- Email: [email protected]
Congenital anal stenosis with anterior sacral meningocele
CASEREPORTS
177
Congenital Anal Stenosis With Anterior Sacral Meningocde By ISSAMJ. SHAKER, M.D., VERNEC. LANIER, M.D., AND RAYMONDA. AMOURY,M.D. Kansas City, MO. This IS-month-old girl was admitted to The Children’s Mercy Hospital because of constipation and recurrent perianal infections with spontaneous drainage and the formation of a fistula-in-ano. She had been constipated since birth with ribbon-like stools and required frequent laxatives and enemas. There was no history of meningitis and neurologic examination was unremarkable. The anal canal was stenotic and rigid and would barely admit the tip of the fifth finger. Roentgenograms demonstrated a hypoplastic sacrum. Contrast material outlined a typical fistula-in-ano from the skin to the lumen of the anorectum. A cystourethrogram and intravenous pyelogram showed bilateral ureteral reflux, nonobstructive hydronephrosis, and a neurogenic bladder. We dilated the anus to a size I3 Hegar dilator, and then palpated a 2 x 3 cm soft cystic mass in the left presacral area. We considered the diagnosis of a presacral teratoma, but a myelogram demonstrated a presacral meningocele (see illustration). We elected not to treat the meningocele, but dilated the childs rectum to a size 18 Hegar and excised a wedge from the fibrous anal ring. The fistula-in-ano was curretted and marsupialized. She was discharged on the 14th hospital ‘day with satisfactory bowel and urinary function. The fistula-in-ano has healed completely and the anal canal remains adequate and pliable. In the 6 months following operation the meningocele has remained stable and there have been no perianal infections and no episodes of meningitis. Ashcraft and Holder bave pointed out the association of anal stenosis with presacral teratomal and Aaronson also reported the association of anal anomalies with anterior sacral meningocele.2 Our case again illustrates the value of the myelogram in differentiating a meningocele from a cystic teratoma in this area. We do not plan to excise the meningocele unless she has further symptoms. REFERENCES 1. Ashcraft, K. W., and Holder, T. M.: Congenital anal stenosis with presacral teratoma. Ann. Surg. 162:1991, 1965. 2. Aaronson, I.: Anterior sacral meningo-
cele, anal canal duplication cyst and covered anus occurring in one family. J. Pediat. Surg. 5559, 1970.
177
Congenital Anal Stenosis With Anterior Sacral Meningocde By ISSAMJ. SHAKER, M.D., VERNEC. LANIER, M.D., AND RAYMONDA. AMOURY,M.D. Kansas City, MO. This IS-month-old girl was admitted to The Children’s Mercy Hospital because of constipation and recurrent perianal infections with spontaneous drainage and the formation of a fistula-in-ano. She had been constipated since birth with ribbon-like stools and required frequent laxatives and enemas. There was no history of meningitis and neurologic examination was unremarkable. The anal canal was stenotic and rigid and would barely admit the tip of the fifth finger. Roentgenograms demonstrated a hypoplastic sacrum. Contrast material outlined a typical fistula-in-ano from the skin to the lumen of the anorectum. A cystourethrogram and intravenous pyelogram showed bilateral ureteral reflux, nonobstructive hydronephrosis, and a neurogenic bladder. We dilated the anus to a size I3 Hegar dilator, and then palpated a 2 x 3 cm soft cystic mass in the left presacral area. We considered the diagnosis of a presacral teratoma, but a myelogram demonstrated a presacral meningocele (see illustration). We elected not to treat the meningocele, but dilated the childs rectum to a size 18 Hegar and excised a wedge from the fibrous anal ring. The fistula-in-ano was curretted and marsupialized. She was discharged on the 14th hospital ‘day with satisfactory bowel and urinary function. The fistula-in-ano has healed completely and the anal canal remains adequate and pliable. In the 6 months following operation the meningocele has remained stable and there have been no perianal infections and no episodes of meningitis. Ashcraft and Holder bave pointed out the association of anal stenosis with presacral teratomal and Aaronson also reported the association of anal anomalies with anterior sacral meningocele.2 Our case again illustrates the value of the myelogram in differentiating a meningocele from a cystic teratoma in this area. We do not plan to excise the meningocele unless she has further symptoms. REFERENCES 1. Ashcraft, K. W., and Holder, T. M.: Congenital anal stenosis with presacral teratoma. Ann. Surg. 162:1991, 1965. 2. Aaronson, I.: Anterior sacral meningo-
cele, anal canal duplication cyst and covered anus occurring in one family. J. Pediat. Surg. 5559, 1970.