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Shigella dysenteriae I: A forgotten cause of pseudomembranous colitis
Volume 89 Number 4
Brief clinical and laboratory observations
SI: Vasoactive intestinal peptide stimulation of adenylate cyclase and active electrolyte secretion in intestinal mucosa, J Clin Invest 54:536, 1974. 6. Said SI, and Faloona GR: Elevated plasma and tissue levels of vasoactive intestinal polypeptide in the watery-diarrhea syndrome due to pancreatic, bronchogenic and other tumors, N Engl J Med 293:155, 1975. 7. Swift PGF, Bloom SR, and Harris F: Watery diarrhea and ganglioneuroma with secretion of vasoactive intestinal
Shigella dysenteriae I: A forgotten cause of pseudomembranous colitis M. Kelber, M.D., and M. E. Ament, M.D.,* Los Angeles, Calif.
S H I G E L L A DYSENTERIAE type I is k n o w n to cause severe d i a r r h e a which tnay rapidly lead to d e h y d r a t i o n , shock, and death. The pathologic changes in the colon can show extensive mucosal ulceration a n d destruction with h e m o r r h a g e ? Older pathology textbooks described pseud o m e m b r a n o u s colitis in association with shigellosis.-' R e c e n t textbooks of gastroenterology :~-:' a n d gastrointestinal pathology G have excluded it. P s e u d o m e m b r a n o u s colitis has been principally described in association with certain antibiotics (ampicillin, tetracycline, clindamycin, c h l o r a m p h e n i c o l , cephalothin, a n d streptomycin) ..... a n d following prolonged complicated surgery. This report describes a case o f p s e u d o m e m b r a n o u s colitis secondary to infection with Shigella dysenteriae type 1 w h i c h progressed to toxic m e g a c o l o n requiring colectomy. C A S E REPORT Patient R.B., a 2 10/12-year-old Mexican-American boy, was admitted to the hospital with a 10-hour history of fever and profuse bloody mucoid diarrhea. The patient had never traveled out of the Los Angeles area and had not received any antibiotics or other medications. Physical examination disclosed weight, 16 kg; height, 97.5 cm; temperature, 39 ~ (rectal); pulse, 160/minute; blood pressure, 120/70. Bowel sounds were hyperactive, and the abdomen was
595
peptide, Arch Dis Child 50:896, 1975. 8. Rambaud J-C, Modigliani R, Matuchansky C, Bloom S, Said S, Pessayre D, and Bernier J-J: Pancreatic cholera. Studies on tumoral secretions and pathophysiology of diarrhea, Gastroenterology 69"110, 1975. 9. Rambaud JC, and Matuchansky C: Diarrhoeas and digestive endocrine tumours, Clin Gastroenterol 3:657, 1974.
diffusely tender, but the patient did not have abdominal guarding. Laboratory data revealed hemoglobin, 12.7 gm/dl; hematocrit, 36.1%; white blood count, 11,000/mm~; segmented neutrophils, 30%; band forms, 14%; metamyelocytes, 3%; lymphocytes, 4%; monocytes, 10%; eosinophils, 3%; serum sodium~ 136 mEq/1; potassium, 3.6 mEq/1; chloride, 100 mEq/l; bicarbonate, 19 mEq/1; glucose, 107 mg/dl; serum albumin, 4.0 gm/dl. Microscopic examination of the stool demonstrated many polymorphonuclear leukocytes and red blood cells. Multiple stool examinations for ova and parasites were negative. Initial impression was infectious diarrhea, but six stool cultures grew only mixed flora: nonenteropathogenic Escherichia coli and Staphylococcus aureus. Treatment was supportive and consisted of intravenous fluids. Diarrheal stool excreted weighed an average of 1,000 gm/day. A barium enema done on fifth hospital day was interpreted as consistent with ulcerative colitis. Rectosigmoidoscopy was done the next day to 8.0 cm level and showed typical yellow-white plaques characteristic of pseudomembranous colitis. Macroscopic changes were confirmed by rectal biopsy. Rectal crypts were destroyed and those remaining were depleted of mucus. Surface epithelium was denuded and mucosal surface was covered with fibrin and polymorphonuclear leukocytes. Stool losses increased to 1,600 gin/day on the ninth day. Hemoglobin and serum albumin decreased to 10.5 and 1.4 gm/dl, respectively. The paiient became toxic; the abdomen was distended and bowel signs were hypoactive. Abdominal flat plate was interpreted as showing toxic megacolon. Colectomy with retention of rectal stump and ileostomy was performed. Pathologic examination of the resected colon was interpreted as acute pseudomembranous and ulcerative colitis with terminal ileitis. Stool culture obtained on day of surgery grew S. dysenteriae type I. It was resistant to tetracycline, sulfathiazole, and chloramphenicol, but sensitive to gentamicin, cephalothin, and ampicillin. Two weeks postoperatively proctoscopy of rectal stump showed friable mncosa but pseudomembranes were absent. Culture obtained at procedure grew Shigella dysenteriae type I. Patient was treated wit h anrpicillin 500 mg orally, four times a day, for one week~ Rectal cultures became Shigella tree. DISCUSSION
From the Departments of Pediatrics and Family Medicine, UCLA CenterJbr Health. Sciences, Santa Monica Hospital Medical Centen *Reprint address: Departmenzof Pediatrics, MDCC-22-442, UCLA-CH& Los Angeles, Calif. 90024.
Shigella dysenteriae accounts for less t h a n 1% of all Shigella infections in the U n i t e d States. 1~ It causes the most severe diarrhea of all species a n d the greatest n u m b e r of extraintestinal complications. 1:'- 1'~
596
Brief clinical and laboratory observations
Infectious diarrhea was originallysuspected because of acute onset with fever and leukocytosis, the severity of the diarrhea, and the presence of sheets of polymorphonuclear leukocytes in stool. The difficulty in culturing S. dysenteriae from the stool was a major factor in our failure to establish a correct diagnosis. This is not u n c o m m o n since 20% of patients known to be infected with Shigella may not have microorganisms cultured from their stool. The barium enema interpreted as ulcerative colitis added to confusion in diagnosisY H a d proctosigmoidoscopy been done initially, diagnostic dilemma would have been avoided, and a course of antimicrobial therapy might have reversed the pathologic,process before colectomy became necessary.
Shigella dysenteriae type I. must be ~eturned to the list of causes of pseudomembranous colitis. REFERENCES
1. Formal SB, Gemski R, Gianella RA, et al: Mechanism at Shigella pathogenesis, Am J Clin Nutr 25:1427, 1972. 2. Saphir O: A text on systemic pathology, New York 1959, Grune & Stratton, Inc, pp 950-952; 962-963. 3. Sleisenger M, and Fordtran J: Gastrointestinal disease, Philadelphia, 1973, WB Saunders Company, pp 13731377. 4. Silverman A, Roy CC, and Cozzetto FJ: Pediatric clinical gastroenterology, St. Louis, 1971, The CV Mosby Company, pp 140-141. 5. Boekus HL, et al: Gastroenterology, in Dearing WH, editor: Infectious diarrheas, Philadelphia, 1976, WB Saunders Company, pp 956-957.
The "'locked-in'" syndrome in children Gerald S. Golden, M.D.,* Norman Leeds, M.D., Bronx, N. ii., Martin W. Kremenitzer, M.D., New York, N. Y., and Barry S. Russman, M.D.,
Hartford, Conn. From the Departments of Pediatrics and Neurology, Albert Einstein College of Medicine; Department of Radiology, Montefiore Hospital and Medical Center," Departments of Pediatrics and Neurology, New York Medical College," and Departments of Pediatrics (end Neurology, University of Connecticut Medical School. *Reprint address: 1410 Pelham ParkwaySouth, Bronx, N. Y. 10461.
The Journal of Pediatrics October 1976
6. Morson BC, and Dawson IMP: Gastrointestinal pathology, ed 2, Oxford, England, 1974, Blackwell Scientific Publications, p 259. 7. Reiner L, Schlesinger MJ, Miller GM: Pseudomembranous colitis following aureomycin and chloramphenicol, Arch Pathol 54:39, 1972. 8. Pettet JD, Baggenstoss AH, Dearins HH, et al: Postoperative pseudomembranous enterocolitis, Surg Gynecol Obstet 98:546, 1954. 9. Scott AJ, Nicholson GI, and Ken AR: Lincomycin as a cause of pseudomembranous colitis, Lancet 2:1232, 1973. 10. Cohen IE, McNeill CJ, and Wells RF: Clindamycin associated colitis, JAMA 223:1379, 1973. 11. Tedesco FJ, Barton RW, and Alpers DH: Clindamycin associated colitis, Ann Intern Med 81:429, 1974. 12. Goulston SH, and McGovern VJ: Pseudomembranous colitis, Gut 6:207, 1973. 13. Tedesco FJ, Stanley RJ, and Alpers DH: Diagnostic features of clindamycin associated pseudomembranous colitis, N Engl J Med 290:841, 1974. 14. Reller B, Gangarosa EI, and Brachman PS: Shigellosis in the United States-five-years review of nationwide surveillance, 1964-1968, Am J Epidemiol 91:161, 1970. 15. Levine VD, Overturf GD, and Mathies AW: Shigella dysenteriae type I--severe dysentery and sepsis with hematologic hepatic and renal complications, West J Med 121:501, 1974. 16. Willis KC, and Rosenblatt RM: Shiga bacillus dysentery complicated by bacteremia and disseminated intravascular coagulation, J PEmATR 83:90, 1973. 17. Shimkin PM, and Link RJ: Pseudomembranous colitis: A consideration in the barium enema differential diagnosis of acute generalized ulceration colitis, Br J Radiol 46:437, i973.
THE TERM " L O C K E D - I N " SYNDROME refers to the condition of total motor paralysis with preservation of mesencephatic-controlled vertical eye movements and an intact sensorium. This has been described as "alert wakefulness accompanied by m u t e tetraplegia. TM As evidence for intact cerebral cortical function, the patient can be taught to communicate by means of a set of coded eye blinks. The syndrome differs from that of the akinetic mute. In the latter condition, the patient can move, although he rarely does, and despite a superficial appearance of alert wakefulness, communication cannot be established. No previous reports o f this condition in children have been found. CASE REPORTS Case 1. This 13-year-old boy had cyanotic congenital heart disease consisting of tricuspid atresia, ventricular septal defect, and pulmonic stenosis. At age 10 years a Blalock-Taussig anastamosis had been performed.
Brief clinical and laboratory observations
SI: Vasoactive intestinal peptide stimulation of adenylate cyclase and active electrolyte secretion in intestinal mucosa, J Clin Invest 54:536, 1974. 6. Said SI, and Faloona GR: Elevated plasma and tissue levels of vasoactive intestinal polypeptide in the watery-diarrhea syndrome due to pancreatic, bronchogenic and other tumors, N Engl J Med 293:155, 1975. 7. Swift PGF, Bloom SR, and Harris F: Watery diarrhea and ganglioneuroma with secretion of vasoactive intestinal
Shigella dysenteriae I: A forgotten cause of pseudomembranous colitis M. Kelber, M.D., and M. E. Ament, M.D.,* Los Angeles, Calif.
S H I G E L L A DYSENTERIAE type I is k n o w n to cause severe d i a r r h e a which tnay rapidly lead to d e h y d r a t i o n , shock, and death. The pathologic changes in the colon can show extensive mucosal ulceration a n d destruction with h e m o r r h a g e ? Older pathology textbooks described pseud o m e m b r a n o u s colitis in association with shigellosis.-' R e c e n t textbooks of gastroenterology :~-:' a n d gastrointestinal pathology G have excluded it. P s e u d o m e m b r a n o u s colitis has been principally described in association with certain antibiotics (ampicillin, tetracycline, clindamycin, c h l o r a m p h e n i c o l , cephalothin, a n d streptomycin) ..... a n d following prolonged complicated surgery. This report describes a case o f p s e u d o m e m b r a n o u s colitis secondary to infection with Shigella dysenteriae type 1 w h i c h progressed to toxic m e g a c o l o n requiring colectomy. C A S E REPORT Patient R.B., a 2 10/12-year-old Mexican-American boy, was admitted to the hospital with a 10-hour history of fever and profuse bloody mucoid diarrhea. The patient had never traveled out of the Los Angeles area and had not received any antibiotics or other medications. Physical examination disclosed weight, 16 kg; height, 97.5 cm; temperature, 39 ~ (rectal); pulse, 160/minute; blood pressure, 120/70. Bowel sounds were hyperactive, and the abdomen was
595
peptide, Arch Dis Child 50:896, 1975. 8. Rambaud J-C, Modigliani R, Matuchansky C, Bloom S, Said S, Pessayre D, and Bernier J-J: Pancreatic cholera. Studies on tumoral secretions and pathophysiology of diarrhea, Gastroenterology 69"110, 1975. 9. Rambaud JC, and Matuchansky C: Diarrhoeas and digestive endocrine tumours, Clin Gastroenterol 3:657, 1974.
diffusely tender, but the patient did not have abdominal guarding. Laboratory data revealed hemoglobin, 12.7 gm/dl; hematocrit, 36.1%; white blood count, 11,000/mm~; segmented neutrophils, 30%; band forms, 14%; metamyelocytes, 3%; lymphocytes, 4%; monocytes, 10%; eosinophils, 3%; serum sodium~ 136 mEq/1; potassium, 3.6 mEq/1; chloride, 100 mEq/l; bicarbonate, 19 mEq/1; glucose, 107 mg/dl; serum albumin, 4.0 gm/dl. Microscopic examination of the stool demonstrated many polymorphonuclear leukocytes and red blood cells. Multiple stool examinations for ova and parasites were negative. Initial impression was infectious diarrhea, but six stool cultures grew only mixed flora: nonenteropathogenic Escherichia coli and Staphylococcus aureus. Treatment was supportive and consisted of intravenous fluids. Diarrheal stool excreted weighed an average of 1,000 gm/day. A barium enema done on fifth hospital day was interpreted as consistent with ulcerative colitis. Rectosigmoidoscopy was done the next day to 8.0 cm level and showed typical yellow-white plaques characteristic of pseudomembranous colitis. Macroscopic changes were confirmed by rectal biopsy. Rectal crypts were destroyed and those remaining were depleted of mucus. Surface epithelium was denuded and mucosal surface was covered with fibrin and polymorphonuclear leukocytes. Stool losses increased to 1,600 gin/day on the ninth day. Hemoglobin and serum albumin decreased to 10.5 and 1.4 gm/dl, respectively. The paiient became toxic; the abdomen was distended and bowel signs were hypoactive. Abdominal flat plate was interpreted as showing toxic megacolon. Colectomy with retention of rectal stump and ileostomy was performed. Pathologic examination of the resected colon was interpreted as acute pseudomembranous and ulcerative colitis with terminal ileitis. Stool culture obtained on day of surgery grew S. dysenteriae type I. It was resistant to tetracycline, sulfathiazole, and chloramphenicol, but sensitive to gentamicin, cephalothin, and ampicillin. Two weeks postoperatively proctoscopy of rectal stump showed friable mncosa but pseudomembranes were absent. Culture obtained at procedure grew Shigella dysenteriae type I. Patient was treated wit h anrpicillin 500 mg orally, four times a day, for one week~ Rectal cultures became Shigella tree. DISCUSSION
From the Departments of Pediatrics and Family Medicine, UCLA CenterJbr Health. Sciences, Santa Monica Hospital Medical Centen *Reprint address: Departmenzof Pediatrics, MDCC-22-442, UCLA-CH& Los Angeles, Calif. 90024.
Shigella dysenteriae accounts for less t h a n 1% of all Shigella infections in the U n i t e d States. 1~ It causes the most severe diarrhea of all species a n d the greatest n u m b e r of extraintestinal complications. 1:'- 1'~
596
Brief clinical and laboratory observations
Infectious diarrhea was originallysuspected because of acute onset with fever and leukocytosis, the severity of the diarrhea, and the presence of sheets of polymorphonuclear leukocytes in stool. The difficulty in culturing S. dysenteriae from the stool was a major factor in our failure to establish a correct diagnosis. This is not u n c o m m o n since 20% of patients known to be infected with Shigella may not have microorganisms cultured from their stool. The barium enema interpreted as ulcerative colitis added to confusion in diagnosisY H a d proctosigmoidoscopy been done initially, diagnostic dilemma would have been avoided, and a course of antimicrobial therapy might have reversed the pathologic,process before colectomy became necessary.
Shigella dysenteriae type I. must be ~eturned to the list of causes of pseudomembranous colitis. REFERENCES
1. Formal SB, Gemski R, Gianella RA, et al: Mechanism at Shigella pathogenesis, Am J Clin Nutr 25:1427, 1972. 2. Saphir O: A text on systemic pathology, New York 1959, Grune & Stratton, Inc, pp 950-952; 962-963. 3. Sleisenger M, and Fordtran J: Gastrointestinal disease, Philadelphia, 1973, WB Saunders Company, pp 13731377. 4. Silverman A, Roy CC, and Cozzetto FJ: Pediatric clinical gastroenterology, St. Louis, 1971, The CV Mosby Company, pp 140-141. 5. Boekus HL, et al: Gastroenterology, in Dearing WH, editor: Infectious diarrheas, Philadelphia, 1976, WB Saunders Company, pp 956-957.
The "'locked-in'" syndrome in children Gerald S. Golden, M.D.,* Norman Leeds, M.D., Bronx, N. ii., Martin W. Kremenitzer, M.D., New York, N. Y., and Barry S. Russman, M.D.,
Hartford, Conn. From the Departments of Pediatrics and Neurology, Albert Einstein College of Medicine; Department of Radiology, Montefiore Hospital and Medical Center," Departments of Pediatrics and Neurology, New York Medical College," and Departments of Pediatrics (end Neurology, University of Connecticut Medical School. *Reprint address: 1410 Pelham ParkwaySouth, Bronx, N. Y. 10461.
The Journal of Pediatrics October 1976
6. Morson BC, and Dawson IMP: Gastrointestinal pathology, ed 2, Oxford, England, 1974, Blackwell Scientific Publications, p 259. 7. Reiner L, Schlesinger MJ, Miller GM: Pseudomembranous colitis following aureomycin and chloramphenicol, Arch Pathol 54:39, 1972. 8. Pettet JD, Baggenstoss AH, Dearins HH, et al: Postoperative pseudomembranous enterocolitis, Surg Gynecol Obstet 98:546, 1954. 9. Scott AJ, Nicholson GI, and Ken AR: Lincomycin as a cause of pseudomembranous colitis, Lancet 2:1232, 1973. 10. Cohen IE, McNeill CJ, and Wells RF: Clindamycin associated colitis, JAMA 223:1379, 1973. 11. Tedesco FJ, Barton RW, and Alpers DH: Clindamycin associated colitis, Ann Intern Med 81:429, 1974. 12. Goulston SH, and McGovern VJ: Pseudomembranous colitis, Gut 6:207, 1973. 13. Tedesco FJ, Stanley RJ, and Alpers DH: Diagnostic features of clindamycin associated pseudomembranous colitis, N Engl J Med 290:841, 1974. 14. Reller B, Gangarosa EI, and Brachman PS: Shigellosis in the United States-five-years review of nationwide surveillance, 1964-1968, Am J Epidemiol 91:161, 1970. 15. Levine VD, Overturf GD, and Mathies AW: Shigella dysenteriae type I--severe dysentery and sepsis with hematologic hepatic and renal complications, West J Med 121:501, 1974. 16. Willis KC, and Rosenblatt RM: Shiga bacillus dysentery complicated by bacteremia and disseminated intravascular coagulation, J PEmATR 83:90, 1973. 17. Shimkin PM, and Link RJ: Pseudomembranous colitis: A consideration in the barium enema differential diagnosis of acute generalized ulceration colitis, Br J Radiol 46:437, i973.
THE TERM " L O C K E D - I N " SYNDROME refers to the condition of total motor paralysis with preservation of mesencephatic-controlled vertical eye movements and an intact sensorium. This has been described as "alert wakefulness accompanied by m u t e tetraplegia. TM As evidence for intact cerebral cortical function, the patient can be taught to communicate by means of a set of coded eye blinks. The syndrome differs from that of the akinetic mute. In the latter condition, the patient can move, although he rarely does, and despite a superficial appearance of alert wakefulness, communication cannot be established. No previous reports o f this condition in children have been found. CASE REPORTS Case 1. This 13-year-old boy had cyanotic congenital heart disease consisting of tricuspid atresia, ventricular septal defect, and pulmonic stenosis. At age 10 years a Blalock-Taussig anastamosis had been performed.