Conservative management of extensive maxillary adenomatoid odontogenic tumor with a customized cyst plug: A case report emphasizing differential diagnosis and bone regeneration

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Abstract Background This case report aims to present a misdiagnosed adenomatoid odontogenic tumor (AOT) initially suspected as a dentigerous cyst (DC), and to evaluate the effectiveness of a customized cyst plug in facilitating spontaneous bone regeneration without the need for bone grafting. Case presentation: We report a 13-year-old female with anterior maxillary swelling and a unilocular radiolucent lesion surrounding an impacted cuspid. Surgical decompression revealed abnormal tissue, confirmed as AOT histologically. Postoperatively, a palatal bone defect healed completely after 17 months of using a customized cyst plug, avoiding bone grafting. Conclusions This case highlights the diagnostic difficulty in distinguishing AOT from dentigerous cysts. In this single adolescent patient, a minimally invasive approach with a customized cyst plug was associated with radiographic bone fill of the postoperative defect. This graft-sparing strategy may be considered in selected cases; however, efficacy cannot be inferred from a single case, and longer-term comparative data are needed.
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Case presentation: We report a 13-year-old female with anterior maxillary swelling and a unilocular radiolucent lesion surrounding an impacted cuspid. Surgical decompression revealed abnormal tissue, confirmed as AOT histologically. Postoperatively, a palatal bone defect healed completely after 17 months of using a customized cyst plug, avoiding bone grafting. Conclusions This case highlights the diagnostic difficulty in distinguishing AOT from dentigerous cysts. In this single adolescent patient, a minimally invasive approach with a customized cyst plug was associated with radiographic bone fill of the postoperative defect. This graft-sparing strategy may be considered in selected cases; however, efficacy cannot be inferred from a single case, and longer-term comparative data are needed. Adenomatoid Odontogenic Tumor Dentigerous Cyst misdiagnosis bone defects cyst plug Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 1. BACKGROUND AOT is a relatively rare benign odontogenic tumor, first described by Steensland in 1905 1 . AOT accounts for approximately 3–7% of all odontogenic tumors and is characterized clinically as a slow-growing 2 , well-demarcated lesion predominantly affecting adolescents, especially in the maxillary cuspid region. Although rare, Philipsen et al. suggest that AOT ranks fourth among all odontogenic tumors in terms of frequency 3 . The precise pathogenesis of AOT remains unclear; however, it is widely accepted that it originates from the reduced enamel epithelium associated with unerupted teeth 4 . Clinically and radiographically, AOT frequently resembles odontogenic cysts, particularly DC, resulting in misdiagnosis rates reported between 20–30% 5 . Such diagnostic confusion can lead to inappropriate surgical approaches, increasing the risk of postoperative complications, particularly due to the fragility of the thin palatal bone plate. This case report presents the clinical management of a 13-year-old female patient with extensive maxillary AOT, emphasizing the differential diagnosis between AOT and DC, exploring the efficacy of conservative surgical approaches, and highlighting the role of a personalized cyst plug in facilitating effective postoperative bone regeneration. 2. CASE PRESENTATION 2.1 Patient information A 13-year-old female patient presented with swelling in the right maxillary region lasting over three months. One day prior to consultation, the patient underwent cone-beam computed tomography (CBCT) at another hospital, revealing a cystic lesion in the right maxilla, prompting referral to our facility for further management. 2.2 Clinical examination Asymmetry of the maxillofacial region, no boils or eczema on the surface skin, no obvious abnormality in the opening type and degree of opening, no obvious abnormality in the bilateral temporomandibular joints and facial nerve function. Intraoral examination showed permanent teeth, general oral hygiene, normal overjet and overbite, and a swelling of about 4.0cm×3.8cm in size extending from the right maxillary central incisor to the second molar region. On palpation the swelling was firm, slightly tender, clear boundary, and slight redness and swelling of the gingiva.The pulp vitality of the related teeth were normal.TM = 0°. 2.3 Auxiliary examination CBCT showed a cystic lesion of approximately 4.0 cm × 3.8 cm in the right maxilla, with well-defined and low-density areas. The lesion extended to the maxillary sinus, nasolacrimal duct, and the roots of teeth #16, #15, #14, and #12. An embedded, impacted tooth #13 was seen in the area of the lesion, located near the nasolacrimal duct (Fig. 1 ). 2.4 Diagnostic and therapeutic procedures The preoperative diagnosis was a DC. Under local anesthesia, open decompression surgery and extraction of impaction #13 were performed. A square incision was made along the hard palate in the area of the lesion, the flap was flipped, the bone wall was intact, the ultrasonic bone cutter removed the bone wall of the hard palate, the cyst wall was exposed, the cyst wall was intact, the quality of the cyst was tough, and the cyst cavity had a large amount of yellowish liquid outflow, which was scraped off thoroughly, the bone surface was trimmed, the hemostasis was fully achieved, and impaction #13 was exposed, which was extracted by forceps, washed with large amounts of physiological saline, there was no active bleeding, and iodine imitation gauze was placed, and silk suture was applied. Postoperative part of the capsule wall specimens sent for pathological examination: epithelial cells were arranged in a tubular or duct-like structure, which is a characteristic of AOT (Fig. 2 ). Immunohistochemical staining was negative for mesenchymal ectoderm, further supporting the diagnosis of AOT rather than other odontogenic tumors. Pathologic diagnosis: right maxilla Adenomatoid Odontogenic Tumor. Sutures were removed 7 days postoperatively, and the wound healed well with no signs of infection. At the 20-day postoperative follow-up, a cavernous bone defect was noted in the palatal side of the trauma, probably due to the large extent of the lesion and its proximity to adjacent anatomical structures. It was decided to continue to monitor the site and wear a cyst plug to promote bone healing. At 18-month postoperative follow-up, the bone defect had completely healed and the patient reported no discomfort (Fig. 3 ). Final CBCT confirmed complete closure of the surgical site and pulp vitality testing was normal in the lesion area (Fig. 4 ). 3. DISCUSSION AND CONCLUSION AOT was first described by Steensland in 1905 but was not recognized as a distinct pathological entity until the mid-20th century 6 , 7 . In 1969, Philipsen and Bern coined the term "adenoid odontogenic tumor," which has since been adopted by the World Health Organization (WHO). Initially referred to as "adenoameloblastoma", current classifications distinguish AOT clearly from ameloblastoma 7 . The present case highlights the clinical specificity and complexity involved in managing AOT. Epidemiologically, AOT constitutes approximately 3–4% of odontogenic tumors, predominantly affecting adolescents, with a female-to-male ratio of about 2:1. It typically occurs in the anterior maxillary region, commonly involving impacted teeth, particularly maxillary cuspids 8 . AOT is classified into three subtypes: follicular (most common, associated with unerupted teeth), extra-follicular intraosseous (not tooth-related, predominantly anterior jaw), and peripheral (rare, located in gingival or mucosal tissues) 9 . Notably, approximately 71% of AOT cases present as follicular type, contributing significantly to frequent misdiagnoses as DC 10 . In terms of pathogenesis, there are two main hypotheses: the plate remnant theory suggests that the tumor originated from the plate or its residual epithelium, which could explain the close relationship between follicular AOT and impacted teeth 11 . Some scholars have suggested that AOT may have originated from the transformation of the plate's cystic epithelium 12 , 13 . In the present case, the tumor encircled the cusp of the rooted tooth, which supports the plate remnant theory and does not completely exclude the possibility of cystic transformation. At the same time, this phenomenon precisely reflects the complexity of the origin of AOT. 3.1 Differential diagnosis of AOT and DC The differential diagnosis of AOT and DC is of great value in clinical work. The diagnostic and therapeutic process of this case, from the initial misdiagnosis of DC to the final diagnosis of AOT, provides us with valuable lessons. Through a systematic review of the literature and an in-depth analysis of the features of this case, we found that these two lesions differ significantly in several aspects. In terms of pathogenesis, AOT is a true odontogenic tumor originating from the odontogenic epithelium, specifically the reduced enamel epithelium (REE) 14 . In contrast, DC are developmental cysts that originate from the degeneration of the REE after crown formation 15 . This intrinsic difference determines their differences in biological behavior and pathological features. In terms of clinical presentation, patients with AOT usually present with a slow-growing, painless swelling with an average duration of up to 2–3 years 16 . In the present case, the patient came to the clinic with a complaint of a 3-month history, which is relatively rare in AOT and may have contributed to the initial misdiagnosis. In contrast, DC grows relatively quickly and may be accompanied by signs of infection. It is noteworthy that AOT exhibits a significant gender predisposition with a prevalence twice as high in females as in males, whereas there is no significant gender difference in DC 17 . In this case, unilocular radiolucency accompanied by calcified dots or contained teeth demonstrated by CBCT, is a typical manifestation of AOT. These calcified foci correspond histologically to amyloid deposits within the tumor. In contrast, DC usually shows homogeneous translucent shadows and rarely calcifications. In addition, AOT usually has thicker walls (> 2 mm) and is not as closely related to the impacting teeth as DC. In this case, the atypical relationship of the lesion to the obstructing 13 teeth was one factor that led to the initial misdiagnosis. Histopathologic examination is the gold standard for confirming the diagnosis of AOT. In the present case, an abnormally thickened cyst wall was observed intraoperatively and was immediately sent for histopathological examination, a worthwhile practice. The characteristic pathologic manifestations of AOT are the formation of a duct-like structure by the tumor epithelium surrounded by spindle cells 18 . Immunohistochemical staining showed a strong positivity for CK19 19 , whereas DC lacked these characteristic manifestations. Notably, approximately 20–30% of AOT cases are initially misdiagnosed as DC6, a statistic suggesting the clinician's need for vigilance 5 . 3.2 Treatment and surgical considerations 3.2.1 Conservative surgery: the treatment of choice The mainstay of treatment for AOT is surgical excision or curettage 20 . Conservative local excision is usually sufficient because AOT is usually peritumoral and slow-growing, and non-invasive. For larger AOTs, open decompression may be considered to reduce the size of the tumor before resection 21 . In the present case, due to a preoperative misdiagnosis of a DC, open decompression surgery was used, but after abnormal thickening of the cyst wall was detected intraoperatively, the plan was promptly adjusted to thoroughly scrape the diseased tissue and send it for pathological examination, which is in line with the principle of treatment for AOTs. In adolescent patients, surgery requires special consideration for the protection of the permanent dental embryo and the impact on jaw development. In this case, despite the involvement of the lesion in the nasolacrimal duct and maxillary sinus region, the adjacent pulp viability was successfully preserved through delicate operation (ultrasonic bone knife debridement, avoiding excessive stripping of soft tissues), and the 18-month postoperative follow-up showed normal root development. 3.2.2 Application of a personalized Cyst Plug AOT occurs in the maxillary anterior region, where the bone plate is thin and is prone to intraoperative cavitation defects. The successful application of a personalized cyst plug in this case promotes the healing of the postoperative bone defect and provides an important reference for the management of similar cases. The effect of open decompression is related to smooth drainage, and the cyst plug can keep the drainage port open, balance the pressure inside and outside the capsule, and promote the therapeutic effect 22 . Through in-depth analysis, we found that the combination of cyst plugs after open decompression surgery to prevent premature healing of the wound is a mainstream practice, and the patient in the present case has been wearing a prosthetic-type plug for 17 months after the surgery 23 (Fig. 5 ). The cyst plug can ensure the effect of continuous drainage of capsule fluid, to avoid premature healing and formation of a dead space. It also prevents food from entering the cavity, makes cleaning easier for the patient, increases compliance, decreases swelling, and reduces the risk of secondary infections and postoperative complications. Compared with invasive methods such as bone grafting, plug therapy is less invasive and less costly, and is particularly suitable for adolescent patients in their growth period. From a biomechanical point of view, the plug provides a stable mechanical environment for the defect area 24 . The 2-mm-thick acrylic resin plate used in this case can effectively disperse the occlusal force and control the local strain within the optimal osteogenic range of 200–800 microstrain 25 . Finite element analysis shows that this design can reduce the stress concentration at the defect edge by 30%, creating favorable conditions for bone regeneration 26 . In conclusion, AOT is typically a benign lesion with excellent outcomes after conservative surgical management, although conclusions should be limited to the context of individual cases. This case highlights two key findings. First, the lesion was initially misdiagnosed as a dentigerous cyst and managed with decompression. However, intraoperative recognition of atypical tissue led to a timely revision of the surgical plan, and the diagnosis of AOT was subsequently confirmed by histopathology. Second, the lesion was unusually extensive with maxillary sinus involvement, a presentation that is less commonly reported. The postoperative use of a personalized cyst plug resulted in substantial radiographic bone fill of the palatal cavitation and satisfactory clinical healing. There was no clinical or radiographic evidence of recurrence at the 18-month follow-up. As a single-case report without a comparator and with limited follow-up, causality between the cyst plug and bone regeneration cannot be established, and spontaneous remodeling in adolescents cannot be excluded. Imaging-based assessments may also be subject to measurement variability. Therefore, our findings should be interpreted as hypothesis-generating rather than definitive. In adolescents with large AOTs, a conservative approach combined with customized postoperative support (e.g., a cyst plug) may be considered after histologic confirmation and with close follow-up. Prospective series or registries comparing plug-assisted healing with bone grafting are needed to define indications and long-term outcomes. Abbreviations AOT adenomatoid odontogenic tumor DC dentigerous cyst CBCT cone-beam computed tomography WHO World Health Organization REE reduced enamel epithelium Declarations Ethics approval and consent to participate This case report was approved by the Ethics Committee of Affiliated Stomatology Hospital of Guangzhou Medical University (LCYJ20250423010). Consent for publication We have obtained written informed consent from the patient's parents for the publication of the clinical details and clinical images. Competing interests The authors declare that they have no competing interests. Funding There is no funding for this report. Author Contribution Zuwen Ma and Guangwei Chen: conception; design of study; drafting of the manuscript;Huilin Wu: Investigation;Weifa Li: Investigation;Libin Zhou: Conceptualization, Supervision; Writing-Review and Editing. Acknowledgements: Not applicable Data Availability All data generated or analysed during this study are included in this manuscript. The datasets used or analyzed during the current study are available from the corresponding author upon reasonable request. References Steensland HS. Epithelioma adamantinum. J Exp Med. 1905;6(4–6):377–89. Santosh ABR, Coard KCM, Williams EB, Jones T. Adenomatoid odontogenic tumor: Clinical and radiological diagnostic challenges. J Pierre Fauchard Acad (india Sect). 2017;31:115–20. 10.1016/j.jpfa.2017.06.001 . Philipsen HP, Reichart PA. Adenomatoid odontogenic tumour: Facts and figures. Oral Oncol. 1999;35(2):125–31. Francisconi NdaS, Silva BB, Araújo LTC, Mariani TR, Gil LF, Rivero ERC. Adenomatoid odontogenic tumor: A case report. 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15:32:47","extension":"png","order_by":18,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":46016,"visible":true,"origin":"","legend":"","description":"","filename":"Onlinegroupimage3.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/644559f3c821a1f9602ca572.png"},{"id":92010614,"identity":"b42a13e4-fb8b-4e49-9f9b-b8b28295b1bc","added_by":"auto","created_at":"2025-09-23 15:40:47","extension":"xml","order_by":19,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":66652,"visible":true,"origin":"","legend":"","description":"","filename":"f387dae18b344a8780af5644556101e41structuring.xml","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/9c4b0b3dfe3c17204550f0ab.xml"},{"id":92010615,"identity":"9f50fcf4-ea78-4f82-b80e-e55594735ada","added_by":"auto","created_at":"2025-09-23 15:40:47","extension":"html","order_by":20,"title":"","display":"","copyAsset":false,"role":"acdc-reference","size":73987,"visible":true,"origin":"","legend":"","description":"","filename":"earlyproof.html","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/274ad7cf0f7f2ab251b91da5.html"},{"id":92010597,"identity":"95eb2ee8-4262-4d35-876c-f6b0a2b7c0ff","added_by":"auto","created_at":"2025-09-23 15:40:47","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1523579,"visible":true,"origin":"","legend":"\u003cp\u003eCBCT images showing impacted maxillary right cuspid with well-circumscribed radiolucency\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/7eacca7289479c558b45ca7f.png"},{"id":92011739,"identity":"b261266b-97a7-40ec-a5c2-0b716bb6ba5f","added_by":"auto","created_at":"2025-09-23 15:48:47","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":3540048,"visible":true,"origin":"","legend":"\u003cp\u003eTumor cells arranged in duct-like patterns\u003c/p\u003e","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/5558a9f03fcaadf4fe8aaf2e.png"},{"id":92010604,"identity":"9c0f66b4-e61d-4e45-b7a3-2ac0995bc8b2","added_by":"auto","created_at":"2025-09-23 15:40:47","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":824295,"visible":true,"origin":"","legend":"\u003cp\u003ePostoperative reconstruction of palatal bone defects\u003c/p\u003e","description":"","filename":"3.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/0e1f34ab870ecd28b9496e0d.png"},{"id":92008796,"identity":"4dcda9fe-309f-4663-8fc2-d27aa6b32a23","added_by":"auto","created_at":"2025-09-23 15:32:47","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":1481858,"visible":true,"origin":"","legend":"\u003cp\u003e17-month postoperative CBCT\u003c/p\u003e","description":"","filename":"4.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/89b5544a4cf142d7f7facf76.png"},{"id":92008808,"identity":"86748bdf-b28a-4b77-86c3-172c798e836e","added_by":"auto","created_at":"2025-09-23 15:32:47","extension":"png","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":2740899,"visible":true,"origin":"","legend":"\u003cp\u003eA prosthetic-type plug for 17-month postoperative\u003c/p\u003e","description":"","filename":"5.png","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/d70d0d1c60645880e87881be.png"},{"id":92012845,"identity":"a4ee978b-102e-4b07-82dc-d1b9aba4befe","added_by":"auto","created_at":"2025-09-23 15:56:57","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":15229751,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-7281237/v1/4e661205-5ff6-4e71-bbc4-7957cb534694.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Conservative management of extensive maxillary adenomatoid odontogenic tumor with a customized cyst plug: A case report emphasizing differential diagnosis and bone regeneration","fulltext":[{"header":"1. BACKGROUND","content":"\u003cp\u003eAOT is a relatively rare benign odontogenic tumor, first described by Steensland in 1905\u003csup\u003e1\u003c/sup\u003e. AOT accounts for approximately 3\u0026ndash;7% of all odontogenic tumors and is characterized clinically as a slow-growing\u003csup\u003e\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e\u003c/sup\u003e, well-demarcated lesion predominantly affecting adolescents, especially in the maxillary cuspid region. Although rare, Philipsen et al. suggest that AOT ranks fourth among all odontogenic tumors in terms of frequency\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u003c/sup\u003e. The precise pathogenesis of AOT remains unclear; however, it is widely accepted that it originates from the reduced enamel epithelium associated with unerupted teeth\u003csup\u003e\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e\u003c/sup\u003e. Clinically and radiographically, AOT frequently resembles odontogenic cysts, particularly DC, resulting in misdiagnosis rates reported between 20\u0026ndash;30%\u003csup\u003e5\u003c/sup\u003e. Such diagnostic confusion can lead to inappropriate surgical approaches, increasing the risk of postoperative complications, particularly due to the fragility of the thin palatal bone plate. This case report presents the clinical management of a 13-year-old female patient with extensive maxillary AOT, emphasizing the differential diagnosis between AOT and DC, exploring the efficacy of conservative surgical approaches, and highlighting the role of a personalized cyst plug in facilitating effective postoperative bone regeneration.\u003c/p\u003e"},{"header":"2. CASE PRESENTATION","content":"\u003cdiv id=\"Sec3\" class=\"Section2\"\u003e\u003ch2\u003e2.1 Patient information\u003c/h2\u003e\u003cp\u003eA 13-year-old female patient presented with swelling in the right maxillary region lasting over three months. One day prior to consultation, the patient underwent cone-beam computed tomography (CBCT) at another hospital, revealing a cystic lesion in the right maxilla, prompting referral to our facility for further management.\u003c/p\u003e\u003c/div\u003e\u003cdiv id=\"Sec4\" class=\"Section2\"\u003e\u003ch2\u003e2.2 Clinical examination\u003c/h2\u003e\u003cp\u003eAsymmetry of the maxillofacial region, no boils or eczema on the surface skin, no obvious abnormality in the opening type and degree of opening, no obvious abnormality in the bilateral temporomandibular joints and facial nerve function. Intraoral examination showed permanent teeth, general oral hygiene, normal overjet and overbite, and a swelling of about 4.0cm\u0026times;3.8cm in size extending from the right maxillary central incisor to the second molar region. On palpation the swelling was firm, slightly tender, clear boundary, and slight redness and swelling of the gingiva.The pulp vitality of the related teeth were normal.TM\u0026thinsp;=\u0026thinsp;0\u0026deg;.\u003c/p\u003e\u003c/div\u003e\u003cdiv id=\"Sec5\" class=\"Section2\"\u003e\u003ch2\u003e2.3 Auxiliary examination\u003c/h2\u003e\u003cp\u003eCBCT showed a cystic lesion of approximately 4.0 cm \u0026times; 3.8 cm in the right maxilla, with well-defined and low-density areas. The lesion extended to the maxillary sinus, nasolacrimal duct, and the roots of teeth #16, #15, #14, and #12. An embedded, impacted tooth #13 was seen in the area of the lesion, located near the nasolacrimal duct (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003c/div\u003e\u003cdiv id=\"Sec6\" class=\"Section2\"\u003e\u003ch2\u003e2.4 Diagnostic and therapeutic procedures\u003c/h2\u003e\u003cp\u003eThe preoperative diagnosis was a DC. Under local anesthesia, open decompression surgery and extraction of impaction #13 were performed. A square incision was made along the hard palate in the area of the lesion, the flap was flipped, the bone wall was intact, the ultrasonic bone cutter removed the bone wall of the hard palate, the cyst wall was exposed, the cyst wall was intact, the quality of the cyst was tough, and the cyst cavity had a large amount of yellowish liquid outflow, which was scraped off thoroughly, the bone surface was trimmed, the hemostasis was fully achieved, and impaction #13 was exposed, which was extracted by forceps, washed with large amounts of physiological saline, there was no active bleeding, and iodine imitation gauze was placed, and silk suture was applied. Postoperative part of the capsule wall specimens sent for pathological examination: epithelial cells were arranged in a tubular or duct-like structure, which is a characteristic of AOT (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). Immunohistochemical staining was negative for mesenchymal ectoderm, further supporting the diagnosis of AOT rather than other odontogenic tumors. Pathologic diagnosis: right maxilla Adenomatoid Odontogenic Tumor.\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eSutures were removed 7 days postoperatively, and the wound healed well with no signs of infection. At the 20-day postoperative follow-up, a cavernous bone defect was noted in the palatal side of the trauma, probably due to the large extent of the lesion and its proximity to adjacent anatomical structures. It was decided to continue to monitor the site and wear a cyst plug to promote bone healing. At 18-month postoperative follow-up, the bone defect had completely healed and the patient reported no discomfort (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e). Final CBCT confirmed complete closure of the surgical site and pulp vitality testing was normal in the lesion area (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e).\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003c/div\u003e"},{"header":"3. DISCUSSION AND CONCLUSION","content":"\u003cp\u003eAOT was first described by Steensland in 1905 but was not recognized as a distinct pathological entity until the mid-20th century\u003csup\u003e\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e,\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e\u003c/sup\u003e. In 1969, Philipsen and Bern coined the term \"adenoid odontogenic tumor,\" which has since been adopted by the World Health Organization (WHO). Initially referred to as \"adenoameloblastoma\", current classifications distinguish AOT clearly from ameloblastoma\u003csup\u003e\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e\u003c/sup\u003e. The present case highlights the clinical specificity and complexity involved in managing AOT.\u003c/p\u003e\u003cp\u003eEpidemiologically, AOT constitutes approximately 3\u0026ndash;4% of odontogenic tumors, predominantly affecting adolescents, with a female-to-male ratio of about 2:1. It typically occurs in the anterior maxillary region, commonly involving impacted teeth, particularly maxillary cuspids\u003csup\u003e\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u003c/sup\u003e. AOT is classified into three subtypes: follicular (most common, associated with unerupted teeth), extra-follicular intraosseous (not tooth-related, predominantly anterior jaw), and peripheral (rare, located in gingival or mucosal tissues)\u003csup\u003e\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e\u003c/sup\u003e. Notably, approximately 71% of AOT cases present as follicular type, contributing significantly to frequent misdiagnoses as DC\u003csup\u003e\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e\u003cp\u003eIn terms of pathogenesis, there are two main hypotheses: the plate remnant theory suggests that the tumor originated from the plate or its residual epithelium, which could explain the close relationship between follicular AOT and impacted teeth\u003csup\u003e\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e\u003c/sup\u003e. Some scholars have suggested that AOT may have originated from the transformation of the plate's cystic epithelium\u003csup\u003e\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e,\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e. In the present case, the tumor encircled the cusp of the rooted tooth, which supports the plate remnant theory and does not completely exclude the possibility of cystic transformation. At the same time, this phenomenon precisely reflects the complexity of the origin of AOT.\u003c/p\u003e\u003cdiv id=\"Sec8\" class=\"Section2\"\u003e\u003ch2\u003e3.1 Differential diagnosis of AOT and DC\u003c/h2\u003e\u003cp\u003eThe differential diagnosis of AOT and DC is of great value in clinical work. The diagnostic and therapeutic process of this case, from the initial misdiagnosis of DC to the final diagnosis of AOT, provides us with valuable lessons. Through a systematic review of the literature and an in-depth analysis of the features of this case, we found that these two lesions differ significantly in several aspects.\u003c/p\u003e\u003cp\u003eIn terms of pathogenesis, AOT is a true odontogenic tumor originating from the odontogenic epithelium, specifically the reduced enamel epithelium (REE)\u003csup\u003e\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e. In contrast, DC are developmental cysts that originate from the degeneration of the REE after crown formation\u003csup\u003e\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e\u003c/sup\u003e. This intrinsic difference determines their differences in biological behavior and pathological features.\u003c/p\u003e\u003cp\u003eIn terms of clinical presentation, patients with AOT usually present with a slow-growing, painless swelling with an average duration of up to 2\u0026ndash;3 years\u003csup\u003e\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e\u003c/sup\u003e. In the present case, the patient came to the clinic with a complaint of a 3-month history, which is relatively rare in AOT and may have contributed to the initial misdiagnosis. In contrast, DC grows relatively quickly and may be accompanied by signs of infection. It is noteworthy that AOT exhibits a significant gender predisposition with a prevalence twice as high in females as in males, whereas there is no significant gender difference in DC\u003csup\u003e\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e\u003cp\u003eIn this case, unilocular radiolucency accompanied by calcified dots or contained teeth demonstrated by CBCT, is a typical manifestation of AOT. These calcified foci correspond histologically to amyloid deposits within the tumor. In contrast, DC usually shows homogeneous translucent shadows and rarely calcifications. In addition, AOT usually has thicker walls (\u0026gt;\u0026thinsp;2 mm) and is not as closely related to the impacting teeth as DC. In this case, the atypical relationship of the lesion to the obstructing 13 teeth was one factor that led to the initial misdiagnosis.\u003c/p\u003e\u003cp\u003eHistopathologic examination is the gold standard for confirming the diagnosis of AOT. In the present case, an abnormally thickened cyst wall was observed intraoperatively and was immediately sent for histopathological examination, a worthwhile practice. The characteristic pathologic manifestations of AOT are the formation of a duct-like structure by the tumor epithelium surrounded by spindle cells\u003csup\u003e\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e\u003c/sup\u003e. Immunohistochemical staining showed a strong positivity for CK19\u003csup\u003e19\u003c/sup\u003e, whereas DC lacked these characteristic manifestations. Notably, approximately 20\u0026ndash;30% of AOT cases are initially misdiagnosed as DC6, a statistic suggesting the clinician's need for vigilance\u003csup\u003e\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e\u003c/div\u003e\u003cdiv id=\"Sec9\" class=\"Section2\"\u003e\u003ch2\u003e3.2 Treatment and surgical considerations\u003c/h2\u003e\u003cdiv id=\"Sec10\" class=\"Section3\"\u003e\u003ch2\u003e3.2.1 Conservative surgery: the treatment of choice\u003c/h2\u003e\u003cp\u003eThe mainstay of treatment for AOT is surgical excision or curettage\u003csup\u003e\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e\u003c/sup\u003e. Conservative local excision is usually sufficient because AOT is usually peritumoral and slow-growing, and non-invasive. For larger AOTs, open decompression may be considered to reduce the size of the tumor before resection\u003csup\u003e\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e\u003c/sup\u003e. In the present case, due to a preoperative misdiagnosis of a DC, open decompression surgery was used, but after abnormal thickening of the cyst wall was detected intraoperatively, the plan was promptly adjusted to thoroughly scrape the diseased tissue and send it for pathological examination, which is in line with the principle of treatment for AOTs. In adolescent patients, surgery requires special consideration for the protection of the permanent dental embryo and the impact on jaw development. In this case, despite the involvement of the lesion in the nasolacrimal duct and maxillary sinus region, the adjacent pulp viability was successfully preserved through delicate operation (ultrasonic bone knife debridement, avoiding excessive stripping of soft tissues), and the 18-month postoperative follow-up showed normal root development.\u003c/p\u003e\u003c/div\u003e\u003cdiv id=\"Sec11\" class=\"Section3\"\u003e\u003ch2\u003e3.2.2 Application of a personalized Cyst Plug\u003c/h2\u003e\u003cp\u003eAOT occurs in the maxillary anterior region, where the bone plate is thin and is prone to intraoperative cavitation defects. The successful application of a personalized cyst plug in this case promotes the healing of the postoperative bone defect and provides an important reference for the management of similar cases. The effect of open decompression is related to smooth drainage, and the cyst plug can keep the drainage port open, balance the pressure inside and outside the capsule, and promote the therapeutic effect\u003csup\u003e\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e\u003cp\u003eThrough in-depth analysis, we found that the combination of cyst plugs after open decompression surgery to prevent premature healing of the wound is a mainstream practice, and the patient in the present case has been wearing a prosthetic-type plug for 17 months after the surgery\u003csup\u003e\u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e\u003c/sup\u003e(Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e5\u003c/span\u003e). The cyst plug can ensure the effect of continuous drainage of capsule fluid, to avoid premature healing and formation of a dead space. It also prevents food from entering the cavity, makes cleaning easier for the patient, increases compliance, decreases swelling, and reduces the risk of secondary infections and postoperative complications. Compared with invasive methods such as bone grafting, plug therapy is less invasive and less costly, and is particularly suitable for adolescent patients in their growth period. From a biomechanical point of view, the plug provides a stable mechanical environment for the defect area\u003csup\u003e\u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e\u003c/sup\u003e. The 2-mm-thick acrylic resin plate used in this case can effectively disperse the occlusal force and control the local strain within the optimal osteogenic range of 200\u0026ndash;800 microstrain\u003csup\u003e\u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e\u003c/sup\u003e. Finite element analysis shows that this design can reduce the stress concentration at the defect edge by 30%, creating favorable conditions for bone regeneration\u003csup\u003e\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003eIn conclusion, AOT is typically a benign lesion with excellent outcomes after conservative surgical management, although conclusions should be limited to the context of individual cases. This case highlights two key findings. First, the lesion was initially misdiagnosed as a dentigerous cyst and managed with decompression. However, intraoperative recognition of atypical tissue led to a timely revision of the surgical plan, and the diagnosis of AOT was subsequently confirmed by histopathology. Second, the lesion was unusually extensive with maxillary sinus involvement, a presentation that is less commonly reported. The postoperative use of a personalized cyst plug resulted in substantial radiographic bone fill of the palatal cavitation and satisfactory clinical healing. There was no clinical or radiographic evidence of recurrence at the 18-month follow-up. As a single-case report without a comparator and with limited follow-up, causality between the cyst plug and bone regeneration cannot be established, and spontaneous remodeling in adolescents cannot be excluded. Imaging-based assessments may also be subject to measurement variability. Therefore, our findings should be interpreted as hypothesis-generating rather than definitive. In adolescents with large AOTs, a conservative approach combined with customized postoperative support (e.g., a cyst plug) may be considered after histologic confirmation and with close follow-up. Prospective series or registries comparing plug-assisted healing with bone grafting are needed to define indications and long-term outcomes.\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eAOT\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003eadenomatoid odontogenic tumor\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eDC\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003edentigerous cyst\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eCBCT\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003econe-beam computed tomography\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eWHO\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003eWorld Health Organization\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003cdiv class=\"DefinitionListEntry\"\u003e\u003cdiv class=\"Term\"\u003eREE\u003c/div\u003e\u003cdiv class=\"Description\"\u003e\u003cp\u003ereduced enamel epithelium\u003c/p\u003e\u003c/div\u003e\u003c/div\u003e\u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003ch2\u003eEthics approval and consent to participate\u003c/h2\u003e\u003cp\u003eThis case report was approved by the Ethics Committee of Affiliated Stomatology Hospital of Guangzhou Medical University (LCYJ20250423010).\u003c/p\u003e\u003c/p\u003e\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003cp\u003eWe have obtained written informed consent from the patient's parents for the publication of the clinical details and clinical images.\u003c/p\u003e\u003c/p\u003e\u003cp\u003e\u003ch2\u003eCompeting interests\u003c/h2\u003e\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\u003c/p\u003e\u003ch2\u003eFunding\u003c/h2\u003e\u003cp\u003eThere is no funding for this report.\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eZuwen Ma and Guangwei Chen: conception; design of study; drafting of the manuscript;Huilin Wu: Investigation;Weifa Li: Investigation;Libin Zhou: Conceptualization, Supervision; Writing-Review and Editing.\u003c/p\u003e\u003ch2\u003eAcknowledgements:\u003c/h2\u003e\u003cp\u003eNot applicable\u003c/p\u003e\u003ch2\u003eData Availability\u003c/h2\u003e\u003cp\u003eAll data generated or analysed during this study are included in this manuscript. The datasets used or analyzed during the current study are available from the corresponding author upon reasonable request.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eSteensland HS. Epithelioma adamantinum. J Exp Med. 1905;6(4\u0026ndash;6):377\u0026ndash;89.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003eSantosh ABR, Coard KCM, Williams EB, Jones T. Adenomatoid odontogenic tumor: Clinical and radiological diagnostic challenges. J Pierre Fauchard Acad (india Sect). 2017;31:115\u0026ndash;20. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1016/j.jpfa.2017.06.001\u003c/span\u003e\u003cspan address=\"10.1016/j.jpfa.2017.06.001\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e\u003cli\u003e\u003cspan\u003ePhilipsen HP, Reichart PA. Adenomatoid odontogenic tumour: Facts and figures. 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Published online January 2012.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-oral-health","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ohea","sideBox":"Learn more about [BMC Oral Health](http://bmcoralhealth.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ohea/default.aspx","title":"BMC Oral Health","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Adenomatoid Odontogenic Tumor, Dentigerous Cyst, misdiagnosis, bone defects, cyst plug","lastPublishedDoi":"10.21203/rs.3.rs-7281237/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-7281237/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eBackground\u003c/h2\u003e\u003cp\u003eThis case report aims to present a misdiagnosed adenomatoid odontogenic tumor (AOT) initially suspected as a dentigerous cyst (DC), and to evaluate the effectiveness of a customized cyst plug in facilitating spontaneous bone regeneration without the need for bone grafting.\u003c/p\u003e\u003ch2\u003eCase presentation:\u003c/h2\u003e\u003cp\u003eWe report a 13-year-old female with anterior maxillary swelling and a unilocular radiolucent lesion surrounding an impacted cuspid. Surgical decompression revealed abnormal tissue, confirmed as AOT histologically. Postoperatively, a palatal bone defect healed completely after 17 months of using a customized cyst plug, avoiding bone grafting.\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e\u003cp\u003eThis case highlights the diagnostic difficulty in distinguishing AOT from dentigerous cysts. In this single adolescent patient, a minimally invasive approach with a customized cyst plug was associated with radiographic bone fill of the postoperative defect. This graft-sparing strategy may be considered in selected cases; however, efficacy cannot be inferred from a single case, and longer-term comparative data are needed.\u003c/p\u003e","manuscriptTitle":"Conservative management of extensive maxillary adenomatoid odontogenic tumor with a customized cyst plug: A case report emphasizing differential diagnosis and bone regeneration","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-09-23 15:32:42","doi":"10.21203/rs.3.rs-7281237/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2026-04-20T13:11:03+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-12-24T05:37:59+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-12-22T06:44:22+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"232663812029863998900093510597353377645","date":"2025-12-22T05:30:32+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-12-19T04:47:55+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"134039691783525618075179243368937992893","date":"2025-12-19T04:43:33+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"311121126633667111282252769355974085911","date":"2025-12-19T04:11:42+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-09-23T14:17:29+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"295220600365573114533462562432508999354","date":"2025-09-16T06:32:06+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-09-15T11:57:22+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-09-15T11:55:48+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2025-09-05T04:25:10+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-09-04T14:15:40+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Oral Health","date":"2025-09-04T13:46:07+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-oral-health","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ohea","sideBox":"Learn more about [BMC Oral Health](http://bmcoralhealth.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ohea/default.aspx","title":"BMC Oral Health","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"8a321c5b-878b-4339-9ee3-ad92d7f643ab","owner":[],"postedDate":"September 23rd, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2026-05-02T10:38:31+00:00","versionOfRecord":[],"versionCreatedAt":"2025-09-23 15:32:42","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-7281237","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-7281237","identity":"rs-7281237","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

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