NDT Advance Access originally published online on April 23, 2007
Nephrology Dialysis Transplantation 2007 22(8):2398-2399; doi:10.1093/ndt/gfm232
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© The Author [2007]. Published by Oxford University Press on behalf of ERA-EDTA. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org
(Section Editor: G. Neild)
Renal osteodystrophy manifesting as localized enlargement of the jaw
Disciplina de Patologia Bucal, Faculdade de Odontologia da Universidade de São Paulo
Correspondence and offprint requests to: Patrícia Leite de Godoi Adachi, DDS, MS, Department of Oral Pathology, School of Dentistry-FOUSP, Av: Prof. Lineu Prestes, 2227 Cidade Universitária, CEP 05508-900, São Paulo, Brazil. Email: paricardo{at}usp.br
Keywords: jaw enlargement; osteitis fibrosa; renal osteodystrophy; secondary hyperparathyroidism
A 39-year-old woman presented with a 6-month history of expanding masses in both the upper and lower jaw. The lesions had enlarged slowly without pain. She had chronic renal disease (CRD) and had been receiving haemodialysis three times a week for 4 years.
On examination, circumscribed swellings covered by normal mucosa were seen in three different sites: lower vestibular molars region of the right side and palatal incisors region on upper and lower jaws (Figure 1A and B). The lesions were hard and painless on palpation. The radiographic examination showed ill-defined radiolucencies in the affected areas. Blood tests revealed anaemia, elevated serum creatinine, urea and parathyroid hormone (2013 pg/ml).
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Incisional biopsies from the three affected areas were performed. Microscopic examination of the biopsy specimens revealed a cellular fibrous lesion containing spindle fibroblastic cells permeated by numerous osteoid trabeculae. Marked osteoblastic riming of the trabeculae was noted. A few scattered multinucleated giant cells (Figure 2) and haemorrhage were also observed. The histopathological diagnosis was consistent with osteitis fibrosa.
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Discussion
Osteitis fibrosa is the most common type of renal osteodystrophy and it appears mainly as the result of secondary hyperparathyroidism [1]. This lesion occurs most often in the long bones, ribs and pelvis. Involvement of the jaw occurs with relative frequency and it may be the earliest clinical manifestation of the renal disease. However, renal osteodystrophy associated with jaw enlargement, especially when localized, is uncommon and appears to be confined to long-term dialysis patients [2].
Osteitis fibrosa develops by bone remodelling, characterized by proliferation of osteoblasts covering bone surfaces, as well as an increase in osteoblastic cells with cuboidal type of cellular morphology and an increased number of osteoclastic resorption lacunae. In addition, there is a prominent fibrous tissue accumulation around the trabecular surfaces produced by fibroblastic cells [1]. In some areas of the skeleton, such as jaws, tumour-like lesions consisting of vascular fibrous stroma with giant cells may develop [3]. Oral radiographic findings in renal osteodystrophy include altered trabecular pattern, diffuse ground glass appearance and bone resorption with loss of lamina dura [2].
As giant cell lesions of hyperparathyroidism cannot be distinguished from certain other giant cell lesions of the jaws, a biochemical study of the patient's serum to measure calcium, phosphorus and alkaline phosphatase levels is required [3]. Renal osteodystrophy must be considered in the diagnosis of any diffuse fibro-osseous enlargements seen in patients undergoing chronic renal dialyses [2]. Jaw enlargement tends to cease after the correction of the secondary hyperparathyroidism, however, sometimes surgery is necessary [2]. Our patient has undergone cosmetic surgery and is waiting for a parathyroidectomy.
Conflict of interest statement. None declared.
References
- Hruska K. New concepts in renal osteodystrophy. Nephrol Dial Transplant (1998) 13:2755–2760.
[Free Full Text] - Kalyvas D, Tosios KI, Leventis MD, Tsiklakis K, Angelopoulos AP. Localized jaw enlargement in renal osteodystrophy: report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod (2004) 97:68–74.[Web of Science][Medline]
- Phillips VM, Breytenbach HS, Grotepass F, Nortjé C, van Wyk CM, van Buuren AJ. Secondary hyperparathyroidism manifesting in the mandible. J Dent Assoc S Afr (1982) 37:373–375.[Medline]
Accepted in revised form: 26. 3.07
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