Pelvic gliomatosis within foci of endometriosis.

The third reported case of pelvic gliomatosis found within foci of endometriosis is documented 16 years after the removal of a benign cystic teratoma. Grossly at laparoscopy the lesions appear as typical deep fibrotic endometriotic implants.

INTRODUCTION

Glial nodules (gliomatosis) involving the peritoneum or omentum are an unusual finding attributed to rupture or a defect in the capsule of an ovarian teratoma. This case study describes a woman who demonstrated this rare histologic finding in close association with foci of endometriosis. It was discovered 16 years following the removal of a benign cystic teratoma. Two similar cases of peritoneal gliomatosis associated with endometriosis have been reported.[1,2] These women also had ovarian teratomas removed and years later had surgery for endometriosis. Both had peritoneal implants containing mixed foci of gliomatosis and endometriosis.

CASE REPORT

A 30 year old, gravida 0 woman was referred for laparoscopic hysterectomy indicated for persistent pelvic pain secondary to pelvic endometriosis and adhesions. She reported constant, mainly right-sided pelvic and low back pain with radiation to her legs. Intensity was described as ranging from moderate to severe with exacerbation from activity, coitus, and defecation.

Prior surgical history was extensive, beginning at age 12 (1973) with an exploratory laparotomy, left salpingooophorectomy for an ovarian teratoma. In 1985, a right ovarian cyst was drained laparoscopically. Two years later she underwent laparotomy with right ovarian cystectomy, right fimbrioplasty, left salpingostomy, and uterine suspension. In 1989, an episode of pelvic inflammatory disease with ultrasound demonstrating a complex right adnexal mass (10.5 cm x 6.5 cm) precipitated an exploratory laparotomy for presumed tubo-ovarian abscess. This procedure included right oophorectomy, extensive adhesiolysis, right salpingectomy, and appendectomy. Microscopic examination confirmed acute right salpingitis with fibrous serosal adhesions and peritubular endometriosis, right ovarian endometrioma with subacute oophoritis, and acute periappendicitis.

In 1992 she underwent hysteroscopy, laparoscopy via ninth intercostal space insufflation and subcostal incision, extensive small bowel and omental enterolysis, cul-de-sac dissection with excision of deep fibrotic endometriosis from multiple areas on the rectum, left uterosacral ligament, and rec tovaginal septum, bilateral ureterolysis, and hysterectomy using McCall culdoplasty for vaginal cuff suspension (HR). Seven of eight biopsies confirmed endometriosis including the uterine specimen (). The pathologist noted that in all specimens, except that of the left uterosacral ligament, there was a peculiar fibrillary change reminiscent of neuroglia (, ). Consultation with Mayo Clinic confirmed peritoneal gliomatosis associated with endometriosis as the peritoneal implants were strongly positive for glial fibrillary acidic protein (GFAP).

Peritoneal nodule containing endometriotic glands with associated glial implants (Hematoxylin-eosin times 100)

Higher power view of peritoneal nodule with glands and associated glial tissues (Hematoxylin-eosin times 400)

Slides from the patient's previous surgery (1989) were reviewed by the same pathologist. No glial cells were found.

COMMENT

Peritoneal gliomatosis is presumed to be the result of gradual maturation of the neuroepithelial elements that escape from a tear in the capsule of an ovarian teratoma.[3] Albukerk and associates favor coelomic metaplasia as the etiology of the endometriosis in their case and suggest that this developed independent of the peritoneal gliomatosis.[3] Our findings do not confirm or dispute this explanation. However, since this represents the third case of gliomatosis and associated endometriosis, we believe the findings may not be as rare as originally proposed. Interestingly, the rectal, uterosacral ligament, and rectovaginal septal lesions grossly appear as typical deep fibrotic endometriotic implants.

Table 1.

Biopsy location	Endometriosis	GFAP
1. Deep rectal nodule	+	+
2. Very deep rectum	−	+
3. Left uterosacral / left rectum	+	+
4. Left deep rectum	+	+
5. Rectovaginal septum	+	+
6. Left uterosacral ligament / anterior rectum	+	−
7. Major specimen / deep rectum	+	+
8. Uterus	+	+