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Lymphangioleiomyomatosis observed in the dissected lymph node and myometrium in a patient that underwent radical operation for endometrial cancer

  • H. Morita1,2,*,
  • Y. Miyahara2
  • Y. Ueno3
  • K. Otani4
  • F. Kawakami4
  • Y. Ebina2
  • H. Yamada2

1Department of Community Medicine and Medical Network, Kobe University Graduate School of Medicine, Kusunokicho Chuo-ku Kobe Hyogo, Japan

2Department of Obstetrics and Gynecology, Kobe University Graduate School of Medicine, Kusunokicho Chuo-ku Kobe Hyogo, Japan

3Department of Pathology, Kobe University Graduate School of Medicine, Kusunokicho Chuo-ku Kobe Hyogo, Japan

4Department of Radiology, Kobe University Graduate School of Medicine, Kusunokicho Chuo-ku Kobe Hyogo, Japan

DOI: 10.12892/ejgo3709.2017 Vol.38,Issue 5,October 2017 pp.815-817

Published: 10 October 2017

*Corresponding Author(s): H. Morita E-mail: hmorita@med.kobe-u.ac.jp

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Abstract

Lymphangioleiomyomatosis (LAM) is a rare disease that afflicts mostly young women. It is characterized pathologically by the appearance of interstitial collections of atypical smooth muscle cells and cyst formation in the lungs, lymph node, kidney, and so on. A case was presented that underwent radical operation with the diagnosis of endometrial cancer, in which were found LAM cells in dissected lymph node and myometrium. Case: A 40-year-old woman underwent radical operation with the diagnosis of endometrial cancer. Although the dissected pelvic and para-aortic lymph nodes did not show the metastases of cancer, immunohistochemical study revealed the LAM cells among the dissected lymph nodes. Further histological study showed the LAM cells were in the uterine myometrium as well. Physical examination excluded the diagnosis of tuberous sclerosis. Since pulmonary LAM were observed as well, the physician followed the patient. Discussion: Sporadic LAM, without a complication of tuberous sclerosis, rarely shows LAM cells systemically as in this case. Conclusions: When LAM is observed coincidentally in operated dissected lymph node, it is important to examine for tuberous sclerosis and also to follow up the findings in the lung.

Keywords

Lymphangioleiomyomatosis; Endometrial cancer; Lymph node; Myometrium; Tuberous sclerosis.

Cite and Share

H. Morita, Y. Miyahara, Y. Ueno, K. Otani, F. Kawakami, Y. Ebina, H. Yamada. Lymphangioleiomyomatosis observed in the dissected lymph node and myometrium in a patient that underwent radical operation for endometrial cancer. European Journal of Gynaecological Oncology. 2017; 38(5): 815-817. doi: 10.12892/ejgo3709.2017

References

[1] “NHLBI Workshop Summary. Report of workshop on lymphangioleiomyomatosis. National Heart, Lung, and Blood Institute”. Am. J. Respir. Crit. Care Med., 1999, 159, 679.

[2] Johnson S.: “Rare diseases. 1. Lymphangioleiomyomatosis: clinical features, management and basic mechanisms”. Thorax, 1999, 54, 254.

[3] Hornick J.L., Pan C-C.: “PEComa”. In: Fletcher C.D.M., Brodge J.A., Hogendoorn P.C.W., Mertens F. (eds). World Health Organization classification of tumours of soft tissue and bone”. 4thed. Lyon: IARC, 2013, 230.

[4] Kalassian K.G., Doyle R., Kao P., Ruoss S., Raffin T.A.: “Lymphangioleiomyomatosis: new insights”. Am. J. Respir. Crit. Care Med., 1997, 155, 1183.

[5] Matsui K., Takeda K., Yu Z.X., Travis W.D., Moss J., Ferrans V.J.: “Role for activation of matrix metalloproteinases in the pathogenesis of pulmonary lymphangioleiomyomatosis”. Arch. Pathol. Lab. Med., 2000, 124, 267.

[6] Lee K.M., Wong C., Russell P.: ”Pelvic lymphangioleiomyomatosis associated with endosalpingiosis”. Pathology, 2009, 41, 500.

[7] Iwasa Y., Tachibana M., Ito H., Iwami S., Yagi H., Yamada S., et al.: “Extrapulmonary lymphangioleiomyomatosis in pelvic and paraaortic lymph nodes associated with uterine cancer: a report of 3 cases”. Int. J. Gynecol. Pathol., 2011, 30, 470.

[8] Rabban J.T., Firetag B., Sangoi A.R., Post M.D., Zaloudek C.J.: “Incidental pelvic and para-aortic lymph node lymphangioleiomyomatosis detected during surgical staging of pelvic cancer in women without symptomatic pulmonary lymphangio -leiomyomatosis or tuberous sclerosis complex”. Am. J. Surg. Pathol., 2015, 39, 1015.

[9] Crausman R.S., Jennings C.A., Mortenson R.L., Ackerson L.M., Irvin C.G., King T.E. Jr.: “Lymphangioleiomyomatosis: the pathophysiology of diminished exercise capacity”. Am. J. Respir. Crit. Care Med., 1996, 153, 1368.

[10] Berger U., Khaghani A., Pomerance A., Yacoub M.H., Coombes R.C.: “Pulmonary lymphangioleiomyomatosis and steroid receptors. An immunocytochemical study”. Am. J. Clin. Pathol., 1990, 93, 609.

[11] Ohori N.P., Yousem S.A., Sonmez-Alpan E., Colby T.V.: “Estrogen and progesterone receptors in lymphangioleiomyomatosis, epithelioid hemangioendothelioma, and sclerosing hemangioma of the lung”. Am. J. Clin. Pathol., 1991, 96, 529.

[12] Matsui K., Takeda K., Yu Z.X., Valencia J., Travis W.D., Moss J., et al.: “Downregulation of estrogen and progesterone receptors in the abnormal smooth muscle cells in pulmonary lymphangioleiomyomatosis following therapy. An immunohistochemical study”. Am. J. Respir. Crit. Care Med., 2000, 161, 1002.

[13] McCormack F.X., Inoue Y., Moss J., Singer L.G., Strange C., Nakata K., et al.: “Efficacy and safety of sirolimus in lymphangioleiomyomatosis”. N. Engl. J. Med., 2011, 364, 1595.

[14] Pechet T.T., Meyers B.F., Guthrie T.J., Battafarano R.J., Trulock E.P., Cooper J.D., Patterson G.A.: “Lung transplantation for lymphangioleiomyomatosis”. J. Heart Lung Transplant., 2004, 23, 301.

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