Phosphaturic mesenchymal tumour of the mandible – the useful criteria for a diagnosis on fine needle aspiration cytology

Monappa, Vidya; Naik, Monappa A; Mathew, Mary; Rao, Lakshmi; Rao, S. B.; Ramachandra, Lakshmi; Jaiprakash, Padmapriya

doi:10.1111/cyt.12030

Cited by 8 publications

(14 citation statements)

References 10 publications

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“…Case 3 was a 13 year old female and case 12 was a 9 year old female. The most common locations are the extremities, followed by head and neck sites, with isolated lesions also found in the chest wall, pharynx, breast, and spinal nerve [50,59]. In the H&N, the paranasal sinuses were the most favored location for PMT [3].…”

Section: Discussionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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Phosphaturic mesenchymal tumor (PMT) is a rare neoplasm that secretes fibroblast growth factor-23 (FGF-23) and causes oncogenic osteomalacia. It occurs in adults with equal gender distribution and the most common location is the lower extremities, followed by the head and neck. Besides osteomalacia, the clinical presentation includes bone pain and multiple bone fractures. Microscopic features consist of spindle cells, multinucleated giant cells, and calcifications embedded in a chondromyxoid matrix. Laboratory findings indicate normal calcium and parathyroid levels, hypophosphatemia, and increased levels of FGF-23 that usually revert to normal after surgical removal. Due to its rarity, the purpose of the study was to report 2 new oral cases of PMT and to review the literature in the head and neck. The first case occurred in the gingiva and had been present for 6 years. The second case was a recurrence of a previously diagnosed PMT in the right mandible that metastasized to the lung and soft tissue. The literature review included 53 cases in the head and neck. There was a predilection for extra-oral sites (76 %) compared to intra-oral sites (24 %) with paranasal sinuses considered the most common location (38 %) followed by the mandible (15 %). There were 9 recurrences that included 3 malignant cases indicating a potentially aggressive tumor. Due to the indeterminate biological behavior of PMT and its rarity, a comprehensive evaluation of medical, laboratory, radiographic, and histological findings are crucial for a definitive diagnosis and treatment.

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Section: Discussionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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show abstract

“…Twelve of these reported cases were noted in intraoral sites, with the mandible being the most common, followed by the floor of the mouth, tongue, gingiva, and maxilla . Involvement of the mandible is exceptionally rare, with only eight reported cases in the literature to date …”

Section: Discussionmentioning

confidence: 99%

“…Of the 55 cases of head and neck TIO reported in the medical literature, 9 had recurrence . Of the eight cases of mandibular TIO, six were treated with surgery . One was treated with curettage because of its proximity to the inferior alveolar nerve canal …”

Section: Discussionmentioning

confidence: 99%

“…After considering several therapeutic options, we elected to surgically resect the tumor using a segmental mandibulectomy, a procedure that, to our knowledge, has not been previously used for this disorder . Segmental mandibulectomy ensures a complete surgical resection with wide margins.…”

Section: Discussionmentioning

confidence: 99%

“…23 Involvement of the mandible is exceptionally rare, with only eight reported cases in the literature to date. 7,[23][24][25][26][27][28][29] Our first major challenge was to understand the reason for the patient's chief complaint. The patient was seen with 1 year of nonspecific worsening symptoms of bone pain, muscle weakness, and fractures before he was diagnosed.…”

Section: Discussionmentioning

confidence: 99%

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Tumor‐induced hypophosphatemic osteomalacia caused by a mesenchymal tumor of the mandible managed by a segmental mandibulectomy and microvascular reconstruction with a free fibula flap

et al. 2019

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Background Tumor‐induced osteomalacia is a rare paraneoplastic syndrome in which patients develop hypophosphatemia and osteomalacia. Methods and Results Here, we report a unique case of a 42‐year‐old man who presented to our institution with a 1‐year history of pain in his ribs, hips, lower back, and feet. Radiologic examination revealed a decrease in bone density and multiple insufficiency fractures. Laboratory evaluation revealed hypophosphatemia, low serum 1,25 dihydroxy vitamin D3, and elevated fibroblast growth factor 23 (FGF23). A positron emission tomography/CT scan showed increased uptake in the right mandibular third molar region. Panoramic radiography and CT scanning showed a lytic expansile bone lesion. A mandibular bone biopsy revealed a mixed connective tissue tumor. A right segmental mandibulectomy was performed, followed by microvascular reconstruction. The resection was confirmed by normalization of serum phosphate and FGF23. Conclusion Successful management of this condition was achieved, with complete surgical resection of the tumor and reconstructive surgery.

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Cytological findings of phosphaturic mesenchymal tumor: Report of a case with summary of prior published cases

Das,

Mohin,

Dey

et al. 2023

Diagnostic Cytopathology

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Phosphaturic mesenchymal tumor (PMT) is a rare neoplasm causing tumor‐induced osteomalacia (TIO) and is characterized by secretion of FGF23, renal phosphate wasting and hypophosphataemia. It can be completely cured by resection and therefore its diagnosis is of utmost importance. Although the histology is well described, there is sparse literature on cytology of PMT and only three cases have been described so far. A 45‐year‐old lady presented with a non‐tender mass in hard palate for 2 years from which fine‐needle aspiration was done. The smears were paucicellular and showed bland spindle cells embedded in osteoid‐like stromal matrix in a hemorrhagic background. Here we take the opportunity to describe the cytological findings of PMT along with its cytological differentials and a summary of prior published cases.

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Phosphaturic mesenchymal tumour of the mandible – the useful criteria for a diagnosis on fine needle aspiration cytology

Cited by 8 publications

References 10 publications

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Tumor‐induced hypophosphatemic osteomalacia caused by a mesenchymal tumor of the mandible managed by a segmental mandibulectomy and microvascular reconstruction with a free fibula flap

Cytological findings of phosphaturic mesenchymal tumor: Report of a case with summary of prior published cases

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