Adenomyomas of the Gallbladder: An Analysis of Frequency, Clinicopathologic Associations, and Relationship to Carcinoma of a Malformative Lesion.

CONTEXT.­: The nature and associations of gallbladder (GB) "adenomyoma" (AM) remain controversial. Some studies have attributed up to 26% of GB carcinoma to AMs. OBJECTIVE.­: To examine the true frequency, clinicopathologic characteristics, and neoplastic changes in GB AM. DESIGN.­: Cholecystectomy cohorts analyzed were 1953 consecutive cases, prospectively with specific attention to AM; 2347 consecutive archival cases; 203 totally embedded GBs; 207 GBs with carcinoma; and archival search of institutions for all cases diagnosed as AM. RESULTS.­: Frequency of AM was 9.3% (19 of 203) in totally submitted cases but 3.3% (77 of 2347) in routinely sampled archival tissue. A total of 283 AMs were identified, with a female to male ratio = 1.9 (177:94) and mean size = 1.3 cm (range, 0.3-5.9). Most (96%, 203 of 210) were fundic, with formed nodular trabeculated submucosal thickening, and were difficult to appreciate from the mucosal surface. Four of 257 were multifocal (1.6%), and 3 of 257 (1.2%) were extensive ("adenomyomatosis"). Dilated glands (up to 14 mm), often radially converging to a point in the mucosa, were typical. Muscle was often minimal, confined to the upper segment. Nine of 225 (4%) revealed features of a duplication. No specific associations with inflammation, cholesterolosis, intestinal metaplasia, or thickening of the uninvolved GB wall were identified. Neoplastic change arising in AM was seen in 9.9% (28 of 283). Sixteen of 283 (5.6%) had mural intracholecystic neoplasm; 7 of 283 (2.5%) had flat-type high-grade dysplasia/carcinoma in situ. Thirteen of 283 cases had both AM and invasive carcinoma (4.6%), but in only 5 of 283 (1.8%), carcinoma arose from AM (invasion was confined to AM, and dysplasia was predominantly in AM). CONCLUSIONS.­: AMs have all the features of a malformative developmental lesion, and may not show a significant muscle component (ie, the name "adeno-myoma" is partly a misnomer). While most are innocuous, some pathologies may arise in AMs, including intracholecystic neoplasms, flat-type high-grade dysplasia or carcinoma in situ, and invasive carcinoma (1.8%, 5 of 283). It is recommended that gross examination of GBs include serial slicing of the fundus for AM detection and total submission if one is found.

Extrauterine adenomyoma: A case report and systematic review of the literature.

OBJECTIVE: To describe a case of extrauterine adenomyoma (EA) and review all the cases of EA in the literature. METHODS: Pubmed/MEDLINE, Embase, and Google Scholar from 1807 to December 2022. All studies reporting the histologic diagnosis of an EA. We collected the following data: patient's age, size and location of adenomyoma, presence of endometriosis and adenomyosis, past gynecologic treatment, symptoms, diagnostic imaging, surgical intervention, alternative/adjuvant treatment, associated malignancy, and follow up. RESULTS: Sixty-seven studies with 85 patients were included. Pain was the most frequent symptom (69.5%). Among diagnostic examinations, ultrasonography was used in 60 out of 81 reported cases, with several radiologic features described. EA was located inside the pelvis in 77.6% of patients. Adnexa were the most frequent site of the disease (24, 28.2%). History of endometriosis or adenomyosis was described in 35 patients (35, 41.2%). Uterine tissue morcellation was reported in 6 of the 85 patients (7.1%). Associated malignancy was detected in 9 out of 85 patients with available data (10.6%). There were two recurrences of disease. CONCLUSION: Specific imaging features of EA are yet to be described in the literature. History of endometriosis and adenomyosis or uterine tissue morcellation may be suggestive of EA. Histologic examination can give a definitive diagnosis and exclude malignant transformation.

Adenomyoma of the small intestine in children: a 12-year experience in a tertiary referral center.

BACKGROUND: Adenomyoma of the small intestine is rare in children and the clinical characteristics is not clear. The study was to document the clinical characteristics and treatment of children with adenomyoma of the small intestine. METHODS: A retrospective study was conducted in children with intestinal adenomyoma from 2010 to 2022. We recorded age, gender, symptoms, location, tumour size and treatment options. RESULTS: Thirteen patients with adenomyoma of the small bowel were included. The median age was 20 months with a male-to-female ratio of 10:3 and more than half of the patients were younger than 2 years old. The mean tumour size was 2.0 cm. The lesion was found accidentally in one patient, and the others presented with symptoms of intussusception. A pathological lead point was found on ultrasound in seven patients. All tumours were located in the ileum, ranging from 24 to 260 cm proximal to the ileocecal valve. The tumour was found in an antimesenteric site in eight patients. Three patients suffered intestinal necrosis, and segmental resection of the ileum was performed. Three patients without intestinal necrosis underwent tumour rection, while intestinal resection and anastomosis were performed in the remaining seven. All patients recovered well except one, who developed intussusception 7 days after surgery; that patient underwent surgery and recovered uneventfully. CONCLUSIONS: Adenomyoma of the small intestine has a male predominance in children and intussusception is a common presentation. The ultrasound feature is a mass of mixed echogenicity containing several small cystic areas. Surgery is the primary treatment option and the procedure should be chosen based on intraoperative findings.

Surgical treatment of a rare case of atypical polypoid adenomyoma of the uterus in a menopausal patient: a case report.

Atypical polypoid adenomyoma is a rare benign tumor of the uterus that usually affects women of reproductive age and has an increased risk of progression into endometrial cancer. The pathogenetic mechanism has not been completely clarified. Due to the rarity of the tumor, current experience regarding the diagnostic and therapeutic approach is limited. For menopausal patients, hysterectomy seems to be the main treatment option. Our case concerns an asymptomatic menopausal patient with a vaginal delivery in her obstetric history and no hereditary history of gynecological cancer who came to the outpatient clinic for a gynecological examination. Transvaginal ultrasound revealed the presence of a large, round solid mass with increased vascularity within the endometrial cavity. A diagnostic dilation and curettage of the endometrium was performed. Histological examination of the endometrial biopsy showed an atypical polypoid adenomyoma, and it was decided to perform a total abdominal hysterectomy with bilateral adnexectomy. Histological examination of the surgical specimen of the uterus revealed no residual disease, no coexisting foci of atypical endometrial hyperplasia or endometrial cancer. The postoperative course was uneventful. The patient remains to this day under regular follow-up. The present case report highlights the significant difficulties involved in the preoperative diagnosis of atypical polypoid adenomyoma of the uterus and the difficult differential diagnosis from atypical endometrial hyperplasia and endometrial cancer, particularly in menopausal patients. At the same time, it is pointed out that despite its rarity, the early diagnosis of atypical polypoid adenomyoma, especially in young women, must be the main concern of the modern gynaecologist, in order to design the optimal treatment aimed at preserving fertility while avoiding the risk of recurrence of damage or malignant progression into endometrial cancer.

Laparoscopic intervention for solid pelvic tumours in Mayer-Rokitansky-Küster-Hauser syndrome: a case of bilateral uterine adenomyomas of the rudimentary uterus.

Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome, a condition caused by Müllerian anomalies, is characterised by congenital vaginal aplasia and a rudimentary uterus. Case reports concerning uterine fibroids associated with MRKH syndrome are limited, and differentiating between uterine fibroids and ovarian solid tumours prior to surgical intervention is often challenging. Here, we present the case of a patient with MRKH syndrome and asymptomatic bilateral pelvic solid tumours located close to both ovaries. Based on intraoperative and histopathological findings, the tumours were diagnosed as adenomyomas of the rudimentary uterus. This is the first reported case of a uterine adenomyoma associated with MRKH syndrome. Moreover, our report highlights the fact that diagnostic laparoscopy is a valuable method to evaluate pelvic tumours in MRKH syndrome.

Endometrioid Adenocarcinoma Resembling Cutaneous Pilomatrix Carcinoma: A Case Report of an Aggressive Neoplasm in a Young Woman With Diffusely Aberrant Beta-catenin Expression and Associated Morular Metaplasia and Atypical Polypoid Adenomyoma-type Change.

Endometrioid carcinoma with histopathologic resemblance to cutaneous pilomatrix carcinoma with mutations in the gene encoding beta-catenin, CTNNB1 are rare. There are minimal numbers of reports of high-grade tumors with this divergent differentiation in the literature. We report the case of a 29-yr-old female with an unusual presentation of endometrial cancer with overall histologic appearance indicative of a recently reported aggressive subtype of Federation of Gynecology and Obstetrics (FIGO) IVB grade 3 endometrioid carcinoma with features resembling cutaneous pilomatrix carcinoma. She was treated with a primary chemotherapy regimen with an initial significant response to treatment before developing symptomatic brain metastasis for which she underwent whole-brain radiotherapy. We discuss the unusual histologic and radiologic presentation as well as her individual management throughout this case report. The apparent association with morular metaplasia and atypical polypoid adenomyoma suggests that this rare carcinoma is within a spectrum of lesions associated with aberrant beta-catenin expression/beta-catenin mutation. Its aggressive nature highlights the importance of early recognition of this rare lesion.

When Aunt Minnie turns out to be a Zebra: distal bile duct adenomyoma stimulating pancreas head tumor.

A 55-year-old female patient with a history of ethyl and nicotine abuse presented with chronic diarrhoea, significant weight loss and abnormal liver tests. Ultrasound examination revealed dilatation of the bile ducts and computed tomography scan with IV contrast further established simultaneous dilatation of the common bile duct and pancreatic duct (double duct sign). The dilatation abruptly stopped at the level of the pancreatic head which demonstrated a small irregular hypovascular zone (1.2 cm), suspected for malignancy. The following week, a radical pancreaticoduodenectomy was performed. Anatomopathological analysis revealed an adenomyoma (12 mm) located distal to the common bile duct, without arguments for malignancy.

Extrauterine adenomyoma of the lesser omentum: A case report and review of the literature.

RATIONALE: The extrauterine adenomyoma is rare and it is extremely rare outside the pelvic cavity. Herein, we reported the first case of a single extrauterine adenomyoma occurring in the lesser omentum. PATIENTS CONCERNS: This case involved a 55-year-old woman who had undergone subtotal gastrectomy and omentectomy for gastric carcinoma. During postoperational pathological examination, 1 lymph node-like mass was coincidentally found in the lesser omentum. The patient had a history of hysterectomy for uterine leiomyoma 8 years ago. DIAGNOSES: The resected 17 "lymph nodes" from the lesser omentum were routinely checked for possible metastasis of gastric carcinoma. One of lymph node-like mass was microscopically showed that it was composed of benign smooth muscle components, endometrial glands and stroma by HE staining. Therefore, adenomyoma was initially considered. INTERVENTIONS: The lymph node-like mass was removed together with the lesser omentum during the subtotal gastrectomy and omentectomy for gastric carcinoma. No special intervention was performed for the adenomyoma. OUTCOMES: Immunohistochemical staining confirmed that smooth muscle tissue was diffusely and strongly positive for Desmin, smooth muscle actin, estrogen receptor, and progesterone receptor, and negative for CD117, Dog-1, S100, and CD34. Endometrial glands and stroma were positive for estrogen receptor and progesterone receptor, and the endometrium interstitium was also positive for CD10. The final diagnosis of extrauterine adenomyoma occurring in the lesser omentum was established. LESSONS: So far, to the best of our knowledge, total 53 cases of extrauterine adenomyoma have been reported in 45 English reports. The most common location for a single mass was pelvic cavity (37 cases), but rarely outside the pelvic cavity. This is the first case of a single extrauterine adenomyoma occurring in the lesser omentum.

Surgical techniques for excision of juvenile cystic adenomyoma.

OBJECTIVE: To review causes of pelvic pain among adolescents and discuss surgical techniques for safe and effective resection of juvenile cystic adenomyomas. DESIGN: Case report. SETTING: Academic medical center. PATIENTS: We present a 16-year-old patient with chronic pelvic pain and ultrasound evidence of a 2.4 cm adenomyoma. The lesion was thought specifically to represent a juvenile cystic adenomyoma, defined as a cystic lesion >1 cm occurring in women younger than 30 years with severe dysmenorrhea that is distinct from the uterine cavity and surrounded by hypertrophic myometrium. INTERVENTION: Given minimal relief from medical therapy and high suspicion for coexistent endometriosis, our patient elected to undergo laparoscopic resection of adenomyoma and excision of pelvic lesions. MAIN OUTCOME MEASURES: Preoperative considerations discussed in this video include imaging to identify the location of the lesion and adjacent structures, such as the uterine vessels, discontinuation of gonadotropin-releasing hormone agonist for adequate intraoperative visualization, and the high likelihood of encountering endometriosis at operation. RESULTS: We review the following surgical techniques: maximize visualization with the use of a uterine manipulator and temporary oophoropexy, optimize hemostasis via temporary uterine artery ligation and control of collateral blood vessels, complete ureterolysis, meticulous enucleation of adenomyoma, and excision of coexistent endometriotic lesions. Surgical findings demonstrated a 2 cm lesion along the left lower uterine segment and red-brown lesions along bilateral ovarian fossa, pathologically confirmed as adenomyoma and superficial endometriosis, respectively. CONCLUSION: This video presents strategies for safe and effective adenomyoma resection and treatment of refractory chronic pelvic pain in an adolescent.

Clinical analysis of 44 cases of atypical polypoid adenomyoma of the uterus.

BACKGROUND: Atypical polypoid adenomyoma (APA) is a rare intrauterine polypoid lesion that occurs predominantly in premenopausal women. Although APA was previously considered a benign lesion and treated conservatively, an increasing number of cases show that APA has a high rate of recurrence or residual disease and that it precedes the development of carcinoma. The clinical management of APA remains to be established. The aim of this study was to analyse the clinicopathological features of APA and discuss its diagnosis and prognosis. METHODS: Forty-four patients with APA were admitted to Beijing Obstetrics and Gynecology Hospital from 2005 to 2019, and their clinical and histopathologic features were evaluated. B-ultrasound was performed, and all the patients (n = 44) underwent hysteroscopy. Endometrium excision was performed by means of the four-step diagnosis and treatment method. Hysteroscopic transcervical resection (TCR) was performed in 5 patients with APA-H and 11 with APA-L. Except for one patient who underwent transcervical endometrial resection, all the patients underwent hysterectomy and salpingectomy or salpingo-oophorectomy. Data from a median follow-up of 42 months (ranging from 3 to 174 months) were available for these patients. RESULTS: Pathological diagnosis were made according to the degree of abnormality of the APA surface glands, resulting in APA-L in 36 patients and APA-H in 8 patients. Among these patients, 28 (25 APA-L and 3 APA-H) were treated conservatively. The effect of the four-step diagnosis and treatment method as an APA therapy was excellent. During the follow-up, no evidence of recurrence was found. CONCLUSIONS: For patients with intracavitary lesions > 1 cm, the hysteroscopic four-step diagnosis and treatment method and pathological diagnosis are the basis of clinical treatment. More than 30% of APA surface glands have complex structures characterized by branching and budding or other high-risk factors, such as endometrial hyperplasia, which are indications for hysterectomy. For patients who desire to become pregnant or to preserve the uterus, hysteroscopy with complete excision of the lesions should be the preferred treatment method. The patients should be treated and followed up closely with regular hysteroscopy and endometrial biopsy.

Hysteroscopic treatment assisted by photodynamic diagnosis for atypical polypoid adenomyoma: A report of two cases.

Atypical polypoid adenomyoma (APA) is an intrauterine tumor for which hysteroscopic tumor resection allows for fertility preservation. Complete resection is important because of the high recurrence rate of APA, but is difficult due to the lack of characteristic hysteroscopic findings. We previously reported a case in which photodynamic diagnosis (PDD) was useful for detection of APA. Here, we report two additional cases of APA treated by hysteroscopic resection with PDD. The procedure was approved by the ethical committee. Case 1: A 35-year-old female who underwent hysteroscopic surgery for a submucosal tumor suspected to be APA with hypermenorrhea. Case 2: A 37-year-old female in whom hysteroscopic surgery was performed for a residual APA lesion after hormone therapy. In Case 1, PDD identified the tumor borders and this enabled as complete resection as possible. In Case 2, lesions could not be identified clearly under white light, but some areas were PDD-positive and were excised. Among 19 specimens from these two cases and the previously reported case, all PDD-positive specimens were pathologically diagnosed as APA. The sensitivity and specificity of PDD for APA were 76.9% and 100%, respectively. These results suggest that PDD can contribute to identification of APA.

Adenomiosis apendicular: reporte de dos casos / Appendicular adenomiosis: report of two cases

Resumen Los adenomiomas son tumoraciones benignas constituidas por un agregado nodular de músculo liso, glándulas endometriales y estroma endometrial. La presencia de adenomiomas fuera del útero es un hallazgo infrecuente. Presentamos dos casos de adenomiomas extrapélvicos localizados en el apéndice. El estudio histológico resultó esencial para el diagnóstico.

Abstract Adenomyomas are a benign tumor compound of smooth muscle nodular aggregate, endometrial glands and endometrial stroma. Adenomyomas presenting outside uterus are a rare finding. Here we report two extrapelvic adenomyomas of the appendix. Histological examination was essential for diagnosis.

Patterns of SATB2 and p16 reactivity aid in the distinction of atypical polypoid adenomyoma from myoinvasive endometrioid carcinoma and benign adenomyomatous polyp on endometrial sampling.

AIMS: Atypical polypoid adenomyoma (APAM) is an uncommon uterine lesion composed of complex endometrioid glands with frequent squamous morular metaplasia and fibromuscular stroma. On endometrial curettage, biopsy or polypectomy specimens, the admixture of endometrioid glands and smooth muscle raises the differential diagnosis of myoinvasive endometrioid carcinoma. Reproductive-age APAM patients may opt for fertility preservation, whereas myoinvasive carcinoma is treated surgically. One previous study reported an incidental finding that the stroma of APAM, in contrast to that of other polypoid lesions, was SATB2-positive. APAM has also been reported to show increased stromal p16 staining. We aimed to assess whether SATB2 and p16 are useful stains for the distinction of APAM from myoinvasive carcinoma and benign adenomyomatous polyps. METHODS AND RESULTS: Cases of 'atypical polypoid adenomyoma' (n = 32), 'adenomyomatous polyp' (n = 39) and 'myoinvasive endometrioid carcinoma' (n = 30) were identified. Morphological features were assessed, along with the intensity and extent of SATB2 and p16 staining in the stromal component of each lesion. SATB2 expression was seen in the stromal components of 30 of 32 (94%) APAMs, versus none of 39 (0%) benign adenomyomatous polyps and five of 30 (17%) myoinvasive endometrioid carcinomas. Stromal p16 expression was seen in 31 of 31 (100%) APAMs, versus 20 of 39 (51%) benign adenomyomatous polyps and 12 of 30 (40%) myoinvasive endometrioid carcinomas. CONCLUSIONS: Patchy to diffuse SATB2 and block-type p16 staining of fibromuscular stroma separating atypical endometrioid glands is more consistent with APAM than with myoinvasive endometrioid carcinoma. These stains are potentially useful adjuncts to careful morphological evaluation of endometrial biopsies/curettings.

Lesion-targeted cytology to improve cytological sampling for atypical polypoid adenomyomas of the uterus: A case series and review of the literature.

OBJECTIVE: Atypical polypoid adenomyomas (APAs) are uncommon tumours consisting of atypical endometrioid glands and fibromyomatous stroma. Identifying the biphasic nature of atypical glandular components and spindle mesenchymal components without atypia is crucial for the cytological diagnosis of APA. We investigated the utility of lesion-targeted cytology (LTC) to directly collect firm spindle components. METHODS: We recruited seven consecutive surgical patients who underwent cytological examinations before surgery and were diagnosed with APA on postoperative histological examinations. Cytological smears were obtained by routine sampling in five cases and by targeted sampling using transvaginal ultrasonography, that is, LTC, in two cases. We retrospectively analysed the cytological findings from our cases and compared them to those of APA cases previously reported in the English literature. RESULTS: Among 5/7 cases that involved routine cytological sampling, normal cytological findings were found in 2 and atypical glandular cells were found in 3, but spindle cells from mesenchymal components were not detected. In contrast, among 2/7 cases in which sampling involved LTC, spindle cells without atypia, in addition to atypical glandular cells were found. CONCLUSIONS: Lesion-targeted cytology is useful to assess mesenchymal components of APAs and may improve the cytological diagnosis of APA.

Juvenile Cystic Adenomyoma vs Blind Uterine Horn: Challenges in the Diagnosis and Surgical Management.

BACKGROUND: Juvenile cystic adenomyomas (JCAs) are rare uterine lesions. Differential diagnosis might be difficult. We present the case of an adolescent who was diagnosed with JCA and was managed with laparoscopic excision. CASE: A 14-year-old patient with complaint of menarche with excruciating dysmenorrhea, was diagnosed using magnetic resonance imaging with a uterine anomaly consisting of a normal right hemiuterus, and a left cystic lesion with surrounding hypotense myometrium. She was managed with laparoscopic excision of the left side, and uterine reconstruction. Histology was suggestive of JCA, associated with diffuse adenomyosis. Dysmenorrhea improved considerably after surgery. SUMMARY AND CONCLUSION: Differential diagnosis between cystic uterine lesions relies on clinical, imaging, and perioperative clues that might assist in their formal classification. Doubt might still remain in some cases.