Case Report |
Corresponding author: Deyan Neychev ( dneitchev@yahoo.com ) © 2022 Deyan Neychev, Bozhidar Pilichev, Denitsa Serteva, Petya Kanazirska.
This is an open access article distributed under the terms of the Creative Commons Attribution License (CC BY 4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Citation:
Neychev D, Pilichev B, Serteva D, Kanazirska P (2022) A rare case of a giant cell tumour caused by vitamin D deficiency. Folia Medica 64(4): 676-681. https://doi.org/10.3897/folmed.64.e63623
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Reparative giant cell granulomas are benign masses of multi-etiological nature, which account for 1%–7% of all benign lesions of the jaws. The objective of this case report is to present the relationship between isolated vitamin D deficiency and the development of reparative giant cell granuloma.
Herein, we present the case of a 70-year-old female patient with a painless mass of increased mobility in the mandibular region, and pain in the involved teeth. After histological confirmation and laboratory screening, а reparative giant cell granuloma caused by serious deficiency of vitamin D3 - (25-OH)D was diagnosed. The treatment protocol included surgical removal of the lesion and vitamin D replacement therapy.
In rare cases, this type of lesion can be a primary manifestation of vitamin D deficiency; therefore, it is extremely important to be aware of this pathology.
mandible, reparative giant cell granuloma, vitamin D deficiency
The giant cell tumour is not a true neoplasm, but rather a reactive condition caused by factors such as injury, inflammation, or hormonal factors.[
Despite being a benign lesion, giant cell granuloma can occur in two clinical subforms - non-aggressive and aggressive.[
A clinically and histologically similar lesion may occur in the event of elevated parathyroid hormone (PTH) levels with impaired calcium-phosphorus metabolism, which necessitates differentiation from the classic brown tumour in hyperparathyroidism.[
A 70-year-old female patient visited our clinic with complaints of swelling and spontaneous pain in the right mandible. Her past history was unremarkable; she had had well-controlled arterial hypertension for 2 years, and before the initiation of systemic treatment, her arterial blood pressure was up to 180/90. Clinical examination revealed a tumour in the mandible, located in the right premolar region. The formation had firm texture and smooth surface, and was painless on palpation. Increased mobility of teeth 43 and 44 was found (Fig.
Panoramic radiograph showed a mono-lacunar formation involving the canine and the premolar in this area, without root resorption of the adjacent teeth. Cone-beam computed tomography (CBCT), more specifically images in coronal, sagittal, and axial plane and 3D reconstruction showed a hypodense oval lesion around the roots of teeth 43 and 44, which had homogeneous structure, dimensions of 11.35×9.43 mm and density of 200 HU (Fig.
The histological analysis found stroma rich in spindle-shaped fibroblasts, among which multinucleate giant osteoclast-like cells were disseminated, with less than 20 nuclei in the cytoplasm (Fig.
Under local anesthesia with 4% articaine, the patient underwent surgery - resection with margins in healthy bone tissue and extraction of the teeth adjacent to the lesion. No changes were found during the further examinations performed to assess the gland. Elevated levels of PTH and alkaline phosphatase were found by laboratory screening tests. After further measurement of the level of 25-(OH)D, deficiency was confirmed and replacement therapy was prescribed. During the postoperative period, the surgical field was calm. No changes in the jawbone were found six months postoperatively (Fig.
Fibrous stroma with multiple giant osteoclast-like cells in and around a fresh hemorrhage and osteoid trabeculae with a zone of ossification, H&E stain, ×40.
Fibrous stroma with multiple giant osteoclast-like cells near and in a fresh haemorrhage, H&E stain, ×10.
The giant cell reparative granuloma of the jaws is a rare lesion, more common in females (in a 2:1 ratio), which can be explained with the relationship between hormone secretion and its manifestation in females.[
Histologically, the lesion consists of fibrous tissue composed of areas of hemorrhage, aggregations of multinucleate giant cells and, in some places, osteoid trabeculae - findings corresponding to the findings in the presented clinical case. Giant cells are commonly located on vascular stroma with hemosiderin deposits. Giant cells are also found in other giant cell lesions such as aneurysmal bone cysts, giant cell tumours, cherubism, and brown tumours. In giant cell tumours, giant cells are evenly distributed, in contrast to giant cell granulomas in which clusters of these cells are observed.[
Giant cell granuloma as a primary manifestation of vitamin D deficiency is a relatively rare condition described in the literature. In most such cases, impairment of parathyroid glands is found, therefore, it is necessary to assess their function. Primary hyperparathyroidism in 80% of cases is associated with an adenoma, more commonly located in the lower pair of glands or ectopically – in the thyroid gland, the mediastinum or the thymus, and, in rare cases, it is due to glandular hyperplasia (15% to 20%) or carcinoma (<0.5%).[
The differential diagnosis should also include a brown tumour due to secondary hyperparathyroidism, which may be due to chronic renal failure, vitamin D deficiency, lower intestinal calcium absorption, and liver disease.[
When treating giant cell granuloma, a number of factors must be taken into account such as the anatomical characteristics, the clinical behavior of the lesion[
Giant cell granuloma can be caused by multiple factors related to patient’s general health. In rare cases, this lesion may be a primary manifestation of vitamin D deficiency, therefore, it is essential to be aware of this pathology, to carry out the necessary diagnostic tests and to administer adequate and timely treatment. Vitamin D should be taken into account as an etiological factor for the occurrence of this condition with the view of making correct differential diagnosis with other possible diseases or similar conditions.