Adenoid cystic carcinoma of the Bartholin's gland is underpinned by MYB- and MYBL1- rearrangements.

OBJECTIVE: Adenoid cystic carcinoma (AdCC) of the Bartholin's gland (AdCC-BG) is a very rare gynecologic vulvar malignancy. AdCC-BGs are slow-growing but locally aggressive and are associated with high recurrence rates. Here we sought to characterize the molecular underpinning of AdCC-BGs. METHODS: AdCC-BGs (n = 6) were subjected to a combination of RNA-sequencing, targeted DNA-sequencing, reverse-transcription PCR, fluorescence in situ hybridization (FISH) and MYB immunohistochemistry (IHC). Clinicopathologic variables, somatic mutations, copy number alterations and chimeric transcripts were assessed. RESULTS: All six AdCC-BGs were biphasic, composed of ductal and myoepithelial cells. Akin to salivary gland and breast AdCCs, three AdCC-BGs had the MYB::NFIB fusion gene with varying breakpoints, all of which were associated with MYB overexpression by IHC. Two AdCC-BGs were underpinned by MYBL1 fusion genes with different gene partners, including MYBL1::RAD51B and MYBL1::EWSR1 gene fusions, and showed MYB protein expression. Although the final AdCC-BG studied had MYB protein overexpression, no gene fusion was identified. AdCC-BGs harbored few additional somatic genetic alterations, and only few mutations in cancer-related genes were identified, including GNAQ, GNAS, KDM6A, AKT1 and BCL2, none of which were recurrent. Two AdCC-BGs, both with a MYB::NFIB fusion gene, developed metastatic disease. CONCLUSIONS: AdCC-BGs constitute a convergent phenotype, whereby activation of MYB or MYBL1 can be driven by the MYB::NFIB fusion gene or MYBL1 rearrangements. Our observations further support the notion that AdCCs, irrespective of organ site, constitute a genotypic-phenotypic correlation. Assessment of MYB or MYBL1 rearrangements may be used as an ancillary marker for the diagnosis of AdCC-BGs.

Bilateral bartholin's gland abscesses in a 4-year-old girl with vitamin a deficiency: a case report.

BACKGROUND: A Bartholin's gland abscess is one of the most common infections in women of reproductive age. Although Bartholin's gland abscesses have been reported in prepubertal children, they are rarer in prepubertal children than in adults. Herein, we report a case of bilateral Bartholin's gland abscesses in a 4-year-old girl with vitamin A deficiency. CASE PRESENTATION: A 4-year-old girl diagnosed with autism spectrum disorder was admitted to the hospital for close examination and treatment because of persistent fever and malaise. The child was a marked fussy eater and was diagnosed with corneal ulceration and night blindness secondary to vitamin A deficiency. Both of the patient's labia were swollen, and a diagnosis of a bilateral Bartholin's gland abscess was made using computed tomography. Incisional drainage was performed under general anesthesia. The patient's postoperative course was uneventful, and she was discharged from the hospital on day 8 after the surgery. During hospitalization, attempts were made to correct the vitamin deficiency by adding nutritional supplements to the diet. Three months after the surgery, no recurrence of abscesses was noted. CONCLUSIONS: Decreased immunocompetence and mucosal barrier function due to vitamin A deficiency is thought to be the underlying cause of Bartholin's gland abscesses. Although prepubertal Bartholin's gland abscesses have been reported, they are rare. To the best of our knowledge, no reports of bilateral Bartholin's gland abscesses potentially caused by vitamin A deficiency have been reported. When prepubertal girls present with Bartholin's gland abscesses, the presence of immunodeficiency due to vitamin or trace element deficiency should also be considered.

Comparison of Word Catheter and Marsupialization in the Management of Bartholin's Glands: A Systematic Review and Meta-Analysis.

OBJECTIVE: There is no consensus on the best option for the management of Bartholin's gland abscesses. This systematic review and meta-analysis aimed to compare the Word catheter placement and marsupialization methods in the management of women with a cyst or abscess of the Bartholin's glands. DATA SOURCES: The PubMed, Scopus, Embase, Web of Science, and Cochrane Library databases, as well as Google Scholar, were searched to retrieve articles published between January 1990 and July 2023, comparing the Word catheter treatment with marsupialization for women with a cyst or abscess of the Bartholin's glands. STUDY SELECTION: Both observational studies and randomized controlled trials (RCTs) were included in this meta-analysis. DATA EXTRACTION AND SYNTHESIS: After the review, out of 9 relevant articles, only 4 (2 observational studies and 2 RCTs) were included in this meta-analysis, providing the data of 735 patients (396 in the Word catheter group vs. 339 in the marsupialization group). The data was extracted from the selected articles, using a data extraction form. Comparison of the Word catheter and marsupialization methods suggested that the risk of recurrence was approximately 7.6% in the Word catheter group and 9.4% in the marsupialization group. The findings indicated no significant difference in the recurrence of cysts or abscesses when comparing the Word catheter treatment with marsupialization (odds ratio = 0.99, 95% confidence interval: 0.29-3.43, P = 0.990, I2 = 77%). CONCLUSION: This meta-analysis found no significant difference in the rate of recurrence between the Word catheter and marsupialization methods. In general, marsupialization and Word catheter placement in the treatment of Bartholinitis seem to be equally effective.

Bartholin's gland cyst caused by Sneathia amnii: a case report.

BACKGROUND: Sneathia amnii is a conditional pathogen of the female genital tract that is involved in bacterial vaginosis and poor reproductive and perinatal outcomes. Few studies have reported subcutaneous cysts following invasive infection caused by S amnii. CASE PRESENTATION: Here we report the case of a 27-year-old woman who presented with Bartholin's gland cyst due to S amnii infection, and was successfully treated with surgical neostomy and antibiotic agents. The isolate was gram-negative, bacillary, anaerobic, and was identified by polymerase chain reaction (PCR) amplification of the 16 S rRNA. CONCLUSIONS: S amni is an important but underappreciated pathogen that needs further investigation. This report describes the microbial and pathogenic characteristics of S amnii and is expected to provide a valuable reference in obstetric and gynecologic clinical practice.

Adenoid Cystic Carcinoma (ACC) of the Bartholin's gland misdiagnosed for three times: a case report.

BACKGROUND: Adenoid cystic carcinoma (ACC) in Bartholin's gland is an uncommon malignant tumor. These tumors have a vague clinical feature, so they are diagnosed late and discovered at a high-level stage. Our case presented Three Recurrences and Three times Misdiagnosis of Adenoid Cystic Carcinoma (ACC). CASE PRESENTATION: We report a case of adenoid cystic carcinoma arising in Bartholin's gland of a 64-year-old female patient that appeared after three previous vulvar tumors were excised. The patient underwent bilateral radiotherapy which was performed on the perineum. CONCLUSION: ACC of the vulvar sweat glands is prone to misdiagnosis and delay in both diagnosis and treatment. As seen in our case, it was misdiagnosed three times as Chondroid Syringoma. Further studies need to be conducted to better understand the tumor prognosis, and its optimal treatment options.

Acinar Melanosis of the Bartholin Gland: A Report of 3 Cases.

Melanocytic pigmentation occurs in multiple sites in the lower female genital tract, but is rare within benign cysts of the vulva. We report 3 patients with multiloculated cystic lesions of the vulvar vestibule exhibiting prominent melanocytic pigmentation. The current cases differ from a previous report of melanosis in a Bartholin gland cyst in that the population of melanocytes occupies the acinar structures of the gland, rather than a squamous-lined surface. A similar cell population is demonstrated by immunoperoxidase methods in a fourth patient's nonpigmented gland, suggesting that melanin production may arise in a native, rather than metaplastic, cell population.

Enteric Type Bartholin Gland Adenocarcinoma: An Unusual Variant of a Rare Neoplasm.

Vulval cancer is rare. With Bartholin gland carcinomas representing <5% of all vulval carcinomas they present both diagnostic and management challenges. There are a small number of cases in the literature describing Bartholin gland carcinomas with unusual histology which necessitates the need to explore the possibility of metastases from elsewhere. We present a case of a 55-yr-old woman presenting with a vulval lesion within the Bartholin gland. Morphology demonstrated enteric type adenocarcinoma and the immunohistochemistry profile was positive for CK7, CK20, CDX2, CEA, and CA19-9. There was no evidence of an alternative primary cancer and the tumor was excised with negative regional sentinel node assessment. Genotyping showed no detectable mutations in KRAS, BRAF or NRAS suggesting a possible future role for anti-EGFR therapy.

Association of Bartholin cysts and abscesses and sexually transmitted infections.

INTRODUCTION: Bartholin gland cysts or abscesses account for many gynecologic visits in the emergency department (ED). Previous smaller studies have suggested a link between Bartholin cysts/abscesses and sexually transmitted infections (STIs), but few studies have involved the ED. METHODS: We retrospectively identified patients aged 18 years or older seen in 1 ED between January 2012 and March 2017 who had urinalysis and urine culture and/or were tested for gonorrhea, chlamydia, or trichomonas by nucleic acid amplification testing. Univariate and multivariate analyses were used to evaluate associations between Bartholin cysts/abscess and demographics, laboratory findings, and ED diagnoses. RESULTS: Data were collected for 75,000 ED patients; 64 patients had a diagnosis of Bartholin cyst or abscess, 40 of whom were also tested for Neisseria gonorrhoeae or Chlamydia trachomatis. Ten percent of patients with a Bartholin cyst/abscess were infected with N gonorrhoeae, compared with 3% of those without a Bartholin cyst/abscess (P = .008). The rates of C trachomatis and Trichomonas vaginalis infections were 13% and 26%, respectively, among patients with a Bartholin cyst/abscess, compared with 8% and 30%, respectively, among those without a Bartholin cyst/abscess (P > .05 for both). On regression analysis, only increased urobilinogen level (ß, 0.31; odds ratio, 1.36; 95% CI, 1.11-1.66; P = .003) and infection with N gonorrhoeae (ß, 1.69; odds ratio, 5.40; 95% CI, 1.43-20.35; P = .01) were associated with a Bartholin cyst/abscess. CONCLUSIONS: Clinicians in the ED should consider testing patients with a Bartholin cyst/abscess for gonorrhea.

Primary adenocarcinomas of the vulva and related structures: An enigmatic and diverse group of tumors✰.

Approximately half of adenocarcinomas that involve the vulva are secondary, either through direct extension or metastases from elsewhere. Primary vulvar adenocarcinomas are rare and encompass a diverse array of neoplasms that are nominally classified based on the presumed tissue or organ of origin, the tumoral phenotype, or both. In this review, we summarize the clinicopathologic features of adenocarcinomas that originate from the vulva and related structures, including the terminal urethra. Adenocarcinomas of this region encompass lesions that are defined by their primary site (such as adenocarcinomas of the Bartholin gland, which by definition must be in the region of the Bartholin gland), histomorphology and immunophenotype (such as clear cell carcinoma and adenocarcinoma of intestinal [cloacogenic] type), or both (such as adenocarcinoma of skene gland origin, which is associated with that specific organ but which also displays a distinctive phenotype that is similar to the phenotype of high grade prostatic adenocarcinoma). Other types, such as mammary-type adenocarcinomas, are presumed to originate from the putative mammary-like glands of the vulva and display a spectrum of pathologic features that are similar to their mammary counterparts. Similarly, vulvar carcinomas of sweat gland origin are pathologically similar to their counterparts in the non-vulvar skin and include a variety of cutaneous adnexal-type malignancies such as apocrine adenocarcinoma and eccrine adenocarcinoma. Some tumors, such as adenoid cystic carcinoma, may represent a Bartholin gland adenocarcinoma, a carcinoma of sweat gland origin, or a carcinoma arising from extramammary Paget disease (EMPD), depending on the context. Invasive carcinomas of various types have been reported in 7-12.7% of EMPD, and these are likely the most common primary glandular malignancy of the vulva. Occasional vulvar adenocarcinomas have been reported to be HPV-associated, although this association has not been established for the broader group of vulvar adenocarcinomas. Rare adenocarcinomas are not classifiable by the aforementioned nosologic scheme, and are designated as vulvar adenocarcinoma NOS.

Surgical Treatment of Bartholin's Gland Abscess: Is Word Catheter Superior to Marsupialization?

STUDY OBJECTIVE: Bartholin's gland abscess may occur in up to 2% of the women. Surgical drainage using the Word catheter application or marsupialization is the treatment of choice in the management of Bartholin's gland abscess. We aimed to compare the abscess recurrence rates between these 2 surgical methods. DESIGN: A retrospective cohort database study. SETTING: A university-affiliated, high-volume teaching hospital in southern Israel. PATIENTS: All women who were surgically treated for Bartholin's gland abscess. INTERVENTIONS: Different clinical and postoperative characteristics were retrieved from the patients' records. A univariate analysis was conducted, and p <.05 was considered significant. MEASUREMENTS AND MAIN RESULTS: During the study period, 321 women were admitted to our center with Bartholin's gland abscess and were managed surgically. Of these, 215 (67%) were treated using the Word catheter and 106 (33%) by drainage and marsupialization. No differences were found in clinical and microbiologic features between the study groups. In addition, recurrence rates as well as recurrent admissions did not differ significantly. Postoperative complications were similar between the groups. CONCLUSION: Our study reassures that both the Word catheter application and marsupialization are appropriate and safe when treating Bartholin's gland abscess.

Bartholin gland carcinoma: A case report.

Bartholin gland carcinomas are rare types of tumours, accounting for less than 1% among females. This case report is of a 20-years patient in her 16th week of pregnancy who presented with pain and swelling in the left groin for two weeks. Examination showed a well-defined fixed swelling with induration in the left vulva, with an irregular margin and a diameter of about 5 cm, just at the Bartholin's gland site with two palpable ipsilateral lymph nodes. Bartholin's gland abscess was suspected. The decision was to drain the pus under general anaesthesia. During the procedure, a hard mass was felt at the left Bartholin glands region. The incision was extended, which unraveled a 5*5 cm abnormal-looking mass; it was hard and fixed to the adjacent tissues. Histopathology confirmed adenocarcinoma of Bartholin gland in stage III. The oncologist advised pregnancy termination and chemoradiation. Although surgical excision is essential for treating tumours, still, a multimodal approach can prove to be more effective.

Adenoid Cystic Carcinoma of Bartholin's Gland: What Is the Best Approach?

Background and summary: Among all vulvar cancers, primary adenoid cystic carcinoma (ACC) of Bartholin's gland is a very rare tumor characterized by a slow growth, a high local aggressiveness, and a remarkable recurrence rate. Due to its rarity, treatment remains a challenge for oncologists and gynecological surgeons. Key message: The present paper reports clinical, radiological, and histological features of ACC of Bartholin's gland and reviews the literature data on the treatment options with a particular focus on the potential role of particle radiation therapy.

Evaluation of treatments for Bartholin's cyst or abscess: a systematic review.

BACKGROUND: No consensus on the management of symptomatic cysts or abscesses of the Bartholin's gland exists. OBJECTIVES: To assess the effectiveness and safety of surgical interventions for a symptomatic Bartholin's cyst or abscess. SEARCH STRATEGY: We searched bibliographical databases from inception to April 2019. SELECTION CRITERIA: Randomised trials evaluating a surgical intervention for the treatment of a symptomatic Bartholin's cyst or abscess. DATA COLLECTION AND ANALYSIS: Eight trials, reporting data from 699 women, were included. Study characteristics and methodological quality were recorded for each trial. Summary estimates were calculated using random-effects methods. MAIN RESULTS: When considering the recurrence of a symptomatic Bartholin's cyst or abscess, the evidence was consistent with notable effects in either direction (risk ratio [RR] 0.76; 95% confidence interval [CI] 0.41-1.40) when comparing marsupialisation with incision, drainage and insertion of a Word catheter. Limited inference could be made when comparing marsupialisation with incision, drainage and silver nitrate insertion (RR 1.00; 95% CI 0.57-1.75), and incision, drainage and cavity closure (RR 0.25; 95% CI 0.01-4.89). There was limited reporting of secondary outcomes, including haematoma, infectious morbidity and persistent dyspareunia. CONCLUSIONS: Current randomised trial evidence does not support the use of any single surgical intervention for the treatment of a symptomatic cyst or abscess of the Bartholin's gland. PROSPECTIVE REGISTRATION: PROSPERO: International Prospective Register of Systematic Reviews; CRD42018088553. TWEETABLE ABSTRACT: Further research is needed to identify an effective treatment for #Bartholin's cyst or abscess. @jamesmnduffy.

Clitoral leiomyoma in a premenopausal woman: a case report.

BACKGROUND: Extrauterine leiomyomata is an uncommon lesion that can lead to several problems of differential diagnosis, especially when localized in the external genitalia. There are few reports in the English literature and a novel association with Alport's syndrome has been investigated since the 1980s. CASE PRESENTATION: Here, we describe the case of a premenopausal woman who presented with an indolent swelling of the right interlabial fossa that resulted in a Bartholin cyst. In addition to this cyst, a benign leiomyoma of the right side of the clitoris was also found and removed. Our patient refused any further examination, although she was informed that genetic counselling could be organized to rule out an association with Alport's syndrome. CONCLUSIONS: Extrauterine leiomyomata localized in the external genitalia is an uncommon lesion arising from smooth muscle cells around vascular epithelium or erectile tissue. Since an association between extrauterine leiomyomata and Alport's syndrome has been described, genetic testing can be proposed to these patients. Upper intestinal tract symptoms such as dysphagia should prompt a gastroenterological evaluation as an association with an esophageal leiomyomatosis has been described.

Risk factors associated with recurrent referral to the emergency room following surgical treatment of Bartholin's gland abscess.

There is no consensus regarding the best surgical modality for the treatment of Bartholin's gland abscess. The aim of the study was to evaluate the risk factors associated with the recurrent referral of the emergency room (ER) following surgical treatment for a Bartholin's gland abscess. A retrospective cohort study was done. Clinical and microbiological characteristics were retrieved from the patients' hospital records. A univariate analysis was followed using multiple logistic regression model. During the study period, 320 women were managed surgically, of those 54 (37.0%) had had a recurrent referral to the ER. The rate of positive previous cultured abscesses was significantly higher among patients with a recurrent referral to the ER (66.7% vs. 51.3%, p value < .05). The mode of a previous abscess drainage (Word catheterisation or marsupialisation) was not associated with recurrent referral to the ER or with recurrent hospitalisation. The possible association between positive cultures and recurrence warrants re-consideration of routine antimicrobial administration for Bartholin's gland abscess.IMPACT STATEMENTWhat is already known on this subject? A recurrence of a Bartholin's gland abscess following surgical treatment varies greatly and there is no consensus regarding the best surgical modality for treatment. None of the studies have examined a recurrent referral to the emergency room (ER) as a primary outcome.What do the results of this study add? Our study strengthens previous studies and reassures that recurrence is not associated with surgical modality. Specifically, a recurrent referral to the ER and hospitalisation were not found to be associated with surgical modality, both which may be attributed to various reasons other than the recurrence of the abscess. An association was found between positive culture results and a recurrent referral to the ER.What are the implications of these findings for clinical practice and/or further research? The association found that positive results warrant further larger studies in order to determine which of the patients may benefit from antibiotic treatment in addition to the surgical treatment.

A different strategy for management of a case of adenoid cystic carcinoma of Bartholin gland.

Adenoid cystic carcinoma of the Bartholin gland is a rare vulval cancer. Available literature suggests an aggressive nature with protracted clinical symptoms and local recurrence despite adequate surgical excision with or without adjuvant radiotherapy. This case demonstrates the role of neoadjuvant chemotherapy and interstitial brachytherapy with less radical surgery, in the treatment of adenoid cystic carcinoma of the Bartholin gland. A 63-year-old woman presented in the clinic four months after the removal of vulvar mass with histopathology report showing adenoid cystic carcinoma of the Bartholin gland with positive margins. Clinical examination and imaging showed local disease extension up to the levator ani and abutting anal margin. She was given four cycles of neoadjuvant chemotherapy followed by ipsilateral hemivulvectomy with inguino-femoral lymphadenectomy followed by 25 cycles of external beam radiation and three cycles of interstitial brachytherapy. The patient has been disease free for more than 30 months. The use of neoadjuvant chemotherapy in treatment of Bartholin carcinoma along with conser vative surgery and radiotherapy can be a good treatment strategy as it reduces the morbidity associated with radical surgery without recurrence to date.

The impact of CO2 laser for treatment of Bartholin's gland cyst or abscess on female sexual function: a pilot study.

The aim of this study is to evaluate the sexual functionality before and after treatment of Bartholin's gland diseases (BGD) with CO2 laser and to compare our results to patients who underwent surgical cold knife and to a healthy control group (HCG). Consecutive patients (n = 15) affected by BG cyst or abscess who underwent CO2 laser treatment were evaluated. Patients were asked to complete the Italian translation of the Female Sexual Functioning Index (FSFI) before and 4 weeks after treatment. Results after CO2 laser were compared with two control groups: patients affected by BG cyst (n = 15) or abscess treated with surgical cold knife treatment and a HCG (n = 18). A statistically significant advantage of CO2 laser versus cold knife treatment in terms of lubrication, pain and global score were recorded. Both the single scores of five domains and total score of FSFI were globally higher after any treatment compared to before (CO2 and cold knife) of BGD. According to our data, CO2 laser therapy is often well tolerated by patients and correlated with a favorable sexual health recovery.

Bartholin Duct Cyst and Gland Abscess: Office Management.

The Bartholin glands, located in the base of the labia minora, have a role in vaginal lubrication. Because of the presence of other glands, removal of a Bartholin gland does not affect lubrication. Ductal blockage of these typically pea-sized structures can result in enlargement of the gland and subsequent development of Bartholin duct cysts or gland abscesses. Two percent of women will develop a cyst or an abscess in their lifetime, and physicians should be familiar with the range of treatment options. Bartholin duct cysts and gland abscesses can be treated in the office. The healing and recurrence rates are similar among fistulization, marsupialization, and silver nitrate and alcohol sclerotherapy. Needle aspiration and incision and drainage, the two simplest procedures, are not recommended because of the relatively increased recurrence rate.

Nodular Hyperplasia of the Bartholin Gland, A Benign Mimicker of Aggressive Angiomyxoma: A Case Series and Literature Review.

Nodular hyperplasia (NH) of the Bartholin gland is an exceedingly rare benign solid lesion of the female genital tract that can mimic the Bartholin gland cyst clinically. The histologic criteria for NH were established in 1998 by Koenig and Tavassoli. In this case series, we describe 4 cases of NH from Women and Infants Hospital in Rhode Island. All cases have microscopic features of lobular proliferation of acini and inspissated mucin. One case especially has extensive mucin extravasation mimicking an aggressive angiomyxoma. In this case series, we call attention to NH as another entity to consider in the differential diagnosis of an enlarged Bartholin gland. We also discuss ways to distinguish it from other benign and malignant solid lesions of the vulvar vestibule.