E n d o m e t r i o m a M a s q u e r a d i n g as an Anorectal Abscess" Report of a Case* DAvE B. SWERDLOW, M.D. Section of Colon and Rectal Surgery, Department of Surgery, Mountainside Hospital, Montclair, New Jersey
T r a m p u z , 20 1962, 7 cases; Binder, a 1965, 1 case; Murray, 12 1959, 1 case; Stingl, 19 1960, 1 case; M c G i v n e y a n d Mazuji, 1~ 1966, 1 case; Beischer,2 1966, I case. P a u l l a n d Tedeschi la reported 15 more cases i n 1971. McElwain 9 has treated two cases i n sisters. A l t h o u g h these 57 cases are from the literature, my obstetric colleagues report that they all t h i n k they have seen cases b u t have n o t reported them.
SEVERE PERIANAL PAIN is a f r e q u e n t complaint. T h e etiology is usually obvious. Some entities, such as i n t e r m u s c u l a r abscess, levator spasm syndrome, pelvic hematoma, a n d herpes, may be more difficult to diagnose. T h i s is the report of a case i n which perineal endometriosis caused severe anorectal pain. Endometriosis is defined as e n d o m e t r i a l tissue i n a b n o r m a l situations. E n d o m e t r i oma is a t u m o r made u p of ectopic endom e t r i a l fragments. It occurs in w o m e n of c h i l d b e a r i n g age. It is c o m m o n l y seen i n the ovaries, uterus, tubes, u t e r i n e ligament, and small a n d large bowel. Other areas i n c l u d e the p e r i t o n e u m , o m e n t u m , bladder, pouch of Douglas, rectovaginal septum, vagina, vulva, cervix, femoral a n d i n g u i n a l canals, umbilicus, a n d a b d o m i n a l wall. O t h e r unusual sites include extrapelvic structures such as lungs a n d extremities. Endometriosis developing i n areas of the p e r i n e u m a n d elsewhere following surgical or obstetric t r a u m a is b e i n g reported with increasing frequency.5, 7, a2-1g, 19 Cheleden, s i n 1967, reported two cases of perineal endometriosis, reviewed the literature, a n d quoted the following cases: Prince a n d Abrams, 14 1957, 26 cases;
R e p o r t of a Case T h e patient, a 27-year-old woman, was first seen on September 19, 1973, with the chief complaint of severe rectal pain. The pain was of increasing severity, of one week's duration. Examination revealed slight swelling anteriorly, with tenderness. Using local infiltration anesthesia, a small amount of clot, but n o pus, was obtained upon incision, and the pain was relieved. No pus was seen over several days of soaking. Ten days later, the wound healed. No swelling or tenderness was present. She was seen on March 8, 1974, with a six-week history of pain in the anterior perianal region. Examination revealed the area to be swollen and tender. No redness was present. Incision again was performed and no pus was obtained. No purulent drainage ensued following the incision, and the wound healed. Ten days later, pain and swelling recurred. On April 24, 1974, an acute fissure developed. The fissure responded to conservative therapy. On April 30, 1974, severe pain recurred, and at this time, two small bluish areas were present in the wound. Biopsy of this area, under local anesthesia, confirmed the diagnosis of endometriosis of the perineum. The patient did have a perineal episiotomy scar in the right anterior quadrant, but this area was definitely in the left anterior quadrant. On June 26, 1974, a
* Received for publication February 20, 1975. Address reprint requests to Dr. Swerdlow: 44 Fairfield St., Montclair, New Jersey 07042. 620 Dis. Col. & Rect. October 1975
Volume 18 Number 7
Volume 18 Number 7
E N D O M E T R I O M A MhMICKING ABSCESS
total a b d o m i n a l hysterectomy with bilateral salpingo-oophorectomy was performed. T h e pain was temporarily relieved. However, about three m o n t h s after surgery, the tenderness again recurred. T h e patient was admitted to the hospital, where excision of a 3-cm mass from the left anterior q u a d r a n t of the p e r i n e u m was accomplished. She has h a d an u n e v e n t f u l postoperative course, and the symptoms have disappeared.
Etiology Many theories have attelnpted to explain endometriosis. Sampson 16 proposed a transplantation theory that viable endometrial cells were transferred to extrauterine locations by retrograde menstruation (direct mechanical transport) or by way of the blood or lymph stream. Gruenwald s proposed that certain embryonic primitive cells have the potential to undergo metaplasia and, with the appropriate environmental circumstances and stimulation, endometrial cells will develop. No one theory seems to explain the etiology. Perhaps, a combination of theories such as that proposed by Steck and Helwig is may be necessary to explain the mechanism of enclometriosis of the perineum. It is interesting to note that in the series of Paull and Tedeschi, 15 cases occurred only after curettage was performed immediately postpartum. During the same period, 13,000 infants were also delivered with episiotomy without development of endometriosis in the mothers. Therapy T h e r e is no general agreement about the nature of therapy to use with endometriosis. If no annoying symptom is present, it is probably best left alone. If a lesion is symptomatic, various hormonal and ablative treatments have been recommended. Pregnancy: Pregnancy e is frequently followed by an involution of the ectopic endometrial tissue similar to the involution that takes place in the uterus. This is an ideal solution in the cases of young women desirous of starting or increasing their families.
621
Hormonal: Within the past 10 years, with the development of more potent progestational compounds, reliable production of a more effective prolonged pseudodecidual change in the areas of endometriosis has been rendered easier and more tolerable to the patient. T h e resulting decidual change is followed by necrosis and atrophy. It is the atrophy that is responsible for the beneficial effect on the disease, and at least temporary inhibition of its further progression. T h e exact biochemical mechanism by which either normal pregnancy or hormonal therapy with estrogens or progesterone exerts this beneficial effect is not precisely known. T h e endometrial atrophy probably results from both pituitary blockage of production of follicle-stimulating hormone and interference with metabolism of the endometrial tissue locally. An initial swelling of endometrial tissue takes place following initiation of hormone therapy, with a possible increase in symptoms. Once treatment is instituted, it is continued for 6 to 12 months. Objective regressions are reporteda to occur in as many as 80 per cent 0[ patients treated. T h e subsequent pregnancy rates have ranged from 85 to 50 per cent in some series. Surgery: Female Organs: T h e distressing symptoms of endometriosis may be alleviated by surgery. Removal of the uterus, with or without the tubes and ovaries, has been performed for this disease. A more conservative approach that preserves hormonal function is desirable. Hysterectomy alone has provided excellent results t, 4, 6 in the treatment of endometriosis, even though some or all ovarian flmction has been preserved. Cashman 4 reported none of 85 patients who underwent hysterectomy alone required additional surgical treatment for their endometriosis. Sheets et al.*7 reported that 39 of 40 patients were relieved of the symptoms due to endometriosis by hysterectomy alone. In the other patient, a symptomatic local implant in a scar in the vaginal vault
SWERDLOW
622
r e s p o n d e d to r a d i u m (800 m g / h o u r ) app l i e d to the area. O v a r i a n f u n c t i o n cont i n u e d unaffected. L o c a l : Cheleden,5 M i n v i e l l e a n d D e L a Cruz, 11 a n d R a m s e y 15 t r e a t e d t h e i r p a t i e n t s w i t h local e x c i s i o n of the p e r i n e a l or peria n a l masses. T h e y r e p o r t e d p r o m p t h e a l i n g a n d r e l i e f of symptoms. T h i s is a less r a d i c a l p r o c e d u r e w i t h c o n c o m i t a n t decreases in m o r b i d i t y a n d m o r t a l i t y . Also f e r t i l i t y is left intact. O t h e r : E n d o m e t r i o s i s is f r e q u e n t l y f o u n d in m a n y areas of the b o d y r a t h e r t h a n o n l y one. F o r this reason, M i n v i e l l e 1I r e c o m m e n d s t h a t h o r m o n a l t h e r a p y be i n s t i t u t e d for six m o n t h s f o l l o w i n g local excision. Sheets et al. 17 t r e a t e d a local r e c u r r e n c e or a c o n c o m i t a n t lesion w i t h r a d i u m i m p l a n t s . Discussion a n d S u m m a r y E n d o m e t r i o s i s of t h e p e r i n e u m m a y cause severe p e r i a n a l p a i n . S u r g e o n s a n d gynecologists s h o u l d be a w a r e of this e n t i t y in the d i f f e r e n t i a l d i a g n o s i s of such p a i n . It m a y o c c u r in o l d scars or n o n t r a u m a t i z e d tissue. I t occurs m o r e f r e q u e n t l y a f t e r curettage. T h e t r e a t m e n t of choice i n sympt o m a t i c lesions is p r o b a b l y local surgical e x c i s i o n r a t h e r t h a n a n a t t e m p t at h o r m o n a l r e g u l a t i o n . H o r m o n a l or r a d i a t i o n t h e r a p y is a v a i l a b l e for s u p p l e m e n t a l t h e r a p y if necessary. References 1. Beecham CT: Changing concepts in the treatment of endometriosis. J Mich Med Soc 59: 104, 1960 2. Beischer NO: Endometriosis of an episiotomy scar cured by pregnancy. Obstet Gynecol 28: 15, 1966
Dis. Col. &Reet. October 1975
3. Binder SS: Endometriosis of the vulva and perineum: Report of a case. Pacific Med Surg 73: 294, 1965 4. Cashman BZ :Hysterectomy with preservation of ovarian tissue in the treatment of endometriosis. Am J Obstet Gynecol 49: 484, 1945 5. Cheleden J: Endometriosis of the perineum: Report of two cases. South Med J 61: 1313, 1968 6. Gruenwald P: Origin of endometriosis from the mesenchyrne of the celomic walls. Am J Obstet Gynecol 44: 470, 1942 7. Kale S, Shuster M, Shangold J: Endometrioma in a cesarean scar: Case report and review of the literature. Am J Obstet Gynecol 111: 596, 1971 8. Kistner RW: Current status of the hormonal treatment of endometriosis. Clin Obstet Gynecol 9: 271, 1966 9. McElwain J: Personal communication to the author 10. McGivney J, Mazuji MK: Endometriosis of episiotomy scar: Case report. Am Surg 32: 469, 1966 11. Minvielle L, De La Cruz JV: Endometriosis of the anal canal: Presentation of a case. Dis Colon Rectum 11: 32, 1968 12. Murray RR: Endometriosis of an episiotomy scar. US Armed Forces Med J 10: 1463, 1959 13. Paull J, Tedeschi LG: Perineal endometriosis at the site of episiotomy scar. Obstet Gynecol 40: 28, 1972 14. Prince LN, Abrams J: Endometriosis of the perineum: Review of the literature and case report. Am J Obstet Gynecol 73: 890, 1957 15. Ramsey WH: Endometrioma involving the perianal tissues: Report of a case. Dis Colon Rectum 14: 366, 1971 16. Sampson JA: Peritoneal endometriosis due to the menstrual dissemination of endometrial tissue into the peritoneal cavity. Ara J Obstet Gynecol 14:422, 1927 17. Sheets JL, Symmonds RE, Banner EA: Conservative surgical management of endometriosis. Obstet Gynecol 23: 625, 1964 18. Steck WD, Helwig EB: Cutaneous endometriosis. Clin Obstet Gynecol 9: 373, 1966 19. Stingl A: Ein seltener Fall yon Endometriose in der Dammnarbe. Klin Med (Wien) 15: 325, 1960 20. Trampuz V: Endometriosis of the perineum: A report of 5 new cases. Am J Obstet Gynecol 84: 1522, 1962
T r a m p u z , 20 1962, 7 cases; Binder, a 1965, 1 case; Murray, 12 1959, 1 case; Stingl, 19 1960, 1 case; M c G i v n e y a n d Mazuji, 1~ 1966, 1 case; Beischer,2 1966, I case. P a u l l a n d Tedeschi la reported 15 more cases i n 1971. McElwain 9 has treated two cases i n sisters. A l t h o u g h these 57 cases are from the literature, my obstetric colleagues report that they all t h i n k they have seen cases b u t have n o t reported them.
SEVERE PERIANAL PAIN is a f r e q u e n t complaint. T h e etiology is usually obvious. Some entities, such as i n t e r m u s c u l a r abscess, levator spasm syndrome, pelvic hematoma, a n d herpes, may be more difficult to diagnose. T h i s is the report of a case i n which perineal endometriosis caused severe anorectal pain. Endometriosis is defined as e n d o m e t r i a l tissue i n a b n o r m a l situations. E n d o m e t r i oma is a t u m o r made u p of ectopic endom e t r i a l fragments. It occurs in w o m e n of c h i l d b e a r i n g age. It is c o m m o n l y seen i n the ovaries, uterus, tubes, u t e r i n e ligament, and small a n d large bowel. Other areas i n c l u d e the p e r i t o n e u m , o m e n t u m , bladder, pouch of Douglas, rectovaginal septum, vagina, vulva, cervix, femoral a n d i n g u i n a l canals, umbilicus, a n d a b d o m i n a l wall. O t h e r unusual sites include extrapelvic structures such as lungs a n d extremities. Endometriosis developing i n areas of the p e r i n e u m a n d elsewhere following surgical or obstetric t r a u m a is b e i n g reported with increasing frequency.5, 7, a2-1g, 19 Cheleden, s i n 1967, reported two cases of perineal endometriosis, reviewed the literature, a n d quoted the following cases: Prince a n d Abrams, 14 1957, 26 cases;
R e p o r t of a Case T h e patient, a 27-year-old woman, was first seen on September 19, 1973, with the chief complaint of severe rectal pain. The pain was of increasing severity, of one week's duration. Examination revealed slight swelling anteriorly, with tenderness. Using local infiltration anesthesia, a small amount of clot, but n o pus, was obtained upon incision, and the pain was relieved. No pus was seen over several days of soaking. Ten days later, the wound healed. No swelling or tenderness was present. She was seen on March 8, 1974, with a six-week history of pain in the anterior perianal region. Examination revealed the area to be swollen and tender. No redness was present. Incision again was performed and no pus was obtained. No purulent drainage ensued following the incision, and the wound healed. Ten days later, pain and swelling recurred. On April 24, 1974, an acute fissure developed. The fissure responded to conservative therapy. On April 30, 1974, severe pain recurred, and at this time, two small bluish areas were present in the wound. Biopsy of this area, under local anesthesia, confirmed the diagnosis of endometriosis of the perineum. The patient did have a perineal episiotomy scar in the right anterior quadrant, but this area was definitely in the left anterior quadrant. On June 26, 1974, a
* Received for publication February 20, 1975. Address reprint requests to Dr. Swerdlow: 44 Fairfield St., Montclair, New Jersey 07042. 620 Dis. Col. & Rect. October 1975
Volume 18 Number 7
Volume 18 Number 7
E N D O M E T R I O M A MhMICKING ABSCESS
total a b d o m i n a l hysterectomy with bilateral salpingo-oophorectomy was performed. T h e pain was temporarily relieved. However, about three m o n t h s after surgery, the tenderness again recurred. T h e patient was admitted to the hospital, where excision of a 3-cm mass from the left anterior q u a d r a n t of the p e r i n e u m was accomplished. She has h a d an u n e v e n t f u l postoperative course, and the symptoms have disappeared.
Etiology Many theories have attelnpted to explain endometriosis. Sampson 16 proposed a transplantation theory that viable endometrial cells were transferred to extrauterine locations by retrograde menstruation (direct mechanical transport) or by way of the blood or lymph stream. Gruenwald s proposed that certain embryonic primitive cells have the potential to undergo metaplasia and, with the appropriate environmental circumstances and stimulation, endometrial cells will develop. No one theory seems to explain the etiology. Perhaps, a combination of theories such as that proposed by Steck and Helwig is may be necessary to explain the mechanism of enclometriosis of the perineum. It is interesting to note that in the series of Paull and Tedeschi, 15 cases occurred only after curettage was performed immediately postpartum. During the same period, 13,000 infants were also delivered with episiotomy without development of endometriosis in the mothers. Therapy T h e r e is no general agreement about the nature of therapy to use with endometriosis. If no annoying symptom is present, it is probably best left alone. If a lesion is symptomatic, various hormonal and ablative treatments have been recommended. Pregnancy: Pregnancy e is frequently followed by an involution of the ectopic endometrial tissue similar to the involution that takes place in the uterus. This is an ideal solution in the cases of young women desirous of starting or increasing their families.
621
Hormonal: Within the past 10 years, with the development of more potent progestational compounds, reliable production of a more effective prolonged pseudodecidual change in the areas of endometriosis has been rendered easier and more tolerable to the patient. T h e resulting decidual change is followed by necrosis and atrophy. It is the atrophy that is responsible for the beneficial effect on the disease, and at least temporary inhibition of its further progression. T h e exact biochemical mechanism by which either normal pregnancy or hormonal therapy with estrogens or progesterone exerts this beneficial effect is not precisely known. T h e endometrial atrophy probably results from both pituitary blockage of production of follicle-stimulating hormone and interference with metabolism of the endometrial tissue locally. An initial swelling of endometrial tissue takes place following initiation of hormone therapy, with a possible increase in symptoms. Once treatment is instituted, it is continued for 6 to 12 months. Objective regressions are reporteda to occur in as many as 80 per cent 0[ patients treated. T h e subsequent pregnancy rates have ranged from 85 to 50 per cent in some series. Surgery: Female Organs: T h e distressing symptoms of endometriosis may be alleviated by surgery. Removal of the uterus, with or without the tubes and ovaries, has been performed for this disease. A more conservative approach that preserves hormonal function is desirable. Hysterectomy alone has provided excellent results t, 4, 6 in the treatment of endometriosis, even though some or all ovarian flmction has been preserved. Cashman 4 reported none of 85 patients who underwent hysterectomy alone required additional surgical treatment for their endometriosis. Sheets et al.*7 reported that 39 of 40 patients were relieved of the symptoms due to endometriosis by hysterectomy alone. In the other patient, a symptomatic local implant in a scar in the vaginal vault
SWERDLOW
622
r e s p o n d e d to r a d i u m (800 m g / h o u r ) app l i e d to the area. O v a r i a n f u n c t i o n cont i n u e d unaffected. L o c a l : Cheleden,5 M i n v i e l l e a n d D e L a Cruz, 11 a n d R a m s e y 15 t r e a t e d t h e i r p a t i e n t s w i t h local e x c i s i o n of the p e r i n e a l or peria n a l masses. T h e y r e p o r t e d p r o m p t h e a l i n g a n d r e l i e f of symptoms. T h i s is a less r a d i c a l p r o c e d u r e w i t h c o n c o m i t a n t decreases in m o r b i d i t y a n d m o r t a l i t y . Also f e r t i l i t y is left intact. O t h e r : E n d o m e t r i o s i s is f r e q u e n t l y f o u n d in m a n y areas of the b o d y r a t h e r t h a n o n l y one. F o r this reason, M i n v i e l l e 1I r e c o m m e n d s t h a t h o r m o n a l t h e r a p y be i n s t i t u t e d for six m o n t h s f o l l o w i n g local excision. Sheets et al. 17 t r e a t e d a local r e c u r r e n c e or a c o n c o m i t a n t lesion w i t h r a d i u m i m p l a n t s . Discussion a n d S u m m a r y E n d o m e t r i o s i s of t h e p e r i n e u m m a y cause severe p e r i a n a l p a i n . S u r g e o n s a n d gynecologists s h o u l d be a w a r e of this e n t i t y in the d i f f e r e n t i a l d i a g n o s i s of such p a i n . It m a y o c c u r in o l d scars or n o n t r a u m a t i z e d tissue. I t occurs m o r e f r e q u e n t l y a f t e r curettage. T h e t r e a t m e n t of choice i n sympt o m a t i c lesions is p r o b a b l y local surgical e x c i s i o n r a t h e r t h a n a n a t t e m p t at h o r m o n a l r e g u l a t i o n . H o r m o n a l or r a d i a t i o n t h e r a p y is a v a i l a b l e for s u p p l e m e n t a l t h e r a p y if necessary. References 1. Beecham CT: Changing concepts in the treatment of endometriosis. J Mich Med Soc 59: 104, 1960 2. Beischer NO: Endometriosis of an episiotomy scar cured by pregnancy. Obstet Gynecol 28: 15, 1966
Dis. Col. &Reet. October 1975
3. Binder SS: Endometriosis of the vulva and perineum: Report of a case. Pacific Med Surg 73: 294, 1965 4. Cashman BZ :Hysterectomy with preservation of ovarian tissue in the treatment of endometriosis. Am J Obstet Gynecol 49: 484, 1945 5. Cheleden J: Endometriosis of the perineum: Report of two cases. South Med J 61: 1313, 1968 6. Gruenwald P: Origin of endometriosis from the mesenchyrne of the celomic walls. Am J Obstet Gynecol 44: 470, 1942 7. Kale S, Shuster M, Shangold J: Endometrioma in a cesarean scar: Case report and review of the literature. Am J Obstet Gynecol 111: 596, 1971 8. Kistner RW: Current status of the hormonal treatment of endometriosis. Clin Obstet Gynecol 9: 271, 1966 9. McElwain J: Personal communication to the author 10. McGivney J, Mazuji MK: Endometriosis of episiotomy scar: Case report. Am Surg 32: 469, 1966 11. Minvielle L, De La Cruz JV: Endometriosis of the anal canal: Presentation of a case. Dis Colon Rectum 11: 32, 1968 12. Murray RR: Endometriosis of an episiotomy scar. US Armed Forces Med J 10: 1463, 1959 13. Paull J, Tedeschi LG: Perineal endometriosis at the site of episiotomy scar. Obstet Gynecol 40: 28, 1972 14. Prince LN, Abrams J: Endometriosis of the perineum: Review of the literature and case report. Am J Obstet Gynecol 73: 890, 1957 15. Ramsey WH: Endometrioma involving the perianal tissues: Report of a case. Dis Colon Rectum 14: 366, 1971 16. Sampson JA: Peritoneal endometriosis due to the menstrual dissemination of endometrial tissue into the peritoneal cavity. Ara J Obstet Gynecol 14:422, 1927 17. Sheets JL, Symmonds RE, Banner EA: Conservative surgical management of endometriosis. Obstet Gynecol 23: 625, 1964 18. Steck WD, Helwig EB: Cutaneous endometriosis. Clin Obstet Gynecol 9: 373, 1966 19. Stingl A: Ein seltener Fall yon Endometriose in der Dammnarbe. Klin Med (Wien) 15: 325, 1960 20. Trampuz V: Endometriosis of the perineum: A report of 5 new cases. Am J Obstet Gynecol 84: 1522, 1962
