Treatment of juvenile cystic adenomyoma by sclerotherapy with alcohol instillation: A case report.

Adenomyosis is characterized by the presence of ectopic endometrial tissue within the myometrium. Treatment options ranges from use of non-steroidal anti-inflammatory drugs and hormonal suppression for symptomatic relief, to endometrial ablation or even hysterectomy. In this paper we report the case of successful ultrasound-guided aspiration of focal adenomyosis with intracavitary alcohol instillation in a young patient with symptomatic juvenile cystic adenomyoma. This is the second report of the treatment of sclerotherapy by alcohol instillation, which may be considered as an alternative modality in treating the cases of symptomatic adenomyoma.

Atypical polypoid adenomyoma follow-up and management: Systematic review of case reports and series and meta-analysis.

BACKGROUND: Atypical polypoid adenomyoma (APA) is a rare uterine tumor typically found in fertile age and associated with infertility. Among young nullipara women, conservative treatment is proposed despite the high recurrence rate and the association with endometrial cancer.Our aim was to assess the risk of recurrence with different conservative treatments in fertile ages and the prevalence of malignant or pre-malignant associated lesions to better address an adequate patient counselling when treatment modalities are discussed. METHODS: This study is a systematic review and meta-analysis of case reports and case series about APA management and follow-up. A literature search was carried from Medline and Scopus for studies published from January 1, 1980 to December 31, 2018. RESULTS: We included 46 observational studies and 296 cases in fertile women. The prevalence of APA relapse was 44% (CI.95 33-57%) and was lower in cases treated with operative hysteroscopy (22%; CI.95 11-39%) than in cases treated with blind curettage and polypectomy (38%; CI.95 15-67%). The prevalence of the concomitant or during the follow-up diagnosis of endometrial carcinoma was 16% (CI.95 9-29%). The risk of cancer development during follow-up was significantly less in cases treated with histeroscopy (10.56% new cumulative diagnosis at 5 years follow up; CI.95 0-23.7%) than blind curettage and polypectomy (35.5% new cumulative diagnosis at 5 years; CI.95 11.65-52.92%; P < .05). Medical treatment with medroxyprogesterone acetate after surgery does not reduce APA recurrence. Pregnancy was observed in 79% cases in which the desire was expressed. CONCLUSION: This review suggests that conservative treatment performed by operative hysteroscopy is the optimal choice because it lowers the risk of recurrence, improves the accuracy of concomitant carcinoma or hyperplasia diagnosis, and leaves the possibility of future pregnancies.

Tratamiento de adenomioma polipoide atípico en paciente nuligesta mediante resección histeroscópica y dispositivo intrauterino de levonorgestrel / Treatment of atypical polypoid adenomyoma in a nulligravida patient by hysteroscopic resection and levonorgestrel-releasing intrauterine system

El adenomioma polipoide atípico uterino fue descrito por primera vez por Mazur. Tradicionalmente ha sido considerado un tumor benigno, sin embargo, recientes estudios han demostrado su potencial de malignización y una posible relación con la hiperplasia endometrial atípica y el carcinoma endometrial. La edad media de aparición es de 35-40 años, por lo que es fundamental el tratamiento conservador, ya que en muchos casos se trata de pacientes nuligestas. Así pues, una opción terapéutica en estos casos puede ser la resección histeroscópica transcervical, que permite de forma concomitante tanto el tratamiento como el diagnóstico definitivo, asociado a tratamiento hormonal con gestágenos. En este artículo, reportamos el caso clínico de una mujer nuligrávida con el diagnóstico de adenomioma polipoide atípico, que se trata mediante una resección histeroscópica y la inserción posterior de un dispositivo intrauterino liberador de levonorgestrel

Atypical polypoid adenomyoma of the uterus was first reported by Mazur. Traditionally it was considered to be a benign tumor, but recent studies demonstrated a possible relation with atypical endometrial hyperplasia, endometrial carcinoma and its potential for malignancy. The common age of onset is approximately 35-40 years, so conservative treatment is essential, since in many cases these patients are nulligravida. So, a man option can be a hysteroscopic resection transcervical for the treatment and the diagnosis at the same time, associated with hormonal treatment with gestagens. In this article, we report a case of a nulligravida woman with an atypical polypoid adenomyoma, which is treated by hysteroscopic resection and the postinsertion of a levonorgestrel-releasing intrauterine system

Clinical presentation and management of atypical polypoid adenomyomas: Systematic review of the literature.

The aim of this study is to investigate clinical behavior of Atypical Polypoid Adenomyomas (APAs) and to describe the rates of (i) recurrences, (ii) their association with endometrial hyperplasia and (iii) with endometrial cancer. All studies that reported the outcome of the clinical management of patients with histologically proven APAs were included. A review of the English literature since 1970 was systematically performed (PROSPERO No CRD42018080003). A quality assessment tool was used to assess the scientific value of all the studies. Main contribution of this review is the proposal of new definitions regarding the clinical behaviour of APAs: Cure, Residual or persistent APA, Recurrent APA, Synchronous endometrial hypeprasia, Subsequent endometrial hyperplasia, Synchronous endometrial cancer, and Subsequent endometrial cancer, are terms elucidated in the context of this review. Their rates after initial diagnosis and treatment of APAs are presented as the main outcome measures. 63 studies and 350 patients were included in the systematic review. Fifteen studies that reported 208 patients who did not have hysterectomy as initial treatment were included for further quantitative assessment. The cure rate of APAs in cases where uterus was preserved was 51.0% (106/208), the residual rate of APA was 20.2% (42/208), the recurrence rate of APA was 35.1% (73/208), the concurrent endometrial hyperplasia rate was 7.2% (15/208), the concurrent endometrial cancer rate was 4.8% (10/208), the subsequent endometrial hyperplasia rate of was 6.7% (14/208), and the subsequent endometrial cancer rate of was 10.1% (21/208). Moreover, 56.4% of the patients with APA who opted for uterine sparing treatment and wished to conceive they had a viable pregnancy. The use of hysteroscopic techniques at the initial management of APAs is related with significantly decreased residual rate, and signicantly increased cure rates. In this review, the recurrence rate and the association of APAs with cancer appeared to be higher compared to the previously reported in the literature rates. APAs comprise an intriguing clinical entity that needs individualized treatment, considering the increased association to serious gynecological diseases. Hysterectomy is the appropriate treatment, however uterus-sparing surgery can be offered to selected patients.

Management of women with atypical polypoid adenomyoma of the uterus: A quantitative systematic review.

INTRODUCTION: Atypical polypoid adenomyoma is an uncommon uterine lesion which can coexist with endometrial atypical hyperplasia and/or cancer. Atypical polypoid adenomyoma affects premenopausal women in most cases, but it shows high recurrence rate if conservatively treated. To date, the management of patients is based on low-quality evidence and is not standardized. Our primary aim was to explore the optimal management of atypical polypoid adenomyoma, with particular regard to the fertility-sparing approach. The secondary aim was to define clinicopathologic features of atypical polypoid adenomyoma. MATERIAL AND METHODS: Medline, Embase, Web of Sciences, Scopus, ClinicalTrial.gov, OVID, Google Scholar and Cochrane Library were searched for studies reporting outcomes of atypical polypoid adenomyoma treatments. Univariate comparisons among outcomes of fertility-sparing treatments (rates of initial response, progression, recurrence, final complete response, pregnancy) were performed with Fisher's exact test (α = .05). RESULTS: Eleven retrospective studies with 237 patients were included; 85.5% of patients were premenopausal and 62.9% were nulliparous. Atypical polypoid adenomyoma coexisted with atypical hyperplasia in 5.5% of cases and with endometrial cancer in 5.9%. Overall risks of recurrence and progression to cancer were 28.9% and 16.6%, respectively. Fertility-sparing treatments included hormonal therapy with or without maintenance, hysteroscopic transcervical resection, dilation and curettage, and hormonal therapy combined with transcervical resection or dilation and curettage. Transcervical resection showed significantly higher initial response rates (P from <0.001 to 0.023) than any other treatment. Transcervical resection and transcervical resection+hormonal therapy showed significantly lower progression rates (P < 0.001), and higher final complete response rates (P < 0.001) than any other treatment. No significant differences were found in the rates of pregnancy (P = 0.533 - 0.647) or recurrence (P = 0.052 - 0.475). Among the different transcervical resection techniques, the 4-step transcervical resection showed significantly lower rates of progression (P = 0.002) and recurrence (P = 0.013) than other techniques. Limitations to our results were the retrospective design of the studies and the relatively small sample size, due to the rarity of atypical polypoid adenomyoma. CONCLUSIONS: Based on its effectiveness and safety, transcervical resection may be the first-line fertility-sparing treatment for atypical polypoid adenomyoma. In particular, 4-step transcervical resection showed the best results. Given the risk of recurrence, progression and coexistent atypical hyperplasia or cancer, follow-up biopsies are advisable. When fertility preservation is not required, hysterectomy might be advisable.

Clinicopathological Characteristics of Primary Ovarian Adenomyoma: A Single-institutional Experience.

Adenomyoma is a benign neoplasm composed of endometrial-type glands, specialized endometrial-type stroma, and well-formed smooth muscle bundles. This tumor typically originates within the uterus, whereas extrauterine adenomyoma is an unusual presentation. The ovary is the most common site of extrauterine adenomyoma. In this study, we describe the clinical and pathological features and immunohistochemical findings of primary ovarian adenomyoma in patients at our Institution. In addition, we provide a thorough review of previously published cases of primary ovarian adenomyoma and clarify their clinicopathological characteristics. The most common clinical presentations of ovarian adenoma were abdominopelvic pain and abnormal menstruation. Imaging features of ovarian adenomyoma varied, showing mixed solid and cystic, solid, or cystic masses. Frequently associated conditions included congenital anomalies of the urinary tract and endometriosis. Although most cases of ovarian adenomyoma exhibit benign histopathological features, we observed one case of endometrioid carcinoma arising in ovarian adenomyoma. Clinical follow-up data indicated that simple excision of the mass is curative for ovarian adenomyoma, but certain patients underwent unnecessary surgical procedures due to lack of awareness of primary ovarian adenomyoma and high index of suspicion for malignancy in patients with solid ovarian masses. Although ovarian adenomyoma is rare, awareness of this tumor type aids pathologists in making correct diagnoses and clinicians in avoiding unwarranted therapeutic procedures.

A literature review of radiological findings to guide the diagnosis of gallbladder adenomyomatosis.

BACKGROUND: Gallbladder adenomyomatosis (GA) is a benign gallbladder entity discovered as an asymptomatic gallbladder mass. Since gallbladder cancer is in the differential diagnosis for gallbladder masses, the ability to differentiate benign disease avoids a more extensive oncologic resection. This study sought to review imaging modalities used to diagnose GA. METHODS: PubMed and SciVerse Scopus were systematically searched using the terms: "gallbladder adenomyomatosis" and "gallbladder imaging" for articles published between January 2000 and January 2015. RESULTS: A total of 14 articles were reviewed in this analysis. Contemporary series report the use of ultrasound (US), computed tomography (CT) or magnetic resonance imaging (MRI) in GA imaging. Ultrasound detection of Rokitansky-Aschoff sinuses, visualized as small cystic spaces with associated "comet-tail" or "twinkling" artifact, is pathognomonic for GA. A "Pearl-Necklace" sign of small connected sinuses on MRI or "Rosary" sign on CT are additional characteristics that may assist in establishing a diagnosis. CONCLUSION: Ultrasound is the most commonly used tool to investigate GA. If not diagnostic, CT or MRI are effective in attempting to differentiate a benign or malignant cholecystic mass. Characteristic signs should lead the surgeon to perform a laparoscopic cholecystectomy in symptomatic patients or manage non-operatively in asymptomatic patients.

[Atypical polypoid adenomyoma: retrospective study about 8 cases from Jeanne-de-Flandre Hospital between 1996 and 2008]. / Adénomyome polypoïde atypique: série rétrospective de 8 cas survenus à l'hôpital Jeanne-de-Flandre entre 1996 et 2008.

OBJECTIVES: To propose a therapeutic approach and follow-up of patients with atypical polypoid adenomyoma (APA) wishing to remain fertile. PATIENTS AND METHOD: We are presenting a retrospective study of eight APA cases. RESULTS: Two patients immediately received radical treatment, and one patient had an endometrial thermocoagulation. The remaining five patients received a conservative treatment with ultrasound and hysteroscopic monitoring. Among them, two had full-term pregnancies, one is being treated for infertility, in one case, the APA disappeared after three hysteroscopy-curettages and macroprogestative treatment. Lastly, one of our patients died from an aggressive endometrioid ovarian bilateral tumour associated with an endometrial adenocarcinoma. DISCUSSION AND CONCLUSION: Radical treatment is necessary for cases of APA in women no longer wishing to remain fertile. For those wishing to remain fertile, we can offer a conservative treatment once they have been informed regarding the associated risk of relapse, degeneration, and ovarian lesions which necessitate stricter follow-up.

Reproductive performance of severely symptomatic women with uterine adenomyoma who wanted preservation of the uterus and underwent combined surgical-medical treatment.

OBJECTIVE: To assess the factors associated with future pregnancy and successful delivery in women who were treated for uterine adenomyoma with combination (surgical-medical) therapy using ultramini- or mini-laparotomy conservative surgery and gonadotropin-releasing hormone agonist. MATERIALS AND METHODS: One hundred and two women were evaluated. Items for analysis included: age, body mass index, and conception history; clinical symptoms of dysmenorrhea and menorrhagia; tumor location and preoperative serum level of cancer antigen 125 (CA125); the intraoperative findings of the weight of the removed tumor, and the uterine cavity opening. RESULTS: After excluding those patients using contraception or searching for an assisted reproductive technique, a total of 56 women were enrolled for analysis. Twenty-three (41.1%) women had 27 clinical pregnancies after 3 years of follow-up; 15 went on to deliver a healthy live-born child; two delivered preterm but healthy babies; seven had elective abortions; four had spontaneous abortions; and one had an ectopic pregnancy. The women who had a successful delivery during the 3-year follow-up after treatment tended to be younger, with a lower body mass index, lower baseline analgesic usage score, and lower preoperative serum level of CA125, be nulliparous, and with an adenoma in an anterior location. The linear regression model showed that age and baseline analgesic usage score were independent predictors of successful delivery and accounted for 56.5% of the total variance related to successful delivery. CONCLUSION: Age was an important factor associated with future successful delivery, therefore, caution should be taken in considering the maintenance of future fertility in older women treated with surgical-medical therapy.

Long-term follow-up of severely symptomatic women with adenomyoma treated with combination therapy.

OBJECTIVE: The aim of our study was to assess the long-term efficacy of conservative surgery combined with gonadotropin-releasing hormone agonist therapy for uterine adenomyoma. MATERIALS AND METHODS: We carried out an uncontrolled descriptive study of 285 women who had symptomatic uterine adenomyoma. A total of 186 women with pathologically proven adenomyoma underwent ultramini-laparoscopic adenomyomectomy and a 6-month course of goserelin acetate treatment, and were evaluated semi-annually during a follow-up period of at least 3 years. RESULTS: Patient scores for dysmenorrhea using a self-reported six-point verbal numeric rating scale significantly declined compared with the baseline assessment, from 3.84 ± 0.65 to 0.33 ± 0.57, 0.52 ± 0.86, and 0.88 ± 1.29 at the end of the 1-, 2-, and 3-year follow-up visits, respectively (p < 0.001). Similar reductions were observed for analgesic usage scores. Menorrhagia scores significantly decreased compared with the baseline assessment, from 3.45 ± 1.46 to 0.42 ± 0.59, 0.65 ± 0.83, and 1.1 ± 1.34 at the end of the 1-, 2-, and 3-year follow-up visits, respectively (p < 0.001). CONCLUSION: Combination therapy for adenomyoma provides an effective treatment option for long-term symptom control and uterine preservation in severely symptomatic women for whom previous long-term drug therapy has failed or proven to be intolerable.

A multiple perspectives on atypical polypoid adenomyoma of uterus.

Atypical polypoid adenomyoma (APA) is a rare benign uterine tumor, with less than 200 cases have been reported in English literature. Although, it is considered as a benign lesion and treated conservatively previously, more and more cases show that APA has a high rate of recurrence or residual, and is found to precede the development of carcinoma. Given the data from present research on APA, the therapy of APA becomes more complex and must be cautious, especially for the nulliparous and premenopausal patients. In addition, because of the low incidence, studies on this disease are less, and the etiology and pathogenesis of APA is still unclear. In this review, we aim to summarize recent researches concerning APA from multiple perspectives, including clinical presentation, histogenesis, immunohistochemistry and molecular features, diagnosis and differential diagnosis, treatment opinion and prognosis, which may provide theory and clinical basis for the future clinical treatment and research of this rare disease.

Adenomyoma associated with high level of CA 125 and CA 19-9: case report.

PURPOSE OF INVESTIGATION: A rare case of increasing CA 125 and CA 19-9 levels increasing in a woman with adenomyoma is described. METHODS: A 39-year-old nullipara woman with CA 125 = 1,796 U/ml and CA 19-9 = 177 U/ml was submitted to abdominal and pelvic MRI, gastric endoscopy, colonoscopy, hysteroscopy, pelvic Doppler and PET scan. None of the exams revealed any apparent malignant disease. RESULTS: Six months of gonadotropin releasing hormone agonist treatment reduced CA 125 and CA 19-9 levels. However, after contraceptive pill use the markers were again elevated, and a laparoscopic hysterectomy was performed, and normal CA 125 and CA 19-9 levels were achieved. CONCLUSIONS: Adenomyoma may be associated with high levels of CA 125 and CA 19-9.

Gastric adenomyoma presenting as melena: a case report and literature review.

Gastric adenomyoma (AM) is a rare benign tumor characterized by gland-like structures embedded within a smooth muscle stroma. We report a case of a 68-year-old man with gastric AM admitted to our hospital for melana. Endoscopic examination revealed a gastric mass of about 4 cm in diameter, located in the antrum. Histologic examination of the excised specimen showed irregularly arranged glands and interlacing smooth muscle bundles surrounding the glandular elements. Although gastric AM is rare, it should be considered in differential diagnosis of extramucosal gastric tumor.

Multiple extrauterine adenomyomas and uterus-like masses: case reports and review of the literature.

OBJECTIVE: To describe two rare cases of multiple extrauterine adenomyomas, their clinical management, and their response to surgical and hormone therapy with GnRH agonist. DESIGN: Case report and review of the English-language scientific literature. SETTING: Cases presented in a tertiary health care academic institution. PATIENT(S): Two patients without urogenital malformations diagnosed with extrauterine adenomyoma, which is a benign tumor composed of smooth muscle and endometrium, typically originating within the uterus. INTERVENTION(S): Surgical radical excision of adenomyomas followed by a long-term hormone therapy with GnRH agonist. MAIN OUTCOME MEASURE(S): Anatomical and clinical outcomes. RESULT(S): Surgical treatment followed by long-term GnRH agonist therapy appeared effective in keeping the disease stable. After a long-term follow-up of 10 and 4 years, the two patients are still asymptomatic and stable. CONCLUSION(S): Only 19 other cases of extrauterine adenomyomas are reported in the English-language scientific literature, and no cases of multiple masses are described until now. It is probable that these extrauterine adenomyomas arose from a metaplastic transformation of the subcelomic mesenchyme. Long-term GnRH agonist therapy after surgery appeared effective in keeping the disease stable.

Early results of magnetic resonance-guided focused ultrasound surgery of adenomyosis: analysis of 20 cases.

STUDY OBJECTIVE: To evaluate the thermal ablative effects of magnetic resonance-(MR) guided focused ultrasound surgery (MRgFUS) on adenomyosis and to assess improvement in clinical parameters. DESIGN: Twenty patients with adenomyosis were treated with MRgFUS. Extensive adenomyosis (6 cases) was treated with 2 applications. Uterine volume was evaluated by MR imaging before and immediately after MRgFUS. Ablation of adenomyosis and the architecture of nonperfused areas were evaluated immediately after MRgFUS. Improvement in patient symptoms was assessed through the symptom severity score questionnaire (Canadian Task Force classification II-3). SETTING: Department of gynecology at a Japanese general hospital. PATIENTS: Premenopausal women at least 18 years of age with symptomatic adenomyosis. INTERVENTIONS: Thermal ablation by MRgFUS. MEASUREMENTS AND MAIN RESULTS: We classified the nonperfused lesions on contrast-enhanced MR images immediately after MRgFUS into 3 types: lesions with round margins (type R), serrated margins (type S), and honeycomb architecture (type H). Type R was the most common (16/20 patients). Most adenomyosis lesions could be sufficiently ablated close to the serosal surface or to the endometrium by MRgFUS. The mean uterine volume 6 months after therapy was decreased by 12.7%. Symptom severity score improved significantly during 6 months of follow-up. No serious complications were observed. CONCLUSION: These early results indicate the safe and effective ablation of adenomyosis tissue by MRgFUS. The procedure also resulted in the improvement in clinical symptoms during the 6 months of follow-up.

Gallbladder polyps, cholesterolosis, adenomyomatosis, and acute acalculous cholecystitis.

Acute acalculous cholecystitis is characterized by acute inflammation of the gallbladder in the absence of stones, usually occurring in elderly and critically ill patients with atherosclerosis, recent surgery or trauma, or hemodynamic instability. Patients may present with only unexplained fever, leukocytosis, and hyperamylasemia without right upper quadrant tenderness. If untreated, rapid progression to gangrene and perforation occurs. Surgical cholecystectomy and cholecystostomy provide the most definitive treatment although recent studies indicate success with percutaneous or endoscopic cholecystostomy. Cholesterolosis and adenomyomatosis of the gallbladder are usually clinically silent and incidental findings at the time of cholecystectomy. Cholesterolosis is characterized by mucosal villous hyperplasia with excessive accumulation of cholesterol esters within epithelial macrophages. Usually clinically silent, the condition rarely is associated with biliary symptoms or idiopathic pancreatitis and cannot reliably be detected by ultrasonography. Adenomyomatosis describes an acquired, hyperplastic lesion of the gallbladder characterized by excessive proliferation of surface epithelium with invaginations into a thickened muscularis propria. Ultrasonography may reveal a thickened gallbladder wall with intramural diverticula. Adenomyomatosis may portend a higher risk of gallbladder malignancy. Most cases of cholesterolosis and adenomyomatosis identified by imaging require no specific treatment. Gallbladder polyps include all mucosal projections into the gallbladder lumen and include cholesterol polyps, adenomyomas, inflammatory polyps, adenomas, and other miscellaneous polyps. Most polyps are nonneoplastic and rarely cause symptoms. Cholecystectomy is advocated for polyps greater than 10 mm in size because of increased risk of adenomatous or carcinomatous features.