Dataset Information

Neoadjuvant chemotherapy followed by radical vulvectomy for adenoid cystic carcinoma of Bartholin's gland: a case report.

ABSTRACT:

Background

Adenoid cystic carcinoma (ACC) of the Bartholin's gland is a rare cancer of the female genital tract for which there is no consensus on treatment. As there are no current reports on neoadjuvant chemotherapy followed by surgery for this disease, our case explores the effectiveness of neoadjuvant chemotherapy.

Case description

We report a case of ACC of Bartholin's gland. The patient is a 69-year-old woman with a left vulvar mass. Pelvic magnetic resonance imaging (MRI) showed a left perineal occupying lesion with indistinct boundaries to the surrounding tissues. The patient received two cycles of neoadjuvant chemotherapy with a regimen of paclitaxel combined with cisplatin and the mass was significantly reduced. Subsequently, she received a radical vulvectomy and left inguinal lymph node dissection. The operation was successful, and the patient was treated with simultaneous radiotherapy and chemotherapy after the surgery. The patient is currently 14 months postoperative and reports no significant pain with normal urination and defecation. The pelvic MRI was reviewed regularly, and there was no sign of recurrence.

Conclusions

After neoadjuvant chemotherapy, the extent of the lesion was significantly reduced, which reduced the difficulty of surgery, thus reducing surgical complications and enabling complete resection of the tumor tissue. Based on the treatment of this patient, we speculate that preoperative neoadjuvant chemotherapy may be a better choice for patients with extensive and fixed lesions.

SUBMITTER: Gao Y

PROVIDER: S-EPMC9641131 | biostudies-literature | 2022 Oct

REPOSITORIES: biostudies-literature

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Publications

Neoadjuvant chemotherapy followed by radical vulvectomy for adenoid cystic carcinoma of Bartholin's gland: a case report.

Gao Yuewen Y Li Hongqi H Li Changzhong C

Translational cancer research 20221001 10

<h4>Background</h4>Adenoid cystic carcinoma (ACC) of the Bartholin's gland is a rare cancer of the female genital tract for which there is no consensus on treatment. As there are no current reports on neoadjuvant chemotherapy followed by surgery for this disease, our case explores the effectiveness of neoadjuvant chemotherapy.<h4>Case description</h4>We report a case of ACC of Bartholin's gland. The patient is a 69-year-old woman with a left vulvar mass. Pelvic magnetic resonance imaging (MRI) s ...[more]

PMID: 36388019

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Project description:PurposeTo report the therapeutic efficacy of integrating neoadjuvant chemotherapy with conventional bimodal therapies for lacrimal gland adenoid cystic carcinoma by providing an additional 8 years of follow-up data on the same cohort of patients whose cumulative 10-year disease-free survival outcomes were reported in 2013.DesignNon-randomized, retrospective, interventional case series.MethodsNineteen consecutive patients treated with neoadjuvant intra-arterial cytoreductive chemotherapy (IACC), orbital exenteration, chemoradiotherapy, and adjuvant intravenous chemotherapy at a single institution were included. Analyses were undertaken of locoregional recurrences and distant metastases, disease-free survival time, TNM tumor stage at presentation, response to IACC, and prognostic impact of positive resection margins. The main outcome measures were overall survival, disease-free survival, disease relapse, positive tumor resection margins, and tumor stage at presentation.ResultsEight patients with an intact lacrimal artery (group 1), 7 with AJCC stage T4a-c, had significantly better overall survival (87.5% versus 14.3% at 15 years), disease-specific mortality, and recurrences (all < .001, log-rank test) than prior conventionally treated patients from the Bascom Palmer Eye Institute. Group 1 was superior to group 2, patients lacking an intact lacrimal artery, concerning overall survival (P = .042) and recurrence (P = .017), but with no significant difference in disease-specific mortality (P = .23). Group 2 was associated with a significantly lower cause-specific mortality than the institutional comparator group (P = .039). Prior tumor resection with lateral wall osteotomy and failure to adhere to all protocol elements were adverse prognostic factors for suboptimal outcomes. Positive tumor margins increased the risk of all-cause mortality 4.1 times (P = .036, stratified Cox proportional hazards regression) and disease-specific mortality 8.0 times (P = .043, stratified Cox proportional hazards regression) than a patient with negative margins.ConclusionsExtended follow-up supplemented with AJCC staging data supports neoadjuvant IACC as an integral component of a trimodal treatment strategy in patients with an intact lacrimal artery. Protocol elements implemented as designed appear to have improved overall survival and decreased disease relapse in this cohort. This extended long-term IACC dataset suggests that a critical bar of at least 15 years of follow-up is appropriate for assessing the efficacy of current conventional and future globe-sparing bimodal therapies.

Project description:Adenoid cystic carcinoma (ACC) is a slow growing, but relentless cancer. Due to its rarity and lack of understanding of its molecular etiology, no standard chemotherapy for ACC currently exists and many patients suffer from recurrent and/or metastatic disease. As such, development of safe and effective therapies is imperative. To describe and summarize existing clinical trial studies and preclinical discoveries, we surveyed the PubMed on developmental therapeutics for ACC. Objective response rates to monotherapy with cytotoxic agents were approximately 10% with cisplatin, 5-FU, gemcitabine, mitoxantrone, epirubicin, vinorelbine and paclitaxel. The most studied combination therapies were cyclophosphamide-doxorubicin-cisplatin (CAP) and cisplatin-vinorelbine, with an objective response rate of 18-31%. Among molecularly targeted drugs, the most studied drugs are inhibitors targeting the vascular endothelial growth factor receptor (VEGFR) to inhibit tumor angiogenesis. Among those, lenvatinib and axitinib showed a relatively high objective response rate of 11-16% and 9-17%, respectively. Given high recurrence rates and chemoresistance of ACC, treatments targeting cancer stem cells (CSC), which function as tumor-initiating cells and drive chemoresistance, may be particularly valuable. CSC have been shown to be targetable via MYB, Notch1, p53 and epigenetic mechanisms. Myb overexpression is characteristic in ACC but was previously thought to present a difficult target due to its nature as a transcription factor. However, due to the development Myb-targeted inhibitors and an ongoing clinical trial of MYB-targeted cancer vaccine therapy, MYB is becoming an increasingly attractive therapeutic target. Drugs targeting NOTCH signaling demonstrated 5-17% response rate in phase I clinical trials. Within the field of epigenetics, treatment with PRMT5 inhibitors has shown 21% partial response rate in phase I clinical trial. Immunotherapies, such as PD-1 inhibitors, are also associated with CSC, but have not been effective against ACC. However, clinical trials of cancer vaccine therapies are actively being conducted. In addition to conventional chemotherapies and inhibitors of angiogenesis, the emergence of new therapies such as immunotherapy and those targeting cancer stemness is expected to bring clinical benefits to patients in the future.

Dataset Information

Neoadjuvant chemotherapy followed by radical vulvectomy for adenoid cystic carcinoma of Bartholin's gland: a case report.

Background

Case description

Conclusions

Publications

Neoadjuvant chemotherapy followed by radical vulvectomy for adenoid cystic carcinoma of Bartholin's gland: a case report.

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