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  Table of Contents    
CASE REPORT  
Year : 2015  |  Volume : 58  |  Issue : 4  |  Page : 524-527
Serous papillary cystadenofibroma of the fallopian tube: A case report and short review of literature


1 Department of Pathology, R. N. Cooper Hospital, Mumbai, Maharashtra, India
2 Department of Gynecology, R. N. Cooper Hospital, Mumbai, Maharashtra, India

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Date of Web Publication4-Nov-2015
 

   Abstract 

Serous papillary cystadenofibromas (SPCAFs) of the fallopian tube are very rare benign tumors of the female genital tract. They are usually asymptomatic and are found incidentally. Until now, only 18 cases of this tumor have been reported in the world literature. We report a case of SPCAF of the left fallopian tube in a 30-year-old female who presented with a large abdominal mass and pain. On computed tomography, a diagnosis of ovarian neoplasm was given. However, during surgery the tumor was found to arise from the fallopian tube and was treated with tubal cystectomy with sparing of the ovary. We present this unique case on account of its rarity, unusual presentation, and huge size along with a short review of literature.

Keywords: Benign, cystadenofibroma, fallopian tube

How to cite this article:
Khatib Y, Patel RD, Kashikar AS, Chavan K. Serous papillary cystadenofibroma of the fallopian tube: A case report and short review of literature. Indian J Pathol Microbiol 2015;58:524-7

How to cite this URL:
Khatib Y, Patel RD, Kashikar AS, Chavan K. Serous papillary cystadenofibroma of the fallopian tube: A case report and short review of literature. Indian J Pathol Microbiol [serial online] 2015 [cited 2019 May 25];58:524-7. Available from: http://www.ijpmonline.org/text.asp?2015/58/4/524/168865



   Introduction Top


Tumors of the  Fallopian tube More Details are uncommon and are the rarest tumors of the female genital tract. According to the World Health Organization (WHO) classification, benign tumors of the fallopian tube comprise papilloma, cystadenoma, adenofibroma, cystadenofibroma (CAF), metaplastic papillary tumor and endometrioid polyp.[1] Serous papillary CAF (SPCAF) is a rare tumor of the fallopian tube which is usually found at the fimbrial end of the tube and is considered to be of mullerian origin.[2] Most women with these tumors are asymptomatic, and majority are incidental findings during work up or surgery for other gynecological disorders.[3] Preoperative diagnosis is usually missed. Even though they are rare, they have to be differentiated from other tumors of the fallopian tube like borderline and malignant tumors as management and prognosis is different.[4] Only, a cystectomy is needed in these cases, and fertility can be preserved especially in young patients.


   Case Report Top


A 30-year-old female, para 2, live 2, presented with the complaints of abdominal mass and pain. Her menstrual history was regular. She had two full term normal deliveries with the last childbirth 3 years back. Her previous gynecological and medical history was uneventful. Abdominal examination revealed an ill-defined mass arising from the pelvis and reaching up to the xiphisternum [Figure 1]a. It was firm in consistency, nontender and had restricted mobility. On per vaginal examination, uterus could not be felt separately from the mass. Both fornices were full and nontender. Contrast enhanced computed tomography revealed a large well-defined cystic lesion of 17.8 cm × 16 cm × 14 cm size arising from the pelvis and extending into the abdomen up to the subhepatic space. A regular nodular enhancing soft tissue component was seen along the inferior margin of the cyst. Left ovary could not be separately identified from the lesion [Figure 1]b. An impression of cystic ovarian neoplasm was given. All routine investigations were normal. Tumor markers were cancer antigen - 125 = 52.98 IU/ml (normal 0–35 IU/ml), l actate dehydrogenase = 651 µ/l (normal 313–318 µ/l), carcinoembryonic antigen = 1.03 ng/ml (normal 0–5 ng/ml), cancer antigen - 19.9 = 4.84 µ/l (normal < 39 µ/l), and alpha-feto protein = 0.6 IU/ml (normal 0.56–2.64 IU/ml). An exploratory laparotomy was done. Intraoperatively, a huge cystic mass was seen occupying the entire abdomen. 2 L of straw colored fluid was drained from the cyst. The cyst was found to arise from the left fallopian tube, and the left ovary was found to be completely separate from the tube. A left tubal cystectomy was done. Grossly, an already partially cut open specimen of the cyst arising from the serosal surface of the fallopian tube was received measuring 12 cm × 10 cm along with the tube measuring 8 cm long [Figure 2]a. On cut section, the cyst was smooth with a solid area measuring 6 cm × 4.5 cm showing multiple, coarse granular papillary excrescences protruding within the lumen [Figure 2]b. Microscopy showed a cyst with many branching papillary projections of varying sizes lined by cuboidal lining with focal stratification. The core of the papillae and cyst wall showed abundant, fibrous stroma of dense connective tissue. No atypia or invasion was seen [Figure 3] and [Figure 4]. Based on the histopathological findings, a diagnosis of SPCAF of the fallopian tube was given. The patient is doing well 2 months after surgery.
Figure 1: (a) Huge ill-defined abdominal mass arising from pelvis and reaching upto the xiphisternum. (b) Contrast enhanced computed tomography showing a large well defined cystic lesion along with a solid component arising from the pelvis and extending upto sub-hepatic region

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Figure 2: (a) Gross photograph showing cyst arising from the serosal surface of the fallopian tube. (b) Gross photograph of cut section of fallopian tube cyst showing multiple, coarse granular papillary excrescences

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Figure 3: Section showing a serous papillary cystadenofibroma arising from the wall of the fallopian tube (H and E, ×40)

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Figure 4: Section showing tumour with many branching papillary projections of varying sizes with core of the papillae and cyst wall having abundant fibrous stoma of dense

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   Discussion Top


Adenofibromas of the fallopian tube are rare tumors, which usually present as small asymptomatic incidental findings. CAF was first diagnosed by Iwanow in 1909 who termed it as CAF papilleferum.[3] Since then 18 cases have been reported in world literature. A review of literature of earlier cases was done. Clinicopathological presentation of the reported cases along with the present case is summarized [Table 1].[2],[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13] The age of presentation showed a wide range from 19 to 73 years. Mean age reported is 49 years. Clinical presentation was variable. In 1914 Kustner described a tubal CAF associated with a hydrosalpinx.[3] In four cases, they were found incidentally during a hysterectomy for leiomyoma [2],[3],[5],[6] and in one case for prolapse.[7] One case was found during lower segment cesarean section [4] and other during a tubal ligation.[8] One case was found associated with normal pregnancy [4],[9] and another occurred concurrently with an ectopic pregnancy.[10] A case of bilateral tumors presented with primary infertility [11] while another occurred during embryo transfer following in vitro fertilization.[12],[14] A case of CAF with torsion presented as an acute abdomen and was mistaken for appendicitis.[13],[15] The present case was unusual for its large size and presentation as an abdominal mass. CAF presents as a round solitary mass and can be found on the fimbrial end, intraluminally or on the serosal surface of the tube. Most are present at the fimbrial end. They are usually small and measure about 0.5–3 cm in diameter. Most are cystic with coarse papillary excrescences as seen in the present case. Histologically two components are present, a connective tissue stroma without nuclear pleomorphism or mitosis and papillary structures or tubal structures lined by epithelial cells. The epithelial cell type has been serous in most cases but occasionally may be endometrioid.[2]. Gürbüz and Ozkara demonstrated that the topographic location of the lesion, histopathological findings, immunoprofile of vimentin, cytokeratin coexpression, and diffuse epithelial membrane antigen immunoreactivity suggested that this tumor is an embryologic remnant originating from the mullerian duct.[3] Tumor seems to have a benign course, and no malignant potential has been described. The differential diagnosis of a tumor of tubal origin includes tubal carcinoma, a serous papillary tumor of low malignant potential (STLMP) and borderline tumor. Malignant neoplasms constitute 0.18–1.6% of all malignant tumors of the female genital tract. Most are seen in postmenopausal women. Microscopy shows cellular pleomorphism with nuclear atypia. STLMP and borderline tumors show stratification with nuclear atypia. Hence, histology is necessary to confirm the diagnosis. Management is conservative, tubal cystectomy being adequate. Laparoscopy may also be used in the diagnosis and even treatment of this condition as demonstrated by Sills et al.[12] The cyst was decompressed and removed intact without incident via a 5 mm laparoscopic cannula.
Table 1: Reported cases of cystadenofibroma of the fallopian tube

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   Conclusion Top


SPCAF of the fallopian tube is a rare lesion which is usually small and asymptomatic. Hence, it is usually diagnosed during surgery for other gynecological disorders. In larger symptomatic cases as in the present case, a preoperative diagnosis is difficult. As they are benign tumors, only a cystectomy is required for their treatment.

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   References Top

1.
Alvarado-Cabrero I, Cheung A, Caduff R. Tumours of the fallopian tube. In: World Health Organization Classification of Tumors: Tumours of the Breast and Female Genital Organs. Lyon, France: IARC Press; 2003. p. 206-11.  Back to cited text no. 1
    
2.
de la Fuente AA. Benign mixed Müllerian tumour – Adenofibroma of the fallopian tube. Histopathology 1982;6:661-6.  Back to cited text no. 2
[PUBMED]    
3.
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.  Back to cited text no. 3
    
4.
Pandey A, Nandini NM, Sarkar A, Tangri R, Bhattacharya S. Benign cystadenofibroma of the fallopian tube: A case report. J Physiol Pathophysiol 2012;3:29-30.  Back to cited text no. 4
    
5.
Iwanow WM. Cystadenofibroma papilliferum tubae fallopie. Zentalbl Gyneakol 1909;33:745.  Back to cited text no. 5
    
6.
Küster I. Einen tumor der fimbria overica. Berl Klin Wochenschr 1914;51:1486.  Back to cited text no. 6
    
7.
Erra S, Costamagna D. Serous cystadenofibroma of the fallopian tube: Case report and literature review. G Chir 2012;33:31-3.  Back to cited text no. 7
[PUBMED]    
8.
Valerdiz Casasola S, Pardo Mindan J. Cystadenofibroma of fallopian tube. Appl Pathol 1989;7:256-9.  Back to cited text no. 8
    
9.
Kanbour AI, Burgess F, Salazar H. Intramural adenofibroma of the fallopian tube. Light and electron microscopy. Cancer 1973;31:1433-9.  Back to cited text no. 9
[PUBMED]    
10.
Silverman AY, Artinian B, Sabin M. Serous cystadenofibroma of the fallopian tube: A case report. Am J Obstet Gynecol 1978;130:593-5.  Back to cited text no. 10
[PUBMED]    
11.
Fukushima A, Shoji T, Tanaka S, Sugiyama T. A case of fallopian tube adenofibroma: Difficulties associated with differentiation from ectopic pregnancy. Clin Med Insights Case Rep 2014;7:135-7.  Back to cited text no. 11
    
12.
Mondal SK. Adenofibroma and ectopic pregnancy of left fallopian tube: A rare coexistence. J Obstet Gynaecol Res 2010;36:690-2.  Back to cited text no. 12
    
13.
Chen KT. Bilateral papillary adenofibroma of the fallopian tube. Am J Clin Pathol 1981;75:229-31.  Back to cited text no. 13
[PUBMED]    
14.
Sills ES, Kaplan CR, Perloe M, Tucker MJ. 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.  Back to cited text no. 14
    
15.
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.  Back to cited text no. 15
    

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Correspondence Address:
Dr. Richa D Patel
Department of Pathology, R. N. Cooper Hospital, Mumbai - 400 056, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.168865

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