Table 4 Characteristics of case reports and case series included
Reference/Country/Language | Nº patients Type of disease Sex* (M/F) Age** (I/F) | Orthodontic and/or dentofacial orthopedic treatment Treatment of permanent or deciduous dentition Success in orthodontic and/or dentofacial orthopedic treatment Esthetic and functional rehabilitation | Pattern of tooth agenesis Missing teeth Disturbances of the teeth Jaws affected | Radiologic study Cephalometric study |
|---|---|---|---|---|
Karwetzky et al. [41]/Germany/German | N = 1 ED Sex: M Age: I: 8 y F: 2 y | Orthopedic: U-Bar activator type I to stimulate maxillary growth Orthodontic: canine bands to increase intercanine width Treatment of permanent canines Outcomes: intercanine distance increased to 18–20 mm (7 months) Upper and lower removable prostheses | Mandibular anodontia/Maxillary oligodontia All permanent teeth missing, except two maxillary canines No disturbances in the two erupted canines Maxilla and mandible affected | No OPG No cephalometric study |
Waggoner et al. [45]/New York, USA/English | N = 1 HED Sex: M Age: I: 34 m F: 7 weeks | Orthodontic: Brackets for dental alignment to close 6 mm wide inter-incisive diastema, twice due to recurrence. Two brackets, rectangular wire and elastics changed at 3-week intervals Treatment of deciduous dentition Successful treatment: closed diastema and at 8 months X-ray showed no radicular resorption Upper and lower removable prostheses | Mandibular oligodontia/Maxillary oligodontia All permanent teeth were missing, except two maxillary central incisors and mandibular first molars Conically-shaped primary maxillary central incisors Maxilla and mandible affected | OPG. Mandibular occlusal X-ray No cephalometric study |
Itthagarun et al. [46]/Hong Kong/English | N = 1 HED Sex: F Age: I: 7 y F: 6 m | Orthodontic: (1) maxillary removable orthodontic appliance with finger spring to close the space between the inter-incisive diastema; (2) fixed appliance with open coil spring, due to loss of removable appliance Treatment of permanent dentition Successful treatment: closed diastema Upper and lower removable prosthesis | Mandibular oligodontia/Maxillary oligodontia Missing deciduous teeth: 62, 64, 71, 72, 81, 82 Missing permanent teeth: 11, 12, 13, 14, 16, 17, 22, 23, 24, 25, 26, 27, 31,32, 33, 35, 36, 37, 41, 42, 43, 45, 46, 47 Conical teeth: 51, 52, 53, 61, 63. 61 showed a shorter root compared with its antimere Maxilla and mandible affected | OPG No cephalometric study |
Köymen et al. [64]/Turkey/Turkish | N = 1 ED Sex: M Age: I: 16 y F: - | Orthodontic: brackets to close inter-incisor diastema Treatment in permanent dentition Successful treatment: closed diastema Upper and lower removable prosthesis | Mandibular oligodontia/Maxillary hypodontia Missing permanent teeth: 12, 13, 22, 23, 31, 32, 33, 41, 42, 43 No disturbances of the teeth Maxilla and mandible affected | OPG No cephalometric study |
Yenisey et al. [48]/Turkey/English | N = 1 HED Sex: M Age: I: 10 y F: 7 m | Orthodontic: a button was placed on the retained tooth in the palate after the mucosa was opened surgically. Another button was placed on the upper removable prosthesis. Elastics were attached between the buttons to apply a 60-g force Treatment of permanent dentition Successful treatment: closed diastema Upper and lower removable prosthesis | Mandibular anodontia/Maxillary oligodontia All mandibular permanent teeth were missing. Radiographic examination showed a conical impacted tooth in the middle of the right anterior maxilla, the rest of the teeth were missing There were two conical, hypoplastic anterior teeth in the left maxilla Maxilla and mandible affected | OPG No cephalometric study |
Gruber et al. [49]/New York/English | N = 1 HED Sex: M Age: Phase 1: I: 7 y F: 7 m Phase 2: I: 8 y F: 3 y | Phase 1: Orthopedic treatment: palatal expander and protraction headgear—retainers with spring Orthodontic diagnosis: anterior crossbite, severe class III malocclusion, retrusion maxillary, deficient maxillary and mandibular height Goal: to enhance alveolar growth and establish a class I occlusion Outcomes: overjet 3–4 mm Phase 2: protraction headgear Goal: To correct rapid growth of the mandible and a current edge-to-edge anterior bite relationship Negative sequelae: gingival recession of the vestibular aspect of 72 Outcomes: Class I relationship and 4 mm overjet Treatment of deciduous and permanent dentition No prosthetic treatment has been performed so far | Mandibular and maxillary oligodontia Missing permanent teeth: 12, 13, 15, 22, 23, 24, 25, 31, 33, 34, 35, 37, 41, 42, 44, 45, 47 Deciduous teeth: 20 conical shaped microdontic teeth, amelogenesis imperfecta. The roots were unusually long Permanent teeth: 11 hypocalcified, microdontic and conically shaped Maxilla and mandible affected | OPG (5 ½, 8 and 10 years) Lateral skull X-ray (6, 7 ¾ and 10 years) No cephalometric study |
Altug-Atac et al. [50]/Turkish/English | N = 1 EDW Sex: M Age: Phase I: I: 6 y. and 4 m F: 6 m Phase II: I: 9 y. and 3 m F: 6 m. + 6 m. for retention Phase III: I: 10 y. and 3 m F: 21 m | Phase I: Removable tongue shield was inserted to rehabilitate tongue Phase II: Class II activator was applied to stimulate mandibular growth and eliminate the 12.5-mm overjet Phase III: Orthodontic treatment. Dental alignment with fixed brackets. Intermaxillary class II elastics to achieve and maintain the skeletal class I relationship. Coil springs to close the spaces Treatment of deciduous and permanent dentition Success achieved in all phases of treatment No prosthetic treatment indicated | Mandibular hypodontia Missing permanent teeth: 41, 42, 31 53, 63, 73, 83, 12, 22, 31, 32, 41, 42 were conically shaped Class II, division I malocclusion. Retrognathic mandible | OPG (before and after treatment) 4 cephalometric studies Phase II: Skeletal class II (ss-n-sm: 7.71°) severe hyperdivergent growth pattern (NSL/ML: 39.82°) (ILs/NL: 114.4º and ILi/ML: 92.4º) End of treatment: ss-n-sm: 6.06° NSL/ML: 43.36° ILs/NL: 92.71° ILi/ML: 85.42° |
Ioannidou-Marathiotou et al. [51]/Greece/English | N = 1 HED Sex: M Age: I: 10 years F: 6 y. and 8 m | Orthopedic: removable partial prothesis with palatal expansion screw. Goal: to enhance the transverse growth of the maxilla Orthodontic: brackets with a segmental arch wire and elastic chain for dental alignment. Goal: to close the midline diastema Treatment in permanent and deciduous dentition There was an increase in the transverse dimensions of the upper dental arch Removable upper and lower prostheses | Mandibular anodontia, maxillary oligodontia All mandibular deciduous and permanent teeth were missing. In maxillary arch 53, 63, 11, 16, 21, 26 were present 53, 63, 11, 21 showed conical shaped Mandible and maxilla affected | OPG (before and after treatment) 2 cephalometric studies 10 years: concave skeletal profile (ANB: − 6.7°, retrognathic maxilla (SNA: 72.7°) decreased lower face height. Midline diastema of 5 mm 16 years and 8 months: face had normal vertical dimensions and the anteroposterior skeletal discrepancy had improved. (SNA: 73.8º; ANB: − 0.4º) |
Oblack et al. [66]/Slovenia/Slovene | N = 1 ED Sex: M Age: I: 12 y F: – | Orthodontic: brackets to dental alignment and maintenance of space Treatment of permanent and deciduous dentition Orthodontic treatment allowed rehabilitation with 4 maxillary implants, crowns and bridges | Maxillary and mandibular oligodontia Missing permanent teeth: 12, 13, 14, 15, 22, 24, 25, 31, 32, 34, 35, 41, 42, 44 53, 11, 21, 23, 33, 43 conical shaped Maxilla and mandible affected | OPG No cephalometric study |
Priya et al. [54]/India/English | N = 1 HED Sex: F Age: I: 22 y F: – | Orthodontic: brackets for dental alignment Treatment of permanent and deciduous dentition Orthodontic treatment allowed rehabilitation with 2 maxillary and 1 mandibular implants, crowns and bridges, to recover the reduced vertical dimension | Maxillary and mandibular hypodontia Missing permanent teeth: 15, 12, 22, 27, 38, 45 Generalized Microdontia. Canines had conical shaped. Molars appeared malformed with obliterated occlusal tables Maxilla and mandible affected | OPG No cephalometric study |
Shah et al. [43]/India/English | N = 1 XHED Sex: M Age: I: 21 y F: 6 m | Orthodontic: brackets for erupted canine tooth retained in the palate and dental alignment Treatment of permanent dentition The tooth retained was exposed Rehabilitation was done with 3 mandibular implants, crowns and bridges | Maxillary and mandibular oligodontia Missing permanent teeth: 14, 16, 17, 22, 24, 25, 27, 31, 32, 33, 36, 37, 41, 42, 43, 46 Two impacted teeth in the right maxillary canine region and one in the left maxillary canine region Maxilla and mandible affected | OPG No cephalometric study |
Suja et al. [56]/India/English | N = 1 HED Sex: F Age: I: 24 y F: - | Orthodontic: brackets for dental leveling and aligning Treatment of permanent and deciduous dentition Orthodontic treatment allowed rehabilitation with crowns and fixed prothesis in permanent and primary retained teeth | Mandibular and maxillary oligodontia Missing permanent teeth: 12, 14, 15, 17, 22, 24, 25, 27, 31, 32, 33, 37, 41, 42, 43, 47 Anterior teeth were conically shaped Maxilla and mandible affected | OPG No cephalometric study |
Bergendal et al. [57]/Sweden/English | N = 1 ED Sex: M Age: I: 21 y F: 12 mo | Orthodontic: dental leveling and aligning Treatment of permanent and deciduous dentition At 6 y and 4 mo placement of two mandibular implants to retain overdenture. Maxillary removable denture At 19 y and 10 mo. Two additional mandibular implants to support fixed complete prosthesis After orthodontic treatment two maxillary tooth supported prostheses, preserving a small midline diastema | Mandibular anodontia and maxillary oligodontia All deciduous teeth were missing except two maxillary incisors and canines All permanent teeth were missing except 11, 21, 16 and 26 Deciduous teeth were conically shaped. Permanent teeth were malformed Maxilla and mandible affected | OPG (5y and 6mo, 7y, 33y) Cephalometric studies: 7y and 3mo: Normal maxilla and normal sagittal and vertical relation with the mandible 10y and 4mo: slight class III with ANB -1° and ANPg -2° 20y and 4mo: slight class III 32y and 5mo: the angulated abutments of mandibular prosthesis compensated for the slight class III and the profile looked similar to a class I |
Kościelska et al. [58]/Poland/English | N = 2 P2 ED Sex: M Age: I: 5 y F: - P3 ED Sex: M Age: I: 12 y F: - | P2 Orthopedic: maxillary and mandibular prostheses with Fisher screws to expand Treatment of deciduous dentition Success in esthetic and functional rehabilitation with the removable prosthesis with screws P3 Orthopedic: acrylic denture for maxilla and mandible with Fisher screws Treatment of permanent and deciduous dentition Success in esthetic and functional rehabilitation with the removable prosthesis with screws | P2 Mandibular oligodontia/Maxillary oligodontia All deciduous teeth were missing except 55, 65, 85 55, 65, 85 microdontics Maxilla and mandible affected P3 Mandibular oligodontia/Maxillary oligodontia Missing permanent teeth: 12, 13, 14, 17, 22,23, 24, 25, 31, 32, 33, 34, 35, 37, 41, 42, 43, 45, 47 No disturbance in the erupted teeth Maxilla and mandible affected | P2 No OPG No cephalometric study P3 OPG No cephalometric study |
Knobloch et al. [59]/Canada/English | N = 1 HED Sex: M Age: Phase I: I: 11 y F: – Phase II: I: 18–9 y F: 2–3 y | Phase I (11 y.): Presurgical orthodontics with brackets. Retention with Hawley plate Goal: To address the impacted left posterior segment and the midline diastema and consolidate the spaces of anterior maxillary teeth Orthodontic treatment allowed consolidation of anterior maxillary spaces, but the left impacted teeth could not be repositioned, and the midline diastema could not be totally closed Phase II: Fixed postsurgical orthodontics. (The orthognathic surgery was Le Fort type I to correct maxillary AP hypoplasia at end of growth) Goal: to control tooth position and aid the surgeon in maintaining proper maxillary stability Orthodontic phase II was successful Esthetic functional rehabilitation: 10 y. Two mandibular anterior implants to support overdenture 20 y. 3 additional implants in anterior mandible and 2 implants in posterior maxilla 21 y. In maxilla, crowns and removable prosthesis tooth-implant supported. In mandible, fixed prosthesis implant supported | Mandibular oligodontia/Maxillary oligodontia All deciduous teeth were missing except 65. Permanent missing teeth: 13, 14, 15, 17, 23, 24, 25, 27, 31, 32, 33, 34, 35, 37, 41, 42, 43, 44, 45, 47 11, 12, 21, 22 were conically shaped. 16 were dysmorphic Maxilla and mandible affected | OPG. (10, 18–19, 20, 21 y.) Lateral skull X-ray (17 y.) Cephalometric study: maxillary AP hypoplasia |
Celli et al. [15]/Italy/English | N = 1 HED Sex: M Age: I: 6 y F: 4 y | Phase 1: Maxilla and mandible: Removable orthopedic/prosthesis with repositioning of teeth and expansion screw Phase 2: Maxilla: maxillary rapid expander partially removable and partially fixed. The device repositioned the anterior missing teeth Mandible: Fixed orthopedics with reposition of teeth and telescopic screw Treatment of permanent and deciduous dentition Success in dentofacial orthopedic treatment in both phases Rehabilitation was with the same prosthesis | Maxillary hypodontia and Mandibular oligodontia Deciduous missing teeth: 51, 52, 61, 62, 71, 72, 81, 82 Permanent missing teeth: 11, 12, 15, 21, 22, 31, 32, 35, 37, 41, 42, 45, 47 Deciduous canines were conically shaped Maxilla and mandible affected | OPG Cephalometric study (Steiner and Rickets) Skeletal class I with a trend to class III Maxillary and mandibular retrognathism Vertical hypodivergent skeletal pattern Class I molar and canine Overjet and overbite could not be estimated because all front teeth were absent |
Kuźniarski et al. [16]/Poland/English | N = 1 ED Sex: M Age: I: 3 y F: - | Orthopedic: removable plate with reposition of teeth and Fisher expansion screws in the maxilla and mandible Treatment in deciduous dentition No information on expansion success. The diastema was not treated Rehabilitation was with the same removable prosthesis | No information on permanent teeth Deciduous missing teeth: 52, 53, 54, 62, 63, 64, 71, 72, 73, 74, 75, 81, 82, 83, 84, 85 Deciduous incisors with a conical, strongly sharpened shape Maxilla and mandible affected | No OPG No cephalometric study |
Schanbl et al. [60]/Austria/English | N = 2 P1 XHED Sex: M Age: I: 10 y F: - P2 XHED Sex: M Age: I: 17 y F: – | P1 Orthodontic: the diastemas between the anterior maxillary teeth were closed by a fixed orthodontic device Treatment in deciduous dentition The diastemas were closed At 10 y. rehabilitation was made with a mandibular tooth-supported overdenture and a maxillary removable two-implant-retained prosthesis P2 Orthodontic: pre-post orthodontic gap closure between 21 and 22 by means of a spring attached to the removable denture Treatment in permanent dentition The gap was closed Rehabilitation was carried out with 4 maxillary and 4 mandibular implants, crowns, and partial removable dentures tooth and implant attached | P1 Mandibular oligodontia/Maxillary oligodontia Deciduous missing teeth: 52, 54, 55, 62, 64, 65, 71, 72, 73, 74, 75, 81, 82, 83, 84, 85 Permanent missing teeth: 12, 13, 14, 15, 16, 17, 22, 23, 24, 25, 26, 27, 31, 32, 34, 35, 37, 41, 42, 44, 45, 46, 47 All anterior teeth erupted were conically shaped Maxilla and mandible affected P2 Mandibular oligodontia/Maxillary oligodontia Permanent missing teeth: 11, 12, 14, 15, 16, 17, 23, 24, 25, 26, 27, 31, 32, 33, 34, 35, 37, 41, 42, 43, 44, 45, 47 All erupted anterior teeth were conically shaped Maxilla and mandible affected | P1 OPG (6, 10 y.) No cephalometric study P2 OPG (17, 23 y.) No cephalometric study |
Ierardo et al. [62]/Italy/English | N = 1 HED Sex: M Age: I: 8 y F: 12 days activation + 6 m. retention | Orthopedic: hybrid modified rapid palatal expander with braces in 6 + 6 and two mini-screws in the anterior palatal region Treatment of permanent and deciduous dentition Successful resolution of the transverse contraction Definitive treatment not indicated | Mandibular oligodontia/Maxillary oligodontia Deciduous missing teeth: 52, 54, 62, 64, 71, 72, 73, 74, 81, 82, 83, 84 Permanent missing teeth: 12, 13, 14, 15, 16, 17, 22, 23, 24, 25, 26, 27, 31, 32, 33, 34, 35, 37, 41, 42, 43, 44, 45, 47 All erupted anterior teeth were conically shaped Maxilla and mandible affected. Hypotrophy of both jaws | OPG Occlusal X-ray. To verify the diastases of the median palatal suture CBCT to verify the quantity of available bone and space for correct insertion of miniscrews Cephalometric study Class I with a light trend to skeletal class III Hypo-divergence Counterclockwise growth Reduced anterior facial height |
Szemraj-Folmer et al. [63]/Poland/English | N = 1 ED Sex: F Age: Phase I I: 8 y F: 2 y Phase II I: 11 y F: 58 m | Phase 1. Removable orthodontic appliance for distalization of the maxillary right first molar prior to autologous transplantation Phase 2: Fixed orthodontics for dental alignment, space maintenance and occlusion after autologous transplantation The objectives of orthodontic treatment were achieved: orthodontic alignment of the teeth (including the transplanted premolars) closing spaces at the donor sites reducing the space between the mandibular left first molar and canine The end of rehabilitation treatment not explained | Maxillary hypodontia/mandibular hypodontia Missing teeth 12, 22, 23, 24, 25, 34, 35, No disturbances of the teeth Maxilla and mandible affected | OPG (8, 15 and 16 y.) No cephalometric study |
Yajing Liu et al. [61] /China/English | N = 1 ED Sex: F Age: I: 17 y F: 12 m | Orthodontic: brackets for dental alignment and inter-incisor diastema closure Treatment of permanent dentition Orthodontic treatment achieved closure of the diastema Definitive rehabilitation was carried out in maxilla with 2 esthetic crowns on central incisors and 2 partial fixed prostheses 6-implant-supported. In mandible with a complete prosthesis supported by 6 implants | Mandibular oligodontia/Maxillary oligodontia Permanent missing teeth: 12, 13, 14, 15, 17, 22, 23, 24, 25, 27, 31, 32, 33, 34, 35, 37, 41, 42, 43, 44, 45, 47 Erupted maxillary and mandibular anterior teeth were conically shaped Maxilla and mandible affected | OPG No cephalometric study |
Wimalarathna et al. [14]/Sri Lanka/English | N = 1 HED Sex: M Age: I: 11 y F: 5 y | Orthodontic: brackets for dental alignment and space maintenance The objective of orthodontic treatment was achieved Treatment in permanent and deciduous dentition Final rehabilitation was achieved with esthetic crowns | Mandibular hypodontia/maxillary hypodontia Permanent missing teeth: 12, 15, 22, 23, 31, 41 53, 62, 63, 73, 81, 83, 32, 42 conically shaped. First lower molars with taurodontism Maxilla and mandible affected | OPG No cephalometric study |
Gonzaga Luiz et al. [42]/Brazil/English | N = 1 ED Sex: F Age: I: 19 y F: 8 m | Orthodontic: DSD (digital smile design) aligners program-based Treatment in permanent and deciduous dentition Success of orthodontic treatment was partial, as the tooth alignment was achieved but not the extrusion needed to reestablish the vertical dimension Rehabilitation was made with esthetic crowns on natural tooth and implants (2 in maxilla and 2 in mandible) | Mandibular oligodontia/maxillary hypodontia Permanent missing teeth: 12, 13, 22, 23, 31, 32, 37, 41, 42, 43, 47 Erupted maxillary and mandibular anterior teeth were conically shaped Maxilla and mandible affected | OPG CBCT Cephalometric study, before and after orthodontic treatment, but not described |