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ORIGINAL ARTICLE |
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| Year : 2021 | Volume
: 26
| Issue : 5 | Page : 287-293 |
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Surgical management of adnexal masses in the pediatric and adolescent age group: Our experience
Meera Luthra1, Chiranjiv Kumar2
1 Department of Pediatric Surgery, Medanta-the Medicity, Gurugram, Haryana, India
2 Department of Pediatric Surgery, Holyfamily Hospital, New Delhi, India
| Date of Submission | 01-May-2020 |
| Date of Decision | 13-Jun-2020 |
| Date of Acceptance | 27-Nov-2020 |
| Date of Web Publication | 16-Sep-2021 |
Correspondence Address:
Dr. Meera Luthra
D-9, Geetanjali Enclave, New Delhi - 110 017
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jiaps.JIAPS_136_20
 Abstract | | |
Context: Adnexal masses in girls represent a wide pathological spectrum. We performed a retrospective analysis of clinical and diagnostic aspects of adnexal masses in girls.
Aims: To analyze the clinical characteristics and surgical management of adnexal masses in children and adolescents.
Settings and Design: Retrospective observational analysis from 2000 to 2020.
Subjects and Methods: This analysis was performed on 28 children under the age of 17 years with adnexal masses who were managed by us at tertiary care centers of Delhi-NCR. Patients were analyzed based on their age at the time of admission, their presenting complaints, clinical and radiological findings, tumor markers, management, and follow-up.
Results: The age ranged between newborn and 17 years. Abdominal pain, nausea with vomiting, and abdominal lump was the most common presenting complaints. Pelvic ultrasound with or without color Doppler done in all patients and computed tomography, magnetic resonance imaging was performed in seven patients preoperatively. Adnexal masses were unilateral in 26 (92.85%) patients and bilateral in 2 (7.15%) patients. Five babies had antenatal diagnosis of ovarian cysts. Serum alpha fetoprotein (AFP) and beta-human chorionic gonadotropin (b-HCG) were normal in all 19 tested patients. Cancer antigen 125 (CA-125) was raised in three ovarian tumors out of 9 tested patients. Laparoscopy was performed in 23 out of 28 patients (30 ovaries). Ovarian cyst torsion was present in 50% patients. Ovarian cystectomy was done in 10 (33.3%) patients. Six (20%) ovaries were saved by detorsion of the ovary and deroofing of the cyst with or without ovarian biopsy. Oophorectomy was done in five (16.7%) patients. Salpingo-oopherectomy had to be done in seven (23.3%) patients. Histopathology revealed functional nonneoplastic lesions in 18 cases (66.7%), benign neoplastic lesions in 8 cases (29.6%), and malignant neoplastic lesion in 1 case (3.7%).
Conclusions: Adnexal masses in children are generally benign. AFP, b-HCG, and CA-125 in ovarian torsion are within normal limits. Urgent surgery is required if torsion is established. The ovary should be preserved wherever possible.
Keywords: Adnexal masses, adolescent, benign, malignant, pediatric, torsion
How to cite this article:
Luthra M, Kumar C. Surgical management of adnexal masses in the pediatric and adolescent age group: Our experience. J Indian Assoc Pediatr Surg 2021;26:287-93 |
How to cite this URL:
Luthra M, Kumar C. Surgical management of adnexal masses in the pediatric and adolescent age group: Our experience. J Indian Assoc Pediatr Surg [serial online] 2021 [cited 2023 Mar 30];26:287-93. Available from: https://www.jiaps.com/text.asp?2021/26/5/287/326055 |
| Introduction | |  |
Adnexal masses are most commonly observed in adults; they rarely occur in children. The majority of the adnexal masses encountered in children or patients of premenarchal age are nonneoplastic lesions. The clinical signs and symptoms of adnexal masses are usually nonspecific. Early management is necessary to preserve fertility.[1] Ovarian cysts are the most common causes of intra-abdominal cysts detected antenatally by ultrasound (US) studies.[2] Nussbaum et al.[3] classified them as simple or uncomplicated and complex or complicated cysts suggesting ovarian torsion. Controversy remains regarding the best treatment for these cysts ranging from observation, aspiration to surgical exploration in cyst <5 cm. Ovarian tumors in children account for only 1% of childhood malignancies.[4] We share our experience in managing these ovarian masses in pediatric and adolescent age group patients.
| Subjects and Methods | | |
The records of 28 girls under the age of 17 years with adnexal masses who were treated by us (first author was the primary surgeon in all cases) at tertiary referral centers of Delhi-NCR over last 20 years, from 2000 to 2020 were reviewed and retrospectively analyzed based on their age at the time of admission, presenting complaints, clinical and radiological findings, tumor markers, management, and follow-up. All of the patients underwent transabdominal pelvic US with or without color Doppler preoperatively. Additional imaging such as computed tomography (CT), magnetic resonance imaging (MRI) or positron emission tomography (PET) scan was performed as necessary. Ovarian mass characteristics (structure, size, and presence of torsion) were evaluated. An ovarian lesion was described as large when its diameter was 10 cm or more in girls aged between 1 and 18 years and 5 cm or more in new-borns and infants. Such classification was based on the previous experience of other authors, to obtain comparable results.[5],[6] Tumor markers such as serum alpha fetoprotein (AFP), beta-human chorionic gonadotropin (b-HCG), and cancer antigen 125 (CA-125) were tested along with lactate dehydrogenase (LDH) in cases of suspected ovarian tumor. All symptomatic and complicated ovarian cysts were managed by laparoscopy or laparotomy followed by ovarian detorsion, deroofing of ovarian cyst with or without ovarian biopsy, ovarian cystectomy, oophorectomy, or salpingo-oopherectomy. Complex lesions were surgically excised. The patient with malignant lesion – Juvenile granulosa cell tumor (JGCT) underwent complete surgical excision of tumor followed by chemotherapy with bleomycin, etoposide, and cisplatin (BEP regimen). The patient serum AFP levels were monitored. The diagnoses of patients with ovarian tumor were histopathologically confirmed. Simple cysts measuring <5 cm was managed conservatively using US scans performed monthly for 3 months and were excluded from this study.
| Results | | |
- Demographics – the median patient's age at the time of admission in our cohort was 11 years and 9 months (ranged between newborn to 17 years) [Figure 1]. Five neonates were antenatally diagnosed, as confirmed by postnatal US scans
- Symptomatology – Abdominal pain (n = 23), nausea with vomiting (n = 18), and abdominal lump (n = 17) were the most common presenting complaints. Half of the patients were premenarchal. Mean duration of hospital stay in our study was 4.38 days (range: 2–9 days).
Diagnosis
- Imaging – transabdominal pelvic US with or without color Doppler done in all patients and additional imaging such as CT/PET-CT or MRI scan was performed in seven patients preoperatively [Figure 2]. US revealed torsion of ovarian masses in 20 (66.7%) ovaries [Figure 3]. Complex cystic mass were present in 16 cases, simple cystic mass was present in 12 cases, and solid mass were present in two patients. Large ovarian mass was present in 10 (33.3%) cases. Out of these 10 cases, five patients were antenatally diagnosed simple cysts. Of the other five, 2 were functional nonneoplastic lesions, which included 1 hemorrhagic cyst and 1 simple cyst. Two were benign neoplastic lesions. Of the benign neoplastic lesions, one was serous cystadenoma and the other was mucinous cystadenoma. One patient had malignant lesion, i.e., JGCT (malignant sex cord stromal tumor) [Figure 4]
- Tumor markers such as serum AFP and b-HCG were tested in 19 patients and CA-125 were tested in nine patients of ovarian masses along with LDH in cases of suspected ovarian tumor. Levels of AFP and b-HCG were within the normal limits in all 19 patients. Levels of CA-125 were raised in all three cases of diagnosed ovarian tumor which include mucinous cystadenoma, fibroma, and JGCT. LDH was also raised in one case of JGCT.
 | Figure 2: (a) Computed tomography and (b) positron emission tomography – Computed tomography scan image of malignant neoplastic ovarian mass (Juvenile granulosa cell tumor)
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 | Figure 3: Excised specimen of the solid ovarian mass (Juvenile granulosa cell tumor)
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 | Figure 4: Pelvic ultrasound showing right-sided hypoechoic enlarged ovary with no internal vascularity suggestive of torsion
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Management- Surgical, adjuvant therapy, and follow-up
Ovarian lesions were unilateral in 26 (92.85%) patients, out of which 12 (42.85%) were on the right side and 14 (50%) were on the left side. Bilateral ovarian lesions were present in two (7.15%) patients [Figure 5]. Out of these two cases, one girl was operated on both sides in one sitting laparoscopically where she underwent left salpingo-oopherectomy and on right side deroofing/drilling of ovarian cyst was performed. Second girl initially presented with right ovarian torsion with a gangrenous right ovary for which laparoscopic right salpingo-oopherectomy was performed. Left ovary was normal at that time. She recovered uneventfully and 6 months later she came with left ovarian torsion for which laparoscopic detorsion and deroofing of left ovary was done. After this procedure, the ovary became pink. She was well for the next 5 years. After 5 years follow-up, she again presented with recurrent left-sided lower abdomen pain. US pelvis revealed left ovarian torsion with hemorrhagic cyst. Laparotomy and left oophoropexy done with anterior abdominal wall. She recovered uneventfully.
One more patient who had laparoscopic right oophorectomy for right ovarian cyst had again presented 1 year later with pain abdomen and vomiting. US pelvis showed left ovary bulky; however, there was no ovarian torsion; hence, she was managed conservatively. She is now doing well.
Five babies had antenatal diagnosis of ovarian cysts. Four were operated at birth because of the large cyst causing abdominal distension and/or increased respiratory rate. One baby was followed up for 3 months, as it was a simple 5 cm cyst at birth and was operated because the cyst persisted in size and contained debris. This was an auto-amputated cyst.
Laparoscopy was performed in 23 out of 28 patients (30 ovaries). The most common indication for emergency surgery was ovarian cyst torsion [Figure 6]. In one patient of ovarian cyst, laparoscopy was converted to laparotomy due to bleeding from the edges of the cyst. In five patients, laparotomy was done. Three of these five patients had ovarian tumor.
Ovarian cystectomy was done in 10 (33.3%) patients [Figure 7]. Paraovarian cystectomy was done in 1 (3.33%) patient. Six (20%) ovaries were saved by detorsion of the ovary and deroofing of the cyst with or without ovarian biopsy. Oophorectomy had to be done due to gangrene following ovarian torsion in 5 (16.7%) patients. Salpingo-oophorectomy had to be done in total seven patients, of which, four (13.3%) patients had gangrene due to ovarian torsion and three (10%) patients had ovarian tumor. One (3.33%) patient had right ovarian torsion in an irreducible right inguinal hernia for which detorsion of right ovary and right herniotomy was performed. | Figure 6: Laparoscopy showing (a) right ovary with two twists (b) Right ovary twisted and left normal ovary
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In two patients, right side ovarian torsion was initially mistaken for appendicitis. The ovaries were bulky and multicystic. All other patients had a preoperative diagnosis and ovary conserving surgery was done laparoscopically wherever possible.
Histopathology reports of these patients revealed functional nonneoplastic lesions in 18 cases (66.7%), which include ovarian stroma with hemorrhagic area (hemorrhagic cyst) in 10 cases, simple cyst in three cases, follicular cyst in 3, inclusion cyst in 1 case, and paraovarian cyst in 1 case. Benign neoplastic lesions were present in eight cases (29.6%), which include serous cystadenoma in five cases, mucinous cystadenoma in one case, benign mature cystic teratoma in one case, and benign sex-cord stromal tumor favoring fibroma in one case. One patient in our cohort had presented with malignant neoplastic lesion (3.7%), which was confirmed as JGCT (malignant sex cord stromal tumor). In two patients of ovarian cyst torsion, detorsion and deroofing of the cyst was done without ovarian biopsy; however, intraoperatively, they appeared to be benign. Out of the five antenatally diagnosed ovarian cysts, two were serous cystadenoma, two were serous cyst, and one was follicular cyst.
Three patients presented with large abdominal lump and were managed by laparotomy and salpingo-oophorectomy with peritoneal and lymph node biopsies. Liver metastases were ruled out. Histopathology in one case revealed JGCT-pT1NX, for which she received three cycles of adjuvant chemotherapy (BEP regimen), and the patients' tumor markers levels were monitored. PET-CT scan was performed 9 months postoperatively to look for any residual/recurrent disease which was normal. In two other patients, histopathologic results revealed benign sex-cord stromal tumor favoring fibroma and mucinous cystadenoma required no additional treatment.
Overall survival rate is 100% in our series. One adnexal torsion patient postsalpingo-oopherectomy had twice recurrence of adnexal torsion on opposite side after 6 months and 5 years follow-up which was managed with ovarian preserving surgery. Of the three patients of adnexal tumor who underwent laparotomy in our series, had follow-up of 6 years, 4 years, and 1 year, respectively, and have no complaints. All other patients had uneventful follow-up for minimum of 1 year.
| Discussion | | |
Adnexal masses may originate from the ovaries, Fallopian tube More Detailss, and other pelvic organs. The types of adnexal masses include tumors, inflammatory, or functional cysts. Approximately one third of adnexal masses are ovarian tumors. Malignant ovarian tumors are rare, particularly in patients under 5 years of age.[1],[4] In this study, all patients with ovarian mass undergoing surgical intervention were included. One patient was histopathologically diagnosed with JGCT, and she received adjuvant chemotherapy.
Neonatal ovarian cysts are nearly always benign and self-limiting, and many of them go unreported.[7] In girls, the frequency of such cysts was reported as approximately 5% of all abdominal masses in the 1st month of life.[8]
Follicular cysts of ovary are commonly detected on antenatal US. This is possibly due to ovarian stimulation of the baby's ovary by maternal hormones. Majority of fetal cysts are unilateral. Spontaneous regression of both simple and complex cyst can occur postnatally. Half of them regress by 1 month of age, 75% by 2 months, and 90% by 3 months.[9]
The complications include hemorrhage within the cyst, rupture of the cyst, cyst causing gastrointestinal, or urinary tract obstruction. The most common complication is ovarian torsion and necrosis. In one of our patients who had one ovary removed for gangrene due to ovarian torsion came back with pain on the other side and was diagnosed to have ovarian torsion in the solitary ovary. Because she was diagnosed early, we were able to salvage the ovary. Classically, the child with ovarian torsion has spasmodic recurrent pain and folding of her legs. The pain is usually localized to the side of the ovarian cyst. The plain X-ray is often unremarkable, and there may be a few air fluid levels. The blood counts are normal. A routine US abdomen is normal or may show a bulky ovary. When this happens in a girl child with no accompanying tenderness, a color Doppler must be done to look for ovarian torsion. Most cysts are simple and have very few complications. Malignant changes are extremely rare and are seen only in complex cysts. Other complications which are more common include torsion of the ovarian pedicle, hemorrhage into the cyst or into the abdominal cavity or as a wandering mass in the abdomen which was an autoamputated cyst.[10]
The sonographic appearance of an ovarian cyst is a fluid-filled mass which may be found in the lower abdomen but occasionally also found in the upper abdomen, since the ovarian pedicle in new-born babies is long. This allows it to be mobile, making it more likely to undergo torsion. The differential diagnosis of a cystic mass includes renal or vesical malformation, cystic lesion of mesentery, lymphangioma, and even a pelvic teratoma. US revealed torsion of ovarian masses in 20 (66.7%) ovaries in our series as opposed to 25% in the series reported by Khedkar et al.[4] Color Doppler is a useful investigation but is very operator dependant and can be false negative as decreased blood flow is not always seen.[11] However, it is must for early detection of ovarian cyst/tumor. In a girl having severe right-sided flank pain with normal leukocyte counts, differential diagnosis of right ovarian torsion must be kept in mind along with acute appendicitis. The use of a CT/MRI scan of abdomen in these cases more or less allows for accurate diagnosis.[3],[12]
Once detected the purpose of treatment of ovarian cysts is to preserve ovarian parenchyma as much as possible while avoiding complications.[3],[12] Several reports have been suggested that unilateral simple cysts which are echo free (<5 cm) can be managed conservatively with serial USs as a good majority may resolve spontaneously. The interval between each US should be individualized for each patient. The decision to operate depends on clinical symptoms: abdominal distension, severe pain, respiratory distress, etc., The US appearance of a large complex cyst warrants early surgery.[13] All simple cysts larger than 5 cm in diameter present a greater problem as they are more prone to complications if left untreated.[3] Ovarian torsion is the most common complication which requires laparoscopy along with ovarian de-torsion. The cyst puncture and de-roofing should be performed at the same time. Cystectomy or even oophorectomy/salpingo-oopherectomy should be reserved for complicated or intractable cysts. The most important risk with cyst puncture is cyst rupture leading to peritonitis which is very rare. This conservative approach thus prevents the removal of normal viable ovarian tissue and preserves the reproductive potential of the candidate.
Teratomas are the most common germ cell tumors observed in the majority of published series.[4],[14] This subgroup of tumors may be further divided into mature teratomas, which are benign or immature teratomas, which may be either malignant or benign. Teratomas are composed of recognizable tissues of ectodermal, mesodermal, and endodermal origin, in any combination. Immature teratomas are common germ cell tumors comprising two or more germ cell layers (ectoderm, mesoderm, or endoderm) derived from a pluripotent malignant precursor cell. Mature teratomas account for approximately 15% of all ovarian tumors,[15] whereas immature tumors are rare, representing less than 1% of ovarian tumors.[16] Mature teratomas are classified as cystic, solid or mono-dermal. Immature teratomas show only solid mass. In immature teratoma cases, AFP is widely used. It has been suggested that the AFP level in immature teratoma is not correlated to either stage or grade of the tumor.[17]
Granulosa cell tumors (GCT) are rare sex cord stromal tumor, encompassing only 2% of all the ovarian tumors.[18] Only 0.1% of all ovarian tumors and 4%–5% of GCT occur in children. JGCT a subtype of ovarian sex cord stromal tumor has a favorable prognosis if diagnosed at an early stage.[19]
Templeman et al.[20] retrospectively reviewed the charts of 140 girls and young women under 21-year-old with noninflammatory ovarian masses treated at their institution and reported that most frequent cause of an ovarian mass requiring surgery is an ovarian cyst, which justifies consideration of a laparoscopic approach. The patient's age rather than time of surgery predict operative approach and type of surgery. Caution should be exercised in patients over age 12 months with a complex mass on US and clinical evidence of hormonal activity, as these masses are usually malignant.
In our study, functional cysts (66.7%) were most common adnexal masses followed by benign neoplasms (29.6%) and malignant neoplasms (3.7%), which are similar to the study by Spinelli et al.[21] who also reported functional cysts (48%) as most common adnexal masses followed by benign neoplasms (42.5%) and malignant neoplasms (4.0%).
Łuczak and Bagłaj[5] retrospectively analyzed 214 patients operated on for ovarian masses and reported that predominantly solid structures noted on imaging studies, large dimension, and positive tumor markers are the clinical predictors of malignancy. A diagnosis of purely cystic lesion with negative markers or of a small size should be an indication for a gonad-sparing procedure. Until more prospective clinical trials regarding this topic are conducted, efforts should be made to improve operative techniques and treatment planning with the aim of increasing the rate of ovarian-sparing procedures. Given the above mentioned characteristics of malignant and non-malignant lesions we propose a simplified algorithmic management approach to ovarian masses in girls [Figure 8]. | Figure 8: Management algorithm for ovarian masses in pediatric and adolescent patients
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| Conclusions | | |
Adnexal masses in children include a broad array of pathologic diagnoses that have variable clinical presentations. In our series, majority of patients present either in infants <6 months age group or near puberty. The most common pathology in our series was ovarian cyst undergoing torsion. AFP, b-HCG, and CA-125 are normal in all our cases of ovarian torsion, so we can avoid doing these tests in cases of ovarian torsion. Diagnosis of ovarian cysts with/without torsion requires high index of suspicion. Color Doppler, done in preoperative period, helps in decision-making and safe management by laparoscopic route. Ovarian conservation is possible when diagnosed early. Detorsion is safe and should always be attempted to salvage the adnexa. Prompt diagnosis and early intervention cannot be over emphasized. If cystectomy is difficult in an ischemic ovary the patient can be re-examined and reoperated in 6–8 weeks. Simple cyst can be monitored safely with close follow-up. Surgery is indicated if the cyst fails to regress after several months or is symptomatic. Ovarian cysts in children are generally benign. With accurate staging, complete resection, and chemotherapy for malignant tumors, patients have demonstrated excellent survival rates.
Acknowledgment
The authors would like to thank Jyoti Badola for typing the manuscript and spending hours on getting the photos just as we wanted. Dr. N.R. Shastri who assisted me on the first few surgeries and kindled my interest in the subject.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
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