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A rare case of regression of brown tumors of tertiary hyperparathyroidism after parathyroidectomy and renal transplant: A 5-year follow-up

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ABSTRACT

Tertiary hyperparathyroidism (HPT) is a rare condition that affects patients with secondary HPT, which develop hyperplasia of the parathyroid glands, thus causing an increase in parathyroid hormone levels. Bone alterations are the main consequences of this condition including the development of osteolytic lesions called brown tumor. This article reports an unusual case of brown tumors located in the maxilla and mandible in a 19-year-old man with chronic renal failure with hyperplasia of the parathyroid glands. The lesions regressed approximately 5 months after the parathyroidectomy. At this same time, the patient underwent renal transplant. The patient was followed for 5 years, showing improvement in overall clinical status. There was also improvement of the results of laboratory tests and the pattern of trabecular bone. The correct diagnosis of oral lesions was of great relevance for the conservative treatment could have been chosen.

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(a) Intraoral photograph of maxillary lesion showing healing with a small residual deformity; (b) Intraoral photograph of mandibular lesion showing healing
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Figure 11: (a) Intraoral photograph of maxillary lesion showing healing with a small residual deformity; (b) Intraoral photograph of mandibular lesion showing healing

Mentions: A 19-year-old man, Caucasian, was referred to our Oral and Maxillofacial Surgery Service for evaluation of swellings in the maxilla and mandible. The patient had been diagnosed with secondary HPT due to chronic renal failure in the final stage, whose cause was a urinary tract infection. The patient was in a generally debilitated state, making hemodialysis sessions 3 times a week [Figure 1]. Physical examination revealed that he was short of stature and had vertebral scoliosis. Intraoral inspection revealed an asymptomatic swelling in the anterior palate with a bony consistency, with crackling on some points, measuring approximately 5.0 cm in diameter [Figure 2], which had commenced 2 years previously. Intraoral examination also showed an increase in volume in the anterior region of the mandible with a bony consistency, asymptomatic, measuring approximately 4.0 cm in diameter [Figure 3], also with the same time course. The teeth-related injuries had no mobility, had vitality, no resorption and were displaced. Radiographic examination demonstrated generalized demineralization of the bones. A skull radiograph disclosed a “ground-glass” or “salt and pepper” appearance of the calvarium and loss of cortical lines [Figure 4]. The panoramic radiograph revealed a thinning of the trabecular bone, resulting in an appearance similar to “ground-glass” or “salt and pepper” [Figure 5]. The intraoral radiograph showed a generalized loss of lamina dura of the most teeth of the maxilla and mandible [Figure 6a and b]. The X-rays of hands showed metastatic calcifications in the joints and subperiosteal resorption of the phalanges [Figure 7]. Laboratory tests were abnormal, confirming the diagnosis of HPT. Initial investigations showed elevated levels of PTH 1490 pg/ml, calcium 12.0 mg/dl, phosphate 6.1 mg/dl, and alkaline phosphatase 320 U/L. The hemoglobin level was 6.8 g/dl due to decreased levels of erythropoietin. From the above data, we opted for the incisional biopsy of lesions. The histopathology report showed giant cell lesion (brown tumors of HPT) [Figure 8]. The diagnosis was suggested by the clinical history and confirmed by biochemical, radiological, and histopathologic examinations. Parathyroid glands fine needle aspiration was performed. The result, associated with clinical and laboratory findings, revealed a case of hyperplasia of the parathyroid glands. Because of parathyroidectomy has already been scheduled, it was decided to follow the lesions with the expectation of remission after surgery of the parathyroid glands. Approximately, 2 months after our evaluation, after regression of the symptoms of anemia, the patient underwent parathyroidectomy. The biopsy performed confirmed the diagnosis of glandular hyperplasia. One of the parathyroid glands was reimplanted in the right forearm to maintain PTH levels within normal levels. Laboratory tests were within the normal levels 5 months after parathyroid surgery, with regression of the lesions, and the patient underwent renal transplantation. The patient was followed up for 5 years, showing improvement in overall clinical status [Figure 9]. The pattern of trabecular bone of the jaw has also improved [Figure 10a–d], and the lesions of the maxilla and mandible healed [Figure 11a and b]. We also observed the disappearance of metastatic calcification and supraperiosteal resorption of the phalanges [Figure 12a and b].


A rare case of regression of brown tumors of tertiary hyperparathyroidism after parathyroidectomy and renal transplant: A 5-year follow-up
(a) Intraoral photograph of maxillary lesion showing healing with a small residual deformity; (b) Intraoral photograph of mandibular lesion showing healing
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4979328&req=5

Figure 11: (a) Intraoral photograph of maxillary lesion showing healing with a small residual deformity; (b) Intraoral photograph of mandibular lesion showing healing
Mentions: A 19-year-old man, Caucasian, was referred to our Oral and Maxillofacial Surgery Service for evaluation of swellings in the maxilla and mandible. The patient had been diagnosed with secondary HPT due to chronic renal failure in the final stage, whose cause was a urinary tract infection. The patient was in a generally debilitated state, making hemodialysis sessions 3 times a week [Figure 1]. Physical examination revealed that he was short of stature and had vertebral scoliosis. Intraoral inspection revealed an asymptomatic swelling in the anterior palate with a bony consistency, with crackling on some points, measuring approximately 5.0 cm in diameter [Figure 2], which had commenced 2 years previously. Intraoral examination also showed an increase in volume in the anterior region of the mandible with a bony consistency, asymptomatic, measuring approximately 4.0 cm in diameter [Figure 3], also with the same time course. The teeth-related injuries had no mobility, had vitality, no resorption and were displaced. Radiographic examination demonstrated generalized demineralization of the bones. A skull radiograph disclosed a “ground-glass” or “salt and pepper” appearance of the calvarium and loss of cortical lines [Figure 4]. The panoramic radiograph revealed a thinning of the trabecular bone, resulting in an appearance similar to “ground-glass” or “salt and pepper” [Figure 5]. The intraoral radiograph showed a generalized loss of lamina dura of the most teeth of the maxilla and mandible [Figure 6a and b]. The X-rays of hands showed metastatic calcifications in the joints and subperiosteal resorption of the phalanges [Figure 7]. Laboratory tests were abnormal, confirming the diagnosis of HPT. Initial investigations showed elevated levels of PTH 1490 pg/ml, calcium 12.0 mg/dl, phosphate 6.1 mg/dl, and alkaline phosphatase 320 U/L. The hemoglobin level was 6.8 g/dl due to decreased levels of erythropoietin. From the above data, we opted for the incisional biopsy of lesions. The histopathology report showed giant cell lesion (brown tumors of HPT) [Figure 8]. The diagnosis was suggested by the clinical history and confirmed by biochemical, radiological, and histopathologic examinations. Parathyroid glands fine needle aspiration was performed. The result, associated with clinical and laboratory findings, revealed a case of hyperplasia of the parathyroid glands. Because of parathyroidectomy has already been scheduled, it was decided to follow the lesions with the expectation of remission after surgery of the parathyroid glands. Approximately, 2 months after our evaluation, after regression of the symptoms of anemia, the patient underwent parathyroidectomy. The biopsy performed confirmed the diagnosis of glandular hyperplasia. One of the parathyroid glands was reimplanted in the right forearm to maintain PTH levels within normal levels. Laboratory tests were within the normal levels 5 months after parathyroid surgery, with regression of the lesions, and the patient underwent renal transplantation. The patient was followed up for 5 years, showing improvement in overall clinical status [Figure 9]. The pattern of trabecular bone of the jaw has also improved [Figure 10a–d], and the lesions of the maxilla and mandible healed [Figure 11a and b]. We also observed the disappearance of metastatic calcification and supraperiosteal resorption of the phalanges [Figure 12a and b].

View Article: PubMed Central - PubMed

ABSTRACT

Tertiary hyperparathyroidism (HPT) is a rare condition that affects patients with secondary HPT, which develop hyperplasia of the parathyroid glands, thus causing an increase in parathyroid hormone levels. Bone alterations are the main consequences of this condition including the development of osteolytic lesions called brown tumor. This article reports an unusual case of brown tumors located in the maxilla and mandible in a 19-year-old man with chronic renal failure with hyperplasia of the parathyroid glands. The lesions regressed approximately 5 months after the parathyroidectomy. At this same time, the patient underwent renal transplant. The patient was followed for 5 years, showing improvement in overall clinical status. There was also improvement of the results of laboratory tests and the pattern of trabecular bone. The correct diagnosis of oral lesions was of great relevance for the conservative treatment could have been chosen.

No MeSH data available.


Related in: MedlinePlus