Volume 78 Number 2
Brief clinical and laboratory observations
with acute lymphoblastic leukemia than with acute myelogenous leukemia# Cytosine arabinoside (Cytosar, The Upjohn Co., Kalamazoo, Mich.), a pyrimidine nucleoside, has been reported to have in vitro activity against the herpes varicella virus, 4 as well as to be beneficial in other disseminated viral infections2 The use of cytosine arabinoside in a leukemic patient with varicella has been reported by Seligman and Rosner# They observed a child with acute myelogenous leukemia who developed varicella while receiving weekly subcutaneous injections of cytosine arabinoside; they regarded the occurrence of varicella in a leukemic patient as "fortuitous and unrelated to the leukemia, for cytosine arabinoside may not have in vivo activity against the varicella virus in man, even though in vitro activity has been demonstrated, ''~ Alternatively, Juel-Jensen 6 suggested that the dose of the drug employed by Seligman and Rosner 4 to maintain a leukemic remision may not have been adquate to prevent or treat varicella. It would appear
Intramural bematoma of the duodenum: An unusual complication of small bowel biopsy
that the role of cytosine arabinoside in the treatment of varicella has not been established and that reports such as this one may be helpful in assessing its adequacy. Though cytosine arabinoside is commercially available, it must be emphasized that its use as an antiviral agent is on an investigational basis only.
REFERENCES 1. Merselie, J. G., Kaye, D., and Hook, E. W.: Disseminated herpes zoster, Arch. Intern. Med. 113: 679, 1961. 2. Shanbrom, E., Miller, S., and Haar, H.: Herpes zoster in hematologic neoplasms. Some unusual manifestations, Ann. Intern. Med. 53: 523, 1960. 3. McKelvey, E., and ttan, H. C.: Cytosine arabinoside therapy for disseminated herpes zoster in a patient with IgG pyroglobulinemia, Blood 34: 706, 1969. 4. Seligman, B. R, and Rosner, F.: Varicella and cytosine arabinoside, Lancet l: 307, 1970. 5. Howard, J. P., Cerik, H., and Murphy, M. L.: Cytosine arabinoside in acute leukemia in children, Cancer Chemother. Rep. 50: 287, 1966. 6. Juel-Jensen, B. E.: Varicella and cytosine arabinoside, Lancet 1: 572, 1970.
variety of instruments has been developed for obtaining a biopsy. 1-~ Although complications such as perforation or hemorrhage have been recorded for both adults and children, we could find only one reported case of duodenal hematoma following small bowel biopsy12; for that reason we record the following case. CASE
M. Mullinger, M.D., B. J. Wood, M.D., M. R. Kliman, M.D., and G. C. Robinson, M.D. VANCOUVER~
I N
R E C E N T
B.
C.~ C A N A D A
Y E AR S ,
peroraI
intestinal
biopsy has become an accepted method for investigation of malabsorption syndromes; a From the Department of Paediatrics, University of British Columbia.
323
REPORT
A 10-year-old Chinese boy~ was admitted July 3 at 8:00 A.~., after an overnight fast, for a small bowel biopsy as part of the investigation of a possible malabsorption syndrome. At 9:00 A.~., he was given 50 mg. of meperidine HC1 and 50 mg. of promethazine HC1, intramuscularly. A multipurpose suction biopsy tube (W. E. Quinton Instrument Co., Seattle, Wash.) was passed at 10:00 A.z~., and a biopsy was obtained without untoward incident at 12:00 noon. Two small pieces of transverse duodenum were obtained; one for microscopic examination and one *The detailed investigation of this boy is to be reported separately.
324
Brie[ clinical and laboratory observations
for analysis of disaccharidase content. The microscopic sections revealed normal papillary fronds lined with intact unremarkable epithelium. The underlying glands were normal, and there was no significant increase in inflammatory cells. No serosa was seen. Hemoglobin was 12.4 Gin. per cent, the white blood count was 7,900 per cubic millimeter with a normal differential. The patient was discharged home on July 4. At home on the evening of July 4, he felt tired and nauseated. At 5:00 P.M., he vomited a small amount of clear fluid. He began to have intermittent nonradiating moderately severe pain in the right upper quadrant, lasting a few minutes at a time. He had a few potato chips and a carbonated beverage for dinner. Subsequently, he vomited small amounts 2 or 3 times. Normal urine was passed without any pain. He did not pass any stools for 24 hours. On readmission to hospital July 5, he looked sick but was not in acute distress. His pulse was 90 per minute and regular. Blood pressure was 90/60, respirations were 20 per minute, and temperature was 36.7 ~ C. There was a small area of mild-to-moderate tenderness in the right upper quadrant with well-localized muscle guarding over this area, but no rebound tenderness, no release discomfort, and no abnormality to percussion or auscultation. No other abnormalities were noted on physical examination. A roentgenogram of the abdomen failed to reveal free air. Hemoglobin was 13 Gm. per cent, white blood cell count was 8,400 per cubic millimeter, the serum electrolytes were within normal limits, and the serum amylase was 730 Caraway units (normal range = 80 to 100 Caraway units). The tentative diagnosis was posterior penetrating ulcer-equivalent, with localized pancreatitis. He was treated conservatively with continuous nasogastric suction, intravenous fluids, antibiotics, probantheline bromide, phenobarbital, and bed rest. Two days later the hemoglobin had fallen to 9.8 Gin. per cent; the white blood cell count was 7,300 per cubic millimeter with a normal differential. All stools examined were negative for blood. The total serum bilirubin was less than i mg. per cent, serum glutamie oxaloacetic transaminase was 33 units, lactate dehydrogenase was 70 units, serum glutamic pyruvate transaminase was 18 units, and the serum amylase had fallen to 250 Caraway units. Since blood factors involved in clotting and bleeding were not abnormal in April of the same year, these were not re-examined at this time. The right upper quadrant pain gradually subsided. Intravenous
I'he Journal o[ Pediatrics February 1971
fluid therapy was discontinued after 4 days, and the patient was started on an ulcer regimen. On the seventh hospital day a roentgenographic study of the gastrointestinal tract revealed abnormal findings limited to the second and third portions of the duodenum. In the second portion which was mildly dilated, barium was noted to move up and down, and there was delay in the passage of barium beyond this area. An intrinsic mass 6.5 cm. in transverse dimension and 4 cm. in superior-inferior dimension was outlined, and the lumen of the third portion of the duodenum was narrowed to 2 or 3 ram. by it (Fig. 1 ). It was thought that this mass most likely represented an intramural hematoma of the duodenum because of the drop in the hemoglobin concentration and that the transient rise in serum amylase might have been due to partial obstruction of the pancreatic duct by the duodenal hematoma. 12 A second radiographic study of the stomach and duodenum on the fourteenth hospital day revealed a diminution in the size of the duodenal lesion. The patient was discharged home much improved. DISCUSSION
S h w a c h m a n a n d associates 6 recently reviewed their experience with 800 biopsies in children; the C r o s b y - K u g l e r capsule was used. T h e y biopsied the t h i r d p a r t of the d u o d e n u m because they believed t h a t the risk of perforation was m i n i m a l there a n d t h a t the information o b t a i n e d was adequate. T h e y h a d only 2 h e m o r r h a g e s serious enough to require transfusion. I n their review of the literature, they f o u n d only a few r e p o r t e d complications, including only one perforation in a severely debilitated baby. P a r t i n and Schubert 7 r e p o r t e d 6 intestinal perforations in 83 biopsies in children, for each of which the porthole of the capsule was 3 mm. in diameter. N o difficulties were encountered when a capsule with a p o r t h o l e of 2 mm. was used. I n I73 ~ u n a I biopsies in children, M c N i c h o l l a n d Egan, 8 using the CrosbyK u g l e r capsule, h a d only one serious complic a t i o n - - a perforation. Sheehy a n d Floch '~ r e p o r t e d 2 instances of significant h e m o r rhage a n d one intestinal p e r f o r a t i o n in 2,374 intestinal biopsies with the Crosby capsule, largely in adults. T o b i n a n d associates ~0
Volume 78 Number 2
Brief clinical and laboratory observations
325
Fig. 1. Large radiolucent filling defect in duodenum at junction of second and third portions causing incomplete obstruction.
reported a duodenal h e m a t o m a in a 21-yearold Caucasian male following biopsy with the Ross biopsy instrument which consists of a 4 lumen tube, a biopsy capsule, and a balloon which m a y be cuffed about the tube. T h e lumen of the d u o d e n u m m a y be occluded by an enlarging hematoma. T h e mass can usually be demonstrated roentgenographically. I n addition, the involved segment usually narrows gradually, and the edematous mucosa creates a "picket fence" outline of the contrast material. A '~coiled-spring" appearance arising from the crowding together of the thickened mueosal folds of the distal end of the invplved area is considered pathognomonlc. 11 Most successflfl reports of nonoperative m a n a g e m e n t have been of children in w h o m there was only partial obstruction of the duodenum. Nasogastric decompression shouM be followed by cautious dietary measures. Since the majority of duodenal hematomas occur as isolated lesions and do not compromise the integrity of the duodenal wall, a nonoperative approach m a y be effective in some instances? ~
SUMMARY
T h e occurrence of an intramural hematoma in the transverse portion of the duod e n u m in a 10-year-old Chinese boy following a peroral small bowel biopsy is reported. REFERENCES
1. Royer, M., Oroxatto, O., Biempica, I., and Batcazar, M.: Biopsia duodenal pot aspiraction bafo control radioscopir Prensa Med. Argent. 42: 2515, 1955. 2. Shiner, M.: Duodenal biopsy, Lancet I: 17, 1956, 3. Crosby, W. I-I., and Kugler, I-I. W.: Intraluminal biopsy of the small intestine. The intestinal biopsy capsule, Amer. J. Digest. Dis. 2: 236, 1957. 4. Ross, J. R., and Moore, V. A.: Small intestinal biopsy capsule utifizing hydrostatic and suction principles, Gastroenterology 40: 113, 1961. 5. Brandborg, L. L., Rubin, C. E., and Quinton, W. E.: A multipurpose instrument for suction biopsy of esophagus, stomach, small bowel and colon, Gastroenterology 37: 1, 1959. 6. Shwachman, H., Khaw, T.-T., and Antonowicz, I.: Diagnosis and treatment: Peroral intestinal biopsy, Pediatrics 43: 460, 1969. 7. Partln, J. O., and Schubert, W. K.: Precautionary note on the use of the intestinal biopsy capsule in infants and emaciated children, New Eng. J. Med. 274: 44, 1966.
326
Brie[ clinical and laboratory observations
8. McNicholl, B., and Egan, B.: Jejunal biopsy in celiac disease, Clin. Pediat. 7: 544, 1968. 9. Sheehy, T. W., and Floch, M. H.: The small intestine: Its function and diseases, New York, 1964, Paul B. Hoebm5 Inc., Medical Book Division of Harper & Row, PubIishers, p. 37. 10. Tobin, J. J., Schlang, H. A., and James, D. R.: Intramural hematoma of the duodenum associated with peroral small bowel biopsy, J. A. M. A. 198: 211, 1966.
The coagulation defect in Reye's syndrome Allen D. Schwartz, M . D . NEW
HAVEN,
GONN.
S I N c E Reye a n d associates '~ initial report, over 100 cases of the syndrome of acute e n c e p h a l o p a t h y a n d fatty degeneration of the viscera have a p p e a r e d in the literature. Gastrointestinal h e m o r r h a g e has often been n o t e d d u r i n g the course of the illness, a n d at p o s t m o r t e m examination of m a n y patients, large amounts of blood have been f o u n d in the gastrointestinal t r a c t ? "5 T h e r e is a paucity of d a t a to explain the h e m o r r h a g i c diathesis. C o a g u l a t i o n studies were p e r f o r m e d in a child with Reye's syndrome in an a t t e m p t *o better characterize the pathogenesis of the bleeding. These studies are the subject of this report. CASE REPORT
A 6-year-old Caucasian gM was considered well until 5 days prior to admission when she complained of a sore throat and a mild cough. She seemed to be recovering until 2 days prior From the Department of Pediatrics, Yale University School of Medicine, and The Yale-New Haven Hospital. Supported in part by United States Public Health Service Grant 5 TO1 HD00177-03. Reprint address: Department of Ped~atrles. Yale University School of Medlc~ne, 833 Cedar St., New Haven, Conn. 06510.
The Journal o[ Pediatrics February 1971
11. Nelson, B., and Levin, E. J.: IntramuraI hematoma of the duodenum: A diagnostic roentgen sign, Radiology 63: 823, 1954. 12. Babbitt, D. P., Hussain, M. I., Gavino, P., Sakaguchi, S., and Pequet, A. R.: Intramural duodenal hematoma in children, Amer. J. Dis. Child. 115: 37, 1968. 13. Freeark, R. J.: Intramural hematoma of the duodenum, Arch. Surg. 92: 463, 1966.
to admission when she began to vomit repeatedly. The following day she did not recognize her parents and had progressive deterioration in the level of consciousness with sporadic episodes of excitement and screaming. She was admitted to another hospital where examination of the cerebrospinal fluid showed no celIs and a protein concentration of 18 rag. per 100 ml. Blood glucose concentration was 118 rag. per 100 ml. She was transferred to the Yale-New Haven Hospital. At the time of admission she was a well-developed, well-nourished child who was unresponsive to verbal stimuli and thrashed about in bed. The pupils were widely dilated but responded to Iight. The liver was palpable 2 cm. below the right costal margin. The spleen was not felt. Deep tendon reflexes were brisk and symmetrical. Plantar responses were extensor. There was no jaundice. Laboratory data and hospital course. Hemoglobin concentration was t3.5 Gin. per 100 ml. White blood cell count was 15,900 per cubic millimeter with a normal differential. The red cell morphology was unremarkable. Platelet count was 540,000 per cubic millimeter. Lumbar puncture revealed clear cerebrospinal fluid with i3I red cells and 2 Iymphocytes. Spinal fluid protein concentration was 10 mg. per 100 ml. Blood glucose level was 95 mg. per 100 ml. Laboratory, studies outlined in Table I indicated marked hepatic dysfunction. Coagulation studies were performed during the second hospital day; the results are summarized in Table II. Stool was negative for occult blood. Studies for Australian antigen and antibody were negative in the child and her mother. The father's serum contained antibody to Australian antigen. A myxovirus was grown from the patient's throat. The child was treated for hepatic failure with cIeansing enemas, neomycin sulfate, orally and