Advances in Plastic & Reconstructive Surgery © All rights are reserved by Łukasz Krakowczyk. et al.

*Address for Correspondence: Dr. Łukasz Krakowczyk, Maria Sklodowska-Curie Institute-Oncology Center Gliwice Branch, Department of Oncological and Reconstructive Surgery, Ul.Wybrzeże AK 15, 44-100 Gliwicce, Warsaw; Tel: +48322788417; E-Mail: lukaszkrakowczyk@wp.pl

Received: May 20th, 2019; Date Accepted: July 6th, 2019; Date Published: July 8th, 2019.

Ł. Krakowczyk1, K. Dowgierd3, M. Borowiec3, D. Smyczek2, D.Walczak1, A. Maciejewski1

1Maria Sklodowska-Curie Institute-Oncology Center Gliwice Branch, Department of Oncological and Reconstructive Surgery Warsaw.2John Paul II Upper Silesian Child Health Centre, Medical University of Silesia in Katowice, Department of Pediatric Surgery and Urology, Warsaw.3Voivoid Children Hospital, Department of Head and Neck Surgery-Maxillofacial Surgery in Olsztyn, Warsaw.

AbstractBackground and Aims: Central Giant Cell Granuloma (CGCG) accounts for 1-7% of all benign lesions of the head and neck region. It often arises in the maxilla followed by mandible and affects children and young adults. Free flap surgery in the pediatric population has gained widespread acceptance regarding its technical utility and reliability. One-stage reconstruction combining osseous free flaps with virtual surgical planning are becoming the standard for mandibular defects. The aims of the study are to present the cases of aggressive type of CGCG and surgical procedure strategy.

Materials and Methods: Six children (5 boys, 1 girl, and age ranging from 9 to 14 y.o) were treated in Children Hospital in Olsztyn during the period 2013-2017. In the present material, the clinical features, diagnosis, and surgical treatment of the giant cell tumor found in the cheek with extensions into mandible, in one case on the both side, as a multiple tumors of the mandible. Because of aggressive, histopathological type of CGCG, in all cases we performed an extended tumor resections with one stage reconstructions using fibula free flap with virtual surgical planning.

Results: The success rate of free flaps was 100%. In one case we note the venous occlusion on the 2nd postoperative day, requiring revision of the vascular anastomosis. There were no donor site complications. Partial skin island necrosis was seen in one case which not required surgical intervention.

Conclusions: In aggressive histopathological type of CGCG, we have to performed an extended tumor resections with one stage reconstructions. The advantages of free flap with virtual surgical planning in children, which include better adaptation of the flap growth and more ideal reconstruction of lost parts, provide the surgeon with more satisfactory functional and aesthetic results.

Keywords: Giant Cell Granuloma; Head and Neck; Computer-aided Surgical Planning; Reconstruction, Fibula Free Flap.

useful characterization of serotonin receptor subtypes in the treatment of

Research Article ISSN: 2572-6684

Central Giant Cell Granuloma of the Mandible in Children with One Stage Reconstructions Using Fibula Free Flaps with Virtual Surgical Planning

middle and lower face defects, even in the pediatric population [1]. Initial concerns as to the feasibility and reliability of the procedure inchildren were readily overcome by the fact that the size of the pedicle vessels is big enough to make anastomosis and that the donor sites are sufficient [2, 3].

Virtual surgical planning has proved an invaluable tool to better understand the anticipated defect when creating a reconstructive plan and restoring lost parts. The models are used in this regard and custom titan plates and implants to promote increased intra-operative quality.

The aim of this study was to review our experience with fibula free flap with virtual surgical planning for reconstruction after rese-ction of central giant cell granuloma of the mandible in children and to assess the feasibility and safety of this technique. Only the cases of children treated for aggressive variety of CGCG were reviewed so that we could focus on the unique challenges of excisional and reconstru-ctive surgery in these children.

Materials and Methods

The study included all patients who underwent extended rese-

Adv Plast Reconstr Surg, 2019 Page 252 of 256

Introduction Central Giant Cell Granuloma (CGCG) is not a true neoplasm, but rather a reactive process; its origin can be triggered by trauma or inflammation. It is a rare bony lesion in the head and neck region, and it most commonly affects mandible followed by the maxilla and often seen in children and young adults, in the second and third decades of life. Fast-growing lesions have rarely been reported. In these six cases, CGCG are characterized by an aggressive behavior against an innocent histological appearance, pain, and rapid facial swelling. The present cases illustrates aggressive variety of CGCG, with an a typical clinical presentation in six children. Diagnosis of the disease was made in particular on computed tomography and biopsy.

Free flaps are a useful reconstructive option in the management

Ł. Krakowczyk, K. Dowgierd, M. Borowiec, D. Smyczek, D.Walczak, A. Maciejewski. Venous Central Giant Cell Granuloma of the Mandible in Children with One Stage Reconstructions Using Fibula Free Flaps with Virtual Surgical Planning. Adv Plast Reconstr Surg, 2019; 3(3): 252-256.

ction of the tumors in mandible following with primary fibula free flap planning at the Children Hospital in Olsztyn between 2013-2017 (5 boys, 1 girl; age ranging from 9 to 14 y.o). The study was approved by the institutional review board of the Maria Sklodowska-Curie Institute-Oncology Center Gliwice Branch. All of the selected patients had pathology reports confirming the aggressive variety of CGCG. Follow-up notes from surgical and medical outpatient clinics were reviewed for late complications, evaluation of growth and function at the reconstructed and donor sites, and outcome. The mean follow-up was 20.5 months (range 6-58). As a part of our workup imaging studies were obtained, including a craniofacial CT and lower extremity CT. Virtual surgical planning was use to provide fibular cutting guides and to provide custom contoured plates and jigs. The plan of the surgery include the proper sequence of the resection of the tumor, marking the edges and specimen handoff.

Results

In three cases resection of the mandible includes the body, angle and half of the ramus was performed and in one case almost whole mandible without mental protuberance [Figure 1]. In two cases only the body of the mandible was performed. A total of six fibula free flap for reconstruction of surgical defects following surgery for giant cell granuloma of the mandible were executed. Having displayed the steps of the surgery on the wall inside the operating theatre (printed, colorful pictures as well as donor site and tumor resection from virtual surgical planning) it is possible, that free flap harvesting can occur simultaneo-usly with tumor resection. As far as well designed complicated case is concerned all participants from the surgical team are well prepared for the surgery, know exactly the steps of the procedure which help it to prevent from mistakes, and do it exactly as in the plan.

In all cases fibula osteo septocutaneous flap was designed with small 2x5cm skin paddle as a survival monitor with isolated about 5-10 cm peroneal vessel pedicle [Figure 2].

Prior to pedicle division the fibula cutting guide was attached to the fibula. Proximal and distal osteotomies were then performed and connected to individual titanium plate [Figure 3]. A limited neck dissection exposing the facial artery and vein were performed in all cases. After osteosynthesis fibula with mandible the microvascular anastomosis were performed, connecting the facial artery with the peroneal artery using coupler size from 1.5 to 2.5mm, and facial vein with peroneal vein using coupler size from 2.0 to 3.0mm. In 4 cases the skin islands were sutured as a survival monitor to the submental region, in two cases were placed in floor of the month, none of them required the tracheostomy. The success rate of free flaps was 100%. In one case we note the venous occlusion on the 2nd postoperative day, requiring revision of the anastomosis. There was no impairment of donor site growth or function, and no any complications. Partial skin island necrosis was seen in one case which did not require surgical intervention. Functional outcome, including mastication, deglutition, and speech was very satisfactory [Figure 4]. Following the early postoperative period, all children resumed a full oral diet and were able to masticate and swallow solid food. In any of the cases there was no need for a feeding gastrostomy. All children had intelligible speech and the cosmetic results were excellent. All children could resume normal physical activities, and there were no reports of limb length discrepancies. At last follow-up six children were free of disease in operated region, but in two cases the giant cell granuloma appear in another different localization of the body: in the iliac crest bone and in maxilla.

Figure 1: Pre-operative appearance, multifocal giant cell tumors of the mandible in virtual surgical planning, final stereolithografic models with actual custom plates used intraoperatively and fibula cutting guides.

Adv Plast Reconstr Surg, 2019 Page 253 of 256

Ł. Krakowczyk, K. Dowgierd, M. Borowiec, D. Smyczek, D.Walczak, A. Maciejewski. Venous Central Giant Cell Granuloma of the Mandible in Children with One Stage Reconstructions Using Fibula Free Flaps with Virtual Surgical Planning. Adv Plast Reconstr Surg, 2019; 3(3): 252-256.

Figure 3: Giant cell tumors of the right side of the mandible, the postoperative specimen and preparation of fibula free flap with skin island.

Figure 2: Fibula free flap preparation, CT after tumors resection and reconstruction and three month post-operative appearance and functional outcome.

Adv Plast Reconstr Surg, 2019 Page 254 of 256

Ł. Krakowczyk, K. Dowgierd, M. Borowiec, D. Smyczek, D.Walczak, A. Maciejewski. Venous Central Giant Cell Granuloma of the Mandible in Children with One Stage Reconstructions Using Fibula Free Flaps with Virtual Surgical Planning. Adv Plast Reconstr Surg, 2019; 3(3): 252-256.

Figure 4: X-ray and CT after tumors resection and reconstruction and three month post-operative appearance and functional outcome

Discussion Central Giant Cell Granuloma (CGCG) is found in 75% in the

mandible, usually near the mental foramen and often crossing the midline, also in the region of molar teeth [4].

From slow and asymptomatic growth, associated with non-aggre-ssive form, which applies to 60-80% of cases, that stay unnoticed for a long time, because of their growth inside of the trabecular bone and no pain or neurological disorders. To the rarely occurring aggressive type, usually found in 19 to 40% of all CGCG cases, which causes bone mutilation, migration and loss of teeth, root resorption and infiltration of the soft tissues. Frequent symptoms are pain and pare-sthesia, pathological fractures and recurrences.

Ultrastructural and immunohistochemical examinations perfo-rmed to estimate the type of the tumor, showed no difference between aggressive an non-aggressive types. Whereas in patient with aggre-ssive type tumor, high level of proliferation marker-Ki 67 was observed [1].

The use of a vascularized composite bone flap for functional mandible reconstruction represents the current state-of the-art technique. Satisfactory aesthetic results can be achieved using the fibula flap with virtual surgical planning, which is the ideal choice for mandible reconstruction [5, 6]. In the present cases, the autologous reconstruction utilized digital technology and custom-made models were performed with excellent functional and aesthetic results. However, the short-term outcome particularly if this does not extend through puberty-may differ significantly from the final result following the adolescent growth spurt and resultant anatomical changes and asymmetries [7].

There are few article comparing the reconstruction using virtual surgical planning and performing them without it.

It is commonly known, that the effect relied on the surgeons’ subjecti-ve assessment of aestethic and future function. Although acceptable outcomes are achievable, the personal competences results in variable results. All surgeons who are using VSP are consentaneous: the time of the surgery is shorter and what is more important, the aesthetical and functional result is better [8, 9].

Conclusions

In aggressive, histopathological type of CGCG, we have to performed an extended tumor resections with one stage reconstru-ctions. The surgical result of this cases demonstrate that free flap reconstruction using virtual planning is an efficient and relatively safe technique for reconstructing surgical defects of the head and neck in children undergoing extensive surgery. Preoperative virtual planning along with use of prefabricated cutting jigs allows for precise contou-ring and positioning of microvascular free flaps (fibula, iliac crest, medial condyle of femur flaps) head and neck region reconstructions. Using this technique, the authors have achieved high rate (100%) of dental rehabilitation along with reduced operative times. The authors believe that virtual planning technologies are becoming a gold standard technique in mandible surgery and this technology in selected patients can significantly improve the quality of reconstruction.

Acknowledgements

All authors confirm that this research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

Declaration of conflicting interests

The Authors declares that there is no conflict of interest.

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Adv Plast Reconstr Surg, 2019 Page 256 of 256

Ł. Krakowczyk, K. Dowgierd, M. Borowiec, D. Smyczek, D.Walczak, A. Maciejewski. Venous Central Giant Cell Granuloma of the Mandible in Children with One Stage Reconstructions Using Fibula Free Flaps with Virtual Surgical Planning. Adv Plast Reconstr Surg, 2019; 3(3): 252-256.

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2 Thompson SH, Bischoff P, Bender S. Central giant cell granuloma of the mandible. J Oral Maxillofac Surg. 1983; 41:743-746. [Crossref]

3 3.Roasa FV, Castañeda SS, Mendoza DJC. Pediatric free flap reconstruction for head and neck defects. Curr Opin Otolaryngol Head Neck Surg. 2018; 26:334-339. [Crossref]

4 4. Akçal A, Karşıdağ S, Sucu DÖ, Turgut G, Uğurlu K. Microsurgical reconstruction in pediatric patients: a series of 30 patients. Ulus Trauma Acil Cerr Derg. 2013;19:411-416. [Crossref]

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6 Runyan CM, Sharma V, Staffenberg DA, Levine JP, Brecht LE, Wexler LH, et al. Jaw in a day-state of the art in maxillary reconstruction. J Craniofac Surg. 2016; 27:2101-2104. [Crossref]

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8 Khechoyan DY, Saber NR, Burge J, Fattah A, Drake J, Forrest CR, Phillips JH. Surgical outcomes in craniosynostosis reconstruction: The use of prefabricated templates in cranial vault remodeling. J Plast Reconstr Aesthet Surg. 2014; 67:9-16. [Crossref]

Funding statement No funding

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