Primary neoplastic lesions of the temporomandibular joint (TMJ) are rare, with malignant tumors in this region being even more uncommon. The prognosis of maxillofacial malignancies is often poor due to challenges in achieving an adequate surgical margin. Heein, we report a case of TMJ osteosarcoma with an exceptional 34-year disease-free survival following surgery. A 26-year-old patient underwent preoperative chemotherapy and radiation therapy, followed by surgical resection and adjuvant postoperative radiation therapy. Despite significant postoperative cheek deformity, the patient remained free of recurrence or metastasis. We propose that the favorable outcome may be attributed to complete tumor resection facilitated by the TMJ joint capsule, which may have acted as a barrier to tumor spread. This case highlights the potential protective role of the joint capsule in achieving successful outcomes in TMJ osteosarcoma.
{"title":"A primary osteosarcoma of the mandibular articular process with long-term follow-up: A case report","authors":"Tomomi Sakuma , Hiroyuki Yoshitake , Namiaki Takahara , Tohru Ikeda , Tetsuya Yoda","doi":"10.1016/j.ajoms.2025.05.005","DOIUrl":"10.1016/j.ajoms.2025.05.005","url":null,"abstract":"<div><div><span>Primary neoplastic lesions of the temporomandibular joint<span><span> (TMJ) are rare, with malignant tumors in this region being even more uncommon. The prognosis of maxillofacial malignancies is often poor due to challenges in achieving an adequate surgical margin. Heein, we report a case of TMJ </span>osteosarcoma<span> with an exceptional 34-year disease-free survival following surgery. A 26-year-old patient underwent preoperative chemotherapy and radiation therapy, followed by surgical resection and adjuvant postoperative radiation therapy. Despite significant postoperative cheek deformity, the patient remained free of recurrence or </span></span></span>metastasis<span>. We propose that the favorable outcome may be attributed to complete tumor resection facilitated by the TMJ joint capsule, which may have acted as a barrier to tumor spread. This case highlights the potential protective role of the joint capsule in achieving successful outcomes in TMJ osteosarcoma.</span></div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1226-1231"},"PeriodicalIF":0.4,"publicationDate":"2025-05-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907844","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
This study investigated the role of osteopontin (OPN), a secreted phosphoglycoprotein, in radioresistance in oral squamous cell carcinoma (OSCC).
Methods
Samples were collected from 66 patients with OSCC. Immunohistochemistry and double staining were performed on tissue sections to assess OPN levels in stromal and tumor cells and OPN and CD163 colocalization, respectively. Cell lines were treated to simulate cancer-associated environments. Macrophage and fibroblast differentiation were induced; enzyme-linked immunosorbent assays, in situ hybridization, and western blotting were performed; and reactive oxygen species (ROS) were detected to evaluate protein levels and cell responses. Cells were also irradiated for survival analysis.
Results
Immunohistochemical analysis of OSCC tissue samples revealed OPN localization predominantly in the tumor stroma, particularly in tumor-associated macrophages (TAMs). In vitro experiments demonstrated that TAM-like cells differentiated from THP-1 monocytes secreted higher OPN levels compared with cancer-associated fibroblast-like cells. Clinicopathological analysis revealed a significant correlation between high OPN expression and poor chemoradiotherapy response. The addition of recombinant OPN to OSCC cell lines decreased radiosensitivity, which was reversed by blocking OPN signaling using anti-CD44 and anti-integrin αVβ3 antibodies. Furthermore, OPN treatment reduced radiation-induced DNA double-strand breaks and ROS production and increased glutathione levels, which were inhibited by blocking OPN-CD44 and OPN-integrin αVβ3 pathways.
Conclusions
OPN secreted by the tumor stroma, particularly TAMs, contributes to OSCC radioresistance by regulating ROS and GSH levels via the OPN-CD44 and OPN-integrin αVβ3 axes. Therefore, targeting OPN signaling may represent a novel therapeutic strategy for overcoming radioresistance in OSCC.
目的探讨骨桥蛋白（OPN）在口腔鳞状细胞癌（OSCC）放射耐药中的作用。方法对66例OSCC患者进行标本采集。对组织切片进行免疫组化和双染色，分别评估间质细胞和肿瘤细胞中的OPN水平以及OPN和CD163的共定位。细胞系被处理以模拟癌症相关的环境。诱导巨噬细胞和成纤维细胞分化；进行酶联免疫吸附试验、原位杂交和western blotting；并检测活性氧（ROS）来评估蛋白水平和细胞反应。细胞也被照射以进行存活分析。结果OSCC组织样本的免疫组化分析显示，OPN主要定位于肿瘤间质，特别是肿瘤相关巨噬细胞（tam）。体外实验表明，与癌症相关的成纤维细胞样细胞相比，THP-1单核细胞分化的tam样细胞分泌更高的OPN水平。临床病理分析显示高OPN表达与放化疗反应差有显著相关性。在OSCC细胞系中添加重组OPN可降低放射敏感性，而使用抗cd44和抗整合素αVβ3抗体阻断OPN信号传导可逆转这一现象。此外，通过阻断OPN- cd44和OPN-整合素α v - β3通路，OPN处理降低了辐射诱导的DNA双链断裂和ROS产生，并增加了谷胱甘肽水平。结论肿瘤基质分泌的sopn，特别是tam，通过OPN-CD44和OPN-integrin α v - β3轴调控ROS和GSH水平，参与OSCC的耐辐射作用。因此，靶向OPN信号可能是克服OSCC放射耐药的一种新的治疗策略。
{"title":"Osteopontin derived from tumor-associated macrophages modulates radiosensitivity of oral squamous cell carcinoma via reactive oxygen species production","authors":"Hikaru Nakashima , Keisuke Yamana , Ryoji Yoshida , Yuichiro Matsuoka , Hidetaka Arita , Shunsuke Gohara , Masatoshi Hirayama , Kenta Kawahara , Akiyuki Hirosue , Yoshikazu Kuwahara , Tomohiko Wakayama , Hideki Nakayama","doi":"10.1016/j.ajoms.2025.05.008","DOIUrl":"10.1016/j.ajoms.2025.05.008","url":null,"abstract":"<div><h3>Objective</h3><div>This study investigated the role of osteopontin (OPN), a secreted phosphoglycoprotein, in radioresistance in oral squamous cell carcinoma (OSCC).</div></div><div><h3>Methods</h3><div>Samples were collected from 66 patients with OSCC. Immunohistochemistry and double staining were performed on tissue sections to assess OPN levels in stromal and tumor cells and OPN and CD163 colocalization, respectively. Cell lines were treated to simulate cancer-associated environments. Macrophage and fibroblast differentiation were induced; enzyme-linked immunosorbent assays, in situ hybridization, and western blotting were performed; and reactive oxygen species (ROS) were detected to evaluate protein levels and cell responses. Cells were also irradiated for survival analysis.</div></div><div><h3>Results</h3><div>Immunohistochemical analysis of OSCC tissue samples revealed OPN localization predominantly in the tumor stroma, particularly in tumor-associated macrophages (TAMs). In vitro experiments demonstrated that TAM-like cells differentiated from THP-1 monocytes secreted higher OPN levels compared with cancer-associated fibroblast-like cells. Clinicopathological analysis revealed a significant correlation between high OPN expression and poor chemoradiotherapy response. The addition of recombinant OPN to OSCC cell lines decreased radiosensitivity, which was reversed by blocking OPN signaling using anti-CD44 and anti-integrin αVβ3 antibodies. Furthermore, OPN treatment reduced radiation-induced DNA double-strand breaks and ROS production and increased glutathione levels, which were inhibited by blocking OPN-CD44 and OPN-integrin αVβ3 pathways.</div></div><div><h3>Conclusions</h3><div>OPN secreted by the tumor stroma, particularly TAMs, contributes to OSCC radioresistance by regulating ROS and GSH levels via the OPN-CD44 and OPN-integrin αVβ3 axes. Therefore, targeting OPN signaling may represent a novel therapeutic strategy for overcoming radioresistance in OSCC.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1186-1195"},"PeriodicalIF":0.4,"publicationDate":"2025-05-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907841","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-05-14DOI: 10.1016/j.ajoms.2025.05.006
Hideki Ichikawa , Junko Yagisawa , Junichi Tanaka , Hiroyuki Kato , Aki Ito
Melanotic neuroectodermal tumor of infancy (MNTI) is a rare pigmented tumor of neural crest origin, commonly occurring in the head and neck region. Despite being a benign tumor, MNTI is characterized by high cell proliferation activity and can sometimes enlarge while destroying surrounding tissues. Herein, we report a case of MNTI in a 3-month-old girl who exhibited rapid tumor growth following a biopsy; we have included a discussion of the case and review of the relevant literature. Upon initial examination, a 20-mm (long axis) elastic hard mass was observed in the left upper alveolar region extending to the oral vestibule. The mucosal surface was smooth, and submucosal blue-gray tissue was visible. A biopsy was performed under general anesthesia, but the lesion rapidly enlarged into a grayish-white, irregular mass within a few days. Subsequently, a diagnosis of MNTI was made, and 14 days after the biopsy, the patient underwent a left maxillary tumor resection and surrounding bone removal under general anesthesia. Following the surgery, a pediatric prosthesis was timely fabricated by the Department of Pediatric Dentistry, resulting in both morphological and functional recovery. Treatment options should carefully consider factors related to quality of life to address potential facial deformities that may become more prominent with the subsequent growth of the child.
{"title":"Rapid growth of a melanotic neuroectodermal tumor after biopsy in a 3-month-old infant","authors":"Hideki Ichikawa , Junko Yagisawa , Junichi Tanaka , Hiroyuki Kato , Aki Ito","doi":"10.1016/j.ajoms.2025.05.006","DOIUrl":"10.1016/j.ajoms.2025.05.006","url":null,"abstract":"<div><div>Melanotic neuroectodermal tumor of infancy<span><span><span> (MNTI) is a rare pigmented tumor of neural crest origin, commonly occurring in the head and neck region. Despite being a </span>benign tumor<span><span>, MNTI is characterized by high cell proliferation activity and can sometimes enlarge while destroying surrounding tissues. Herein, we report a case of MNTI in a 3-month-old girl who exhibited rapid tumor growth following a biopsy; we have included a discussion of the case and review of the relevant literature. Upon initial examination, a 20-mm (long axis) elastic hard mass was observed in the left upper alveolar region extending to the oral </span>vestibule<span>. The mucosal surface was smooth, and submucosal blue-gray tissue was visible. A biopsy was performed under </span></span></span>general anesthesia<span>, but the lesion rapidly enlarged into a grayish-white, irregular mass within a few days. Subsequently, a diagnosis of MNTI was made, and 14 days after the biopsy, the patient underwent a left maxillary tumor<span><span><span> resection and surrounding bone removal under general anesthesia. Following the surgery, a pediatric<span> prosthesis was timely fabricated by the Department of Pediatric Dentistry, resulting in both morphological and functional recovery. Treatment options should carefully consider factors related to </span></span>quality of life to address potential </span>facial deformities that may become more prominent with the subsequent growth of the child.</span></span></span></div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1232-1238"},"PeriodicalIF":0.4,"publicationDate":"2025-05-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907845","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-05-10DOI: 10.1016/j.ajoms.2025.04.003
S. Sowmya , R. Sangavi , Pradeep kumar yadalam
Objective
Forensic odontology is a field of dentistry that applies dental science to the legal system, examining and analyzing dental evidence in criminal investigations, civil litigation, and other legal proceedings. It plays a crucial role in determining the gender of individuals whose bones have been significantly deformed in a catastrophic event. Forensic dentists use morphological and genetic studies to determine an individual's gender, relying on sexual dimorphism and remnants. The mandible and gonial angle, formed by the lower mandible and ramus, aid in gender determination and age estimation. The study aimed to assess the comparison of neural networks and gradient boosting in the prediction of gender based on gonial angle measurements.
Methods
Two hundred CBCT and 200 OPG images were retrieved from Oral and Maxillofacial Radiology archives, involving 100 males and 100 females. Before evaluation, CBCT scans underwent manual reorientation for standardization. The coronal view was adjusted by aligning the software's vertical reference line with the median sagittal plane. The axial reconstruction line was aligned with the mandibular body. The sagittal reconstruction image thickness was increased to 35 millimeters, with two lines for demarcation of the Gonion point. After obtaining the dataset, outliers were removed and normalized, and data were split into 80 % percent and 20 % percent test data and subjected to gradient boosting and neural networks.
Result
The study compares Neural Networks' and gradient-boosting models' performance on a task, finding that the Neural Network outperformed the latter with an Area Under the Curve (AUC) of 0.922 and a higher F1 score (Harmonic mean of Precision and Recall).
Conclusion
The study demonstrates that the gonial angle, a mandibular measure, can accurately determine gender, with conventional statistical methods and machine learning models predicting it, but with limitations
目的法医牙医学是将牙科学应用于法律体系的一个领域，在刑事调查、民事诉讼和其他法律诉讼中检查和分析牙科学证据。它在确定在灾难性事件中骨骼严重变形的个体的性别方面起着至关重要的作用。法医牙医利用形态学和遗传学研究来确定一个人的性别，依靠两性二态性和残余物。由下颌骨和支组成的下颌骨和角角有助于确定性别和估计年龄。该研究旨在评估神经网络和梯度增强在基于卵形角测量的性别预测中的比较。方法从口腔颌面放射学档案中检索CBCT图像200张，OPG图像200张，男、女各100张。在评估之前，CBCT扫描进行了手动重新定位以标准化。通过将软件的垂直参考线与正中矢状面对齐来调整冠状视图。轴向重建线与下颌骨体对齐。矢状面重建图像厚度增加到35毫米，用两条线划分Gonion点。获得数据集后，对异常值进行去除和归一化处理，将数据分成80% % %和20% % %的测试数据，并进行梯度增强和神经网络处理。结果比较了神经网络和梯度增强模型在某一任务上的表现，发现神经网络的曲线下面积（AUC）为0.922，F1分数（Precision and Recall的调和平均值）更高，优于梯度增强模型。结论下颌角是下颌的一个测量指标，可以准确地判断性别，传统的统计方法和机器学习模型可以预测，但存在局限性
{"title":"Gradient boosting based prediction of gender based on gonial angle measurements","authors":"S. Sowmya , R. Sangavi , Pradeep kumar yadalam","doi":"10.1016/j.ajoms.2025.04.003","DOIUrl":"10.1016/j.ajoms.2025.04.003","url":null,"abstract":"<div><h3>Objective</h3><div>Forensic odontology is a field of dentistry that applies dental science to the legal system, examining and analyzing dental evidence in criminal investigations, civil litigation, and other legal proceedings. It plays a crucial role in determining the gender of individuals whose bones have been significantly deformed in a catastrophic event. Forensic dentists use morphological and genetic studies to determine an individual's gender, relying on sexual dimorphism and remnants. The mandible and gonial angle, formed by the lower mandible and ramus, aid in gender determination and age estimation. The study aimed to assess the comparison of neural networks and gradient boosting in the prediction of gender based on gonial angle measurements.</div></div><div><h3>Methods</h3><div>Two hundred CBCT and 200 OPG images were retrieved from Oral and Maxillofacial Radiology archives, involving 100 males and 100 females. Before evaluation, CBCT scans underwent manual reorientation for standardization. The coronal view was adjusted by aligning the software's vertical reference line with the median sagittal plane. The axial reconstruction line was aligned with the mandibular body. The sagittal reconstruction image thickness was increased to 35 millimeters, with two lines for demarcation of the Gonion point. After obtaining the dataset, outliers were removed and normalized, and data were split into 80 % percent and 20 % percent test data and subjected to gradient boosting and neural networks.</div></div><div><h3>Result</h3><div>The study compares Neural Networks' and gradient-boosting models' performance on a task, finding that the Neural Network outperformed the latter with an Area Under the Curve (AUC) of 0.922 and a higher F1 score (Harmonic mean of Precision and Recall).</div></div><div><h3>Conclusion</h3><div>The study demonstrates that the gonial angle, a mandibular measure, can accurately determine gender, with conventional statistical methods and machine learning models predicting it, but with limitations</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 5","pages":"Pages 921-928"},"PeriodicalIF":0.4,"publicationDate":"2025-05-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144653043","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Abnormal craniofacial morphology in patients with cleft lip and/or palate has been attributed to defects in craniofacial growth potential and to the effects of surgical and orthodontic treatment. The influence of these factors was examined by analyzing craniofacial growth patterns throughout the growth process from infancy to adulthood.
Design
Retrospective longitudinal study
Participants
The subjects were 46 Japanese female CL/P patients who underwent surgical lip and/or palatal repairs and orthodontic treatment at the Kyushu University Dental Hospital, Fukuoka, Japan.
Methods
Using serial cephalometric radiographs from 4 months to 15 + years of age, growth patterns of craniofacial morphology were compared for three cleft types (12 patients with UCLA, 13 CP, and 21 UCLP) using repeated measures ANOVA.
Results
The growth patterns of the patients with UCLP differed from those of the patients with UCLA in the maxillofacial angle (S-N-A), maxillary basal length (A’-Ptm’), posterior maxillary height (S-Ptm’), maxillofacial widths (NC-NC’ and Mx-Mx’), and three maxillofacial ratios (Cd-Go/S-N, Pog-Go/S-N, N-PP/Cd-Go). The growth patterns of the patients with UCLP differed from those of the patients with CP in cranial base lengths (N-Ba and S-Ba), the maxillary basal length, maxillofacial angle, maxillofacial width, and the ratio S-Ptm'/N-PP.
Conclusion
Craniofacial growth patterns varied by the cleft types, the presence of both cleft lip and cleft palate inhibited forward and downward growth of the posterior maxillofacial complex, and surgical intervention on the lips, alveolar bone, and palate had a cumulative inhibitory effect on maxillofacial growth.
{"title":"Craniofacial growth patterns of female patients with cleft lip and/or palate: A longitudinal study from infancy to adolescence","authors":"Akira Suzuki , Naoko Tamura , Masaaki Sasaguri , Takeshi Mitsuyasu , Seiji Nakamura","doi":"10.1016/j.ajoms.2025.05.004","DOIUrl":"10.1016/j.ajoms.2025.05.004","url":null,"abstract":"<div><h3>Objectives</h3><div>Abnormal craniofacial morphology<span><span> in patients with cleft lip and/or palate has been attributed to defects in craniofacial growth potential and to the effects of surgical and </span>orthodontic treatment. The influence of these factors was examined by analyzing craniofacial growth patterns throughout the growth process from infancy to adulthood.</span></div></div><div><h3>Design</h3><div>Retrospective longitudinal study</div></div><div><h3>Participants</h3><div>The subjects were 46 Japanese female CL/P patients who underwent surgical lip and/or palatal repairs and orthodontic treatment at the Kyushu University Dental Hospital, Fukuoka, Japan.</div></div><div><h3>Methods</h3><div>Using serial cephalometric<span> radiographs from 4 months to 15 + years of age, growth patterns of craniofacial morphology were compared for three cleft types (12 patients with UCLA, 13 CP, and 21 UCLP) using repeated measures ANOVA.</span></div></div><div><h3>Results</h3><div><span>The growth patterns of the patients with UCLP differed from those of the patients with UCLA in the maxillofacial angle (S-N-A), maxillary basal length (A’-Ptm’), posterior maxillary height (S-Ptm’), maxillofacial widths (NC-NC’ and Mx-Mx’), and three maxillofacial ratios (Cd-Go/S-N, Pog-Go/S-N, N-PP/Cd-Go). The growth patterns of the patients with UCLP differed from those of the patients with CP in cranial </span>base lengths (N-Ba and S-Ba), the maxillary basal length, maxillofacial angle, maxillofacial width, and the ratio S-Ptm'/N-PP.</div></div><div><h3>Conclusion</h3><div>Craniofacial growth patterns varied by the cleft types, the presence of both cleft lip and cleft palate inhibited forward and downward growth of the posterior maxillofacial complex, and surgical intervention on the lips, alveolar bone, and palate had a cumulative inhibitory effect on maxillofacial growth.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1167-1180"},"PeriodicalIF":0.4,"publicationDate":"2025-05-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907817","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Osteocartilaginous choristomas are benign bone lesion composed of hyaline cartilage that undergo ossification and arise when osteochondral cells proliferate into surrounding tissues. In the oral and maxillofacial regions, these lesions usually occur on the dorsum of the tongue, with no reported cases of osteocartilaginous choristomas occurring on the oral floor. We report the case of a 57-year-old Japanese male referred to our hospital in late November 2021 with the chief complaint of tongue hypoaesthesia and a mass on the right side of the oral floor. Clinical examination revealed that the lesion was a 22 × 13 mm elastic hard mass with an ulcer at the centre of the lesion on the right side of the oral floor behind Walton's canal orifice. Computed tomography images showed an opaque mass with a major diameter of 20 mm, internal heterogeneity, and a well-defined border located slightly to the right of the floor of the oral cavity. Surgical resection was performed through an intraoral incision under local anaesthesia. The imaging and surgical findings at our hospital showed a 22 × 15 mm lesion with clear boundaries, which could be removed as a single mass from the surrounding connective tissue. Histological investigation revealed that lesion has the characteristics of hyaline cartilage with some areas showing ossification, leading to a diagnosis of osteocartilaginous choristoma of the oral floor. The patient has progressed well and remains recurrence-free 2 years postoperatively.
{"title":"A case of osteocartilaginous choristoma at the floor of the oral cavity","authors":"Takasuke Usuki , Soju Seki , Toshihiro Uchihashi , Tomonori Sasahira , Shingo Kodama , Kohei Kawamura , Susumu Tanaka , Syoichiro Ishii , Munehiro Hamaguchi","doi":"10.1016/j.ajoms.2025.05.003","DOIUrl":"10.1016/j.ajoms.2025.05.003","url":null,"abstract":"<div><div><span>Osteocartilaginous choristomas<span><span> are benign bone lesion<span> composed of hyaline cartilage that undergo ossification and arise when osteochondral cells proliferate into surrounding tissues. In the oral and maxillofacial regions, these lesions usually occur on the dorsum of the </span></span>tongue<span>, with no reported cases of osteocartilaginous choristomas occurring on the oral floor. We report the case of a 57-year-old Japanese male referred to our hospital in late November 2021 with the chief complaint of tongue hypoaesthesia and a mass on the right side of the oral floor. Clinical examination revealed that the lesion was a 22 × 13 mm elastic hard mass with an ulcer at the centre of the lesion on the right side of the oral floor behind Walton's canal orifice. Computed tomography images showed an opaque mass with a major diameter of 20 mm, internal heterogeneity, and a well-defined border located slightly to the right of the floor of the </span></span></span>oral cavity<span>. Surgical resection was performed through an intraoral incision<span> under local anaesthesia. The imaging and surgical findings at our hospital showed a 22 × 15 mm lesion with clear boundaries, which could be removed as a single mass from the surrounding connective tissue. Histological investigation revealed that lesion has the characteristics of hyaline cartilage with some areas showing ossification, leading to a diagnosis of osteocartilaginous choristoma of the oral floor. The patient has progressed well and remains recurrence-free 2 years postoperatively.</span></span></div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1299-1303"},"PeriodicalIF":0.4,"publicationDate":"2025-05-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907754","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
In a patient with hematological malignancy, oral microbiome is disturbed during chemotherapy including hematopoietic stem cell transplantation (HSCT). A critical role of salivary microbiota has not been fully investigated.
Methods
We collected saliva of 6 HSCT recipients before and after (around 30 day) transplantation, and characterized patterns of oral microbiota. We set up a healthy control cohort (N = 6) as a normal reference. A two-step PCR protocol was used to amplify a 16S rDNA in the V3-V4 region. Sequencing of the amplicons was performed on an Illumina MiSeq (Illumina). A significance of alpha and beta diversity of microbiota, and taxonomic composition were investigated.
Results
The patients’ characteristics were median age 43 years old (range 39–58) and 4 female patients. The diversity of microorganisms in saliva were significantly decreased after HSCT. Shannon index (t-test, P = 0.03) and Simpson index (P = 0.06) were significantly lower in recipients after HSCT comparing to before HSCT. In recipients experienced severe (>grade 2) oral mucositis, alpha index was less diverse compared to recipients with mild or moderate oral mucositis, and in whose saliva genus Lautropia were predominant after HSCT. A common observation of salivary microbiota after HSCT were significant increase of phylum Actinomycetacea and genus Actinomyces. Genus Neisseria was significantly decreased after HSCT.
Conclusions
We observed substantial decreases in the number and diversity of salivary microbiota through HSCT in recipient with hematological malignancy. Lautropia is newly found microorganism, which is possible to linked to mitigation of severe oral mucositis.
{"title":"Alternation of salivary microbiome and Lautropia dominancy after allogenic hematopoietic stem cell transplantation","authors":"Osamu Imataki , Akihiro Takeuchi , Tomoya Ishida , Makiko Uemura , Minoru Miyake , Yumiko Ohbayashi","doi":"10.1016/j.ajoms.2025.05.001","DOIUrl":"10.1016/j.ajoms.2025.05.001","url":null,"abstract":"<div><h3>Objective</h3><div><span>In a patient with hematological malignancy<span>, oral microbiome is disturbed during chemotherapy including </span></span>hematopoietic stem cell transplantation<span> (HSCT). A critical role of salivary microbiota has not been fully investigated.</span></div></div><div><h3>Methods</h3><div><span>We collected saliva of 6 HSCT recipients before and after (around 30 day) transplantation, and characterized patterns of oral microbiota<span>. We set up a healthy control cohort (N = 6) as a normal reference. A two-step PCR protocol was used to amplify a 16S rDNA<span> in the V3-V4 region. Sequencing of the amplicons was performed on an </span></span></span>Illumina MiSeq<span> (Illumina). A significance of alpha and beta diversity of microbiota, and taxonomic composition were investigated.</span></div></div><div><h3>Results</h3><div><span>The patients’ characteristics were median age 43 years old (range 39–58) and 4 female patients. The diversity of microorganisms in saliva were significantly decreased after HSCT. Shannon index (t-test, P = 0.03) and Simpson index (P = 0.06) were significantly lower in recipients after HSCT comparing to before HSCT. In recipients experienced severe (>grade 2) oral mucositis<span>, alpha index was less diverse compared to recipients with mild or moderate oral mucositis, and in whose saliva genus </span></span><em>Lautropia</em> were predominant after HSCT. A common observation of salivary microbiota after HSCT were significant increase of phylum <em>Actinomycetacea</em> and genus <span><span>Actinomyces</span><em>.</em></span> Genus <span><span>Neisseria</span></span> was significantly decreased after HSCT.</div></div><div><h3>Conclusions</h3><div><span>We observed substantial decreases in the number and diversity of salivary microbiota through HSCT in recipient with hematological malignancy. </span><em>Lautropia</em> is newly found microorganism, which is possible to linked to mitigation of severe oral mucositis.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1251-1256"},"PeriodicalIF":0.4,"publicationDate":"2025-05-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907734","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-05-02DOI: 10.1016/j.ajoms.2025.05.002
Ayumi Yamamoto, Sho Ishida, Yasuo Hanazawa
Juvenile xanthogranuloma (JXG) is a rare non-Langerhans cell histiocytosis that typically presents as a self-limiting dermatological condition in young children, and oral mucosal involvement is exceedingly rare. We report the case of a 2-year-old boy, in whom a pink exophytic nodule gradually enlarged for 3 months and subsequently changed to an orange submucosal mass in approximately 1 month. Excisional biopsy was performed to obtain a definitive diagnosis. Histopathological examination revealed diffuse proliferation of foamy histiocytes and Touton giant cells within the lesion. Based on these results, the patient was diagnosed with JXG. Herein, we present an uncommon case of oral JXG to increase awareness about this lesion. The patient is currently healthy, and no recurrence has been observed 2 years after surgical excision.
{"title":"Juvenile xanthogranuloma of the tongue: A case report","authors":"Ayumi Yamamoto, Sho Ishida, Yasuo Hanazawa","doi":"10.1016/j.ajoms.2025.05.002","DOIUrl":"10.1016/j.ajoms.2025.05.002","url":null,"abstract":"<div><div><span>Juvenile xanthogranuloma<span> (JXG) is a rare non-Langerhans cell histiocytosis that typically presents as a self-limiting dermatological condition<span> in young children, and oral mucosal involvement is exceedingly rare. We report the case of a 2-year-old boy, in whom a pink exophytic nodule gradually enlarged for 3 months and subsequently changed to an orange submucosal mass in approximately 1 month. Excisional biopsy was performed to obtain a definitive diagnosis. Histopathological examination revealed diffuse proliferation of foamy </span></span></span>histiocytes and Touton giant cells within the lesion. Based on these results, the patient was diagnosed with JXG. Herein, we present an uncommon case of oral JXG to increase awareness about this lesion. The patient is currently healthy, and no recurrence has been observed 2 years after surgical excision.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 6","pages":"Pages 1294-1298"},"PeriodicalIF":0.4,"publicationDate":"2025-05-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144907740","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Primary Intraosseous Squamous Cell Carcinoma (PIOSCC) may develop from the dentigerous cyst (DC). The present systematic review aimed at characterizing and analyzing the so-far-published cases of PIOSCC arising from DC.
Methods
A comprehensive search was conducted utilizing PubMed, Web of Science, and Google Scholar for all relevant studies using a variety of relevant keywords. All studies that reported PIOSCC arising from DC were eligible for inclusion. The quality of the included studies was assessed using the JBI Critical Appraisal Tools for case reports.
Results
Twenty-seven case report studies including 29 patients with DC were analyzed. Approximately 79 % of the patients were males, with a mean age of 54 years. One case involved a child who was just 1.4-year-old. The mandible was the most frequently affected jaw, and associated with impacted lower third molars. Two crucial criteria for diagnosis of PIOSCC originating from DC was the transition from normal to abnormal cyst epithelium, and the absence of cystic ameloblastoma, and they were reported in 28 cases (96.6 %). Other two criteria, exclusion of the presence of primary carcinoma elsewhere, and communication with the oral mucosa, were reported less frequently (6 and 18 cases, respectively). Several studies incorporated additional evaluation methods such as immunohistochemical, CT, MRI, and PET-CT. In terms of treatment, surgery alone was the first choice, followed by radiation therapy.
Conclusions
Despite its rarity, PIOSCC can arise from DC. Once DC is suspected, a prompt surgical excision with robust serial microscopic examination of the whole excised lesion should be done.
目的原发性骨内鳞状细胞癌（PIOSCC）可由牙性囊肿（DC）发展而来。本系统综述旨在描述和分析迄今为止发表的由DC引起的PIOSCC病例。方法利用PubMed、Web of Science、b谷歌Scholar等多种相关关键词对所有相关研究进行综合检索。所有报道DC引起的PIOSCC的研究都符合纳入条件。使用JBI病例报告关键评估工具评估纳入研究的质量。结果共分析了27例病例报告，其中29例为DC。约79 %的患者为男性，平均年龄54岁。其中一个案例涉及一个只有1.4岁的孩子。下颌骨是最常受影响的颌骨，并与阻生的下第三磨牙有关。诊断源自DC的PIOSCC的两个关键标准是囊肿上皮从正常向异常的转变，以及没有囊性成釉细胞瘤，28例（96.6% %）报告了这两个标准。其他两个标准，排除其他地方原发癌的存在，以及与口腔粘膜的沟通，报告的频率较低（分别为6例和18例）。一些研究纳入了额外的评估方法，如免疫组织化学、CT、MRI和PET-CT。在治疗方面，手术是首选，其次是放射治疗。结论尽管罕见，PIOSCC也可由DC引起。一旦怀疑DC，应立即手术切除，并对整个切除病变进行严格的连续显微镜检查。
{"title":"Primary intra-osseous squamous cell carcinoma arising from dentigerous cysts: A systematic review of the published case reports","authors":"Esam Halboub , Gamilah Al-Qadhi , Layla Hafed , Faisal Mehsen Alali","doi":"10.1016/j.ajoms.2025.04.013","DOIUrl":"10.1016/j.ajoms.2025.04.013","url":null,"abstract":"<div><h3>Objectives</h3><div>Primary Intraosseous Squamous Cell Carcinoma (PIOSCC) may develop from the dentigerous cyst (DC). The present systematic review aimed at characterizing and analyzing the so-far-published cases of PIOSCC arising from DC.</div></div><div><h3>Methods</h3><div>A comprehensive search was conducted utilizing PubMed, Web of Science, and Google Scholar for all relevant studies using a variety of relevant keywords. All studies that reported PIOSCC arising from DC were eligible for inclusion. The quality of the included studies was assessed using the JBI Critical Appraisal Tools for case reports.</div></div><div><h3>Results</h3><div>Twenty-seven case report studies including 29 patients with DC were analyzed. Approximately 79 % of the patients were males, with a mean age of 54 years. One case involved a child who was just 1.4-year-old. The mandible was the most frequently affected jaw, and associated with impacted lower third molars. Two crucial criteria for diagnosis of PIOSCC originating from DC was the transition from normal to abnormal cyst epithelium, and the absence of cystic ameloblastoma, and they were reported in 28 cases (96.6 %). Other two criteria, exclusion of the presence of primary carcinoma elsewhere, and communication with the oral mucosa, were reported less frequently (6 and 18 cases, respectively). Several studies incorporated additional evaluation methods such as immunohistochemical, CT, MRI, and PET-CT. In terms of treatment, surgery alone was the first choice, followed by radiation therapy.</div></div><div><h3>Conclusions</h3><div>Despite its rarity, PIOSCC can arise from DC. Once DC is suspected, a prompt surgical excision with robust serial microscopic examination of the whole excised lesion should be done.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 5","pages":"Pages 1089-1095"},"PeriodicalIF":0.4,"publicationDate":"2025-04-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144654025","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Eagle’s syndrome is characterized by laryngopharyngeal hypersensitivity, such as sore throat and dysphagia in otorhinolaryngology. It is caused by mechanical pressure from the styloid process that extends to ≥ 25 mm. In most cases, the symptoms appear unilaterally. There are two primary symptoms as follows: (i) classic stylohyoid syndrome, which occurs after tonsillectomy; (ii) stylo-carotid artery syndrome, which involves compression of the carotid artery or sympathetic nerve. We experienced a case of repeated cerebral infarctions due to stylo-carotid artery syndrome. Details of the case are shown below. An elongated styloid process caused three times cerebral infarctions due to internal carotid artery dissection. Internal carotid artery dissection treated with carotid artery stent. But after that, the carotid artery stent was fractured by the styloid process compression. As a result, he suffered a fourth cerebral infarction due to embolization caused by the stent fragments. Using CT angiography and angiography, we diagnosed carotid artery dissection and carotid stent fracture due to an elongated styloid process. In addition to medical therapy for cerebral infarction, we surgically resected the styloid process to prevent recurrence. The scattered stent fragments in the cerebral artery were difficult to remove and were left behind. However, he has not had a recurrence two and a half years. In order to prevent repeated or iatrogenic cerebral infarction, we believe that an elongated styloid process should be resected before stent treatment.
{"title":"A case of cerebral infarction in which a carotid artery stent was fractured by an elongated styloid process(eagle syndrome)","authors":"Nobuhiro Yoshimi , Hiromitsu Hatakeyama , Kazuo Matsui , Noriyuki Katsumata , Satoshi Yamada , Kiyoshi Misawa","doi":"10.1016/j.ajoms.2025.04.012","DOIUrl":"10.1016/j.ajoms.2025.04.012","url":null,"abstract":"<div><div>Eagle’s syndrome is characterized by laryngopharyngeal hypersensitivity, such as sore throat and dysphagia in otorhinolaryngology. It is caused by mechanical pressure from the styloid process that extends to ≥ 25 mm. In most cases, the symptoms appear unilaterally. There are two primary symptoms as follows: (i) classic stylohyoid syndrome, which occurs after tonsillectomy; (ii) stylo-carotid artery syndrome, which involves compression of the carotid artery or sympathetic nerve. We experienced a case of repeated cerebral infarctions due to stylo-carotid artery syndrome. Details of the case are shown below. An elongated styloid process caused three times cerebral infarctions due to internal carotid artery dissection. Internal carotid artery dissection treated with carotid artery stent. But after that, the carotid artery stent was fractured by the styloid process compression. As a result, he suffered a fourth cerebral infarction due to embolization caused by the stent fragments. Using CT angiography and angiography, we diagnosed carotid artery dissection and carotid stent fracture due to an elongated styloid process. In addition to medical therapy for cerebral infarction, we surgically resected the styloid process to prevent recurrence. The scattered stent fragments in the cerebral artery were difficult to remove and were left behind. However, he has not had a recurrence two and a half years. In order to prevent repeated or iatrogenic cerebral infarction, we believe that an elongated styloid process should be resected before stent treatment.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 5","pages":"Pages 1027-1030"},"PeriodicalIF":0.4,"publicationDate":"2025-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144654122","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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