Case Study
Cavernous hemangioma arising from the diaphragm
Asian Cardiovascular & Thoracic Annals 21(6) 735–738 ß The Author(s) 2012 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492312469361 aan.sagepub.com
Katsuhito Ueno, Eriho Takeuchi, Haruaki Hino and Mitsuaki Kawashima
Abstract A 51-year-old man was referred to our hospital with an abnormal nodule in the right lung field. Computed tomography revealed a homogeneous nodule adjacent to the diaphragm, which appeared to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected, and positron-emission tomography showed no significant uptake. At surgery, 2 red papillary tumors were found, originating from the right diaphragm, and tumor extirpation was performed. The pathological diagnosis was cavernous hemangioma.
Keywords Diaphragm, hemangioma, muscle neoplasms, thoracic surgical procedures
Introduction Primary diaphragmatic tumors are quite rare. Hemangioma commonly arises from soft tissue. Hemangioma arising from diaphragm is extremely rare.1 The description of a case of primary diaphragmatic hemangioma is presented in this report.
Case report A 51-year-old man suffering from reflux esophagitis and discomfort in the back of the throat was referred to our hospital because of an abnormal nodule in the right lower lung field. He had never smoked and had no exceptional medical history. Physical examination revealed no abnormal finding. Chest radiographs appeared normal, and computed tomography showed a homogeneous nodular opacity without calcification and an unclear border between the lesion and the diaphragm. The lesion measured 17 10 mm and was not opacified with contrast medium. On sagittal multiplanar reconstructed images, the tumor seemed to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected (Figure 1). Laboratory examinations detected no abnormalities, and serum tumor maker levels including carcinoembryonic antigen and cytokeratin 19 fragment were normal. Arterial blood gases on room air revealed pO2 of 97.3 torr,
pCO2 of 40.7 torr, pH 7.378, and oxygen saturation 97.4%. Spirometry indicated pulmonary function was well preserved. Positron-emission tomography demonstrated no significant uptake in the whole body. The patient underwent surgical resection. Two red papillary tumors originated from the right diaphragm and protruded into the thoracic cavity. There was no adhesion between the tumors and the lung. The vascularity around the tumors was increased (Figure 2(a)). Partial resection of the diaphragm was performed with staplers to achieve complete tumor extirpation, and the staple line was reinforced with running sutures. The tumor was cavernous and filled with blood (Figure 2(b)). Microscopic examination revealed that the wall of the tumor consisted of hyperplasia of the vessel, and included smooth muscle and fibrous tissue (Figure 3). Blood vessels in the muscle layer were dilated, and the intraluminal surface of the vessel was CD31-positive and D2-40-negative. The pathological Center for Pulmonary Diseases, Department of Thoracic Surgery, National Hospital Organization Tokyo National Hospital, Tokyo, Japan Corresponding author: Katsuhito Ueno, MD, PhD, Department of Thoracic Surgery, National Hospital Organization Tokyo National Hospital, 3-1-1,Takeoka, Kiyose City, Tokyo 204-8585, Japan. Email: [email protected]
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
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Figure 1. Preoperative computed tomography. Axial images revealed a homogeneous nodular opacity without calcification or a distinct border between the lesion and the diaphragm. The lesion measured 17 10 mm and was not opacified with contrast medium. On sagittal multiplanar reconstructed images, the tumor appeared to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected.
Figure 2. (a) Intraoperative view. The patient underwent surgical resection during which 2 red papillary tumors were found that appeared to have originated from the right diaphragm and subsequently protruded into the thoracic cavity. There was increased vascularity around the tumors. (b) Macroscopic view. Tumor extirpation was performed with mechanical staplers; it was cavernous and filled with blood.
diagnosis was cavernous hemangioma. The postoperative course was uneventful, and the patient was well 3 months after the operation.
Discussion Primary diaphragmatic tumors are rare, with fewer than 200 cases reported in the literature.2 Most patients were between 40 and 50 years of age at the time of
diagnosis, although the range is quite extensive. The ratio of men to women was found to be 1:1.1, and the incidence of right- versus left-sided lesions was equal.3 The ratio of benign to malignant tumors is approximately 1.6:1, and patients with benign diaphragmatic tumors usually have no symptoms.1 Hemangioma is one of the most common soft tissue tumors, constituting 7% of all benign tumors in the whole body. Only 9 cases of diaphragmatic
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
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Figure 3. Microscopic view (Hematoxylin and Eosin Stain). Microscopic examination revealed that the wall of the tumor consisted of the hyperplasia of the vessel and its wall included the smooth muscle and fibrous tissue. Blood vessels in the muscle layer were dilated, and the intraluminal surface of the vessel was CD31-positive and D2-40-negative.
hemangioma have been reported previously in the literature.1,4–11 Hemangiomas are classified into 3 types: capillary, cavernous, and venous. Capillary hemangiomas are the most common, and cavernous hemangiomas are less frequent than capillary hemangiomas, although they share common age and anatomic distributions.1 Radiologically, most diaphragmatic tumors appear as a smooth or lobulated soft tissue mass protruding into the inferior portion of the lung.4 Tumors arising in the region of the diaphragm most likely originate from an adjacent organ rather than the diaphragm itself. It is important to differentiate a primary diaphragmatic tumor from an invasive tumor emanating from an adjacent structure or a metastatic deposit from a distant neoplasm. However, the structure and position of the diaphragm make diagnostic imaging difficult. Multidetector-row computed tomography seems to be the best examination to differentiate a primary diaphragmatic tumor from a tumor originating from an adjacent organ. Because the diaphragm is difficult to visualize on cross-sectional imaging, axial images may be complemented by multiplanar reformatting to increase the diagnostic yield.12 Magnetic resonance imaging might be useful to establish the diagnosis, but it seems to be difficult to differentiate hemangioma from other vascular-rich tumors using this modality.1 Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflicts of interest statement None declared.
References 1. Kono R, Terasaki H, Fujimoto K, et al. Venous hemangioma arising from the diaphragm: a case report of computed tomography and magnetic resonance imaging findings. J Thorac Imaging 2006; 21: 231–234. 2. Weksler B and Ginsberg RJ. Tumors of the diaphragm [Review]. Chest Surg North Am 1998; 8: 441–447. 3. Olafsson G, Rausing A and Holen O. Primary tumors of the diaphragm. Chest 1971; 59: 568–570. 4. Ohsaki Y, Morimoto H, Osanai S, et al. Extensively calcified hemangioma of the diaphragm with increased 99mTchydroxymethylene diphosphonate uptake. Intern Med 2000; 39: 576–578. 5. Tsang FH, Lun KS and Cheng LC. Hemangioma of the diaphragm presenting with cardiac tamponade. J Card Surg 2011; 26: 620–623. 6. Ino H, Naitou M, Hato S, et al. A rare primary diaphragmatic hemangioma successfully treated by laparoscopic surgery: report of a case. Surg Today 2010; 40: 654–657. 7. Cacciaguerra S, Vasta G, Benedetto AG, et al. Neonatal diaphragmatic hemangioma. J Pediatr Surg 2001; 36: E21. 8. Kaniklides C and Dimopoulos PA. Diaphragmatic haemangioma. A case report. Acta Radiol 1999; 40: 329–332. 9. Saito H, Nomura F, Murate T, et al. Case of primary hemangioma of the diaphragm. Nihon Kyobu Shikkan Gakkai Zasshi 1983; 21: 587–590.
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
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10. Beppu M, Fujimoto K, Hikita K, Kurita K and Hirai T. Primary diaphragmatic hemangioma. Kyobu Geka 1977; 30: 801–805. 11. Timmermans M, Lagadec B, Poilleux J and Hivet M. Primary tumors of the diaphragm (apropos of 2 angiomas). Ann Chir 1971; 25: 1315–1324.
12. Limmer KK, Kernstine KH, Grannis Jr FW and Weiss LM. Diaphragmatic diseases, benign or malignant: overview. In: Sugarbaker DJ, Bueno R, Krasna MJ, Mentzer SJ, Zellos L (eds) Adult Chest Surgery, 1st edn. New York: McGraw-Hill Professional, 2009, pp. 1054–1067.
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
Cavernous hemangioma arising from the diaphragm
Asian Cardiovascular & Thoracic Annals 21(6) 735–738 ß The Author(s) 2012 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492312469361 aan.sagepub.com
Katsuhito Ueno, Eriho Takeuchi, Haruaki Hino and Mitsuaki Kawashima
Abstract A 51-year-old man was referred to our hospital with an abnormal nodule in the right lung field. Computed tomography revealed a homogeneous nodule adjacent to the diaphragm, which appeared to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected, and positron-emission tomography showed no significant uptake. At surgery, 2 red papillary tumors were found, originating from the right diaphragm, and tumor extirpation was performed. The pathological diagnosis was cavernous hemangioma.
Keywords Diaphragm, hemangioma, muscle neoplasms, thoracic surgical procedures
Introduction Primary diaphragmatic tumors are quite rare. Hemangioma commonly arises from soft tissue. Hemangioma arising from diaphragm is extremely rare.1 The description of a case of primary diaphragmatic hemangioma is presented in this report.
Case report A 51-year-old man suffering from reflux esophagitis and discomfort in the back of the throat was referred to our hospital because of an abnormal nodule in the right lower lung field. He had never smoked and had no exceptional medical history. Physical examination revealed no abnormal finding. Chest radiographs appeared normal, and computed tomography showed a homogeneous nodular opacity without calcification and an unclear border between the lesion and the diaphragm. The lesion measured 17 10 mm and was not opacified with contrast medium. On sagittal multiplanar reconstructed images, the tumor seemed to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected (Figure 1). Laboratory examinations detected no abnormalities, and serum tumor maker levels including carcinoembryonic antigen and cytokeratin 19 fragment were normal. Arterial blood gases on room air revealed pO2 of 97.3 torr,
pCO2 of 40.7 torr, pH 7.378, and oxygen saturation 97.4%. Spirometry indicated pulmonary function was well preserved. Positron-emission tomography demonstrated no significant uptake in the whole body. The patient underwent surgical resection. Two red papillary tumors originated from the right diaphragm and protruded into the thoracic cavity. There was no adhesion between the tumors and the lung. The vascularity around the tumors was increased (Figure 2(a)). Partial resection of the diaphragm was performed with staplers to achieve complete tumor extirpation, and the staple line was reinforced with running sutures. The tumor was cavernous and filled with blood (Figure 2(b)). Microscopic examination revealed that the wall of the tumor consisted of hyperplasia of the vessel, and included smooth muscle and fibrous tissue (Figure 3). Blood vessels in the muscle layer were dilated, and the intraluminal surface of the vessel was CD31-positive and D2-40-negative. The pathological Center for Pulmonary Diseases, Department of Thoracic Surgery, National Hospital Organization Tokyo National Hospital, Tokyo, Japan Corresponding author: Katsuhito Ueno, MD, PhD, Department of Thoracic Surgery, National Hospital Organization Tokyo National Hospital, 3-1-1,Takeoka, Kiyose City, Tokyo 204-8585, Japan. Email: [email protected]
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
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Asian Cardiovascular & Thoracic Annals 21(6)
Figure 1. Preoperative computed tomography. Axial images revealed a homogeneous nodular opacity without calcification or a distinct border between the lesion and the diaphragm. The lesion measured 17 10 mm and was not opacified with contrast medium. On sagittal multiplanar reconstructed images, the tumor appeared to be an extrapulmonary lesion. No hilar or mediastinal lymph node swelling was detected.
Figure 2. (a) Intraoperative view. The patient underwent surgical resection during which 2 red papillary tumors were found that appeared to have originated from the right diaphragm and subsequently protruded into the thoracic cavity. There was increased vascularity around the tumors. (b) Macroscopic view. Tumor extirpation was performed with mechanical staplers; it was cavernous and filled with blood.
diagnosis was cavernous hemangioma. The postoperative course was uneventful, and the patient was well 3 months after the operation.
Discussion Primary diaphragmatic tumors are rare, with fewer than 200 cases reported in the literature.2 Most patients were between 40 and 50 years of age at the time of
diagnosis, although the range is quite extensive. The ratio of men to women was found to be 1:1.1, and the incidence of right- versus left-sided lesions was equal.3 The ratio of benign to malignant tumors is approximately 1.6:1, and patients with benign diaphragmatic tumors usually have no symptoms.1 Hemangioma is one of the most common soft tissue tumors, constituting 7% of all benign tumors in the whole body. Only 9 cases of diaphragmatic
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
Ueno et al.
737
Figure 3. Microscopic view (Hematoxylin and Eosin Stain). Microscopic examination revealed that the wall of the tumor consisted of the hyperplasia of the vessel and its wall included the smooth muscle and fibrous tissue. Blood vessels in the muscle layer were dilated, and the intraluminal surface of the vessel was CD31-positive and D2-40-negative.
hemangioma have been reported previously in the literature.1,4–11 Hemangiomas are classified into 3 types: capillary, cavernous, and venous. Capillary hemangiomas are the most common, and cavernous hemangiomas are less frequent than capillary hemangiomas, although they share common age and anatomic distributions.1 Radiologically, most diaphragmatic tumors appear as a smooth or lobulated soft tissue mass protruding into the inferior portion of the lung.4 Tumors arising in the region of the diaphragm most likely originate from an adjacent organ rather than the diaphragm itself. It is important to differentiate a primary diaphragmatic tumor from an invasive tumor emanating from an adjacent structure or a metastatic deposit from a distant neoplasm. However, the structure and position of the diaphragm make diagnostic imaging difficult. Multidetector-row computed tomography seems to be the best examination to differentiate a primary diaphragmatic tumor from a tumor originating from an adjacent organ. Because the diaphragm is difficult to visualize on cross-sectional imaging, axial images may be complemented by multiplanar reformatting to increase the diagnostic yield.12 Magnetic resonance imaging might be useful to establish the diagnosis, but it seems to be difficult to differentiate hemangioma from other vascular-rich tumors using this modality.1 Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflicts of interest statement None declared.
References 1. Kono R, Terasaki H, Fujimoto K, et al. Venous hemangioma arising from the diaphragm: a case report of computed tomography and magnetic resonance imaging findings. J Thorac Imaging 2006; 21: 231–234. 2. Weksler B and Ginsberg RJ. Tumors of the diaphragm [Review]. Chest Surg North Am 1998; 8: 441–447. 3. Olafsson G, Rausing A and Holen O. Primary tumors of the diaphragm. Chest 1971; 59: 568–570. 4. Ohsaki Y, Morimoto H, Osanai S, et al. Extensively calcified hemangioma of the diaphragm with increased 99mTchydroxymethylene diphosphonate uptake. Intern Med 2000; 39: 576–578. 5. Tsang FH, Lun KS and Cheng LC. Hemangioma of the diaphragm presenting with cardiac tamponade. J Card Surg 2011; 26: 620–623. 6. Ino H, Naitou M, Hato S, et al. A rare primary diaphragmatic hemangioma successfully treated by laparoscopic surgery: report of a case. Surg Today 2010; 40: 654–657. 7. Cacciaguerra S, Vasta G, Benedetto AG, et al. Neonatal diaphragmatic hemangioma. J Pediatr Surg 2001; 36: E21. 8. Kaniklides C and Dimopoulos PA. Diaphragmatic haemangioma. A case report. Acta Radiol 1999; 40: 329–332. 9. Saito H, Nomura F, Murate T, et al. Case of primary hemangioma of the diaphragm. Nihon Kyobu Shikkan Gakkai Zasshi 1983; 21: 587–590.
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
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Asian Cardiovascular & Thoracic Annals 21(6)
10. Beppu M, Fujimoto K, Hikita K, Kurita K and Hirai T. Primary diaphragmatic hemangioma. Kyobu Geka 1977; 30: 801–805. 11. Timmermans M, Lagadec B, Poilleux J and Hivet M. Primary tumors of the diaphragm (apropos of 2 angiomas). Ann Chir 1971; 25: 1315–1324.
12. Limmer KK, Kernstine KH, Grannis Jr FW and Weiss LM. Diaphragmatic diseases, benign or malignant: overview. In: Sugarbaker DJ, Bueno R, Krasna MJ, Mentzer SJ, Zellos L (eds) Adult Chest Surgery, 1st edn. New York: McGraw-Hill Professional, 2009, pp. 1054–1067.
Downloaded from aan.sagepub.com at GEORGIAN COURT UNIV on March 22, 2015
