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Oncogenic osteomalacia caused by occult nasal mesenchymal tumor: a monster in the cave.

Ray S, Chakraborty PP, Biswas K, Beatrice AM, Ghosh S, Mukhopadhyay S, Chowdhury S - Oxf Med Case Reports (2015)

Bottom Line: A whole-body functional imaging failed to reveal any areas of increased activity.The tumor was surgically removed.After surgery, his symptoms were relieved and biochemical parameters normalized.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Endocrinology and Metabolism , Institute of Post Graduate Medical Education and Research (IPGMER) and SSKM Hospital , Kolkata , India.

ABSTRACT
Authors describe a case of oncogenic osteomalacia in a 35-year-old man, who presented with a 2-year history of generalized pain and progressive weakness of lower limbs, eventually became bedbound. At admission, he had severe hip pain resulting from bilateral femoral neck fractures. Laboratory investigations revealed hypophosphatemia, hyperphosphaturia, normocalcemia, elevated alkaline phosphatase and normal serum levels of parathormone and 25-hydroxyvitamin D. Serum fibroblast growth factor 23 (FGF23) level was elevated. A radiographic skeletal survey showed osteoporosis and insufficiency fractures of the femoral neck. A whole-body functional imaging failed to reveal any areas of increased activity. However, on computed tomography and magnetic resonance imaging of the head and neck region, a tumor was discovered at left nasal cavity. The tumor was surgically removed. After surgery, his symptoms were relieved and biochemical parameters normalized. We stress that careful clinical examination including nose and paranasal sinuses may be rewarding in cases with hypophosphatemic osteomalacia.

No MeSH data available.


Related in: MedlinePlus

(A) Frontal pelvic radiograph showing generalized osteopenia, and bilateral impacted subcapital fractures of the femoral neck with coxa vara deformity. (B) Axial non-contrast CT scan of pelvis at the level of hip joint showing bilateral impacted fractures of the femoral neck and fracture of the left quadrilateral plate of acetabulum.
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OMV031F1: (A) Frontal pelvic radiograph showing generalized osteopenia, and bilateral impacted subcapital fractures of the femoral neck with coxa vara deformity. (B) Axial non-contrast CT scan of pelvis at the level of hip joint showing bilateral impacted fractures of the femoral neck and fracture of the left quadrilateral plate of acetabulum.

Mentions: Laboratory evaluation revealed a decreased serum phosphorus level of 1.5 mg/dl (normal, 2.5–4.5), an elevated serum total alkaline phosphatase concentration of 1756 U/l (normal, 98–251) and a low normal serum 1,25-dihydroxyvitamin D value of 16 pg/ml (normal, 19.6–54.3). Both serum intact parathormone and 25-hydroxyvitamin D levels were normal. Urine biochemistry confirmed a low renal tubular resorption of phosphate. Serum FGF23 measured using a carboxy-terminal FGF-23 ELISA was more than five times the upper limit of normal. A radiographic skeletal survey revealed diffuse osteopenia and insufficiency fractures of bilateral femoral neck and right ischiopubic ramus. A non-contrast computed tomography (CT) scan of pelvis showed intracortical and trabecular bone resorption (Fig. 1A and B). DXA scan of his lumbar spine showed a bone density of 0.592 g/cm2 with a Z-score of −4.0, and hip showed 0.458 g/cm2 with a Z-score of −3.8. The clinical presentation and laboratory findings suggested a diagnosis of OOM. Therefore, an effort was made to localize the tumor. Surprisingly, a whole-body 99mTc-sestamibi scintigraphy failed to detect any area with increased tracer uptake. However, on CT and magnetic resonance imaging (MRI), a tumor was discovered in the superior aspect of left nasal cavity (Fig. 2A and B).Figure 1:


Oncogenic osteomalacia caused by occult nasal mesenchymal tumor: a monster in the cave.

Ray S, Chakraborty PP, Biswas K, Beatrice AM, Ghosh S, Mukhopadhyay S, Chowdhury S - Oxf Med Case Reports (2015)

(A) Frontal pelvic radiograph showing generalized osteopenia, and bilateral impacted subcapital fractures of the femoral neck with coxa vara deformity. (B) Axial non-contrast CT scan of pelvis at the level of hip joint showing bilateral impacted fractures of the femoral neck and fracture of the left quadrilateral plate of acetabulum.
© Copyright Policy - creative-commons
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4664848&req=5

OMV031F1: (A) Frontal pelvic radiograph showing generalized osteopenia, and bilateral impacted subcapital fractures of the femoral neck with coxa vara deformity. (B) Axial non-contrast CT scan of pelvis at the level of hip joint showing bilateral impacted fractures of the femoral neck and fracture of the left quadrilateral plate of acetabulum.
Mentions: Laboratory evaluation revealed a decreased serum phosphorus level of 1.5 mg/dl (normal, 2.5–4.5), an elevated serum total alkaline phosphatase concentration of 1756 U/l (normal, 98–251) and a low normal serum 1,25-dihydroxyvitamin D value of 16 pg/ml (normal, 19.6–54.3). Both serum intact parathormone and 25-hydroxyvitamin D levels were normal. Urine biochemistry confirmed a low renal tubular resorption of phosphate. Serum FGF23 measured using a carboxy-terminal FGF-23 ELISA was more than five times the upper limit of normal. A radiographic skeletal survey revealed diffuse osteopenia and insufficiency fractures of bilateral femoral neck and right ischiopubic ramus. A non-contrast computed tomography (CT) scan of pelvis showed intracortical and trabecular bone resorption (Fig. 1A and B). DXA scan of his lumbar spine showed a bone density of 0.592 g/cm2 with a Z-score of −4.0, and hip showed 0.458 g/cm2 with a Z-score of −3.8. The clinical presentation and laboratory findings suggested a diagnosis of OOM. Therefore, an effort was made to localize the tumor. Surprisingly, a whole-body 99mTc-sestamibi scintigraphy failed to detect any area with increased tracer uptake. However, on CT and magnetic resonance imaging (MRI), a tumor was discovered in the superior aspect of left nasal cavity (Fig. 2A and B).Figure 1:

Bottom Line: A whole-body functional imaging failed to reveal any areas of increased activity.The tumor was surgically removed.After surgery, his symptoms were relieved and biochemical parameters normalized.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Endocrinology and Metabolism , Institute of Post Graduate Medical Education and Research (IPGMER) and SSKM Hospital , Kolkata , India.

ABSTRACT
Authors describe a case of oncogenic osteomalacia in a 35-year-old man, who presented with a 2-year history of generalized pain and progressive weakness of lower limbs, eventually became bedbound. At admission, he had severe hip pain resulting from bilateral femoral neck fractures. Laboratory investigations revealed hypophosphatemia, hyperphosphaturia, normocalcemia, elevated alkaline phosphatase and normal serum levels of parathormone and 25-hydroxyvitamin D. Serum fibroblast growth factor 23 (FGF23) level was elevated. A radiographic skeletal survey showed osteoporosis and insufficiency fractures of the femoral neck. A whole-body functional imaging failed to reveal any areas of increased activity. However, on computed tomography and magnetic resonance imaging of the head and neck region, a tumor was discovered at left nasal cavity. The tumor was surgically removed. After surgery, his symptoms were relieved and biochemical parameters normalized. We stress that careful clinical examination including nose and paranasal sinuses may be rewarding in cases with hypophosphatemic osteomalacia.

No MeSH data available.


Related in: MedlinePlus