1995
DOI: 10.1097/00000478-199501000-00007
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Feminizing Sertoli Cell Tumors in Boys with Peutz-Jeghers Syndrome

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Cited by 132 publications
(73 citation statements)
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“…A markedly increased incidence of carcinomas of the gastrointestinal tract including stomach, small bowel, colon and pancreatic cancer, as well as breast, ovary, uterus, cervix, lung and testis cancer has been observed through longitudinal studies of affected kindreds (Giardiello et al, 1987(Giardiello et al, , 2000Spigelman et al, 1989;Gruber et al, 1998;Lim et al, 2004;Hearle et al, 2006). In addition, rare tumors have been associated with PJS including those of a reproductive origin-including testicular and ovarian sex cord tumors, Sertoli cell tumors and adenoma malignum of the cervix-as well as non-adenocarcinoma pancreatic tumors, including pancreatic intraductal papillary mucinous neoplasia and serous cystadenomas (Podczaski et al, 1991;Young et al, 1995;Tomlinson and Houlston, 1997;Su et al, 1999;Sato et al, 2001;Yee et al, 2003).…”
Section: Peutz-jeghers Syndrome and Human Cancer Geneticsmentioning
confidence: 99%
“…A markedly increased incidence of carcinomas of the gastrointestinal tract including stomach, small bowel, colon and pancreatic cancer, as well as breast, ovary, uterus, cervix, lung and testis cancer has been observed through longitudinal studies of affected kindreds (Giardiello et al, 1987(Giardiello et al, , 2000Spigelman et al, 1989;Gruber et al, 1998;Lim et al, 2004;Hearle et al, 2006). In addition, rare tumors have been associated with PJS including those of a reproductive origin-including testicular and ovarian sex cord tumors, Sertoli cell tumors and adenoma malignum of the cervix-as well as non-adenocarcinoma pancreatic tumors, including pancreatic intraductal papillary mucinous neoplasia and serous cystadenomas (Podczaski et al, 1991;Young et al, 1995;Tomlinson and Houlston, 1997;Su et al, 1999;Sato et al, 2001;Yee et al, 2003).…”
Section: Peutz-jeghers Syndrome and Human Cancer Geneticsmentioning
confidence: 99%
“…83 In terms of the testis, the sex cords are the site of tumorigenesis and a total of 35 boys with PJS have presented with testicular Sertoli cell tumors. 36,37,39,59,81,[84][85][86][87][88][89][90][91][92][93][94][95][96][97][98][99][100] In boys with PJS, there is an increase of aromatase expression and activity in these tumors, resulting in excess estradiol production that not only acts to feed local tumor growth, 36,39 but also accelerates testicular and extragonadal morphogenesis. For instance, the obvious clinical manifestations are enlarged testes, pre-pubertal gynecomastia and feminizing precocious puberty, and it has become increasingly numbers and the fact that spermatozoa in these mice display decreased motility, as evidenced by the proven inability to fertilize oocytes in vitro.…”
Section: Clinical Casesmentioning
confidence: 99%
“…67 Microscopically, SCTATs result in clusters of enlarged annular seminiferous tubules nestled among seminiferous tubules of normal size and appearance. 100 The enlarged seminiferous tubules are composed of hypertrophied Sertoli cells enclosed by a thickened basement membrane, with no evidence of germ cells. 100 Moreover, the tumors in boys with PJS can in some instances show signs of calcification distributed throughout the tumor, which is a hallmark for the diagnosis of a large cell calcifying Sertoli cell tumor (LCCSCT).…”
Section: Arommentioning
confidence: 99%
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“…Testicular sex cord and Sertoli-cell tumours may occur in prepubertal boys affected with PJS leading to sexual precocity and gynaecomastia (Wilson et al, 1986;Coen et al, 1991;Young et al, 1995). The production of oestrogen in ovarian tumours in girls with PJS has also been reported causing isosexual precocity (Sohl et al, 1983).…”
mentioning
confidence: 99%