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An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma
G Model REMNIM-788; No. of Pages 4
ARTICLE IN PRESS Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
Clinical note
An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma Y. Basaran b , S. Ince a , E. Alagoz a,∗ , C. Meric b , A. Taslipinar b a b
Gulhane Military Medical Academy and School of Medicine, Department of Nuclear Medicine, Etlik, Ankara, Turkey Gulhane Military Medical Academy and School of Medicine, Department of Endocrinology and Metabolism, Etlik, Ankara, Turkey
a r t i c l e
i n f o
Article history: Received 11 January 2016 Accepted 22 February 2016 Available online xxx Keywords: Parathyroid adenoma Brown tumor Bone scintigraphy FDG PET/CT
a b s t r a c t We present a patient with a complex clinical picture of primary hyperparathyroidism with multiple destructive skeletal lesions suspicious of bone metastases and concomitant multifocal papillary thyroid carcinoma with a metastatic central lymph node. He presented with progressively worsening right hip pain and restricted motion. Magnetic resonance imaging revealed multiple lytic lesions involving predominantly the right trochanter minor and the left inferior and posterior pubic rami. Biochemical tests were consistent with primary hyperparathyroidism. Neck ultrasound and parathyroid scintigraphy revealed a single parathyroid adenoma and a thyroid nodule, preoperative cytology of which confirmed papillary thyroid carcinoma, as did the final surgical specimen. Biochemical results, regarding hyperparathyroidism, declined to normal levels and his complaints gradually decreased after surgery. Postoperative whole body bone scintigraphy showed increased tracer uptakes at multiple sites, but they were proved to be metabolically inactive by fluorodeoxyglucose positron emission tomography/computed tomography. © 2016 Elsevier España, S.L.U. and SEMNIM. All rights reserved.
Presentación inusual de hiperparatiroidismo primario: múltiples tumores pardos y coexistencia de carcinoma tiroideo r e s u m e n Palabras clave: Adenoma paratiroideo Tumor pardo Gammagrafía ósea FDG PET/TC
Presentamos el caso de un paciente con un cuadro clínico de hiperparatiroidismo primario, con muchas lesiones óseas destructivas sospechosas de metástasis óseas y carcinoma tiroideo multifocal concomitante con un ganglio linfático metastásico central. Se presentó con agravamiento progresivo de dolor y restricción de movimiento en la cadera derecha. La resonancia magnética reveló múltiples lesiones líticas que implicaban principalmente al trocánter menor derecho y a las ramas púbicas izquierdas inferior y posterior. Las pruebas bioquímicas fueron consistentes con un hiperparatiroidismo primario. La ecografía cervical y la gammagrafía paratiroidea revelaron un único adenoma paratiroideo y un nódulo tiroideo, cuya citología preoperatoria confirmó un carcinoma papilar de tiroides, que fue confirmado también por la muestra final obtenida quirúrgicamente. Los resultados bioquímicos, en relación con el hiperparatiroidismo, descendieron a niveles normales tras la cirugía, y los dolores fueron remitiendo gradualmente. La gammagrafía ósea postoperatoria de cuerpo entero reflejó un incremento de captación del radiotrazador en múltiples localizaciones, que resultaron ser metabólicamente inactivas en la tomografía por emisión de positrones con fluorodesoxiglucosa/tomografía computarizada. © 2016 Elsevier España, S.L.U. y SEMNIM. Todos los derechos reservados.
Introduction Brown tumors are rare non-neoplastic skeletal manifestations of primary and secondary hyperparathyroidism1 . Common sites of involvement are pelvis, femur and ribs, but may appear in any bone2 . Since the incidence of thyroid disease is higher among patients with hyperparathyroidism than the general population3 , these
lesions must be distinguished from metastases from thyroid carcinoma. Thus, a comprehensive preoperative evaluation, including fine-needle aspiration of thyroid nodules even not reported to have malignant features on ultrasound (US), should be carried out. We present a case of a 38-year-old male with multiple brown tumors caused by primary hyperparathyroidism and coexisting metastatic multifocal papillary thyroid carcinoma. Clinical case
∗ Corresponding author. E-mail address: [email protected] (E. Alagoz).
The patient was admitted with expansile lytic lesions occupying mostly trochanter minor of the right femoral neck, and the left
http://dx.doi.org/10.1016/j.remn.2016.02.005 2253-654X/© 2016 Elsevier España, S.L.U. and SEMNIM. All rights reserved.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
G Model REMNIM-788; No. of Pages 4 2
ARTICLE IN PRESS Y. Basaran et al. / Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
Fig. 1. Preoperative coronal T1-weighted MRI demonstrated a hypointense lytic expansile mass lesion at the trochanter minor of the right femoral neck (arrow). Significant contrast enhancement in the mass lesion and some degree of soft tissues expansion was noted.
inferior and posterior parts of pubic ramus (30 mm × 16 mm × 5.5 mm, 16 mm × 6.5 mm and 23 mm × 17 mm in diameter, respectively) on magnetic resonance imaging (MRI). They were reported to be hypointense on T1 and hyperintense on T2 weighted images, with various amounts of bone destruction, cortical thinning and some degree of soft tissue expansion around. Brown tumor was the most probable radiologic diagnosis (Fig. 1). At admission, he complained of severe hip pain, which had persisted for one year and progressively worsened over time, limiting his daily activities. On physical examination, tenderness with flexion and rotation of the right hip was detected, resulting in reduced range of motion in the hip joint. He had a 3-year history of kidney stones treatment with ureteral stenting and extracorporeal shock wave lithotripsy. But the rest of his medical history was unremarkable with no history of recent trauma. Biochemical tests of note were PTH of 896.9 pg/mL (normal: 11.1–79.5), calcium of 15.06 mg/dL (normal: 8.5–10.5), phosphate of 1.87 mg/dL (normal: 2.6–4.5) and vitamin D3 of 18.4 mcg/L (normal: 11–70). Thyroid US displayed a 16 mm × 15 mm hypoechoic mass in the lower right pole of the gland, suggested for a parathyroid adenoma, and a thyroid nodule with irregular borders which was suspicious for malignancy (8 mm × 4 mm in diameter) in the left lobe. There were no grossly defined lymph nodes. Technetium-99m methoxyisobutylisonitrile (99m Tc MIBI) scan revealed focal increased radiopharmaceutical uptake just
below the lower pole of right thyroid lobe (Fig. 2). Bone density by DXA revealed a distal radius T-score of −3.7. An ultrasoundguided fine-needle aspiration biopsy (FNAB) of the thyroid nodule was performed, which was suggestive of a malignant cytology. Preoperative exploration for exclusion of multiple endocrine neoplasia did not find evidence of pituitary, pancreatic or adrenal tumors. To promote tumor regression and reduce hungry bone syndrome calcium and vitamin D treatment was initiated. Subsequently, the patient underwent bilateral total thyroidectomy with central compartment neck dissection and right inferior parathyroidectomy. Histological examination confirmed the presence of a parathyroid adenoma and three papillary micro-carcinomas of thyroid (with dimensions of 6 mm and 2 mm in the left lobe and 2 mm in the right lobe) with a central lymph node metastasis. Postoperatively, the patient received an ablative dose of 100 mCi (3700 MBq) iodine131. Post-radioactive iodine whole body scan showed an uptake in the thyroid bed, but no ectopic uptake suggestive of local or distant metastases was visualized. The patient was followed-up on levothyroxine 150 mcg daily and was in a complete remission, with undetectable levels of serum thyroglobulin (Tg) and anti-Tg postoperatively. PTH and calcium levels decreased to 53 pmol/L and 9.21 mg/dL, respectively. Moreover, the biochemistry regarding primary hyperparathyroidism remained normal during 3-monthly follow-ups and his symptoms of hip pain considerably weakened, but did not completely resolve. At the 18-month final follow-up, the patient persisted to experience mild symptoms of hip pain during activity and was subjected to whole-body bone scan, which revealed increased tracer uptakes in left sternoclavicular joint region, left proximal forearm, left pubic ramus and right femoral neck (Fig. 3). Single photon emission computed tomography/CT (SPECT/CT) images of the left ulna demonstrated an expansile lytic lesion with internal septas and SPECT/CT images of the pelvis showed increased tracer uptakes with peripheral sclerosis in the left pubic ramus and right femoral neck, the same osteolytic lesions seen on the preoperative MRI and postoperative whole body bone scans. In order to assess their metabolic activity, 18 F-FDG PET/CT was performed. There was an increased 18 F-FDG uptake only in the left sternoclavicular joint, which was highly suggestive of degeneration. No 18 F-FDG uptake in the aforementioned lesions was detected (Fig. 4). Discussion Hyperparathyroidism is a relatively common disorder. However, brown tumor, also known as osteoclastoma, is a rare benign bone lesion with incidence ranging from 1.5% in primary hyperparathyroidism to 13% in secondary hyperparathyroidism4 . It results from disturbed bone remodeling due to the long-lasting
Fig. 2. 99m Tc MIBI parathyroid scintigraphy planar (A) and SPECT/CT coronal (B) images showed a focus of intense radiopharmaceutical uptake (arrow) just below lower pole of the right thyroid lobe.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
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ARTICLE IN PRESS Y. Basaran et al. / Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
3
Fig. 3. 99m Tc whole-body bone scan after parathyroidectomy (A) displayed increased tracer uptakes in left sternoclavicular joint (thick arrow), left proximal forearm (arrowhead), left pubic ramus (short arrow) and right femoral neck (long arrow). CT image of left ulna (B) revealed expansile lytic lesions with lamellar style septas, a finding suggestive of brown tumor (arrowhead). Axial CT (C) and SPECT/CT (D) images of pelvis demonstrated increased tracer uptakes with peripheral sclerosis at the right femoral neck (long arrow) and left pubic ramus (short arrow), all consistent with brown tumors.
increase in PTH levels.5 Brown tumor can be confused with more serious bone lesions, including aneurysmal bone cyst, giant cell tumor, myeloma, Paget’s disease,6 leukemia, histiocytosis,7 and even metastasis.8 Although the guidelines do not consider primary hyperparathyroidism a risk factor for thyroid carcinoma, several data suggest that this risk may be increased when compared to the general population,3 which was in accordance with the present case. Concomitant non-medullary thyroid carcinoma was found in approximately 3% of patients operated on for primary hyperparathyroidism.9 We believe that a preoperative evaluation of all thyroid nodules should be considered among patients with hyperparathyroidism, regardless of their US features. The present case presented with a complaint of severe pain in the right hip. MRI revealed multiple lytic bone lesions suggestive of brown tumors, but also suspicious of metastases. Laboratory tests
confirmed the presence of the hyperparathyroidism. Thyroid US and parathyroid scintigraphy revealed a suspicious thyroid nodule and a single parathyroid adenoma. Preoperative examination of the thyroid nodule was consistent with a malignant cytology. The patient underwent a complete surgical removal of whole thyroid gland and right inferior parathyroid. Interestingly, histological examination confirmed the presence of a metastatic multifocal papillary thyroid carcinoma. Subsequently, he was subjected to radioactive iodine ablation and followed with TSH suppression therapy. Further exploration depicted multiple sites of increased uptake on whole-body bone scan. However, 18 F-FDG PET/CT did not exhibit an abnormal uptake throughout the skeletal system, which proved these osteolytic lesions to be metabolically inactive. Unlike bone scan, 18 F-FDG PET/CT is expected to revert to normal within 3 months after fracture or trauma.10 Additionally, the right hip pain noticeably attenuated with dramatically increased range of motion
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
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4
Fig. 4. Maximum intensity projection (MIP) image of 18 F-FDG PET/CT after parathyroidectomy (A) showed increased 18 F-FDG uptake in the left sternoclavicular joint region (arrow), but no 18 F-FDG uptake was noted in the other bone lesions. Axial CT (B) and axial fusion (C) images revealed increased 18 F-FDG uptake in the left sternoclavicular joint suggestive of degeneration (arrow). The lytic lesion on the left side of sternum, which was previously thought as a brown tumor had no 18 F-FDG uptake. All these lesions were confirmed to be metabolically inactive. Coronal CT (D) image demonstrated calculi in the right renal calyx probably caused by hyperparathyroidism (arrow).
after the surgery. Based on both imaging results and the improved clinical status of the patient, the aforementioned lesions were suspected to be brown tumors, although not confirmed histologically. Conclusion The diagnosis of brown tumor should be considered in patients with hyperparathyroidism and multiple destructive bone lesions. Moreover, as the concomitant thyroid carcinoma is not uncommon among primary hyperparathyroidism patients, prior to surgical intervention thyroid nodules should be further investigated with FNAB if possible. Conflicts of interests None. References 1. Knowles NG, Smith DL, Outwater EK. MRI diagnosis of brown tumor based on magnetic susceptibility. J Magn Reson Imaging. 2008;28:759–61.
2. Irie T, Mawatari T, Ikemura S, Matsui G, Iguchi T, Mitsuyasu H. Brown tumor of the patella caused by primary hyperparathyroidism: a case report. Korean J Radiol. 2015;16:613–6. 3. Cinamon U, Levy D, Marom T. Is primary hyperparathyroidism a risk factor for papillary thyroid cancer. An exemplar study and literature review. Int Arch Otorhinolaryngol. 2015;19:42–5. 4. Jackson W, Sethi A, Carp J, Talpos G, Vaidya R. Unusual spinal manifestation in secondary hyperparathyroidism: a case report. Spine (Phila Pa 1976). 2007;32:E557–60. 5. Hoshi M, Takami M, Kajikawa M, Teramura K, Okamoto T, Yanagida I, et al. A case of multiple skeletal lesions of brown tumors, mimicking carcinoma metastases. Arch Orthop Trauma Surg. 2008;128:149–54. 6. Fineman I, Johnson JP, Di-Patre PL, Sandhu H. Chronic renal failure causing brown tumors and myelopathy. Case report and review of pathophysiology and treatment. J Neurosurg. 1999;90:242–6. 7. Mateo L, Massuet A, Sola M, Perez Andres R, Musulen E, Sanchez Torres MC. Brown tumor of the cervical spine: a case report and review of the literature. Clin Rheumatol. 2011;30:419–24. 8. Kaya RA, Cavusoglu H, Tanik C, Kahyao˘glu O, Dilbaz S, Tuncer C, et al. Spinal cord compression caused by a brown tumor at the cervicothoracic junction. Spine J. 2007;7:728–32. 9. Leitha T, Staudenherz A. Concomitant hyperparathyroidism and nonmedullary thyroid cancer, with a review of the literature. Clin Nucl Med. 2003;28: 113–7. 10. Uslu L, C¸alıs¸kan A, Sa˘ger S, Teksöz S, Halac¸ M. Preoperative and postoperative 18 FDG PET/CT and bone scinigraphy images in a patient with parathyroid adenoma. Clin Nucl Med. 2013;38:456–8.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
ARTICLE IN PRESS Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
Clinical note
An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma Y. Basaran b , S. Ince a , E. Alagoz a,∗ , C. Meric b , A. Taslipinar b a b
Gulhane Military Medical Academy and School of Medicine, Department of Nuclear Medicine, Etlik, Ankara, Turkey Gulhane Military Medical Academy and School of Medicine, Department of Endocrinology and Metabolism, Etlik, Ankara, Turkey
a r t i c l e
i n f o
Article history: Received 11 January 2016 Accepted 22 February 2016 Available online xxx Keywords: Parathyroid adenoma Brown tumor Bone scintigraphy FDG PET/CT
a b s t r a c t We present a patient with a complex clinical picture of primary hyperparathyroidism with multiple destructive skeletal lesions suspicious of bone metastases and concomitant multifocal papillary thyroid carcinoma with a metastatic central lymph node. He presented with progressively worsening right hip pain and restricted motion. Magnetic resonance imaging revealed multiple lytic lesions involving predominantly the right trochanter minor and the left inferior and posterior pubic rami. Biochemical tests were consistent with primary hyperparathyroidism. Neck ultrasound and parathyroid scintigraphy revealed a single parathyroid adenoma and a thyroid nodule, preoperative cytology of which confirmed papillary thyroid carcinoma, as did the final surgical specimen. Biochemical results, regarding hyperparathyroidism, declined to normal levels and his complaints gradually decreased after surgery. Postoperative whole body bone scintigraphy showed increased tracer uptakes at multiple sites, but they were proved to be metabolically inactive by fluorodeoxyglucose positron emission tomography/computed tomography. © 2016 Elsevier España, S.L.U. and SEMNIM. All rights reserved.
Presentación inusual de hiperparatiroidismo primario: múltiples tumores pardos y coexistencia de carcinoma tiroideo r e s u m e n Palabras clave: Adenoma paratiroideo Tumor pardo Gammagrafía ósea FDG PET/TC
Presentamos el caso de un paciente con un cuadro clínico de hiperparatiroidismo primario, con muchas lesiones óseas destructivas sospechosas de metástasis óseas y carcinoma tiroideo multifocal concomitante con un ganglio linfático metastásico central. Se presentó con agravamiento progresivo de dolor y restricción de movimiento en la cadera derecha. La resonancia magnética reveló múltiples lesiones líticas que implicaban principalmente al trocánter menor derecho y a las ramas púbicas izquierdas inferior y posterior. Las pruebas bioquímicas fueron consistentes con un hiperparatiroidismo primario. La ecografía cervical y la gammagrafía paratiroidea revelaron un único adenoma paratiroideo y un nódulo tiroideo, cuya citología preoperatoria confirmó un carcinoma papilar de tiroides, que fue confirmado también por la muestra final obtenida quirúrgicamente. Los resultados bioquímicos, en relación con el hiperparatiroidismo, descendieron a niveles normales tras la cirugía, y los dolores fueron remitiendo gradualmente. La gammagrafía ósea postoperatoria de cuerpo entero reflejó un incremento de captación del radiotrazador en múltiples localizaciones, que resultaron ser metabólicamente inactivas en la tomografía por emisión de positrones con fluorodesoxiglucosa/tomografía computarizada. © 2016 Elsevier España, S.L.U. y SEMNIM. Todos los derechos reservados.
Introduction Brown tumors are rare non-neoplastic skeletal manifestations of primary and secondary hyperparathyroidism1 . Common sites of involvement are pelvis, femur and ribs, but may appear in any bone2 . Since the incidence of thyroid disease is higher among patients with hyperparathyroidism than the general population3 , these
lesions must be distinguished from metastases from thyroid carcinoma. Thus, a comprehensive preoperative evaluation, including fine-needle aspiration of thyroid nodules even not reported to have malignant features on ultrasound (US), should be carried out. We present a case of a 38-year-old male with multiple brown tumors caused by primary hyperparathyroidism and coexisting metastatic multifocal papillary thyroid carcinoma. Clinical case
∗ Corresponding author. E-mail address: [email protected] (E. Alagoz).
The patient was admitted with expansile lytic lesions occupying mostly trochanter minor of the right femoral neck, and the left
http://dx.doi.org/10.1016/j.remn.2016.02.005 2253-654X/© 2016 Elsevier España, S.L.U. and SEMNIM. All rights reserved.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
G Model REMNIM-788; No. of Pages 4 2
ARTICLE IN PRESS Y. Basaran et al. / Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
Fig. 1. Preoperative coronal T1-weighted MRI demonstrated a hypointense lytic expansile mass lesion at the trochanter minor of the right femoral neck (arrow). Significant contrast enhancement in the mass lesion and some degree of soft tissues expansion was noted.
inferior and posterior parts of pubic ramus (30 mm × 16 mm × 5.5 mm, 16 mm × 6.5 mm and 23 mm × 17 mm in diameter, respectively) on magnetic resonance imaging (MRI). They were reported to be hypointense on T1 and hyperintense on T2 weighted images, with various amounts of bone destruction, cortical thinning and some degree of soft tissue expansion around. Brown tumor was the most probable radiologic diagnosis (Fig. 1). At admission, he complained of severe hip pain, which had persisted for one year and progressively worsened over time, limiting his daily activities. On physical examination, tenderness with flexion and rotation of the right hip was detected, resulting in reduced range of motion in the hip joint. He had a 3-year history of kidney stones treatment with ureteral stenting and extracorporeal shock wave lithotripsy. But the rest of his medical history was unremarkable with no history of recent trauma. Biochemical tests of note were PTH of 896.9 pg/mL (normal: 11.1–79.5), calcium of 15.06 mg/dL (normal: 8.5–10.5), phosphate of 1.87 mg/dL (normal: 2.6–4.5) and vitamin D3 of 18.4 mcg/L (normal: 11–70). Thyroid US displayed a 16 mm × 15 mm hypoechoic mass in the lower right pole of the gland, suggested for a parathyroid adenoma, and a thyroid nodule with irregular borders which was suspicious for malignancy (8 mm × 4 mm in diameter) in the left lobe. There were no grossly defined lymph nodes. Technetium-99m methoxyisobutylisonitrile (99m Tc MIBI) scan revealed focal increased radiopharmaceutical uptake just
below the lower pole of right thyroid lobe (Fig. 2). Bone density by DXA revealed a distal radius T-score of −3.7. An ultrasoundguided fine-needle aspiration biopsy (FNAB) of the thyroid nodule was performed, which was suggestive of a malignant cytology. Preoperative exploration for exclusion of multiple endocrine neoplasia did not find evidence of pituitary, pancreatic or adrenal tumors. To promote tumor regression and reduce hungry bone syndrome calcium and vitamin D treatment was initiated. Subsequently, the patient underwent bilateral total thyroidectomy with central compartment neck dissection and right inferior parathyroidectomy. Histological examination confirmed the presence of a parathyroid adenoma and three papillary micro-carcinomas of thyroid (with dimensions of 6 mm and 2 mm in the left lobe and 2 mm in the right lobe) with a central lymph node metastasis. Postoperatively, the patient received an ablative dose of 100 mCi (3700 MBq) iodine131. Post-radioactive iodine whole body scan showed an uptake in the thyroid bed, but no ectopic uptake suggestive of local or distant metastases was visualized. The patient was followed-up on levothyroxine 150 mcg daily and was in a complete remission, with undetectable levels of serum thyroglobulin (Tg) and anti-Tg postoperatively. PTH and calcium levels decreased to 53 pmol/L and 9.21 mg/dL, respectively. Moreover, the biochemistry regarding primary hyperparathyroidism remained normal during 3-monthly follow-ups and his symptoms of hip pain considerably weakened, but did not completely resolve. At the 18-month final follow-up, the patient persisted to experience mild symptoms of hip pain during activity and was subjected to whole-body bone scan, which revealed increased tracer uptakes in left sternoclavicular joint region, left proximal forearm, left pubic ramus and right femoral neck (Fig. 3). Single photon emission computed tomography/CT (SPECT/CT) images of the left ulna demonstrated an expansile lytic lesion with internal septas and SPECT/CT images of the pelvis showed increased tracer uptakes with peripheral sclerosis in the left pubic ramus and right femoral neck, the same osteolytic lesions seen on the preoperative MRI and postoperative whole body bone scans. In order to assess their metabolic activity, 18 F-FDG PET/CT was performed. There was an increased 18 F-FDG uptake only in the left sternoclavicular joint, which was highly suggestive of degeneration. No 18 F-FDG uptake in the aforementioned lesions was detected (Fig. 4). Discussion Hyperparathyroidism is a relatively common disorder. However, brown tumor, also known as osteoclastoma, is a rare benign bone lesion with incidence ranging from 1.5% in primary hyperparathyroidism to 13% in secondary hyperparathyroidism4 . It results from disturbed bone remodeling due to the long-lasting
Fig. 2. 99m Tc MIBI parathyroid scintigraphy planar (A) and SPECT/CT coronal (B) images showed a focus of intense radiopharmaceutical uptake (arrow) just below lower pole of the right thyroid lobe.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
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ARTICLE IN PRESS Y. Basaran et al. / Rev Esp Med Nucl Imagen Mol. 2016;xxx(xx):xxx–xxx
3
Fig. 3. 99m Tc whole-body bone scan after parathyroidectomy (A) displayed increased tracer uptakes in left sternoclavicular joint (thick arrow), left proximal forearm (arrowhead), left pubic ramus (short arrow) and right femoral neck (long arrow). CT image of left ulna (B) revealed expansile lytic lesions with lamellar style septas, a finding suggestive of brown tumor (arrowhead). Axial CT (C) and SPECT/CT (D) images of pelvis demonstrated increased tracer uptakes with peripheral sclerosis at the right femoral neck (long arrow) and left pubic ramus (short arrow), all consistent with brown tumors.
increase in PTH levels.5 Brown tumor can be confused with more serious bone lesions, including aneurysmal bone cyst, giant cell tumor, myeloma, Paget’s disease,6 leukemia, histiocytosis,7 and even metastasis.8 Although the guidelines do not consider primary hyperparathyroidism a risk factor for thyroid carcinoma, several data suggest that this risk may be increased when compared to the general population,3 which was in accordance with the present case. Concomitant non-medullary thyroid carcinoma was found in approximately 3% of patients operated on for primary hyperparathyroidism.9 We believe that a preoperative evaluation of all thyroid nodules should be considered among patients with hyperparathyroidism, regardless of their US features. The present case presented with a complaint of severe pain in the right hip. MRI revealed multiple lytic bone lesions suggestive of brown tumors, but also suspicious of metastases. Laboratory tests
confirmed the presence of the hyperparathyroidism. Thyroid US and parathyroid scintigraphy revealed a suspicious thyroid nodule and a single parathyroid adenoma. Preoperative examination of the thyroid nodule was consistent with a malignant cytology. The patient underwent a complete surgical removal of whole thyroid gland and right inferior parathyroid. Interestingly, histological examination confirmed the presence of a metastatic multifocal papillary thyroid carcinoma. Subsequently, he was subjected to radioactive iodine ablation and followed with TSH suppression therapy. Further exploration depicted multiple sites of increased uptake on whole-body bone scan. However, 18 F-FDG PET/CT did not exhibit an abnormal uptake throughout the skeletal system, which proved these osteolytic lesions to be metabolically inactive. Unlike bone scan, 18 F-FDG PET/CT is expected to revert to normal within 3 months after fracture or trauma.10 Additionally, the right hip pain noticeably attenuated with dramatically increased range of motion
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005
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Fig. 4. Maximum intensity projection (MIP) image of 18 F-FDG PET/CT after parathyroidectomy (A) showed increased 18 F-FDG uptake in the left sternoclavicular joint region (arrow), but no 18 F-FDG uptake was noted in the other bone lesions. Axial CT (B) and axial fusion (C) images revealed increased 18 F-FDG uptake in the left sternoclavicular joint suggestive of degeneration (arrow). The lytic lesion on the left side of sternum, which was previously thought as a brown tumor had no 18 F-FDG uptake. All these lesions were confirmed to be metabolically inactive. Coronal CT (D) image demonstrated calculi in the right renal calyx probably caused by hyperparathyroidism (arrow).
after the surgery. Based on both imaging results and the improved clinical status of the patient, the aforementioned lesions were suspected to be brown tumors, although not confirmed histologically. Conclusion The diagnosis of brown tumor should be considered in patients with hyperparathyroidism and multiple destructive bone lesions. Moreover, as the concomitant thyroid carcinoma is not uncommon among primary hyperparathyroidism patients, prior to surgical intervention thyroid nodules should be further investigated with FNAB if possible. Conflicts of interests None. References 1. Knowles NG, Smith DL, Outwater EK. MRI diagnosis of brown tumor based on magnetic susceptibility. J Magn Reson Imaging. 2008;28:759–61.
2. Irie T, Mawatari T, Ikemura S, Matsui G, Iguchi T, Mitsuyasu H. Brown tumor of the patella caused by primary hyperparathyroidism: a case report. Korean J Radiol. 2015;16:613–6. 3. Cinamon U, Levy D, Marom T. Is primary hyperparathyroidism a risk factor for papillary thyroid cancer. An exemplar study and literature review. Int Arch Otorhinolaryngol. 2015;19:42–5. 4. Jackson W, Sethi A, Carp J, Talpos G, Vaidya R. Unusual spinal manifestation in secondary hyperparathyroidism: a case report. Spine (Phila Pa 1976). 2007;32:E557–60. 5. Hoshi M, Takami M, Kajikawa M, Teramura K, Okamoto T, Yanagida I, et al. A case of multiple skeletal lesions of brown tumors, mimicking carcinoma metastases. Arch Orthop Trauma Surg. 2008;128:149–54. 6. Fineman I, Johnson JP, Di-Patre PL, Sandhu H. Chronic renal failure causing brown tumors and myelopathy. Case report and review of pathophysiology and treatment. J Neurosurg. 1999;90:242–6. 7. Mateo L, Massuet A, Sola M, Perez Andres R, Musulen E, Sanchez Torres MC. Brown tumor of the cervical spine: a case report and review of the literature. Clin Rheumatol. 2011;30:419–24. 8. Kaya RA, Cavusoglu H, Tanik C, Kahyao˘glu O, Dilbaz S, Tuncer C, et al. Spinal cord compression caused by a brown tumor at the cervicothoracic junction. Spine J. 2007;7:728–32. 9. Leitha T, Staudenherz A. Concomitant hyperparathyroidism and nonmedullary thyroid cancer, with a review of the literature. Clin Nucl Med. 2003;28: 113–7. 10. Uslu L, C¸alıs¸kan A, Sa˘ger S, Teksöz S, Halac¸ M. Preoperative and postoperative 18 FDG PET/CT and bone scinigraphy images in a patient with parathyroid adenoma. Clin Nucl Med. 2013;38:456–8.
Please cite this article in press as: Basaran Y, et al. An unusual presentation of primary hyperparathyroidism: multiple brown tumors and coexisting thyroid carcinoma. Rev Esp Med Nucl Imagen Mol. 2016. http://dx.doi.org/10.1016/j.remn.2016.02.005