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A symptomatic intercalated duct lesion of the parotid gland: a case report with immunohistochemical and genetic analyses

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Abstract

Intercalated duct lesions (IDLs) are usually asymptomatic. We report a case of IDL, in which a palpable mass formed. The patient was a 45-year-old Japanese male, who noticed a mass in the left parotid region. The nodular lesion was well-circumscribed, but did not have a fibrous capsule or exhibit infiltrative growth. It contained a small cystic space and consisted of basaloid cells arranged in a cribriform pattern and inner ductal cells. It had some solid areas of nest-like proliferation displaying mild cellular atypia. Immunohistochemically, the luminal cells were positive for cytokeratin (CK)7 and epithelial membrane antigen, and the abluminal cells were positive for CK5/6, p63, and DOG1. S-100 protein-positive stromal cells were also seen. The lesion’s cells were all positive for SOX10, and the nuclei of some basaloid cells were positive for β-catenin. The Ki-67 labeling index was 3.8%. The ductal cells contained diastase-digestion-resistant, Periodic acid Schiff-positive zymogen granules. Genetically, the lesion harbored a missense mutation in the CTNNB1 gene. We diagnosed the lesion as an IDL. As IDLs are usually small non-neoplastic lesions, symptomatic cases are rare. Based on its common immunohistochemical and genetic features, IDL may be a precursor of basal cell adenoma/adenocarcinoma, such as intercalated duct adenoma.

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Availability of data and materials

The data sets used and analyzed during the current study are available from the corresponding author on reasonable request.

Abbreviations

IDL:

Intercalated duct lesion

CK:

Cytokeratin

ADP/H:

Adenomatous ductal proliferation/hyperplasia

IDH:

Intercalated duct hyperplasia

BCA:

Basal cell adenoma

BCAC:

Basal cell adenocarcinoma

EMC:

Epithelial-myoepithelial carcinoma

D-PAS:

Diastase-digested, Periodic acid Schiff

FFPE:

Formalin-fixed, paraffin-embedded

EMA:

Epithelial membrane antigen

ASMA:

Alpha-smooth muscle actin

PLAG1:

Pleomorphic adenoma gene 1

SOX10:

SRY-related HMG-box 10

DOG1:

Discovered of GIST 1

PCR:

Polymerase chain reaction

IDA:

Intercalated duct adenoma

LEF-1:

Lymphoid enhancer-binding factor 1

H & E:

Hematoxylin and eosin

References

  1. Chiosea S, Seethala R, Williams MD (2017) Intercalated duct hyperplasia. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ (eds) WHO Classification of Head and Neck Tumours, 4th edn. IARC Press, Lyon, France, p p197

    Google Scholar 

  2. Yu GY, Donath K (2001) Adenomatous ductal proliferation on the salivary gland. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 911:215–221

    Article  Google Scholar 

  3. Di Palma S (1994) Epithelial-myoepithelial carcinoma with co-existing multifocal intercalated duct hyperplasia of the parotid gland. Histopathology 25:494–496

    Article  PubMed  Google Scholar 

  4. Chetty R (2000) Intercalated duct hyperplasia: possible relationship to epithelial-myoepithelial carcinoma and hybrid tumours of salivary gland. Histopathology 37:260–263

    Article  CAS  PubMed  Google Scholar 

  5. Weinreb I, Seethala RR, Hunt JL, Chetty R, Dardick I, Perez-Ordonez B (2009) Intercalated duct lesions of salivary gland: a morphologic spectrum from hyperplasia to adenoma. Am J Surg Pathol 33:1322–1329

    Article  PubMed  Google Scholar 

  6. Montalli VA, Martinez E, Tincani A, Martins A, Abreu Mdo C, Neves C, Costa AF, Araújo VC, Altemani A (2014) Tubular variant of basal cell adenoma shares immunohistochemical features with normal intercalated ducts and is closely related to intercalated duct lesions of salivary gland. Histopathology 64:880–889

    Article  PubMed  Google Scholar 

  7. Ortolani E, Polimeni A, Lauriola L, Coli A (2016) Basal cell adenoma with S-100 positive stroma; a case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol 1211:e62–e64

    Article  Google Scholar 

  8. Jo VY, Sholl LM, Krane JF (2016) Distinctive pattern of CTNNB1 (β-catenin) alterations in salivary gland basal cell adenoma and basal cell adenocarcinoma. Am J Surg Pathol 40:1143–1150

    Article  PubMed  Google Scholar 

  9. Kawahara A, Harada H, Abe H, Yamaguchi T, Taira T, Nakashima K, Mihashi H, Akiba J, Kage M (2011) Nuclear beta-catenin expression in basal cell adenomas of salivary gland. J Oral Pathol Med 40:460–466

    Article  CAS  PubMed  Google Scholar 

  10. Sato M, Yamamoto H, Hatanaka Y, Nishijima T, Jiromaru R, Yasumatsu R, Taguchi K, Masuda M, Nakagawa T, Oda Y (2018) Wnt/beta-catenin signal alteration and its diagnostic utility in basal cell adenoma and histologically similar tumors of the salivary gland. Pathol Res Prac 214:586–592

    Article  CAS  Google Scholar 

  11. Seki N, Yamazaki N, Ikeda T, Harada H, Himi T (2017) A symptomatic case of adenomatous ductal proliferation/hyperplasia with a large cystic lesion. Case Rep Oncol 10:676–682

    Article  PubMed  PubMed Central  Google Scholar 

  12. Adhikari BR, Nishimura M, Takimoto K, Harada F, Onishi A, Hiraki D, Paudel D, Neopan P, Utsunomiya M, Morikawa T, Yoshida K, Sato J, Shakya M, Nakayama E, Nagayasu H, Abiko Y (2018) Adenomatous ductal proliferation/hyperplasia in the parotid gland associated without any other pathological lesions; a report and survey of the literatures. Med Mol Morphol 51:244–248

    Article  PubMed  Google Scholar 

  13. Skalova A, Hyrcza MD, Leivo I (2022) Update from the 5th edition of the world health organization classification of head and neck tumors salivary glands. Head Neck Pathol 16:40–53

    Article  PubMed  Google Scholar 

  14. Mok Y, Pang YH, Teh M, Petersson F (2016) Hybrid intercalated duct lesion of the parotid: diagnostic challenges of a recently described entity with fine needle aspiration finding. Head Neck Pathol 10:269–274

    Article  PubMed  Google Scholar 

  15. Adi MM, Chisholm DM, Waterhouse JP (1994) Stereological and immunohistochemical study of development of human fetal labial salivary glands and their S-100 protein reactivity. J Oral Pathol Med 23:36–40

    Article  CAS  PubMed  Google Scholar 

  16. Ohtomo R, Mori T, Shibata S, Tsuta K, Maeshima AM, Akazawa C, Watabe Y, Honda K, Yamada T, Yoshimoto S, Asai M, Okano H, Kanai Y, Tsuda H (2013) SOX10 is a novel marker of acinus and intercalated duct differentiation in salivary gland tumors: a clue to the histogenesis for tumor diagnosis. Mod Pathol 26:1041–1050

    Article  CAS  PubMed  Google Scholar 

  17. Rooper LM, McCuiston AM, Westra WH, Bishop JA (2019) SOX10 immunoexpression in basaloid squamous cell carcinomas: a diagnostic pitfall for ruling out salivary differentiation. Head Neck Pathol 13:543–547

    Article  PubMed  Google Scholar 

  18. Hsieh MS, Lee YH, Chang YL (2016) SOX10-positive salivary gland tumors: a growing list, including mammary analogue secretory carcinoma of the salivary gland, sialoblastoma, low-grade salivary duct carcinoma, basal cell adenoma/adenocarcinoma, and a subgroup of mucoepidermoid carcinoma. Hum Pathol 56:134–142

    Article  CAS  PubMed  Google Scholar 

  19. Chenevert J, Duvvuri U, Chiosea S, Dacic S, Cieply K, Kim J, Shiwarski D, Seethala RR (2012) DOG1: a novel marker of salivary acinar and intercalated differentiation. Mod Pathol 25:919–929

    Article  CAS  PubMed  Google Scholar 

  20. Bilodeau EA, Acquafondata M, Barnes EL, Seethala RR (2015) A comparative analysis of LEF-1 in odontogenic and salivary tumors. Hum Pathol 46:255–259

    Article  CAS  PubMed  Google Scholar 

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Acknowledgements

The authors thank Mr. Naofumi Ishikawa; Ms. Yoko Yamazaki; Ms. Chinatsu Tsuchiya; Mr. Kensuke Shimazaki; Ms. Aki Kubota; Mr. Koji Takahashi; Ms. Nobuyo Tsujino; Mr. Yohei Saguchi; Mr. Jun-ichi Sakano; and the medical staff at the Department of Pathology, Shizuoka General Hospital, Shizuoka, Japan, for the technical assistance they provided. We are also grateful to Ms. Mamiko Uemura, Ms. Tomo Terao, and Ms. Misato Iwama, doctors’ assistants at the Department of Pathology, Shizuoka General Hospital, Shizuoka, Japan, for their help with the preparation of the manuscript.

Funding

This study was supported in part by a Grant-in-Aid from the Medical Research Support Project of Shizuoka Prefectural Hospital Organization in 2021 (to KK).

Author information

Authors and Affiliations

  1. Department of Pathology, Shizuoka General Hospital, 4-27-1 Kita-Ando, Aoi-ku, Shizuoka, Shizuoka, 420-8527, Japan

    Kimihide Kusafuka, Kazuki Hirata, Akinori Murakami, Aya Muramatsu, Kazumori Arai & Makoto Suzuki

  2. Department of Diagnostic Pathology, Hamamatsu University Hospital, Hamamatsu, Shizuoka, Japan

    Satoshi Baba

  3. Department of Otorhinolaryngology-Head and Neck Surgery, Shizuoka General Hospital, Shizuoka, Shizuoka, Japan

    Yoshiharu Kitani

Authors
  1. Kimihide Kusafuka
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  2. Satoshi Baba
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  3. Yoshiharu Kitani
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  4. Kazuki Hirata
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  5. Akinori Murakami
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  6. Aya Muramatsu
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  7. Kazumori Arai
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  8. Makoto Suzuki
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Contributions

KK designed and drafted the manuscript, and KK, AM, AM, KA, and MS made the histopathological diagnoses and analyzed the immunohistochemical results. KH performed the excellent immunohistochemistry. SB performed the molecular analysis using PCR-based direct sequencing. MS supervised the manuscript. All of the authors have read and approved the final manuscript.

Corresponding author

Correspondence to Kimihide Kusafuka.

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Competing interests

The authors declare that they have no competing interests relating to this study.

Ethical approval and consent to participate

This study was approved by the institutional review board of Shizuoka General Hospital (SGHIRB#2019007). All subjects signed informed consent forms before participating.

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All subjects signed informed consent forms before participating.

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Kusafuka, K., Baba, S., Kitani, Y. et al. A symptomatic intercalated duct lesion of the parotid gland: a case report with immunohistochemical and genetic analyses. Med Mol Morphol 55, 329–336 (2022). https://doi.org/10.1007/s00795-022-00328-7

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  • DOI: https://doi.org/10.1007/s00795-022-00328-7

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