Ectopic eruption of the second premolar: an analysis of four different treatment approaches.

PURPOSE: Ectopic second premolars may lead to impaction and loss of space in the jaws, and in rare cases even to resorption of the first permanent molar. The aim of this study was to analyse different treatment strategies of ectopic second premolars and if possible give guidelines on when to favour different treatment approaches. MATERIALS AND METHODS: The study was a retrospective, non-randomised, outcome analysis of treatment on 41 ectopic second premolars in 37 patients (24 females and 13 males). In all cases oral examination, radiographs (pre-, peri-, and post) and full medical history were obtained. The treatment options included: (a) spontaneous eruption, (b) spontaneous eruption + extraction of primary tooth, (c) surgical exposure, (d) surgical uprighting, and (e) surgical uprighting + orthodontic extrusion. For evaluation each tooth was scored according to: (1) stage of root development, (2) distance between edges of the premolar and first permanent molar, (3) depth of impaction, (4) inclination, (5) horizontal position of the tooth. The level of significance was set to 5%. RESULTS: Only mild cases of ectopic second premolars are self-correcting. Based on the position of the tooth in the jaw different treatment options may be chosen, these may include: extraction of primary predecessor (impaction depth < 5 mm, inclination < 55°), surgical exposure of tooth germ (impaction depth < 5.5 mm, inclination < 95°) or surgical uprighting (impaction depth > 5.5 mm with no inclination limit). CONCLUSION: If there is no sign of self-correction after a short observation period, it is important to consider active treatment to help guiding the tooth into the correct eruption pathway.

Primary failure of eruption of teeth in two siblings with a novel mutation in the PTH1R gene.

BACKGROUND: Primary failure of tooth eruption (PFE) is a rare non-syndromic disorder with prematurely ceased eruption of the posterior teeth, despite clearance by bone resorption of the eruption path. It is generally agreed that most of the impacted teeth are second molars that are deeply seated, and without symptoms. Traditionally, patients with failure of tooth eruption undergo surgical and/or orthodontic treatment. However, patients with PTH1R mutations have no beneficial effect of such a regime and PFE is therefore important to diagnose. CASE REPORT AND FOLLOW-UP: A family with three PFE affected members in two generations, involving both the primary and permanent dentitions, and a novel mutation in the PTH1R gene are reported. Furthermore, the treatment of the eruption failure was documented in one of the cases. CONCLUSION: In the present study, the proband initially only had a minor clinical problem, lack of eruption of the primary second left mandibular molar. However, over time several problems appeared in the permanent dentition. Clinical signs of PFE should lead one to look for similar dental problems in related family members and to molecular DNA testing. Confirmation of the diagnosis PFE in young children has the advantage that unnecessary treatment can be avoided, since early orthodontic intervention for these patients is futile. Once growth is complete, several multidisciplinary treatment strategies can partially solve the posterior open bite malocclusion that is characteristic of this disorder. Treatment should be planned in cooperation with specialists who are used to treating PFE patients.

Early post-natal development of the mandibular permanent first molar in infants with unilateral complete cleft lip and palate.

OBJECTIVES: Studies have shown that the mandibular permanent first molar (M1inf ) in young children with isolated cleft palate is characterized by delay in maturation and has reduced crown width. Consequently, it is of interest to investigate the early maturation and width of the follicle and crown of M1inf in children with combined cleft lip and palate. DESIGN: Retrospective, longitudinal study. Cephalometric X-rays of 47 consecutive Danish children with UCCLP (37 males; 10 females) and 44 with unilateral incomplete cleft lip (UICL) (29 males; 15 females) examined at 2 and 22 months of age. UICL served as control group. Maturation (according to Haavikko), width of follicle (FW) and crown (CW) of M1inf were assessed. RESULTS: The maturation of the first mandibular molar was delayed in both genders at 2 and 22 months of age. FW and CW were smaller in children with UCCLP at both 2 and 22 months of age. There was a positive correlation between maturation and FW. CONCLUSIONS: Maturation of the first mandibular molar is delayed in both genders, and FW and CW were reduced in UCCLP compared to controls. Maturation was correlated with FW.

A Pilot Study on the Influence of Facial Expression on Measurements in Three-Dimensional Digital Surfaces of the Face in Infants With Cleft Lip and Palate.

OBJECTIVE: Three-dimensional surface imaging is an increasingly popular modality for face measurements in infants with cleft lip and palate. Infants are noncompliant toward producing specific facial expressions, and selecting the appropriate moment of acquisition is challenging. The objective was to estimate amount and spatial distribution of deformation of the face due to facial expression in infants with cleft lip and palate and provide recommendations for an improved acquisition protocol, including a method of quality control in terms of obtaining images with true neutral expression. MATERIAL AND METHODS: Three-dimensional surface images of ten 4-month-old infants with unrepaired cleft lip and palate were obtained using a 3dMDface stereophotogrammetric system. For each subject, five surface images judged as representing a neutral expression were obtained during the same photo session. Mean and maximum deformations were calculated. A formalized review was performed, allowing the image exhibiting the "best" neutral expression to be selected, thus decreasing errors due to residual facial expression. RESULTS: Deformation due to facial expression generally increased from forehead to chin. The amount of deformation in three selected regions were determined: nose (mean, 1 mm; maximum = 3 mm); cleft region (mean, 2 mm; maximum = 5 mm); chin region (mean, 5 mm; maximum = 12 mm). Analysis indicated that introduction of a formalized review of images could reduce these errors by a factor of 2. CONCLUSIONS: The continuous change of facial expression in infants represents a substantial source of error; however, this may be reduced by incorporating a formalized review into the acquisition protocol.

Maxillary length in 11- to 26-week-old normal fetuses studied by 3D ultrasound.

OBJECTIVES: The objective of this article is to investigate normal prenatal maxillary length using 3D ultrasound and to correlate this with previously reported results for the mandible and the biparietal diameter (BPD). METHODS: Seventy-two 3D ultrasound volumes from normal pregnancies in 52 volunteers (gestational age: 11-26 weeks) were obtained using a GE Voluson 730 Expert 3D scanner. Maxillary length and BPD were measured. Growth velocity and rate were calculated. Maxillary values were correlated with BPD and previously reported mandibular values. RESULTS: The mean total maxillary length ranged from 7.4 ± 2.7 mm at 11 weeks to 33.1 ± 2.7 mm at 26 weeks. Maxillary growth velocity can be described using a linear model. The correlation between maxillary and mandibular length, and BPD was high (R = 0.85-0.88). There was no statistically significant difference in the growth velocity between maxilla and mandible, whereas the velocity of the increase in BPD growth was significantly larger than that of the jaws. However, the growth rate was larger for the jaws than for the BPD. CONCLUSIONS: Normative measures for the maxilla in 11- to 26-week-old fetuses are presented. Change in maxillary length was described using a linear model. The maxillary and mandibular growth velocities and rates were similar but differ from those of the BPD.

Short mandible - a possible risk factor for cleft palate with/without a cleft lip.

OBJECTIVES: To estimate the influence of a short mandible on the risk of developing a cleft palate with/without a cleft lip (CP). SETTING AND SAMPLE POPULATION: The retrospective sample consisted of 115 2-month-old Danish infants with CP, and 70 control infants with unilateral incomplete cleft lip (UICL). MATERIAL AND METHODS: Cephalometric X-rays were obtained. Mandibular length (Lm ) was measured and corrected for body length (Lb ) to remove influence of varying body length in the sample. Logistic regression was applied to the corrected mandibular length (Lmc ) to calculate the risk of having a cleft palate. RESULTS: The mean mandibular length in the group with CP was about 4 mm shorter than in the control group. Odds ratio (OR) was calculated to be 0.58 (95% confidence interval 0.48-0.68), implying that an individual's risk of cleft palate with/without a cleft lip increases about 50% per mm decrease in mandibular length. CONCLUSIONS: A special facial type including a short mandible is a possible risk factor for cleft palate, and it was found that the risk of cleft palate increases 58% per mm decreases in mandibular length.

Prenatal 3D ultrasound diagnostics in cleidocranial dysplasia.

A 34-year-old Caucasian woman with cleidocranial dysplasia (CCD) and a known family history of CCD was referred for an ultrasound examination in the first trimester of her second pregnancy. Molecular genetic analysis of the RUNX2 gene was non-informative. A routine 2D ultrasound examination carried out at a local hospital at gestational age 12 weeks showed no signs of CCD. A 3D ultrasound examination in week 15+4 showed a fetus with typical CCD features including large fontanelles, lack of nasal bones, clavicles without the typical S-form, as well as severe delay in calvarial ossification, especially in the midline. Serial 3D ultrasound examinations during pregnancy confirmed the diagnosis, and over time the manifestations became even more distinct. The diagnosis was clinically confirmed at birth. This case suggests that the typical craniofacial CCD traits, including wide unmineralized areas in the calvarial midline and missing nasal bones, are easily recognizable using 3D ultrasound as early as in week 15.

An echo-poor spine at 13 weeks: an early sign of cleidocranial dysplasia.

OBJECTIVE: Early prenatal diagnosis of cleidocranial dysplasia (CCD) in a case in which molecular genetic analysis of the RUNX2 gene was non-informative. METHODS: 2D ultrasound examination. RESULTS: At week 13+6, a 2D ultrasound examination revealed a fetus with severely delayed ossification of the vertebral spine. The clavicles were barely seen and the calvarial bones were significantly less ossified than expected for gestational age. The fetus had otherwise normal anatomy and biometry. Serial ultrasound examinations during pregnancy confirmed the diagnosis, but the manifestations became less distinct. The diagnosis was confirmed clinically at birth. CONCLUSION: This case illustrates an early easily recognizable pattern of severely delayed ossification of the vertebral spine, which is probably a characteristic of CCD.

Early postnatal development of the mandible in children with isolated cleft palate and children with nonsyndromic Robin sequence.

OBJECTIVE: Analysis of early postnatal mandibular size and growth velocity in children with untreated isolated cleft palate (ICP), nonsyndromic Robin sequence (RS), and a control group of children with unilateral incomplete cleft lip (UICL). MATERIAL: 114 children (66 isolated cleft palate, 7 Robin sequence, 41 unilateral incomplete cleft lip) drawn from a group representing all Danish cleft children born from 1976 through 1981. All children were examined at both 2 and 22 months of age. METHODS: Cephalometric x-rays and maxillary plaster casts. Mandibular length and height were measured and mandibular growth velocity (mm/year) was calculated. Cleft width was measured on the casts at 2 months of age. RESULTS: Mean mandibular length and posterior height were significantly smaller in isolated cleft palate and Robin sequence, compared with unilateral incomplete cleft lip. Mandibular length in Robin sequence was also significantly shorter, compared with isolated cleft palate. No significant difference was found between mean mandibular growth velocities in the three groups. No significant correlation was found between mandibular length and cleft width in either isolated cleft palate or Robin sequence at 2 months of age. CONCLUSION: The children with isolated cleft palate and Robin sequence had small mandibles shortly after birth, but with a relatively normal growth potential. No true mandibular catch-up growth was found up to 22 months of age in either group. No significant correlation was found between mandibular size and cleft width in either group at 2 months of age. However, there was a significant trend toward the shorter the mandible, the more severe the sagittal extension of the cleft.

Early craniofacial morphology and growth in children with bilateral complete cleft lip and palate.

PURPOSE: Analysis of craniofacial morphology and growth in children with bilateral complete cleft lip and palate (BCCLP), compared with a control group with unilateral incomplete cleft lip (UICL), before any treatment as well as 20 months after lip closure. MATERIAL: The children were drawn from a group representing all Danish children with cleft born 1976 to 1981. Sixty-four children were included in the study (19 BCCLP and 45 UICL). The ages were 2 and 22 months at examinations 1 and 2, respectively. METHOD: The method of investigation was infant cephalometry in three projections. The craniofacial morphology was analyzed using linear, angular, and area variables. Growth was defined as the displacement vector from the coordinate of the corresponding landmark in the x-ray at examination 1 to its coordinate at examination 2, corrected for x-ray magnification. The growth of an anatomical region in a patient was assessed by investigating the growth pattern formed by a collection of individual growth vectors in that region. RESULTS: The BCCLP group differed significantly from the UICL group. The most striking findings in BCCLP were an extremely protruding premaxilla; markedly increased posterior maxillary width; increased width of the nasal cavity; short maxilla with reduced posterior height; short mandible; bimaxillary retrognathia; severe reduction in the size of the pharyngeal airway; and a more vertical facial growth pattern. CONCLUSION: Our findings indicate that a facial type including a wide and posterior short maxilla, short mandible, and bimaxillary retrognathia might be a liability factor that increases the probability of developing cleft lip and palate.

Craniofacial morphology and growth comparisons in children with Robin Sequence, isolated cleft palate, and unilateral complete cleft lip and palate.

OBJECTIVE: Comparison of early craniofacial morphology and growth in children with nonsyndromic Robin Sequence (RS), isolated cleft palate (ICP), and unilateral complete cleft lip and palate (UCCLP). SUBJECTS: One hundred eight children with cleft: 7 with RS, 53 with ICP, and 48 with UCCLP were included in the study. The children were drawn from the group of all Danish children with cleft born 1976 through 1981. METHOD: Three-projection infant cephalometry. RESULTS: The craniofacial morphology in the RS, ICP, and UCCLP groups had some common characteristics: a wide maxilla with decreased length and posterior height, wide nasal cavity, short mandible, bimaxillary retrognathia, and reduced pharyngeal airway. The shortest mandible was found in RS followed by ICP and UCCLP; the pharyngeal airway was reduced in RS and ICP, compared with UCCLP; and the maxillary complex and nasal cavity were wider in UCCLP than in the other groups. The amount of facial growth in all three groups was similar; however, the direction was more vertical in UCCLP than in RS and ICP. CONCLUSION: Except for a shorter RS mandible, the facial morphology of infants with RS and ICP was similar, as was the amount of facial growth and the growth pattern. The differences in facial morphology can be ascribed to the difference in the primary anomaly. The amount of facial growth was similar in the three groups; however, the growth pattern showed a more vertical direction in UCCLP than in RS and ICP. It is hypothesized that the mandibular retrognathia in RS represents the outer end of that of the ICP distribution.

Early craniofacial morphology and growth in children with nonsyndromic Robin Sequence.

PURPOSE: Craniofacial morphology and growth comparisons in children with untreated nonsyndromic Robin Sequence (RS) and a control group with unilateral incomplete cleft lip (UICL) in which the lip was surgically closed at 2 months of age. MATERIAL: The 52 children (7 RS and 45 UICL) included in the study were drawn from a group representing all Danish cleft children born 1976 through 1981. The ages of the children were 2 and 22 months at the time of examination 1 and 2, respectively. METHOD: The method of investigation was three-projection cephalometry. Craniofacial morphology was analyzed by means of linear, angular, and area variables. Growth at a specific anatomical location in a patient was defined as the displacement vector from the coordinate of the corresponding landmark at examination 1 to its coordinate at examination 2. RESULTS: The most striking findings in the RS group were markedly increased posterior maxillary width, increased width of the nasal cavity, short maxilla with reduced posterior height, short mandible, bimaxillary retrognathia, and severe reduction in size of the pharyngeal airway. The amount of facial growth was similar in the two groups; however, a tendency toward a more vertical growth direction was observed in the RS group. CONCLUSION: Facial morphology in children with RS differed significantly from that of children with UICL at both 2 and 22 months of age. The magnitude of facial growth was similar in the two groups, whereas a tendency toward a more vertical facial growth direction was observed in the RS group.

Early craniofacial morphology and growth in children with unoperated isolated cleft palate.

OBJECTIVE: Analysis of craniofacial morphology and growth in children with untreated isolated cleft palate (ICP) (cleft of the secondary palate only) at 2 and 22 months of age and comparison of the morphology and growth to that of a control group with unilateral incomplete cleft lip (UICL). MATERIAL AND METHODS: A total of 98 cleft children (53 with ICP and 45 with UICL) drawn from a larger group representing all Danish children with cleft born in the period 1976 to 1981 were included in the study. Craniofacial morphology and growth were analyzed using three-projection infant cephalometry. RESULTS: The ICP group differed significantly from the UICL group. The most striking findings in the ICP group were: short maxilla; reduced posterior maxillary height; increased posterior maxillary width (in the 2-month-old); short mandible; reduced posterior height of the mandible; bimaxillary retrognathia; and reduced pharyngeal depth, height, and area. The facial growth pattern was fairly similar in the two groups except for a somewhat more vertical growth direction in the ICP group. CONCLUSION: The facial morphology in ICP children differs significantly from that of children with UICL of the same age. The differences in facial morphology can be ascribed to the difference in the primary anomaly in the ICP group. The facial growth pattern was fairly similar in the ICP and UICL group; however, a somewhat more vertical growth direction was observed in the ICP group.

A method for three-projection infant cephalometry.

OBJECTIVE: To assess morphology and growth in infants and children with craniofacial anomalies based on comprehensive digitization of radiographic films in three, mutually orthogonal projections. METHOD: The method consists of (1) acquisition of radiographic films in a highly standardized three-projection (lateral, frontal, and axial) cephalometer, (2) marking and digitization of a total of 279 anatomical landmarks in the three projections, and (3) computation and presentation (tabular and graphical) of 356 linear and angular variables describing the craniofacial morphology, including soft tissue. Computation of statistical entities describing a patient, a group of patients, the differences between patients or groups of patients was carried out. Error assessment of the method involved investigation of error distribution among a number of error sources. Duplicate digitization of radiographic films from 30 randomly selected patients, and from 10 dry skulls, was carried out to determine the errors contributed by the procedure of landmark digitization and the distribution of error among landmarks and variables, as well as between projections. RESULTS: The average error due to landmark digitization, s(i), determined by duplicate digitization and calculated by use of Dahlberg's formula was 0.8 mm for linear variables and 1.6 degrees for angular variables. CONCLUSION: This method of infant cephalometry has been shown to be highly accurate and reproducible, and it adds significant new potential for, e.g., asymmetry detection, population comparison, and growth measurements compared to other cephalometric techniques due to its standardized acquisition and digitization protocol, inclusion of an axial projection, and the large number of well-defined landmarks and variables involved.

Craniofacial comparisons in 22-month-old lip-operated children with unilateral complete cleft lip and palate and unilateral incomplete cleft lip.

OBJECTIVE: The objective of the study was to analyze the craniofacial morphology in infants with unilateral complete cleft lip and palate (UCCLP) in which the lip and the anterior part of the palate had been surgically closed at 2 months of age and to compare the morphology with that of a control group with unilateral incomplete cleft lip (UICL) in which the lip had also been surgically closed at 2 months of age. DESIGN: The sample consisted of a total of 108 cleft children all fulfilling the entry criteria, besides diagnosis, as follows: The child was of Danish origin; the age of the child was between 650 and 750 days (approximately 22 months) at the time of examination; the child was healthy except for its single cleft malformation; the surgical procedure in each group had been performed at about 2 months of age by the same surgeon. The surgical methods used were a Tennison procedure (UICL group) and a Tennison procedure supplemented by palatovomer plasty (UCCLP group). METHODS: The method of investigation was infant cephalometry in the lateral, frontal, and axial projections. Linear, angular, and area variables describing the craniofacial morphology were calculated and supplemented by mean plots from the cephalometric projections in the two groups. RESULTS AND CONCLUSIONS: Statistical analysis based on Student's t test showed that the facial morphology in the 22-month-old UCCLP group differed significantly from that of the UICL group. The most pronounced differences were found in the maxillary complex and the mandible. The deviations observed in the UCCLP group at 22 months of age were similar to those previously observed at 2 months of age. However, several of the dysmorphic traits had become less pronounced; some had remained the same; and a few had become worse with time.

Craniofacial growth in subjects with unilateral complete cleft lip and palate, and unilateral incomplete cleft lip, from 2 to 22 months of age.

This paper reports a longitudinal quantitative cephalometric analysis of the craniofacial growth in subjects with unilateral complete cleft lip and palate (UCCLP), and unilateral incomplete cleft lip (UICL), from 2 to 22 months of age. The purpose of the study was to determine the amount and direction of growth in UCCLP compared to UICL (control group) from 2 months of age (just prior to lip repair) to 22 months of age, 20 months later. The sample comprised of 49 subjects with UCCLP (37 males and 11 females) and 45 with UICL (29 males and 16 females). The cephalometric analysis of the craniofacial morphology included lateral, frontal, and axial projections. The data were presented as mean plots of the craniofacial region including the calvaria, cranial base, orbits, nasal bone, maxilla, mandible, cervical column, pharynx, and soft-tissue profile. A valid common coordinate system (registration according to the n-s line in the lateral projection, latero-orbitale line in the frontal projection, and meatus acusticus externus line in the axial projection for the landmark positions at examination 1 and 2) was ascertained. The growth at a specific anatomical location in a patient was defined as the displacement vector from the coordinate of the corresponding landmark in the X-ray at examination 1 to its coordinate at examination 2, corrected for X-ray magnification. The growth of an anatomical region in a patient was assessed by investigating the growth pattern formed by a collection of individual growth vectors in that region. The amount of growth in the UCCLP and UICL group was very similar. The general craniofacial growth pattern, in terms of the direction of growth, was also fairly similar in the UCCLP group and the control group. However, the maxilla and mandible showed a more vertical growth pattern than that observed in the control group. This study confirms that UCCLP is a localized deviation, and not a craniofacial anomaly, due to the fact that a normal growth potential has been observed in all craniofacial regions, except where the growth had been directly influenced by surgical intervention. Furthermore, the vertical growth pattern of the maxilla and mandible supports the hypothesis of a special facial type in cleft lip and palate individuals, and the facial type as a liability factor increasing the probability of cleft lip and palate.

A comparison of the craniofacial morphology in 2-month-old unoperated infants with unilateral complete cleft lip and palate, and unilateral incomplete cleft lip.

This paper reports a cephalometric analysis of the craniofacial morphology in infants with unoperated unilateral complete cleft lip and palate (UCCLP) and unoperated unilateral incomplete cleft lip (UICL). The purpose of the study was to determine the nature and extent of the craniofacial deviations in UCCLP as compared to the morphology in UICL, which has previously been shown to be close to normal. The samples comprised 82 infants with UCCLP (58 males and 24 females) and 75 with UICL (48 males and 27 females). The mean age was about 2 months in both groups. The cephalometric analysis of craniofacial morphology included the lateral, frontal, and axial projections. The data were presented as mean plots of the craniofacial region including the calvaria, cranial base, orbits, nasal bone, maxilla, mandible, cervical column, pharynx, and soft-tissue profile. The most pronounced deviations in the UCCLP group were observed in the maxillary complex and the mandible. The most striking findings were: markedly increased width of the maxilla, a short mandible, and bimaxillary retrognathia except for the premaxillary area, which was relatively protruding and asymmetric. The study did not support the hypothesis previously suggested in the literature that cleft lip and palate is a craniofacial anomaly as size and shape of the calvaria and cranial base were found to be normal. The etiology of cleft lip and palate is still incompletely understood. Based on the present study, we suggest that facial type may be a liability factor that could represent a developmental threshold increasing the probability of cleft lip and palate.