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Longo R, Colin S, Streiff C, Benichou CG, Wendel C, Campitiello M, Plastino F, Bastien C, Egea J. A primary small cell neuroendocrine carcinoma (SCNC) of the oral cavity (cheek mucosa): Description of a case report. Oral Oncol 2023; 137:106295. [PMID: 36565487 DOI: 10.1016/j.oraloncology.2022.106295] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/10/2022] [Accepted: 12/18/2022] [Indexed: 12/24/2022]
Abstract
BACKGROUND Small cell neuroendocrine carcinoma (SCNC) of the oral cavity is a poorly differentiated, high-grade and very aggressive tumor with a poor prognosis. CASE DESCRIPTION A 64-year-old, Caucasian, smoker man consulted for an ulcero-necrotic, exophytic, lesion of the right retromolar trigone. Haed&neck CT scan showed a right tonsillar tumor lesion. The 18F-PET scan confirmed the presence of a right, highly hypermetabolic tonsillar lesion and two homolateral, cervical lymph nodes. Histology and immunohistochemistry were consisted with the diagnosis of a primary SCNC of the oral cavity. As the tumor was locally advanced and unresectable, the patient underwent a definitive radio-chemotherapy with a cisplatin/etoposide combined regimen (4 cycles). The treatment was well tolerated and led to a complete tumor response. CONCLUSION The particularity of this case relies on the rarity of the oral SCNC, its difficult and challenging diagnosis, and the complexity of its management that is not validated by large clinical trials, data being extrapolated from small cell lung cancer. In our case, the patient presenting a locally advanced tumor was treated by a combined radio-chemiotherapy leading to a complete tumor regression. The patient's follow up is too short to assess the real benefit of this treatment on overall survival.
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Affiliation(s)
- Raffaele Longo
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Sebastien Colin
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Camille Streiff
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Claire Gamelon Benichou
- Division of Radiotherapy, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Chloé Wendel
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Marco Campitiello
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Francesca Plastino
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Claire Bastien
- Division of Pathology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
| | - Julie Egea
- Division of Medical Oncology, "CHR Metz-Thionville", 1 Allée du Château, 57085 Ars-Laquenexy, France.
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2
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Archibald H, Keel S, Becker E. Large cell neuroendocrine carcinoma of the floor of mouth and retromolar trigone in association with basaloid squamous cell carcinoma. Int J Oral Maxillofac Surg 2022; 51:1520-1524. [PMID: 35523694 DOI: 10.1016/j.ijom.2022.01.020] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/04/2021] [Revised: 11/11/2021] [Accepted: 01/26/2022] [Indexed: 11/24/2022]
Abstract
Large cell neuroendocrine carcinoma and basaloid squamous cell carcinoma are each rare but aggressive diseases of the head and neck. A case of large cell neuroendocrine carcinoma occurring simultaneously with a basaloid squamous cell carcinoma originating from the oral cavity is described in this report. The diagnosis of these pathologies can be challenging but is important, as staging and treatment recommendations differ from those for oral cavity squamous cell carcinoma.
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Affiliation(s)
- Hunter Archibald
- Department of Otolaryngology-Head and Neck Surgery, University of Minnesota, Minneapolis, Minnesota, USA.
| | - Suzanne Keel
- Department of Pathology, Regions Hospital, St. Paul, Minnesota, USA
| | - Emily Becker
- Department of Otolaryngology, Regions Hospital, St. Paul, Minnesota, USA
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3
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Zeng C, Ling X, Tang Y, Xu H. 18F-FDG PET/CT and PET/MRI fusion imaging for neuroendocrine carcinoma of the tongue: A case report. Radiol Case Rep 2022; 17:1491-1495. [PMID: 35265247 PMCID: PMC8898757 DOI: 10.1016/j.radcr.2021.12.040] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2021] [Revised: 12/17/2021] [Accepted: 12/19/2021] [Indexed: 11/15/2022] Open
Abstract
Neuroendocrine carcinoma (NEC) involving the tongue is a rare and aggressive disease that is more common in middle-aged and elderly males. We report a case of a 56-year-old male who presented to our hospital with sore throat and was found to have a mass in the left root of the tongue. 18F-FDG PET/CT revealed intense FDG uptake in the mass of the tongue base, as well as different uptake of FDG in the mid-posterior mediastinal mass, right adrenal gland, and enlarged lymph nodes in the neck and mediastinum. Gadolinium-enhanced MRI clearly showed the extent of the tongue lesion, additionally suggesting the presence of brain metastases. 18F-FDG PET/MRI fusion images of the neck were obtained on the workstation, which may have a higher diagnostic value for tongue NEC. The patient underwent a biopsy of the mass in the left root of the tongue and was pathologically diagnosed with NEC. Whole-body 18F-FDG PET/CT and regional PET/MRI fusion images have complementary roles in the diagnosis of tongue NEC, and the former is mainly applied to determine the clinical stage of the disease and to guide treatment.
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Affiliation(s)
| | | | | | - Hao Xu
- Correspondence to Hao Xu, Address: No. 613 West Huangpu Avenue, Guangzhou, 510630, China. Tel: +86-20-3868 8404; Fax: +86-20-3868 8000.
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4
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Schuch LF, Schmidt TR, de Oliveira Zigmundo G, Kirschnick LB, Silveira FM, Martins MAT, Carlos R, Dos Santos JN, Fonseca FP, Vargas PA, Wagner VP, Martins MD. Oral and Maxillofacial Neuroendocrine Carcinoma: A Systematic Review. Head Neck Pathol 2021; 16:525-537. [PMID: 34870796 PMCID: PMC9187797 DOI: 10.1007/s12105-021-01398-2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/13/2021] [Accepted: 11/23/2021] [Indexed: 10/19/2022]
Abstract
The aim of the present study was to integrate the available data published in the literature on oral and maxillofacial neuroendocrine carcinomas concerning the demographic, clinical and histopathological features of this condition. An electronic search with no publication date restriction was undertaken in April 2021 in four databases. Eligibility criteria included reports published in English having enough data to confirm a definite diagnosis, always showing a neuroendocrine marker. Cases originating in the oropharynx, including base of the tongue and tonsils, were excluded. Outcomes were evaluated by the Kaplan-Meier method along with Cox regression. Twenty-five articles (29 cases) from nine different countries were detected. Mean patient age was 56.3 (± 17.5) years, with a slight male predilection. Symptomatology was present in 72.2% of informed cases. Regarding clinical presentation, a non-ulcerated nodule located in the gingiva with a mean size of 3.4 (± 2.0) cm was most frequently reported. Concomitant metastasis was identified in seven individuals. Histopathologically, most neoplasms were of the small cell type, and immunohistochemistry for both epithelial and neuroendocrine differentiation was used in 65.5% cases. Radical surgery was the treatment of choice in almost all cases, with or without adjuvant therapy. Mean follow-up was 20.5 (± 21.2) months, and only four patients developed recurrences. Eleven (44.0%) individuals died due to the disease. Ulcerated lesions were a prognostic factor. This study provides knowledge that can assist surgeons, oncologists, and oral and maxillofacial pathologists with the diagnosis and management of neuroendocrine carcinomas. Our findings demonstrated that the long-term prognosis of this lesion continues to be poor.
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Affiliation(s)
- Lauren Frenzel Schuch
- grid.411087.b0000 0001 0723 2494Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas, Piracicaba, São Paulo Brazil
| | - Tuany Rafaeli Schmidt
- grid.8532.c0000 0001 2200 7498Department of Oral Pathology, Faculdade de Odontologia, School of Dentistry, Universidade Federal do Rio Grande do Sul, R. Ramiro Barcelos, 2492, room 503, Porto Alegre, Rio Grande do Sul CEP: 90035-003 Brazil
| | - Gisele de Oliveira Zigmundo
- grid.8532.c0000 0001 2200 7498Department of Oral Pathology, Faculdade de Odontologia, School of Dentistry, Universidade Federal do Rio Grande do Sul, R. Ramiro Barcelos, 2492, room 503, Porto Alegre, Rio Grande do Sul CEP: 90035-003 Brazil
| | - Laura Borges Kirschnick
- grid.411087.b0000 0001 0723 2494Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas, Piracicaba, São Paulo Brazil
| | - Felipe Martins Silveira
- grid.11630.350000000121657640Molecular Pathology Area, School of Dentistry, Universidad de la República (UDELAR), Montevideo, Uruguay
| | - Marco Antonio Trevizani Martins
- grid.8532.c0000 0001 2200 7498Department of Oral Pathology, Faculdade de Odontologia, School of Dentistry, Universidade Federal do Rio Grande do Sul, R. Ramiro Barcelos, 2492, room 503, Porto Alegre, Rio Grande do Sul CEP: 90035-003 Brazil ,grid.8532.c0000 0001 2200 7498Department of Oral Medicine, Hospital de Clínicas de Porto Alegre, Universidade Federal do Rio Grande do Sul, Porto Alegre, Rio Grande do Sul Brazil
| | - Roman Carlos
- Pathology Section, Centro Clínico de Cabeza y Cuello/Hospital Herrera Llerandi, Guatemala City, Guatemala
| | - Jean Nunes Dos Santos
- grid.8399.b0000 0004 0372 8259Department of Pathology, Postgraduate Program in Dentistry and Health, School of Dentistry, Universidade Federal da Bahia, Salvador, Bahia Brazil
| | - Felipe Paiva Fonseca
- grid.8430.f0000 0001 2181 4888Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Minas Gerais Brazil
| | - Pablo Agustin Vargas
- grid.411087.b0000 0001 0723 2494Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas, Piracicaba, São Paulo Brazil
| | - Vivian Petersen Wagner
- grid.11835.3e0000 0004 1936 9262Academic Unit of Oral and Maxillofacial Medicine and Pathology, Department of Clinical Dentistry, University of Sheffield, Sheffield, UK
| | - Manoela Domingues Martins
- grid.8532.c0000 0001 2200 7498Department of Oral Pathology, Faculdade de Odontologia, School of Dentistry, Universidade Federal do Rio Grande do Sul, R. Ramiro Barcelos, 2492, room 503, Porto Alegre, Rio Grande do Sul CEP: 90035-003 Brazil ,grid.411087.b0000 0001 0723 2494Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas, Piracicaba, São Paulo Brazil
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Huang D, Peng Y, Xiong H, Hu X, Wang C, Wang Z, Su T, Huang L. Primary poorly differentiated neuroendocrine carcinoma of the oral cavity. Oral Dis 2021; 28:1811-1815. [PMID: 33694290 DOI: 10.1111/odi.13839] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/14/2021] [Revised: 02/12/2021] [Accepted: 03/06/2021] [Indexed: 11/28/2022]
Affiliation(s)
- Danni Huang
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Ying Peng
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Haofeng Xiong
- Center for Medical Genetics & Hunan Key Laboratory of Medical Genetics, School of Life Sciences, Central South University, Changsha, China
| | - Xin Hu
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Can Wang
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Zijia Wang
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Tong Su
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
| | - Long Huang
- Department of Oral and Maxillofacial Surgery, Center of Stomatology, Xiangya Hospital, Central South University, Changsha, China.,Research Center of Oral and Maxillofacial Tumor, Xiangya Hospital, Central South University, Changsha, China.,Institute of Oral Precancerous Lesions, Central South University, Changsha, China
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6
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Zhou Y, Zhou HC, Peng H, Zhang ZH. Primary small cell neuroendocrine carcinoma of the right posterior tongue. World J Meta-Anal 2020; 8:285-291. [DOI: 10.13105/wjma.v8.i4.285] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/11/2020] [Revised: 07/30/2020] [Accepted: 08/21/2020] [Indexed: 02/06/2023] Open
Abstract
Small cell neuroendocrine carcinoma (SNEC) is an extremely aggressive tumor and mainly occurs in the lung. Primary extra-pulmonary SNEC is rare. To date, only 11 primary SNECs occurring in the oral cavity have been reported in the English literature. We describe a case of primary SNEC of the right posterior tongue in a 46-year-old man. The patient had stage IVA disease and received adjuvant chemotherapy, followed by radical surgery and radiotherapy. He remained tumor-free for 20 mo before death due to gastrointestinal metastasis. The relevant literature on the 11 previously reported patients was reviewed, and the clinical features, histopathological characteristics, differential diagnosis and therapeutic strategies of this rare tumor were analyzed.
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Affiliation(s)
- Yu Zhou
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230001, Anhui Province, China
| | - Hang-Cheng Zhou
- Department of Pathology, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230001, Anhui Province, China
| | - Hui Peng
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230001, Anhui Province, China
| | - Zhi-Hong Zhang
- Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230001, Anhui Province, China
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7
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Tatullo M, Codispoti B, Makeeva I, Benincasa C, Spagnuolo G. From Mouth to Brain: Neuroendocrine Markers Play as a Crosstalk Among Oral and Neurodegenerative Diseases. Front Endocrinol (Lausanne) 2019; 10:378. [PMID: 31263455 PMCID: PMC6584809 DOI: 10.3389/fendo.2019.00378] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/13/2019] [Accepted: 05/28/2019] [Indexed: 11/24/2022] Open
Abstract
The neuroendocrine system consists of various cells distributed in non-endocrine functional structures, able to synthesize amines and peptides with both local (paracrine) and systemic (endocrine) effects. The presence of such cells, belonging to the neuroendocrine system, is highlighted by the presence of neuroendocrine markers: the most suggestive are chromogranin A, synaptophysin, S-100B protein and glial fibrillary acidic protein. The presence of neuroendocrine markers is commonly associated to the occurrence of neuroendocrine cancers, currently representing the 0.5 percent of all malignant tumors. Nevertheless, neuroendocrine markers have been found to be overexpressed in rare oral neuroendocrine tumors, but also in quite common inflammatory conditions, such as severe periodontitis. The monitoring of neuroendocrine markers is, thus, a common factor of interest among dentistry and neurology: the analysis of neuroendocrine markers in oral diseases may be predictive and prognostic about the severity of neurological diseases, such as lateral amyotrophic sclerosis and traumatic brain injuries. The aim of this mini-review is to highlight the role of neuroendocrine molecules as advantageous diagnostic and prognostic markers for both oral diseases and neurodegenerative disorders.
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Affiliation(s)
- Marco Tatullo
- Biomedical Section, Tecnologica Research Institute, Crotone, Italy
- Department of Therapeutic Dentistry, Sechenov University Russia, Moscow, Russia
- *Correspondence: Marco Tatullo
| | - Bruna Codispoti
- Biomedical Section, Tecnologica Research Institute, Crotone, Italy
| | - Irina Makeeva
- Department of Therapeutic Dentistry, Sechenov University Russia, Moscow, Russia
| | | | - Gianrico Spagnuolo
- Department of Therapeutic Dentistry, Sechenov University Russia, Moscow, Russia
- Department of Neurosciences, Reproductive and Odontostomatological Sciences, Università di Napoli Federico II, Naples, Italy
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Jha V, Sharma P, Yadav AK, Mandal AK. Postradiotherapy small cell neuroendocrine carcinoma of base of tongue: An unusual clinical and site presentation. J Postgrad Med 2016; 63:50-52. [PMID: 27853043 PMCID: PMC5394820 DOI: 10.4103/0022-3859.194209] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/28/2023] Open
Abstract
Small cell neuroendocrine carcinoma (NEC) of the tongue is an extremely rare entity with only seven cases reported in literature till date. These are high-grade tumors with a dismal prognosis. There is much ambiguity regarding the classification, treatment protocols and prognostic factors of these tumors due to the paucity of studies. We describe an exceptionally rare and unusual case of small cell NEC of the base of tongue arising in an elderly man 6 years after receiving radiotherapy for the treatment of squamous cell carcinoma at the same site. The diagnosis was confirmed on histopathological examination and supported by immunohistochemical positivity for neuron-specific enolase, synaptophysin, chromogranin, pan-cytokeratin, and cytokeratin 7. To the best of our knowledge, this is the first case of intraoral NEC arising many years later within the irradiated field of the initial tumor.
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Affiliation(s)
- V Jha
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
| | - P Sharma
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
| | - A K Yadav
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
| | - A K Mandal
- Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India
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