Expansive jaw lesions in chronic kidney disease: review of the literature and a report of two cases

doi:10.1016/j.oooo.2014.11.002

Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology

Volume 119, Issue 3, March 2015, Pages 340-345

https://doi.org/10.1016/j.oooo.2014.11.002 Get rights and content

Chronic kidney disease is an increasing public health problem, with a worldwide prevalence estimated to be between 8% and 16%. The metabolic alterations induce bone and soft tissue changes, and the encompassing term chronic kidney disease–mineral and bone disorder syndrome (CKD-MBD) is used to describe them. The cardinal manifestations of the syndrome are bone catabolism and soft tissue calcifications, which ultimately compromise the cardiovascular and skeletal systems. In rare cases, tumorous enlargement of the craniofacial bones occurs. This article provides a brief review of the pathogenesis and imaging of craniofacial changes in CKD-MBD and reports on two cases of expansive jaw lesions. The term expansive renal osteitis fibrosa is recommended to describe these lesions.

Section snippets

Case 1

A 30-year-old female had been suffering from CKD for 11 years and was on hemodialysis for 2 years and ambulatory peritoneal dialysis for 9 years. She had noted a slowly progressing painless swelling of her palate (Figure 1). A posteroanterior skull radiograph showed a multilocular lesion affecting the palate and indiscrete foci of thickening bone of the calvaria, resulting in a cotton-wool appearance (Figure 2). Panoramic radiography demonstrated loss of cortical definition and lamina dura and

Case 2

A 31-year-old female was referred by her physician with a main complaint of a tumor involving the anterior mandible. The lesion had enlarged slowly over 3 years and showed significant labial expansion and tooth displacement (Figure 6). She had been diagnosed with chronic kidney disease 13 years previously and has been on peritoneal dialysis since. Figure 7 shows the radiologic features of the well-defined, expansive, multilocular, mixed radiolucent–radiopaque lesion with displacement of teeth.

Discussion

The pivotal event in the pathogenesis of CKD-MBD is suspension of the activation of vitamin D by the diseased kidneys, which results in the reduction of the renal retention and gastrointestinal absorption of calcium. The resulting hypocalcemia induces secondary hyperparathyroidism with the release of parathyroid hormone (PTH), which activates osteoclasts, releasing skeletal calcium⁸ and fibroblast growth factor 23.⁹ Fibroblast growth factor 23 increases the catabolism of active vitamin D and

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Radiographic and microscopic features of a case of brown tumor and osteitis fibrosa simultaneously occurring in a patient are shown in Figure 4. Moreover, diffuse jaw enlargement in renal osteodystrophy has been classified as uremic leontiasis ossea or just leontiasis ossea.46,49 However, because “leontiasis ossea” is a nonspecific clinical term and is also applied to other causes of facial bone enlargement, such as fibrous dysplasia and Paget disease of bone, some authors recommend the term “expansive renal osteitis fibrosa” as a more appropriate term regarding the CKD-MBD context.49
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Moreover, if alkaline phosphatase is elevated, it indicates high bone turnover. The latter may also be present in Paget disease.7 Approximately 8%-16% of the worldwide population suffers from a chronic kidney disease,8 and 4.3% of them present with craniofacial brown tumors as a manifestation of hyperparathyroidism.9
We present the case of a 32-year-old male patient complaining of recurrent mandibular pain for 3.5 years. Panoramic radiography indicated increased cortical density of the mandibular lower border. Scintigraphy and single-photon emission computed tomography revealed metabolic hyperactivity in that region without pathologic lymph nodes. A bone biopsy specimen of the mandibular lower border did not have inflammation or cytologic atypia. Endocrinologic investigation confirmed secondary hyperparathyroidism as a result of hypovitaminosis D. Several weeks after starting therapy with oral vitamin D supplements, the symptoms completely disappeared. Increased cortical density is a rare manifestation of secondary hyperparathyroidism, which normally causes the lamina dura to vanish and produces a ground-glass appearance as a result of blurring of the trabecular bone pattern. Because focal hyperostosis can have multiple benign or malignant causes, radiologic examination of the jaw bones is indispensable for evaluating orofacial pain. Increased cortical density may be caused by metabolic diseases, requiring further investigations, including biopsy and blood analysis.

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Oral and maxillofacial radiologyExpansive jaw lesions in chronic kidney disease: review of the literature and a report of two cases

Section snippets

Case 1

Case 2

Discussion

Lancet

J Am Coll Cardiol

Am J Kidney Dis

Int J Oral Maxillofac Surg

Int J Oral Maxillofac Surg

Oral Surg Oral Med Oral Pathol Oral Radiol Endod

Am J Otolaryngol

KDIGO 2012 Clinical practice guidelines for the evaluation and management of chronic kidney disease

Kidney Int Suppl

Unresolved lytic lesions following parathyroidectomy in a patient with chronic renal failure

J Bone Joint Surg Br