Calcifying epithelial odontogenic tumor

牙源性钙化上皮肿瘤
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    文章类型: English Abstract
    目的:应用螺旋CT(CT)和锥形束CT(CBCT)分析牙源性钙化囊肿和牙源性钙化上皮肿瘤的三维影像学特征。
    方法:临床记录,组织病理学报告,回顾性获得了19例连续的钙化性牙源性囊肿(COC)患者和16例连续的钙化性上皮牙源性肿瘤(CEOT)患者的CBCT或非增强螺旋CT图像,和射线照相特征,包括位置,尺寸,扩展,内部结构和钙化,进行了分析。
    结果:在19例COC中(男性12例,女性7例,平均年龄27岁),89.5%(17/19)的病变起源于前磨牙区,其中100.0%表现为皮质扩张,78.9%的人停用皮质。在16例CEOT病例中(男性3例,女性13例,平均年龄36岁),81.3%(13/16)的病灶位于前磨牙和磨牙区,其中56.3%表现为皮质扩张,96.8%的患者出现皮质中断。根据内部钙化的分布,这些病变分为:Ⅰ(非钙化型):无钙化;Ⅱ(偏心边缘型):沿病变一侧散布的多个钙化;Ⅲ(弥散型):大量钙化弥散分布在病变中;Ⅳ(斑块型):钙化斑块≥5mm;Ⅴ(冠状周围型):多个钙化聚集在阻生牙齿周围。73.7%的COC病变存在钙化,其中Ⅱ型9个,3个Ⅲ型和2个Ⅳ型病变,42.8%的CEOT病变有钙化图像,其中Ⅲ型病变2例,Ⅴ型病变5例。六个COC病变具有牙瘤样图像。此外,9例Ⅰ型CEOTs中有8例组织学上为Langerhans细胞丰富亚型,其具有较小的尺寸(平均中远侧直径为17.8mm)并且与阻生牙齿无关。
    结论:COC病变倾向于起源于颌骨的前部,并表现出皮质扩张,有时与牙瘤有关。CEOT通常发生在后颌骨,并已中断皮质。两个病变的钙化图明显不同。超过70%的COC病变有钙化图像,大部分分散在囊肿的一侧,远离受影响的牙齿。大约60%的CEOT病变表现出较小的尺寸和非钙化,其余的CEOT病例通常有钙化图像聚集在受累牙齿周围。
    OBJECTIVE: To analyze the three-dimensional radiographic characteristics of calcifying odontogenic cyst and calcifying epithelial odontogenic tumor using spiral computed tomography (CT) and cone-beam computed tomography (CBCT).
    METHODS: Clinical records, histopathological reports, and CBCT or non-enhanced spiral CT images of 19 consecutive patients with calcifying odontogenic cyst (COC) and 16 consecutive patients with calcifying epithelial odontogenic tumor (CEOT) were retrospectively acquired, and radiographic features, including location, size, expansion, internal structure and calcification, were analyzed.
    RESULTS: Among the 19 COC cases (12 males and 7 females, with an average age of 27 years), 89.5% (17/19) of the lesions originated from the anterior and premolar areas, 100.0% of them exhibited cortex expansion, and 78.9% had discontinued cortex. Among the 16 CEOT cases (3 males and 13 females, with an average age of 36 years), 81.3% (13/16) of the lesions were in the premolar and molar areas, 56.3% of them exhibited cortex expansion, and 96.8% had discontinued cortex. According to the distribution of internal calcifications, these lesions were divided into: Ⅰ (non-calcification type): absence of calcification; Ⅱ (eccentric marginal type): multiple calcifications scattered along one side of the lesion; Ⅲ (diffused type): numerous calcifications diffusely distributed into the lesion; Ⅳ (plaque type): with a ≥ 5 mm calcified patch; Ⅴ (peri-coronal type): multiple calcifications clustered around impacted teeth. Calcifications were present in 73.7% of COC lesions, including 9 type Ⅱ, 3 type Ⅲ and 2 type Ⅳ lesions, and 42.8% of CEOT lesions had calcification images, including 2 type Ⅲ and 5 type Ⅴ lesions. Six COC lesions had odontoma-like images. Moreover, 8 of 9 type Ⅰ CEOTs were histologically Langerhans cell-rich subtype, which had a smaller size (with an average mesiodistal diameter of 17.8 mm) and were not associated with impacted teeth.
    CONCLUSIONS: COC lesions tended to originate from the anterior part of the jaw and exhibit cortex expansion, and were sometimes associated with odontoma. CEOT commonly occurred in the posterior jaw and had discontinued cortex. Two lesions had significantly different calcification map. Over 70% of COC lesions had calcification images, which were mostly scattered along one side of the cysts, far from the impacted teeth. Approximately 60% of CEOT lesions exhibited smaller size and non-calcification, and the remaining CEOT cases often had calcification images clustered around the impacted teeth.
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  • 文章类型: Case Reports
    最近,随着WHO2022年头颈部肿瘤分类,CEOT非钙化变体的报告激增。本病例描述了一种罕见的CEOT组织病理学变异,其中报道了少数病例。
    这项工作旨在报告一例罕见的非钙化型Pindborg肿瘤累及右侧下颌骨。钙化上皮牙源性肿瘤中钙化的缺乏提出了诊断挑战和预后意义。对16例报道的这种罕见变异的临床病例进行了文献综述。
    UNASSIGNED: There has been a recent spike in reporting of noncalcifying variants of CEOT with the WHO 2022 classification of head and neck tumors. The present case describes a rare histopathological variant of CEOT of which a handful of cases have been reported.
    UNASSIGNED: This work aimed to report a rare case of noncalcifying type of Pindborg Tumor involving the right mandible. The absence of calcifications in the calcifying epithelial odontogenic tumor presents a diagnostic challenge and prognostic implications. A literature review of the 16 reported clinical cases of this scarce variant was performed.
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  • 文章类型: Case Reports
    钙化上皮牙源性肿瘤(CEOT),也被称为Pindborg肿瘤,是一种罕见的牙源性良性肿瘤.它于1946年由Thoma和Goldman首次报道,并于1957年被Pindborg定义为独立的肿瘤。在这里,我们报道了一例CEOT病例,涉及一名53岁男性患者I-125植入后大部分下颌骨.我们与政府和医院部门合作切除肿瘤,用腓骨皮瓣移植重建下颌骨,并妥善处理放射性粒子.
    Calcifying Epithelial Odontogenic Tumor (CEOT), also known as Pindborg tumor, is a rare odontogenic benign tumor. It was first reported by Thoma and Goldman in 1946 and defined as an independent tumor by Pindborg in 1957. Herein, we reported a CEOT case involving most of the mandible after I-125 implantation in a 53-year-old man. We cooperated with governmental and hospital departments to resect the tumors, reconstruct the mandible with a fibular flap graft, and properly dispose of the radioactive particles.
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  • 文章类型: Case Reports
    良性纤维骨病变是一组病理状况,其特征是用经历矿化的细胞纤维结缔组织代替正常骨。最常见的良性纤维骨病变类型包括纤维发育不良,骨化性纤维瘤,骨发育不良.然而,诊断这些病变可能具有挑战性,因为它们的临床重叠,放射学,和组织学特征,这会给外科医生带来诊断困境,放射科医生,和病理学家。一种罕见的良性纤维骨性病变是骨水泥骨化性纤维瘤(COF),这是一种影响颅面区域的良性纤维骨性肿瘤的确定形式,尤其是下巴(70%)。这里,我们介绍了一名61岁的上颌前区女性患者的COF病例。由于病变和健康骨骼之间有明确的区别,病灶采用保守性手术切除,然后进行刮宫和一期闭合。然而,由于COF与Paget病和纤维发育不良等其他纤维骨性病变具有重叠特征,因此其鉴别诊断对临床医生来说具有很大的挑战性。骨化性纤维瘤和纤维发育不良通常表现为组织病理学,临床,和放射学重叠。八个月后的术后随访是不可预测的,放射学照片显示额骨厚度增加,顶骨,上颌骨骨髓间隙闭塞,用棉绒/毛玻璃外观改变小梁图案,上颌窦空间缩小.在得出最终结论之前,必须对纤维骨病变进行适当的评估和诊断。颌面部骨骼中的骨化性纤维瘤并不常见,八个月后,复发率是罕见的。该病例强调了将COF作为颌面部纤维骨病变的鉴别诊断的重要性,以及正确评估和诊断以确定适当的治疗计划和预后的必要性。总之,良性纤维骨病变的诊断可能是具有挑战性的,由于它们的重叠特征,但早期诊断和正确评估对于成功的治疗结果至关重要.COF是一种罕见的良性纤维骨性病变,应将颌面部区域的其他纤维骨性病变视为鉴别诊断。在得出最终结论之前,应采取必要的步骤确认诊断。
    Benign fibro-osseous lesions are a group of pathological conditions characterized by the replacement of normal bone with cellular fibrous connective tissue that undergoes mineralization. The most common types of benign fibro-osseous lesions include fibrous dysplasia, ossifying fibroma, and osseous dysplasia. However, diagnosing these lesions can be challenging due to their overlapping clinical, radiological, and histological features, which can cause a diagnostic dilemma for surgeons, radiologists, and pathologists. One rare type of benign fibro-osseous lesion is the cemento-ossifying fibroma (COF), which is a definitive form of a benign fibro-osseous tumor that affects the craniofacial region, particularly the jaws (70%). Here, we present a case of COF in a 61-year-old female patient in the maxillary anterior region. Due to a clear distinction between the lesion and healthy bone, the lesion was treated with conservative surgical excision followed by curettage and primary closure. However, differential diagnosis of COF can be highly challenging for clinicians due to its overlapping features with other fibro-osseous lesions like Paget\'s disease and fibrous dysplasia. Ossifying fibroma and fibrous dysplasia often present a histopathological, clinical, and radiological overlap. The post-operative follow-up after eight months was unpredictable, with a radiological picture showing the increased thickness of the frontal bone, parietal bone, and maxilla with obliteration of marrow spaces, alteration of the trabecular pattern with a cotton wool/ground glass appearance, and reduced maxillary sinus space. Proper evaluation and diagnosis of fibro-osseous lesions are necessary before arriving at a final conclusion. Cemento-ossifying fibroma in the maxillofacial skeleton is uncommon, and after eight months, the recurrence rate is rare. This case highlights the importance of considering COF as a differential diagnosis for fibro-osseous lesions in the maxillofacial region and the necessity for proper evaluation and diagnosis to determine the appropriate treatment plan and prognosis. In summary, the diagnosis of benign fibro-osseous lesions can be challenging due to their overlapping features, but early diagnosis and proper evaluation are essential for successful treatment outcomes. COF is a rare type of benign fibro-osseous lesion where other fibro-osseous lesions in the maxillofacial region should be considered as a differential diagnosis, and the necessary steps should be taken to confirm the diagnosis before arriving at a final conclusion.
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  • 文章类型: Journal Article
    多项研究表明,胰岛素样生长因子1(IGF-I)水平的升高或其结合蛋白水平的改变与最常见癌症的风险增加有关。包括结直肠癌,肺,乳房和前列腺。这项研究的目的是研究IGF-1在钙化上皮牙源性肿瘤(CEOT)和成釉细胞瘤中的表达。
    研究样本由来自口腔病理科的23块石蜡组成,牙科学院,大马士革大学,包括六个CEOT活检,对丛状成釉细胞瘤进行了两次活检,对滤泡性成釉细胞瘤进行了14次活检。制备所有标本并用兔IGF-1多克隆抗体进行免疫染色。根据德国半定量评分系统评估免疫染色,然后使用SPSS13.0版对结果进行汇总和统计分析(对两个独立样本进行学生检验,单向方差分析,Kruskal-Wallis检验和Mann-WhitneyU检验),显著性水平P值小于0.05被认为具有统计学意义.
    所有CEOT和成釉细胞瘤样本的IGF-1染色呈阳性,除了一个成釉细胞瘤样本,这是负面的。结果显示,CEOT和成釉细胞瘤之间的IGF-1表达值(P=0.993)和IGF-1表达率(P=0.874),IGF-1表达频率(P=0.761)和IGF-1染色强度评分(P=0.731)均无统计学差异。
    IGF-1在牙源性肿瘤的生长中起重要作用,CEOT和成釉细胞瘤之间的IGF-1表达没有差异。
    Several studies have indicated that increased levels of insulin-like growth factor 1 (IGF-I) or altered levels of its binding proteins are associated with an increased risk of the most common cancers, including cancers of the colorectal, lung, breast and prostate. The aim of this study is to investigate the expression of IGF-1 in both calcifying epithelial odontogenic tumor (CEOT) and ameloblastoma.
    UNASSIGNED: The research sample consisted of 23 paraffin block from Oral Pathology Department, Faculty of Dentistry, Damascus University, including six biopsies of CEOT, two biopsies of the plexiform ameloblastoma and 14 biopsies of the follicular ameloblastoma. All specimen were prepared and immunostained with rabbit polyclonal antibody to IGF-1. Immunostaining was assessed according to German-semiquantitative scoring system, and then the results were aggregated and statistically analyzed using SPSS version 13.0 (Student\'s test for two independent samples, one-way analysis of variance, Kruskal-Wallis test and Mann-Whitney U-test), the significance level P value less than 0.05 was considered statistically significant.
    UNASSIGNED: All CEOT and ameloblastoma samples were positive for IGF-1 staining, except one sample of ameloblastoma, which was negative. The results showed that there were no statistically significant differences between CEOT and ameloblastoma in the values of IGF-1 expression (P=0.993) and IGF-1 expression rates (P=0.874) and in the frequency of IGF-1 expression (P=0.761) and IGF-1 staining intensity score (P=0.731).
    UNASSIGNED: IGF-1 plays an important role in the growth of odontogenic tumors and there are no differences in IGF-1 expression between CEOT and ameloblastoma.
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  • 文章类型: Case Reports
    牙源性肿瘤是可以影响任何个体的无声疾病。一种这样的病变是钙化性上皮牙源性肿瘤(CEOT)。这是一种罕见的病变,具有局部侵袭性。在过去的7-8个月中,一名33岁的男性出现鼻塞和左眼持续流泪。临床检查显示左眼球略微突出,眼球水平改变,持续有水样放电。赫斯图表证实了正常的眼球运动。口内发现并不显著。根据组织病理学方面确定CEOT的最终诊断。CEOT主要出现在生命的第三个到第五个十年,没有性别偏好。表现出的症状和体征包括无痛的膨胀性肿块,尽管有与疼痛有关的报道,鼻塞,鼻出血,和突起。在这篇文章中,我们想提出一个CEOT的情况下,以泪液和鼻塞为主要和唯一的表现,一个不寻常的发现.此外,讨论并提出了一个问题,即需要适当的随访期才能消除复发的存在。人们必须保持警惕,不仅要证明与之相关的最常见疾病的单一症状,而且还要探索鲜为人知的可能性。此外,我们需要进一步深入研究,以确定一定的持续时间,之后复发的可能性最小。
    Odontogenic tumors are silent ailments which can affect any individual. One such lesion is a calcifying epithelial odontogenic tumor (CEOT). It is a rare lesion with a locally aggressive nature. A 33-year-old male presented with nasal blockage and continuous flow of tears from the left eye for the past 7-8 months. Clinical examination revealed slightly protruded left eyeball and altered level of eyeballs with continuous watery discharge. Hess chart confirmed normal eye movements. Intraoral findings were not significant. The final diagnosis of CEOT was established based on the histopathological aspects. CEOT is mostly found in the third to fifth decade of life without gender predilection. Presenting symptoms and signs comprise painless expansile mass, although there are reports associated with pain, nasal obstruction, epistaxis, and proptosis. In this article, we would like to present a case of CEOT with epiphora and nasal blockage as the main and only presentation, an uncommon finding. Furthermore, discussing and posing a question of an adequate period of follow-up required to negate the presence of recurrence. One must stay vigilant enough not only to attest a singular symptom to the commonest ailment related to it but also to explore the possibility of the less known. Furthermore, we need to further research in depth to establish a certain duration after which the likeliness of recurrence is to the minimum.
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  • 文章类型: Case Reports
    颌面部囊性病变的诊断和治疗需要仔细评估并结合临床表现和放射学研究。Pindborg肿瘤,也被称为钙化上皮牙源性肿瘤,是一种局部侵袭性良性肿瘤,到目前为止,只有大约300个案例被公布。这项研究提出了这种已经不常见的肿瘤的新病例,与受影响的牙齿无关,并描述了这种罕见实体的临床病理特征,以及对其他报告病例的审查。尽管手术被认为是Pindborg肿瘤的首选治疗方法,关于手术切除的扩展尚无明确的共识。
    The diagnosis and treatment of maxillofacial cystic lesions requires careful evaluation and correlation of the clinical presentation and radiological studies. The Pindborg tumor, also known as the calcifying epithelial odontogenic tumor, is a locally invasive benign neoplasm, with only around 300 cases being published to date. This study presents a new case of this already uncommon neoplasm, not associated with an impacted tooth, and describes the clinicopathological features of this rare entity, along with a review of other reported cases. Despite surgery having been recognized as the treatment of choice for the Pindborg tumor, no firm consensus exists concerning the extension of surgical resection.
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  • DOI:
    文章类型: Journal Article
    一名60岁的女性出现无症状的下颌牙种植体失败。计算机断层扫描(CT)显示部分钙化,软组织内的低信号损伤,沿着颊皮层测量1.3x0.8x1.0厘米。切开活检显示为基底细胞样肿瘤,由具有卵圆形核的细胞片组成,不同的核仁,和缺乏嗜酸性粒细胞的细胞质。出现了Mitoses,平均每10个高功率场约有2个,有分散的单个凋亡细胞。观察到许多层状钙化体(Liesegang环),这些体汇合形成更大的营养不良矿化灶。这些特征清楚地确立了该肿瘤的恶性性质。免疫组织化学,肿瘤的突触素呈阳性,CAM5.2为局灶性阳性,Ki-67增殖指数约为25%.这是首次报道具有钙化上皮牙源性肿瘤和神经内分泌分化的恶性变体特征的肿瘤。
    A 60-year-old female presented with asymptomatic failing mandibular dental implants. Computed tomography (CT) showed a partially calcified, hypointense lesion within the soft tissues, measuring 1.3 x 0.8 x 1.0 cm along the buccal cortex. Incisional biopsy demonstrated a basaloid type of tumor composed of sheets of cells with plump ovoid nuclei, distinct nucleoli, and scant eosinophilic cytoplasm. Mitoses were present, averaging about 2 per 10 high power fields with scattered individual apoptotic cells. Numerous laminated calcified bodies (Liesegang rings) were observed with confluence of these bodies to form larger foci of dystrophic mineralization. These features clearly established the malignant nature of this tumor. Immunohistochemically, the tumor was positive for synaptophysin, focally positivity for CAM 5.2 and had a Ki-67 proliferation index of approximately 25%. This is the first report of a tumor with features of a malignant variant of calcifying epithelial odontogenic tumor and neuroendocrine differentiation.
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  • 文章类型: Journal Article
    钙化上皮牙源性肿瘤是一种罕见的良性肿瘤,约占所有牙源性肿瘤的1%。大多数病例发生在下颌骨后部,还有一些涉及上颌骨。尽管它们的生物学行为相对惰性,上颌骨的肿瘤倾向于快速生长。我们报告了一名33岁的女性患者右上颌骨肿胀的情况。在影像学检查中发现了与受影响的多余牙齿相关的等密度区域。组织病理学诊断为钙化上皮牙源性肿瘤。选择的治疗方法是手术切除病变和相关的牙科元件。患者随访11个月,无复发迹象。除了描述这个案子,我们回顾了有关钙化性上皮牙源性肿瘤与多生牙的关系的文献,并发现了两个针对该主题的病例报告。
    The calcifying epithelial odontogenic tumor is a rare benign neoplasm that accounts for approximately 1% of all odontogenic tumors. Most of the cases occur in the posterior mandible, and a few involve the maxilla. Despite their relatively indolent biological behavior, tumors in the maxilla tend to grow fast. We report the case of a 33-year-old female patient exhibiting swelling in the right maxilla. An isodense area associated with an impacted supernumerary tooth was found on imaging examination. The histopathologic diagnosis was a calcifying epithelial odontogenic tumor. The treatment of choice was surgical removal of the lesion and associated dental elements. The patient has been followed up for 11 months and shows no signs of recurrence. Besides describing this case, we reviewed the literature on the association of calcifying epithelial odontogenic tumors with supernumerary teeth and found two case reports addressing this subject.
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  • 文章类型: Journal Article
    最近已经认识到钙化上皮牙源性肿瘤的非钙化朗格汉斯细胞丰富变体(NCLC-CEOT)和中央牙源性纤维瘤的淀粉样蛋白丰富变体(AR-COF)的重叠临床病理特征。这两种疾病是否确实是一个独特的疾病实体还是属于CEOT和COF,仍在争论中,分别。为了澄清这个问题,我们进行了文献综述,比较了NCLC-CEOT之间临床病理特征的异同,AR-COF,经典CEOT,经典COF我们旨在调查NCLC-CEOT和AR-COF是否可能是相同的和一个独特的疾病实体,或CEOT或COF的变体(或多个变体);或COF,NCLC-CEOT/AR-COF,CEOT代表一种疾病的组织病理学谱。我们的结果表明,NCLC-CEOT和AR-COF病例具有许多相似的临床病理特征。因此,我们认为它们是同一疾病实体。由于NCLC-CEOT/AR-COF病例几乎没有复发报告,保守手术治疗是合适的。NCLC-CEOT/AR-COF病例与COF而非CEOT病例表现出一些重叠的临床病理特征。然而,临床病理特征的差异仍然在NCLC-CEOT/AR-COF之间得到认可,COF,CEOT案件。未来的研究,特别是分子生物学研究,可以进一步阐明它们之间的关系,并有助于NCLC-CEOT/AR-COF病例的正确分类。
    Overlapping clinicopathological features of non-calcifying Langerhans cell rich variant of calcifying epithelial odontogenic tumor (NCLC-CEOT) and the amyloid rich variant of the central odontogenic fibroma (AR-COF) have been recognized recently. It is still under debate whether these two diseases are indeed one unique disease entity or belong to CEOT and COF, respectively. To clarify this issue, we have performed a literature review to compare the similarities and differences in clinicopathological features among NCLC-CEOT, AR-COF, classic CEOT, and classic COF. We aimed to investigate whether NCLC-CEOT and AR-COF might be the same and one distinctive disease entity, or a variant (or variants) of either CEOT or COF; or whether COF, NCLC-CEOT/AR-COF, and CEOT represented a histopathological spectrum of one disease. Our results indicate that NCLC-CEOT and AR-COF cases share many similar clinicopathological features. Thus, we suggest that they are the same disease entity. Due to nearly no reported recurrence of NCLC-CEOT/AR-COF cases, the conservative surgical treatment is appropriate. The NCLC-CEOT/AR-COF cases show some overlapping clinicopathological features with COF rather than the CEOT cases. However, differences in the clinicopathological features are still recognized among the NCLC-CEOT/AR-COF, COF, and CEOT cases. Future research, particularly molecular biological studies, may further elucidate their relationships and assist proper classification of the NCLC-CEOT/AR-COF cases.
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