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5. Kilic D, Sahin E, Gulcan O, Bolat B, Turkoz R, Hatipoglu A. Octreotide for treating chylothorax after cardiac surgery. Tex Heart Inst J 2005;32:437–9. 6. Lim KA, Kim SH, Huh J, et al. Somatostatin for postoperative chylothorax after surgery for children with congenital heart disease. J Korean Med Sci 2005;20:947–51. 7. Grosman I, Simon D. Potential gastrointestinal uses of somatostatin and its synthetic analogue octreotide. Am J Gastroenterol 1990;85:1061–72. 8. Lai F, Chen L, Tu Y, Lin M, Li X. Prevention of chylothorax complicating extensive esophageal resection by mass ligation of thoracic duct: a random control study. Ann Thorac Surg 2011;91:1770–4.
Spontaneous Hemothorax Caused by Pulmonary Micro-Venous Hemangioma
Division of Thoracic Surgery, Joetsu General Hospital, Joetsu, Niigata; Departments of Diagnostic Pathology, and General Thoracic and Cardiovascular Surgery, Graduate School of Medicine and Pharmaceutical Sciences, University of Toyama, Toyama, Toyama; and Department of Anatomic and Diagnostic Pathology, Dokkyo Medical University, Mibu-machi, Tochigi, Japan
Various etiologies of spontaneous hemothorax have been reported, though the cause remains unidentified in some patients despite an exploratory thoracotomy. We report on an 89-year-old man with spontaneous hemothorax resolved by partial resection of the lung using complete video-assisted thoracoscopic surgery. The histopathologic findings revealed a ruptured micro-venous hemangioma located just below the bleeding visceral pleura. To the best of our knowledge, this is the first report of spontaneous hemothorax caused by a pulmonary micro-venous hemangioma. (Ann Thorac Surg 2015;100:299–301) Ó 2015 by The Society of Thoracic Surgeons
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An 89-year-old man was admitted to our hospital with general fatigue that developed in the morning. Decreased breathing sounds were heard on the left. Blood pressure was 124/70 mm Hg and pulse rate was regular at 80 beats per minute. There was no dementia or recall of any kind of trauma. Comorbidities included hypertension, hyperlipidemia, and arteriosclerosis obliterates. The patient had been taking ticlopidine and limaprost alfadex for 10 years for arteriosclerosis obliterates. There was a past history of right non-small cell lung cancer for which he underwent curative surgery (pathologic stage IA, negative surgical margin). There was no bleeding tendency in the operative findings and the course was excellent. A chest X-ray performed on the day of admission showed left pleural effusion (Fig 1). In addition, that had also been performed incidentally 5 days prior as a routine follow-up examination for the previous lung cancer, though no abnormal findings suggestive of left pleural effusion or recurrence were noted. A left thoracocentesis yielded bloody effusion, which confirmed the diagnosis of hemothorax, while cytology did not reveal any malignant findings. An enhanced computed tomography (CT) examination did not show the cause or bleeding site. White blood cell count was 9.4 109/L with a normal differential cell count, hemoglobin at 114 g/L with normal red cell indexes, and platelet count at 19.3 109/L. Bleeding time and clotting results were normal. We considered a wait and see approach because vital signs were stable. However, the following morning chest X-ray findings showed increasing effusion and a blood test revealed progressive anemia (hemoglobin at 94 g/L).
H
emothorax is a relatively common condition reported to be associated with trauma, iatrogenesis, coagulopathy, aortic dissection, pulmonary infarction, malignant neoplasm, or spontaneous pneumothorax [1, 2]. In contrast, a spontaneous hemothorax is rare and the etiology can remain unknown in some patients even after an exploratory thoracotomy [1, 3]. Here we present the first case of spontaneous hemothorax caused by a pulmonary micro-venous hemangioma.
Accepted for publication Aug 29, 2014. Address correspondence to Dr Homma, Division of Thoracic Surgery, Joetsu General Hospital, 148-1 Daido-Fukuda, Joetsu, Niigata 943-8507 Japan; e-mail:
[email protected].
Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
Fig 1. Chest X-ray image obtained on admission showing left pleural effusion. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.08.065
FEATURE ARTICLES
Takahiro Homma, MD, Yutaka Yamamoto, MD, Johji Imura, MD, PhD, Yoshinori Doki, MD, PhD, Naoki Yoshimura, MD, PhD, Kazutaka Senda, MD, PhD, Masayoshi Toge, MD, Toshihiro Ojima, MD, Yoshifumi Shimada, MD, and Nobuhide Masawa, MD, PhD
CASE REPORT HOMMA ET AL SPONTANEOUS HEMOTHORAX
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CASE REPORT HOMMA ET AL SPONTANEOUS HEMOTHORAX
Ann Thorac Surg 2015;100:299–301
results were revealed, we found no evidence of hemangioma in those images.
Comment
FEATURE ARTICLES
Fig 2. Photograph showing blood oozing from the visceral pleura of the upper segment (arrow).
Thus, we performed surgery at 20 hours after the admission using 3-port access video-assisted thoracoscopic surgery. Although several adhesions between the parietal pleura and visceral pleura were found in the thorax, no ruptured adhesion or obvious bleeding point was identified and no dissemination was observed. Blood clots were evacuated using a sucker, after which we could identify blood oozing from the visceral pleura of the upper segment (Fig 2). We resected that portion of the lung using an endoscopic stapler (Endo GIA; United States Surgical Corporation, Norwalk, CT). No bleeding was observed on the opposite side of the parietal pleura or along the staple lines. The total amount of bleeding was 1,150 mL. The postoperative course was uneventful and excellent, and the patient was discharged 6 days after surgery. A histopathologic examination revealed dilated engorged neoplastic vessels located just below the bleeding visceral pleura, which was surrounded with a large quantity of red blood cells. This finding corresponded with micro-venous hemangioma and was identifiable as the cause of hemothorax (Fig 3A, 3B). No atypical cells or findings of pulmonary arteriovenous malformation were observed. Although we reviewed the CT scans after these Fig 3. (A) Histologic view showing a pulmonary venous hemangioma filled with a large amount of red blood cells (hematoxylin-eosin staining; magnification 20). (B) Histologic view showing an abnormally dilated, thin-walled pulmonary vein (immunostaining with factor VIII-related antigen; magnification 20).
Spontaneous hemothorax is uncommon and often conservatively treated [1, 3]. In some patients, no source of hemorrhage can be identified and no etiology determined despite an exploratory thoracotomy. Although hemothorax associated with antithrombotic therapy has been reported, no description of the underlying histopathologic conditions has been presented [4–7]. This is the first case of spontaneous hemothorax caused by a ruptured pulmonary micro-venous hemangioma. Antithrombotic therapy may have been a factor related to the bleeding in our patient. Such therapy is known to cause bleeding complications, with an intracerebral hemorrhage the most critical. A leading hypothesis to explain the etiology of intracerebral hemorrhage is intracerebral micro-hemangioma. Wakai and colleagues [8] reported that some cases have an angiographically occult vascular malformation. Immunostaining with factor VIII-related antigen is very useful for identification of micro-hemangioma in a histopathologic examination, as shown in the present case. Although intracerebral hemorrhage and hemothorax are different in regard to systemic and pulmonary circulation, and the blood-brain barrier, they have 2 interesting similarities; microhemangioma as the etiology and onset during antithrombotic therapy. As for why the etiology of spontaneous hemothorax remains unknown even after an exploratory thoracotomy, we consider difficulty with identification of the bleeding point and a pathologic examination without immunostaining as 2 possible reasons. In the present case, identification of the bleeding spot was difficult because of its small size with oozing. Thoracoscopic observation, which is multidirectional and provides a magnified view, enabled identification in our patient. In summary, we report the first case of spontaneous hemothorax caused by a pulmonary micro-venous hemangioma, which should be considered as an etiologic factor when enhanced CT does not reveal a cause or bleeding site. Careful thoracoscopic observation and
Ann Thorac Surg 2015;100:301–2
CASE REPORT BRICHON ET AL GIANT PURELY POLYPOID ESOPHAGEAL LEIOMYOMA
immunostaining with factor VIII-related antigen were helpful for diagnosis in our case.
References
A Giant Purely Polypoid Esophageal Leiomyoma
muscularis mucosae and should not be confused with a giant fibrovascular polyp that is postulated to arise at the pharyngoesophageal junction and may cause asphyxia when protruding into the mouth. A 62-year-old woman had symptoms of dysphagia for several months. Endoscopy showed a huge polypoid mass, probably pedunculated from the left side of the esophageal wall (Fig 1). Endoscopic biopsies were suggestive of leiomyoma. A chest computed tomographic scan confirmed that it was a pure polypoid tumor of 13-cm length (Fig 2). An attempt at endoscopic removal was unsuccessful. A right posterolateral thoracotomy was performed in the fifth interspace and the azygos vein was divided. The esophageal wall was distended without external tumor. A 10-cm right esophagomyotomy gave access to a tumor pedunculated along 5 cm of the left side. After complete resection, the left wall was closed by a 2-layer (muscular and then mucosa) running absorbable suture. The right opening was then closed with a 3-layer running absorbable suture (mucosal, muscular, and pleural). Postoperative follow-up was uneventful and endoscopy was normal at 6 months. Pathologic examination revealed fusiform cells from a large (14 7 5 cm) leiomyoma originating from the submucosal layer (Fig 3) and partially covered by the mucosa.
Comment
Pierre-Yves Brichon, MD, Marie-H elene Laverriere, MD, Sebastien Guigard, MD, and Philippe Chaffanjon, MD ˇ
Departments of Thoracic Surgery and Pathology, Hopital Universitaire de Grenoble, Grenoble, France
We present a case of a giant (13-cm length) purely polypoid esophageal leiomyoma without any intramural development. This form of leiomyoma is rare and develops due to proliferation originating from the muscularis mucosae, although the intramural type originates in the muscularis propria. This should not be confused with giant fibrovascular polyps, which are postulated to arise at the pharyngoesophageal junction when a flap of mobile, redundant submucosa prolapses distally and may cause asphyxia when protruding into the mouth. Our case was successfully removed by a right thoracotomy. (Ann Thorac Surg 2015;100:301–2) Ó 2015 by The Society of Thoracic Surgeons
Leiomyoma is the most common benign tumor of the esophagus and most of them are intramural [1]. Polypoid leiomyomas are very rare. Four cases were reported by Bona and colleagues [2] as originating from the gastroesophageal junction and protruding into the stomach cavity. All 4 (4 to 12 cm long) were submucosally dissected with a gastroscopic diathermic electrosurgical knife and then removed. Operative time was rather long
E
sophageal leiomyomas are usually intramural and originate in the muscularis propria. The purely polypoid type is less frequent and originates from the
Accepted for publication Sept 9, 2014. Address correspondence to Dr Brichon, Department of Thoracic Surgery, Hopital Universitaire A. Michallon, La Tronche, 38700 France; e-mail:
[email protected]. ˇ
Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
Fig 1. Endoscopy showing the head of the polyp. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.09.064
FEATURE ARTICLES
1. Yung CM, Bessen SC, Hingorani V, Clements NC Jr, Caruso AA. Idiopathic hemothorax. Chest 1993;103:638–9. 2. Homma T, Sugiyama S, Kotoh K, Doki Y, Tsuda M, Misaki T. Early surgery for treatment of spontaneous hemopneumothorax. Scand J Surg 2009;98:160–3. 3. Chen CY, Hsu CL, Chang CH, Chen KY, Yu CJ, Yang PC. Hemothorax in a medical intensive care unit: incidence, comorbidity and prognostic factors. J Formos Med Assoc 2010;109:574–81. 4. Rostand RA, Feldman RL, Block ER. Massive hemothorax complicating heparin anticoagulation for pulmonary embolus. South Med J 1977;70:1128–30. 5. Ganguli A, Walker L, FitzGerald RJ, Pirmohamed M. Spontaneous hemothorax following anticoagulation with lowmolecular-weight heparin. Ann Pharmacother 2009;43:1528–31. 6. Wang CC, Ng CJ, Seak CK, Seak CJ. Spontaneous hemothorax following thrombolytic and anticoagulant therapy for massive pulmonary embolism. Am J Emerg Med 2013;31:999. € Pamukc¸u G€ 7. Do gan NO1, unaydın G, Tekin M, Cevik Y. Nontraumatic massive spontaneous hemothorax with concomitant warfarin use. Case Rep Emerg Med 2013;2013:546024. 8. Wakai S, Kumakura N, Nagai M. Lobar intracerebral hemorrhage. A clinical, radiographic, and pathological study of 29 consecutive operated cases with negative angiography. J Neurosurg 1992;76:231–8.
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