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 ¤  Abstract
 ¤ Introduction
 ¤ Case Report
 ¤ Discussion
 ¤ Conclusion
 ¤  References
 ¤  Article Figures

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 Table of Contents     
UNUSUAL CASE
Year : 2018  |  Volume : 14  |  Issue : 2  |  Page : 158-160
 

A case report of cardia cancer complicated with idiopathic muscular hypertrophy of the oesophagus treated with thoracoscopic surgery


1 Department of General Surgery, Jiyang People's Hospital, Jinan, China
2 Department of Thoracic, The Second Hospital of Shandong University, Jinan, China

Date of Submission15-Aug-2017
Date of Acceptance29-Aug-2017
Date of Web Publication12-Mar-2018

Correspondence Address:
Dr. Chuanliang Peng
Department of Thoracic, The Second Hospital of Shandong University, 247 Beiyuan Street, Jinan 250033
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmas.JMAS_164_17

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 ¤ Abstract 

The incidence of idiopathic muscular hypertrophy of oesophagus (IMHE) is low, and <100 cases of IMHE have been reported. IMHE is a benign oesophageal disease, characterised by hyperplasia of all layers of the wall and in particular, muscle layer. Only a few cases have been reported regarding its clinical symptoms and images. In this present case, we report a cardia cancer with IMHE, showing significant hypertrophy of muscular layer of middle part of the oesophagus and successfully treated with minimally invasive thoracoscopic surgery.


Keywords: Cardia cancer, idiopathic muscular hypertrophy of esophagus, minimally invasive thoracoscopic surgery


How to cite this article:
Ren J, Hao Y, Peng C. A case report of cardia cancer complicated with idiopathic muscular hypertrophy of the oesophagus treated with thoracoscopic surgery. J Min Access Surg 2018;14:158-60

How to cite this URL:
Ren J, Hao Y, Peng C. A case report of cardia cancer complicated with idiopathic muscular hypertrophy of the oesophagus treated with thoracoscopic surgery. J Min Access Surg [serial online] 2018 [cited 2021 Sep 21];14:158-60. Available from: https://www.journalofmas.com/text.asp?2018/14/2/158/217063



 ¤ Introduction Top


The incidence of idiopathic muscular hypertrophy of oesophagus (IMHE) is low, the first case of IMHE was reported by Baillie in 1799.[1] Up to present, <100 cases of IMHE have been reported. IMHE is a benign oesophageal disease, characterised by hyperplasia of all layers of the wall and in particular, muscle layer. The aetiology of this disease is not clearly elucidated, most reported cases have been diagnosed at autopsy, and the final diagnosis was confirmed by pathological examination. Only a few cases have been reported regarding its clinical symptoms and images.

In this present case, we report a cardia cancer with IMHE, showing significant hypertrophy of muscular layer of middle part of the oesophagus, and successfully treated with minimally invasive thoracoscopic surgery.


 ¤ Case Report Top


A 66-year-old male was admitted to hospital with the symptoms of progressive dysphagia and weight loss of 10 pounds in 2 months. His medical and family histories were unremarkable, and the physical exam was non-contributory. Endoscopy revealed a non-circumferential mass 35 cm from the incisors. Biopsy demonstrated poorly differentiated adenocarcinoma. The barium meal examination showed that the upper oesophagus was dilated. Thoracic computed tomography (CT) demonstrated obvious thickening of oesophagus wall at middle and lower part [Figure 1]. Positron emission tomography-CT showed hypermetabolic activity at lower part of the oesophagus and no evidence of metastatic disease. From above analysis, we preoperatively diagnosed oesophageal or cardia cancer.
Figure 1: The oesophagography (a) showed contraction of oesophagus, computed tomography showed the thickened muscle layer (b)

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Surgery was performed under general anaesthesia; a double-lumen endotracheal intubation was used. Patients were placed in a supine position and underwent laparoscopy abdominal surgery with five abdominal ports. The 1-cm incision besides the umbilicus was used as the observation port, and the other four ports were positioned at the incisions in the subcostal region of the left and right midclavicular lines, and the incisions 3 cm superior to the umbilicus in the left and right parasternal lines. The stomach was routinely mobilised, the abdominal lymph node dissected, and the pedicled omental flap was reserved at the site close to the fundus of the stomach. Gastric conduit of about 4 cm in diameter was made using a linear stapler. Then he was positioned to the left lateral decubitus position and was anteverted moderately. Thoracoscopy was performed with four thoracic ports. A 1-cm incision was made at the 7th intercostal space on the posterior axillary line for observation, the operational ports located at the 4th intercostal space on the anterior axillary line, 6th and 9th intercostal space on the scapular line, respectively. The oesophagus was mobilised at the site 4 cm superior to the azygos vein, and thoracic lymph nodes were systematically dissected, including the left and right recurrent laryngeal nerve chain lymph nodes. The attached gastric conduit was pulled into the thoracic cavity through the diaphragmatic hiatus for intrathoracic anastomosis.[2]

On gross examination, the tumour located at the junction of the stomach and oesophagus (32 mm × 18 mm), and the oesophagus was invaded. Longitudinal section along the oesophagus showed the gradual increase in hypertrophy towards oral direction. Microscopic examination demonstrated poorly differentiated adenocarcinoma, hypertrophy of muscularis without degeneration of muscular layer and plexus, ganglionic cells [Figure 2]. The final diagnosis was made as cardia cancer complicated with IMHE.
Figure 2: Dissection of the oesophagus during thoracoscopic operation

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 ¤ Discussion Top


IMHE is rare benign disease, and only a few cases have been reported in English literature. Baillie described the first case in 1799, and later reviewed by Woods, Sloper and so on.[3],[4],[5] Up to present, <100 cases of IMHE could be found in English literature. Most were made diagnosis at post-mortem examination, and the reports describing symptoms and images have been very sparse. Hence, we hereby reported one case and reviewed the literature of IMHE.

Of the cases described, there is a predominance for this disease entity in male, and the aged ranged from 2 to 68. The clinical symptoms of IMHE were dysphagia, regurgitation, post-sternal pain, and early morning or nocturnal mucus-vomits. However, the presence of an oesophageal lesion in most cases was unsuspected until revealed at necropsy.[6] In this case, dysphagia may be caused by cardia tumour, not by the hypertrophy of the oesophagus. The oesophagography shows spasm, contraction and stenosis of the oesophagus. CT shows the thickened muscle layer and large nodules, firstly reported by Agostini et al.,[7] but large nodules were not found in the present case. The thickness was variable but always greater than normal. Strictly on a clinical and radiologic basis, this entity is impossible to definitively diagnose, imaging examination characteristic and preoperative gastroscopy are nonspecific. Diagnosis is made on histological results. In addition to cardia adenocarcinoma, the essential histological change included significant thickness of inner circular muscular layer without abnormal degeneration of muscle fibre. Sometimes there was also a lymphocytic infiltration of the intermuscular septa in the region of Auerbach's plexus. The treatment to IMHE seems to be myotomy. The indications of oesophagectomy have been already reported to include the following occasions: (1) accompanying lesion exists on the oesophagus besides IMHE; (2) myotomy was tried but not effective; (3) the malignant lesion cannot be ruled out. We resected the cardia tumour and thickened muscle together through minimally invasive approach.


 ¤ Conclusion Top


We have reported a relatively rare case of cardia cancer with IMHE. Preoperative diagnosis does not affect the surgical treatment, the final diagnosis was confirmed by pathological examination. The thoracoscopic approach is a good treatment option.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 ¤ References Top

1.
Shimada H, Kise Y, Chino O, Nishi T, Tanaka H, Kenmochi T, et al. Acase of superficial esophageal cancer complicated with idiopathic muscular hypertrophy of the esophagus. Tokai J Exp Clin Med 2003;28:103-8.  Back to cited text no. 1
    
2.
Ai B, Zhang Z, Liao Y. Laparoscopic and thoracoscopic esophagectomy with intrathoracic anastomosis for middle or lower esophageal carcinoma. J Thorac Dis 2014;6:1354-7.  Back to cited text no. 2
    
3.
Marston EL, Bradshaw HH. Idiopathic muscular hypertrophy of the esophagus. J Thorac Cardiovasc Surg 1959;38:248-52.  Back to cited text no. 3
    
4.
Peison B. Idiopathic muscular hypertrophy of the lower esophagus and pylorus in an adult. Chest 1971;59:682-7.  Back to cited text no. 4
    
5.
Zeller R, McLelland R, Myers B, Thompson WM. Idiopathic muscular hypertrophy of the esophagus: A case report. Gastrointest Radiol 1979;4:121-5.  Back to cited text no. 5
    
6.
Sloper JC. Idiopathic diffuse muscular hypertrophy of the lower oesophagus. Thorax 1954;9:136-46.  Back to cited text no. 6
    
7.
Agostini S, Grimaud JC, Salducci J, Clement JP. Idiopathic muscular hypertrophy of the esophagus: CT features. J Comput Assist Tomogr 1988;12:1041-3.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2]



 

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