Clinical, morphological and immunohistochemical analysis of 13 cases of phosphaturic mesenchymal tumor - A holistic diagnostic approach

Chatterjee, Debajyoti; Bardia, A; Pal, Rimesh; Saikia, Uma Nahar; Bhadada, Sanjay Kumar; Radotra, Bishan Dass

doi:10.1016/j.anndiagpath.2021.151783

Cited by 10 publications

(8 citation statements)

References 16 publications

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“…S-100 is a marker of neuroendocrine tumors. Chatterjee et al reported that an immunophenotype of PMT (SATB2 + /ERG + /CD56 + /S-100 − /STAT6 − ) helped distinguish PMT from its histological mimics (34). We believe that a negative staining of S-100 is useful in differential diagnosis, although several cases reported positive staining of S-100.…”

Section: Discussionmentioning

confidence: 80%

Sphenoid sinus is a rare site for tumor-induced osteomalacia: A case report and literature review

Wang

et al. 2023

Front. Endocrinol.

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BackgroundIn this paper, we present a rare case of tumor-induced osteomalacia (TIO) and a literature review of this rare disease.MethodsA case of TIO of the isolated sphenoid sinus was reported. Furthermore, the clinical features of TIO in the sphenoid sinus and other sinonasal sinuses were also reviewed and summarized.ResultsA 35-year-old man with muscle weakness and lower back pain came to the Department of Neurology. No obvious neurological disease was found; however, magnetic resonance imaging of the extremities accidentally showed a tumor in the axilla. Bone scintigraphy showed suspicious bone metastasis. Hypophosphatemia was neglected. Interestingly, 2-deoxy-2-[fluorine-18]fluoro-d-glucose positron emission tomography/computed tomography (18F-FDG PET/CT) detected a tumor in the axilla and another in the sphenoid sinus, but only the tumor in the sphenoid sinus had somatostatin receptor (SSTR) expression in 68-gallium 1,4,7,10-tetraazacyclododecane-1,4,7,10-tetraacetic acid octreotate (Ga-68 DOTATATE) PET/CT. The sphenoid sinus tumor was proven to be a phosphaturic mesenchymal tumor (PMT), and the phosphate levels returned to normal after surgery. The literature review showed only 17 cases of TIOs that occurred in the sphenoid sinus, with an average age of 43.3 ± 13.7 years. Only three cases of TIOs in the sphenoid sinus did not invade the nasal cavity or other paranasal sinuses, which could be identified as isolated sphenoid sinus diseases. We compared the clinical features of sphenoid TIOs with those of non-sphenoid sinonasal TIOs, and it was found that the concentration of 1,25-dihydroxy vitamin D in the group with sphenoid TIOs was much higher than that in the group with non-sphenoid sinonasal TIOs. A total of 153 cases of TIOs in the sinonasal sinus were reviewed. The ethmoid sinus was found to be the major site (64.7%), followed by the nasal cavity (50.3%), maxillary sinus (19.0%), frontal sinus (16.4%), and sphenoid sinus (11.8%). There were 66 patients (43.1%) who showed tumors invading more than one sinus. Most of the tumors (69.3%) were diagnosed as PMTs by pathology, followed by hemangiopericytoma (14.3%). Immunostaining was beneficial in the differential diagnosis of these tumors; however, larger sample sizes are needed for better accuracy.ConclusionTIO in the sinonasal sinus, especially in the sphenoid sinus, is rare. Moreover, isolated sphenoid sinus disease can be easily misdiagnosed. When the clinical manifestation of osteomalacia is atypical, associating it with sphenoid sinus disease is even more difficult. Thus, TIO in the sphenoid sinus needs further exploration.

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Section: Discussionmentioning

confidence: 80%

Sphenoid sinus is a rare site for tumor-induced osteomalacia: A case report and literature review

Wang

et al. 2023

Front. Endocrinol.

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“…PMTs display hemangiopericytoma (HPC)‐like vasculature and adipose tissue 997,1000,1001 . IHC helps differentiate from other lesions; PMTs are usually positive for CD56, ERG, and SATB2 1000,1001 …”

Section: Other Rare Benign Neoplasms and Lesionsmentioning

confidence: 99%

International Consensus Statement on Allergy and Rhinology: Sinonasal Tumors

Kuan

Wang

Adappa

et al. 2024

Int Forum Allergy Rhinol

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BackgroundSinonasal neoplasms, whether benign and malignant, pose a significant challenge to clinicians and represents a model area for multidisciplinary collaboration in order to optimize patient care. The International Consensus Statement on Allergy and Rhinology: Sinonasal Tumors (ICSNT) aims to summarize the best available evidence and presents 48 thematic and histopathology‐based topics spanning the field.MethodsIn accordance with prior ICAR documents, ICSNT assigned each topic as an Evidence‐Based Review with Recommendations, Evidence‐Based Review, and Literature Review based on level of evidence. An international group of multidisciplinary author teams were assembled for the topic reviews using the Preferred Reporting Items for Systematic Reviews and Meta‐Analyses format, and completed sections underwent a thorough and iterative consensus‐building process. The final document underwent rigorous synthesis and review prior to publication.ResultsThe ICNST document consists of 4 major sections: general principles, benign neoplasms and lesions, malignant neoplasms, and quality of life and surveillance. It covers 48 conceptual and/or histopathology‐based topics relevant to sinonasal neoplasms and masses. Topics with a high level of evidence provided specific recommendations, while other areas summarized the current state of evidence. A final section highlights research opportunities and future directions, contributing to advancing knowledge and community intervention.ConclusionAs an embodiment of the multidisciplinary and collaborative model of care in sinonasal neoplasms and masses, ICSNT was designed as a comprehensive, international, and multidisciplinary collaborative endeavor. Its primary objective is to summarize the existing evidence in the field of sinonasal neoplasms and masses.This article is protected by copyright. All rights reserved

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“…Phosphaturic mesenchymal tumor (PMT) is a rare tumor of soft tissue and bone of uncertain histiogenesis. It secretes FGF‐23, which causes renal phosphate wasting leading to tumor induced osteomalacia (TIO) 1 . The term “phosphaturic mesenchymal tumor” was coined by Weidner and Santa Cruz in 1987 2 .…”

Section: Introductionmentioning

confidence: 99%

“…Here we take the opportunity to describe the cytological findings of PMT along with its cytological differentials and a summary of prior published cases. 1 The term "phosphaturic mesenchymal tumor" was coined by Weidner and Santa Cruz in 1987. 2 They described this distinct mesenchymal neoplasm as a tumor composed of unusual mix of bland spindle cells in a highly vascular stroma along with the presence of microcysts, grungy calcified matrix and occasional osteoclast-like giant cells.…”

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confidence: 99%

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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Clinical, morphological and immunohistochemical analysis of 13 cases of phosphaturic mesenchymal tumor - A holistic diagnostic approach

Cited by 10 publications

References 16 publications

Sphenoid sinus is a rare site for tumor-induced osteomalacia: A case report and literature review

Sphenoid sinus is a rare site for tumor-induced osteomalacia: A case report and literature review

International Consensus Statement on Allergy and Rhinology: Sinonasal Tumors

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

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