Adenoid cystic carcinoma of Bartholin's gland: a case report

Adenoid cystic carcinoma is an uncommon histological type of the already rare carcinoma of Bartholin's gland with 51 cases described in the literature. We present a case of a 66-year-old woman who was admitted with severe pelvic pain. In an examination under anesthesia a 10x5 cm apparently fixed mass at the left vaginal wall originating from the area of Bartholin's gland was found. and biopsy indicated carcinoma. The patient underwent wide local excision. Although clinically inoperable, the tumor did not infiltrate the bony pelvis and no evidence of metastasis was found. Pathology examination revealed adenoid cystic carcinoma of Bartholin's gland. PCR did not detect human papillomavirus DNA in the specimen. The patient has been treated with adjuvant radiotherapy, and is alive with no evidence of disease after ten months.     

Adenoid cystic carcinoma of the Bartholin’s gland: a rare tumor unmarked by persistent vulvar pain in a postmenopausal women

INTRODUCTION: Adenoid cystic carcinoma (ACC) of the Bartholin's gland is a rare malignancy characterized by slow growth, local invasion and perineural infiltration. CASE: A 64-year-old postmenopausal woman presented with persistent vulvar pain. Local examination revealed a 2 x 2 cm painful vulvar nodule. Nodule was excised and ACC originating in the Bartholin's gland with positive resection margin was determined in pathological examination. We performed hemivulvectomy and ipsilateral inguinal lymph node dissection. Pathology showed that the resection margins were free of tumor and there was no lymph node metastasis. DISCUSSION: In cases of persistent vulvar pain without swelling, complete vulvovaginal evaluation should be done to prevent delay in diagnosis of Bartholin's gland cancer. Treatment modality must be tailored to each patient, though there is no consensus on the treatment.     

Adenoid cystic carcinoma of Bartholin gland

Five cases of adenoid cystic carcinoma of the Bartholin gland, a rare vulvar tumor, are reviewed with respect to clinical and pathological characteristics. Histologic transition from normal Bartholin gland to adenoid cystic carcinoma was evident in two cases. Two patients developed the tumor in association with pregnancy. Local recurrences are common and may precede distant metastases, pulmonary being the most common. Patients with repetitive local recurrence or pulmonary metastases may have slowly progressive disease and survive for many years. This is reflected in the disparity between the progression-free interval and survival curves. The recommended primary treatment is wide local excision, obtaining clear margins, and an ipsilateral inguinal lymphadenectomy.     

Adenoid cystic carcinoma of the Bartholin’s gland:a case report and review of the literature

Adenoid cystic carcinoma(ACC) of the Bartholin's gland is a rare malignant tumor of vulvar which is characterized by slow growth,local invasion and perineural infiltration.The survival rates for 10 years range from 50% to 100%.The disease free interval for 10 years range from 33% to 38%.Currently,there is no consensus on the treatment of ACC of the Bartholin's gland.Primary surgery includes wide local excision or radical vulvectomy with or without lymph node dissection.Adjuvant radiotherapy and chemotherapy are advocated for the treatment of this cancer.Work is still needed to identify an effective systemic therapy.     

Primary adenoid cystic carcinoma of sweat glands in vulva: report of an unusual case and review of the literature

Adenoid cystic carcinoma arising from the vulvar sweat glands is a rare malignancy of the female genital tract. We report a case of adenoid cystic carcinoma of sweat glands occurring in the left labia majora of a 52-year-old female patient. The patient underwent radical hemivulvectomy and left inguinal lymph node dissection with negative surgical margins and negative inguinal lymph node metastasis. Then, four episodes of combined chemotherapy without further radiotherapy were given. However, the tumor recurred after 3?months. Currently, the patient has been followed up for 2?years with no distant metastasis. According to our experience, although the tumor has a high tendency of local recurrence after resection, an acceptable survival time of the patient can be achieved with primary surgery.     

Adenoid cystic carcinoma of Bartholin's gland: a review of the literature and report of a patient with widespread metastases to the bone

Flam F, Larson B. Adenoid cystic carcinoma of Bartholin's gland: are view of the literature and report of a patient with widespread metastases to the bone. Int J Gynecol Cancer 1997; 7 :458–460.
We report the case of a 41-year-old woman with adenoid cystic carcinoma of Bartholin's gland. Approximately 50 cases of this tumor have been described in the world literature. Local recurrences are common in this disease as well as distant metastases, primarily to the lungs. On the basis of earlier reports this patient was treated with surgery and external radiation. The patient is unique in that she developed widespread bone metastases in the absence of local recurrence.     

First detection of sentinel node in adenocarcinoma of Bartholin's gland

We report the first case of detection of sentinel node in a 54 year-old woman presenting an adenocarcinoma of Bartholin's gland. Primary carcinoma of Bartholin's gland is rare and represents 2-7% of vulvar malignant lesions; this could explain the lack of consensus about treatment. The best attitude could be vulvectomy and inguinal lymphadenectomy. Pelvic lymphadenectomy is not required when no pelvic sentinel node is observed or when no metastatic inguinal node can be detected.     

Adenoid cystic carcinoma of the Bartholin's gland: report of two cases and review of the literature

BACKGROUND: Adenoid cystic carcinoma (ACC) of the Bartholin's gland is a rare malignancy of the female genital tract and there have been 62 cases of ACC of the Bartholin's gland in the literature. CASES: Two cases of ACC of the Bartholin's gland are reported. CONCLUSION: There is no consensus on optimal treatment of ACC of the Bartholin's gland. Most commonly, wide local excision and radical vulvectomy with or without lymph node dissection, are performed. More long-term follow up is recommended to evaluate optimal primary treatment and roles of radiotherapy and chemotherapy because ACC of the Bartholin's gland recurs and metastasizes long after primary treatment.     

Adenoid cystic carcinoma of Bartholin's gland: a clinical, immunohistochemical and ultrastructural study of a case with regard to its histogenesis.

Adenoid cystic carcinoma of Bartholin's gland is a rare tumor, of which 45 cases including the present one have been reported in the literature. Although the histogenesis of this tumor is still controversial, the tumor is suspected to be of myoepithelial origin, and most studies have been performed on the salivary glands with few reports concerning Bartholin's gland. We report a case of adenoid cystic carcinoma of Bartholin's gland with regard to its histogenesis. A typical tumor nest had three main components: true luminary structures containing sialomucins, solid monotonous cells of a myoepithelial nature, and numerous pseudocysts containing abundant proteoglycans and basal membrane-like materials. From these results we speculate that reserve cells located in the intercalated small ducts of Bartholin's gland may have the potential to differentiate into two cell types, myoepithelial and luminary cells, the former forming the pseudocysts.     

Adenoid cystic carcinoma of Bartholin's gland: a report of five new cases treated with surgery and radiotherapy

Adenoid cystic carcinoma (ACC) is a slow-growing tumor with a marked tendency for perineural and local invasion. The neoplasm occurs more frequently in the head and neck region and only 39 previously described cases of adenoid cystic carcinoma of Bartholin's gland are found in the world literature. Most authors advocate surgery as the primary treatment. Several cytostatic drugs have been tried but the results have been poor. The benefit of radiotherapy in the treatment of this type of tumor is not yet established. This is a report of five new cases all treated with surgery and postoperative radiotherapy. Three patients had histopathologically proven evidence of residual disease after surgery and were subsequently treated with radiotherapy. They are now living without evidence of disease after 28, 51, and 138 months.     

Liver metastasis in a case of adenoid cystic carcinoma of the Bartholin’s gland: a rare presentation

Introduction  Adenoid cystic carcinoma of the Bartholin’s gland accounts for a minority of all Bartholin’s gland malignancies. Although local recurrences commonly occur distant metastasis is rare. Case report  We present the first reported case of Adenoid cystic carcinoma of the Bartholin’s gland with metastasis to the liver. Discussion  There is no consensus on the treatment of adenoid cystic carcinoma of the Bartholin’s gland. Wide local excision and/or vulvectomy and radiotherapy are advocated for treatment of this cancer. There is not much data on treatment of metastasis and it varies according to site. An erratum to this article can be found at     

The influence of some prognostic factors on the clinical outcome of patients with squamous cell carcinoma of the vulva.

The present paper examined the influence of patient age, surgical T stage, tumor size, tumor differentiation and lymphnodal status on the clinical outcome of 29 patients with primary vulvar squamous cell carcinoma treated with radical surgery. Eighteen patients underwent radical vulvectomy with bilateral inguinal-femoral lymphadenectomy alone; 10 patients had additional bilateral pelvic lymphadenectomy; another patient had additional bilateral pelvic lymphadenectomy and anterior pelvic exenteration for a carcinoma of the clitoris involving the urethra. Nine patients developed relapsing disease; the site of recurrence was local in 4 patients, inguinal in 2, both local and inguinal in one patient, pelvic in one, both pelvic and distant in one. Eight recurrences occurred within 24 months from surgery; another patient developed an inguinal recurrence 45 months after operation. The actuarial 5-year disease-free survival rates were as follows: 64% for patients younger than 70 years and 63% for patients 70 years of age or older (p = not significant); 79% for patients with surgical T1-T2 stage disease and 30% for those with surgical T3 stage disease (p = 0.01); 88% for patients with tumor size less than 3 cm and 31% for those with tumor size greater than 3 cm (p less than 0.001); 66% for patients with well or moderately differentiated tumor and 51% for those with poorly differentiated tumor (p = not significant); 82% for patients with negative groin lymph nodes and 39% for those with positive groin lymph nodes (p less than 0.001).(ABSTRACT TRUNCATED AT 250 WORDS)     

Skull metastasis in primary vulvar adenocarcinoma of the Bartholin's gland: a case report

BACKGROUND: Metastatic bone involvement in vulvar squamous carcinoma is very rare. Here, we present a case of primary adenocarcinoma of the Bartholin's gland giving rise to isolated skull metastasis. CASE: A 74-year-old woman was referred with a diagnosis of primary vulvar adenocarcinoma of the right labium minus. A right hemivulvectomy was performed with ipsilateral inguinofemoral lymphadenectomy, revealing a primary adenocarcinoma of the Bartholin's gland and metastatic involvement in 6 out of 13 lymphnodes. During chemotherapy administration, the patient started complaining of a worsening of right hypoacousis. CT scan and MRI of the brain showed the presence of a metastatic lesion on the right side of the skullbone. The diagnosis was metastatic poorly differentiated adenocarcinoma. CONCLUSION: In adenocarcinoma of the Bartholin's gland at an advanced stage of disease, a bone scan or survey in the preoperative as well as follow up staging procedures could be useful.     

外阴癌淋巴结切除适应证和争议

外阴局部广泛切除术+腹股沟淋巴结切除术是目前外阴癌的基本手术方式。FIGO和NCCN指南均推荐FIGOⅠA期可不行腹股沟淋巴结切除术,所有ⅠB期或Ⅱ期患者,应该行腹股沟淋巴结切除术。晚期外阴癌在确定总体治疗方案前,应先明确腹股沟淋巴结状态,再确定后续处理方案。如果术前未发现可疑转移淋巴结,行双侧腹股沟、股淋巴结切除术;术前已明确淋巴结阳性者,建议仅切除肿大的淋巴结,术后给予腹股沟和盆腔放疗,最好避免系统性淋巴结切除术。在有关淋巴结切除的争议中,切除腹股沟、股淋巴结及采用三切口腹股沟横切口技术、保留大隐静脉等被大多数学者认可;但对于靠近中线但不侵犯中线的病灶是否可不切除双侧腹股沟淋巴结及外阴黑色素瘤、前庭大腺癌等少见病理类型的淋巴结切除指征尚有争议。     

Merkel cell carcinoma of the Bartholin's gland

BACKGROUND: There are a few cases of "small cell neuroendocrine carcinoma" of the Bartholin's gland as well as Merkel cell carcinoma of the vulva reported in the gynecologic oncology literature, most with aggressive clinical behavior. Merkel cells were originally described in the epidermis with neuroendocrine properties. Merkel cell carcinoma is different from the neuroendocrine small cell (oat cell) tumor arising in the lung and other internal organs based on morphology and immunohistochemistry. CASE: A 49-year-old female was diagnosed with a FIGO stage I Merkel cell carcinoma of the Bartholin's gland. The tumor originated from the duct and stained with endocrine markers and cytokeratin 7 and 20. She was treated with radical wide local excision and bilateral inguinal lymph node dissection followed by radiation therapy to the pelvis, perineum, vulva, and inguinal regions. Two years after the diagnosis, she is alive with no evidence of recurrent disease. CONCLUSION: This is a case of Merkel cell carcinoma of the Bartholin's gland discovered in the early stage with a survival of 2 years following diagnosis and treatment with no evidence of disease recurrence.     

Cloacogenic adenocarcinoma of the vulva presenting as recurrent Bartholin's gland infection.

Primary adenocarcinoma of the vulva of the cloacogenic type is extremely rare. We report the case of a 49-year-old patient with a 6-year history of recurrent left Bartholin's gland infection and a 1-year history of an ulcerated tumor on the similar lower aspect of the left labium majus. Fine needle biopsy revealed adenocarcinoma and wide local excision with bilateral superficial inguinal lymph node dissection was performed. The patient was well and disease-free at 24 months after the operation. This case provides a good example of why an early biopsy should be performed for any persistent anogenital lesion. Although the reported data on cloacogenic adenocarcinoma are limited, a wide local excision appears to be the most widely used treatment strategy for any vulvar cancer of 2 cm or less in diameter.     

Adenoid cystic carcinoma of Bartholin's gland: a case report

OBJECTIVE: A case of adenoid cystic carcinoma (ACC) of the Bartholin's gland in a 34-year-old woman with unusual presentation and early recurrence is reported. METHODS: Clinical and histologie features were recorded. Immunohistochemical stains and cell-cycle analysis by flow cytometry technique on paraffin-embedded tumor tissue were performed. RESULTS: The tumor presented as a painful nodule in the episiotomy scar three months after delivery. Initial treatment included only wide local excision. Six months later local recurrence occurred despite clear surgical margins. Histologically a predominant "classic" cribriform growth pattern was identified. Immunoreactivity in tumor cells supported dual epithelial-myoepithelial differentiation. Estrogen and progesterone receptors were negative. The DNA histogram revealed a diploid stemline and a low S-phase fraction. CONCLUSION: ACC of the Bartholin's gland is a rare malignant tumor with great propensity for local recurrence. The optimal therapeutic approach has not been established due to the lack of well-defined prognostic parameters.     

Advanced primary carcinoma of the Bartholin gland: report of 18 patients

Conservative surgery plus radiotherapy for vulvar cancer has been established as a therapeutic alternative to extensive radical surgery and produces a similar cumulative 5-year survival. We retrospectively analyzed the cases of 18 patients with advanced primary carcinoma of the Bartholin gland treated with wide local excision (WLE) or radical vulvectomy and lymphadenectomy followed by radiotherapy (RT) at the University of Texas M. D. Anderson Cancer Center from January 1978 through December 1990. All patients have been observed for a minimum of 7 months (maximum follow-up, 15 years; median follow-up, 9 years). Of the 18 patients, 7 were treated with wide local excision (WLE) followed by radiation therapy (RT) (Group 1), 9 had radical vulvectomy (RV) followed by RT to the vulvar and inguinal-femoral and pelvic node areas (Group II), and 2 were treated with RT alone after biopsy of the tumor (Group III). The 5-year disease-free survival rates were 86%, 78%, and 50% for groups I, II, and III, respectively, and 83% for the whole group. Of 2 patients treated with RT alone, one lived for 6 years with no evidence of disease, and the other lived for 20 months. The rate of local tumor control was 100% for all three treatment groups. There were no significant differences among the treatment groups in rate of primary tumor control or 5-year disease-free survival rate (p=0.1300). The present study demonstrated WLE followed by RT is the best treatment for advanced primary carcinoma of the Bartholin gland. Less radical surgery plus RT produces good long-term survival and has fewer complications.     

Sentinel lymph node identification in a primary ductal carcinoma arising in the vulva

Primary or metastatic breast-like carcinoma of the vulva is a rare event. Because of the similarity with breast ductal carcinoma, we think that the same principles used for treatment of orthotopic breast cancer can be applied, as well as the use of sentinel lymph node technique, which is widely accepted in the management of early-stage breast cancer. We report a 49-old-year postmenopausal woman who was referred to our institution after small biopsy of a 3.5- x 3-cm right vulvar tumor. Histopathologically, infiltration of the vulvar dermis by a ductal carcinoma of mammary gland type was reported. At operation, the sentinel node technique revealed two sentinel nodes in the right inguinal area. Although these nodes proved negative for malignancy, the patient underwent wide local excision of tumor and complete ipsilateral inguinofemoral lymphadenectomy. The remaining excised nodes were negative. Surgical specimen proved estrogen- and progesterone-positive receptors, the reason for which the patient received tamoxifen adjuvant therapy. This report represents the first case in the world literature of primary breast carcinoma arising in the vulva in which sentinel lymph node identification has been possible. Because of the rarity of this condition, the pathologic similarity of this tumor along with currently accepted guidelines for the management of breast cancer supports the possibility of local excision and sentinel lymph node identification as a possible alternative to inguinofemoral lymphadenectomy.     

Conservative surgical management of superficially invasive stage I vulvar carcinoma

Fifty patients with stage I squamous cell carcinoma of the vulva were treated by means of wide local excision and either unilateral or bilateral superficial inguinal lymphadenectomy. Depth of invasion per se was not an exclusionary criterion; however, 36 of 37 patients for whom depth of invasion could be assessed had tumors invasive to a maximum depth of 5 mm. Factors investigated included recurrences and survival in addition to the early and delayed morbidity associated with this operative approach. Recurrent intraepithelial or minimally invasive cancer was documented in six patients, five of whom were treated successfully by a subsequent wide local excision following initial surgery. Only one patient died of recurrent carcinoma 16 months following surgery. The morbidity with this operation was appreciably less than that generally reported with more extensive operations commonly employed in the management of vulvar cancer and is recommended for management of patients with early invasive disease.