THE ANNALS OF THORACIC SURGERY. Journal of The Society of Thoracic Surgeons and the Southern Thoracic Surgical Association

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1 THE ANNALS OF THORACIC SURGERY Journal of The Society of Thoracic Surgeons and the Southern Thoracic Surgical Association VOLUME 3 NUMBER 2 FEBRUARY 1967 Application of the Belsey Hiatal Hernia Repair to Infants and Children with Recurrent Bronchitis, Bronchiolitis, and Pneumonitis Due to Regurgitation and Aspiration * Milton V. Davis, M.D., and Javad Fiuzat, M.D. U pper and lower respiratory tract inflammations remain the most common medical problem in infants and children. They generally have been considered to be due to infection. Most respond well to treatment, but a few either fail to respond or have a significant number of recurrent attacks, and many fail to thrive. Gastroesophageal regurgitation is quite common in infants and has been considered to be normal by some. It appears that some of the children who regularly regurgitate gastric contents into the esophagus and pharynx will also aspirate gastric contents from the pharynx into the tracheobronchial tree [4, 6, 71. Apparently many who regurgitate From the Thoracic and Cardiovascular Surgery Service, Children s Medical Center, and the University of Texas Southwestern Medical School, Dallas, Tex. Presented at the Thirteenth Annual Meeting of the Southern Thoracic Surgical Association, Asheville, N.C., Nov. 3-5, *Address: 3707 Gaston Ave., Dallas, Tex VOL. 3, NO. 2, FEB.,

2 DAVIS AND FIUZAT either do not aspirate or do not develop subsequent bronchial or bronchopulmonary inflammation. This is a report of a total of 18 patients who were referred because of severe recurrent bronchitis, bronchiolitis, or pneumonitis. They were studied for gastroesophageal reflux, and 9 were operated upon. AGE AND SEX INCIDENCE The youngest patient in this group was 3 months of age when first seen in consultation, and the oldest was 12 years of age. The ages of those operated upon compared with the ages of those not operated upon were about the same. Approximately half were males and half were females in both the operative and nonoperative groups. Seven of the 9 patients in each group were 4 years of age or younger. SELECTION OF CASES The operative and nonoperative cases make up a highly selected group of patients. They were originally selected by their pediatricians or family doctors on the basis of either failure to thrive, recurrent broncho ulmonary inflammation, or both and were referred for study in most instances a F ter a great deal of treatment. The selection of cases for surgery was based on the severity of their clinical problem and whether or not definite gastroesophageal reflux could be demonstrated using the water test. This has been described by several authors [2, 3, 51. The first case was referred for lung biopsy because of severe recurrent pneumonitis. After careful study it was decided to do a simultaneous lung biopsy and Belsey hiatal hernia repair to determine whether prevention of gastroesophageal reflux could assist the patient. None of these patients had x-ray evidence of a hiatal hernia. PAST EXPERIENCE WITH THIS GROUP OF CASES In the past it has been customary to consider bronchography and bronchoscopy for children with recurrent bronchitis, bronchiolitis, and pneumonitis. These procedures have been carried out in numerous cases, usually without yielding a definite diagnosis other than bronchitis. SURGICAL TECHNIQUE The method of repair was patterned after the descriptions of Belsey [l, 41 and of Urschel and Paulson [71. It consisted basically of the following steps: 1. Posterolateral thoracotomy through the left sixth intercostal space. 2. Lung biopsy by subsegmental resection of the lateral basal segment of the left lower lobe, unless gross pneumonitis was found elsewhere. 3. Sharp dissection to free the distal esophagus from the mediastinal pleura and periesophageal areolar tissue up to the level of the inferior pulmonary vein or higher. 4. Sharp dissection to separate the esophagogastric junction from the hiatal ring. 5. Identification of both vagus nerves followed by suturing of the esophagus to the upper end of the phrenoesophageal ligament and to the stomach with approximately four interrupted sutures extending on the anterolateral left surface of the esophagogastric junction from one vagus nerve to the other. 6. The second and final layer of mattress sutures is added from the esophagus to the stomach to the peritoneal surface of diaphragm; then this suture layer is brought out on pleural surface of diaphragm. When this second row is tied down, it accomplishes the following: 100 THE ANNALS OF THORACIC SURGERY

3 Regurgitation in Children a. A considerable segment of the lower end of the esophagus is pulled down into the infradiaphragmatic (abdominal) position. b. The fundus of the stomach then comes to lie along the left and anterolateral surface of the esophagus, creating a sharp esophagofundic angle and a buttress. The fundus also forms a pouch. Positive pressure in this area tends to close the esophagogastric junction. c. The mattress sutures coming through the diaphragm tend to hold the esophagus and stomach in the proper anatomical position. 7. If the hiatal ring is large, and it usually is, one or two sutures are placed to pull the two limbs of the right crus together behind the esophagus. DATA ON INDIVIDUAL PATIENTS Tabulation of all previous treatment would be too detailed; however, mention of some of the salient points in several representative cases in each group will show the nature of the clinical problems being dealt with. CASE 1 (OPERATIVE GROUP) K. W. was seen at 3 months of age after having had four visits to the outpatient clinic and two hospital admissions for bilateral bronchopneumonia. When seen, she had been hospitalized for 30 days. She had failed to gain weight, had continued to cough, had bilateral rhonchi on auscultation of the lungs, and had x-ray evidence of persistent bilateral bronchopneumonia (Fig. IA). Cinefluorography revealed gastroesophageal reflux to be marked. The referring pediatrician had requested lung biopsy for histological, bacteriological, and virological study. Even though the patient did not have a hiatal hernia, the gastroesophageal junction was considered to be large (chalasia), and the insertion of the esophagus into the stomach was high on the lesser curvature. This resulted in a wide esophagofundic angle and relatively little esophagus below the diaphragm. It was decided to perform the lung biopsy and also to do a Belsey hiatal hernia repair in the hope of preventing gastroesophageal reflux and the recurrent pneumonitis. This operation was carried out in December, By request of the medicai staff, not because the child was having further trouble, she was seen five times in the next six months in the outpatient clinic. On each occasion she was found A FIG. 1. K. W., Case 1. (A) Preoperative chest x-ray. (B) Six weeks postoperative chest x-ray showing clearing lung fields and Belsey fundic air bubble. B VOL. 3, NO. 2, FEB.,

4 DAVIS AND FIUZAT FIG. 2. D. K., Case 2. Chest x-ray showing bilateral aspiration pneumonitis. to be free of cough, free of pneumonia, eating well, gaining weight and thriving. She remains well. Figure 1B was taken six weeks postoperatively. CASE 2 (NONOPERATIVE GROUP) D. K. was first seen at 7 weeks of age because of recurrent bilateral bronchopneumonia which had never really cleared (Fig. 2). He was admitted to the hospital three times in three months, and was hospitalized the last of these three admissions for 45 days. Following discharge he was seen in the outpatient clinic five times because of recurrent cough and pneumonitis. Cinefluorography revealed marked gastroesophageal reflux. Surgery was recommended but not accepted. The patient has not done well, and his bronchopneumonia persists. CASE 3 (OPERATIVE GROUP) S. C., an 11-year-old female referred in March, 1966, because of recurrent bilateral bronchopneumonia, cough, bronchitis, and occasional asthmatic bronchitis. Wheezing at night had been noticed by her parents for two years. The child was a large, healthy-appearing girl who had been restricted from physical education at school for the past two years because of cough and bronchitis. Cinefluorography revealed a small amount of reflux into the distal esophagus when the test was performed with the stomach empty. Testing after a full meal revealed marked gastroesophageal reflux to the level of the clavicles. A wide gastroesophageal junction and probable large hiatal ring was identified as well as a high insertion of the esophagus to the lesser curvature and a wide esophagofundic angle. Surgery was performed in July, Figure 3 is the preoperative chest x-ray on this child. Postoperative chest x-rays revealed: (1) a longer length of intraabdominal esophagus, (2) insertion of the esophagus lower on the lesser curvature or in the normal position, (3) the fundic air bubble characteristic of a satisfactory Belsey repair, and (4) clearing of the basal infiltrate. The child has been entirely free of symptoms since surgery. CASE 4 (NONOPERATIVE GROUP) C. W. was seen at the age of 12 years in March, 1966, because ol recurrent bilateral pneumonia and x-ray findings seen on several occasions of left lower lobe pneumonia and right middle lobe atelectasis. At that time the child was hospitalized for 63 days, during which period she ran a low-grade fever, had moderate cough, and had x-ray findings of bilateral inflammation. Cinefluorog- 102 THE ANNALS OF THORACIC SURGERY

5 Regurgitation in Children FIG. 3. S. C., Case 3. Preoperative chest x-ray. raphy revealed moderate gastroesophageal reflux. Bronchoscopic examination revealed marked bronchitis, and normal flora (Neisseria catarrhalis and Streptococcus viridans) were recovered. Surgery was recommended but declined by the pediatric service and the parents. This child s problem remains, with chronic interstitial pneumonitis in the right middle lobe and the left lower lobe, recurrent cough, and low-grade fever. Figure 4A is a posteroanterior chest roentgenogram of this child; Figure 4B is a lateral view. CASE 5 (NONOPERATIVE GROUP) P. S. was first seen (Fig. 5) in November, 1964, at 17 months of age, at which time he weighed 24 pounds. Thirteen months later he weighed 31% pounds, and the x-rays were unchanged. He had had two hospital admissions and over twenty clinic visits for bilateral bronchopneumonia, cough, and wheezing. He had ten visits since the last hospital admission with the same complaints. While in the A FZG. 4. C. W., Case 4. (A) The x-ray shows pneumonitis in the right middle lobe and left lower lobe and bilateral peribronchial inflammation. (B) Lateral view. B VOL. 3, NO. 2, FEB.,

6 DAVIS AND FIUZAT FIG. 5. P. S., Case 5. Posteroanterior x-ray showing severe bilateral bronchopneumonia. hospital he had a temperature as high as 104", and he has had a total of 27 courses of antibiotics. He has marked gastroesophageal reflux by water testing; surgery was advised but was not accepted. This child does considerably better when he sleeps in an upright or semiupright position, although this is getting to be more difficult to accomplish now that he is older. CASE 6 (NONOPERATIVE GROUP) C. C. was seen at 14 months of age in March, At that time he weighed 18 pounds, and he had had recurrent pneumonia three times in the previous 3 months. X-rays (Fig. 6) at that time and on subsequent clinic visits continued to show bilateral bronchopneumonia. Cinefluorography revealed severe gastroesophageal reflux. Surgery was recommended but not carried out. CASE 7 (OPERATIVE GROUP) B. G. S., a 10-year-old boy, was seen in the spring of 1966 for recurrent asthmatic bronchitis. His chest roentgenogram showed enlarged hilar shadows and probably some degree of interstitial fibrosis. Cinefluorography revealed moderate reflux into the distal esophagus during water testing on an empty FIG. 6. C. C., Case 6. Posteroanterior x-ray. 104 THE ANNALS OF THORACIC SURGERY

7 Regurgitation in Children FIG. 7. B. G. S., Case 7. pnezim onia. The x-ray shows postoperative right upper lobe stomach. Considerably more reflux was seen when the test was performed after a full meal. Surgery was performed in June, This child s chief complaint was recurrent wheezing. Pneumonia, first in the right upper lobe (Fig. 7) and later in the left lower lobe, occurred in the postoperative period and required vigorous nebulization therapy, suctioning, and antibiotics before it cleared. After discharge this child s postoperative course was excellent for approximately six weeks, after which he began to wheeze again. This patient must be considered, at least at this time, a treatment failure. CASE 8 (OPERATIVE GROUP) S. W., a 4%-year-old girl, was first seen in late winter of for almost constant recurrent bronchopneumonia. Cinefluorography on the first test revealed marked gastroesophageal reflux. This was repeated five days later and showed only moderate reflux. This illustrates that the same individual will show varying degrees of reflux at different times and under different conditions for no apparent reason. The child was operated on in March, 1966, and has remained perfectly well since that time. Figure 8 is a postoperative roentgenogram showing a satisfactory Belsey repair. CASE 9 (OPERATIVE GROUP) R. C. B., a 3%-year-old boy, was seen in the winter of for recurrent bilateral bronchopneumonia. Cinefluorography revealed a marked spontaneous gastroesophageal reflux without water testing and severe reflux on water testing. This child was operated upon in March, He has had no pneumonia since and has been gaining weight and doing very well clinically. Reexamination with cinefluorography reveals very mild but definite gastroesophageal reflux, suggesting an inadequate repair, although the patient is very much improved. CASE 10 (OPERATIVE GROUP) T. D., a 2-year-old girl, was first seen in October, 1965, for kerosene ingestion. In the next four months she had four clinic visits and one hospital admission for recurrent bilateral bronchopneumonia. She was hospitalized for 47 days the last time. Water testing during cinefluorography revealed marked gastroesophageal VOL. 3, NO. 2, FEB.,

8 DAVIS AND FIUZAT FIG. 8. S. W., Case 8. This x-ray, taken four days postoperatively, shows (1) adequate subdiaphragmatic length of esophagzu, (2) acute esophagofirndic angle, and (3) buttress of stomach at esophagogastric junction. reflux; she regurgitated often while lying in bed. Surgery was performed in February, The interstitial pneumonitis cleared, and she has remained well. CASE 11 (OPERATIVE GROUP) S. D., was seen at the age of 1 year for recurrent bilateral bronchopneumonia. Water testing during cinefluorography showed marked gastroesophageal reflux. Surgery was recommended but refused. The child was discharged from the hospital at the parents' request. Later, the parents requested that surgery be performed because he was coughing a great deal. Surgery was performed in February, The child had moderate bronchitis and some pneumonitis, requiring intensive nebulization therapy and bronchial suctioning postoperatively. He was discharged in good condition and has remained well. CASE 12 (OPERATIVE GROUP) W. B. E., a 17-month-old boy, was chronically ill with persistent bronchopneumonia as he had had fourteen clinic visits in the two months prior to admission and four hospital admissions in 18 months of life for pneumonia and failure to thrive. His fever had run as high as 104.4' in the hospital. Cinefluorography revealed regurgitation with spontaneous reflux and marked regurgitation with water testing. The patient was operated upon in April, He was discharged on the seventh postoperative day and was examined in the clinic only twice in the next month for what appeared to be a mild pharyngitis. He began to gain weight immediately, and he has remained well. CASE 13 (OPERATIVE GROUP) G. B. was a 2%-year-old boy who had had two admissions for bilateral bronchopneumonia in four months, with an average of 22 hospital days per admission. Cinefluorography revealed marked spontaneous gastroesophageal reflux and marked reflux during water testing. Surgery was performed in June, His postoperative course was uneventful, and he was discharged a week later. He has remained well. 106 THE ANNALS OF THORACIC SURGERY

9 Regurgitation in Children A FIG. 9. K. H., Case 14. (A) The posteroanterior chest x-ray shows left lower lobe atelectasis. (B) Lower lobe pneumonitis and atelectasis is shozun in the lateral view. B CASE 14 (NONOPERATWE GROUP) K. H., a 7-year-old girl, was referred for evaluation of left lower lobe atelectasis. Figures 9A and B are her chest roentgenograms. Bronchoscopy was carried out and revealed bronchitis and normal bacterial flora on culture. Cinefluorography revealed mild gastroesophageal reflux during water testing. Surgery was not recommended because of the benign clinical course. DISCUSSION This is a preliminary report. It will be important to follow these cases, see how well they do in the future, and compare the operative and nonoperative groups. However, it is the opinion of the authors, as well as the pediatricians who have worked with us on these patients, that the striking improvement in some warrants a further consideration of this treatment. Even though this has been a brief experience, certain salient points have emerged which seem worth mentioning: 1. The water test has been variable in the same patient from one time to another and in the same patient on the same day if done on an empty stomach and then repeated after a full meal. As stressed by Urschel and Paulson [7], reflux is much more likely to be demonstrated following a full meal. Many times spontaneous gastroesophageal reflux occurs without water ingestion. 2. While we do.not have a large series of so-called normals who have had cinefluorography, gastroesophageal reflux unquestionably VOL. 3, NO. 2, IXB.,

10 DAVIS AND FIUZAT does occur in many children and in quite a large number of infants who do not have abnormal digestive, bronchial, or pulmonary symptoms or signs. It would be helpful to know the results of water testing in a large series of normal subjects and to classify them carefully; however, radiologists seem to be a bit reluctant to subject children to cinefluorography without a medical indication. 3. The anatomy of the lower esophagus, the hiatal ring, and the junction of the esophagus and stomach probably are far more important than has previously been appreciated, at least by the authors of this paper. A wide hiatal ring, a wide esophagogastric junction, insertion of the esophagus so high on the lesser curvature that the esophagogastric junction could be compared to an inverted funnel (Fig. 10) all seem to contribute to the gastroesophageal reflux syndrome. Older reports describing so-called chalasia have included some of these points. 4. There is a striking difference in the index of suspicion for this condition as well as interpretation among different radiologists. 5. Postoperative cinefluorographic studies and follow-up of these patients have indicated that there can be encouraging clinical improvement even though some degree of gastroesophageal reflux persists. LUNG BIOPSY Lung biopsy was performed in all of the operative cases. Histological, bacteriological, and virological studies of the specimens were nondiagnostic. It was kept in mind that the syndromes studied all were associated with a clinical problem characterized by recurrent bronchitis and pneumonia. FIG. 10. K. W., Case 1. Inverted funnel contour of esophagogastric junction and gastroesophageal reflux on water testing. 108 THE ANNALS OF THORACIC SURGERY

11 Regurgitation in Children THE POSTOPERATIVE COURSE Bearing in mind that all of these patients had severe bilateral recurrent bronchopulmonary inflammation, it would be expected that they would not have an entirely smooth postoperative course. They all required nebulization therapy and bronchial suctioning, considerably more of both than would be required after, for example, a thoracotomy for an uncomplicated persistent ductus arteriosus. There were no wound infections, pleural infections, or deaths in the group. Postoperative prophylactic antibiotics were not used routinely. Case 7, who had postoperative lobar pneumonitis, had antibiotics used for treatment. Gastric retention of air was present in all, but nasogastric suction or decompression was thought to be necessary in only 1. FAILURE OF THE METHOD There have been two areas of apparent initial failure in this effort to date: 1. Those patients who had definite clinical bronchial asthma preoperatively have not attained as good results as those who have had bronchitis and pneumonitis [6]. 2. We must question the adequacy of the repair if the postoperative cinefluorographic studies reveal any significant degree of gastroesophageal reflux. This has occurred in 2 of our cases out of 9. Urschel and Paulson [7] feel that one cause of this is failure to free the esophagus sufficiently high enough in the mediastinum to attain adequate length of esophagus below the diaphragm. CONCLUSION 1. It would appear that some of the patients who have the combination of a severe gastroesophageal reflux and recurrent bronchopulmonary problems can be helped by performing a Belsey-type hernia repair even though there is no herniation of the stomach through the hiatus or above the diaphragm. 2. These patients are probably not normal anatomically. Careful studies will usually reveal little, if any, of the esophagus below the diaphragm. A wide esophagofundic angle causes an inverted funnel appearance at the esophagogastric junction. This is usually associated with a large hiatus. 3. This preliminary study would seem to lend support to the idea that all so-called respiration infections are not infections at all, but that some, or perhaps many, are due to chemical bronchitis and chemical pneumonitis, especially in those who run a protracted course with numerous recurrences. This may account in part for the large number of failures of treatment and prophylaxis by antibiotics. VOL. 3, NO. 2, EEB., 1967 log

12 DAVIS AND FIUZAT 4. Our initial experience is encouraging and would seem to support a continuation of the above-outlined program. SUMMARY 1. A total of 18 patients who presented with severe recurrent bilateral bronchopulmonary inflammation and who also were found to have gastroesophageal reflux have been reviewed. 2. Nine of these patients have had thoracotomies and a Belseytype hiatal hernia repair even though there was no herniation of the stomach. 3. The criteria for selection of these cases and some comments about the series up to the present time have been made, and some of the cases have been presented in brief summary to bring out certain points of interest. REFERENCES Belsey, R. Diaphragmatic Hernia: Modern Trends in Gastroenterology. London: Butterworth, P Cavvalho, M. de Chirurgie de syndrome hiato-oesophagien (communication prbalable). Arch. Mal. Appar. Dig. 40:280, Crummy, A. B. The water test in the evaluation of gastroesophageal reflux. Radiology 78:501, Hiebert, C. A., and Belsey, R. Incompetency of the gastric cardia without radiologic evidence of hiatal hernia. J. Thorac. Surg. 42:352, Linsman, J. F. Gastroesophageal reflux elicited while drinking water (watersiphonage test): Its clinical correlation with pyrosis. Amer. J. Roentgenol. 94:325, Overholt, R. H., and Voorhees, R. J. Esophageal reflux as a trigger in asthma. Dis. Chest 49:464, Urschel, H. C., Jr., and Paulson, D. L. Gastroesophageal reflux and hiatal hernia complications and therapy. J. Thorac. Cardiou. Surg. (in press). 110 THE ANNALS OF THORACIC SURGERY