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Bulky mesonephric adenocarcinoma of the uterine cervix treated with neoadjuvant chemotherapy and radical surgery: report of the first case

Abstract

Aims and background

Malignant mesonephric adenocarcinoma of the uterine cervix is a rare occurrence with few cases described in the literature. Although surgery seems to be effective in the treatment of early-stage tumor, no cases describing outcomes of locally advanced stage are available.

Methods

We report the first case of a patient with International Federation of Obstetrics and Gynecologists stage IIB mesonephric adenocarcinoma undergoing neoadjuvant chemotherapy and radical surgery.

Conclusions

Despite the inherent limitation of a single description of a case, our experience supports the utilization of neoadjuvant chemotherapy in patients with malignant mesonephric adenocarcinoma of the uterine cervix. Further prospective multi-institutional studies are needed.

Tumori 2016; 102(Suppl. 2): e82 - e83

Article Type: CASE REPORT

DOI:10.5301/tj.5000386

Authors

Antonino Ditto, Fabio Martinelli, Giorgio Bogani, Maria L. Gasparri, Violante Di Donato, Biagio Paolini, Maria L. Carcangiu, Domenica Lorusso, Francesco Raspagliesi

Article History

Disclosures

Financial support: None.
Conflict of interest: None.

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Introduction

Malignant mesonephric adenocarcinoma of the uterine cervix represents an uncommon malignancy, arising from the vestigial remnants of the Wolffian ducts (mesonephric remnants) (1, 2). To date, fewer than 30 cases have been described in the medical literature (3). Owing to its rarity, several biological and clinical features are unclear. Hence, no mature data on different therapeutic strategies exist and patients are subjected to a great variety of treatments. Although surgery seems to be the mainstay of treatment for early stage of disease, no mature data supporting the efficacy of chemotherapy or radiotherapy in locally advanced stage are available. A lesson learned from typical carcinoma of the uterine cervix (i.e., squamous cell and adenocarcinoma) suggested that the administration of neoadjuvant chemotherapy (NACT) might be useful to reduce tumor diameter and improve patient outcomes (4). However, no case describing this approach in the setting of mesonephric adenocarcinoma has been described. We present the first case of mesonephric ­adenocarcinoma of the uterine cervix managed with NACT followed by radical surgery.

Case report

A 51-year-old nulliparous woman was diagnosed with a bulky (about 5-6 cm) cervical lesion involving the vagina due to the occurrence of abnormal uterine bleeding. Her medical history was unremarkable. Cervical biopsy showed the presence of a well-differentiated adenocarcinoma. The patient was staged as IIB cervical cancer according to the International Federation of Obstetrics and Gynecologists (FIGO) staging system (4). Nuclear magnetic resonance imaging (MRI) showed a 6 × 5.3 lesion involving the uterine cervix and the posterior vaginal wall. The patient underwent 3 cycles of NACT with cisplatin-adriamycin-paclitaxel. Clinical and radiologic (MRI) examinations showed partial response, with maximum tumor diameters of 3.8 cm. The patient underwent open abdominal nerve-sparing radical hysterectomy (type C1 according to the Querleu and Morrow classification), bilateral salpingo-oophorectomy, and pelvic lymphadenectomy. Histologic examination showed a poor differentiated FIGO grade 3 mesonephric adenocarcinoma of the uterine cervix. Histologically, mesonephric adenocarcinoma exhibits a variety of patterns of growth, as in this case. This endocervical tumor is mostly composed of glands, many of them with intraluminal small villous papillae, lined by one or more layers of columnar cells. In other areas, a tubular pattern, characterized by back-to-back, small, round, uniform tubules, lined by cuboidal cells, predominates. Sometimes a densely eosinophilic secretion is seen in the glandular lumens simulating non-neoplastic mesonephric remnants. A minor component with a so-called retiform pattern is seen. This is characterized by elongated, slit-like branching tubules lined by one layer of cuboidal cells. A focal spindle cell component was also present. Figure 1 displays histologic features. Lymph vascular space invasion was identified. Moreover, tumor dissemination was detected in the vaginal stroma. No lymph node metastasis was detected among 57 nodes yielded. External beam radiotherapy and vaginal brachytherapy were delivered. The patient was disease-free at 6-month follow-up.

Histologic features of malignant mesonephric adenocarcinoma of the uterine cervix: (A) retiform pattern; (B) spindle cell component among small neoplastic tubules; (C) tubular pattern with dense eosinophilic intraluminal secretion; (D) glandular pattern with small villous papillae resembling endometrioid carcinoma.

Discussion

Mesonephric adenocarcinoma of the uterine cervix is rare, with fewer than 30 cases described in the medical literature. Mesonephric adenocarcinoma represents the malignant transformation of the mesonephric ducts. Generally, mesonephric remnants are located deep to endocervical glands (1). Pathologic transformation of mesonephric remnants includes both benign (i.e., hyperplasia) and carcinoma. While, generally, hyperplasic lesions are incidental findings, mesonephric carcinoma tends to form mass lesions (1).

As aforementioned, no mature data suggesting the best beneficial therapeutic strategies are available. Few authors suggested that radical surgery achieves oncologic control in patients with early stage of disease (3, 5). However, data on patients diagnosed with bulky lesion are scant. In 2006, Yap et al (3) reported a case of mesonephric adenocarcinoma of the uterine cervix and reviewed the available literature. Silver et al (5) reported 11 cases of mesonephric adenocarcinoma of the uterine cervix. Among 11 patients, only 1 patient had locally advanced stage (stage IIB) mesonephric adenocarcinoma. In this case, the patient was treated with radiotherapy and developed a pelvic recurrence after 2.2 years (5). This paucity of data reflects the need to report larger experiences on the management of this malignancy.

In conclusion, we report the first case of bulky mesonephric adenocarcinoma of the uterine cervix treated with NACT and radical surgery. In our experience, NACT was effective in reducing tumor diameter, thus allowing radical resection of the tumor. Further prospective multi-institutional experiences are warranted in order to improve outcomes of patients with rare neoplasms.

Disclosures

Financial support: None.
Conflict of interest: None.
References
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  • 3. Yap OW Hendrickson MR Teng NN Kapp DS Mesonephric adenocarcinoma of the cervix: a case report and review of the literature. Gynecol Oncol 2006 103 1155 1158 Google Scholar
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Authors

Affiliations

  • Department of Gynecologic Oncology, IRCCS National Cancer Institute, Milan - Italy
  • Department of Pathology, IRCCS National Cancer Institute, Milan - Italy

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