Mark Mintline , Jeffrey Elo , Nickan Zarrabi , Sahar Mirfarsi , Sevin Barghan , Setareh Lavasani
{"title":"Simple bone cyst with unusual root resorption: a case report","authors":"Mark Mintline , Jeffrey Elo , Nickan Zarrabi , Sahar Mirfarsi , Sevin Barghan , Setareh Lavasani","doi":"10.1016/j.oooo.2025.04.047","DOIUrl":"10.1016/j.oooo.2025.04.047","url":null,"abstract":"<div><div>This case report presents an unusual case of a gnathic simple bone cyst (SBC) with significant tooth resorption of a mandibular second molar. A 30-year-old male presented to the dental school clinic for further evaluation of an incidental periapical radiographic finding of a radiolucent lesion in the left mandibular second molar region. The lesion was not identifiable on his panoramic radiographic imaging 1 year prior. Plain films and cone-beam computed tomography (CBCT) images demonstrated a relatively well-demarcated radiolucent lesion in the left posterior body of the mandible with substantial resorption of the second molar. Based on the aggressive behavior of the lesion, a biopsy was immediately performed. Surgical exploration revealed a bone cavity containing sanguineous fluid with no evidence of a cystic lining or mass lesion consistent with an SBC. Sparse soft tissue fragments were curetted, and pieces of the bone cavity walls were submitted for histopathologic analysis. Histopathologic examination revealed a thin layer of fibrous tissue without an epithelial lining and lamellar bone consistent with an SBC. SBCs of the jaws commonly extend between the roots of teeth without resorption, and a minority of cases show bone expansion. Rarely, gnathic SBCs substantially resorb tooth roots.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e81-e82"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670598","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Solitary fibrous tumor of the buccal mucosa: case presentation and review of the literature","authors":"Eduard Munarov , Tzvi Krupka , Aryeh Mahana , Aaron Yancoskie","doi":"10.1016/j.oooo.2025.04.020","DOIUrl":"10.1016/j.oooo.2025.04.020","url":null,"abstract":"<div><h3>Introduction</h3><div>Solitary fibrous tumor (SFT) occurs primarily in adults and is considered an entity of intermediate potential. Most cases follow a benign clinical course, with select tumors exhibiting malignant features. While SFT can occur at any site, the pleural lining of the thorax is the most common. Occurrence in the oral cavity is infrequent. Although histopathology is diverse from case to case, consistent characteristics include a monomorphic spindle cell population in a fibrous stroma with endothelial-lined channels of differing caliber that may show a stag-horn appearance. Treatment is dependent upon the clinical and microscopic features with complete excision and clinical follow-up being appropriate in most cases. We report an SFT of the right buccal mucosa in a female of 29 years and review the literature.</div></div><div><h3>Materials and Methods</h3><div>A 29-year-old female presented with an oral lesion that had gradually increased in size over 6 months. A nonpainful 1.5 × 1.5 cm spherical mass was identified on the right buccal mucosa.</div></div><div><h3>Results</h3><div>Incisional biopsy was performed under local anesthesia and the tissue was submitted to pathology. Microscopic features included an intact surface mucosa and underlying moderately-dense proliferation of monomorphic spindle cells possessing inconspicuous cytoplasmic borders arranged haphazardly in a fibrovascular stroma. The spindle cells stained strongly positive for CD-34 and STAT-6 and were negative for S-100 protein, confirming a diagnosis of SFT. The patient underwent excision under general anesthesia. Final histopathology showed tumor-free margins. The patient is free of disease 4-months postoperatively. The overall features of this case (patient age of less than 55 years, tumor size less than five cm, and few mitotic figures), predict a favorable clinical course based on Demiccio and colleagues’ criteria.</div></div><div><h3>Conclusion</h3><div>The clinical, histopathological, and immunohistochemical features of an SFT in a 29-year-old female are presented and the literature is reviewed. Management and follow-up are discussed.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e73"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670785","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Wattawan Wongpattaraworakul, Allen Choi, Marisa Buchakjia, Emily Lanzel, Andrean Simons-Burnett, Anand Rajan
{"title":"Prognostic value of PD-L1 expression in oral squamous cell carcinoma","authors":"Wattawan Wongpattaraworakul, Allen Choi, Marisa Buchakjia, Emily Lanzel, Andrean Simons-Burnett, Anand Rajan","doi":"10.1016/j.oooo.2025.04.021","DOIUrl":"10.1016/j.oooo.2025.04.021","url":null,"abstract":"<div><h3>Background</h3><div>Programmed cell death protein ligand-1 (PD-L1) is an immune checkpoint protein. In cancer, PD-L1 expression inhibits immunological control leading to tumor cells avoiding immune destruction. In oral squamous cell carcinoma (OSCC), previous literature showed conflicting prognostic value of PD-L1 among studies potentially due to different clones of antibody, differences in scoring methods, and definitions of positive expression. Additionally, PDL1 could be expressed both on immune cells and cancer cells. The distribution of the cell positivity is different across different OSCC cases. Here, we aim to evaluate the prognostic role of PD-L1 expression in OSCC using an FDA-approved antibody and the associated recommended scoring criteria. We also evaluated prognostic significant of PDL1 based on the localization of positive cells.</div></div><div><h3>Methods</h3><div>We analyzed PD-L1 expression by immunohistochemistry in tissue microarrays containing 347 OSCC using antibody clone 22C3 pharmDx and combined positive score (CPS) criteria. Different cutoff points of PD-L1 expression were used to assess progression-free (PFS) and overall patient survival (OS). Survival outcomes were analyzed using Kaplan–Meier plots and log-rank tests.</div></div><div><h3>Results</h3><div>PD-L1 expression had no impact on survival in patients treated with surgery and radiation. However, in OSCC treated with surgery alone, OSCC with CPS ≥ 70 was associated with worse PFS. Interestingly, regardless of CPS, OSCC with PDL1 positivity exclusively/mostly on cancer cells was also associated with poor PFS. Even in cases with PDL1 < 70, if the PDL1 positivity exclusively/mostly expressed on cancer cells, the PFS is as worse as OSCC with CPS ≥ 70.</div></div><div><h3>Conclusion</h3><div>PD-L1 expression using anti-PD-L1 antibody (22C3) with the associated CPS scoring criteria can predict PFS in OSCC patients who received surgery alone. Additionally, the cellular localization of PD-L1 levels appears to influence PFS: high levels of expression in cancer cells alone appear to portend a worse prognosis.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e73-e74"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670786","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Subacute necrotizing sialadenitis: 3 new cases with review of literature","authors":"Nagamani Narayana","doi":"10.1016/j.oooo.2025.04.017","DOIUrl":"10.1016/j.oooo.2025.04.017","url":null,"abstract":"<div><div>The main objective of this poster is to inform clinical dentists regarding subacute necrotizing sialadenitis (SAN). It was first described in 1985 as an unusual inflammatory condition affecting minor salivary glands primarily seen in the palate. It has also shown to present in the buccal mucosa, upper lip, and ventral tongue. SANs present as a symptomatic mass of few days duration, resolving in a week in contrast to necrotizing sialometaplasia which presents with ulceration and takes about 3 weeks to heal. Though there is no consensus regarding the etiology of SAN, all cases resolve without therapy. On searching UNMC College of Dentistry oral pathology service for cases of SANs from 1990 to 2021 (31 years) three cases were found. The age ranged from 16 to 77 years and presented in two males and a female. Two cases were from the palate while one case was from the upper lip. All cases presented with painful palpable masses with no ulceration. Two cases of the palate had a clinical diagnosis of lipoma and adenoma, while the case from the upper lip was called a fibroma. All cases microscopically presented with focal acinar degeneration with a mixed inflammatory infiltrate. No metaplasia of the ductal epithelium was noted. In conclusion, if an erythematous, painful, nonulcerated, palpable palatal mass is noted clinicians should consider SAN in the differential diagnosis.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e72"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144672096","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Andres Flores-Hidalgo , Fatima Zahra Aly , Megumi Williamson , Ricardo Padilla
{"title":"Immunohistochemical and molecular analysis of the inflammatory microenvironment of oral mucosal potentially malignant disorders","authors":"Andres Flores-Hidalgo , Fatima Zahra Aly , Megumi Williamson , Ricardo Padilla","doi":"10.1016/j.oooo.2025.04.010","DOIUrl":"10.1016/j.oooo.2025.04.010","url":null,"abstract":"<div><h3>Introduction</h3><div>Recent studies have shown that the nature of the tumor microenvironment and infiltrating immune cells can significantly modify the outcome of genetic aberration in the oral epithelium. Although their role in cancer progression is not fully understood, these infiltrating cells have been strongly correlated with a poor or better prognosis for the patient in many solid tumors. This study analyzes the phenotypic and molecular expression of immune infiltrating cells in OPMD and known histopathologic mimickers.</div></div><div><h3>Materials and Methods</h3><div>Twenty archived tissue samples were selected from ECU, and UNC Oral and Maxillofacial Pathology Laboratories diagnosed with moderate to severe oral epithelial dysplasia (OED). Dual IHC/DIF staining was carried out on each case with CD4/CD8 and CD163/STAT1 antibodies, and automated histomorphometric digital analysis was performed. The second part of the study involves spatial transcriptomics to identify genes and cell types in FFPE tissue. Bulk RNA sequencing (Illumina, CA) is currently being performed to measure the average gene expression across the population of cells and identify differences between the study groups.</div></div><div><h3>Results</h3><div>Preliminary results show that STAT1/CD163+ macrophages were mainly found underlying the epithelium in OED with similar distribution to CD4+ cells in the same compartment. Intraepithelial CD8+ lymphocyte distribution correlated with the degree of dysplasia in high-grade OED, and that finding correlates with a small presence of STAT1/CD163+ cells. Compared with OLP, an increased presence of inflammatory intraepithelial cells in OED (CD8+ cells and STAT1/CD163+ macrophages) was observed. A significant increase in transcriptional signals appears in OED, specifically from cytotoxic cells.</div></div><div><h3>Conclusions</h3><div>The preliminary results of our study suggest that the inflammation elicited by premalignant conditions could be used as a surrogate indicator of malignant transformation and its microscopic separation from other mucosal inflammatory diseases.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e70"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144672098","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Primordial odontogenic tumor: report of a rare case with a review of literature","authors":"Navneet Kaur Sehgal , Dwight Rice , Tsungju O-Lee , Mia Nepomuceno-Perez , Anupama Grandhi","doi":"10.1016/j.oooo.2025.04.049","DOIUrl":"10.1016/j.oooo.2025.04.049","url":null,"abstract":"<div><div>The primordial odontogenic tumor (POT) was first described in 2014. The term was used due to the histologic similarity of the tumor with the elements of the early stages of tooth development. It is a benign odontogenic tumor with epithelial and mesenchymal components. It occurs in the first and second decades of life. It is prevalent in the tooth-bearing areas of the mandible. Almost 18 cases have been published in peer-reviewed journals so far. It commonly presents as a well-defined pericoronal unilocular radiolucency. It is composed of fibromyxoid tissue with areas resembling the dental papilla and lined by epithelium that resembles the inner enamel epithelium of the enamel organ. Focal stellate reticulum-like cells can be seen external to the lining epithelium. A few cases in the literature have shown small focal calcifications. Treatment with conservative surgical excision appears to be curative, with no recurrence potential. Here we report a case of a primordial odontogenic tumor.</div></div><div><h3>Case</h3><div>An 8-year-old male patient presented to the Loma Linda University Ear, Nose, and Throat Surgery Department with a soft tissue mass in the right maxillary sinus along with a free-floating devitalized tooth. A clinical diagnosis of a maxillary cyst was made. A Caldwell-Luc (transantral) approach was utilized for enucleation of the lesion in the right maxillary sinus. A right maxillary antrostomy was also performed. The histopathology showed a biphasic tumor with an epithelial lining that was composed of columnar cells with focal stellate reticulum. A diagnosis of primordial odontogenic tumor was made based on the microscopic features. Upon a 2-year follow-up, the patient appears to have responded well to treatment with no evidence of recurrence.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e82"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670600","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Prokopios P. Argyris , Gabrielle R. Dennis , Rajaram Gopalakrishnan , Ioannis Koutlas , John R. Kalmar , Kristin K. McNamara
{"title":"Oral soft tissue and jawbone sarcomas (OSTJS): a retrospective clinicopathologic analysis of 128 cases from two institutions","authors":"Prokopios P. Argyris , Gabrielle R. Dennis , Rajaram Gopalakrishnan , Ioannis Koutlas , John R. Kalmar , Kristin K. McNamara","doi":"10.1016/j.oooo.2025.04.053","DOIUrl":"10.1016/j.oooo.2025.04.053","url":null,"abstract":"<div><h3>Introduction</h3><div>Head/neck sarcomas comprise a heterogeneous group of malignant mesenchymal neoplasms accounting for 1%-2% of all head/neck cancers and 4%-10% of all sarcomas in adults. Head/neck sarcomas exhibit aggressive biologic behavior with 5-year survival of 46%-60%. Studies regarding OSTJS are scarce. We present the collaborative experience with OSTJS from two academic institutions.</div></div><div><h3>Materials and Methods</h3><div>Archived OSTJS cases (2000-2022) were retrieved from the electronic laboratory databases of the oral pathology services at Ohio State University and University of Minnesota. Patient age and gender, anatomic site, and histopathologic subtype were recorded. Cases with incomplete information and/or diagnosis of spindle cell carcinoma or myeloid sarcoma were excluded.</div></div><div><h3>Results</h3><div>From a total of 128 OSTJS cases, 123 (96.1%) were primary and 5 (3.9%) metastatic. Seventy-seven (60.2%) cases affected men and 51 (39.8%) women (age range: 4-102 years). Favored sites included the mandible (48, 37.5%), maxilla (39, 30.4%), gingiva (15, 11.7%), palate (13, 10.2%), and tongue (4, 3.1%). OSTJS showed a predilection for adults (113, 88.3%; mean age: 52.2 years) with only 15 pediatric examples (11.7%; age range: 4-18 years, mean: 12.8 years). All but one pediatric OSTJS were intraosseous (14, 93.3%). The pediatric OSTJS included osteosarcoma (8, 53.3%), Ewing sarcoma (4, 26.7%), and 1 each (6.7%) of mesenchymal chondrosarcoma, <em>TFCP2::EWSR1</em> rhabdomyosarcoma, and alveolar soft part sarcoma. In adults, the most frequent tumors were osteosarcoma (58, 51.3%), Kaposi sarcoma (KS; 18, 15.9%), leiomyosarcoma (7, 6.2%), chondrosarcoma (6, 5.3%), angiosarcoma (5, 4.4%), and 4 each (3.5%) of rhabdomyosarcoma and undifferentiated pleomorphic sarcoma. The most common oral soft tissue sarcoma was KS (18 of 39, 46.2%).</div></div><div><h3>Conclusions</h3><div>OSTJS are uncommon with a strong predilection for the jawbones of males in their 4th decade. Osteosarcoma is the predominant sarcoma in both the pediatric and adult populations. Although OSTJS are chiefly primary tumors, metastatic sarcomas to the oral cavity may infrequently occur.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e83"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670604","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Soo-Yeon Sohn , Brandon Gold , Janine Westra , William Westra
{"title":"Human papillomavirus genotype distribution in head and neck cancer as a function of human Immunodeficiency virus status","authors":"Soo-Yeon Sohn , Brandon Gold , Janine Westra , William Westra","doi":"10.1016/j.oooo.2025.04.077","DOIUrl":"10.1016/j.oooo.2025.04.077","url":null,"abstract":"<div><h3>Introduction</h3><div>Among the most transformative events in head and neck oncology over the past two decades has been the recognition that a significant fraction of head and neck squamous cell carcinoma (HNSCC) is caused by the human papillomavirus (HPV). The distribution of HPV genotypes in these HPV-HNSCCs diverges from patterns observed in anogenital cancers, but little is known about how complex interactions between viral exposures, modes of transmission, tissue susceptibilities, and other factors such as immunosuppression impact on genotype distribution. For example, patients who are human immunodeficiency virus (HIV) positive are at increased risk of developing HPV-HNSCC, but it is not known if these tumors are infected with different HPV genotypes compared to patients without HIV.</div></div><div><h3>Methods</h3><div>Data was collected for all patients with HNSCCs that had undergone HPV testing at an academic hospital as part of clinical care (2012-2019). Screening was performed using real-time PCR targeting L1 of low and high-risk HPV types, followed by genotyping of positive cases. Genotype status was correlated with HIV status. For HPV-HNSCCs, genotype distribution was compared for patients with and without HIV<em>.</em></div></div><div><h3>Results</h3><div>HPV testing had been performed on HNSCCs from 29 patients who were HIV positive. A total of 28 of 29 (97%) tumors were HPV positive. Of these, 18 (64%) harbored HPV16 and 10 (36%) harbored a non-16 variant. Compared to HPV-HNSCCs from patients who were HIV-negative, those from patients with HIV were more likely to be related to a non-16 variant.</div></div><div><h3>Conclusion</h3><div>For patients with HPV-HNSCCs, HPV genotypes are unevenly distributed across different patient populations as a function of HIV status. The finding challenges a prevailing assumption that HPV16 eclipses non-16 subtypes as the prevailing etiologic agent for all HPV-HNSCCs, and may inform developing therapeutic and surveillance strategies that depend on precise delineation of HPV genotype distribution across specific patient populations.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e91-e92"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670536","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Noncaseating granulomatous inflammation of the orofacial region and its potential association with underlying systemic disease: a clinicopathological analysis of 19 cases","authors":"Carter Milne, Bryan Trump","doi":"10.1016/j.oooo.2025.04.071","DOIUrl":"10.1016/j.oooo.2025.04.071","url":null,"abstract":"<div><h3>Introduction</h3><div>Noncaseating granulomatous inflammation can be an inconclusive finding in many cases involving the oral cavity that may steer clinicians toward forming a differential diagnosis including (1) Crohn’s disease, (2) sarcoidosis, (3) chronic oral infection, (4) foreign material, or (5) allergies. Nineteen clinical cases were identified from the biopsy database at the University of Utah between October 2021 and March 2023. The main objective was to compare the findings and diagnoses of these cases through the lens of similar studies in today’s literature to leave clinicians better informed about trends of both systemic involvement and the most common location of clinical presentation. Materials and Methods. Five parameters were chosen to be evaluated: (1) patient’s age, (2) gender, (3) diagnosis, (4) location differing from the most common site of the upper lip, and (5) the absence of any diagnostic tests performed to arrive at proper diagnosis.</div></div><div><h3>Results</h3><div>Ages ranged from 6 to 83 years old—median being 52—nine male, 10 female. None of the lesions were associated with Crohn’s or allergies. One specimen was associated with sarcoidosis, one tested positive for infection, two for foreign material contamination, and two that were consistent with lichen planus. The remaining 13 had no definitive diagnosis but were signed off less-specifically as orofacial granulomatosis. Only one case presented on the upper lip.</div></div><div><h3>Conclusions</h3><div>Although only six of the 19 specimens were determined to have a more definitive cause, the other 13 were only given a diagnosis of either noncaseating granulomatous inflammation or orofacial granulomatosis because further testing for systemic factors was not performed. The hope is that future studies will highlight how much more common systemic involvement proves to be when patients elect to follow through with further systemic testing—allowing clinicians to treatment plan better for patients with these conditions.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e90"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670558","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Moni Ahmadian , Sonal Shah , Jean Shen , Joseph Lasky
{"title":"Dentoalveolar manifestations as the presenting sign of Gardner syndrome","authors":"Moni Ahmadian , Sonal Shah , Jean Shen , Joseph Lasky","doi":"10.1016/j.oooo.2025.04.009","DOIUrl":"10.1016/j.oooo.2025.04.009","url":null,"abstract":"<div><h3>Background</h3><div>Gardner syndrome (GS) is a rare variant of familial adenomatous polyposis (FAP) associated with highly penetrant mutations involving the <em>adenomatous polyposis coli (APC)</em> tumor suppressor gene. Although GS is typically inherited as an autosomal dominant trait, approximately 30% of cases may represent de novo mutations. GS is classically characterized by multiple premalignant gastrointestinal polyps, osteomas, epidermoid cysts, and both cutaneous and subcutaneous soft tissue tumors. However, in correlation with the specific position of the mutations within the <em>APC</em> gene, GS demonstrates variable clinical expressivity. Between 22% and 30% of patients with GS present with dental abnormalities consisting of multiple impacted teeth, supernumerary teeth, and odontomas. These dental abnormalities may precede the intestinal manifestations and may serve as a key feature in early diagnosis of this syndrome.</div></div><div><h3>Case Summary</h3><div>A 14-year-old, otherwise healthy and asymptomatic, female patient presented to the orthodontic clinic with complaint of over-retained primary teeth. Clinical and radiographic evaluation revealed multiple impacted permanent teeth, micro-odontomas, and areas of sclerotic dense bone without significant facial deformity. On further inquiry, no familial pattern is identified within the patient’s immediate family. Subsequent medical and imaging workup led to identification of multiple adenomatous and tubular gastrointestinal polyps and a paraspinal desmoid fibromatosis. Further genetic studies confirmed a diagnosis of GS.</div></div><div><h3>Conclusion</h3><div>This case highlights the significance of recognition of the dentoalveolar manifestations in the early diagnosis of GS emphasizing on the importance of interdisciplinary collaboration and maintaining open communication with other healthcare practitioners. Dental clinicians may serve a key role in the early diagnosis and instigation of adequate treatment of this life-threatening rare syndrome.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e69-e70"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144672097","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}