Cystic and solitary nodular pulmonary metastases in a patient with low‑grade endometrial stromal sarcoma: A case report and literature review

Xu,Yang; Liang,Zhi‑Xin; Guo,Jun‑Tang; Su,Xin; Lu,Yun‑Long; Guan,Xi‑Zhou

doi:10.3892/ol.2019.10409

Cystic and solitary nodular pulmonary metastases in a patient with low‑grade endometrial stromal sarcoma: A case report and literature review

Authors:
- Yang Xu
- Zhi‑Xin Liang
- Jun‑Tang Guo
- Xin Su
- Yun‑Long Lu
- Xi‑Zhou Guan
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Affiliations: Department of Respiratory Diseases, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China, Department of Thoracic Surgery, Chinese People's Liberation Army General Hospital, Beijing 100853, P.R. China, Department of Respiratory Diseases, Hainan Branch of Chinese People's Liberation Army General Hospital, Sanya, Hainan 572013, P.R. China, Department of Pathology, Hainan Branch of Chinese People's Liberation Army General Hospital, Sanya, Hainan 572013, P.R. China
Published online on: May 30, 2019 https://doi.org/10.3892/ol.2019.10409
Pages: 1133-1144
Copyright: © Xu et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Pulmonary metastases of endometrial stromal sarcoma (ESS) are uncommon and can be difficult to diagnose. The aims of the present study were to investigate the clinical and pathological features, and enhance the awareness of pulmonary metastases in patients with low‑grade ESS. The study reports a case of low‑grade ESS that resulted in cystic and nodular pulmonary metastases. Furthermore, the PubMed database was searched using ‘pulmonary metastases of low‑grade endometrial stromal sarcoma’ as the key phrase. The literature on pulmonary metastases of low‑grade ESS was reviewed and 35 cases were included in the present study. The clinical manifestations, imaging data, pathological features, treatment and prognosis of the 35 previously reported cases and the current case were retrospectively analyzed. The age range of the 36 patients diagnosed with low‑grade ESS was 28‑65 years. The time period from confirmation of ESS to lung metastases was 1.5‑27 years. In 50% of the patients, the pulmonary metastases were asymptomatic. The most common pulmonary symptom was dyspnea, followed by chest pain, pneumothorax and coughing. The most common chest imaging presentation was multiple pulmonary nodules, followed by a solitary nodule or mass. Histology was used to identify that the pulmonary metastases had the pathological features of low‑grade ESS. The immunohistochemical results demonstrated strong diffuse immunoreactivity for cluster of differentiation 10, estrogen receptor and progesterone receptor in almost all the specimens. The review of the literature revealed that pulmonary metastases from low‑grade ESS are rare but not negligible. Furthermore, the detailed clinical information, imaging findings and immunohistochemical detection are important for making a diagnosis.

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1	Ali RH and Rouzbahman M: Endometrial stromal tumours revisited: An update based on the 2014 WHO classification. J Clin Pathol. 68:325–332. 2015. View Article : Google Scholar : PubMed/NCBI
2	Prat J and Mbatani: Uterine sarcomas. Int J Gynaecol Obstet. 131 (Suppl 2):S105–S110. 2015. View Article : Google Scholar : PubMed/NCBI
3	Xue WC and Cheung AN: Endometrial stromal sarcoma of uterus. Best Pract Res Clin Obstet Gynaecol. 25:719–732. 2011. View Article : Google Scholar : PubMed/NCBI
4	Oliva E, Clement PB and Young RH: Endometrial stromal tumors: An update on a group of tumors with a protean phenotype. Adv Anat Pathol. 7:257–281. 2000. View Article : Google Scholar : PubMed/NCBI
5	Cohen I: Endometrial pathologies associated with postmenopausal tamoxifen treatment. Gynecol Oncol. 94:256–266. 2004. View Article : Google Scholar : PubMed/NCBI
6	Ashraf-Ganjoei T, Behtash N, Shariat M and Mosavi A: Low grade endometrial stromal sarcoma of uterine corpus, a clinico-pathological and survey study in 14 cases. World J Surg Oncol. 4:502006. View Article : Google Scholar : PubMed/NCBI
7	Pekindil G, Tuncyurek O, Orguc S, Inceboz U, Kandiloglu AR and Caglar H: A case of endometrial stromal sarcoma with curvilinear calcification. Gynecol Oncol. 98:318–321. 2005. View Article : Google Scholar : PubMed/NCBI
8	Aubry MC, Myers JL, Colby TV, Leslie KO and Tazelaar HD: Endometrial stromal sarcoma metastatic to the lung: A detailed analysis of 16 patients. Am J Surg Pathol. 26:440–449. 2002. View Article : Google Scholar : PubMed/NCBI
9	Itoh T, Mochizuki M, Kumazaki S, Ishihara T and Fukayama M: Cystic pulmonary metastases of endometrial stromal sarcoma of the uterus, mimicking lymphangiomyomatosis: A case report with immunohistochemistry of HMB45. Pathol Int. 47:725–729. 1997. View Article : Google Scholar : PubMed/NCBI
10	Kim GY, Sung CO, Han J, Park JO and Lee KS: Pulmonary metastases of uterine endometrial stromal sarcoma: Diffuse micronodular and ground glass opacities: A case report. J Korean Med Sci. 19:901–903. 2004. View Article : Google Scholar : PubMed/NCBI
11	Abrams J, Talcott J and Corson JM: Pulmonary metastases in patients with low-grade endometrial stromal sarcoma. Clinicopathologic findings with immunohistochemical characterization. Am J Surg Pathol. 13:133–140. 1989. View Article : Google Scholar : PubMed/NCBI
12	Inayama Y, Shoji A, Odagiri S, Hirahara F, Ito T, Kawano N and Nakatani Y: Detection of pulmonary metastasis of low-grade endometrial stromal sarcoma 25 years after hysterectomy. Pathol Res Pract. 196:129–134. 2000. View Article : Google Scholar : PubMed/NCBI
13	Qian ZD, Huang LL and Zhu XM: An immunohistochemical study of CD83- and CD1a-positive dendritic cells in the decidua of women with recurrent spontaneous abortion. Eur J Med Res. 20:22015. View Article : Google Scholar : PubMed/NCBI
14	Satoh Y, Ishikawa Y, Miyoshi T, Mukai H, Okumura S and Nakagawa K: Pulmonary metastases from a low-grade endometrial stromal sarcoma confirmed by chromosome aberration and fluorescence in-situ hybridization approaches: A case of recurrence 13 years after hysterectomy. Virchows Arch. 442:173–178. 2003.PubMed/NCBI
15	Murakami A, Hayashi T, Terao Y, Mori T, Kumasaka T, Seyama K and Takahashi K: Cystic, nodular and cavitary metastases to the lungs in a patient with endometrial stromal sarcoma of the uterus. Intern Med. 53:1001–1005. 2014. View Article : Google Scholar : PubMed/NCBI
16	Miyamoto H, Jones CE, Raymond DP, Wandtke JC, Strang JG, Bourne PA, Bonfiglio TA and Xu H: Pulmonary metastases from uterine neoplasms after long tumour-free interval: Four cases and review of the literature. Pathology. 41:234–241. 2009. View Article : Google Scholar : PubMed/NCBI
17	Lim MC, Lee S and Seo SS: Megestrol acetate therapy for advanced low-grade endometrial stromal sarcoma. Onkologie. 33:260–262. 2010. View Article : Google Scholar : PubMed/NCBI
18	Mahadeva R, Stewart S and Wallwork J: Metastatic endometrial stromal sarcoma masquerading as pulmonary lymphangioleiomyomatosis. J Clin Pathol. 52:147–148. 1999. View Article : Google Scholar : PubMed/NCBI
19	Akhavan A, Shishebor F, Moghimi M and Binesh F: Endometrial stromal sarcoma of uterus with metastasis to the lung and brain. BMJ Case Rep. 2012(pii): bcr20120069542012.PubMed/NCBI
20	Binesh F, Zahir ST, Akhavan A and Bovanlu TR: Endometrial stromal sarcoma of the uterus presenting as pulmonary metastasis. BMJ Case Rep. 2013(pii): bcr20130085652013.PubMed/NCBI
21	Kang DO, Choi SI, Oh JY, Sim JK, Choi JH, Choo JY, Hwang JW, Lee SH, Lee JH, Lee KY, et al: Endometrial stromal sarcoma presented as an incidental lung mass with multiple pulmonary nodules. Tuberc Respir Dis (Seoul). 76:131–135. 2014. View Article : Google Scholar : PubMed/NCBI
22	Ota S, Shinagawa K, Ueoka H, Tada S, Tabata M, Hamazaki S, Kondo E, Kiura K, Mannami T, Shibayama T, et al: Spontaneous regression of metastatic endometrial stromal sarcoma. Jpn J Clin Oncol. 32:71–74. 2002. View Article : Google Scholar : PubMed/NCBI
23	Steele SJ, Scott JM and Stephens TW: Endometrial stromal sarcoma. Report of a case with a pulmonary metastasis extending through the heart. Br J Surg. 55:943–945. 1968. View Article : Google Scholar : PubMed/NCBI
24	Nakamura K, Nakayama K, Ishikawa M, Ishikawa N, Katagiri H, Katagiri A, Ishibashi T, Sato E, Iida K, Sultana R and Kyo S: Letrozole as second-line hormonal treatment for recurrent low-grade endometrial stromal sarcoma: A case report and review of the literature. Oncol Lett. 12:3856–3860. 2016. View Article : Google Scholar : PubMed/NCBI
25	Giin Chong S, Mitchell P, Fabre A and McDonnell TJ: Recurrent pneumothoraces in a 65-year-old female: An unusual case of cystic lung disease. Eur Respir Rev. 23:271–272. 2014. View Article : Google Scholar : PubMed/NCBI
26	Spano JP, Soria JC, Kambouchner M, Piperno-Neuman S, Morin F, Morere JF, Martin A and Breau JL: Long-term survival of patients given hormonal therapy for metastatic endometrial stromal sarcoma. Med Oncol. 20:87–93. 2003. View Article : Google Scholar : PubMed/NCBI
27	Hwang H, Matsuo K, Duncan K, Pakzamir E, Pham HQ, Correa A, Fedenko A and Mhawech-Fauceglia P: Immunohistochemical panel to differentiate endometrial stromal sarcoma, uterine leiomyosarcoma and leiomyoma: Something old and something new. J Clin Pathol. 68:710–717. 2015. View Article : Google Scholar : PubMed/NCBI
28	Conklin CM and Longacre TA: Endometrial stromal tumors: The new WHO classification. Adv Anat Pathol. 21:383–393. 2014. View Article : Google Scholar : PubMed/NCBI
29	Micci F, Panagopoulos I, Bjerkehagen B and Heim S: Consistent rearrangement of chromosomal band 6p21 with generation of fusion genes JAZF1/PHF1 and EPC1/PHF1 in endometrial stromal sarcoma. Cancer Res. 66:107–112. 2006. View Article : Google Scholar : PubMed/NCBI
30	Micci F, Gorunova L, Gatius S, Matias-Guiu X, Davidson B, Heim S and Panagopoulos I: MEAF6/PHF1 is a recurrent gene fusion in endometrial stromal sarcoma. Cancer Lett. 347:75–78. 2014. View Article : Google Scholar : PubMed/NCBI
31	Panagopoulos I, Thorsen J, Gorunova L, Haugom L, Bjerkehagen B, Davidson B, Heim S and Micci F: Fusion of the ZC3H7B and BCOR genes in endometrial stromal sarcomas carrying an X;22-translocation. Genes Chromosomes Cancer. 52:610–618. 2013.PubMed/NCBI
32	Dewaele B, Przybyl J, Quattrone A, Finalet Ferreiro J, Vanspauwen V, Geerdens E, Gianfelici V, Kalender Z, Wozniak A, Moerman P, et al: Identification of a novel, recurrent MBTD1-CXorf67 fusion in low-grade endometrial stromal sarcoma. Int J Cancer. 134:1112–1122. 2014. View Article : Google Scholar : PubMed/NCBI
33	Amant F, Floquet A, Friedlander M, Kristensen G, Mahner S, Nam EJ, Powell MA, Ray-Coquard I, Siddiqui N, Sykes P, et al: Gynecologic cancer InterGroup (GCIG) consensus review for endometrial stromal sarcoma. Int J Gynecol Cancer. 24 (Suppl 3):S67–S72. 2014. View Article : Google Scholar : PubMed/NCBI
34	Koh WJ, Greer BE, Abu-Rustum NR, Apte SM, Campos SM, Cho KR, Chu C, Cohn D, Crispens MA, Dizon DS, et al: Uterine Sarcoma, Version 1.2016: Featured updates to the NCCN guidelines. J Natl Compr Canc Netw. 13:1321–1331. 2015. View Article : Google Scholar : PubMed/NCBI
35	Denschlag D, Thiel FC, Ackermann S, Harter P, Juhasz-Boess I, Mallmann P, Strauss HG, Ulrich U, Horn LC, Schmidt D, et al: Sarcoma of the Uterus. Guideline of the DGGG (S2k-Level, AWMF registry No. 015/074, August 2015). Geburtshilfe Frauenheilkd. 75:1028–1042. 2015. View Article : Google Scholar : PubMed/NCBI
36	Yamaguchi M, Erdenebaatar C, Saito F, Motohara T, Miyahara Y, Tashiro H and Katabuchi H: Long-term outcome of aromatase inhibitor therapy with Letrozole in patients with advanced low-grade endometrial stromal sarcoma. Int J Gynecol Cancer. 25:1645–1651. 2015. View Article : Google Scholar : PubMed/NCBI
37	Cuppens T, Tuyaerts S and Amant F: Potential therapeutic targets in uterine sarcomas. Sarcoma. 2015:2432982015. View Article : Google Scholar : PubMed/NCBI