Child’s Nerv Syst (2000) 16:184–189 © Springer-Verlag 2000
Chul-Kee Park Kyu-Chang Wang Jeong Kee Seo Byung-Kyu Cho
Received: 15 May 1999 C.-K. Park · K.-C. Wang · B.-K. Cho Division of Pediatric Neurosurgery, Seoul National University Children’s Hospital and Neuroscience Research Institute, SNUMRC, 28 Yongon-dong, Chongno-gu, Seoul 110-744, Korea J.K. Seo Department of Pediatrics, Seoul National University Children’s Hospital, 28 Yongon-dong, Chongno-gu, Seoul 110-744, Korea B.-K. Cho (✉) Division of Pediatric Neurosurgery, Seoul National University Children’s Hospital, 28 Yongon-dong, Chongno-gu, Seoul 110-744, Korea e-mail:
[email protected] Tel.: +82-2-7602350 Fax: +82-2-7473648
C A S E R E P O RT
Transoral protrusion of a peritoneal catheter: a case report and literature review
Abstract Transoral protrusion of a peritoneal catheter is rare. Only two cases have been reported in the English literature. We now report the case of a 5-year-old girl who presented with a catheter that had been inserted 4 years previously, protruding from her mouth. Signs of cerebrospinal fluid infection or peritonitis were absent. The peritoneal catheter was cut and externalized at the chest. The distal portion, which had perforated the stomach wall, was removed using endoscopic procedures. After 3 weeks of antibiotic treatment, a new shunt was inserted. Analysis of 50 cases of bowel perforation extracted from the English literature showed that among the suggested factors such as age, gender, nutritional state, history of abdomi-
Introduction Although ventriculoperitoneal (VP) diversion of the cerebrospinal fluid (CSF) using artificial shunt devices is an accepted, innovative method for the management of hydrocephalus, high rates of various complications have been reported, ranging from 24% to 47% [7, 13, 14], and abdominal complications account for approximately 25% of them [3]. The incidence of bowel perforation by a shunt catheter is known to be as low as 0.1–0.7% [15, 20]. We describe a case of transoral protrusion of a peritoneal shunt catheter that had spontaneously perforated the stomach wall. To our knowledge this is the third such case to be reported in the English literature [8, 12]. We found 49 documented cases of bowel perforation by shunt cathe-
nal surgery, and length and type of the catheter, age was the only predisposing factor. In the treatment of bowel perforation by a peritoneal catheter, suspected shunt infection should be managed properly and contamination be minimized during removal of the peritoneal catheter. Key words Cerebrospinal fluid shunt · Bowel perforation · Stomach · Peritoneal catheter · Transoral protrusion · Endoscopy
ters in the English literature and reviewed the clinical characteristics, including predisposing factors, pathogenesis, and prognostic factors.
Case report On 6 September 1997, a 5-year-old girl was admitted to Seoul National University Children’s Hospital with a distal shunt catheter protruding from her mouth. She had been delivered by cesarean section at 32 weeks of gestational age and with a birthweight of 1.75 kg following premature rupture of the amniotic membrane. At the age of 6 months, she underwent endoscopic III ventriculostomy for presumed aqueductal stenosis, which was caused by germinal matrix hemorrhage. At the age of 1 year, her head continued to enlarge and a VP shunt (low-pressure silicone distal catheter with a slit: AccuFlo, Codman, Goleta, USA) was inserted. Follow-up imaging revealed collapsed ventricles with asymptomatic spontaneous bi-
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frontal subdural fluid collection, which regressed gradually. Her development was normal. One month before admission, the patient had developed abdominal pain and redness along the shunt catheter tract on her chest. Physical examination and laboratory tests including CSF studies performed at outpatient clinic were normal. However, she experienced several episodes of abdominal pain which spontaneously improved. On the day of admission, her vomiting was accompanied by regurgitation of 15 cm of the distal shunt catheter through her mouth. Her mother held it on the way to our hospital, only to lose hold of it as soon as she arrived. The patient was alert and complained only of headache. Neck stiffness, fever, abdominal pain and tenderness were absent. Redness on the right anterior chest wall and along the subcutaneous tract of the shunt catheter was noted. Laboratory findings of peripheral blood were as follows: white blood cells (WBCs), 12,490/mm3; hemoglobin, 12.2 g/dl; erythrocyte sedimentation rate (ESR), 3 mm/h; C-reactive protein (CRP), 9.1 mg/dl. CSF findings from reservoir tap were: red blood cells (RBCs), 0/mm3; WBCs, 0/mm3; protein, 11 mg/dl; glucose, 56 mg/dl. CSF and blood culture studies showed no growth of organisms. Upward migration of a shunt catheter through the esophagus was documented in the plain chest
and abdomen radiographs (Fig. 1). The peritoneal catheter was cut just proximal to the area of redness on the anterior chest and was externalized for CSF drainage. There was subcutaneous pus along the reddened area, which was irrigated. Cefotaxime and amikacin were started prophylactically for CSF infection. Cefotaxime and amikacin were chosen for their broad-spectrum cover of skinborne Gram-positive organisms as well as Gram-negative bowel organisms, respectively. Coagulase-negative staphylococcus was cultured from the pus. Two days later, the rest of the peritoneal catheter was easily removed using upper gastrointestinal endoscopy, and the perforation site in the upper body of the stomach was confirmed (Fig. 2). Clinical and sonographic signs of peritoneal spillage were absent. The patient was restrained from oral intake for 2 weeks, and nutrients were supplied parenterally. No evidence of infection was observed on clinical examinations during that period. Three weeks after endoscopic catheter removal (delayed for an additional week because of drug-induced fever and leukocytopenia), the shunt was revised. The patient remained well up to a follow-up examination 1 year later.
Discussion
Fig. 1 A plain chest radiograph taken on arrival at our hospital. The peritoneal catheter was looped along the stomach curvature, and the tip of the catheter was located at the esophagus Fig. 2 A gastrofiberscopic view. A shunt catheter which perforated the upper body of the stomach wall is seen (left). The catheter was grasped by means of an endoscopic device (right) and removed
Wilson and Bertrand [29] were the first authors to report a case of bowel perforation by a distal shunt catheter. We have found 50 such cases in the English literature, including ours (Table 1). Of the patients concerned, 26 were male and 22, female; 2 were of unknown gender. The mean age at perforation was 7.7 years, ranging from 1 month to 69 years. Thirty-five patients were younger than 5 years. The entity affects predominantly young patients. A couple of plausible explanations can be supposed: (1) children have weak bowel musculature, which is prone to perforation by a rather stiff catheter tip, (2) young patients have more vigorous peristaltic activity of the bowel than older patients [18]. The most common symptom of bowel perforation is protrusion of distal shunt catheters through the anus or the shunt catheter passage. This accounts for 27 of the 50 cases reported. Oral protrusion of the distal catheter was observed in 3 cases, including ours. Other common presentations were infectious signs and neurological deteri-
1975 1975 1975
1976
1982
1983
1983 1984 1987 1987 1987 1987 1988 1989 1991
[5]
[18]
[1]
[3] [17] [8] [11] [12] [19] [26] [9] [20]
30 months 10 years 14 years 15 years 18 years 10 months 3 years 23 years 69 years 6 months 8 years 18 months 29 years 2 years 18 months 1 months 9.5 years 3 years 10 months 8 years 37 months
F F F M M M F F F M M M M M F M F M M M M
F M M F
Shunt catheter passage Protrusion (anus) Fv, HA, N/V Scalp injury Protrusion (anus) ND Protrusion (anus) Fv, abdominal pain Protrusion (anus) Fv, N/V Protrusion (anus) Fv, HA, N/V Protrusion (anus) Protrusion (oral) Protrusion (anus) Protrusion (oral) Fv, HA, N/V Protrusion (anus) Protrusion (anus) Fv, abdominal pain
Protrusion (anus) N Y N Y N N Y N N Y N N N Y ? N Y Y Y Y
Y N Y N Y N N N N N N N N N N ? N Y N N N
Y S S ? SC ? ? ? S S ? ? ? SC ? SC SC ? ? PE S
S
N 1 N N N Many ? N N N N N 1 N ? N 2 N N N
N
? ? N 2 1 4 N N N N N 2 N 1 N
84 2 4 25 10 36 24 7 6 2 17 ? 27 10 ? 3 24 11 48 25
2
3 0 9 19
4 10 2 8 48 4 Mean=10
C tC dC dC SigC SB ? St tC aC SigC SB C St ? St St tC SB St
dC
tC ? SB SB SB ? dC tC tC C C SB SB ? tC
N N N E. cloacae N E. coli N Staphylococcus N N N N E. coli N E. coli, K. pneumoniae P. mirabilis E. coli, P. mirabilis, P. aerugi nosa N N E. coli, K. pneumoniae S. aureus N N Y E. coli Diphtheroids E. coli, Klebsiella E. coli, P. mirabilis E. coli N N ? Y S. epidermidis K. pneumoniae, S. fecalis N S. epidermidis
R R D R R R R D R R R ? R R R D R R R ?
R
D R D D D D R D R R R R R R R
OC
[10] [21] [24]
PE ? ? S S SC ? ? ? S ? SC SC PE S
CSF infection
1973 1974
Y N N N N Y N N N N N N N Y N
Site
[25] [13]
Y N N N N Y N Y N N N N Y Y N
Duration (months)
1971 1972
Shunt catheter passage Protrusion (anus) Autopsy found Lethargy, N/V Pulmonary embolism Shunt catheter passage Asymptomatic Abdominal pain Skin necrosis Shunt catheter passage Shunt catheter passage Protrusion (anus) Shunt malfunction Protrusion (anus) ND
Rev.Hx
[28] [22]
5 months 6 months 48 months 30 months 4 years 3 years 3 months 5 years 19 months 2 years 10 years 5 months 1 months 2 years 42 months
? F F F F F 3F, 2M
Device
1966
GI sx
[29]
CC
Peritonitis
Age
Publication year
Reference no.
Sex
coiled, S silicone, ? unmentioned, aC ascending colon, tC transverse colon, dC descending colon, SigC sigmoid colon, C colon, Rt rectum, SB small bowel, St stomach, R recovered, D died)
Table 1 Summary of reviewed cases (CC chief complaint, Rev.Hx revision history, Dur duration from operation to perforation, GI gastrointestinal, sx symptom, N no, Y yes, OC outcome, N/V nausea/vomiting, Fv fever, Asx asymptomatic, HA headache, CSF cerebrospinal fluid, ND neurological deterioration, PE polyethylene, SC spring-
186
? D R R R R R R R R ? R R R Enterobacter E. cloacae N N E. coli, K. pneumoniae N S. aureus N N E. coli N N N N St St St St SigC C C ? dC ? ? ? Rt St 32 42 4 1 day 5 6 8 12 24 4 144 8 8 48 1 N N N N N N N N N N N N N S ? ? ? ? S S S SC ? ? S SC S N N N N N N N N N N N N N N Y N N Y Y N N N N N Y N N N Fv, abdominal pain Fv, altered mentality ND Fv, N/V Protrusion (anus) Protrusion (anus) Protrusion (anus) Protrusion (anus) Protrusion (anus) Protrusion (anus) Shunt catheter passage Protrusion (anus) Protrusion (anus) Protrusion (oral) 1996 1996 1996 1997
1999
[6] [16] [27] [2]
Current
69 months 68 years 4 months 5 months 6 years 32 months 15 months 24 months 16 years 4.5 months 14 years 14 months 14 months 5 years M M F ? M F M M M M F M M F 1992 1994 1995 1995 1995 [14] [4] [7] [15] [23]
Age Sex Publication year Reference no.
Table 1 (continued)
CC
GI sx
Peritonitis
Device
Rev.Hx
Duration (months)
Site
CSF infection
OC
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oration. Gastrointestinal symptoms were present in 19 of the 50 cases, though only 7 of the patients revealed clinical evidence of peritonitis. CSF infection was present in 23 cases, and the most common organism identified was Escherichia coli (11 cases), followed by staphylococcus, klebsiella, E. cloacae, and proteus. There were 23 cases with CSF infection, and in 15 of these the causative organisms were Gram-negative organisms; in 7 of the 15 the perforation site was in the colon. Bearing in mind that bacterial colonization of the colon is higher than that of any other hollow organ of the body, it is not surprising that there is a significant correlation between colon perforation and CSF infection by Gram-negative bacteria. Therefore, the possibility of bowel perforation should be considered if Gram-negative organisms are detected in association with shunt infection. CSF infection was roughly 3 times as common as peritonitis. Among the 26 cases with infection, 4 had both CSF infection and peritonitis. Interestingly, this suggests that when a shunt catheter perforates the bowel segment, some mechanisms come into play that prevent direct extension of infection from bowel to peritoneal cavity. The infection may manifest itself at a distance from the original source. Two possible mechanisms of peritoneal protection have been suggested: (1) fibrous encasement of the catheter along its intraperitoneal course, which has been pathologically demonstrated in reported cases of bowel perforation [1, 2, 5, 15, 20, 22], and (2) coverage of the perforation site by the omentum. The mean interval between the last shunting procedure and perforation was 18.7 months, ranging from 1 day to 12 years. It was less than 3 months in only 5 cases. Therefore, it is suggested that spontaneous bowel perforation by a shunt catheter is a chronic process. Two possible mechanisms of bowel perforation have been postulated. One is that the catheter adheres to the serosal surface of the bowel by way of a foreign body reaction and subjects a fixed point to repeated pressure. This slowly produces an ulcer at the intestinal wall, eventually leading to perforation. The other is direct injury to the bowel wall during the shunting procedure [1]. This mechanism may apply in the cases in which the interval from shunt operation to perforation is very short. Although Christoph [7] and other authors suggested that bowel perforation by a shunt catheter is probably associated with previous abdominal operations, only 9 patients had a history of operations involving shunt revision or other abdominal surgery. It has been widely accepted that spring-coiled catheters are more prone to bowel perforation. Of the 30 cases in which the shunt devices used were specified, silicon catheters were used in 16, spring-coiled catheters in 11, and polyethylene catheters in 3. Because spring-coiled catheters were not as widely used as the other types, it seems likely that the spring-coiled catheter is more prone to perforation. To test the validity of this impression, we
188
need to know the numbers of cases in which each type of catheter was used. Although many authors have suggested various predisposing factors, such as male gender, malnutrition and poor general condition, length and type of catheter, and history of previous abdominal surgery, age stands out as the main risk factor for bowel perforation. The diagnosis is easily made if a shunt catheter emerges from the body. Otherwise, clinical suspicion and plain radiographs are important. The injection of radiopaque dye into the shunt device, followed by a check on the site of dye leakage is helpful. The followings are recommendations for treatment. Firstly, puncture the reservoir to collect CSF for culture and then externalize the shunt catheter. Secondly, remove the remaining distal shunt catheter. It can be removed endoscopically if the catheter tip is inside the viscus, or by pulling on it if the tip is exposed. It should not be removed proximally, because of possible contamination of the peritoneal cavity from the distal catheter segment. Invasive procedures such as laparotomy or laparoscopy are not necessary except in special circumstances. To our knowledge, there has been only 1 case report
of removal of a distal catheter by an endoscopic method which involved a sigmoidoscope for distal colon perforation [6]. Thirdly, ensure that infection is absent or controlled by laboratory findings. In our case, we tried to keep the patient in a fasting state for 2 weeks to let the perforated site heal. Oral intake may be allowed sooner, however, because the perforation site is so small and 2 or 3 days would be enough for regeneration of the gastrointestinal mucosa. External ventricular drainage is necessary when CSF infection is uncontrollable. Fourthly, the shunt should be revised. Bowel perforation by a shunt catheter has resulted in relatively serious outcomes. Ten of the 50 patients died, and 7 of them had CSF infection. Azimi et al. [5] proposed possible life-threatening complications of bowel perforation: ventriculitis, meningitis, intraperitoneal abscess, fecal fistulae, and generalized peritonitis. Sharma [26] also mentioned septic complications as the most important prognostic factors. The majority of cases with poor outcome were in old reports. In recent cases the results are not so disappointing owing to effective treatment with antibiotics.
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17. Miserocchi G, Sironi VA, Ravagnati L (1984) Anal protrusion as a complication of ventriculo-peritoneal shunt. Case report and review of the literature. J Neurosurg Sci 28:43–46 18. Nishijima M, Endoh S, Ohyama H, Higuchi H (1982) Gastric perforation by a ventriculoperitoneal shunt. Neurosurgery 10:754–756 19. Oi S, Shose Y, Asano N, Oshio T, Matsumoto S (1987) Intragastric migration of a ventriculoperitoneal shunt catheter. Neurosurgery 21:255–257 20. Oshio T, Matsumura C, Kirino A, Go M, Bando Y, Manabe Y, Nakagawa Y (1991) Recurrent perforations of viscus due to ventrculoperitoneal shunt in a hydrocephalic child. J Pediatr Surg 26:1404–1405 21. Peirce KR, Loeser JD (1975) Perforation of the intestine by a Raimondi peritoneal catheter. J Neurosurg 43:112–113 22. Rubin RC, Ghatak NR, Visudhipan P (1972) Asymptomatic perforated viscus and gram-negative ventriculitis as a complication of valve-regulated ventriculoperitoneal shunts. J Neurosurg 37:616–618
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23. Sami A, Ait Ben Ali S, Choukry M, Achouri M, Naja A, Ouboukhlik A, Elkamar A, Elazhari A, Boucetta M (1995) Anal migration of ventriculoperitoneal shunt catheter. A propos of 3 cases. Neurochirurgie 41:315–318 24. Schulhof LA, Worth RM, Kalsbeck JE (1975) Bowel perforation due to peritoneal shunt. A report of seven cases and review of the literature. Surg Neurol 3:265–269
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