Benign Multicystic Mesothelioma Research Articles

Adenomatoid Tumor: A Review of Pathology With Focus on Unusual Presentations and Sites, Histogenesis, Differential Diagnosis, and Molecular and Clinical Aspects With a Historic Overview of Its Description.

Adenomatoid tumors have been described almost a century ago, and their nature has been the subject of debate for decades. They are tumors of mesothelial origin usually involving the uterus, the Fallopian tubes, and the paratesticular region. Adenomatoid tumors of the adrenal gland, the liver, the extragenital peritoneum, the pleura, and the mediastinum have been rarely reported. They are usually small incidental findings, but large, multicystic and papillary tumors, as well as multiple tumors have been described. Their pathogenesis is related to immunosuppression and to TRAF7 mutations. Despite being benign tumors, there are several macroscopic or clinical aspects that could raise diagnostic difficulties. The aim of this review was to describe the microscopic and macroscopic aspects of adenomatoid tumor with a special focus on its differential diagnosis and pathogenesis and the possible link of adenomatoid tumor with other mesothelial lesions, such as the well-differentiated papillary mesothelioma and the benign multicystic mesothelioma, also known as multilocular peritoneal cysts.

Complete laparoscopic pelvic peritonectomy plus hyperthermic intraperitoneal chemotherapy.

The aim of our study was to present the technique for, and early results of complete laparoscopic pelvic peritonectomy (LPP) plus hyperthermic intraperitoneal chemotherapy (HIPEC). We conducted a study on consecutive patients who had LPP for limited peritoneal carcinomatosis (peritoneal carcinomatosis index < 10) from ovarian cancer, colon cancer and benign multicystic mesothelioma, from January 2017 to November 2019 at 2 referral centers in Spain. Perioperative, pathologic, 30-day major morbidity and mortality characteristics were analyzed. The surgical technique is shown in the attached video. Twelve LPP + HIPEC were performed. Complete cytoreduction was achieved in 100% of the patients, the median duration of the operation was 450min (range 360-600min). There were 2 cases (16%) of IIIa morbidity (trocar hernia and pleural effusion), and no mortality. The median length of hospital stay was 5.5days (range 4-10days). The median length of follow-up was 10months (range 2-30months). There was a recurrence at the splenic hilum in 1 patient which was treated by laparoscopic splenectomy and one nodal recurrence at 13months while all other patients are alive and free of disease at last follow-up. This is the first technical video of a minimally invasive approach for complete pelvic peritonectomy plus omentectomy associated with HIPEC. For highly selected patients, this procedure presents a feasible and safe alternative to the maximally invasive approach.

A minimally invasive approach for peritonectomy procedures and hyperthermic intraperitoneal chemotherapy (HIPEC) in limited peritoneal carcinomatosis: The American Society of Peritoneal Surface Malignancies (ASPSM) multi-institution analysis.

Minimally invasive surgery is playing an increasing role in the treatment of patients with gastrointestinal and gynaecological malignancies as the data show reduced morbidity, faster recovery and similar oncological outcome when compared to open procedures. The American Society of Peritoneal Surface Malignancies (ASPSM) conducted a retrospective study to analyse peritonectomy procedures and HIPEC done via the laparoscopic route. A database with standard clinical and pathological parameters was set up and distributed amongst ASPSM members. Rate of relapse, morbidity and mortality were the primary endpoints of the study. A total of 90 patients from 7 centres around the world were identified. Sixty percent were female. Mean age was 50years. Peritoneal carcinomatosis from appendiceal origin was the most common diagnosis in a 64.9% of patients and colon origin was diagnosed in 16.5% of patients. Mean peritoneal cancer index (PCI) was 4.1 (0-10). Forty-one percent of patients had a bowel resection. Mean operative time was 4.7h (2.5-8). All patients had a complete cytoreduction and HIPEC. Grade 3 and 4 morbidity was 3.0 and 6.5%, respectively. The most common reason for re-operation was an internal hernia in 2 out of 5 cases. Operative mortality and re-admission rates were 0 and 5%, respectively. Mean hospital stay was 7.4days (1-18). At a mean follow-up of 31.6months, 15/90 patients have a disease relapse but loco-regional relapse was identified in only five patients. Analysis of these data suggests that minimally invasive approach for peritonectomy procedures and HIPEC is feasible, safe and should be considered as part of the armamentarium for highly selected patients with peritoneal surface malignancies with limited tumour burden, defined as PCI of 10 or less and borderline tumours as low-grade pseudomyxoma and benign multicystic mesothelioma.

Cytoreductive Surgery and Intraperitoneal Hyperthermic Chemotherapy (HIPEC) by Minimally Invasive Approach, an Initial Experience.

The cytoreductive surgery (CRS) associated with hyperthermic intraperitoneal chemotherapy (HIPEC) has become the standard treatment in patients with carcinomatosis peritoneal from different origins. The use of a minimally invasive approach for this high complex procedure might be an alternative that provides them less morbidity and faster recovery with similar oncologic outcomes. We describe the initial experience of CRS and HIPEC done via the laparoscopic route in patients with minimal peritoneal metastases in our Unit from March 2016 to January 2018. A total of eight patients were operated by this minimally invasive approach. The different diagnoses were low-grade pseudomyxoma peritonei (2), benign multicystic mesothelioma (2), primary epithelial ovarian carcinomatosis (2) and locally advanced colon carcinoma T4 (2). The median age was 54 (20-62) years, the median PCI was 3 (2-4), the median operative time was 287min (240-360), complete cytoreduction CC0 was achieved in all the patients, and no major morbidity was observed. The median length of stay was 4.75days (4-5). After a median follow-up of 9.5months, no relapse has been observed. The results suggest that this minimally invasive approach for CRS and HIPEC is feasible and safe in a highly selected group of patients with peritoneal surface malignancies.

Benign multicystic mesothelioma and peritoneal inclusion cysts: are they the same clinical and histopathological entities? A systematic review to find an evidence-based management.

Peritoneal mesothelial cysts (PMC) are a clinical dilemma because of their true pathogenic nature. Many definitions have been associated with PMC, including "benign multicystic mesothelioma", "cystic mesothelioma", "multilocular peritoneal inclusion cysts", ''inflammatory cysts of the peritoneum" or "postoperative peritoneal cyst". We herein performed a systematic review of the literature focusing on clinical and histopathological aspects of PMC, diagnosis, and therapies. Moreover, we described our experience with a case of PMC in a young female. Since there is often a history of prior surgery or inflammatory disease, most authors consider PMC of reactive origin. However, in some cases they occur without any documentable signs of disease or injury. A variety of clinical findings can complicate the preoperative assessment and a multitude of histological pictures may potentially lead to a misdiagnosis. The absence of a uniform treatment strategy and lack of long-term follow-up often hinder the accurate definition leading to unnecessary or unnecessarily aggressive therapy. PMC are more common than had previously been thought. Most authors consider them non-neoplastic; thus the designation of "peritoneal inclusion cyst" is preferable. The term "mesothelioma" should be used only in cases of histological evidences of atypia. The high rates of recurrence suggest that the goal of treatment should not be necessarily complete eradication, but symptomatic relief through individualized treatment. This is a topic of particular importance, especially in young female where recurrence rates could be lower than those reported in adults and where an improperly aggressive treatment could have repercussions on fertility.

Robotik Yardımlı Benign Multikistik Mezotelyoma Rezeksiyonu

Robotik Yardımlı Benign Multikistik Mezotelyoma Rezeksiyonu

Recurrent benign multicystic peritoneal mesothelioma: Approach to this rare condition

Abstract Background Benign multicystic mesothelioma is a rare benign tumour derived from the peritoneal mesothelium. The aim of this paper is to present a case of this rare tumour and review the clinical features, diagnosis and treatment of this disease. Clinical case The case is presented of a 22-year-old female diagnosed with multicystic mesothelioma after an urgent resection of intra-abdominal tumour in the context of acute abdominal pain. In the subsequent follow-up, the patient had a recurrence of the lesion, and at 2 years was treated by further resection. Conclusions Benign multicystic mesothelioma is a benign tumour of unknown origin, and with a non-specific clinical manifestation. The most effective treatment is surgical, although there is a high tendency to local recurrence.

A Rare Form of Intraabdominal Mass: Benign Multicystic Mesothelioma

Benign multicystic mesothelioma of pleura and periton is a rare tumor, which is seen in middle-aged women mostly. It was defined in 1979. Even though it is a clinically benign tumor, 50% relapse is seen after surgery. It is rare in men and generally diagnosed after the examinations following surgery because of the difficulties on diagnosing.

Benign Multicystic Omental Mesothelioma in Pregnancy: A Case Report and Review of the Literature

Benign Multicystic Peritoneal Mesothelioma (BMPM) is a very rare condition of unknown pathogenesis, that occurs mainly in women in their reproductive age (5:1 female/male ratio). The pathogenesis of BMPM is unclear and doubts regarding its neoplastic and reactive nature exist. Furthermore, the differential diagnosis with other diseases remains uncertain. In most cases the diagnosis is accidental, during a laparotomy performed for other reasons, since the diagnostic imaging does not allow a definite diagnosis preoperatively. Radical surgical resection is the “gold standard” for treatment of BMPM, despite a high rate of recurrence (about 50%). We report on a case of a 30-year-old pregnant woman with multilocular omental cystic mass, 36 x 22 cm in size, underwent elective caesarean section at term of pregnancy. The definitive histologic diagnosis was benign multicystic mesothelioma of the omentum. BMPM detected in pregnancy or during cesarean delivery is very rare; in fact only three other cases have been reported. Particularly, we present the only case, described in the literature, of a young female with benign multicystic omental mesothelioma in pregnancy that was treated solely by surgery, without recurrence after Case Study D’Agate et al.; BJMMR, 7(2): 138-144, 2015; Article no.BJMMR.2015.217 139 three years follow-up. In this article we also describe diagnostic evaluation, treatment, prognosis of this rare disease as the possible positive effects of adjuvants medical treatments.

A rare omental tumor presenting as pelvic mass – A case report

Abstracts Benign multicystic mesothelioma (BMCM) of omentum and peritonium is a rare intra-abdominal lesion of unknown etiology. Incidence is more common in females of child-bearing age group. Rarity of this tumor and non-specific symptoms causes preoperative diagnostic dilemma. Precise diagnosis requires immunohistochemistry study. Despite of high recurrence rate, aggressive surgical excision is the treatment of choice. Our case report of BMCM is a rare as it was detected in a young male patient and there was involvement of omentum only.

Benign multicystic mesothelioma: Is histological aggressiveness a prognostic of local recurrence?

Objectives: Benign multicystic mesothelioma is an exceptionally rare entity (0.15/100,000/year) that mostly affects pre-menopausal women. It is suspected to arise from mesothelial cells exposed to repetitive inflammatory stimulus (previous surgery, endometriosis, inflammatory pelvic disease, Mediterranean familial fever). Radical surgical resection is the standard of care but local recurrence may be as high as 50%. Rarely malignant progression may occur (2 out of 130). The characterization of clinical and histological features predictive of local recurrence of malignant transformation may play a major role in therapeutic guidance.

Peritoneal Mesothelioma: The Site of Origin Matters

The etiology, gender distribution, pathology, natural history, and treatment options for mesothelioma (MM) differ substantially depending on the site of origin. Peritoneal mesothelioma (MPeM) is a rare disease, comprising only approximately 10% to 15% of the 2,500 cases of MM diagnosed in the United States each year. Patients with MPeM are younger than patients with pleural MM, and a higher proportion, mostly women, are long-term survivors. Most MPeM is caused by asbestos exposure. Germ-line mutations of BAP1 (BRCA associated protein 1) can predispose to MM, uveal melanoma, and potentially other cancers. MPeM can be challenging to diagnose, and cytology is rarely helpful. Review by an experienced pathologist using a panel of at least two positive and two negative immunohistochemical stains is essential. The three major pathologic subtypes are epithelial, sarcomatoid, and biphasic. Most cases are epithelial; the others have a dismal prognosis. Two indolent subtypes of borderline malignant potential-well-differentiated papillary mesothelioma and benign multicystic mesothelioma-are more common in the peritoneum and are treated surgically. In highly selected patients receiving treatment at experienced referral centers, an aggressive locoregional strategy that combines cytoreductive surgery to remove all gross disease and hyperthermic intraperitoneal chemotherapy to treat residual microscopic tumors yields a 3-year survival of 60% and a median survival approaching 5 years, far better than expected from historic controls. This approach also provides durable palliation of malignant ascites in nearly all patients. Pemetrexed is the only U.S. Food and Drug Administration (FDA)-approved systemic chemotherapy for pleural MM. Largely on the basis of data from pharmaceutical registry studies, the activity of pemetrexed-based chemotherapy appears to be similar in pleural MM and MPeM.

Peritoneal Mesothelioma: The Site of Origin Matters

The etiology, gender distribution, pathology, natural history, and treatment options for mesothelioma (MM) differ substantially depending on the site of origin. Peritoneal mesothelioma (MPeM) is a rare disease, comprising only approximately 10% to 15% of the 2,500 cases of MM diagnosed in the United States each year. Patients with MPeM are younger than patients with pleural MM, and a higher proportion, mostly women, are long-term survivors. Most MPeM is caused by asbestos exposure. Germ-line mutations of BAP1 (BRCA associated protein 1) can predispose to MM, uveal melanoma, and potentially other cancers. MPeM can be challenging to diagnose, and cytology is rarely helpful. Review by an experienced pathologist using a panel of at least two positive and two negative immunohistochemical stains is essential. The three major pathologic subtypes are epithelial, sarcomatoid, and biphasic. Most cases are epithelial; the others have a dismal prognosis. Two indolent subtypes of borderline malignant potential—well-differentiated papillary mesothelioma and benign multicystic mesothelioma—are more common in the peritoneum and are treated surgically. In highly selected patients receiving treatment at experienced referral centers, an aggressive locoregional strategy that combines cytoreductive surgery to remove all gross disease and hyperthermic intraperitoneal chemotherapy to treat residual microscopic tumors yields a 3-year survival of 60% and a median survival approaching 5 years, far better than expected from historic controls. This approach also provides durable palliation of malignant ascites in nearly all patients. Pemetrexed is the only U.S. Food and Drug Administration (FDA)–approved systemic chemotherapy for pleural MM. Largely on the basis of data from pharmaceutical registry studies, the activity of pemetrexed-based chemotherapy appears to be similar in pleural MM and MPeM.

Benign Multicystic Mesothelioma Presenting as a Cause of Chronic Pelvic Pain

Background: Benign multicystic mesothelioma occurs predominantly in women of reproductive age. The clinical presentation is varied, from being asymptomatic, an incidental finding during surgery, to lower abdominal pain, abdominal or pelvic mass, or abdominal distension. Case: The case of a 40-year-old woman who presented with chronic pelvic pain caused by benign multicystic mesothelioma is presented. Results: After a 4 cm×4 cm pelvic mass adherent to the left pelvic side wall, small bowel, and appendix, and obliterating the posterior cul-de-sac, was found through laparoscopy, the procedure was converted to a laparotomy, excision of the pelvic mass, appendectomy, and enterolysis. Pathology confirmed a benign multicystic mesothelioma. Conclusions: Benign multicystic mesothelioma is a rare lesion of uncertain pathogenesis or malignant potential. When diagnosed, complete excision and cytoreductive surgery is required, to avoid local recurrence. (J GYNECOL SURG 28:294)

Lymphangioma of the small bowel mesentery: A case report and review of the literature

Lymphangioma is a rare benign condition characterized by proliferation of lymphatic spaces. It is usually found in the head and neck of affected children. Lymphangioma of the small-bowel mesentery is rare, having been reported for less than 1% of all lymphangiomas. Importantly, it can cause fatal complications such as volvulus or involvement of the main branch of the mesenteric arteries, requiring emergency surgery. Moreover, the gross and histopathologic findings may resemble benign multicystic mesothelioma and lymphangiomyoma. Immunohistochemical study for factor VIII-related antigen, D2-40, calretinin and human melanoma black-45 (HMB-45) are essential for diagnosis. Factor VIII-related antigen and D2-40 are positive in lymphangioma but negative in benign multicystic mesothelioma. HMB-45 shows positive study in the smooth-muscle cells around the lymphatic spaces of the lymphangiomyoma. We report a case of small-bowel volvulus induced by mesenteric lymphangioma in a 2-year-and-9-mo-old boy who presented with rapid abdominal distension and vomiting. The abdominal computed tomography scan showed a multiseptated mass at the right lower quadrant with a whirl-like small-bowel dilatation, suggestive of a mesenteric cyst with midgut volvulus. The intraoperative findings revealed a huge, lobulated, yellowish pink, cystic mass measuring 20 cm × 20 cm × 10 cm, that was originated from the small bowel mesentery with small-bowel volvulus and small-bowel dilatation. Cut surface of the mass revealed multicystic spaces containing a milky white fluid. The patient underwent tumor removal with small-bowel resection and end-to-end anastomosis. Microscopic examination revealed that the cystic walls were lined with flat endothelial cells and comprised of smooth muscle in the walls. The flat endothelial cells were positive for factor VIII-related antigen and D2-40 but negative for calretinin. HMB-45 showed negative study in the smooth-muscle cells around the lymphatic spaces. Thus, the diagnosis was lymphangioma of the small bowel mesentery with associated small bowel volvulus.

Benign multicystic mesothelioma with concurrent colonic adenocarcinoma: a report of two cases

Benign multicystic mesothelioma (BMM) is a rare tumoral lesion of the peritoneum often discovered incidentally upon surgery, laparoscopy, or radiographical imaging in females of childbearing age. The etiology and question as to whether these lesions occur via a neoplastic or reactive process still remain unknown. In this report, we present two cases of BMM detected in females with concurrent colonic adenocarcinoma arising in the ileocecal region. The tumors were present in the serosa surrounding the colonic adenocarcinoma in one case, and in the omentum in the other case, which grossly mimicked peritoneal seeding of carcinoma. An immunohistochemical analysis revealed that the cells lining the cystic spaces in both lesions were positive for keratin and calretinin, confirming their mesothelial origin. To the best of our knowledge, these cases represent the first documented examples of multicystic mesothelioma concurrent with colonic adenocarcinoma.

Unusual histological findings after partial pancreaticoduodenectomy including benign multicystic mesothelioma, adenomyoma of the ampulla of Vater, and undifferentiated carcinoma, sarcomatoid variant: a case series

IntroductionThe standard operation for carcinoma of the pancreatic head is a partial pancreaticoduodenectomy. Unusual histological findings may occasionally occur in the surgical specimen. We present three unusual histologic diagnoses after pancreaticoduodenectomy.Case presentationsIn the first case, an 86-year-old Caucasian woman was admitted with abdominal pain and nausea. Preoperative evaluation showed a 3 cm cystic lesion in the head of the pancreas. Pathology revealed a benign multicystic mesothelioma. In the second case, a 45-year-old Caucasian man complained of nausea, vomiting and general malaise for several months. Endoscopic retrograde cholangiopancreatographic examination and a computed tomography scan showed a stenosis of the distal bile duct secondary to a mass in the head of the pancreas and duodenum. Histology showed an adenomyoma of the ampulla. In the third case, a 59-year-old Caucasian man presented with chronic alcoholic pancreatitis. A computed tomography scan revealed a 3.5 cm lesion in the head of the pancreas with cystic and solid components. Pathology showed an undifferentiated carcinoma, sarcomatoid variant.ConclusionPartial pancreaticoduodenectomy is usually performed for ductal adenocarcinomas, neuroendocrine tumors or chronic pancreatitis. Compared to the majority of the above diagnoses, the three cases in our study are very rare. Benign multicystic mesothelioma is a very rare tumor that originates from the peritoneum. Although it demonstrates a benign clinical behaviour, it frequently recurs after resection. Adenomyoma of the bile duct or ampullary region is a very unusual, benign, localized lesion characterized by adenomyomatous hyperplasia. Undifferentiated carcinoma, sarcomatoid variant, is an aggressive tumor and is characterized by spindle cells. As the lesions were suspicious for carcinoma, partial pancreaticoduodenectomy was justified in all three patients. The histologic diagnosis after partial pancreaticoduodenectomy may differ from the preoperative and intraoperative findings. These cases demonstrate that a definitive diagnosis may only be obtained by a pathologic examination of the surgical specimen.

Laparoscopic Diagnosis and Treatment of Pelvic Benign Multicystic Mesothelioma Associated with High CA19.9 Serum Concentration

We report a case of benign multicystic mesothelioma in a 20-year-old woman referred because of amenorrhea. She underwent pelvic transabdominal ultrasound, which disclosed a micropolycystic appearance of the ovaries and a fluid collection in the pouch of Douglas. Tumor serum markers revealed an increase in CA19.9. Abdominal and pelvic computed tomography scans confirmed the presence of ascites. Laparoscopy disclosed small, thin-walled, translucent cysts in the Douglas cavity. The cysts were free-floating in a yellowish, sticky, gelatinous material. Microscopically, cystic lesions showed mesothelium-lined cystic spaces surrounded by a delicate thin fibrovascular wall. With immunohistochemistry, the tumor cells were strongly positive for cytokeratin and calretinin. These aspects were suggestive of benign multicystic mesothelioma. Electron microscopy confirmed the mesothelial nature of this tumor. Serial evaluation of the CA19.9 concentration showed a progressive decrease in the serum marker in the normal range. The patient is now well and symptom-free with no recurrence 24 months after surgery. The association between benign multicystic mesothelioma and increased CA19.9 serum concentration has been described only once, in a man. To our knowledge, this is the second case of benign multicystic mesothelioma associated with increased CA19.9 serum concentration and the first diagnosed in a woman. In the present case, a minimally invasive laparoscopic approach enabled not only histologic diagnosis of benign multicystic mesothelioma but also its surgical treatment. Although benign multicystic mesothelioma is a rare pathologic entity, it is important that sonologists include it in the differential diagnosis of diseases that manifest with ascites. Furthermore, all surgeons should be aware of the macroscopic and laparoscopic appearance of the lesion, and its generally benign course.

Benign Multicystic Mesothelioma in the Left Round Ligament: Case Report

Benign multicystic mesothelioma is a rare mesothelial lesion that forms multicystic masses in the upper abdomen, pelvis, and retroperitoneum. Most cases have a benign course. We present the ultrasound and MR findings of benign multicystic mesothelioma in the left round ligament, which caused a left inguinal hernia in a 46-year-old woman.

ダグラス窩に認められた良性多嚢胞性中皮腫の１例

We report an extremely rare case of benign multicystic mesothelioma (BMM) focusing on fine-needle aspiration cytology (FNAC). A 28-year-old woman undergoing abdominal ultrasonography was found to have a multilocular lesion of the Douglas pouch. FNAC smear results showed numerous sheets of mesothelial-like cells without atypia, together with scattered isolated metaplastic squamous cells with single pyknotic nuclei. These findings yielded a diagnosis of BMM. FNAC smears can thus be made based on laparoscopic findings.