Journal of Pathology and Translational Medicine

Case Studies

Adenocarcinoma of the minor salivary gland with concurrent MAML2 and EWSR1 alterations: Sangjoon Choi, Junhun Cho, Seung Eun Lee, Chung-Hwan Baek, Yi-Kyung Kim, Hyung-Jin Kim, Young Hyeh Ko; J Pathol Transl Med. 2021;55(2):132-138. Published online January 22, 2021; DOI: https://doi.org/10.4132/jptm.2020.12.11

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Salivary gland tumors are histologically diverse, and each entity has distinctive histopathological and molecular features. We report two cases of salivary gland tumors with unique histological and molecular findings, which have not been documented previously. The tumors were located in the base of the tongue in both patients. Most tumor cells were arranged in cords and nests, giving a trabecularlike appearance. Focally, glandular structures with intraluminal mucin and perivascular pseudorosette-like configurations were identified. Tumor cells had eosinophilic to clear cytoplasm, and showed mild nuclear atypia. They were positive for pancytokeratin and negative for S-100, p63, c-KIT, androgen receptor, and neuroendocrine markers. Multiple foci of capsular or lymphovascular invasion were identified, but the Ki-67 labeling index was low (< 5%). Fluorescence in situ hybridization revealed concurrent alterations of MAML2 and EWSR1 gene. Further investigations with a larger number of cases with similar histological and molecular features will accurately classify this tumor.

Citations

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Salivary Gland Neoplasms With a Unique Trabecular Histology and MAML2 Translocation
Bokyung Ahn, Seung-Ho Choi, Doeun Kim, Deokhoon Kim, Kyung-Ja Cho
American Journal of Surgical Pathology.2023; 47(10): 1085. CrossRef
Mesonephric-like Adenocarcinoma of the Ovary: Clinicopathological and Molecular Characteristics
Hyun Hee Koh, Eunhyang Park, Hyun-Soo Kim
Diagnostics.2022; 12(2): 326. CrossRef
The evolving role of molecular pathology in the diagnosis of salivary gland tumours with potential pitfalls
Kanwalpreet Kaur, Shailee Mehta, Sangita Vanik, Priti Trivedi, Nirmalya Banerjee, Harsh Dhar, Sourav Datta, Subhadeep Karanjai
European Archives of Oto-Rhino-Laryngology.2022; 279(8): 3769. CrossRef
Alveolar Soft Part Sarcoma of the Uterus: Clinicopathological and Molecular Characteristics
Yurimi Lee, Kiyong Na, Ha Young Woo, Hyun-Soo Kim
Diagnostics.2022; 12(5): 1102. CrossRef
Endometrioid Carcinomas of the Ovaries and Endometrium Involving Endocervical Polyps: Comprehensive Clinicopathological Analyses
Jihee Sohn, Yurimi Lee, Hyun-Soo Kim
Diagnostics.2022; 12(10): 2339. CrossRef
Mesonephric-like Differentiation of Endometrial Endometrioid Carcinoma: Clinicopathological and Molecular Characteristics Distinct from Those of Uterine Mesonephric-like Adenocarcinoma
Sujin Park, Go Eun Bae, Jiyoung Kim, Hyun-Soo Kim
Diagnostics.2021; 11(8): 1450. CrossRef
Mesonephric-like Adenocarcinoma of the Uterine Corpus: Comprehensive Immunohistochemical Analyses Using Markers for Mesonephric, Endometrioid and Serous Tumors
Hyunjin Kim, Kiyong Na, Go Eun Bae, Hyun-Soo Kim
Diagnostics.2021; 11(11): 2042. CrossRef

A Rare Case of Adenosquamous Carcinoma Arising in the Background of IgG4-Related Lung Disease: Sangjoon Choi, Sujin Park, Man Pyo Chung, Tae Sung Kim, Jong Ho Cho, Joungho Han; J Pathol Transl Med. 2019;53(3):188-191. Published online March 11, 2019; DOI: https://doi.org/10.4132/jptm.2019.02.21

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Abstract PDF

IgG4-related disease is a systemic inflammatory disease and is known as IgG4-related lung disease (IgG4-RLD) when it involves the respiratory system. Primary lung cancer arising from a background of IgG4-RLD is very rare. Herein, we report a case of adenosquamous carcinoma arising from the background of IgG4-RLD and presenting as an interstitial lung disease pattern. A 66-year-old man underwent lobectomy under the impression of primary lung cancer. Grossly, the mass was ill-defined and gray-tan colored, and the background lung was fibrotic. Microscopically, tumor cells showed both squamous and glandular differentiation. Dense lymphoplasmacytic infiltration with fibrosis and obliterative phlebitis were seen in the background lung. IgG4 immunohistochemical stain showed diffuse positivity in infiltrating plasma cells. Primary lung adenosquamous carcinoma has not been reported in a background of IgG4-RLD. Due to the rarity of IgG4-RLD, physicians must follow patients with IgG4-RLD over long periods of time to accurately predict the risk of lung cancer.

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Endometrioid Carcinomas of the Ovaries and Endometrium Involving Endocervical Polyps: Comprehensive Clinicopathological Analyses
Jihee Sohn, Yurimi Lee, Hyun-Soo Kim
Diagnostics.2022; 12(10): 2339. CrossRef
A Case of IgG4-related Disease Composed of a Paravertebral Tumor Alone with Multiple Lung Cancers
Mutsumi Ozasa, Toyomitsu Sawai, Yosuke Harada, Sumako Yoshioka, Nobuko Matsuo, Hiroshi Mukae
Haigan.2021; 61(3): 213. CrossRef
Serous Carcinoma of the Endometrium with Mesonephric-Like Differentiation Initially Misdiagnosed as Uterine Mesonephric-Like Adenocarcinoma: A Case Report with Emphasis on the Immunostaining and the Identification of Splice Site TP53 Mutation
Sangjoon Choi, Yoon Yang Jung, Hyun-Soo Kim
Diagnostics.2021; 11(4): 717. CrossRef
A Case of IgG4-related Thyroiditis Diagnosed by Total Thyroidectomy
Daiki Sakamoto, Masao Yagi, Hiroshi Iwai
Practica Oto-Rhino-Laryngologica.2021; 114(7): 547. CrossRef
Mesonephric-like Differentiation of Endometrial Endometrioid Carcinoma: Clinicopathological and Molecular Characteristics Distinct from Those of Uterine Mesonephric-like Adenocarcinoma
Sujin Park, Go Eun Bae, Jiyoung Kim, Hyun-Soo Kim
Diagnostics.2021; 11(8): 1450. CrossRef
Mesonephric-like Adenocarcinoma of the Uterine Corpus: Comprehensive Immunohistochemical Analyses Using Markers for Mesonephric, Endometrioid and Serous Tumors
Hyunjin Kim, Kiyong Na, Go Eun Bae, Hyun-Soo Kim
Diagnostics.2021; 11(11): 2042. CrossRef
Not Cancer After All: Two Rare Cases of IgG4-Related Lung Disease
Josué Pinto, Carla Damas, António Morais
Archivos de Bronconeumología.2020; 56(1): 53. CrossRef
Not Cancer After All: Two Rare Cases of IgG4-Related Lung Disease
Josuèc) Pinto, Carla Damas, António Morais
Archivos de Bronconeumología (English Edition).2020; 56(1): 52. CrossRef
Axillary lymphadenopathy with IgG4 positive plasma cell infiltration as differential diagnosis of metastatic lung adenocarcinoma
Yutaro Ito, Masanori Harada, Namio Kagoo, Tsutomu Kubota, Koshiro Ichijyo, Eisuke Mochizuki, Masahiro Uehara, Shun Matsuura, Masaru Tsukui, Naoki Koshimizu
Respiratory Medicine Case Reports.2020; 31: 101196. CrossRef

Abrupt Dyskeratotic and Squamoid Cells in Poorly Differentiated Carcinoma: Case Study of Two Thoracic NUT Midline Carcinomas with Cytohistologic Correlation: Taebum Lee, Sangjoon Choi, Joungho Han, Yoon-La Choi, Kyungjong Lee; J Pathol Transl Med. 2018;52(5):349-353. Published online July 27, 2018; DOI: https://doi.org/10.4132/jptm.2018.07.16

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Abstract PDF

Cytologic diagnosis of nuclear protein in testis (NUT) midline carcinoma (NMC) is important due to its aggressive behavior and miserable prognosis. Early diagnosis of NMC can facilitate proper management, and here we report two rare cases of thoracic NMC with cytohistologic correlation. In aspiration cytology, the tumor presented with mixed cohesive clusters and dispersed single cells, diffuse background necrosis and many neutrophils. Most of the tumor cells had scanty cytoplasm and medium-sized irregular nuclei, which had fine to granular nuclear chromatin. Interestingly, a few dyskeratotic cells or squamoid cell clusters were present in each case. Biopsy specimen histology revealed more frequent squamous differentiation, and additional immunohistochemistry tests showed nuclear expression of NUT. Because this tumor has a notorious progression and has been previously underestimated in terms of its prevalence, awareness of characteristic findings and proper ancillary tests should be considered in all suspicious cases.

Citations

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Diagnostic significance and cytological features of NUT carcinoma by EBUS‐FNA, a case report and literature review
Yaping Ju, Miriam Velazquez, Andy Sherrod, Tiannan Wang
Cytopathology.2024; 35(4): 497. CrossRef
Exploring cytologic features and potential diagnostic challenges of metastatic NUT carcinoma to the parotid gland: A case report and a comprehensive literature review
Crystal Y. Li, Salih Salihoglu, Francisco J. Civantos, Jaylou M. Velez Torres
Diagnostic Cytopathology.2024;[Epub] CrossRef
Nuclear Protein in Testis (NUT) Carcinoma: A Comprehensive Immunohistochemical Analysis of 57 Cases With Consideration of Interpretation and Pitfall Recognition
Ayesha Farooq, Allison L. Kerper, Jennifer M. Boland, Ying-Chun Lo
Archives of Pathology & Laboratory Medicine.2024; 148(8): 898. CrossRef
BRD3‐NUTM1‐expressing NUT carcinoma of lung on endobronchial ultrasound‐guided transbronchial needle aspiration cytology, a diagnostic pitfall
Sameer Chhetri Aryal, Shereen Zia, Shannon Rodgers, Yulei Shen, Kyle Perry, Lisi Yuan
Diagnostic Cytopathology.2022;[Epub] CrossRef
Nuclear protein of the testis midline carcinoma of the thorax
Ayae Saiki, Keita Sakamoto, Yuan Bee, Takehiro Izumo
Japanese Journal of Clinical Oncology.2022; 52(6): 531. CrossRef
Approach to Mediastinal Fine Needle Aspiration Cytology
Zaibo Li, Huihong Xu, Fang Fan
Advances in Anatomic Pathology.2022; 29(6): 337. CrossRef
Diagnosis, Treatment and Prognosis of Primary Pulmonary NUT Carcinoma: A Literature Review
Jiaqian Yuan, Zhili Xu, Yong Guo
Current Oncology.2022; 29(10): 6807. CrossRef
Case report: Immunovirotherapy as a novel add-on treatment in a patient with thoracic NUT carcinoma
Linus D. Kloker, Branko Calukovic, Katrin Benzler, Alexander Golf, Sebastian Böhm, Sven Günther, Marius Horger, Simone Haas, Susanne Berchtold, Julia Beil, Mary E. Carter, Tina Ganzenmueller, Stephan Singer, Abbas Agaimy, Robert Stöhr, Arndt Hartmann, Tho
Frontiers in Oncology.2022;[Epub] CrossRef
Cytomorphology of primary pulmonary NUT carcinoma in different cytology preparations
Rimlee Dutta, Aruna Nambirajan, Saurabh Mittal, Sinchita Roy‐Chowdhuri, Deepali Jain
Cancer Cytopathology.2021; 129(1): 53. CrossRef
Update on genetically defined lung neoplasms: NUT carcinoma and thoracic SMARCA4-deficient undifferentiated tumors
Kyriakos Chatzopoulos, Jennifer M. Boland
Virchows Archiv.2021; 478(1): 21. CrossRef
Immunotherapy and Targeting the Tumor Microenvironment: Current Place and New Insights in Primary Pulmonary NUT Carcinoma
Xiang Li, Hui Shi, Wei Zhang, Chong Bai, Miaoxia He, Na Ta, Haidong Huang, Yunye Ning, Chen Fang, Hao Qin, Yuchao Dong
Frontiers in Oncology.2021;[Epub] CrossRef
Prevalence of NUT carcinoma in head and neck: Analysis of 362 cases with literature review
Taebum Lee, Junhun Cho, Chung‐Hwan Baek, Young‐Ik Son, Han‐Sin Jeong, Man Ki Chung, Sang Duk Hong, Yong Chan Ahn, Dong Ryul Oh, Jae Myoung Noh, Keunchil Park, Myung‐Ju Ahn, Hyung‐Jin Kim, Yi Kyung Kim, Young Hyeh Ko
Head & Neck.2020; 42(5): 924. CrossRef
Lung nuclear protein in testis carcinoma in an elderly Korean woman: A case report with cytohistological analysis
Hwa Jin Cho, Hyun‐Kyung Lee
Thoracic Cancer.2020; 11(6): 1724. CrossRef
Clinicopathological characteristics of primary lung nuclear protein in testis carcinoma: A single‐institute experience of 10 cases
Yoon Ah Cho, Yoon‐La Choi, Inwoo Hwang, Kyungjong Lee, Jong Ho Cho, Joungho Han
Thoracic Cancer.2020; 11(11): 3205. CrossRef

Original Article

Pulmonary Nodular Lymphoid Hyperplasia with Mass-Formation: Clinicopathologic Characteristics of Nine Cases and Review of the Literature: Jongmin Sim, Hyun Hee Koh, Sangjoon Choi, Jinah Chu, Tae Sung Kim, Hojoong Kim, Joungho Han; J Pathol Transl Med. 2018;52(4):211-218. Published online June 15, 2018; DOI: https://doi.org/10.4132/jptm.2018.04.27

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Abstract PDF

Background
Pulmonary nodular lymphoid hyperplasia (PNLH) is a non-neoplastic pulmonary lymphoid disorder that can be mistaken for malignancy on radiography. Herein, we present nine cases of PNLH, emphasizing clinicoradiological findings and histological features.
Methods
We analyzed radiological and clinicopathological features from the electronic medical records of nine patients (eight females and one male) diagnosed with PNLH. IgG and IgG4 immunohistochemical staining was performed in three patients.
Results
Two of the nine patients had experienced tuberculosis 40 and 30 years prior, respectively. Interestingly, none were current smokers, although two were ex-smokers. Three patients complaining of persistent cough underwent computed tomography of the chest. PNLH was incidentally discovered in five patients during examination for other reasons. The remaining patient was diagnosed with the disease following treatment for pneumonia. Imaging studies revealed consolidation or a mass-like lesion in eight patients. First impressions included invasive adenocarcinoma and mucosal-associated lymphoid tissue‒type lymphoma. Aspergillosis was suspected in the remaining patient based on radiological images. Resection was performed in all patients. Microscopically, the lesions consisted of nodular proliferation of reactive germinal centers accompanied by infiltration of neutrophils and macrophages in various degrees and surrounding fibrosis. Ultimately, all nine patients were diagnosed with PNLH and showed no evidence of recurrence on follow-up.
Conclusions
PNLH is an uncommon but distinct entity with a benign nature, and understanding the radiological and clinicopathological characteristics of PNLH is important.

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Clinical and Imaging Features of Pulmonary Nodular Lymphoid Hyperplasia
Dong-Lei Nie, Yan-Hong Shi, Xin-Min Li, Xiao-Jiang Wang, Bao-Li Han, Guo-Fu Zhang
Journal of Thoracic Imaging.2025;[Epub] CrossRef
Utilizing Immunoglobulin G4 Immunohistochemistry for Risk Stratification in Patients with Papillary Thyroid Carcinoma Associated with Hashimoto Thyroiditis
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Endocrinology and Metabolism.2024; 39(3): 468. CrossRef
Pulmonary Nodular Lymphoid Hyperplasia Evaluated with Bronchoalveolar Lavage Fluid Findings: A Case Report and Review of the Literature on Japanese Patients
Sakiko Moriyama, Takashi Kido, Noriho Sakamoto, Mai Fuchigami, Takatomo Tokito, Daisuke Okuno, Takuto Miyamura, Shota Nakashima, Atsuko Hara, Hiroshi Ishimoto, Yoshitaka Imaizumi, Kazuto Tsuruda, Katsunori Yanagihara, Junya Fukuoka, Hiroshi Mukae
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Jonathan Teow Koon Goh, Issam Al Jajeh, Jessica Han Ying Tan
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Pulmonary nodular lymphoid hyperplasia presenting as multifocal subsolid nodules: A case report and literature review
Yoon Jin Cha, Duk Hwan Moon, Ji Hyun Park, Sungsoo Lee, Ji Ae Choi, Tae Hoon Kim, Chul Hwan Park
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The diagnostic challenge of adenocarcinoma in pulmonary nodular lymphoid hyperplasia
Anita Savić Vuković, Melita Kukuljan, Morana Dinter, Ksenija Jurinović, Nives Jonjić
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