Obstet Gynecol Sci.  2018 Sep;61(5):590-597. 10.5468/ogs.2018.61.5.590.

Minimal deviation adenocarcinoma (adenoma malignum) of the uterine cervix: clinicopathological analysis of 17 cases

• Lee MH
• Kim ES
• Choi MC
• Heo JH
• Jang JH
• Jung SG
• Park H
• Joo WD
• Lee C
• Lee JH

• KMID: 2426024
• DOI: http://doi.org/10.5468/ogs.2018.61.5.590

Abstract

OBJECTIVE
The aim of this study was to evaluate the clinicopathological features of minimal deviation adenocarcinoma (MDA) and to analyze its prognostic factors.
METHODS
We retrospectively analyzed the medical records of 17 patients who were diagnosed with MDA at a single institution between January 2005 and December 2015.
RESULTS
The median age of the patients was 47.7 years (33-75 years). MDA was diagnosed in 7 patients (41.2%) before performing definitive surgery. Stage IB disease was diagnosed in 12 patients (70.6%) and advanced stage disease (stage II: 3, stage III: 2) in 5. MDA was incidentally diagnosed following hysterectomy for benign conditions in 6 patients. Adjuvant therapy was administered to 13 patients (76.5%). During median follow-up over 33.6 months (7-99 months), 11 patients (64.7%) showed no evidence of disease, 6 (35.3%) showed persistent or recurrent disease and 5 died of the disease. Peutz-Jeghers syndrome was not suspected in any patient, and no mutation was detected in the 3 patients who underwent genetic testing. Univariate analysis showed that advanced stage disease (P=0.016) and lymphovascular space invasion (P=0.002) demonstrated a statistically significant association with poor overall survival (OS) rates. Advanced stage disease continued to show a significant association with poor OS rates (hazard ratio, 2.92; 95% confidence interval, 1.097-7.746; P=0.032) even after multivariate analysis.
CONCLUSION
Early diagnosis is important to manage MDA. Clinicians should consider MDA among the differential diagnoses in patients with a suspicious clinical presentation even with negative cervical screening tests.

Keyword

Cervical cancer; Minimal deviation adenocarcinoma; Peutz-Jeghers syndrome; Prognostic factors

MeSH Terms

Adenocarcinoma*
Cervix Uteri*
Diagnosis, Differential
Early Diagnosis
Female
Follow-Up Studies
Genetic Testing
Humans
Hysterectomy
Mass Screening
Medical Records
Multivariate Analysis
Peutz-Jeghers Syndrome
Retrospective Studies
Uterine Cervical Neoplasms

Figure

• 

  Fig. 1 (A) Kaplan-Meier curve showing the overall survival rate based on stages of minimal deviation adenocarcinoma (P=0.016). (B) Kaplan-Meier curve showing the overall survival rate based on lymphovascular space invasion (P=0.002).

Reference

1. Li G, Jiang W, Gui S, Xu C. Minimal deviation adenocarcinoma of the uterine cervix. Int J Gynaecol Obstet. 2010; 110:89–92.

2. Naucler P, Ryd W, Törnberg S, Strand A, Wadell G, Elfgren K, et al. Human papillomavirus and Papanicolaou tests to screen for cervical cancer. N Engl J Med. 2007; 357:1589–1597.

3. Gilks CB, Young RH, Aguirre P, DeLellis RA, Scully RE. Adenoma malignum (minimal deviation adenocarcinoma) of the uterine cervix. A clinicopathological and immunohistochemical analysis of 26 cases. Am J Surg Pathol. 1989; 13:717–729.

4. Kaku T, Enjoji M. Extremely well-differentiated adenocarcinoma (“adenoma malignum”) of the cervix. Int J Gynecol Pathol. 1983; 2:28–41.

5. Hemminki A, Markie D, Tomlinson I, Avizienyte E, Roth S, Loukola A, et al. A serine/threonine kinase gene defective in Peutz-Jeghers syndrome. Nature. 1998; 391:184–187.

6. Kurman RJ. International Agency for Research on Cancer. World Health Organization. WHO classification of tumours of female reproductive organs. Lyon: International Agency for Research on Cancer;2014.

7. Colombo N, Carinelli S, Colombo A, Marini C, Rollo D, Sessa C, et al. Cervical cancer: ESMO Clinical Practice Guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2012; 23:vii27–vii32.

8. Kaminski PF, Norris HJ. Minimal deviation carcinoma (adenoma malignum) of the cervix. Int J Gynecol Pathol. 1983; 2:141–152.

9. Silverberg SG, Hurt WG. Minimal deviation adenocarcinoma (“adenoma malignum”) of the cervix: a reappraisal. Am J Obstet Gynecol. 1975; 121:971–975.

10. Hart WR. Symposium part II: special types of adenocarcinoma of the uterine cervix. Int J Gynecol Pathol. 2002; 21:327–346.

11. Massad LS, Einstein MH, Huh WK, Katki HA, Kinney WK, Schiffman M, et al. 2012 updated consensus guidelines for the management of abnormal cervical cancer screening tests and cancer precursors. J Low Genit Tract Dis. 2013; 17:S1–S27.

12. McKelvey JL, Goodlin RR. Adenoma malignum of the cervix. A cancer of deceptively innocent histological pattern. Cancer. 1963; 16:549–557.

13. 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; 70:219–223.

14. Xu JY, Hashi A, Kondo T, Yuminamochi T, Nara M, Hashi K, et al. Absence of human papillomavirus infection in minimal deviation adenocarcinoma and lobular endocervical glandular hyperplasia. Int J Gynecol Pathol. 2005; 24:296–302.

15. An HJ, Kim KR, Kim IS, Kim DW, Park MH, Park IA, et al. Prevalence of human papillomavirus DNA in various histological subtypes of cervical adenocarcinoma: a population-based study. Mod Pathol. 2005; 18:528–534.

16. Odashiro AN, Odashiro DN, Nguyen GK. Minimal deviation endometrioid adenocarcinoma of the cervix: report of three cases with exfoliative cytology. Diagn Cytopathol. 2006; 34:119–123.

17. Schiffman M, Wentzensen N. Human papillomavirus infection and the multistage carcinogenesis of cervical cancer. Cancer Epidemiol Biomarkers Prev. 2013; 22:553–560.

18. Pirog EC, Kleter B, Olgac S, Bobkiewicz P, Lindeman J, Quint WG, et al. Prevalence of human papillomavirus DNA in different histological subtypes of cervical adenocarcinoma. Am J Pathol. 2000; 157:1055–1062.

19. McGowan L, Young RH, Scully RE. Peutz-Jeghers syndrome with “adenoma malignum” of the cervix. A report of two cases. Gynecol Oncol. 1980; 10:125–133.

20. Kuragaki C, Enomoto T, Ueno Y, Sun H, Fujita M, Nakashima R, et al. Mutations in the STK11 gene characterize minimal deviation adenocarcinoma of the uterine cervix. Lab Invest. 2003; 83:35–45.

21. Lee JY, Dong SM, Kim HS, Kim SY, Na EY, Shin MS, et al. A distinct region of chromosome 19p13.3 associated with the sporadic form of adenoma malignum of the uterine cervix. Cancer Res. 1998; 58:1140–1143.

22. Young RH, Welch WR, Dickersin GR, Scully RE. Ovarian sex cord tumor with annular tubules: review of 74 cases including 27 with Peutz-Jeghers syndrome and four with adenoma malignum of the cervix. Cancer. 1982; 50:1384–1402.

23. Srivatsa PJ, Keeney GL, Podratz KC. Disseminated cervical adenoma malignum and bilateral ovarian sex cord tumors with annular tubules associated with Peutz-Jeghers syndrome. Gynecol Oncol. 1994; 53:256–264.