gastrointestinal tract duplications are uncommon congenital anomalies and found anywhere along the alimentary tract from the mouth to the anus .
almost all of the reported cases were detected with respiratory distress manifestation in the early days of life .
most eds are found in the distal third of the esophagus and are frequently incidental findings on routine chest radiography .
ed is divided into three types : cystic ( the most common type ) , tubular , and diverticular .
ed can be associated with other congenital anomalies such as small intestine duplications , esophageal atresia , bronchoesophageal fistulas ( befs ) , and spinal abnormalities .
less than 20% of eds have communication with the main lumen through a tubular tract .
communicating tubular ed is extremely rare , and only one case report of communicating tubular ed combined with bef in an adult has been published in the japanese literature ( in 1994 ) .
here , we report a case of communicating tubular ed combined with bef , diagnosed on the basis of endoscopy and esophagography findings during the postoperational evaluation of bef .
a 49-year - old male patient was admitted to our hospital s emergency center because of herbicide poisoning . after drinking several glasses of alcohol and herbicide , he presented with persistent nausea and vomiting .
he had been drinking about 54 g alcohol daily and had 50-pack - year history of cigarette smoking .
he had a history of pulmonary tuberculosis and received antituberculosis medication 8 years ago . at that time
on admission , initial physical examination revealed no specific finding except for an elevated respiratory rate of 28/min .
the results of laboratory tests were as follows : white blood cell count 15,660/mm , hemoglobin 12.9 g / dl , hematocrit 38.1% , platelet count 155,000/mm , total protein 7.9 g / dl , albumin 3.7 g / dl , aspartate aminotransferase 80 u / l , alanine aminotransferase 37 u / l , prothrombin time 1.03 , and c - reactive protein 3.48 mg / dl .
the results of blood gas analysis were ph 7.513 , pco2 35.2 mm hg , po2 80.5 mm hg , and hco3 27.7 mmol / l .
after aspiration pneumonia was diagnosed , the patient was treated with conservative management . on the second hospital day
on endoscopic and bronchoscopic examinations , a fistula opening was found at the mid - esophagus , and the opening had a whitish surface with a slightly screwed pattern without inflammatory sign or discharge ( fig .
we could not localize the other opening or a fistula tract . a previous esophagography image ( fig .
1b ) taken at a local hospital revealed a communicating fistula tract between the bronchus and the lower esophagus ( bef ) ; however , a chest computed tomography ( ct ) scan , also taken at the local hospital , did not show discrete bef . on the 6th hospital day ,
his pneumonic condition was well improved and he could ambulate . on the 10th hospital day
, he underwent surgical operation for the repair of the bef . during the operation ,
a 2-cm - long fistula tract between the lower esophagus and the medial basal segment of the left lung was noted , which was successfully removed and repaired .
his condition was stable , and he had no complication . on the 3rd postoperational day , a follow - up esophagography for the postoperational evaluation revealed contrast leakage at the left side of the mid - esophagus and drainage to the distal esophagus ( fig .
we thought that the leakage resulted from an unidentified condition , such as ed with esophago - esophageal fistula ( fig .
a proximal fistula opening was found at the mid - esophagus and the distal opening was found on the cardia of the stomach , located in the hiatal hernia ( fig .
an about 7-mm air - filled tract was noted , one end of which was connected to the distal esophagus and the other end was connected to the cardia of the stomach ( fig .
initially , he was operated for the symptomatic bef ; however , a communicating ed was later detected incidentally during a postoperational follow - up .
the symptoms of bef were improved without any unusual complication , and he wanted to be discharged without any further treatment or surgery . at 2 years of follow - up
the only case of a communicating tubular ed with bef was published in the japanese literature .
the patient was a 51-year - old woman who chronically experienced coughing when drinking water .
esophagoscopy revealed tubular ed with bef , and endoscopy showed one small opening of ed .
most eds are found in the distal third of the esophagus and are frequently asymptomatic ; however , dysphagia , respiratory distress , recurrent pneumonia , vomiting , failure to thrive , gastroesophageal reflux , melena , and anemia may also be present in rare cases . at the time of admission , our patient had chronic intermittent cough and knew about his bef .
pneumonia might be considered a complication of drug intoxication or bef . on admission , persistent cough developed abruptly ,
chest ct and endoscopic examination did not reveal a communicating ed at that time ; however , postoperational follow - up esophagography for the confirmation of fistula closure revealed another remaining fistula tract .
this fistula tract communicated not with the bronchus but with the distal esophagus as a tubular structure , which is an evidence of the possibility of ed .
therefore , endoscopic reexamination and follow - up chest ct were done , and we finally confirmed ed .
later , we incidentally found that he had a communicating tubular ed with bef , which we could not find on the initial evaluation .
the ed was initially not detected because we thought the cough symptom was caused by the bef that was detected on esophagography at the previous hospital .
the ed was congenitally accompanied by the bef , and they shared the same opening , which might have caused the relatively less influx of the contrast medium into the ed than into the bef , making the ed undetectable on esophagography . according to the assumption from interdisciplinary discussion , it is highly likely that the removal of the bef might have made the ed easily accessible for the contrast medium and thus more detectable on esophagography .
this is also supported by the fact that ed was suspected on the review of the previous ct images ( images not presented here ) .
one major limitation of this case report is that the possibility of an esophagogastric fistula , not an ed , could not be confirmed by histology . in another report with a similar case to ours ,
ed was verified on the basis of the histological findings . however , as the endoscopic findings suggest ( fig . 3b ) , the orifice inside the hiatal hernia appeared to be covered by the epithelium of the esophagus , suggesting the possibility of an orifice from the ed , rather than an esophagogastric fistula .
a histological examination would be helpful for differential diagnosis in patients showing similar findings in the future .
upper gastrointestinal endoscopy can help in the diagnosis through direct vision ; however , it can not determine whether the ed is a communicating or a noncommunicating type . in our case , the initial endoscopic examination for the confirmation of bef could not find a communicating tubular ed
. however , the result of the follow - up esophagography revealed the possibility of a communicating tubular ed , and we conducted repeat endoscopy carefully .
3 , the distal orifice of the ed was at the sliding hiatal hernia sac , and it was very difficult to find .
the opening could be seen along the whole esophagus as a whitish or pinkish hole not discriminated from the surrounding opening tissue .
esophagography may show displacement of the esophagus due to a paraesophageal mass ; however , a tubular ed may not influence the esophagus .
contrast esophagographic evaluation could be useful in diagnosing a communicating ed preoperatively , and to confirm that a leak is not present during the postoperative follow - up period .
ct could demonstrate the exact anatomic position of the fistular tract and influence the decision making about resectability .
magnetic resonance imaging , endoscopic ultrasonography , and tc-99 m pertechnetate scintigraphy could be helpful for the diagnosis of this disease . however , these methods yield mainly inconclusive results .
the management of ed is dependent on the type and size of the ed , and the severity of the symptoms .
complete resection is a well - documented treatment for duplication of the esophagus ; however , conservative management can be an option for nonsymptomatic patients .
symptomatic ed has mainly been managed with extensive surgery through a thoracotomy . with recent advances in minimally invasive surgery , surgical treatment
has improved in efficacy and offers advantages to patients . a clear exposure and identification of the duplication are important for a successful operation .
as our patient received primary repair of bef without knowledge of ed , and his symptoms disappeared , we did not conduct additional surgery for ed .
after surgery , he has been doing well during the follow - up period . in conclusion ,
identifying a communicating ed with bef is difficult . on the diagnostic examination for a bef ,
the possibility of ed should be considered and careful endoscopic examination is also important for the detection of fistula orifices .
 esophageal duplication ( ed ) is rarely diagnosed in adults and is usually asymptomatic . 
 especially , ed that is connected to the esophagus through a tubular communication and combined with bronchoesophageal fistula ( bef ) is extremely rare and has never been reported in the english literature . 
 this condition is very difficult to diagnose . 
 although some combinations of several modalities , such as upper gastrointestinal endoscopy , esophagography , computed tomography , magnetic resonance imaging , and endoscopic ultrasonography , can be used for the diagnosis , the results might be inconclusive . here 
 , we report on a patient with communicating tubular ed that was incidentally diagnosed on the basis of endoscopy and esophagography during the postoperational evaluation of bef . 
INTRODUCTION
CASE REPORT
DISCUSSION