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Injury to the common iliac artery during suction rectal biopsy
CASE REPORTS
Injury to the Common Iliac Artery During Suction Rectal Biopsy By E.L. Cusick a n d R.G. Buick
Birmingham, England • A case of injury to the common iliac artery during suction rectal biopsy is described. Factors that might influence the incidence of complications after suction rectal biopsy are considered. Suction rectal biopsy, although invaluable in establishing the diagnosis of Hirschsprung's disease, is a potentially dangerous technique and should not be deb egated to an inexperienced operator. In neonates it is unwise to take biopsy specimens more than 4 cm from the anal verge. A posterior approach is likely to be safer than an anterior or lateral one.
Copyright © 1995 by W.B. Saunders Company
The patient was extubated on the second postoperative day, and her recovery period was uncomplicated. The rectal biopsy specimens showed a negative acetylcholinesterase reaction; ganglia were found in the submucosa of all biopsy specimens, thus excluding the diagnosis of Hirschsprung's disease. She tolerated oral feeding well and was discharged on the 14th postoperative day. She had no other gastrointestinal problems, and the result of a sweat test (performed on an outpatient basis) was negative. She is now 3 years old and has had no problems related to the vascular injury. Although slightly diminished in volume, her left femoral pulse is palpable, there are no audible bruits, and there has been no discrepancy in limb growth. She continues to have annual follow-uP examinations.
INDEX WORDS: Rectal biopsy, suction, complications. DISCUSSION
UCTION RECTAL biopsy is widely accepted as a safe accurate method of diagnosing Hirschsprung's disease. The technique can be used without anesthesia from the neonatal period onward, using the apparatus devised by Noblett. 1 Although in early studies there were no significant complications, 1,2 more recently there have been serious complications 3,4including at least one death) We present a case of iliac artery injury, a potentially fatal complication of suction rectal biopsy that has not been reported previously.
S
CASE REPORT An infant girl born at 33 weeks' gestation was transferred to Birmingham Children's Hospital at 4 days of age. She had not passed meconium until 36 hours after birth, at which time abdominal distension and umbilical flare were observed. The abdominal distension persisted in the absence of other symptoms, and triple antibiotics were begun. Physically she appeared healthy and had a distended nontender abdomen. Abdominal x-rays did not confirm the suspected diagnosis of necrotizing enterocolitis, but she received parenteral nutrition and triple antibiotics for 10 days. Thereafter enteral feeding was begun, which she was slow to tolerate. Her abdomen remained slightly distended, and bowels opened infrequently. An upper gastrointestinal contrast series proved normal, and a suction rectal biopsy was undertaken on day 20 t o exclude the diagnosis of Hirschsprung's disease; the specimens were taken 2, 4, and 6 cm from the anal verge, which was our usual practice for older children. Immediately after the procedure she became grey and apneic, with an impalpable pulse and rapidly distending abdomen. She was resuscitated, and emergency investigations showed a hemoglobin level of 3.8 g / d E A further circulatory/respiratory collapse precipitated intubation and ventilation before emergency surgery. During lapar0tomy, the abdomen was found to be full of blood, Pressure was applied at the aortic bifurcation, and a 3- x l-ram hole was found in the left common iliae artery, just proximal to its bifurcation. The rectum was perforated above the peritonea ! reflection, which was repaired by primary suture. The initial attempt to repair the arterial defect by primary suturing resulted in significant narrowing of the artery. Satisfactory repair was achieved with a patch taken from the umbilical artery.
Journalof Pediatric Surgery, Vo130, No 1 (January), 1995: pp 111-112
In 1965, Dobbins and Bill described a technique of suction rectal biopsy to exclude the diagnosis of Hirschsprung's disease by demonstrating the presence of ganglion cells within the rectal submucosa. 5 Unlike the older technique of wedge biopsy, the procedure was performed without anesthesia. By Specifically aiming to avoid full-thickness specimens, it was expected that the recognized complications of serious bleeding, perforation, sepsis, and perirectal fibrosis 6,7 would be avoided. The technique was refined by Noblett in 1969 with the development of a specific rectal suction biopsy tube.1 Using this apparatus, it was believed that rectal biopsy was simple and safe in the hands of relatively inexperienced operators. 1 Initial reports encompassing large numbers of biopsies confirmed this view, ie, no serious complications wer e reported among 208 consecutive biopsies. 2 However, more recently there have been several cases that highlight the potential hazards of suction rectal biopsy. In 1983, Rees et al reported five cases of inadvertent full-thickness biopsy specimens taken in neonates, including three patients who had evidence of perforation, one of whom died of overwhelming pelvic sepsis? They also reported three cases of rectal hemorrhage necessitating transfusion) In 1989, Jani et al reported a case of peripheral limb gangrene accompanying a severe toxic episode that developed 2 days afte r suction rectal biopsy? In assessing the complications arising from suction From the Department of Paediatric Surgery, Birmingham Children's Hospital, Birmingham, England. Address reprint requests to E.L. Cusick, Senior Registrar in Paediatric Surgery, Clarendon Wing, Leeds General Infirmary, Leeds LS2 9NS, England. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3001-0028503. 00/0 111
112
CUSICK AND BUICK
Pressure Drop (mmHg) 500 400 300
*
1~
200 100
1
2
3
4
5
Volume Aspirated r-I 5ml Syringe
A1OmlSyringe
o2OmlSyringe
*Calculated
Fig 1. Rectal suction biopsy device: syringe size versus pressure drop.
rectal biopsy, two factors should be considered: the technique applied and the indications for its use. The suction device was fully described by Noblett in 1969.1 In brief, a small portion of tissue is sucked into a side apperture of the instrument and excised by the action of a hollow cylindrica ! knife that lies within the device. Using a graduated manometer, Noblett limited the negative pressure to 20 to 25 inches of mercury) She acknowledged that this value was high but believed that the risk of perforation was minimal because none of the specimens in the study group contained longitudinal or circular muscle. ~ Lower pressures were found to yield an inadequate depth of submucosa. Common Practice involves use of a 20- or 50-mL syringe as the source of suction, without an intervening manometer. We wished to determine whether this could produce an excessive negative pressure and tested the pressures induced. As stated in Boyles' law, for a given temperature, pressure x volume is a constant. Thus, the negative pressure created by using a syringe can be calculated easily. Using a manometer calibrated to measure negative pressure, we applied suction to a standard suction rectal biopsy device (volume, 4 mL) using 5-, 10-, and 20-mL syringes (successively), with the plunger withdrawn to 1, 2, 3, 4, and 5 mL, in turn. In addition, we calculated the theoretical negative pressure achieved at these volumes, this being independent of syringe size (Fig 1). The calculated negative pressure that can be produced by a standard rectal suction biopsy device (volume, 4 mL) at 20 mL and 50
mL is 643 mm Hg (25 inches of Hg) and 700 mm Hg (28 inches of Hg). In practice, as is seen from the experimental situation, a lower pressure is likely to be achieved. We achieved pressures of approximately 3~ of the expected value. The pressures achieved with syringes are within the range recommended by Noblett, and use of a graduated manometer should not be necessary. Performing multiple biopsie s is required to increase the likelihood of obtaining an adequate specimen; specimens taken too low can yield equivocal results. Dobbins and Bill stressed the need to guard against perforation of the rectosigmoid in infants by limiting biopsies to below the level of the peritoneal reflection, 5 and Noblett recommended that the specimens be taken 3 to 4 cm from the anal valves. 1 In the present case, the specimens were taken 2, 4, and 6 cm from the anal verge; the latter proved to be fullthickness and was the presumed source of perforation and vessel injury. Rees et al questioned the advisability of their standard procedure of taking biopsy specimens at 2, 4, and 5 cm in neonates, 3 while observing that perforation below the level of the peritoneal reflection (as in their fatal case) may be more dangerous than at a higher level because of the difficulties of treatment. We suggest that al! biopsies be taken from the posterior rectal wall, thereby limiting the potential for damage to intraperitoneal structures. In neonates, we no longer take specimens from further than 4 cm from the anal verge. Through review of all the reported cases, it is evident that none of the neonates with bowel perforation had Hirschsprung's disease. Whether this reflects some intrinsic difference in the bowel wall in Hirschsprung's disease, which renders perforation less likely, or reflects the larger number of negative biopsy specimens is unclear. It appears likely that premature infants (as in our case) have a greater risk of perforation, but information on gestational age is not included in previous publications. It should be recognized that the very useful technique of suction rectal biopsy is potentially dangerous in neonates. ACKNOWLEDGMENT The authors acknowledge the assistance of Dr Tom Cluttenbrock of the Department of Anaesthesia at Queen Elizabeth Hospital, Birmingham.
REFERENCES 1. Noblett HR: A rectal suction biopsy tube for use in diagnosis of Hirschsprung's Disease. J Pediatr Surg 4:406-409, 1969 2. Campbell PE, Noblett HR: Experience with rectal suction biopsy in the diagnosis of Hirschsprung's disease. J Pediatr Surg 4:410-415, 1969 3. Rees BI, Azmy A, Nigram M, et al: Complications of rectal suction biopsy. J Pediatr Surg 18:273-275, 1983
4. Jani BR, Brereton RJ, Dillon MJ: Peripheral limb gangrene following rectal biopsy. Clin Pediatr 28:585-588, 1989 5. Dobbins WO, Bill AH: Diagnosis of Hirschsprung's disease excluded by rectal suction biopsy. N Engl J Med 272:990-993, 1965 6. Shandling B: New technique in diagnosis of Hirsehsprung's disease. Can J Surg 4:298-305, 1961 7. Fraser GC, Wilkinson AW: Neonatal Hirschsprung's disease. Br Med J 3:7-10, 1967
Injury to the Common Iliac Artery During Suction Rectal Biopsy By E.L. Cusick a n d R.G. Buick
Birmingham, England • A case of injury to the common iliac artery during suction rectal biopsy is described. Factors that might influence the incidence of complications after suction rectal biopsy are considered. Suction rectal biopsy, although invaluable in establishing the diagnosis of Hirschsprung's disease, is a potentially dangerous technique and should not be deb egated to an inexperienced operator. In neonates it is unwise to take biopsy specimens more than 4 cm from the anal verge. A posterior approach is likely to be safer than an anterior or lateral one.
Copyright © 1995 by W.B. Saunders Company
The patient was extubated on the second postoperative day, and her recovery period was uncomplicated. The rectal biopsy specimens showed a negative acetylcholinesterase reaction; ganglia were found in the submucosa of all biopsy specimens, thus excluding the diagnosis of Hirschsprung's disease. She tolerated oral feeding well and was discharged on the 14th postoperative day. She had no other gastrointestinal problems, and the result of a sweat test (performed on an outpatient basis) was negative. She is now 3 years old and has had no problems related to the vascular injury. Although slightly diminished in volume, her left femoral pulse is palpable, there are no audible bruits, and there has been no discrepancy in limb growth. She continues to have annual follow-uP examinations.
INDEX WORDS: Rectal biopsy, suction, complications. DISCUSSION
UCTION RECTAL biopsy is widely accepted as a safe accurate method of diagnosing Hirschsprung's disease. The technique can be used without anesthesia from the neonatal period onward, using the apparatus devised by Noblett. 1 Although in early studies there were no significant complications, 1,2 more recently there have been serious complications 3,4including at least one death) We present a case of iliac artery injury, a potentially fatal complication of suction rectal biopsy that has not been reported previously.
S
CASE REPORT An infant girl born at 33 weeks' gestation was transferred to Birmingham Children's Hospital at 4 days of age. She had not passed meconium until 36 hours after birth, at which time abdominal distension and umbilical flare were observed. The abdominal distension persisted in the absence of other symptoms, and triple antibiotics were begun. Physically she appeared healthy and had a distended nontender abdomen. Abdominal x-rays did not confirm the suspected diagnosis of necrotizing enterocolitis, but she received parenteral nutrition and triple antibiotics for 10 days. Thereafter enteral feeding was begun, which she was slow to tolerate. Her abdomen remained slightly distended, and bowels opened infrequently. An upper gastrointestinal contrast series proved normal, and a suction rectal biopsy was undertaken on day 20 t o exclude the diagnosis of Hirschsprung's disease; the specimens were taken 2, 4, and 6 cm from the anal verge, which was our usual practice for older children. Immediately after the procedure she became grey and apneic, with an impalpable pulse and rapidly distending abdomen. She was resuscitated, and emergency investigations showed a hemoglobin level of 3.8 g / d E A further circulatory/respiratory collapse precipitated intubation and ventilation before emergency surgery. During lapar0tomy, the abdomen was found to be full of blood, Pressure was applied at the aortic bifurcation, and a 3- x l-ram hole was found in the left common iliae artery, just proximal to its bifurcation. The rectum was perforated above the peritonea ! reflection, which was repaired by primary suture. The initial attempt to repair the arterial defect by primary suturing resulted in significant narrowing of the artery. Satisfactory repair was achieved with a patch taken from the umbilical artery.
Journalof Pediatric Surgery, Vo130, No 1 (January), 1995: pp 111-112
In 1965, Dobbins and Bill described a technique of suction rectal biopsy to exclude the diagnosis of Hirschsprung's disease by demonstrating the presence of ganglion cells within the rectal submucosa. 5 Unlike the older technique of wedge biopsy, the procedure was performed without anesthesia. By Specifically aiming to avoid full-thickness specimens, it was expected that the recognized complications of serious bleeding, perforation, sepsis, and perirectal fibrosis 6,7 would be avoided. The technique was refined by Noblett in 1969 with the development of a specific rectal suction biopsy tube.1 Using this apparatus, it was believed that rectal biopsy was simple and safe in the hands of relatively inexperienced operators. 1 Initial reports encompassing large numbers of biopsies confirmed this view, ie, no serious complications wer e reported among 208 consecutive biopsies. 2 However, more recently there have been several cases that highlight the potential hazards of suction rectal biopsy. In 1983, Rees et al reported five cases of inadvertent full-thickness biopsy specimens taken in neonates, including three patients who had evidence of perforation, one of whom died of overwhelming pelvic sepsis? They also reported three cases of rectal hemorrhage necessitating transfusion) In 1989, Jani et al reported a case of peripheral limb gangrene accompanying a severe toxic episode that developed 2 days afte r suction rectal biopsy? In assessing the complications arising from suction From the Department of Paediatric Surgery, Birmingham Children's Hospital, Birmingham, England. Address reprint requests to E.L. Cusick, Senior Registrar in Paediatric Surgery, Clarendon Wing, Leeds General Infirmary, Leeds LS2 9NS, England. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3001-0028503. 00/0 111
112
CUSICK AND BUICK
Pressure Drop (mmHg) 500 400 300
*
1~
200 100
1
2
3
4
5
Volume Aspirated r-I 5ml Syringe
A1OmlSyringe
o2OmlSyringe
*Calculated
Fig 1. Rectal suction biopsy device: syringe size versus pressure drop.
rectal biopsy, two factors should be considered: the technique applied and the indications for its use. The suction device was fully described by Noblett in 1969.1 In brief, a small portion of tissue is sucked into a side apperture of the instrument and excised by the action of a hollow cylindrica ! knife that lies within the device. Using a graduated manometer, Noblett limited the negative pressure to 20 to 25 inches of mercury) She acknowledged that this value was high but believed that the risk of perforation was minimal because none of the specimens in the study group contained longitudinal or circular muscle. ~ Lower pressures were found to yield an inadequate depth of submucosa. Common Practice involves use of a 20- or 50-mL syringe as the source of suction, without an intervening manometer. We wished to determine whether this could produce an excessive negative pressure and tested the pressures induced. As stated in Boyles' law, for a given temperature, pressure x volume is a constant. Thus, the negative pressure created by using a syringe can be calculated easily. Using a manometer calibrated to measure negative pressure, we applied suction to a standard suction rectal biopsy device (volume, 4 mL) using 5-, 10-, and 20-mL syringes (successively), with the plunger withdrawn to 1, 2, 3, 4, and 5 mL, in turn. In addition, we calculated the theoretical negative pressure achieved at these volumes, this being independent of syringe size (Fig 1). The calculated negative pressure that can be produced by a standard rectal suction biopsy device (volume, 4 mL) at 20 mL and 50
mL is 643 mm Hg (25 inches of Hg) and 700 mm Hg (28 inches of Hg). In practice, as is seen from the experimental situation, a lower pressure is likely to be achieved. We achieved pressures of approximately 3~ of the expected value. The pressures achieved with syringes are within the range recommended by Noblett, and use of a graduated manometer should not be necessary. Performing multiple biopsie s is required to increase the likelihood of obtaining an adequate specimen; specimens taken too low can yield equivocal results. Dobbins and Bill stressed the need to guard against perforation of the rectosigmoid in infants by limiting biopsies to below the level of the peritoneal reflection, 5 and Noblett recommended that the specimens be taken 3 to 4 cm from the anal valves. 1 In the present case, the specimens were taken 2, 4, and 6 cm from the anal verge; the latter proved to be fullthickness and was the presumed source of perforation and vessel injury. Rees et al questioned the advisability of their standard procedure of taking biopsy specimens at 2, 4, and 5 cm in neonates, 3 while observing that perforation below the level of the peritoneal reflection (as in their fatal case) may be more dangerous than at a higher level because of the difficulties of treatment. We suggest that al! biopsies be taken from the posterior rectal wall, thereby limiting the potential for damage to intraperitoneal structures. In neonates, we no longer take specimens from further than 4 cm from the anal verge. Through review of all the reported cases, it is evident that none of the neonates with bowel perforation had Hirschsprung's disease. Whether this reflects some intrinsic difference in the bowel wall in Hirschsprung's disease, which renders perforation less likely, or reflects the larger number of negative biopsy specimens is unclear. It appears likely that premature infants (as in our case) have a greater risk of perforation, but information on gestational age is not included in previous publications. It should be recognized that the very useful technique of suction rectal biopsy is potentially dangerous in neonates. ACKNOWLEDGMENT The authors acknowledge the assistance of Dr Tom Cluttenbrock of the Department of Anaesthesia at Queen Elizabeth Hospital, Birmingham.
REFERENCES 1. Noblett HR: A rectal suction biopsy tube for use in diagnosis of Hirschsprung's Disease. J Pediatr Surg 4:406-409, 1969 2. Campbell PE, Noblett HR: Experience with rectal suction biopsy in the diagnosis of Hirschsprung's disease. J Pediatr Surg 4:410-415, 1969 3. Rees BI, Azmy A, Nigram M, et al: Complications of rectal suction biopsy. J Pediatr Surg 18:273-275, 1983
4. Jani BR, Brereton RJ, Dillon MJ: Peripheral limb gangrene following rectal biopsy. Clin Pediatr 28:585-588, 1989 5. Dobbins WO, Bill AH: Diagnosis of Hirschsprung's disease excluded by rectal suction biopsy. N Engl J Med 272:990-993, 1965 6. Shandling B: New technique in diagnosis of Hirsehsprung's disease. Can J Surg 4:298-305, 1961 7. Fraser GC, Wilkinson AW: Neonatal Hirschsprung's disease. Br Med J 3:7-10, 1967