|
1
|
Han F, Liu F, Wang H, Qin Y, Lu Q, Wu X, Guo Z, Nan X. Clinicopathologic characterization of secretory carcinoma of salivary gland. World J Surg Oncol 2024; 22:282. [PMID: 39456022 PMCID: PMC11515321 DOI: 10.1186/s12957-024-03561-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/29/2024] [Accepted: 10/16/2024] [Indexed: 10/28/2024] Open
Abstract
BACKGROUND To investigate the clinicopathologic characteristics, therapeutic methods, and prognosis of secretory carcinoma of salivary gland (SCSG). METHODS The clinicopathologic data of 13 patients with SCSG admitted to Shanxi Cancer Hospital from January 2018 to June 2023 were retrospectively analyzed, and a literature review was performed. RESULTS A total of eight males and five females aged 22-78 years old were enrolled, and they commonly presented with painless masses in the parotid or submandibular gland. They all underwent surgical treatment, accompanied by typical pathological examinations postoperatively. Fluorescence in situ hybridization (FISH) was conducted in seven cases, the results were all positive, and no gene fusion other than ETV6-NTRK3 was found. Two patients developed local relapse during follow-up, both of which were in the surgical area. By the end of the follow-up, 12 patients survived and one patient died. CONCLUSIONS SCSG is a rare low-grade malignancy with a good prognosis. Pathological and immunohistochemical characteristics are the key to secretory carcinoma (SC) diagnosis, and surgical excision is the major treatment means for SCSG. Whether to perform simultaneous cervical lymph node dissection and other adjuvant therapies should be determined based on the pathological stage and the presence or absence of high-risk factors.
Collapse
Affiliation(s)
- Fei Han
- First Clinical Medical School, Shanxi Medical University, Taiyuan, 030001, China
- Department of Head and Neck Surgery, Shanxi Hospital Affiliated to Cancer Hospital, Shanxi Provincial Cancer Hospital, Chinese Academy of Medical Sciences/Cancer HospitalAffiliated to Shanxi Medical University, Taiyuan, 031000, Shanxi Province, China
| | - Feng Liu
- Department of Head and Neck Surgery, Shanxi Hospital Affiliated to Cancer Hospital, Shanxi Provincial Cancer Hospital, Chinese Academy of Medical Sciences/Cancer HospitalAffiliated to Shanxi Medical University, Taiyuan, 031000, Shanxi Province, China
| | - Hao Wang
- First Clinical Medical School, Shanxi Medical University, Taiyuan, 030001, China
| | - Yanchao Qin
- Department of Head and Neck Surgery, Shanxi Hospital Affiliated to Cancer Hospital, Shanxi Provincial Cancer Hospital, Chinese Academy of Medical Sciences/Cancer HospitalAffiliated to Shanxi Medical University, Taiyuan, 031000, Shanxi Province, China
| | - Qian Lu
- Second Clinical Medical School, Shanxi Medical University, Taiyuan, 030001, China
| | - Xuesong Wu
- Department of Head and Neck Surgery, Shanxi Hospital Affiliated to Cancer Hospital, Shanxi Provincial Cancer Hospital, Chinese Academy of Medical Sciences/Cancer HospitalAffiliated to Shanxi Medical University, Taiyuan, 031000, Shanxi Province, China
| | - Zhen Guo
- Department of Head and Neck Surgery, Shanxi Hospital Affiliated to Cancer Hospital, Shanxi Provincial Cancer Hospital, Chinese Academy of Medical Sciences/Cancer HospitalAffiliated to Shanxi Medical University, Taiyuan, 031000, Shanxi Province, China
| | - Xinrong Nan
- Department of Oral and Maxillofacial Surgery, First Hospital of Shanxi Medical University, No. 56 Jiefang South Road, Taiyuan, 030001, Shanxi Province, China.
| |
Collapse
|
|
2
|
Wang Y, Sun J, Sun B, Zhang C, Tian Z, Wang L, Li J. The genetic and immune features of salivary gland secretory carcinoma with high-grade transformation. Oral Dis 2024; 30:4320-4330. [PMID: 38263601 DOI: 10.1111/odi.14876] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/09/2023] [Revised: 12/26/2023] [Accepted: 01/11/2024] [Indexed: 01/25/2024]
Abstract
OBJECTIVES To compare the clinicopathological, molecular, and immune features of conventional and high-grade transformation (HGT) secretory carcinoma (SC) in salivary glands. MATERIALS AND METHODS The clinicopathological data of 88 cases including 74 conventional SCs and 14 SCs with HGT were reviewed. Targeted next-generation sequencing was performed in 11 SCs with HGT and 7 conventional SCs. The level of PD-L1 and CD8+ TILs was determined by immunohistochemistry. RESULTS Compared with the conventional group, the rates of nodal metastasis, local recurrence, distant metastasis and mortality were significantly higher in the HGT cohort. Mutations of ARID1A/B, KMT2A, HOXD13, NRG1 and ETV6 genes were identified in HGT SCs. A recurrent E307G mutation in GATA6 gene was also observed in two cases. Two deceased HGT patients with distant metastasis harboured NOTCH3 mutations. ETV6-RET translocation was prone to occur in the HGT SCs. Additionally, PD-L1 expression was low, and CD8+ TILs were sparse in most HGT cases. CONCLUSION Our findings reveal novel gene alterations involved in the progression of HGT in SCs. Most HGT SCs patients cannot benefit from PD-L1 blocking and may be approached with a distinct treatment strategy including the lymph node dissection and application of molecular target drugs in precision oncology.
Collapse
Affiliation(s)
- Yu Wang
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Jingjing Sun
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Bao Sun
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Chunye Zhang
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Zhen Tian
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Lizhen Wang
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Jiang Li
- Shanghai Key Laboratory of Stomatology, Department of Oral Pathology, Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| |
Collapse
|
|
3
|
Asuquo J, Blavo C, Bhatta R. Secretory Carcinoma of the Philtrum of the Upper Lip: A Case Study. Cureus 2024; 16:e68823. [PMID: 39371716 PMCID: PMC11456339 DOI: 10.7759/cureus.68823] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/04/2024] [Indexed: 10/08/2024] Open
Abstract
The parotid, submandibular, and sublingual salivary glands are the major salivary glands in the mouth. Cancers that arise in these glands are relatively uncommon, usually benign, and rarely metastasize. We present a unique case of a 17-year-old male diagnosed with high-grade secretory carcinoma of the salivary gland that was generally asymptomatic except for a persistent rash. The patient reported no significant past medical, family, or social history. A multidisciplinary team efficiently diagnosed and treated the cancer with histopathology, MRI, tumor excision, lymphadenectomy, and adjuvant treatment. Despite the rarity of the cancer, which was found in a high-grade regionally advanced disease, in an uncharacteristically young patient, the patient was effectively treated with adjuvant chemoradiotherapy without treatment-related complications.
Collapse
Affiliation(s)
- Jessica Asuquo
- Department of Clinical Education, Nova Southeastern University Dr. Kiran C. Patel College of Osteopathic Medicine, Davie, USA
| | - Cyril Blavo
- Department of Preclinical Education, Nova Southeastern University Dr. Kiran C. Patel College of Osteopathic Medicine, Davie, USA
| | - Rabi Bhatta
- Internal Medicine, Southwest Healthcare Medical Education Consortium, Temecula, USA
| |
Collapse
|
|
4
|
Mahomed F, de Bruin J, Ngwenya S, Masango Z, Hodkinson K. ETV6 Molecular Heterogeneity in Salivary Secretory Carcinoma: A Case Series Report and Literature Review. Head Neck Pathol 2024; 18:66. [PMID: 39101978 PMCID: PMC11300731 DOI: 10.1007/s12105-024-01673-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/30/2024] [Accepted: 07/03/2024] [Indexed: 08/06/2024]
Abstract
BACKGROUND ETV6 gene rearrangement is the molecular hallmark of secretory carcinoma (SC), however; the nature, frequency, and clinical implications of atypical ETV6 signal patterns by fluorescence in situ hybridization (FISH) has not yet been systematically evaluated in salivary gland neoplasms. METHODS The clinical, histopathologic, immunohistochemical and molecular features of seven salivary SCs, including four cases with atypical ETV6 FISH patterns, were retrospectively analyzed along with a critical appraisal of the literature on unbalanced ETV6 break-apart in SCs. RESULTS The patients were four males and three females (31-70 years-old). Five presented with a painless neck mass and two patients with recurrent disease had a history of a previously diagnosed acinic cell carcinoma of the buccal mucosa. Histologically, there were varied combinations of microcystic, papillary, tubular, and solid patterns. All tumors were diffusely positive for S100 and/or SOX10, while 2 cases also showed luminal DOG1 staining. Rearrangement of the ETV6 locus was confirmed in 5/7 cases, of which 3 cases showed classic break-apart signals, 1 case further demonstrated duplication of the ETV6 5`end and the other loss of one copy of ETV6. Two cases harbored ETV6 deletion without rearrangement. Two of the 4 cases with atypical ETV6 FISH patterns represented recurrent tumors, one with widespread skeletal muscle involvement, bone and lymphovascular invasion. Surgical treatment resulted in gross-total resection in all 7 cases, with a median follow up of 9.5 months post-surgery for primary (n = 3) and recurrent disease (n = 1). CONCLUSION Duplication of the distal/telomeric ETV6 probe represented the most common (26/40; 65%) variant ETV6 break-apart FISH pattern in salivary SC reported in the literature and appears indicative of an aggressive clinical course.
Collapse
Affiliation(s)
- Farzana Mahomed
- Department of Oral Pathology, School of Oral Health Sciences, Faculty of Health Sciences, University of the Witwatersrand, Private Bag 3, Johannesburg, 2050, South Africa.
| | - Jana de Bruin
- Department of Anatomical Pathology, School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa
| | - Sizakele Ngwenya
- Department of Oral Pathology, School of Oral Health Sciences, Faculty of Health Sciences, University of the Witwatersrand, Private Bag 3, Johannesburg, 2050, South Africa
| | - Zinhle Masango
- Department of Molecular Medicine and Haematology, National Health Laboratory Services, School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa
| | - Katherine Hodkinson
- Department of Molecular Medicine and Haematology, National Health Laboratory Services, School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa
| |
Collapse
|
|
5
|
Zhao YY, Ge HJ, Yang WT, Shao ZM, Hao S. Secretory breast carcinoma: clinicopathological features and prognosis of 52 patients. Breast Cancer Res Treat 2024; 203:543-551. [PMID: 37897648 DOI: 10.1007/s10549-023-07153-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/02/2023] [Accepted: 09/29/2023] [Indexed: 10/30/2023]
Abstract
PURPOSE Secretory breast carcinoma is a rare histological subtype of invasive breast cancer and considered with an indolent clinical behavior. This study was conducted to analyze the clinicopathological features of patients with secretory breast carcinoma (SBC), explore the outcome, and compare the prognostic difference with invasive ductal breast carcinoma (IDC). METHODS AND MATERIALS: Patients with SBC diagnosed between 2006 and 2017 from Fudan University Shanghai Cancer Center were included in the study, excluding patients with previous malignant tumor history and incomplete clinical data or follow-up records. Peculiar clinicopathological and immunohistochemical features of the cases were fully described. Clinical data of 4979 cases of IDC were also evaluated during this period. After propensity score matching, prognostic analysis of SBCs and IDCs was calculated by Kaplan-Meier method and landmark analysis method. RESULTS The data of 52 patients diagnosed with SBC were identified from the pathological files. Among them, 47 patients were women, and 5 were men. The median age of the 52 SBCs was 46 years (mean, 48.1 years; range, 10-80 years). The tumor sizes ranged from 0.3 to 6.8 cm, with a mean of 3.5 cm. Eight patients (15.4%) had positive axillary lymph node involvement. The molecular classification was mostly triple-negative breast cancer (65.4%). Fluorescence in situ hybridization confirmed the presence of ETV6::NTRK3 rearrangement in 16 of 18 cases (88.9%). Furthermore, Kaplan-Meier survival analysis and landmark analysis demonstrated that there were no statistically significant differences in DFS and OS between SBC and IDC patients. CONCLUSION Although SBCs are generally associated with a favorable prognosis, our work exhibited that the clinicopathological features of SBC were partly different from former understandings, indicating that therapeutic procedure should be prudent. Further studies are necessary to fully identify the clinical behavior and predictive markers to improve diagnosis and management in this unique subtype of breast cancer.
Collapse
Affiliation(s)
- Yuan-Yuan Zhao
- Department of Breast Surgery, Fudan University Shanghai Cancer Center, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China
- Department of General Surgery, Huashan Hospital, Fudan University, 12 Urumqi Road (M), Shanghai, 200040, People's Republic of China
| | - Hui-Juan Ge
- Department of Oncology, Shanghai Medical College, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China
- Department of Pathology, Fudan University Shanghai Cancer Center, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China
| | - Wen-Tao Yang
- Department of Oncology, Shanghai Medical College, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China
- Department of Pathology, Fudan University Shanghai Cancer Center, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China
| | - Zhi-Ming Shao
- Department of Breast Surgery, Fudan University Shanghai Cancer Center, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China.
- Department of Oncology, Shanghai Medical College, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China.
- Institutes of Biomedical Science, Fudan University, Shanghai, People's Republic of China.
| | - Shuang Hao
- Department of Breast Surgery, Fudan University Shanghai Cancer Center, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China.
- Department of Oncology, Shanghai Medical College, Fudan University, 270 Dong-An Road, Shanghai, 200032, People's Republic of China.
| |
Collapse
|
|
6
|
Nocini R, Sanna G, Trotolo A, Fior A, Brunelli M, Manfrin E, Favero V. Secretory carcinoma of salivary glands with NTRK3 break-apart molecular rearrangement: Potential misdiagnosis with mucoepidermoid carcinoma. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2023; 124:101635. [PMID: 37709146 DOI: 10.1016/j.jormas.2023.101635] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/06/2023] [Accepted: 09/11/2023] [Indexed: 09/16/2023]
Abstract
A woman presented a right submandibular gland lesion with cytologic diagnosis of mucoepidermoid carcinoma. Patient underwent sialoadenectomy en bloc with supraomohyoid neck dissection. Positivity for ETV6-NTRK3 genes fusion on surgical sample led to final diagnosis of secretory carcinoma (SC). Secretory carcinoma has been renamed by WHO in 2017 from mammary-analogue-secretory carcinoma (MASC). Only 649 have been reported until 2019. While cytologic alteration are shared with other neoplasms as the acinic cell and mucoepidermoid carcinomas, ETV6-NTRK3 rearrangement is pathognomonic of SC. Although usually indolent and with low-stage presentation, SC has higher rate of local recurrences and nodal involvement than ACC. Surgical treatment represent the gold standard. Real prevalence of SC is probably underestimated due to the recent WHO 2017 reclassification. While cytologic analysis does not allow to discriminate SC from other malignancies, chromosomal examination is recommended. When low-grade SC is diagnosed, complete surgical resection assures good prognosis.
Collapse
Affiliation(s)
- Riccardo Nocini
- Head and Neck Department, Division of Otolaringology, AOUI Verona, P.le L.A. Scuro.10, Verona 37134, Italy
| | - Giangiacomo Sanna
- Head and Neck Department, Division of Maxillo-Facial Surgery, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy
| | - Alessandro Trotolo
- Head and Neck Department, Division of Maxillo-Facial Surgery, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy
| | - Andrea Fior
- Head and Neck Department, Division of Maxillo-Facial Surgery, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy
| | - Matteo Brunelli
- Department of Diagnostics and Public Health, Section of Pathology, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy
| | - Erminia Manfrin
- Department of Diagnostics and Public Health, Section of Pathology, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy
| | - Vittorio Favero
- Head and Neck Department, Division of Maxillo-Facial Surgery, AOUI Verona, P.le L.A. Scuro. 10, Verona 37134, Italy.
| |
Collapse
|
|
7
|
Richardson ET, Jo VY, Schnitt SJ. Salivary Gland-like Tumors of the Breast. Arch Pathol Lab Med 2023; 147:1014-1024. [PMID: 37651394 DOI: 10.5858/arpa.2023-0038-ra] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/19/2023] [Indexed: 09/02/2023]
Abstract
CONTEXT The World Health Organization classification of tumors of the breast recognizes several special type carcinomas and benign lesions with features comparable to those of salivary gland tumors. OBJECTIVE To discuss the histologic, immunophenotypic, molecular, and clinical features of salivary gland-like carcinomas of the breast. These breast tumors are often negative for hormone receptors and human epidermal growth factor receptor 2 (HER2), that is, triple-negative, but they generally have a much better prognosis than triple-negative breast carcinomas of no special type. We compare the immunophenotypic, molecular, and clinical features of these breast tumors with their salivary gland counterparts, highlighting similarities and differences. We also discuss benign salivary gland-like breast tumors. Finally, we highlight recent developments in understanding the molecular pathogenesis of these breast tumors and novel ancillary studies that can be used to support their diagnosis. DATA SOURCES A literature review was conducted, and papers were selected for further analysis and discussion by the authors of this review based on their novelty, applicability, and impact in the field. CONCLUSIONS Breast tumors that exhibit morphologic overlap with salivary gland tumors have been recognized by pathologists for decades, but the similarities and differences in their molecular pathogenesis have not been understood until more recently. These developments have led to novel diagnostic tools and further knowledge of these rare breast lesions.
Collapse
Affiliation(s)
- Edward T Richardson
- From the Department of Pathology, Brigham and Women's Hospital and Harvard Medical School, Boston, Massachusetts
- The Dana-Farber Cancer Institute, Boston, Massachusetts
| | - Vickie Y Jo
- From the Department of Pathology, Brigham and Women's Hospital and Harvard Medical School, Boston, Massachusetts
- The Dana-Farber Cancer Institute, Boston, Massachusetts
| | - Stuart J Schnitt
- From the Department of Pathology, Brigham and Women's Hospital and Harvard Medical School, Boston, Massachusetts
- The Dana-Farber Cancer Institute, Boston, Massachusetts
| |
Collapse
|
|
8
|
Baněčková M, Thompson LDR, Hyrcza MD, Vaněček T, Agaimy A, Laco J, Simpson RHW, Di Palma S, Stevens TM, Brcic L, Etebarian A, Dimnik K, Majewska H, Stárek I, O'Regan E, Salviato T, Helliwell T, Horáková M, Biernat W, Onyuma T, Michal M, Leivo I, Skalova A. Salivary Gland Secretory Carcinoma: Clinicopathologic and Genetic Characteristics of 215 Cases and Proposal for a Grading System. Am J Surg Pathol 2023; 47:661-677. [PMID: 37070739 DOI: 10.1097/pas.0000000000002043] [Citation(s) in RCA: 14] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/19/2023]
Abstract
Salivary gland secretory carcinoma (SC), previously mammary analog SC, is a low-grade malignancy characterized by well-defined morphology and an immunohistochemical and genetic profile identical to SC of the breast. Translocation t(12;15)(p13;q25) resulting in the ETV6 :: NTRK3 gene fusion is a characteristic feature of SC along with S100 protein and mammaglobin immunopositivity. The spectrum of genetic alterations for SC continues to evolve. The aim of this retrospective study was to collect data of salivary gland SCs and to correlate their histologic, immunohistochemical, and molecular genetic data with clinical behavior and long-term follow-up. In this large retrospective study, we aimed to establish a histologic grading scheme and scoring system. A total of 215 cases of salivary gland SCs diagnosed between 1994 and 2021 were obtained from the tumor registries of the authors. Eighty cases were originally diagnosed as something other than SC, most frequently acinic cell carcinoma. Lymph node metastases were identified in 17.1% (20/117 cases with available data), with distant metastasis in 5.1% (6/117). Disease recurrence was seen in 15% (n=17/113 cases with available data). The molecular genetic profile showed ETV6 :: NTRK3 gene fusion in 95.4%, including 1 case with a dual fusion of ETV6 :: NTRK3 and MYB :: SMR3B . Less frequent fusion transcripts included ETV6 :: RET (n=12) and VIM :: RET (n=1). A 3-tiered grading scheme using 6 pathologic parameters (prevailing architecture, pleomorphism, tumor necrosis, perineural invasion (PNI), lymphovascular invasion (LVI), and mitotic count and/or Ki-67 labeling index) was applied. Grade 1 histology was observed in 44.7% (n=96), grade 2 in 41.9% (n=90), and grade 3 in 13.5% (n=29) of cases. Compared with low-grade and intermediate-grade SC, high-grade tumors were associated with a solid architecture, more prominent hyalinization, infiltrative tumor borders, nuclear pleomorphism, presence of PNI and/or LVI, and Ki-67 proliferative index >30%. High-grade transformation, a subset of grade 2 or 3 tumors, seen in 8.8% (n=19), was defined as an abrupt transformation of conventional SC into high-grade morphology, sheet-like growth, and a tumor lacking distinctive features of SC. Both overall survival and disease-free survival (5 and 10 y) were negatively affected by tumor grade, stage, and TNM status (each P <0.0001). SC is a low-grade malignancy with predominantly solid-microcystic growth patterns, driven by a gene fusion, most commonly ETV6 :: NTRK3 . There is a low risk for local recurrence and a good overall long-term survival, with a low risk for distant metastasis but a higher risk for locoregional lymph node metastasis. The presence of tumor necrosis, hyalinization, PNI and/or LVI, and positive resection margins correlate with higher tumor grade, less favorable prognosis, and increased mortality. The statistical results allowed us to design a 3-tiered grading system for salivary SC.
Collapse
Affiliation(s)
- Martina Baněčková
- Department of Pathology, Charles University, Faculty of Medicine in Plzen
- Bioptic Laboratory Ltd Plzen
| | | | - Martin D Hyrcza
- Department of Pathology and Laboratory Medicine, University of Calgary, Arnie Charbonneau Cancer Institute
| | - Tomáš Vaněček
- Molecular Genetic Laboratory, Bioptic Laboratory Ltd, Plzen
| | - Abbas Agaimy
- Institute of Pathology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Comprehensive Cancer Center (CCC) Erlangen-EMN, Erlangen, Germany
| | - Jan Laco
- The Fingerland Department of Pathology, Charles University, Faculty of Medicine and University Hospital Hradec Kralove, Hradec Kralove
| | - Roderick H W Simpson
- Department of Pathology and Laboratory Medicine, University of Calgary, Calgary Laboratory Services, Foothills Medical Centre, Calgary, AB, Canada
| | - Silvana Di Palma
- Division of Clinical Medicine, Department of Histopathology, University of Surrey, Royal Surrey County Hospital, Guildford, Surrey
| | - Todd M Stevens
- Department of Pathology, University of Alabama at Birmingham, Birmingham, AL
| | - Luka Brcic
- Diagnostic and Research Institute of Pathology, Medical University of Graz, Graz, Austria
| | - Arghavan Etebarian
- Department of Oral and Maxillofacial Pathology, School of Dentistry, Alborz University of Medical Sciences, Karaj, Iran
| | - Katarina Dimnik
- Institute of Pathology, Faculty of Medicine, University of Ljubljana, Ljubljana, Slovenia
| | - Hanna Majewska
- Department of Pathology, Warmia and Mazury University, Olsztyn
| | - Ivo Stárek
- Department of Otorhinolaryngology, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University Olomouc, Olomouc, Czech Republic
| | - Esther O'Regan
- Department of Histopathology, St. James's Hospital & Dublin Dental Hospital, Trinity College Dublin, Dublin, Ireland
| | - Tiziana Salviato
- Division of Pathology, Department of Medical and Surgical Sciences for Children & Adults, University-Hospital of Modena and Reggio Emilia, Modena, Italy
| | - Tim Helliwell
- Department of Cellular Pathology, University of Liverpool, Liverpool, UK
| | - Markéta Horáková
- Department of Pathology, Charles University, Faculty of Medicine in Plzen
- Bioptic Laboratory Ltd Plzen
| | - Wojciech Biernat
- Department of Pathology, Medical University of Gdansk, Gdansk, Poland
| | - Timothy Onyuma
- Department of Pathology, Kenyatta National Hospital, Nairobi, Kenya
| | - Michal Michal
- Department of Pathology, Charles University, Faculty of Medicine in Plzen
- Bioptic Laboratory Ltd Plzen
| | - Ilmo Leivo
- Institute of Biomedicine, Pathology, University of Turku and Turku University Hospital, Turku, Finland
| | - Alena Skalova
- Department of Pathology, Charles University, Faculty of Medicine in Plzen
- Bioptic Laboratory Ltd Plzen
| |
Collapse
|
|
9
|
Mahendru R, Kakkar A, Cipriani NA, Sarma C, Ghosh V, Kaur K, Gupta S, Mohan A. Pleural metastasis from parotid secretory carcinoma: First report of morphology on effusion cytology, and role of pan-TRK immunohistochemistry. Diagn Cytopathol 2023; 51:E28-E37. [PMID: 36148935 DOI: 10.1002/dc.25057] [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: 06/22/2022] [Revised: 09/03/2022] [Accepted: 09/14/2022] [Indexed: 12/13/2022]
Abstract
Distant metastasis from salivary gland secretory carcinoma (SC) is rare, with lung and pleura being the most frequent site. While cytological features of SC on fine needle aspirates are well documented, its morphology in serous effusions has not been described. We describe the cytomorphological features on effusion cytology of two patients with ETV6::NTRK3 fusion-positive SC, who subsequently developed pleural metastases. Cytospin preparations of pleural fluid showed tightly cohesive, irregularly shaped and ball-like clusters of large tumor cells with scant to abundant uni- and multi-vacuolated cytoplasm. Nuclei were eccentrically placed, round to oval, vesicular, with finely granular chromatin, irregular nuclear membranes and conspicuous to prominent nucleoli. With these features, the tumors resembled an adenocarcinoma, indistinguishable from a lung primary. Cell blocks from both cases showed tumor fragments, some of which had the hollow appearance of transversely sectioned cell spheres as seen in lung and breast adenocarcinomas. Immunohistochemistry on cell blocks revealed nuclear pan-TRK positivity in both cases. Case 1 also showed focal mammaglobin staining, and TTF1 negativity. Pleural metastases from SC may mimic other adenocarcinomas. As targeted therapy, that is, selective TRK inhibitors are available for treatment of metastatic disease, NTRK3 fusion status is not only diagnostic, but also required to plan treatment. Pan-TRK immunohistochemistry serves as a viable cost-effective, easy to apply surrogate marker for NTRK3 fusion, particularly in diagnostic laboratories lacking easy access to molecular testing on cytological material.
Collapse
Affiliation(s)
- Ria Mahendru
- Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
| | - Aanchal Kakkar
- Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
| | - Nicole Anne Cipriani
- Department of Pathology, The University of Chicago Medicine, Chicago, Illinois, USA
| | - Chetna Sarma
- Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
| | - Vivek Ghosh
- Department of Radiation Oncology, BRA IRCH, All India Institute of Medical Sciences, New Delhi, India
| | - Kavneet Kaur
- Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
| | - Subhash Gupta
- Department of Radiation Oncology, BRA IRCH, All India Institute of Medical Sciences, New Delhi, India
| | - Anant Mohan
- Department of Pulmonary, Critical Care and Sleep Medicine, All India Institute of Medical Sciences, New Delhi, India
| |
Collapse
|
|
10
|
Mannelli G, Comini LV, Sacchetto A, Santoro R, Spinelli G, Bonomo P, Desideri I, Bossi P, Orlandi E, Alderotti G, Franchi A, Palomba A, Eccher A, Marchioni D, Nocini R, Piazza C, Molteni G. Estimating survival after salvage surgery for recurrent salivary gland cancers: Systematic review. Head Neck 2022; 44:1961-1975. [PMID: 35441406 PMCID: PMC9545583 DOI: 10.1002/hed.27062] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/07/2021] [Revised: 03/28/2022] [Accepted: 04/04/2022] [Indexed: 11/10/2022] Open
Abstract
Recurrent salivary gland carcinomas (RSCs) are poorly characterized and their clinical features and treatment options have not yet been fully described. The goal of this study was to analyze the therapeutic strategies and oncological outcomes of RSC patients through a literature review analysis. This systematic review was performed according to the PRISMA statements. Inclusion criteria for the systematic review were based on the population, intervention, comparison, and outcomes according to (PICO) framework. Two thousand seven hundred and four records were selected and 1817 recurrences were studied. Three hundred and sixty-five patients underwent salvage surgery (20.1%) and their 5-year mortality rate, overall survival and disease-free survival were 35%, 70%, and 42%, respectively. RSCs are aggressive neoplasms with a high rate of distant metastases (28.9%). Salvage surgery can be considered in patients with limited local and/or regional recurrences, even in case of single distant relapse, appearing within the first 3 years of follow-up.
Collapse
Affiliation(s)
- Giuditta Mannelli
- Department of Experimental and Clinical MedicineUniversity of FlorenceFlorence
| | - Lara V. Comini
- Head and Neck Oncology and Robotic Surgery, Department of Experimental and Clinical MedicineUniversity of FlorenceFlorenceItaly
| | - Andrea Sacchetto
- Otorhinolaryngology‐Head and Neck Surgery, Department of Surgery, Dentistry, Gynecology, and PediatricsUniversity of Verona, University Hospital of VeronaVeronaItaly
| | - Roberto Santoro
- Department of Experimental and Clinical MedicineUniversity of FlorenceFlorence
| | - Giuseppe Spinelli
- Department of Maxillo Facial SurgeryAzienda Ospedaliero‐Universitaria CareggiFlorenceItaly
| | - Pierluigi Bonomo
- Radiation OncologyAzienda Ospedaliero‐Universitaria CareggiFlorenceItaly
| | - Isacco Desideri
- Radiation OncologyAzienda Ospedaliero‐Universitaria CareggiFlorenceItaly
| | - Paolo Bossi
- Department of Medical and Surgical Specialties, Radiological Sciences and Public HealthUniversity of Brescia, ASST (Azienda Socio Sanitaria Territoriale) Spedali CiviliBresciaItaly
| | - Ester Orlandi
- Radiation Oncology, Clinical DepartmentNational Center for Oncological Hadrontherapy (CNAO)PaviaItaly
| | - Giammarco Alderotti
- Department of Statistics, Computer ScienceApplications “G. Parenti” (DiSIA), University of FlorenceFlorenceItaly
| | - Alessandro Franchi
- Department of Translational ResearchSchool of Medicine, University of PisaPisaItaly
| | - Annarita Palomba
- Unit of Histopathology and Molecular Diagnostics, Careggi University HospitalFlorenceItaly
| | - Albino Eccher
- Pathology Unit, Department of Pathology and DiagnosticsUniversity Hospital of VeronaVeronaItaly
| | - Daniele Marchioni
- Otorhinolaryngology‐Head and Neck Surgery, Department of Surgery, Dentistry, Gynecology, and PediatricsUniversity of Verona, University Hospital of VeronaVeronaItaly
| | - Riccardo Nocini
- Otorhinolaryngology‐Head and Neck Surgery, Department of Surgery, Dentistry, Gynecology, and PediatricsUniversity of Verona, University Hospital of VeronaVeronaItaly
| | - Cesare Piazza
- Unit of Otorhinolaryngology—Head and Neck Surgery, ASST Spedali Civili of BresciaUniversity of BresciaBresciaItaly
| | - Gabriele Molteni
- Otorhinolaryngology‐Head and Neck Surgery, Department of Surgery, Dentistry, Gynecology, and PediatricsUniversity of Verona, University Hospital of VeronaVeronaItaly
| |
Collapse
|
|
11
|
Wiles AB, Gabrielson M, Baloch ZW, Faquin WC, Jo VY, Callegari F, Kholova I, Song S, Centeno BA, Ali SZ, Tommola S, Fadda G, Petrone G, Wang H, Rossi ED, Pantanowitz L, Maleki Z. Secretory carcinoma of the salivary gland, a rare entity: An international multi-institutional study. Cancer Cytopathol 2022; 130:684-694. [PMID: 35385604 PMCID: PMC9545582 DOI: 10.1002/cncy.22574] [Citation(s) in RCA: 21] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/31/2021] [Revised: 02/16/2022] [Accepted: 02/28/2022] [Indexed: 01/29/2023]
Abstract
BACKGROUND Secretory carcinoma (SC) of the salivary gland is a rare entity with limited published literature on cytomorphology. The authors present the largest cohort to date of SC fine‐needle aspiration (FNA) cases. METHODS FNA cases of histologically confirmed SC were retrospectively retrieved from 12 academic institutions in the United States, Italy, Finland, and Brazil. The collated data included patient demographics, imaging findings, cytopathologic diagnoses according to the Milan System for Reporting Salivary Gland Cytopathology, cytomorphologic characteristics, and immunohistochemical/molecular profiles. RESULTS In total, 40 SCs were identified (male‐to‐female ratio, 14:26) in patients with a mean age of 52 years (age range, 13‐80 years). Ultrasound imagining revealed a hypoechoic, ovoid, poorly defined, or lobulated mass. The most common primary site was the parotid gland (30 of 40 tumors). Regional lymph node metastasis (9 patients) and distant metastasis (4 patients; brain, liver, lungs, and mediastinum) were noted. Two patients died of disease. FNA smears were cellular and demonstrated mainly large, round cells with intracytoplasmic vacuoles or granules and round‐to‐oval nuclei with smooth nuclear contour, minimal irregularities, and prominent nucleoli arranged predominantly in clusters, papillary formations, and single cells. The background was variable and contained inflammatory cells, mucin, or proteinaceous material. The diagnoses were malignant (19 of 38 tumors; 50%), suspicious for malignancy (10 of 38 tumors; 26%), salivary gland neoplasm of uncertain malignant potential (7 of 38 tumors; 18%), and atypia of undetermined significance (2 of 38 tumors; 6%) according to the Milan System for Reporting Salivary Gland Cytopathology. Two malignant cases (2 of 40 tumors; 5%) were metastases. The neoplastic cells were immunoreactive for S100 (23 of 24 tumors), mammaglobin (18 of 18 tumors), GATA‐3 (13 of 13 tumors), AE1/AE3 (7 of 7 tumors), and vimentin (6 of 6 tumors). ETV6‐NTRK3 fusion was detected in 32 of 33 tumors by fluorescence in situ hybridization (n = 32) and next‐generation sequencing (n = 1). CONCLUSIONS Familiarity with cytomorphologic features and the immunohistochemical/molecular profile of SC can enhance diagnostic accuracy.; Secretory carcinoma of the salivary gland, which may manifest with diverse cytomorphology, mammaglobin expression, and ETV6 rearrangement or ETV6‐NTRK3 fusion, was investigated along with cytomorphologic features to facilitate an accurate diagnosis. The results indicated that familiarity with these features and with the immunohistochemical/molecular profile of secretory carcinoma of the salivary gland enhanced diagnostic accuracy.
Collapse
Affiliation(s)
- Austin B Wiles
- Department of Pathology, Virginia Commonwealth University, Richmond, Virginia
| | - Matthew Gabrielson
- Department of Pathology, The Johns Hopkins Hospital, Baltimore, Maryland
| | - Zubair W Baloch
- Department of Pathology, Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania
| | - William C Faquin
- Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts
| | - Vickie Y Jo
- Department of Pathology, Brigham and Women's Hospital, Boston, Massachusetts
| | - Fabiano Callegari
- Department of Pathology, Federal University of Sao Paulo, Sao Paulo, Brazil
| | - Ivana Kholova
- Department of Pathology, Tampere University, Faculty of Medicine and Health Technology, Tampere, Finland.,Pathology, Fimlab Laboratories, Tampere, Finland
| | - Sharon Song
- Department of Pathology, Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania
| | | | - Syed Z Ali
- Department of Pathology, The Johns Hopkins Hospital, Baltimore, Maryland
| | - Satu Tommola
- Department of Pathology, Tampere University, Faculty of Medicine and Health Technology, Tampere, Finland.,Pathology, Fimlab Laboratories, Tampere, Finland
| | - Guido Fadda
- Department of Pathology, Catholic University of Rome, Rome, Italy
| | | | - He Wang
- Department of Pathology, Yale University, New Haven, Connecticut
| | - Esther D Rossi
- Department of Pathology, Catholic University of Rome, Rome, Italy
| | - Liron Pantanowitz
- Department of Pathology, University of Michigan, Ann Arbor, Michigan
| | - Zahra Maleki
- Department of Pathology, The Johns Hopkins Hospital, Baltimore, Maryland
| |
Collapse
|
|
12
|
Alves LDB, de Melo AC, Farinha TA, de Lima Araujo LH, Thiago LDS, Dias FL, Antunes HS, Amaral Eisenberg AL, Santos Thuler LC, Cohen Goldemberg D. A systematic review of secretory carcinoma of the salivary gland: where are we? Oral Surg Oral Med Oral Pathol Oral Radiol 2021; 132:e143-e152. [PMID: 32493686 DOI: 10.1016/j.oooo.2020.04.007] [Citation(s) in RCA: 19] [Impact Index Per Article: 4.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/10/2019] [Revised: 03/22/2020] [Accepted: 04/11/2020] [Indexed: 12/12/2022]
Abstract
OBJECTIVE The aim of this systematic review was to describe the epidemiology, diagnostic criteria, differential diagnosis, treatment, prognostic factors, and treatment outcomes of secretory carcinoma. STUDY DESIGN A comprehensive search of Lilacs, PubMed, Science Direct, and Web of Science databases was conducted to identify all case reports, letter to the editor, and histopathologic reclassifications regarding salivary gland secretory carcinoma published in English, Spanish, French, and Portuguese. RESULTS The final analysis included 119 studies, which totaled 642 secretory carcinoma diagnoses, with 239 case reports and 403 diagnostic reclassifications, mostly in the United States. The age range was 5 to 87 years, and cases were predominantly in males (58.7%) and mostly affecting the parotid glands (73.7%). The disease usually presents as a slow-growing, painless mass. The main differential diagnosis is acinic cell carcinoma, and the tumor is usually treated with surgery. The prognosis is considered favorable, although there have been reports of local recurrences, distant metastases, and deaths. CONCLUSIONS It is important that clinicians become aware of this salivary gland neoplasm and report clinical data, clinical course, management and long-term follow-up. There is an urgent need to conduct more clinical trials, especially on tropomyosin receptor kinase (TRK) inhibitors and other potential target therapy modalities.
Collapse
Affiliation(s)
| | | | - Thayana Alves Farinha
- Scientific Initiation Student, Clinical Research Division, National Cancer Institute of Brazil, Rio de Janeiro, Brazil (INCA)
| | | | - Leandro de Souza Thiago
- Clinical Research Division, National Cancer Institute of Brazil, Rio de Janeiro, Brazil (INCA)
| | - Fernando Luiz Dias
- Head and Neck Surgery Service, National Cancer Institute of Brazil, Rio de Janeiro, Brazil (INCA)
| | | | | | | | - Daniel Cohen Goldemberg
- Clinical Research Division, National Cancer Institute of Brazil, Rio de Janeiro, Brazil (INCA).
| |
Collapse
|
|
13
|
Silver E, Lazow SK, Peters SM, Solomon M, Yusupov S. Secretory Carcinoma of the Salivary Gland - Systematic Review of the Literature and Report of 2 Cases. J Oral Maxillofac Surg 2021; 80:101-112. [PMID: 34653372 DOI: 10.1016/j.joms.2021.08.155] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/29/2021] [Revised: 08/19/2021] [Accepted: 08/19/2021] [Indexed: 01/16/2023]
Abstract
PURPOSE Secretory carcinoma (SC) of the salivary gland, formerly known as mammary analogue secretory carcinoma, is an uncommon and fairly newly described low grade malignant neoplasm of the salivary gland. Given the small number of cases reported in the literature to date, treatment guidelines are scarce. This study aimed to describe the clinical characteristics of SC, discuss prior management strategies, and provide recommendations for future treatment. METHODS We performed a systematic review of all the cases of SC reported in the literature since it was first recognized in 2010. Using Pubmed, Crossref, and Google Scholar, we identified all articles reporting cases of SC. RESULTS We identified 657 cases of SC in 109 articles. In addition, we provided 2 new cases, for a total of 659 cases in 110 articles. To our knowledge, this is the largest review of cases of SC in the literature to date. We summarized the clinical characteristics of SC, as well as the nodal status, clinical management, recurrence rate, and death rate. CONCLUSIONS SC occurs on average in middle age (with a large age range), presents most often initially as localized disease without metastasis, and has a low but not insignificant recurrence rate. Deaths have been reported. The generalized recommendations for treatment of SC are in line with those of other low-grade salivary gland malignancies.
Collapse
Affiliation(s)
- Eric Silver
- Resident, Oral and Maxillofacial Surgery, Kings County Hospital Center, Brooklyn, NY.
| | - Stewart K Lazow
- Professor and Vice-Chairman, Oral Maxillofacial Surgery Program Director, Retired, Kings County Hospital Center/SUNY Brooklyn, Brooklyn, NY
| | - Scott M Peters
- Assistant Professor, Oral Pathology, Columbia University, New York, NY
| | - Marshall Solomon
- Professor Emeritus, Oral Maxillofacial Pathology, Kings County Hospital Center, Brooklyn, NY
| | - Steve Yusupov
- Director, Oral and Maxillofacial Oncology, Staten Island University Hospital/Northwell Health, Staten Island, NY
| |
Collapse
|
|
14
|
Pang Y, Sun L, Liu H, Ma J. Differential diagnosis and treatment of salivary secretory carcinoma and acinic cell carcinoma. Oral Oncol 2021; 119:105370. [PMID: 34157553 DOI: 10.1016/j.oraloncology.2021.105370] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2021] [Revised: 05/10/2021] [Accepted: 05/25/2021] [Indexed: 02/08/2023]
Abstract
Secretory carcinoma (SC) and acinic cell carcinoma (AciCC) are two rare tumors originating in the salivary gland of the head and neck. Before the World Health Organization (WHO) classified SC as a new entity in 2017, the majority of SC cases were incorrectly diagnosed as AciCC. Indeed, they are similar in biological behaviors, clinical manifestations and histomorphological features. Especially, SC and zymogen granule-poor AciCC are difficult to differentiate, which brings a tough challenge in clinical diagnosis. This article provides an updated understanding of the differential diagnosis in SC and AciCC from two main perspectives: histopathology and molecular genetics. The targeted therapies for both tumors are also mentioned. It aims to give some hints in clinical diagnosis and treatment, in hopes that patients with adequate diagnosis could obtain the opportunityformore effective treatment.
Collapse
Affiliation(s)
- Yu Pang
- Department of Medical Oncology, West China Hospital, Sichuan University, Chengdu, Sichuan Province 610041, PR China; State Key Laboratory of Oral Diseases, National Clinical Research Center for Oral Diseases, West China Hospital of Stomatology, Sichuan University, Chengdu, Sichuan Province 610041, PR China.
| | - Lingqi Sun
- Department of Neurology, Air Force Hospital of Western Theater Command, Chengdu, Sichuan Province 610041, PR China
| | - Huijia Liu
- Department of Medical Oncology, West China Hospital, Sichuan University, Chengdu, Sichuan Province 610041, PR China
| | - Ji Ma
- Department of Medical Oncology, West China Hospital, Sichuan University, Chengdu, Sichuan Province 610041, PR China.
| |
Collapse
|
|
15
|
Min FH, Li J, Tao BQ, Liu HM, Yang ZJ, Chang L, Li YY, Liu YK, Qin YW, Liu WW. Parotid mammary analogue secretory carcinoma: A case report and review of literature. World J Clin Cases 2021; 9:4052-4062. [PMID: 34141766 PMCID: PMC8180236 DOI: 10.12998/wjcc.v9.i16.4052] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/18/2021] [Revised: 02/17/2021] [Accepted: 03/13/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Mammary analogue secretory carcinoma (MASC) is a rare low-grade malignant salivary gland tumor. The morphological and immunohistochemical features of MASC closely resemble those of breast secretory carcinoma. The key characteristics of the lesion are a lack of pain and slow growth. There is no obvious specificity in the clinical manifestations and imaging features. The diagnosis of the disease mainly depends on the detection of the MASC-specific ETV6-NTRK3 fusion gene.
CASE SUMMARY This report describes a rare case of a 32-year-old male patient who presented with a gradually growing lesion that was initially diagnosed as breast-like secretory carcinoma of the right parotid gland. Imaging and histological investigations were used to overcome the diagnostic difficulties. The lesion was managed with right parotidectomy, facial nerve preservation, biological patch implantation to restore the resulting defect, and postoperative radiotherapy. On postoperative follow-up, the patient reported a mild facial deformity with no complications, signs of facial paralysis, or Frey’s syndrome.
CONCLUSION The imaging and histological diagnostic challenges for MASC are discussed.
Collapse
Affiliation(s)
- Feng-He Min
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Jia Li
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
| | - Bo-Qiang Tao
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Hui-Min Liu
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Zhi-Jing Yang
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Lu Chang
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Yu-Yang Li
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
- Department of Oral Implant, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
| | - Ying-Kun Liu
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| | - Yi-Wen Qin
- Department of Stomatology, Chongqing Medical University, Chongqing 400016, China
| | - Wei-Wei Liu
- Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Jilin University, Changchun 130021, Jilin Province, China
- Jilin Provincial Key Laboratory of Tooth Development and Bone Remodeling, Changchun 130021, Jilin Province, China
| |
Collapse
|