Extreme maxillomandibular discrepancy in unilateral cleft lip and palate: Longitudinal follow-up in a patient with mandibular prognathism

Growth deficiency of the maxilla is a frequent finding in patients with complete unilateral cleft lip and palate. When the sagittal discrepancy is severe, orthodontic treatment combined with orthognathic surgery is required. This article reports the treatment of a girl born with unilateral cleft lip and palate who had lip and palate repair at 3 and 12 months of age, respectively. At 3 years of age, she already showed a severe anteroposterior maxillary deficiency with an anterior crossbite in the deciduous dentition. A Class III skeletal pattern progressively increased during the mixed dentition period. Mandibular prognathism coupled with an extremely hyperdivergent growth pattern was observed. An alveolar bone graft was performed at 10 years of age. At 16 years of age, the ANB angle was −13.7° with a negative overjet of −9.8 mm. Comprehensive orthodontic treatment was conducted with extraction of the mandibular first premolars and maxillary lateral incisors due to dental crowding. Orthognathic surgery was performed at 18.9 years of age involving maxillary advancement of 7.4 mm and mandibular setback of 6.6 mm. Facial and occlusal changes were dramatic. Final nose repair was conducted at 19.7 years of age. At 22 years of age and 3 years after debonding, stability of the occlusal and skeletal results was observed, clearly demonstrating that the objectives established for the rehabilitation have been achieved.

Highlights

  • A patient had severe maxillary deficiency associated with mandibular prognathism.

  • Occlusal development was monitored.

  • Treatment protocol for unilateral cleft lip and palate was followed.

  • Individualized treatment planning produced good esthetic results.

The rehabilitation process of patients with complete unilateral cleft lip and palate (UCLP) starts with the primary surgeries. Lip and palate repairs are usually performed at early ages. Upper lip tension and the scar of the primary surgeries are considered etiologic factors of gradual maxillary anteroposterior growth restriction. The severity of the maxillary deficiency varies among patients, depending on the initial cleft width, the technique and number of the primary surgeries, the age when the surgeries were performed, and the growth pattern. Maxillary growth also depends on surgical variations.

LeFort I osteotomy with maxillary advancement surgery is required in approximately 25% of patients with UCLP. The interarch relationship in the deciduous and mixed dentitions may provide an early prognosis for orthodontic treatment as well as the need for orthognathic surgery. Comprehensive orthodontic treatment has distinct objectives and is conducted in different ages, when orthognathic surgery or compensatory treatment is required. For this reason, longitudinal follow-ups of facial growth are important in patients with UCLP.

Orthognathic surgery in noncleft patients may demonstrate some relapse. In patients with cleft lip and palate, studies have shown that 5 to 6 mm of maxillary advancement with the LeFort I technique relapsed in 25% to 30% of patients. A greater amount of maxillary advancement was previously associated with instability.

The aim of this study was to describe the 3-phase orthodontic treatment of a girl with complete UCLP who developed an extreme anteroposterior maxilomandibular discrepancy caused by a severe maxillary deficiency coupled with mandibular prognathism. Her complete rehabilitation was conducted at 1 center in Brazil from 4 months to 22 years of age.

Diagnosis and etiology

The patient was brought at 28 days of age by her parents for an initial consultation at the Hospital for Rehabilitation of Craniofacial Anomalies, at the University of São Paulo. She had a complete left UCLP with no associated syndromes. Her rehabilitation process started with primary plastic surgeries. Lip repair was performed at 4 months of age using the Spina technique. Palate repair was performed at 10 months of age using the Furlow technique ( Fig 1 ). Because of a palatal fistula, 2 additional palatal repairs were performed at 17 months and 3 years of age ( Fig 2 ).

Fig 1
A, Facial frontal view before lip repair at 28 days of age; B, maxillary dental model taken after lip repair and before palate repair; C, nasolabial region after lip repair.

Fig 2
Facial and intraoral photographs in the deciduous dentition at 3 years of age.

At 8.3 years of age, the patient had her first orthodontic appointment. A skeletal Class III malocclusion with severe maxillary deficiency and a prognathic and hyperdivergent mandible were observed. In the frontal view, the face showed mild assymetry with the chin deviated to the left. The facial profile photograph demonstrated a marked midfacial deficiency, a tipped down nose, a retruded upper lip, and a strong chin. The nose also showed assymetry with flattening of the alar base on the cleft side. The sagittal interach relationship score was 5 according to the Goslon Yardstick. The patient was in the mixed dentition with a negative overjet, bilateral crossbite, and maxillary constriction ( Fig 3 ). At this stage, severe crowding was observed in the maxillary arch with the right permanent lateral incisor palatally displaced. The maxillary left permanent lateral incisor mesial to the alveolar cleft was absent, as well as the maxillary left second premolar. In the mandible, moderate dental crowding was observed. The maxillary dental midline was shifted to the right.

Fig 3
Facial and intraoral photographs in the mixed dentition at 8.3 years of age.

The maxilla was short and retruded, and the mandible was large and well positioned with a hyperdivergent skeletal pattern. The maxillary and mandibular incisors were lingually tipped. The skeletal and soft tissue profile was severely concave.

The etiology of the malocclusion was most probably atributed to both genetic and enviromental factors. The maxillary deficiency was caused by an enviromental factor from the primary plastic surgeries. The mandibular prognathism was probably determined by a genetic pattern.

Treatment objectives

Based on the initial orthodontic diagnosis, the objectives of treatment were to (1) correct the maxillary arch constriction; (2) rehabilitate the alveolar cleft with bone grafting; (3) level and align the maxillary and mandibular teeth in their basal bones, correcting the maxillary dental midline deviation; (4) coordinate the maxillary and mandibular dental arches; (5) correct the skeletal Class III relationship; (6) achieve adequate overjet and overbite; and (7) correct the asymmetry and improve the facial esthetics.

Treatment objectives

Based on the initial orthodontic diagnosis, the objectives of treatment were to (1) correct the maxillary arch constriction; (2) rehabilitate the alveolar cleft with bone grafting; (3) level and align the maxillary and mandibular teeth in their basal bones, correcting the maxillary dental midline deviation; (4) coordinate the maxillary and mandibular dental arches; (5) correct the skeletal Class III relationship; (6) achieve adequate overjet and overbite; and (7) correct the asymmetry and improve the facial esthetics.

Treatment alternatives

Treatment options were established in accordance with the objectives. The first option consisted of secondary alveolar bone grafting, followed by midface osteogenic distraction to promote maxillary advancement and comprehensive orthodontic treatment. The second option involved secondary alveolar bone grafting, followed by eruption and aligment of the permanent canine on the cleft side, comprehensive orthodontic treatment, and orthognathic surgery with maxillary advancement and mandibular setback. The patient and her parents chose the second option.

Treatment progress

Interceptive orthodontics started at 8.3 years of age to prepare the maxillary arch for secondary alveolar bone grafting. The maxillary right lateral incisor was extracted. Rapid maxillary expansion was performed with a Hyrax appliance and screw activation of approximately 7 mm. After a 6-month retention period, a fixed palatal arch was installed as a retainer. Secondary alveolar bone grafting was performed with autogenous bone from the iliac crest at 10 years of age.

After secondary alveolar bone grafting, orthodontic appointments with a 4-month interval were performed to follow the maxillary left permanent canine eruption path through the grafted area. Eruption of the left canine with a severe mesioangulation was observed after secondary alveolar bone grafting. At this stage, fixed appliances were bonded only in the maxillary arch for leveling and aligning of the maxillary teeth and to mesially move the left canine root to stimulate bone formation at the grafted area. Nickel-titanium and stainless steel round archwires were used. This second orthodontic intervention lasted 9 months. After debonding, a Hawley appliance was installed in the maxillary arch.

Comprehensive orthodontic treatment before the orthognathic surgery was postponed until the end of growth. Figure 4 shows the growth follow-up from 8 to 16 years of age. From 12 to 16 years of age, the patient returned for follow-up visits at the center once a year. At 16 years of age, the anteroposterior skeletal discrepancy increased, impairing facial esthetics ( Figs 5 and 6 ). The intraoral examination showed a severe bilateral Class III malocclusion and a mandibular tooth size-arch length discrepancy of −11.6 mm. Overjet and overbite were −9.8 and 1.8 mm, respectively. The maxillary dental midline was deviated 4 mm to the right. The mandibular dental midline was coincident with the center of the chin, and the ANB angle was −13.7° ( Table ).

Fig 4
Superimposition of 8-year and pretreatment cephalometric tracings.

Fig 5
Facial and intraoral photographs before comprehensive orthodontic treatment at 16 years of age.

Fig 6
Pretreatment lateral headfilm and panoramic radiograph.

Dec 10, 2018 | Posted by in Orthodontics | Comments Off on Extreme maxillomandibular discrepancy in unilateral cleft lip and palate: Longitudinal follow-up in a patient with mandibular prognathism
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