Pub Date : 2024-08-05DOI: 10.1007/s12105-024-01674-x
Ana Cláudia Garcia Rosa, André Machado de Senna, Fabrício Passador Santos, Andresa Borges Soares, Vera Cavalcanti de Araújo
Purpose: This case report examines the clinical presentation, diagnosis, treatment, and outcomes of mucocutaneous leishmaniasis with primary oral involvement in HIV-positive and HIV-negative patients diagnosed in Brazil.
Methods: We discuss the clinical manifestations, diagnostic methods, and therapeutic strategies, highlighting the clinical and histopathologic diagnostic features and distinct progression patterns based on HIV status. Our findings are compared with patterns observed in other countries, emphasizing the differences between the Americas and Europe, Asia, and Africa.
Results: In the Americas, particularly in Brazil, mucocutaneous leishmaniasis often presents with localized oral lesions, even in the presence of systemic immunosuppression, whereas in the Europe, Asia, and Africa, oral involvement is typically associated with visceral leishmaniasis in immunocompromised patients. These differences were due to variations in the parasite species involved.
Conclusion: This comparison underscores the importance of regional and immunological factors in diagnosing and managing this neglected infectious disease.
目的:本病例报告探讨了巴西确诊的 HIV 阳性和 HIV 阴性患者原发性口腔受累的皮肤黏膜利什曼病的临床表现、诊断、治疗和结果:我们讨论了临床表现、诊断方法和治疗策略,强调了临床和组织病理学诊断特征以及基于 HIV 感染状况的不同进展模式。我们的研究结果与其他国家观察到的模式进行了比较,强调了美洲与欧洲、亚洲和非洲之间的差异:结果:在美洲,尤其是巴西,粘膜利什曼病通常表现为局部口腔病变,即使存在全身免疫抑制,而在欧洲、亚洲和非洲,口腔受累通常与免疫功能低下患者的内脏利什曼病有关。这些差异是由于所涉及的寄生虫种类不同造成的:这一比较强调了地区和免疫因素在诊断和管理这种被忽视的传染病中的重要性。
{"title":"Oral Leishmaniasis in HIV-Positive and HIV-Negative Patients: A Comparative Analysis with Two New Case Reports.","authors":"Ana Cláudia Garcia Rosa, André Machado de Senna, Fabrício Passador Santos, Andresa Borges Soares, Vera Cavalcanti de Araújo","doi":"10.1007/s12105-024-01674-x","DOIUrl":"10.1007/s12105-024-01674-x","url":null,"abstract":"<p><strong>Purpose: </strong>This case report examines the clinical presentation, diagnosis, treatment, and outcomes of mucocutaneous leishmaniasis with primary oral involvement in HIV-positive and HIV-negative patients diagnosed in Brazil.</p><p><strong>Methods: </strong>We discuss the clinical manifestations, diagnostic methods, and therapeutic strategies, highlighting the clinical and histopathologic diagnostic features and distinct progression patterns based on HIV status. Our findings are compared with patterns observed in other countries, emphasizing the differences between the Americas and Europe, Asia, and Africa.</p><p><strong>Results: </strong>In the Americas, particularly in Brazil, mucocutaneous leishmaniasis often presents with localized oral lesions, even in the presence of systemic immunosuppression, whereas in the Europe, Asia, and Africa, oral involvement is typically associated with visceral leishmaniasis in immunocompromised patients. These differences were due to variations in the parasite species involved.</p><p><strong>Conclusion: </strong>This comparison underscores the importance of regional and immunological factors in diagnosing and managing this neglected infectious disease.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11300730/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141890480","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-08-05DOI: 10.1007/s12105-024-01670-1
Tarek Abi-Saab, Taja Lozar, Yi Chen, Alex P Tannenbaum, Heather Geye, Menggang Yu, Paul Weisman, Paul M Harari, Randall J Kimple, Paul F Lambert, Ricardo V Lloyd, Rong Hu
Background: High-risk human papillomavirus (HR-HPV) infection has been increasingly recognized as a risk factor for sinonasal tract carcinomas. However the prevalence and prognostic significance of HPV-associated sinonasal carcinomas is not well known due to limited studies and inconsistency in HPV testing modalities in literatures. Morphologically, HPV-associated sinonasal carcinomas encompass a diverse group of tumors. HPV-associated sinonasal adenocarcinoma has not been reported. The purpose of this study was to determine the prevalence, morphologic spectrum and prognostic implication of HPV-associated sinonasal carcinomas.
Methods: This cohort included 153 sinonasal carcinomas. Tissue microarrays were constructed. P16 immunohistochemistry and HR-HPV E6/7 in-situ Hybridization (ISH) were performed. Carcinomas were deemed HPV-associated based on a positive ISH testing. Clinicopathologic data was collected.
Results: 28/153 (18%) sinonasal carcinomas were HPV-associated. HPV-associated carcinomas consisted of 26 (93%) squamous cell carcinomas and variants, 1 (3.5%) HPV-related multiphenotypic sinonasal carcinoma and 1 (3.5%) adenocarcinoma. The HPV-associated adenocarcinoma closely resembled HPV-associated endocervical adenocarcinoma morphologically. HPV-associated carcinomas occurred in 8 (29%) women and 20 (71%) men with a median age of 66 years old. HPV-associated carcinomas were predominantly located at nasal cavity. A trend toward improved overall survival and progression free survival in HPV-associated carcinomas patients was observed, yet without statistical significance.
Conclusion: Our study identifies a novel HPV-associated sinonasal adenocarcinoma subtype, highlights the broad morphologic spectrum of HPV-associated sinonasal carcinomas, and supports routine p16 testing during pathology practice regardless of tumor subtype followed by a confirmatory HR-HPV testing. This practice is critical for studying the clinical behavior of HPV-associated sinonasal carcinomas.
{"title":"Morphologic Spectrum of HPV-associated Sinonasal Carcinomas.","authors":"Tarek Abi-Saab, Taja Lozar, Yi Chen, Alex P Tannenbaum, Heather Geye, Menggang Yu, Paul Weisman, Paul M Harari, Randall J Kimple, Paul F Lambert, Ricardo V Lloyd, Rong Hu","doi":"10.1007/s12105-024-01670-1","DOIUrl":"10.1007/s12105-024-01670-1","url":null,"abstract":"<p><strong>Background: </strong>High-risk human papillomavirus (HR-HPV) infection has been increasingly recognized as a risk factor for sinonasal tract carcinomas. However the prevalence and prognostic significance of HPV-associated sinonasal carcinomas is not well known due to limited studies and inconsistency in HPV testing modalities in literatures. Morphologically, HPV-associated sinonasal carcinomas encompass a diverse group of tumors. HPV-associated sinonasal adenocarcinoma has not been reported. The purpose of this study was to determine the prevalence, morphologic spectrum and prognostic implication of HPV-associated sinonasal carcinomas.</p><p><strong>Methods: </strong>This cohort included 153 sinonasal carcinomas. Tissue microarrays were constructed. P16 immunohistochemistry and HR-HPV E6/7 in-situ Hybridization (ISH) were performed. Carcinomas were deemed HPV-associated based on a positive ISH testing. Clinicopathologic data was collected.</p><p><strong>Results: </strong>28/153 (18%) sinonasal carcinomas were HPV-associated. HPV-associated carcinomas consisted of 26 (93%) squamous cell carcinomas and variants, 1 (3.5%) HPV-related multiphenotypic sinonasal carcinoma and 1 (3.5%) adenocarcinoma. The HPV-associated adenocarcinoma closely resembled HPV-associated endocervical adenocarcinoma morphologically. HPV-associated carcinomas occurred in 8 (29%) women and 20 (71%) men with a median age of 66 years old. HPV-associated carcinomas were predominantly located at nasal cavity. A trend toward improved overall survival and progression free survival in HPV-associated carcinomas patients was observed, yet without statistical significance.</p><p><strong>Conclusion: </strong>Our study identifies a novel HPV-associated sinonasal adenocarcinoma subtype, highlights the broad morphologic spectrum of HPV-associated sinonasal carcinomas, and supports routine p16 testing during pathology practice regardless of tumor subtype followed by a confirmatory HR-HPV testing. This practice is critical for studying the clinical behavior of HPV-associated sinonasal carcinomas.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11300749/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141890479","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-08-05DOI: 10.1007/s12105-024-01672-z
Saygo Tomo, José Alcides Almeida de Arruda, Brendo Vinicius Rodrigues Louredo, Pablo Agustin Vargas, Luciana Estevam Simonato
Ectomesenchymal chondromyxoid tumor (ECT) is a rare soft tissue tumor with peculiar histogenesis, exhibiting a predilection for the dorsum of the tongue. Molecular evidence suggests that it may originate from the migration of ectomesenchymal pluripotent cells from the neural crest to the tongue, where these cells may eventually proliferate and undergo myxoid and chondroid differentiation. This article illustrates a case of a 16-year-old female patient who presented with a nodule on the dorsum of her tongue, which had been present for four years. Surgical excision was performed, and histopathological analysis revealed a myxoid neoplasia composed of polygonal and spindle cells within a loose stroma containing chondroid areas. Tumor cells were positive for GFAP and S-100 proteins on immunohistochemical study, confirming the diagnosis of ECT. After a 5-year follow-up, the patient has shown no evidence of recurrence. Although rare, ECT can be diagnosed straightforwardly due to its distinctive clinical, histopathological, and immunohistochemical features. Clinicians and pathologists should become familiar with this tumor in order to avoid misdiagnosis.
{"title":"Ectomesenchymal Chondromyxoid Tumor of the Anterior Tongue: A Case Image.","authors":"Saygo Tomo, José Alcides Almeida de Arruda, Brendo Vinicius Rodrigues Louredo, Pablo Agustin Vargas, Luciana Estevam Simonato","doi":"10.1007/s12105-024-01672-z","DOIUrl":"10.1007/s12105-024-01672-z","url":null,"abstract":"<p><p>Ectomesenchymal chondromyxoid tumor (ECT) is a rare soft tissue tumor with peculiar histogenesis, exhibiting a predilection for the dorsum of the tongue. Molecular evidence suggests that it may originate from the migration of ectomesenchymal pluripotent cells from the neural crest to the tongue, where these cells may eventually proliferate and undergo myxoid and chondroid differentiation. This article illustrates a case of a 16-year-old female patient who presented with a nodule on the dorsum of her tongue, which had been present for four years. Surgical excision was performed, and histopathological analysis revealed a myxoid neoplasia composed of polygonal and spindle cells within a loose stroma containing chondroid areas. Tumor cells were positive for GFAP and S-100 proteins on immunohistochemical study, confirming the diagnosis of ECT. After a 5-year follow-up, the patient has shown no evidence of recurrence. Although rare, ECT can be diagnosed straightforwardly due to its distinctive clinical, histopathological, and immunohistochemical features. Clinicians and pathologists should become familiar with this tumor in order to avoid misdiagnosis.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11300727/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141890476","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-07-29DOI: 10.1007/s12105-024-01669-8
Ondina Karla Mousinho Rocha Torres, Natália Rodrigues Silva, Caio César da Silva Barros, Guilherme de Carvalho Wanderley, Olavo Hoston Gonçalves Pereira, Márcia Cristina da Costa Miguel
Adenoid ameloblastoma is a newly recognized epithelial odontogenic tumor. Herein, we present the case of a 24-year-old male patient who exhibited swelling in the anterior region and right hemi-mandible. Computed tomography demonstrated the presence of a hypodense osteolytic lesion associated with an impacted tooth. Based on the clinical hypotheses of the dentigerous cyst, odontogenic keratocyst, and ameloblastoma, an incisional biopsy was performed, and the diagnosis of ameloblastoma was rendered. A surgical resection of the tumor was performed. Histopathological examination of the specimen revealed typical areas of ameloblastoma associated with ductiform structures and cell proliferation in a solid storiform pattern, features resembling those found in adenomatoid odontogenic tumor. Based on these findings, the diagnosis of adenoid ameloblastoma was rendered. The accurate diagnosis of this locally infiltrative tumor is essential due to its similarity to other odontogenic neoplasms.
{"title":"Adenoid Ameloblastoma: The Newly Recognized Odontogenic Tumor - A Case Report.","authors":"Ondina Karla Mousinho Rocha Torres, Natália Rodrigues Silva, Caio César da Silva Barros, Guilherme de Carvalho Wanderley, Olavo Hoston Gonçalves Pereira, Márcia Cristina da Costa Miguel","doi":"10.1007/s12105-024-01669-8","DOIUrl":"10.1007/s12105-024-01669-8","url":null,"abstract":"<p><p>Adenoid ameloblastoma is a newly recognized epithelial odontogenic tumor. Herein, we present the case of a 24-year-old male patient who exhibited swelling in the anterior region and right hemi-mandible. Computed tomography demonstrated the presence of a hypodense osteolytic lesion associated with an impacted tooth. Based on the clinical hypotheses of the dentigerous cyst, odontogenic keratocyst, and ameloblastoma, an incisional biopsy was performed, and the diagnosis of ameloblastoma was rendered. A surgical resection of the tumor was performed. Histopathological examination of the specimen revealed typical areas of ameloblastoma associated with ductiform structures and cell proliferation in a solid storiform pattern, features resembling those found in adenomatoid odontogenic tumor. Based on these findings, the diagnosis of adenoid ameloblastoma was rendered. The accurate diagnosis of this locally infiltrative tumor is essential due to its similarity to other odontogenic neoplasms.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-07-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11286602/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141789488","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-07-03DOI: 10.1007/s12105-024-01662-1
Mollie Clark, Hannah Walsh, India Stephens-Laborde, Syed Ali Khurram
Purpose: There are a number of diagnostic criteria that can be used to support a diagnosis of Sjögren's syndrome (SS), a chronic autoimmune condition often characterised by xerostomia and xerophthalmia. Of the available investigations, the most invasive is the labial gland biopsy (LGB) for histopathology, which is associated with a risk of long-term altered sensation to the lip. A positive histological diagnosis is currently considered to be one of the most objective criteria, however there is debate about the interobserver agreement between pathologists, as well as the sensitivity and specificity of this test. We aim to determine if the diagnostic value of the LGB is significant enough to warrant the surgical procedure and its associated risks.
Methods: This study involved assessing the degree of agreement between members of a pathology team for a cohort of 50 LGBs taken for the purpose of confirming or excluding SS. The Tarpley system was used, which involves the allocation of a 'focus score'. Additionally, the histological diagnoses were compared to the relevant serological findings where available.
Results: All cases within the cohort had adequate tissue for assessment. 84% agreement (Cohen's Kappa = 0.585) was seen between the current team's consensus and the original reporting pathologist on whether the appearance was supportive of SS. However, only 58% agreement was seen for focus scores (Weighted Kappa = 0.496). The agreement between the serology result and whether the histology was supportive of SS was 79% (Cohen's Kappa = 0.493).
Conclusion: The findings raise the possibility that undue emphasis is placed on the value of a histological SS diagnosis. The current system for assessing and grading these biopsies is ambiguous in nature, with a low threshold considered indicative of SS. Due to the risk of complications associated with a LGB, alternative minimally invasive investigations should always be considered. The histological findings in isolation, particularly when a low focus score is seen, may not be predictive of a diagnosis of SS.
{"title":"The Value of Labial Gland Biopsies as a Diagnostic Test for Sjögren's Syndrome.","authors":"Mollie Clark, Hannah Walsh, India Stephens-Laborde, Syed Ali Khurram","doi":"10.1007/s12105-024-01662-1","DOIUrl":"10.1007/s12105-024-01662-1","url":null,"abstract":"<p><strong>Purpose: </strong>There are a number of diagnostic criteria that can be used to support a diagnosis of Sjögren's syndrome (SS), a chronic autoimmune condition often characterised by xerostomia and xerophthalmia. Of the available investigations, the most invasive is the labial gland biopsy (LGB) for histopathology, which is associated with a risk of long-term altered sensation to the lip. A positive histological diagnosis is currently considered to be one of the most objective criteria, however there is debate about the interobserver agreement between pathologists, as well as the sensitivity and specificity of this test. We aim to determine if the diagnostic value of the LGB is significant enough to warrant the surgical procedure and its associated risks.</p><p><strong>Methods: </strong>This study involved assessing the degree of agreement between members of a pathology team for a cohort of 50 LGBs taken for the purpose of confirming or excluding SS. The Tarpley system was used, which involves the allocation of a 'focus score'. Additionally, the histological diagnoses were compared to the relevant serological findings where available.</p><p><strong>Results: </strong>All cases within the cohort had adequate tissue for assessment. 84% agreement (Cohen's Kappa = 0.585) was seen between the current team's consensus and the original reporting pathologist on whether the appearance was supportive of SS. However, only 58% agreement was seen for focus scores (Weighted Kappa = 0.496). The agreement between the serology result and whether the histology was supportive of SS was 79% (Cohen's Kappa = 0.493).</p><p><strong>Conclusion: </strong>The findings raise the possibility that undue emphasis is placed on the value of a histological SS diagnosis. The current system for assessing and grading these biopsies is ambiguous in nature, with a low threshold considered indicative of SS. Due to the risk of complications associated with a LGB, alternative minimally invasive investigations should always be considered. The histological findings in isolation, particularly when a low focus score is seen, may not be predictive of a diagnosis of SS.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-07-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11222351/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141493897","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-07-03DOI: 10.1007/s12105-024-01663-0
Faseela Beegum, Akshay Trimukhe
In 1977, the American Joint Committee on Cancer (AJCC) introduced the inaugural Cancer Staging Manual, which implemented the T (tumor extent), N (regional lymph node status), and M (presence or absence of distant metastasis) staging system. This systematic approach aimed to convey the extent of disease across various cancer types, providing clinicians with a practical framework to plan treatment strategies, predict prognosis, and assess outcomes. The AJCC 8th edition, effective from January 1, 2018, continues this tradition. However, certain shortcomings persist in the AJCC 8th edition, as identified through clinical experience. Specifically, challenges arise in accurately assessing depth of invasion in unique histological variants of oral squamous cell carcinoma (e.g., Oral verrucous carcinoma, Carcinoma cuniculatum, and Papillary squamous cell carcinoma) and minor salivary gland tumors. Additionally, discrepancies exist in the perception of bone invasion patterns and in reporting practices. There is also a need for staging guidelines for malignant odontogenic tumors and multifocal tumors of the oral cavity, supplemented by diagrammatic representations. Lastly, there is a call for comprehensive staging criteria for carcinomas of the ear, external auditory canal, and temporal bone. We advocate for the inclusion of these considerations in future editions of the AJCC Cancer Staging Manual.
{"title":"Traversing the Terrain: Potential Pitfalls within the AJCC 8th Edition Staging System for Lip and Oral Cavity Cancers.","authors":"Faseela Beegum, Akshay Trimukhe","doi":"10.1007/s12105-024-01663-0","DOIUrl":"10.1007/s12105-024-01663-0","url":null,"abstract":"<p><p>In 1977, the American Joint Committee on Cancer (AJCC) introduced the inaugural Cancer Staging Manual, which implemented the T (tumor extent), N (regional lymph node status), and M (presence or absence of distant metastasis) staging system. This systematic approach aimed to convey the extent of disease across various cancer types, providing clinicians with a practical framework to plan treatment strategies, predict prognosis, and assess outcomes. The AJCC 8th edition, effective from January 1, 2018, continues this tradition. However, certain shortcomings persist in the AJCC 8th edition, as identified through clinical experience. Specifically, challenges arise in accurately assessing depth of invasion in unique histological variants of oral squamous cell carcinoma (e.g., Oral verrucous carcinoma, Carcinoma cuniculatum, and Papillary squamous cell carcinoma) and minor salivary gland tumors. Additionally, discrepancies exist in the perception of bone invasion patterns and in reporting practices. There is also a need for staging guidelines for malignant odontogenic tumors and multifocal tumors of the oral cavity, supplemented by diagrammatic representations. Lastly, there is a call for comprehensive staging criteria for carcinomas of the ear, external auditory canal, and temporal bone. We advocate for the inclusion of these considerations in future editions of the AJCC Cancer Staging Manual.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-07-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11222341/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141493898","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-07-03DOI: 10.1007/s12105-024-01666-x
Sibele Nascimento de Aquino, Cinthia Veronica Bardález López de Cáceres, Hélen Kaline Farias Bezerra, João Paulo Gonçalves de Paiva, Brendo Vinícius Rodrigues Louredo, Alan Roger Santos-Silva, Márcio Ajudarte Lopes, Pablo Agustin Vargas
Purpose: This study describes a large, well-documented case series of salivary gland polymorphous adenocarcinomas (PAC) from a single Brazilian center.
Methods: Demographic data, clinical presentation, histopathological and immunohistochemical features from 26 cases of PAC were analyzed and discussed in detail.
Results: Most patients were females (n = 21), with a ratio of 1:4.2 (male: female) with a mean age of 58.8 years (ranging from 36 to 84 years). The most common clinical presentation was a fibrocollagenous, firm nodular lesion, with a mean size of 2.46 cm (ranging from 0.5 to 3 cm). Most lesions occurred on the palate (n = 16), followed by buccal mucosa (n = 3), upper lip (n = 3), buccal vestibule (n = 2) and alveolar ridge (n = 1). Histologically, various growth patterns were observed, including tubular, solid, cribriform, papillary, and cystic. Additionally, glomeruloid slit-like structures, mucous, and clear cells were noted. Surface papillary epithelial hyperplasia was observed in a few cases. Nine cases exhibited myxoid and collagenous areas, while two cases showed fusiform areas and another case demonstrated squamous differentiation. Clear cell predominance was noted in two cases, and peri- and intraneural invasion was seen in eight cases. Immunohistochemical analysis revealed positivity for S-100, p63 and CK7, and negativity for p40 in all cases. The Ki-67 proliferation index was markedly low in most cases, with a mean of 2.5%.
Conclusion: We have provided a broad, detailed description of the clinical and microscopic features of PAC in a large, Brazilian cohort. These findings, in a resource-limited area, may be quite useful for establishing a proper diagnosis.
{"title":"Clinicopathological Features of 26 Intraoral Polymorphous Adenocarcinomas from a Single Brazilian Institution.","authors":"Sibele Nascimento de Aquino, Cinthia Veronica Bardález López de Cáceres, Hélen Kaline Farias Bezerra, João Paulo Gonçalves de Paiva, Brendo Vinícius Rodrigues Louredo, Alan Roger Santos-Silva, Márcio Ajudarte Lopes, Pablo Agustin Vargas","doi":"10.1007/s12105-024-01666-x","DOIUrl":"10.1007/s12105-024-01666-x","url":null,"abstract":"<p><strong>Purpose: </strong>This study describes a large, well-documented case series of salivary gland polymorphous adenocarcinomas (PAC) from a single Brazilian center.</p><p><strong>Methods: </strong>Demographic data, clinical presentation, histopathological and immunohistochemical features from 26 cases of PAC were analyzed and discussed in detail.</p><p><strong>Results: </strong>Most patients were females (n = 21), with a ratio of 1:4.2 (male: female) with a mean age of 58.8 years (ranging from 36 to 84 years). The most common clinical presentation was a fibrocollagenous, firm nodular lesion, with a mean size of 2.46 cm (ranging from 0.5 to 3 cm). Most lesions occurred on the palate (n = 16), followed by buccal mucosa (n = 3), upper lip (n = 3), buccal vestibule (n = 2) and alveolar ridge (n = 1). Histologically, various growth patterns were observed, including tubular, solid, cribriform, papillary, and cystic. Additionally, glomeruloid slit-like structures, mucous, and clear cells were noted. Surface papillary epithelial hyperplasia was observed in a few cases. Nine cases exhibited myxoid and collagenous areas, while two cases showed fusiform areas and another case demonstrated squamous differentiation. Clear cell predominance was noted in two cases, and peri- and intraneural invasion was seen in eight cases. Immunohistochemical analysis revealed positivity for S-100, p63 and CK7, and negativity for p40 in all cases. The Ki-67 proliferation index was markedly low in most cases, with a mean of 2.5%.</p><p><strong>Conclusion: </strong>We have provided a broad, detailed description of the clinical and microscopic features of PAC in a large, Brazilian cohort. These findings, in a resource-limited area, may be quite useful for establishing a proper diagnosis.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-07-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11222357/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141493896","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-28DOI: 10.1007/s12105-024-01665-y
Carla Isabelly Rodrigues-Fernandes, Danielle Machado Farias, Jurema Freire Lisboa de Castro, Luciano P Dos Santos, Oslei Paes de Almeida, Danyel Elias da Cruz Perez
Myoepithelioma is a benign salivary gland tumor. Central myoepitheliomas are very rare. The aim of this report was to describe a case of maxillary myoepithelioma. A 14-year-old female patient presented with an multilocular lesion in the anterior maxilla, with nearly 8 months of duration. The lesion was asymptomatic, and the patient's dental history was unremarkable. The diagnostic hypothesis was an odontogenic tumor. Biopsy specimen consisted of nests of plasmacytoid cells in a myxoid stroma without duct formation. No cellular atypia or bone and cartilage formation were noted. The neoplastic cells were positive for Pan-cytokeratin, S100, CK7, and CK8. The final diagnosis was myoepithelioma. The patient was treated by surgical excision followed by bone curettage, and no signs of recurrence were found after 8 years of treatment.
{"title":"Central Myoepithelioma of the Maxilla.","authors":"Carla Isabelly Rodrigues-Fernandes, Danielle Machado Farias, Jurema Freire Lisboa de Castro, Luciano P Dos Santos, Oslei Paes de Almeida, Danyel Elias da Cruz Perez","doi":"10.1007/s12105-024-01665-y","DOIUrl":"10.1007/s12105-024-01665-y","url":null,"abstract":"<p><p>Myoepithelioma is a benign salivary gland tumor. Central myoepitheliomas are very rare. The aim of this report was to describe a case of maxillary myoepithelioma. A 14-year-old female patient presented with an multilocular lesion in the anterior maxilla, with nearly 8 months of duration. The lesion was asymptomatic, and the patient's dental history was unremarkable. The diagnostic hypothesis was an odontogenic tumor. Biopsy specimen consisted of nests of plasmacytoid cells in a myxoid stroma without duct formation. No cellular atypia or bone and cartilage formation were noted. The neoplastic cells were positive for Pan-cytokeratin, S100, CK7, and CK8. The final diagnosis was myoepithelioma. The patient was treated by surgical excision followed by bone curettage, and no signs of recurrence were found after 8 years of treatment.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-06-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11213834/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141471610","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-28DOI: 10.1007/s12105-024-01664-z
Camila Oliveira Barbeiro, Heitor Albergoni Silveira, Roberto Henrique Barbeiro, Karina Helen Martins, Andreia Bufalino, Fernando Chahud, Jorge Esquiche León
Background: Previous studies have shown that at least a of intraoral eosinophilic ulcer is best classified as a CD30 + T-cell lymphoproliferative disorder (LPD), with histopathology reminiscent of lymphomatoid papulosis (LyP) of the skin. Microscopically, a mixed population of inflammatory cells, often including eosinophils and varying numbers of atypical lymphoid cells, frequently expressing CD30, is typical for LyP, whose clinicopathological spectrum includes type A, B, C, D, E, and LyP with DUSP22/IRF4 rearrangement. To date, about 27 intraoral LyP cases have been reported. Of them, 7 cases were diagnosed as LyP type C, which is frequently confused with anaplastic large cell lymphoma (ALCL) on histopathology.
Methods: A 60-year-old male was referred for a one-month history of a tongue ulcer.
Results: Microscopy showed numerous subepithelial atypical large lymphoid cells, which expressed CD4 (with partial loss of CD3, CD5, and CD7), CD8 (few cells), CD30 (about 50%, in non-diffuse pattern with size variability), TIA-1, and Ki-67 (85%), without staining for CD56, ALK, LMP1, and EBER1/2, concerning for a diagnosis of ALCL. However, after three weeks, the lesion completely healed.
Conclusion: We present here a rare case of intraoral CD30+ T-cell LPD that we believe is the oral counterpart of cutaneous LyP type C.
{"title":"Intraoral CD30+ T-Cell Lymphoproliferative Disorder with Lymphomatoid Papulosis Type C Features Mimics Lymphoma Histopathologically and Immunohistochemically.","authors":"Camila Oliveira Barbeiro, Heitor Albergoni Silveira, Roberto Henrique Barbeiro, Karina Helen Martins, Andreia Bufalino, Fernando Chahud, Jorge Esquiche León","doi":"10.1007/s12105-024-01664-z","DOIUrl":"10.1007/s12105-024-01664-z","url":null,"abstract":"<p><strong>Background: </strong>Previous studies have shown that at least a of intraoral eosinophilic ulcer is best classified as a CD30 + T-cell lymphoproliferative disorder (LPD), with histopathology reminiscent of lymphomatoid papulosis (LyP) of the skin. Microscopically, a mixed population of inflammatory cells, often including eosinophils and varying numbers of atypical lymphoid cells, frequently expressing CD30, is typical for LyP, whose clinicopathological spectrum includes type A, B, C, D, E, and LyP with DUSP22/IRF4 rearrangement. To date, about 27 intraoral LyP cases have been reported. Of them, 7 cases were diagnosed as LyP type C, which is frequently confused with anaplastic large cell lymphoma (ALCL) on histopathology.</p><p><strong>Methods: </strong>A 60-year-old male was referred for a one-month history of a tongue ulcer.</p><p><strong>Results: </strong>Microscopy showed numerous subepithelial atypical large lymphoid cells, which expressed CD4 (with partial loss of CD3, CD5, and CD7), CD8 (few cells), CD30 (about 50%, in non-diffuse pattern with size variability), TIA-1, and Ki-67 (85%), without staining for CD56, ALK, LMP1, and EBER1/2, concerning for a diagnosis of ALCL. However, after three weeks, the lesion completely healed.</p><p><strong>Conclusion: </strong>We present here a rare case of intraoral CD30+ T-cell LPD that we believe is the oral counterpart of cutaneous LyP type C.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-06-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11213839/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141471612","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-28DOI: 10.1007/s12105-024-01658-x
W W B de Kort, R Millen, E Driehuis, L A Devriese, R J J van Es, S M Willems
Introduction: Patient derived organoids (PDOs) are 3D in vitro models and have shown to better reflect patient and tumor heterogeneity than conventional 2D cell lines. To utilize PDOs in clinical settings and trials for biomarker discovery or drug response evaluation, it is valuable to determine the best way to optimize sample selection for maximum PDO establishment. In this study, we assess patient, tumor and tissue sampling factors and correlate them with successful PDO establishment in a well-documented cohort of patients with head and neck squamous cell carcinoma (HNSCC).
Methods: Tumor and non-tumorous adjacent tissue samples were obtained from HNSCC patients during routine biopsy or resection procedures at the University Medical Center Utrecht. The tissue was subsequently processed to establish PDOs. The sample purity was determined as the presence of epithelial cells in the culture on the day of organoid isolation as visualized microscopically by the researcher. PDO establishment was recorded for all samples. Clinical data was obtained from the medical records and was correlated to PDO establishment and presence of epithelial cells.
Results: Organoids could be established in 133/250 (53.2%) primary tumor site tissues. HNSCC organoid establishment tended to be more successful if patients were younger than the median age of 68 years (74/123 (60.2%) vs. 59/127 (46.5%), p = 0.03). For a subset of samples, the presence of epithelial cells in the organoid culture on the day of organoid isolation was recorded in 112/149 (75.2%) of these samples. When cultures were selected for presence of epithelial cells, organoid establishment increased to 76.8% (86/112 samples).
Conclusion: This study found a trend between age and successful organoid outgrowth in patients with HNSCC younger than 68 years and emphasizes the value of efficient sampling regarding PDO establishment.
{"title":"Clinicopathological Factors as Predictors for Establishment of Patient Derived Head and Neck Squamous Cell Carcinoma Organoids.","authors":"W W B de Kort, R Millen, E Driehuis, L A Devriese, R J J van Es, S M Willems","doi":"10.1007/s12105-024-01658-x","DOIUrl":"https://doi.org/10.1007/s12105-024-01658-x","url":null,"abstract":"<p><strong>Introduction: </strong>Patient derived organoids (PDOs) are 3D in vitro models and have shown to better reflect patient and tumor heterogeneity than conventional 2D cell lines. To utilize PDOs in clinical settings and trials for biomarker discovery or drug response evaluation, it is valuable to determine the best way to optimize sample selection for maximum PDO establishment. In this study, we assess patient, tumor and tissue sampling factors and correlate them with successful PDO establishment in a well-documented cohort of patients with head and neck squamous cell carcinoma (HNSCC).</p><p><strong>Methods: </strong>Tumor and non-tumorous adjacent tissue samples were obtained from HNSCC patients during routine biopsy or resection procedures at the University Medical Center Utrecht. The tissue was subsequently processed to establish PDOs. The sample purity was determined as the presence of epithelial cells in the culture on the day of organoid isolation as visualized microscopically by the researcher. PDO establishment was recorded for all samples. Clinical data was obtained from the medical records and was correlated to PDO establishment and presence of epithelial cells.</p><p><strong>Results: </strong>Organoids could be established in 133/250 (53.2%) primary tumor site tissues. HNSCC organoid establishment tended to be more successful if patients were younger than the median age of 68 years (74/123 (60.2%) vs. 59/127 (46.5%), p = 0.03). For a subset of samples, the presence of epithelial cells in the organoid culture on the day of organoid isolation was recorded in 112/149 (75.2%) of these samples. When cultures were selected for presence of epithelial cells, organoid establishment increased to 76.8% (86/112 samples).</p><p><strong>Conclusion: </strong>This study found a trend between age and successful organoid outgrowth in patients with HNSCC younger than 68 years and emphasizes the value of efficient sampling regarding PDO establishment.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2,"publicationDate":"2024-06-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11213837/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141471611","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}