Giant serous adenofibroma of the fallopian tube.

Serous adenofibroma of the fallopian tube is a rare, benign tumour of the female genital tract. They are usually small, asymptomatic and incidentally diagnosed during a surgery for another gynaecological condition. This report presents an atypical case of a 17-year-old girl with a tubal serous adenofibroma that presented with a palpable mass occupying the entire abdomen accompanied by urinary symptoms. She underwent a laparoscopic surgery with drainage of 1800 mL of yellow, citrine liquid from the cyst and left salpingectomy with no complications.

obstetrics, gynaecology and fertility; pathology

Tubal serous adenofibroma is a rare, benign, tumour with less than 20 cases reported in medical literature.1–11 It is normally small, asymptomatic and discovered incidentally during the work-up or surgery for another gynaecological disorder.1 We are reporting on a clinical case of a 17-year-old girl with a tubal serous adenofibroma presenting with an abdominal mass and urinary symptoms. As far as we know, this is the youngest patient ever described in the literature with this condition and one of the largest serous adenofibromas identified to date. Case presentation A 17-year-old girl, presented with symptoms of abdominal distension, that evolved over a 4-month period, associated with urinary symptoms including dysuria and stress incontinence. The patient experienced menarche at 12 years of age and was not yet sexually active. Her periods were painful and irregular. She had a medical history of epilepsy and asthma, both stable, with no need of medication. On presentation she was apyrexic and haemodynamically stable. The physical examination revealed an enlarged abdomen with a firm, non-tender mass with unrestricted mobility, localised to the umbilical region and left lumbar quadrant. The mass was well-delineated and slightly painful to palpation but with no signs of an acute abdomen. The external genitalia were normal. Investigations Pelvic ultrasound scan revealed a cystic, anechoic, unilocular formation arising from the pelvis (origin unknown) and reaching up to the xiphisternum measuring 159×87×18 mm. No sinister features such as septum, vegetations, solid components, calcifications or papillary formations were detected. Both ovaries were visible and apparently normal. The MRI (figure 1) showed a cystic unilocular lesion measuring 210×170×75 mm with thin regular walls, no signs of malignancy and apparent origin in the left ovarian. The MRI did not detect any gastrointestinal or urological anomalies. Full blood count was normal as were tests for pregnancy, inflammation and tumour markers. The summary urine and urine culture tests were negative. Differential diagnosis An abdomino-pelvic mass in a teenager requires a prompt careful investigation and an accurate differential diagnosis. The main priority is to exclude malignancy. An important cause of a pelvic mass development in a patient is a leiomyofibroma, however, it is infrequent in teenagers and the ultrasound and MRI promptly excluded this diagnosis. An advanced pregnancy was ruled out by a negative serum hCG (human chorionic gonadotropin). Pelvic inflammatory disease was excluded by (1) clinical history (no previous sexual intercourse), (2) clinical examination (apyrexic and no vaginal discharge), (3) blood tests (negative serum inflammation markers) and (4) radiological examinations. The patient’s urinary symptoms instigated investigations to exclude urinary tract disorders which were excluded by normal urine analysis and imaging. This led us to believe that the symptoms were due to compression caused by the occupying space lesion. Although a high percentage of abdomino-pelvic masses in teenagers are gynaecological (table 1), other organ-related conditions should be excluded (table 2). In the presented case, the patient did not manifest any gastrointestinal symptoms or systemic signs and her clinical examination was not compatible with an acute abdomen, impaction, inflammatory bowel disease, aneurysm or ascites. Table 1. | Site | Disease | | Ovary | Organic and functional cystsFollicular cyst Luteal cyst Haemorrhagic cyst | | Ovarian torsion | | | Endometriosis | | Benign tumoursTeratoma | | | Malignant tumours | | | Fallopian tube | Tubo-ovarian abscess; pelvic inflammatory disease | | Para-ovarian cysts | | | Hydrossalpinx | | | Ectopic pregnancy | | | Benign tumours | | | Malignant tumours | | | Uterus | Pregnancy | | Uterus body neoplasm | | | Fibroma | | | Müllerian anomalies | | | Haemato-pyometra | Table 2. | Site | Disease | | Gastrointestinal tract | Appendicular abscess | | Neoplasms | | | Diverticulitis, peridiverticular abscess | | | Crohn’s disease, segmental ileitis | | | Impaction | | | Mesenteric cysts | | | Hepatoblastoma | | | Hepatocellular carcinoma | | | Choledochal cyst | | | Urinary tract | Pelvic kidney | | Bladder globe | | | Hydronephrosis | | | Polycystic kidney | | | Urachus cyst | | | Bladder tumours | | | Miscellany | Lymphadenopathy | | Peritoneal carcinomatosis | | | Musculo-skeletal tumours | | | Organ ectopia (migrant spleen) | | | Pelvic vessel aneurysms | | | Foreign bodies | | | Pelvic dysmorphisms | | | Complications of previous surgery | | | Haematomas | | | Musculoskeletal inflammations | Thus, an adnexal mass was the most likely cause for the clinical findings. When investigating this type of clinical picture, ultrasound (suprapubic and endovaginal) is the most important examination as it is specific and accessible.12 Good quality imaging allows identification of the origin (ovary, fallopian tube, uterine, extra-gynaecological), and can distinguish between benign and malignant adnexal masses. In the case presented, despite the large volume of the tumour, suprapubic ultrasound revealed a pelvic mass with mainly benign characteristics (anechoic cystic lesion without any calcifications, septum, papillary formations). The MRI provided additional characterisation of the mass and the surrounding tissues, which was useful for surgical planning. Serum Ca125 is an important marker when evaluating an adnexal mass and its risk of malignancy but it can also be elevated in benign conditions such as tubo-ovarian abscess, endometriosis, peritonitis and diverticulitis.12 During the investigation of this clinical case the clinical history, physical examination and imaging directed the diagnosis to a pelvic, most probably gynaecological, mass with benign characteristics, despite its size. The true origin of the mass (ovary, fallopian tube or para-ovarian) was not possible to ascertain from ultrasound and MRI. Treatment Due to the large tumour dimensions, it was not possible to perform a diagnostic laparoscopy. Therefore, before the surgery, 1800 mL of yellow, citrine fluid was drained from the cyst using a Veress needle inserted through the umbilicus into the cyst. Afterwards, the Veress needle was removed and inserted again in the umbilical incision to create the CO2 pneumoperitoneum. This approach allowed insertion of a 10 mm trocar and subsequent execution of a diagnostic laparoscopy. Inspection of the abdomino-pelvic cavity revealed a collapsed, simple cyst arising from the left fallopian tube. The uterus, right fallopian tube, both ovaries, liver, omentum and peritoneal and diaphragmatic surfaces appeared normal. There was no ascitic fluid nor other signs of malignancy identified intraoperatory. The surgeons inserted three accessory 5 mm trocars (lower abdominal quadrants and supra-pubic) and tried to perform a cystectomy. However, due to the large volume of the cyst and its adherence to the fallopian tissue, it was impossible to find a cleavage plane between the fallopian tube and the cyst. As a result, the cystectomy revealed itself very difficult to achieve and a left salpingectomy had to be performed (figure 2). The fallopian tube was removed using an endo-bag and sent for anatomopathological examination. Outcome and follow-up The liquid cytology was compatible with a benign cyst. Histology results revealed a fallopian tube with 77×100 mm with oedematous layers and with a cystic adherent structure with irregular external surface and internal surface with trabecular areas and four papillary projections with 4–9 mm, compatible with a tubal serous adenofibroma. The patient recovered uneventfully and was discharged 1-day postoperatively. Twenty months after surgery, the patient is clinically stable and asymptomatic.

Adnexal masses are uncommon in adolescent females with an estimated incidence of 2.6 per 100 000.13 Although the accurate incidence is difficult to calculate, it appears that 9%–11% of adnexal masses in adolescents are malignant.14 Regarding very large pelvic masses, there is no direct correlation with malignancy in teenagers and, as with adults’, size has not proven to be a good predictor of malignancy.13 Traditionally, the size of an adnexal tumour was a limiting factor when evaluating the possibility of a laparoscopic approach. Nowadays, the contraindications to a laparoscopic approach are not clearly defined. Larger tumours, however, may indicate a higher risk of malignancy and greater operative difficulties. According to Talwar et al15 the limiting factor in performing a laparoscopy instead of a laparotomy in voluminous tumours is mostly the risk of malignancy and not the tumour size. Furthermore, laparoscopic surgery provides better visualisation, shorter hospital stay, faster recovery, less postoperative pain and better cosmetic results.15 There are many reports in the literature regarding laparoscopic management of a voluminous adnexal mass. A significant number recommend decompressing the cyst by aspiration under direct visualisation, drainage using a mini-laparotomy or direct aspiration with a Veress needle with or without ultrasound guidance.16 17 The advantage of decompression is not only to provide adequate space to perform the surgery, but also to avoid inadvertent spillage of the cyst contents. In our case, as the tumour characteristics were benign and there was no clinical, laboratorial or imagiological suspicion of a malignant mass, the spillage of the cyst contents was not a concern. The large dimensions of the tumour in relation to the patient’s abdominal cavity made it impossible to perform a drainage under direct visualisation. As the mass was easily palpable, well delimited and with dimensions above the umbilicus, it was easy to introduce the Veress needle through the umbilicus and directly aspirate the cyst content before the diagnostic laparoscopy. Thereby, we avoided larger incisions and still performed the laparoscopy safely, with all the advantages associated with that approach. Tubal serous adenofibroma is a rare tumour with low malignant potential.2 As far as we know, it was first reported by Iwanov.3 Although its origin is not clear, it was hypothesised to be an embryological remnant originated from the müllerian duct rather than a neoplastic proliferation process.4 Benign epithelial fallopian tube tumours are categorised into papilloma and serous adenofibromas.18 The tumour is usually a round, small (0.5–3 cm), solitary mass that grows intraluminally or attached to the tubal serous surface or fimbriae. It is composed of stromal connective tissue without nuclear pleomorphism or mitosis, and papillary or tubal structures lined by epithelial cells.5 Unlike most reported cases, our case reveals a large mass occupying the majority of the abdomen and causing compressive urinary symptoms. Most reported cases were diagnosed incidentally in patients aged 30–50 years old. Only two cases, besides ours, occurred in patients under 21 years old.1–11 There are some reported cases, like ours, that are symptomatic and may present as acute abdominal pain or subfertility. Symptoms associated with this condition include: abdominal pain, palpable mass, vaginal bleeding and urinary or bowel symptoms. Preoperative diagnosis can be difficult as the tumour may appear malignant macroscopically or on ultrasound and histology is necessary to confirm the diagnosis. Also, in respect to larger tumours it may be difficult to define the true starting point of this lesion. In this case, the MRI did not offer additional information since it could not define the origin of the mass and the ultrasound had already shown benign characteristics. Most of the reported cases were submitted to a radical surgery (adnexectomy or salpingectomy) because it was not possible to diagnose the condition preoperatively. There were two cases associated with infertility with tumours smaller than 6 cm, where the diagnosis was suspected prior to surgery and a cystectomy was performed.1 6 In our case, due to technical difficulty it was not possible to perform a conservative procedure and therefore, a salpingectomy was performed. Learning points. An abdomino-pelvic mass in a teenager requires a prompt, thorough investigation to exclude malignancy or serious complications such as adnexal torsion. Large cystic lesions with benign characteristics may be treated by laparoscopy with reduced morbidity. Tubal serous adenofibromas are rare benign tumours that may appear malignant macroscopically or on ultrasound. Given their low malignant potential, it is important to consider diagnosis before planning a radical surgery, especially in patients who wish to preserve their fertility. Footnotes Contributors: AA conducted the patient clinical evaluation and treatment. MAT and AA conceived the idea and designed this article. RSC and ML were responsible for collection of the supporting data and literature revision. MAT analysed the data and was in charge of the article drafting and writing. AA supervised the findings of this work. All authors reviewed the content and contributed to the final manuscript approval. The four authors agreed to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work were appropriately investigated and resolved. Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors. Competing interests: None declared. Patient consent for publication: Obtained. Provenance and peer review: Not commissioned; externally peer reviewed.

- 1.Grigore M, Popovici R, Furnica C, et al. Three-Dimensional ultrasound and HDlive in tubal serous cystadenofibroma: a case report and literature review. Med Ultrason 2017;19:444–6. 10.11152/mu-986 [DOI] [PubMed] [Google Scholar] - 2.de Silva TS, Patil A, Lawrence RN. Acute presentation of a benign cystadenofibroma of the fallopian tube: a case report. J Med Case Rep 2010;4:181. 10.1186/1752-1947-4-181 [DOI] [PMC free article] [PubMed] [Google Scholar] - 3.Iwanow WM. Cystadenofibroma papilliferum tubae Fallopie. Zentalbl Gynaekol 1909;33:745. [Google Scholar] - 4.Gürbüz Y, Ozkara SK. Immunohistochemical profile of serous papillary cystadenofibroma of the fallopian tube: a clue of paramesonephritic origin. Appl Immunohistochem Mol Morphol 2003;11:153–5. [PubMed] [Google Scholar] - 5.Khatib Y, Patel RD, Kashikar AS, et al. Serous papillary cystadenofibroma of the fallopian tube: a case report and short review of literature. Indian J Pathol Microbiol 2015;58:524–7. 10.4103/0377-4929.168865 [DOI] [PubMed] [Google Scholar] - 6.Sills ES, Kaplan CR, Perloe M, et al. Laparoscopic approach to an uncommon adnexal neoplasm associated with infertility: serous cystadenofibroma of the fallopian tube. J Am Assoc Gynecol Laparosc 2003;10:545–7. 10.1016/S1074-3804(05)60165-X [DOI] [PubMed] [Google Scholar] - 7.Silverman AY, Artinian B, Sabin M. Serous cystadenofibroma of the fallopian tube: a case report. Am J Obstet Gynecol 1978;130:593–5. 10.1016/0002-9378(78)90088-1 [DOI] [PubMed] [Google Scholar] - 8.Valerdiz Casasola S, Pardo Mindan J. Cystadenofibroma of fallopian tube. Appl Pathol 1989;7:256–9. [PubMed] [Google Scholar] - 9.Erra S, Costamagna D. Serous cystadenofibroma of the fallopian tube: case report and literature review. G Chir 2012;33:31–3. [PubMed] [Google Scholar] - 10.Terada T. Serous surface papillary cystadenoma of fallopian tube clinically presenting with ectopic pregnancy. Case Reports in Clinical Pathology 2016;3:2331–726. 10.5430/crcp.v3n3p48 [DOI] [Google Scholar] - 11.Pandey, A, et al. Benign cystadenofibroma of the fallopian tube: a case report. J Physiol Pathophysiol 2012;3:29–30. 10.5897/JPAP11.021 [DOI] [Google Scholar] - 12.Alessandrino F, Dellafiore C, Eshja E, et al. Chiara Cassani and Alfredo La Fianza. differential diagnosis for female pelvic masses. medical imaging in clinical practice, Okechukwu Felix Erondu, IntechOpen, 2013. Available: https://www.intechopen.com/books/medical-imaging-in-clinical-practice/differential-diagnosis-for-female-pelvic-masses - 13.Zolton JR, Maseelall PB. Evaluation of ovarian cysts in adolescents. Open J Obstet Gynecol 2013;03:12–16. 10.4236/ojog.2013.37A1004 [DOI] [Google Scholar] - 14.Eskander RN, Bristow RE. Adnexal masses in pediatric and adolescent females: a review of the literature. Curr Obstet Gynecol Rep 2012;1:25–32. 10.1007/s13669-011-0001-4 [DOI] [Google Scholar] - 15.Talwar P, Talwar K, C. A. PS, Pranidha Shree CA. Large adnexal mass: is laparoscopic surgery a safe option? Int J Reprod Contracept Obstet Gynecol 2019;8:1431–5. 10.18203/2320-1770.ijrcog20191194 [DOI] [Google Scholar] - 16.Nagele F, Magos AL. Combined ultrasonographically guided drainage and laparoscopic excision of a large ovarian cyst. Am J Obstet Gynecol 1996;175:1377–8. 10.1016/S0002-9378(96)70060-1 [DOI] [PubMed] [Google Scholar] - 17.Rabbani I, Wynn JS, Hickling DJ. Laparoscopic excision of a large ovarian cyst. Gynecol Surg 2007;4:225–7. 10.1007/s10397-007-0277-6 [DOI] [Google Scholar] - 18.Kurman J, Carcangiu L, Herrington S, et al. Who classification of tumours of female reproductive organs. WHO Press, 2014. [Google Scholar]