CAPT. WILLIAM S. MOSKOVITZ, MC, USA, LT. COL. CARL W. HUGHES, MC, USA AND COL. WARNER F. BOWERS, MC, USA, Honolulu, Hawaii
From the Department of Surgery and General Surgery Service, Tripler U. S. Army Hospital, Honolulu, Hawaii.
Army HospitaI on October 27, 1958, for diagnostic studies because of recurrent episodes of pneumonitis. He was born at an Army station hospital on December 15, 1956. The mother was eighteen years oId, gravida I, para o. Her pregnancy was uneventfu1 and deIivery was precipitous. The patient’s birth weight was 6 pounds, 4 ounces. Excessive salivation and abdominal distention were noted. X-ray IiIms of the chest reveaIed ateIectasis and pneumonia of the upper lobe of the right lung, and a IipiodaI swaIIow as we11 as a barium enema demonstrated no abnormaIities. The infant had then been transferred to a genera1 hospita1 where a diagnosis of “H” type tracheoesophagea1 IistuIa was made. At the age of six days, the child underwent thoracotomy with repair of the “H” type tracheoesophageal fistuIa. He had an uneventfu1 postoperative course unti1 twenty days after surgery when he vomited, became dyspneic and had recurrence of abdomina1 distention. X-ray fiIms were obtained which reveaIed pneumonia of the upper Iobe of the right Iung and after two to three days of administration of antibiotics his condition improved. SubsequentIy, there were repeated episodes of coughing, vomiting and abdominaI distention at ten- to fourteen-day intervaIs. AI1 of these responded to treatment of antibiotics, intravenous fluids and aspiration. The possibiIity of a recurrent tracheoesophagea1 IistuIa was considered but a11 studies, including esophagograms and bronchoscopy, revealed no abnormaIities. Because of recurrent attacks of vomiting, dyspnea and cyanosis, gastrostomy was performed on February 22, 1957. Subsequent feedings were uneventfu1, and except for one episode of cyanosis, flaccidity and puIse eIevation to 200 which occurred on March 12, and responded to digitaIis therapy, the child had no further episodes of coughing, vomiting and abdomina1 distention. Laboratory tests for mucoviscidosis were negative. On ApriI 2 the gastrostomy was cIosed, and on May 6 the child was discharged to his parents’ care. He was admitted to a university hospita1 on JuIy 3 with a twenty-four-hour history of cough, vomiting and fever. His weight was 4.38 kg. PhysicaI examination reveaIed the previous surgica1 scars; respiratory rate, 45 per minute; puIse, 150
Iiterature of recent years has contained numerous reports of successfu1 surgica1 repair of tracheoesophagea1 f%stuIa. The majority of cases are associated with esophagea1 atresia, onIy four patients in Gross’  series not having associated atresia (“H” type of tracheoesophagea1 fistuIa). There have been very few cases of recurrent tracheoesophagea1 fistuIa foIIowing initia1 repair and in many of the Iarger series, this compIication is not reported. The few cases reported were recognized in the earIy postoperative period and apparentIy a striking cIinica1 change in the patient Iead to recognition of the condition. Lyon and Johnson  in 1948 reported a recurrent fistuIa occurring on the fourth postoperative day. Bigger [I] in 1949 reported on one chiId who died seven months after initia1 surgery for recurrent fistuIa. Potts’  series of thirty-five cases reported in 1950 made no mention of In Gross’  extensive recurrent fistuIa. experience, recurrence was never mentioned aIthough he states that diffIcuIties in swaIIowing, coughing and recurrent attacks of pneumonitis pIague this fIeId of surgery. HewIett  in 1955 reported a case of recurrent tracheoesophagea1 fistuIa which was successfuIIy repaired twentytwo and a haIf months after initia1 repair. At that time he found onIy four other cases of recurrent fistuIa in the Iiterature. FataIity rate foIIowing operation for recurrent fistuIa has been extremeIy high, approaching 50 per cent. The rarity of recurrence of tracheoesophagea1 fistuIa foIIowing primary repair Ieads us to report the foIIowing case of successful repair of a recurrent fistuIa twenty-three months after initia1 operation.
CASE REPORT A twenty-two month oId white maIe infant was admitted to the SurgicaI Service of TripIer U. S. American
Journal of Surgery,
returned to the SurgicaI Service for surgery. AIthough a defmite diagnosis of recurrent tracheoesophageal fistuIa could not be proved by x-ray studies or bronchoscopy, cIinicaIIy it was believed to be the cause of the recurrent attacks of pneumonia, and thoracotomy was performed on November 14. At operation, the chest was entered through the third interspace, excising the old scar. Dissection revealed the old suture Iine and a smaI1 fistulous tract at this Ievel between the esophagus and trachea. It was noted that as the anesthetist controhed respirations, the esophagus would distend with air much Iike inIIating a balloon. The Iistula was divided and the esophagus coIIapsed, remaining so. Openings of the tract were closed with No. 440 silk sutures in two layers, and mediastina1 pleura was placed so as to separate the two suture Iines. The lung then was reinhated, repair checked by pouring saline into the thoracic cavity and when air-tight repair was proved, the chest was closed with a multi-holed catheter in pIace for water-sea1 drainage. PostoperativeIy, intravenous feedings were maintained for forty-eight hours. At that time an esophagogram was performed which showed no evidence of spiIIage into the trachea. The patient was then started on fluids by mouth and progressed to a reguIar diet which he toIerated weII. Antibiotics were continued for seven days. The chest tube was removed on the sixth postoperative day and skin sutures on the seventh postoperative day. Repeat IiIms of the chest were taken which reveaIed that the chronic inflammatory process in the Iungs was cIearing. He was discharged on November 26 (tweIve days postoperativeIy), weighing 20 pounds. The patient was seen subsequentIy on December 5 and continued to do weI1. He then weighed 21 pounds which was the heaviest he had ever been. He was eating weI1, his appetite was tremendous and there were no further episodes of coughing and vomiting with his feedings. He has been folIowed up for more than one year postoperativeJy and has had no further respiratory diffrcultics.
per minute; rales throughout the lung heIds; and cyanosis. The remainder of the physica examination was within normal limits. X-ray IiIms were interpreted as showing an aspiration pneumonia. Treatment consisted of intravenous fluids, oxygen and antibiotics. Three esophagograms were performed and considered within normal Iimits. He was discharged on September 4, weighing 4.75 kg. On March 7, 1958, the child was First admitted to Tripler U. S. Army HospitaI Pediatric Service because of respiratory distress, cough, vomiting and fever. A diagnosis of interstitial pneumonitis was made on the basis of roentgenographic and physical findings. He responded to treatment with oxygen and antibiotics and was discharged on March 19. At this time, repeated x-ray films of the chest revcaIed persistent perihilar pneumonitis. The child’s weight on discharge was 16 pounds, 3 ounces. He was readmitted to TripIer U. S. Army HospitaI on May I 8 with similar history and physical findings, and after treatment for pneumonitis was discharged on May 22, weighing 16 pounds, 11 ounces. On May 25 he was admitted for the third time, and after pneumonitis had cleared, diagnostic studies were performed. Esophagogram revealed no abnormalities. Bronchography was performed with the patient under genera1 anesthesia on July 24 and, except for atelectasis of the middle Iobe of the right Iung, was within norma Iimits. There was no evidence of a tracheoesophageal IistuIa. Tests for mucoviscidosis were negative. The chiId was then discharged on JuIy 26 with a diagnosis of recurrent pneumonitis. He again was admitted on September 6 but was discharged the next day without any treatment. He was subsequentiy readmitted to the SurgicaI Service of the hospita1 on October 27 for evaIuation of recurrent pneumonitis. An esophagogram performed while an outpatient on September 26 showed spillage of the [email protected]
into the tracheobronchial tree but no t%tuIa couId be demonstrated. Except for excessive salivation and repeated episodes of coughing and vomiting, poor appetite and weight gain, the history was within normal limits. PhysicaI examination reveaIed no abnormalities except for previous surgica1 scars, malnutrition and coarse rhonchi throughout both lung fieIds. Laboratory data were norma except for hemoglobin, I 1.5 gm. per cent; and hematocrit, 38 per cent. On October 3 I bronchoscopy was performed with the child under genera1 anesthesia. No fistuIa couId be seen; however, excessive amounts of bronchial secretions were noted. Severe respiratory distress then deveIoped with cyanosis and substerna and intercostal retraction with breathing. He was started on antibiotics and oxygen and transferred to the Pediatric Service for treatment of pneumonitis. He responded to treatment and subsequentIy was
COMMENTS It is apparent by history that the patient had a recurrence of the fistuIa during his postoperative course. Although it was suspected, actuaI presence of the fistuIa couId not be demonstrated. SeveraI factors may Iead to the re-estabIishment of a fistuIa: (I) a leaking esophagea1 suture line with direct extension into the site of the trachea1 closure; (2) direct contact between the esophagea1 and trachea1 suture lines; (3) mechanica trauma resulting from early postoperative dilatation of the esophagus. The contiguous position of the suture Iines and a leak in the esophagea1 suture Iine appear III
to have been the factors present in the recurrent fistuIa reported herein. The true pathoIogic condition was masked because of the response of respiratory difhcuIties to antibiotics. AIthough antibiotics probabIy kept the chiId aIive, they aIso aided in substantiating the tentative diagnosis of recurrent pneumonitis. Repeated efforts to prove a tracheoesophageal fistula by x-ray studies and bronchoscopy were not successfu1, and it is understandable why it was beIieved that these were recurrent attacks of pneumonitis. It is interesting to specuIate whether the recurrent attacks of pneumonitis and diffrcuIty in degIutition, which Gross  states are common in patients with postoperative tracheoesophagea1 IistuIa, are not reaIIy due to recurrence of the IistuIa without associated esophageal stenosis. Many accurate descriptions have been written deaIing with the diagnosis of tracheoesophagea1 IistuIa with associated esophagea1 fistuIa. The existence of a fistula without esophageal stenosis or atresia is reIativeIy uncommon and may be overIooked. HoIt et a1.  in 1946 and Leigh et a1. [T] in 195 I wrote about the diffrcuIties in diagnosing tracheoesophagea1 IistuIa without esophagea1 stenosis or atresia. Some cases have masqueraded under the diagnosis of fauIty degIutition and recurrent pneumonitis. The authors have suggested the use of additiona x-ray studies, not generaIIy used in the evaIuation of patients with suspected tracheoesophagea1 IistuIa, in order to demonstrate the IistuIa. It seems justiIiabIe to incIude the studies in the diagnostic survey of any infant suffering repeated episodes of pneumonitis, especiahy in those with dilliculty following surgery for tracheoesophagea1 IistuIa. Literature on the operative technic for tracheoesophagea1 hstuIa has not stressed any
particmar point to prevent the recurrence of fistula. In the case presented herein, as we11 as that of HewIett [J], interposition of a mediastinal Aap between the suture Iines may have prevented recurrence. The use of the interposed mediastina1 ffap shouId be a routine part of the operative procedure for repair of tracheoesophagea1 hstula. SUMMARY
A case of recurrent tracheoesophagea1 fistuIa without associated esophagea1 stenosis is presented. Corrective surgery was performed twenty-three months after initia1 repair of a tracheoesophagea1 fistuIa. The rarity of the condition, the fact that it is amenabIe to surgery, and beIief that many cases of recurrent pneumonitis and fauIty degIutition foIIowing tracheoesophageal surgery in infants may well be due to smaI1 recurrent tracheoesophagea1 IistuIas without esophagea1 stenosis, have compeIIed us to report our case. REFERENCES I.
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