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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 5  |  Issue : 2  |  Page : 124-127

Surgical management of a vomeropremaxillary fracture in a patient with complete bilateral cleft lip and palate following trauma


1 Department of Orthodontics and Dentofacial Orthopedics, Institute of Dental Sciences and Hospital, Modinagar, Uttar Pradesh, India
2 Department of Plastic Surgery, Sant Parmanand Hospital, New Delhi, India

Date of Web Publication26-Jul-2018

Correspondence Address:
Dr. Karan Sharma
Institute of Dental Sciences and Hospital, Modinagar, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_10_18

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  Abstract 


Timing of treatment of protruding premaxilla in a bilateral cleft lip and palate (BCLP) patient is an issue of dilemma. Fracture of the premaxillary segment following trauma in a patient with complete BCPL is presented. Salvaging the mobile premaxilla becomes an emergency. Surgical management of such a fracture has been discussed in a patient with repaired cleft lip and palate in the light of recent literature.

Keywords: Bilateral cleft lip and palate, fracture, premaxilla, surgical management, vomer


How to cite this article:
Sharma K, Singh S, Batra P, Sood S C. Surgical management of a vomeropremaxillary fracture in a patient with complete bilateral cleft lip and palate following trauma. J Cleft Lip Palate Craniofac Anomal 2018;5:124-7

How to cite this URL:
Sharma K, Singh S, Batra P, Sood S C. Surgical management of a vomeropremaxillary fracture in a patient with complete bilateral cleft lip and palate following trauma. J Cleft Lip Palate Craniofac Anomal [serial online] 2018 [cited 2019 Jan 20];5:124-7. Available from: http://www.jclpca.org/text.asp?2018/5/2/124/237630




  Introduction Top


Cleft lip and palate is the second most frequent major congenital anomaly (1:750–1:1000 live births), with clubfoot being the most common.[1] The incidence appears to be slowly rising, as the incidence has increased from 1.45/1000 live birth in 1942 to 1.89/1000 live birth in 1981. Clefts of lip and palate are more common in American Indians (3.7/1000 live birth) followed by Japanese (2.7/1000), Maoris and Chinese (2.0/1000), Caucasians (1.7/1000), and Negroids (0.4/1000).[2] Complete bilateral cleft lip and palate (BCLP) is the most severe of the common orofacial cleft subtypes. Failure of the premaxillary segment to fuse with the lateral maxillary segments results in a complete bilateral cleft of the lip. Subsequent forward growth of the premaxilla, attached only to the vomer above, leads to its projection beyond the lateral segments. In infancy and before surgery, the craniofacial morphology is characterized by a prominent premaxilla, a retrognathic maxilla, reduced posterior maxillary height, and a small, retruded mandible.[3] The prolabium is devoid of any muscle fibers. The nostrils are stretched, and the tip of the nose is broad. The columella appears to be shortened or nonexistent, and the prolabium often seems to be joined directly to the tip of the nose.[4]

Fractures of the premaxillary complex are rarely seen, and diagnosis, management, and follow-up have been rarely reported, even though trauma to the premaxillary segment and teeth within is seen commonly.[5] Da Silva et al[6] conducted a study to analyze the prevalence of oral trauma in subjects with complete bilateral clefts, with anterior projection of premaxilla. A sample of 106 children aged between 6 and 9 years was analyzed. It was found that 53% of the sample had suffered from oral trauma, the majority sustained soft tissue lesions (91%), and dental trauma including avulsion, luxation, and intrusion of teeth was observed in the rest. Trauma specific to the premaxillary region was also assessed. Around 75% trauma affected the soft tissue of premaxilla, 16% of trauma involved the maxillary incisors, followed by 9% of cases in which there was an association between lesion in premaxilla and maxillary incisors. Traumatic injuries to the premaxillary segment and teeth within the premaxilla have often been reported, but the fracture of the premaxillary-vomer region has been rarely reported.


  Case Report Top


A 3-year-old boy with repaired BCPL and protruding premaxilla had suffered a fall at his home while playing. Patient experienced mild discomfort at the site of repair of lip. Patient's parents noticed slight bleeding and swelling along with movement in upper front tooth region. They presented to the dental outpatient department a day after the incident. The patient was previously operated at 8 months for lip repair and at 12 months for palatal repair; 6 months later, a revision surgery was done for the palate. Upon examination, swelling was noted in upper lip philtrum region with no associated laceration. There was slight bleeding from the premaxillary region which was controlled by applying moist saline packs. There was no loss of sensorimotor functions of the patient; palpation was done to rule out fractures of the orbital rim and the mandible. The premaxillary segment was mobile with no subluxation and luxation of the primary central incisors [Figure 1].
Figure 1: Displaced and mobile premaxilla

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Patient was referred for radiological examination. A noncontrasted cone-beam computed tomography (CBCT) was done which revealed an obliquely oriented minimally displaced fracture involving the premaxillary vomer segment [Figure 2]. Along with fracture, CBCT also revealed repaired cleft defect. A bilateral osseous defect/cleft was present involving the anterior maxillary alveolus and floor of the nasal cavity. It extended posteriorly to involve the anterior and posterior hard palate till the posterior margin bilaterally. The transverse dimension of the defect was approximately 3.6 mm and 6 mm, respectively, on the right and left side in the anterior hard palate region, 1.8 cm in the mid-palatal region, and 1.0 cm at the junction of hard and soft palate.
Figure 2: Fracture (indicated by the arrow) in the sagittal section of cone-beam computed tomography

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Surgery

Since the fracture was minimally displaced, it was decided to manage the mobile premaxilla and the lateral segments with just the help of 4-0 chromic sutures. Four weeks later, the premaxilla was still mobile, so a decision was made for open reduction and fixation. After the initial preanesthetic consultations, the patient was placed under general anesthesia. Patient was placed in Rose position, and a mouth gag was applied by placing its prongs carefully on the lateral segments. After draping and disinfection, the fracture site was accessed by a very small midline intramucosal incision. Care was taken to avoid reflecting the mucoperiosteum as much as possible. The margins at the fracture site were smoothened and a small ostectomy was performed on the vomer bone to enable the setback of the premaxilla, which was then set in alignment with the lateral alveolar arches.

The repositioned premaxilla was fixed using Kirschner wire pins (0.028). The patient was put on soft diet in the postoperative period. The approach incisions were closed using 3-0 Vicryl interrupted sutures. Healing of the repositioned segment was uneventful [Figure 3] and mucosal fusion had started between the repositioned premaxilla and the lateral segments.
Figure 3: Healed fracture and reduced premaxillary segment

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Six weeks after the wire fixation, the K pins were removed after making sure that the premaxilla was fixed and mobility was not present.


  Discussion Top


Cleft lip and palate is a second most common birth defect, with only 10% of patients having bilateral deformity. Usually, patients with repaired bilateral clefts have a higher incidence of oral trauma than the general population.[6] Complete BCLP patients show a characteristic anterior projection of the premaxilla which often persists even after lip repair.[4] The protruding premaxilla, unrestrained by either of the maxillary alveoli, is only attached to the nasal septum by the septomaxillary ligament. In normal children, the cartilaginous septum must slide forward in relation to the premaxillary region because of the restraint on the premaxilla by the lip and lateral maxillary segments. In the bilateral cleft, the premaxilla is carried forward at the same rate as that of the growing septum to which it is firmly held. The premaxilla has only one restraining connection, the vomer. This restraint is realized as a tension between these bones borne by the vomero-premaxillary suture, thus creating the conditions for bone formation.[7] Often, there is disproportion between the size of the premaxilla and the size of the gap where it should lie between the maxillary segments.[8] Premaxillary protrusion is the result of excessive bone apposition at the vomero-premaxillary junction and the absence of restraint by the maxillary elements.[9] Latham [8] showed histological evidence that rapid hard-tissue formation during growth takes place at this site. This anatomical feature in BCLP children together with an increased overjet predisposes this region to higher incidence of oral trauma than general population. Although trauma to the premaxillary region in bilateral cleft cases has been reported, the fractures at the premaxillary vomer area have been rare.

Presurgical orthopedics procedures such as nasoalveolar molding (NAM) have shown promising results to manage a protruding premaxilla. NAM can be employed early so that the lip repair can be done easily.[10] Even though few studies have questioned the efficacy of NAM,[11],[12] any procedure, which could prevent the need for an additional surgical intervention, would be better in guiding the growth of the midface.[13] Many patients, unfortunately, lose out on the benefit from the timely execution of presurgical orthopedics and thus end up requiring surgical correction. Patients requiring the osteotomy of premaxilla for setback in bilateral cleft palate cases are usually carried out after the age of 6 years to avoid any deleterious effect on the growth of the nasomaxillary complex.[14] The patient presented with the fracture at the age of 3 years, so a decision was made to manage the premaxillary segment more conservatively and avoid the need for an extensive surgical procedure. An attempt was made to fix the premaxilla with the lateral segments using stabilizing sutures. The premaxilla was mobile even after 4 weeks, so open reduction and fixation was the only option left to stabilize the premaxilla.

Open reductions usually require approach incisions and reflection of full-thickness flaps. This is routine for normal patients, but this could be problematic in bilateral cleft patients as extensive periosteal stripping could jeopardize the blood supply of the premaxilla. Care was taken for the patient to minimize the need for extensive reflection of the mucoperiosteum for the access to the fracture site. Three-dimensional imaging in the form of CBCT also helped in the accurate assessment of the fracture site.

Many studies in the literature have documented the importance of the vomero-premaxillary suture as the primary growth site for the midface.[15],[16] Any surgical manipulation of this area before the age of 8 years might hamper the growth of the entire midface.[15] In the present case since an open reduction was done to reduce and fix the fracture segments, it was decided to perform a minor ostectomy of the vomer bone to move the premaxilla dorsally. Even though this might be deleterious to the overall growth of the midface, repeated risk of trauma to the premaxilla and eventual loss of the premaxillary segment outweighed the risk of growth retardation.

Various techniques have been described for the fixation of the premaxillary segment. Kirschner wires, cortical bone grafts, occlusal splints, and direct interdental wiring and rigid internal fixation have been used.[17],[18],[19] In this case, Kirschner wire was used for fixation The Kirschner wire was first introduced in 1909 by Martin Kirschner. Kirschner or K-wires or pins are smooth stainless pins. They come in different sizes and are used to hold bone fragments together or provide an anchor for skeletal traction.

Kirschner wires provide a stable fixation and have the added advantage that a second surgical procedure is not usually required to remove them. They can be easily removed under local anesthesia or conscious sedation.


  Conclusion Top


A rare case of fracture of the vomer-premaxillary complex is presented. Bilateral cleft cases with a protruding premaxilla are a challenge to treat. The premaxilla in some cases might be prone to repeated trauma and could require an early setback and alignment with the lateral segments. Management of the fracture and reduction of the premaxillary segment dorsally have been described.

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.
Mossey PA, Little J, Munger RG, Dixon MJ, Shaw WC. Cleft lip and palate. Lancet 2009;374:1773-85.  Back to cited text no. 1
    
2.
Vanderas AP. Incidence of cleft lip, cleft palate, and cleft lip and palate among races: A review. Cleft Palate J 1987;24:216-25.  Back to cited text no. 2
    
3.
McComb H. Anatomy of the unilateral and bilateral cleft lip nose. In: Bardach J, Morris HL, editors. Multidisciplinary Management of Cleft Lip and Palate. 1st ed. Philadelphia: W.B. Saunders; 1990. p. 144-9.  Back to cited text no. 3
    
4.
Dahl E, Kreiborg S, Jensen BL. Roentgencephalometric studies of infants with untreated cleft lip and palate. In: Kriens O, editor. What Is a Cleft Lip and Palate? A Multidisciplinary Update. 1st ed. Stuttgart: Georg Thieme Verlag; 1989. p. 113-5.  Back to cited text no. 4
    
5.
Iizuka T, Thorén H, Annino DJ Jr., Hallikainen D, Lindqvist C. Midfacial fractures in pediatric patients. Frequency, characteristics, and causes. Arch Otolaryngol Head Neck Surg 1995;121:1366-71.  Back to cited text no. 5
    
6.
da Silva JY, Aranha AM, Peixoto V, Costa B, Gomide MR. Prevalence of oral trauma in children with bilateral clefts. Dent Traumatol 2005;21:9-13.  Back to cited text no. 6
    
7.
Haug RH, Foss J. Maxillofacial injuries in the pediatric patient. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:126-34.  Back to cited text no. 7
    
8.
Latham RA. Development and structure of the premaxillary deformity in bilateral cleft lip and palate. Br J Plast Surg 1973;26:1-1.  Back to cited text no. 8
    
9.
Wallace AF. The problem of the premaxilla in bilateral clefts. Br J Plast Surg 1963;16:32-6.  Back to cited text no. 9
    
10.
Shetty V, Vyas HJ, Sharma SM, Sailer HF. A comparison of results using nasoalveolar moulding in cleft infants treated within 1 month of life versus those treated after this period: Development of a new protocol. Int J Oral Maxillofac Surg 2012;41:28-36.  Back to cited text no. 10
    
11.
Papadopoulos MA, Koumpridou EN, Vakalis ML, Papageorgiou SN. Effectiveness of pre-surgical infant orthopedic treatment for cleft lip and palate patients: A systematic review and meta-analysis. Orthod Craniofac Res 2012;15:207-36.  Back to cited text no. 11
    
12.
Prahl C, Kuijpers-Jagtman AM, Van 't Hof MA, Prahl-Andersen B. A randomized prospective clinical trial of the effect of infant orthopedics in unilateral cleft lip and palate: Prevention of collapse of the alveolar segments (Dutchcleft). Cleft Palate Craniofac J 2003;40:337-42.  Back to cited text no. 12
    
13.
Grabowski R, Kopp H, Stahl F, Gundlach KK. Presurgical orthopaedic treatment of newborns with clefts – Functional treatment with long-term effects. J Craniomaxillofac Surg 2006;34 Suppl 2:34-44.  Back to cited text no. 13
    
14.
Padwa BL, Sonis A, Bagheri S, Mulliken JB. Children with repaired bilateral cleft lip/palate: Effect of age at premaxillary osteotomy on facial growth. Plast Reconstr Surg 1999;104:1261-9.  Back to cited text no. 14
    
15.
Bishara SE, Olin WH. Surgical repositioning of the premaxilla in complete bilateral cleft lip and palate. Angle Orthod 1972;42:139-47.  Back to cited text no. 15
    
16.
Heidbuchel KL, Kuijpers-Jagtman AM, Van't Hof MA, Kramer GJ, Prahl-Andersen B. Effects of early treatment on maxillary arch development in BCLP. A study on dental casts between 0 and 4 years of age. J Craniomaxillofac Surg 1998;26:140-7.  Back to cited text no. 16
    
17.
Posnick JC, Tompson B. Modification of the maxillary le fort I osteotomy in cleft-orthognathic surgery: The unilateral cleft lip and palate deformity. J Oral Maxillofac Surg 1992;50:666-75.  Back to cited text no. 17
    
18.
Carlini JL, Biron C, Gomes KU, Da Silva RM. Surgical repositioning of the premaxilla with bone graft in 50 bilateral cleft lip and palate patients. J Oral Maxillofac Surg 2009;67:760-6.  Back to cited text no. 18
    
19.
Batra P, Agrawal V, Kiran HJ, Madanagowda SB. Treatment of a patient with a bilateral cleft lip and palate with implants and surgery of the maxillary anterior region. World J Orthod 2010;11:380-6.  Back to cited text no. 19
    


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