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Epidural abscess complicating swenson procedure: A case report and a review of the literature
Epidural A b s c e s s Complicating S w e n s o n Procedure: A Case Report and a Review of the Literature By D. A. K. Watters, S. A. Moussa, and N. Buyukpamukcu Edinburgh, Scotland
9 A case is reported in which an anastomotic leak following the Swenson procedure for Hirschsprung's disease was complicated by the development of a pelvic abscess that communicated freely with the epidural space. The child presented with signs and symptoms of an epidural abscess, but his myelogram was normal. The diagnosis was made by urografin enema. The child was treated by defunctioning colostomy and drainage of the pelvic abscess by enlarging the defect at the anastomosis site digitally. The epidural space drained freely to the pelvis and therefore laminectomy was not required. The possible etiology of such a communication is discussed.
H E r a t e of a n a s t o m o t i c l e a k a g e f o l l o w i n g t h e S w e n s o n p r o c e d u r e for t h e t r e a t m e n t o f H i r s c h s p r u n g ' s d i s e a s e is r e p o r t e d as 5%. 1 T h e i n c i d e n c e o f pelvic abscess in t h e s a m e series was 2.9%. A r e s e r v o i r o f sepsis c a u s e d by a n y p a t h o g e n i c o r g a n i s m m a y l e a d to t h e d e v e l o p m e n t of an e p i d u r a l abscess. 2 I n f e c t i o n m a y s p r e a d d i r e c t l y f r o m an o s t e o m y e l i t i c v e r t e b r a , via B a t s o n ' s v e n o u s plexus, by t h e a r t e r i a l c i r c u l a t i o n or by spread along lymphatic channels from a pre v e r t e b r a l or post v e r t e b r a l abscess. 2'4
T
CASE REPORT
Our patient Richard first presented when 12 days old with abdominal distension and intestinal obstruction. A diagnosis of Hirschsprung's disease was made, confirmed by rectal biopsy, and a defunctioning transverse colostomy carried out. At the age of 6 months the aganglionic segment was excised and a Swenson pull-through procedure was performed. Postoperatively he developed a pelvic abscess secondary to anastomotic breakdown which on healing led to stenosis. This required regular dilatations before his colostomy was closed at the age of 1 year. At the age of 21/2 Richard was admitted with a 10-day history of pyrexia which had failed to respond to ampicillin. He complained of lower back pain, but there were no other symptoms. Examination revealed an ill, irritable child with From the Surgical Paediatric Registrar, Western General Hospital, Edinburgh, and the Professor o f Paediatric Surgery, Hacettepe University Medical Faculty, Ankara, Turkey. Address reprint requests to Mr. D. A. K. Watters, Surgical Registrar, Western General Hospital, Crewe Road, Edinburgh EH4 2XU, Scotland. 9 1984 by Grune & Stratton, Inc. 0022-3468/84/1902-002350t.00/0 218
no neck stiffness, a negative Kernig's sign but with back rigidity and resistance to spinal flexion. There was stenosis at his coloanal anastomosis and foul-smelling watery stools. Full rectal examination was not possible because of his stricture. The WBC was 22.7, showing a neutrophil leucocytosis. There was also a microcytic, hypochromic anemia with an Hb of 8.2 g / d E The chest x-ray was normal. A lumbar puncture was performed and a small quantity of pus was obtained. A repeat lumbar puncture also yielded pus. Mixed organisms were identified by microscopy and E. coli was later grown on culture. A diagnosis of spinal abscess, possibly originating from bowel in view of the mixed flora on microscopy, was made. A cysternomyelogram was carried out which showed clear CSF with 8 WBC/mm 3 and no growth on culture. The myelogram revealed no abnormality. Treatment commenced with daily rectal washouts in view of the foul-smelling liquid feces and the risk of enterocolitis. Richard failed to show improvement with antibiotics over the next three days. He developed bilateral psoas signs and his WBC and ESR remained high. Bone scan and ultrasound did not show any abnormality. Eight days after his original admission a urografin enema demonstrated a fistula from the site of coloanal anastomosis to the pre-sacral space and thence to the epidural space (Fig. 1). A colostomy was carried out and examination under anesthetic revealed the site of breakdown in the anastomosis on the posterior wall, and pus was drained. The child remained on an antibiotic combination of gentamicin, metronidazole, cloxacillin, and benzyl penicillin and made a steady recovery. Three months after his colostomy a repeat gastrografin enema showed no communication with the epidural space despite persistence of the anastomotic defect (Fig. 2) that will require further surgical treatment. DISCUSSION
T h i s c a s e is r e m a r k a b l e in two respects: first, to o u r k n o w l e d g e , a c u t e spinal e p i d u r a l abscess s e c o n d a r y to a n a s t o m o t i c l e a k a g e in t h e pelvis has not b e e n p r e v i o u s l y r e p o r t e d . S e c o n d , w h i l e the s t a n d a r d t r e a t m e n t of e p i d u r a l collections of pus is l a m i n e c t o m y 4 this was not n e c e s s a r y in this child b e c a u s e t h e r e was f r e e c o m m u n i c a t i o n b e t w e e n t h e e p i d u r a l s p a c e a n d t h e p e l v i c presacral abscess w h i c h was d r a i n e d rectally. T h e m o d e o f d e v e l o p m e n t o f the fistula b e t w e e n t h e p r e - s a c r a l abscess and t h e e p i d u r a l s p a c e is u n c e r t a i n . T h e r e was no n e u r o e n t e r i c cyst or fistula found w h e n t h e initial S w e n s o n p r o c e d u r e was c a r r i e d out. S i n c e the b o n e scan a n d plain films o f t h e v e r t e b r a e a n d s a c r u m w e r e n o r m a l t h e e p i d u r a l c o l l e c t i o n did not a r i s e by Journal of Pediatric Surgery, Vol. 19, No. 2 (April), 1984
SWENSON PROCEDURE
219
Fig. 1. Contrast enema demonstrating pre-sacral abscess and communication with the epidural space.
Fig. 2. Contrast enema 3 months later shows no communication with epidural space.
means of an intervening osteomyelitis which is the most common mode of spread. 2"4 A recently reported case 5 of epidural abscess at the level of T2 following pelvic infection due to an intrauterine contraceptive device, emphasized the importance of Batson's venous plexus in the pathogenesis of epidural abscess. However, a blood-born spread is unlikely to lead to the development of a two-day communication between the pelvis and epidural space. There are lymphatic channels between the pre-vertebral and epidural spaces which enter the foramina with the nerve roots. 4 It is recognized that an epidural abscess may present with postvertebral pus since the abscess may drain via the lymphatics communicating with this space. Another theoretical mode of spread would be via an anterior sacral defect (spina bifida). Sacral and pelvic x-rays have failed to demonstrate any sacral defect. It is possible that the pressure of pus from the abscess opened up tissue planes through the sacral foramina but if this
were so we would have expected other cases to have been reported. Thus the mode of development of this fistula is uncertain. The case we have presented had back pain and various reports have repeatedly emphasized the importance of considering the diagnosis of epidural abscess in patients with back pain. 2'4'7'8However, epidural abscess is rare in young children. Baker 6 found only six cases in the world literature and reported on a seventh. She emphasized that in young children the symptoms are nonspecific (fever, irritability) and that the classical presentation of back, leg, or abdominal pain with root signs is less likely, as children are poor historians. Our difficulty in making the correct diagnosis was due to the normal myelogram despite the presence of epidural pus and this is very unusual. The stricture at the anastomotic site prevented adequate rectal examination which otherwise might have suggested the presence of a pelvic abscess sooner.
REFERENCES
1. Swenson O, Sherman JO. Fisher JR, et al: The treatment and post-operativecomplications of congenital mega-
colon; a twenty-fiveyear follow-up. Ann Surg 182:266-273, 1975
220
2. Heusner AP: Non-tuberculosis spinal epidural infections. New Engl J Med 239:845-854, 1948 3. Hulme A, Dott NM: Spinal epidural abscess. Br Med J
1:64-68, 1954 4. Baker AS, Ojemann RG, Swartz MN, et al: Spinal epidural abscess. N Engl J Med 293:463 468, 1975 5. Atkinson L, Deambrosis W, Sheehy J, et al: A spinal
WAI-FERS, MOUSSA, AND BUYUKPAMUKCU
epidural abscess complicating an intra-uterine contraceptive device. Aust N Z J Obstet Gynaecol 18:272-273, 1978 6. Baker C J: Primary spinal epidural abscess. Am J Dis Child 121:337-339, 1971 7. Hakin RN, Burt AA, Cook JB: Acute spinal epidural abscess. Paraplegia 17:330-336, 1979 8. Hancock DO: A study of 49 patients with acute spinal extradural abscess. Paraplegia 10:285-288, 1973
9 A case is reported in which an anastomotic leak following the Swenson procedure for Hirschsprung's disease was complicated by the development of a pelvic abscess that communicated freely with the epidural space. The child presented with signs and symptoms of an epidural abscess, but his myelogram was normal. The diagnosis was made by urografin enema. The child was treated by defunctioning colostomy and drainage of the pelvic abscess by enlarging the defect at the anastomosis site digitally. The epidural space drained freely to the pelvis and therefore laminectomy was not required. The possible etiology of such a communication is discussed.
H E r a t e of a n a s t o m o t i c l e a k a g e f o l l o w i n g t h e S w e n s o n p r o c e d u r e for t h e t r e a t m e n t o f H i r s c h s p r u n g ' s d i s e a s e is r e p o r t e d as 5%. 1 T h e i n c i d e n c e o f pelvic abscess in t h e s a m e series was 2.9%. A r e s e r v o i r o f sepsis c a u s e d by a n y p a t h o g e n i c o r g a n i s m m a y l e a d to t h e d e v e l o p m e n t of an e p i d u r a l abscess. 2 I n f e c t i o n m a y s p r e a d d i r e c t l y f r o m an o s t e o m y e l i t i c v e r t e b r a , via B a t s o n ' s v e n o u s plexus, by t h e a r t e r i a l c i r c u l a t i o n or by spread along lymphatic channels from a pre v e r t e b r a l or post v e r t e b r a l abscess. 2'4
T
CASE REPORT
Our patient Richard first presented when 12 days old with abdominal distension and intestinal obstruction. A diagnosis of Hirschsprung's disease was made, confirmed by rectal biopsy, and a defunctioning transverse colostomy carried out. At the age of 6 months the aganglionic segment was excised and a Swenson pull-through procedure was performed. Postoperatively he developed a pelvic abscess secondary to anastomotic breakdown which on healing led to stenosis. This required regular dilatations before his colostomy was closed at the age of 1 year. At the age of 21/2 Richard was admitted with a 10-day history of pyrexia which had failed to respond to ampicillin. He complained of lower back pain, but there were no other symptoms. Examination revealed an ill, irritable child with From the Surgical Paediatric Registrar, Western General Hospital, Edinburgh, and the Professor o f Paediatric Surgery, Hacettepe University Medical Faculty, Ankara, Turkey. Address reprint requests to Mr. D. A. K. Watters, Surgical Registrar, Western General Hospital, Crewe Road, Edinburgh EH4 2XU, Scotland. 9 1984 by Grune & Stratton, Inc. 0022-3468/84/1902-002350t.00/0 218
no neck stiffness, a negative Kernig's sign but with back rigidity and resistance to spinal flexion. There was stenosis at his coloanal anastomosis and foul-smelling watery stools. Full rectal examination was not possible because of his stricture. The WBC was 22.7, showing a neutrophil leucocytosis. There was also a microcytic, hypochromic anemia with an Hb of 8.2 g / d E The chest x-ray was normal. A lumbar puncture was performed and a small quantity of pus was obtained. A repeat lumbar puncture also yielded pus. Mixed organisms were identified by microscopy and E. coli was later grown on culture. A diagnosis of spinal abscess, possibly originating from bowel in view of the mixed flora on microscopy, was made. A cysternomyelogram was carried out which showed clear CSF with 8 WBC/mm 3 and no growth on culture. The myelogram revealed no abnormality. Treatment commenced with daily rectal washouts in view of the foul-smelling liquid feces and the risk of enterocolitis. Richard failed to show improvement with antibiotics over the next three days. He developed bilateral psoas signs and his WBC and ESR remained high. Bone scan and ultrasound did not show any abnormality. Eight days after his original admission a urografin enema demonstrated a fistula from the site of coloanal anastomosis to the pre-sacral space and thence to the epidural space (Fig. 1). A colostomy was carried out and examination under anesthetic revealed the site of breakdown in the anastomosis on the posterior wall, and pus was drained. The child remained on an antibiotic combination of gentamicin, metronidazole, cloxacillin, and benzyl penicillin and made a steady recovery. Three months after his colostomy a repeat gastrografin enema showed no communication with the epidural space despite persistence of the anastomotic defect (Fig. 2) that will require further surgical treatment. DISCUSSION
T h i s c a s e is r e m a r k a b l e in two respects: first, to o u r k n o w l e d g e , a c u t e spinal e p i d u r a l abscess s e c o n d a r y to a n a s t o m o t i c l e a k a g e in t h e pelvis has not b e e n p r e v i o u s l y r e p o r t e d . S e c o n d , w h i l e the s t a n d a r d t r e a t m e n t of e p i d u r a l collections of pus is l a m i n e c t o m y 4 this was not n e c e s s a r y in this child b e c a u s e t h e r e was f r e e c o m m u n i c a t i o n b e t w e e n t h e e p i d u r a l s p a c e a n d t h e p e l v i c presacral abscess w h i c h was d r a i n e d rectally. T h e m o d e o f d e v e l o p m e n t o f the fistula b e t w e e n t h e p r e - s a c r a l abscess and t h e e p i d u r a l s p a c e is u n c e r t a i n . T h e r e was no n e u r o e n t e r i c cyst or fistula found w h e n t h e initial S w e n s o n p r o c e d u r e was c a r r i e d out. S i n c e the b o n e scan a n d plain films o f t h e v e r t e b r a e a n d s a c r u m w e r e n o r m a l t h e e p i d u r a l c o l l e c t i o n did not a r i s e by Journal of Pediatric Surgery, Vol. 19, No. 2 (April), 1984
SWENSON PROCEDURE
219
Fig. 1. Contrast enema demonstrating pre-sacral abscess and communication with the epidural space.
Fig. 2. Contrast enema 3 months later shows no communication with epidural space.
means of an intervening osteomyelitis which is the most common mode of spread. 2"4 A recently reported case 5 of epidural abscess at the level of T2 following pelvic infection due to an intrauterine contraceptive device, emphasized the importance of Batson's venous plexus in the pathogenesis of epidural abscess. However, a blood-born spread is unlikely to lead to the development of a two-day communication between the pelvis and epidural space. There are lymphatic channels between the pre-vertebral and epidural spaces which enter the foramina with the nerve roots. 4 It is recognized that an epidural abscess may present with postvertebral pus since the abscess may drain via the lymphatics communicating with this space. Another theoretical mode of spread would be via an anterior sacral defect (spina bifida). Sacral and pelvic x-rays have failed to demonstrate any sacral defect. It is possible that the pressure of pus from the abscess opened up tissue planes through the sacral foramina but if this
were so we would have expected other cases to have been reported. Thus the mode of development of this fistula is uncertain. The case we have presented had back pain and various reports have repeatedly emphasized the importance of considering the diagnosis of epidural abscess in patients with back pain. 2'4'7'8However, epidural abscess is rare in young children. Baker 6 found only six cases in the world literature and reported on a seventh. She emphasized that in young children the symptoms are nonspecific (fever, irritability) and that the classical presentation of back, leg, or abdominal pain with root signs is less likely, as children are poor historians. Our difficulty in making the correct diagnosis was due to the normal myelogram despite the presence of epidural pus and this is very unusual. The stricture at the anastomotic site prevented adequate rectal examination which otherwise might have suggested the presence of a pelvic abscess sooner.
REFERENCES
1. Swenson O, Sherman JO. Fisher JR, et al: The treatment and post-operativecomplications of congenital mega-
colon; a twenty-fiveyear follow-up. Ann Surg 182:266-273, 1975
220
2. Heusner AP: Non-tuberculosis spinal epidural infections. New Engl J Med 239:845-854, 1948 3. Hulme A, Dott NM: Spinal epidural abscess. Br Med J
1:64-68, 1954 4. Baker AS, Ojemann RG, Swartz MN, et al: Spinal epidural abscess. N Engl J Med 293:463 468, 1975 5. Atkinson L, Deambrosis W, Sheehy J, et al: A spinal
WAI-FERS, MOUSSA, AND BUYUKPAMUKCU
epidural abscess complicating an intra-uterine contraceptive device. Aust N Z J Obstet Gynaecol 18:272-273, 1978 6. Baker C J: Primary spinal epidural abscess. Am J Dis Child 121:337-339, 1971 7. Hakin RN, Burt AA, Cook JB: Acute spinal epidural abscess. Paraplegia 17:330-336, 1979 8. Hancock DO: A study of 49 patients with acute spinal extradural abscess. Paraplegia 10:285-288, 1973