Complete oral rehabilitation of a deep bite patient with Ewing’s sarcoma; surgical and prosthetic phases: A clinical report :Mehran Bahrami, Moeen H Shirazi, Mohammed Hussein M Alsharbaty, Dental Hypotheses

CASE REPORT
Year : 2017 | Volume : 8 | Issue : 3 | Page : 80--84

Complete oral rehabilitation of a deep bite patient with Ewing’s sarcoma; surgical and prosthetic phases: A clinical report

Mehran Bahrami, Moeen H Shirazi, Mohammed Hussein M Alsharbaty
Dental Research Center, Department of Maxillofacial Prosthodontics, School of Dentistry, Tehran University of Medical Sciences, Tehran, Iran

Correspondence Address:
Mohammed Hussein M Alsharbaty
Dental Research Center, Department of Prosthodontics, School of Dentistry, Tehran University of Medical Sciences, Kargar Street, Tehran
Iran

Abstract

Introduction: Ewings sarcoma (ES) is an unusual, aggressive malignant neoplasm occurring primarily in the long bones of the lower and upper extremities followed by pelvis, ribs, vertebrae, skull, and jaws-bones. Patients with ES tumor need long-term complete oral reconstruction. The location of the primary tumor in the face is predominantly in the mandible, and usually in the posterior mandible. Case Report: Here, both surgical procedures and prosthetic reconstruction in three stages of a 10-year-old childs life presented. Definitive prosthetic reconstruction is extremely challenging in such patients after previous excessive surgical resection, especially when the patient has complex dental abnormality. Fixed dental prostheses and implant-supported fixed restorations have been selected as the preferred treatment option in this case after increasing occlusal vertical dimension (OVD) to create sufficient interocclusal space (IOS). Discussion: In this case, ES patient treated in three phases. In the first-stage, only surgical resection of tumor and bone graft reconstruction with titanium plate fixation was performed. In the second-stage, Lefort I surgery for maxilla with dental implants insertion in mandible was done simultaneously. For definitive restorations fabrication, it was mandatory to increase occlusal vertical dimension (OVD) to create sufficient inter-occlusal space (IOS) for the restorations. OVD has been altered to achieve more stable relationship of mandibular teeth with maxillary teeth, and to obtain an optimum esthetic and functional result. Several factors should be considered as determinants for increasing the OVD such as remaining tooth structure, the space available for the restorations such as the current situation, occlusal variables, and esthetics.

How to cite this article:
Bahrami M, Shirazi MH, Alsharbaty MM. Complete oral rehabilitation of a deep bite patient with Ewing’s sarcoma; surgical and prosthetic phases: A clinical report.Dent Hypotheses 2017;8:80-84

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Bahrami M, Shirazi MH, Alsharbaty MM. Complete oral rehabilitation of a deep bite patient with Ewing’s sarcoma; surgical and prosthetic phases: A clinical report. Dent Hypotheses [serial online] 2017 [cited 2023 Jun 2 ];8:80-84
Available from: http://www.dentalhypotheses.com/text.asp?2017/8/3/80/212430

Full Text

Introduction

Ewing’s sarcoma (ES) is an unusual, aggressive malignant neoplasm occurring primarily in the long bones of the lower and upper extremities followed by pelvis, ribs, vertebrae, skull, and jaws-bones.[1] ES is a malignant, small, round, blue-cell tumor with metastatic potential that usually occurs in older children and young adults. At early diagnosis, approximately 25% of patients with ES present with clinically noticeable metastases.[2] The location of the primary tumor in the face occurs in approximately 2% of the cases, predominantly in the mandible,[3],[4],[5] and less frequently in the maxilla about one-eighth as often as the mandible.[6] This tumor usually appears in the posterior mandible;[3],[7] however, it could originate in the anterior mandible, crossing the midline. This tumor was originally described by James Ewing in 1921, and was considered to arise from undifferentiated osseous mesenchymal cells, however, the cell of origin is undeniably recognized. This lesion comprises 4–15% of all primary bone tumors and accounts for 3% of all childhood cancers.[8]

The common signs and symptoms are swelling, paresthesia, and pain. Radiographic examination may show “onion skin,” “sun ray,” or “honey comb” features.[9] ES, mainly in children, demonstrates difficulty in arriving at a specific diagnosis, although this can be handled with the aid of histopathological and immunohistochemical assessment.[9],[10] There is no formal staging system available for this tumor, and it is usually classified as localized, metastatic, or recurrent. The management of the lesion is by multimodal therapy and depends on the extent of the lesion. In cases of localized ES of mandible, surgery with chemotherapy is the treatment of choice.[4],[6],[11],[12]

Here, a case of ES of mandible in a 10-year-old patient managed by aggressive surgical resection (hemimandibulectomy) followed by iliac crest autogenous bone graft reconstruction is reported. After 9 years, the patient was recalled for pursuing the definitive treatment; complete rehabilitation with implant insertion surgery and prosthesis fabrication has been established.

Case Report

First treatment intervention

A 10-year-old child was referred to the Maxillofacial and Implants Department in Tehran University of Medical Sciences. The chief complaint was painful swelling and periapical lesion in the mandible of 2-month duration. Clinical examination revealed large firm mass fixed to the underlying bone in the left mandible with painful tenderness extending from #31 to #37. There was also expansion in both buccal and lingual cortex. Radiological examination revealed an ill-defined radiolucent region involving the body of the left side of the mandible.

Computed tomography (CT) scan revealed lytic lesion in the left body of the mandible with associated destruction of the medial cortical plate and adjacent soft tissue mass. The surgeon had performed a frozen section procedure, and the mass was sent to the pathological laboratory for further assessment. Histological examination of biopsy specimen showed a neoplastic tissue composed of proliferated elongated cells with slightly pleomorphic spindle-shaped nuclei arranged in twisted bundles. The final diagnosis was ES; the final decision was to conduct surgical resection followed by bone graft. First, the patient was managed preoperatively by chemotherapy.

The surgical procedure consisted of type IV mandibular surgical resection of the involved side by hemimandibulectomy, followed by iliac crest autogenous bone graft with titanium plate fixation. Intraoperatively, margins of both buccal and lingual soft tissue and bony margins from the labial cortex of the opposite side were sent for frozen section examination. Histological examination did not demonstrate tumor cells on any of the specimens. Patient was then referred to the department of oncology for postoperative chemotherapy. The postoperative result was pleasing with no recurrence. Three months later, the patient recalled for mandibular transitional removable denture fabrication to improve the mastication efficiency. Every 6 months, the patient was followed-up.

Second treatment intervention

After 8 years, the patient returned for definitive treatment initiation. Intraoral examination showed that the patient had class II dental relationship with deep bite occlusion and without stable occlusal contact. Further assessment revealed maxillary occlusal surfaces contacting the lower gingival tissue, occlusal canting, crowded teeth in both jaws, enlarged tongue, and missing teeth #(31–37) due to the previous surgical resection [Figure 1] and [Figure 2]. The main dilemma in this situation was the absence of interocclusal space (IOS) and maxillary occlusal canting with lingualized teeth. There were three treatment options to resolve maxillary occlusal canting, i.e., orthodontic intrusion using temporary anchorage device (TAD), crown lengthening followed by teeth preparation, and Lefort I orthognathic surgery. Lefort I surgery was considered as a valuable treatment for correcting maxillary occlusal canting. For the mandible, the decision was to simultaneously insert seven dental implants with maxillary surgery to complete oral rehabilitation.{Figure 1}{Figure 2}

The surgeon performed the Lefort I-one piece osteotomy procedure for maxilla. Seven two-stage implants were placed in the mandible in the position of teeth #41, 33, 34, 35, 36, 37 (Dentium/Superline, 3.8 × 10 mm, internal hexagon, Seoul, South Korea) and #38 (Dentium/Superline, 3.8 × 7 mm, internal hexagon, Seoul, South Korea). The surgeon extracted #41 during implant insertion because of severe tooth mobility, and implant was inserted in its place immediately. The most distal implant was too far posteriorly near the ramus with no benefit to be used in the future prosthesis, hence, there was no choice just to leave it submerged in the bone.

Definitive treatment phase

Six months after the surgery, the patient returned for definitive prosthetic phase initiation. After making preliminary impressions, stone casts were mounted. Wax-rim on temporary-record-base was used for establishing OVD and recording centric relation (CR). Using an arbitrary face-bow (Dentatus, New York, USA) and CR record, preliminary casts were mounted in a semiadjustable articulator (AB Dentatus, New York, USA). Diagnostic wax up was accomplished on these casts. The decision was to fabricate FDP for the remaining teeth #(42–47) on the right side and implant-supported fixed prostheses (ISFP) on the left side of mandible. Because the patient had deep bite with no enough IOS to fabricate the restorations, the decision was to increase the OVD in the posterior region with 2 mm.

The mandibular teeth from #(42–47) have been prepared for metal ceramic restorations (MCR). Using diagnostic wax up, chair-side provisional crowns were fabricated with self-cure acrylic resin (Tempron, GC Crop, USA) according to the designated OVD. The provisional crowns were temporarily cemented (Tempbond, Kerr Corp, Italy). Interim-acrylic-resin partial denture prosthesis for the edentulous left side was fabricated at the predetermined VDO, using canine-protected concept of occlusion. During this provisional phase, patient’s oral hygiene, speech, lip-closure path, anterior guidance, and mastication were monitored. Absence of any problems after approximately 2 months confirmed the patient’s satisfaction of both the interim prosthesis and the provisional restorations that have been fabricated at the predetermined OVD. The customized anterior guidance was fabricated by making impression from these provisional and interim prostheses. The related stone casts were mounted. Following 2-month period with the provisional crowns and interim prosthesis, definitive dental and implants restorations fabrication was preceded. Open-tray one-step impression was made with custom tray and elastomeric impression material (Panasil, Kettenbach GmbH, Germany). Both implants and prepared teeth in mandibular arch were recorded in this impression, simultaneously.

Metal frameworks of MCR restorations were tried. Then, porcelain (Ceramco 3, Dentsplay, USA) was applied. Canine-protected concept of occlusion was used in both sides during lateral excursions. For delivery appointment, all dental and implants restorations were cemented with temporary cement (Tempbond, Kerr Corp, Italy), and panoramic radiograph after initial cementation was obtained [Figure 3],[Figure 4],[Figure 5]. After 3 weeks, the patient was recalled for checking the occlusion, and only the teeth MCRs were cemented with RMGI permanent cement (FujiCEM 2, GC Corp, USA).{Figure 3}{Figure 4}{Figure 5}

Oral hygiene was meticulously instructed, and the patient was motivated for application of chlorhexidine mouthwash. The patient has been followed-up for more than 1 year with no complication.

Discussion

In this case report, ES patient was treated in three phases. In the first-stage, only surgical resection of tumor and bone graft reconstruction with titanium plate fixation was performed. In the second-stage, Lefort I surgery for maxilla with dental implants insertion in mandible was done simultaneously. As the maxillary arch had occlusal canting with no IOS available, three treatment options were available. One option was crown lengthening for maxillary teeth and elective root canal treatment (RCT), which could be destructive option and then crowns fabrication. Because the crown-root ratio was not in optimal situation, this option was excluded. Another option was orthodontic intrusion with temporary anchorage devices (TADs), but it has been ruled out due to several risks and complication. These complications could be summarized such as root trauma, which might affect the dental root or periodontal ligament, and may lead to loss of tooth vitality or ankylosis.[13] Stationary anchorage failure may occur because TADs may become loose. When miniscrews become mobile, they will not regain stability and may need to be removed or could be fractured.[14],[15] Placement of TADs in the maxillary palatal slope may increase injury risks to the greater palatine nerve.[16] Another complication could include soft-tissue irritation or sinus perforation.[17] The last rational option is Lefort I one-piece osteotomy surgery, which was the preferred treatment option in this case. Model surgery and references marks have been done successfully on the articulator to aid in intraoperative positioning.

For definitive restorations fabrication, it was mandatory to increase OVD to create sufficient IOS for the restorations. OVD has been altered to achieve more stable relationship of mandibular teeth with maxillary teeth, and to obtain an optimum esthetic and functional result. Increasing the OVD from the clinical perspective has been reported to facilitate the treatment of patients presenting with complex dental abnormalities such as generalized tooth wear and significant occlusal irregularities.[18],[19],[20]

Several factors should be considered as determinants for increasing the OVD such as remaining tooth structure, the space available for the restorations such as the current situation, occlusal variables, and esthetics. Minimizing the increase in VDO is useful to reduce the overall complexity of the prosthodontic treatment. Several studies revealed that increased OVD with fixed prostheses indicated less symptoms severity than removable prostheses.[21],[22],[23],[24],[25] This result could be related to the fixed restorations having the advantages of being fixed in the mouth, imitating natural teeth morphology, decreasing bulkiness with reduced interferences with speech, and improved general comfort. In addition, the fixed nature of the prostheses may improve patient compliance and approval of the treatment. The limitations related to this case were to leave the most distal implant submerged with no benefits in the treatment plan. Another constraint was to prepare the left mandibular teeth to increase the OVD and restorations fabrication. The patient was followed for more than 1 year, and no signs or symptoms of muscular or tempromandibular joint disorders were noticed.

Conclusion

In this case report, clinical steps have been explained for treating patient with mandibular ES in three stages, from surgical resection procedure till the definitive prosthetic phase. The patient was completely satisfied with the outcome and has been followed-up for more than 1 year.

Financial support and sponsorship

None.

Conflicts of interest

There are no conflicts of interest.

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