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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) Microscopically, the tumor composed of cells arranged in sheets in a hyalinized stroma and multiple stag horn-shaped blood vessels lined by flat endothelial cells within the mass (hematoxylin and eosin, ×100). (B) Photomicrograph showing round to oval cells with minimal pleomorphism/mitotic activity, nuclei are vesicular with the presence of prominent small nucleoli in a hyalinized stroma; part of respiratory epithelium with pseudostratified ciliated columnar cells also present (hematoxylin and eosin, ×400).
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OMV031F3: (A) Microscopically, the tumor composed of cells arranged in sheets in a hyalinized stroma and multiple stag horn-shaped blood vessels lined by flat endothelial cells within the mass (hematoxylin and eosin, ×100). (B) Photomicrograph showing round to oval cells with minimal pleomorphism/mitotic activity, nuclei are vesicular with the presence of prominent small nucleoli in a hyalinized stroma; part of respiratory epithelium with pseudostratified ciliated columnar cells also present (hematoxylin and eosin, ×400).

Mentions: Meanwhile, we have to start phosphate (1.5 g/day) and calcitriol (0.75 µg/day) supplementations considering his debilitating symptoms. A decision was made to remove the possible causative tumor. The Otorhinolaryngology department was consulted and this time nasal endoscopy revealed a smooth pinkish mass in the region of left choana. Complete removal of the tumor was performed through lateral rhinotomy and at operation, the mass was found to be highly vascular. On subsequent microscopic examination, the specimen showed characteristic features of a hemangiopericytoma with the presence of branching staghorn-like blood vessels lying in a loose textured hyalinized stroma, surrounded by round- to spindle-shaped cells with scanty cytoplasm and vesicular nuclei (Fig. 3A and B). Post-surgery, he experienced clinical improvement of his musculoskeletal pains with normalization of the serum phosphorus and tubular phosphate reabsorption. Table 1 summarizes the laboratory findings before and after 4 weeks following the surgical tumor resections.Table 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) Microscopically, the tumor composed of cells arranged in sheets in a hyalinized stroma and multiple stag horn-shaped blood vessels lined by flat endothelial cells within the mass (hematoxylin and eosin, ×100). (B) Photomicrograph showing round to oval cells with minimal pleomorphism/mitotic activity, nuclei are vesicular with the presence of prominent small nucleoli in a hyalinized stroma; part of respiratory epithelium with pseudostratified ciliated columnar cells also present (hematoxylin and eosin, ×400).
© Copyright Policy - creative-commons
Related In: Results  -  Collection

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

OMV031F3: (A) Microscopically, the tumor composed of cells arranged in sheets in a hyalinized stroma and multiple stag horn-shaped blood vessels lined by flat endothelial cells within the mass (hematoxylin and eosin, ×100). (B) Photomicrograph showing round to oval cells with minimal pleomorphism/mitotic activity, nuclei are vesicular with the presence of prominent small nucleoli in a hyalinized stroma; part of respiratory epithelium with pseudostratified ciliated columnar cells also present (hematoxylin and eosin, ×400).
Mentions: Meanwhile, we have to start phosphate (1.5 g/day) and calcitriol (0.75 µg/day) supplementations considering his debilitating symptoms. A decision was made to remove the possible causative tumor. The Otorhinolaryngology department was consulted and this time nasal endoscopy revealed a smooth pinkish mass in the region of left choana. Complete removal of the tumor was performed through lateral rhinotomy and at operation, the mass was found to be highly vascular. On subsequent microscopic examination, the specimen showed characteristic features of a hemangiopericytoma with the presence of branching staghorn-like blood vessels lying in a loose textured hyalinized stroma, surrounded by round- to spindle-shaped cells with scanty cytoplasm and vesicular nuclei (Fig. 3A and B). Post-surgery, he experienced clinical improvement of his musculoskeletal pains with normalization of the serum phosphorus and tubular phosphate reabsorption. Table 1 summarizes the laboratory findings before and after 4 weeks following the surgical tumor resections.Table 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