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ACTA OTO-LARYNGOLOGICA CASE REPORTS 2023, VOL. 8, NO. 1, 22–28 https://doi.org/10.1080/23772484.2023.2174439 CASE REPORT Odontogenic myxoma involving the right nasal cavity, orbital floor, and skull base in a 20-year-old woman: Removal and review of the literature a,b a,b a,b a,c a,b a,b a,b Danlin Huang , Fei Liu , Junyi Liang , Xiao Xing , Xingsha Wu , Shuai Yang , Xinfeng Wei and a,b Shuo Li Department of Otolaryngology, The 6th Affiliated Hospital of Shenzhen University Health Science Center, Shenzhen, China; Department of Otolaryngology, Huazhong University of Science and Technology Union Shenzhen Hospital, Shenzhen, China; Department of Otolaryngology, Affiliated Shenzhen Sixth Hospital of Guangdong Medical University, Shenzhen, China ABSTRACT ARTICLE HISTORY Received 31 August 2022 Odontogenic myxoma (OM) is a rare benign tumor in the jaws. It is considered locally aggres- Revised 25 January 2023 sive but non-metastatic. Here, we report a case of Odontogenic myxoma observed at a relatively Accepted 25 January 2023 rare site, which occurs in the maxillary posterior interdental region and maxillary sinus, as well as involves the right nasal cavity, orbital floor, and skull base. A 20-year-old young female KEYWORDS patient was referred to institution for right-sided nasal congestion and facial swelling. The right Odontogenic myxoma; inferior nasal tract and common nasal tract of the patient were blocked with a reddish neo- operative therapy plasm. The clinical history and preoperative examination and iconography findings were indica- tive of an odontogenic cyst. The patient received Right-sided nasal sinus tumor resection via end nasal endoscopic anterior lacrimal saphenous fossa combined with Kollu’s approach. The postoperative pathological findings were suggestive of an Odontogenic myxoma. Conservative surgical treatment is less surgically invasive, more acceptable to patients, and results in a better quality of survival. Abbreviations: OM: odontogenic myxoma; CT: computed tomography; MRI: magnetic reson- ance imaging Introduction College of Stomatology at Sichuan University con- ducted a retrospective analysis of 1642 cases of odon- Odontogenic myxoma (OM) was firstly described by togenic tumors between 1952 and 2004 and Thoma and Goldman in 1947, and they attributed it discovered that only 4.6 per cent of odontogenic to mesenchymal and/or odontogenic ectodermal mes- tumors in the Chinese population were caused by enchyme [1,2]. OM is a rare benign tumor in the OM [6]. Argentinean teenagers and Egyptian daiquiri jaws and is mainly seen in the tooth-bearing zone [3]. had a higher incidence of OM, making up 8.5% of It is considered locally aggressive but non-metastatic. individuals with odontogenic tumors [7,8] and in a The maxilla OM was more aggressive than the man- California survey of the United States performed that dible OM. Maxillary Odontogenic myxomafrequently accounted for 2.2% [9]. In this study, we reported a affects the paranasal sinus [4,5]. The dispersed stellate and spindle-shaped cells in an abundant mucus-like 20 years old female with OM, which occurs in the extracellular matrix is one of the outstanding charac- maxillary posterior interdental region and maxillary teristics of OM, which may contain odontogenic epi- sinus, as well as involves the right nasal cavity, orbital thelial cells [3]. The overall incidence of OM is floor, and skull base. reported to be between 0.2% and 17.7% in all odonto- genic tumors. The age at OM diagnosis is usually Case report between 20 to 39 years, especially in females. According to previous studies, the overall incidence The patient, a 20-year-old female, was presented to the of OM was an ethnic disparity. The West China hospital with the primary complaint of ‘right-sided CONTACT Shuo Li shuoli@email.szu.edu.cn Department of Otolaryngology, The 6th Affiliated Hospital of Shenzhen University Health Science Center, Shenzhen, China These authors contributed equally to this work. 2023 The Author(s). Published by Informa UK Limited, trading as Taylor & Francis Group. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ACTA OTO-LARYNGOLOGICA CASE REPORTS 23 nasal congestion for 2 years, exacerbated by facial swel- the right nasal cavity inward, and had a tooth visible at ling for 6 months.’ The patient developed right-sided the top. The cyst’s CT value was about 26 HU, and its nasal congestion 2 years ago for no apparent reason, density was more uniform, with intact sinus wall bone and it steadily intensified, accompanied by a runny (Figure 1) A MRI of the sinuses revealed a soft tissue nose, no headache, exophthalmos, and facial swelling. mass with a low signal in T1WI and a high signal in The patient had been visited at numerous other hos- T2W that was about 5.2 cm by 4.7 cm and was located pitals and was discovered to have a right paranasal in the right maxillary sinus, sieve sinus, and middle sinus mass by nasal endoscopy and CT evaluation of and lower nasal passages. The surrounding bone was the paranasal sinuses, and surgery was indicated. The destroyed, and the mass was enlarged adjacent to the aforementioned symptoms have been worse over the ostium of the maxillary sinus. The lesion affected the last six months and are now accompanied by facial maxilla, and protruded medially into the nasal cavity, swelling, protrusion of ophthalmoparesis, soreness absorbing the middle and inferior turbinate. Enhanced when pressing on the face and eye, self-conscious scan: The mass showed markedly heterogeneous ‘gyrus- slightly decreased right visual acuity, occasionally like’ enhancement (Figure 2). For diagnostic and thera- blurred vision, no movement restriction or diplopia, peutic purposes, we surgically resected the lesion by no loss of smell, etc. Examination: The right nasal performing a transnasal endoscopic anterior lacrimal inferior and common nasal tracts are filled with a light fossa combined with Collis’s approach to the right nasal red tumor; the tumor has compressed and flattened sinus tumor (Figure 3), and a histological examination the inferior turbinate medially and superiorly; the of the resected tissue was conducted. Macroscopically, mass’s surface is smooth, and its root cannot be seen. the specimen was off-white in color and consisted of CT and MRI of the paranasal sinuses were done in soft and hard tissues. Microscopic examination showed order to determine the size, origin, and kind of tumor. a polyp-like mass with scattered epithelial strips of The right maxilla had a large cystic low-density lesion odontogenic origin visible in the mucus-like intersti- that was significantly distended, unevenly enlarged, tium, with no obvious heterogeneity, and the lesion was convened upward into the maxillary sinus significantly considered to be an Odontogenic myxoma tumor thinned adjacent sinus wall bone structure, obstructed (Figure 4). Due to the excessive enormous lacuna, we Figure 1. CT of the paranasal sinuses shows a large cystic hypodense lesion visible in the right maxilla, which projects upward and occupies the maxillary sinus to the point where the bone structure of the adjacent sinus wall is significantly thinned and obstructs the right nasal cavity inward, with a tooth visible on top. 24 D. HUANG ET AL. Figure 2. MRI of sinus showed a soft tissue mass of about 5.2 cm 4.7 cm in size in the right maxillary sinus, septal sinus and middle and lower nasal passages, with low signal in T1WI and high signal in T2W. Enhancement scan: the mass showed signifi- cant heterogeneous ‘brain gyrus-like’ enhancement. employed Vaseline gauze and an expansion sponge to preoperative diagnosis should be validated by patho- logic biopsy. fill the nasal cavity and systemic intravenous antibiotic Because the mucinous tumor in this patient had infusion to reduce the dead space and avoid postopera- occupied the entire maxillary sinus cavity and was bulg- tive infection. ing toward the nasal septal surface, a trans maxillary sinus conventional opening approach was unable to expose the mass. In combination with CT and MRI, Discussion the right Odontogenic myxoma tumor occupied the OM is commonly observed in the area of the mouth entire maxillary sinus cavity; in order to better expose where teeth are present. The maxilla OM was more the mass and gain room for manipulation, we per- aggressive than the mandible OM and more likely to formed the procedure using a transnasal endoscopic progress into the sinuses. For CT and MRI could not anterior lacrimal fossa approach. A greyish-white mass provide microcosmic details, the diagnosis of OM is that could not be reached by instruments was still pre- often dependent on pathology. sent after using the 70-degree nasal endoscope to In this case, CT exhibited a large cystic hypodense explore the anterior medial aspect of the maxillary sinus lesion in the right maxilla, with an intracapsular CT and the floor wall. so the mass was completely removed value of about 26 HU. Because of the lesion, the right endoscopically in combination with the lacrimal gin- maxilla was clearly distended and unevenly enlarged, gival sulcus approach. The postoperative pathology was convexed upward and occupied the maxillary sinus. reported as an Odontogenic myxoma tumor. The bone structure of the adjacent sinus wall was sig- Mucinous tumors are often removed surgically. nificantly thinned and obstructed the right nasal cav- The tumor has no envelope and is locally infiltrative, ity inward as a result of the lesion. so if the procedure is not removed entirely, it may According to the previous study, OM is a rare come back. When the tumor is big, a partial mandible benign tumor and had overlapped clinical features or maxilla excision is the best surgical option to min- with other diseases, which leads to being easily con- imize recurrence. There are no clear guidelines for fused with other jaw tumors. However, the lesion the surgical treatment of Odontogenic myxoma causes the CT to show a cystic component with low tumors. Saalim et al. [11] performed a statistical ana- signal separation in the focal area, which suggests a lysis of papers related to the treatment of 39 patients high possibility of OM [10]. Thepreoperativeclinical with Odontogenic myxoma tumors, Treatments were data could not provide efficient evidence to identify the classified as conservative (curettage, enucleation with patient as an Odontogenic myxoma tumor. Besides, curettage, excision curettage, and excision) and resec- this cystic hypodense lesion was more likely to be diag- tion. They classified curettage, enucleation with curet- nosed as an odontogenic cyst according to its features tage, excision curettage, and excision as ‘conservative in preoperative CT. To exclude intraosseous cystic treatment’. During a mean follow-up period of 10 years, lesions, central giant cell lesions, ameloblastomas, odon- the overall recurrence rate was 5 out of 39 patients togenic keratotic cysts, or other osteolytic lesions, the (13%), the difference in the recurrence rate of ACTA OTO-LARYNGOLOGICA CASE REPORTS 25 Figure 3. (A) Transnasal endoscopic right anterior lacrimal saphenous approach. A1: The white dashed line shows the curved inci- sion at the anterior edge of the inferior turbinate head by the anterior lacrimal saphenous approach. A2: grinding drill to grind away the bone of the inner wall of the maxillary sinus from anterior to posterior. A3: Greyish-white papillary neoplasm in the maxillary sinus. (B) Excision of maxillary sinus massþ opening of the right inferior nasal tract. B1: a molar located in the postero- superior wall of the maxillary sinus and encased in a greyish-white neoplasm. B2: After exfoliation of the swelling, the bone of the orbital floor wall was seen to be significantly thinned, and the blue-purple orbital tissue could have peered through the bone wall with the perichondrium intact and bluish. B3: a few bony defects in the posterior external wall of the maxillary sinus, with the dura mater of the inferior temporal fossa visible. (C) Endoscopic transtibial gingival sulcus section Lu’s approach. C1: an approximately 3-cm-long incision in the right labiogingival sulcus. C2: Biting forceps to remove part of the bone of the anterior maxillary sinus wall until the floor wall of the maxillary sinus and the mass are completely exposed. C3: closure of the anterior aspect of the inferior turbinate and the lateral wall of the nasal cavity with 6-0 sutures. IT: inferior turbinate; SP: nasal septum; MS: maxillary sinus; LG: labiogingival groove; OF: orbital floor. Odontogenic myxoma tumors by conservative resec- uninvolved surrounding tissue, or even peripheral oste- tion or radical surgery was not statistically significant otomy, with no way to preserve vital structures and (p¼ 0.51), and maxillary lesions were more likely to maintain oral function. Boffano et al. [13] suggested recur than mandibular lesions. Zanetti et al. [12]strongly conservative treatment when Odontogenic myxoma recommended conservative treatment with an exten- tumors are less than 3 cm in diameter, while segmental sive scraping of normal tissue or extensive apparently resection and immediate reconstruction are preferred 26 D. HUANG ET AL. Figure 4. We can see odontogenic epithelial stripes. Figure 5. The nasal endoscopy A 45 endoscopic examination of the right anterior lacrimal saphenous fossa and the maxillary sinus at 3 months following surgery revealed excellent mucosal development and no indications of tumor growth. The right nasal cavity was still unobstructed. Figure 6. The CT examination of the paranasal sinuses was repeated 3 months after surgery, showing enlargement of the right maxillary sinus cavity, thinning of the sinus wall, and absence of the medial wall part and middle turbinate, showing postopera- tive changes; no obstruction or narrowing of the nasal cavity was seen now. No recurrence was found in the paranasal sinus MRI at 9 months after surgery. for larger patients. However, Yoko Kawase-Koga et al. physiology and aesthetics as much as possible while [14] performed the removal and extensive scraping of minimizing the risk of recurrence. However, radical the surrounding normal tissue in a patient with an surgery is still required after conservatively treating Odontogenic myxoma tumor larger than 3 cm in OM recurrence [15,16]. We surgically resected the lesion by performing a diameter without recurrence at 10 years of follow-up. Conservative treatments are much less invasive, so we transnasal endoscopic anterior lacrimal fossa combined can use surgical approaches that preserve normal with Collus’s approach to the right nasal sinus tumor. ACTA OTO-LARYNGOLOGICA CASE REPORTS 27 Figure 7. On the first postoperative day the patient showed swelling at the right nasolabial fold. And a postoperative image of the patient 3 months after the removal of the tumour. During the 3-month postoperative follow-up, this patient had a repeat nasal endoscopy (Figure 5) and CT of the paranasal sinuses (Figure 6), and no clinical or imaging signs of recurrence were found. The patient’s maxillofacial swelling and nasal congestion were significantly better than before (Figure 7). We will follow up on this case for a long time. According to the nasal endoscopic image, the tumor treatments is less surgically invasive, more acceptable was totally removed and the surrounding normal tissue to patients, and own a better quality of survival. The mucosa was extensively scraped to preserve part of the clinical management of this case, as well as our com- middle turbinates, skull base, orbital floor wall, and prehensive evaluation of the literature, could assist bone wall in the maxillary sinus. Due to the large inform treatment decisions for odontogenic myxoma, cumulative range of Odontogenic myxoma tumors in with the goal of reducing the risk of recurrence while this patient, we opted for conservative treatment . using a less intrusive surgical technique wherever pos- Because the segmental resection and reconstruction are sible. Although conservative and marginal surgery has more traumatic to the organism, and the patient is now proven effective, the risk of recurrence remains con- only 20 years old, a female patient, we used a minimally siderable and long-term follow-up is indispensable. invasive surgical approach as much as possible while More evidence of long-term outcomes after conserva- ensuring a reduced risk of recurrence. tive surgery for OM is needed. Eman Kheir et al. [17] Performed MRI was effective in displaying thetrueextension andcontents of OMs. Informed consent CT scans demonstrated the extensions of OMs, expan- sion, growth pattern, and rendered it possible to com- The patient has provided written informed consent for pub- pare density of OM with that of surrounding muscles. lication of the case. The datasets generated during and/or analyzed during On MRI, the walls of the tumors and patterns of the the current study are available from the corresponding growth were clearly depicted. Although the tumors dis- author on reasonable request. played predominantly a smooth wall, however in focal areas scalloping, crevices, budding, and/or lobulations were detected. These features supported the distinctive Disclosure statement infiltrative nature of the tumor. Three months after sur- The authors have no conflicts of interest to disclose. gery, we reviewed sinus CT, which showed no evidence of tumor growth. The same results were obtained with sinus MRI 9 months after surgery. Funding This work was supported by the The Science and Technology Foundation of Shenzhen (JCYJ20210324112607020). Conclusion Odontogenic myxoma tumors are clinically treated ORCID with conservative and marginal surgery, both with low recurrence rates. However, conservative surgical Shuo Li http://orcid.org/0000-0001-6025-3948 28 D. HUANG ET AL. 1,088 cases from Northern California and compari- References son to studies from other parts of the world. J Oral [1] Neville BW, Damm DD, Allen CM, et al. Maxillofac Surg. 2006;64(9):1343–1352. Odontogenic cysts and tumors. In: Oral and max- [10] Xue Z, Jingbo W, Xiaofeng T. Spiral CT and MRI illofacial pathology, 3rd ed. Philadelphia: WB manifestations of Odontogenic Myxoma tumors of Saunders; 2010. p. 678–740. the Jaws. Chin Med Imag Technol. 2020;36(01):42–45. [2] Noffke CE, Raubenheimer EJ, Chabikuli NJ, et al. [11] Saalim M, Sansare K, Karjodkar FR, et al. 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Acta Oto-Laryngologica Case Reports – Taylor & Francis
Published: Dec 31, 2023
Keywords: Odontogenic myxoma; operative therapy
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