Revista Adolescência e Saúde

Revista Oficial do Núcleo de Estudos da Saúde do Adolescente / UERJ

NESA Publicação oficial
ISSN: 2177-5281 (Online)

Vol. 16 nº 3 - Jul/Sep - 2019

Original Article Imprimir 

Páginas 32 a 39


Ovarian lesions in children and adolescents: 10 years histological analysis

Lesiones ováricas en niños y adolescentes: análisis histológico a 10 años

Lesões de ovário em crianças e adolescentes: análise histológica de 10 anos

Autores: Sônia Maria Fior1; Liliane Diefenthaeler Herter2; Noadja Tavares de França3; Luciana Tomkowski Cancian4; Felipe Luzzatto5

1. Medical Residency in Gynecology and Obstetrics, Federal University of Health Sciences of Porto Alegre (UFCSPA). Porto Alegre, RS, Brazil
2. Doctor in Medical Sciences from the Federal University of Rio Grande do Sul (UFRGS). Master in Medical Clinic from the Federal University of Rio Grande do Sul (UFRGS). Teacher at the Federal University of Health Sciences of Porto Alegre (UFCSPA). Porto Alegre, RS, Brazil
3. Medical Residency in Gynecology and Obstetrics, Federal University of Health Sciences of Porto Alegre (UFCSPA). Porto Alegre, RS, Brazil
4. Medical Residency in Cancer Surgery by the National Cancer Institute (INCA). Porto Alegre, RS, Brazil
5. Master in Pathology by the Federal University of Health Sciences of Porto Alegre (UFCSPA). Porto Alegre, RS, Brazil

Sônia Maria Fior
(sonia_fior@hotmail.com)
Universidade Federal de Ciências da Saúde de Porto Alegre (UFCSPA), Ginecologia
Rua Sarmento Leite, 245, Centro Histórico
Porto Alegre, RS, Brasil. CEP: 90050-170

Submitted on 01/25/2019
Approved on 05/10/2019

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Keywords: Ovary; Ovariectomy; Ovarian Neoplasms; Child; Adolescent.
Palabra Clave: Ovario; Ovariotomía; Neoplasias ováricas; Niño; Adolescente
Descritores: Ovário; Ovariectomia; Neoplasias Ovarianas; Criança; Adolescente.

Abstract:
OBJECTIVE: Evaluate the causes of oophorectomies, oophoroplasty and ovarian biopsies performed in children and adolescents.
METHODS: A descriptive and retrospective cross - sectional study was carried out through anatomopathological reports of the Pathology Service of the Santa Casa de Misericórdia Hospital Complex in Porto Alegre. Were included all female patients with ages from 0 to 19 years, who underwent ovarian surgery during the period of January 2006 to December 2016.
RESULTS: 117 patients were included in the study where both ovaries were equally affected (p = 0,926). Surgical specimens ranged from 4 to 3000 grams and from 1,6 to 32 cm in diameter. Most patients (75,2%) underwent oophorectomy and the mean age was 11,78 years (SD ± 5,56 years), the majority being adolescent (70,1%). From 117 cases, 108 were due to benign causes (92,3%) and nine cases (7,7%) due to malignant causes. Of the benign causes, the most common etiology was mature teratoma (26,4%). Of the nine cases of cancer, five (71,4%) had slightly increased tumor markers.
CONCLUSIONS: Despite only 7,7% had malignant disease, 75% underwent oophorectomy. This finding is an alert for surgeons and gynecologists to create more defined criteria when performing oophorectomy, since most of the lesions are benign and this organ plays an important role in their puberty and reproductive function.

Resumen:
OBJETIVO: Avaluar las causas de las ooforectomías, la ooforoplastia y las biopsias de ovario realizadas en niños y adolescentes.
MÉTODOS: Estudio transversal descriptivo y retrospectivo a través de la revisión de informes patológicos del Servicio de Patología del Complejo Hospitalario Santa Casa de Misericordia de Porto Alegre. Se incluyeron pacientes de sexo femenino entre 0 y 19 años que se sometieron a cirugía ovárica durante el período comprendido entre enero de 2006 y diciembre de 2016.
RESULTADOS: se incluyeron un total de 117 pacientes, donde ambos ovarios fueron igualmente afectados (p = 0.926). Las muestras quirúrgicas variaron de 4 a 3000 gramos y de 1,6 a 32 cm de diámetro. La mayoría de los pacientes (75,2%) se sometieron a ooforectomía, y la edad promedio fue de 11,78 años (DE ± 5,56 años), siendo la mayoría adolescentes (70,1%). De los 117 casos, 108 se debieron a causas benignas (92.3%) y nueve casos (7.7%) a causas malignas. De las causas benignas, la etiología más común fue el teratoma maduro (31 casos = 26.4%). De los nueve casos de cáncer, cinco (71.4%) tenían marcadores tumorales ligeramente agrandados.
CONCLUSIONES: aunque solo el 7.7% de los pacientes tenían enfermedad maligna, el 75% se sometió a una ooforectomía. Este hallazgo es una advertencia para que los cirujanos y ginecólogos creen criterios más definidos sobre cuándo realizar la ooforectomía, ya que la mayoría de las lesiones son benignas y este órgano desempeña un papel importante en la pubertad y la función reproductiva.

Resumo:
OBJETIVO: Avaliar as causas das ooforectomias, ooforoplastias e biópsias ovarianas realizadas em crianças e adolescentes.
MÉTODOS: Estudo transversal descritivo e retrospectivo através da revisão de laudos anatomopatológicos do Serviço de Patologia do Complexo Hospitalar Santa Casa de Misericórdia de Porto Alegre. Foram incluídas as pacientes femininas, entre 0 e 19 anos, que realizaram procedimento cirúrgico ovariano durante o período de janeiro de 2006 a dezembro de 2016.
RESULTADOS: Foram incluídas 117 pacientes, onde ambos ovários foram acometidos igualmente (p=0,926). As peças cirúrgicas variaram de 4 a 3000 gramas e de 1,6 a 32 cm de diâmetro. A maioria das pacientes (75,2%) foi submetida à ooforectomia, e a idade média foi de 11,78 anos (DP ± 5,56 anos), sendo a maioria adolescente (70,1%). Dos 117 casos, 108 foram por causas benignas (92,3%) e nove casos (7,7%) por causas malignas. Das causas benignas, a etiologia mais comum foi o teratoma maduro (31 casos = 26,4%). Dos nove casos de câncer, cinco (71,4%) tinham marcadores tumorais levemente aumentados.
CONCLUSÕES: Apesar de apenas 7,7% das pacientes apresentarem doença maligna, 75% realizaram ooforectomia. Esse achado é um alerta para que cirurgiões e ginecologistas criem critérios mais definidos de quando realizar ooforectomia, já que a maioria das lesões é benigna e esse órgão desempenha importante papel na puberdade e função reprodutiva das mesmas.

INTRODUCTION

Ovarian diseases in children and adolescents rarely require surgical treatment, since most lesions are functional and do not require intervention1. The ovary is responsible for puberty, reproductive function and also optimizes peak bone mass2. As such, it is of great importance and, whenever possible, should be preserved. Usually, oophorectomy in young patients is indicated in cases of malignant ovarian tumors or in the annex torsion with necrosis.

Ovarian lesions are represented by neoplastic (benign and malignant tumors) or non-neoplastic (functional cysts3, attachment torsion, endometriomas, and infectious processes) changes. Although ovarian tumor is the most common gynecological tumor in childhood and adolescence, it is still quite rare in women under 21 years4. Fortunately, these tumors, when malignant and properly treated (conservative surgery and combined chemotherapy), have low morbidity and good fertility rate5. The greater availability of ultrasound led to an increase in the number of cyst detection in children, most of them being small and functional (1-3 cm)6.

Ovarian torsion is an uncommon condition in a pediatric patient7, but should be diagnosed early. Initially, lymphatic and venous congestion occurs, which may be followed by arterial ischemia, often culminating in tissue necrosis8. However, it is important to note that the bluish appearance due to venous stasis should not be confused with necrosis, and in these cases the ovary can be preserved.

The aim of this study is to know the causes of oophorectomies, oophoroplasty and ovarian biopsies performed in children and adolescents of the Santa Casa de Misericórdia Hospital Complex of Porto Alegre.


METHODS

A descriptive and retrospective cross-sectional study was conducted with female patients aged 0 to 19 years, who underwent ovarian surgery between January 2006 and December 2016 at the Santa Casa de Misericórdia Hospital Complex of Porto Alegre. Data were obtained through the review of the pathological reports of the Pathology Service of the referred complex and also through the analysis of medical records.

Quantitative variables were described as mean and standard deviation or median and interquartile range, and qualitative variables as absolute and relative frequencies. To assess the association between categorical variables, Pearson's chi-square or Fischer's exact tests were applied. In the comparison of medians the Mann-Whitney or Kruskal-Wallis tests complemented by Dunn were used. The adopted significance level was 5% (p ≤ 0.05) and the analyzes were performed using the SPSS version 21.0 software. The study was duly approved by the Research Ethics Committee of the Santa Casa de Misericórdia Sisterhood of Porto Alegre under Opinion No. 2,297,918.

According to the World Health Organization (WHO) classification, patients were classified as children (0 to 9 years) and adolescents (10 to 19 years).


RESULTS

The sample consisted of 117 patients, with a mean age of 11.78 years (SD ± 5.56 years) operated by different professionals. The age group with the highest prevalence of ovarian surgical procedure was newborns and after six years of age (Figure 1).


Figure 1 - Age distribution of the patients analyzed in the present study. (n = 117 patients)



Regarding the procedure performed, the majority (75.2%) underwent oophorectomy, as many surgeons opt for this procedure when there is adnexal mass, followed by oophoroplasty (24.8%).

There was no difference regarding the affected ovarian side: 59 patients (50.4%) underwent procedure in the left ovary and 58 patients (49.6%) in the right ovary (p = 0.926). Forty-two girls (35.9%) underwent salpingectomy at the same surgical time, 88.1% due to benign causes. Of these, only five (11.9%) patients had pathology compatible with malignant lesions: two cases of sexual cord tumor, an immature teratoma, a lymphoblastic leukemia and a choriocarcinoma.

Thirty patients (25.6%) had ovarian torsion and all reported abdominal pain. There was no association between the age of the patients and the occurrence of ovarian torsion (p = 0.48), nor was there an association between the ovarian volume described in the imaging exams and the diagnosis of ovarian torsion (p = 0.869). Of these 30 patients, 18 (60%) had a history of abdominal pain and vomiting prior to diagnosis. From this same group, 16 (53.4%) patients presented torsion of the annex without underlying lesion where the mean age of the patients was 10 years (SD ± 5.4 years). The remaining 14 (46.6%) patients had torsion with ovarian disease and averaged 12.5 years (SD ± 4.8 years), with the following underlying ovarian lesions: six mature teratomas, three hemorrhagic cysts, two cysts simple serosa, a follicular cyst, a cystoadenofibroma and a fibrotecoma. There was no statistically significant difference between the two groups.

Regarding the pathological results (Table 1), the majority (31/117 = 26.5%) presented mature cystic teratoma, followed by 16 cases (13.6%) of ovarian torsion without underlying lesion, 14 cases (12%). of hemorrhagic luteinic cyst and 11 cases (9.4%) of cystoadenomafibroma. Sixty-four patients (54.7%) had ovarian tumor. Nine of the 117 cases (7.7%) were malignant lesions, being three cases of immature teratoma.




Forty-six pathological findings had the weight described in the pathology report ranging from 4 to 3,000 grams. The lowest weight value was equivalent to a cystoadenofibroma (4g) and the largest to a serous cystoadenoma (3000g).

Seventy-two patients (61.5%) had imaging exams listed in their medical records. Most (46.2%) had ultrasound, followed by tomography (11.1%) and magnetic resonance imaging (8.5%).

Only 39 (33.3%) patients were screened for tumor markers: 38 patients were screened for alpha-alpha-protein protein (AFP), 32 for serum HCG (human chorionic gonadotropin), 17 for LDH (lactic dehydrogenase), 9 for Ca -125 and CEA (carcinoembryonic antigen) and only five patients dosed alkaline phosphatase (AF). Of these patients, none had altered serum HCG, CEA or AF. Three patients had increased Ca-125, one due to mature teratoma and two due to endometrioma, and in one 19-year-old patient Ca-125 was 787 U / mL (reference value = up to 35 U / mL). The highest value found for LDH was 357 U / L (reference value = 100-190 U / L) in a 10-year-old girl with pathology compatible with mature cystic teratoma. The sensitivity of tumor markers ranged from 0 to 75% and the specificity from 23.1 to 100% (Table 2).




DISCUSSION

Currently, the literature has suggested that most ovarian cancers originate in the fallopian tube, as a gradual transformation of the epithelium would have the ability to invade and metastasize within the fallopian tube itself and reach the ovary9. However, it is not yet known whether salpingectomy will lead to reduced ovarian cancer mortality9. In our study, 42 (35.9%) girls underwent salpingectomy at the same surgical time. Of these, only five (11.9%) had cancer and six of these (14.5%) were twisted. While prophylactic salpingectomy could contribute to the reduction of ovarian cancer, on the other hand, it would increase cases of infertility. Thus, there is still a need for further discussion in the literature about the best course of action in cases of removing the ovary in patients without complete offspring: removing or not concomitantly removing the ipsilateral tube.

According to the National Comprehensive Cancer Network (NCCN Guideline 2017)10, patients with tumors limited to one or both ovaries and having the intact capsule (IA and IB respectively) do not necessarily need chemotherapy. However, even if they have ovarian-limited tumor, capsule rupture (as possible in the case of an ovarian biopsy, for example) the stage automatically shifts to HF (tumor limited to one or both ovaries with either of the following: capsule rupture) , ovarian surface tumor or malignant cells with ascites), which will require the need for adjuvant chemotherapy surgery10. Thus, it is necessary to reflect on the advantage and risk of biopsies in ovaries with the possibility of containing a malignant tumor. Many services opt for oophorectomy in these cases, but prior discussion with the patient and family members of the risk / benefit of a biopsy x cystectomy x oophorectomy is required.

Ovarian torsion is a relatively rare event in the pediatric age group.11 It is an emergency condition that affects 4.9 / 100,000 women between 1 and 20 years of age, and most cases are accompanied by an ovarian mass or cyst1,11. However, in childhood, torsion in an ovary with no underlying lesion is more common due to the greater length of the utero-ovarian ligament12. In our study, the age difference between pure ovarian torsions and those with underlying injury were not statistically significant, although we observed a tendency for pure ovarian torsions to affect younger patients (10 years, SD ± 5,4 years). The same was true of the study by Karaman et al.12 where, of the 29 girls with ovarian torsion, eight (27.5%) had normal and average ovaries of 13 years and 21 (72.5%) had mass or adjacent cyst and an average age of 14 years. Also, in the study by Jourjon et al13, which evaluated 65 girls with ovarian torsion, 60.6% twisted with adjacent mass, which shows a higher proportion of torsion in injured ovaries compared with a previous study that showed 27% 14.

In our study, all patients with ovarian torsion (11/307) reported abdominal pain (p <0.001). Of these, the majority (60%) had vomiting, which was similar to the study by Karaman et al.12 where the most common symptom was abdominal pain with the majority of cases presenting with nausea and vomiting. Appelbaum et al15 state that vomiting is commonly associated with torsion in several studies of children and adolescents, agreeing with Jourjon et al13 who found nausea and vomiting as the only statistically significant predictor of torsion.

There was no association between the ovarian volume described in the imaging exams of our study and the diagnosis of ovarian torsion (p = 0.869). This result contrasts with that found by Jourjon et al13 where mass size predicts torsion because it occurred more frequently with lesions ≥ 5cm. The finding of this study is supported by the literature since it was also found that tumors> 5cm in patients older than 1 year are likely to twist, with sensitivity of 83% 16.

Ovarian tumors constitute 1% to 2% of all tumors reported in children and adolescents5. In this same age group, female genital tract tumors are rare, but among them, ovarian cancer is the most frequent representing 1% of all childhood cancers11. In our study, 64 (54.7%) patients had ovarian tumor, where the most common type was the germ cell tumor represented by mature teratoma in 31 cases (26.5%). Germ cell tumor is the most common ovarian neoplasia in childhood and adolescence, with mature cystic teratomas accounting for 55-70% of cases17. Al Jama et al18 evaluated 52 patients, aged 6 to 20 years, diagnosed with ovarian tumor and found 87% of germ cell tumors. In addition, Akakpo et al5 published a study of 706 patients diagnosed with ovarian tumor, 67 cases (9.5%) in patients aged 0 to 19 years. Of these, 53.7% had mature teratomas.

In our sample, of the 64 tumor patients, 45 (70.3%) were adolescents. This was similar to the study by Akakpo and colleagues5 who reported that 48/67 patients (71.6%) were over nine years old. In our study, nine (7.7%) patients had a malignant tumor, a result similar to Piippo et al19, who analyzed the treatment performed in 79 girls under 17 years old who had ovarian mass and found that in seven (8, 8%) of them the pathological examination was compatible with malignant tumor.

The evaluation of tumor markers may increase the accuracy in the differential diagnosis of pediatric ovarian pathology, but their role is still controversial as it presents false positive and false negative results3. Although not validated, tumor markers commonly evaluated in the pediatric population include AFP, HCG, LDH, and inhibin A and B20. When the suspicion is high for malignancy, the use of these markers may facilitate preoperative planning and, in addition, assist in the follow-up of patients with postoperative remission evidence20.

In our study, 39 (33.3%) patients dosed tumor markers and they were elevated in 16 cases (41%): four with malignant ovarian tumor and 12 with benign tumors. Eleven girls with cancer had negative tumor markers. We can compare these results with those of Spinelli et al. who evaluated 130 children and adolescents with ovarian lesions, 110 dosed serum tumor marker levels (Ca-125, AFP, HCG, and CA-19.9). Of these, 18 (16.4%) had elevated levels: five with malignant ovarian tumors and 13 with benign tumors. In the same study, Ca-125 was increased in seven cases of benign lesions: four mature teratomas, two serous cystoadenomas and one fibroma. Already, in our work, we had three cases of elevated Ca-125 and all due to benign causes: two endometrioid cysts and one mature teratoma. Some markers (CEA, HCG, FA, AFP) were very specific (97-100%) but very insensitive (0-20%). On the other hand, in our sample, LDH had a sensitivity of 75% but specificity of 23%. Therefore, markers may assist in decision making, but have a higher value in follow-up when positive.

Although the vast majority of lesions of the patients analyzed were benign (92.3%) in the present study, oophorectomy was performed in 75% of cases, and this data deserves discussion. This finding is a warning for surgeons and gynecologists to discuss more precise criteria for ovarian removal in young patients, as this organ plays an important role in puberty and reproductive function. The risk of cystectomy (capsule rupture in malignant lesion, bleeding) and oophorectomy for benign disease should be assessed with the patient and family prior to surgery.


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