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Saudi Journal of Kidney Diseases and Transplantation
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RENAL DATA FROM THE ARAB WORLD Table of Contents   
Year : 2010  |  Volume : 21  |  Issue : 4  |  Page : 772-777
Brown tumors in patients with chronic renal failure and secondary hyperparathyroidism: Report of 12 cases


1 Department of Nephrology, La Rabta Hospital, and Laboratory of Kidney Pathology, Tunis, Tunisia
2 Department of Nephrology and Internal Medicine, Charles Nicolle Hospital, and Laboratory of Kidney Pathology, Tunis, Tunisia

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Date of Web Publication26-Jun-2010
 

   Abstract 

Brown tumors are unusual but serious complications of renal osteodystrophy. We retrospectively studied 12 patients presenting with chronic renal failure and brown tumor related to secondary hyperparathyroidism. Eleven patients were on chronic hemodialysis. The median duration between renal failure and end stage renal failure was 36 months (range: 12-190 months) and the median duration in dialysis for 11 cases: 92 months (range: 72-252 months). The bone pain was noted in all cases (100%), pathological fracture in one case (8%) and a palpable bone tumor in 10 cases (83%). Elevated serum Calcium (> 2.35 mmol/L) was noted in four cases (33%), elevated serum Phosphate (> 1.78 mmol/L) in ten cases (80%), elevated serum Alkaline Phosphate (> 290 UI/L) in all cases and intact PTH was > 300 pg/mL in all cases with a serum median rate at 1475 pg/mL (range: 682-3687 pg/L). Subtotal parathyroidectomy was performed in all cases with a resultant decrease in size of brown tumors. We report here patient with CKD with unusual frequency and variable locations. This may be attributed tothe lack of the new calcium free phosphate binders and calcimimetics.

How to cite this article:
Fatma LB, Barbouch S, Fethi BH, Imen BA, Karima K, Imed H, Fethi E, Fatma BM, Rim G, Taieb BA, Maiz HB, Adel K. Brown tumors in patients with chronic renal failure and secondary hyperparathyroidism: Report of 12 cases. Saudi J Kidney Dis Transpl 2010;21:772-7

How to cite this URL:
Fatma LB, Barbouch S, Fethi BH, Imen BA, Karima K, Imed H, Fethi E, Fatma BM, Rim G, Taieb BA, Maiz HB, Adel K. Brown tumors in patients with chronic renal failure and secondary hyperparathyroidism: Report of 12 cases. Saudi J Kidney Dis Transpl [serial online] 2010 [cited 2018 Sep 23];21:772-7. Available from: http://www.sjkdt.org/text.asp?2010/21/4/772/64655

   Introduction Top


Brown tumors are unusual but serious com­plications of renal osteodystrophy. Brown tu­mors associated with secondary hyperparathy­ roidism (HPT) in patients with chronic renal failure has been increasingly documented of late and are more commonly observed with increased longevity of hemodialysis patients and can be found in any bone. The rate of brown tumor in secondary HPT due to chronic kidney disease (CKD) ranges from 1.5% to more than 13%. [1],[2],[3] The brown tumors result from the destruction of local bone occurring especially in regions where bone resorption is particularly rapid with hemorrhage and repa­rative granulation tissue containing giant multi­nucleated cells replacing the normal marrow content, resulting in a brown tumor. [4],[5],[6]

The mandible is commonly involved compared to the maxillary bone. [5],[7],[8],[9],[10],[11]

Radiological studies report different bone lo­cation of theses lesions and suggest that it may be an incidental finding in an asymptomatic pa­tient although bone pain has been described. [6],[9],[12] Phosphate binders and vitamin D are useful preventive measures of secondary HPT and brown tumors. [13],[14] Although parathyroidectomy is curative, surgical removal of the brown tu­mor mass may sometimes be required.

The purpose of this study is to analyze cli­nical, biologic and radiologic characteristics of brown tumors in our patients.


   Patients and Methods Top


Patients

This is a retrospective study including 12 pa­tients hospitalized in our department between 1990 and 2006 because of brown tumor.

There were four males and eight females with a mean age of 39.9 ΁ 14.3 years (range: 19-61years). All patients had chronic renal failure and 11 of them were on regular maintenance hemodialysis (HD). Their median duration bet­ween renal failure and end stage renal failure was 36 months (range: 12-190 months) and the median duration on dialysis was 92 months (72-252 months).

The underlying renal disease was nephroangio­sclerosis in four cases, interstitial involvement in five cases, glomerular involvement in one case and unknown in two cases.

Methods

Blood samples collected at the same time of the diagnosis of brown tumor, were analyzed at Charles Nicolle Hospital laboratory using standard clinical laboratory methods. Intact PTH was measured by immunologic assay (upper limit of normal is 65 pg/mL). Hypercalcemia is defined by a serum calcium level of more than 2.35 mmol/L, hyperphosphatemia by a se­rum phosphorus level of more than 1.78 mmol/ L, elevated serum Alkaline Phosphate by a serum level of more than 290 UI/L and an elevated serum calcium phosphorus product by a serum level of more than 4.5 mmol 2 /L 2 .


   Results Top


In this report, all patients were symptomatic: the bone pain was noted in all cases, bone deformation in ten cases (83%), pathological fracture in one case (8%) and a palpable bone tumor in ten cases (83%). Biologic serum mean data are summarized in [Table 1]. Hyper­calcemia was noted in four cases (33%), hyper­phosphatemia in ten cases (80%), elevated se­rum Bone Alkaline Phosphate and Intact PTH was noted in all cases.

The radiologic features were mainly the de­mineralization noted in all cases, the subpe­riosteal bone erosions in the distal phalanx in 11 cases (91%), the "Pepper pot" appearance and resorptive defects of the diploes in 11 cases (91%), pathologic fracture in one case and metastatic calcification in five cases (41%).

The brown tumors were localized in man­dible in three cases [Figure 1], in the maxillary bone in four cases, in femur in two cases and it was multiple in three cases with shoulder (hu­merus and omoplatus) and phalangeal involve­ment in two cases [Figure 2], [Figure 3], [Figure 4] and [Figure 5] and femur and clavicle involvement in one case.

Treatment performed was surgical in all cases with a subtotal parathyroidectomy. Recovery was observed in 11 cases with a decrease of mean intact PTH serum level from 1771 to 234 pg/mL. In one case, the results were insufficient and required a total parathyroidectomy with heterotopic autotransplantation (resection of the remaining parathyroid and implantation of its minifragments in the right forearm muscle (LEAL). The histological examination showed a single adenoma in 12% of cases and multiple gland hyperplasia in 88% of cases.

A decrease in size of brown tumors was ob­served in all cases. However, it was insuffi­cient in two cases requiring a surgical resec­tion of brown tumor with mandible involve­ment in one case and maxillary involvement in one case. The histological examination per­formed in the two cases, with light microscopy showed many osteoclastic giant cells arranged in groups adjacent to hemosiderin granules, within a fibrovascular hemorrhagic stroma which conclude on a "giant cell tumor".

Clinical, biologic characteristics of the 12 pa­tients and their evolution after treatment were summarized in [Table 1],[Table 2] and [Table 3].


   Discussion Top


To our knowledge, our data is one of the lar­gest reported in literature. The particularities of our study are the presence of symptoms such as the bone pain in all cases, the fre­quency of the maxillary and the multiple in­volvements which are reported to be rare.

The effective control of HPT is crucial to prevent and sometimes treat brown tumors. Vitamin D improves serum PTH levels. In fact, intravenous calcitriol is often sufficient in high serum PTH level if hyperphosphatemia is kept under control. [13],[14],[15],[16],[17] In our country, calcium free phosphate binders are not available resul­ting in frequent episodes of hyperphosphaemia and hypercalcemia precluding the use of vita­min D analogues This could probably explain the unusual frequency and the highly expanded lesions of brown tumors in our data.

Medical treatment of brown tumor based on high doses of vitamin D is effective in many cases [18],[19],[20] nevertheless, most of these lesions require total or subtotal parathyroidectomy. [1] The recovery is fast [6],[7] and the lesions are later seen as sclerotic on radiographs. [21]

Surgical resection of brown tumor should be considered if the mass does not regress after correction of the inciting hyperparathyroidism after a mean follow up period of one to two years or if the patient is symptomatic. [22],[23],[24]

Surgical resection and decompression of the brown tumors is urgently needed in anatomical sites that can compromise the local area by continuous expansion, such as maxillary brown tumor in our case causing serious deformity and breathing difficulty. [1],[24],[25] In these cases, usually parathyroidectomy and aesthetic osteo­plasty. [24],[26],[27] or decompressive surgery in pa­tients with vertebral involvement and spinal cord compression. [22],[28]


   Conclusion Top


In conclusion due to the lack of claicum free binders preventive measures of secondary HPT are insufficient and result in the unusual high rate of brown tumor.

Parathyroidectomy remains the first choice treatment and leads often in a decrease in size of brown tumors even when it is multiple. Remaining tumor mass may sometimes need local surgical removal after parathyroidectomy.

 
   References Top

1.Marrone LF, Ettore GC, Passavati G, et al. Maxillary brown tumor in secondary hyper­parathyroidism requiring urgent parathyroid­dectomy. J Nephrol 2001;14:415-9.  Back to cited text no. 1      
2.Sargent MA, Fleming SJ, Chattopadhyay C, Ackrill P, Sambrook P. Bone cysts and haemo­dialysis-related amyloidosis. Clin Radiol 1989; 40:277-81.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]  
3.Katz AL, Hampers CL, Merril JP. Secondary hyperparathyroidism and renal osteodystrophy. Medicine 1969;48:333-74.  Back to cited text no. 3      
4.Catizone L, Casolino D, Santoro A, Casanova S, Cerasoli PT, Zucchelli P. An unusual mani­festation of renal osteodystrophy. Nephron 1984;37:133-6.  Back to cited text no. 4  [PUBMED]    
5.Brown TW, Genant HK, Hattner RS, Orloff S, Potter DE. Multiple brown tumors in patient with chronic renal failure and secondary hyper­parathyroidism. AJR Am J Roentgenol 1977; 128:131-4.  Back to cited text no. 5  [PUBMED]  [FULLTEXT]  
6.Marini M, Vidiri A, Guerrisi R, Campodonico F, Ponzio R. Progress of brown tumors in patients with chronic renal insufficiency under­going dialysis. Eur J Radiol 1992;14:67-71.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]  
7.Korzets A, Ori Y, Chagnac A. Brown tumor and secondary hyperparathyroidism: A conti­nuing saga. Nephrol Dial Transplant 1996;11: 1669.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]  
8.Prado FO, Rosales AC, Rodriguez CI, Colette RD, Lopez MA. Brown tumor of the mandible associated with secondary hyperparathyroidism: a case report and review of the literature. Gen Dent 2006;54:341-3.  Back to cited text no. 8      
9.Margini F, Lusenti T, Ambrosetti F, Nicoli F, Borgetti PP, Rustichelli R. Effects of para­thyroidectomy in a case of hyperparathyroi­dism secondary to chronic renal failure insu­fficiency with a multiple brown fat tumor. Radiol Med 1988;75:247-50.  Back to cited text no. 9      
10.Orejas G, Rey C, Vicente SG, Fernandez L, Santos F, Malaga S. Maxillary brown tumor as manifestation of renal osteodystrophy. Nephron 1993;64:483-4.  Back to cited text no. 10      
11.Haitami S, Khazana M, Rifki C, Benyahya I. Brown tumor of the jaws. Rev Stomatol Chir Maxillofac 2006;107:481-2.  Back to cited text no. 11      
12.Bandeira F. From mild primary hyperparathy­roidism: the Brazilian experience. Arq Bras Endocrinol Metab 2006;50:657-63.  Back to cited text no. 12      
13.Liach F, Hervas J, Cerezo S. The importance of dosing intravenous calcitriol in dialysis patients with severe hyperparathyroidism. Am J Kidney Dis 1995;26:845-51.  Back to cited text no. 13      
14.Potter DE, Wilson CJ, Ozonoff MB. Hyper­parathyroid bone disease in children under­going long term hemodialysis: treatment with vitamin D. J Pediatr 1974;85:60-6.  Back to cited text no. 14  [PUBMED]    
15.Salusky IB, Goodman WG, Sahney S, et al. Sevelamer controls parathyroid hormone-in­duced bone disease as efficiently as calcium carbonate without increasing serum calcium levels during therapy with active vitamin D serols. J Am Soc Nephrol 2005;16:2501-8.  Back to cited text no. 15  [PUBMED]  [FULLTEXT]  
16.Moe SM, Chertow GM, Coburn JW, et al. Achieving NKF-K/DOQI bone metabolism and disease treatment goals with cinacalcet HCI. Kidney Int 2005;67:760-1.  Back to cited text no. 16  [PUBMED]  [FULLTEXT]  
17.Moe SM, Cunningham J, Bommer J, et al. Long­term treatment of secondary hyperparathyroi­dism with the calcimimetic cinacalcet HCI. Nephrol Dial Transplant 2005;20:2186-93.  Back to cited text no. 17  [PUBMED]  [FULLTEXT]  
18.Ben Hamida F, Ben Abdallah T, Ben Moussa F, Abderrahi E, El Matri A, Ben Maiz H. Treatment of severe secondary hyperpara­thyroidism by oral pulse alfacalcitriol (one alpha) in a patient on chronic hemodialysis. Saudi J Kidney Transplant 1996;7:S82-4.  Back to cited text no. 18      
19.Garcia GM, Leanza H, Najun ZC, Barreneche M. Medical parathyroidectomy. Its efficacy in treatment of lower maxillary brown tumor. Medicina B Aires 1996;56:59.  Back to cited text no. 19      
20.Francisco AL. Secondary hyperparathyroidism: review of the disease and its treatment. Clin Ther 2004;26:1976-93.  Back to cited text no. 20  [PUBMED]  [FULLTEXT]  
21.Takeshita T, Takeshita K, Abe S, Takami H, Imamura T, Furui S. Brown tumors with fluid­fluid levels in a patient with primary hyper­parathyroidism: radiologic findings. Radiat Med 2006;24:631-4.  Back to cited text no. 21  [PUBMED]  [FULLTEXT]  
22.Terrass F, Ayad A, Benjalloun M, et al. Cauda equine compression revealing brown tumor of the spine in a long-term hemodialysis patient. Joint Bone Spine 2006;73:748-50.  Back to cited text no. 22      
23.Lessa MM. Brown tumor of the facial bones: case report and literature review. Ear Nose Throat J 2005;84:42-4.  Back to cited text no. 23      
24.Leal CT, Lacativa PG, Gomes EM, et al. Sur­gical approach and clinical outcome of a defor­ming brown tumor at the maxilla in a patient with secondary hyperparathyroidism due to chronic renal failure. Arq Bras Endocrinol Metabol 2006;50:963-7.  Back to cited text no. 24  [PUBMED]  [FULLTEXT]  
25.Dantas M, Costa RS, Jorgetti V, et al. Facial leontiasis ossea: a rare presentation of hyper­parathyroidism secondary to chronic renal in­sufficiency. Nephron 1991;58:475-8.  Back to cited text no. 25  [PUBMED]    
26.Desigan S, Syed R, Conway GS, Kurzawinski TR, Bomanji JB. Giant cervical parathyroid adenoma mimicking a sternocleidomastoid mass and presenting as a brown tumor of the man­dible. Clin Nucl Med 2007;32:306-8.  Back to cited text no. 26  [PUBMED]  [FULLTEXT]  
27.Pinto LP, Cherubinim K, Salum FG, Yurgel LS, de Figueiredo MA. Highly aggressive brown tumor in the jaw associated with tertiary hyper­parathyroidism. Pediatr Dent 2006;28:543-6.  Back to cited text no. 27  [PUBMED]  [FULLTEXT]  
28.Haddad FH, Malkawi OM, Sharbaji AA, Jbara IF, Rihani HR. Primary hyperparathyroidism. A rare cause of spinal cord compression. Saudi Med J 2007;28:783-6.  Back to cited text no. 28  [PUBMED]    

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Correspondence Address:
Samia Barbouch
Department of Nephrology and Internal Medicine, Charles Nicolle Hospital, 09 Avril Avenue, Tunis
Tunisia
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PMID: 20587894

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]

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