The Influence of Sidedness in Unilateral Cleft Lip and Palate on Mid Facial Growth at Five Years of Age.

OBJECTIVE: To determine whether facial growth at five years is different for children with a left versus right sided cleft lip and palate. DESIGN: Retrospective cohort study. SETTING: Seven UK regional cleft centres. PATIENTS: Patients born between 2000-2014 with a complete unilateral cleft lip and palate (UCLP). MAIN OUTCOMES MEASURE: 5-Year-Old's Index scores. RESULTS: 378 children were included. 256 (68%) had a left sided UCLP and 122 (32%) had a right sided UCLP. 5-Year-Old's index scores ranged from 1 (good) to 5 (poor). There was a higher proportion of patients getting good scores (1 and 2) in left UCLP (43%) compared to right UCLP (37%) but there was weak evidence for a difference (Adjusted summary odds ratio 1.27, 95% CI 0.87 to 1.87; P = .22). CONCLUSIONS: Whilst maxillary growth may be different for left versus right sided UCLP, definitive analysis requires older growth indices and arch forms.

Sagittal Growth Restriction of the Midface Following Isolated Cleft Lip Repair: A Systematic Review and Meta-Analysis.

Midface hypoplasia (MFH) is a long-term sequela of cleft lip and palate repair, and is poorly understood. No study has examined the aggregate data on sagittal growth restriction of the midface following repair of the lip, but not palate, in these patients.A systematic review of 3780 articles was performed. Twenty-four studies met inclusion criteria and 11 reported cephalometric measurements amenable to meta-analysis. Patients with Veau class I-III palatal clefts were included so long as they had undergone only lip repair. Groups were compared against both noncleft and unrepaired controls.Cephalometrics were reported for 326 patients (31.3% female). Noncleft controls had an average SNA angle of 81.25° ± 3.12°. The only patients demonstrating hypoplastic SNA angles were those with unilateral CLP with isolated lip repair (77.4° ± 4.22°). Patients with repaired CL had SNA angles similar to noncleft controls (81.4° ± 4.02°). Patients with unrepaired CLP and CL tended toward more protruding maxillae, with SNA angles of 83.3° ± 4.04° and 87.9° ± 3.11°, respectively. Notably, when comparing SNA angles between groups, patients with CLP with isolated lip repair had significantly more hypoplastic angles compared to those with repaired CL (P < .0001). Patients with CLP with isolated lip repair were also more hypoplastic than noncleft controls (P < .0001). In contrast, there was no significant difference between the SNA of patients with repaired CL and controls (P = .648).We found that cleft lip repair only appeared to contribute to MFH in the setting of concurrent cleft palate pathology, suggesting that scarring from lip repair itself is unlikely to be the predominant driver of MFH development. However, studies generally suffered from inadequate reporting of timing, technique, follow-up time, and cleft severity.

Sexual dimorphism in human midfacial growth patterns from newborn to 5 years old based on computed tomography.

Recent studies have supported the presence and varying nature of craniofacial sexual dimorphism (SD) from the very first stages of ontogeny. But the exact patterns of between-sex differences during the first years of life remain obscure despite the importance of these data for craniofacial surgery treatment and forensic studies. Our study employs a large dataset of clinical computed tomography scans of individuals of East Slavonic descent from birth to 5 years of age (247 males and 184 females) to address the pattern of age-related between-sex differences in 22 linear measurements of the mid-face. At birth, SD of most dimensions is low, but it increases significantly during the first year of life. The level of SD of most variables fluctuates in both directions during the second year and peaks during the third and fourth years of life. During the sixth year, SD of about half of the variables markedly decreases. In adults, SD of all variables increases, but to a very different extent: from 2% to 13%. Most sexually dimorphic features of the facial skeleton begin to develop early in postnatal ontogeny and then may or may not become accentuated during puberty. Importantly, the patterns of age changes in the level of SD differ strongly between various dimensions, and so cannot be expressed by a single value for the whole face. Additionally, the level of SD for a particular variable is not ontogenetically stable during the first years of life.

LeFort III/I for Beckwith-Wiedemann Syndrome: A Case Report.

This case presents a facially mature patient with Beckwith-Wiedemann Syndrome (BWS) who presented with severe class III malocclusion. Computed tomography imaging revealed an anterior crossbite of 19 mm and a narrow pharyngeal airway at the level of the tongue base precluding mandibular setback surgery. The patient was indicated for a LeFort III combined with a LeFort I advancement, each of 10 mm, for a 20 mm combined advancement. Stable, functional occlusion was achieved without airway compromise. This novel use of the combined LeFort III/I can restore stable class I occlusion in patients with BWS at risk for tongue base airway compromise.

Does the Transversus Abdominis Plane Block Reduce Morbidity Following Iliac Crest Bone Harvest in Pediatric Patients With Alveolar Clefts?

OBJECTIVE: The purpose of this study is to determine whether a Transversus Abdominis Plane (TAP) block can reduce donor site morbidity among pediatric patients undergoing iliac crest bone grafting for repair of their alveolar cleft. DESIGN: This retrospective cohort study was carried out at the Division of Dentistry at the Montreal Children's Hospital. Medical charts of patients who underwent alveolar cleft bone grafting between January 2011 and January 2021 were reviewed and they were divided into two groups, intraoperative TAP block and intraoperative local anesthesia infiltration (control group). The outcomes measured were patients' post-operative pain at the donor site, in-hospital narcotics requirements and length of stay. RESULTS: A total of 66 patients were included. There were no significant differences in pain scale among the TAP group and control group [1.9 (SD 2.5) and 1.3 (SD 2.1), respectively (p = 0.23)]. The mean length of stay for both groups was 1 day. Interestingly, there was a significant higher proportion of patients who required in-hospital opioids (morphine) in the TAP block group when compared to the control group (p = 0.03). CONCLUSIONS: The results of our study suggest there may be no role for a TAP block in reducing pain and improving opioid stewardship.

Early Secondary Alveolar Bone Grafting and Facial Growth of Patients with Complete Unilateral Cleft Lip and Palate.

OBJECTIVE: To investigate the craniofacial growth outcomes of early secondary alveolar bone grafting(ABG) around 6 years of age. DESIGN: Retrospective cohort study. SETTING: 1 North-American and 5 Northern-European cleft centers. SUBJECTS: 33 subjects with CUCLP consecutively treated with secondary ABG around 6 years of age were compared to 105 subjects from 4 centers treated with late secondary ABG and 19 subjects from 1 center with primary ABG. METHODS: Preorthodontic standardized lateral cephalometric radiographs taken after 12 years of age were traced and analyzed according to the Eurocleft Study protocol. Fourteen angular and two proportional measurements were performed. Measurement means from the Study Center(SC) were compared to 5 Northern-European centers using analysis of variance and Welch's modified t-tests, and P < .05 was considered statistically significant. RESULTS: For the SC, the mean age ± SD at the time of bone graft was 5.85 ± 0.71 years and the mean age at the time of the lateral cephalogram was 13.4 ± 1.8 years. The sagittal maxillary prominence of the SC was favorably comparable to the 5 Northern-European centers. The mean SNA (78.1 ± 4.3) for the SC was significantly higher compared to 4 of the 5 Northern-European centers(all P < .05), and the mean ANB angle was comparable to 4 of the 5 centers. Similarly, the mean soft tissue ANB angle was not significantly different to the 5 centers. The soft tissue vertical proportions compared favorably to all 5 Northern-European centers(all P < .01). CONCLUSIONS: Craniofacial growth outcomes of early secondary ABG around 6 years compare favorably to the outcomes of late secondary ABG.

Evaluation of Radiographic Outcomes of Alveolar Graft Associated with Premaxillary Osteotomy Performed with rhBMP-2.

OBJECTIVE: To evaluate the influence of filling material and timing of surgery on radiograph outcomes of alveolar grafting with premaxillary osteotomy. The null hypothesis was that radiographic outcomes would be similar with both rhBMP-2 (rhBMP-2G) and cancellous bone from the iliac crest (IG), regardless of the timing of surgery. DESIGN: Cross-sectional study with consecutive sampling of 56 periapical or occlusal radiographs taken 12 months after surgery. SETTING: A single tertiary craniofacial center. PATIENTS/PARTICIPANTS: Twenty-eight patients with complete bilateral cleft lip and palate and mean age of 13 years. The individuals underwent bilateral alveolar grafting associated with premaxillary osteotomy (AG + PO) with rhBMP-2 or cancellous bone from the iliac crest. INTERVENTIONS: Experienced maxillofacial surgeons used the same surgical technique in both groups. AG + PO were assigned as success or failure by 3 blinded raters based on modified Bergland and SWAG scales. MAIN OUTCOME MEASURES: The influence of filling materials and timing of surgery on radiographic outcomes was verified by Fisher's exact test and chi-square test (P < .05). RESULTS: There was no significance variation between the mean age of participants in the rhBMP-2G and IG (P = .471). Scales showed almost perfect reliability (agreement rate = 96.4%; K = 0.85). rhBMP-2G and IG had similar success rates with modified Bergland scale (85.7% and 82.1%) and SWAG scale (92.9% and 82.1%), respectively. However, only modified Bergland scale found influence of age on radiographic outcomes (P = .025). CONCLUSIONS: AG + PO performed with rhBMP-2 and iliac crest bone showed similar radiographic success rates, regardless of the timing of surgery.

Review of Diet Protocols Following Orthognathic Surgery and Analysis of Postoperative Weight Loss.

INTRODUCTION: Orthognathic surgery is routinely practiced, yet little comparative data exists to evaluate post-orthognathic surgery diet protocols. OBJECTIVE: To evaluate which postoperative diet protocols are recommended and to quantify post-orthognathic surgery weight changes in our institutional cohort. METHODS: An internet search was carried out on Google for "orthognathic surgery diet" and the postoperative diet recommendations from centers worldwide were quantified. Additionally, a retrospective analysis of patients that underwent orthognathic surgery at our institution was performed, and their preoperative and postoperative weights were recorded. RESULTS: The internet search yielded 58 centers that met our inclusion criteria. Most centers were in the United States (n = 37, 63.8%) and were oral and maxillofacial surgeon (OMFS)-led centers (n = 39, 67.2%). Postoperative diets were categorized into 7 distinct protocols, ranging from most to least restrictive-the most popular was liquid diet for 2 to 4 weeks followed by soft diet for 2 to 6 weeks. There were no significant patterns observed across different geographical regions or specialties.In our institution, 135 patients were identified. Overall, there was an average maximum weight loss of 4.1 kg by week 4, followed by a gradual increase in weight. Linear regression analysis showed that patients with greater preoperative body mass index (BMI) lost more weight postoperatively than patients with lower BMI (R2 = 0.25, P < .001). CONCLUSION: There is a significant variability in recommended postoperative diets following orthognathic surgery. Following a moderately restrictive diet at our institution, patients returned to their preoperative weight after approximately 4 months.

Does Early Secondary Alveolar Bone Grafting Influence Need for Additional Maxillary Advancement Procedures in Cleft Lip and Palate?

OBJECTIVE: To determine if secondary alveolar bone grafting (SABG) timing in patients with cleft lip and palate (CLP) influences the future need for additional maxillary advancement procedures, particularly Le Fort I osteotomy with rigid external distraction (RED). DESIGN: Retrospective cohort study. Groups were separated by SABG timing: early mixed dentition (ages 68 years) or late mixed dentition (ages 9-11 years). The criterion for RED was negative overjet ≥8 mm, and sufficient dental development for RED. SETTING: Single tertiary care institution. PATIENTS: Patients with CLP that underwent SABG from 2010 to 2015. Exclusion criteria included syndromic conditions, SABG surgery at age >12 years, current age <12 years, and <2 years follow-up. 104 patients were included. MAIN OUTCOME MEASURES: The number of RED candidates and treated patients. RESULTS: There was no statistical difference in the number of RED candidates (P = .0718) nor treated patients (P = .2716) based on SABG timing; stratification by laterality was also insignificant. Early SABG is associated with higher odds of being a RED candidate (pooled, unilateral, bilateral) and treated patient (pooled and unilateral); however, there were no statistically significant associations between SABG timing and the number of RED candidates and treated patients as determined by logistic regression models. CONCLUSION: There is no statistically significant association between SABG timing and the odds of being a RED candidate or treated patient. Future prospective studies are recommended to assess the relationship between SABG timing and maxillary growth in patients with CLP.

Interdisciplinary Approach for the Treatment of Complex Bilateral Cleft Lip and Palate With Missing Premaxilla.

Complex craniofacial deformities such as complete bilateral cleft lip and palate require interdisciplinary approach for proper diagnosis and treatment. A severe skeletal discrepancy caused by bilateral cleft lip and palate and missing premaxilla was successfully managed with orthodontic preparation and distraction osteogenesis. Conventional prosthodontic treatment combined with orthodontic preparation was proven to be a viable option to manage multiple missing teeth in cleft lip and palate.

Three-Dimensional Analysis of Bilateral Cleft Lip and Palate Nasal Deformity.

OBJECTIVE: This cross-sectional study utilizes 3-dimensional analysis to assess nasal morphology in patients with bilateral cleft lip and palate (BCLP) compared to controls across the timeline of cleft care. DESIGN: Retrospective comparative cross-sectional study. SETTING: Tertiary pediatric academic institution. PATIENTS AND PARTICIPANTS: One hundred and twelve patients with BCLP and an equal number of age and sex-matched control participants. MAIN OUTCOME MEASURE(S): Nasolabial angle, nasal length, nasal protrusion, columella length, columella width, nasal tip width, alar width, and alar base width were collected at each time point. The measurements were collected pre-nasoalveolar molding (NAM) therapy, post-NAM therapy, post-primary cleft rhinoplasty, 1 year, 5 years, 10 years, and 15 years of age. RESULTS: Nasolabial angle and nasal tip width were significantly different from controls from pre-NAM through 15 years of age time points. Nasal length was not significantly different at any time point. Alar width and alar base width were significantly different from pre-NAM through 10 years of age time points. Nasal protrusion, columella length, and columella width were significantly different from pre-NAM through 5 years of age time points. CONCLUSIONS: This study demonstrates that three-dimensional photogrammetry is effective in assessing the changes in nasal morphology that occur throughout the course of care in patients with BCLP from before cleft lip repair to the completion of nasal growth.

Treatment of Unilateral Cleft Lip and Palate Patient With Intraoral Tooth Tissue Borne Distractor and Facemask Therapy: A Case Report.

This case report demonstrates the successful use of intraoral distractor/hygenic rapid expander (HYRAX) for rapid maxillary expansion in anteroposterior direction with an adjunctive use of face mask therapy for anterior orthopedic traction of maxillary complex in a cleft patient with concave profile. The patient was a 13-year-old girl who reported with a chief complaint of backwardly positioned upper jaw and a severely forward positioned lower jaw. Therefore, a treatment was chosen in which acrylic bonded rapid maxillary expansion was done with tooth tissue borne intraoral distractor/HYRAX having a different activation schedule along with Dr Henri Petit facemask to treat maxillary retrognathism. As a result, crossbite got corrected and attained a positive jet with no bone loss in cleft area over a period of 5 months which was followed by fixed mechanotherapy achieving a well settled occlusion in 1 year. After completion of expansion and fixed mechanotherapy, ANB became +1 post-treatment which was -4 pretreatment. The prognathic profile was markedly improved by expansion and taking advantage of the remaining growth potential, thus minimizing the chances of surgery later in life. This provided a viable alternative to orthognathic surgery with good long-term stability.

The Effects of Nasoalveolar Molding on Nasal Proportions at the Time of Nasal Maturity.

BACKGROUND: The aim of this study is to assess the effect of nasoalveolar molding (NAM) versus no-NAM on nasal morphology in patients with unilateral cleft lip and palate (UCLP) at the time of nasal maturity. METHODS: A retrospective, single-institution review was conducted on all non-syndromic patients with UCLP. Inclusion criteria included age 14 years or above, unilateral cleft repair at the time of infancy, and adequate photography taken at nasal maturity and prior to rhinoplasty. Exclusion criteria included age less than 14 years, syndromic diagnosis, and rhinoplasty prior to nasal maturity. Ten parameters were measured twice from standardized clinical photographs using the Dolphin Imaging Software for establishment of intrarater reliability. Subjective analysis was achieved through completion of the Asher McDade grading scale by 3 expert cleft practitioners. RESULTS: Nostril height, columellar angle, alar cant, vertical alar height, alar height angle, nasofacial angle, and nasolabial angle were found to be significantly less severe in patients who had undergone NAM in conjunction with surgical repair when compared with those who had undergone surgical repair alone. Asher McDade grading revealed significant improvement in nasal form, nasal symmetry/deviation, nasal profile, vermillion border, and overall score in patients who underwent NAM compared to no-NAM. CONCLUSION: The use of presurgical NAM during infancy can improve nasal symmetry and nasal proportions at the time of nasal maturity.

Bilateral Craniofacial Cleft (Tessier Type 4): Case Report and Oculoplastic Reconstruction.

Bilateral Tessier type 4 craniofacial clefts are extremely rare and disfiguring malformations with vision-threatening ramifications. To date, there is no consensus in the literature with respect to the ideal surgical technique and management of these patients. Emergent eyelid reconstruction and additional procedures may be required to protect the cornea and avoid further ophthalmic and surgical complications. We present our experience and challenges of managing a case of bilateral Tessier type 4 clefting with an emphasis on oculoplastic considerations.

Circummaxillary Sutures in Patients With Apert, Crouzon, and Pfeiffer Syndromes Compared to Nonsyndromic Children: Growth, Orthodontic, and Surgical Implications.

OBJECTIVE: To evaluate patency of circummaxillary sutures in children with Apert, Crouzon, and Pfeiffer Syndromes and to compare it to a nonsyndromic matched control group. DESIGN: Case-control study. SETTING: Tertiary care public hospital. MATERIALS AND METHODS: Thirty-eight computed tomography (CT) scans of patients affected by syndromic craniofacial synostosis (13 patients with Apert syndrome, 20 patients with Crouzon syndrome, and 5 patients with Pfeiffer syndrome), average age 5 ± 2.8 years, range 1.9 to 12 years, were compared to age- and sex-matched control CTs of 38 nonsyndromic children. Computed tomography scans of the study group had to be performed prior to any midfacial surgery. MAIN OUTCOME MEASURES: Midpalatal suture, zygomaticomaxillary sutures, and pterigomaxillary sutures were evaluated and scored. RESULTS: The syndromic group showed a significant earlier ossification of all sutures compared to the nonsyndromic group. Significant differences were already present in early childhood and continued through adolescence. CONCLUSIONS: Based on the differences in terms of maxillary sutural ossification identified, midfacial hypoplasia does not seem to be only secondary to premature cranial base ossification, but also to primary synostosis of facial sutures, thus providing new insights into the pathogenesis of midface deficiency in children with craniofacial-synostosis. Care should be taken when planning any maxillary orthopedics, such as expansion or maxillary protraction, given the high frequency of early fusion of circummaxillary sutures.

NAM Therapy-Evidence-Based Results.

Many orthodontists working on patients with cleft lip and palate (CLP) have shown great enthusiasm for presurgical infant orthopedics (PSIO) to improve surgical outcomes with minimal intervention. Even though every clinician aims to use the best treatment modality for their patients, PSIO effects can be confounded by surgical type and timing of the primary repair, as is discussed in many studies. In such cases, one should be cautious when evaluating the particular outcomes for patients with CLP since it is difficult to differentiate the sole effect of an individual surgical or orthodontic intervention. As with any treatment methodology, nasoalveolar molding (NAM) has both benefits and limitations. Commonly cited concerns with NAM, and PSIO in general, include increased cost, increased burden of care, and a negative impact on maxillary growth. However, NAM cannot be deemed as having apparent long-term negative or positive effects on skeletal or soft tissue facial growth, based on previous studies. A review of the literature suggests that NAM does not alter skeletal facial growth when compared with the samples that did not receive PSIO. Nevertheless, the published studies on NAM show evidence of benefits to the patient, caregivers, the surgeon, and society. These benefits include documented reduction in severity of the cleft deformity prior to surgery and as a consequence improved surgical outcomes, reduced burden of care on the care givers, reduction in the need for revision surgery, and consequent reduced overall cost of care to the patient and society.

Facial Growth of Patients With Complete Unilateral Cleft Lip and Palate Treated With Alveolar Bone Grafting at 6 Years.

OBJECTIVE: To investigate the effect that alveolar bone grafting (ABG) around 6 years of age has on facial growth by assessing craniofacial growth outcomes. DESIGN: Retrospective cohort study. SETTING: North American cleft centers. PARTICIPANTS: A total of 33 children with complete unilateral cleft lip and palate who were consecutively treated with secondary ABG around 6 years of age were compared to 148 participants from 4 centers with late secondary ABG. METHODS: Preorthodontic standardized lateral cephalometric radiographs were analyzed and traced according to the Americleft Study protocol. Sixteen angular and 2 proportional measurements were performed. The outcomes of all ABG were assessed using the Standardized Way to Assess Graft scale. Measurement means from the study center (SC) were compared to 4 North American centers using analysis of variance and Welch modified t tests, and P < .05 was considered statistically significant. RESULTS: For the SC, the mean age (SD) at the time of bone graft was 5.85 (0.71) years and the mean age at the time of the lateral cephalogram was 13.4 (1.8) years. The sagittal maxillary prominence of the SC was comparable to the 4 other centers. The mean SNA (78.1 [4.3]) for the SC was significantly higher compared to one center that used primary bone grafting ( P = .03). The soft tissue mean ANB (3.52 [4.09]) for the SC was significantly lower compared to 3 of the centers. CONCLUSIONS: Early secondary ABG around 6 years of age did not result in reduced midface projection as assessed by SNA and thus did not compromise anterior maxillary growth.

Short- and Long-Term Effects of Late Maxillary Advancement With the Liou-Alt-RAMEC Protocol in Unilateral Cleft Lip and Palate.

OBJECTIVE: The objective of this retrospective longitudinal study was to evaluate short- and long-term results of the application of the Liou Alt-RAMEC (alternate rapid maxillary expansion and constriction) technique, a late orthopedic maxillary protraction technique, with intraoral anchorage, in patients with cleft. MATERIALS AND METHODS: Twenty-six patients with unilateral cleft lip and palate (UCLP) were consecutively treated with the Alt-RAMEC technique. The average age of the patients was 11.7 years (10.3-13.2 years) before protraction and 18.3 years (17.4-21.1 years) at long-term follow-up. A sample of nontreated patients with UCLP was used as a control group. It was matched for sex, skeletal class III, and age (11.3 years). The control sample had records at the end of growth (18.7 years). RESULTS: The sagittal advancement of A-point, after the application of the technique, was 5.7 (2.17) mm. Some mandibular dentoalveolar and positional adaptation was noted. The position of the maxilla was stable in the long term. On the other hand, the UCLP control group showed hardly any growth at the maxillary level during the long-term follow-up period. CONCLUSION: Our results showed that the Alt-RAMEC technique, performed at the correct time, with a double-hinged expander, followed by class III spring or elastic traction, 24 hours per day, allows for satisfactory maxillary protraction, with, at this stage, apparently stable long-term results. Nevertheless, as only 50% of the patients had long-term follow-up data, we are still unable to predict the percentage of patients which will not eventually need orthognathic surgery.

Cranio-Maxillofacial and Dental Findings in Albright's Hereditary Osteodystrophy and Pseudohypoparathyroidism.

INTRODUCTION: The clinical phenotype of pseudohypoparathyroidism (PHP) is caused by Albright's Hereditary Osteodystrophy (AHO). Often, "round face" the only facial clinical sign reported in the literature. The aim of this study was to highlight various cranio-maxillofacial clinical findings associated with AHO. RESULTS: Four patients presented with PHP type 1a. Only one patient exhibited the classical round face. All patients exhibited dental anomalies, class III malocclusion with maxillary retrusion, and a copper beaten appearance of the skull. One suffered from craniosynostosis. CONCLUSION: The frequency of craniofacial and dental features associated with malocclusion should prompt careful follow-up, particularly during facial growth, in patients with AHO.

A Programmable Expander for Patients With Cleft Lip and Palate.

This report describes a method to expand cleft segments of patients with cleft lip and palate prior to the secondary alveolar bone graft (ABG) using a reprogrammable nickel titanium expander. The expander can be reprogrammed with electric current to the desired width for each patient and delivers a gradual slow expansive spring force that expands and aligns the segments without further adjustments. Reprogramming the expander eliminates the need for multiple appliances and reduces chair time and patient appointments. The expander can be left in place during the ABG, because it does not interfere with a palatal flap. This alternative approach may be useful for patients who travel long distances to obtain cleft-related care.