Benign pulmonary metastasizing leiomyomatosis: case report

The authors report a rare case of leiomyomatosis of the lung diagnosed in a 43-year-old woman, with uterine intravenous leiomyomatosis. Benign metastasizing leiomyoma (BML) is an extremely rare lesion characterized by usually multiple, benign-appearing smooth muscle tumors of the lung in females with coexisting uterine leiomyoma. On the basis of their histological and immunohistological features, a unified histogenetic view of leiomyomas with vascular invasion (LWVI) and BML of the uterus is proposed. LWVI and BML may be the same pathological entity and microscopic vascular invasion may represent the metastatic mechanism of BML. LWVI seems to be the precursor of BML.

Benign metastasizing leiomyoma (BML); Uterine intravenous leiomyomatosis (LWVI); Pulmonary nodules.

[1] Alessi G., Lemmerling M., Vereecken L., De Waele L.: “Benign metastasizing leiomyoma to skull base and spine: a report of two cases”. Clin. Neurol. Neurosurg., 2003, 105, 170.

[2] Joseph V., Chacko G., Raghuram L., Rajshekhar V.: “Benign metastasizing leiomyoma causing spinal cord compression”. Surg. Neurol., 2003, 60, 575.

[3] Patton K.T., Cheng L., Papavero V., Blum M.G, Yeldandi A.V., Adley B.P., et al. Benign metastasizing leiomyoma: clonality,telomere length and clinicopathologic analysis; Mod Pathol, 2006; 19, 130–140.

[4] Agnos J.W., Starkey G.W.: “Primary leiomyosarcoma and leiomyoma of the lung”. N. Engl. J. Med., 1958, 258, 12.

[5] Orlowski T.M., Stasiak K., Kolodziej J.: “Leiomyoma of the lung”. J. Tjrac Cadiovas. Surg., 1978, 76, 257.

[6] White S.H., Ibrahim N.B., Forrester-Wood C.P., Jeyasingham K.: “Leiomyoma of the lower respiratory tract”. Thorax, 1985, 40, 306.

[7] Gal A.A., Brooks J.S., Pietra G.G.: “Leiomyomatous neoplasms of the lung: a clinical, histologic and immunohistochemical study”. Mod. Pathol., 1990, 2, 209.

[8] Tadashi T.: “Case Report: Vascular leiomyoma of the lung arising from pulmonary artery”. Int. J. Clin. Exp. Pathol., 2013, 6, 97.

[9] Steiner P.: “Metastasizing fibroleiomyoma of the uterus: a report of a case and review of the literature”. Am. J. Pathol., 1939, 15, 89.

[10] Cho K.R., Woodruff J.D., Epstein J.I.: “Leiomyoma of the uterus with multiple extrauterine smooth muscle tumors: a case report suggesting multifocal origin”. Hum. Pathol., 1989, 20, 80.

[11] Arai T., Yasuda Y., Takay T., Shibayama M.: “Natural decrease of benign metastasizing leiomyoma”. Chest, 2000, 117, 921.

[12] Uchida T., Tokumaru T., Kojima H., Nakagawaji K., Imaizumi M., Abe T.: “A case of multiple leiomyomatous lesions of the lung: an analysis of flow cytometry and hormone receptors”. Surg. Today, 1992, 22, 265.

[13] Kwon Y.I., Kim T.H., Sohn J.W., Yoon H.J., Shin D.H., Park S.S.: “Benign pulmonary metastasizing leiomvomatosis: case report and a review of the literature”. Korean J. Intern. Med., 2006, 21, 173.

[14] Radzikowska E., Szczepulska-Wójcik E., Langfort R., Oniszh K., Wiatr E.: “Benign pulmonary metastasizing leiomyoma uteri. Case report and review of literature”. Pneumonol. Alergol. Pol., 2012, 80, 560.

[15] Abramson S., Gilkeson R.C., Goldstein J.D., Woodard P.K., Eisenberg R., Abramson N.l.: “Benign metastasizing leiomyoma: clinical, imaging, and pathologic correlation”. AJR Am. J. Roentgenol., 2001, 176, 1409.

[16] Horstmann J.P., Pietra G.G., Harman J.A., Cole N.G., Grinspan S.: “Spontaneous regression of pulmonary leiomyomas during pregnancy”. Cancer, 1977, 39, 314.

[17] Robboy S.J., Bentley R.C., Butnor K., Anderson M.C.: “Pathology and pathophysiology of uterine smooth muscle tumors”. Environ. Health Perspect., 2000, 5 Suppl., 779.

[18] Kim Y.S., Kim E.J., Park C.H., Park J.S., Jee Y.K., Lee K.Y.: “A case of benign metastasizing pulmonary leiomyomatosis”. Tuberc. Respir. Dis., 2002, 53, 190.

[19] Esteban J.M., Allen W.M., Schaerf R.H.: “Benign metastasizing leiomyoma of the uterus: histological and immunohistochemical characterization of primary and metastatic lesions”. Arch. Pathol. Lab. Med., 1999, 123, 960.

[20] Kayser K., Zink S., Schneider T. Dienemann H, André S., Kaltner H., et al.: “Benign metastasizing leiomyoma of the uterus: docu-mentation of clinical, immunohistochemical and lectin-histochemical data of ten cases”. Virchows Arch., 2000, 437, 284.

[21] Shneider T., Kugler C., Kayser K., Dienemann H.: “Benign, pulmonary metastatic leiomyoma of the uterus”. Chirurg., 2001, 72, 308.

[22] Salminen U.S., Halttunen P., Miettinen M., Mattila S.: “Benign mesenchymal tumours of the lung including sclerosing hemangiomas”. Ann. Chir. Gynaecol., 1990, 79, 85.

[23] Rivera J.A., Christopoulos S., Small D., Trifiro M.: “Hormonal manipulation of benign metastasizing leiomyomas ; Report of two cases and review of the literature”. J. Clin. Endocrinol. Metab., 2004, 89, 3183.

[24] Tietze L., Günther K., Hörbe A., Pawlik C., Klosterhalfen B., Handt S., Merkelbach-Bruse S.: “Benign metastasizing leiomyoma: a cytogenetically balanced but clonal disease”. Hum. Pathol., 2000, 31, 126.

[25] Abu-Rustum N.R., Curtin J.P., Burt M., Jones W.B.: “Regression of uterine low–grade smooth–muscle tumors metastatic to the lung after oophorecetomy”. Obstet. Gynecol., 1997, 89, 850.

[26] Jacobson T.Z, Rainey E.J, Turton C.W.: “Pulmonary benign metastasizing leiomyoma: response to treatment with goserelin”. Thorax, 1995, 50, 1225.

[27] Saynajakangas O., Maiche A.G., Liakka K.A.: “Multiple progressive pulmonary leiomyomatous metastases treated with tamoxifen: a case report with a review of the literature”. Acta. Oncol., 2004, 43, 113.

[28] Ishikawa H., Fenkci V., Marsh E.E., Yin P., Chen D., Cheng Y.H., et al.: “CCAAT/Enhancer Binding Protein b regulates aromatase expression via multiple and novel cisregulatory sequences in uterine leiomyoma”. J. Clin. Endocrinol. Metab., 2008, 93, 981. doi: 10.1210/jc.2007-2507. Epub 2008 Jan 8.

[29] Jo J.H., Lee J.H., Kim D.C., Kim S.H., Kwon H.C., Kim J.S., Kim H. J.: “A case of benign metastasizing leiomyoma with multiple metastasis to the soft tissue, skeletal muscle lung and breast”. Korean J. Intern. Med., 2006, 21, 199.

[30] Canzonieri V., D’Amore E.S., Bartoloni G., Piazza M., Blandamura S., Carbone A.: “Leiomyomatosis with vascular invasion. A unified pathogenesis regarding leiomyoma with vascular microinvasion, benign metastasizing leiomyoma and intravenous leiomyomatosis”. Virchows Arch., 1994, 425, 541.