Implant-Prosthetic Rehabilitation of a Patient With EEC Syndrome Using Additively Manufactured Custom-Made Subperiosteal Implants: A Case Report

Giacomo De Riu, Andrea Biglio, Giovanni Spano, Giuseppe Consorti, Jerome R. Lechien, Luigi Angelo Vaira

Full text link: https://pubmed.ncbi.nlm.nih.gov/40518851/

Implant-Prosthetic Rehabilitation of a Patient With EEC Syndrome Using Additively Manufactured Custom-Made Subperiosteal Implants: A Case Report

1. Scientific Reference

• Study Title: Implant-Prosthetic Rehabilitation of a Patient With EEC Syndrome Using Additively Manufactured Custom-Made Subperiosteal Implants: A Case Report
• Authors: Giacomo De Riu, Andrea Biglio, Giovanni Spano, Giuseppe Consorti, Jerome R. Lechien, Luigi Angelo Vaira
• Journal: Journal of Oral Implantology
• Year of Publication: 2025
• DOI: 10.1177/10556656251350443

2. Scientific Background

Ectrodactyly-Ectodermal Dysplasia-Cleft (EEC) syndrome is a rare inherited disorder frequently associated with craniofacial abnormalities that can significantly complicate oral rehabilitation. In affected patients, severe maxillary atrophy combined with altered anatomical structures may prevent the placement of conventional endosseous dental implants, even after extensive reconstructive procedures. These challenging clinical situations require individualized treatment strategies capable of overcoming anatomical limitations while providing adequate prosthetic support.

Recent advances in digital implantology, including cone beam computed tomography (CBCT), computer-aided design and manufacturing (CAD/CAM), and additive manufacturing technologies, have renewed interest in patient-specific subperiosteal implants. Unlike traditional implant-supported rehabilitation, these custom devices are designed to conform precisely to the patient’s residual bone anatomy, offering a potential solution for cases where conventional implant placement is not feasible. This case report illustrates the application of this digital workflow in the rehabilitation of a patient with severe maxillary deficiency associated with EEC syndrome.

3. Study Objective

The primary objective of this case report was to describe the implant-prosthetic rehabilitation of a 23-year-old female patient affected by EEC syndrome who presented with severe maxillary atrophy after multiple unsuccessful bone grafting procedures. The authors aimed to document the clinical feasibility of a patient-specific titanium subperiosteal implant designed through a fully digital CAD/CAM workflow and manufactured using additive manufacturing technology. The report focuses on the short- to medium-term clinical outcome of this personalized treatment approach rather than comparing it with alternative reconstructive techniques.

4. Methodology

This publication is a single-patient case report, representing a descriptive clinical study with a low level of scientific evidence.

The reported case involved a 23-year-old woman diagnosed with EEC syndrome and presenting with severe maxillary atrophy. Previous reconstructive bone grafting procedures had failed to provide sufficient bone volume for conventional endosseous implant placement. A digital workflow was therefore implemented using cone beam computed tomography (CBCT) and intraoral digital scans to design a patient-specific subperiosteal implant. The implant was manufactured from titanium using CAD/CAM technology and laser-based additive manufacturing to achieve precise adaptation to the patient’s maxillary anatomy.

Clinical follow-up extended to 18 months after surgery. The abstract does not specify additional evaluation criteria, functional outcome measures, or statistical analyses, which is consistent with the descriptive nature of a case report.

5. Main Results

According to the authors, the customized subperiosteal implant remained clinically stable throughout the 18-month follow-up, and no postoperative complications were reported during this period. The personalized implant enabled implant-supported prosthetic rehabilitation despite anatomical conditions that precluded the use of conventional dental implants.

The report demonstrates the practical feasibility of combining digital planning with additive manufacturing to fabricate a patient-specific implant adapted to a severely resorbed maxilla. However, the findings remain limited to a single clinical observation. The available abstract does not provide detailed information regarding peri-implant tissue conditions, prosthetic performance, patient-reported outcomes, functional assessments, or quantitative success parameters. Consequently, the reported outcomes should be interpreted as descriptive clinical observations rather than evidence of predictable long-term treatment performance.

6. Clinical Analysis

This case report illustrates a highly individualized implant rehabilitation strategy for a patient in whom conventional treatment options were no longer feasible. The severe maxillary atrophy associated with EEC syndrome, combined with the failure of multiple bone grafting procedures, created a clinical scenario in which standard endosseous implants could not be considered. Rather than pursuing additional reconstructive surgery, the clinicians selected a patient-specific subperiosteal implant designed to conform to the existing maxillary anatomy.

One of the most relevant aspects of this report is the integration of a fully digital workflow. The combination of CBCT imaging, digital impressions, CAD/CAM design, and additive manufacturing demonstrates how contemporary technologies can be applied to develop customized implant solutions for anatomically complex cases. The individualized design aims to maximize adaptation to the residual bone while supporting implant-prosthetic rehabilitation in circumstances where traditional approaches are unsuitable.

The absence of reported complications and the maintenance of implant stability over an 18-month follow-up suggest that this treatment approach may be clinically feasible in carefully selected patients. Nevertheless, these observations should be interpreted cautiously. As a single case report, the study does not establish the predictability, long-term survival, or reproducibility of custom-made subperiosteal implants. Furthermore, the abstract does not provide detailed information regarding prosthetic function, peri-implant soft tissue health, patient satisfaction, or objective clinical performance indicators.

Consequently, this publication should be viewed primarily as a proof of clinical feasibility rather than definitive evidence supporting routine use of customized subperiosteal implants. Larger clinical studies with extended follow-up will be required to determine the long-term role of this technology in managing severe maxillary atrophy and complex craniofacial deformities.

7. Clinical Applications

The findings presented in this case report suggest that additively manufactured patient-specific subperiosteal implants may represent a valuable treatment option for selected patients with severe maxillary atrophy in whom conventional implant placement is not possible. The reported approach may be particularly relevant for individuals with complex craniofacial conditions, such as EEC syndrome, especially after unsuccessful bone grafting procedures.

The study also highlights the potential clinical value of integrating digital planning, CBCT imaging, CAD/CAM design, and additive manufacturing into implant rehabilitation workflows. These technologies enable the production of implants tailored to each patient’s anatomy, potentially expanding treatment possibilities in highly challenging reconstructive cases. However, given the limited level of evidence provided by a single case report, these applications should be considered exploratory and require confirmation through larger clinical investigations before broader clinical adoption can be recommended.

8. Level of Evidence, Limitations and Transparency

This publication is a single case report, representing a low level of clinical evidence. Its findings describe the outcome of an individualized treatment performed in one patient and should therefore be interpreted as descriptive rather than confirmatory evidence.

Several methodological limitations are either explicit or inherent to the study design. These include the inclusion of only one patient, the absence of a control group, the lack of comparison with alternative treatment modalities, and the descriptive nature of the reported outcomes. Although an 18-month follow-up provides useful medium-term information, it does not allow conclusions regarding long-term implant survival or biological performance.

The abstract does not report any conflicts of interest, financial disclosures, or relationships with implant manufacturers. Since these details are not included in the available information, no conclusions can be drawn regarding potential competing interests. Overall, the clinical observations presented should be regarded as preliminary evidence that warrants validation through prospective clinical studies involving larger patient cohorts.

9. Key Study Highlights

• Study design: Single case report.
• Level of evidence: Low (descriptive clinical evidence).
• Study population: One 23-year-old female patient diagnosed with Ectrodactyly-Ectodermal Dysplasia-Cleft (EEC) syndrome.
• Number of patients: 1.
• Implant used: One patient-specific titanium subperiosteal implant manufactured using CAD/CAM design and additive manufacturing. Additional implant specifications are not provided in the available abstract.
• Follow-up period: 18 months.
• Primary outcome evaluated: Clinical feasibility and stability of a customized subperiosteal implant for implant-prosthetic rehabilitation in the presence of severe maxillary atrophy.
• Main finding: The implant remained stable throughout the 18-month follow-up, with no reported complications.
• Scientific conclusion: The authors suggest that digitally designed, custom-made subperiosteal implants may represent a viable alternative for selected patients requiring complex craniofacial reconstruction when conventional implant therapy is not feasible.
• Study limitations: Single-patient design, no control group, no comparison with alternative treatment approaches, and limited follow-up duration. Additional methodological limitations are not detailed in the available abstract.

10. Scientific Impact

This case report contributes to the growing body of literature exploring personalized implant rehabilitation through digital technologies and additive manufacturing. Rather than providing evidence for widespread clinical implementation, it demonstrates how patient-specific subperiosteal implants can be integrated into the management of exceptionally challenging anatomical situations in which conventional implant therapy is no longer possible.

The report also reflects the ongoing evolution of modern subperiosteal implants. Unlike earlier generations, contemporary designs benefit from advanced digital workflows that combine CBCT imaging, CAD/CAM planning, and three-dimensional metal printing to achieve a highly individualized fit. This technological evolution may broaden reconstructive possibilities for patients with severe maxillary atrophy or rare craniofacial disorders while minimizing the need for additional reconstructive procedures when conventional options have been exhausted.

From a scientific perspective, the publication should be regarded as hypothesis-generating rather than practice-changing. It supports further investigation into customized subperiosteal implant systems and highlights the need for prospective studies with larger patient populations, standardized outcome measures, and long-term follow-up to establish their clinical predictability and durability.

11. Editorial Conclusion

This case report presents an individualized implant rehabilitation strategy for a patient with EEC syndrome and severe maxillary atrophy in whom conventional implant therapy was not achievable. The successful medium-term outcome observed after placement of a custom-made, additively manufactured subperiosteal implant illustrates the potential of digital technologies to address highly complex reconstructive challenges. However, because the evidence is limited to a single clinical case, the findings should be interpreted cautiously. Further clinical research involving larger cohorts and longer follow-up periods will be necessary to determine the long-term reliability, safety, and clinical indications of customized subperiosteal implants in advanced implant rehabilitation.