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| RENAL DATA FROM THE ARAB WORLD |    | | | | Year : 2010 | Volume : 21 | Issue : 4 | Page : 772-777 | | | Brown tumors in patients with chronic renal failure and secondary hyperparathyroidism: Report of 12 cases | | | Lilia Ben Fatma1, Samia Barbouch2, Ben Hamida Fethi2, Ben Abdallah Imen2, Khiari Karima2, Helal Imed2, El Younsi Fethi2, Ben Moussa Fatma1, Goucha Rim2, Ben Abdallah Taieb2, Hedi Ben Maiz1, Khedher Adel2
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
Click here for correspondence address and email | Date of Web Publication | 26-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 2014 Mar 4];21:772-7. Available from: http://www.sjkdt.org/text.asp?2010/21/4/772/64655 |
| Introduction | |  |
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 | | |
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 | | |
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 | | |
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 | | |
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 | | |
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Correspondence Address:
Samia Barbouch
Department of Nephrology and Internal Medicine, Charles Nicolle Hospital, 09 Avril Avenue, Tunis
Tunisia
PMID: 20587894
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1], [Table 2], [Table 3] | |
| This article has been cited by | | 1 | Pathological fracture of the thigh-bone due to brown tumor | | | Yankova, M. and Todorov, V. and Todorov, S. | | Nephrology, Dialysis and Transplantation. 2011; 17(3-4): 56-62 | | [Pubmed] | |
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