Two Cases of Hypercalcemia in Pediatric Ovarian Dysgerminoma

Hosseini, B.; Leibl, Marli; Stoffman, Jayson; Morris, Amanda Sheffield

doi:10.1016/j.jogc.2018.05.004

Cited by 8 publications

(6 citation statements)

References 14 publications

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“…Previous data reported associations of dysgerminoma with paraneoplastic hypercalcemia [1,11,21,46,47]. Furthermore, a recently published study sustains that hypercalcemia is associated with elevated parathyroid hormone, parathyroid hormone-related protein (PTH-rP), 1,25 dihydroxyvitamin D [47] and hemophagocytic lymphohistiocytosis. We had no information regarding PTH level in our patient.…”

Section: Discussionmentioning

confidence: 92%

Uncommon Metastasis of Ovarian Dysgerminoma: A Case Report and Review of the Literature

et al. 2021

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Ovarian malignant germ cell tumors (OMGCT) represent less than 10% of all ovarian tumors. Dysgerminoma is the most common malignant primitive germ cell tumor in young women, known for its curability and low propensity to invade and metastasize when diagnosed early. Herein, we report an unusual type of ovarian dysgerminoma (OD) metastasis with a brief review of the literature, lacking similar reported cases. To our knowledge, although there are several case reports of dysgerminoma metastases with variable anatomic location and presentation, vaginal metastasis has not been previously described. The local or systemic relapse together with local and distant metastasis is considered as an independent predictor of poor survival in patients with OD. In light of the absence of mutations status, our patient successfully responded to therapy. Currently, the patient remains in clinical remission. A specific follow-up plan is ongoing knowing that ovarian dysgerminomas tend to recur most often in the first 2–3 years after treatment.

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Section: Discussionmentioning

confidence: 92%

Uncommon Metastasis of Ovarian Dysgerminoma: A Case Report and Review of the Literature

et al. 2021

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“…Previous reports have indicated that lymphomas secrete calcitriol ectopically, while some reports suggest that calcitriol secreting tumors lead to hypercalcemia in dysgerminoma. 6 , 11 , 19 Furthermore, some studies report that 1α-hydroxylase is focally expressed in dysgerminoma tumor cells. 9 , 11 Consequently, it has been suspected that 1α-hydroxylase, which is expressed excessively in tumor cells, is the primary cause of hypercalcemia.…”

Section: Discussionmentioning

confidence: 99%

Hypercalcemia associated with dysgerminoma and elevation of calcitriol: A case report and review of the literature

Hara

Suwanai

Abe

et al. 2022

SAGE Open Medical Case Reports

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We report on the case of a patient with dysgerminoma, a rare germ cell tumor, which showed hypercalcemia with an elevation of 1α,25-dihydroxycholecalciferol (calcitriol). A 27-year-old nulliparous woman presented with hypercalcemia during the examination of a right ovarian tumor with an elevation of calcitriol, lactate dehydrogenase, and alkaline phosphatase. Fractional excretion of calcium was elevated, and intact parathyroid hormone was suppressed. After undergoing right salpingo-oophorectomy, the patient’s serum calcium and calcitriol returned to the normal range within a week. A literature search was conducted on the topic by reviewing databases for dysgerminoma showing hypercalcemia. We identified 14 patients from the literature and performed a pooled analysis, including the results of our case. However, most cases lack data that can help investigate the potential association between parathyroid hormone, parathyroid hormone-related protein, calcitriol, and phosphorus in hypercalcemia. Thus, more case reports that include additional information are required to fully elucidate the mechanism of hypercalcemia associated with dysgerminoma.

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“…La hipercalcemia relacionada con tumores puede ser causada por diferentes mecanismos fisiopatológicos, entre los que se encuentran la producción de un péptido similar a la PTH, el aumento de la producción de vitamina D, la producción de PTH por el tumor o la resorción ósea por metástasis. 4,8,9 El más frecuente es la producción de PTHrP o PTH-like, que genera un aumento del intercambio mineral óseo, de la reabsorción de calcio renal y de la absorción intestinal de calcio por incremento de la actividad de la alfa-1-hidroxilasa. Además, la inmovilización prolongada secundaria a la astenia puede actuar como agravante.…”

Section: Discussionunclassified

“…La hipercalcemia asociada a tumores malignos se encuentra descripta en 10-30 % de los pacientes adultos, pero sólo en 0,5-1,3 % de los cánceres pediátricos, generalmente en leucemia linfoblástica aguda, y aparece en menos del 5 % de los tumores ováricos. [3][4][5] La presentación clínica es inespecífica y aparece con valores de calcemia mayores a 12 mg/dl. El estreñimiento, la anorexia y las náuseas y vómitos suelen ser los síntomas iniciales, seguidos por manifestaciones renales (poliuria y polidipsia secundarios a hipercalciuria al inicio), neuromusculares (hipotonía y mialgias) y en último lugar, cardiovasculares (hipertensión arterial [HTA] y arritmias).…”

Section: Introductionunclassified

Hipercalcemia en paciente pediátrica con disgerminoma ovárico: a propósito de un caso

et al. 2021

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Presentación de casos clínicos RESUMENLa hipercalcemia asociada a tumores malignos es una entidad poco frecuente en pediatría (0,5-1,3 % de los cánceres pediátricos). Es causada por diferentes mecanismos fisiopatológicos y los síntomas de presentación suelen ser inespecíficos, pero potencialmente graves. Presentamos un caso clínico de una paciente de 12 años con diagnóstico de disgerminoma ovárico bilateral. La enfermedad se presentó con hipercalcemia grave, tratada con hiperhidratación asociada a diuréticos de asa, bifosfonatos y, por último, la resección quirúrgica del tumor, que permitió la resolución definitiva del cuadro. A pesar de tratarse de un trastorno hidroeléctrico poco habitual en pediatría, dada la potencial gravedad de la hipercalcemia, es importante la detección y el tratamiento tempranos, con el fin de evitar complicaciones en el corto y el largo plazo.

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Two Cases of Hypercalcemia in Pediatric Ovarian Dysgerminoma

Cited by 8 publications

References 14 publications

Uncommon Metastasis of Ovarian Dysgerminoma: A Case Report and Review of the Literature

Uncommon Metastasis of Ovarian Dysgerminoma: A Case Report and Review of the Literature

Hypercalcemia associated with dysgerminoma and elevation of calcitriol: A case report and review of the literature

Hipercalcemia en paciente pediátrica con disgerminoma ovárico: a propósito de un caso

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