Journal of Pathology and Translational Medicine

Original Articles

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

Cholesteatoma of the Renal Pelvis: A case report.: Nam Hoon Kim, Young Chun Moon, Moon Hyang Park; Korean J Pathol. 1995;29(5):691-693.

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Abstract: Cholesteatoma in the kidney and ureter are accumulations of waxy, gray flakes of keratin materials, secondary to squamous metaplasia of the transitional epithelium. Herein, we describe a case of cholesteatoma in the renal pelvis of a 69-year-old woman, and give a brief review of the literature. In the upper pole of the left kidney was a 6 cm cystic lesion filled with a thick, flaky, grayish, comified material. Microscopically, the cystic area showed calyceal and pelvic structures being replaced by keratinizing stratified squamous epithelium. The surrounding renal parenchyma was atrophic with features of chronic pyelonephritis.

Case Reports

Sarcomatoid Transitional Cell Carcinoma of the Renal Pelvis A report of two cases.: Kyo Young Lee, Mi seon Kwon, Yeong Jin Choi, Chang Suk Kang, Seok Jin Kang, Baying Kee Kim, Sang In Shim; Korean J Pathol. 1999;33(2):128-132.

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Abstract: Sarcomatoid carcinomas are malignant epithelial neoplasms in which the tumor cells assume a partial or complete spindle cell pattern of growth, leading to the erroneous classification of some true carcinomas as sarcomas. These spindle cells are malignant and manifest various amount of both vimentin and cytokeratin. Positive reaction of some of the spindle cells for cytokeratin antibodies is confirmatory. Clinical features do not differ significantly from those of patients with high-grade transitional cell carcinoma. So far, 13 cases of sarcomatoid transitional cell carcinoma of the renal pelvis have been reported in English and Korean literature. In this report, we describe clinicopathologic features of recently observed two cases of sarcomatoid transitional cell carcinoma of the renal pelvis and summarize the pathologic findings of previously reported cases with review of the literature.

Ependymoma Arising from the Pelvis: A Case Report.: Kyungji Lee, Jihan Jung, Youn Soo Lee, Kyo Young Lee, Byung Kee Kim, Chang Suk Kang; Korean J Pathol. 2005;39(2):120-124.

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Abstract PDF: We report here on a case of an ependymoma arising from the pelvis in a 25-year-old woman. She had no evidence of abnormality in her brain and bilateral ovaries. The diagnosis was based on light microscopic, immunohistochemical, and ultrastructural features of a typical ependymoma, including the patterns of pseudorosette or true ependymal rosette, the strong immunopositivity for glial fibrillary acid protein and intermediate filaments, and cilia of tumor cells. The mass was over 20 cm in maximum diameter, and it was located between the uterus and rectum without any connection to bilateral ovaries. There were many metastatic nodules in the pelvis and omentum. In addition, the proliferation index in the most active area was 10% by immunohistochemistry using monoclonal antibody MIB-1. Although the prognosis of the pelvic ependymoma is known to be difficult to evaluate, this case may serve to illustrate the poor prognostic course, according to the size of the tumor, the evidence of metastasis, and the MIB-1 labelling index.

Epidermal Cyst in the Renal Pelvis: A Case Report with Review of the Literature.: Sun Zoo Kim, Tae In Park, Sang Han Lee, Jung Sik Kwak; Korean J Pathol. 2004;38(4):270-272.

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Abstract PDF: We report here on a case of an epidermal cyst arising in the kidney. This cyst occurred in a 61-year-old woman with a past history of several attacks of ureteral stones and she received treatments of extracoporeal shock wave lithotripsy and open nephrolithostomy. On the intravenous pyelogram, a relatively well demarcated, 5x5 cm-sized lesion with calcification was detected in the renal pelvis and calices. The lesion was removed by percutaneous nephrolithotomy. Histologically, the lesion had the same morphologic feature as a typical epidermal cyst arising in the skin. It has been postulated that the intrarenal epidermal cyst arises either from epidermal remnants or it results from traumatic implantation of transformed epithelium. Considering the past history of the patient, it might well be suspected that the present case occurred as a result of traumatic implantation of metaplastic squamous epithelial cells. We report here on a very interesting case of an epidermal cyst in the renal pelvis with a review of the relevant literatures.

Localized Amyloidosis of the Ureter: A Report of Two Cases.: Ho Jung Lee, Dong Eun Song, Jong Eun Joo, Won Mi Lee, Dong Hoon Kim, Eun Kyung Kim, Jae Y Ro; Korean J Pathol. 2004;38(3):184-187.

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Abstract PDF: We report on two cases of localized amyloidosis involving the ureter. The patients were a 64-year-old woman with right upper quadrant pain (case 1) and a 36-year-old woman suffering from left flank pain and intermittent gross hematuria (case 2). An intravenous pyelography of case 1 revealed multiple filling defects in the entire right ureter, whereas retrograde pyelography in case 2 showed diffuse narrowing in the mid and lower portions of the left ureter. Localized amyloidosis of the ureter was diagnosed in the two cases, and both had amyloid deposit in the renal pelvis and the urinary bladder in case 1, and in the contralateral ureter and the renal pelvis in case 2. Right nephroureterectomy was performed in case 1, but a segmental resection of the ureter with preservation of the kidney was administered in case 2. These two cases demonstrate that ureteral amyloidosis can be associated with amyloid deposition in the renal pelvis and the urinary bladder. Although ureteral amyloidosis is a rare occurrence, it should be considered in the differential diagnosis of ureteral obstruction to avoid unnecessary radical surgery.

Parapelvic Renal Cyst (Pericalyceal Lymphangiectasis): A case report.: Weon Seo Park, Je Geun Chi; Korean J Pathol. 1994;28(2):210-212.

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Abstract PDF: Parapelvic renal cyst, also designated as pericalyceal lymphangiectasis, is an unusual lesion that is usually brought to light during surgery for ureteropelvic junction obstruction or recurrent pyelonephritis. Grossly, the renal pelvis is enveloped by a multilocular cystic mass filled with clear fluid. This lesion is confined to the peripelvic tissues and does not extend into the parenchyma, which, however, may show the effects of hydronephrosis or pyelonephritis. A 50-year-old man presented with hydronephrosis. An ultrasonography revealed hydronephro-sis of the left kidney. Intravenous pyelography and DMSA ("Tc-Dimercaptosuccinic acid) scan showed nonfunctioning kidney of the same side. Simple left nephrectomy was done. The renal pelvis was mildly dilated and a cyst was found buldging into the renal pelvis. The content was watery clear and the cyst was not connected to the renal pelvis or calyces. The cyst was round unilocular and lined by attenuated single layer of endothelial cells. The endothelial cells showed no reactivity to factor-VIII related antigen. With these findings, we concluded that this cystic lesion is basically lymphatic cyst and hydronephrosis was caused by the compression of pelvic out-flow of the kidney.

Original Article

Pseudosarcomatous Variant of Transitional Cell Carcinoma of the Renal Pelvis.: Yun Kyung Kang, Ta Jin Kim, Yong Il Kim, Si Whang Kim; Korean J Pathol. 1992;26(6):610-614.

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Abstract PDF: We report a case of pseudosarcomatous variant of transitional cell carcinoma of the renal pelvis which showed grossly and microscopically the distinct biphasic growth patterns. Grossly, most part of the tumor showed solid growth protruding into the renal pelvic cavity as well as infiltrating into the parenchyma of lower pole. The overlying pelvic mucosa was replaced by a diffuse, papillary transitional cell carcinoma, and the solid mass was composed of pleomorphic spindle cell sarcomatoid component with frequent myxoid change and a few foci of osteoid deposit. Ultrastructural study of the spindle cells revealed epithelial differentiation featured with rich cytoplasmic organelles, basal lamina and basement membrane-like structures, although immunohistochemistry failed to detect epithelial differentiation.

Case Report

Keratinizing Desquamative Squamous Metaplasia of the Upper Urinary Tract: A case report.: Soo Min Kang, Sun Young Kim; Korean J Pathol. 1991;25(1):76-80.

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Abstract PDF: Keratinizing desquamative squamous metaplasia of the transitional epithelium occurs rarely in the upper urinary tract, especially in the ureter. It is associated in most cases with long-standing chronic inflammation but the exact pathogenesis is unknown, and the relationship to cancer has continued to be controversial. More cases should be accumulated for a better assessmnt of this lesion. Recently, we experienced a case of keratinizing desquamative squamous metaplasia in the renal pelvis, adjacent calyces and ureter following acute and chronic pyelonephritis. Herein, we are presenting our case with a review of literatures.

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