Uterine Leiomyomas in Adolescents: A Diagnostic and Treatment Dilemma

Abstract

Background: Despite being the most common pelvic tumor in women during the reproductive age, uterine leiomyoma is a rare pathology in adolescents. However, diagnosis and treatment of this benign tumor is more challenging in younger patients because of being unexpected and associated with fertility-related hazards following surgery. Cases: This case series describes 22 adolescents with uterine leiomyomas and their variable clinical presentations; among these patients, uterine size ranged from 14 weeks and 36 weeks of gestation. One of these patients was eventually diagnosed with leiomyosarcoma. Management of these cases was individualized depending on their presentations and operative indications. The patients underwent appropriate surgical procedures. Results: The postoperative course was smooth in these cases, except for 4 women who had transient fever on the first postoperative day; this was resolved quickly. No recurrences were reported at follow-ups. Hysterosalpingography was conducted in 9 women, and 3 showed abnormalities of dye localization or tubal obstruction. Only 1 patient had infertility as a primary complaint. Conclusions: According to this case series, considerations and precautions should be taken when adolescent patients are surgically treated to minimize the risk of fertility impairment or conversion to hysterectomy, which are both catastrophic in this age group. (J GYNECOL SURG 31:357)

Introduction

Uterine leiomyoma is the most common benign tumor affecting women during of reproductive age. This tumor is found in >50% of women between 35 and 50 years of age, and the incidence increases further with age.^1,2 The pathogenesis of leiomyoma is thought to be influenced in a complex fashion by certain growth factors, estrogen, and progesterone. This explains the tendency of uterine leiomyomas to develop and grow during the reproductive years and to regress after menopause. Unlike what occurs in adults, leiomyoma in adolescents are quite rare; only a few cases have been reported in the literature.³ Current knowledge of the possible pathogenesis of uterine myomas may not explain the occurrence of leiomyoma fully in the adolescent population.

Uterine leiomyomata in adults most commonly present with menstrual abnormalities. Patients may also complain of pelvic pressure and discomfort, infertility, and obstetric complications. Many women with myomas may also be asymptomatic. Available reports from the literature are not adequate for describing the presentation of uterine myomas in younger patients. However, most girls have presented with masses that were initially mistaken for ovarian cysts.³ While there are universally accepted guidelines for treating uterine leiomyomas in adults that recommend minimally invasive surgical approaches,⁴ a management approach for this condition in the adolescent population is not well-established.

In this study of 22 cases, one goal was to recognize the prevalence of leiomyoma in adolescents and to describe the most common clinical scenarios that can help other clinicians to recognize this rare diagnosis. Another goal was to identify plans for treatment that may be appropriate in this young population.

Case Series

This case series spanned the time period from January 2012 to March 31, 2013. The analysis involved selecting cases from young women who were admitted to the department of obstetrics and gynecology of one of three tertiary care centers: the Qena University Hospital; the Women's Health Hospital of Assiut University; or the South Egypt Cancer Institute, all in Assiut, Egypt. These women were admitted for gynecologic evaluation, and possible surgical interventions were considered. Women who were younger than 18, who had pelvi-abdominal masses >12 weeks' gestation size consistent with uterine enlargement, who had suspicious presentations (abnormal menstrual bleeding, pelvic pain, or masses), or who had been already diagnosed with uterine or cervical leiomyomas were initially recruited for the analysis.

These women were evaluated thoroughly; for each patient, a history was taken and a full physical and gynecologic examination, including a pelvic ultrasound (PUS), was conducted. Women with inconclusive ultrasound results underwent magnetic resonance imaging (MRI). Infusion sonography (IS) was also indicated in some patients in whom the PUS results were inconclusive, particularly for women complaining of infertility. Parts of the examinations and diagnostic procedures were restricted by patients' virginity. Each patient's nemoglobin (Hb) level was also measured. Eventually, 22 young women <18 years of age were diagnosed with uterine or cervical leiomyomas, which indicated a need for surgical intervention in one of the three tertiary care centers that participated in the study. An institutional review board approved this study.

Consent for participation was obtained from each of the 22 women, and their data were collected on a comprehensive sheet that was designed by the current authors for this study. Data included age at presentation, marital status, major complaint, breast abnormalities, uterine size, sonographic approach, preoperative Hb level, type of surgery, operative time, intraoperative estimated blood loss (EBL), histopathology, and postoperative complications. The women were followed for 6 months after surgery and hysterosalpingography (HSG) were conducted for nonvirgin women to check the impact of surgery on fertility issues. These data were also reported. Statistical analysis was performed, using the SPSS software package, version 13.0 (SPSS, Chicago, IL).

Twenty-two young women with preoperative diagnosis of uterine leiomyoma were included in this multicenter case series. All were <18 years old (average age: 17.77). Six women (27.3%) complained of abdominal swelling only, 6 women had leiomyomas that were incidentally discovered during infertility workups (27.3%), 5 women had irregular uterine bleeding (22.7%), and 5 women complained of pelvic pain and abdominal swelling (22.7%). Uterine size among these patients ranged from 14 weeks to 36 weeks for uterine-body leiomyomas and ranged from 3 cm to 8 cm for cervical polypoid leiomyomas. Leiomyomas were single in 7 cases (31.9%) and multiple in 15 cases (68.1%). The results of breast examinations was negative in all but 1 patient who was diagnosed with fibroadenoma. Preoperative Hb concentration ranged from 6.5 gm/dL to 12 gm/dL (9.09±1.72 gm/dL). Table 1 summarizes the principal data for these patients.

Table 1.

Summary of Clinical Data, Management & Follow-Up of Study Population

Pt. #	Age (in years)	Sexual status	Chief complaint	Mass size	Type of leiomyoma	Hb level	US approach	Histopathology	Postoperative complications	Operative time	Blood loss	Operation	HSG results	Other comments
1	18	Married	Bleeding	3×4 cm	Single cervical polypoid leiomyoma	7.00	TV	Leiomyoma	No	30.00	100	Excision	.	_
2	18	Married	Infertility	14 weeks	Interstitial	9.00	TV	Leiomyoma	Yes	90.00	500	Myomectomy	.	–
3	18	Married	Mass	14 weeks	Interstitial	9.00	TV	Leiomyoma	No	120.00	500	Myomectomy	.	–
4	18	Married	Infertility	14weeks	Interstitial encroaching on the cavity	8.00	TV	Leiomyoma	Yes	90.00	600	Myomectomy	.	–
5	18	Married	Bleeding	24 weeks	Multiple; 1 was SM	8.00	TV	Leiomyoma	.	90.00	450	Myomectomy	.	–
6	18	Married	Infertility	14 weeks	Interstitial	9.00	TA	Leiomyoma	.	90.00	500	Myomectomy	.	–
7	18	Virgin	Mass & pain	24 weeks	Subserous & interstitial	8.00	TA	Leiomyoma	.	90.00	600	Myomectomy	.	–
8	17	Married	Infertility	14 weeks	Interstitial	11.00	TV	Leiomyoma	.	90.00	500	Myomectomy	Abnormal	–
9	18	Married	Mass	14 weeks (10×9 cm)	Interstitial	12.00	TV	Leiomyoma	.	80.00	500	Myomectomy	Normal	Posterior wall myoma with hyaline degeneration
10	18	Married	Mass	14 weeks	Interstitial	10.00	TV	Leiomyoma	.	60.00	600	Myomectomy	Normal	–
11	18	Virgin	Mass	24 weeks	Interstitial & subserous	8.00	TA	Leiomyoma	Yes	120.00	700	Myomectomy	Abnormal	Localized dye
12	18	Virgin	Mass	28 weeks	2 large subserous & 1 intramural	9.00	TA	Leiomyoma	Yes	180.00	600	Myomectomy	.	–
13	18	Married	Bleeding	5×5 cm (SM)	SM	7.00	TV	Leiomyoma	No	120.00	400	Myomectomy	Normal	Hysteroscopic myomectomy
14	17	Virgin	Mass and pain	16 weeks	Interstitial	12.00	TA	Leiomyoma	No	100.00	500	Myomectomy	.	Anterior wall interstitial myoma
15	18	Married	Infertility	–	Interstitial	12.00	TV	Leiomyoma	No	110.00	450	Myomectomy	Normal	–
16	18	Virgin	Bleeding	4×4 cm (SM)	SM	6.50	TA	Leiomyoma	No	100.00	400	Myomectomy	.	Abdominal myomectomy for an SM myoma
17	18	Married	Mass & pain	36 weeks	Multiple intramural & subserous	9.00	TA	Leiomyoma	No	200.00	1500	Subtotal abdominal hysterectomy	.	–
18	18	Married	Infertility	14 weeks	Interstitial	11.00	TV	Leiomyoma	No	90.00	400	Myomectomy	Normal	Anterior wall myoma in unicorunate uterus
19	18	Married	Mass and pain	16 weeks	Interstitial	9.00	TV	Leiomyoma	No	90.00	600	Myomectomy	Normal	–
20	18	Married	Mass	24 weeks	Interstitial & subserous	10.00	TV	Leiomyoma	No	.	400	Myomectomy	Abnormal	–
21	16	Virgin	Bleeding	8×8 cm	Cervical polypoid leiomyoma with ulcerated tip	7.00	TA	Leiomyoma	No	.	150	Excision	.	–
22	17	Virgin	Mass & pain	36 weeks	Interstitial	8.00	TA	Leiomyosarcoma	No	.	600	TAH-BSO	.	–

. denotes not available.

– denotes no additional comments or missing information; weeks denotes “gestation” size; Hb, hemoglobin, US, ultrasound; HSG, hysterosalpingography, TV, transvaginal; SM, submucous, TA, transabdominal; TAH, transabdominal hysterectomy; BSO, bilateral salpingo-oophorectomy.

Management of these patients was individualized according to their presentations and the characteristics of their myomas; 2 women with cervical polypoid leiomyomas had vaginal polypectomy, 18 women underwent myomectomy one of which was achieved hysteroscopically (Fig. 1), and 2 women underwent hysterectomy. Of the two hysterectomies, one was subtotal because of intractable intraoperative bleeding during the myomectomy (Fig. 2) and the other hysterectomy was total with a bilateral salpingoophrectomy (TAH-BSO) for leiomyosarcoma. Operative time ranged from 30 minutes to 180 minutes (96.66±29.90 minutes), and EBL ranged from 100 mL to 700 mL (472.25±145.54 mL) as estimated by the difference between suctioned and irrigated fluids along with the weight of used towels. No intraoperative complications were reported except for the 1 case with intractable bleeding.

FIG. 1.

Infusion sonography of a single submucous fibroid subsequently treated with hysteroscopic myomectomy.

FIG. 2.

Hugely enlarged uterus (36 weeks) with multiple leiomyomata subsequently managed with hysterectomy.

Results

The postoperative course was smooth for these patients, except for 4 women in whom transient fever was reported on the first postoperative day and that was resolved shortly. No recurrences were reported during the follow-up period. HSG was conducted in 9 women, 3 of these women's results showed abnormalities of dye localization or tubal obstruction. Only 1 of the patients had infertility as a primary complaint.

Discussion

Uterine leiomyoma of the uterus is the most common benign neoplasm of the female pelvis and represents the most common indication for hysterectomy.⁵ However, uterine leiomyomas under the age of 20 years are generally rare.³ To the current authors' knowledge, the first case of uterine leiomyoma in an adolescent 13-year-old girl was reported in 1969 by Wisot and colleagues; this case presented with heavy menstrual bleeding and was treated by myomectomy.⁶ Other reports have described a variety of presentations among this young population, including heavy menstruation, pain, and uremia.⁷ Some patients were eventually diagnosed with uterine sarcomas as well. In the current case series, 1 patient was also diagnosed with uterine leiomyosarcoma. Therefore, a uterine mass in a patient of this age group should be taken seriously, because malignancy is always a possibility.

As the current authors concluded from available reports, the clinical presentation of uterine leiomyomas among young women is variable. Unlike what occurs in adults who have these myomas, there was no single prevalent presentation of this uterine tumor in the current case series; bleeding, pain, pelvi-abdominal mass, and infertility were comparable in prevalence. Unfortunately, all included cases were symptomatic and no data about the prevalence of asymptomatic cases could be retrieved from this study.

According to this case series, adolescents with symptomatic uterine leiomyomas mostly presented with pelvi-abdominal masses at examination; the exception was women with cervical polypoid leiomyomas. Further diagnostic work-ups in this case series was similar to that performed in adults.

However, because virginity-related issues may restrict the use of transvaginal ultrasound, MRI provided precise identification and information about the numbers and location of these tumors. It was helpful to differentiate uterine from adnexal masses, which are more prevalent at that young age. The current authors generally recommend MRI at that age, particularly in women with interstitial leiomyomas that are not perfectly demarcated, because localization of these leiomyomas is critical prior to surgery. Caution should be offered for leiomyomas that approach the cavity. Unfortunately, 3 of 9 women had unsatisfactory HSG results after surgery. MRI also helps to identify potentially malignant features before surgery is conducted.

As in adult population, myomectomy is deemed the standard surgery in symptomatic adolescents. However, because fertility issues are more critical among young women, minimally invasive surgeries are indicated whenever possible. In this series, there was 1 reported case with a single symptomatic submucosal leiomyoma (Type 0) that was managed successfully with hysteroscopic myomectomy. The result of the hysteroscopic approach in this patient was favorable and her postoperative HSG revealed patent tubes and normal uterine cavity.

Unfavorable HSG results were reported in 3 women; 2 of them had not complained of infertility prior to surgery. Interestingly, both women complained only of pelvi-abdominal swelling and their uteri were significantly enlarged (24 weeks' gestation size). One of these women had postoperative fever. However, given that the fever was reported on the first postoperative day, in this case, the fever had mostly no clinical significance. Because leiomyomas in these 2 cases were not submucosal in contrast with the single case that was treated hysteroscopically, the size and number of tumors have more impact on fertility outcomes than the types of leiomyomas. Adolescents with huge myomas should be managed cautiously during surgical intervention because these patients are more liable to have poor fertility outcomes.

A single case of hysterectomy was reported for a nonmalignant indication; this patient had intractable bleeding during myomectomy and had a supracervical hysterectomy after all other measures to control her bleeding had failed. This patient had presented with abdominal swelling and pain. The mass had reached 36 weeks' gestation size. During surgery, the surgeons reported no cleavage plan. Properly speaking, preoperative pain and difficult enucleation were a sequence of myomatous degeneration, which may be expected in huge leiomyomas. For this reason, degenerative changes should be addressed carefully during preoperative imaging especially in patient with huge leiomyomas. Surgeons should be prepared for a bloody operation and the procedure should be assigned to highly qualified surgeons. As in the abovementioned case, surgery may end in a hysterectomy, a sequela that is catastrophic for a patient in this this age group.

Because patients were only followed for 6 months, a definite rate of recurrence in adolescents could not be noted or described. In adults, recurrence has been noted in 23% of women with abdominal myomectomy and in 27% who were treated with laparoscopic myomectomy. However, these results were described after 94 months of follow-up, and ∼5% of recurrent cases were reported 6 months after surgery.⁸ Recurrence has also been also reported in adolescents.³ Nevertheless, there were no recurrences among the patients followed in the current case series.

Conclusions

Uterine leiomyomas should be considered in adolescent women with abnormal uterine bleeding, pelvic pain, or abdominal swelling. MRI is indicated especially when transvaginal ultrasound is not feasible. Minimally invasive surgery is recommended whenever possible. However, surgeons should be cautious about the risk of conversion to hysterectomy or fertility impairment. The size of myomas and the presence of degenerative changes may be the most predictive features to be recognized and reported. Malignancy is still a possibility even in this young population.

Footnotes

Disclosure Statement

All authors state that they have no conflicts of interest.

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Uterine Leiomyomas in Adolescents: A Diagnostic and Treatment Dilemma—A Case Series

Abstract

Abstract

Introduction

Case Series

Results

Discussion

Conclusions

Footnotes

Disclosure Statement

References