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Misdiagnosis Analysis of Cervical Minimal Deviation Adenocarcinoma: a Report of Three Rare Cases and Literature Review

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Misdiagnosis Analysis of Cervical Minimal Deviation Adenocarcinoma: a Report of Three Rare Cases and Literature Review Available online at www.annclinlabsci.org 680 AnnalS of Clinical & Laboratory Science, vol. 46, no. 6, 2016 Misdiagnosis Analysis of Cervical Minimal Deviation Adenocarcinoma: a Report of Three Rare Cases and Literature Review Mingxing Sui1, Yanling Pei2, Dong Li1, Qiaori Li1, Peining Zhu3, Tianmin Xu1, and Manhua Cui1 1Department of Gynecology and Obstetrics, the Second Hospital of Jilin University, Changchun, Jilin, 2Department of Nursing, China-Japan Union Hospital of Jilin University, Changchun, Jilin, and 3Clinical Medicine College, Jilin University, Changchun, Jilin, P. R. China Abstract. Cervical minimal deviation adenocarcinoma (MDA) is a rare variant of cervical adenocarcinoma that is difficult to diagnose due to the deep location, endogenousow gr th pattern, deceptively benign ap- pearance of tumor cells, and lack of connection to human papillomavirus (HPV). Cytological evalua- tion and biopsies offer suboptimal detection and transvaginal sonography or Magnetic Resonance Imag- ing (MRI) only reveal multiple lesions that mimic multiple benign nabothian cysts. Besides, standard screening, diagnostic tools, and treatments are not established. Thus, MDA tends to be misdiagnosed with other gynecological diseases. In this study, we examine three cases with extensive abdominal metastasis and adhesions, which are not initially associated clinically with HPV and cervical malignancies. All cases were misdiagnosed as nabothian cysts, endometrial adenocarcinoma or ovarian cancer, though finally diagnosed as MDA by postoperative pathology. Delay in diagnosis and treatment can result in irreversible outcomes. Misdiagnoses are analyzed and suggestions for improving early detection are discussed with a brief review of the literature. Key words: minimal deviation adenocarcinoma, gastric-type adenocarcinoma, uterine cervix, diagnosis, treatment. Introduction inspection [6]. Differentiating MDA from normal endocervical glands is difficult due to histologically Minimal deviation adenocarcinoma (MDA) or cer- well-differentiated specimens, particularly those vical adenoma malignum (AM) is a subtype of cer- from cytological evaluation and cervical punch bi- vical mucinous adenocarcinoma [1,2] and accounts opsies. MDA often appears as multilocular lesions for only 1 to 3% of all cervical adenocarcinomas [3] which mimic multiple nabothian cysts, benign tu- and 0.15% to 0.45% of all cervical carcinomas re- mors [7,8]. Although MDA has a benign histologi- ported in the literature [4]. Gastric-type adenocar- cal appearance, it is typically aggressive. In addi- cinoma (GTA) is a newly defined subtype of cervi- tion, MDAs are so rare that their true nature and cal mucinous adenocarcinoma in the Classification clinical course has not been fully clarified. This lack of Tumors of Female Reproductive Organs (4th edi- of information delays accurate diagnosis and leads tion, 2014). MDA is described as an extremely to poor patient prognosis. well-differentiated form of GTA [5]. Chief clinical manifestations are profuse watery or mucoid vagi- Here, we examine three rare cases of MDA lacking nal discharge and irregular bleeding. MDA may HPV infection and malignant findings, but with have endophytic rather than exophytic growth pat- extensive abdominal metastases and adhesions in tern and are, therefore, not obvious with clinical the abdominopelvic organs. These cases were vari- ously identified as nabothian cyst, endometrial ad- Address correspondence to Manhua Cui; Department of Gynecology and Obstetrics, the Second Hospital of Jilin University, 218 Zi enocarcinoma, and ovarian cancer. All cases were Qiang Road, Changchun, Jilin 130041, P. R. China. E-mail: finally diagnosed as MDA according to postopera- [email protected] or Tianmin Xu; Department of Gynecology and Obstetrics, the Second Hospital of Jilin University, 218 Zi Qiang tive pathology. This is the first and largest report of Road, Changchun, Jilin 130041, P. R. China. E-mail: xutianmin@126. com cervical MDA with extensive abdominal metastases 0091-7370/16/0600-680. © 2016 by the Association of Clinical Scientists, Inc. Table 1. The characteristicS of our patients and the diagnosiS were summarized. Patient Age Presentation Gynecologic HPV ThinPrep Preoperative Postoperative Treatment Outcome Presen (years) examination infection cytology test diagnosis or diagnosis tation to initial outcome diagnosis (months) 1 55 Vaginal discharge Normal cervix (-) Inflammation(+) Pyometra MDA Surgery Died of cancer 6 Malignant lesions(-) 2 63 Vaginal discharge Hypertrophic (-) Inflammation(+) Nabothian cysts MDA Surgery Died of cancer 20 Lower abdominal colic but smooth cervix Malignant lesions(-) Ovarian tumor Chemotherapy Endometrial (6 courses) adenocarcinoma 3 38 Vaginal discharge Cysts in cervix (-) Inflammation(-) Nabothian cysts MDA Surgery Died of cancer 4 Lower abdominal pain Malignant lesions(-) Ovarian Ovarian Chemotherapy carcinoma mucinous (3 courses) adenocarcinoma MDA: minimal deviation adenocarcinoma tion nal discharge sinceprimiparous) menopause. presented with one Ca Ca tion for treating patients withto MDA. better diagnose MDA and ideallycinoma lay the or founda- that mimic nabothian cysts, endometrial adenocar- planned. pingooophorectomy, and pelvic lymphadenectomy fication and total abdominal hysterectomy,The bilateral cancer sal- was staged accordingas IB1 cinoma classi- toFIGO area indicated irregular glands consistent withcharge adenocar- source, colposcopy wasphic used cervix and and SCC biopsyGy was of normal. the and a uterine cavity massuterine (3.5 enlargement cm similar x to 2.0 the cm) after previous presentation treatment. discharge occurred for two months, instead ofHy impr inflammatorytreatment. cells. genic fluids Pa we lowish-white pyogenic fluid. adhesionsThe patient and finally pyometra under- withthesia. a copious amount of the patient was under aThus, combined hysteroscopy spinal-epidural anes- examinationcult to was performed navigate while outpatient procedure, beyond the the cervicalnant canals external lesions. cervical While hysteroscopy orifice. moderate was inflammation anticipated and as nointraepithelial an infection was negative.malig- or A ThinP uterine cavity mass measuringslightly 5.6 enlarged cm and transvaginal x sonographycervix 2.7 except sho for cm. minor HPV noted pelvicascites adhesions not be visualized.um The of surface the uterus in of thethe abdominopelvic middle ofBoth the uterine pelvic cavity. sides The parametri- tightly adherent to the uterus andceeded infiltrated 1 with cancer. cm. The bladderand and mesenteric surface intestines. fixed to the transverse colonomentum and partially wasadherent hard tothe and nodular thologic examination of the endometrium and py nt internal cervix adhesiolysis and pyometra cleaning. se 1. necologic examination pogastralgia and abnormal odorous bloody se re Di Re ve we In (Figure 1a) latation and curettage aled normal genitalia and a normally appearing A 55-year-old Chinese woman (primigravida, ports traoperative findings sho re Ap Mi Se ro aspirated from the abdominal cavity and the ov pr ve re sdiagnosis analysis of cervical MDA und ligament and the whole uterus could arian cancer. ve ox ral neoplastic focal tissues on the intestine aled inflammation without malignant imately, 800 ml faint Tr . we ansvaginal sonography confirmed re we va thick and hard, stabilizing the ginal discharge. The uterus was re we re ve apparent and the largest ex- re We aled a firm and hypertr re re we per ctum p cytology test (Figure 2a) Gy ye present information re re we ar of abnormal va To necologic examina- d internal cervical sive. Thegreater we quired after anti- evaluate the dis- ye re we llow mucous firm, brittle, re re which was appeared. too diffi- re va ve we ov ginal va we 68 aled ye ing d a gi- o- o- re 1 l- 682 AnnalS of Clinical & Laboratory Science, vol. 46, no. 6, 2016 Figure 1. Histological findings of cervix. a Photomicrographs of cervical punch biopsy revealed adenocarcinoma with ir- regular glands (H&E, 100x); b Numerous cervical mucilaginous glands irregularly sized and shaped, infiltrating cervical wall and invading veSSelS (H&E, 100x). Figure 2. Surgically resected tissues. a Greater omentum waS hard with several nodules; b Right ovary waS enlarged (ap- proximate 5 x 4 cm) and cystic; c Lesion specimenS on peritoneum, mesentery, and bladder wall; d Enlarged uterus and hard cervix. Misdiagnosis analysis of cervical MDA 683 drugs were ineffective. The patient had moderate watery vaginal discharge for 8 months and experienced nausea, vomiting, and occasional abdominal discomfort. Weight loss over 8 months was 3 kg and no bowel movement for 4 days was reported. A hypertrophic but smooth cervix was confirmed and a right adnexal mass of ~7 cmx4 was palpable with poor mobility, fine limits, and mild tenderness. A left attachment area was thickening with mild tenderness. There waswe lo r abdominal tenderness and muscular tension. Transvaginal sonography showed multiple cervical internal cystic foci resembling a honey- comb, right adnexa with multiple septations sans echo cystic mass (6.7 cm x 4.1 cm) and a left adnexa with Figure 3. Pelvic CT of soft tissue maSS located in the right multiple septations sans echo cystic mass (2.5 cm x 1.6 side of pelvis with low density lesion necrosis, unclear boundary between the lesion and surrounding intestine. cm). HPV infection was negative. ThinPrep cytology re- vealed moderate inflammation, but no intraepithelial or malignant lesions. CA125 was normal, though CA199
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