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一名儿科患者上颌前部罕见的成釉细胞纤维牙瘤的诊断与治疗病例报告。

A Case Report on the Diagnosis and Management of a Rare Ameloblastic Fibro-Odontoma in the Anterior Maxilla of a Pediatric Patient.

作者信息

Ash Hiralal, Singh Rajeev Kumar, Anwar Eram, Banerjee Abhishek, Chatterjee Ananjan, Ramalingam Karthikeyan

机构信息

Oral and Maxillofacial Surgery, Buddha Institute of Dental Sciences and Hospital, Patna, IND.

Oral and Maxillofacial Pathology, Awadh Dental College and Hospital, Jamshedpur, IND.

出版信息

Cureus. 2025 May 18;17(5):e84343. doi: 10.7759/cureus.84343. eCollection 2025 May.

DOI:10.7759/cureus.84343
PMID:40530215
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12173212/
Abstract

Ameloblastic fibro-odontoma (AFO) is an uncommon, non-cancerous odontogenic tumor that predominantly affects children and young adolescents. This case report details a nine-year-old male patient who presented with a firm swelling accompanied by intermittent serous discharge in the upper right jaw following a fall. Clinical assessment revealed incomplete mouth closure, facial asymmetry, and dentoalveolar extrusion. Radiographic analysis via cone beam computed tomography revealed a heterogeneous radiolucent lesion with focal radiopacities, thinning of the cortical plates, and a minor breach in the nasal floor, indicative of an osteolytic lesion. Differential diagnoses considered included adenomatoid odontogenic tumor and desmoplastic ameloblastoma. Routine blood tests and fine-needle aspiration cytology (FNAC) yielded negative results. The lesion was surgically enucleated under general anesthesia, with preservation of the affected teeth. Histopathological analysis confirmed the diagnosis of AFO, characterized by hyperchromatic columnar ameloblast-like cells, stellate reticulum-like cells, and basophilic dentinoid-like formation within an immature connective tissue stroma. Postoperative recovery was smooth, resulting in notable improvement in facial aesthetics and oral function. AFO is typically located in the posterior mandible, making its occurrence in the anterior maxilla unusual. Conservative surgical management is the recommended strategy and is associated with low recurrence rates. Although malignant transformation has been documented, extensive treatment is generally reserved for cases exhibiting dysplastic changes or aggressive recurrence. This case underscores the importance of early diagnosis, imaging, and histological verification for effective treatment planning. Regular follow-up is crucial to monitor for potential recurrence and to ensure favorable long-term outcomes in patients with AFO.

摘要

成釉细胞纤维牙瘤(AFO)是一种罕见的非癌性牙源性肿瘤,主要影响儿童和青少年。本病例报告详细介绍了一名9岁男性患者,该患者在跌倒后右上颌出现质地坚硬的肿胀,并伴有间歇性浆液性分泌物。临床评估显示张口受限、面部不对称和牙槽骨突出。通过锥形束计算机断层扫描进行的影像学分析显示,存在一个异质性的透射性病变,伴有局灶性阻射性区域、皮质骨板变薄以及鼻底轻度破损,提示为溶骨性病变。考虑的鉴别诊断包括腺样牙源性肿瘤和促结缔组织增生性成釉细胞瘤。常规血液检查和细针穿刺细胞学检查(FNAC)结果均为阴性。在全身麻醉下对病变进行了手术摘除,并保留了患牙。组织病理学分析证实了AFO的诊断,其特征为在未成熟的结缔组织基质内有深染的柱状成釉细胞样细胞、星网状细胞样细胞以及嗜碱性牙本质样结构。术后恢复顺利,面部美观和口腔功能有显著改善。AFO通常位于下颌骨后部,因此发生在上颌前部较为罕见。推荐的治疗策略是保守手术治疗,其复发率较低。尽管已有恶性转化的报道,但广泛的治疗通常仅适用于表现出发育异常改变或侵袭性复发的病例。本病例强调了早期诊断、影像学检查和组织学验证对于有效治疗计划的重要性。定期随访对于监测潜在复发以及确保AFO患者获得良好的长期预后至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/99e1f38de559/cureus-0017-00000084343-i08.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/24236e090c09/cureus-0017-00000084343-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/00a3fb766866/cureus-0017-00000084343-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/bd75ae80a3d4/cureus-0017-00000084343-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/4dd6e9aa3513/cureus-0017-00000084343-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/1e59c38456ae/cureus-0017-00000084343-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/99f9547f1aaa/cureus-0017-00000084343-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/9e5ecbe01967/cureus-0017-00000084343-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/99e1f38de559/cureus-0017-00000084343-i08.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/24236e090c09/cureus-0017-00000084343-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/00a3fb766866/cureus-0017-00000084343-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/bd75ae80a3d4/cureus-0017-00000084343-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/4dd6e9aa3513/cureus-0017-00000084343-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/1e59c38456ae/cureus-0017-00000084343-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/99f9547f1aaa/cureus-0017-00000084343-i06.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/9e5ecbe01967/cureus-0017-00000084343-i07.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d94b/12173212/99e1f38de559/cureus-0017-00000084343-i08.jpg

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本文引用的文献

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Ameloblastic Fibro-odontoma - A Case Report of Two Uncommon Cases.成釉细胞纤维牙瘤——两例罕见病例报告
Ann Maxillofac Surg. 2024 Jan-Jun;14(1):116-119. doi: 10.4103/ams.ams_7_24. Epub 2024 May 24.
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Ameloblastic fibro‑odontoma in the posterior mandible: A case report.下颌骨后部的成釉细胞纤维牙瘤:一例报告。
Med Int (Lond). 2023 Nov 20;3(6):63. doi: 10.3892/mi.2023.123. eCollection 2023 Nov-Dec.
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Ameloblastic Fibro-Odontoma of the Posterior Mandible: A Rare Pathological Entity.下颌骨后部的成釉细胞纤维牙瘤:一种罕见的病理实体。
Cureus. 2023 Sep 30;15(9):e46264. doi: 10.7759/cureus.46264. eCollection 2023 Sep.
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Ameloblastic Fibro-Odontoma.成釉细胞纤维牙瘤
Fetal Pediatr Pathol. 2023 Apr;42(2):281-284. doi: 10.1080/15513815.2022.2088910. Epub 2022 Jun 24.
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Ameloblastic Fibro-Odontoma: A Journey of Progression?成釉细胞纤维牙瘤:进展历程?
J Oral Maxillofac Pathol. 2022 Feb;26(Suppl 1):S40-S45. doi: 10.4103/jomfp.jomfp_184_21. Epub 2022 Feb 28.
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A Locally Aggressive Ameloblastic Fibro-Odontoma: A Case Report and Literature Review.一例局部侵袭性成釉细胞纤维牙瘤:病例报告及文献综述
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Ameloblastic Fibrodentinoma and Ameloblastic Fibro-Odontoma: An Updated Systematic Review of Cases Reported in the Literature.成釉细胞纤维牙本质瘤和成釉细胞纤维牙瘤:文献报道病例的最新系统评价
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Ameloblastic fibro-odontoma.成釉细胞纤维牙瘤
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