Authors:
Paul D. Silva, MD Department of Obstetrics and Gynecology, Gundersen Lutheran Medical Center La Crosse, Wis Lauren A. Ross Summer Fellow Department of Medical Research Gundersen Lutheran Medical Foundation La Crosse, Wis
Long-term Management of Adenomyosis in a 15-Year-Old
ABSTRACT
The authors present what they believe to be the first reported case of pediatric adenomyosis. The adenomyosis was successfully controlled after resection and long-term treatment with medroxyprogesterone acetate (DMPA) injectable suspension, and fertility was conserved.
Address for correspondence: Paul D. Silva, MD Department of Obstetrics and Gynecology C03-002 Gundersen Lutheran Medical Center 1900 South Avenue La Crosse, WI 54601 Telephone: (608) 775-2306 Fax: (608) 775-6611 Email: pdsilva@gundluth.org
T
he purpose of this article is to describe the first reported case of pediatric adenomyosis treated by conservative surgery and medroxyprogesterone acetate injectable suspension (DMPA, Depo-Provera Contraceptive Injection, Pfizer, New York) with long-term successful reproductive outcome. Adenomyosis is a clinical condition characterized by the presence of the endometrial glands and stroma within the myometrium. Cystic areas containing old blood may often be seen grossly within the myometrium. Dysmenorrhea, menorrhagia, and an enlarged uterus characterize adenomyosis.1,2 The depth of penetration of the myometrium has been used to assess the severity.1,2 Adenomyosis is most commonly reported in middle-aged women,3 and cases in young women are extremely rare. Hysterectomy is the most common treatment for adenomyosis when childbearing has been completed.4,5 In women who wish to conserve fertility, alternative treatments have been used, including gonadotropin-releasing hormone agonists, contraceptives, and conservative surgeries.4 To our knowledge, and after a Medline search from 1966 to 2004, we present what is thought to be the first documented case of pediatric adenomyosis.
CASE REPORT
A 15-year-old virginal female with normal monthly menses presented with an episode of acute lower abdominal pain. She underwent a laparotomy under the care of a general surgeon, with removal of a normal appendix. While in surgery she was noted to have a ruptured ovarian cyst, which was thought to be
contributing to her pain. The surgeon also noted an intrauterine mass on the right side of the uterus. He found no evidence of a bicornuate or double uterus. Postoperatively, she was referred for gynecologic evaluation. A pelvic examination was performed the following month in which the external genitalia, vagina, and cervix appeared within normal limits. The uterus and adnexa did not palpate as enlarged or tender. A pelvic ultrasound revealed a right intramural mass measuring 28x29 mm, with a central cystic portion measuring 8x9 mm. At that time it was decided to monitor the mass ultrasonographically to check for growth (Figure 1). Two months later the patient presented with severe right pelvic pain. By examination the uterus was mildly enlarged. An ultrasound revealed that the mass in the uterine wall had increased in size to 50x34 mm and displayed 2 cystic areas (Figure 2). Due to the pain, growth of the mass, and its atypical ultrasound appearance, exploratory surgery was recommended. At laparotomy, the uterus was found to have a 5-cm right myometrial mass containing 2 cystic areas. The mass was removed by wedging out the involved section of the uterine wall. After closing the myometrium, the uterus had a normal external conformation. Although the endometrial cavity had not been entered, a deep myometrial resection had been performed. Therefore, a recommendation was made for cesarean section in any future pregnancies, since there was potential weakening of the uterine wall. Pathologic examination of the mass demonstrated extensive adenomyosis with a “chocolate cyst” in the myometrium. After surgery, intermittent DMPA injections were successfully used with
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penetrate into the myometrium. Though hysterectomy is the most common treatment for symptomatic adenomyosis,4 we chose a more conservative route due to the patient’s young age and desire to maintain fertility potential. Our treatment consisted of resection of the adenomyosis and long-term treatment with DMPA. The main differential diagnosis of a uterine mass in the adolescent includes uterine anomalies, interstitial pregnancy, and leiomyoma. Although in our case the adenomyosis was imaged on sonogram, in most cases the diagnosis has not been made until hysterectomy.1,5 However, with improvements in magnetic resonance imaging and ultrasound resolution, the diagnosis is increasingly being made without requiring a hysterectomy specimen. Therefore, in women wishing to avoid hysterectomy, conservative treatments may be attempted.1 Conservative surgical treatment may include resection, laparoscopic myometrial endomyometrial
Figure 1. Sonogram of pelvis demonstrating cystic mass within uterine wall.
good pain control and cessation of menses. Serial ultrasounds showed no recurrence of the mass (Figure 3). Approximately 8 years after the resection of the adenomyosis, the patient stopped receiving DMPA and spontaneously conceived at age 23. However, a miscarriage resulted shortly after an ultrasound diagnosis of a blighted ovum. A few months later the patient once again spontaneously conceived and this time maintained an uncomplicated pregnancy. Nine years after her adenomyosis resection, at the age of 24, the patient gave birth to a healthy, term infant via a planned primary low transverse cesarean section. No defects were noted in the uterine wall at the time of surgery.
COMMENT
Adenomyosis, most commonly found in middle-aged women, is defined by endometrial glands and stroma that
Figure 3. One of a series of sonograms taken since surgical removal of the mass showing no sign of recurrence.
Figure 2. A second sonogram taken 2 months after the first (Figure 1), illustrating growth of the mass within the uterine wall.
electrocoagulation, and myometrial excision.4 In this patient, who desired future fertility, the authors used myometrial excision because the endometrium is not generally damaged and the limits of myometrial damage are clearly defined, allowing for precise repair. Suppression of the endometrial glands of adenomyosis can be obtained by hormonal contraceptives, danazol, and gonadotropin-releasing hormone analogues (GnRH-As),4,1 although ovulation is also inhibited. The pain of adenomyosis can also be symptomatically treated through analgesics or presacral neurectomy.1 Conservative treatments are often only temporizing measures, and many patients will eventually go on to hysterectomy.3,4 We present what we believe to be the first reported case of pediatric adenomyosis. With conservative treatment, 2 desired outcomes were reached. The adenomyosis was successfully controlled after resection and long-term treatment with DMPA. Secondly, fertility was conserved, measured by the spontaneous pregnancy and term birth of a normal, female infant.
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ACKNOWLEDGMENT
The authors thank the Gundersen Lutheran Medical Foundation, La Crosse, Wis, for its support of this project.
REFERENCES
1. Wood C. Surgical and medical treatment of adenomyosis. Hum Reprod Update. 1998;4:323-336. 2. Munro MG. Abnormal uterine bleeding in the reproductive years. Part IImedical management. J Am Assoc Gynecol Laparosc. 2000;7:19-32. 3. Silva PD, Perkins HE, Schauberger CW. Live birth after treatment of severe adenomyosis with a gonadotropin-releasing hormone agonist. Fertil Steril. 1994;61:171-172. 4. Chen FP, Soong YK. The efficacy and complications of laparoscopic presacral neurectomy in pelvic pain. Obstet Gynecol. 1997;90:974-977.
Canoes
Photograph by Kraig Schuster Gundersen Lutheran
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