Oral Surgery Oral Medicine Oral Pathology Oral Radiology最新文献

{"title":"Intraoral salivary lymphoepithelial carcinoma: clinicopathologic and immunophenotypic characterization of 3 examples indicates consistently elevated PD-L1 expression","authors":"Prokopios P. Argyris ,&nbsp;Carter Lukenda ,&nbsp;Emilian V. Racila ,&nbsp;James Midtling ,&nbsp;Mansur Ahmad ,&nbsp;Rajaram Gopalakrishnan ,&nbsp;Paul Freedman ,&nbsp;Ioannis Koutlas","doi":"10.1016/j.oooo.2025.04.054","DOIUrl":"10.1016/j.oooo.2025.04.054","url":null,"abstract":"<div><h3>Introduction</h3><div>Head/neck lymphoepithelial carcinoma (LEC) represents a morphologically undifferentiated carcinoma with associated, nonneoplastic, lymphoplasmacytic infiltration. LEC chiefly arises in the nasopharynx and major salivary glands (MSGs), primarily the parotid. Although there is approximately 80% 5-year survival rate, aggressive surgical resection may cause debilitating morbidity. Overexpression of the targetable immunotherapy marker PD-L1 was recently reported in MSG LEC and pulmonary lymphoepithelioma-like carcinoma. PD-L1 expression in intraoral salivary LEC (ISLEC) is vastly understudied.</div></div><div><h3>Materials and Methods</h3><div>Three examples of ISLEC diagnosed between 1985 and 2022 were retrieved from the archives of two oral and maxillofacial pathology laboratories. The specimens were immunohistochemically studied with antibodies against AE1/AE3, CK7, CD3, CD20, p16, p53, Ki67, and PD-L1 (SP263). EBER in situ hybridization (ISH) was also performed.</div></div><div><h3>Results</h3><div>All three cases affected men (age range = 42-84 years; median = 61 years), and involved the floor of mouth, soft palate/uvula, and tongue. Microscopically, the lesions were unencapsulated and showed diffuse infiltration by nonkeratinizing sheets and islands of undifferentiated carcinoma cells with associated dense lymphoplasmacytic inflammation. The overlying mucosal epithelium was unremarkable. By immunohistochemistry, all three tumors showed strong and diffuse AE1/AE3 positivity and selective p53 staining. CK7 and p16 were negative. Ki67 highlighted 20%-80% of lesional cells. The lymphocytic inflammatory infiltrate comprised of a mixed population of T and B lymphocytes. EBER ISH was positive in one case. Notably, all tumors displayed membranous, focal-to-diffuse, PD-L1 staining with tumor proportion score (TPS) &gt; 95% in two and 40%-50% in the third case, and combined positive score (CPS) &gt; 20.</div></div><div><h3>Conclusions</h3><div>ISLECs are rare. Overall, PD-L1 appears to be highly expressed in ISLEC, thus rendering such patients potential candidates for targeted α-PD-1/PD-L1 immunotherapy.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e83-e84"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670605","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":"Clinicopathological analysis of odontogenic keratocysts with dysplasia","authors":"Julia Yu Fong Chang ,&nbsp;Yi-Ping Wang ,&nbsp;Pei-Hsuan Lu","doi":"10.1016/j.oooo.2025.04.036","DOIUrl":"10.1016/j.oooo.2025.04.036","url":null,"abstract":"<div><h3>Introduction</h3><div>Odontogenic keratocyst (OKC) with dysplasia is one of the precursor lesions of primary intraosseous carcinoma (PIOC). The aim of this study is to perform clinicopathological analysis of OKC with dysplasia. Be cautious with the clinicopathologic features of OKC with dysplasia and close follow-up might be helpful for early detection and improvement of the prognosis of PIOC.</div></div><div><h3>Materials and Methods</h3><div>This retrospective study included 6 cases. Patients’ age, gender, lesion location, symptoms, radiographic and microscopic findings were reviewed and analyzed.</div></div><div><h3>Results</h3><div>This study included 4 male and 2 female patients of mean age 51 (range 16-81) years. Lesions were slightly more common on the maxilla than the mandible, esp. posterior maxilla. The most common chief complaint was pain and swelling. Two-third of the cases showed an unilocular radiolucency with cortical bone perforation and were associated with impacted tooth. Root resorption was seen in 3 cases while tooth displacement was present in one case. In addition to the classic OKC histopathologic features, budding of the epithelium, basal cell hyperplasia and crowding, increased number and upward located mitotic figures, high columnar palisaded basal cells with frequent reverse polarity, and occasional basal vacuolation were also noted. Heavy inflammation was also frequently present in focal areas.</div></div><div><h3>Conclusions</h3><div>The dysplastic changes in OKC were infrequently concomitant occurred within classic OKC. Therefore, be familiar with the clinicopathologic features of high-risk cases, and careful examination are required for accurate diagnosis.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e78-e79"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670784","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":"Poorly differentiated spindle cell carcinoma with rhabdomyoblastic differentiation: highlights on immunohistochemistry interpretation","authors":"Rania Younis ,&nbsp;Shahd Alajaji ,&nbsp;Brian Moore ,&nbsp;Joshua Lubek ,&nbsp;Amy Plotkin ,&nbsp;Williams Twaddell ,&nbsp;Jason Molitoris ,&nbsp;John Papadimitriou ,&nbsp;John R. Basile ,&nbsp;Ahmed Sultan","doi":"10.1016/j.oooo.2025.04.070","DOIUrl":"10.1016/j.oooo.2025.04.070","url":null,"abstract":"<div><h3>Introduction</h3><div>Tumors of the head and neck that exhibit rhabdomyoblastic differentiation include rhabdomyosarcoma, leiomyosarcoma, synovial sarcoma, spindle cell carcinoma (SpCC), malignant peripheral nerve sheath tumor, or metastatic disease. Spindle cell carcinoma (SpCC) is a rare subtype of squamous cell carcinoma (SCC) typically seen in the oral cavity and larynx. Cases with ulcerated surface epithelium completely devoid of surface dysplastic epithelium that present entirely of a spindle cell proliferation can pose a diagnostic challenge. Additionally, immunohistochemistry (IHC) can demonstrate evidence of differentiation to variable mesenchymal components. Here we present a poorly differentiated SpCC with rhabdomyoblastic differentiation as evidenced by positivity for the rhabdomyoblastic biomarker MyoD-1.</div></div><div><h3>Case Presentation</h3><div>A 54-year-old female presented to her dentist with a nodular mass on the anterior mandibular gingiva mimicking a pyogenic granuloma. Upon histopathological examination, H&amp;E tissue sections showed an ulcerated mass devoted of any surface epithelium with malignant spindle cells invading the connective tissue. The spindle cells showed several malignant criteria including pleomorphism, hyperchromatism, increased nuclear-to-cytoplasmic ratio, and atypical mitotic figures. They were arranged in short interlacing fascicles. Abundant eosinophilic cytoplasm with rhabdomyoblastic differentiation was featured. Initial IHC analysis showed diffuse positivity for Myo-D1, focal positivity for Desmin, and negativity for Myogenin, SMA, h-Caldesmon. However, Pancytokeratin, was diffusely positive confirming the diagnosis of SpCC. Other cytokeratins, p53, p63, p40, TLE-1, CD99, and neural markers were all negative. Surgical management at the University of Maryland Medical Center included composite mandibulectomy and bilateral neck dissection, followed by adjuvant radiotherapy and chemotherapy. Surgical pathology findings were positive for perineural invasion, negative for all lymph nodes and tissue margins.</div></div><div><h3>Conclusion</h3><div>This case highlights the challenge in diagnosing cases of SpCC devoid of surface epithelium. Pathologists should also be aware of the possibility of rhabdomyoblastic differentiation in SpCC and cautiously interpret skeletal muscle biomarkers given the variable expression patterns.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e89"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670387","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":"A case report of benign fibrous histiocytoma","authors":"Hajir Aldaod,&nbsp;Naomi Ramer","doi":"10.1016/j.oooo.2025.04.067","DOIUrl":"10.1016/j.oooo.2025.04.067","url":null,"abstract":"<div><h3>Introduction</h3><div>The benign fibrous histiocytoma is a rare tumor in the oral cavity. It was recognized by the WHO in 1960 as a lesion composed of cells that resemble round histiocytic and spindled fibroblastic morphology. We present a case of a 33-year-old female with extensive carious decay and soft tissue mass.</div></div><div><h3>Materials and Methods</h3><div>Full-text case reports and case series in the English language were searched in PubMed, Ovid, Scopus, and Google Scholar Databases from 1960 to 2023. Results regarding benign fibrous histiocytoma relevant to the case report were reviewed.</div></div><div><h3>Results</h3><div>We present a 33-year-old female with grossly decayed carious teeth in the buccal gingival area of the mandible. The patient presented with a 6-month history of a tissue-colored, pedunculated gingival mass. Immunohistochemistry CD68 and Factor XIIIa were performed and were positive and supportive of the diagnosis. A definitive diagnosis of benign fibrous histiocytoma was made based on the histopathologic findings.</div></div><div><h3>Conclusion</h3><div>The benign fibrous histiocytoma is a rare soft tissue entity in the oral cavity. The age of our patient fits the classical clinical presentation of this entity, however, the gingival location of the lesion is less commonly reported. Histologically, a nodular core of dense and well-vascularized fibrous connective tissue with a stroma of a diffuse infiltrate of mixed inflammatory cell infiltrate was seen. The submucosa demonstrated a benign proliferation of multinucleated giant cells and delicate spindle cells, characteristic for benign fibrous histiocytoma.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e88"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670384","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":"Comparison between NOR-1 and DOG-1 immunohistochemistry staining in the diagnosis of salivary acinic cell carcinoma","authors":"Shawki Abed,&nbsp;Saja Alramadhan,&nbsp;Indraneel Bhattacharyya,&nbsp;Sarah Fitzpatrick,&nbsp;Mohammed N. Islam","doi":"10.1016/j.oooo.2025.04.061","DOIUrl":"10.1016/j.oooo.2025.04.061","url":null,"abstract":"<div><h3>Introduction</h3><div>Acinic cell carcinoma (ACC) of the salivary gland presents diagnostic challenges due to its wide histomorphological variability. DOG-1 has been shown to be sensitive in the diagnosis of ACC but is not specific. DOG-1 positivity can be seen in normal salivary acini, polymorphous adenocarcinoma, and luminal cells of pleomorphic adenoma. A novel rearrangement t(4;9) (q13;q31) leading to the upregulation of nuclear receptor subfamily 4 group A member 3 (NR4A3) has recently been identified in ACC. Immunostaining using an antibody targeting this rearrangement, neuron-derived orphan receptor 1 (NOR-1), has been investigated for reliable diagnosis of ACC. The purpose of this study was to evaluate and compare the performance of NOR-1 antibody with DOG-1.</div></div><div><h3>Materials and Methods</h3><div>An IRB-approved retrospective search of the University of Florida Oral Pathology biopsy service database between 2000 and 2022 was performed for ACC cases. DOG-1, mammaglobin, and NR4A3 immunohistochemical staining was performed on all included cases with appropriate control using the commercially available antibody NOR-1 (sc-393902 [H-7], Santa Cruz Biotechnology Inc.).</div></div><div><h3>Results</h3><div>Eight cases of low-grade ACC were included. Patient age ranged between 40 and 95 years with equal gender distribution. One case was from parotid gland while all other cases were from buccal and labial minor salivary glands. Five cases were conventional solid variant, two were microcystic, and one papillary-cystic variants. Six cases out of eight stained positive for NOR-1 with variable intensity, while DOG-1 stained five cases out of eight. All cases were negative for mammaglobin.</div></div><div><h3>Conclusions</h3><div>NOR-1 staining revealed more sensitivity than DOG-1 for diagnosis of ACC within our case series. However, evaluation of this marker in additional studies with larger number of cases is recommended.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e86"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670528","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":"Mucous membrane pemphigoid related to nivolumab immunotherapy: report of a rare case and review of the literature","authors":"Konstantinos Tzanavaris ,&nbsp;Efstathios Pettas ,&nbsp;Erofili Papadopoulou ,&nbsp;Maria Georgaki ,&nbsp;Evangelia Piperi ,&nbsp;Nikolaos Nikitakis","doi":"10.1016/j.oooo.2025.04.079","DOIUrl":"10.1016/j.oooo.2025.04.079","url":null,"abstract":"<div><h3>Background</h3><div>Immune-checkpoint inhibitors (ICIs) have been successfully used in the management of patients suffering from various malignancies; however, these agents have been also blamed for a wide spectrum of adverse events involving several anatomic sites, including the oral mucosa. Herein, we describe a rare case of mucous membrane pemphigoid (MMP) related to PD-1 inhibitor nivolumab and review the English-language literature on immunotherapy-associated MMP.</div></div><div><h3>Case Description</h3><div>A 74-year-old female presented for the evaluation of widespread painful ulcerations affecting the buccal mucosae, lower lip, and tongue. Nine months ago, the patient was diagnosed with lung adenocarcinoma and underwent 4 cycles of chemotherapy followed by radiation therapy; she was concurrently receiving nivolumab, which was also continued as monotherapy. The oral lesions developed after the last nivolumab injection, approximately 8 months after diagnosis and initiation of immunotherapy. With a clinical provisional diagnosis of a vesiculobullous disease of autoimmune or immunotherapy-associated origin, a perilesional diagnostic oral biopsy was performed. Microscopically, subepithelial clefting was observed, while direct immunofluorescence showed linear deposition of immunoreactants at the basement membrane zone. No circulating autoantibodies in patient’s serum were detected by ELISA and a final diagnosis of MMP was rendered. Considering the temporal relationship between the onset of the symptoms and nivolumab administration, an etiologic correlation between immunotherapy and MMP was considered very likely. Systemic prednisolone treatment, along with nivolumab withdrawal following consultation with patient’s oncologist, resulted in rapid remission of oral lesions.</div></div><div><h3>Conclusions</h3><div>Although ICIs have an established role in treatment of several cancers, they have also been associated with side effects of variable severity. Especially, ICI-related MMP seems to be quite rare with only a few cases reported in the English-language literature. Oral health care professionals should be aware of such adverse events, as their prompt recognition, accurate diagnosis, and appropriate management improves patients’ quality of life.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Pages e92-e93"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670533","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":"Oral lesions in a dyskeratosis congenital patient treated with bone marrow transplantation: challenges in differential diagnosis","authors":"Efstathios Pettas ,&nbsp;Erofili Papadopoulou ,&nbsp;Maria Kouri ,&nbsp;Konstantinos Tzanavaris ,&nbsp;Ioanna Tsouri ,&nbsp;Emmanouil Vardas ,&nbsp;Maria Georgaki ,&nbsp;Evangelia Piperi ,&nbsp;Nikolaos Nikitakis","doi":"10.1016/j.oooo.2025.04.078","DOIUrl":"10.1016/j.oooo.2025.04.078","url":null,"abstract":"<div><h3>Background</h3><div>Dyskeratosis congenita (DKC) represents a rare hereditary disorder resulting from mutations in DKC-1 gene usually displaying an X-linked recessive trait. The classic clinical triad of DKC includes hyperpigmentation of the skin, nail dystrophy, and white plaques of the oral mucosa, along with a high incidence of aplastic anemia development. Herein, a well-documented case of DKC is described with emphasis on differential diagnostic dilemmas.</div></div><div><h3>Case Description</h3><div>A 25-year-old male, nonsmoker, and nonalcohol drinker, was referred for evaluation of long-standing oral white lesions. The patient had a molecularly-confirmed diagnosis of DKC since age 8 for which he underwent hematopoietic stem cell transplantation (HSCT) at age 11. A history of oral painful lesions, first noticed a few years after birth, was also reported; following HSCT, oral white and ulcerative lesions were periodically managed with topical immunosuppressive regimens. On clinical intraoral examination, multifocal nondetached oral white patches and prominent atrophy of the dorsal tongue were evident; in addition, loss of elasticity of the oral tissues with fibrous bands of the buccal mucosa and limited mouth opening were also observed, raising the possibility of sclerodermatous chronic graft-versus-host disease (cGVHD). Further, cutaneous reticular hyperpigmentation, dystrophic nails, and alopecia were present. Biopsies of tongue white lesions showed dysplastic changes. Laser ablation of the residual white lesions and close follow-up appointments were suggested.</div></div><div><h3>Discussion and Conclusions</h3><div>Oral white plaques in DKC patients exhibit a significant potential for malignant transformation; concomitant cGVHD development in HSCT recipients is possible and might perplex the diagnosis of oral lesions. Biopsy and histopathologic evaluation of suspicious lesions are essential for diagnosis and risk assessment of malignant transformation, while close monitoring is also of high importance. Oral manifestations DKC and cGVHD may show significant overlap, while both conditions may coexist and potentially synergistically contribute to the risk of malignant transformation.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e92"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670537","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":"Interobserver agreement in histopathologic diagnosis of oral lichen planus","authors":"Leen AlQudah,&nbsp;Si On Lim,&nbsp;Valerie Murrah,&nbsp;Ricardo Padilla","doi":"10.1016/j.oooo.2025.04.058","DOIUrl":"10.1016/j.oooo.2025.04.058","url":null,"abstract":"<div><h3>Introduction</h3><div>Oral lichen planus (OLP) is a chronic immune-mediated disorder with unknown etiology. Optimal OLP diagnosis takes both histopathological and clinical aspects into consideration. Diagnostic criteria for OLP have varied over the years. This study aimed to evaluate interobserver agreement using the 2016 American Academy of Oral and Maxillofacial Pathology (AAOMP) diagnostic criteria.</div></div><div><h3>Methods</h3><div>This retrospective observational cohort study included 102 archived tissue samples received at the UNC Oral and Maxillofacial Pathology Laboratory and diagnosed as OLP or oral epithelial dysplasia (OED). Seventy-four OLP and 28 OED cases were retrieved. Three board certified oral and maxillofacial pathologists assessed the blinded cases individually. Each observer (observer A, B, C) was provided with the following clinical information: anatomic location, number of lesions (multiple vs solitary), sex, and age. Statistical analyses were conducted using SAS (SAS Institute Inc.) version 9.4 with unweighted Cohen’s kappa (k) test utilized to measure interobserver agreement in pairs.</div></div><div><h3>Results</h3><div>Responses were categorized as OLP vs OED. This study demonstrated a concordance with signout diagnosis of OLP using the 2016 AAOMP criteria of 77%, 43%, 91% for observers A, B, and C, respectively; whereas the concordance with signout diagnosis of OED for all observers was 93%. Interobserver agreement using Cohen’s categories of k grading were as follows: Observers A and B: 0.42 (moderate), Observers B and C: 0.35 (fair), Observers A and C: 0.71 (substantial).</div></div><div><h3>Conclusion</h3><div>This study illustrates the range of variation that can occur between pathologists in the histopathologic diagnosis of OLP. Although some variation in interobserver agreement in OLP diagnosis was shown to occur in the study, the overall moderate agreement supports the use of 2016 AAOMP criteria. Importantly, the congruency of observers concerning the diagnosis of mild OED is significant as it supports the reliability of adequate existing histopathologic criteria for a premalignant condition.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e85"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670555","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":"Phosphaturic mesenchymal tumor of the mandible presenting with an epithelial component in a patient with kidney dysfunction and tumor-induced osteomalacia","authors":"Jesse Bissell ,&nbsp;Carter Bruett ,&nbsp;Bryan Trump ,&nbsp;Mason Borsch ,&nbsp;David Adams ,&nbsp;Leslie Halpern","doi":"10.1016/j.oooo.2025.04.075","DOIUrl":"10.1016/j.oooo.2025.04.075","url":null,"abstract":"<div><div>Phosphaturic mesenchymal tumor (PMT) is a benign neoplasm that causes tumor-induced osteomalacia via secretion of fibroblast growth factor-23 (FGF-23). The FGF-23 secreted by osteocytes acts on the renal tubules causing phosphaturia. Patients exhibit hypophosphatemia and bone demineralization and endure significant loss of function and pain as a result. The patients have abnormal lab values before tumor resection such as an elevation in FGF-23 and serum alkaline phosphatase, low phosphorus, and low or unusually normal 1-25 dihydroxyvitamin D. However, in some cases, osteomalacia may not be apparent at the time of biopsy. Histologically, most PMTs present as nonspecific masses with difficult to discern features. The tumor contains a rich vascular supply which is often hemangiopericytoma-like and features a “grungy” calcifying matrix. Fibrohistiocytic spindled cells proliferate and can produce areas reminiscent of giant cell tumors of bone. Histological subtypes have been identified but appear to be minor morphologic variants. The diagnosis is challenging, and it can take years due to symptomatology, tumor localization, and diagnostic challenges. It is rarely reported in the head and neck, and when it involves the jaws, it can contain a variable epithelial component which complicates the diagnostic process. Here we report a case of a phosphaturic mesenchymal tumor presenting in the mandible with an epithelial component. We discuss the diagnostic challenges and the clinical outcome.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e91"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670562","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":"Recreational drugs induced pemphigus: an atypical clinical and histologic presentation","authors":"Anwar AlMuzaini ,&nbsp;Jassem Bastaki","doi":"10.1016/j.oooo.2025.04.035","DOIUrl":"10.1016/j.oooo.2025.04.035","url":null,"abstract":"<div><h3>Introduction</h3><div>Pemphigus is a group of autoimmune blistering disease processes affecting the skin and mucosal surfaces. Pemphigus vulgaris is the most common form of pemphigus encountered in the oral cavity. Many causes have been attributed to the initiation of the disease process including drug use. Herein, we present a case of recreational Drug-Induced Pemphigus with atypical clinical and histologic features.</div></div><div><h3>Case Report</h3><div>A 39-year-old Kuwaiti male presented with a 2-year history of multiple recreational drug abuse, including hashish, ethanol, methamphetamine, and chemical, that triggered schizophrenia. Prior to admission to the psychiatric hospital, he developed oral ulcerations of 1-year onset. ELISA showed elevated DSG-3 and indirect immunofluorescence testing was “suggestive of pemphigus.” On exam, the patient had 2 small deep ulcers in the left buccal mucosa, white keratotic, and rough mucosal changes, and crusted labial verrucous lesions not typical of pemphigus. Multiple lesional samples were examined histologically and showed acantholysis, focal suprabasal epithelial clefting, focal superficial epithelial separation, and dyskeratosis. Focally, there was extensive dyskeratosis and tier keratosis, reminiscent of Darier disease. Direct immunofluorescence (DIF) testing was negative. Additional biopsies were performed from recently developing lesions with less hyperplasia and crusting to further asses the disease process and progression. Dyskeratosis was not as prominent and the diagnosis rendered was suggestive of Pemphigus with clinical and histologic features of Pemphigus vegetans and foliaceous. Systemic corticosteroid therapy was commenced and the patient started showing improvements a week later.</div></div><div><h3>Conclusions</h3><div>Recreational drug-induced pemphigus can pose a diagnostic challenge when the lesions clinically are not typical of pemphigus. Thorough history-taking and appropriate diagnostic modalities can facilitate early diagnosis and management.</div></div>","PeriodicalId":49010,"journal":{"name":"Oral Surgery Oral Medicine Oral Pathology Oral Radiology","volume":"140 3","pages":"Page e78"},"PeriodicalIF":2.0,"publicationDate":"2025-07-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144670632","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}