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. 2025 Nov 21;25:1818. doi: 10.1186/s12903-025-07230-5

Outcomes of dental implant treatment in 3–18 year old children with ectodermal dysplasia: a systematic review spanning three decades

Shuyu Zhu 1, Rongkun Chen 2, Shu Zhang 1, Zhiya Wang 1, Zhigang Xie 1,
PMCID: PMC12639673  PMID: 41272663

Abstract

Background

Alveolar hypoplasia in Ectodermal Dysplasia (ED) children poses unique challenges for implant treatment compared to those with missing teeth from trauma or other congenital disorders. This study aimed to summarize and evaluate the clinical evidence regarding the feasibility of implant treatment in young patients with ED.

Materials and methods

The systematic review (PROSPERO registration number: CRD42024600172) adhered to PRISMA guidelines and conducted searches on PubMed, Scopus, Web of Science, and Google Scholar to retrieve articles published between 1994 and 2024. Inclusion criteria focused on identifying studies specifically addressing implant treatment in children with ED. The risk of bias was evaluated using the Joanna Briggs Institute checklist.

Results

The review encompassed 3 retrospective studies, 1 prospective study, 4 case series, and 15 case reports, involving a total of 46 patients aged 3 to 18 years. Among these patients, the highest implant survival rates were observed in the age group of 8 to 13 years, while the lowest rates were noted in the 3 to 8 years age group. The predominant complications identified included the necessity for prosthesis remake or relining and alterations in implant positioning, which can typically be addressed through surgical interventions and adjustments to the prosthesis.

Conclusions

Throughout the dental implant treatment of ED children, the complications relevant to growth and development are inevitable yet manageable, potentially leading to a favorable treatment success rate. However, given the substantial constraints posed by the existing clinical data, the benefit-to-risk ratio of implant therapy in ED children warrants further observation and evaluation.

Supplementary Information

The online version contains supplementary material available at 10.1186/s12903-025-07230-5.

Keywords: Dental implants, Ectodermal dysplasia, Growing patients, Systematic review

Introduction

Ectodermal dysplasia (ED) represents a group of genetic syndromes characterized by congenital anomalies affecting two or more ectodermal structures in the body [1]. These syndromes typically present with mesodermal abnormalities involving hair, teeth, nails, sweat glands, craniofacial structures, and digits [2]. The influence of ED on the oral and maxillofacial region involves restricted growth of the mandible and maxilla, insufficient development of the maxillary and mandibular alveolar ridges, reduced salivary flow, as well as abnormalities and variations in the number and morphology of both primary and permanent dentition [3].

Childhood represents a critical period for psychological and physical development, where effective vocal communication and nutrient intake play vital roles in a child’s overall growth. Compared to healthy individuals of the same age and sex, children with ED often exhibit severe dental anomalies, such as various degrees of tooth agenesis from hypodontia to complete anodontia and malformed, small, and conical teeth [46]. These anomalies lead to underdeveloped alveolar ridges, compromising facial structure and future prosthetic options [4]. Furthermore, the absence of teeth significantly impairs masticatory function, resulting in reduced muscle activity during chewing, inefficient chewing efficiency, nutritional challenges, and mealtime fatigue [7, 8]. Speech impairments are common due to the lack of anterior teeth and reduced salivary flow, with objective phonatory alterations detected in acoustic analyses [9]. In addition, temporomandibular joint disorders have been associated with ED-related dental anomalies; reported issues include correctable disc displacement and congenital structural anomalies [10]. Untreated tooth agenesis in ED may lead to systemic and developmental deficiencies, including nutritional deficits and compromised craniofacial development [11, 12]. ED children often experience a lower quality of life related to oral health, with studies indicating a particular susceptibility to psychological depression around the age of nine when they become acutely aware of their condition [13, 14]. Therefore, early restorative interventions before a child’s school-going age are recommended to address aesthetic, functional, and psychological needs, promoting personal self-esteem and social acceptance [15, 16].

Oral reconstruction for children with ED is essential yet highly challenging. Tooth loss in ED children is predominantly treated with removable partial dentures or complete dentures. Nonetheless, the application of removable dentures over insufficient residual alveolar ridge in individuals with ED could precipitate functional problems. Furthermore, the prevalent hypoplasia of the salivary glands often results in mucosal desiccation, compromising the retention of removable dentures and making their use particularly challenging in children. The advent of oral implant technology over three decades ago provided a promising solution to this dilemma. Nevertheless, concerns regarding the potential impact of implants on maxillofacial development and the risk of implant failure due to alveolar hypoplasia in ED children led many dentists to advise against the use of oral implants in young children. Historically, it has been recommended that the minimum age for implant placement in boys should not be younger than 18 years, and slightly earlier for girls, typically above 16–18 years of age [17].

In reality, young children with ED encounter greater challenges with removable denture usage, making oral implant treatment more essential and beneficial for them [18]. Consequently, some dental professionals advocate for the early utilization of dental implants before growth cessation, and they have been actively investigating this feasibility in clinical studies for over three decades [1921]. Landmark studies by Albert D in 1997 and Alcan in 2006, which included patients as young as 3 or 4 years old [22, 23], demonstrated promising clinical outcomes. These findings prompted Taisse to assert that implantation before the traditional age thresholds is viable, provided that the benefits outweigh the risks [24]. Further clinical studies exploring implant treatment in young children by Toomarian in 2014, even though with a limited sample size, indicated no discernible correlation between implant survival rates and age [25]. These study results indicate that the predominant age for implant placement tends to be around 8 years (early adolescence) [26, 27].

As a result of continuous advancements in implantation techniques and preceding pioneering clinical investigations, the age-related considerations regarding the application of oral implants in pediatric patients have gradually eased over the past decade. The 2013 International Delphi Conference delivered a consensus on oral rehabilitation in children with ED, indicating that implants could potentially be placed in the mandibular anterior region starting from the age of 7–8 years in “necessary cases” [28]. Several systematic reviews have suggested a minimum age of 10 for the placement of oral implants in developing patients to avoid structural, growth-related complications [29, 30].

Due to the rarity of ED, the absence of randomized controlled clinical trials necessitates the reliance on data from various study designs to synthesize clinical experiences with practical implications. The objective is to systemically evaluate the existing clinical evidence concerning the suitability of implant therapy for enhancing the outcomes in young ED patients and to compile valuable insights that can assist pediatric dental clinicians in formulating tailored treatment strategies to maximize the benefits of advancements in implant technique.

Materials and methods

Protocol

A systematic review was meticulously conducted in alignment with the PRISMA guidelines (PRISMA 2020), specifically adhering to “The PRISMA 2020 statement: an updated guideline for reporting systematic reviews.”

Focused questions

  1. Is a younger age at implantation associated with an increased risk of implant failure in ED patients?

  2. What is the severity of complications following implant treatment in pediatric ED patients? Furthermore, how effectively can these complications be addressed or resolved, and what specific techniques are available for their management?

Eligibility criteria

The inclusion criteria were as follows:

  1. Studies reflecting the utilization of implants in patients diagnosed with ED.

  2. Research including suitable subjects, devoid of systemic diseases that could adversely affect the prognosis of dental implants.

  3. Publications comprising encompassing original research articles such as randomized controlled trials (RCTs), observational studies (cohort and case-control studies and case series), and retrospective studies.

  4. Studies reporting on at least one patient who underwent their initial dental implant placement before reaching 18 years of age.

The exclusion criteria were as follows:

  1. Non-ED patients.

  2. Non-Implants.

  3. Pre-clinical studies, review articles, conference proceedings, protocol articles, letters to the editor, or book chapters.

  4. Studies lacking information on patients who received their first dental implant before turning 18 years old.

Search strategy and data extraction

The electronic databases (PubMed, Scopus, Web of Science, and Google Scholar) were systematically searched by two independent reviewers for articles published between January 1994 and January 2024. In case of discrepancies, a third reviewer was consulted. The search terms included (“Ectodermal Dysplasia” OR “hypohidrotic ectodermal dysplasia” OR “ectodermal defect” OR “congenital ectodermal defect”) AND (implant OR “dental implants”). Duplicate entries were eliminated using EndNote X9.2 software. Initial screening of articles based on titles and abstracts was conducted using the same software. Eligible articles meeting the inclusion criteria underwent full-text assessment. Additionally, reference lists were manually scanned for additional relevant studies. One reviewer extracted data including patient details, implantation timing, site of placement, implant characteristics, number of implants, follow-up duration, implant failures, complications, etc., which were cross-verified by the second reviewer. Missing information was sought via email, with non-responsive authors leading to study exclusion. Data extraction and analysis focused exclusively on implant procedures performed on patients under 18 years of age. In cases where a patient received implants both as a minor and as an adult, only data related to implants placed before turning 18 were included. Similarly, for studies involving multiple patients, only those receiving implants before the age of 18 were considered for outcome analysis, while data from patients receiving their first implant as adults were excluded from the analysis.

Risk of bias within studies

The case reports and case series were evaluated by two researchers independently using the Joanna Briggs Institute checklist, a critical appraisal tool designed for the evaluation of case reports. Responses on the checklist were categorized as yes, no, unclear, or not available (Supplementary Material 1).

Results

Study selection

A total of 2517 potentially relevant articles were collected through database and manual searches. Following deduplication (n = 22) and initial abstract screening, 2446 articles were excluded as they did not align with the review objectives. Subsequently, 49 full-text studies underwent detailed evaluation, leading to the exclusion of 26 studies that did not meet the inclusion criteria. As a result, 23 studies were rigorously selected for inclusion in the systematic review. The research selection process and literature search outcomes are illustrated in Fig. 1, adhering to the PRISMA guidelines.

Fig. 1.

Fig. 1

Systematic pattern of the search strategy

Characteristics of the included studies

Among the 23 articles included in the systematic review, there were 3 retrospective studies, 1 prospective study, 4 case series and 15 case reports. A total of 46 patients and 192 implants were included, with patient ages ranging from 3 to 18 years. Detailed information on the eligible studies can be found in Supplementary Material 2.

Implant survival rate

Within the cohort of 46 patients included in this study, there were 5 patients aged between 13 and 18, 21 patients aged between 8 and 13, and 12 patients aged between 3 and 8. Given the relatively limited number of patients in each age subgroup, the analysis focuses on the survival rates of implants across the three age categories under 18 years (Table 1).

Table 1.

Implant survival rate and complication incidence across different age groups

Age range Number of patients Gender Number of implants Average follow-up time Implant survival rate The number of patients with complications
3–7 years 12

M:8

F:2

NA:2

48 5.7y 97.9% 8 (66.7%)
8–12 years 21

M:6

F:5

NA:10

70 4.2y 100% 5 (23.8%)
13–18 years 5

M:3

W:2

23 8y 91.3% 1 (20%)

Abbreviation: M male, F female, NA not available, y year

Table 1 does not include the results of two studies due to incomplete age group information [31, 32]

The collective data analysis of the present study suggests that the incidence of complications in younger children is higher than that of older children, which suggests that implant procedures in younger patients indeed carry a higher risk and should be approached with caution. On the other hand, the fact that this higher risk did not diminish the implant survival rate as shown above, indicates that technological efforts in the past 30 years are able to effectively manage those potential risks.

Complications and management

The complications observed in the included studies were categorized into biological and mechanical complications (Table 2). The most frequently reported issues were the need for denture remaking or relining (17.4%) and implant position changing (17.4%), both attributable to the unique challenge of managing craniofacial growth in pediatric patients.

Table 2.

Implant-related complications and follow-up time based on timing of implantation

Complications Number of patients Timing of implantation (mean age) Average follow-up time
Biological complications Failure of osseointegration 2 (4.3%) 10.5y 10y
Peri-implant diseases (Peri-implantitis, Peri-implant mucositis, Peri-implant soft tissue hyperplasia) 2 (4.3%) 13y 16y
Implant position changing 8 (17.4%) 7.75y 5.5y
Mechanical complications Need for remaking or relining denture 8 (17.4%) 7y 6.3y
Porcelain fracture 1 (2.2%) 8y 1y
Prosthesis fracture 1 (2.2%) 4y 6y
Screw loosening 1 (2.2%) 11y 17y
Detachment of components 1 (2.2%) NA NA
No complication / 29 (63%) 10.69y 4.6y

Abbreviation: y year, NA not available

In the limited case reports available, management strategies for implant position changing (a biological complication resulting from growth-related infraposition) have included surgical reduction of overgrown alveolar bone to improve hygiene access, the use of longer abutments to prosthetically compensate for implant submergence, and, in one complex case, distraction osteogenesis to reposition the implant and alveolar segment [22, 31, 33, 34]. However, it is essential to underscore that these techniques are based on individual cases, and their long-term effectiveness and indications remain incompletely established. Two cases of failure of osseointegration (4.3%) led to implant removal [22, 35]. Furthermore, two cases (4.3%) of peri-implant diseases were reported. One patient in the 8–13 years age group was diagnosed with peri-implantitis affecting four anterior implants. This patient subsequently underwent a protracted peri-implant and periodontal treatment, achieving outcomes that were judged to be stable [36]. Another patient in the 13–18 years age group presented with hyperplastic ulcerated tissue (peri-implant soft tissue hyperplasia) beneath the distal extension of the fixed prosthesis, which was successfully resolved by revising the prosthetic design [37].

Mechanical complications were the most frequently reported type, affecting 12 patients (26.1%). The most common issue was the need for denture remaking or relining (17.4%), directly linked to alveolar growth and dimensional alterations in the developing jaw. Additionally, four other distinct mechanical complications were each reported in a separate patient: screw loosening [38], prosthesis fracture [23], porcelain fracture [39], and detachment of components [40]. It is important to note that these two groups (reline/remake and other complications) involved different patients, indicating that 12 unique patients experienced at least one mechanical complication. These modifications are typically performed during regular follow-up appointments to maintain prosthetic function and fit.

Implant-threatening complications refers to uncontrollable or severe adverse events that directly jeopardize the survival of the dental implant, ultimately leading to the failure of the entire implant treatment [41, 42]. These complications mainly include osseointegration failure, peri-implantitis, implant fracture, and damage to important anatomical structures. In the present study, the incidence of such complications was 6.5% (3/46) at the patient level and 3.6% (7/192) at the implant level [22, 35, 36]. This comprised two cases of failure of osseointegration leading to implant removal [22, 35] and one case of peri-implantitis that posed a direct threat to implant survival [36]. Notably, the most severe complications were the two cases of osseointegration failure, leading to the conversion of restorations from implant-supported to conventional mucosa-supported removable dentures [22, 35]. Notably, the case of peri-implantitis, initially critical, was effectively managed through prolonged treatment, resulting in stabilized outcomes and preventing implant loss [36]. No complications related to implant fracture or structural damage were documented.

The relevance of implant placement to complication

The mandible, particularly the mandibular interforaminal region, was the predominant site selected for implant placement in this study (Table 3). Among the 46 patients included, all received implants in the mandible, with 43 patients receiving implants in the interforaminal region and one patient receiving implants in both the interforaminal and molar regions [16, 22, 23, 25, 3134, 3638, 4353]. Although 34.9% of these patients experienced complications, none of these involved the severe complication of implant failure. The observed complications primarily included implant submerge, implant rotation, and the need for prosthesis revision or replacement [25, 3134, 5153]. These complications, influenced by the craniofacial development of the patients, appeared unavoidable but were effectively managed through appropriate adjustments.

Table 3.

Patient gender, number of implants, follow-up time, survival rate, and complication incidence across different implant sites

Placement site Number of patients Gender Number of implants Average follow-up time Implant survival rate The number of patients with complications
Mandible interforaminal region 43

M:17

F:8

NA:18

128 5.5y 100% 15(34.9%)
Mandible molar region 1 M 3 17y 100% 1(100%)
Maxillary anterior region 6

M:3

F:2

NA:1

15 2.6y 93.3% 2(33.3%)
Maxillary premolar region 5

M:1

F:3

NA:1

12 5.5y 83.3% 3(60%)
Maxillary molar region 1 NA 2 2 m 100% 0

Abbreviation: M male, F female, NA not available, y year, m month

In addition to mandible implants, 14 patients also underwent maxillary implant placement. None of the patients solely received maxillary implants. The implant survival rate for maxillary implants was lower compared to mandibular implants. Out of the 32 maxillary implants, three were removed due to implant failure, hindering the attainment of an implant-supported denture [37]. Three patients reported implant submersion, and one patient required prosthesis remaking [22, 33, 51]. These complications stemmed from growth and development factors but were subsequently resolved.

The relevance of implant size and bone augmentation techniques to complication

Alveolar hypoplasia in ED children makes the implant treatment more difficult than that in other teeth-missing children due to trauma or other congenital disorders. It was suggested that this problem can be alleviated by using mini-implants with diameters ranging from 1.8 to 3 mm for transitional or permanent restorations [54]. This cognition is supported by the collective data in the present study that implantation of mini-implants, narrow-diameter implants, and short implants in children with ED achieved a high implant survival rate with lower complication rate in comparison with standard size implants (Table 4) [25, 37, 4447, 49, 50, 53]. During follow-up, there were no mechanical complications, such as implant fracture, occurred.

Table 4.

Survival rate, complications, and bone augmentation techniques for different types of dental implants

Size of implant Number of patients Number of implants Average follow-up time Implant survival rate The number of patients with complications The number of patients with bone augmentation techniques
Mini-implant 8 28 2.4y 100% 2(25%) 0
Narrow-diameter implant 2 14 7.6y 100% 0 2(100%)
Short implant 3 8 2.3y 100% 0 0
Standard implant 12 40 9.7y 95% 6(50%) 2(16.7%)
NA 14 86 6y 97.7% 8(57.1%) 3(21.4%)

Abbreviation: y: year

Mini-implant (diameter < 3 mm), Narrow-diameter implant (3 mm < diameter < 3.5 mm), Short implant (length < 8 mm). The results of two studies were not included due to incomplete information [16, 31]

Prosthesis and special design for rehabilitation

Choosing implant-supported overdenture and implant-supported fixed restoration in the mandible is a common and safe option [16, 22, 23, 25, 33, 34, 3638, 43, 4553, 55]. Although complications are present, they are generally manageable. Implant-supported single crowns in both the upper and lower jaws demonstrate excellent outcomes with no complications and a survival rate of 100%, making them suitable for partial tooth loss (Table 5) [44, 50].

Table 5.

Implant survival rates, follow-up durations, and complication rate across different rehabilitation methods

Rehabilitation Jaw position Number of patients Number of implants Average follow-up time Implant survival rate since rehabilitation The number of patients with complications
Implant-supported overdenture Mandible 20 45 3.7y 100% 6
Maxilla 3 10 7.2y 80% 2
Implant-supported fixed restoration Mandible 13 58 6.6y 100% 6
Maxilla 2 11 3.1y 100% 1
Implant-supported single crown Mandible 3 9 1.8y 100% 0
Maxilla 2 6 2y 100% 0

Abbreviation: y: year

The detail information of Tables 1–5 is listed in Supplementary Material 3

In addressing the growth and development of the facial cranium, certain pediatric dentists have employed specialized designs. In four studies, prostheses were split in the midline to avoid constricting the mandible growth [16, 22, 36, 37]. Montanari et al. utilized a sliding bar to connect implants and the removable prosthesis [16, 37]. The sliding movement is able to follow the deformation of the mandible during mastication or the opening and closing of the mouth, thus preventing horizontal disturbances to jaw growth [56, 57]. Clarke et al. and Montanari et al. recommend to use of resilient silicone liners and the Seeger Rings to absorb shocking energy and allow for the physiological movement of the prostheses upon loading and functioning [16, 51, 5860].

Discussion

This systematic review assessed the existing clinical evidence regarding the feasibility of implant therapy for improving outcomes in young patients with ED. It synthesized valuable insights aimed at assisting pediatric dental practitioners in formulating tailored treatment strategies for young ED patients to optimize the benefits of advancements in implant technique.

The prevalence of ED is rare, estimated at approximately 7 cases per 100,000 newborns [61]. Despite its rarity, ED poses a significant concern for child welfare globally. With a stable global newborn population of around 140 million annually over the past three decades, an estimated 10,000 ED cases are reported each year, resulting in an accumulated total of approximately 300,000 cases from 1994 to the present [62]. Even within developed countries, which represent about 10–20% of the world’s population, an estimated 30,000–60,000 ED cases of ED have been identified during this period [62]. While it is assumed that proper dental services are available to these children with ED, only a limited number of cases involving implant treatments have been documented in the dental community, underscoring the cautious approach of pediatric dental practitioners towards exploring the potential benefits and risks associated with this advanced technique in pediatric patients.

Implant failure attributed to facial cranium development during childhood remains a significant concern when treating young patients with ED using implants. Data collected in this study revealed a 98% implant survival rate in ED patients who underwent implant placement before the age of 18, with no discernible variance in implant failure rates across different age groups in pediatric patients. Conversely, studies have reported a 95% implant survival rate in young adult ED patients who received implants after the age of 18 [63]. The consistency in implant survival rates between patients under 18 and those over 18 suggested that the risks associated with implant treatment can be effectively managed even in young children with ED, provided that complications are well addressed.

Indeed, the data presented in this study verified a common concern that complications related to growth and development are apparently inevitable in pediatric patients receiving implants for ED. The primary complication observed is implant positioning changes, predominantly ascribed to the posterior and superior rotational growth of the mandible. The rotational growth alters the spatial relationship between the implant and adjacent teeth [64]. Implants placed in the anterior mandible will move with the mandible as growth occurs in the condyles and rami. The mandibular rotation that occurs during growth has not notably posed significant challenges concerning the angulation of these implants and the prosthodontic occlusal plane. While these growth and development-related complications are seemingly in pediatric patients receiving implants for ED, most of them are effectively manageable. Strategies for addressing implant position changes have included surgical reduction of overgrown alveolar bone to enhance hygiene access, utilization of longer abutments for prosthetic compensation of implant submersion, and, in a complex case, distraction osteogenesis for realigning the implant and alveolar segment [22, 31, 33, 34]. However, it is important to note that these techniques are based on individual cases, and their long-term efficacy and indications are not yet fully established. Regular analyses of growth patterns using cephalometric, panoramic radiographs, and CBCT, combined with periodic review, can effectively manage these potential risks.

While mechanical complications like screw loosening and implant or prosthesis fractures are common in adults, they also manifest in younger patients [65, 66]. However, a notable challenge in children is the frequent necessity for denture replacement or relining, primarily due to ongoing alveolar growth and dimensional changes in the developing jaw. This growth-related requirement represents a fundamental difference in the nature and cause of mechanical complications between pediatric and adult populations.

Given the limited sample size, establishing a direct correlation between age and implant failure appears challenging. Conversely, both failed implants originated from the maxilla, suggesting that the implant placement site may be more susceptive to implant failure than the age of implant received.

The belief that mandibular implants are safer than maxillary implants is commonly held and supported by the aggregated data in this study [29, 63]. The mandible, especially the mandible interforaminal region, is frequently preferred for implant placement. Disparities in survival rates may be attributed to the distinct developmental characteristics of the maxilla and mandible. Maxillary implants are susceptible to vertical and anteroposterior displacements due to resorption at the nasal floor and anterior surface, potentially leading to implant failures [22, 43]. Maxillary implants do not move in tandem with the maxilla’s downward and forward growth or the eruption of adjacent structures, often resulting in infraposition [22, 33]. Transversal growth in the maxilla occurs mostly at the mid-palatal suture, particularly before the age of 10 [67]. Implants crossing this suture can restrict transverse growth, thus it is advisable to avoid maxillary implants until early adulthood [68]. In contrast, the mandible experiences less dramatic transverse and vertical skeletal changes. Transverse development ceases before the eruption of permanent canines, with minimal changes thereafter. While continuous forward growth in the premolar region could theoretically lead to lingual implant displacement, this phenomenon was not observed in the current study [67]. The eruption of adjacent teeth and increased mandibular loading from prostheses may elevate vertical alveolar height, a factor that can be effectively managed as mentioned above.

The present study indicates that implantation of mini-implants, narrow-diameter implants, and short implants in children with ED achieved better performance compared to conventional-sized implants. These specialized implants not only effectively withstood occlusal forces in young ED patients but also reduced the necessity for additional bone augmentation procedures, thereby minimizing surgical trauma [69]. The intuitive perception that mini-implants could offer enhanced benefits to ED children with alveolar hypoplasia was validated by the study findings. However, it is surprising to note that out of the 176 implants analyzed, only 50 were special implants, 40 were standard implants, and 86 lacked size documentation. This suggests that the optimal strategy of utilizing mini-implants has not received sufficient attention in cautious clinical practices over the past three decades. While studies have indicated that alterations in implant diameter significantly impact stress distribution, and the survival rates of mini-implants and narrow-diameter implants may not be as favorable as those of conventional implants in adult patients, these conclusions are specific to adults and may not apply to pediatric patients [70]. Further research is warranted to explore the potential benefits and drawbacks of utilizing these special implants in pediatric populations. Considering the promising outcomes observed with mini-implants in ED children, a more proactive consideration of their use is recommended.

Limitations and prospective

The limitations are obvious. As ED is a rare disease, relevant clinical studies are rare and most results are from case reports. Thus, our study may be affected significantly by the so-called “Survivor bias”, namely, successful cases are more likely to be reported and accepted for publication by the academic community, should be noted [71]. While our data seems to support the usage of implants in ED children, the foreseeable survivor bias necessitates caution in drawing definitive conclusions. Improved research designs are hoped to further clarify the findings.

Even though the presence of the above drawback, the collective analysis of all the eligible data during the three decades is able to answer some crucial questions for implant treatment in children with ED.

  1. Is a younger age at implantation associated with an increased risk of implant failure in ED patients?

    The data collected in this study indicated that there is apparently no significant difference in implant survival rates between the ages over and under age 18. However, the foreseeable survivor bias necessitates caution in drawing definitive conclusions.

  2. What is the severity of complications following implant treatment in pediatric ED patients? Furthermore, how effectively can these complications be addressed or resolved, and what specific techniques are available for their management?

    Upon receiving implant treatment, complications may indeed be prevalent in ED children, whereas most complications are manageable and do not diminish the implant survival rate. The most common complications were the need for the denture to be remade or relined and changes in implant position due to craniofacial growth, which might be dealt with surgical reduction of overgrown alveolar bone to improve hygiene access, the use of longer abutments to prosthetically compensate for implant submergence. Special designs of prostheses, such as split in the midline or combining sliding connection, to accommodate the ongoing development of the patient’s jawbone have significant potential for further development. Adjustments to prostheses and radiographic monitoring were required periodically. Mini-implants, narrow-diameter implants, and short implants may be good choices for ED children to avoid complications. It must be emphasized that these are described techniques from individual cases, and their long-term efficacy and indications are not yet fully established.

Conclusions

During the dental implant treatment of ED children, the complications relevant to growth and development are inevitable but manageable, thus the justified successful rate of the treatment may be achieved. However, due to the significant limitation of the clinical data currently available, the benefit-to-risk ratio of the implant treatment of ED children is still to be further observed.

Supplementary Information

12903_2025_7230_MOESM1_ESM.docx (33.1KB, docx)

Supplementary Material 1: Comprises three tables utilizing the Joanna Briggs Institute checklist to assess the Risk of Bias in Case Reports, Case Series, and Cohort Studies included in this systematic review.

12903_2025_7230_MOESM2_ESM.docx (45.5KB, docx)

Supplementary Material 2: Contains two tables that meticulously document the detailed information of eligible studies included in the research. 

12903_2025_7230_MOESM3_ESM.docx (68.9KB, docx)

Supplementary Material 3: Includes five tables that provide detailed records corresponding to Tables 1-5.

Acknowledgements

Not applicable.

Registry and the registration no. of the review

PROSPERO registration number: CRD42024600172.

Abbreviations

PRISMA

Preferred Reporting Items for Systematic Reviews and Meta-Analyses

ED

Ectodermal Dysplasia

Authors’ contributions

Conception and design of the study: Shuyu Zhu, Zhigang Xie; Acquisition of data: Shuyu Zhu, Rongkun Chen, Shu Zhang, Zhiya Wang; Analysis and interpretation of data: Shuyu Zhu, Rongkun Chen, Shu Zhang, Zhiya Wang; Assessed the risk of bias, Graded the evidence: Rongkun Chen, Shu Zhang; Drafting the article, Revising it critically for important intellectual content: Shuyu Zhu, Zhigang Xie; Final approval of the version to be submitted: Shuyu Zhu, Rongkun Chen, Shu Zhang, Zhiya Wang, Zhigang Xie.

Funding

This research was supported by Xingdian Talents-Medical and Health Care Talent Project (XDYC-YLWS-2023-049); the Key Joint Special Project of Yunnan Provincial Science and Technology Department and Kunming Medical University (202101AY070001-025); the Key Project of Yunnan Clinical Research Center for Oral Diseases (2022ZD005).

Data availability

All data generated or analysed during this study are included in this published article and its supplementary materials.

Declarations

Ethics approval and consent to participate

Not applicable.

Consent for publication

Not applicable.

Competing interests

The authors declare no competing interests.

Footnotes

Publisher’s note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

References

  • 1.Asano N, Yasuno S, Hayashi R, Shimomura Y. Characterization of EDARADD gene mutations responsible for hypohidrotic ectodermal dysplasia. J Dermatol. 2021;48(10):1533–41. 10.1111/1346-8138.16044. [DOI] [PubMed] [Google Scholar]
  • 2.Clarke A. Hypohidrotic ectodermal dysplasia. J Med Genet. 1987;24(11):659–63. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 3.Schneider H. Ectodermal dysplasias: new perspectives on the treatment of so Far immedicable genetic disorders. Article. Front Genet Sep. 2022;13:7. 10.3389/fgene.2022.1000744. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 4.Anuroopa A, Abdulla J, Lovely M. Oral rehabilitation of a young patient with hypohidrotic ectodermal dysplasia: A clinical report. Contemp Clin Dent Apr. 2012;3(Suppl 1):S33–6. 10.4103/0976-237x.95101. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 5.AlNuaimi R, Mansoor M. Prosthetic rehabilitation with fixed prosthesis of a 5-year-old child with hypohidrotic ectodermal dysplasia and oligodontia: a case report. J Med Case Rep Nov. 2019;8(1):329. 10.1186/s13256-019-2268-4. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 6.Puttaraju GH, Visveswariah PM. Ectodermal dysplasia in identical twins. J Pharm Bioallied Sci Jul. 2013;5(Suppl 2):S150–3. 10.4103/0975-7406.114314. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 7.Ding M, Kang Y, Qin M, Zhu J. Masticatory function in growing individuals with hypohidrotic ectodermal dysplasia: a longitudinal study. Int J Paediatr Dent. 2025;35(3):598–607. 10.1111/ipd.13271. [DOI] [PubMed] [Google Scholar]
  • 8.Lexner MO, Bardow A, Hertz JM, Nielsen LA, Kreiborg S. Anomalies of tooth formation in hypohidrotic ectodermal dysplasia. Int J Paediatr Dent. 2007;17(1):10–8. 10.1111/j.1365-263X.2006.00801.x. [DOI] [PubMed] [Google Scholar]
  • 9.Semmler M, Kniesburges S, Pelka F, Ensthaler M, Wendler O, Schützenberger A. Influence of reduced saliva production on phonation in patients with ectodermal dysplasia. J Voice. 2023;37(6):913–23. 10.1016/j.jvoice.2021.06.016. [DOI] [PubMed] [Google Scholar]
  • 10.Goyal M, Pradhan G, Gupta S, Kapoor S. Hypohidrotic ectodermal dysplasia with ankylosis of temporomandibular joint and cleft palate: a rare presentation. Contemp Clin Dent. 2015;6(1):110–2. 10.4103/0976-237x.149304. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 11.Sfeir E, Nahass MG, Mourad A. Evaluation of masticatory stimulation effect on the maxillary transversal growth in ectodermal dysplasia children. Int J Clin Pediatr Dent Jan-Mar. 2017;10(1):55–61. 10.5005/jp-journals-10005-1408. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 12.Hsieh YL, Razzoog M, Garcia Hammaker S. Oral care program for successful long-term full mouth habilitation of patients with hypohidrotic ectodermal dysplasia. Case Rep Dent. 2018;2018:4736495. 10.1155/2018/4736495. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 13.Taoufik K, Divaris K, Kavvadia K, Koletsi-Kounari H, Polychronopoulou AJTODJ. Development and validation of the Greek version of the early childhood oral health impact scale (ECOHIS). Open Dent J. 2020;14(1):88–96. [Google Scholar]
  • 14.Gupta P, Agrawal S, Grover CJIP. Hypohidrotic Ectodermal Dysplasia: Classical Clin Features. 2024;61(5):503–503. [PubMed] [Google Scholar]
  • 15.Schnabl D, Grunert I, Schmuth M. Rehabilitation IKJJO. Prosthetic rehabilitation of patients with hypohidrotic ectodermal dysplasia. Syst Rev. 2018;45(7)555–570. [DOI] [PubMed]
  • 16.Montanari M, Grande F. Catapano PSJCid, research. r. Rehabilitation with implant-supported overdentures in preteens patients with ectodermal dysplasia: a cohort study. 2023;25(6):1187–96. [DOI] [PubMed]
  • 17.Chrcanovic, Ramos BJJoC-MS. Dental implants in patients with ectodermal dysplasia: a systematic review. 2018:S1010518218302865. [DOI] [PubMed]
  • 18.Najam YA, Tahmaseb A, Wiryasaputra D, Wolvius E, Dhamo BJIJID. Outcomes of dental implants in young patients with congenital versus non-congenital missing teeth. 2021;7(1):92. [DOI] [PMC free article] [PubMed]
  • 19.Bhattacharjee B, Saneja R, Bhatnagar A, et al. A comparative evaluation of neurophysiological activity, active tactile sensibility and stereognostic ability of complete denture prosthesis, and implant-supported prosthesis wearer-a pilot study. Clin Implant Dent Relat Res. 2022;24(4):510–21. 10.1111/cid.13094. [DOI] [PubMed] [Google Scholar]
  • 20.Sermsiripoca K, Pisarnturakit PP, Mattheos N, Pimkhaokham A, Subbalekha KJCID, Research R. Comparing pre- and post-treatment patients’ perceptions on dental implant therapy. 2021. [DOI] [PubMed]
  • 21.Chatrattanarak W, Aunmeungtong W, Khongkhunthian P. Comparative clinical study of conventional dental implant and mini dental implant-retained mandibular overdenture: a 5- to 8-year prospective clinical outcomes in a previous randomized clinical trial. Clin Implant Dent Relat Res. 2022;24(4):475–87. 10.1111/cid.13098. [DOI] [PubMed] [Google Scholar]
  • 22.Guckes AD, McCarthy GR, Brahim J. Use of endosseous implants in a 3-year-old child with ectodermal dysplasia: case report and 5-year follow-up. Pediatr Dent. 1997;19(4):282–5. [PubMed] [Google Scholar]
  • 23.Alcan T, Basa S, Kargul B. Growth analysis of a patient with ectodermal dysplasia treated with endosseous implants: 6-year follow-up. J Oral Rehabil. 2006;33(3):175–82. 10.1111/j.1365-2842.2005.01566.x. [DOI] [PubMed] [Google Scholar]
  • 24.Waltzman SB, Cohen NL, Gomolin RH, Shapiro WH, Hoffman RAJAJO. Long-term results of early cochlear implantation in congenitally and prelingually deafened children. 1994;15:9. [PubMed]
  • 25.Toomarian L, Ardakani MR, Ramezani J, Adli AR, Tabari ZA. Using implants for prosthodontic rehabilitation of a 4-year-old with ectodermal dysplasia. Gen Dent. 2014;62(5):e1–5. [PubMed] [Google Scholar]
  • 26.Filius MA, Vissink A, Raghoebar GM, Visser A. Implant-retained overdentures for young children with severe oligodontia: a series of four cases. J Oral Maxillofac Surg. 2014;72(9):1684–90. 10.1016/j.joms.2014.04.034. [DOI] [PubMed] [Google Scholar]
  • 27.Mello BZ, Silva TC, Rios D, Machado MA, Valarelli FP, Oliveira TM. Mini-implants: alternative for oral rehabilitation of a child with ectodermal dysplasia. Braz Dent J. 2015;26(1):75–8. 10.1590/0103-6440201300111. [DOI] [PubMed] [Google Scholar]
  • 28.Klineberg I, Cameron A, Hobkirk J, et al. Rehabilitation of children with ectodermal dysplasia. Part 2: an international consensus meeting. Int J Oral Maxillofac Implants. 2013;28(4):1101–9. 10.11607/jomi.2981. [DOI] [PubMed] [Google Scholar]
  • 29.Bohner L, Hanisch M, Kleinheinz J, Jung SJ. Dental implants in growing patients: a systematic review. Br J Oral Maxillofac Surg. 2019;57(5):397–406. [DOI] [PubMed] [Google Scholar]
  • 30.Elagib MFA, Alqaysi MAH, Almushayt MOS, Nagate RR, Gokhale ST, Chaturvedi S. Dental implants in growing patients: a systematic review and meta-analysis. Technol Health Care. 2023;31(3):1051–64. [DOI] [PubMed] [Google Scholar]
  • 31.Kearns G, Sharma A, Perrott D, Schmidt B, Vargervik KJOSOMOPOR. Endodontology. Placement of endosseous implants in children and adolescents with hereditary ectodermal dysplasia. 1999;88(1):5–10. [DOI] [PubMed]
  • 32.Dds MAPF, Arjan Vissink D, Dds GMR, Anita Visser DDS, PJJoO, Surgery M. Implant-retained overdentures for young children with severe oligodontia: a series of four Cases - ScienceDirect. 2014;72(9):1684–90. [DOI] [PubMed]
  • 33.Celar AG, Durstberger G, Zauza K. Use of an individual traction prosthesis and distraction osteogenesis to reposition osseointegrated implants in a juvenile with ectodermal dysplasia: a clinical report. J Prosthet Dent. 2002;87(2):145–8. [DOI] [PubMed] [Google Scholar]
  • 34.Victor E. and, Alveolar bone growth in response to endosteal implants in two patients with ectodermal dysplasia. 1998. [DOI] [PubMed]
  • 35.Huang PY, Driscoll CF. From childhood to adulthood: oral rehabilitation of a patient with ectodermal dysplasia. J Prosthet Dent. 2014;112(3):439–43. 10.1016/j.prosdent.2014.04.012. [DOI] [PubMed] [Google Scholar]
  • 36.Singer SL, Henry PJ, Liddelow G, Rosenberg IJJPD. Long-term follow-up of implant treatment for oligodontia in an actively growing individual: a clinical report. 2012;108(5). [DOI] [PubMed]
  • 37.Peterson Y, Huang, Carl F. Dentistry DJJoP. From childhood to adulthood: oral rehabilitation of a patient with ectodermal dysplasia. 2014. [DOI] [PubMed]
  • 38.McMillan AS, Nunn JH, Postlethwaite KR. Implant-supported prosthesis in a child with hereditary mandibular anodontia: the use of ball attachments. Int J Paediatr Dent. 1998;8(1):65–9. [DOI] [PubMed] [Google Scholar]
  • 39.Singer SL, Henry PJ, Liddelow G, Rosenberg I. Long-term follow-up of implant treatment for oligodontia in an actively growing individual: a clinical report. J Prosthet Dent. 2012;108(5):279–85. 10.1016/S0022-3913(12)60176-0. [DOI] [PubMed] [Google Scholar]
  • 40.Montanari M, Grande F, Lepidi L, Piana G, Catapano S. Rehabilitation with implant-supported overdentures in preteens patients with ectodermal dysplasia: a cohort study. Clin Implant Dent Relat Res. 2023;25(6):1187–96. [DOI] [PubMed] [Google Scholar]
  • 41.Albrektsson T, Zarb GA, Worthington P, Eriksson ARJIJO, Implants M. The long-term efficacy of currently used dental implants: a review and proposed criteria of success. 1986;1(1):11. [PubMed]
  • 42.Buser D, Weber H, Lang NJCOIR. Tissue integration of non-submerged implants. 1-year results of a prospective study with 100 ITI hollow-cylinder and hollow-screw implants. Clin Oral Implants Res. 1990;1(1):33–40. [DOI] [PubMed] [Google Scholar]
  • 43.Kramer FJ, Baethge C, Tschernitschek HJCOIR. Implants in children with ectodermal dysplasia: a case report and literature review. 2010;18(1):140–6.  [DOI] [PubMed]
  • 44.Artopoulou II, Martin JW, Suchko GDJPD. Prosthodontic rehabilitation of a 10-year-old ectodermal dysplasia patient using provisional implants. 2009. [PubMed]
  • 45.Heuberer S, Dvorak G, Zauza K, Watzek GJCOIR. The use of onplants and implants in children with severe oligodontia: a retrospective evaluation. Clin Oral Implants Res. 2012;23(7):827–31. [DOI] [PubMed] [Google Scholar]
  • 46.Paulus C, Martin P. %J revue de stomatologie dCM-fedCO. Hypodontia due to ectodermal dysplasia: rehabilitation with very early dental implants. 2013;114(3):e5–8. [DOI] [PubMed]
  • 47.Fernandes MBZ, Cruvinel ST, Daniela R, Moreira MMAA, Pinelli VF, Marchini OTJBDJ. Mini-implants: alternative for oral rehabilitation of a child with. Ectodermal Dysplasia. 2015;26(1):75. [DOI] [PubMed] [Google Scholar]
  • 48.Bergendal B, Bjerklin K, Bergendal T, Prosthodontics GKJIJ. Dental implant therapy for a child with X-linked hypohidrotic ectodermal Dysplasia—. Three Decades Managed Care. 1900;28(4):348–56. [DOI] [PubMed] [Google Scholar]
  • 49.Güler N, ?ildir u, Iseri U, Sandalli N. ?zkan Dilek %J oral surgery OM, oral Pathology, oral Radiology, Endodontology. Hypohidrotic ectodermal dysplasia with bilateral impacted teeth at the coronoid process: a case rehabilitated with mini dental implants. 2005;99(5):E34–8. [DOI] [PubMed]
  • 50.Sfeir E, Nassif N, Moukarzel CJEJPD. Use of mini dental implants in ectodermal dysplasia children: follow-up of three cases. 2014;15(2 suppl):207–12. [PubMed]
  • 51.Clarke L, Bowyer L, Noone J, Stevens C, Ashley MJOS. Britain’s youngest implant patients? – A Case Series of implant treatment in children with ectodermal dysplasia. 2020.
  • 52.Cezaria Triches T, Ximenes M, Oliveira de Souza JG, Rodrigues Lopes Pereira Neto A, Cardoso AC, Bolan M. Implant-supported oral rehabilitation in child with ectodermal dysplasia – 4-year Follow-up. Bull Tokyo Dent Coll. 2017;58(1):49–56. 10.2209/tdcpublication.2016-0012. [DOI] [PubMed] [Google Scholar]
  • 53.Kilic S, Altintas SH, Yilmaz Altintas N, et al. Six-year survival of a mini dental implant-retained overdenture in a child with ectodermal dysplasia. J Prosthodont. 2017;26(1):70–4. 10.1111/jopr.12366. [DOI] [PubMed] [Google Scholar]
  • 54.Sweeney IP, Ferguson JW, Heggie AA, Lucas JOJIJPD. Treatment outcomes for adolescent ectodermal dysplasia patients treated with dental implants. 2010;15(4):241–8. [DOI] [PubMed]
  • 55.A MM, B CWA BBA, et al. Rehabilitation of ectodermal dysplasia patients presenting with hypodontia: outcomes of implant rehabilitation part 1 - ScienceDirect. 2018;62(4):473–8. [DOI] [PubMed]
  • 56.Azpiazu-Flores FX, Lee DJ, Mata-Mata SJ, Zheng F. Rehabilitation of a patient with mandibular flexure using contemporary glass-infiltrated high performance CAD-CAM polymers: a clinical report with 1-year follow-up. J Prosthet Dent. 2024;132(3):477–83. 10.1016/j.prosdent.2022.12.014. [DOI] [PubMed] [Google Scholar]
  • 57.Mijiritsky E, Shacham M, Meilik Y, Dekel-Steinkeller M. Clinical influence of mandibular flexure on oral rehabilitation: narrative review. International J Environ Res Public Health Dec. 2022;13(24). 10.3390/ijerph192416748. [DOI] [PMC free article] [PubMed]
  • 58.Pozzan MC, Grande F, Mochi Zamperoli E, Tesini F, Carossa M, Catapano S. Assessment of preload loss after cyclic loading in the OT bridge system in an "All-on-Four" rehabilitation model in the absence of one and two prosthesis Screws. Materials (Basel, Switzerland). 2022;15(4). 10.3390/ma15041582. [DOI] [PMC free article] [PubMed]
  • 59.Grande F, Pozzan MC, Marconato R, Mollica F, Catapano S. Evaluation of load distribution in a mandibular model with four implants depending on the number of prosthetic screws used for OT-Bridge system: a finite element analysis (FEA). Materials (Basel, Switzerland). 2022;15(22). 10.3390/ma15227963. [DOI] [PMC free article] [PubMed]
  • 60.Grande F, Cesare PM, Mochi Zamperoli E, Gianoli CM, Mollica F, Catapano S. Evaluation of tension and deformation in a mandibular Toronto Bridge anchored on three fixtures using different framework materials, abutment systems, and loading conditions: a FEM analysis. Eur J Dent. 2023;17(4):1097–105. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 61.Julia RR, Isabel M-RM, Efraín G-G F, M-CC R. M-CA, Glustein PMJIJoD. Hypohidrotic ectodermal dysplasia: clinical and molecular review. 2018. 
  • 62.Sood A, Mishra D. Ectodermal dysplasia: a case report and molecular review. 2020.
  • 63.Guckes AD, Scurria MS, King TS, Mccarthy GR, Brahim JSJJPD. Prospective clinical trial of dental implants in persons with ectodermal dysplasia. 2002;88(1):21–5. [PubMed]
  • 64.Krarup S, Darvann TA, Larsen P, Marsh JL, Kreiborg SJJA. Three-dimensional Anal Mandibular Growth Tooth Eruption. 2005;207(5):669–82. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 65.Alhammadi SH, Burnside G, Milosevic AJBOH. Clinical outcomes of single implant supported crowns versus 3-unit implant-supported fixed dental prostheses in Dubai health authority: a retrospective study. BMC Oral Health. 2021;21(1):171. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 66.Gupta A, Kale B, Masurkar D, Jaiswal P. Etiology of dental implant complication and failure—an overview. AIMS Bioeng. 2023;10(2):141–52. 10.3934/bioeng.2023010. [Google Scholar]
  • 67.Carmichael RP, Am GKBSJAOMSCN, Dental, Implants. Growth Jaws Determ Skeletal Maturity. 2008;16(1):1–9. [DOI] [PubMed] [Google Scholar]
  • 68.Cronin RJJ, Oesterle LJJDCNA. Implant use in growing patients. Treat Plann Concerns. 1998;42(1):1–34. [PubMed] [Google Scholar]
  • 69.Jayakumar P, FelsyPremila G, Muthu MS, Kirubakaran R, Panchanadikar N, Al-Qassar SS. Bite force of children and adolescents: a systematic review and meta-analysis. J Clin Pediatr Dent. 2023;47(3):39–53. [DOI] [PubMed] [Google Scholar]
  • 70.de Oliveira Rigotti RL, Tardelli JDC, Dos Reis AC, da Valente MLC. Influence of dental implant/mini-implant design on stress distribution in overdentures: a systematic review. Oral Maxillofac Surg. 2024;28(2):515–27. [DOI] [PubMed] [Google Scholar]
  • 71.Brown SJ, Goetzmann W, Ibbotson RG, Ross SAJAER. Survivorship bias in performance studies. 1992;79.

Associated Data

This section collects any data citations, data availability statements, or supplementary materials included in this article.

Supplementary Materials

12903_2025_7230_MOESM1_ESM.docx (33.1KB, docx)

Supplementary Material 1: Comprises three tables utilizing the Joanna Briggs Institute checklist to assess the Risk of Bias in Case Reports, Case Series, and Cohort Studies included in this systematic review.

12903_2025_7230_MOESM2_ESM.docx (45.5KB, docx)

Supplementary Material 2: Contains two tables that meticulously document the detailed information of eligible studies included in the research. 

12903_2025_7230_MOESM3_ESM.docx (68.9KB, docx)

Supplementary Material 3: Includes five tables that provide detailed records corresponding to Tables 1-5.

Data Availability Statement

All data generated or analysed during this study are included in this published article and its supplementary materials.


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