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Hemorrhage from Meckel's diverticulum
H E M O R R H A G E FROM M E C K E L ' S DIVERTICULUM R. A. th~GoNs, M.D., AND ~I. E. PORT CHESTER,
GUNDY,M.D.
N. Y.
1701 Ruysch first described the existence of an anatomic structure I N which later was described more fully by Meckel in 1812, and has since been called by his name. According to Arey, in the early embryo of 1.5 ram. to 2 mm., with the development of the fore-and hind-gut, the entodermal vesicle is divided into a dorsal intestine and a ventral yolk sac, with a narrowed portion between. With the growth of the embryo and the yolk sac, the connecting portion does not increase much in size, and eventually becomes a slender yolk stalk. This yolk stalk later becomes a slender thread extending from the junction of the fore- and hind-gut to the yolk sac, or umbilical vesicle. It loses its attachment to the gut in the 7 ram. embryo. A blind pocket may persist .at the point of its union with the intestine. This is known as Meckel's diverticulum. The blind end of this pouch may remain adherent to the umbilicus, or may separate and lie free in the abdominal cavity. The lumen of the pouch is continuous with that of the ileum, about 80 cm. above the ileocecal vane. We feel that hemorrhage from such diverticulum is of sufficient rari t y to warrant this report of the following two cases. CASE 1.--M. U., a male, 6 years old, of Polish extraction, was first seen Jan. 25, 1937. About one year previouw the child was taken to a physician because of a small amount of clark blood noticed in the stool. This was thought to originate in a rectal fissure, and did not recur. Uncomplicated chickenpox occurred three weeks before admission. Four days previous to admission the mother noticed some dark red blood in the stool The child showed no other symptoms, and had no increase in the number of stools. Two days before admission the blood recurred in the stool and was more plentiful. The child still showed no other untoward symptoms. The day preceding admission he had no stools and seemed well. On the day of admisMon he had a large stool in the morning which consisted almost entirely of dark blood. He showed no evidence of acute illness until noon, when he became pale and weak. I-Ie refused luncheon, vomited once during the afternoon, and became rapidly paler and weaker, although he had no further stools, tie had no pain at any t~me. The child was seen by the family physician who referred him to the hospital. Physiea~ Examination.--On admission at 5 p.~. on /January 25, the child appeared dangerously ill. The facies were drawn and anxious, and the skin, lips, and mucous membranes were pale and dry. The abdomen was flat, soft, not tender, and no masses or rigidity could be palpated. Rectal examination was negative except for the presence of considerable old blood. The temperature w~s 101 ~ F.; the puls% 130; and the respirations, 22. The remainder of the physical examination was entirely negative. Laboratory Findings.--Itemeglobin, 25 per cent (Sahli); red blood cells, 2,270,000; white blood cells, 23,000; polymorphonuclears, 82 per cent; lymphoeytes, 18 per cent; no abnormal white cells; red cells showed poikilocytosls and anisocytosis, 563
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b u t no n o r n m b l a s t s . P l a t e l e t s 184,000; n o r m a l clot r e t r a c t i o n ; c o a g u l a t i o n time, three a n d one-half m i n u t e s . B l e e d i n g time w a s n o t increased. Blood was t y p e 2 Morse. T h e u r i n e w a s n o r m a l except f o r h i g h specific g r a v i t y a n d 3 plus acetone. Clinical Course.--The child w a s given a blood t r a n s f u s i o n a t once, receiving 360 c.c. of whole blood b y direct method. A low e n e m a yielded n o t h i n g b u t clotted, a n d p a r t i a l l y d e s t r o y e d blood. The l a b o r a t o r y reported t h a t this stool showed red blood cells in all s t a g e s of d i s i n t e g r a t i o n , w i t h a f e w a p p a r e n t l y n o r m a l red cells. The stool was n e g a t i v e f o r ova a n d p a r a s i t e s . T h e n e x t m o r n i n g the color w a s improved, a n d t h e h e m o g l o b i n was 39 p e r cent ( S a h l i ) w i t h red blood cells n u m b e r i n g 2,850,000. T e m p e r a t u r e was 9 9 ~ pulse, 105; respiration, 18. A n e n e m a yielded a l a r g e a m o u n t of old blood. There w a s no s p o n t a n e o u s stool. I I e received 400 c.c. of whole blood b y direct t r a n s f u s i o n . T h a t e v e n i n g he h a d a large, d a r k red, liquid stool a n d a g a i n showed s i g n s of shock. I t w a s decided n o t to postpone operation a n y longer, as he was suspected of h a v i n g f r e s h b l e e d i n g into t h e bowel f r o m a p e r s i s t e n t M e c k e l ' s diverticulum. A t i A.M. on J a n . 27, 1937, the a b d o m e n w a s opened. The p e r i t o n e a l c a v i t y cont a i n e d a s m a l l a m o u n t of clear fluid. T h e whole colon a p p e a r e d relaxed a n d atonie a n d was d i s t e n d e d w i t h d a r k blood, which showed t h r o u g h its wall. A b o u t 15 inches p r o x i m a l to the ileocecal valve, w a s f o u n d a l a r g e d i v e r t i c u l u m a b o u t 4 inches long, a n d of the s a m e d i a m e t e r as the a d j a c e n t ileum. T h e d i v e r t i c u l u m w a s clamped, ligated, a n d removed. I t s s t u m p was cauterized a n d inverted w i t h a p u r s e - s t r i n g
suture. T h e t e m p e r a t u r e dropped to n o r m a l t w e n t y - f o u r h o u r s a f t e r operation a n d t h e p o s t o p e r a t i v e course w a s u n e v e n t f u l . T h e h e m o g l o b i n rose to 64 per cent ( S a h l i ) w i t h a red blood c o u n t of 4,400,000 on t h e n i n t h day.' The child was d i s c h a r g e d in good condition on t h e eleventh day, a n d h a s r e m a i n e d well since. Pathologic Report by Dr. Margaret Loder.--Macroscopically t h e s p e c i m e n consisted of a d i v e r t i c u l u m m e a s u r i n g 50 to 60 ram. in l e n g t h . A p p r o x i m a t e l y 30 ram. f r o m the closed end, t h e m u e o s a showed a n u l c e r a t e d a r e a 8 to 10 ram. in d i a m e t e r , w i t h a n increase of fibrous t i s s u e in t h e wall, a n d some constriction. Definite bleeding p o i n t s a p p e a r e d in t h e floor of t h e ulcer. T h e m u c o s a a r o u n d t h e edges of t h e ulcer a p p e a r e d congested. Microscopically t h e sections showed a definite u l c e r a t i o n of t h e mucosa, w i t h a n increase of a d u l t fibrous tissue in the s u b m u c o s a a n d m u s c u l a r i s . Leucocytie infiltration w a s p r e s e n t at t h e base o f t h e ulcer, p e r i v a s c n l a r a n d m o d e r a t e diffuse infiltration in the s t r o m a of the s u b m u c o s a , a n d infiltration to a lesser e x t e n t in t h e m u s c u l a r i s . The m u c o s a a n d l i n i n g e p i t h e l i u m were typical of t h a t f o u n d in the a d j a c e n t ileum. A n a r e a in which the s u r f a c e g l a n d s were lined by a l a r g e clear cell, u s u a l l y f o u n d i n t h e peptic g l a n d s of t h e stomach, was f o u n d n e a r t h e edge of the ulcer i n t h e sections examined. Pathologic D~agnosis.--Bleeding ulcer in a p e r s i s t e n t MeckeI's diverticulum. CASE 2 . - - M a c M . , a male two y e a r s of age, of A m e r i c a n p a r e n t a g e .
Present Illness.--On J a n . 28, 1937, t h e child w a s given E x q a x because of constipation. O n J a n u a r y 29 he h a d f o u r bowel m o v e m e n t s , three of w h i c h c o n t a i n e d blood. T h e f a t h e r t h o u g h t ' t h a t the blood was ~ ' s o m e w h a t b r i g h t . " The next day t h e child h a d only one stool which contained blood. H i s a p p e t i t e r e m a i n e d f a i r l y n o r m a l a n d he h a d no s y m p t o m s t h a t a t t r a c t e d t h e p a r e n t s ' notice except f o r one emesis. H e h a d no stools on J a n u a r y 3], nor o~L F e b r u a r y 1. The child s u d d e n l y became v e r y weak, pale, a n d l a n g u i d t o w a r d e v e n i n g of F e b r u a r y :[. H e was adm i t t e d to t h e hospital a t 5 P.~L Physical Examination.--Temperature was 100 ~ F . ; pulse, 140; respiration, 30. Color w a s v e r y p a l e ; t h e child, v e r y quiet a n d a p a t h e t i c ; lips, skin, a n d m u c o u s m e m b r a n e s were v e r y pale a n d dry. B l o w i n g systolic m u r m u r w a s h e a r d at the
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cardiac apex. A b d o m e n was flat, sol% n o t tender, and showed no palpable m a s s or rigidity. Rectal e x a m i n a t i o n was n e g a t i v e except f o r t h e presence of old blood in l a r g e a m o u n t s . The r e m a i n d e r of the physicM e x a m i n a t i o n was irrelevant. Laboratory Findings.--ltemoglobin, 33 per cent ( S a h l i ) ; red blood cells, 2,300,000; white blood cells, 10~200; p o l y m o r p h o n u c l e a r s , 39 per c e n t ; lymphocytes~ 51 per cent; n o r m o b l a s t s , 10 per cent. Blood was type 4 Morse. The u r i n e showed a l b u m i n plus, a n d occasional h y M i n casts. Clinical Course.--One h u n d r e d f o r t y cubic c e n t i m e t e r s of whole blood w a s g i v e n by direct t r a n s f u s i o n on a d m i s s i o n , a n d 150 e.c. t h e next m o r n i n g . A d i a g n o s i s of h e m o r r h a g e f r o m a p e r s i s t e n t M e e k e l ' s d i v e r t i c u l u m was made, a n d the a b d o m e n w~s opened at 4 L~z. on F e b r u a r y 2. A smM1 diverticulum, a b o u t the size o f t h e distal p h a l a n x of t h e t h u m b , was f o u n d a b o u t 10 inches p r o x i m a l to t h e ileocecat valve. This was clamped, ligated~ removed , a n d t h e base cauterized a n d i n v e r t e d by a p u r s e - s t r i n g s u t u r e . The d i v e r t i c u l u m was opened i n t h e o p e r a t i n g r o o m a n d clotted Mood f o u n d in its lumen. The appendix~ which a p p e a r e d normM, w a s removed in t h e u s u a l m a n n e r . T h e i m m e d i a t e p o s t o p e r a t i v e course was u n e v e n t f u l , a n d the h e m o g l o b i n rose to 45 per cent (Sahli) i~l t w e n t y - f o u r hours. O n t h e t h i r d p o s t o p e r a t i v e d a y he seemed restless, r e f u s e d food, v o m i t e d once, a n d the t e m p e r a t u r e rose to 102.6 ~. E x a m i n a t i o n showed a n acute n a s o p h a r y n g i t i s . On the f o u r t h d a y p o s t o p e r a t i v e he developed loose g r e e n stools w i t h m u c u s b u t no blood. T h i s condition p e r s i s t e d for f o u r d a y s with t e m p e r a t u r e f l u c t u a t i n g b e t w e e n 100 a n d 102 ~ a n d g r a d u a l l y f a l l i n g to n o r m a l on t h e e i g h t h p o s t o p e r a t i v e day. H e was d i s c h a r g e d on F e b r u a r y 16, f o u r t e e n d a y s following operation, a p p a r e n t l y in good condition. T h r o u g h some oversight, t h e blood count was n o t r e p e a t e d before discharge. Pathologic l~eport by Dr. Loder.--Macroseopieally the speeiinens s u b m i t t e d consisted o f : ( a ) A short, narrow, d i v e r t i c u l u m m e a s u r i n g 20 ram. in length. T h e specimen h a d been sectioned lengthwise, ~nd t h e c o n t e n t s lost. The m u e o s a was smooth t h r o u g h o u t . T h e wall ~ p p e a r e d to be m o d e r a t e l y thickened. There w a s no evidence of acute i n f l a m m a t i o n . ( b ) A n a p p e n d i x s h o w i n g some h y p e r p l a s i a of the l y m p h o i d tissue~ a n d a n otherwise g r o s s l y n o r m a l wall. D a r k feces was f o u n d in the h m e n . Microscopically t h e sections f r o m the d i v e r t i c u l u m showed a mucosa, lined by epithelium similar to t h a t of t h e a d j a c e n t ileum. The s u r f a c e e p i t h e l i u m of t h e sections e x a m i n e d a p p e a r e d intact. There was art increase of fibrous tissue i n t h e submucosa, m u s e u l a r i s , a n d subser0sa. P e r i v a s e u l a r a n d m o d e r a t e l y diffuse infiltration, c o n s i s t i n g of small r o u n d cells, p l a s m a cells, a n d a few p o l y n u e l e a r leucocytes, w a s f o u n d in the s t r o m a of t h e mucosa, s u b m u c o s a , m u s c u l a r i s , and subserosa. Sections f r o m t h e a p p e n d i x showed p e r i v a s e u l a r infiltration in t h e outer m u s c u l a r , a n d s u b s e r o u s layers. The l y m p h o i d tissue was hyperplastic. Diagnosis. Chronic M e c k e l ' s diverticulitis. Mild chronic appendicitis. COMMENT
We feel that these cases should be reported because they are typical of one of the true pediatric emergencies. W i t h o u t adequate t r e a t m e n t these children m i g h t well be lost, as has been reported by Callender ~ and Taylor. 2 The first ease reported was certainly in a very critical condition when admitted, and a guarded prognosis was given. I t was considered poor surgical j u d g m e n t to operate on this child immediately u p o n admission, due to the existing condition of shock. A p p a r e n t l y this child lost his blood very rapidly, as is indicated by the Complete absence of any signs of blood regeneration in the blood smear.
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The second case reported was in better physical condition upon admission. H e had apparently lost less blood, and had probably lost it more slowly as indicated by the normoblasts seen in the blood smear. There are certain similarities in these two cases which may be considered as typical of cases of severe hemorrhage from a persistent Meckel's diverticulum. These might be enumerated as follows: 1. The onset is often insidious, and usually without subjective symptoms. Mason and Graham 3 noted that the bleeding m a y extend intermittently over a period of months or years, if not severe. 2. Neither of these cases showed diarrhea or tenesmus. Chesterman 4 remarks that diarrhea is unusual in these cases, and occurs only with severe, rapid hemorrhage. We believe that this is due to the fact that the bleeding occurs far enough above the ileocecal valve, so that the blood enters the colon slowly. Also the walls of the colon lose their tonicity, probably as a result of the increasing blood loss. This lack of tone probably increases as the loss of blood continues, so that our two cases had no spontaneous stools just before admission, in spite of the presence of considerable old blood in the colon and rectum. 3. The blood in the stools was mostly old but some fresh blood cells were found microscopically. This localizes the bleeding point above the lower bowel but not as high as the duodenum. 4. In our cases there was no evidence of any appreciable peritoneal irritation, as indicated by the absence of pain, tenderness, or rigidity. This was helpful in ruling out duodenal ulcer and appendicitis. Cobb ~ in reporting a series of 100 cases of peptic ulcer in Meckel's diverticulum, found that pain was a symptom in 80 per cent. Usually this was vague abdominal pain, often of a colicky nature r e f e r r e d to the umbilicus, and only rarely simulating the discomfort of duodenal ulcer. It is interesting to note that 55 of these cases eventually perforated. 5. These children show clinical and laboratory evidence of a much more rapid and severe loss of blood than is indicated by the amount passed in the stool. This is helpful in ruling out bleeding from lesions of the lower bowel such a papillomata, colonic ulcers, or hemorrhoids. 6. These. children did not vomit blood as might happen in gastric or duodenal ulcer hemorrhage. 7. They remained relatively well for a considerable period after the bleeding started. This resulted in a delay in medical attention. When a certain level of blood loss was reached, they suddenly became dangerously ill, and prompt action was needed. 8. The response to surgical removal was p r o m p t and absolute. The cases are interesting in that they illustrate two different pathologic types of bleeding Meckel's diverticulum. The first case showed a definite ulceration to explain the hemorrhage, whereas the pathologist was unable to demonstrate any source of the bleeding in the second case, although clotted blood was definitely present in the diverticulum at the
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time of operation. This latter condition has been reported frequently in the literature. Chesterman 4 suggests that the hemorrhage in these cases must be due to recurrent inflammation of a hemorrhagic nature, or possibly to acute microscopic ulceration. F a r r and Penke 6 give a more plausible explanation by suggesting that the hemorrhage is due to ulceration in the wall of the adjacent ileum. A confirmation of this opinion is f o u n d in Cobb's 5 analysis of 45 cases in which the location of the ulcer was described. H e found that in 11 per cent, the ulceration occurred outside of the diverticulum, in the wall of the adjacent ileum. It is also interesting to note that our first case showed cells in the mucosa near the margin of the ulcer, which resembled cells f o u n d normally in gastric mueosa. K a u f m a n n 7 states that islands of gastric mucosa, complete gastric mucosa, or even pancreatic tissues, m a y be found in the mucosa of Meckel's diverticula. Greenwald s reports t h a t 22 in a series of 23 cases of ulceration of Meckel's diverticulum, examined microscopically, showed the presence of gastric~'mucosa. It has been stated that it is probably impossible to demonstrate Meckel's diverticulum by roentgenogram. This was discussed in a conference at the Massachusetts General Hospital in March, 1936, 0 and also by Golden. 1~ Caffey 11 informs me that he believes the reason to be due to the relationship existing between the opening of the diverticulum, and the plane of the ileum. We believe that it is highly probable t h a t many cases of bleeding Meckel's diverticula are falsely diagnosed as duodenal ulcers, papillomata, internal hemorrhoids, and other conditions, when the blood loss is not severe or long continued. In the past two years 28 cases of Meckel's diverticulum with hemorrhage have been reported in the American literature b y several authors2, o, 12, 1~, 1~, ~5, ~6 This condition should be kept in mind whenever blood is found in the stool which cannot be explained by an obvious cause. REFERENCES 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13.
Callender, G . R . : Am. J. M. Sc. 150: 69, 1915. Taylor, A . L . : J. P a t h . & ]3act. 30: 415, 1927. Mason, J. ]Y[., ~nd Graham, G . S . : Ann. Surg. 96: 893, 1939.. Chesterman, J, T . : Brit. J. Surg. 23: 267, 1935. Cobb, D. ]3.: Ann. Surg. 103: 747, 1936. Farr, C. E., and Penke, M.: Ann. Surg. 101: ]026, 1935. K a u f m a n n ' s Pathology, Vol. 1, p. 726. Greenwald, tI. iVf., and Steiner, M.: Am. Y. Dis. Child. 42: 1176, 1931. New E n g l a n d J. Med. 214: 481, 1936. Golden, I~.: Diagnostic Roentgenology, ed. 1, Thomas Nelson & Son, p. 310. Caffey, J.: P e r s o n a l communication. Montgomery, A. II.: I n t e r n a t . Clin. 1: 216, 1935. Greenb]att, R. ]3., Pund, E. R., and Chancy, R. I-I.: Am. J. Surg. 31: 285, 1936. 14. New E n g l a n d J. Med. 213: 878, 1935. 15. Brown, P. W., a n d Pemberton, J. de Y.: Ann. Int. Med. 9: 1684, 1936. 16. Christopher, F., a n d Blessing, 1~.: Am. J. Surg. 31: 556, 1936. 264 KING STRE~
GUNDY,M.D.
N. Y.
1701 Ruysch first described the existence of an anatomic structure I N which later was described more fully by Meckel in 1812, and has since been called by his name. According to Arey, in the early embryo of 1.5 ram. to 2 mm., with the development of the fore-and hind-gut, the entodermal vesicle is divided into a dorsal intestine and a ventral yolk sac, with a narrowed portion between. With the growth of the embryo and the yolk sac, the connecting portion does not increase much in size, and eventually becomes a slender yolk stalk. This yolk stalk later becomes a slender thread extending from the junction of the fore- and hind-gut to the yolk sac, or umbilical vesicle. It loses its attachment to the gut in the 7 ram. embryo. A blind pocket may persist .at the point of its union with the intestine. This is known as Meckel's diverticulum. The blind end of this pouch may remain adherent to the umbilicus, or may separate and lie free in the abdominal cavity. The lumen of the pouch is continuous with that of the ileum, about 80 cm. above the ileocecal vane. We feel that hemorrhage from such diverticulum is of sufficient rari t y to warrant this report of the following two cases. CASE 1.--M. U., a male, 6 years old, of Polish extraction, was first seen Jan. 25, 1937. About one year previouw the child was taken to a physician because of a small amount of clark blood noticed in the stool. This was thought to originate in a rectal fissure, and did not recur. Uncomplicated chickenpox occurred three weeks before admission. Four days previous to admission the mother noticed some dark red blood in the stool The child showed no other symptoms, and had no increase in the number of stools. Two days before admission the blood recurred in the stool and was more plentiful. The child still showed no other untoward symptoms. The day preceding admission he had no stools and seemed well. On the day of admisMon he had a large stool in the morning which consisted almost entirely of dark blood. He showed no evidence of acute illness until noon, when he became pale and weak. I-Ie refused luncheon, vomited once during the afternoon, and became rapidly paler and weaker, although he had no further stools, tie had no pain at any t~me. The child was seen by the family physician who referred him to the hospital. Physiea~ Examination.--On admission at 5 p.~. on /January 25, the child appeared dangerously ill. The facies were drawn and anxious, and the skin, lips, and mucous membranes were pale and dry. The abdomen was flat, soft, not tender, and no masses or rigidity could be palpated. Rectal examination was negative except for the presence of considerable old blood. The temperature w~s 101 ~ F.; the puls% 130; and the respirations, 22. The remainder of the physical examination was entirely negative. Laboratory Findings.--Itemeglobin, 25 per cent (Sahli); red blood cells, 2,270,000; white blood cells, 23,000; polymorphonuclears, 82 per cent; lymphoeytes, 18 per cent; no abnormal white cells; red cells showed poikilocytosls and anisocytosis, 563
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b u t no n o r n m b l a s t s . P l a t e l e t s 184,000; n o r m a l clot r e t r a c t i o n ; c o a g u l a t i o n time, three a n d one-half m i n u t e s . B l e e d i n g time w a s n o t increased. Blood was t y p e 2 Morse. T h e u r i n e w a s n o r m a l except f o r h i g h specific g r a v i t y a n d 3 plus acetone. Clinical Course.--The child w a s given a blood t r a n s f u s i o n a t once, receiving 360 c.c. of whole blood b y direct method. A low e n e m a yielded n o t h i n g b u t clotted, a n d p a r t i a l l y d e s t r o y e d blood. The l a b o r a t o r y reported t h a t this stool showed red blood cells in all s t a g e s of d i s i n t e g r a t i o n , w i t h a f e w a p p a r e n t l y n o r m a l red cells. The stool was n e g a t i v e f o r ova a n d p a r a s i t e s . T h e n e x t m o r n i n g the color w a s improved, a n d t h e h e m o g l o b i n was 39 p e r cent ( S a h l i ) w i t h red blood cells n u m b e r i n g 2,850,000. T e m p e r a t u r e was 9 9 ~ pulse, 105; respiration, 18. A n e n e m a yielded a l a r g e a m o u n t of old blood. There w a s no s p o n t a n e o u s stool. I I e received 400 c.c. of whole blood b y direct t r a n s f u s i o n . T h a t e v e n i n g he h a d a large, d a r k red, liquid stool a n d a g a i n showed s i g n s of shock. I t w a s decided n o t to postpone operation a n y longer, as he was suspected of h a v i n g f r e s h b l e e d i n g into t h e bowel f r o m a p e r s i s t e n t M e c k e l ' s diverticulum. A t i A.M. on J a n . 27, 1937, the a b d o m e n w a s opened. The p e r i t o n e a l c a v i t y cont a i n e d a s m a l l a m o u n t of clear fluid. T h e whole colon a p p e a r e d relaxed a n d atonie a n d was d i s t e n d e d w i t h d a r k blood, which showed t h r o u g h its wall. A b o u t 15 inches p r o x i m a l to the ileocecal valve, w a s f o u n d a l a r g e d i v e r t i c u l u m a b o u t 4 inches long, a n d of the s a m e d i a m e t e r as the a d j a c e n t ileum. T h e d i v e r t i c u l u m w a s clamped, ligated, a n d removed. I t s s t u m p was cauterized a n d inverted w i t h a p u r s e - s t r i n g
suture. T h e t e m p e r a t u r e dropped to n o r m a l t w e n t y - f o u r h o u r s a f t e r operation a n d t h e p o s t o p e r a t i v e course w a s u n e v e n t f u l . T h e h e m o g l o b i n rose to 64 per cent ( S a h l i ) w i t h a red blood c o u n t of 4,400,000 on t h e n i n t h day.' The child was d i s c h a r g e d in good condition on t h e eleventh day, a n d h a s r e m a i n e d well since. Pathologic Report by Dr. Margaret Loder.--Macroscopically t h e s p e c i m e n consisted of a d i v e r t i c u l u m m e a s u r i n g 50 to 60 ram. in l e n g t h . A p p r o x i m a t e l y 30 ram. f r o m the closed end, t h e m u e o s a showed a n u l c e r a t e d a r e a 8 to 10 ram. in d i a m e t e r , w i t h a n increase of fibrous t i s s u e in t h e wall, a n d some constriction. Definite bleeding p o i n t s a p p e a r e d in t h e floor of t h e ulcer. T h e m u c o s a a r o u n d t h e edges of t h e ulcer a p p e a r e d congested. Microscopically t h e sections showed a definite u l c e r a t i o n of t h e mucosa, w i t h a n increase of a d u l t fibrous tissue in the s u b m u c o s a a n d m u s c u l a r i s . Leucocytie infiltration w a s p r e s e n t at t h e base o f t h e ulcer, p e r i v a s c n l a r a n d m o d e r a t e diffuse infiltration in the s t r o m a of the s u b m u c o s a , a n d infiltration to a lesser e x t e n t in t h e m u s c u l a r i s . The m u c o s a a n d l i n i n g e p i t h e l i u m were typical of t h a t f o u n d in the a d j a c e n t ileum. A n a r e a in which the s u r f a c e g l a n d s were lined by a l a r g e clear cell, u s u a l l y f o u n d i n t h e peptic g l a n d s of t h e stomach, was f o u n d n e a r t h e edge of the ulcer i n t h e sections examined. Pathologic D~agnosis.--Bleeding ulcer in a p e r s i s t e n t MeckeI's diverticulum. CASE 2 . - - M a c M . , a male two y e a r s of age, of A m e r i c a n p a r e n t a g e .
Present Illness.--On J a n . 28, 1937, t h e child w a s given E x q a x because of constipation. O n J a n u a r y 29 he h a d f o u r bowel m o v e m e n t s , three of w h i c h c o n t a i n e d blood. T h e f a t h e r t h o u g h t ' t h a t the blood was ~ ' s o m e w h a t b r i g h t . " The next day t h e child h a d only one stool which contained blood. H i s a p p e t i t e r e m a i n e d f a i r l y n o r m a l a n d he h a d no s y m p t o m s t h a t a t t r a c t e d t h e p a r e n t s ' notice except f o r one emesis. H e h a d no stools on J a n u a r y 3], nor o~L F e b r u a r y 1. The child s u d d e n l y became v e r y weak, pale, a n d l a n g u i d t o w a r d e v e n i n g of F e b r u a r y :[. H e was adm i t t e d to t h e hospital a t 5 P.~L Physical Examination.--Temperature was 100 ~ F . ; pulse, 140; respiration, 30. Color w a s v e r y p a l e ; t h e child, v e r y quiet a n d a p a t h e t i c ; lips, skin, a n d m u c o u s m e m b r a n e s were v e r y pale a n d dry. B l o w i n g systolic m u r m u r w a s h e a r d at the
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cardiac apex. A b d o m e n was flat, sol% n o t tender, and showed no palpable m a s s or rigidity. Rectal e x a m i n a t i o n was n e g a t i v e except f o r t h e presence of old blood in l a r g e a m o u n t s . The r e m a i n d e r of the physicM e x a m i n a t i o n was irrelevant. Laboratory Findings.--ltemoglobin, 33 per cent ( S a h l i ) ; red blood cells, 2,300,000; white blood cells, 10~200; p o l y m o r p h o n u c l e a r s , 39 per c e n t ; lymphocytes~ 51 per cent; n o r m o b l a s t s , 10 per cent. Blood was type 4 Morse. The u r i n e showed a l b u m i n plus, a n d occasional h y M i n casts. Clinical Course.--One h u n d r e d f o r t y cubic c e n t i m e t e r s of whole blood w a s g i v e n by direct t r a n s f u s i o n on a d m i s s i o n , a n d 150 e.c. t h e next m o r n i n g . A d i a g n o s i s of h e m o r r h a g e f r o m a p e r s i s t e n t M e e k e l ' s d i v e r t i c u l u m was made, a n d the a b d o m e n w~s opened at 4 L~z. on F e b r u a r y 2. A smM1 diverticulum, a b o u t the size o f t h e distal p h a l a n x of t h e t h u m b , was f o u n d a b o u t 10 inches p r o x i m a l to t h e ileocecat valve. This was clamped, ligated~ removed , a n d t h e base cauterized a n d i n v e r t e d by a p u r s e - s t r i n g s u t u r e . The d i v e r t i c u l u m was opened i n t h e o p e r a t i n g r o o m a n d clotted Mood f o u n d in its lumen. The appendix~ which a p p e a r e d normM, w a s removed in t h e u s u a l m a n n e r . T h e i m m e d i a t e p o s t o p e r a t i v e course was u n e v e n t f u l , a n d the h e m o g l o b i n rose to 45 per cent (Sahli) i~l t w e n t y - f o u r hours. O n t h e t h i r d p o s t o p e r a t i v e d a y he seemed restless, r e f u s e d food, v o m i t e d once, a n d the t e m p e r a t u r e rose to 102.6 ~. E x a m i n a t i o n showed a n acute n a s o p h a r y n g i t i s . On the f o u r t h d a y p o s t o p e r a t i v e he developed loose g r e e n stools w i t h m u c u s b u t no blood. T h i s condition p e r s i s t e d for f o u r d a y s with t e m p e r a t u r e f l u c t u a t i n g b e t w e e n 100 a n d 102 ~ a n d g r a d u a l l y f a l l i n g to n o r m a l on t h e e i g h t h p o s t o p e r a t i v e day. H e was d i s c h a r g e d on F e b r u a r y 16, f o u r t e e n d a y s following operation, a p p a r e n t l y in good condition. T h r o u g h some oversight, t h e blood count was n o t r e p e a t e d before discharge. Pathologic l~eport by Dr. Loder.--Macroseopieally the speeiinens s u b m i t t e d consisted o f : ( a ) A short, narrow, d i v e r t i c u l u m m e a s u r i n g 20 ram. in length. T h e specimen h a d been sectioned lengthwise, ~nd t h e c o n t e n t s lost. The m u e o s a was smooth t h r o u g h o u t . T h e wall ~ p p e a r e d to be m o d e r a t e l y thickened. There w a s no evidence of acute i n f l a m m a t i o n . ( b ) A n a p p e n d i x s h o w i n g some h y p e r p l a s i a of the l y m p h o i d tissue~ a n d a n otherwise g r o s s l y n o r m a l wall. D a r k feces was f o u n d in the h m e n . Microscopically t h e sections f r o m the d i v e r t i c u l u m showed a mucosa, lined by epithelium similar to t h a t of t h e a d j a c e n t ileum. The s u r f a c e e p i t h e l i u m of t h e sections e x a m i n e d a p p e a r e d intact. There was art increase of fibrous tissue i n t h e submucosa, m u s e u l a r i s , a n d subser0sa. P e r i v a s e u l a r a n d m o d e r a t e l y diffuse infiltration, c o n s i s t i n g of small r o u n d cells, p l a s m a cells, a n d a few p o l y n u e l e a r leucocytes, w a s f o u n d in the s t r o m a of t h e mucosa, s u b m u c o s a , m u s c u l a r i s , and subserosa. Sections f r o m t h e a p p e n d i x showed p e r i v a s e u l a r infiltration in t h e outer m u s c u l a r , a n d s u b s e r o u s layers. The l y m p h o i d tissue was hyperplastic. Diagnosis. Chronic M e c k e l ' s diverticulitis. Mild chronic appendicitis. COMMENT
We feel that these cases should be reported because they are typical of one of the true pediatric emergencies. W i t h o u t adequate t r e a t m e n t these children m i g h t well be lost, as has been reported by Callender ~ and Taylor. 2 The first ease reported was certainly in a very critical condition when admitted, and a guarded prognosis was given. I t was considered poor surgical j u d g m e n t to operate on this child immediately u p o n admission, due to the existing condition of shock. A p p a r e n t l y this child lost his blood very rapidly, as is indicated by the Complete absence of any signs of blood regeneration in the blood smear.
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The second case reported was in better physical condition upon admission. H e had apparently lost less blood, and had probably lost it more slowly as indicated by the normoblasts seen in the blood smear. There are certain similarities in these two cases which may be considered as typical of cases of severe hemorrhage from a persistent Meckel's diverticulum. These might be enumerated as follows: 1. The onset is often insidious, and usually without subjective symptoms. Mason and Graham 3 noted that the bleeding m a y extend intermittently over a period of months or years, if not severe. 2. Neither of these cases showed diarrhea or tenesmus. Chesterman 4 remarks that diarrhea is unusual in these cases, and occurs only with severe, rapid hemorrhage. We believe that this is due to the fact that the bleeding occurs far enough above the ileocecal valve, so that the blood enters the colon slowly. Also the walls of the colon lose their tonicity, probably as a result of the increasing blood loss. This lack of tone probably increases as the loss of blood continues, so that our two cases had no spontaneous stools just before admission, in spite of the presence of considerable old blood in the colon and rectum. 3. The blood in the stools was mostly old but some fresh blood cells were found microscopically. This localizes the bleeding point above the lower bowel but not as high as the duodenum. 4. In our cases there was no evidence of any appreciable peritoneal irritation, as indicated by the absence of pain, tenderness, or rigidity. This was helpful in ruling out duodenal ulcer and appendicitis. Cobb ~ in reporting a series of 100 cases of peptic ulcer in Meckel's diverticulum, found that pain was a symptom in 80 per cent. Usually this was vague abdominal pain, often of a colicky nature r e f e r r e d to the umbilicus, and only rarely simulating the discomfort of duodenal ulcer. It is interesting to note that 55 of these cases eventually perforated. 5. These children show clinical and laboratory evidence of a much more rapid and severe loss of blood than is indicated by the amount passed in the stool. This is helpful in ruling out bleeding from lesions of the lower bowel such a papillomata, colonic ulcers, or hemorrhoids. 6. These. children did not vomit blood as might happen in gastric or duodenal ulcer hemorrhage. 7. They remained relatively well for a considerable period after the bleeding started. This resulted in a delay in medical attention. When a certain level of blood loss was reached, they suddenly became dangerously ill, and prompt action was needed. 8. The response to surgical removal was p r o m p t and absolute. The cases are interesting in that they illustrate two different pathologic types of bleeding Meckel's diverticulum. The first case showed a definite ulceration to explain the hemorrhage, whereas the pathologist was unable to demonstrate any source of the bleeding in the second case, although clotted blood was definitely present in the diverticulum at the
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time of operation. This latter condition has been reported frequently in the literature. Chesterman 4 suggests that the hemorrhage in these cases must be due to recurrent inflammation of a hemorrhagic nature, or possibly to acute microscopic ulceration. F a r r and Penke 6 give a more plausible explanation by suggesting that the hemorrhage is due to ulceration in the wall of the adjacent ileum. A confirmation of this opinion is f o u n d in Cobb's 5 analysis of 45 cases in which the location of the ulcer was described. H e found that in 11 per cent, the ulceration occurred outside of the diverticulum, in the wall of the adjacent ileum. It is also interesting to note that our first case showed cells in the mucosa near the margin of the ulcer, which resembled cells f o u n d normally in gastric mueosa. K a u f m a n n 7 states that islands of gastric mucosa, complete gastric mucosa, or even pancreatic tissues, m a y be found in the mucosa of Meckel's diverticula. Greenwald s reports t h a t 22 in a series of 23 cases of ulceration of Meckel's diverticulum, examined microscopically, showed the presence of gastric~'mucosa. It has been stated that it is probably impossible to demonstrate Meckel's diverticulum by roentgenogram. This was discussed in a conference at the Massachusetts General Hospital in March, 1936, 0 and also by Golden. 1~ Caffey 11 informs me that he believes the reason to be due to the relationship existing between the opening of the diverticulum, and the plane of the ileum. We believe that it is highly probable t h a t many cases of bleeding Meckel's diverticula are falsely diagnosed as duodenal ulcers, papillomata, internal hemorrhoids, and other conditions, when the blood loss is not severe or long continued. In the past two years 28 cases of Meckel's diverticulum with hemorrhage have been reported in the American literature b y several authors2, o, 12, 1~, 1~, ~5, ~6 This condition should be kept in mind whenever blood is found in the stool which cannot be explained by an obvious cause. REFERENCES 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13.
Callender, G . R . : Am. J. M. Sc. 150: 69, 1915. Taylor, A . L . : J. P a t h . & ]3act. 30: 415, 1927. Mason, J. ]Y[., ~nd Graham, G . S . : Ann. Surg. 96: 893, 1939.. Chesterman, J, T . : Brit. J. Surg. 23: 267, 1935. Cobb, D. ]3.: Ann. Surg. 103: 747, 1936. Farr, C. E., and Penke, M.: Ann. Surg. 101: ]026, 1935. K a u f m a n n ' s Pathology, Vol. 1, p. 726. Greenwald, tI. iVf., and Steiner, M.: Am. Y. Dis. Child. 42: 1176, 1931. New E n g l a n d J. Med. 214: 481, 1936. Golden, I~.: Diagnostic Roentgenology, ed. 1, Thomas Nelson & Son, p. 310. Caffey, J.: P e r s o n a l communication. Montgomery, A. II.: I n t e r n a t . Clin. 1: 216, 1935. Greenb]att, R. ]3., Pund, E. R., and Chancy, R. I-I.: Am. J. Surg. 31: 285, 1936. 14. New E n g l a n d J. Med. 213: 878, 1935. 15. Brown, P. W., a n d Pemberton, J. de Y.: Ann. Int. Med. 9: 1684, 1936. 16. Christopher, F., a n d Blessing, 1~.: Am. J. Surg. 31: 556, 1936. 264 KING STRE~