Gracilis
Muscle
Transposition for Anal Incontinence Long-Term Follow-Up
Roberta E. Sonnino, Olivier Reinberg, Arib L. Bensoussan, Jean-Martin
in Children:
Laberge, and Hen+ Blanchard
Montreal, Quebec 0 We report a series of 7 patients, aged 6.5 to 19 years (mean, 12.g), who have been treated for uncontrollable fecal incontinence since 1976: 5 had imperforate anus and multiple subsequent operative procedures, 1 had a low myelomeningocele with bisphincteric incontinence, and 1 had a traumatic destruction of the sphincter apparatus. A modified Pickrell procedure was performed, with the gracilis muscle transposed subcutaneously, without constructing a pulley through the median raphe as originally described. All patients were evaluated by anorectal manometry preoperatively and postoperatively. They were followed-up for a period of 0.5 to 12.5 years (mean, 4.4). All patients were continent at follow-up, with a normal defecation pattern and no enemas required. None of the patients had evidence of fibrosis of the muscle or anal canal, and tension in the transposed muscle was maintained. Voluntary contractions remain efficient in all cases. Age was thought to be an important factor: personal motivation and compliance with physiotherapy, essential for a good outcome, is unlikely to be present in the younger child. We conclude that the gracilis sling procedure is an excellent long-term alternative for total fecal incontinence when time and other therapeutic measures have failed. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Fecal incontinence; sition; anorectal manometry.
gracilis muscle transpo-
S
INCE THE ORIGINAL description by Pickrell et al’ of the gracilis muscle transposition for fecal incontinence, few long-term manometric studies in children have been reported. Corman followed-up 14 patients for 5 years or more, but only 1 patient was less than 20 years of age.’ Leguit et al reported 10 cases with 3 patients under 20 years of age and a follow-up of 2 to 17 yeaq3 whereas Scharli reported a long-term follow-up of anomyoplasties in children, but without a precise description of the manometric studies.4 Other series5x6 have included manometric data in follow-up periods of approximately 4 years. We report a long-term follow-up study of 7 children who underwent a gracilis sling procedure with preoperative and postoperative manometric studies. Subjective outcome is compared with the objective manometric data. These parameters reflect the presence or absence of perianal fibrosis and maintenance of proper tension in the transposed muscle. MATERIALS AND METHODS Between 1976 and 1989, 7 patients (4 girls and 3 boys), underwent a gracilis muscle transposition. Ages ranged from 6.5 to 19 years (mean, 12.9). Etiology of the incontinence included unsuccessful repair of high imperforate anus, often after trials of conservative measures and multiple surgical procedures (5 cases), Journa/offediafric
Surgery, Vol26, No 10 (October). 1991: pp 1219-1223
low myelomeningocele (MMC) (1 case). and severe pelvic trauma in 1 patient. Table 1 summarizes the clinical history for all patients.
Case Repotis Case 1. Despite numerous surgical procedures for high imperforate anus, this lo-year-old boy remained totally incontinent of stool. No sphincter activity could be felt on rectal examination. Case 2. A boy, evaluated at age 11 years, had undergone multiple procedures for high imperforate anus, associated with a VATER syndrome. On rectal examination, his anus was patulous, with no appreciable sphincter contraction and extensive perianal fibrosis. Case 3. A 6-month-old girl underwent closure of a MMC (L,-S,). When evaluated at age 12 years, she was totally incontinent of stool and urine, but had no motor deficit in her lower extremities. She performed intermittent urinary catheterization every 4 hours, and digital evacuation of stools, but had persistent, continuous soiling. The anus was patulous and lacked muscle tone. There was no perineal sensation, but rectal filling sensation appeared intact. Case 4. An infant girl underwent repair of a high imperforate anus, associated with a VATER syndrome. At age 12 years she was totally incontinent with an associated rectal mucosal prolapse. On rectal examination, only weak, ineffective contractions of the puborectalis could be felt. Case 5. A 19-year-old girl suffered major pelvic trauma with a left lower extremity avulsion and a large perineal wound. It is not clear whether the entire sphincteric apparatus was destroyed by the initial trauma or by subsequent infection. She was totally incontinent, with a patulous anus. There were no contractions of either the puborectalis or external sphincter muscles. Rectal sensation was preserved. Case 6. A 17-year-old girl sought medical attention for socially embarrassing incontinence, present since pull-through for imperforate anus in infancy. The incontinence had recently worsened during pregnancy and delivery of her first child. Multiple attempts, both surgical and medical, to improve sphincter tone, had failed. On rectal examination, her anus was widely patent, with minimal sphincter contractions. No puborectalis tone could be appreciated. Computed tomography scan confirmed severe atrophy of all muscles of the pelvic floor. Case 7. A 15year-old boy remained totally incontinent after pull-through for imperforate anus, associated with a VATER syndrome. The patient also had congenital left hemiplegia. He had been followed-up carefully for 5 years with conservative measures and biofeedback, but continued to be incontinent despite signifi-
From the Department of Pediatric Surgery, The Montreal Children’s Hospital, and Hapital Sainte-Justine, Montreal, Quebec. Date accepted: June 20, 1990. Address reprint requests to A% L. Bensoussan, MD, Hapita Sainte-Justine, 3175 C&e Sainte-Catherine, Montreal, Quebec H3T I C5, Canada. Copyright o I991 by W. B. Saunders Company 0022-3468/91/2610-0019$03.00/0
1219
SONNINO ET AL
1220
Operative Procedures
Table 1. Preoperative Clinical Evaluation Case
NO.
Age
SEX
Etiology
w
status
Incontinent
M
lmperforate anus
IO
M
lmperforate anus
11
F
Myelomeningocele
Incontinent
6.5
Incontinent
F
lmperforate anus
12
Continuous soiling
F
Trauma
19
Colostomy
F
lmperforate anus
17
Incontinent
M
lmperforate anus
15
Incontinent
cant voluntary contractions of the puborectalis and external sphincters.
Manomettic Studies and Preoperative Preparation Preoperative and postoperative manometry were performed in all patients. No bowel preparation was given and standard manometric technique was followed. The patient is allowed to lie comfortably on an examining table and anorectal pressures are recorded in three different positions: (1) supine with legs extended; (2) supine with legs crossed; and (3) erect with legs crossed. A double balloon catheter is introduced in the anus so that the distal balloon will be located in the anal canal and the proximal balloon in the rectum. Pressures are transmitted by a Statham P 23 DB pressure gauge (sensitivity from -50 to +300 mm Hg; Hato Key, Puerto Rico) to a four-channel Beckman R 511a recorder (Schiller Park, IL). The paper speed is set at 1 mm/s. Manometric evaluation begins by recording the lower level of sensitivity to rectal filling. The rectal balloon is inflated with increasing volumes of air. The value recorded is the lowest balloon volume that the patient is able to perceive repeatedly. Presence of the rectoanal inhibitory reflex may be documented at the same time, as demonstrated by a decrease in pressure at the level of the internal sphincter with inflation of the rectal balloon. This reflex is represented graphically by a relaxation wave. The patient is then encouraged to voluntarily contract the sphincter muscles and pressures are recorded in the different positions. The mean recorded pressure is used for data interpretation. The values recorded preoperatively in these patients are summarized in Table 2. Preoperative electromyographic studies of the gracilis and adductor muscles are also obtained in candidates for a gracilis sling procedure. Isometric voluntary contraction exercises, under the guidance of a physiotherapist, are carried out in the 2 to 3 weeks before surgery, to increase muscle strength.
Table 2. Preoperative and Long-Term Manometric Preoperative Perception Case NO.
Etiology
Evaluation Maximal
Evaluation Last Evaluation
Perception
Maximal
LWd
PWISSUW
LWd
PreSSUW
ImL)
(mm Hd
bnL)
(mm Hg)
Muscle
Transposition for Anal Incontinence Long-Term Follow-Up
Roberta E. Sonnino, Olivier Reinberg, Arib L. Bensoussan, Jean-Martin
in Children:
Laberge, and Hen+ Blanchard
Montreal, Quebec 0 We report a series of 7 patients, aged 6.5 to 19 years (mean, 12.g), who have been treated for uncontrollable fecal incontinence since 1976: 5 had imperforate anus and multiple subsequent operative procedures, 1 had a low myelomeningocele with bisphincteric incontinence, and 1 had a traumatic destruction of the sphincter apparatus. A modified Pickrell procedure was performed, with the gracilis muscle transposed subcutaneously, without constructing a pulley through the median raphe as originally described. All patients were evaluated by anorectal manometry preoperatively and postoperatively. They were followed-up for a period of 0.5 to 12.5 years (mean, 4.4). All patients were continent at follow-up, with a normal defecation pattern and no enemas required. None of the patients had evidence of fibrosis of the muscle or anal canal, and tension in the transposed muscle was maintained. Voluntary contractions remain efficient in all cases. Age was thought to be an important factor: personal motivation and compliance with physiotherapy, essential for a good outcome, is unlikely to be present in the younger child. We conclude that the gracilis sling procedure is an excellent long-term alternative for total fecal incontinence when time and other therapeutic measures have failed. Copyright o 1991 by W.B. Saunders Company INDEX WORDS: Fecal incontinence; sition; anorectal manometry.
gracilis muscle transpo-
S
INCE THE ORIGINAL description by Pickrell et al’ of the gracilis muscle transposition for fecal incontinence, few long-term manometric studies in children have been reported. Corman followed-up 14 patients for 5 years or more, but only 1 patient was less than 20 years of age.’ Leguit et al reported 10 cases with 3 patients under 20 years of age and a follow-up of 2 to 17 yeaq3 whereas Scharli reported a long-term follow-up of anomyoplasties in children, but without a precise description of the manometric studies.4 Other series5x6 have included manometric data in follow-up periods of approximately 4 years. We report a long-term follow-up study of 7 children who underwent a gracilis sling procedure with preoperative and postoperative manometric studies. Subjective outcome is compared with the objective manometric data. These parameters reflect the presence or absence of perianal fibrosis and maintenance of proper tension in the transposed muscle. MATERIALS AND METHODS Between 1976 and 1989, 7 patients (4 girls and 3 boys), underwent a gracilis muscle transposition. Ages ranged from 6.5 to 19 years (mean, 12.9). Etiology of the incontinence included unsuccessful repair of high imperforate anus, often after trials of conservative measures and multiple surgical procedures (5 cases), Journa/offediafric
Surgery, Vol26, No 10 (October). 1991: pp 1219-1223
low myelomeningocele (MMC) (1 case). and severe pelvic trauma in 1 patient. Table 1 summarizes the clinical history for all patients.
Case Repotis Case 1. Despite numerous surgical procedures for high imperforate anus, this lo-year-old boy remained totally incontinent of stool. No sphincter activity could be felt on rectal examination. Case 2. A boy, evaluated at age 11 years, had undergone multiple procedures for high imperforate anus, associated with a VATER syndrome. On rectal examination, his anus was patulous, with no appreciable sphincter contraction and extensive perianal fibrosis. Case 3. A 6-month-old girl underwent closure of a MMC (L,-S,). When evaluated at age 12 years, she was totally incontinent of stool and urine, but had no motor deficit in her lower extremities. She performed intermittent urinary catheterization every 4 hours, and digital evacuation of stools, but had persistent, continuous soiling. The anus was patulous and lacked muscle tone. There was no perineal sensation, but rectal filling sensation appeared intact. Case 4. An infant girl underwent repair of a high imperforate anus, associated with a VATER syndrome. At age 12 years she was totally incontinent with an associated rectal mucosal prolapse. On rectal examination, only weak, ineffective contractions of the puborectalis could be felt. Case 5. A 19-year-old girl suffered major pelvic trauma with a left lower extremity avulsion and a large perineal wound. It is not clear whether the entire sphincteric apparatus was destroyed by the initial trauma or by subsequent infection. She was totally incontinent, with a patulous anus. There were no contractions of either the puborectalis or external sphincter muscles. Rectal sensation was preserved. Case 6. A 17-year-old girl sought medical attention for socially embarrassing incontinence, present since pull-through for imperforate anus in infancy. The incontinence had recently worsened during pregnancy and delivery of her first child. Multiple attempts, both surgical and medical, to improve sphincter tone, had failed. On rectal examination, her anus was widely patent, with minimal sphincter contractions. No puborectalis tone could be appreciated. Computed tomography scan confirmed severe atrophy of all muscles of the pelvic floor. Case 7. A 15year-old boy remained totally incontinent after pull-through for imperforate anus, associated with a VATER syndrome. The patient also had congenital left hemiplegia. He had been followed-up carefully for 5 years with conservative measures and biofeedback, but continued to be incontinent despite signifi-
From the Department of Pediatric Surgery, The Montreal Children’s Hospital, and Hapital Sainte-Justine, Montreal, Quebec. Date accepted: June 20, 1990. Address reprint requests to A% L. Bensoussan, MD, Hapita Sainte-Justine, 3175 C&e Sainte-Catherine, Montreal, Quebec H3T I C5, Canada. Copyright o I991 by W. B. Saunders Company 0022-3468/91/2610-0019$03.00/0
1219
SONNINO ET AL
1220
Operative Procedures
Table 1. Preoperative Clinical Evaluation Case
NO.
Age
SEX
Etiology
w
status
Incontinent
M
lmperforate anus
IO
M
lmperforate anus
11
F
Myelomeningocele
Incontinent
6.5
Incontinent
F
lmperforate anus
12
Continuous soiling
F
Trauma
19
Colostomy
F
lmperforate anus
17
Incontinent
M
lmperforate anus
15
Incontinent
cant voluntary contractions of the puborectalis and external sphincters.
Manomettic Studies and Preoperative Preparation Preoperative and postoperative manometry were performed in all patients. No bowel preparation was given and standard manometric technique was followed. The patient is allowed to lie comfortably on an examining table and anorectal pressures are recorded in three different positions: (1) supine with legs extended; (2) supine with legs crossed; and (3) erect with legs crossed. A double balloon catheter is introduced in the anus so that the distal balloon will be located in the anal canal and the proximal balloon in the rectum. Pressures are transmitted by a Statham P 23 DB pressure gauge (sensitivity from -50 to +300 mm Hg; Hato Key, Puerto Rico) to a four-channel Beckman R 511a recorder (Schiller Park, IL). The paper speed is set at 1 mm/s. Manometric evaluation begins by recording the lower level of sensitivity to rectal filling. The rectal balloon is inflated with increasing volumes of air. The value recorded is the lowest balloon volume that the patient is able to perceive repeatedly. Presence of the rectoanal inhibitory reflex may be documented at the same time, as demonstrated by a decrease in pressure at the level of the internal sphincter with inflation of the rectal balloon. This reflex is represented graphically by a relaxation wave. The patient is then encouraged to voluntarily contract the sphincter muscles and pressures are recorded in the different positions. The mean recorded pressure is used for data interpretation. The values recorded preoperatively in these patients are summarized in Table 2. Preoperative electromyographic studies of the gracilis and adductor muscles are also obtained in candidates for a gracilis sling procedure. Isometric voluntary contraction exercises, under the guidance of a physiotherapist, are carried out in the 2 to 3 weeks before surgery, to increase muscle strength.
Table 2. Preoperative and Long-Term Manometric Preoperative Perception Case NO.
Etiology
Evaluation Maximal
Evaluation Last Evaluation
Perception
Maximal
LWd
PWISSUW
LWd
PreSSUW
ImL)
(mm Hd
bnL)
(mm Hg)
