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Case Study
A Rare Case of Angioleiomyoma Arising in the Subglottic Area to Upper Trachea of a Patient with Underlying Asthma
Yeoun Eun Sung, Chin Kook Rhee, Kyo Young Lee
J Pathol Transl Med. 2017;51(1):92-95.   Published online August 22, 2016
DOI: https://doi.org/10.4132/jptm.2016.06.21
  • 7,572 View
  • 106 Download
  • 4 Web of Science
  • 3 Crossref
AbstractAbstract PDF
Angioleiomyoma is a rare disease that is histologically characterized by smooth muscle cells arranged around vascular spaces. Although angioleiomyomas occur rarely in the head and neck region, they can cause various symptoms according the site involved. Here, we present a 44-yearold male patient with a 15-year history of asthma, who presented with recent onset of chest discomfort, globus sensation and throat pain. Medication was not effective in relieving his symptoms, and further evaluation revealed a polypoid ovoid mass, almost obstructing the airway at the border of the larynx and upper trachea on chest computed tomography. The mass was completely resected via a rigid bronchoscopy procedure. Histopathologic examination revealed that the excised mass was angioleiomyoma, which was immunohistochemically positive for smooth muscle actin and negative for desmin.

Citations

Citations to this article as recorded by  
  • Angioleiomyoma of the Epiglottis Mimicking Epiglottic Hemangioma: Clinical Experience and Literature Review
    Yang-Yang Bao, Xiao-Jie Shi, Li-Bo Dai, Yu Guo, Hong-Tian Yao, Shui-Hong Zhou
    Ear, Nose & Throat Journal.2022; : 014556132211000.     CrossRef
  • Angioleiomyoma of the Larynx: A Case Report and Literature Review
    Federica Perardi, Giuseppe Abbate, Leonardo R. Iannuzzelli, Rossella Contini, Manuela De Munari, Francesco G. Sciuto, Monica Leutner, Antonio Scotti
    Ear, Nose & Throat Journal.2020; 99(10): 658.     CrossRef
  • Flexible bronchoscopy and cryoextraction for critical airway obstruction caused by an endobronchial angioleiomyoma
    Sumit Chatterji, Efrat Ofek, Tiberiu Shulimzon
    Respirology Case Reports.2019;[Epub]     CrossRef
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
  • 7,687 View
  • 56 Download
  • 9 Crossref
AbstractAbstract PDF
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 Report
Angioleiomyoma of the Nasal Cavity: A Case Report.
Su Jin Kim, Sook Hee Hong, Mee Sook Roh
Korean J Pathol. 2004;38(3):181-183.
  • 1,564 View
  • 14 Download
AbstractAbstract PDF
Angioleiomyoma of the sinonasal area is an extremely rare benign neoplasm. To the best of our knowledge, only 26 cases have been described. Here, we report a case of angioleiomyoma arising in the nasal cavity of a 60-year-old woman. Microscopically, the tumor consisted of proliferating smooth muscle cells punctuated with thick-walled vessels with slit-like lumina. The tumor was negative for estrogen and progesterone receptor by immunohistochemical study. Further studies are needed to clarify whether the growth of this tumor is sex steroid-dependent.
Original Article
Pulmonary Lymphangioleiomyomatosis: Pathologic Analysis of Eight Korean Cases.
Seung Sook Lee, Jeong Wook Seo, Eul Keun Ham, Yong Il Kim, Nam Hee Won, Jung Gi Im, Young Soo Shim
Korean J Pathol. 1994;28(4):358-367.
  • 1,418 View
  • 11 Download
AbstractAbstract PDF
Histopathology of pulmonary lymphangioleiomyomatosis(LAM) is studied using four new cases and six previously reported cases, which include two cases without definite evidence of LAM. The important diagnostic features of this lesion were nodular proliferation of immature smooth muscle and cleft or cyst formation within the nodules of smooth muscle cells. The nuclei of the smooth muscle cells were bigger than those of blood vessels or fibrotic lung, and the direction of nuclei was irregular. The lung parenchyma showed little inflammatory change but there were multiple air cysts with smooth muscle nodules at their margin. There were two cases with exuberant proliferation of smooth muscle nodules and two cases with papilliferous projections of the cells into lymphatic lumen. Whereas, three cases had only a few small slender nodules of smooth muscle cells at the margin of air cyst. The lymphatic lumen with smooth muscle nodules is dilated in four cases but other four cases show collapsed lumen. Pulmonary hemorrhage and hemosiderosis were prominent in three cases. There were variety of histology in terms of the cellularity of smooth muscle nodules, the size of the lymphatic lumen and the degree of pulmonary destruction, which may have significance on the clinical presentation and prognostication.
Case Report
Pulmonary Lymphangioleiomyomatosis: A case report.
Won Bo Jo, Nam Hee Won, Seung Yong Paik, Hae Kyung Ahn
Korean J Pathol. 1991;25(3):269-274.
  • 1,761 View
  • 18 Download
AbstractAbstract PDF
Lymphangioleiomyomatosis(LAM) is a rare disease of women of child-bearing age in which there is progressive hyperplasia of atypical smooth mucle along lymphatics in the lung, and/or axial lymphatics in the thorax and abdomen, resulting in honeycombing of lung. Interestingly there has been a speculation that it represents a forme furste or incomplete expression of tuberous sclerosis complex. This is based on the observation that patients with tuberous sclerosis can manifest pulmonary lesions indistinguishable from LAM. We report a case of LAM occuring in a 39-year-old female, who complained of recurrent pneumothorax, chest pain and shortness of breath. Three years ago, the patient had right nephrectomy under the diagnosis of ruptured angiomyolipoma. A X-ray film of the chest showed honeycombing with a diffusely reticulonodular pattern and cyst-like spaces. She had a characteristic facial appearance of adenoma sebaceum, which her father and uncle had. Microscopically, the lung showed a marked smooth muscle proliferation around the slit-like lymphatic spaces and also some respiratory bronchioles.

J Pathol Transl Med : Journal of Pathology and Translational Medicine