|Year : 2018 | Volume
| Issue : 1 | Page : 1-5
Primary failure of eruption, A review
Uma B Dixit, Mahak C Jain
Department of Pedodontics and Preventive Dentistry, D. Y. Patil Deemed to be University, School of Dentistry, Navi Mumbai, Maharashtra, India
|Date of Web Publication||19-Nov-2018|
Dr. Uma B Dixit
Department of Pedodontics and Preventive Dentistry, D. Y. Patil Deemed to be University, School of Dentistry, Nerul, Navi Mumbai, Maharashtra
Source of Support: None, Conflict of Interest: None
Primary failure of tooth eruption (PFE) is defined as a non-syndromic condition in which non-ankylosed teeth fail to erupt fully or partially due to malfunction of the eruption mechanism. Although the eruption path is clear without any mechanical obstruction like cyst or supernumerary tooth, affected teeth fail to erupt. Patients diagnosed with PFE have revealed mutation in the gene encoding the G protein of parathyroid hormone 1 receptor (PTH1R). The condition is characterized by progressive posterior open bite due to involvement of all the teeth distal to the first affected tooth. These teeth fail to respond to orthodontic forces and attempts to correct open bite orthodontically cause the affected teeth to ankylose, thus worsening the open bite. Diagnosis of PFE is critical as management differs from the infra-occluded teeth due to mechanical obstruction or systemic etiology. This review attempts to discuss etiology, clinical features, diagnosis and management of the teeth affected with primary failure of tooth eruption.
Keywords: Delayed eruption, eruption disorder of teeth, posterior open bite, primary failure of eruption, parathyroid hormone 1 receptor gene
|How to cite this article:|
Dixit UB, Jain MC. Primary failure of eruption, A review. Indian J Oral Health Res 2018;4:1-5
| Introduction|| |
Tooth eruption is defined as the movement of a tooth from its site of development within the alveolar process to its functional position in the oral cavity. Tooth eruption is an intricate and complex process which involves coordinated signaling events between the dental follicle, osteoblasts, and osteoclasts. Disruption of this process may lead to delayed eruption or complete failure of eruption.
Failure of eruption can be classified as follows:
- Impaction – cessation in the eruption of the tooth which is caused by clinically or radiographically detectable physiologic barrier in the eruption path or due to abnormal positioning of the tooth. However, impaction is a very rare phenomenon in primary dentition. Prevalence of impaction of primary teeth has been reported to be 1:10,000. The most commonly seen primary impacted tooth is the second molar.
- Primary retention – cessation in the eruption of a normally placed and a normally developed tooth before gingival emergence without a physical barrier in the eruption path. The eruption of tooth in such cases is delayed for more than 2 years and there is no specified local or systemic factor. A disturbance in the metabolic event of the dental follicle causes primary retention or primary failure of eruption (PFE) as bone resorption is not initiated.
- Secondary retention – cessation of the eruption after emergence without the presence of a physical barrier or ectopic position of tooth. This occurs after tooth has penetrated the oral mucosa. Earlier, Raghoebar et al. claimed trauma, infection, disturbed local metabolism, and genetic factors to be responsible for secondary retention. Further studies conducted by Raghoebar et al. demonstrated histologic evidence of ankylosis in these teeth, mostly seen in the bifurcation and interradicular surfaces., The proposed mechanism was the replacement of cementoblasts by osteoblasts due to a local disturbance in the periodontal ligament during the repair process of local physiologic resorption.
Various systemic factors including syndromes are associated with delayed or failure of tooth eruption:
- Cleidocranial dysplasia and Gardener's syndrome are the most common syndromes that are associated with delayed or failed tooth eruption. In these syndromes, the eruption disturbances are thought to occur because of physical interference by multiple supernumerary teeth and reduced eruptive potential
- Patients with ectodermal dysplasia can have hypodontia or anodontia with delayed eruption of permanent teeth
- In Down syndrome, tooth eruption is delayed and can occur 2–3 years later than in normal dentition
- Delay in both development of teeth and eruption, ectopic eruption, and abnormalities in incisors and molars are frequently associated with Apert syndrome
- GAPO syndrome is a rare multiple congenital anomalies syndrome involving connective tissue characterized by growth retardation, alopecia, pseudoanodontia due to the failure of tooth eruption, and ocular manifestations
- Delayed eruption is often reported in patients who are deficient in some essential nutrients. The high metabolic demand on the growing tissues might influence the eruptive process. Disturbance of the endocrine glands usually has a profound effect on the entire body, including the dentition. Hypothyroidism, hypoparathyroidism, hypopituitarism, and pseudohypoparathyroidism are the most common endocrine disorders associated with the delayed eruption
- Rare disease like mucopolysaccharidosis is also associated with delayed tooth eruption. Ichthyosis vulgaris is a rare type of hereditary disorder which is characterized by delayed primary and secondary tooth eruption. Cherubism is a skeletal dysplasia characterized by anodontia of the permanent molars and may show delayed eruption. In a rare disease like de Lange syndrome, there is delayed tooth eruption and the teeth are smaller in size.
| Primary Failure of Eruption|| |
Secondary retention described by Raghoebar et al., has many features common with PFE, a term and the concept credited to Proffit and Vig and more commonly used in the literature. They originally defined PFE as a condition in which nonankylosed teeth fail to erupt fully or partially due to malfunction of the eruption mechanism. As reported by Proffit and Vig, this condition was first described by Ballard. Proffit and Vig also described a report by Kaban et al. discussing five cases of molar eruption failure in 1976, one of which might have represented primary failure of eruption. PFE has been defined as a localized failure of secondary tooth eruption which exists without systemic involvement.
| Epidemiology of Primary Failure of Eruption|| |
Literature shows the scarcity of reported epidemiologic studies of PFE in the overall population. Frazier-Bowers et al. analyzed 112 cases of unusual problems of tooth eruption and PFE was confirmed in 39% of the cases. However, this cannot be considered as a true prevalence. Baccetti reported the prevalence of failure of eruption of the 1st and 2nd permanent molars to be 1.7% in the sample of 1520 nonsyndromic patients.
Strickland et al. conducted a retrospective study on the prevalence of various eruption disturbances. A total of 1237 panoramic radiographs of patients between the age of 6 and 25 years were reviewed. The authors reported that 12.4% of the reviewed radiographs showed eruption disturbances. Prevalence of PFE was reported to be 8.4%. With all the available information, it seems that the PFE either has a very low prevalence or it may be under-reported or misdiagnosed with a different entity.
Some studies have shown equal sex predilection of prevalence of PFE in males and females. Whereas, more predilection for females than males was reported by Bacchetti and Vijesh et al. Ahmad et al. published a meta-analysis of identifiable cases of PFE from literature and reported higher predilection for females (60%) than in males (40%).
No significant differences have been found in PFE affected teeth between maxilla and mandible and between the left and right side.,
| Etiology|| |
Earlier studies and case reports published in the literature have revealed familial nature of the primary failure of eruption.,, Analysis of some of these families strongly suggested an autosomal dominant inheritance pattern.,
Patients diagnosed with PFE have revealed mutation in the gene encoding the G protein of parathyroid hormone 1 receptor (PTH1R). PTH1R is present on the small arm of chromosome 3 (3p22-21.1) and encodes a G-coupled protein receptor for both parathyroid hormone (PTH) and parathyroid hormone-related protein (PTHrP). PTH regulates calcium metabolism and is found in the osteoblastic site in the alveolar bone near the tooth bud. PTHrP regulates chondrocyte proliferation and differentiation, playing a key role in skeletal development during early bone growth. Stellate reticulum cells in the dental follicle secrete PTHrP; which induces overexpression of colony-stimulating factor 1 and receptor activator of nuclear factor kappa B ligand responsible for osteoclastogenesis. A concomitant overexpression of bone morphogenic protein 2, which leads to osteogenesis, occurs at the apical end of the dental follicle in a chronologic and spatial fashion.
In a retrospective study by Rhoads, Hendricks, Frazier-Bowers, 16 out of 27 patients diagnosed with PFE showed unclassified nonfunctional single nucleotide polymorphism in PTH1R. With this finding of the absence of mutation in PTH1R gene in these patients, the authors proposed that at least one other gene would be probably responsible for PFE. Genetic analysis study by Frazier-Bowers et al. ruled out AMELX, RUNX2, POSTN, and AMBN as candidate genes for PFE in families and individuals.
A novel mutation in PTH1R (1092delG, which results in a frameshift and premature termination of the PTH1R protein) has been noted in patients diagnosed with PFE. This novel mutation was associated with the clinical finding of infra-occluded deciduous molars along with permanent molars.
| Clinical Features|| |
The estimated age of onset of PFE is suggested to be between the ages of 4 and 21 years. Unerupted teeth are usually unnoticed and asymptomatic until they are detected by a routine radiograph. There is a growth deficiency of the alveolar process in the affected area. Roots of the affected teeth are fully formed and usually do not show any signs of resorption. Tooth is usually seen at the base of a vertical bony defect, indicating a clear eruption path.
Some of the clinical and radiographic characteristics of PFE described by various authors are as follows:
- Involvement of more than one permanent tooth, where, posterior teeth are more commonly affected than anterior teeth, and first permanent molars are more likely involved, followed seldom by premolars or canines
- Varying degree of eruption abnormality is seen distal to the first involved tooth
- The condition is frequently seen unilateral. However, bilateral involvement has been reported
- Involved teeth may erupt partially and then cease to erupt in spite of having clear eruption path and not being ankylosed. However, it is observed that after the eruption failure, these teeth may become ankylosed
- Although it is suggested that PFE does not affect primary teeth, few reports in the literature contradict these observations., Both primary and permanent dentitions are reported to be affected
- The involved teeth do not respond to orthodontic force. If orthodontic force is applied with an attempt to bring the submerged tooth in the arch, it may lead to ankylosis rather than facilitating normal tooth movement
- Frazier-Bowers et al. reported that a high percentage of subjects with PFE had skeletal Class III relationships. However, the study sample was not a representative sample of the population, as it was a collection of records sent by other dentists for diagnosis of failure of eruption.
| Types of Primary Failure of Eruption|| |
Frazier-Bowers et al. from their observations, classified PFE according to the phenotypic characteristics that could be related to the timing of onset. They are as follows:,
- Type 1 is marked by a progressive open bite from anterior to posterior, eruption defect originating at the same developmental time for all affected teeth. Thus, it is related to specific chronological time
- Type 2 also presents as a progressive open bite from the anterior to posterior with varied expression of failure of eruption in more than one quadrant and with greater, however, inadequate eruption of second molar. This defect was earlier related to the stage of root development; however, later with genetic findings, this phenotype was attributed to coordinated series of molecular events that act in a temporally and spatially specific manner such that posterior rather than anterior alveolar bone is affected
- Type 3 was later added by Frazier-Bowers et al. as a combination of Type 1 and Type 2
- Similarly, Type 4 was added as PFE not caused by mutation of PTHR1 gene.
Type 2 is difficult to identify than Type 1; because in Type 2, partial eruption of the second molar might misguide the clinician in recognizing failure of eruption. Differentiation between Types 1 and 2 can be made with surety after the age of 14–15 years when eruption of second molars should be complete under normal circumstances.
| Diagnosis of Primary Failure of Eruption|| |
A significant challenge in the accurate diagnosis of PFE is the high degree of clinical variability observed in familial and isolated cases.
As management of failure of eruption due to mechanical obstruction differs from that of PFE, the first step should be to rule out mechanical obstruction. Any of the factors associated with unerupted or partially erupted tooth-like cyst, tumor or supernumerary teeth or biologic factor (family history, developmental pathology) or oral habits (lateral tongue thrust) should be ruled out. Furthermore, as PFE is a nonsyndromic condition, it is imperative to investigate if failure of eruption is associated with any other syndromic features.
Involvement of permanent first molar along with other adjacent teeth and supracrestal presentation of the affected teeth have been suggested to be hallmark diagnostic features of PFE. As progressive lateral open bite can also be caused by mechanical failure of eruption of posterior teeth due to unlevelled curve of spee or lateral tongue thrust, these reasons should be ruled out. In the absence of such discrepancies progressive lateral open bite remains a key diagnostic feature of PFE. Frazier-Bowers and Tucker proposed the most important features of PFE that aid in diagnosis, namely, infra-occlusion of affected teeth, significant posterior open bite accompanied by normal vertical facial growth and inability of the tooth to move orthodontically. A recent systematic review by Hanisch et al. identified bilateral infraocclusion of the posterior teeth as a hallmark feature of PFE.
Although genetic testing and identification of mutation in PTH1R gene would confirm the diagnosis of PFE, absence of mutation would not necessarily rule out PFE.
| Treatment of Primary Failure of Eruption|| |
Teeth with PFE do not respond to orthodontic forces. An attempt to correct the lateral open bite with orthodontic forces leads to ankylosis of the involved teeth, which could then act as an anchor and further forces to extrude these teeth result in intrusion of the adjacent teeth. Eventually, such attempts will worsen the already existing open bite.
Treatment choice for PFE must be considered after evaluating the patient's age and the severity of PFE. For young children, definitive treatment may not be rendered until the vertical growth is completed. In adult patients with only mild infraocclusion, no treatment is required; however, regular observation is necessary. In young patients, direct or indirect composite build-ups could ensure occlusal stability and preserve alveolar bone level until an implant placement is possible. When affected teeth are partially erupted in the oral cavity, overlay crowns or overlay dentures can be given.,
Extraction of the affected tooth may be the treatment of choice in some cases. Yasumura and Sueishi reported a case of maxillary first molar that failed to erupt after fenestration and responded negatively to orthodontic forces. Extraction of the affected first molar resulted in mesial migration and spontaneous eruption of the unerupted second molar. Further orthodontic treatment resulted in functional occlusion.
Another approach that has been suggested to correct the open bite related to PFE is single tooth osteotomies (corticotomies) with immediate elastic traction taking advantage of the regional acceleratory phenomenon. Shirota et al. reported a case of 24-year-old male with unilateral posterior open bite secondary to PFE of maxillary premolars and molars. The patient was treated successfully with segmental osteotomy along with alveolar distraction.
An alternative orthodontic treatment approach is to avoid treatment with a continuous arch wire and employing a segmented approach from premolar to premolar and leaving the infraocclusion and related open bite in the molar region uncorrected.
| Conclusions|| |
PFE is characterized by nonsyndromic eruption failure of permanent teeth in the absence of mechanical obstruction. Diagnosis of PFE is critical as these teeth fail to respond to orthodontic forces. As the first permanent molars are involved most of the times, a pediatric dentist has the responsibility to diagnose PFE. Careful scrutiny of clinical and radiographic features and whenever possible genetic testing will confirm the diagnosis of PFE. Once diagnosed, above-mentioned treatment modalities may be used to treat the open bite related to PFE.
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