Pediatr Radiol (1992) 22:142-144
Pediatric Radiology 9 Springer-Verlag 1992
"Brown tumor" of the maxilla S. Fasanelli 1, M. Graziani 1, R. Boldrini 2, and C. Bosman 2 1Department of Radiology and 2Department of Pathology, Hospital Bambino Gesfi, Rome, Italy Received: 14 October 1991; accepted: 10 January 1992
Abstract. A case of brown tumor of hyperparathyroidism in a young girl with a long history of chronic renal failure and hemodialysis therapy is reported. The diagnosis can be reached with the aid of the classic skeletal radiological sign which address the evaluation of both CT and M R imaging.
Giant cell tumors in the head and neck are very uncommon, especially in children. Three types of "giant cell lesions" occur in the skeleton. Many bone tumor variants can also demonstrate large numbers of giant cells [1]. Because of their histologic similarities, diagnostic problems may arise among the "reparative giant-cell granuloma", the "true giant-cell tumor", otherwise called osteoclastoma, and the so called "brown tumor of hyperparathyroidism" [2]. The "brown tumors", so called because of the reddish-brown color of their tissue [3], occur both with primary and
Fig.1. a Axial CT after contrast administration revealed an expansile lesion in the ethmoidal region with bone destruction. Interruption of maxillary sinuses and orbital walls. Non-homogeneous appearance of the mass showing central areas of diminished density, b Coronal CT after contrast administration revealed the destruction of the wall of the sphenoid sinus and of the ethmoidal roof. The lateral dislocation of the ocular globes is clearly evident Fig.2. a Axial MR scan proton-density weighted (TR 2500, TE 25), shows an isointense large massinthe ethmoidal sinus, b AxialMRscanT2weighted (TR 2500,TE l l0), shows the mass disomogeneous and slightly hyperintense rounded by hyperintense tissue probably due to inflammation component or mucoid impaction, e Coronal MR scan Tl-weighted (TR 400, TE 25), shows slight hypointensity of the mass. d Coronal MR scan Tl-weighted (TR 400, TE 25) after gadolinium-DTPA shows noticeable enhancement of the signal
secondary hyperparathyroidism in patients undergoing long term dialysis or have endstage renal disease [4, 5]. In the head and neck the lesions develop chiefly in the mandible and maxilla[l]. To plan the initial treatment and longterm management of these kinds of tumors an accurate diagnosis is needed and an appraisal of the clinical course, biochemical laboratory data, radiographic appearance are all necessary [1]. Histologic differentiation between the brown tumor and the giant-cell granuloma may be impossible, therefore the role of the imaging procedures for diagnosis is of main importance [6, 7]. Special attention must be reserved to locate the presence of smaller multiple cystic areas in bones, generalized osteoporosis, subperiosteal bone resorption, especially of distal phalanges and the distal end of the clavicle and disappearance of the lamina dura. All these signs indicate hyperparathyroidism [8] and lead to the correct diagnosis of "brown tumor" [2, 3, 9].
Case report V.A. is a 19-year-old girl with multiple congenital malformations. She began hemodialysis therapy 21/2years ago in chronic renal failure related to left kidney agenesis and right vesicoureteral reflux which was surgically corrected at the age of two. At that time biochemical tests already demonstrated impairment of renal function. She presented with a 1 month history of increasing headaches, epiphora and bilateral exophthalmos. Axial and coronal CT (Fig. 1) (Somatom DRH) showed a central mass with irregular contours causing destruction of the ethmoid and nasal cavity bones, and the adjacent walls of the frontal, sphenoid and maxillary sinuses. The mass had a nonhomogeneous density with central areas of less value, and with enhancement after contrast. Magnetic resonance imaging (Fig.2) (MRI) (Permanent unit PM400 Ansaldo 0.2 T) showed a round mass, 5 cm in diameter, in the ethmoid. Contrast-enhancement took place following gadolinium DTPA. Skeletal X-ray showed signs of secondary hyperparathyroidism consistent with renal osteodystrophy. Subperiostealphalangeal resorption, loss of the outer corticallining in the distal tufts of the hand, subperiosteal and subchondral resorptionin the necks of femur and humerus, lateral end of the clavicle, distal radius and ulna, sacroiliac joints, and pelvic bones, were all present. A provisional diagnosis of "brown tumor" was made on the evidence of the widespread skeletal and biochemical abnormalities.
Fig.3. Photomicrographs showingthe histologic pattern of the "brown tumor". The giant-cells are sparse and unevenly distributed in a stroma composed of spindleshaped cells. ME 25 x
144 and the true giant-cell tumor especially when the pathologist does not possess the necessary clinical and biochemical information [3]. Brown tumors do occur in renal osteodystrophy and will probably be encountered more frequently in renal osteodystrophy, than in primary hyperparathyroidism because of the large numbers of patients with renal failure being kept alive by dialysis and due to early biochemical diagnosis of primary hyperparathyroidism by serum biochemistry [8]. A radiological correct diagnosis can be made only if supported by the presence of the characteristic skeletal findings of hyperparathyroidism [2, 9]. In our case the mass was expansile and bone destroying, which is in contrast with others' findings of localized, ovoidshaped lesion [2]. Although iron deposits around the tumoral tissue have been shown to produce changes on M R I [1, 3] this case did not show the expected hypointense signals in the T1 and T2 weighted images. The cause of this is probably due to either the absence of hemosiderin deposits or the low strength of the magnetic field used.
References
Ng,4. Histologic pattern of parathyroids showing clear cell hyperplasia. HE 40 x
An open biopsy confirmed our hypothesis with the tissue samples showing a reddishbrown color. The histologic pattern of the lesion was a rather distinctive one (Fig. 3). The stroma was composed of spindle-shaped cells with hemorrhagic extravasation. Numerous multinuclear giant-cells were present, sparsely distributed or, more often, clumped especially around areas of hemorrhage. Some osseous trabeculae were formed in the organizing spindle-cell stroma. Because of the secondary hyperparathyroidism, parathyroidectomy
was undertaken. Eight glands were found in the laterocervical and submandibular regions, two ofwhich were particulary enlarged. Histological examination disclosed clear cell hyperplasia (Fig. 4).
Discussion Because of histologic similarities, diagnostic problems may arise between giant-cell reparative granuloma, the "brown tumor of hyperparathyroidism"
1. Quick CA, Anderson R, Stool S (1980) Giant cell tumors of the maxilla in children. Laryngoscope 90:784 2. Som PM, Lawson W, Cohen BA (1983) Giant-cell lesions of the facial bones. Radiology 147:129 3. Schajowicz F (1981) Tumors and tumorlike lesions of bone and joints. Springer, New York, p 500 4. Brown TW, Genant HK, Hattner RS, Orloft S, Potter DE (1977) Multiplebrown tumors in a patient with chronic renal failure and secondary hyperparathyroidism. A JR 128:131 5. Rao R Solomon M, Avramides A et al (1978) Brown tumors associated with secondary hyperparathyroidism of chronic renal failure. J Oral Surg 36:154 6. Batsakis JG (1979) Tumors of the head and neck. Clinical and pathological considerations, 2rid edn. Williams and Wilkins, Baltimore 7. Newton TH, Hasso AN, Dillon WP (1988) Computed tomography of the head and neck,vol 3. ClavadelPress,NewYork,p 11 8. Sundaram M (1989) Renal osteodystrophy. Skeletal Radio118: 415 9. Rhea JT, Weber AL (1983) Giant-cell granuloma of the sinuses. Radiology 147:135 S. Fasanelli Department of Radiology Hospital Bambino Gesfi Piazza S. Onofrio, 4 1-00165 Rome Italy
Pediatric Radiology 9 Springer-Verlag 1992
"Brown tumor" of the maxilla S. Fasanelli 1, M. Graziani 1, R. Boldrini 2, and C. Bosman 2 1Department of Radiology and 2Department of Pathology, Hospital Bambino Gesfi, Rome, Italy Received: 14 October 1991; accepted: 10 January 1992
Abstract. A case of brown tumor of hyperparathyroidism in a young girl with a long history of chronic renal failure and hemodialysis therapy is reported. The diagnosis can be reached with the aid of the classic skeletal radiological sign which address the evaluation of both CT and M R imaging.
Giant cell tumors in the head and neck are very uncommon, especially in children. Three types of "giant cell lesions" occur in the skeleton. Many bone tumor variants can also demonstrate large numbers of giant cells [1]. Because of their histologic similarities, diagnostic problems may arise among the "reparative giant-cell granuloma", the "true giant-cell tumor", otherwise called osteoclastoma, and the so called "brown tumor of hyperparathyroidism" [2]. The "brown tumors", so called because of the reddish-brown color of their tissue [3], occur both with primary and
Fig.1. a Axial CT after contrast administration revealed an expansile lesion in the ethmoidal region with bone destruction. Interruption of maxillary sinuses and orbital walls. Non-homogeneous appearance of the mass showing central areas of diminished density, b Coronal CT after contrast administration revealed the destruction of the wall of the sphenoid sinus and of the ethmoidal roof. The lateral dislocation of the ocular globes is clearly evident Fig.2. a Axial MR scan proton-density weighted (TR 2500, TE 25), shows an isointense large massinthe ethmoidal sinus, b AxialMRscanT2weighted (TR 2500,TE l l0), shows the mass disomogeneous and slightly hyperintense rounded by hyperintense tissue probably due to inflammation component or mucoid impaction, e Coronal MR scan Tl-weighted (TR 400, TE 25), shows slight hypointensity of the mass. d Coronal MR scan Tl-weighted (TR 400, TE 25) after gadolinium-DTPA shows noticeable enhancement of the signal
secondary hyperparathyroidism in patients undergoing long term dialysis or have endstage renal disease [4, 5]. In the head and neck the lesions develop chiefly in the mandible and maxilla[l]. To plan the initial treatment and longterm management of these kinds of tumors an accurate diagnosis is needed and an appraisal of the clinical course, biochemical laboratory data, radiographic appearance are all necessary [1]. Histologic differentiation between the brown tumor and the giant-cell granuloma may be impossible, therefore the role of the imaging procedures for diagnosis is of main importance [6, 7]. Special attention must be reserved to locate the presence of smaller multiple cystic areas in bones, generalized osteoporosis, subperiosteal bone resorption, especially of distal phalanges and the distal end of the clavicle and disappearance of the lamina dura. All these signs indicate hyperparathyroidism [8] and lead to the correct diagnosis of "brown tumor" [2, 3, 9].
Case report V.A. is a 19-year-old girl with multiple congenital malformations. She began hemodialysis therapy 21/2years ago in chronic renal failure related to left kidney agenesis and right vesicoureteral reflux which was surgically corrected at the age of two. At that time biochemical tests already demonstrated impairment of renal function. She presented with a 1 month history of increasing headaches, epiphora and bilateral exophthalmos. Axial and coronal CT (Fig. 1) (Somatom DRH) showed a central mass with irregular contours causing destruction of the ethmoid and nasal cavity bones, and the adjacent walls of the frontal, sphenoid and maxillary sinuses. The mass had a nonhomogeneous density with central areas of less value, and with enhancement after contrast. Magnetic resonance imaging (Fig.2) (MRI) (Permanent unit PM400 Ansaldo 0.2 T) showed a round mass, 5 cm in diameter, in the ethmoid. Contrast-enhancement took place following gadolinium DTPA. Skeletal X-ray showed signs of secondary hyperparathyroidism consistent with renal osteodystrophy. Subperiostealphalangeal resorption, loss of the outer corticallining in the distal tufts of the hand, subperiosteal and subchondral resorptionin the necks of femur and humerus, lateral end of the clavicle, distal radius and ulna, sacroiliac joints, and pelvic bones, were all present. A provisional diagnosis of "brown tumor" was made on the evidence of the widespread skeletal and biochemical abnormalities.
Fig.3. Photomicrographs showingthe histologic pattern of the "brown tumor". The giant-cells are sparse and unevenly distributed in a stroma composed of spindleshaped cells. ME 25 x
144 and the true giant-cell tumor especially when the pathologist does not possess the necessary clinical and biochemical information [3]. Brown tumors do occur in renal osteodystrophy and will probably be encountered more frequently in renal osteodystrophy, than in primary hyperparathyroidism because of the large numbers of patients with renal failure being kept alive by dialysis and due to early biochemical diagnosis of primary hyperparathyroidism by serum biochemistry [8]. A radiological correct diagnosis can be made only if supported by the presence of the characteristic skeletal findings of hyperparathyroidism [2, 9]. In our case the mass was expansile and bone destroying, which is in contrast with others' findings of localized, ovoidshaped lesion [2]. Although iron deposits around the tumoral tissue have been shown to produce changes on M R I [1, 3] this case did not show the expected hypointense signals in the T1 and T2 weighted images. The cause of this is probably due to either the absence of hemosiderin deposits or the low strength of the magnetic field used.
References
Ng,4. Histologic pattern of parathyroids showing clear cell hyperplasia. HE 40 x
An open biopsy confirmed our hypothesis with the tissue samples showing a reddishbrown color. The histologic pattern of the lesion was a rather distinctive one (Fig. 3). The stroma was composed of spindle-shaped cells with hemorrhagic extravasation. Numerous multinuclear giant-cells were present, sparsely distributed or, more often, clumped especially around areas of hemorrhage. Some osseous trabeculae were formed in the organizing spindle-cell stroma. Because of the secondary hyperparathyroidism, parathyroidectomy
was undertaken. Eight glands were found in the laterocervical and submandibular regions, two ofwhich were particulary enlarged. Histological examination disclosed clear cell hyperplasia (Fig. 4).
Discussion Because of histologic similarities, diagnostic problems may arise between giant-cell reparative granuloma, the "brown tumor of hyperparathyroidism"
1. Quick CA, Anderson R, Stool S (1980) Giant cell tumors of the maxilla in children. Laryngoscope 90:784 2. Som PM, Lawson W, Cohen BA (1983) Giant-cell lesions of the facial bones. Radiology 147:129 3. Schajowicz F (1981) Tumors and tumorlike lesions of bone and joints. Springer, New York, p 500 4. Brown TW, Genant HK, Hattner RS, Orloft S, Potter DE (1977) Multiplebrown tumors in a patient with chronic renal failure and secondary hyperparathyroidism. A JR 128:131 5. Rao R Solomon M, Avramides A et al (1978) Brown tumors associated with secondary hyperparathyroidism of chronic renal failure. J Oral Surg 36:154 6. Batsakis JG (1979) Tumors of the head and neck. Clinical and pathological considerations, 2rid edn. Williams and Wilkins, Baltimore 7. Newton TH, Hasso AN, Dillon WP (1988) Computed tomography of the head and neck,vol 3. ClavadelPress,NewYork,p 11 8. Sundaram M (1989) Renal osteodystrophy. Skeletal Radio118: 415 9. Rhea JT, Weber AL (1983) Giant-cell granuloma of the sinuses. Radiology 147:135 S. Fasanelli Department of Radiology Hospital Bambino Gesfi Piazza S. Onofrio, 4 1-00165 Rome Italy
