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Inicio Clínica e Investigación en Ginecología y Obstetricia Uterine leiomyoma in adolescents: A case report and a review of the literature
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Vol. 51. Núm. 3.
(julio - septiembre 2024)
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Vol. 51. Núm. 3.
(julio - septiembre 2024)
Case report
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Uterine leiomyoma in adolescents: A case report and a review of the literature
Leiomioma uterino en adolescentes: reporte de un caso y revisión de la literatura
Visitas
1098
M. Dermea,
Autor para correspondencia
martina.derme@uniroma1.it

Corresponding author.
, M. Briantea, G. Massellib
a Department of Maternal and Child Health and Urological Sciences, Umberto I Hospital, Sapienza University, Rome, Italy
b Department of Radiological Sciences, Oncology and Anatomo-Pathology, Umberto I Hospital, Sapienza University, Rome, Italy
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Table 1. Literature report of myomas in adolescents.
Abstract
Introduction

Fibroids are common benign gynecological tumors but a rare finding in adolescents. Although infrequent, some symptomatic cases have been described in literature.

Main symptoms and/or clinical findings

A 16-year-old Caucasian patient came to our attention for abdominal pain and dysmenorrhea appeared two months before. Her gynecological history was characterized by regular menstrual cycles, normal in quantity, with dysmenorrhea. Bimanual pelvic examination revealed an anteverted mobile uterus, no adnexal tenderness or masses. Speculum examination showed a normal cervix. No vaginal bleeding or discharge was observed during the visit.

Main diagnoses

Transabdominal/transvaginal ultrasound demonstrated an anteverted uterus of 71mm×44×48mm, with a heterogeneous myometrial structure and a hypoechoic subserosal-intramural mass (FIGO leiomyoma subclassification system: O-4) localized in the posterior uterine wall, measuring 26mm×19mm×16mm, slightly vascularized at the Color-Doppler (Color Score 2). Magnetic resonance imaging confirmed the ultrasound diagnosis.

Therapeutic interventions and outcomes

Considering pelvic mass dimension and the patient age, a “wait and see” approach was chosen and the patient was re-evaluated a month and three months after the first ultrasound. The second and the third transvaginal ultrasound exam showed an unchanged picture.

Conclusion

The management of leiomyoma in young patients should be targeted to dimension and symptoms of the mass. When facing myomas of small dimension, paucisymptomatic or asymptomatic, with no signs of malignity, we suggest an expectant management.

Keywords:
Uterine myoma
Adolescent
Ultrasound
Dysmenorrhea
Abdominal pain
Resumen
Introducción

Los fibromas son tumores ginecológicos benignos comunes, pero son un hallazgo raro en los adolescentes. Aunque son poco frecuentes, en la literatura se han descrito algunos casos sintomáticos.

Principales síntomas y/o hallazgos clínicos

Paciente caucásica de 16 años que acudió a nuestra consulta por dolor abdominal y dismenorrea de aparición 2 meses antes. Sus antecedentes ginecológicos se caracterizaron por ciclos menstruales regulares, normales en cantidad, con dismenorrea. El examen pélvico bimanual reveló un útero móvil en anteversión, sin dolor a la palpación ni masas anexiales. El examen con espéculo mostró un cuello uterino normal. Durante la visita no se observó sangrado ni secreción vaginal.

Diagnósticos principales

La ecografía transabdominal/transvaginal demostró un útero antevertido de 71×44×48mm, con una estructura miometrial heterogénea y una masa subseroso-intramural hipoecoica (sistema de subclasificación de leiomiomas FIGO: O-4) localizada en la pared posterior del útero, de 26×19×16mm, ligeramente vascularizado en el Doppler color (Color Score 2). La resonancia magnética confirmó el diagnóstico ecográfico.

Intervenciones terapéuticas y resultados

Teniendo en cuenta la dimensión de la masa pélvica y la edad de la paciente, se optó por un enfoque de «esperar y ver» y la paciente fue reevaluada un mes y 3 meses después de la primera ecografía. El último examen de ultrasonido transvaginal mostró una imagen sin cambios.

Conclusión

El tratamiento del leiomioma en los pacientes jóvenes debe centrarse en el tamaño y los síntomas de la masa. Ante miomas de pequeño tamaño, paucisintomáticos o asintomáticos, sin signos de malignidad, sugerimos un manejo expectante.

Palabras clave:
Mioma uterino
Adolescente
Ecografía
Dismenorrea
Dolor abdominal
Texto completo
Introduction

Uterine leiomyomas, also known as fibroids, are neoformations originating from smooth muscle cells of the myometrium. Fibroids are extremely heterogeneous in their location, dimension and clinical presentation. When symptomatic, myomas can present themselves, depending on their size and location in the uterus, with heavy bleeding or abnormal uterine bleeding (AUB), dysmenorrhea, dyspareunia, bulk symptoms (pelvic pressure, back or abdominal pain), urinary or gastrointestinal symptoms (pollakiuria or constipation).1 Fibroids can also be associated with infertility and other poor obstetrical outcomes (increased risk of preterm labor, cesarean delivery, fetal malpresentation and growth restriction).2,3

Myomas are the most common form of benign tumors of the uterus but notable differences are found in their prevalence and presentation. Fibroids are more frequent, tend to present at a younger age, are greater in number and larger in size in women of African ancestry.4 Other risk factors identified include: nulliparity, hypertension, obesity, late menopause and early menarche, familiarity.5

Considering that fibroid development and growth is strongly influenced by female hormones, leiomyomas mainly interest women during reproductive years and typically regress following menopause.6 Therefore, fibroids are a common finding in women between the ages of 30 and 50 but are extremely rare in adolescents. At 25–30 years the incidence of fibroids is only 0.31 per 1000 women years, but by ages 45–50 the incidence has increased 20-fold to 6.20 per 1000 women years.7 From 1969, only 35 cases in young women have been reported.

In this work we present the case of a 16-year-old patient addressed to the gynecologist for abdominal pain and dysmenorrhea, who was diagnosed with a uterine leiomyoma. In addition, we provide an updated review of the literature, including the management of myomas in adolescent patients.

Case reportInformation of the patient

A 16-year-old Caucasian patient came to our attention for abdominal pain and dysmenorrhea appeared two months before. She had an unremarkable medical, surgical and family history. Her gynecological history was also mute and was characterized by regular menstrual cycles, normal in quantity, with a dysmenorrhea 8 out of 10 on the Visual Analogue Scale.

Clinical findings

Patient vital signs were normal. On examination, Tanner staging was appropriate for age. Her body mass index was 26. Bimanual pelvic examination revealed an anteverted mobile uterus, no adnexal tenderness or masses. Speculum examination showed a normal cervix. No vaginal bleeding or discharge was observed during the visit.

Diagnostic evaluation

Transabdominal/transvaginal ultrasound (Mindray DC-80A, convex ultrasound transducer, 1.3–5.7MHz, China) demonstrated an anteverted uterus of 71mm×44mm×48mm, with a heterogeneous myometrial structure and a hypoechoic subserosal-intramural mass (FIGO leiomyoma subclassification system: O-4) localized in the posterior uterine wall, measuring 26mm×19mm×16mm, slightly vascularized at the Color-Doppler (Color Score 2). The endometrium had a three-layers pattern, synchronized to her menstrual cycle. Ovaries were unremarkable bilaterally. No pelvic fluid was observed. The pelvis MRI (Siemens AVANTO 1.5T) showed an oval mass on the upper left wall of the uterine myometrium (axial dimensions 25mm×15mm), with a low to intermediate signal intensity compared with the normal myometrium in the T1-weighted sequences acquired on axial and sagittal planes and an intermediate to high signal intensity compared with the normal myometrium in the T2-weighted sequences acquired on an axial and sagittal planes, compatible with an hypercellular variant of an uterine leiomyoma. The post-contrast administration sequences acquired during arterial and venous phases showed an homogenous enhancement similar to the myometrium which is consistent with non-degenerated fibroids; the finding was confirmed with DWI sequences (b1000) and ADC mapping in which there was no evidence of significant restriction in the diffusion pattern inside the fibroid mass.

Therapeutic intervention

Considering pelvic mass dimension, a “wait and see” approach was chosen and the patient was re-evaluated a month after and three months after the first ultrasound. In this period, in order to alleviate patient's symptoms (abdominal pain and dysmenorrea), we prescribed paracetamol 1000mg orally twice a day during menstruation.

Monitoring and results

The second and the third transvaginal ultrasound exam (GE Voluson S8, micro-convex transducer, 4–10MHz, South Korea), a month and three months after the first exam respectively, showed an unchanged picture (Fig. 1A and B): a hypoechoic subserosal-intramural mass (FIGO leiomyoma subclassification system: O-4) localized in the posterior uterine wall, measuring 23mm×19mm×13mm, slightly vascularized at the Color-Doppler (Color Score 2). 3-dimensional (3-D) ultrasound revealed a normal uterine cavity.

Figure 1.

(A and B). Transvaginal ultrasound described a hypoechoic subserosal-intramural mass (FIGO leiomyoma subclassification system: O-4) localized in the posterior uterine wall, measuring 23mm×19mm×13mm, slightly vascularized at the Color-Doppler (Color Score 2).

(0.03MB).
Discussion

In this report we add another case of symptomatic uterine fibroid in an adolescent patient to the very few described in the last 53 years (Table 1).

Table 1.

Literature report of myomas in adolescents.

Authors, reference  Year of publication  Age  Ethnicity  Clinical manifestation  Myoma size (cm)  Evaluation  Management  Outcome 
Wisot et al.  1969  15  Caucasian  AUB*  12×7×Abdominal flat plate  Abdominal myomectomy  No evidence of disease at 6 months 
Augensen S  1981  15  Not specified  AUB, pelvic mass, urinary retention  10  Pelvic ultrasound  Abdominal myomectomy  No evidence of disease at 1 year 
De Rooy et al.  1986  15  Not specified  Abdominal pain and mass, hemorrhagic shock  16.5×11×8.5  Pelvic ultrasound  Abdominal myomectomy  No evidence of disease at 5 years 
Horejsi et al.  1988  15  Not specified  Not specified  Unknown  Pelvic ultrasoundPelvic arteriographyCT scan  Abdominal hysterectomy+BSO  Not specified 
Heimer et al.  1991  15  Not specified  Abdominal pain and fatigue  20×12  Pelvic ultrasoundCT scan  Abdominal myomectomy  No evidence of disease at 6 months 
Morad et al.  1993  15  Saudi Arabian  AUB, abdominal mass  Pelvic ultrasound  Abdominal myomectomy  Pregnant after 8 months, deliver by cesarean delivery 
Fields et al.199616  Hispanic  22 weeks pregnant, periumbilical pain  Unknown  Pelvic ultrasound  Expectant  Not specified 
20  Hispanic  Pregnancy with fetal distress/amnionitis  6–7  Pelvic ultrasound  Expectant  Not specified 
19  Hispanic  Increased abdominal girth, dysmenorrhea, back pain  790Pelvic ultrasoundCT scan  Myomectomy  Not specified 
19  Hispanic  Pregnancy with labor pain  Not specified  Pelvic ultrasound  Expectant  Not specified 
21  Hispanic  Vaginal discharge and abdominal pain  11×4×Pelvic ultrasound  Expectant  Not specified 
Nguyen-Duc et al.  2003  15  Not specified  AUB, abdominal pain  11×13  TA ultrasound§  Abdominal myomectomy  No evidence of the disease at 6 months 
Bekker et al.  2004  15  African American  AUB, abdominal pain  26×21×Pelvic ultrasound  Abdominal myomectomy  Not specified 
Grapsa et al.  2006  16  Not specified  AUB, abdominal pain  30×23×10  Pelvic ultrasoundCT scan  Abdominal myomectomy  No evidence of disease at 6 years 
Diesen et al.  2008  14  African American  Dysmenorrhea, abdominal distension  15×15×10  Pelvic ultrasoundMRI||  Abdominal myomectomy  No evidence of disease at 3 months 
Perkins et al.  2009  17  African American  Pelvic mass  10  Pelvic ultrasoundMRI  Abdominal myomectomy  Pregnant after unspecified time, delivered at term by cesarean delivery 
Karim et al.  2010  16  Not specified  Pelvic mass, increased abdominal volume  25×15×10  TV#/TA ultrasound  Abdominal myomectomy  Not specified 
Tsili et al.  2010  16  Not specified  Abdominal pain and distension  Multiple, largest 13  TV/TA ultrasoundMRI  Laparoscopic myomectomy  Recurrence of multiple myomas at 2 years 
Khorrami et al.  2011  17  Caucasian  AUB, refractory anemia  2.2×4.2×1.7  TV ultrasound  Hysteroscopic myomectomy  No evidence of the disease at unspecified time 
Taskin et al.  2011  16  Not specified  Pelvic mass protruding through vagina  4×TA ultrasound  Hysteroscopic myomectomy  No evidence of the disease at 9 months 
Wright et al.  2011  14  Caucasian  AUB, abdominal pain, increased abdominal volume  16  Pelvic ultrasoundMRI  Abdominal myomectomy  Asymptomatic 3cm myoma at 1 year follow-up 
Naiditch et al.  2011  15  Not specified  Pelvic mass, abdominal pain, associated ovarian teratoma  TV ultrasoundMRI  Abdominal myomectomy and teratoma resection  Not specified 
Pérez-Colon et al.  2011  15  Not specified  AUB  Not specified  Not specified  Not specified 
Maggiore et al.  2013  14  Caucasian  AUB, abdominal and back pain  13×10×10  TA ultrasoundMRI  Abdominal myomectomy  No evidence of the disease at 6 months 
Kayadibi et al.  2014  15  Not specified  Pelvic mass, increased abdominal volume  17×20×16  Pelvic ultrasoundMRI  Abdominal myomectomy  No evidence of the disease at 6 months 
Salehi et al.  2016  15  Caucasian  Pelvic pain  17.5×14.8×4.5  Pelvic ultrasoundCT scan  Laparoscopic myomectomy  Not specified 
Giannella et al.  2016  18  Black  Dyspareunia  TV ultrasoundCT scan  Laparoscopic myomectomy  No evidence of disease at 8 months 
Zigman et al.  2018  16  Not specified  Vaginal discharge  MRI  Vaginal myomectomy  Not specified 
Morita et al.  2019  13  Not specified  Abdominal distension  11  TAultrasoundMRI  Laparoscopic myomectomy  No evidence of disease at 18 months 
Vaz et al.  2019  16  Hispanic  Abdominal pain and distension  15×10×12  TV ultrasoundCT scanMRI  Abdominal myomectomy  Leiomyosarcoma in the benign myomectomy site 
Kumura et al.  2020  12  Asian  AUB  2.9×2.1×2.7  TA ultrasound  Hysteroscopic myomectomy  Not specified 
Murphy et al.  2020  16  Not specified  AUB, anemia, pelvic mass  TA ultrasound  Abdominal myomectomy  No evidence of the disease at 5 months 
Gong et al.  2021  19  Not specified  Pelvic pain  962  TA ultrasoundMRI  Single-incision laparoscopic myomectomy  Lost in follow-up 
Foo et al.  2021  21  Black  Menorrhagia and dysmenorrhoea  9.4×8.3×8.73.7×2.7×3.2  TV ultrasoundMRI  Abdominal myomectomy  Not specifiedFH tumor predisposition syndrome 
Derme et al.  2023  16  Caucasian  Abdominal pain  2.6×1.9×1.6  TV and TA US  Expectant  Ongoing follow-up 
*

Abnormal uterine bleeding.

Computer tomography scan.

Bilateral salpingo-ophorectomy.

§

Transabdominal ultrasound.

||

Magnetic resonance imaging.

#

Transvaginal ultrasound.

The first case was presented by Wisot et al. in 1969. Since then, a total of 35 patients, including the one whom case we describe, were identified. The youngest was aged 12-year-old and the oldest aged 21-year-old (mode 15-year-old). Most of them reported abnormal uterine bleeding, followed by abdominal pain. Other patients described abdominal mass and distention, anemia and dysmenorrhea. All of them, except one, performed at least an ultrasound (transabdominal US or transvaginal US). In 13 cases the diagnostic was completed with a magnetic resonance imaging (MRI) and in 7 cases a computer tomography scan (CT) was performed. Myomectomy was the most common surgical treatment chosen (28 cases), either approached via hysteroscopy (3 cases), laparoscopy (5 cases), or laparotomy (18 cases). One patient underwent a vaginal myomectomy and only one patient underwent a hysterectomy. The expectant management was preferred in 5 patients, of whom 4 were pregnant. The histological examination did not reveal a malignity in any cases of the ones reported. In 14 cases, there was no evidence of the disease in a variable period of follow-up time (3 months–6 years). In 2 cases there was a recurrence of the fibroids, respectively, at the 1 year follow-up and the 2 years follow-up. In one case a leiomyosarcoma was described in the previous benign myomectomy site. In 2 cases a pregnancy followed the leiomyoma diagnosis and the surgical treatment and both patients underwent a cesarean section.

Our case presents the same characteristics of the ones reported in literature, concerning symptoms and methods of diagnostic. Considering the dimension of the fibroid which affected our patient and the absence of any sign of malignity at the ultrasound and MRI performed, an expectant management was preferred, despite the vast majority of the cases described.

Gynecological symptoms in adolescents often recognize a different etiology from the adult population. Abnormal uterine bleeding is typically due to anovulation secondary to the immaturity of the hypothalamic–pituitary–ovarian axis.8 The majority of cases of dysmenorrhea are primary or functional and, when secondary, dysmenorrhea is typically attributed to endometriosis.9 In the adolescent age group, the distinct possibility of a müllerian anomaly must also be considered. Abdominal distension, when due to gynecological disorders, is often reconducted to an ovarian mass. As shown, uterine fibroids can be found in adolescent patients, although rare. For this reason the diagnosis of leiomyoma should be taken into account even in younger population.

The approach to a woman presenting the described symptoms should always include the evaluation of family history (familiarity for gynecological cancers and fibroids) and a gynecological visit. Sonography is the first line imaging exam. When possible, the combined transabdominal and transvaginal approach is preferred. Transvaginal method is generally chosen as it allows better evaluation of patients with retroverted uterus, inadequate bladder distension, significant bowel gas or obese patients. Transabdominal method, however, is superior for the assessment of large and fundal leiomyomas and it is appropriate even for virgo patients. The examinator should provide a detailed description of the position and the diameter of the lesions, in order to facilitate the follow-up of the mass, according to the FIGO classification system. In addition to ultrasound, MRI can improve further characterization of soft tissues and of mass features such as margins, vascularization, necrosis and growth. The differential diagnosis between leiomyomas and leiomyosarcomas remains, however, difficult.

Different options are currently available for the treatment of uterine leiomyomas. Medical strategies include contraceptive steroids, selective progesterone receptor modulators, gonadotropin-releasing hormone (GnRH) agonists and antagonists, aromatase inhibitors. Other surgical and non-surgical approaches include hysterectomy, myomectomy by hysteroscopy, laparotomy or laparoscopy, uterine artery embolization (UAE) and other interventions performed under radiologic or ultrasound.10 Regarding the management of leiomyomas in adolescents, there are no specific guidelines for this age group. Although fertility preservation is crucial, the treatment proposed should always be targeted to patient desire, symptoms and to dimension of the fibroid.

In the cases reported in literature, most of the patients underwent myomectomy, which can be a valid option for large, symptomatic fibroid not responsive to medical therapy. Extensive counseling regarding recurrence after surgical treatment and future obstetrical implications, such as the possible need to perform a cesarean section, should be provided to the patients. When facing myomas of small dimension, paucisymptomatic or asymptomatic, with no signs of malignity, we suggest, however, an expectant management. A strict echographic follow-up is mandatory to monitor increase in dimension or number of fibroid or the appearance of suspicious signs.

In conclusion, uterine leiomyomas are uncommon yet encountered in younger fertile women and their treatment should be targeted.

Ethical disclosuresProtection of human and animal subjects

The authors declare that no experiments were performed on humans or animals for this study.

Confidentiality of data

The authors declare that they have followed the protocols of their work center on the publication of patient data.

Right to privacy and informed consent

The authors declare that no patient data appear in this article.

Patient consent

The authors declare that the protocols of their institutions on the publication of patient data have been followed and the privacy has been respected.

Funding

The authors certify that there is no financial support.

Conflict of interest

The authors certify that there is no conflict of interest with any financial organization regarding the material discussed in the manuscript.

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