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Piórek A, Płużański A, Wiśniewski P, Tabor S, Winiarczyk K, Knetki-Wróblewska M, Kowalski DM, Krzakowski M. Pulmonary Benign Metastasizing Leiomyoma in a Postmenopausal Woman: A Case Report and Review of the Literature. Diseases 2024; 12:181. [PMID: 39195180 DOI: 10.3390/diseases12080181] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/31/2024] [Revised: 08/02/2024] [Accepted: 08/09/2024] [Indexed: 08/29/2024] Open
Abstract
Pulmonary benign metastasizing leiomyoma (PBML) is a rare condition characterized by the spread of uterine leiomyomas to the lungs, typically observed in premenopausal women with a history of hysterectomy or myomectomy. This report presents a unique case of a postmenopausal woman, aged 65, that emphasizes the clinical, radiological, histologic, and immunohistochemical aspects of the disease. On presentation, the patient suffered from severe pain. On imaging, a sizable lung tumor was found. Histopathological examination and immunoprofiling confirmed PBML. The patient underwent various treatments, including surgery, radiation therapy, and hormonal therapy, illustrating the challenges in managing PBML. A literature review underscores the rarity of PBML and its diverse clinical manifestations. This study provides valuable insights into the complexities of PBML.
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Affiliation(s)
- Aleksandra Piórek
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Adam Płużański
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Piotr Wiśniewski
- Department of Pathology, Military Institute of Medicine-National Research Institute, 04-141 Warsaw, Poland
| | - Sylwia Tabor
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Kinga Winiarczyk
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Magdalena Knetki-Wróblewska
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Dariusz M Kowalski
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
| | - Maciej Krzakowski
- Department of Lung Cancer and Thoracic Tumors, Maria Sklodowska-Curie National Research Institute of Oncology, 02-781 Warsaw, Poland
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Tu W, Yano M, Schieda N, Krishna S, Chen L, Gottumukkala RV, Alencar R. Smooth Muscle Tumors of the Uterus at MRI: Focus on Leiomyomas and FIGO Classification. Radiographics 2023; 43:e220161. [PMID: 37261965 DOI: 10.1148/rg.220161] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/03/2023]
Abstract
Leiomyomas are smooth muscle tumors of the uterus and are the most common uterine neoplasm. Although leiomyomas are usually asymptomatic, they can manifest with symptoms such as pain or uterine bleeding. Leiomyomas are classified on the basis of their anatomic location and morphology. Localization of leiomyomas relative to the endometrium, myometrium, and uterine serosa with use of the International Federation of Gynecology and Obstetrics (FIGO) classification system is helpful for guiding management in symptomatic patients. The FIGO system is a practical and universally accepted approach for classifying leiomyomas to guide radiologists and clinicians in deciding management. The MRI appearance of conventional leiomyomas is related to their tissue contents of smooth muscle and fibrous tissue and is well established. The MRI features of some leiomyoma subtypes and forms of degeneration also have been described. Other smooth muscle tumors of the uterus recognized in the 2020 World Health Organization classification system include intravenous leiomyomatosis, smooth muscle tumors of uncertain malignant potential, and metastasizing leiomyoma. At the far end of the spectrum are leiomyosarcomas, which are frankly malignant and therefore must be managed accordingly. Although MRI features that suggest a diagnosis of leiomyosarcoma have been proposed, these features overlap with those of some leiomyoma subtypes and degeneration. © RSNA, 2023 See the invited commentary by Fennessy and Gargiulo in this issue. Online supplemental material and the slide presentation from the RSNA Annual Meeting are available for this article. Quiz questions for this article are available through the Online Learning Center.
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Affiliation(s)
- Wendy Tu
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Motoyo Yano
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Nicola Schieda
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Satheesh Krishna
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Longwen Chen
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Ravi V Gottumukkala
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
| | - Raquel Alencar
- From the Department of Medical Imaging, University of Alberta, 116 St and 85 Ave, Edmonton, Alberta, Canada T6G 2R3 (W.T.); Department of Radiology (M.Y.) and Department of Laboratory Medicine and Pathology (L.C.), Mayo Clinic Arizona, Phoenix, AZ; Department of Radiology, University of Ottawa, Ottawa, Ontario, Canada (N.S.); Department of Medical Imaging, University Medical Imaging Toronto, University of Toronto, Toronto, Ontario, Canada (S.K.); and Department of Radiology, Brigham and Women's Hospital, Harvard University, Boston, MA (R.V.G., R.A.1)
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Tong T, Fan Q, Wang Y, Li Y. Benign metastasizing uterine leiomyoma with lymphatic and pulmonary metastases: a case report and literature review. BMC Womens Health 2023; 23:154. [PMID: 37005604 PMCID: PMC10068149 DOI: 10.1186/s12905-023-02237-y] [Citation(s) in RCA: 9] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/06/2022] [Accepted: 02/21/2023] [Indexed: 04/04/2023] Open
Abstract
BACKGROUND Benign metastasizing leiomyoma (BML) is a rare disease usually observed in women of reproductive or premenopausal age with a history of uterine myomectomy or hysterectomy. The most common sites of metastases are the pulmonary, and other sites include heart, bones, liver, lymph nodes, bladder, skeletal muscles, and central nervous system. Here, we report a case of a 50 year-old woman with a history of hysterectomy who was initially suspected of uterine sarcoma but was finally confirmed to have BML with lung and lymph node metastases, and discuss the treatment and prognosis of BML. CASE PRESENTATION A 50 year-old woman with a history of total abdominal hysterectomy presented with mild but persistent abdominal pain for more than 3 months. She was suspected of having uterine sarcoma before surgery and laparoscopic extensive debulking surgery including bilateral oophorectomy, pelvic and para-aortic lymph node dissection to the level of the left renal vein, and transcutaneous dissection of the right inguinal lymph nodes. Pathology confirmed a benign leiomyoma, and the patient was diagnosed with BML. No medication was administered after the surgery, and the follow-up was of no significance. CONCLUSION Benign metastasizing leiomyoma (BML) is a rare disorder in which histologically benign smooth muscle tumors metastasize to extrauterine sites. Metastases are commonly observed in the lung, liver, lymph nodes, skin, bladder, esophagus, and skeletal muscles. BML is usually misdiagnosed as a malignant tumor before surgery until the pathology confirms its benign nature. However, this treatment remains controversial and undetermined. The prognosis is usually favorable owing to its benign nature.
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Affiliation(s)
- Tong Tong
- Department of Gynecological Oncology, The International Peace Maternity and Child Health Hospital, School of Medicine, Shanghai Jiao Tong University, Hengshan Road No. 910, Shanghai, 200030, China
| | - Qiong Fan
- Department of Gynecological Oncology, The International Peace Maternity and Child Health Hospital, School of Medicine, Shanghai Jiao Tong University, Hengshan Road No. 910, Shanghai, 200030, China
| | - Yudong Wang
- Department of Gynecological Oncology, The International Peace Maternity and Child Health Hospital, School of Medicine, Shanghai Jiao Tong University, Hengshan Road No. 910, Shanghai, 200030, China.
| | - Yuhong Li
- Department of Gynecological Oncology, The International Peace Maternity and Child Health Hospital, School of Medicine, Shanghai Jiao Tong University, Hengshan Road No. 910, Shanghai, 200030, China
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Libertini M, Hallin M, Thway K, Noujaim J, Benson C, van der Graaf W, Jones RL. Gynecological Sarcomas: Molecular Characteristics, Behavior, and Histology-Driven Therapy. Int J Surg Pathol 2020; 29:4-20. [PMID: 32909482 DOI: 10.1177/1066896920958120] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Gynecological sarcomas represent 3% to 4% of all gynecological malignancies and 13% of all sarcomas. The uterus is the most frequent primary site (83%); less frequently sarcomas are diagnosed originating from the ovary (8%), vulva and vagina (5%), and other gynecologic organs (2%). As the classification of gynecologic sarcomas continues to diversify, so does the management. Accurate histopathologic diagnosis, utilizing appropriate ancillary immunohistochemical and molecular analysis, could lead to a more personalized approach. However, there are subtypes that require further definition, with regard to putative predictive markers and optimal management. The aim of this review is to highlight the importance of accurate diagnosis and classification of gynecologic sarcoma subtypes by the surgical pathologist in order to provide more tailored systemic treatment, and to highlight the increasing importance of close collaboration between the pathologist and the oncologist.
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Affiliation(s)
| | - Magnus Hallin
- Royal Marsden Hospital/Institute of Cancer Research, London, UK
| | - Khin Thway
- Royal Marsden Hospital/Institute of Cancer Research, London, UK
| | | | | | | | - Robin L Jones
- Royal Marsden Hospital/Institute of Cancer Research, London, UK
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Dai HY, Guo SL, Shen J, Yang L. Pulmonary benign metastasizing leiomyoma: A case report and review of the literature. World J Clin Cases 2020; 8:3082-3089. [PMID: 32775390 PMCID: PMC7385613 DOI: 10.12998/wjcc.v8.i14.3082] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/20/2020] [Revised: 06/16/2020] [Accepted: 06/30/2020] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Pulmonary benign metastatic leiomyoma (PBML), which is very rare, is a type of benign metastatic leiomyoma (BML). Here, we report a case of PBML, finally diagnosed through multidisciplinary team (MDT) discussions, and provide a literature review of the disease.
CASE SUMMARY A 55-year old asymptomatic woman was found to have bilateral multiple lung nodules on a chest high-resolution computed tomography (HRCT) scan. Her medical history included total hysterectomy for uterine leiomyoma. The patient was diagnosed with PBML, on the basis of her clinical history, imaging manifestations, and computed tomography (CT)-guided percutaneous lung puncture biopsy, via MDT discussions. As the patient was asymptomatic, she received long-term monitoring without treatment. A follow-up of chest HRCT after 6 mo showed that the PBML lung nodules were stable and there was no progression.
CONCLUSION For patients with a medical history of hysterectomy and uterine leiomyoma with lung nodules on chest CT, PBML should be considered during diagnosis based on the clinical history, imaging manifestations, CT-guided percutaneous lung puncture biopsy, and MDT discussions.
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Affiliation(s)
- Hai-Yun Dai
- Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400010, China
| | - Shu-Liang Guo
- Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400010, China
| | - Jian Shen
- Department of Cardiology, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400010, China
| | - Li Yang
- Department of Respiratory and Critical Care Medicine, The First Affiliated Hospital of Chongqing Medical University, Chongqing 400010, China
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Abstract
RATIONALE Pulmonary benign metastasizing leiomyoma (PBML) is rare, usually occurs in women who underwent hysterectomy during the reproductive years, and has no obvious clinical symptoms. A full understanding of the characteristics of PBML is important for its sequential treatment and prognosis. PATIENT CONCERNS In this report, a 36-year-old female patient with previous uterine leiomyoma who underwent 3 surgical resections of the uterus, bilateral fallopian tubes, and partial omentum was investigated. The physical examination revealed a tumor in the right lower lobe and mediastinum and a solid nodule in the right middle lobe. DIAGNOSES Chest computed tomography (CT) confirmed a tumor in the right lower lobe and mediastinum and a solid nodule in the right middle lobe. Further positron-emission tomography computed tomography (PET-CT) with 18F-fluorodeoxyglucose (FDG) of the whole body showed mildly intense accumulation of 18F-FDG in the tumor (maximum standardized uptake value [SUV max], 2.6). A pathological examination then confirmed the presence of fibrous and vascular tissue after CT-guided percutaneous biopsy of the tumor in the right lower lobe. Additionally, surgical resection of the tumor and nodule was performed for histological analysis and immunohistochemical assays for estrogen receptor (ER) and progesterone receptor (PR). INTERVENTIONS The patient underwent complete tumor surgical resection and nodule wedge resection. OUTCOMES No postoperative complications occurred. No recurrence or other signs of metastasis were found during an 18-month follow-up observation period. CONCLUSION In this case, lung and mediastinal metastasis of uterine fibroids was observed. However, depending on only a postoperative histological analysis is insufficient for the diagnosis of PBML. Histological analysis combined with an evaluation of the expression levels of ER and PR is crucial for the diagnosis and treatment of PBML.
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Affiliation(s)
| | | | | | - Yuwei Guo
- Department of pathology, Hebei Medical University Fourth Hospital
| | - Mengdi Cong
- Department of pathology, Children's hospital of Hebei Province, Shijiazhuang, China
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Abstract
RATIONALE Multiple pulmonary leiomyomatous hamartoma (MPLH) is an extremely rare benign disease that mostly occurs in women of reproductive age. PATIENT CONCERNS A 32-year-old female patient recently diagnosed with multiple bilateral pulmonary nodules. She has the symptoms of dry cough, chest tightness, dyspnea on exertion. Chest X-ray identified multiple bilateral pulmonary nodules in the lung, and the diameter of the largest nodule was about 3.1 cm. DIAGNOSES Pathology confirmed the diagnosis of MPLH based on morphology and immunohistochemical staining. INTERVENTIONS The patient presented with multiple well-defined nodular shadows in chest computed tomography (CT), atypical image and symptoms were detected. Positron emission tomography/CT scan showed mild fluorine-18 fluorodeoxyglucose uptake in the lesions and no abnormal foci in any other parts of her body. She subsequently underwent a video-assisted thoracoscopic surgery with wedge resection of the biggest one of the nodules. Then the patient given symptomatic treatment, without hormone, no further treatment was prescribed. OUTCOMES The patient is in the good general condition and without obvious pulmonary symptoms after the follow-up of 1 year, chest CT scan showed no significant changes in the sizes and locations of her bilateral pulmonary nodules. LESSONS Due to its rare presentation, the primary MPLH may be undiagnosed. Awareness of main morphologic and immunohistochemical features of MPLH is critical for the recognition of this uncommon disease.
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Affiliation(s)
- Dan Cheng
- Department of Respiratory and Critical Care Medicine, Renmin Hospital of Wuhan University, Wuhan, China
- Lung Biology Center, Department of Medicine, University of California, San Francisco, CA
| | - Fangcheng Zhang
- Center of Ultrapathology, Renmin Hospital of Wuhan University, Wuhan, China
| | - Ke Hu
- Department of Respiratory and Critical Care Medicine, Renmin Hospital of Wuhan University, Wuhan, China
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Affiliation(s)
- Boris I Medarov
- Division of Pulmonary, Critical Care and Sleep Medicine, Albany Medical College, Albany, New York
| | - Malik M H S Khan
- Division of Pulmonary, Critical Care and Sleep Medicine, Albany Medical College, Albany, New York
| | - Aakash Modi
- Division of Pulmonary, Critical Care and Sleep Medicine, Albany Medical College, Albany, New York
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Abu Saadeh F, Riain CO, Cormack CM, Gleeson N. Lung metastases from benign uterine leiomyoma: does 18-FDG-PET/CT have a role to play? Ir J Med Sci 2018; 188:619-624. [DOI: 10.1007/s11845-018-1876-0] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/13/2018] [Accepted: 07/16/2018] [Indexed: 12/26/2022]
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Rizzo V, Parissis H. A rare case of benign metastasizing leiomyoma†. J Surg Case Rep 2017; 2017:rjx190. [PMID: 28959432 PMCID: PMC5610580 DOI: 10.1093/jscr/rjx190] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/12/2017] [Accepted: 09/16/2017] [Indexed: 11/20/2022] Open
Abstract
Benign metastasizing leiomyoma is a rare disorder involving distant metastases secondary to a smooth muscle tumour of the myometrium. This case report describes a 48-year-old woman with multiple pulmonary nodules noted on post-operative chest x-ray. Further investigation revealed a history of total abdominal hysterectomy for benign fibroids of the uterus, 6 years prior to the scan. CT-guided fine needle aspirate of the pulmonary nodules showed smooth muscle proliferation within lung parenchyma, suggesting smooth muscle tumour confirmed with immunohistochemistry. Management is still controversial in view of low case numbers; however, treatment with hormone therapy and castration has been attempted.
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Affiliation(s)
- Victoria Rizzo
- Royal Victoria Hospital, Belfast BT12 6BA, UK
- Belfast Health and Social Care Trust, Trust Headquarters, Belfast City Hospital, Belfast BT9 7AB, UK
- Correspondence address. Royal Victoria Hospital, 274 Grosvenor Road, Belfast BT126BA, UK. E-mail:
| | - Haralambos Parissis
- Royal Victoria Hospital, Belfast BT12 6BA, UK
- Belfast Health and Social Care Trust, Trust Headquarters, Belfast City Hospital, Belfast BT9 7AB, UK
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Lee SR, Choi YI, Lee SJ, Shim SS, Lee JH, Kim YK, Sung SH. Multiple cavitating pulmonary nodules: rare manifestation of benign metastatic leiomyoma. J Thorac Dis 2017; 9:E1-E5. [PMID: 28203428 DOI: 10.21037/jtd.2016.11.112] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Benign metastasizing leiomyoma (BML) is a rare disease of pathologically benign, but the tumor metastasizes to other organs. The most common organ involved in BML is lung. Pulmonary involvement usually manifested as multiple nodules on chest X-ray, however we experienced an interesting case of a 52-year-old premenopausal woman who presented with multiple bilateral lung cavitations and nodules on a chest X-ray without any respiratory symptoms. Chest computed-tomography identified multiple cavitary lesions of 5-12 mm in diameter and well-defined nodules of 5-10 mm in diameter in both lung fields. Transthoracic needle biopsy was performed and the resected lesion consisted of benign spindle cells was positive for estrogen receptor (ER) and progesterone receptor (PR) and was diffuse positive for actin and desmin by immunohistochemical (IHC) staining, suggesting leiomyoma. The final diagnosis was benign pulmonary metastasizing leiomyoma (BPML) and the patient underwent subcutaneous injection of a gonadotrophin releasing hormone (GnRH) agonist for 12 months, follow-up low-dose chest computed tomography (CT) scan at 15 months revealed decreased cavitations and nodular lesions. We should take into consideration the possibility of BPML when we encounter multiple cystic or cavitary lesions on chest X-ray, although the common form of BPML is nodulary lesions on imaging studies.
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Affiliation(s)
- Sa Ra Lee
- Departments of Obstetrics and Gynecology, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Youn-I Choi
- Department of Internal Mediciney, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Seok Jeong Lee
- Department of Internal Mediciney, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Sung Shine Shim
- Department of Radiology, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Jin Hwa Lee
- Department of Internal Mediciney, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Yoo Kyung Kim
- Department of Radiology, Ewha Womans University School of Medicine, Seoul, Republic of Korea
| | - Soon Hee Sung
- Department of Pathology, Ewha Womans University School of Medicine, Seoul, Republic of Korea
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Huang BS, Yang MH, Wang PH, Li HY, Chou TY, Chen YJ. Oestrogen-induced angiogenesis and implantation contribute to the development of parasitic myomas after laparoscopic morcellation. Reprod Biol Endocrinol 2016; 14:64. [PMID: 27716434 PMCID: PMC5053344 DOI: 10.1186/s12958-016-0200-y] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/27/2016] [Accepted: 09/29/2016] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Iatrogenic parasitic myomas (PMs), caused by intra-corporeal power morcellation during laparoscopy is gradually increasing. However, the pathogenesis and medical treatment of PMs remain largely unelucidated. METHODS Laparoscopically-induced PM xenografted mouse model was conducted by xenografting human uterine myoma fragments into the abdominal cavity of SCID mice and hormonal manipulation was performed using this mouse model to demonstrate the role of oestrogen in the development of implanted PMs. Immunohistochemistry of oestrogen receptor α (ERα), progesterone receptor (PR), vimentin, vascular endothelial growth factor (VEGF), microvessel density (MVD) and Ki-67 index was performed and compared. RESULTS In the patient with PMs, ERα, PR, angiogenesis and proliferative property expression were upregulated in PM lesions compared to uterine myomas. In the laparoscopically-induced PM mouse model, implanted myomas had more steroid receptor expressions, angiogenesis and proliferative property compared with pre-xenografted or non-implanted myoma. Depletion of oestrogen in the ovariectomized (OVX) mice decreased laparoscopically-induced PM implantations. In comparison, the implantations of PMs were increased with additional E2 supplement. Hormonal manipulation in the PM mouse model, including AI, GnRHa and SERM groups, were compared and AI significantly decreased the implantations, steroid receptor, angiogenesis, cell density, and proliferative index of PMs compared with control group. Furthermore, GnRHa significantly decreased VEGF and MVD expressions compared with control group. CONCLUSIONS These data highlight the crucial role of oestrogen in the development of laparoscopically-induced PMs and suggest that hormone manipulation may be a potential therapeutic agent. TRIAL REGISTRATION This protocol was approved by the Human and Animal Institutional Review Board of Taipei Veterans General Hospital ( VGHIRB No 2014-10-002C on Nov. 17th, 2014; IACUC 2014-119 on Aug. 22nd, 2014).
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Affiliation(s)
- Ben-Shian Huang
- Department of Obstetrics and Gynaecology, Taipei Veterans General Hospital, No.201, Sec. 2, Shih-Pai Road, Taipei, 112 Taiwan
- Department of Obstetrics and Gynaecology, National Yang-Ming University Hospital, No.169, Siaoshe Road, Yilan, 260 Taiwan
- Department of Obstetrics and Gynaecology, School of Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
- Institute of Clinical Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
| | - Muh-Hwa Yang
- Institute of Clinical Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
| | - Peng-Hui Wang
- Department of Obstetrics and Gynaecology, Taipei Veterans General Hospital, No.201, Sec. 2, Shih-Pai Road, Taipei, 112 Taiwan
- Department of Obstetrics and Gynaecology, School of Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
- Institute of Clinical Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
| | - Hsin-Yang Li
- Department of Obstetrics and Gynaecology, Taipei Veterans General Hospital, No.201, Sec. 2, Shih-Pai Road, Taipei, 112 Taiwan
| | - Teh-Ying Chou
- Institute of Clinical Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
| | - Yi-Jen Chen
- Department of Obstetrics and Gynaecology, Taipei Veterans General Hospital, No.201, Sec. 2, Shih-Pai Road, Taipei, 112 Taiwan
- Department of Obstetrics and Gynaecology, School of Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
- Institute of Clinical Medicine, National Yang-Ming University, No.155, Sec.2, Li-Nong Street, Taipei, 112 Taiwan
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Abstract
Benign metastasizing leiomyoma (BML) is a rare and poorly characterized disease affecting primarily premenopausal women. Asymptomatic patients are often diagnosed incidentally by radiographs or other lung-imaging procedures performed for other indications, and the diagnosis is eventually confirmed by biopsy. Patients with BML are usually treated pharmacologically with antiestrogen therapies or surgically with oophorectomy or hysterectomy. Antiestrogen therapy is typically efficacious and, in general, most patients have a favorable prognosis. Asymptomatic patients with a confirmed diagnosis of BML, may be followed conservatively without treatment.
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Affiliation(s)
- Gustavo Pacheco-Rodriguez
- Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Building 10, Room 5N307, 9000 Rockville Pike, Bethesda, MD 20892-1434, USA
| | - Angelo M Taveira-DaSilva
- Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Building 10, Room 6D05, MSC-1590, 9000 Rockville Pike, Bethesda, MD 20892-1590, USA
| | - Joel Moss
- Cardiovascular and Pulmonary Branch, National Heart, Lung, and Blood Institute, National Institutes of Health, Building 10, Room 6D05, MSC-1590, 9000 Rockville Pike, Bethesda, MD 20892-1590, USA.
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Wei WT, Chen PC. Benign metastasizing leiomyoma of the lung: A case report and literature review. Oncol Lett 2015; 10:307-312. [PMID: 26171020 DOI: 10.3892/ol.2015.3224] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/07/2014] [Accepted: 04/14/2015] [Indexed: 11/06/2022] Open
Abstract
Pulmonary benign metastasizing leiomyoma (BML) is a rare event characterized by benign soft-tissue tumors that occur when uterine leiomyomas metastasize to the lung. The present study reports the case of a 47-year-old female patient who presented with multiple bilateral pulmonary nodules on a chest X-ray during a health checkup nine years after a hysterectomy due to uterine fibroids. Chest computed tomography (CT) revealed multiple well-defined nodular shadows in the lung. One tumor of the left upper lung was resected by thoracoscopic surgery. Pathologically, the resected lesion consisted of benign spindle cells and was diagnosed as BML. The post-operative course was uneventful. Other lung nodules have been meticulously monitored at follow-up, and repeat CT two years later showed that these nodules had not increased at all in size and that no new lobe nodules had appeared. The present study indicates that pulmonary BML occurs in a low proportion of female with a history of uterine leiomyoma and treatment methods for it are diverse and controversial.
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Affiliation(s)
- Wei-Tian Wei
- Department of Oncological Surgery, Zhejiang Cancer Hospital, Hangzhou, Zhejiang 310000, P.R. China
| | - Peng-Cheng Chen
- Department of Oncological Surgery, Zhejiang Cancer Hospital, Hangzhou, Zhejiang 310000, P.R. China
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15
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Alraiyes AH, Kheir F, Hirsh S, Salerno D, Bernal-Green L, Daroca P. A 40-year-old woman with multiple lung nodules. Chest 2013; 143:1826-1829. [PMID: 23732596 DOI: 10.1378/chest.12-1795] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/12/2023] Open
Affiliation(s)
- Abdul Hamid Alraiyes
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA.
| | - Fayez Kheir
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA
| | - Sharon Hirsh
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA
| | - Daniel Salerno
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA
| | - Lynn Bernal-Green
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA
| | - Philip Daroca
- Department of Pulmonary, Critical Care and Environmental Medicine, Tulane University Health Sciences Center, New Orleans, LA
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16
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17
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Chen S, Zhang Y, Zhang J, Hu H, Cheng Y, Zhou J, Shen L, Chen H. Pulmonary benign metastasizing leiomyoma from uterine leiomyoma. World J Surg Oncol 2013; 11:163. [PMID: 23866077 PMCID: PMC3722006 DOI: 10.1186/1477-7819-11-163] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/12/2012] [Accepted: 07/07/2013] [Indexed: 11/10/2022] Open
Abstract
Background Benign metastasizing leiomyoma (BML) occurs in a low proportion of uterine leiomyomas and treatment methods for BML are diverse and controversial. The study introduces preliminary experiences in the diagnosis and treatment of BML with the purpose of finding a suitable management strategy for these patients. Methods Three patients with BML were treated in our department from April 2008 to July 2012. Each of these patients presented with multiple nodules in both lungs, where we performed video-assisted thoracoscopic wedge resection to harvest enough tissue for histopathologic and immunohistochemical examination. The patients were treated with medical castration or surgical castration after the diagnosis of BML. Results The ultimate pathologic results ruled out the possibility of leiomyosarcoma and other metastatic diseases, and confirmed that the pulmonary lesions were BML. The lung lesions remained stable in two patients who were treated by surgical castration, and the lung nodules regressed in one patient treated with gonadotropin-releasing hormone analogues. Conclusions The diagnosis of BML is based on the medical history of uterine myomas and histopathologic and immunohistochemical examination of lung nodules. Video-assisted thoracoscopic wedge resection is the best way to harvest tissue for diagnosis. The better outcomes in BML seem to call for medical intervention, either chemical or surgical, after diagnosis is made.
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Affiliation(s)
- Sufeng Chen
- Department of Thoracic Surgery, Fudan University Shanghai Cancer Center, 270 Dong'an Road, Shanghai 200032, China
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18
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Lewis EI, Chason RJ, DeCherney AH, Armstrong A, Elkas J, Venkatesan AM. Novel hormone treatment of benign metastasizing leiomyoma: an analysis of five cases and literature review. Fertil Steril 2013; 99:2017-24. [PMID: 23465706 PMCID: PMC3672263 DOI: 10.1016/j.fertnstert.2013.01.147] [Citation(s) in RCA: 55] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/06/2011] [Revised: 01/30/2013] [Accepted: 01/30/2013] [Indexed: 01/04/2023]
Abstract
OBJECTIVE To evaluate novel hormonal therapies in patients with unresectable benign metastasizing leiomyoma (BML) disease. DESIGN Case series. SETTING National Institutes of Health (NIH). PATIENT(S) Five subjects with the diagnosis of BML based on imaging and/or histopathologic diagnosis. INTERVENTION(S) Four patients were treated with single or combination therapy of leuprolide acetate and/or an aromatase inhibitor. One patient was treated with an antiprogestin (CDB-2914). MAIN OUTCOME MEASURE(S) Response to therapy was measured by tumor burden on cross-sectional imaging employing RECIST (Response Evaluation Criteria in Solid Tumors) 1.1 guidelines. RESULT(S) Four patients treated with single or combination therapy of leuprolide acetate and/or an aromatase inhibitor demonstrated stable disease with reduction in tumor burden. The fifth patient treated with antiprogestin (CDB-2914) had degeneration of her tumor, progression of its size, and an improvement in symptoms. CONCLUSION(S) Hormone treatment with GnRH agonist and/or aromatase inhibition may be a therapeutic option to reduce tumor burden in unresectable BML disease or for those patients who wish to avoid surgical intervention. RECIST 1.1 guidelines, while traditionally used to evaluate tumor response to cancer therapeutics, may be useful in evaluating BML tumor burden response to hormone therapy.
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Affiliation(s)
- Erin I Lewis
- Department of Obstetrics and Gynecology, UCLA Medical Center, Los Angeles, California 90095, USA.
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19
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Okabe R, Shoji T, Huang CL. Benign metastasizing leiomyoma of the lung with spontaneous pneumothorax. Thorac Cardiovasc Surg Rep 2013; 2:26-8. [PMID: 25360407 PMCID: PMC4176076 DOI: 10.1055/s-0033-1345266] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/17/2013] [Accepted: 04/09/2013] [Indexed: 11/28/2022] Open
Abstract
A 47-year-old woman was admitted with recurrent pneumothorax. Preoperative chest computed tomography (CT) showed multiple lung nodules and cysts bilaterally. She had undergone enucleatic myomectomy 12 years earlier. Video-associated thoracoscopic biopsy was performed. Histopathologically, there were bulla-like dilated cystic changes, the walls of which showed spindle cell proliferation, causing pneumothorax. Hormone therapy was started after benign metastasizing leiomyoma resection; pneumothorax has not recurred in 7 months. Multiple residual lung nodules have decreased or disappeared on CT.
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Affiliation(s)
- Ryo Okabe
- Division of Thoracic Surgery, Matsue Red Cross Hospital, Matsue, Japan
| | - Tsuyoshi Shoji
- Department of Thoracic Surgery, Kitano Hospital, Osaka, Japan
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20
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Jiang CY, Wang W, Yuan ZR. A rare pancreatic tumor in a 52-year-old Chinese woman. Pancreatic benign metastasizing leiomyoma. Gastroenterology 2013; 144:511, 659-60. [PMID: 23333466 DOI: 10.1053/j.gastro.2012.10.016] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/08/2012] [Revised: 09/29/2012] [Accepted: 10/10/2012] [Indexed: 12/30/2022]
Affiliation(s)
- Chong-Yi Jiang
- Department of Surgery, Huadong Hospital, Fudan University, Shanghai, PR China
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21
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Rege AS, Snyder JA, Scott WJ. Benign metastasizing leiomyoma: a rare cause of multiple pulmonary nodules. Ann Thorac Surg 2012; 93:e149-51. [PMID: 22632533 DOI: 10.1016/j.athoracsur.2011.12.047] [Citation(s) in RCA: 27] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/09/2011] [Revised: 11/22/2011] [Accepted: 12/12/2011] [Indexed: 01/04/2023]
Abstract
Benign metastasizing leiomyoma (BML) is a rare cause of pulmonary nodules that occurs when uterine leiomyomas metastasize to the lung. The management of these lesions varies from resection and hysterectomy to nonsurgical treatments such as hormonal therapy. We report a case of a 45-year-old woman with multiple nodules of the right lung identified during preoperative imaging before her hysterectomy for uterine fibroids.
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Affiliation(s)
- Aparna S Rege
- Department of Surgical Oncology, Fox Chase Cancer Center, Philadelphia, Pennsylvania 19111, USA
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22
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Silva I, Tomé V, Oliveira J. Benign metastasising leiomyoma: a progressive disease despite chemical and surgical castration. BMJ Case Rep 2012; 2012:bcr.01.2012.5505. [PMID: 22605795 DOI: 10.1136/bcr.01.2012.5505] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Benign metastasising leiomyoma (BML) is a rare entity characterised by uterine leiomyoma that, later on, develops slow-growing metastasis mainly to the lung. In general, these lung metastases are incidentally discovered, but sometimes can become symptomatic with dyspnoea, cough and chest pain. The expression of oestrogen and progesterone receptors by these tumours supports the idea that they respond to hormone therapy (chemical, with oestrogen receptor modulators, aromatase inhibitors or luteinising hormone releasing hormone analogues and surgical, with bilateral adnexectomy). The authors present a case report of BML with two peculiarities: a less common pattern of metastisation (soft tissue), in addition to lung; and disease progression despite treatment with chemical and surgical castration.
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Affiliation(s)
- Inês Silva
- Department of Clinical Oncology, Instituto Português de Oncologia, Lisboa, Portugal, Lisbon, Portugal.
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23
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Pulmonary benign metastasizing leiomyoma from the uterus in a postmenopausal woman: report of a case. Indian J Thorac Cardiovasc Surg 2010. [DOI: 10.1007/s12055-010-0067-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022] Open
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24
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25
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Abstract
Pulmonary benign metastasizing leiomyoma (BML) is a rare disease occurring predominantly in women of reproductive age and usually develops several years after the resection of a uterine leiomyoma. A 52-year-old postmenopausal woman was admitted to our hospital because of a right-sided empyema. Contrast-enhanced computed tomography showed a multiloculated pleural effusion on the right side and multiple small nodules in the left lung. A wedge biopsy revealed the pulmonary nodule consisting of branching glandular structures surrounded by abundant smooth muscle cells with no atypia. We performed a gynecologic examination to identify the primary origin of the pulmonary smooth muscle tumors. A uterine leiomyoma was found, and the patient underwent a total hysterectomy. Both pulmonary nodules and uterine leiomyoma were positive for estrogen and progesterone receptors. Therefore, we diagnosed the pulmonary lesions as BMLs. This is an interesting case of pulmonary BML identified simultaneously with uterine myoma in a postmenopausal woman. BML should be considered in women with multiple pulmonary nodules, even though it is rare.
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26
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Lee EJ, Jeong HC, Lee SY, Kim JH, Lee SY, Shin C, Shim JJ, In KH, Kang KH, Yoo SH, Lee SH, Kim HK, Oh YW. 2 Cases of a Benign Pulmonary Metastasizing Leiomyoma. Tuberc Respir Dis (Seoul) 2009. [DOI: 10.4046/trd.2009.67.6.551] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/24/2022] Open
Affiliation(s)
- Eun Joo Lee
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Hye Cheol Jeong
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Sung Yong Lee
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Je Hyeong Kim
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Sang Yeub Lee
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Chol Shin
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Jae Jeong Shim
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Kwang Ho In
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Kyung Ho Kang
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Se Hwa Yoo
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Korea University Hospital, Seoul, Korea
| | - Sang Hoon Lee
- Department of Obstetrics and Gynecology, Korea University Hospital, Seoul, Korea
| | - Han-Kyeom Kim
- Department of Pathology, Korea University Hospital, Seoul, Korea
| | - Yu Whan Oh
- Department of Radiology, Korea University Hospital, Seoul, Korea
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27
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Fasih N, Prasad Shanbhogue AK, Macdonald DB, Fraser-Hill MA, Papadatos D, Kielar AZ, Doherty GP, Walsh C, McInnes M, Atri M. Leiomyomas beyond the Uterus: Unusual Locations, Rare Manifestations. Radiographics 2008; 28:1931-48. [PMID: 19001649 DOI: 10.1148/rg.287085095] [Citation(s) in RCA: 238] [Impact Index Per Article: 14.0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/02/2023]
Affiliation(s)
- Najla Fasih
- Department of Diagnostic Imaging, Ottawa Hospital, Ottawa, ON, Canada.
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28
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Multiple benign metastasizing leiomyomas in the pelvic lymph nodes and biceps muscle: report of a case. Surg Today 2008; 38:432-5. [PMID: 18560966 DOI: 10.1007/s00595-007-3609-2] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/08/2007] [Accepted: 07/09/2007] [Indexed: 10/22/2022]
Abstract
A 47-year-old woman with an earlier history of uterine leiomyoma suffered from multiple recurrent tumors in the retroperitoneal lymph nodes and biceps muscle of the right upper arm. The woman with a right lower abdominal tumor was referred to our hospital. An abdominal computed tomography scan revealed two round nodules with well-defined margins in the retroperitoneum in the pelvis, and echography revealed a similar nodule in the biceps of the right upper arm. A biopsy of the abdominal retroperitoneal tumor demonstrated benign metastasizing leiomyoma (BML). An extirpation of the abdominal tumors was therefore performed. After the operation, false climacteric medical treatment was performed for 3 years and no recurrence has since been observed. This is the first reported case of multiple BML in the lymph nodes and muscle occurring simultaneously.
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29
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Kwon YI, Kim TH, Sohn JW, Yoon HJ, Shin DH, Park SS. Benign pulmonary metastasizing leiomvomatosis: case report and a review of the literature. Korean J Intern Med 2006; 21:173-7. [PMID: 17017666 PMCID: PMC3890720 DOI: 10.3904/kjim.2006.21.3.173] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/27/2022] Open
Abstract
The authors report here on a case of a nearly asymptomatic 51-year-old Korean woman who was found to have diffuse, multiple nodules of the lungs on a routine chest radiograph. She had undergone hysterectomy 16 years previously for uterine myoma. An open lung biopsy revealed tumor that was composed of interlacing bundles of spindle cells with cigar shaped nucleus and eosinophilic myofibrils in the cytoplasm; consistent with multiple leiomyomas. The stains for SMA, desmin, MSA and Ki-67 were positive and the stain for c-kit was negative. The other stains for estrogen and progesterone receptor were positive. During the open lung biopsy procedure, all the nodules were excised. We report here on an interesting case of benign metastasizing leiomyoma (BML) in 51-year-old patient. To the best of our knowledge, this case showed the longest period of clinical progression in Korea. This is also one of a few cases in which curative excision was successfully performed.
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Affiliation(s)
- Young-Il Kwon
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
| | - Tae-Hyung Kim
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
| | - Jang Won Sohn
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
| | - Ho Joo Yoon
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
| | - Dong Ho Shin
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
| | - Sung Soo Park
- Department of Internal Medicine, Hanyang University College of Medicine, Seoul, Korea
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30
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Wentling GK, Sevin BU, Geiger XJ, Bridges MD. Benign metastasizing leiomyoma responsive to megestrol: case report and review of the literature. Int J Gynecol Cancer 2005; 15:1213-7. [PMID: 16343217 DOI: 10.1111/j.1525-1438.2005.00190.x] [Citation(s) in RCA: 23] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022] Open
Abstract
Benign metastasizing leiomyoma (BML) is an unusual condition typically treated by surgical resection and hormonal therapy. A 37-year-old woman presented with dyspnea 6 years after uterine myomectomy. Computed tomographic scans showed pelvic and right-sided lung masses. Total abdominal hysterectomy and right salpingo-oophorectomy revealed only leiomyomas and benign ovarian cysts. Thoracotomy revealed multiple nodules consistent with leiomyomas. Estrogen and progesterone receptors on lung and uterine tissue were positive. A 21- x 18-mm residual area of neoplasm in the lung completely resolved 3 months after treatment with megestrol. The patient remains free of disease 3 years after lung resection. We present a rare case of BML in which the lung neoplasm responded to megestrol alone in the setting of intact ovarian function.
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Affiliation(s)
- G K Wentling
- Section of General Surgery, Mayo Clinic, Jacksonville, Florida 32224, USA
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31
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Pitts S, Oberstein EM, Glassberg MK. Benign metastasizing leiomyoma and lymphangioleiomyomatosis: sex-specific diseases? Clin Chest Med 2004; 25:343-60. [PMID: 15099894 DOI: 10.1016/j.ccm.2004.01.014] [Citation(s) in RCA: 32] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
The striking similarity between benign metastasizing leiomyoma and lymphangioleiomyomatosis is that they both affect young women. This observation underscores the importance of gender-dependent variables in disease and our lack of understanding of gender differences. Many researchers are currently investigating the link between disease and hormone levels in the body. The beneficial role of estrogens remains in question in pulmonary medicine, and further ongoing research should help to define the role of hormones in the lung.
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Affiliation(s)
- Shannon Pitts
- Department of Internal Medicine, University of Rochester School of Medicine, Strong Memorial Hospital, 6704 Setters Run, Rochester, NY 14564, USA
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32
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Säynäjäkängas O, Maiche AG, Liakka KA. Multiple progressive pulmonary leiomyomatous metastases treated with tamoxifen--a case report with a review of the literature. Acta Oncol 2004; 43:113-4. [PMID: 15068329 DOI: 10.1080/02841860310020159] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/26/2022]
Affiliation(s)
- O Säynäjäkängas
- Department of Pulmonary Diseases, Lapland Central Hospital, Oulu University, Finland.
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33
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Tucakovic M, Bascom R, Bascom PB. Pulmonary medicine and palliative care. Best Pract Res Clin Obstet Gynaecol 2001; 15:291-304. [PMID: 11358403 DOI: 10.1053/beog.2000.0169] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Gynaecological malignancies affect the respiratory system both directly and indirectly. Malignant pleural effusion is a poor prognostic factor: management options include repeated thoracentesis, chemical pleurodesis, symptomatic relief of dyspnoea with oxygen and morphine, and external drainage. Parenchymal metastases are typically multifocal and respond to chemotherapy, with a limited role for pulmonary metastatectomy. Pulmonary tumour embolism is frequently associated with lymphangitic carcinomatosis, and is most common in choriocarcinoma. Thromboembolic disease, associated with the hypercoagulable state of cancer, is treated with anticoagulation. Inferior vena cava filter placement is indicated when anticoagulation cannot be given, or when emboli recur despite adequate anticoagulation. Palliative care has a major role for respiratory symptoms of gynaecological malignancies. Treatable causes of dyspnoea include bronchospasm, fluid overload and retained secretions. Opiates are effective at relieving dyspnoea associated with effusions, metatases, and lymphangitic tumour spread. Non-pharmacological therapies include energy conservation, home redesign, and dyspnoea relief strategies, including pursed lip breathing, relaxation, oxygen, circulation of air with a fan, and attention to spiritual suffering. Identification and treatment of gastroesophageal reflux, sinusitis, and asthma can improve many patients' coughs. Chest wall pain responds to local radiotherapy, nerve blocks or systemic analgesia. Case examples illustrate ways to address quality of life issues.
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Affiliation(s)
- M Tucakovic
- Pulmonary, Allergy and Critical Care Medicine, the Department of Medicine, Penn State College of Medicine, The Milton S. Hershey Medical Center, Hershey, PA, USA
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