In this case report, we would like to present an alternative surgical technique for the treatment of the bilateral prominent mandibular angles by using a custom-made resin splint (template). The prefabricated template provided a precise outline for the osteotomy, which alleviated the uncertainty of the osseous cut. Furthermore, masseter muscle myomectomy was not necessary in this case which decreased morbidity, and procedure and recovery time, while assuring a more symmetric outcome

Almeida, Luis Eduardo

Costa, Davani

Filho, Marco Antonio de Oliveira

Wang, Kingsley

English

epublications@Marquette

Marquette Universitye-Publications@MarquetteSchool of Dentistry Faculty Research andPublications Dentistry, School of7-1-2005Treatment of the Prominent Mandibular Angle byUsing a Custom Made Surgical SplintLuis Eduardo AlmeidaMarquette University, luis.almeida@marquette.eduKingsley WangThomas Jefferson UniversityDavani CostaHospital Angelina Caron, Curitiba, Parana, BrazilMarco Antonio de Oliveira FilhoHospital Evalgelico e CuritibaAccepted version. Journal of Oral and Maxillofacial Surgery, Vol. 63, No. 7 ( July 2005): 1052-1056.DOI. © 2005 Elsevier B.V. Used with permission.Luis Eduardo Almeida was affiliated with Hospital Evangelico de Curitiba, Curitiba, Parana, Brazil atthe time of publication.                                                                                 Marquette Universitye-Publications@MarquetteDentistry Faculty Research and Publications/School of DentistryThis paper is NOT THE PUBLISHED VERSION; but the author’s final, peer-reviewed manuscript.The published version may be accessed by following the link in the citation below.Journal of Oral and Maxillofacial Surgery, Vol. 60, No. 6 (2002): 681-684. DOI. This article is ©Elsevier (WB Saunders) and permission has been granted for this version to appear in e-Publications@Marquette. Elsevier (WB Saunders) does not grant permission for this article tobe further copied/distributed or hosted elsewhere without the express permission fromElsevier (WB Saunders).Mandible Cleft: Report of a Case and Review of the Literature*Luis Eduardo AlmeidaDepartment of Oral and Maxillofacial Deformities Center, Hospital Angelina Caron, BrazilMarquette University School of Dentistry, Milwaukee, WILucienne UlbrichDepartment of Oral and Maxillofacial Deformities Center, Hospital Angelina Caron, BrazilFabrício TogniDepartment of Oral and Maxillofacial Deformities Center, Hospital Angelina Caron, BrazilMedian cleft of the lower lip and associated structures is a relatively rare condition.1 We report the case of a patient with mandibular cleft. Unlike other reported cases of similar disorders, there was no cleft ofthe lower lip. The literature on median clefts of the lower lip and mandible is reviewed, and the etiologyand treatment are discussed.Report of a caseA 10-year-old girl was referred to our unit at Angelina Caron Hospital on July 27, 1999. She was the firstchild of healthy parents. The patients gave no history of former abnormalities in the family. The                                                      pregnancy was uneventful, and as far as can be ascertained, there was no exposure of the mother toteratogenic substances during the pregnancy.A general clinical examination revealed normal contour of the upper face and head (Figs 1, 2).Fig. 1. Frontal view of the patient.Fig. 2. Lateral view of the patient, showing the lack of chin prominence.The upper and lower lips were intact, but there was a midline cleft of the mandible, and the 2 halveswere freely movable. The morphology and size of the tongue were within normal limits, but the tip wasbound with soft tissue overlying the midline (ankyloglossia). The lower incisors were not present at the time of the examination. In addition, there was evidence of other skeletal deformities: larger spacesamong the fingers of the hands and the absence of chin prominence clinically and radiologically.When asked to occlude, the patient, at a first trial, could occlude the left side. Then, when asked toclench hard, she could occlude the right side (Figs 3, 4)Fig. 3. Free movement of the fragments.                                                   Fig. 4. Free movement of the fragments.Radiographs confirmed the median cleft of the mandible. The mandibular left cuspid was in an abnormal position (Figs 5, 6).Fig. 5. Panoramic radiograph showing the mandible cleft and the absence of the inferior incisors.Fig. 6. Mandible occlusal radiograph showing the mandible cleft.There also was agenesia of the lower incisors.Correction of the soft and hard tissues was performed at the same time. With the patient under general anesthesia, the tongue was released with a Z-plasty, and a frenectomy of the lingual frenulum wasperformed (Fig 7).Fig. 7. Ankyloglossia of the patient.After the frenectomy, an incision was made in the mandibular buccal sulcus between the cuspids, and amucoperiosteal flap was raised. At the time of surgery, the left cuspid was extracted (Fig 8).                                                                                       Fig. 8. After debridement, the 2 bone fragments could be observed independently (arrows).Concerning correction of the bony defect, an autogenous iliac crest bone graft of 3 cm was planned. Anocclusion as stable and functional as possible for the child's age was selected.The bony defect was bridged with the autogenous iliac crest bone graft as described earlier. Rigidfixation was performed with 2 plates and 8 screws (Fig 9).Fig. 9. Iliac crest graft positioned and fixed with plates and screws.No intermaxillary fixation was used.The postoperative course of the child was uneventful. After 6 months, the bone graft was well integrated with full union of the mandible. The tongue presents with normal movements. Occlusion isnormal.DiscussionThis case is unusual because the patient had no cleft of the lower lip associated with a cleft mandible. Discussion of the etiology and treatment refers to both defects, and such are reported in the international literature. Monroe2 reviewed the literature, and the first report he found was by Couronnein 1819.Developmental anomalies of structures derived from the upper half of the first branchial arch arecommon, giving rise to deformities such as cleft lip or cleft palate. However, abnormal or incompletedevelopment of structures derived from the lower half of the first branchial arch is rare. This maypresent as a complete3,4,5 or incomplete1,6,7,8,9,10 cleft of the lower lip, mandible,1,2,3,4,7,8,10,11,12 andtongue1, 2, 3, 4, 5, 8,10,12 with occasional associated deformities of soft tissue structures in the neck derivedfrom the lower branchial arch.12 Overall, there is a broad variation in the severity of this defect.11 The most common abnormalitiesrelated to this condition are congenital heart disease,2,3 absence of the hyoid bone,2 and neckcontracture.2,4,6                                                                                                                                                        Facial clefts could be in general part of syndromes. However, in the presented case, we could not identify a specific syndrome.Embryologically, the mandible develops from the cartilage of the first pharyngeal arch, the mandibular process, known as Meckel's cartilage. The mandible is derived from ossification of an osteogenicmembrane formed from ectomesenchymal condensation at 36 to 38 days of development. In themental region, on either side of the symphysis, 1 or 2 small cartilages appear, and endochondralossification commences in the seventh month in utero to form a number of mental ossicles. Theseossicles become incorporated into the intramembranous bone when the symphysis menti is convertedfrom a syndesmosis to a synostosis during the first postnatal year.10 However, several hypotheses concerning the pathogenesis of this deformity have been proposed in theliterature.Most authors presume that failure of fusion of the mandible is probably due to a lack of mesodermal penetration into the midline structures of the mandibular portion of the first branchial arch. This failuremay be a deficiency or delayed growth of the mesenchyme. Why this should occur so frequently inthe upper lip and so seldom in the lower is an unanswered question.2,6,9 Also, consideringthe developmental stageof the organs involved, it appears that the disturbing influence occurs at theend of the fifth or at the beginning of the sixth week of gestation.6 In 1935, Morton and Jordon thought that the failure of the mandibular processes to fuse may keep theventral ends of the succeeding arches from uniting in as much as fusion proceeds from above.8 This mayexplain the absence of the hyoid bone, thyroid cartilage, strap muscles, and manubrium in some of themore severe cases.In 1981, Parnes et al4 reported that the great variation in the severity of midline clefts of the lower lipand mandible and their association with other congenital defects, which are not necessarily related tobranchial arch defects, suggest that these deformities are not hereditary but instead are produced by 1or more localized influences that can cause the defect or defects. Thus, consideration should be given tothe hypothesis that these deformities are caused by external factors that can result in widespreaddisturbances in embryologic development.4 This theory, however, can be easily excluded because of thefact that localization of the clefting deformity of the mandible involves the midline in all casesdescribed.11 In 1995, however, Oostrom et al11 proposed a pathogenesis of clefts of the lip and mandible. Instead ofpaired branchial arches they reported that only 1 branchial arch develops during the early embryonicperiod, that is, the first 7 weeks of development, and that within this first branchial arch, 2 mandibular processes grow out, separated by a midline groove. Furthermore, they reported that the mandibularprocesses merge during the late embryonic period and that during the same period, the alveolar processand lip develop, together with the outgrowth of 1 membrane bone center in each mandibular process,resulting in the formation of the mandible with its symphysis.11 Therefore, hypoplasia of the mandibular process during the early embryonic period leads to the mostsevere cleft of the mandible, which extends into the neck, whereas during the late embryonic period,the less severe median clefts develop. Oostrom et al11 also proposed that disturbances of the outgrowthof bone centers of the mandible result in mandibular and soft tissue clefting, whereas defects in the                                                                                                                                                                                                      -merging process produce a notch of the vermilion or a higher cleft of the lower lip with or withoutinvolvement of the alveolar process of the mandible.Concerning treatment, the rarity and variation of severity of the condition are responsible for the lack ofa consensus on the nature and timing of the corrective surgery. The majority of the authors proposecorrection of the soft tissue structures as soon as possible, so as not to cause feeding and/or speakingproblems, and mandibular bone grafting in later stages of life. The timing of treatment of mandibular clefts by most surgeons when the child is about 8 to 10 years to avoid damaging developing toothbuds.1,2,6,7,8,10,12 However, Sherman and Goulian3 described the successful management of a completecleft of the lower lip and mandible in a 1-stage procedure in a 20-month-old child.In our patient, we corrected both deformities in a 1-stage procedure in consideration of the age and thestage of development of the patient.References1 CC Knowles, AHM Littlewood, PG Bush. Incomplete median cleft of the lower lip and chin with complete cleft of the mandible. Br Dent J, 127 (1969), p. 3372 CWMonroe. Midline cleft of the lower lip, mandible and tongue with flexion contracture of theneck: Case report and review of the literature. Plast Reconstr Surg, 38 (1966), p. 3123 JE Sherman, D Goulian. The successful one-stage surgical management of a midline cleft of the lowerlip, mandible and tongue. Plast Reconstr Surg, 66 (1980), p. 7564 EI Parnes, CN Galvão, Y Silva. Midline cleft of the lower lip and mandible: Report of a case. J OralSurg, 39 (1981), p. 9585 MM Chidzonga, JK Shija. Congenital median cleft of the lower lip, bifid tongue with ankyloglossia,cleft palate, and submental epidermoid cyst: Report of a case. J Oral MaxillofacSurg, 46 (1988), p. 8096 H Fujino, Y Kyoshoin, T Katsuki. Median cleft of the lower lip, mandible, and tongue with midlinecervical cord: A case report. Cleft Palate J, 7 (1970), p. 6797 DR Millard, SA Wolfe, S Berkowitz. Median cleft of the lower lip and mandible: Correction of themandibular defect. Br J Plast Surg, 32 (1979), p. 3458 DR Millard, JA Lehman, M Deane, et al.Median cleft of the lower lip and mandible: A case report. Br JPlast Surg, 24 (1971), p. 3919 R Ranta. Incomplete median cleft of the lower lip associated with cleft palate, the Pierre Robinanomaly or hypodontia. Int J Oral Surg, 13 (1984), p. 55510 AP Armstrong, N Waterhouse. Tessier 30 median mandibular cleft: Case report and literaturereview. Br J Plast Surg, 49 (1996), p. 53611CAM Oostrom, C Vermeij-Keers, PM Gilbert, et al.Median cleft of the lower lip and mandible. Casereports, a new embryologic hypothesis, and subdivision. Plast Reconstr Surg, 97 (1996), p. 31312 MM Chidzonga, VM Lopez Perez, S Mzezewa. Treatment of median cleft of the lower lip, mandible,and bifid tongue with ankyloglossia: A case report. Int J Oral Maxillofac Surg, 25 (1996), p. 272

Treatment of the Prominent Mandibular Angle by Using a Custom Made Surgical Splint

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Treatment of the Prominent Mandibular Angle by Using a Custom Made Surgical Splint

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