Abstract:There are several reports of pleural adenomatoid (microcystic) mesothelioma, but peritoneal adenomatoid mesothelioma is extremely rare. A 64‐year‐old Japanese woman presented with no symptoms and no asbestos exposure history. An abdominal computed tomography scan revealed multiple hypervascular masses on the liver surface, pelvic cavity and anterior peritoneum. Over 10 pieces of the multiple resected tumors showed numerous microcysts composed of a bland mesothelial cell background with rich capillary vessels. … Show more
“…It is also important to exert caution when making the diagnosis of adenomatoid tumor arising in the peritoneum as this is an extremely rare location for this neoplasm42; therefore, malignant mesothelioma with an adenomatoid/microcystic pattern needs to be excluded through a combination of clinical findings and ancillary testing including loss of BAP-1 immunohistochemistry and CDKN2A homozygous deletion by FISH. The caveats are that a rare case of mesothelioma with adenomatoid pattern can be localized, as described herein, and that some cases of malignant mesothelioma will not show the loss of BAP-1 by immunohistochemistry or the CDKN2A homozygous deletion by FISH 43,44. In contrast, adenomatoid tumors show an intact expression of BAP-1 and a robust membranous staining for L1 cell adhesion molecule (L1CAM) by immunohistochemistry 43,45…”
Section: Discussionmentioning
confidence: 93%
“…The caveats are that a rare case of mesothelioma with adenomatoid pattern can be localized, as described herein, and that some cases of malignant mesothelioma will not show the loss of BAP-1 by immunohistochemistry or the CDKN2A homozygous deletion by FISH. 43,44 In contrast, adenomatoid tumors show an intact expression of BAP-1 and a robust membranous staining for L1 cell adhesion molecule (L1CAM) by immunohistochemistry. 43,45 The patients in this study received no standard treatment, reflecting time-dependent variations, comorbidities and institutional differences, as some of our patients had been initially treated in other centers.…”
Localized malignant peritoneal mesothelioma is a rare tumor with limited information in the literature. In this study, we present our experience with 18 cases seen in our hospital over a period of 43 years (1978 to 2021). Patients' median age was 55 years (y) (range: 33 to 79 y) and most of them were Caucasians. Patients presented with abdominal pain (11), ascites and right leg swelling (1), abdominal mass (1), and as incidental finding (1). Thirty percent of patients reported asbestos exposure, and all patients with available information had family history of tumors; a third had personal history of tumors. Seventy-seven percent had some form of abdominopelvic surgery and/or inflammatory process. Most cases had microscopic features typically seen in malignant mesothelioma; however, some cases had confounding features such as signet-ring cells, spindle cells, clear cell changes, and adenomatoid tumor-like appearance. BAP-1 by immunohistochemistry was lost in 1/3 cases. Only 1 patient underwent genetic testing and had an MSH2 germline mutation. Homozygous deletion of CDKN2A by FISH was not found in 1 tested case, although next-generation sequencing identified a CDKN2A pathogenic mutation. 16/18 (88%) had surgical treatment, and some also received adjuvant chemotherapy. The mean overall survival (OS) of our patients was 80.4 months (95% confidence interval: 54.3-106.52); the 3-year OS was 79%, while the 5-year OS was 52.6%. Fifty-three percent of patients had recurrences and 20% had tumor progression. Although the limited sample precludes definitive conclusions, small tumor size, low-grade cytology, and low mitotic index appeared to be associated with an indolent behavior.
“…It is also important to exert caution when making the diagnosis of adenomatoid tumor arising in the peritoneum as this is an extremely rare location for this neoplasm42; therefore, malignant mesothelioma with an adenomatoid/microcystic pattern needs to be excluded through a combination of clinical findings and ancillary testing including loss of BAP-1 immunohistochemistry and CDKN2A homozygous deletion by FISH. The caveats are that a rare case of mesothelioma with adenomatoid pattern can be localized, as described herein, and that some cases of malignant mesothelioma will not show the loss of BAP-1 by immunohistochemistry or the CDKN2A homozygous deletion by FISH 43,44. In contrast, adenomatoid tumors show an intact expression of BAP-1 and a robust membranous staining for L1 cell adhesion molecule (L1CAM) by immunohistochemistry 43,45…”
Section: Discussionmentioning
confidence: 93%
“…The caveats are that a rare case of mesothelioma with adenomatoid pattern can be localized, as described herein, and that some cases of malignant mesothelioma will not show the loss of BAP-1 by immunohistochemistry or the CDKN2A homozygous deletion by FISH. 43,44 In contrast, adenomatoid tumors show an intact expression of BAP-1 and a robust membranous staining for L1 cell adhesion molecule (L1CAM) by immunohistochemistry. 43,45 The patients in this study received no standard treatment, reflecting time-dependent variations, comorbidities and institutional differences, as some of our patients had been initially treated in other centers.…”
Localized malignant peritoneal mesothelioma is a rare tumor with limited information in the literature. In this study, we present our experience with 18 cases seen in our hospital over a period of 43 years (1978 to 2021). Patients' median age was 55 years (y) (range: 33 to 79 y) and most of them were Caucasians. Patients presented with abdominal pain (11), ascites and right leg swelling (1), abdominal mass (1), and as incidental finding (1). Thirty percent of patients reported asbestos exposure, and all patients with available information had family history of tumors; a third had personal history of tumors. Seventy-seven percent had some form of abdominopelvic surgery and/or inflammatory process. Most cases had microscopic features typically seen in malignant mesothelioma; however, some cases had confounding features such as signet-ring cells, spindle cells, clear cell changes, and adenomatoid tumor-like appearance. BAP-1 by immunohistochemistry was lost in 1/3 cases. Only 1 patient underwent genetic testing and had an MSH2 germline mutation. Homozygous deletion of CDKN2A by FISH was not found in 1 tested case, although next-generation sequencing identified a CDKN2A pathogenic mutation. 16/18 (88%) had surgical treatment, and some also received adjuvant chemotherapy. The mean overall survival (OS) of our patients was 80.4 months (95% confidence interval: 54.3-106.52); the 3-year OS was 79%, while the 5-year OS was 52.6%. Fifty-three percent of patients had recurrences and 20% had tumor progression. Although the limited sample precludes definitive conclusions, small tumor size, low-grade cytology, and low mitotic index appeared to be associated with an indolent behavior.
“…We conducted a literature search of the PubMed database to understand the current status of adenomatoid mesothelioma in the abdominal cavity. We came across six cases, including our case, that reported an “adenomatoid mesothelioma” (Table 1 ) [ 9 – 13 ]. There was one man and five women, and none had a history of asbestos exposure.…”
Section: Discussionmentioning
confidence: 99%
“…Recurrence was not observed in five out of six patients, but Mori et al . reported a recurrent case after curative resection (patient 5) [ 13 ]. We should also conduct a careful follow-up with our patient, although the optimal follow-up strategy has not been well established.…”
Background
Adenomatoid mesothelioma is a rare subtype of malignant mesothelioma that can be confused with adenomatoid tumors, which are classified as benign. The clinical features and optimal management of adenomatoid mesothelioma have not been elucidated in the literature. In this report, we present an extremely rare case of adenomatoid mesothelioma that developed on the peritoneal surface of the diaphragm as well as a literature review of adenomatoid mesothelioma in the abdominal cavity.
Case presentation
The patient was a 61-year-old Japanese woman who had undergone resection of a malignant peripheral nerve sheath tumor of the hand 18 years prior. She was diagnosed with clinical stage I lung adenocarcinoma on follow-up chest radiography. Simultaneously, a 20-mm enhancing nodule with slow growth on the right diaphragm was detected on contrast-enhanced computed tomography. She presented no specific clinical symptoms. At this point, the lesion was suspected to be a hypervascular tumor of borderline malignancy, such as a solitary fibrous tumor. After a left upper lobectomy for lung adenocarcinoma, she was referred to our department, and laparoscopic tumor resection was performed. Adenomatoid tumors were also considered based on the histopathological and immunohistochemical analyses, but we made the final diagnosis of adenomatoid mesothelioma using the results of the genetic profile. The patient remains alive, with no recurrence noted 6 months after surgery.
Conclusion
We encountered a valuable case of adenomatoid mesothelioma of peritoneal origin. There are some previously reported cases of adenomatoid mesothelioma and adenomatoid tumors that may need to be recategorized according to the current classification. It is important to accumulate and share new findings to clarify the clinicopathological characteristics and genetic status of adenomatoid mesothelioma.
“…The cases presented here highlight the possibility of encountering metastasis of malignant mesothelioma to skin, and definitive pathological diagnosis is important, especially in a patient with relevant history. To our knowledge, there have only been two cases reported 34,35 of unexpected metastatic mesothelioma, one presenting as a gastric polyp 34 and the other as multiple hypervascular masses on the liver surface, pelvic cavity, and anterior peritoneum 35 ; however, none reported to involve skin. Although an uncommon presentation, mesothelioma should be included in the differential diagnosis of tumors that may metastasize to the skin.…”
Malignant mesothelioma is a locally aggressive malignancy most commonly arising from the pleural and/or peritoneal cavity. Distant cutaneous metastasis is extremely rare. Here, we describe two cases of mesothelioma metastatic to the head and neck skin. Case 1: A 64‐year‐old man diagnosed previously with extensive thoracic and abdominal mesothelioma, developed a rapidly growing right upper lip lesion, for which a wedge resection was performed. Case 2: A 77‐year‐old woman with a history of pleural mesothelioma developed a firm, mobile subcutaneous nodule on the right lateral forehead, clinically thought to represent either an epidermal inclusion cyst or a lipoma. A punch biopsy was performed. In both cases, histopathologic evaluation revealed dermal proliferation of epithelioid cells with moderate cytologic atypia and three mitotic figures per mm2 and two mitotic figures per mm2 for Cases 1 and 2, respectively. Immunohistochemical studies revealed the lesional cells to be positive for WT1, mesothelin, D2‐40, CK5/6, while being negative for melanocytic and other keratinocytic markers, supporting a diagnosis of metastatic mesothelioma. Awareness of rare instances of cutaneous metastases from malignant mesothelioma is necessary to avoid possible misdiagnosis and ensure appropriate management.
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