Superimposition and analysis table

Click here to view
Figure 4: Predistraction lateral cephalometric

Click here to view


The panoramic radiograph presented the right-side cleft palate with fistula, ectopic eruption of upper permanent first molars, undermined distal of upper primary second molars due to ectopic eruption of upper permanent first molars, alveolar bone deficiency due to CLP, supernumerary tooth at the upper left side, congenitally missing of upper laterals [Figure 5].
Figure 5: (a) Initial panoramic radiograph. (b) Predistraction progress panoramic radiograph

Click here to view


Clinically, the patient's periodontal tissues were healthy. Three-dimensional computed tomography images showed anteroposterior deficiency of maxilla with narrow and short upper arch [Figure 6].
Figure 6: Cone-beam computed tomography images (a) predistraction. (b) Predistraction progress. (c) Postdistraction

Click here to view


Treatment objective

Because of the midface, hypoplasia patient and her family concern about facial appearance. Thus, the treatment objectives were established as follows; (1) correct the patient's facial concavity by advancing the maxilla and coordinate the facial, maxillary, and mandibular relationship; (2) correct the skeletal Class III anteroposterior jaw relationship; (3) correct the occlusal plan to achieve dental Class I canine and molar; (4) correct the retroclined incisors position and achieve an ideal overjet and overbite relationship.

Treatment alternatives

Based on the treatment objectives, the following treatments' alternatives were considered: (1) Le Fort I maxillary orthognathic surgery advancement with rigid fixation, (2) Le Fort I osteotomy associated with intraoral distractor device, (3) Le Fort I osteotomy associated with RED system. The parents did not want to undergo extensive orthognathic surgery. Therefore, the RED device to advance the maxilla was chosen.

Treatment progress

The treatment plan was to facilitate eruption of permanent teeth to anchor the traction force of the RED system, followed by correction of midface hypoplasia and create normal relationship between upper and lower jaws by DO of the maxilla. The procedure started with leveling and aligning upper central incisors, extract the primary second molars to facilitate eruption of the ectopic upper permanent first molars. Transverse palatal arch wires between U6s extract supernumerary tooth, expose, and align impacted upper canines and upper right first premolars [Figure 7]. After aligning the erupted teeth, transpalatal arch wire removed, 050” headgear tubes are fitted on the first permanent molar, an impression is obtained of the maxillary to fabricate accustom-made device. Head gear (HG) face bow and palatal wires are around the perimeter of the dental arch with considering the incisors brackets and soldered to the upper first molars bands [Figure 8]. The external arms of HG face bow are bent over and anterior to the upper lip and eventually bent into an eyelet figure from which the splint and the RED device are connected by means of surgical wire. The position of the eyelet of the external arms is significantly important to exert control over the anteroposterior movement of the maxilla. The center of the resistance of the maxilla has been estimated to be at the level of the apices of the second of the bicuspids.[11] This guideline can be used to determine the position of the traction hooks. A force vector above the center of mass will create a clockwise rotation, whereas a force vector below it creates counterclockwise rotation, and one through the center of the mass will advance the maxilla in a linear fashion. Therefore, the placement of the traction hooks slightly above the root apices of the teeth and the osteotomy cut will provide a biomechanical favorable position. Surgery and RED system placement: The patient was hospitalized, and surgery of DO was performed under general nasotracheal anesthesia. The maxilla was injected with 1% xylocaine with epinephrine for hemostasis and the scalp was injected with 0.25% marcaine for postoperation pain, vestibular incision was performed, and mucoperiosteum was elevated. A sagittal saw was used to make a Le Fort I osteotomy through the pyriform aperture and the pterygomaxillary fissure. The maxilla was then downfractured and mobilized. The semirigid splint was then cemented with glass ionomer cement to the upper first molars and wired to upper central incisors. The RED frame was then attached to the cranium with three sharp pins bilaterally. This was secured and the lower member of the device was attached to the head frame and 24-gauge wires were used to connect to the extraoral splint arms. The device was activated to 10 mm and the maxillary moved fairly well. It was placed in the latency position. Merocel was packed in the nose and the incision was closed with 3–0 chromic suture. Patient informed to start activation after 1 week latency, 1 turn a day for 7 days with additional 3 days was essential for overcorrection. After achieving correction of sagittal maxillary deficiency, the RED system was maintained for 6 weeks without activation to permit bone consolidation. After the removal of RED device, reverse pull HG fitted and delivered with 3/16in, 6.5 oz face mask elastics. The patient informed to wear it full time to retain the distraction. Using a Facemask after removing the RED has significant stable results.[12] Same fabricated splint was used to apply the elastics force [Figure 9]. The protraction face mask was discontinued after a month and the upper splint was removed. Brackets bonded to upper first molars and full arch wire were placed to continue the dental malocclusion treatment [Figure 10]. The patient moved to another state and she was provided with a referral to continue the orthodontics treatment in Texas.
Figure 7: Predistraction extraoral and intraoral photographs

Click here to view
Figure 8: Custom-made simi-rigid splint

Click here to view
Figure 9: (a) Postdistraction extraoral view. (b) Postdistraction intraoral view. (c) Retention period by reverse headgear

Click here to view
Figure 10: Posttreatment intraoral photos

Click here to view



  Results Top


The posttreatment photographs and radiographs showed that midface advancement with proper overjet and overbite was achieved, and no insufficiency of velopharyngeal or speech function deterioration was observed [Figure 11]. The linear horizontal changes were measured relative to a line perpendicular to S-N plane passing through Sella, and the vertical changes were measured perpendicular to the S-N plane. The cephalometric analysis presented that there is pretty large improvement for maxilla as the overjet was increased by 10.5 mm. The value of SNA improved by 10.4° (A point advanced 10.5 mm anteriorly and 2 mm superiorly), while the nasal tip advanced 4 mm anteriorly and 5 mm superiorly. The upper lip also advanced 6 mm anteriorly and 2 mm superiorly. The deep bite was opened from 7.9 mm to 1.3 mm and the mandibular plane was opened by 3° [Figure 3]. The facial profile changed from concave to convex figure, anterior lower height improved by 9 mm; therefore, the facial balance and esthetics improved, the NLA improved by 26°, the maxillary incisors were proclined and 4 mm of upper centrals incisor displayed in the frontal smile photograph, increased alar base width [Figure 12] and [Figure 13], corrected posterior crossbite [Figure 14]. Posterior open bite presents after maxillary distraction. The patient and her family were satisfied with the result.
Figure 11: Pre- and post-treatment lateral cephalometric

Click here to view
Figure 12: Pre- and post-distraction extraoral photograph

Click here to view
Figure 13: Smile change pre- and post-treatment

Click here to view
Figure 14: Pre- and post-treatment intraoral change

Click here to view


Midfacial hypoplasia is commonly treated by conventional maxillary orthognathic surgery, which included some kind of Le Fort surgery depending on the severity that results in the displacement of the maxilla anteriorly and stabilization afterward with rigid fixation. However, several limitations have been reported with this procedure;[8] the literature documents 25%–40% relapse,[7] excessive blood loss, and edema;[8] it may induce an undesirable fracture, especially in the patient with abnormal bon structure such as CLP patient.[8] The frequent surgical intervention creates a tight surgical scar which is considered as a factor for growth retardation and relapse of the midface after conventional orthognathic surgery. In addition, maxillary advancement more than 6 mm is difficult to achieve using this technique due to maxillary scar.[13] Patients may have pharyngeal flaps that affect stability after conventional maxillary.

McCarthy et al., 1992 reported the first clinical application of distraction osteogeneses, they showed the elongation of hypoplastic mandible by biological process generating new bone between the bony segments.[14] Traction force applied to bone also creates tension in the soft tissue, initiating a sequence of adaptive change termed distraction histogenesis.[15],[16] DO can be performed either by intraoral distractor device or RED system. The major drawbacks of internal distractors are poor victor control, differential movement within the midface, discomfort, infection, and trauma during activation.[7] RED system has provided a significant alternative to treat severe midface hypoplasia with minimally invasive procedure. The gradual advancement and lengthening of the soft tissue in distraction osteogeneses procedure offers proper adaptation and lowers relapse rates 5.56%–10%, with more advancement.[17] This technique can be used to treat skeletal dysplasia at a young age (compared with the need to wait for skeletal maturity if conventional orthognathic surgery is to employed). The surgical procedure affected maxilla without having to operate mandible [18] and it is very simple, no need for blood transfusion or bone grafting nor hard fixation hardware. 2–5 mm overcorrection is very necessary to compensate anticipated relapse after DO procedure. The flexibility of the traction hooks allows for energy storage. This continuous tension is favorable event and is conductive to osteogenesis.[6],[19] However, after ostomizing the maxilla, less force is required to advance it; therefore, the effects on individual tooth position have been minimal.[20] The intraoral splint is custom designed, easily constructed by the orthodontics, which is helpful in patients with unusual arch forms, especially in cleft patients, inexpensive, hygienic, comfortable, none traumatic. It does not interfere with speech and eating. The main advantages of the designer of the intraoral splint are the ability to control the direction of the force vectors at any time during the distraction process without discomfort the patient, adjusting the force to pass through or above the center of the mass of the maxilla alters the rotation movement direction, activating one side more than other allows for midline correction when the maxilla skewed to one side.[19] The limitation of the rigid distraction technique relate to those patients with complete absence of teeth or lack of adequate bone in the cranial vault. Limited patient acceptance of the halo frame due to unaesthetic external component, incompliant patients, or parents may contribute to postoperative problems such as turning the screw to the wrong side, avoidance of daily turning due to pain, and forgetting daily turning of the screw when activating the traction screw.[21] Malocclusion may occur due to traction force through the teeth. The cost of the device is only a minor disadvantage.[12] Some complications related to the use of the cranium fixation have been reported such as device failure, pin loosening after trauma, and infections surrounding the pins.[22] However, patients do not complaint of the pain after placement the halo pins. Patients can shampoo their hair in the usual fashion. The device is removed in the clinic without local anesthesia after the rigid retention (consolidation) phase. Thus, we believe that it is mandatory to the professions to offer special care for patients with CLP such as innovative techniques, regular supervision, motivation to achieve the desired outcome with minor complications.[1]


  Conclusions Top


Our results demonstrate that DO using rigid external distractor allows moving the maxilla in multiple planes, simple, highly effective, stable, and predictable option for treat midface hypoplasia associated with unilateral CLP, especially when maxillary advancement more than 10 mm is required. The therapeutic results showed a significant improvement of the patient's esthetic and occlusal balance.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sudhir R, Ramnathan M. Distraction osteogenesis in the maxilla for a patient with a repaired cleft lip and palate: A case report. Spec Care Dentist 2011;31:182-7.  Back to cited text no. 1
    
2.
Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Cleft Palate J 1987;24:5-77.  Back to cited text no. 2
    
3.
Linton JL. Comparative study of diagnostic measures in borderline surgical cases of unilateral cleft lip and palate and noncleft class III malocclusions. Am J Orthod Dentofacial Orthop 1998;113:526-37.  Back to cited text no. 3
    
4.
Ahluwalia M, Brailsford SR, Tarelli E, Gilbert SC, Clark DT, Barnard K, et al. Dental caries, oral hygiene, and oral clearance in children with craniofacial disorders. J Dent Res 2004;83:175-9.  Back to cited text no. 4
    
5.
Yew CC, Alam MK, Rahman SA. Multivariate analysis on unilateral cleft lip and palate treatment outcome by EUROCRAN index: A retrospective study. Int J Pediatr Otorhinolaryngol 2016;89:42-9.  Back to cited text no. 5
    
6.
Turvey TA, Ruiz RL, Costello BJ. Surgical correction of midface deficiency in cleft lip and palate malformation. Oral Maxillofac Surg Clin North Am 2002;14:491-507.  Back to cited text no. 6
    
7.
Sahoo NK, Jayan B, Roy ID, Desai A. Evaluation of the hard and soft tissue changes after maxillary advancement with rigid external distractor in unilateral cleft lip and palate induced maxillary hypoplasia. Med J Armed Forces India 2015;71 Suppl 2:S355-61.  Back to cited text no. 7
    
8.
Yamauchi K, Mitsugi M, Takahashi T. Maxillary distraction osteogenesis using Le Fort I osteotomy without intraoperative down-fracture. Int J Oral Maxillofac Surg 2006;35:493-8.  Back to cited text no. 8
    
9.
Winters R, Tatum SA. Craniofacial distraction osteogenesis. Facial Plast Surg Clin North Am 2014;22:653-64.  Back to cited text no. 9
    
10.
Li H, Dai J, Si J, Zhang J, Wang M, Shen SG, et al. Anterior maxillary segmental distraction in the treatment of severe maxillary hypoplasia secondary to cleft lip and palate. Int J Clin Exp Med 2015;8:16022-8.  Back to cited text no. 10
    
11.
Nanda R. Biomechanics and Esthetic Strategies in Clinical Orthodontics. St. Louis, Missouri: Elsevier Health Sciences; 2005.  Back to cited text no. 11
    
12.
Wiltfang J, Hirschfelder U, Neukam FW, Kessler P. Long-term results of distraction osteogenesis of the maxilla and midface. Br J Oral Maxillofac Surg 2002;40:473-9.  Back to cited text no. 12
    
13.
Li H, Dai J, Si J, Zhang J, Wang M, Shen SG, et al. Anterior maxillary segmental distraction in the treatment of severe maxillary hypoplasia secondary to cleft lip and palate. Int J Clin Exp Med 2015;8:16022-8.  Back to cited text no. 13
    
14.
McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH. Lengthening the human mandible by gradual distraction. Plast Reconstr Surg 1992;89:1-8.  Back to cited text no. 14
    
15.
Stewart KJ, Lvoff GO, White SA, Bonar SF, Walsh WR, Smart RC, et al. Mandibular distraction osteogenesis: A comparison of distraction rates in the rabbit model. J Craniomaxillofac Surg 1998;26:43-9.  Back to cited text no. 15
    
16.
Triaca A, Minoretti R, Merz BR. Distraction osteogenesis of the mandibular angle and inferior border to produce facial symmetry: Case report. J Oral Maxillofac Surg 2000;58:1051-5.  Back to cited text no. 16
    
17.
Choi SH, Kang DY, Hwang CJ. Adult patient with hemifacial microsomia treated with combined orthodontics and distraction osteogenesis. Am J Orthod Dentofacial Orthop 2014;145:72-84.  Back to cited text no. 17
    
18.
Aduss H. Craniofacial growth in complete unilateral cleft lip and palate. Angle Orthod 1971;41:202-13.  Back to cited text no. 18
    
19.
Figueroa AA, Polley JW, Ko EW. Maxillary distraction for the management of cleft maxillary hypoplasia with a rigid external distraction system. Semin Orthod 1999;5:46-51.  Back to cited text no. 19
    
20.
Polley JW, Figueroa AA. Rigid external distraction: Its application in cleft maxillary deformities. Plast Reconstr Surg 1998;102:1360-72.  Back to cited text no. 20
    
21.
Nout E, Wolvius EB, van Adrichem LN, Ongkosuwito EM, van der Wal KG. Complications in maxillary distraction using the RED II device: A retrospective analysis of 21 patients. Int J Oral Maxillofac Surg 2006;35:897-902.  Back to cited text no. 21
    
22.
Garfin SR, Botte MJ, Waters RL, Nickel VL. Complications in the use of the halo fixation device. J Bone Joint Surg Am 1986;68:320-5.  Back to cited text no. 22
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14]



 

Top
 
 
Search
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)  

 
  In this article
   Abstract
  Introduction
  Subjects and Methods
  Results
  Conclusions
   References
   Article Figures

 Article Access Statistics
    Viewed2173    
    Printed64    
    Emailed0    
    PDF Downloaded268    
    Comments [Add]    

Recommend this journal