Ovarian dysgerminoma presenting with hypercalcaemia. Case report and review of the literature

Anstey, Alexander Vincent; Gowers, L.; Vass, Alex; Robson, A. O.

doi:10.1111/j.1471-0528.1990.tb02556.x

Cited by 9 publications

(4 citation statements)

References 16 publications

(7 reference statements)

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“…Although previous reports have documented hypercalcemia in women with dysgerminomas, [21][22][23][24] the contribution of 1,25(OH) 2 D 3 to this problem has yet to be fully defined. Data presented here suggest that the cause of HHM in dysgerminomas is distinct from that observed for other types of cancer, with tumor-associated 1␣-hydroxylase activity being the putative etiological mechanism.…”

Section: Discussionmentioning

confidence: 97%

“…20 Compared to other tumors, dysgerminomas have a relatively low incidence of hypercalcemia, although there have been previous reports documenting raised serum calcium and 1,25(OH) 2 D 3 with dysgerminomas and seminomas. [21][22][23][24][25] We have used a collection of 12 dysgerminomas to assess expression of 1␣-hydroxylase, and to relate this to circulating hormone levels, macrophage markers, and other components of vitamin D metabolism and signaling. Data indicate that abnormal expression of 1␣-hydroxylase in dysgerminomas may result in a form of HHM distinct from that seen in PTHrP-secreting tumors, and which is associated with increased circulating levels of 1,25(OH) 2 D 3 .…”

Section: Humoral Hypercalcemia Of Malignancy (Hhm) Is a Common Paranementioning

confidence: 99%

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Increased Expression of 25-Hydroxyvitamin D-1α-Hydroxylase in Dysgerminomas

Evans

Taylor

Zehnder

et al. 2004

The American Journal of Pathology

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Humoral hypercalcemia of malignancy (HHM) is a common paraneoplastic disorder usually associated with increased synthesis of parathyroid hormone-related peptide (PTHrP). Unlike non-cancer forms of hypercalcemia, HHM does not routinely involve increased circulating levels of the active form of vitamin D, 1,25-dihydroxyvitamin D3 (1,25(OH)2D3). Dysgerminomas are a notable exception to this rule, previous reports having described hypercalcemia with elevated serum 1,25(OH)2D3. To investigate the etiology of this form of HHM we have characterized expression and activity of the enzyme that catalyzes synthesis of 1,25(OH)2D3, 25-hydroxyvitamin D-1alpha-hydroxylase (1alpha-hydroxylase), in a collection of 12 dysgerminomas. RT-PCR analyses indicated that mRNA for 1alpha-hydroxylase was increased 222-fold in dysgerminomas compared to non-tumor ovarian tissue. Parallel enzyme assays in tissue homogenates showed that dysgerminomas produced fivefold higher levels of 1,25(OH)2D3 compared to normal ovarian tissue. Immunolocalization studies indicated that 1alpha-hydroxylase was expressed by both tumor cells and by macrophages within the inflammatory cell infiltrate associated with dysgerminomas. The immunological nature of the increased 1,25(OH)2D3 production observed in dysgerminomas was further emphasized by correlation between expression of 1alpha-hydroxylase and the endotoxin recognition factors CD14 and toll-like receptor 4 (TLR4). These data suggest that inflammatory mechanisms associated with dysgerminomas are the underlying cause of the increased expression and activity of 1alpha-hydroxylase associated with these tumors. We further postulate that this autocrine/paracrine action of 1alpha-hydroxylase may lead to increased circulating levels of 1,25(OH)2D3 and a form of HHM which is distinct from that seen with PTHrP-secreting tumors.

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Section: Discussionmentioning

confidence: 97%

Section: Humoral Hypercalcemia Of Malignancy (Hhm) Is a Common Paranementioning

confidence: 99%

Increased Expression of 25-Hydroxyvitamin D-1α-Hydroxylase in Dysgerminomas

Evans

Taylor

Zehnder

et al. 2004

The American Journal of Pathology

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“…Dysgerminoma is the most common malignant ovarian tumor in adolescents; 80% of cases occur in the first 2 decades of life. Despite the number of cases each year, it is very infrequently described with hypercalcemia 4–7. Certain ovarian tumors, such as small cell carcinoma of the ovary or ovarian adenocarcinoma, which are rare in children, are more likely to be associated with hypercalcemia.…”

Section: Discussionmentioning

confidence: 99%

Severe malignancy‐associated hypercalcemia in dysgerminoma

Richardson

Otis

Sprinz

2005

Pediatric Blood & Cancer

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Hypercalcemia is a rare complication of pediatric malignant germ cell tumors. The problem may be missed because of unawareness among pediatric oncologists. We describe a 16-year-old girl with an ovarian dysgerminoma associated with severe hypercalcemia, a metabolic abnormality infrequently reported with this disease. We review some of the potential causes of malignancy-associated hypercalcemia and current treatment strategies. It is our recommendation that calcium levels should be monitored in all children with solid ovarian masses. Hypercalcemia seen in these situations may not improve until the tumor is removed.

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“…There have been only 6 previously reported cases of hypercalcemia associated with dysgerminoma in the literature. [7][8][9][10][11][12] To highlight this potentially fatal association, we report an additional case of dysgerminoma associated with hypercalcemia.…”

mentioning

confidence: 99%

Dysgerminoma associated with hypercalcemia: A case report

Okoye

Harmston

Buick

2001

Journal of Pediatric Surgery

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Hypercalcemia is an extremely rare paraneoplastic manifestation of malignancy in children. The authors report a case of dysgerminoma in a 14-year-old girl associated with severe hypercalcemia that only resolved after excision of the tumor. There are only 6 previously reported cases of dysgerminoma associated with hypercalcemia. Serum calcium levels should be checked in all children with solid ovarian tumors. Hypercalcemia in such circumstances may be resistant to medical management, and surgical resection of the tumor results in the normalization of calcium levels.

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Ovarian dysgerminoma presenting with hypercalcaemia. Case report and review of the literature

Cited by 9 publications

References 16 publications

Increased Expression of 25-Hydroxyvitamin D-1α-Hydroxylase in Dysgerminomas

Increased Expression of 25-Hydroxyvitamin D-1α-Hydroxylase in Dysgerminomas

Severe malignancy‐associated hypercalcemia in dysgerminoma

Dysgerminoma associated with hypercalcemia: A case report

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