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.
301
302
CASE REPORT BULAM ET AL YOUNGEST ELASTOFIBROMA PATIENT
Ann Thorac Surg 2015;100:302–4
References 1. Aurea P, Grazia M, Petrella F, Bazzocchi R. Giant leiomyoma of the esophagus. Eur J Cardiothorac Surg 2002;22: 1008–10. 2. Bona D, Aiolfi A, Siboni S, Bernardi D, Bonavina L. Giant leiomyoma of the gastroesophageal junction: technique and results of endoscopic full-thickness resection. Clin Exp Gastroenterol 2011;4:263–7. 3. Koike Y, Obana T, Fujita N, et al. A case of giant esophageal leiomyoma successfully resected by endoscopic submucosal dissection followed by retrieval via laparotomy [Article in Japanese]. Nippon Shokakibyo Gakkai Zasshi 2010;107:1780–5. 4. Sweeney T. Giant fibrovascular polyp causing complete oesophageal obstruction. ANZ J Surg 2011;81:845–6.
FEATURE ARTICLES
Fig 2. Computed tomographic scan confirming a pure polypoid tumor of 13-cm length.
A 1-Year-Old Boy With Paraspinal Elastofibroma: The Youngest Diagnosed Elastofibroma Hakan Bulam, MD, Billur Sezgin, MD, Kemal Findikcioglu, MD, and Nesibe Cesur, MD
(from 150 to 220 minutes). We found in the literature only 1 case of polypoid leiomyoma of the middle esophagus, of 53-mm length and endoscopically removed [3]. As for our case, it derived, histologically, from the muscularis mucosae. This could explain the polypoid type, as intramural types derive from the muscularis propria. Polypoid esophageal leiomyoma should not be confused with giant fibrovascular polyps, which are postulated to arise at the pharyngoesophageal junction in the muscle-deficient Laimer-Haeckermann triangle when a flap of mobile, redundant submucosa prolapses distally [4]. These polyps may cause asphyxia when protruding into the mouth and can be removed by a left cervicotomy.
Department of Plastic Reconstructive and Aesthetic Surgery, Ankara Numune Training and Research Hospital, Ankara; Department of Plastic Reconstructive and Aesthetic Surgery, Erzurum Region Training and Research Hospital, Erzurum; Department of Plastic Reconstructive and Aesthetic Surgery, Gazi University School of Medicine, Ankara; and Department of Pathology, Gazi University School of Medicine, Ankara, Turkey
Elastofibroma is a benign and slow-growing soft tissue tumor that classically presents as an ill-defined solitary mass at the inferior pole of the scapula in the fifth and sixth decades of life for women. This tumor can be rarely seen in childhood, but it has not been reported in the infantile age group to date. To our knowledge, this case represents the first report of elastofibroma in the infantile age group. The aim of this report is to present a paravertebral elastofibroma in a 14-month-old patient and discuss the clinical features and treatment modalities. (Ann Thorac Surg 2015;100:302–4) Ó 2015 by The Society of Thoracic Surgeons
E
lastofibroma is a benign and slow-growing soft-tissue tumor that classically presents as an ill-defined solitary mass at the inferior pole of the scapula in the fifth and sixth decades of life for women. Signs and symptoms include swelling, discomfort, snapping, stiffness, and occasionally pain. As the tumor exhibits benign behavior, it should be surgically removed only in symptomatic patients. Elastofibroma is rare in childhood and has not been reported in the infantile age group to date. This report documents a paravertebral elastofibroma in a 14-month-old boy; the clinical features and treatment modalities are also discussed.
Fig 3. Pathologic specimen. Dense, fasciculated, bland, fusocellular proliferation in the submucosa (arrowhead), originating from the muscularis mucosae (arrow), covered by a normal squamous esophageal epithelium (hematoxylin-eosin-saffron; original magnification 5; [inset] intense cytoplasmic staining for desmin 20). Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
Accepted for publication July 30, 2014. Address correspondence to Dr Bulam, Bestepeler Mah. 31. Sok. No:5/7 Yenimahalle 06500, Ankara, Turkey; e-mail: [email protected].
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.07.086
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.
301
302
CASE REPORT BULAM ET AL YOUNGEST ELASTOFIBROMA PATIENT
Ann Thorac Surg 2015;100:302–4
References 1. Aurea P, Grazia M, Petrella F, Bazzocchi R. Giant leiomyoma of the esophagus. Eur J Cardiothorac Surg 2002;22: 1008–10. 2. Bona D, Aiolfi A, Siboni S, Bernardi D, Bonavina L. Giant leiomyoma of the gastroesophageal junction: technique and results of endoscopic full-thickness resection. Clin Exp Gastroenterol 2011;4:263–7. 3. Koike Y, Obana T, Fujita N, et al. A case of giant esophageal leiomyoma successfully resected by endoscopic submucosal dissection followed by retrieval via laparotomy [Article in Japanese]. Nippon Shokakibyo Gakkai Zasshi 2010;107:1780–5. 4. Sweeney T. Giant fibrovascular polyp causing complete oesophageal obstruction. ANZ J Surg 2011;81:845–6.
FEATURE ARTICLES
Fig 2. Computed tomographic scan confirming a pure polypoid tumor of 13-cm length.
A 1-Year-Old Boy With Paraspinal Elastofibroma: The Youngest Diagnosed Elastofibroma Hakan Bulam, MD, Billur Sezgin, MD, Kemal Findikcioglu, MD, and Nesibe Cesur, MD
(from 150 to 220 minutes). We found in the literature only 1 case of polypoid leiomyoma of the middle esophagus, of 53-mm length and endoscopically removed [3]. As for our case, it derived, histologically, from the muscularis mucosae. This could explain the polypoid type, as intramural types derive from the muscularis propria. Polypoid esophageal leiomyoma should not be confused with giant fibrovascular polyps, which are postulated to arise at the pharyngoesophageal junction in the muscle-deficient Laimer-Haeckermann triangle when a flap of mobile, redundant submucosa prolapses distally [4]. These polyps may cause asphyxia when protruding into the mouth and can be removed by a left cervicotomy.
Department of Plastic Reconstructive and Aesthetic Surgery, Ankara Numune Training and Research Hospital, Ankara; Department of Plastic Reconstructive and Aesthetic Surgery, Erzurum Region Training and Research Hospital, Erzurum; Department of Plastic Reconstructive and Aesthetic Surgery, Gazi University School of Medicine, Ankara; and Department of Pathology, Gazi University School of Medicine, Ankara, Turkey
Elastofibroma is a benign and slow-growing soft tissue tumor that classically presents as an ill-defined solitary mass at the inferior pole of the scapula in the fifth and sixth decades of life for women. This tumor can be rarely seen in childhood, but it has not been reported in the infantile age group to date. To our knowledge, this case represents the first report of elastofibroma in the infantile age group. The aim of this report is to present a paravertebral elastofibroma in a 14-month-old patient and discuss the clinical features and treatment modalities. (Ann Thorac Surg 2015;100:302–4) Ó 2015 by The Society of Thoracic Surgeons
E
lastofibroma is a benign and slow-growing soft-tissue tumor that classically presents as an ill-defined solitary mass at the inferior pole of the scapula in the fifth and sixth decades of life for women. Signs and symptoms include swelling, discomfort, snapping, stiffness, and occasionally pain. As the tumor exhibits benign behavior, it should be surgically removed only in symptomatic patients. Elastofibroma is rare in childhood and has not been reported in the infantile age group to date. This report documents a paravertebral elastofibroma in a 14-month-old boy; the clinical features and treatment modalities are also discussed.
Fig 3. Pathologic specimen. Dense, fasciculated, bland, fusocellular proliferation in the submucosa (arrowhead), originating from the muscularis mucosae (arrow), covered by a normal squamous esophageal epithelium (hematoxylin-eosin-saffron; original magnification 5; [inset] intense cytoplasmic staining for desmin 20). Ó 2015 by The Society of Thoracic Surgeons Published by Elsevier
Accepted for publication July 30, 2014. Address correspondence to Dr Bulam, Bestepeler Mah. 31. Sok. No:5/7 Yenimahalle 06500, Ankara, Turkey; e-mail: [email protected].
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2014.07.086
