Jpn J Radiol (2014) 32:53–57 DOI 10.1007/s11604-013-0266-9
CASE REPORT
Difficulty in complete transarterial embolization for pulmonary benign metastasizing leiomyoma with massive hemoptysis Masaya Miyazaki • Akie Nakayama Daichi Noda • Yasunobu Maehara • Yoshito Tsushima
•
Received: 2 July 2013 / Accepted: 11 November 2013 / Published online: 25 November 2013 Ó Japan Radiological Society 2013
Abstract A 43-year-old woman suffering from massive hemoptysis from pulmonary benign metastasizing leiomyoma (BML) at the left lung hilum underwent bronchial and nonbronchial transarterial embolization (TAE) using gelatin sponge particles and n-butyl 2-cyanoacrylate during three interventional procedures. However, since complete embolization of the tumor was difficult despite decreased tumor size, the tumor was surgically resected 3 months after the last interventional procedure. This case demonstrates the difficulty of complete TAE for pulmonary BML because of its hypervascularity and the rich communications between bronchial and nonbronchial anastomotic arteries.
lung. Most patients are asymptomatic, though some have symptoms such as cough, chest pain, and dyspnea [1, 2]. However, massive hemoptysis caused by BML has not been reported in the literature. Bronchial and nonbronchial transarterial embolization (TAE) is a well-accepted procedure in the management of hemoptysis [3, 4]. To the best of our knowledge, there have been no reports of TAE for BML presenting as massive hemoptysis. An extremely rare case of massive hemoptysis from pulmonary BML treated by TAE, in which complete embolization of the tumor was difficult despite decreased tumor size, is presented.
Keywords Bronchial arterial embolization Pulmonary benign metastasizing leiomyoma n-Butyl 2-cyanoacrylate Gelatin sponge particles
Case report
Introduction Benign metastasizing leiomyomatosis (BML) is a rare phenomenon of women, most commonly affecting the M. Miyazaki (&) Y. Tsushima Department of Diagnostic and Interventional Radiology, Gunma University Hospital, 3-39-15 Showa-machi, Maebashi, Gunma 371-8511, Japan e-mail: [email protected] A. Nakayama Y. Maehara Department of Radiology, Tomioka General Hospital, 2073-1 Tomioka, Tomioka, Gunma 370-2316, Japan D. Noda Department of Surgery, Tomioka General Hospital, 2073-1 Tomioka, Tomioka, Gunma 370-2316, Japan
A 43-year-old woman with massive hemoptysis was admitted to our hospital. She had undergone a total abdominal hysterectomy for uterine leiomyoma at the age of 35 years. She had been receiving hormonal treatment for a 6-year history of pulmonary BML. Contrast-enhanced computed tomography (CT) of the chest was performed immediately, and multiple lung nodules were observed. The largest mass (5 cm) at the left lung hilum had surrounding ground-glass opacity, and enhanced vessels were seen in the mass lesion (Fig. 1a). Laboratory tests revealed mild anemia with a hemoglobin level of 11.5 g/dl; her hemoglobin level was usually about 14 g/dl. Bleeding from the largest mass was suspected as the cause of hemoptysis. Since active bleeding was observed and the size of the mass was relatively large and adjacent to the left pulmonary artery, a TAE rather than surgical resection was selected as a therapy for this patient by consensus between interventional radiologists and thoracic surgeons at a case presentation conference.
123
54
Fig. 1 a Contrast-enhanced CT of the chest shows a mass (5 cm) at the left lung hilum with surrounding ground-glass opacity. Enhanced vessels were seen in the mass. The mass was adjacent to the left pulmonary artery (arrow). b Left bronchial arteriography shows a hypervascular tumor at the left lung hilum, and the tumor was embolized by gelatin sponge (GS) particles via the microcatheter inserted into the distal portion of the cephalic (not shown) and caudal branch (arrow) of the left bronchial artery. Tumor enhancement disappeared after the injection of GS particles. c The second procedure was performed 5 months later. Left subclavian arteriography shows that the tumor was fed by a branch of the left internal thoracic artery. The microcatheter was inserted to the distal portion of the branch artery (arrow); GS particles were injected until the tumor enhancement disappeared
After obtaining informed consent from the patient, a TAE procedure was performed. On bronchial arteriography, a hypervascular tumor fed by a left bronchial artery (BA) was observed at the left lung hilum (Fig. 1b). A 2-F microcatheter (Sniper 2 l7: Terumo Clinical Supply, Tokyo, Japan) was inserted into the distal portion of the left
123
Jpn J Radiol (2014) 32:53–57
BA (Fig. 1b), and 2-mm gelatin sponge (GS) particles (Gelfoam: Pfizer Japan, Tokyo, Japan), cut by scissors, were injected until tumor enhancement disappeared. Twenty days after the procedure, the patient was discharged because the hemoptysis had resolved completely. However, the patient was re-admitted because of hemoptysis 5 months later. At the second interventional radiology procedure, the left hilar tumor was not fed by the left BA; instead, the tumor was fed by a branch of the left internal thoracic artery (ITA) (Fig. 1c). A microcatheter was inserted into the distal portion of the branch artery (Fig. 1c), and 2-mm GS particles were injected until the tumor enhancement disappeared. One week after the procedure, the dilated vessel in the tumor was still observed on enhanced CT. The possibility of recurrent hemoptysis was suspected because of inadequate embolization. A third TAE procedure using n-butyl 2-cyanoacrylate (NBCA: Histoacryl, B. Braun AESCULAP Japan, Tokyo, Japan) as the embolic material was attempted. Using NBCA for this patient was approved by the safety committee of our institution because NBCA had not yet been approved for use as an embolic material via a catheter in our country. First, it was confirmed that tumor enhancement was not observed on pulmonary arteriography. Then, the branch of the left ITA was selected using a microcatheter. The tumor was enhancing from the branch artery, and the drainage vein of the tumor was observed immediately after the injection of the contrast material (Fig. 2a). Then, 1.5 ml of a mixture of NBCA and lipiodol (Guerbet Japan, Tokyo, Japan) (NBCA:lipiodol = 1:3) was injected from the branch artery (Fig. 2b), and the tumor enhancement disappeared after the injection on the left internal thoracic arteriography (Fig. 2b). However, a thin collateral artery feeding the dilated tumor vessel was observed from the left superior intercostal artery (SIA) (Fig. 2c). Therefore, the collateral artery arising from the left SIA was selected and arteriography obtained, and we carefully checked that the spinal cord artery did not arise from the collateral artery. Then, 2.0 ml NBCA/lipiodol (NBCA:lipiodol = 1:5) was injected from the collateral artery (Fig. 2c). After the injection, the tumor enhancement disappeared on the superior intercostal arteriography and aortography (Fig. 2d), and hyperattenuating NBCA/lipiodol deposition and small air density in the dilated vessel were observed in the tumor on the plain CT obtained immediately after the procedure (Fig. 3 left). Based on the CT imaging, it appeared that the tumor had been completely embolized. However, an enhanced CT scan performed 1 week after the procedure revealed that the tumor drug deposition had disappeared, and revascularization of the dilated vessel in the tumor was observed (Fig. 3 right). Alternatively, the tumor had decreased in size (3 cm) compared with the initial CT image. Two months after the third TAE
Jpn J Radiol (2014) 32:53–57
Fig. 2 A third procedure using NBCA as an embolization material was attempted. a Left internal thoracic arteriography shows revascularization of the tumor, and the drainage vein (arrow) of the tumor was observed immediately after the injection of contrast material from the branch artery. b NBCA/lipiodol (1:3) was injected from the branch artery of the internal thoracic artery, and the tumor enhancement
55
disappeared after the procedure. c Left superior intercostal arteriography shows that the thin collateral artery fed the dilated tumor vessel, and NBCA/lipiodol (1:5) was injected via the microcatheter (arrow) inserted into the branch artery of the superior intercostal artery. d Left superior intercostal arteriography and aortography show that the tumor enhancement had completely disappeared
Fig. 3 Left Plain CT performed immediately after the procedure shows that hyperattenuating NBCA/lipiodol deposition and small air density in the dilated vessel were observed in the tumor (white arrow). Right However, 1 week after the third procedure, the tumor drug deposition had disappeared, and revascularization of the dilated vessel was observed in the tumor (white arrowhead). The tumor had decreased in size (3 cm) compared with the initial CT image (Fig. 1a)
procedure, the patient was re-admitted because of hemoptysis, and the thoracic surgeon thought that the tumor should be resected because complete embolization would be difficult using a TAE procedure. Three months after the third TAE procedure, the left lung hilar tumor had been resected with the left upper lung lobe. The tumor was diagnosed pathologically as a pulmonary BML (Fig. 4). The patient has had no episodes of hemoptysis
over the 9 months of follow-up after the surgical resection.
Discussion BML is a rare entity of women aged 35–55 years, occurring years after hysterectomy. The most common site of
123
56
Fig. 4 Small fibrosis and necrosis are observed around the tumor vessel with thrombus (white arrow). Viable smooth muscle cells with high cellularity, minimal atypia, and inconspicuous mitosis are observed in most of the tumor. There is no NBCA/lipiodol deposition in the tumor. (H&E, 91)
metastasis is the lung, although cases with involvement of the lymph nodes, heart, skull, spine, and retroperitoneum have been reported. The lesions were incidentally found in most cases. Although there have been reports of patients with morbidity and mortality from BML, the clinical course is typically indolent [1, 2]. Massive hemoptysis is extremely rare [2], but when it occurs it may be serious in patients with pulmonary BML. No standard treatment guidelines for BML have been published; hence, the therapeutic strategies are largely empirical. Reported treatment modalities include careful observation, surgical resection, hysterectomy and bilateral oophorectomy, progestins, aromatase inhibitors, and medical castration using luteinizing hormone-releasing hormone analogs [5]. Hormonal treatment is a commonly chosen therapy for BML when estrogen and progesterone receptors are identified on tumor histology [1, 2]. However, massive hemoptysis is usually difficult to treat with hormonal treatment, and surgical resection of the tumor might be selected in such a situation. In the present case, TAE was attempted because the thoracic surgeon expected that resection of the tumor might be difficult because of the tumor location. Bronchial and nonbronchial TAE (BAE/nBAE) is well accepted and an established procedure in the management of massive and recurrent hemoptysis [3, 4]. Absorbable GS particles and PVA particles are the most frequently used materials for BAE/nBAE. The clinical success rate has been reported to range from 73 to 99 %, but recurrent bleeding has occurred in from 10 to 55 % of cases [3]. Recently, BAE/nBAE with NBCA has been applied for the treatment of bronchial aneurysm and hemoptysis [6–8].
123
Jpn J Radiol (2014) 32:53–57
According to Razavi et al. [7], recurrent hemoptysis was observed in 2 of 12 patients (17 %) after BAE/nBAE with NBCA for massive hemoptysis compared with 12 of 36 patients (33 %) after BAE/nBAE with PVA particles, but the complication rate was similar in both groups. To the best of our knowledge, there have been no reports of the TAE procedure for BML with massive hemoptysis. We suspect that TAE may become one of the treatment choices for BML with massive hemoptysis since TAE is less invasive than surgical resection. In the present case, we used GS particles in the first and second TAEs because GS particles were the most commonly used embolic material and PVA particles were not available in our country for commercial reasons. Therefore, we changed the embolic material from GS particles to NBCA after complete embolization could not be achieved in the first and second TAEs with GS. The BA usually has anastomoses to the surrounding nonbronchial arteries, such as the ITA and an intercostal artery [3, 9]. Since an anastomotic artery is usually \1 mm in diameter, we thought that GS particles were too large to reach the anastomotic artery and may have led to revascularization of the tumor from the anastomotic artery arising from the nonbronchial arteries. In addition, the drainage vein of the tumor was observed immediately after the injection of contrast material in the present case. Thus, NBCA was chosen because it can reach the more distal arteries more effectively than GS particles and can polymerize before reaching the drainage vein when an appropriate dilution ratio of lipiodol to NBCA is used in the mixture. According to Stoesslein [10], the polymerization time of NBCA can be controlled by adding lipiodol. We changed the NBCA/lipiodol mixture ratio from 1:3 for the ITA branch to 1:5 for the SIA branch, because the polymerization time of the 1:3 mixture ratio might be too early to reach the anastomotic artery. After the 1:5 mixed NBCA/lipiodol was injected, the tumor seemed to be completely embolized on the angiography and CT scan performed immediately after the procedure. However, complete embolization of the tumor was difficult even with NBCA in the present case, which was seen on the enhanced CT performed 1 week after the procedure. According to Ikoma et al. [6], NBCA was detected in the bronchial branch arteries, which ranged from 143 to 1,094 lm on pathological examination in a patient who underwent BAE with NBCA. Therefore, in the present case, we thought that NBCA could reach the anastomotic arteries and embolized the blood supply to the tumor once. However, the NBCA in the anastomotic arteries flowed out 1 week after the procedure because BML is a hypervascular tumor, and many strong blood supplies may have arisen immediately after the procedure. In conclusion, an extremely rare case of massive hemoptysis from pulmonary BML that was treated by TAE, in
Jpn J Radiol (2014) 32:53–57
which complete embolization of the tumor was difficult despite using NBCA, was presented. This case demonstrates the difficulty of complete TAE for pulmonary BML, possibly because of its extreme hypervascularity and the rich communications between the bronchial and nonbronchial anastomotic arteries. Acknowledgments The authors would like to thank Dr. Manabu Honma (Department of Pathology, Tomioka General Hospital) for his experienced review of slides in this case. Conflict of interest of interest.
The authors declare that they have no conflict
References 1. Ogawa M, Hara M, Ozawa Y, Moriyama S, Yano M, Shimizu S, et al. Benign metastasizing leiomyoma of the lung with malignant transformation mimicking mediastinal tumor. Clin Imaging. 2011;35:401–4. 2. Abramson S, Gilkeson RC, Goldstein JD, Woodard PK, Eisenberg R, Abramson N. Benign metastasizing leiomyoma: clinical, imaging, and pathologic correlation. AJR Am J Roentgenol. 2001;176:1409–13.
57 3. Sopko DR, Smith TP. Bronchial artery embolization for hemoptysis. Semin Intervent Radiol. 2011;28:48–62. 4. Mal H, Rullon I, Mellot F, Brugiere O, Sleiman C, Menu Y, et al. Immediate and long-term results of bronchial artery embolization for life-threatening hemoptysis. Chest. 1999;115:996–1001. 5. Yoon G, Kim TJ, Sung CO, Choi CH, Lee JW, Lee JH, et al. Benign metastasizing leiomyoma with multiple lymph node metastasis: a case report. Cancer Res Treat. 2011;43:131–3. 6. Ikoma A, Kawai N, Sato M, Tanaka T, Sonomura T, Sahara S, et al. Pathologic evaluation of damage to bronchial artery, bronchial wall, and pulmonary parenchyma after bronchial artery embolization with N-butyl cyanoacrylate for massive hemoptysis. J Vasc Interv Radiol. 2011;22:1212–5. 7. Razavi MK, Murphy K. Embolization of bronchial arteries with N-butyl cyanoacrylate for management of massive hemoptysis: a technical review. Tech Vasc Interv Radiol. 2007;10:276–82. 8. Aburano H, Kawamori Y, Horiti Y, Kitagawa K, Sanada J, Matsui O. Bronchial artery aneurysm embolization with NBCA. Cardiovasc Intervent Radiol. 2006;29:1141–3. 9. Yoon W, Kim JK, Kim YH, Chung TW, Kang HK. Bronchial and nonbronchial systemic artery embolization for life-threatening hemoptysis: a comprehensive review. Radiographics. 2002;22: 1395–409. 10. Stoesslein F, Ditscherlein G, Romaniuk PA. Experimental studies on new liquid embolization mixtures (histoacryl-lipiodol, histoacryl-panthopaque). Cardiovasc Intervent Radiol. 1982;5:264–7.
123
CASE REPORT
Difficulty in complete transarterial embolization for pulmonary benign metastasizing leiomyoma with massive hemoptysis Masaya Miyazaki • Akie Nakayama Daichi Noda • Yasunobu Maehara • Yoshito Tsushima
•
Received: 2 July 2013 / Accepted: 11 November 2013 / Published online: 25 November 2013 Ó Japan Radiological Society 2013
Abstract A 43-year-old woman suffering from massive hemoptysis from pulmonary benign metastasizing leiomyoma (BML) at the left lung hilum underwent bronchial and nonbronchial transarterial embolization (TAE) using gelatin sponge particles and n-butyl 2-cyanoacrylate during three interventional procedures. However, since complete embolization of the tumor was difficult despite decreased tumor size, the tumor was surgically resected 3 months after the last interventional procedure. This case demonstrates the difficulty of complete TAE for pulmonary BML because of its hypervascularity and the rich communications between bronchial and nonbronchial anastomotic arteries.
lung. Most patients are asymptomatic, though some have symptoms such as cough, chest pain, and dyspnea [1, 2]. However, massive hemoptysis caused by BML has not been reported in the literature. Bronchial and nonbronchial transarterial embolization (TAE) is a well-accepted procedure in the management of hemoptysis [3, 4]. To the best of our knowledge, there have been no reports of TAE for BML presenting as massive hemoptysis. An extremely rare case of massive hemoptysis from pulmonary BML treated by TAE, in which complete embolization of the tumor was difficult despite decreased tumor size, is presented.
Keywords Bronchial arterial embolization Pulmonary benign metastasizing leiomyoma n-Butyl 2-cyanoacrylate Gelatin sponge particles
Case report
Introduction Benign metastasizing leiomyomatosis (BML) is a rare phenomenon of women, most commonly affecting the M. Miyazaki (&) Y. Tsushima Department of Diagnostic and Interventional Radiology, Gunma University Hospital, 3-39-15 Showa-machi, Maebashi, Gunma 371-8511, Japan e-mail: [email protected] A. Nakayama Y. Maehara Department of Radiology, Tomioka General Hospital, 2073-1 Tomioka, Tomioka, Gunma 370-2316, Japan D. Noda Department of Surgery, Tomioka General Hospital, 2073-1 Tomioka, Tomioka, Gunma 370-2316, Japan
A 43-year-old woman with massive hemoptysis was admitted to our hospital. She had undergone a total abdominal hysterectomy for uterine leiomyoma at the age of 35 years. She had been receiving hormonal treatment for a 6-year history of pulmonary BML. Contrast-enhanced computed tomography (CT) of the chest was performed immediately, and multiple lung nodules were observed. The largest mass (5 cm) at the left lung hilum had surrounding ground-glass opacity, and enhanced vessels were seen in the mass lesion (Fig. 1a). Laboratory tests revealed mild anemia with a hemoglobin level of 11.5 g/dl; her hemoglobin level was usually about 14 g/dl. Bleeding from the largest mass was suspected as the cause of hemoptysis. Since active bleeding was observed and the size of the mass was relatively large and adjacent to the left pulmonary artery, a TAE rather than surgical resection was selected as a therapy for this patient by consensus between interventional radiologists and thoracic surgeons at a case presentation conference.
123
54
Fig. 1 a Contrast-enhanced CT of the chest shows a mass (5 cm) at the left lung hilum with surrounding ground-glass opacity. Enhanced vessels were seen in the mass. The mass was adjacent to the left pulmonary artery (arrow). b Left bronchial arteriography shows a hypervascular tumor at the left lung hilum, and the tumor was embolized by gelatin sponge (GS) particles via the microcatheter inserted into the distal portion of the cephalic (not shown) and caudal branch (arrow) of the left bronchial artery. Tumor enhancement disappeared after the injection of GS particles. c The second procedure was performed 5 months later. Left subclavian arteriography shows that the tumor was fed by a branch of the left internal thoracic artery. The microcatheter was inserted to the distal portion of the branch artery (arrow); GS particles were injected until the tumor enhancement disappeared
After obtaining informed consent from the patient, a TAE procedure was performed. On bronchial arteriography, a hypervascular tumor fed by a left bronchial artery (BA) was observed at the left lung hilum (Fig. 1b). A 2-F microcatheter (Sniper 2 l7: Terumo Clinical Supply, Tokyo, Japan) was inserted into the distal portion of the left
123
Jpn J Radiol (2014) 32:53–57
BA (Fig. 1b), and 2-mm gelatin sponge (GS) particles (Gelfoam: Pfizer Japan, Tokyo, Japan), cut by scissors, were injected until tumor enhancement disappeared. Twenty days after the procedure, the patient was discharged because the hemoptysis had resolved completely. However, the patient was re-admitted because of hemoptysis 5 months later. At the second interventional radiology procedure, the left hilar tumor was not fed by the left BA; instead, the tumor was fed by a branch of the left internal thoracic artery (ITA) (Fig. 1c). A microcatheter was inserted into the distal portion of the branch artery (Fig. 1c), and 2-mm GS particles were injected until the tumor enhancement disappeared. One week after the procedure, the dilated vessel in the tumor was still observed on enhanced CT. The possibility of recurrent hemoptysis was suspected because of inadequate embolization. A third TAE procedure using n-butyl 2-cyanoacrylate (NBCA: Histoacryl, B. Braun AESCULAP Japan, Tokyo, Japan) as the embolic material was attempted. Using NBCA for this patient was approved by the safety committee of our institution because NBCA had not yet been approved for use as an embolic material via a catheter in our country. First, it was confirmed that tumor enhancement was not observed on pulmonary arteriography. Then, the branch of the left ITA was selected using a microcatheter. The tumor was enhancing from the branch artery, and the drainage vein of the tumor was observed immediately after the injection of the contrast material (Fig. 2a). Then, 1.5 ml of a mixture of NBCA and lipiodol (Guerbet Japan, Tokyo, Japan) (NBCA:lipiodol = 1:3) was injected from the branch artery (Fig. 2b), and the tumor enhancement disappeared after the injection on the left internal thoracic arteriography (Fig. 2b). However, a thin collateral artery feeding the dilated tumor vessel was observed from the left superior intercostal artery (SIA) (Fig. 2c). Therefore, the collateral artery arising from the left SIA was selected and arteriography obtained, and we carefully checked that the spinal cord artery did not arise from the collateral artery. Then, 2.0 ml NBCA/lipiodol (NBCA:lipiodol = 1:5) was injected from the collateral artery (Fig. 2c). After the injection, the tumor enhancement disappeared on the superior intercostal arteriography and aortography (Fig. 2d), and hyperattenuating NBCA/lipiodol deposition and small air density in the dilated vessel were observed in the tumor on the plain CT obtained immediately after the procedure (Fig. 3 left). Based on the CT imaging, it appeared that the tumor had been completely embolized. However, an enhanced CT scan performed 1 week after the procedure revealed that the tumor drug deposition had disappeared, and revascularization of the dilated vessel in the tumor was observed (Fig. 3 right). Alternatively, the tumor had decreased in size (3 cm) compared with the initial CT image. Two months after the third TAE
Jpn J Radiol (2014) 32:53–57
Fig. 2 A third procedure using NBCA as an embolization material was attempted. a Left internal thoracic arteriography shows revascularization of the tumor, and the drainage vein (arrow) of the tumor was observed immediately after the injection of contrast material from the branch artery. b NBCA/lipiodol (1:3) was injected from the branch artery of the internal thoracic artery, and the tumor enhancement
55
disappeared after the procedure. c Left superior intercostal arteriography shows that the thin collateral artery fed the dilated tumor vessel, and NBCA/lipiodol (1:5) was injected via the microcatheter (arrow) inserted into the branch artery of the superior intercostal artery. d Left superior intercostal arteriography and aortography show that the tumor enhancement had completely disappeared
Fig. 3 Left Plain CT performed immediately after the procedure shows that hyperattenuating NBCA/lipiodol deposition and small air density in the dilated vessel were observed in the tumor (white arrow). Right However, 1 week after the third procedure, the tumor drug deposition had disappeared, and revascularization of the dilated vessel was observed in the tumor (white arrowhead). The tumor had decreased in size (3 cm) compared with the initial CT image (Fig. 1a)
procedure, the patient was re-admitted because of hemoptysis, and the thoracic surgeon thought that the tumor should be resected because complete embolization would be difficult using a TAE procedure. Three months after the third TAE procedure, the left lung hilar tumor had been resected with the left upper lung lobe. The tumor was diagnosed pathologically as a pulmonary BML (Fig. 4). The patient has had no episodes of hemoptysis
over the 9 months of follow-up after the surgical resection.
Discussion BML is a rare entity of women aged 35–55 years, occurring years after hysterectomy. The most common site of
123
56
Fig. 4 Small fibrosis and necrosis are observed around the tumor vessel with thrombus (white arrow). Viable smooth muscle cells with high cellularity, minimal atypia, and inconspicuous mitosis are observed in most of the tumor. There is no NBCA/lipiodol deposition in the tumor. (H&E, 91)
metastasis is the lung, although cases with involvement of the lymph nodes, heart, skull, spine, and retroperitoneum have been reported. The lesions were incidentally found in most cases. Although there have been reports of patients with morbidity and mortality from BML, the clinical course is typically indolent [1, 2]. Massive hemoptysis is extremely rare [2], but when it occurs it may be serious in patients with pulmonary BML. No standard treatment guidelines for BML have been published; hence, the therapeutic strategies are largely empirical. Reported treatment modalities include careful observation, surgical resection, hysterectomy and bilateral oophorectomy, progestins, aromatase inhibitors, and medical castration using luteinizing hormone-releasing hormone analogs [5]. Hormonal treatment is a commonly chosen therapy for BML when estrogen and progesterone receptors are identified on tumor histology [1, 2]. However, massive hemoptysis is usually difficult to treat with hormonal treatment, and surgical resection of the tumor might be selected in such a situation. In the present case, TAE was attempted because the thoracic surgeon expected that resection of the tumor might be difficult because of the tumor location. Bronchial and nonbronchial TAE (BAE/nBAE) is well accepted and an established procedure in the management of massive and recurrent hemoptysis [3, 4]. Absorbable GS particles and PVA particles are the most frequently used materials for BAE/nBAE. The clinical success rate has been reported to range from 73 to 99 %, but recurrent bleeding has occurred in from 10 to 55 % of cases [3]. Recently, BAE/nBAE with NBCA has been applied for the treatment of bronchial aneurysm and hemoptysis [6–8].
123
Jpn J Radiol (2014) 32:53–57
According to Razavi et al. [7], recurrent hemoptysis was observed in 2 of 12 patients (17 %) after BAE/nBAE with NBCA for massive hemoptysis compared with 12 of 36 patients (33 %) after BAE/nBAE with PVA particles, but the complication rate was similar in both groups. To the best of our knowledge, there have been no reports of the TAE procedure for BML with massive hemoptysis. We suspect that TAE may become one of the treatment choices for BML with massive hemoptysis since TAE is less invasive than surgical resection. In the present case, we used GS particles in the first and second TAEs because GS particles were the most commonly used embolic material and PVA particles were not available in our country for commercial reasons. Therefore, we changed the embolic material from GS particles to NBCA after complete embolization could not be achieved in the first and second TAEs with GS. The BA usually has anastomoses to the surrounding nonbronchial arteries, such as the ITA and an intercostal artery [3, 9]. Since an anastomotic artery is usually \1 mm in diameter, we thought that GS particles were too large to reach the anastomotic artery and may have led to revascularization of the tumor from the anastomotic artery arising from the nonbronchial arteries. In addition, the drainage vein of the tumor was observed immediately after the injection of contrast material in the present case. Thus, NBCA was chosen because it can reach the more distal arteries more effectively than GS particles and can polymerize before reaching the drainage vein when an appropriate dilution ratio of lipiodol to NBCA is used in the mixture. According to Stoesslein [10], the polymerization time of NBCA can be controlled by adding lipiodol. We changed the NBCA/lipiodol mixture ratio from 1:3 for the ITA branch to 1:5 for the SIA branch, because the polymerization time of the 1:3 mixture ratio might be too early to reach the anastomotic artery. After the 1:5 mixed NBCA/lipiodol was injected, the tumor seemed to be completely embolized on the angiography and CT scan performed immediately after the procedure. However, complete embolization of the tumor was difficult even with NBCA in the present case, which was seen on the enhanced CT performed 1 week after the procedure. According to Ikoma et al. [6], NBCA was detected in the bronchial branch arteries, which ranged from 143 to 1,094 lm on pathological examination in a patient who underwent BAE with NBCA. Therefore, in the present case, we thought that NBCA could reach the anastomotic arteries and embolized the blood supply to the tumor once. However, the NBCA in the anastomotic arteries flowed out 1 week after the procedure because BML is a hypervascular tumor, and many strong blood supplies may have arisen immediately after the procedure. In conclusion, an extremely rare case of massive hemoptysis from pulmonary BML that was treated by TAE, in
Jpn J Radiol (2014) 32:53–57
which complete embolization of the tumor was difficult despite using NBCA, was presented. This case demonstrates the difficulty of complete TAE for pulmonary BML, possibly because of its extreme hypervascularity and the rich communications between the bronchial and nonbronchial anastomotic arteries. Acknowledgments The authors would like to thank Dr. Manabu Honma (Department of Pathology, Tomioka General Hospital) for his experienced review of slides in this case. Conflict of interest of interest.
The authors declare that they have no conflict
References 1. Ogawa M, Hara M, Ozawa Y, Moriyama S, Yano M, Shimizu S, et al. Benign metastasizing leiomyoma of the lung with malignant transformation mimicking mediastinal tumor. Clin Imaging. 2011;35:401–4. 2. Abramson S, Gilkeson RC, Goldstein JD, Woodard PK, Eisenberg R, Abramson N. Benign metastasizing leiomyoma: clinical, imaging, and pathologic correlation. AJR Am J Roentgenol. 2001;176:1409–13.
57 3. Sopko DR, Smith TP. Bronchial artery embolization for hemoptysis. Semin Intervent Radiol. 2011;28:48–62. 4. Mal H, Rullon I, Mellot F, Brugiere O, Sleiman C, Menu Y, et al. Immediate and long-term results of bronchial artery embolization for life-threatening hemoptysis. Chest. 1999;115:996–1001. 5. Yoon G, Kim TJ, Sung CO, Choi CH, Lee JW, Lee JH, et al. Benign metastasizing leiomyoma with multiple lymph node metastasis: a case report. Cancer Res Treat. 2011;43:131–3. 6. Ikoma A, Kawai N, Sato M, Tanaka T, Sonomura T, Sahara S, et al. Pathologic evaluation of damage to bronchial artery, bronchial wall, and pulmonary parenchyma after bronchial artery embolization with N-butyl cyanoacrylate for massive hemoptysis. J Vasc Interv Radiol. 2011;22:1212–5. 7. Razavi MK, Murphy K. Embolization of bronchial arteries with N-butyl cyanoacrylate for management of massive hemoptysis: a technical review. Tech Vasc Interv Radiol. 2007;10:276–82. 8. Aburano H, Kawamori Y, Horiti Y, Kitagawa K, Sanada J, Matsui O. Bronchial artery aneurysm embolization with NBCA. Cardiovasc Intervent Radiol. 2006;29:1141–3. 9. Yoon W, Kim JK, Kim YH, Chung TW, Kang HK. Bronchial and nonbronchial systemic artery embolization for life-threatening hemoptysis: a comprehensive review. Radiographics. 2002;22: 1395–409. 10. Stoesslein F, Ditscherlein G, Romaniuk PA. Experimental studies on new liquid embolization mixtures (histoacryl-lipiodol, histoacryl-panthopaque). Cardiovasc Intervent Radiol. 1982;5:264–7.
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