Calcitriol-Mediated Hypercalcemia, Somatostatin Receptors Expression and 25-Hydroxyvitamin D3-1α- Hydroxylase in GIST Tumors

Herrera‐Martínez, Yiraldine; González, María José Contreras; Pedraza‐Arevalo, Sergio; Martínez, María del Carmen Guerrero; Martínez, Angela Rodrigo; Menchen, Alberto González; Molina, Maria Angeles Blanco; Gálvez-Moreno, María Ángeles; Vega, A.L. Moreno; Luque, Raúl M; Herrera‐Martínez, Aura D.

doi:10.3389/fendo.2021.812385

Cited by 3 publications

(3 citation statements)

References 23 publications

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“…The underlying mechanisms driving hypercalcaemia in these patients with GIST were identified in six of the previous documented cases. Interestingly, four patients were found to have raised calcitriol levels (1,25(OH)2-vitamin D 3 ) 21 23 25 26. Vitamin D levels were noted to be insufficient in our case.…”

Section: Discussionmentioning

confidence: 53%

“…Conversely, hypercalcaemia resulting from overproduction of calcitriol in solid tumours is very rare and the mechanism remains unclear 27. However, molecular analysis of tumour cells from a patient with GIST presented by Herrera-Martinez et al demonstrated mRNA expression of 1α-hydroxylase and vitamin D receptors in GIST tumour cells, confounding the proposed mechanism of calcitriol-mediated hypercalcaemia in patients with GIST 26. Identifying this underlying aetiology of hypercalcaemia in patients with GIST is imperative for future management, as recent data have suggested patients with calcitriol elevation are less likely to respond to antiresorptive therapy than patients without calcitriol elevation 27…”

Section: Discussionmentioning

confidence: 99%

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Gastrointestinal stromal tumour-induced hypercalcaemia

Trotter,

White

2023

BMJ Case Rep

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Hypercalcaemia is recognised as the most common oncological metabolic emergency, with several proposed underlying mechanisms. Nevertheless, hypercalcaemia has been rarely reported as a complication in patients with gastrointestinal stromal tumours (GISTs). GISTs are uncommon mesenchymal tumours of the gastrointestinal tract. There are only nine previous cases of hypercalcaemia occurring in patients with GIST reported in the literature. We report a case of a man in his 70s with a background of metastatic GIST on fourth-line treatment. The patient presented with new hypercalcaemia and acute kidney injury. Despite medical management, his calcium remained elevated and he deteriorated secondary to significant disease progression.

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Section: Discussionmentioning

confidence: 53%

Section: Discussionmentioning

confidence: 99%

Gastrointestinal stromal tumour-induced hypercalcaemia

Trotter,

White

2023

BMJ Case Rep

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“…The secretion of 1,25(OH)2D accounts for approximately 1% of cases of HM [ 1 ]. It is primarily associated with hematologic malignancies such as Hodgkin and non-Hodgkin lymphoma but has also been reported in some solid tumors such as ovarian dysgerminoma, gastrointestinal stromal tumor (GIST), and seminomas [ 1 , 5 ]. Here, we present a patient with abdominal liposarcoma and HM caused by the production of 1,25(OH)2D.…”

Section: Introductionmentioning

confidence: 99%

Malignant Hypercalcemia Due to the Ectopic Production of Calcitriol by an Abdominal Liposarcoma

Figueiredo¹,

Pereira²,

Cabrera³

et al. 2023

Cureus

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Hypercalcemia of malignancy (HM) is a common form of paraneoplastic syndrome associated with a poor prognosis of the disease. In solid tumors, HM occurs mainly due to the production of parathyroid hormonerelated peptide (PTHrP). We present a case of a 60-year-old male with a 25 cm retroperitoneal liposarcoma diagnosed with severe hypercalcemia (16.8 mg/dL) by a preoperative blood sampling. Hypercalcemia workup showed suppressed parathyroid hormone (PTH), normal PTHrP, and high 1,25-dihydroxyvitamin D (1,25(OH)2D) serum levels. After surgery, hypercalcemia and calcitriol levels normalized. Immunohistochemical analysis of the tumor showed 1α-hydroxylase expression by tumor cells. To our knowledge, this is the first case of liposarcoma-associated hypercalcemia caused exclusively by the ectopic production of calcitriol. Despite being a rare cause of hypercalcemia, measuring 1,25(OH)2D should be considered in the workup of a patient with high serum calcium levels, suppressed PTH, and normal PTHrP.

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Hypercalcaemia in gastrointestinal stromal tumour and sarcoidosis: a case report

Selvananthan,

Fischer,

Lin

2024

BMC Nephrol

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Background Hypercalcaemia is a common manifestation of sarcoidosis but is sparingly described in gastrointestinal stromal tumours (GISTs). We describe a case of acute kidney injury and hypercalcemia resulting from simultaneous diagnosis of GIST and sarcoidosis, the presentation of which has not yet been reported. Case Presentation A 61-year-old male presented with acute kidney injury and hypercalcemia, with elevated 1,25-dihydroxyvitamin D levels. Investigations demonstrated a large gastric antral mass which was resected and proven to be GIST. Histopathology of incidentally found liver nodules revealed non-necrotising epithelioid granulomas consistent with concomitant sarcoidosis. The hypercalcemia was successfully treated with bisphosphonate therapy, resection of the GIST and a four month course of corticosteroids, which was truncated due to a mycobacterial infection. Conclusions Our case report is the first to describe hypercalcemia due to GIST and biopsy-proven sarcoidosis, thereby raising the possibility of a common pathophysiological pathway relating the two entities. We review the literature describing the mechanisms of hypercalcaemia in GIST and the association between GIST and sarcoidosis.

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Calcitriol-Mediated Hypercalcemia, Somatostatin Receptors Expression and 25-Hydroxyvitamin D3-1α- Hydroxylase in GIST Tumors

Cited by 3 publications

References 23 publications

Gastrointestinal stromal tumour-induced hypercalcaemia

Gastrointestinal stromal tumour-induced hypercalcaemia

Malignant Hypercalcemia Due to the Ectopic Production of Calcitriol by an Abdominal Liposarcoma

Hypercalcaemia in gastrointestinal stromal tumour and sarcoidosis: a case report

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