Appendicoumbilical
Fistula as a Sequela of Perforated Appendicitis
By Woo-Hyun
Park, Soon-Ok
Choi, Seong-Ku Taegu, Korea
Woo, and Sang-Sook
Lee
0 This is the first report, we believe, of a 5-year-old boy who developed appendicoumbilical fistula as a sequela of perforated appendicitis. We discuss a proposed explanation of the mechanism in its formation. Copyright o 1991 by WA Saunders Company INDEX WORDS: pendicitis.
Appendicoumbilical
fistula;
perforated
ap-
T
HE UMBILICUS sometimes appears as the outlet of fistula due to various causes such as the presence of vestigial embryonic structure derived from the vitelline duct and urachus, inflammatory bowel disease, or the ligation of a small unrecognized omphalocele. We recently had the opportunity to care for a boy who developed a fistula from the appendix to the umbilicus due to perforated appendicitis. We reviewed English and Korean language literature but could not find similar cases. We report this case with a proposed explanation of the mechanism involved in its formation. CASE
REPORT
A previously healthy 5-year-old boy presented with 2-week history of foul odorous and purulent umbilical discharge. Three weeks prior to admission he developed lower abdominal pain and microscopic pyuria and was treated with antibiotics for “cystitis” at a private clinic. On examination, there was a 0.7 x 0.7 cm, pinkish, and granulomatous umbilical mass with a central opening through which a small amount of purulent discharge was expressed. Admission laboratory data included white blood cell count 12,500/
Fig 1. Midline longitudinal sonogram less uchogenic lesion from the umbilicus dome along the midline, rior aspect of the urinary
JournalofPediafricSurgery,
associated with bladder wall.
Vol26,
shows
a long tubular
and
to the urinary bladder (LIB) thickening of posterosupe-
No 12 (December),
1991: pp 14131416
Fig 2.
Fistulogram
through
the umbilical
opening
shows
a fistu-
lous tract down to the bladder dome, which is continuous with the comma-shaped appendix and cecum. The appendix contains a feclalith in its middle third.
mm’, hemoglobin ll.Sg/dL, hematocrit 34.9 vol%, platelet 417,000, and unremarkable urinalysis. Abdominal ultrasonogram scan showed a long tubular and less echogenic lesion at the lower and anterior abdominal wall from the umbilicus to the urinary bladder dome associated with diffuse thickening of posterosuperior aspect of the urinary bladder wall (Fig 1). A no. 6 feeding tube was introduced into the umbilical opening and fistulogram was taken. It demonstrated a fistulous tract down to the bladder dome along the midline and seemed to be continuous with a comma-shaped barium pocket, with a 1.2 x 1.3 cm filling defect and opacification of barium into the cecum (Fig 2). It was concluded that the boy probably had a patent omphalomesenteric duct emptying into the terminal ileum.
From the Division of Pediatric Surgery and the Departments of Diagnostic Radiology and Anatomic Pathology, Keimyung University Dongsan Medical Center, Taegu, Korea. Address reprint requests to Woo-Hyun Park, MD, Division of Pediatric Surgery, Keimyung University Dongsan Medical Center, 194 Dongsan Dong, Taegu 700-310, Korea. Copyright o 1991 by W.B. Saunders Company 0022-3468191/2612-0020$03.0010 1413
1414
PARK
Fig 4. Microscopic findings of the appendix rative appendicitis (H&E, original magnification
Fig 3. Photograph the tip of the appendix
of the excised specimen. (A); U, umbilicus.
The arrow
indicates
The abdomen was explored through a paramedian musclesplitting skin incision in the right lower quadrant. On exploration, there was a thick, dirty fistulous tract covered by greater omentum along the underside of the parietal peritoneum of the lower anterior abdominal wall down to the dome of the urinary bladder, where it communicated with the tip of the appendix (Fig 3). The appendix contained a 1.2 x 1.3 cm fecalith in its middle third. The entire fistulous tract and the appendix was taken out. Microscopic examination of the appendix showed nearly sloughed-off mucosa, covered by necrotic debris with heavy neutrophilic infiltrates extending to the entire wall with periappendiceal abscess forrnation (Fig 4). Sections of the fistulous tract disclosed the fistula lining partly replaced by granulation tissue and covered by necrotic debris with heavy neutrophilic infiltrates; there were no epithelial elements, suggestive of a urachal remnant (Fig 5). Postoperatively, wound abscess was drained. He was discharged 17 days after the operation without any other problems and has done well so far.
showing x40).
acute
ET AL
suppu-
ably caused by application of a clamp or ligature to unrecognized small omphalocele has been very rare, and only six similar cases have been reported to date in the English language literature.2-6 Internal fistulae such as appendicovesical, appendicorectal, or appendicovaginal fistulae caused by sequelae of perforated appendicitis have been reported,7,8 but we failed to find appendicoumbilical fistulae developed as a sequela of perforated appendicitis in the Korean and English language literature. The mechanism of fistula formation to the umbilicus in perforated appendicitis is not clear, but may occur secondary to the formation of an abscess in the vicinity of the urinary bladder. As the abscess increases in size, it might enter a vestigial structure such
DISCUSSION
Although most abdominal wall fistulae occur in the sites of previous laparotomies, the umbilicus is occasionally the site of spontaneous enterocutaneous fistulae because it is the thinnest part of the lower abdominal wall and offers the path of least resistance. Umbilical fistula have several etiologies. In infancy, the presence of vestigial embryonic structures derived from the vitelline duct or urachus appears to be relatively common.’ Appendicoumbilical fistula prob-
Fig 5. Microscopic findings of the fistula shows a fistulous with acute inflammatory infiltrates and granulation tissue original magnification x40).
tract (H&E,
APPENDICOUMBILICAL
1415
FISTULA
as middle umbilical ligament or other urachal remnants thereby finding a path of less resistance. The cystitis the boy experienced 3 weeks prior to admission thought to be due to perivesical abscess subsequently developed by perforated appendicitis. Several cases of spontaneous enteroumbilical fistula in Crohn’s disease also have been reported.“” The
mechanism of fistula formation in Crohn’s disease seems to be similar to the present case. Treatment for appendicoumbilical fistula secondary to perforated appendicitis should be complete en-bloc excision of the fistula tract and the appendix, and drainage if there is any residual abscess.
REFERENCES 1. Kittle CE, Jenkins HP, Dragstedt LR: Patent omphalomesenteric duct. Arch Surg 54:10-36, 1947 2. Crymble PT: Acase of persistent vitelline duct attached to the vermiform appendix. Br J Surg 19:340-305,1922 3. Singleton A0 Jr, King WB: Persistent vitelline duct continuous with the appendix. Surgery 29:278-280,195l 4. Eckstein HB: Exomphalos-A review of 100 cases. Br J Surg 50:305-410, 1963 5. Sandborn WD: Appendiceal-umbilical fistula. Surgery 2:461463,1967 6. Kadzombe E, Currie ABM: Neonatal fistula from the appendix to the umbilicus. J Pediatr Surg 23:1059-1060,1988
7. Forbes KA, Rose RJ: Appendicovesical fistula. Ann Surg 160:801-805,1964 8. Maingot R: The Treatment of Acute Appendicitis and of Appendix Abscess (ed 6). East Norwalk, CT, Appleton-CenturyCrofts, 1969, pp 1370-1395 9. Hiley PC, Cohen N, Present DH: Spontaneous umbilical fistula in granulomatous (Crohn’s) disease of the bowel. Gastroenterology 60:103-107,197l 10. Rentz TW, Warden CS, Garcia FJ, et al: Crohn’s disease with spontaneous ileoumbilical and ileovesical fistula. Dig Dis Sci 24:316-318, 1979
Fistula as a Sequela of Perforated Appendicitis
By Woo-Hyun
Park, Soon-Ok
Choi, Seong-Ku Taegu, Korea
Woo, and Sang-Sook
Lee
0 This is the first report, we believe, of a 5-year-old boy who developed appendicoumbilical fistula as a sequela of perforated appendicitis. We discuss a proposed explanation of the mechanism in its formation. Copyright o 1991 by WA Saunders Company INDEX WORDS: pendicitis.
Appendicoumbilical
fistula;
perforated
ap-
T
HE UMBILICUS sometimes appears as the outlet of fistula due to various causes such as the presence of vestigial embryonic structure derived from the vitelline duct and urachus, inflammatory bowel disease, or the ligation of a small unrecognized omphalocele. We recently had the opportunity to care for a boy who developed a fistula from the appendix to the umbilicus due to perforated appendicitis. We reviewed English and Korean language literature but could not find similar cases. We report this case with a proposed explanation of the mechanism involved in its formation. CASE
REPORT
A previously healthy 5-year-old boy presented with 2-week history of foul odorous and purulent umbilical discharge. Three weeks prior to admission he developed lower abdominal pain and microscopic pyuria and was treated with antibiotics for “cystitis” at a private clinic. On examination, there was a 0.7 x 0.7 cm, pinkish, and granulomatous umbilical mass with a central opening through which a small amount of purulent discharge was expressed. Admission laboratory data included white blood cell count 12,500/
Fig 1. Midline longitudinal sonogram less uchogenic lesion from the umbilicus dome along the midline, rior aspect of the urinary
JournalofPediafricSurgery,
associated with bladder wall.
Vol26,
shows
a long tubular
and
to the urinary bladder (LIB) thickening of posterosupe-
No 12 (December),
1991: pp 14131416
Fig 2.
Fistulogram
through
the umbilical
opening
shows
a fistu-
lous tract down to the bladder dome, which is continuous with the comma-shaped appendix and cecum. The appendix contains a feclalith in its middle third.
mm’, hemoglobin ll.Sg/dL, hematocrit 34.9 vol%, platelet 417,000, and unremarkable urinalysis. Abdominal ultrasonogram scan showed a long tubular and less echogenic lesion at the lower and anterior abdominal wall from the umbilicus to the urinary bladder dome associated with diffuse thickening of posterosuperior aspect of the urinary bladder wall (Fig 1). A no. 6 feeding tube was introduced into the umbilical opening and fistulogram was taken. It demonstrated a fistulous tract down to the bladder dome along the midline and seemed to be continuous with a comma-shaped barium pocket, with a 1.2 x 1.3 cm filling defect and opacification of barium into the cecum (Fig 2). It was concluded that the boy probably had a patent omphalomesenteric duct emptying into the terminal ileum.
From the Division of Pediatric Surgery and the Departments of Diagnostic Radiology and Anatomic Pathology, Keimyung University Dongsan Medical Center, Taegu, Korea. Address reprint requests to Woo-Hyun Park, MD, Division of Pediatric Surgery, Keimyung University Dongsan Medical Center, 194 Dongsan Dong, Taegu 700-310, Korea. Copyright o 1991 by W.B. Saunders Company 0022-3468191/2612-0020$03.0010 1413
1414
PARK
Fig 4. Microscopic findings of the appendix rative appendicitis (H&E, original magnification
Fig 3. Photograph the tip of the appendix
of the excised specimen. (A); U, umbilicus.
The arrow
indicates
The abdomen was explored through a paramedian musclesplitting skin incision in the right lower quadrant. On exploration, there was a thick, dirty fistulous tract covered by greater omentum along the underside of the parietal peritoneum of the lower anterior abdominal wall down to the dome of the urinary bladder, where it communicated with the tip of the appendix (Fig 3). The appendix contained a 1.2 x 1.3 cm fecalith in its middle third. The entire fistulous tract and the appendix was taken out. Microscopic examination of the appendix showed nearly sloughed-off mucosa, covered by necrotic debris with heavy neutrophilic infiltrates extending to the entire wall with periappendiceal abscess forrnation (Fig 4). Sections of the fistulous tract disclosed the fistula lining partly replaced by granulation tissue and covered by necrotic debris with heavy neutrophilic infiltrates; there were no epithelial elements, suggestive of a urachal remnant (Fig 5). Postoperatively, wound abscess was drained. He was discharged 17 days after the operation without any other problems and has done well so far.
showing x40).
acute
ET AL
suppu-
ably caused by application of a clamp or ligature to unrecognized small omphalocele has been very rare, and only six similar cases have been reported to date in the English language literature.2-6 Internal fistulae such as appendicovesical, appendicorectal, or appendicovaginal fistulae caused by sequelae of perforated appendicitis have been reported,7,8 but we failed to find appendicoumbilical fistulae developed as a sequela of perforated appendicitis in the Korean and English language literature. The mechanism of fistula formation to the umbilicus in perforated appendicitis is not clear, but may occur secondary to the formation of an abscess in the vicinity of the urinary bladder. As the abscess increases in size, it might enter a vestigial structure such
DISCUSSION
Although most abdominal wall fistulae occur in the sites of previous laparotomies, the umbilicus is occasionally the site of spontaneous enterocutaneous fistulae because it is the thinnest part of the lower abdominal wall and offers the path of least resistance. Umbilical fistula have several etiologies. In infancy, the presence of vestigial embryonic structures derived from the vitelline duct or urachus appears to be relatively common.’ Appendicoumbilical fistula prob-
Fig 5. Microscopic findings of the fistula shows a fistulous with acute inflammatory infiltrates and granulation tissue original magnification x40).
tract (H&E,
APPENDICOUMBILICAL
1415
FISTULA
as middle umbilical ligament or other urachal remnants thereby finding a path of less resistance. The cystitis the boy experienced 3 weeks prior to admission thought to be due to perivesical abscess subsequently developed by perforated appendicitis. Several cases of spontaneous enteroumbilical fistula in Crohn’s disease also have been reported.“” The
mechanism of fistula formation in Crohn’s disease seems to be similar to the present case. Treatment for appendicoumbilical fistula secondary to perforated appendicitis should be complete en-bloc excision of the fistula tract and the appendix, and drainage if there is any residual abscess.
REFERENCES 1. Kittle CE, Jenkins HP, Dragstedt LR: Patent omphalomesenteric duct. Arch Surg 54:10-36, 1947 2. Crymble PT: Acase of persistent vitelline duct attached to the vermiform appendix. Br J Surg 19:340-305,1922 3. Singleton A0 Jr, King WB: Persistent vitelline duct continuous with the appendix. Surgery 29:278-280,195l 4. Eckstein HB: Exomphalos-A review of 100 cases. Br J Surg 50:305-410, 1963 5. Sandborn WD: Appendiceal-umbilical fistula. Surgery 2:461463,1967 6. Kadzombe E, Currie ABM: Neonatal fistula from the appendix to the umbilicus. J Pediatr Surg 23:1059-1060,1988
7. Forbes KA, Rose RJ: Appendicovesical fistula. Ann Surg 160:801-805,1964 8. Maingot R: The Treatment of Acute Appendicitis and of Appendix Abscess (ed 6). East Norwalk, CT, Appleton-CenturyCrofts, 1969, pp 1370-1395 9. Hiley PC, Cohen N, Present DH: Spontaneous umbilical fistula in granulomatous (Crohn’s) disease of the bowel. Gastroenterology 60:103-107,197l 10. Rentz TW, Warden CS, Garcia FJ, et al: Crohn’s disease with spontaneous ileoumbilical and ileovesical fistula. Dig Dis Sci 24:316-318, 1979
