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Rupture of the stomach in newborn infants
RUPTURE OF T H E STOMACH IN NEWBORN I N F A N T S REPORT OF TWO CASES ]~THEL C. D U N H A M , ~'[.D. AND RICHARD M . GOLDSTEIN, M . D . N E W HAVEN, CONN.
WO cases of rupture of the stomach occurring in newborn infants T are here reported because of the rarity of the condition. As will be pointed out, only 9 other such cases have been described. Abstracts of the case histories with clinical and pathologic findings follow. CASE 1.--N. S., a white male, was borll Sept. 7~ 1929, in the l'~ew H a v e n Hospital. Tho mother developed a severe toxemia and was delivered at term by eesar~an section. The i n f a n t weighed 4~,166 gm. a n d appeared normal at birth. On the f o u r t h day of life the temperature became elevated. The i n f a n t vomited and about six hours later became acutely ill and was f o u n d to be distended and to have edema of the abdominal wall. The leucocyte count was 4,000, polymorphonuclears 38 per cent, tymphocytes 53 per cent. No definite clinical diagnosis was made at this time, although the symptoms could best be explained by peritonitis. A subcutaneous injection of saline, and a t r a n s f u s i o n of blood were then given and the i n f a n t ' s stomach was lavagcd. The following day, although the i n f a n t ' s condition seemed somewhat improved, the distention and vomiting continued. At this t i m e a roentgenogram, taken a f t e r a rectal injection of barium had been given, showed t h a i there was no evidence of obstruction in the large intestine. Reexamination of the film several days lateT revealed the f a c t t h a t a small amount of air h a d been present in the peritoneal cavity at ~he time the roentgenogram was taken. D u r i n g this period, however, the i n f a n t ' s condition had greatly improved~ the abdominal distention had decreased, stools were passed daily~ fluids were taken well, and there was no vomiting. Th6 temperature remained normal most of the time. A blood culture taken two days a f t e r the onset of the illness showed B. vo/~, as did six subsequent cultures made on the sixth, ninth, fourteenth, slxteenth, seventeenth, and eighteenth days. On the thirteenth day of life a mass was palpated in the left flank and a n exploratory operation was performed. On going through the peritoneum i n the region where the mass had been felt there was an escape of gas and of thin yellowish fluid having the odor of colon bacillus pus. No perforation was found in the intestines, but an accumulation of pus was found walled off near the upper pole of the left kidney. This was t h o u g h t to be an abscess and was opened and drained. On the following day a f t e r the first feeding, undigested milk appeared in the wound. The presence of a fistulous opening was confirmed by fluoroscopic examination after a barium meal. l~rom the Departments of Pediatrics and Pathology, Yale University SChool of Medicine, and U. S. Children's Bureau (Studies of Neonatal !Y[orbidity and !~Iortality). 44
DUI~HAM AI~D GOLDSTEIN : RUPTUI~E OF STOMACH IN NEWBOtCN INFAI~TS
45
A n a t t e m p t w a s m a d e to r e p a i r t h e p e r f o r a t i o n i n t h e stomach, b u t t h i s was unsuccessful. Blood cultures c o n t i n u e d to be positive f o r B. roll, a n d the i n f a n t died on t h e t w e n t y - f i r s t day. T h e final clinical d i a g n o s i s was r u p t u r e of t h e stomach, peritonitis, B. ooli s efp t ~ee~nial. A n a u t o p s y w a s p e r f o r m e d f o u r h o u r s a f t e r death. T h e skin a n d s u b c u t a n e o u s tissue over t h e a b d o m e n were edematous. T h e r e w a s no f r e e fluid in the p e r i t o n e a l cavity. T h e s t o m a c h , t r a n s v e r s e a n d d e s c e n d i n g colon~ a n d several coils of small i n t e s t i n e were b o u n d t o g e t h e r b y fibrinous a n d fibrous adhesions. W h e n t h e s t o m a c h was d i s s e c t e d free, a s u t u r e line m e a s u r i n g 1 am. w a s seen on the g r e a t e r c u r v a t u r e at t h e cardia. O n f r e e i n g t h e spleen several small p o c k e t s o f thick yellow p u s were e n c o u n t e r e d b e t w e e n t h e spleen a n d t h e d i a p h r a g m . S i m i l a r pockets of p u s were s c a t t e r e d b e t w e e n t h e coils of i n t e s t i n e in this region. W h e n t h e s t o m a c h w a s opened, i t w a s seen t h a t t h e m u e o s a , except for the site of r u p t u r e , was i n t a c t a n d t h e u s u a l r u g a e were present. T h e r e m a i n i n g p o r t i o n s of the g a s t r o i n t e s t i n a l t r a c t showed no a b n o r m a l i t i e s aside f r o m t h e serosal a n d p e r i t o n e a l reaction. Serial sections were t a k e n t h r o u g h t h e a r e a of r u p t u r e . The g a s t r i c m u c o s a u p to t h e edge o f t h e r u p t u r e a p p e a r e d n o r m a l . T h e r e was no cellular infiltration or h e m o r r h a g e . T h e p o i n t of rupture, w a s s u r r o u n d e d b y a t h i n wall of h o m o g e n e o u s , necrotic d~bris w h i c h took t h e eosin s t a i n a n d w a s s u r r o u n d e d b y a n a r r o w zone of tissue i n f i l t r a t e d w i t h m o n o n u c l e a r leucocytes a n d e x t r a v a s a t e d red blood cells. F a r t h e r f r o m t h e lesion t h e m u c o s a a n d deeper ]avers of the s t o m a c h were apparently normal. Sections of t h e j e j u n u m a n d ileum showed t h a t t h e serosal s u r f a c e s were t h i c k e n e d b y a n infiltration o f m o n o n u c l e a r leucocytes; t h e m u c o s a e were i n t a c t a n d f r e e f r o m cellular infiltration or h e m o r r h a g e . Sections of t h e l u n g s showed f o c a l p n e u m o n i a . The a n a t o m i c d i a g n o s i s was p e r f o r a t i o n of t h e s t o m a c h ; localized p e r i t o n e a l a b s c e s s ; s u b d i a p h r a g m a t i c a b s c e s s ( l e f t ) ; p a r t i a l o b s t r u c t i o n of t h e s i g m o i d colon; fecal fistula; f o c a l p n e u m o n i a ; recent l a p a r o t o m y f o r d r a i n a g e of p e r i t o n e a l cavity, a n d s u t u r e of s t o m a c h . CASE 2 . - - B . B., a white male, was b o r n p r e m a t u r e l y Sept. ]1, 1930, in t h e _New H a v e n H o s p i t a l . T h e m o t h e r was a h e a l t h y p r i m i p a r a , t w e n t y - o n e y e a r s old. The labor w a s n o r m a l a n d t h e delivery s p o n t a n e o u s . T h e b i r t h w e i g h t was 1,960 g m . T h e i n f a n t ' s g e n e r a l condition was good, b u t t h e t e m p e r a t u r e was s u b n o r m a l (35.6 ~ C.). O n e x a m i n a t i o n t h e i n f a n t , a l t h o u g h small, a p p e a r e d n o r m a l except f o r a b i l a t e r a l h a r e l i p a n d cleft palate. F e e d i n g w i t h modified c o w ' s milk w a s b e g u n a b o u t twelve h o u r s a f t e r birth. T h e i n f a n t v o m i t e d bile-stained m a t e r i a l following t h e first f e e d i n g a n d c o n t i n u e d to v o m i t a f t e r s u b s e q u e n t feedings. Abdominal distention and increased peristalsis appeared about thirty hours after birth. Two h y p o d e r m o c l y s e s were g i v e n a n d at t h l r t y - e i g h t h o u r s l a v a g e a n d g a v a g e were b e g u n a n d c o n t i n u e d t h r o u g h t h e n e x t day. A blood culttu'e t a k e n on t h e t h i r d d a y of life showed colon bacilli in b r o t h a n d plates. A r o e n t g e n o g r a m of t h e a b d o m e n (fiat plate) showed a l a r g e q u a n t i t y o f g a s in t h e s t o m a c h a n d i n t e s t i n a l t r a c t . T h e W a s s e r m a n n t e s t on t h e blood s e r u m was negative. A t t a c k s of c y a n o s i s developed, a n d t h e i n f a n t died at t h e a g e of five days. The clinical imp r e s s i o n w a s s e p t i c e m i a ; p r e m a i u r i t y ; harelip~ a n d cleft palate. A blood culture ( h e a r t ' s blood) t a k e n i m m e d i a t e l y a f t e r d e a t h showed colon bacilli. A n a u t o p s y w a s p e r f o r m e d t h i r t y - s i x h o u r s a f t e r death. The a b d o m e n was dist e n d e d a n d t y m p a n i t i e . The u m b i l i c a l s t u m p w a s d r y a n d firm a n d the periumbilica] t i s s u e w a s p a l e yellow; no e d e m a or c o n g e s t i o n was seen. G a s escaped w h e n t h e p e r i t o n e a l c a v i t y was opened. T h e r e were a p p r o x i m a t e l y 80 c.e. of yellow b r o w n fluid i n t h e p e r i t o n e a l cavity. T h e p e r i t o n e a l s u r f a c e of t h e i n t e s t i n e s w a s red a n d finely g r a n u l a r , a n d t h e loops of i n t e s t i n e were b o u n d t o g e t h e r b y fibrinous
46
T H E J O U R N A L OF FEDIATRICS
exudate. This e x u d a t e e x t e n d e d evenly f r o m t h e d i a p h r a g m into t h e pelvis except a t t h e r e g i o n a b o u t t h e s t o m a c h a n d spleen w h e r e it was t h i c k e r t h a n elsewhere a n d blood-tinged. W h e n t h e liver was t u r n e d back, a defect i n t h e s t o m a c h wall w a s seen e x t e n d i n g f r o m t h e i n s e r t i o n o f t h e e s o p h a g u s for a d i s t a n c e of 1.5 cm. a l o n g t h e g r e a t e r c u r v a t u r e . The e d g e of t h i s defect was d a r k g r e e n black w i t h blood clots in t h e vessels f r i n g i n g t h e opening. E x t e r n a l l y t h e s t o m a c h showed no other a b n o r m a l i t y . I n t e r n a l l y t h e m u c o s a w a s i n t a c t a n d s m o o t h except f o r t h e above a r e a which f o r m e d a n o p e n i n g 3 cm. i n circumference. T h e m u c o s a of t h e lower p o r t i o n of t h e e s o p h a g u s was deeply congested. T h e r e m a i n i n g p o r t i o n s of
Fig. 1 . - - ( C a s e 2.)
Itistologie section t h r o u g h base of ulcer, showing edge of perforation (XI5).
Fig. 2 . - - ( C a s e 2.)
ttistologie section t a k e n f a r t h e r back from b a s e of ulcer, s h o w i n g h e m o r r h a g e into tissues (X15).
9the g a s t r o i n t e s t i n a l t r a c t a p p e a r e d n o r m a l except for the fibrinous e x u d a t e on t h e serosal surfaces. T h e l u n g s showed f o c a l p n e u m o n i a . Microscopic p r e p a r a t i o n s o f t h e s t o m a c h showed no a b n o r m a l i t i e s except a t a n d a d j a c e n t to t h e p o i n t of r u p t u r e . T h e t i s s u e f o r m i n g t h e edge of t h e p e r f o r a t i o n was composed of s u b m u c o s a alone. T h i s t i s s u e s t a i n e d very lightly, only t h e outlines of cells b e i n g visible. S c a t t e r e d t h r o u g h it were s t r a n d s of fibrin a n d s m a l l collections of d a r k b l u e - s t a l n i n g bacilli. F a r t h e r f r o m t h e o p e n i n g t h e s u b m u c o s a w a s t h i c k e r a n d s o m e cellular detM1 could be m a d e o u t ; here a n d t h e r e were h e m o r r h a g e s i n t h e s u b m u c o s a ; t h e blood vessels were d i s t e n d e d ( F i g . 1). I n a section m a d e f u r t h e r f r o m t h e p o i n t of r u p t u r e ( F i g . 2) the s t r u c t u r e o f t h e i n t e s t i n e was clearly m a d e out. T h e r e w a s infiltration of m o n o n u c l e a r leucocytes into t h e m u c o s a ,
DUNI~AM AND GOLDSTEIN : RUPTURE OF STOMACI4 IN NEWBORN INFANTS -47 submucosa, and mnseularis~ hemorrhage into the submucosa~ and edema of all these tissues. Still f a r t h e r away f r o m the point of r u p t u r e the evidences of hemorrhage were less a n d less apparent and the tissue appeared normal except for the fibrinocellular exudate on the serosal surface. Sections t h r o u g h the lower portion of the esophagus showed distended blood vessels in the submucosa and serosa. There were extravasated erythrocytes about one small vessel in the serosa. Sections through the umbilicus showed no cellular reaction or edema; those of the lungs showed focal pneumonia. The anatomic diagnosis was hemorrhage in the wall of the stomach ( e a r d i a ) ; gangrene a n d perforation of the stomach; diffuse peritonitis; p r e m a t u r i t y ; cleft palate, bilateral. REVIEW OF LITERATURE
F r o m a review of the literature it is evident t h a t r u p t u r e of the stomach in y o u n g infants is a rare condition, and that in each case reDorted the r u p t u r e has been considered secondary to the presence of a gastric ulcer. Theile I in 1919 made a comprehensive review of the subject of ulcers of the stomach in children. H e f o u n d reports of 119 cases which occurred in the neonatal period (under one m o n t h ) . Perforation occurred in only 5 of these cases. R i t t e r 2 r e p o r t e d a case in 1882 which was not included in Theile's series. Three additional cases have been reported since 1919 b y Lee and Wells, ~ B u t k a 4 and Stern, P e r k i n s a n d Nesse2 The findings in the 9 cases of r u p t u r e d " g a s t r i c u l c e r " in newborn infants a l r e a d y reported, as well as in the 2 cases reported in this paper, are summarized in Table I. The age of onset of the s y m p t o m s varied between twelve hours and eleven days. F i v e of the infants were males, 2 were females, and in 4 eases the sex of the i n f a n t was not stated. Vomiting occurred in 8 eases, in 6 of which blood, and in 2 bile, was present in the vomitus. I n 4 o~ the eases the stools contained blood. Abdominal distention as a sign of peritonitis was noted in 3 eases. The " u l c e r s " were described in some eases as having r o u n d and sharp cut edges and in others as torn. The p e r f o r a t i o n occurred on the lesser c u r v a t u r e of the stomach in 4 eases, on the g r e a t e r in 6; anteriorly in 3, posteriorly in 5; n e a r the pylorus in 3, n e a r the eardla in 5, and in the middle portion of the stomach in 1 ease. The ulcers were single in all but 2 of the eases reported, those of B u t k a and of Ritter. Butka, in describing the perforation, states t h a t "other areas of necrosis of the mucosa were present but had not progressed to ulcer f o r m a t i o n . " Ritter described, besides the p e r f o r a t e d ulcer, "three black elevations" the n a t u r e of which is not clear. I n only two of the reported eases were roentgenograms of the gastrointestinal t r a c t made. I n the ease r e p o r t e d b y Stern, Perkins and Nesse, the infant, who vomited bile on the d a y a f t e r b i r t h and developed abdominal distention the following day, w~s giveri b a r i u m by stomach tube
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TItE
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JOURNAL
OF PEDIATRICS
DUNHAlYi AND GOLDSTEIN:
RUPTURE OF STOMACH IN NEWBOI~N INFANTS
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and hourly films were taken. A persistent niche was observed on the lesser curvature of the stomach near the pylorus as well as an accumulation of free gas along the lateral abdominal walls. A diag~aosis of gastric perforation was made. A laparotomy was performed and a perforation a quarter of an inch in diameter on the lesser curvature posteriorly was found and closed, but the infant died three days later. In the case reported by Lee and Wells, a diagnosis of pyloric obstruction was made by roentgenogram. At operation, adhesions of the intestine to the pylorus were found, but it was not until postmortem examination was made that a perforation was found on the posterior wall of the stomach. The authors concluded that a~ ulcer o~ the stomach, ruptured during intrauterine life, had become walled off by adhesions. In those cases in which histologic examinations of the stomach at the site of perforation were made, the change~ described were those of necrosis and hemorrhage into the tissue near the point of rupture. A cellular reaction similar to that found in peptic ulcer has not been found nor have evidences of thrombosis or embolism. Blood cultures have not been taken in any of the cases reported. ETIOLOGY
The cause of "ulcers" of the stomach in young infants is obscure. Shore, 6 in discussing these lesions in children, regards the mechanism of their formation as a local disturbance in circulation which, from the action of the gastric juice, leads to necrosis and ulceration, lie mentions as causes of this vascular disturbance congestion, embolism, thrombosis, vascular disease, direct injury to the mueosa, and nervous influences. Weiss and MallorS have recently called attention to vomiting as a cause of lacerations or ulcers in the cardiac end of the stomach. Fulton s has shown a relation between focal lesions in the hypotha]amus and ulcers in the gastrointestinal tract. The possibility of some congenital defect in the structure of the stomach as the underlying cause of rupture must be considered. In the light of the well-known occurrence of socalled "spontaneous rupture of the intestine" in the newborn infant, which is due probably to obstruction and trauma during birth, as reported by RusselP and others, a similar mechanism perhaps should be considered in connection with rupture of the stomach. No case reports of such " s pont aneous " rupture of the stomach have, however, been found. It has not been possible to demonstrate that any of the etiologic factors suggested by these authors played a rSle in b r i n i n g about rupture of the stomach in the cases here reported. There was no history of asphyxia at birth which might lead to congestion nor was there evidence of thrombosis or embolism found postmortem. ~[oreover, in spite of the fact that both of the infants had a bacteremia, evidence was inconclusive
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TI=IE J O U t C N A L OF P E D I A T R I C S
that a general blood stream infection played a rhle, since thrombi were not demonstrable in the bases of the ulcers; and, although organisms were demonstrated in the tissue around the ulcer in one case, there was no polymorphonuelear reaction. Direct t r a u m a to the mucous membrane of the stomach was considered as a possible cause of the ulceration in one of our cases (Case 2) because a tube was used for garage and lavage before the abdominal distention appeared, but it seems highly improbable that a normal mucous membrane could have been damaged by a soft rubber catheter. I n the other case, although a tube was introduced into the stomach, t r a u m a from the tube could not have caused rupture, because signs of peritonitis developed before the use of the tube. Vomiting occurred as one of the initial symptoms in both of the cases reported, but was regarded as probably the result rather t h a n the cause of the abdominal condition. The brain was examined postmortem in one case (Case 2), but no evidence of a lesion of the hypothalamus could be demonstrated. SUI~I 1VIARY
R u p t u r e of the stomach in newborn infants is a rare condition, the etiology of which is obscure. The diagnosis of r u p t u r e or of the lesions which lead to it is difficult, but, in the presence of persistent vomiting and abdominal distention, a roentgenbgram of the abdomen should always be taken without administration of barium. B y this means the presence of free air in the peritoneal cavity--evidence that r u p t u r e has already taken p l a c e - - m a y be determined. I f there is no evidence of free air and the severity of symptoms justifies it, a barium meal m a y be given or an exploratory operation performed with the hope of determining tile cause of the symptoms and of preventing r u p t u r e of the stomach. REFERENCES
1. 2. 3. 4. 5. 6. 7. 8. 9.
Theile, P.: Ergebn. d. inn. Med. u. Kinderh. 16: 302, 1919. Ritter, H.: Aerztl. Mitt. a. Baden 36: 15, 1882. Lee, W. E, and Wells, J. I%.: Ann. Surg. 78: 36, 1923. Butka~ It. E.: J . A . IV[. A. 89: 198, 1927. Stern, M. A., Perkins, E.L., and Nesse, N. S.: Journal-Lancet 49: 492~ 1929. Shore, B. Ir Ann. Sm'g. 92: 234, 1930. Weiss, S, and Mallory, G.X.: J.A. 1VI.A. 98: 1353, ]932. Fulton, J. F.: New England J. Med. 207: 60, 1932. Russell, T. I-I.: Proc. Connecticut M. Soe., 1927, p. 178.
WO cases of rupture of the stomach occurring in newborn infants T are here reported because of the rarity of the condition. As will be pointed out, only 9 other such cases have been described. Abstracts of the case histories with clinical and pathologic findings follow. CASE 1.--N. S., a white male, was borll Sept. 7~ 1929, in the l'~ew H a v e n Hospital. Tho mother developed a severe toxemia and was delivered at term by eesar~an section. The i n f a n t weighed 4~,166 gm. a n d appeared normal at birth. On the f o u r t h day of life the temperature became elevated. The i n f a n t vomited and about six hours later became acutely ill and was f o u n d to be distended and to have edema of the abdominal wall. The leucocyte count was 4,000, polymorphonuclears 38 per cent, tymphocytes 53 per cent. No definite clinical diagnosis was made at this time, although the symptoms could best be explained by peritonitis. A subcutaneous injection of saline, and a t r a n s f u s i o n of blood were then given and the i n f a n t ' s stomach was lavagcd. The following day, although the i n f a n t ' s condition seemed somewhat improved, the distention and vomiting continued. At this t i m e a roentgenogram, taken a f t e r a rectal injection of barium had been given, showed t h a i there was no evidence of obstruction in the large intestine. Reexamination of the film several days lateT revealed the f a c t t h a t a small amount of air h a d been present in the peritoneal cavity at ~he time the roentgenogram was taken. D u r i n g this period, however, the i n f a n t ' s condition had greatly improved~ the abdominal distention had decreased, stools were passed daily~ fluids were taken well, and there was no vomiting. Th6 temperature remained normal most of the time. A blood culture taken two days a f t e r the onset of the illness showed B. vo/~, as did six subsequent cultures made on the sixth, ninth, fourteenth, slxteenth, seventeenth, and eighteenth days. On the thirteenth day of life a mass was palpated in the left flank and a n exploratory operation was performed. On going through the peritoneum i n the region where the mass had been felt there was an escape of gas and of thin yellowish fluid having the odor of colon bacillus pus. No perforation was found in the intestines, but an accumulation of pus was found walled off near the upper pole of the left kidney. This was t h o u g h t to be an abscess and was opened and drained. On the following day a f t e r the first feeding, undigested milk appeared in the wound. The presence of a fistulous opening was confirmed by fluoroscopic examination after a barium meal. l~rom the Departments of Pediatrics and Pathology, Yale University SChool of Medicine, and U. S. Children's Bureau (Studies of Neonatal !Y[orbidity and !~Iortality). 44
DUI~HAM AI~D GOLDSTEIN : RUPTUI~E OF STOMACH IN NEWBOtCN INFAI~TS
45
A n a t t e m p t w a s m a d e to r e p a i r t h e p e r f o r a t i o n i n t h e stomach, b u t t h i s was unsuccessful. Blood cultures c o n t i n u e d to be positive f o r B. roll, a n d the i n f a n t died on t h e t w e n t y - f i r s t day. T h e final clinical d i a g n o s i s was r u p t u r e of t h e stomach, peritonitis, B. ooli s efp t ~ee~nial. A n a u t o p s y w a s p e r f o r m e d f o u r h o u r s a f t e r death. T h e skin a n d s u b c u t a n e o u s tissue over t h e a b d o m e n were edematous. T h e r e w a s no f r e e fluid in the p e r i t o n e a l cavity. T h e s t o m a c h , t r a n s v e r s e a n d d e s c e n d i n g colon~ a n d several coils of small i n t e s t i n e were b o u n d t o g e t h e r b y fibrinous a n d fibrous adhesions. W h e n t h e s t o m a c h was d i s s e c t e d free, a s u t u r e line m e a s u r i n g 1 am. w a s seen on the g r e a t e r c u r v a t u r e at t h e cardia. O n f r e e i n g t h e spleen several small p o c k e t s o f thick yellow p u s were e n c o u n t e r e d b e t w e e n t h e spleen a n d t h e d i a p h r a g m . S i m i l a r pockets of p u s were s c a t t e r e d b e t w e e n t h e coils of i n t e s t i n e in this region. W h e n t h e s t o m a c h w a s opened, i t w a s seen t h a t t h e m u e o s a , except for the site of r u p t u r e , was i n t a c t a n d t h e u s u a l r u g a e were present. T h e r e m a i n i n g p o r t i o n s of the g a s t r o i n t e s t i n a l t r a c t showed no a b n o r m a l i t i e s aside f r o m t h e serosal a n d p e r i t o n e a l reaction. Serial sections were t a k e n t h r o u g h t h e a r e a of r u p t u r e . The g a s t r i c m u c o s a u p to t h e edge o f t h e r u p t u r e a p p e a r e d n o r m a l . T h e r e was no cellular infiltration or h e m o r r h a g e . T h e p o i n t of rupture, w a s s u r r o u n d e d b y a t h i n wall of h o m o g e n e o u s , necrotic d~bris w h i c h took t h e eosin s t a i n a n d w a s s u r r o u n d e d b y a n a r r o w zone of tissue i n f i l t r a t e d w i t h m o n o n u c l e a r leucocytes a n d e x t r a v a s a t e d red blood cells. F a r t h e r f r o m t h e lesion t h e m u c o s a a n d deeper ]avers of the s t o m a c h were apparently normal. Sections of t h e j e j u n u m a n d ileum showed t h a t t h e serosal s u r f a c e s were t h i c k e n e d b y a n infiltration o f m o n o n u c l e a r leucocytes; t h e m u c o s a e were i n t a c t a n d f r e e f r o m cellular infiltration or h e m o r r h a g e . Sections of t h e l u n g s showed f o c a l p n e u m o n i a . The a n a t o m i c d i a g n o s i s was p e r f o r a t i o n of t h e s t o m a c h ; localized p e r i t o n e a l a b s c e s s ; s u b d i a p h r a g m a t i c a b s c e s s ( l e f t ) ; p a r t i a l o b s t r u c t i o n of t h e s i g m o i d colon; fecal fistula; f o c a l p n e u m o n i a ; recent l a p a r o t o m y f o r d r a i n a g e of p e r i t o n e a l cavity, a n d s u t u r e of s t o m a c h . CASE 2 . - - B . B., a white male, was b o r n p r e m a t u r e l y Sept. ]1, 1930, in t h e _New H a v e n H o s p i t a l . T h e m o t h e r was a h e a l t h y p r i m i p a r a , t w e n t y - o n e y e a r s old. The labor w a s n o r m a l a n d t h e delivery s p o n t a n e o u s . T h e b i r t h w e i g h t was 1,960 g m . T h e i n f a n t ' s g e n e r a l condition was good, b u t t h e t e m p e r a t u r e was s u b n o r m a l (35.6 ~ C.). O n e x a m i n a t i o n t h e i n f a n t , a l t h o u g h small, a p p e a r e d n o r m a l except f o r a b i l a t e r a l h a r e l i p a n d cleft palate. F e e d i n g w i t h modified c o w ' s milk w a s b e g u n a b o u t twelve h o u r s a f t e r birth. T h e i n f a n t v o m i t e d bile-stained m a t e r i a l following t h e first f e e d i n g a n d c o n t i n u e d to v o m i t a f t e r s u b s e q u e n t feedings. Abdominal distention and increased peristalsis appeared about thirty hours after birth. Two h y p o d e r m o c l y s e s were g i v e n a n d at t h l r t y - e i g h t h o u r s l a v a g e a n d g a v a g e were b e g u n a n d c o n t i n u e d t h r o u g h t h e n e x t day. A blood culttu'e t a k e n on t h e t h i r d d a y of life showed colon bacilli in b r o t h a n d plates. A r o e n t g e n o g r a m of t h e a b d o m e n (fiat plate) showed a l a r g e q u a n t i t y o f g a s in t h e s t o m a c h a n d i n t e s t i n a l t r a c t . T h e W a s s e r m a n n t e s t on t h e blood s e r u m was negative. A t t a c k s of c y a n o s i s developed, a n d t h e i n f a n t died at t h e a g e of five days. The clinical imp r e s s i o n w a s s e p t i c e m i a ; p r e m a i u r i t y ; harelip~ a n d cleft palate. A blood culture ( h e a r t ' s blood) t a k e n i m m e d i a t e l y a f t e r d e a t h showed colon bacilli. A n a u t o p s y w a s p e r f o r m e d t h i r t y - s i x h o u r s a f t e r death. The a b d o m e n was dist e n d e d a n d t y m p a n i t i e . The u m b i l i c a l s t u m p w a s d r y a n d firm a n d the periumbilica] t i s s u e w a s p a l e yellow; no e d e m a or c o n g e s t i o n was seen. G a s escaped w h e n t h e p e r i t o n e a l c a v i t y was opened. T h e r e were a p p r o x i m a t e l y 80 c.e. of yellow b r o w n fluid i n t h e p e r i t o n e a l cavity. T h e p e r i t o n e a l s u r f a c e of t h e i n t e s t i n e s w a s red a n d finely g r a n u l a r , a n d t h e loops of i n t e s t i n e were b o u n d t o g e t h e r b y fibrinous
46
T H E J O U R N A L OF FEDIATRICS
exudate. This e x u d a t e e x t e n d e d evenly f r o m t h e d i a p h r a g m into t h e pelvis except a t t h e r e g i o n a b o u t t h e s t o m a c h a n d spleen w h e r e it was t h i c k e r t h a n elsewhere a n d blood-tinged. W h e n t h e liver was t u r n e d back, a defect i n t h e s t o m a c h wall w a s seen e x t e n d i n g f r o m t h e i n s e r t i o n o f t h e e s o p h a g u s for a d i s t a n c e of 1.5 cm. a l o n g t h e g r e a t e r c u r v a t u r e . The e d g e of t h i s defect was d a r k g r e e n black w i t h blood clots in t h e vessels f r i n g i n g t h e opening. E x t e r n a l l y t h e s t o m a c h showed no other a b n o r m a l i t y . I n t e r n a l l y t h e m u c o s a w a s i n t a c t a n d s m o o t h except f o r t h e above a r e a which f o r m e d a n o p e n i n g 3 cm. i n circumference. T h e m u c o s a of t h e lower p o r t i o n of t h e e s o p h a g u s was deeply congested. T h e r e m a i n i n g p o r t i o n s of
Fig. 1 . - - ( C a s e 2.)
Itistologie section t h r o u g h base of ulcer, showing edge of perforation (XI5).
Fig. 2 . - - ( C a s e 2.)
ttistologie section t a k e n f a r t h e r back from b a s e of ulcer, s h o w i n g h e m o r r h a g e into tissues (X15).
9the g a s t r o i n t e s t i n a l t r a c t a p p e a r e d n o r m a l except for the fibrinous e x u d a t e on t h e serosal surfaces. T h e l u n g s showed f o c a l p n e u m o n i a . Microscopic p r e p a r a t i o n s o f t h e s t o m a c h showed no a b n o r m a l i t i e s except a t a n d a d j a c e n t to t h e p o i n t of r u p t u r e . T h e t i s s u e f o r m i n g t h e edge of t h e p e r f o r a t i o n was composed of s u b m u c o s a alone. T h i s t i s s u e s t a i n e d very lightly, only t h e outlines of cells b e i n g visible. S c a t t e r e d t h r o u g h it were s t r a n d s of fibrin a n d s m a l l collections of d a r k b l u e - s t a l n i n g bacilli. F a r t h e r f r o m t h e o p e n i n g t h e s u b m u c o s a w a s t h i c k e r a n d s o m e cellular detM1 could be m a d e o u t ; here a n d t h e r e were h e m o r r h a g e s i n t h e s u b m u c o s a ; t h e blood vessels were d i s t e n d e d ( F i g . 1). I n a section m a d e f u r t h e r f r o m t h e p o i n t of r u p t u r e ( F i g . 2) the s t r u c t u r e o f t h e i n t e s t i n e was clearly m a d e out. T h e r e w a s infiltration of m o n o n u c l e a r leucocytes into t h e m u c o s a ,
DUNI~AM AND GOLDSTEIN : RUPTURE OF STOMACI4 IN NEWBORN INFANTS -47 submucosa, and mnseularis~ hemorrhage into the submucosa~ and edema of all these tissues. Still f a r t h e r away f r o m the point of r u p t u r e the evidences of hemorrhage were less a n d less apparent and the tissue appeared normal except for the fibrinocellular exudate on the serosal surface. Sections t h r o u g h the lower portion of the esophagus showed distended blood vessels in the submucosa and serosa. There were extravasated erythrocytes about one small vessel in the serosa. Sections through the umbilicus showed no cellular reaction or edema; those of the lungs showed focal pneumonia. The anatomic diagnosis was hemorrhage in the wall of the stomach ( e a r d i a ) ; gangrene a n d perforation of the stomach; diffuse peritonitis; p r e m a t u r i t y ; cleft palate, bilateral. REVIEW OF LITERATURE
F r o m a review of the literature it is evident t h a t r u p t u r e of the stomach in y o u n g infants is a rare condition, and that in each case reDorted the r u p t u r e has been considered secondary to the presence of a gastric ulcer. Theile I in 1919 made a comprehensive review of the subject of ulcers of the stomach in children. H e f o u n d reports of 119 cases which occurred in the neonatal period (under one m o n t h ) . Perforation occurred in only 5 of these cases. R i t t e r 2 r e p o r t e d a case in 1882 which was not included in Theile's series. Three additional cases have been reported since 1919 b y Lee and Wells, ~ B u t k a 4 and Stern, P e r k i n s a n d Nesse2 The findings in the 9 cases of r u p t u r e d " g a s t r i c u l c e r " in newborn infants a l r e a d y reported, as well as in the 2 cases reported in this paper, are summarized in Table I. The age of onset of the s y m p t o m s varied between twelve hours and eleven days. F i v e of the infants were males, 2 were females, and in 4 eases the sex of the i n f a n t was not stated. Vomiting occurred in 8 eases, in 6 of which blood, and in 2 bile, was present in the vomitus. I n 4 o~ the eases the stools contained blood. Abdominal distention as a sign of peritonitis was noted in 3 eases. The " u l c e r s " were described in some eases as having r o u n d and sharp cut edges and in others as torn. The p e r f o r a t i o n occurred on the lesser c u r v a t u r e of the stomach in 4 eases, on the g r e a t e r in 6; anteriorly in 3, posteriorly in 5; n e a r the pylorus in 3, n e a r the eardla in 5, and in the middle portion of the stomach in 1 ease. The ulcers were single in all but 2 of the eases reported, those of B u t k a and of Ritter. Butka, in describing the perforation, states t h a t "other areas of necrosis of the mucosa were present but had not progressed to ulcer f o r m a t i o n . " Ritter described, besides the p e r f o r a t e d ulcer, "three black elevations" the n a t u r e of which is not clear. I n only two of the reported eases were roentgenograms of the gastrointestinal t r a c t made. I n the ease r e p o r t e d b y Stern, Perkins and Nesse, the infant, who vomited bile on the d a y a f t e r b i r t h and developed abdominal distention the following day, w~s giveri b a r i u m by stomach tube
48
TItE
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JOURNAL
OF PEDIATRICS
DUNHAlYi AND GOLDSTEIN:
RUPTURE OF STOMACH IN NEWBOI~N INFANTS
~9
and hourly films were taken. A persistent niche was observed on the lesser curvature of the stomach near the pylorus as well as an accumulation of free gas along the lateral abdominal walls. A diag~aosis of gastric perforation was made. A laparotomy was performed and a perforation a quarter of an inch in diameter on the lesser curvature posteriorly was found and closed, but the infant died three days later. In the case reported by Lee and Wells, a diagnosis of pyloric obstruction was made by roentgenogram. At operation, adhesions of the intestine to the pylorus were found, but it was not until postmortem examination was made that a perforation was found on the posterior wall of the stomach. The authors concluded that a~ ulcer o~ the stomach, ruptured during intrauterine life, had become walled off by adhesions. In those cases in which histologic examinations of the stomach at the site of perforation were made, the change~ described were those of necrosis and hemorrhage into the tissue near the point of rupture. A cellular reaction similar to that found in peptic ulcer has not been found nor have evidences of thrombosis or embolism. Blood cultures have not been taken in any of the cases reported. ETIOLOGY
The cause of "ulcers" of the stomach in young infants is obscure. Shore, 6 in discussing these lesions in children, regards the mechanism of their formation as a local disturbance in circulation which, from the action of the gastric juice, leads to necrosis and ulceration, lie mentions as causes of this vascular disturbance congestion, embolism, thrombosis, vascular disease, direct injury to the mueosa, and nervous influences. Weiss and MallorS have recently called attention to vomiting as a cause of lacerations or ulcers in the cardiac end of the stomach. Fulton s has shown a relation between focal lesions in the hypotha]amus and ulcers in the gastrointestinal tract. The possibility of some congenital defect in the structure of the stomach as the underlying cause of rupture must be considered. In the light of the well-known occurrence of socalled "spontaneous rupture of the intestine" in the newborn infant, which is due probably to obstruction and trauma during birth, as reported by RusselP and others, a similar mechanism perhaps should be considered in connection with rupture of the stomach. No case reports of such " s pont aneous " rupture of the stomach have, however, been found. It has not been possible to demonstrate that any of the etiologic factors suggested by these authors played a rSle in b r i n i n g about rupture of the stomach in the cases here reported. There was no history of asphyxia at birth which might lead to congestion nor was there evidence of thrombosis or embolism found postmortem. ~[oreover, in spite of the fact that both of the infants had a bacteremia, evidence was inconclusive
50
TI=IE J O U t C N A L OF P E D I A T R I C S
that a general blood stream infection played a rhle, since thrombi were not demonstrable in the bases of the ulcers; and, although organisms were demonstrated in the tissue around the ulcer in one case, there was no polymorphonuelear reaction. Direct t r a u m a to the mucous membrane of the stomach was considered as a possible cause of the ulceration in one of our cases (Case 2) because a tube was used for garage and lavage before the abdominal distention appeared, but it seems highly improbable that a normal mucous membrane could have been damaged by a soft rubber catheter. I n the other case, although a tube was introduced into the stomach, t r a u m a from the tube could not have caused rupture, because signs of peritonitis developed before the use of the tube. Vomiting occurred as one of the initial symptoms in both of the cases reported, but was regarded as probably the result rather t h a n the cause of the abdominal condition. The brain was examined postmortem in one case (Case 2), but no evidence of a lesion of the hypothalamus could be demonstrated. SUI~I 1VIARY
R u p t u r e of the stomach in newborn infants is a rare condition, the etiology of which is obscure. The diagnosis of r u p t u r e or of the lesions which lead to it is difficult, but, in the presence of persistent vomiting and abdominal distention, a roentgenbgram of the abdomen should always be taken without administration of barium. B y this means the presence of free air in the peritoneal cavity--evidence that r u p t u r e has already taken p l a c e - - m a y be determined. I f there is no evidence of free air and the severity of symptoms justifies it, a barium meal m a y be given or an exploratory operation performed with the hope of determining tile cause of the symptoms and of preventing r u p t u r e of the stomach. REFERENCES
1. 2. 3. 4. 5. 6. 7. 8. 9.
Theile, P.: Ergebn. d. inn. Med. u. Kinderh. 16: 302, 1919. Ritter, H.: Aerztl. Mitt. a. Baden 36: 15, 1882. Lee, W. E, and Wells, J. I%.: Ann. Surg. 78: 36, 1923. Butka~ It. E.: J . A . IV[. A. 89: 198, 1927. Stern, M. A., Perkins, E.L., and Nesse, N. S.: Journal-Lancet 49: 492~ 1929. Shore, B. Ir Ann. Sm'g. 92: 234, 1930. Weiss, S, and Mallory, G.X.: J.A. 1VI.A. 98: 1353, ]932. Fulton, J. F.: New England J. Med. 207: 60, 1932. Russell, T. I-I.: Proc. Connecticut M. Soe., 1927, p. 178.