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Case Report

Long-Term Follow-Up after Conservative Surgical Treatment of Odontogenic Myxoma: A Case Report and Literature Review

Hitoshi Sato , Yuji Kurihara, Sunao Shiogama , Kotaro Saka, Yuya Kurasawa, Masakatsu Itose, Atsutoshi Yaso, and Tatsuo Shirota

Department of Oral and Maxillofacial Surgery, School of Dentistry, Showa University, 2-1-1 Kitasenzoku, Ota-ku, Tokyo 145-8515, Japan

Correspondence should be addressed to Hitoshi Sato; Received 2 January 2019; Accepted 29 January 2019; Published 11 February 2019 Academic Editor: Eugenio Maiorano

Copyright © 2019 Hitoshi Sato et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Odontogenic myxoma (OM) is a relatively rare, benign odontogenic tumor with locally aggressive behavior, but it is a nonmetastasizing neoplasm of the jaw bones. Although radical resection with an appropriate surgical margin is recommended, emerging evidence has suggested that a more conservative approach will result in acceptable recurrence rates with less morbidity if careful long-term follow-up is provided. A 56-year-old Japanese man with odontogenic myxoma of the left mandible was conservatively treated by surgical enucleation and curettage because he desired functional and cosmetic preservation. During a follow-up period of 100 months, the patient has remained clinically and radiologically free of recurrence. As far as we can ascertain, 20 reports published after 1990 described 37 patients with mandibular OM that had been treated by conservative surgery. Tumors recurred during a mean follow-up of49 2 ± 42 8 months in 7 (18.9%) patients, and only one of five patients who were followed up for over 100 months developed recurrence. The rate of recurrence decreased from 24.0% to 8.3% when follow-up exceeded 60 months. Although enucleation and curettage have proven effective, the risk of recurrence remains considerable and long-term follow-up is indispensable. More evidence of long-term outcomes after conservative surgery for OM is needed.

1. Introduction

Odontogenic myxoma (OM) is a benign, locally invasive, aggressive, nonmetastasizing neoplasm of the jaw bones. It is the third most common odontogenic lesion with an annual incidence of ~0.07 new patients per million people [1, 2].

Although OM is a benign tumor, radical treatment is needed because the rate of local recurrence ranges from 10% - 33%

[3-5]. Surgical resection with a minimum bone margin of 1 cm has been advocated [6-9], but emerging evidence sug- gests that a more conservative surgical approach will result in acceptable recurrence rates with less patient morbidity if follow-up can be over the long term [7, 10-12]. However, little has been reported about the actual long-term follow-up of patients with OM after conservative surgery.

Here, we describe a mandibular odontogenic myxoma that was treated by conservative surgery and followed up for 100 months thereafter.

2. Case Report

2.1. Patient Information. A 56-year-old Japanese man was referred from a dental clinic for further examination of a radiolucent finding on the left side of his mandible in January 2010. The patient reported having no symptoms in his mouth including the left mandible area. His medical and dental history was noncontributory. An extraoral examina- tion upon initial presentation revealed unremarkable findings and no complaints of paresthesia. An intraoral examination also confirmed the absence of redness and tender swelling of the left mandibular mucosa (Figure 1). Panoramic radiog- raphy revealed an extensive multilocular radiolucent area with imprecise borders and a “soap bubble appearance”

(Figure 2). Computed tomography showed an approximately 39 × 19 × 11 mm tumor that extended to the roots of four teeth (#33 - 36; Figures 3(a) and 3(b)). We considered that the odontogenic tumor was benign and an incisional biopsy

Volume 2019, Article ID 1634842, 6 pages


was performed under local anesthesia. The histopathological findings revealed loosely arranged spindle-shaped cells in a myxoid fibrous stroma, indicating a clinical diagnosis of an odontogenic tumor. Segmental resection of the mandible was planned. The patient was given repeated and careful explanations about the high likelihood of recurrence, but he insisted upon a more conservative approach as he desired functional and cosmetic preservation. Conservative surgery then proceeded under general anesthesia after endodontic treatment of #33 – 36 was completed. The surgery consisted of extracting the second premolar from the left mandible, followed by total enucleation and vigorous curettage of the bone (Figure 4(a)). The surgical specimen (Figure 4(b)) revealed apparently benign, spindled-shaped cells in a loose and abundant mucoid stroma (Figures 5(a) and 5(b)). These findings confirmed the diagnosis of odontogenic myxoma.

The immediate postoperative period and wound healing were uneventful. The patient underwent monthly clinical

examinations for the first year thereafter, then every two months during the second year. Panoramic X-rays were obtained every three months for the first two years. Annual follow-up for eight years included panoramic X-rays and CT imaging (Figures 6(a) and 6(b), respectively), which showed no clinical or radiological signs of recurrence.

3. Discussion

Odontogenic myxomas are very rare (<10% all odontogenic tumors) benign tumors of the ectomesenchymal and/or mes- enchymal origin [13, 14]. They are locally invasive and slow-- growing, and their pathological characteristics in the tooth-bearing areas of the mandible and maxilla are well de fined [13-17]. The radiographic features are described as those of a multilocular lesion with a “soap bubble” or “spider web” appearance [14-17]. Such lesions are often discovered incidentally during routine dental checkups, and about 60%

Figure 1: Intraoral photography before surgery. The image shows no symptoms such as redness or swelling of mucosa in the mandible.

Figure 2: Panoramic dental radiography before surgery. The image shows extensive multilocular radiolucent area with imprecise borders and

“soap bubble appearance”.

(a) (b)

Figure 3: Computed tomography (CT) image before surgery. The axial (a) and coronal (b) CT images show tumor infiltration of cortical bone extending to the inferior mandibular border.


of patients are in their second or third decade of life [4, 18].

Although the present patient was asymptomatic, panoramic radiography of the left mandible revealed an extensive radio- lucent and multilocular area with imprecise borders that extended from the root of tooth #33 to that of tooth #36.

The treatment strategy for OM remains controversial.

Because OM is locally aggressive and it can potentially cause extensive bony destruction, the treatment of choice seems

to be radical surgery such as segmental resection. Indeed, complete surgical removal with curettage and peripheral ostectomy alone seems insufficient since OM is not encapsulated and myxomatous tissue in filtrates adjacent bone [3, 6-9]. Thus, the only initial treatment option for extended OM in principle is surgical resection because of the high risk of recurrence which reportedly ranges from 10% to 33% [4, 5]. In addition, recurrence rates can reach

(a) (b)

Figure 5: Pathophysiological findings of stained specimen. Hematoxylin and eosin stain ×100 (a) and ×400 (b) magnification.

(a) (b)

Figure 4: Surgical procedures and resected specimen. The total enucleation and wide curettage of the surrounding bone (a) and resected specimen (b).

(a) (b)

Figure 6: Images of the jaw at 96 months after surgery. The panoramic dental radiograph (a) and computed tomography image (b) show no signs of recurrence.


25% after simple enucleation and curettage alone [19, 20]. On the other hand, some reports described that the choice of cur- rent recommended therapy depends on the size of the lesion and its nature and behavior between curettage and radical excision [3, 7]. In the present case, CT had shown that the tumor included the #33 - #36 apex and extended to the inferior border of the mandible. Therefore, radical resec- tion such as block resection for mandible was strongly rec- ommended. However, the patient rejected this strategy because he desired functional and cosmetic preservation,

and in fact, conservative treatment with enucleation, curet- tage, and marginal resection would confer several advantages over the radical approach. It is substantially less invasive, it can be surgically implemented via an intraoral approach, and it offers the possibility of preserved nerve function and aesthetics and a shorter stay in the hospital. Recent evidence suggests that a more conservative approach will result in acceptable recurrence rates with less morbidity to patients if long-term follow-up is provided [7, 10-12]. Table 1 describes 20 reports of 37 patients with that was treated by conservative

Table 1: Clinical reports of mandibular odontogenic myxoma published after 1990.

No. Author Year Age Sex Follow-up period (months) Treatment Recurrence Size (mm)

1 Oliveira et al. [21] 2018 9 F 6 E/C None NA

2 Albanese et al. [22] 2012 25 F 6 E None 21 2 × 47 6

3 Mauro et al. [23] 2012 6 M 6 E/C None NA

4 Subramaniam et al. [7] 2016 16 — 7 M None NA

5 Shivashankara et al. [24] 2017 13 M 12 E/N None 40 × 20

6 Subramaniam et al. [7] 2016 18 — 12 E None NA

7 Miranda Rius et al. [25] 2013 55 M 12 E/C None 33 × 28

8 Hammad et al. [26] 2016 45 F 13 M None 50 × 30

9 Francisco et al. [27] 2017 9 F 14 E/C Recurrence NA

10 Francisco et al. [27] 2017 12 F 16 E/C Recurrence NA

11 Sumi et al. [28] 2000 48 M 22 E/C None 70 × 25 × 15

12 Mittal et al. [29] 2016 48 F 24 E/C Recurrence 25 × 20

13 Lin and Basile [30] 2010 25 F 24 E None NA

14 Lo Muzio et al. [4] 1996 21 M 24 E/C None NA

15 Lo Muzio et al. [4] 1996 28 M 24 E/C Recurrence NA

16 Bucci et al. [31] 1993 28 M 24 E/C None 43 × 40

17 Francisco et al. [27] 2017 7 F 26 E/C None NA

18 Francisco et al. [27] 2017 15 F 27 E/C Recurrence NA

19 Lo Muzio et al. [4] 1996 16 F 31 E/C None NA

20 Francisco et al. [27] 2017 30 F 34 E/C None NA

21 Boffano et al. [11] 2011 38 M 38 E/C None 25

22 Boffano et al. [11] 2011 42 F 40 E/C None 30

23 Boffano et al. [11] 2011 20 M 42 E/C None 20

24 Rajasekhar et al. [32] 2008 17 F 48 M None 70 × 30

25 Lo Muzio et al. [4] 1996 22 F 48 E/C Recurrence NA

26 Takahashi et al. [6] 2018 37 F 73 E/C None 40 × 19 × 12

27 Chaudhary et al. [33] 2015 25 F 84 E/C/M None NA

28 Li et al. [18] 2006 7 M 84 E/C None NA

29 Li et al. [18] 2006 32 M 84 E/C None NA

30 Lo Muzio et al. [4] 1996 65 F 84 E/C None NA

31 Francisco et al. [27] 2017 17 M 85 E/C None NA

32 Francisco et al. [27] 2017 11 F 98 E/C None NA

33 Present case 56 M 100 E/C None 39 × 19 × 11

34 Francisco et al. [27] 2017 27 F 117 E/C None NA

35 Kawase-Koga et al. [10] 2014 40 M 120 E/C None 40 × 30 × 15

36 Li et al. [18] 2006 37 M 132 C None NA

37 Kansy et al. [17] 2012 33 F 180 M Recurrence NA

Abbreviations: M: male, F: female, E: enucleation, C: curettage, M: marginal resection, and NA: not available.


surgery [4, 6, 7, 10, 11, 17, 18, 21-33]. The mean age of the patients was 27 0 ± 15 2, and 20 (54%) were female. The tumors in almost all of them were managed by enucleation and curettage and recurred in 7 (18.9%). One and six of the procedures with recurrence had undergone marginal resec- tion and enucleation with curettage, respectively. The patients were followed up for a mean of 49 2 ± 42 9 (range, 2 – 180) months. Including the present patient, only five have been followed up for over 100 months and the tumor recurred in one of them. The recurrence rate among all patients who were treated by conservative surgery was 19.0%. This rate is relatively lower than that previously described [19, 20]. Furthermore, our investigation of the lit- erature indicated that the rate of recurrence decreases from 24.0% to 8.3% when the follow-up period exceeds 60 months.

The main reason for recurrence is thought to be incom- plete removal rather than the intrinsic biological behavior of the OM [34]. Although the tendency is towards a more conservative surgical approach for children and a more rad- ical approach for adults, Kansy et al. does not support this management strategy because the recurrence rates between enucleation and segmental resection are similar [17]. Tumor size has recently been considered to be a factor in the choice of a radical or more conservative surgical approach [4, 10, 11]. Conservative surgical procedures such as enucleation and curettage are recommended when the diameter of OM is <3 cm, whereas a radical approach such as segmental resec- tion with immediate reconstruction is preferred when patients have larger tumors [11]. The conservative surgical recommendation is to enucleate a lesion with wide curettage of normal tissue or a generous amount of apparently intact tissue or even marginal resection of the mandible [12]. This approach has the advantage of preserving vital structures and maintaining oral function, and it could be applied to treat OM that recur after simple surgery [12]. However, one patient who developed recurrent OM 15 years after the initial procedure including tumor resection with the preservation of mandibular continuity has been described [17]. More radical surgery is inevitable for a large number of patients due to a tendency towards more extensive OM with signi ficant destruction of key structures [17, 27]. Moreover, the rate of OM recurrence remains vague because few reports described long-term follow-up after conservative surgery. Thus, more evidence about long-term outcomes after conservative surgi- cal treatment of OM is needed.

4. Conclusions

A conservative surgical approach comprising enucleation and curettage can be effective for OM management. Recur- rence rates decreased from 24.0% to 8.3% among patients who were treated with conservative surgery and followed up for over 60 months. The risk of recurrence is likely to be considerable, and long-term follow-up is indispensable for the conservative management of OM.

Conflicts of Interest

The authors have no conflicts of interest to declare.


[1] E. W. Odell and K. Adebiyi,“Odontogenic myxoma/myxofi- broma,” in El-Naggar AK, J. K. C. Chan, Ed., pp. 229-230, WHO Classification of Head and Neck Tumors. IARC Press, Lyon, 2017.

[2] E. N. M. Simon, M. A. W. Merkx, E. Vuhahula, D. Ngassapa, and P. J. W. Stoelinga,“Odontogenic myxoma: a clinicopatho- logical study of 33 cases,” International Journal of Oral and Maxillofacial Surgery, vol. 33, no. 4, pp. 333–337, 2004.

[3] Y. Leiser, I. Abu-El-Naaj, and M. Peled, “Odontogenic myxoma-a case series and review of the surgical management,” Journal of Cranio-Maxillo-Facial Surgery, vol. 37, no. 4, pp. 206–209, 2009.

[4] L. L. Muzio, P. F. Nocini, G. Favia, M. Procaccini, and M. D.

Mignogna, “Odontogenic myxoma of the jaws: a clinical, radiologic, immunohistochemical, and ultrastructural study,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 82, no. 4, pp. 426–433, 1996.

[5] B. O. Abiose, H. A. Ajagbe, and O. Thomas,“Fibromyxomas of the jawbones–a study of ten cases,” British Journal of Oral &

Maxillofacial Surgery, vol. 25, no. 5, pp. 415–421, 1987.

[6] Y. Takahashi, K. Tanaka, H. Hirai, E. Marukawa, T. Izumo, and H. Harada,“Appropriate surgical margin for odontogenic myxoma: a review of 12 cases,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, vol. 126, no. 5, pp. 404–408, 2018.

[7] S. S. Subramaniam, A. A. Heggie, R. Kumar, and J. M. Shand,

“Odontogenic myxoma in the paediatric patient: a review of eight cases,” International Journal of Oral and Maxillofacial Surgery, vol. 45, no. 12, pp. 1614–1617, 2016.

[8] N. Kumar, M. Kohli, S. Pandey, and P. Agarwal,“Odontogenic myxoma,” Journal of Maxillofacial and Oral Surgery, vol. 13, no. 2, pp. 222–226, 2014.

[9] T. J. King III, J. Lewis, L. Orvidas, and D. Kademani,“Pediatric maxillary odontogenic myxoma: a report of 2 cases and review of management,” Journal of Oral and Maxillofacial Surgery, vol. 66, no. 5, pp. 1057–1062, 2008.

[10] Y. Kawase-Koga, H. Saijo, K. Hoshi, T. Takato, and Y. Mori,

“Surgical management of odontogenic myxoma: a case report and review of the literature,” BMC Research Notes, vol. 7, no. 1, p. 214, 2014.

[11] P. Boffano, C. Gallesio, A. Barreca, F. A. Bianchi, P. Garzino-Demo, and F. Roccia,“Surgical treatment of odon- togenic myxoma,” Journal of Craniofacial Surgery, vol. 22, no. 3, pp. 982–987, 2011.

[12] L. S. S. Zanetti, B. M. de Carvalho, I. R. Garcia, L. A. P. de Barros, P. L. dos Santos, and A. C. R. de Moraes Ferreira,

“Conservative treatment of odontogenic myxoma,” The Journal of Craniofacial Surgery, vol. 22, no. 5, pp. 1939–

1941, 2011.

[13] P. J. Slootweg and A. R. M. Wittkampf,“Myxoma of the jaws.

An analysis of 15 cases,” Journal of Maxillofacial Surgery, vol. 14, no. 1, pp. 46–52, 1986.

[14] G. Martínez-Mata, A. Mosqueda-Taylor, R. Carlos-Bregni et al.,“Odontogenic myxoma: clinico-pathological, immuno- histochemical and ultrastructural findings of a multicentric series,” Oral Oncology, vol. 44, no. 6, pp. 601–607, 2008.

[15] S. Singaraju, S. P. Wanjari, and R. N. Parwani,“Odontogenic myxoma of the maxilla: a report of a rare case and review of the literature,” Journal of Oral and Maxillofacial Pathology, vol. 14, no. 1, pp. 19–23, 2010.


[16] C. E. E. Noffke, E. J. Raubenheimer, N. J. Chabikuli, and M. M.

R. Bouckaert,“Odontogenic myxoma: review of the literature and report of 30 cases from South Africa,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 104, no. 1, pp. 101–109, 2007.

[17] K. Kansy, P. Juergens, Z. Krol et al.,“Odontogenic myxoma:

diagnostic and therapeutic challenges in paediatric and adult patients–a case series and review of the literature,” Journal of Cranio-Maxillo-Facial Surgery, vol. 40, no. 3, pp. 271–276, 2012.

[18] T. J. Li, L. S. Sun, and H. Y. Luo,“Odontogenic myxoma: a clinicopathologic study of 25 cases,” Archives of Pathology &

Laboratory Medicine, vol. 130, no. 12, pp. 1799–1806, 2006.

[19] D. C. Zimmerman and D. C. Dahlin,“Myxomatous tumors of the jaws,” Oral Surgery, Oral Medicine, and Oral Pathology, vol. 11, no. 10, pp. 1069–1080, 1958.

[20] R. E. Barros, F. V. Dominguez, and R. L. Cabrini,“Myxoma of the jaws,” Oral Surgery, Oral Medicine, and Oral Pathology, vol. 27, no. 2, pp. 225–236, 1969.

[21] S. V. Oliveira, A. C. Rocha, M. M. Ceccheti, C. de Barros Gallo, and F. A. Alves,“Odontogenic myxoma in a child treated with enucleation and curettage,” Autopsy & Case Reports, vol. 8, no. 3, article e2018042, 2018.

[22] M. Albanese, P. F. Nocini, A. Fior et al.,“Mandibular recon- struction using fresh frozen bone allograft after conservative enucleation of a mandibular odontogenic myxoma,” Journal of Craniofacial Surgery, vol. 23, no. 3, pp. 831–835, 2012.

[23] A. Mauro, L. Lipari, S. Tortorici, A. Leone, A. Gerbino, and M. Buscemi,“Expression of MMP-2 and MMP-9 in odonto- genic myxoma in a child: report of a clinical case,” Odontology, vol. 101, no. 2, pp. 233–238, 2013.

[24] C. Shivashankara, M. Nidoni, S. Patil, and K. T. Shashikala,

“Odontogenic myxoma: a review with report of an uncommon case with recurrence in the mandible of a teenage male,” The Saudi Dental Journal, vol. 29, no. 3, pp. 93–101, 2017.

[25] J. Miranda Rius, A. Nadal, E. Lahor, B. Mtui, and L. Brunet,

“Unusual presentation of localized gingival enlargement associated with a slow-growing odontogenic myxoma,” Inter- national Journal of Oral Science, vol. 5, no. 3, pp. 172–175, 2013.

[26] H. M. Hammad, Y. M. Hasen, A.-A. M. Odat, A. M. Mikdadi, and R. A. Safadi,“Odontogenic myxoma with diffuse calcifica- tions: a case report and review of a rare histologic feature,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, vol. 122, no. 4, pp. e116–e124, 2016.

[27] A. L. Francisco, T. C. Chulam, F. O. Silva et al.,“Clinicopath- ologic analysis of 14 cases of odontogenic myxoma and review of the literature,” Journal of Clinical and Experimental Den- tistry, vol. 9, no. 4, pp. e560–e563, 2017.

[28] Y. Sumi, O. Miyaishi, K. Ito, and M. Ueda,“Magnetic reso- nance imaging of myxoma in the mandible: a case report,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 90, no. 5, pp. 671–676, 2000.

[29] Y. Mittal, A. Chugh, K. G. Varghese, S. Dwivedi, and V. Goyal,

“Management of recurrent odontogenic myxoma of mandible:

a clinical case report,” Journal of Clinical and Diagnostic Research, vol. 10, no. 10, pp. ZD30–ZD31, 2016.

[30] Y. L. Lin and J. R. Basile,“A case of odontogenic myxoma with unusual histological features mimicking afibro-osseous pro- cess,” Head and Neck Pathology, vol. 4, no. 3, pp. 253–256, 2010.

[31] E. Bucci, L. L. Muzio, M. D. Mignogna, and G. de Rosa,

“Odontogenic myxoma: report of a case with peculiar fea- tures,” Journal of Oral and Maxillofacial Surgery, vol. 49, no. 1, pp. 91–94, 1991.

[32] G. Rajasekhar, M. Mushtaq, N. G. Vura, R. Shekar, and S. Kumar,“Condyloma acuminatum associated with odonto- genic myxoma: a case report,” Journal of Maxillofacial and Oral Surgery, vol. 8, no. 4, pp. 384–387, 2009.

[33] Z. Chaudhary, P. Sharma, S. Gupta, S. Mohanty, M. Naithani, and A. Jain,“Odontogenic myxoma: report of three cases and retrospective review of literature in Indian population,” Con- temporary Clinical Dentistry, vol. 6, no. 4, pp. 522–528, 2015.

[34] J. G. Batsakis, “Myxomas of soft tissues and the facial skeleton,” Annals of Otology, Rhinology & Laryngology, vol. 96, no. 5, pp. 618-619, 1987.



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