Primary peritoneal mesotheliomas in children: a clinicopathological and immunohistochemical study of eight cases

Moran, César A.; Albores‐Saavedra, Jorge; Suster, Saul

doi:10.1111/j.1365-2559.2008.03029.x

Cited by 36 publications

(26 citation statements)

References 38 publications

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“…Among case reports and small series focusing on pediatric patients, the largest to date comprises only 13 patients with histologically confirmed mesothelioma. 7,9,10,12–14 …”

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confidence: 99%

Clinicopathologic and genetic characteristics of young patients with pleural diffuse malignant mesothelioma

2018

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Pleural diffuse malignant mesothelioma typically presents during the seventh decade of life and has poor prognosis. Recent epidemiologic studies have shown differences between young and older mesothelioma patients, but the biology of pleural mesothelioma in young patients is poorly understood. We studied the clinicopathologic and genetic characteristics in pleural mesothelioma patients aged 35 years and younger. Thirty-six consecutive pleural mesothelioma patients aged 35 years and younger were compared with 48 older patients. We examined demographic and clinical characteristics, histologic type, growth patterns, mitotic index, and nuclear grade on hematoxylin and eosin-stained slides, BAP1 protein expression by immunohistochemistry, and CDKN2A and NF2 deletions by fluorescence in situ hybridization. Clinicopathologic and cytogenetic results were compared between young and older groups, and correlated with overall survival. Young patients were more frequently women, reported less asbestos exposure, and had a greater frequency of prior therapeutic radiation and family history of breast cancer than older patients (P<0.05 each). There were no histologic differences between young and older patients (all P>0.05). CDKN2A deletion was less prevalent in young patients (P = 0.01), loss of BAP1 protein expression less frequent in young patients (P = 0.06), and NF2 deletion rates similar between groups (P>0.05 each). Median overall survival was 40 vs 26 months (P = 0.10) in young and older patients, respectively, and 47 vs 31 months (P = 0.04) when comparing patients with epithelioid histology only. High mitotic index and non-epithelioid histology were the only characteristics associated with a poor overall survival in young patients. Young patients with pleural mesothelioma have an equal sex distribution and are more likely to have a history of mantle radiation, family history of breast cancer, and lower rates of CDKN2A deletion than older patients. Our results suggest that pleural mesothelioma in young patients has distinctive clinical and genetic characteristics, despite some similarities to pleural mesothelioma in older patients.

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“…Among case reports and small series focusing on pediatric patients, the largest to date comprises only 13 patients with histologically confirmed mesothelioma. 7,9,10,12–14 …”

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confidence: 99%

Clinicopathologic and genetic characteristics of young patients with pleural diffuse malignant mesothelioma

2018

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“…In contrast to the much more frequent pleural mesotheliomas that occur in adults, peritoneal involvement predominates in children under the age of 15. 1 Only in a fraction of those affected is no history of asbestos exposure elicited. Our patient had no known exposure to asbestos and a negative family history of mesothelioma but had received TBI and imatinib chemotherapy.…”

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confidence: 99%

Metastatic Peritoneal Mesothelioma in a 21-Year-Old Female with Chronic Myelogenous Leukemia

GuptaRohan

NoorudeenShajeer

GallianiCarlos

et al. 2014

Journal of Adolescent and Young Adult Oncology

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We report the case of a 21-year-old female who presented with cervical lymphadenopathy and subsequently diagnosed with peritoneal mesothelioma. She had a medical history of Ph + chronic myelogenous leukemia, treated with total body radiation and bone marrow transplant 13 years earlier, then 10 years of imatinib for a relapse 3 years later. The mesothelioma responded well to combination therapy but recurred locally 3 years after diagnosis. This case report emphasizes that even without asbestos exposure, mesothelioma can present as a second malignancy in survivors of childhood cancer who were exposed to radiation therapy as part of their initial treatment.C hronic myelogenous leukemia (CML) is rarely seen in children. Secondary malignancies can present in patients who receive total body radiation (TBI) as a preparative regimen for bone marrow transplantation (BMT) for CML. We present a case of an unfortunate young female who suffered from two cancers-CML and mesothelioma-at a very young age.A 21-year-old female had been diagnosed with adult-type CML in 1997 at the age of 7. Her past medical history was unremarkable and her family history was positive only for breast cancer in both grandmothers at an advanced age. After confirmation of BCR-ABL translocation, she received an allogeneic matched unrelated BMT preceded by chemotherapy and TBI (1320 cGy in six fractions over 3 days). However, her disease control was temporary, and the patient had relapse of her CML in 2001. Fortunately, imatinib mesylate had just become available via a clinical trial for children, so she was started on this agent and had a complete cytogenetic response. In the years since, her quantitative PCR analysis for BCR-ABL kinase domain mutations has shown persistent major (highest level = 0.008%) or complete (undetectable level) molecular response.In the spring of 2010, the patient presented with left neck swelling. The left external jugular vein was distended and vague fullness was felt over her left lower neck. Computed tomography (CT) scan of the neck, chest, abdomen, and pelvis revealed a clot in the left internal jugular and subclavian veins and several enlarged left supraclavicular lymph nodes. Small bilateral pleural effusions were noted. A questionably abnormal cystic lesion was noted on the left adnexa. She was treated with enoxaparin and underwent left supraclavicular lymph node biopsy (Fig. 1).The biopsy tissue was felt to be a lymph node containing metastatic deposits of a carcinoma of unknown primary. Her immunohistochemical staining was positive for pan-keratin and cytokeratin 5/6 and negative for S-100, TTF-1, Villin, BRST-1, and WT-1. Since family history was positive for breast cancer in both grandmothers at an advanced age, she did not meet criteria for Li-Fraumeni or other genetic predisposition syndromes. Differential diagnosis included an adenocarcinoma (salivary gland, endometrial, pancreatic, or breast), mesothelioma, transitional cell carcinoma, squamous cell carcinoma, thymoma, or basal cell carcinoma. A subsequent posit...

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“…In general, mesotheliomas are more common in adults older than 50 years of age, but these tumors also may occur in children. 23,24 History of long-standing exposure to asbestos, whether confirmed or not, should prompt a careful analysis of the biopsy tissue. Mesotheliomas can occur without a history of asbestos exposure, as evidenced by cases described in children and housewives.…”

Section: Historical Aspectsmentioning

confidence: 99%

Tumors of the Pleura

2010

Tumors and Tumor-Like Conditions of the Lung and Pleura

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Primary peritoneal mesotheliomas in children: a clinicopathological and immunohistochemical study of eight cases

Abstract: Peritoneal malignant mesothelioma in children is a rare condition that can introduce difficulties in histopathological diagnosis.

Cited by 36 publications

References 38 publications

Clinicopathologic and genetic characteristics of young patients with pleural diffuse malignant mesothelioma

Clinicopathologic and genetic characteristics of young patients with pleural diffuse malignant mesothelioma

Metastatic Peritoneal Mesothelioma in a 21-Year-Old Female with Chronic Myelogenous Leukemia

Tumors of the Pleura

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