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Esophageal Cancer With Esophageal Duplication Cyst
Esophageal Cancer With Esophageal Duplication Cyst Zhi-Jun Dai, MD, PhD, Hua-Feng Kang, MD, Shuai Lin, MD, Ming-Hua Bai, MD, Li Ma, MD, Wei-Li Min, MD, Wang-Feng Lu, MD, and Xi-Jing Wang, MD Departments of Oncology and Pathology, the Second Affiliated Hospital, Medical School of Xi’an Jiaotong University, Xi’an, China
Esophageal duplication cysts are benign, asymptomatic anomalies of foregut formation. We report a case of esophageal duplication cyst with esophageal squamous cancer. An upper endoscopy visualized with esophageal scan disclosed a stenotic lesion in the lower esophagus. Computed tomography images revealed a cystic mass in the inferior mediastinum, which was on the right wall of the esophagus. The postoperative pathology report confirmed the diagnosis of esophageal squamous cancer (ulcer type) and esophageal duplication cyst with calcification. (Ann Thorac Surg 2013;96:e15– 6) © 2013 by The Society of Thoracic Surgeons
E
sophageal duplication cyst is a rare benign, congenital cystic mass. Most cases are discovered in children. In adults, almost all of the patients are asymptomatic, and the cysts are diagnosed accidentally during evaluation for other clinical problems. They are frequently located on the right side in the lower third of the esophagus [1]. Respiratory symptoms may be caused by proximal cysts, and distal cysts can cause dysphagia. Malignant degeneration of esophageal duplication is a very rare event [1–3]. We report an interesting case of an esophageal duplication cyst with esophageal cancer in a 61-year-old man with difficulty in eating. A 61-year-old man with dysphagia to both liquids and solids for 2 months was referred for operation. The patient had lost 14 pounds over a 2-month period. His medical history was unremarkable. The results of physical examination were normal. Laboratory examination revealed elevated serum carcinoembryonic antigen (4.42 ng/mL) and cancer antigen-199 (42.72 U/mL). Upper endoscopy visualized with esophageal scanning disclosed a stenotic lesion in the lower esophagus (Fig 1). Computed tomography was performed to better assess the stenotic lesion, and it revealed a cystic mass in the inferior mediastinum, which was on the right wall of the esophagus (Fig 2). Because of the development of symptoms and the results of examination, the diagnosis of esophageal cancer and esophageal duplication cyst was considered but needed to be confirmed. The patient underwent a transthoracic surgical operation. This surgical approach was transthoracic through the left side of the seventh rib bed, open inferior pulmonary ligament to the level of the aortic arch. We could touch a lump about 5 cm ⫻ 5 cm on the wall of the proximal esophagus, surrounded by numerous swelling lymph nodes. Addi-
Accepted for publication Jan 4, 2013. Address correspondence to Dr Wang, Department of Oncology, the Second Affiliated Hospital, Medical School of Xi’an Jiaotong University, Xi’an 710004, China; e-mail: [email protected].
© 2013 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Esophagogram view showing a stenotic lesion in the lower esophagus.
tionally, another lump was found on the esophagus in the right thoracic cavity, which is a part of the esophagus. The lesions were removed completely, and the pathology report confirmed the diagnosis of esophageal squamous cancer (ulcer type) and esophageal duplication cyst with calcification (Fig 3). During the next 12 months of follow-up we observed significant improvement, and the patient had a normal clinical course.
Fig 2. Computed tomography images demonstrating a round cystic mass 5.0 cm ⫻ 4.0 cm in the inferior mediastinum, on the right side of the esophagus (arrow). The esophageal lumen is compressed and thickened. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.01.019
e16
CASE REPORT DAI ET AL ESOPHAGEAL CANCER WITH ESOPHAGEAL DUPLICATION CYST
Ann Thorac Surg 2013;96:e15– 6
Fig 3. (A) Round deflated unilocular cystic mass. (B) Esophageal squamous cancer (ulcer type) in cross-section, esophageal cyst on reverse side. (C) Pseudostratified ciliate mucosa with occasional submucosal glands in the inner layer of the esophageal cyst. (Hematoxylin-phloxine-saffron, ⫻200.) (D) Esophageal squamous cancer. (Hematoxylin-phloxine-saffron, ⫻200.)
Comment The first description of esophageal duplication, in 1711, is credited to Blasius [1]. Although esophageal duplicationconstitutes only 0.5% to 2.5% of all esophageal tumors [4], it is the second most common duplication of the alimentary tract after ileal duplication. The complications include obstruction, infection, hemorrhage, malignancy, or mediastinitis caused by rupture. Esophageal duplication cysts are rare anomalies resulting from a foregut budding error during the third to the sixth week of embryonic development [5]. Several imaging techniques have been used in the diagnosis of esophageal duplication, such as chest roentgenology, computed tomography, magnetic resonance imaging, and barium examination [6]. The preferred treatment for esophageal duplication cysts is complete surgical resection [7]. the location of cysts determined our surgical approach (transthoracic or transabdominal). Cysts above the lower third of the esophagus may not be accessible to the laparoscopic approach. Now, in most cases, a minimally invasive approach, such as thoracoscopy and laparoscopy, is preferred because it provides excellent exposure, a short time in the hospital, minimal postoperative discomfort, and fast recovery. Although malignancy of esophageal cysts is extremely rare, we needed to pay it enough attention. Adenocarcinoma is the most common histologic characteristic of
malignant transformation [8]. In this case, no direct relationship could be found between esophageal cancer and esophageal duplication cyst, through the adjacent tissues and pathology report, But it is rare for both diseases occur simultaneously.
References 1. Arbona JL, Fazzi JG, Mayoral J. Congenital esophageal cysts: case report and review of literature. Am J Gastroenterol 1984;79:177– 82. 2. Lee MY, Jensen E, Kwak S, Larson RA. Metastatic adenocarcinoma arising in a congenital foregut cyst of the esophagus: a case report with review of the literature. Am J Clin Oncol 1998;21:64 – 6. 3. Singh S, Lal P, Sikora SS, Datta NR. Squamous cell carcinoma arising from a congenital duplication cyst of the esophagus in a young adult. Dis Esophagus 2001;14:258 – 61. 4. Whitaker JA, Deffenbaugh LD, Cooke AR. Esophageal duplication cyst. Case report. Am J Gastroenterol 1980;73:329 –32. 5. Cohen SR, Geller KA, Birns JW, Thompson JW, Meyer BW, Lindesmith GG. Foregut cysts in infants and children. Diagnosis and management. Ann Otol Rhinol Laryngol 1982;91: 622–7. 6. Jeung MY, Gasser B, Gangi A, et al. Imaging of cystic masses of the mediastinum. Radiographics 2002;22:S79 –93. 7. Hazelrigg SR, Landreneau RJ, Mack MJ, Acuff TE. Thoracoscopic resection of mediastinal cysts. Ann Thorac Surg 1993; 56:659 – 60. 8. Jacob R, Hawkes ND, Dallimore N, Butchart EG, Thomas GA, Maughan TS. Case report: Squamous carcinoma in an oesophageal foregut cyst. Br J Radiol 2003;76:343– 6.
Esophageal duplication cysts are benign, asymptomatic anomalies of foregut formation. We report a case of esophageal duplication cyst with esophageal squamous cancer. An upper endoscopy visualized with esophageal scan disclosed a stenotic lesion in the lower esophagus. Computed tomography images revealed a cystic mass in the inferior mediastinum, which was on the right wall of the esophagus. The postoperative pathology report confirmed the diagnosis of esophageal squamous cancer (ulcer type) and esophageal duplication cyst with calcification. (Ann Thorac Surg 2013;96:e15– 6) © 2013 by The Society of Thoracic Surgeons
E
sophageal duplication cyst is a rare benign, congenital cystic mass. Most cases are discovered in children. In adults, almost all of the patients are asymptomatic, and the cysts are diagnosed accidentally during evaluation for other clinical problems. They are frequently located on the right side in the lower third of the esophagus [1]. Respiratory symptoms may be caused by proximal cysts, and distal cysts can cause dysphagia. Malignant degeneration of esophageal duplication is a very rare event [1–3]. We report an interesting case of an esophageal duplication cyst with esophageal cancer in a 61-year-old man with difficulty in eating. A 61-year-old man with dysphagia to both liquids and solids for 2 months was referred for operation. The patient had lost 14 pounds over a 2-month period. His medical history was unremarkable. The results of physical examination were normal. Laboratory examination revealed elevated serum carcinoembryonic antigen (4.42 ng/mL) and cancer antigen-199 (42.72 U/mL). Upper endoscopy visualized with esophageal scanning disclosed a stenotic lesion in the lower esophagus (Fig 1). Computed tomography was performed to better assess the stenotic lesion, and it revealed a cystic mass in the inferior mediastinum, which was on the right wall of the esophagus (Fig 2). Because of the development of symptoms and the results of examination, the diagnosis of esophageal cancer and esophageal duplication cyst was considered but needed to be confirmed. The patient underwent a transthoracic surgical operation. This surgical approach was transthoracic through the left side of the seventh rib bed, open inferior pulmonary ligament to the level of the aortic arch. We could touch a lump about 5 cm ⫻ 5 cm on the wall of the proximal esophagus, surrounded by numerous swelling lymph nodes. Addi-
Accepted for publication Jan 4, 2013. Address correspondence to Dr Wang, Department of Oncology, the Second Affiliated Hospital, Medical School of Xi’an Jiaotong University, Xi’an 710004, China; e-mail: [email protected].
© 2013 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Esophagogram view showing a stenotic lesion in the lower esophagus.
tionally, another lump was found on the esophagus in the right thoracic cavity, which is a part of the esophagus. The lesions were removed completely, and the pathology report confirmed the diagnosis of esophageal squamous cancer (ulcer type) and esophageal duplication cyst with calcification (Fig 3). During the next 12 months of follow-up we observed significant improvement, and the patient had a normal clinical course.
Fig 2. Computed tomography images demonstrating a round cystic mass 5.0 cm ⫻ 4.0 cm in the inferior mediastinum, on the right side of the esophagus (arrow). The esophageal lumen is compressed and thickened. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.01.019
e16
CASE REPORT DAI ET AL ESOPHAGEAL CANCER WITH ESOPHAGEAL DUPLICATION CYST
Ann Thorac Surg 2013;96:e15– 6
Fig 3. (A) Round deflated unilocular cystic mass. (B) Esophageal squamous cancer (ulcer type) in cross-section, esophageal cyst on reverse side. (C) Pseudostratified ciliate mucosa with occasional submucosal glands in the inner layer of the esophageal cyst. (Hematoxylin-phloxine-saffron, ⫻200.) (D) Esophageal squamous cancer. (Hematoxylin-phloxine-saffron, ⫻200.)
Comment The first description of esophageal duplication, in 1711, is credited to Blasius [1]. Although esophageal duplicationconstitutes only 0.5% to 2.5% of all esophageal tumors [4], it is the second most common duplication of the alimentary tract after ileal duplication. The complications include obstruction, infection, hemorrhage, malignancy, or mediastinitis caused by rupture. Esophageal duplication cysts are rare anomalies resulting from a foregut budding error during the third to the sixth week of embryonic development [5]. Several imaging techniques have been used in the diagnosis of esophageal duplication, such as chest roentgenology, computed tomography, magnetic resonance imaging, and barium examination [6]. The preferred treatment for esophageal duplication cysts is complete surgical resection [7]. the location of cysts determined our surgical approach (transthoracic or transabdominal). Cysts above the lower third of the esophagus may not be accessible to the laparoscopic approach. Now, in most cases, a minimally invasive approach, such as thoracoscopy and laparoscopy, is preferred because it provides excellent exposure, a short time in the hospital, minimal postoperative discomfort, and fast recovery. Although malignancy of esophageal cysts is extremely rare, we needed to pay it enough attention. Adenocarcinoma is the most common histologic characteristic of
malignant transformation [8]. In this case, no direct relationship could be found between esophageal cancer and esophageal duplication cyst, through the adjacent tissues and pathology report, But it is rare for both diseases occur simultaneously.
References 1. Arbona JL, Fazzi JG, Mayoral J. Congenital esophageal cysts: case report and review of literature. Am J Gastroenterol 1984;79:177– 82. 2. Lee MY, Jensen E, Kwak S, Larson RA. Metastatic adenocarcinoma arising in a congenital foregut cyst of the esophagus: a case report with review of the literature. Am J Clin Oncol 1998;21:64 – 6. 3. Singh S, Lal P, Sikora SS, Datta NR. Squamous cell carcinoma arising from a congenital duplication cyst of the esophagus in a young adult. Dis Esophagus 2001;14:258 – 61. 4. Whitaker JA, Deffenbaugh LD, Cooke AR. Esophageal duplication cyst. Case report. Am J Gastroenterol 1980;73:329 –32. 5. Cohen SR, Geller KA, Birns JW, Thompson JW, Meyer BW, Lindesmith GG. Foregut cysts in infants and children. Diagnosis and management. Ann Otol Rhinol Laryngol 1982;91: 622–7. 6. Jeung MY, Gasser B, Gangi A, et al. Imaging of cystic masses of the mediastinum. Radiographics 2002;22:S79 –93. 7. Hazelrigg SR, Landreneau RJ, Mack MJ, Acuff TE. Thoracoscopic resection of mediastinal cysts. Ann Thorac Surg 1993; 56:659 – 60. 8. Jacob R, Hawkes ND, Dallimore N, Butchart EG, Thomas GA, Maughan TS. Case report: Squamous carcinoma in an oesophageal foregut cyst. Br J Radiol 2003;76:343– 6.