Gastric Duplication By Chumsak Pruksapong, Raymond J. Donovan, Arvoranee Pinit, and Fred J Heldrich Baltimore, Maryland 9 Vomiting and failure to thrive were the presenting complaints of a patient with gastric duplication. The case is presented and features of gastric duplication are summarized with other cases reported in the literature. I N D E X W O R D S : Duplication, gastric.
of the stomach account r only 3.8% of gastrointestinal tract duplications. 1 A review of the literature and case report by BarteF in 1967 revealed 55 recorded cases of gastric duplication. We have found 27 additional recorded cases in the English language literature to 19777 -1~ A double stomach was noted in the cadaver of an 85-yr-old male by Vacchus and Blasius in 1617, but the first clinical documentation was by Wendell in 1911. I~ CASE REPORT This 4-wk-old boy had persistent vomiting since birth with failure to thrive. During his stay in the nursery, occasional spitting was noted with feedings, but he was sent home on the fourth day weighing 3515 g. The vomiting continued and increased in severity, so that just prior to admission, it was occurring with every feeding and consisted of undigested formula. There was no bile or blood in the vomitus and lying in the supine position aggravated the vomiting. Two days prior to admission, it was noted that he would cry and flex his legs up on the abdominal wall when feeding. On admission to the hospital, palpation of the abdomen revealed guarding on pressure over the right upper quadrant and epigastrium which elicited leg flexion. There was also an ill-defined mass in the epigastrium. Low grade fever had been noted on admission, and after cultures of blood, urine, stool, nasopharynx, and spinal fluid were obtained, the infant was placed on Ampicillin, 200 mg/kg/24 hr, and Kanamycin, 15 mg/kg/24 hr, i.v. All cultures were negative after 48 hr, temperature was normal, and antibiotics were discontinued on the third hospital day. Intermittent fever returned by the fifth hospital day, and the initial blood culture was noted to be positive for anerobic diphtheroids after 10 days' incubation. Therapy with Ampicillin and Kanamycin was reinstituted at this time with return of temperature to normal. An upper GI series demonstrated a radiolucent filling defect at the greater curvature of the stomach (Fig_ 1). An exploratory taparotomy revealed a duplication of the stomach, 6 cm in diameter, located along the greater curvature without communication with the lumen of the stomach. After aspiration of a sterile, clark green fluid, the cyst was removed in toto. Microscopic section of the cyst wall revealed normal gastric mucosa (Fig. 2). The postoperative course was uneventful.
Journal of Pediatric Surgery, Vol. 14, No. 1 (February),1979
DISCUSSION
The diagnosis of gastric duplication is usually made in early infancy and the vast majority are discovered by the first year of life. Of those cases we have found recorded, 33 were identified by the third month of life; an additional 19, between the third month and first year; 12, between the 1st to 12th yr; and 19, after the 12th yr. The oldest reported case was a 64-yrold female who developed carcinoma in the duplicated portion. '~ Duplication of the stomach has been noted twice as frequently in the female, but no striking racial differences have been noted. Symptoms usually begin early, but in two patients the diagnosis was made unexpectedly and the patients bad no symptoms referable to the duplication? In order of frequency, the symptoms in the 83 cases reviewed were: palpable abdominal mass, vomiting, weight loss or failure to thrive, abdominal pain, anemia, melena, hematemesis, and fever. ~-t9 Symptoms of abdominal pain, anemia, hematemesis, and melena were seen more frequently in older patients. Bleeding, when noted, was secondary to ulceration of the mucosa in the duplication and requires communication of the duplication with the gastric lumen (Fig. 3). Fever, in our case, can be explained by the positive blood culture of an anerobic organism. Growth of such organisms may require prolonged incubation and, as demonstrated here, a positive culture was not detected until the tenth day of incubation. Any relationship of the gastric duplication to the septicemia in this case remains unclear. Within the stomach, the most common location for the duplication was along the greater curvature (Table l). Only one duplication was noted at the region of the pylorus? Most often, the duplicated portion does not communicate
From the Department of Pediatrics, St. Agnes Hospital, Baltimore, Md. Address reprint requests to Fred J. Heldrich, M.D., Chairman, Department of Pediatrics. St. Agnes Hospital, 900 Caton Avenue. Baltimore, Md. 21229. fr by Grune & Stratton, Inc. 0022 3468/79/1401-0017501.00/0
83
84
PRUKSAPONG ET AL.
Fig. 1. Radiograph showing radiolucent filling defect on greater curvature and antrum of stomach.
with the lumen of the stomach. Duplication of other parts of the gastrointestinal tract has been found in association with gastric duplication. The esophagus, duodenum, ileum, and colon have been reported as additional sites of
Fig. 2. H i s t o l o g i c appearance of duplication cyst showing m u c o s a l w a l t and gastric mucosa.
duplication in a few cases. Duplications involving the esophagus are frequently associated with hemivertebrae, but these have also been reported with gastric duplication? ,7 While the etiology of gastric duplication
GASTRIC DUPLICATION
85
PALPABLE bIASS
~ .........
VOMITING
[
WEIGHT
[
LOSS
ABDObIINAL PAIN ANEblIA GI
BLEEDING
l
'-I
Z0
1 .I
24
F v~3--
Table 1. Location of Duplication of Stomach in 83 Reported Cases
_
55 29
.[ [
55 _
--1
fv- l Fig. 3.
Frequency of symptoms in 83 cases.
remains unexplained, several theories have been proposed, faulty embryonic budding or defective vacuolization being most prominently mentionedY ~ Histologically, duplications present as cystic structures whose walls are composed of a
Location
No of Cases
Greater curvature
54
Lesser curvature
7
Anterior wall Posterior wall
5 9
Pylorus Others
1 7
muscle layer and mucosal epithelium, intimately adjacent to, but not necessarily communicating with, the lumen of the stomach. Surgical excision is considered the treatment of choice for duplication, but the exact surgical procedure is dictated by the individual conditions.
REFERENCES
1. Silverman A, Roy CC, Cozzetto F J: Pediatric Clinical Gastroenterology. St. Louis, C. V. Mosby, 1971, p 71 2. Bartel R J: Duplication of stomach: Case report and review of the literature. Am Surg 33:747 752, 1967 3. Kammerer GT: Duplication of stomach resembling hypertrophic pyloric stenosis. JAMA 207:2101 2102, 1969 4. Grosfeld JL, Boles T, Reiner L: Duplication of pylorus in the newborn: A rare cause of gastric outlet obstruction. J Pediatr Surg 5:365 369, 1970 5. Klidjian A, Sutton PD: Duplication of stomach associated with non-rotation of the gut. Br J Surg 54:731 735, 1967 6. Shochat ST: Perforated gastric duplication with pulmonary communication: A case report. Surgery 70:370-374, 1971 7. White J J, Morgan WW: Improved operative technique for gastric duplication. Surgery 67:522 526, 1970 8. Tabrisky J, Szalay GC, Meade WS: Duplication of stomach: A cause of anemia. Am J G a s t r o e n t e r o l 59:327-331, 1973 9. Kremer RM, Lepoff RB, lzant RJ: Duplication of stomach. J Pediatr Surg 5:361-364, 1970 10. Alschibaja T, Putnam TC, Yablin BA: Duplication of stomach simulating hypertrophic pyloric stenosis. Am J Dis Child 127:120 122, 1974 11. Hawkins ML, Lowery CH, Mullen JT: Gastric duplication. South Med J 67:189, 1974
12. Wilkinson DJ, Wilkinson KW, Hajdu N: Intestinal duplication: A report of two cases. Br J Radiol 46:1070 1072, 1973 13. Hale CR: Radiologic seminar 108. Gastric duplication-Illustrated case with massive hemorrhage. J Miss State Med Assoc 12:303 305, 1971 14. Gray DH: Total reduplication of the stomach: A rare anomaly. Aust NZ J Surg 41:130-133, 1971 15. Mahour GH, Woolley MM, Payne VC Jr: Association of pulmonary segmentation and duplication of stomach. Int Surg 56:224 227, 1971 16. Parker BC, Guthrie J, France NE, et al: Gastric duplication in infancy. J Pediatr Surg 7:294 298, 1972 17. Clement KW, Escamilla HA: Duplication of the stomach. J Natl Med Assoc 66:282 285, 1974 18. Mayo HW Jr: Carcinoma arising in reduplication of the stomach (gastrogenous cyst): A case report. Ann Surg 14l:550 555, 1955 19. Wilkinson DJ, Chir B, Wilkinson KW, et al: Intestinal duplication: A report of two cases. Br J Radiol 46:1070 1072, 1973 20. T o r m a M: Of double stomachs. Arch Surg 109:555 557, 1974 21. Stowens D: Pediatric Pathology. Baltimore, Williams and Wilkins, 1959, p 469
of the stomach account r only 3.8% of gastrointestinal tract duplications. 1 A review of the literature and case report by BarteF in 1967 revealed 55 recorded cases of gastric duplication. We have found 27 additional recorded cases in the English language literature to 19777 -1~ A double stomach was noted in the cadaver of an 85-yr-old male by Vacchus and Blasius in 1617, but the first clinical documentation was by Wendell in 1911. I~ CASE REPORT This 4-wk-old boy had persistent vomiting since birth with failure to thrive. During his stay in the nursery, occasional spitting was noted with feedings, but he was sent home on the fourth day weighing 3515 g. The vomiting continued and increased in severity, so that just prior to admission, it was occurring with every feeding and consisted of undigested formula. There was no bile or blood in the vomitus and lying in the supine position aggravated the vomiting. Two days prior to admission, it was noted that he would cry and flex his legs up on the abdominal wall when feeding. On admission to the hospital, palpation of the abdomen revealed guarding on pressure over the right upper quadrant and epigastrium which elicited leg flexion. There was also an ill-defined mass in the epigastrium. Low grade fever had been noted on admission, and after cultures of blood, urine, stool, nasopharynx, and spinal fluid were obtained, the infant was placed on Ampicillin, 200 mg/kg/24 hr, and Kanamycin, 15 mg/kg/24 hr, i.v. All cultures were negative after 48 hr, temperature was normal, and antibiotics were discontinued on the third hospital day. Intermittent fever returned by the fifth hospital day, and the initial blood culture was noted to be positive for anerobic diphtheroids after 10 days' incubation. Therapy with Ampicillin and Kanamycin was reinstituted at this time with return of temperature to normal. An upper GI series demonstrated a radiolucent filling defect at the greater curvature of the stomach (Fig_ 1). An exploratory taparotomy revealed a duplication of the stomach, 6 cm in diameter, located along the greater curvature without communication with the lumen of the stomach. After aspiration of a sterile, clark green fluid, the cyst was removed in toto. Microscopic section of the cyst wall revealed normal gastric mucosa (Fig. 2). The postoperative course was uneventful.
Journal of Pediatric Surgery, Vol. 14, No. 1 (February),1979
DISCUSSION
The diagnosis of gastric duplication is usually made in early infancy and the vast majority are discovered by the first year of life. Of those cases we have found recorded, 33 were identified by the third month of life; an additional 19, between the third month and first year; 12, between the 1st to 12th yr; and 19, after the 12th yr. The oldest reported case was a 64-yrold female who developed carcinoma in the duplicated portion. '~ Duplication of the stomach has been noted twice as frequently in the female, but no striking racial differences have been noted. Symptoms usually begin early, but in two patients the diagnosis was made unexpectedly and the patients bad no symptoms referable to the duplication? In order of frequency, the symptoms in the 83 cases reviewed were: palpable abdominal mass, vomiting, weight loss or failure to thrive, abdominal pain, anemia, melena, hematemesis, and fever. ~-t9 Symptoms of abdominal pain, anemia, hematemesis, and melena were seen more frequently in older patients. Bleeding, when noted, was secondary to ulceration of the mucosa in the duplication and requires communication of the duplication with the gastric lumen (Fig. 3). Fever, in our case, can be explained by the positive blood culture of an anerobic organism. Growth of such organisms may require prolonged incubation and, as demonstrated here, a positive culture was not detected until the tenth day of incubation. Any relationship of the gastric duplication to the septicemia in this case remains unclear. Within the stomach, the most common location for the duplication was along the greater curvature (Table l). Only one duplication was noted at the region of the pylorus? Most often, the duplicated portion does not communicate
From the Department of Pediatrics, St. Agnes Hospital, Baltimore, Md. Address reprint requests to Fred J. Heldrich, M.D., Chairman, Department of Pediatrics. St. Agnes Hospital, 900 Caton Avenue. Baltimore, Md. 21229. fr by Grune & Stratton, Inc. 0022 3468/79/1401-0017501.00/0
83
84
PRUKSAPONG ET AL.
Fig. 1. Radiograph showing radiolucent filling defect on greater curvature and antrum of stomach.
with the lumen of the stomach. Duplication of other parts of the gastrointestinal tract has been found in association with gastric duplication. The esophagus, duodenum, ileum, and colon have been reported as additional sites of
Fig. 2. H i s t o l o g i c appearance of duplication cyst showing m u c o s a l w a l t and gastric mucosa.
duplication in a few cases. Duplications involving the esophagus are frequently associated with hemivertebrae, but these have also been reported with gastric duplication? ,7 While the etiology of gastric duplication
GASTRIC DUPLICATION
85
PALPABLE bIASS
~ .........
VOMITING
[
WEIGHT
[
LOSS
ABDObIINAL PAIN ANEblIA GI
BLEEDING
l
'-I
Z0
1 .I
24
F v~3--
Table 1. Location of Duplication of Stomach in 83 Reported Cases
_
55 29
.[ [
55 _
--1
fv- l Fig. 3.
Frequency of symptoms in 83 cases.
remains unexplained, several theories have been proposed, faulty embryonic budding or defective vacuolization being most prominently mentionedY ~ Histologically, duplications present as cystic structures whose walls are composed of a
Location
No of Cases
Greater curvature
54
Lesser curvature
7
Anterior wall Posterior wall
5 9
Pylorus Others
1 7
muscle layer and mucosal epithelium, intimately adjacent to, but not necessarily communicating with, the lumen of the stomach. Surgical excision is considered the treatment of choice for duplication, but the exact surgical procedure is dictated by the individual conditions.
REFERENCES
1. Silverman A, Roy CC, Cozzetto F J: Pediatric Clinical Gastroenterology. St. Louis, C. V. Mosby, 1971, p 71 2. Bartel R J: Duplication of stomach: Case report and review of the literature. Am Surg 33:747 752, 1967 3. Kammerer GT: Duplication of stomach resembling hypertrophic pyloric stenosis. JAMA 207:2101 2102, 1969 4. Grosfeld JL, Boles T, Reiner L: Duplication of pylorus in the newborn: A rare cause of gastric outlet obstruction. J Pediatr Surg 5:365 369, 1970 5. Klidjian A, Sutton PD: Duplication of stomach associated with non-rotation of the gut. Br J Surg 54:731 735, 1967 6. Shochat ST: Perforated gastric duplication with pulmonary communication: A case report. Surgery 70:370-374, 1971 7. White J J, Morgan WW: Improved operative technique for gastric duplication. Surgery 67:522 526, 1970 8. Tabrisky J, Szalay GC, Meade WS: Duplication of stomach: A cause of anemia. Am J G a s t r o e n t e r o l 59:327-331, 1973 9. Kremer RM, Lepoff RB, lzant RJ: Duplication of stomach. J Pediatr Surg 5:361-364, 1970 10. Alschibaja T, Putnam TC, Yablin BA: Duplication of stomach simulating hypertrophic pyloric stenosis. Am J Dis Child 127:120 122, 1974 11. Hawkins ML, Lowery CH, Mullen JT: Gastric duplication. South Med J 67:189, 1974
12. Wilkinson DJ, Wilkinson KW, Hajdu N: Intestinal duplication: A report of two cases. Br J Radiol 46:1070 1072, 1973 13. Hale CR: Radiologic seminar 108. Gastric duplication-Illustrated case with massive hemorrhage. J Miss State Med Assoc 12:303 305, 1971 14. Gray DH: Total reduplication of the stomach: A rare anomaly. Aust NZ J Surg 41:130-133, 1971 15. Mahour GH, Woolley MM, Payne VC Jr: Association of pulmonary segmentation and duplication of stomach. Int Surg 56:224 227, 1971 16. Parker BC, Guthrie J, France NE, et al: Gastric duplication in infancy. J Pediatr Surg 7:294 298, 1972 17. Clement KW, Escamilla HA: Duplication of the stomach. J Natl Med Assoc 66:282 285, 1974 18. Mayo HW Jr: Carcinoma arising in reduplication of the stomach (gastrogenous cyst): A case report. Ann Surg 14l:550 555, 1955 19. Wilkinson DJ, Chir B, Wilkinson KW, et al: Intestinal duplication: A report of two cases. Br J Radiol 46:1070 1072, 1973 20. T o r m a M: Of double stomachs. Arch Surg 109:555 557, 1974 21. Stowens D: Pediatric Pathology. Baltimore, Williams and Wilkins, 1959, p 469
