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Saudi Journal of Kidney Diseases and Transplantation
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Table of Contents   
CASE REPORT  
Year : 2016  |  Volume : 27  |  Issue : 1  |  Page : 144-146
Mandibular mass in a child on hemodialysis


1 Department of Pediatrics, Faculty of Medicine, Ain Shams University, Zagazig, Egypt
2 Department of Radio Diagnosis, Faculty of Medicine, Ain Shams University, Zagazig, Egypt
3 Ain Shams University, Zagazig, Egypt

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Date of Web Publication15-Jan-2016
 

   Abstract 

We here with report a 13-year-old female patient on regular hemodialysis for the past five years who presented with a large mandibular mass. This was detected to be a brown tumor due to severe renal osteodystrophy as a complication of secondary hyperparathyroidism. The tumor did not regress even with intensive treatment with intravenous active vitamin D and needed surgical removal.

How to cite this article:
Youssef DM, Mohammed FF, Adham T. Mandibular mass in a child on hemodialysis. Saudi J Kidney Dis Transpl 2016;27:144-6

How to cite this URL:
Youssef DM, Mohammed FF, Adham T. Mandibular mass in a child on hemodialysis. Saudi J Kidney Dis Transpl [serial online] 2016 [cited 2019 Nov 19];27:144-6. Available from: http://www.sjkdt.org/text.asp?2016/27/1/144/174169

   Introduction Top


The term "renal osteodystrophy (ROD)" is often used in a generic sense to include skeletal disorders of patients with chronic renal failure, including ostitis fibrosa, osteomalacia, osteosclerosis and osteoporosis. [1] Brown tumors or osteoclastomas are erosive bony lesions that arise as a complication of hyperparathyroidism (HPT). The initial treatment involves the correction of HPT, which usually results in regression of the tumors. [2] Secondary HPT is described to occur in 92% of the patients undergoing dialysis. Of them, approximately 1.5% develop brown tumors, which may appear in any bone, but are frequently found in the facial bones and jaws, particularly in long-standing cases and with multiple skeletal lesions; these lesions are uncommonly seen these days. [3]

As long as prolonged hemodialysis (HD) is not common in the pediatric population, the occurrence of brown tumor in children is quite rare. [4] We report here a case of brown tumor in a 13-year-old child that was not controlled by medications and needed surgical removal.


   Case Report Top


A 13-year-old child with end-stage renal disease (ESRD) due to chronic interstitial nephritis was on regular HD for the last five years. Dialysis was performed with a Fresenius 2008K machine and hollow fiber polysulfone dialyzers (Fresenius, Bad Homburg, Germany). The dialysis prescription was as follows: three times a week, 3 h/session, blood flow 300 mL/min, with mean urea reduction ration (URR) of 67%. The URR was calculated as follows: [URR = (pre-dialysis urea - post-dialysis urea) χ pre-dialysis urea].

The patient developed a swelling of the jaw on the left side that increased in size gradually. The swelling was soft, painless and minimally tender, and was elastic in consistency. The patient also complained of difficulty in walking due to pain in the left knee joint following a history of fracture of the left femur three years after the onset of dialysis.

The mean laboratory values of the child were as follows: high intact parathormone (iPTH) of 602 pg/mL, serum calcium of 9.7 mg/dL and phosphorus of 5 mg/dL. The vitamin D level was low at 13 ng/mL.

A multi-slice computed tomography (CT) scan showed a uni-locular, expansile osteolytic lesion in the mandibular body at its left paramedian aspect, showing homogeneous attenuation and loss of the bony trabeculae with rarified related alveolar margin, resorption of the outer cortical margin and associated soft tissue component [Figure 1]. The lesion resulted in mal-alignment of the related incisor and canine teeth as seen in the three-dimensional (3D) image [Figure 2].
Figure 1: Coronal computerized tomography scan images displaying unilocular expansile osteolytic lesion in the left paramedian aspect of the mandibular body causing destruction of the outer cortical margin with soft tissue component.

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Figure 2: Three-dimensional volume-rendering image showing manibular osteolyic lesion with mal-alignment of the related teeth.

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A diagnosis of brown tumor of the anterior mandible was made and a trial of intensive course of intravenous active vitamin D was given for three months until the iPTH was reduced to 52 pg/mL. However, the mass did not show any signs of regression. In view of the disfiguring picture and possibility of a behavior mimicking a malignant lesion, [5] surgical treatment was performed with curettage of the affected bone lesion and the mass was excised. Biopsy of the lesion demonstrated a central giant cell lesion between other giant cells that were sheets of plump fibroblasts with a rich vascular network, highly suggestive of a brown tumor.


   Discussion Top


Brown tumors that occur in patients with ESRD represent an extreme form of osteodystrophy. They occur most often in the long bones, ribs and pelvis, but can be found in any bone. Clinically significant lesions in the jaws are rare, especially in the pediatric population. [6]

Radiographic findings of renal osteodystrophy in the jaw include bone resorption with loss of cortical bone, lamina dura and other anatomical landmarks and condensation of trabecular architecture producing a groundglass appearance closely resembling fibrous dysplasia. [7]

Primary or secondary HPT is recognized by the presence of an osteolytic lesion with giant cells. The most useful therapy for patients with brown tumors is surgical excision of the bone lesions along with treatment of the HPT. [8]

In the present study, we found that a CT scan was superior in characterization of the lesion as it revealed unilocular expansile osteolytic lesion with other changes described earlier. The brown tumor can result in mal-alignment of the related lower incisors and canine teeth as seen in the 3D image in our patient, which was responsible for the facial swelling and deformity.


   Conclusion Top


We conclude that brown tumor may be found in children with ESRD on long-term HD with secondary HPT. This tumor may be resistant to treatment with active vitamin D and will need surgical removal.

Conflict of Interest: None declared.

 
   References Top

1.
Habener J, Arnold A, Potts JT. Hyperparathyrodism. In: DeGroot LJ, ed. Endocrinology. 3rd ed. Philadelphia: WB Saunders; 1996. p. 1044-60.  Back to cited text no. 1
    
2.
Nabi Z, Algailani M, Abdelsalam M, Asaad L, Albaqumi M. Regression of brown tumor of the maxilla in a patient with secondary hyperparathyroidism after a parathyroidectomy. Hemodial Int 2010;14:247-9.  Back to cited text no. 2
    
3.
Pinar Sumer A, Arik N, Sumer M, Karagoz F. A rare complication of secondary hyperparathyroidism. Brown tumor of the maxilla and mandible. Saudi Med J 2004;25:2010-2.  Back to cited text no. 3
    
4.
Unlü RE, Abaci E, Kerem M, Aksoy E, Sensöz O. Brown tumor in children with normocalcemic hyperparathyroidism: A report of two cases. J Craniofac Surg 2003;14:69-73.  Back to cited text no. 4
    
5.
Kumar KA, Humayun S, Kumar BP, Rao JB. Reparative giant cell granuloma of the maxilla. Ann Maxillofac Surg 2011;1:181-6.  Back to cited text no. 5
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6.
Krause I, Eisenstein B, Davidovits M, Cleper R, Tobar A, Calderon S. Maxillomandibular brown tumor - A rare complication of chronic renal failure. Pediatr Nephrol 2000;14:499-501.  Back to cited text no. 6
    
7.
Park JW, Choi BR, Gang TI, Huh KH, Yi WJ, Choi SC. Mandibular brown tumor in renal osteodystrophy. Korean J Oral Maxillofac Radiol 2008;38:229-31.  Back to cited text no. 7
    
8.
Triantafillidou K, Zouloumis L, Karakinaris G, Kalimeras E, Iordanidis F. Brown tumors of the jaws associated with primary or secondary hyperparathyroidism. A clinical study and review of the literature. Am J Otolaryngol 2006; 27:281-6.  Back to cited text no. 8
    

Top
Correspondence Address:
Doaa M Youssef
Department of Pediatrics, Faculty of Medicine, Zagazig University, Zagazig
Egypt
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DOI: 10.4103/1319-2442.174169

PMID: 26787582

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    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures
 

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