Journal of Pathology and Translational Medicine

Original Article

Extrapulmonary Lymphangioleiomyoma: Clinicopathological Analysis of 4 Cases: Dae Hyun Song, In Ho Choi, Sang Yun Ha, Kang Min Han, Jae Jun Lee, Min Eui Hong, Yoon-La Choi, Kee-Taek Jang, Sang Yong Song, Chin A Yi, Joungho Han; Korean J Pathol. 2014;48(3):188-192. Published online June 26, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.3.188

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Background

Lymphangioleiomyomatosis (LAM) is a slowly progressive neoplastic disease that predominantly affects females. Usually, LAM affects the lung; it can also affect extrapulmonary sites, such as the mediastinum, the retroperitoneum, or the lymph nodes, although these locations are rare. A localized form of LAM can manifest as extrapulmonary lesions; this form is referred to as extrapulmonary lymphangioleiomyoma (E-LAM). Due to the rare occurrence of E-LAM and its variable, atypical location, E-LAM is often difficult to diagnose. Herein, we report the clinicopathological information from four E-LAM cases, and also review previous articles investigating this disease.

Methods

Four patients with E-LAM were identified at the Samsung Medical Center (Seoul, Korea) from 1995 to 2012. All E-LAM lesions underwent surgical excision.

Results

All patients were females within the age range of 43 to 47 years. Two patients had para-aortic retroperitoneal masses, while the other two patients had pelvic lesions; two out of the four patients also had accompanying pulmonary LAM. In addition, no patient displayed any evidence of tuberous sclerosis. Histologically, two patients exhibited nuclear atypism with cytologic degeneration.

Conclusions

E-LAM should be considered in the differential diagnosis of patients presenting with pelvic or para-aortic masses. We also conclude that further clinical and pathological evaluation is needed in patients with E-LAM and nuclear atypism.

Citations

Citations to this article as recorded by

Lymphangioleiomyomatosis with Tuberous Sclerosis Complex—A Case Study
Aleksandra Marciniak, Jolanta Nawrocka-Rutkowska, Agnieszka Brodowska, Andrzej Starczewski, Iwona Szydłowska
Journal of Personalized Medicine.2023; 13(11): 1598. CrossRef
A case of lymphangioleiomyomatosis with endometrial cancer diagnosed by retroperitoneoscopic para-aortic lymph node dissection
Aiko Ogasawara, Shogo Yamaguchi, Hiroaki Inui, Mieko Hanaoka, Daisuke Shintani, Sho Sato, Masanori Yasuda, Akira Yabuno
JAPANESE JOURNAL OF GYNECOLOGIC AND OBSTETRIC ENDOSCOPY.2022; 38(1): 158. CrossRef
Primary retroperitoneal PEComa: an incidental finding
Bárbara Monteiro Marinho, António Gâmboa Canha, Donzília Sousa Silva, José Davide Pinto Silva
BMJ Case Reports.2022; 15(11): e250466. CrossRef
Imaging Findings of Thoracic Lymphatic Abnormalities
Jingshuo (Derek) Sun, Thomas Shum, Fardad Behzadi, Mark M. Hammer
RadioGraphics.2022; 42(5): 1265. CrossRef
Extrapulmonary uterine lymphangioleiomyomatosis (LAM) and dysfunctional uterine bleeding: the first presentation of LAM in a tuberous sclerosis complex patient
Lucy Grant, Saliya Chipwete, San Soo Hoo, Anjali Bhatnagar
BMJ Case Reports.2019; 12(2): e226358. CrossRef
Summary of the Japanese Respiratory Society statement for the treatment of lung cancer with comorbid interstitial pneumonia
Takashi Ogura, Nagio Takigawa, Keisuke Tomii, Kazuma Kishi, Yoshikazu Inoue, Eiki Ichihara, Sakae Homma, Kazuhisa Takahashi, Hiroaki Akamatsu, Satoshi Ikeda, Naohiko Inase, Tae Iwasawa, Yuichiro Ohe, Hiromitsu Ohta, Hiroshi Onishi, Isamu Okamoto, Kazumasa
Respiratory Investigation.2019; 57(6): 512. CrossRef
Incidental lymphangioleiomyomatosis in the lymph nodes of gynecologic surgical specimens
Ikumi Kuno, Hiroshi Yoshida, Hanako Shimizu, Takashi Uehara, Masaya Uno, Mitsuya Ishikawa, Tomoyasu Kato
European Journal of Obstetrics & Gynecology and Reproductive Biology.2018; 231: 93. CrossRef
Solitary extrapulmonary lymphangioleiomyomatosis of the liver: A case report and literature review
Weiwei Fu, Yujun Li, Hong Li, Ping Yang, Xiaoming Xing
Experimental and Therapeutic Medicine.2016; 12(3): 1499. CrossRef
Incidental Pelvic and Para-aortic Lymph Node Lymphangioleiomyomatosis Detected During Surgical Staging of Pelvic Cancer in Women Without Symptomatic Pulmonary Lymphangioleiomyomatosis or Tuberous Sclerosis Complex
Joseph T. Rabban, Brandie Firetag, Ankur R. Sangoi, Miriam D. Post, Charles J. Zaloudek
American Journal of Surgical Pathology.2015; 39(8): 1015. CrossRef

Case Reports

Adnexal Clear Cell Carcinoma with Comedonecrosis: A Case Report.: Seo Hee Kim, Sun Hee Han, Jung Suk An, Ju Han Lee, Eung Seok Lee, Heum Rye Park, Young Sik Kim; Korean J Pathol. 2010;44(1):92-96.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.1.92

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Abstract PDF

Adnexal clear cell carcinoma with comedonecrosis (ACCCC) is a very rare malignancy of the skin with an aggressive clinical course and a predilection for the scalp. This is the first reported case of ACCCC in Korea. A 79-year-old male presented with left abdominal masses that proved to be two subcutaneous nodules. The tumors histologically consisted of epithelial nests that showed a distinctive zonal arrangement. The centrally located clear cell areas with comedonecroses were merged with the peripheral squamoid cells, often exhibiting retraction artifacts and an infiltrating border. Nuclear pleomorphism and frequent mitoses were prominent. The clear cells were immunopositive for carcinoembryonic antigen and epithelial membrane antigen. We report here on a case of ACCCC involving the abdominal skin, and this tumor should be distinguished from the more indolent squamous cell and tricholemmal carcinomas.

Citations

Citations to this article as recorded by

Adnexal Clear Cell Carcinoma Exhibiting Comedonecrosis of the Ear: A Rare Case Treated With Mohs Micrographic Surgery
Helen Z Chen, Mohamad Jabin, Michelle Tarbox, Russell Akin, Ashley Sturgeon
Cureus.2024;[Epub] CrossRef

Malignant Eccrine Poroma of Abdomen Brief case report.: Jin Ja Park, Young Hee Choi, Kyung Chan Choi, Young Euy Park; Korean J Pathol. 1998;32(4):312-314.

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18 Download

Abstract PDF: Eccrine porocarcinoma is a rare tumor of the skin. A case report of an eccrine porocarcinoma metastasizing to epidural space of spinal cord and inguinal area with a nine year follow up is described. The patient had a nodular growth of the abdomen with both inguinal lymphadenopathy three years before its first excision. After a follow up of nine years, he complained a weakness of lower extremities and back pain. Extradural mass of 10th thoracic vertebra and left inguinal mass were found. Subsequently, the masses histologically identical to the skin tumor were found.

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