- Email: [email protected]
Rehabilitation of medically complex ectodermal dysplasia with novel surgical and prosthodontic protocols
Int. J. Oral Maxillofac. Surg. 2014; 43: 301–304 http://dx.doi.org/10.1016/j.ijom.2013.08.004, available online at http://www.sciencedirect.com
Case Report Craniofacial Anomalies
Rehabilitation of medically complex ectodermal dysplasia with novel surgical and § prosthodontic protocols
M. Dhima1,*, T. J. Salinas1, S. A. Cofer2, K. L. Rieck3 1
Department of Dental Specialties, Division of Prosthetic and Esthetic Dentistry, Mayo Clinic College of Medicine, Rochester, Minnesota, USA; 2Department of Otorhinolaryngology, Division of Pediatric Otorhinolaryngology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA; 3Department of Surgery, Division of Oral and Maxillofacial Surgery, Mayo Clinic College of Medicine, Rochester, Minnesota, USA
M. Dhima, T.J. Salinas, S.A. Cofer, K.L. Rieck: Rehabilitation of medically complex ectodermal dysplasia with novel surgical and prosthodontic protocols. Int. J. Oral Maxillofac. Surg. 2014; 43: 301–304. # 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. The functional and aesthetic needs of a 17-year-old patient afflicted with ectodermal dysplasia, chronic long-term immunosuppression, cleft palate, velopharyngeal insufficiency, hypernasality, maxillary hypoplasia, and oligodontia were met with a multidisciplinary team approach. Predictable functional and aesthetic outcomes were obtained with a combination of injection augmentation of the soft palate and nasopharynx and rigid fixation maxillary external distraction with immediate placement and immediate load protocols. No biological or prosthetic complications were noted after definitive rehabilitation with a mandibular implant-retained fixed prosthesis and a maxillary implant-retained detachable prosthesis.
Introduction
Implant survival rates of 91.3–97.6% have been reported in the mandibular rehabilitation of ectodermal dysplasia (ED) patients when a conventional, two-stage approach, implant-supported prosthesis is used.1 Hypodontia and anodontia are considered to compromise bone growth, affecting the process of osseointegration. In a study of implants placed in children with ED, major risk factors to successful § Presented at the American Academy of Maxillofacial Prosthetics Annual Meeting, Scottsdale, Arizona, USA, November 2011.
0901-5027/030301 + 04 $36.00/0
osseointegration were considered to be low quantity of bone, small dimensions of the mandible, dense cortical bone, and loose cancellous bone.2 An evaluation of bone microarchitecture in the mandibles of ED patients demonstrated a greater trabecular bone connectedness in areas of congenital absence of all teeth compared to areas of up to six teeth missing, which may afford a better resistance to functional loading.3 The hypoplastic maxilla poses a major functional and aesthetic challenge. This can be repaired by maxillary advancement through conventional surgical methods including Le Fort I–III, and often requires multiple subsequent surgeries.4 In cases
Keywords: ectodermal dysplasia; cleft palate; immediate implant placement; immediate implant load; distraction osteogenesis; maxillary hypoplasia; oligodontia; treatment outcome; chronic immune suppression; young adult. Accepted for publication 9 August 2013 Available online 12 September 2013
requiring larger skeletal advancement (more than 10 mm) and soft tissue volume in multiple planes, craniomaxillofacial distraction osteogenesis is considered the primary treatment choice.5 External distraction provides better three-dimensional control of the distraction process, a shorter intraoperative time, and no major operation for device component removal, but has a higher demand with regard to patient compliance.6 Disadvantages include scarring and infection of the skin around the fixation pins, pin loosening, and intracranial pin migration.7 Recently, hyaluronan-based compounds (Deflux; Q-Med, Uppsala, Sweden) have been considered for use as space
# 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
302
Dhima et al.
fillers for posterior pharyngeal wall augmentation.8 Multidisciplinary treatment of a medically complex ED case is presented. Based on recent research findings and careful treatment planning, external distraction osteogenesis was combined with immediate implant placement and immediate load protocols to provide predictable treatment. Materials and methods
In 2010, a 17-year-old Caucasian male presented to the Division of Prosthetic and Aesthetic Dentistry and the Division of Oral and Maxillofacial Surgery for multidisciplinary care. The patient presented with an established diagnosis of ED; a cleft palate had been repaired at another medical centre when the patient was aged 18 months. The medical history was significant for a living donor right kidney transplant at 15 years of age for end-stage renal failure secondary to multicystic dysplasia. The patient was on immunosuppression therapy, which was very well controlled by the transplant team with target levels for immunosuppression with cyclosporine and prednisone. Additional diagnoses included severe maxillary hypoplasia, hypodontia, velopharyngeal insufficiency, non-restorable caries, and generalized moderate periodontitis (Fig. 1a). The patient’s medications included allopurinol, atorvastatin, calcium, vitamin D3, cyclosporine, gabapentin, prednisone, bupropion hydrochloride, and sertraline hydrochloride. The clinical presentation was consistent with a diagnosis of ED and cleft palate; it included
sparse hair, intolerance to light, hypodontia, and maxillary hypoplasia. Treatment planning included a staged approach. It started with the off-label use of a filler injection in the nasopharynx and soft palate to treat the velopharyngeal insufficiency and hypernasality. A paediatric distal chip scope was passed transnasally. A total of 6 ml of off-label filler (Deflux; Q-Med, Uppsala, Sweden) was injected into the soft palate and posterior pharyngeal wall. Clinical and radiographic assessment of the maxilla revealed the need for a significant anterior and vertical advancement. Due to the magnitude of correction required, traditional orthognathic surgery was not performed, but rather a high Le Fort I osteotomy was coupled with the application of a RED device (KLS Martin, Jacksonville, FL) to allow for distraction osteogenesis (Fig. 1b). External distraction osteogenesis was initiated 5 days after device placement at two full revolutions of the anterior ports in a clockwise fashion and two full revolutions of the vertical port in a counterclockwise fashion for 30 days. This was followed by an additional 7 days of distraction with 1 mm distraction for the first 3 days and 0.5 mm distraction for the remaining 4 days. The distraction process was not associated with any complications. The consolidation phase lasted for 3 months. A total of 25 mm of anterior–posterior and 5 mm of vertical displacement were achieved. Post distraction, the patient underwent prosthetic presurgical planning, which revealed the need for a mandibular alveo-
loplasty. This was completed at the time of dental implant placement to accommodate prosthetic and implant components. The intraoral distractor components were removed, and this was followed by full mouth edentulation, alveoloplasty of the mandible, and the placement of a total of eight dental implants (Mark III TiUnite; Nobel Biocare, Yorba Linda, CA, USA) in the maxilla and six implants in the mandible (Fig. 1c). Primary stability to greater than 35 N cm was achieved with all mandibular implants. A prefabricated mandibular complete denture was used as an implant positioning guide during implant placement and was subsequently converted to a fixed implant-supported prosthesis, which was immediately loaded on the same day as the surgery. A two-stage approach was taken for the maxillary implants. The patient underwent uneventful healing for 6 months, uncovering of the maxillary implants, and definitive prosthetic rehabilitation (Fig. 1d). Fig. 2a and b show preoperative and postoperative lateral cephalometric views, respectively. Facial skeletal and soft tissue changes in frontal and lateral views are shown in Fig. 2c–f, respectively. Due to the limited inter-arch distance even after aggressive mandibular alveoloplasty, the mandibular arch was rehabilitated with a metal ceramic fixed prosthesis. The maxillary arch was rehabilitated with an implant-supported overdenture with a primary bar and a metal acrylic overdenture to replace missing dentition and provide lip support. The multidisciplinary approach to his care resulted in both his aesthetic and functional needs being met. Discussion
Fig. 1. Patient from initial presentation to treatment and final rehabilitation.
ED presents with skeletal deficiencies. Oral rehabilitation with the use of endosseous implants often involves orthognathic surgery and ridge or sinus augmentation. The challenge posed here was the planning and sequencing of the surgical and prosthetic steps for predictable management of the functional and aesthetic needs. The complex medical presentation included multiple congenital and acquired conditions such as ED, cleft palate, velopharyngeal insufficiency, hypernasality, maxillary hypoplasia, hypodontia, loss of vertical dimension of occlusion, discrepant occlusal plane, and severely limited restorative space. The perceived concern regarding compromised bone quality and quantity in the jaws of ED patients is in contradiction with the recently described bone microarchitecture.7
Rehabilitation of complex ectodermal dysplasia
303
Fig. 2. Facial and radiographic changes before and after treatment.
In the presented case, implant therapy was central to predictable functional and aesthetic patient rehabilitation. As recommended in the literature, the patient was monitored closely by the provider’s team. Despite this recommendation, the current literature shows a lack of sufficient long-term evidence of the life-expectancy of dental implants in such conditions.9 A recent study on the long-term survival of 45 dental implants placed in 13 liver transplant patients on lifelong immunosuppression showed that at the 3-year follow-up, no implants were lost.10 The off-label use of hyaluronan filler injections resulted in a significantly
decreased hypernasality. The drawback to such an approach is the gradual loss of filler volume, which can occur as early as a few months after application to more than a year. In the treatment planning, sequencing of this step was critical. A concern with the distraction process was that of increased hypernasality, sleep apnea, and difficulty swallowing. To manage this, the filler augmentation was completed prior to the distraction process so that its effect would be ongoing. In this case, the patient did not experience any worsening of hypernasality. Prior augmentation probably had a positive impact as did the slow and gradual distraction process. Other options included repeat injections
or a palatal lift prosthesis to achieve the necessary contact with the pharyngeal wall. After approximately 9 months, increased hypernasality was noted. The patient was made aware of surgical and prosthetic options to improve this and elected not to pursue either option since his hypernasality did not impact his speech intelligibility. Due to the need for large skeletal and surrounding soft tissue augmentation, external distraction osteogenesis in combination with a Le Fort method allowed for better three-dimensional control of the distraction process. The patient’s compliance with the process was high. He did not experience any of the complications reported previously,
304
Dhima et al.
such as pin loosening, infection, or intracranial migration.7 As an effort to compensate for relapse, overcorrection was planned. This was followed by a 3-month period of stabilization prior to prosthetic rehabilitation. An overlap comparison of the patient’s pre- and post-treatment lateral cephalometric radiographs is recommended as a fast and simple method to assess any relapse. Hypodontia and anodontia did not impact the surgical and prosthetic protocols for the mandible. Primary implant stability was achieved in the mandible. Also, during the alveolectomy, the mandibular bone was noted to be very dense but also well vascularized. This seems to support previous evaluations of mandibular bone in ED patients undergoing implant placement.10 The ability to achieve primary stability of mandibular implants allowed for immediate load with the mandibular prosthesis. This, together with uneventful healing contradicts suggestions that hypodontia and anodontia in ED patients may preclude the application of immediate placement and load protocols. These surgical and prosthetic approaches decrease the number of surgical procedures, complications, and prosthetic appointments. They give the patient the chance to evaluate function and aesthetics. Concurrently, providers can utilize this transitional phase to assess patient compliance and mechanical and biological complications. This is vital information that can be used in the design of the final prosthesis to best serve the functional and aesthetic needs of the patient, improve maintenance, and decrease complications. There is, however, a lack of reports in the literature regarding the application of immediate implant placement and immediate load in patients afflicted with ED. Due to the severe maxillary hypoplasia and hypodontia, the patient had experienced considerable supra-eruption of the mandibular dentogingival complex, resulting in limited available restorative space. The significantly limited inter-arch dimen-
sion made an alveolectomy essential in order to accommodate implant and prosthetic components. Further presurgical prosthetic planning revealed the need for lip support. The transitional prostheses were used as surgical guides for implant placement. For both arches, the biomechanical goals were cross-arch implant distribution without the use of cantilevers. The treatment of ED can incorporate the use of orthognathic surgery, distraction osteogenesis, and immediate dental implant placement and loading when planned and sequenced appropriately, to arrive at a predictable, functional, and aesthetic outcome. Additional literature is needed on the application of these surgical and prosthetic approaches in the ED population. Funding
3.
4.
5.
6.
7.
None. Competing interests
8.
None. Ethical approval
None. Acknowledgments. The authors acknowledge Mr Andy Vagle and Mr Thomas Johnson of KLS Martin for their assistance with distraction planning and Dr John E. Volz for his assistance with orthodontic consultation.
References 1. Sweeney IP, Ferguson JW, Heggie AA, Lucas JO. Treatment outcomes for adolescent ectodermal dysplasia patients treated with dental implants. Int J Paediatr Dent 2005;15:241–8. 2. Bergendal B, Ekman A, Nilsson P. Implant failure in young children with ectodermal
9.
10.
dysplasia: a retrospective evaluation of use and outcome of dental implant treatment in children in Sweden. Int J Oral Maxillofac Implants 2008;23:520–4. Silthampitag P, Klineberg I, Jones AS, Austin B, Zee KY, Wallace C, et al. Ultramicroscopy of bone at oral implant sites: a comparison of ED and control patients Part 1—defining the protocol. Int J Prosthodont 2011;24:147–54. Whitaker LA, Bartlett SP. The craniofacial dysostoses: guidelines for management of the symmetric and asymmetric deformities. Clin Plast Surg 1987;14:73–81. McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH. Lengthening the human mandible by gradual distraction. Plast Reconstr Surg 1992;89:9–10. 1-8; discussion. Meling TR, Hogevold HE, Due-Tonnessen BJ, Skjelbred P. Midface distraction osteogenesis: internal vs. external devices. Int J Oral Maxillofac Surg 2011;40:139–45. Kubler AC, Speder B, Zoller JE. Frontoorbital advancement with simultaneous Le Fort III distraction. J Craniomaxillofac Surg 2004;32:291–5. Hallen L, Dahlqvist A. Cross-linked hyaluronan for augmentation of the posterior pharyngeal wall: an experimental study in rats. Scand J Plast Reconstr Surg Hand Surg 2002;36:197–201. Meijer GJ, Cune MS. Surgical dilemmas, Medical restrictions and risk factors. Ned Tijdschr Tandheelkd 2008;15:643–51. Gu L, Yu YC. Clinical outcome of dental implants placed in liver transplant recipients after 3 years: a case series. Transplant Proc 2011;3:2678–82.
Address: M. Dhima Mayo Clinic Department of Dental Specialties Division of Prosthetic and Esthetic Dentistry 200 First Street SW Rochester Minnesota 55905 USA Tel.: +1 507 284 2850 fax: +1 507 284 8082 E-mails: [email protected], [email protected]
Case Report Craniofacial Anomalies
Rehabilitation of medically complex ectodermal dysplasia with novel surgical and § prosthodontic protocols
M. Dhima1,*, T. J. Salinas1, S. A. Cofer2, K. L. Rieck3 1
Department of Dental Specialties, Division of Prosthetic and Esthetic Dentistry, Mayo Clinic College of Medicine, Rochester, Minnesota, USA; 2Department of Otorhinolaryngology, Division of Pediatric Otorhinolaryngology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA; 3Department of Surgery, Division of Oral and Maxillofacial Surgery, Mayo Clinic College of Medicine, Rochester, Minnesota, USA
M. Dhima, T.J. Salinas, S.A. Cofer, K.L. Rieck: Rehabilitation of medically complex ectodermal dysplasia with novel surgical and prosthodontic protocols. Int. J. Oral Maxillofac. Surg. 2014; 43: 301–304. # 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. The functional and aesthetic needs of a 17-year-old patient afflicted with ectodermal dysplasia, chronic long-term immunosuppression, cleft palate, velopharyngeal insufficiency, hypernasality, maxillary hypoplasia, and oligodontia were met with a multidisciplinary team approach. Predictable functional and aesthetic outcomes were obtained with a combination of injection augmentation of the soft palate and nasopharynx and rigid fixation maxillary external distraction with immediate placement and immediate load protocols. No biological or prosthetic complications were noted after definitive rehabilitation with a mandibular implant-retained fixed prosthesis and a maxillary implant-retained detachable prosthesis.
Introduction
Implant survival rates of 91.3–97.6% have been reported in the mandibular rehabilitation of ectodermal dysplasia (ED) patients when a conventional, two-stage approach, implant-supported prosthesis is used.1 Hypodontia and anodontia are considered to compromise bone growth, affecting the process of osseointegration. In a study of implants placed in children with ED, major risk factors to successful § Presented at the American Academy of Maxillofacial Prosthetics Annual Meeting, Scottsdale, Arizona, USA, November 2011.
0901-5027/030301 + 04 $36.00/0
osseointegration were considered to be low quantity of bone, small dimensions of the mandible, dense cortical bone, and loose cancellous bone.2 An evaluation of bone microarchitecture in the mandibles of ED patients demonstrated a greater trabecular bone connectedness in areas of congenital absence of all teeth compared to areas of up to six teeth missing, which may afford a better resistance to functional loading.3 The hypoplastic maxilla poses a major functional and aesthetic challenge. This can be repaired by maxillary advancement through conventional surgical methods including Le Fort I–III, and often requires multiple subsequent surgeries.4 In cases
Keywords: ectodermal dysplasia; cleft palate; immediate implant placement; immediate implant load; distraction osteogenesis; maxillary hypoplasia; oligodontia; treatment outcome; chronic immune suppression; young adult. Accepted for publication 9 August 2013 Available online 12 September 2013
requiring larger skeletal advancement (more than 10 mm) and soft tissue volume in multiple planes, craniomaxillofacial distraction osteogenesis is considered the primary treatment choice.5 External distraction provides better three-dimensional control of the distraction process, a shorter intraoperative time, and no major operation for device component removal, but has a higher demand with regard to patient compliance.6 Disadvantages include scarring and infection of the skin around the fixation pins, pin loosening, and intracranial pin migration.7 Recently, hyaluronan-based compounds (Deflux; Q-Med, Uppsala, Sweden) have been considered for use as space
# 2013 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
302
Dhima et al.
fillers for posterior pharyngeal wall augmentation.8 Multidisciplinary treatment of a medically complex ED case is presented. Based on recent research findings and careful treatment planning, external distraction osteogenesis was combined with immediate implant placement and immediate load protocols to provide predictable treatment. Materials and methods
In 2010, a 17-year-old Caucasian male presented to the Division of Prosthetic and Aesthetic Dentistry and the Division of Oral and Maxillofacial Surgery for multidisciplinary care. The patient presented with an established diagnosis of ED; a cleft palate had been repaired at another medical centre when the patient was aged 18 months. The medical history was significant for a living donor right kidney transplant at 15 years of age for end-stage renal failure secondary to multicystic dysplasia. The patient was on immunosuppression therapy, which was very well controlled by the transplant team with target levels for immunosuppression with cyclosporine and prednisone. Additional diagnoses included severe maxillary hypoplasia, hypodontia, velopharyngeal insufficiency, non-restorable caries, and generalized moderate periodontitis (Fig. 1a). The patient’s medications included allopurinol, atorvastatin, calcium, vitamin D3, cyclosporine, gabapentin, prednisone, bupropion hydrochloride, and sertraline hydrochloride. The clinical presentation was consistent with a diagnosis of ED and cleft palate; it included
sparse hair, intolerance to light, hypodontia, and maxillary hypoplasia. Treatment planning included a staged approach. It started with the off-label use of a filler injection in the nasopharynx and soft palate to treat the velopharyngeal insufficiency and hypernasality. A paediatric distal chip scope was passed transnasally. A total of 6 ml of off-label filler (Deflux; Q-Med, Uppsala, Sweden) was injected into the soft palate and posterior pharyngeal wall. Clinical and radiographic assessment of the maxilla revealed the need for a significant anterior and vertical advancement. Due to the magnitude of correction required, traditional orthognathic surgery was not performed, but rather a high Le Fort I osteotomy was coupled with the application of a RED device (KLS Martin, Jacksonville, FL) to allow for distraction osteogenesis (Fig. 1b). External distraction osteogenesis was initiated 5 days after device placement at two full revolutions of the anterior ports in a clockwise fashion and two full revolutions of the vertical port in a counterclockwise fashion for 30 days. This was followed by an additional 7 days of distraction with 1 mm distraction for the first 3 days and 0.5 mm distraction for the remaining 4 days. The distraction process was not associated with any complications. The consolidation phase lasted for 3 months. A total of 25 mm of anterior–posterior and 5 mm of vertical displacement were achieved. Post distraction, the patient underwent prosthetic presurgical planning, which revealed the need for a mandibular alveo-
loplasty. This was completed at the time of dental implant placement to accommodate prosthetic and implant components. The intraoral distractor components were removed, and this was followed by full mouth edentulation, alveoloplasty of the mandible, and the placement of a total of eight dental implants (Mark III TiUnite; Nobel Biocare, Yorba Linda, CA, USA) in the maxilla and six implants in the mandible (Fig. 1c). Primary stability to greater than 35 N cm was achieved with all mandibular implants. A prefabricated mandibular complete denture was used as an implant positioning guide during implant placement and was subsequently converted to a fixed implant-supported prosthesis, which was immediately loaded on the same day as the surgery. A two-stage approach was taken for the maxillary implants. The patient underwent uneventful healing for 6 months, uncovering of the maxillary implants, and definitive prosthetic rehabilitation (Fig. 1d). Fig. 2a and b show preoperative and postoperative lateral cephalometric views, respectively. Facial skeletal and soft tissue changes in frontal and lateral views are shown in Fig. 2c–f, respectively. Due to the limited inter-arch distance even after aggressive mandibular alveoloplasty, the mandibular arch was rehabilitated with a metal ceramic fixed prosthesis. The maxillary arch was rehabilitated with an implant-supported overdenture with a primary bar and a metal acrylic overdenture to replace missing dentition and provide lip support. The multidisciplinary approach to his care resulted in both his aesthetic and functional needs being met. Discussion
Fig. 1. Patient from initial presentation to treatment and final rehabilitation.
ED presents with skeletal deficiencies. Oral rehabilitation with the use of endosseous implants often involves orthognathic surgery and ridge or sinus augmentation. The challenge posed here was the planning and sequencing of the surgical and prosthetic steps for predictable management of the functional and aesthetic needs. The complex medical presentation included multiple congenital and acquired conditions such as ED, cleft palate, velopharyngeal insufficiency, hypernasality, maxillary hypoplasia, hypodontia, loss of vertical dimension of occlusion, discrepant occlusal plane, and severely limited restorative space. The perceived concern regarding compromised bone quality and quantity in the jaws of ED patients is in contradiction with the recently described bone microarchitecture.7
Rehabilitation of complex ectodermal dysplasia
303
Fig. 2. Facial and radiographic changes before and after treatment.
In the presented case, implant therapy was central to predictable functional and aesthetic patient rehabilitation. As recommended in the literature, the patient was monitored closely by the provider’s team. Despite this recommendation, the current literature shows a lack of sufficient long-term evidence of the life-expectancy of dental implants in such conditions.9 A recent study on the long-term survival of 45 dental implants placed in 13 liver transplant patients on lifelong immunosuppression showed that at the 3-year follow-up, no implants were lost.10 The off-label use of hyaluronan filler injections resulted in a significantly
decreased hypernasality. The drawback to such an approach is the gradual loss of filler volume, which can occur as early as a few months after application to more than a year. In the treatment planning, sequencing of this step was critical. A concern with the distraction process was that of increased hypernasality, sleep apnea, and difficulty swallowing. To manage this, the filler augmentation was completed prior to the distraction process so that its effect would be ongoing. In this case, the patient did not experience any worsening of hypernasality. Prior augmentation probably had a positive impact as did the slow and gradual distraction process. Other options included repeat injections
or a palatal lift prosthesis to achieve the necessary contact with the pharyngeal wall. After approximately 9 months, increased hypernasality was noted. The patient was made aware of surgical and prosthetic options to improve this and elected not to pursue either option since his hypernasality did not impact his speech intelligibility. Due to the need for large skeletal and surrounding soft tissue augmentation, external distraction osteogenesis in combination with a Le Fort method allowed for better three-dimensional control of the distraction process. The patient’s compliance with the process was high. He did not experience any of the complications reported previously,
304
Dhima et al.
such as pin loosening, infection, or intracranial migration.7 As an effort to compensate for relapse, overcorrection was planned. This was followed by a 3-month period of stabilization prior to prosthetic rehabilitation. An overlap comparison of the patient’s pre- and post-treatment lateral cephalometric radiographs is recommended as a fast and simple method to assess any relapse. Hypodontia and anodontia did not impact the surgical and prosthetic protocols for the mandible. Primary implant stability was achieved in the mandible. Also, during the alveolectomy, the mandibular bone was noted to be very dense but also well vascularized. This seems to support previous evaluations of mandibular bone in ED patients undergoing implant placement.10 The ability to achieve primary stability of mandibular implants allowed for immediate load with the mandibular prosthesis. This, together with uneventful healing contradicts suggestions that hypodontia and anodontia in ED patients may preclude the application of immediate placement and load protocols. These surgical and prosthetic approaches decrease the number of surgical procedures, complications, and prosthetic appointments. They give the patient the chance to evaluate function and aesthetics. Concurrently, providers can utilize this transitional phase to assess patient compliance and mechanical and biological complications. This is vital information that can be used in the design of the final prosthesis to best serve the functional and aesthetic needs of the patient, improve maintenance, and decrease complications. There is, however, a lack of reports in the literature regarding the application of immediate implant placement and immediate load in patients afflicted with ED. Due to the severe maxillary hypoplasia and hypodontia, the patient had experienced considerable supra-eruption of the mandibular dentogingival complex, resulting in limited available restorative space. The significantly limited inter-arch dimen-
sion made an alveolectomy essential in order to accommodate implant and prosthetic components. Further presurgical prosthetic planning revealed the need for lip support. The transitional prostheses were used as surgical guides for implant placement. For both arches, the biomechanical goals were cross-arch implant distribution without the use of cantilevers. The treatment of ED can incorporate the use of orthognathic surgery, distraction osteogenesis, and immediate dental implant placement and loading when planned and sequenced appropriately, to arrive at a predictable, functional, and aesthetic outcome. Additional literature is needed on the application of these surgical and prosthetic approaches in the ED population. Funding
3.
4.
5.
6.
7.
None. Competing interests
8.
None. Ethical approval
None. Acknowledgments. The authors acknowledge Mr Andy Vagle and Mr Thomas Johnson of KLS Martin for their assistance with distraction planning and Dr John E. Volz for his assistance with orthodontic consultation.
References 1. Sweeney IP, Ferguson JW, Heggie AA, Lucas JO. Treatment outcomes for adolescent ectodermal dysplasia patients treated with dental implants. Int J Paediatr Dent 2005;15:241–8. 2. Bergendal B, Ekman A, Nilsson P. Implant failure in young children with ectodermal
9.
10.
dysplasia: a retrospective evaluation of use and outcome of dental implant treatment in children in Sweden. Int J Oral Maxillofac Implants 2008;23:520–4. Silthampitag P, Klineberg I, Jones AS, Austin B, Zee KY, Wallace C, et al. Ultramicroscopy of bone at oral implant sites: a comparison of ED and control patients Part 1—defining the protocol. Int J Prosthodont 2011;24:147–54. Whitaker LA, Bartlett SP. The craniofacial dysostoses: guidelines for management of the symmetric and asymmetric deformities. Clin Plast Surg 1987;14:73–81. McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH. Lengthening the human mandible by gradual distraction. Plast Reconstr Surg 1992;89:9–10. 1-8; discussion. Meling TR, Hogevold HE, Due-Tonnessen BJ, Skjelbred P. Midface distraction osteogenesis: internal vs. external devices. Int J Oral Maxillofac Surg 2011;40:139–45. Kubler AC, Speder B, Zoller JE. Frontoorbital advancement with simultaneous Le Fort III distraction. J Craniomaxillofac Surg 2004;32:291–5. Hallen L, Dahlqvist A. Cross-linked hyaluronan for augmentation of the posterior pharyngeal wall: an experimental study in rats. Scand J Plast Reconstr Surg Hand Surg 2002;36:197–201. Meijer GJ, Cune MS. Surgical dilemmas, Medical restrictions and risk factors. Ned Tijdschr Tandheelkd 2008;15:643–51. Gu L, Yu YC. Clinical outcome of dental implants placed in liver transplant recipients after 3 years: a case series. Transplant Proc 2011;3:2678–82.
Address: M. Dhima Mayo Clinic Department of Dental Specialties Division of Prosthetic and Esthetic Dentistry 200 First Street SW Rochester Minnesota 55905 USA Tel.: +1 507 284 2850 fax: +1 507 284 8082 E-mails: [email protected], [email protected]