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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 1  |  Issue : 2  |  Page : 50-52

Cervical adenoma malignum: Diagnostic dilemma of a rare form of cervical cancer


1 Department of Obstetrics and Gynaecology, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna State, Nigeria
2 Department of Histo-Pathology, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna State, Nigeria

Date of Web Publication20-Apr-2017

Correspondence Address:
Fadimatu Bakari
Department of Obstetrics and Gynaecology, Ahmadu Bello University Teaching Hospital, Zaria, Kaduna State
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/archms.archms_6_17

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  Abstract 

Adenoma malignum of the cervix is a rare variant of Adenocarcinoma of the uterine cervix. Its overall incidence rate is low. We report a case of a 51-year-old married multipara who presented with a 4-year history of recurrent, heavy, prolonged menstrual flow, postcoital bleeding, and passage of mucoid vaginal discharge. Pelvic examination revealed a bulky, firm, endophytic cervical mass with ulcerative areas. Ultrasonography showed features of multiple uterine fibroids. Cervical smear cytology and histology of cervical punch biopsy did not show evidence of malignancy. The patient subsequently had a total abdominal hysterectomy and bilateral salpingo-oophorectomy for a purportedly benign pathology of the cervix. Diagnosis of cervical adenoma malignum was made from the surgical specimen postoperatively.

Keywords: Adenoma malignum, minimal deviation adenocarcinoma, uterine cervix


How to cite this article:
Bakari F, Folagbade OB, Umar-Sulayman H, Avidime S, Waziri GD, Ameh N, Adesiyun AG. Cervical adenoma malignum: Diagnostic dilemma of a rare form of cervical cancer. Arch Med Surg 2016;1:50-2

How to cite this URL:
Bakari F, Folagbade OB, Umar-Sulayman H, Avidime S, Waziri GD, Ameh N, Adesiyun AG. Cervical adenoma malignum: Diagnostic dilemma of a rare form of cervical cancer. Arch Med Surg [serial online] 2016 [cited 2019 Jan 23];1:50-2. Available from: http://www.archms.org/text.asp?2016/1/2/50/204804


  Introduction Top


Cervical adenoma malignum refers to a highly well-differentiated adenocarcinoma characterized histologically by distorted and architecturally irregular endocervical glands which penetrate deeply into the wall of the cervix, but without more than minimal and focal evidence of gland cell stratification, anaplasia, or mitotic activity.[1] The tumor appears benign and can be misdiagnosed as multiple nabothian cysts.[2] This rare variant of adenocarcinoma of the cervix accounts for about 1%–3% of all cervical adenocarcinomas.[2]

This case was reported because of the rare incidence of cervical adenoma malignum and the difficulty in making a histopathologic diagnosis because of its deceptively benign histologic appearance and the possibility of being confused with deep nabothian cysts.


  Case Report Top


A 51-year-old married Housewife who was a P4 +[1] and had her last childbirth 20 years before presentation presented to the gynecology emergency unit with 4 years history of recurrent heavy, prolonged menstrual flow, occasional postcoital bleeding, dysmenorrhea, and passage of mucoid vaginal discharge. A day before the presentation, the bleeding per vaginam became excessive with the associated passage of blood clots, generalized body weakness, and easy fatigability. There was, however, no history of syncopal attack, yellowish discoloration of the eyes, cough, difficulty in breathing, abdominal swelling, urinary symptoms, bleeding from other orifices, and weight loss.

She attained menarche at the age of 14 years and used to menstruate for 4–6 days in a regular cycle of 28–30 days before the onset of symptoms.  Pap smear More Details cytology performed about a week before the presentation was negative.

General physical examination revealed a middle-aged woman who was pale but anicteric, acyanosed, well hydrated and had no pedal edema. Her pulse rate was 88 beats/min, small volume and regular. Her blood pressure was 130/80 mmHg. The chest was clinically clear. The abdomen was full and the liver, spleen, and kidneys were not enlarged. The uterus was about 14 weeks pregnant size. Pelvic examination revealed a blood-smeared vulva. The uterus was about 14-weeks pregnancy size on bimanual pelvic examination. A bulky, hyperemic endophytic, firm cervical mass and normal vagina was seen. There was no adnexal mass. Digital rectal examination was normal and there was no parametrial or pelvic wall involvement.

Cervical punch biopsy was taken, and histology showed polypoid lesion comprising tall columnar endocervical epithelium that formed crypts in areas overlying lamina propria with some granulation tissue that contained scattered chronic inflammatory infiltrates. There were no features of malignancy on the histological sections. Abdominopelvic ultrasound scan showed a bulky uterus with anterior uterine wall fibroid that measured 5.09 cm. The packed cell volume was 27%. Bedside clotting time was 8 min. Serum electrolytes, urea, creatinine, and liver function test were within normal limits.

A diagnosis of the cervical polyp was made based on normal Pap smear and punch biopsy histology result that did not show evidence of malignancy. She had a total abdominal hysterectomy with bilateral salpingo-oophorectomy. Intraoperatively, the peritoneal cavity was clean, the uterus was about 14-weeks pregnancy size harboring multiple intramural fibroids and the largest measured about 5 cm in diameter. The cervix was bulky with ulcerative areas. The  Fallopian tube More Detailss appeared grossly normal, and the ovaries looked atretic bilaterally. The patient had an uneventful postoperative recovery and was discharged on the 7th postoperative day.

Histopathological report of surgical specimen showed that sections of the cervix had the following findings; acanthotic ectocervical epithelium in areas with koilocytotic atypia overlying a fibrosed subepithelial stroma within which were numerous deeply dispersed seemingly normal endocervical glands. Other findings are nabothian cysts and sprinklings of mononuclear inflammatory cells [Figure 1] and [Figure 2]. The endometrium was atrophic, the myometrium harbored multiple leiomyoma, the fallopian tubes and ovaries were unremarkable. The histopathologic diagnosis was Cervical Adenoma Malignum.
Figure 1: Micro-photography of adenoma malignum.

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Figure 2: Photomicrograph of adenoma malignum.

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  Discussion Top


Cervical adenoma malignum is a mucinous type of minimal deviation Adenocarcinoma,[3] with unusually well-differentiated glandular cell morphology similar to that of normal cells.[4] The major symptoms it usually presents with are the passage of abnormal, usually mucinous vaginal discharge and vaginal bleeding.[5] The case reported here presented with a history of the passage of mucoid vaginal discharge. Unlike typical cervical cancer, most cases of adenoma malignum exhibit an endophytic rather than an exophytic growth pattern as found in this patient.[2] When the presence of the disease is suspected by cytology of cervical smear which obviously was not the case in this patient, a deep cervical biopsy is usually necessary to make an accurate diagnosis. An ordinary cervical biopsy may be insufficient to detect deeply situated tumor glands,[6] as was the case in this patient. The histopathologic diagnosis of the sections of the cervical punch biopsy taken in this patient was a cervical polyp. Authors have reported minimal deviation Adenocarcinoma of the cervix in association with  Peutz-Jeghers syndrome More Details,[7] and such patient may present with hamartomatous polypoid lesions in the gastrointestinal and/or reproductive tracts.[7],[8] This patient did not have any clinical features suggestive of the syndrome.

The deceptively benign histologic appearance of adenoma malignum may lead to misdiagnosis as the tumor may be confused with deep nabothian cysts.[2],[4],[9] As in this patient, diagnosis of the disease remains difficult and is frequently made only after hysterectomy for a presumed benign indication.[8] The patient discussed had a simple hysterectomy due to the preoperative diagnosis of the cervical polyp. As the case under review, false negative cytology results have also been reported from cervical cytology smears.[10]

There is no standard treatment for this condition. Surgery combined with other adjunct therapy is the main treatment modality depending on the stage of the disease,[10] this ranges from simple hysterectomy to radical hysterectomy with pelvic lymphadenectomy. Adjunctive therapy includes chemotherapy and/or radiotherapy. Patients with the stage IV disease are, however, managed with palliative radiotherapy.[10] The patient was referred to the oncologist for radiotherapy, chemotherapy or both as they may find appropriate. Studies have shown that the prognosis of cervical adenoma malignum may be better than ordinary cervical adenocarcinoma.[6] Early dissemination, poor response to therapy, and an unfavorable prognosis have also been reported by other authors.[10],[11]

In conclusion, minimal deviation Adenocarcinoma (adenoma malignum) of the cervix is a rare disease and may pose a challenge in making a clinicohistopathologic diagnosis. A high index of suspicion should awaken the clinician when there is postcoital bleeding, passage of mucoid vaginal discharge, bulky cervix with endophytic/ulcerative growth with negative cervical smear, and cervical punch biopsy for malignancy.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Silverberg SG, Hurt WG. Minimal deviation adenocarcinoma (“adenoma malignum”) of the cervix: A reappraisal. Am J Obstet Gynecol 1975;121:971-5.  Back to cited text no. 1
    
2.
Kaminski PF, Norris HJ. Minimal deviation carcinoma (adenoma malignum) of the cervix. Int J Gynecol Pathol 1983;2:141-52.  Back to cited text no. 2
    
3.
Ishii K, Hosaka N, Toki T, Momose M, Hidaka E, Tsuchiya S, et al. A new view of the so-called adenoma malignum of the uterine cervix. Virchows Arch 1998;432:315-22.  Back to cited text no. 3
    
4.
Sato S, Ito K, Konno R, Okamoto S, Yajima A. Adenoma malignum. Report of a case with cytologic and colposcopic findings and immunohistochemical staining with antimucin monoclonal antibody HIK-1083. Acta Cytol 2000;44:389-92.  Back to cited text no. 4
    
5.
Lim KT, Lee IH, Kim TJ, Kwon YS, Jeong JG, Shin SJ. Adenoma malignum of the uterine cervix: Clinicopathologic analysis of 18 cases. Kaohsiung J Med Sci 2012;28:161-4.  Back to cited text no. 5
    
6.
Hirai Y, Takeshima N, Haga A, Arai Y, Akiyama F, Hasumi K. A clinicocytopathologic study of adenoma malignum of the uterine cervix. Gynecol Oncol 1998;70:219-23.  Back to cited text no. 6
    
7.
Soeters R, Tiltman A, Learmonth G, Bloch B, Dehaeck K, Levin W. Peutz-Jeghers syndrome in association with adenoma malignum (minimal deviation adenocarcinoma) of the cervix. Case report. Br J Obstet Gynaecol 1989;96:1101-4.  Back to cited text no. 7
    
8.
Podczaski E, Kaminski PF, Pees RC, Singapuri K, Sorosky JI. Peutz-Jeghers syndrome with ovarian sex cord tumor with annular tubules and cervical adenoma malignum. Gynecol Oncol 1991;42:74-8.  Back to cited text no. 8
    
9.
Clement PB, Young RH. Deep nabothian cysts of the uterine cervix. A possible source of confusion with minimal-deviation adenocarcinoma (adenoma malignum). Int J Gynecol Pathol 1989;8:340-8.  Back to cited text no. 9
    
10.
Chang J, Zhang S, Zhou H, Liang JX, Lin ZQ. Clinical analysis of minimal deviation adenocarcinoma of the cervix: A report of five cases. Chin J Cancer 2008;27:1310-4.  Back to cited text no. 10
    
11.
Kaku T, Enjoji M. Extremely well-differentiated adenocarcinoma (“adenoma malignum”) of the cervix. Int J Gynecol Pathol 1983;2:28-41.  Back to cited text no. 11
    


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