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Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir
Case Report
www. AJOG.org
Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir Michael J. Worley Jr, MD; Brian M. Slomovitz, MD; Edyta C. Pirog, MD; Thomas A. Caputo, MD; William J. Ledger, MD
W
orldwide, enterobiasis is a common disease and the causative agent is the roundworm, Enterobius vermicularis.1 Extraintestinal disease is rare and most commonly involves the female reproductive tract. Reports of ectopic enterobiasis involving the vulva, vagina, uterus, fallopian tubes, ovaries, and pelvic peritoneum have been published and diagnosis is often incidental.1-5 We report a patient with a history of multiple abdominal surgeries and a continent, Barnett colonic reservoir (ie, no rectum with an anterior abdominal wall ostomy), incidentally found to have invasive enterobiasis within the myometrium, fallopian tube, and ovary on surgical specimen during a supracervical hysterectomy.
C ASE R EPORT A 43-year-old woman (gravida 2, para 1011) with a history of uterine leiomyomata, presented desiring definitive treatment of severe menorrhagia. Her medical history was significant for ulcerative colitis. She had undergone 8 laparotomies. Her first abdominal surgery was an exploratory laparotomy,
From the Departments of Obstetrics and Gynecology (Drs Worley, Caputo, and Ledger) and Pathology (Dr Pirog), New York Presbyterian Hospital-Cornell University Medical Center, New York, NY; and Department of Obstetrics and Gynecology, Morristown Memorial Hospital, Morristown, NJ (Dr Slomovitz). Received Sept. 8, 2008; revised Nov. 17, 2008; accepted Dec. 1, 2008. Reprints: Brian M. Slomovitz, MD, Division of Gynecologic Oncology, Department of Obstetrics and Gynecology, Women’s Cancer Center, Morristown Memorial Hospital, 100 Madison Ave., Morristown, NJ 07962. [email protected]. 0002-9378/free © 2009 Published by Mosby, Inc. doi: 10.1016/j.ajog.2008.12.001
e6
A 43-year-old woman (gravida 2, para 1011) with a history of uterine leiomyomata and a Barnett colonic reservoir underwent a supracervical hysterectomy. Final pathology revealed Enterobius vermicularis within the myometrium and adnexal vasculature. Infection may have occurred through a modified mode given the presence of a Barnett colonic reservoir and absence of an anus. Key words: ectopic enterobiasis, Enterobius vermicularis, pin worm
which resulted in a total colectomy and the fabrication of an ileostomy. Ten years later, an attempt was made to reverse her ileostomy with the creation of an ileo-anal pouch. This subsequently failed and a Barnett colonic reservoir was created. Twenty years after her first abdominal surgery, she underwent an exploratory laparotomy and left paratubal cystectomy. The remainder of her surgical history included 3 exploratory laparotomies to relieve small bowel obstructions. One month prior to presentation, an unsuccessful uterine artery embolization was conducted. The remainder of her history was unremarkable and on physical examination a 16-cm uterus was present and a 5-cm complex cystic lesion in the left adnexa was seen on computed tomography scan. Her preoperative Papanicolaou test revealed normal findings. A supracervical hysterectomy and left salpingo-oophorectomy were performed. Because of widespread intraabdominal scarring and anatomic distortion, the right ovary and fallopian tube were not visualized. The final pathological report revealed the presence of E vermicularis in subserosal myometrial (Figures 1 and 2), ovarian-hilar (Figure 3), and paratubal vascular channels. Her hospital course was complicated by a partial small bowel obstruction. Resolution was attained with conservative management, and the patient was discharged home on postoperative day 12. An oral dose of mebendazole (100 mg) was administered prior to discharge
American Journal of Obstetrics & Gynecology JUNE 2009
and was repeated 2 weeks later to complete her treatment for E vermicularis.
C OMMENT E vermicularis is a small, threadlike worm that resides primarily in the cecum, appendix, and ascending colon. After fertilization within 1 of the aforementioned locations, the female worm migrates to the anus to deposit eggs. Ova are deposited within a gelatinous material, which in addition to the movement of the migrating worm, contributes to perianal irritation.1,2,6 Hand contamination occurs during scratching. Ingested eggs hatch into larvae within the small bowel and within 2-6 weeks reach maturity, where the cycle continues.
FIGURE 1
Myometrial vascular channel
Low-powered view (original magnification ⫻20) of Enterobius vermicularis infiltrating uterine myometrial vascular channel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
Case Report
www.AJOG.org
FIGURE 2
Myometrial enterobiasis
Low-powered view of Enterobius vermicularis invading uterine myometrial vessel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
FIGURE 3
Ovarian perihilar enterobiasis
Low-powered view of Enterobius vermicularis invading ovarian perihilar vascular channel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
As mentioned previously, the most common site of ectopic enterobiasis is the female reproductive tract. Infestation is thought to occur when either ova are deposited in the genital area by contaminated hands or through the migration of gravid female worms from the perianal region.1,5,6 Worms then travel through the vagina and into the uterus and fallopian tubes and may exit into the pelvis and infect the ovaries, peritoneum, and adjacent structures. Support for this mechanism is in the fact that only female worms have been documented in cervical smears and peritoneal granulomas.1 Our current case would provide a modification on this mode of infection given the presence of a colonic reservoir and absence of an anus and rectum. Rather than perianal hand contamination, transmission of ova may in fact have been facilitated by scratching or manipulation in the area surrounding the stoma. In addition, others have postulated that gravid threadworms have the ability to penetrate the intact bowel and enter the abdominal cavity. This theory is supported by reports of polymicrobial infections of the abdomen involving E vermicularis and enteric bacteria.2,5 However, this mechanism of infection appears less likely in our case after abdominal cultures failed to yield a bacterial organism.
Infection with E vermicularis rarely manifests clinically, making preoperative diagnosis a challenge. Although clinical signs and/or symptoms are often lacking, E vermicularis infestation has been associated with intraabdominal bleeding, peritoneal adhesions, infertility, and tuboovarian abscess.1,7 The treatment of extraintestinal enterobiasis is well documented, and the majority of patients will respond to oral mebendazole.1 f REFERENCES 1. Knuth KR, Fraiz J, Fisch JA, Draper TW. Pinworm infestation of the genital tract. Am Fam Physician 1998;38:127-30. 2. Dundas K, Calder A, Alyusuf R. Enterobius vermicularis thread worm infestation of paraovarian tissue in a woman, who has had a hysterectomy. Br J Obstet Gynecol 1999;106: 605-7. 3. Shnell V, Yandell R, Van Zandt S, Dinh TV. Enterobius vermicularis salpingitis: a distant episode from precipitating appendicitis. Obstet Gynecol 1992;80:553-5. 4. Al-Rufaie HK, Rix GH, Perez Clemente MP, alShawaf T. Pinworms and postmenopausal bleeding. J Clin Pathol 1998;51:401-2. 5. McMahon JN, Connolly CE, Long SV, Meehan FP. Enterobius granulomas of the uterus, ovary and pelvic peritoneum: two case reports. Br J Obstet Gynecol 1984;91:289-90. 6. Kogan J, Alter M, Price H. Bilateral Enterobius vermicularis salpingo-oophoritis. Postgrad Med 1983;73:309-10. 7. Neri A, Tadir Y, Grausbard G, et al. Enterobius (Oxyuris) vermicularis of the pelvic peritoneum–a cause of infertility. Eur J Obstet Gynecol Reprod Biol 1986;23:239-41.
JUNE 2009 American Journal of Obstetrics & Gynecology
e7
www. AJOG.org
Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir Michael J. Worley Jr, MD; Brian M. Slomovitz, MD; Edyta C. Pirog, MD; Thomas A. Caputo, MD; William J. Ledger, MD
W
orldwide, enterobiasis is a common disease and the causative agent is the roundworm, Enterobius vermicularis.1 Extraintestinal disease is rare and most commonly involves the female reproductive tract. Reports of ectopic enterobiasis involving the vulva, vagina, uterus, fallopian tubes, ovaries, and pelvic peritoneum have been published and diagnosis is often incidental.1-5 We report a patient with a history of multiple abdominal surgeries and a continent, Barnett colonic reservoir (ie, no rectum with an anterior abdominal wall ostomy), incidentally found to have invasive enterobiasis within the myometrium, fallopian tube, and ovary on surgical specimen during a supracervical hysterectomy.
C ASE R EPORT A 43-year-old woman (gravida 2, para 1011) with a history of uterine leiomyomata, presented desiring definitive treatment of severe menorrhagia. Her medical history was significant for ulcerative colitis. She had undergone 8 laparotomies. Her first abdominal surgery was an exploratory laparotomy,
From the Departments of Obstetrics and Gynecology (Drs Worley, Caputo, and Ledger) and Pathology (Dr Pirog), New York Presbyterian Hospital-Cornell University Medical Center, New York, NY; and Department of Obstetrics and Gynecology, Morristown Memorial Hospital, Morristown, NJ (Dr Slomovitz). Received Sept. 8, 2008; revised Nov. 17, 2008; accepted Dec. 1, 2008. Reprints: Brian M. Slomovitz, MD, Division of Gynecologic Oncology, Department of Obstetrics and Gynecology, Women’s Cancer Center, Morristown Memorial Hospital, 100 Madison Ave., Morristown, NJ 07962. [email protected]. 0002-9378/free © 2009 Published by Mosby, Inc. doi: 10.1016/j.ajog.2008.12.001
e6
A 43-year-old woman (gravida 2, para 1011) with a history of uterine leiomyomata and a Barnett colonic reservoir underwent a supracervical hysterectomy. Final pathology revealed Enterobius vermicularis within the myometrium and adnexal vasculature. Infection may have occurred through a modified mode given the presence of a Barnett colonic reservoir and absence of an anus. Key words: ectopic enterobiasis, Enterobius vermicularis, pin worm
which resulted in a total colectomy and the fabrication of an ileostomy. Ten years later, an attempt was made to reverse her ileostomy with the creation of an ileo-anal pouch. This subsequently failed and a Barnett colonic reservoir was created. Twenty years after her first abdominal surgery, she underwent an exploratory laparotomy and left paratubal cystectomy. The remainder of her surgical history included 3 exploratory laparotomies to relieve small bowel obstructions. One month prior to presentation, an unsuccessful uterine artery embolization was conducted. The remainder of her history was unremarkable and on physical examination a 16-cm uterus was present and a 5-cm complex cystic lesion in the left adnexa was seen on computed tomography scan. Her preoperative Papanicolaou test revealed normal findings. A supracervical hysterectomy and left salpingo-oophorectomy were performed. Because of widespread intraabdominal scarring and anatomic distortion, the right ovary and fallopian tube were not visualized. The final pathological report revealed the presence of E vermicularis in subserosal myometrial (Figures 1 and 2), ovarian-hilar (Figure 3), and paratubal vascular channels. Her hospital course was complicated by a partial small bowel obstruction. Resolution was attained with conservative management, and the patient was discharged home on postoperative day 12. An oral dose of mebendazole (100 mg) was administered prior to discharge
American Journal of Obstetrics & Gynecology JUNE 2009
and was repeated 2 weeks later to complete her treatment for E vermicularis.
C OMMENT E vermicularis is a small, threadlike worm that resides primarily in the cecum, appendix, and ascending colon. After fertilization within 1 of the aforementioned locations, the female worm migrates to the anus to deposit eggs. Ova are deposited within a gelatinous material, which in addition to the movement of the migrating worm, contributes to perianal irritation.1,2,6 Hand contamination occurs during scratching. Ingested eggs hatch into larvae within the small bowel and within 2-6 weeks reach maturity, where the cycle continues.
FIGURE 1
Myometrial vascular channel
Low-powered view (original magnification ⫻20) of Enterobius vermicularis infiltrating uterine myometrial vascular channel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
Case Report
www.AJOG.org
FIGURE 2
Myometrial enterobiasis
Low-powered view of Enterobius vermicularis invading uterine myometrial vessel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
FIGURE 3
Ovarian perihilar enterobiasis
Low-powered view of Enterobius vermicularis invading ovarian perihilar vascular channel. Worley. Enterobius vermicularis infestation of a hysterectomy specimen in a patient with a colonic reservoir. Am J Obstet Gynecol 2009.
As mentioned previously, the most common site of ectopic enterobiasis is the female reproductive tract. Infestation is thought to occur when either ova are deposited in the genital area by contaminated hands or through the migration of gravid female worms from the perianal region.1,5,6 Worms then travel through the vagina and into the uterus and fallopian tubes and may exit into the pelvis and infect the ovaries, peritoneum, and adjacent structures. Support for this mechanism is in the fact that only female worms have been documented in cervical smears and peritoneal granulomas.1 Our current case would provide a modification on this mode of infection given the presence of a colonic reservoir and absence of an anus and rectum. Rather than perianal hand contamination, transmission of ova may in fact have been facilitated by scratching or manipulation in the area surrounding the stoma. In addition, others have postulated that gravid threadworms have the ability to penetrate the intact bowel and enter the abdominal cavity. This theory is supported by reports of polymicrobial infections of the abdomen involving E vermicularis and enteric bacteria.2,5 However, this mechanism of infection appears less likely in our case after abdominal cultures failed to yield a bacterial organism.
Infection with E vermicularis rarely manifests clinically, making preoperative diagnosis a challenge. Although clinical signs and/or symptoms are often lacking, E vermicularis infestation has been associated with intraabdominal bleeding, peritoneal adhesions, infertility, and tuboovarian abscess.1,7 The treatment of extraintestinal enterobiasis is well documented, and the majority of patients will respond to oral mebendazole.1 f REFERENCES 1. Knuth KR, Fraiz J, Fisch JA, Draper TW. Pinworm infestation of the genital tract. Am Fam Physician 1998;38:127-30. 2. Dundas K, Calder A, Alyusuf R. Enterobius vermicularis thread worm infestation of paraovarian tissue in a woman, who has had a hysterectomy. Br J Obstet Gynecol 1999;106: 605-7. 3. Shnell V, Yandell R, Van Zandt S, Dinh TV. Enterobius vermicularis salpingitis: a distant episode from precipitating appendicitis. Obstet Gynecol 1992;80:553-5. 4. Al-Rufaie HK, Rix GH, Perez Clemente MP, alShawaf T. Pinworms and postmenopausal bleeding. J Clin Pathol 1998;51:401-2. 5. McMahon JN, Connolly CE, Long SV, Meehan FP. Enterobius granulomas of the uterus, ovary and pelvic peritoneum: two case reports. Br J Obstet Gynecol 1984;91:289-90. 6. Kogan J, Alter M, Price H. Bilateral Enterobius vermicularis salpingo-oophoritis. Postgrad Med 1983;73:309-10. 7. Neri A, Tadir Y, Grausbard G, et al. Enterobius (Oxyuris) vermicularis of the pelvic peritoneum–a cause of infertility. Eur J Obstet Gynecol Reprod Biol 1986;23:239-41.
JUNE 2009 American Journal of Obstetrics & Gynecology
e7