“Non-phosphaturic” Variant of Phosphaturic Mesenchymal Tumor of the Middle Ear Expressing Multiple Phosphatonins

Palmisano, Biagio; Pace, Annalisa; Colangelo, Luciano; Polimeni, Roberta; Sonato, Chiara; Messineo, Daniela; Minisola, Salvatore; Magliulo, Giuseppe; Riminucci, Mara; Corsi, Alessandro

doi:10.1177/01455613221149635

Cited by 2 publications

(1 citation statement)

References 19 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…They suggested that these variants may be tumors that secrete inactive or insufficient FGF23. In some reported cases of non-phosphaturic variants ( 10 – 12 ), the patients did not show clinical signs of TIO. In this case, the patient presents only non-phosphaturia, but with clinical and laboratory evidence of TIO.…”

Section: Discussionmentioning

confidence: 84%

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Zhang,

Hu,

Guo

et al. 2024

Front. Endocrinol.

View full text Add to dashboard Cite

Phosphaturic mesenchymal tumors (PMT) are rare and distinctive tumors that typically result in paraneoplastic syndrome known as tumor-induced osteomalacia (TIO). We report a case of bilateral osteoporotic femoral neck fracture caused by PMT. PMT was surgically resected, followed by sequential treatment of bilateral femoral neck fractures with total hip arthroplasty (THA). A 49-year-old perimenopausal woman experienced consistent bone pain with limb weakness persisting for over 2 years. Initially, she was diagnosed with early osteonecrosis of the femoral head and received nonsurgical treatment. However, from 2020 to 2022, her pain extended to the bilateral shoulders and knees with increased intensity. She had no positive family history or any other genetic diseases, and her menstrual cycles were regular. Physical examination revealed tenderness at the midpoints of the bilateral groin and restricted bilateral hip range of motion, with grade 3/5 muscle strength in both lower extremities. Laboratory findings revealed moderate anemia (hemoglobin 66 g/L), leukopenia (2.70 × 109/L), neutropenia (1.28 × 109/L), hypophosphatemia (0.36 mmol/L), high alkaline phosphatase activity (308.00 U/L), and normal serum calcium (2.22 mmol/L). After surgery, additional examinations were performed to explore the cause of hypophosphatemic osteomalacia. After definitive diagnosis, the patient underwent tumor resection via T11 laminectomy on August 6, 2022. Six months after the second THA, the patient regained normal gait with satisfactory hip movement function without recurrence of PMT-associated osteomalacia or prosthesis loosening. By providing detailed clinical data and a diagnostic and treatment approach, we aimed to improve the clinical understanding of femoral neck fractures caused by TIO.

show abstract

Section: Discussionmentioning

confidence: 84%

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Zhang,

Hu,

Guo

et al. 2024

Front. Endocrinol.

View full text Add to dashboard Cite

show abstract

Occipital bone and tumor-induced osteomalacia: a rare tumor site for an uncommon paraneoplastic syndrome

et al. 2023

Self Cite

View full text Add to dashboard Cite

Introduction Tumor-induced osteomalacia (TIO) is an uncommon paraneoplastic syndrome due to the overproduction of fibroblast growth factor 23 (FGF23). It is predominantly caused by mesenchymal tumors and cured upon their complete removal. Non-surgical treatment is an alternative option but limited to specific clinical conditions. Methods We report a challenging case of TIO caused by a tumor involving the occipital bone. We also performed a literature review of TIO caused by tumors localized at this site, focusing on clinical findings, treatment, and outcomes. Results The patient, a 62-year-old male, presented with a long-lasting history of progressive weakness. Biochemical evaluation revealed severe hypophosphatemia due to low renal tubular reabsorption of phosphate with raised intact FGF23 values. A 68 Ga-DOTATATE PET/TC imaging showed a suspicious lesion located in the left occipital bone that MRI and selective venous catheterization confirmed to be the cause of TIO. Stereotactic gamma knife radiosurgery was carried out, but unfortunately, the patient died of acute respiratory failure. To date, only seven additional cases of TIO have been associated to tumors located in the occipital bone. Furthermore, the tumor involved the left side of the occipital bone in all these patients. Conclusion The occipital region is a difficult area to access so a multidisciplinary approach for their treatment is required. If anatomical differences could be the basis for the predilection of the left side of the occipital bone, it remains to be clarified.

show abstract

“Non-phosphaturic” Variant of Phosphaturic Mesenchymal Tumor of the Middle Ear Expressing Multiple Phosphatonins

Cited by 2 publications

References 19 publications

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Phosphaturic mesenchymal tumor-induced bilateral osteomalacia femoral neck fractures: a case report

Occipital bone and tumor-induced osteomalacia: a rare tumor site for an uncommon paraneoplastic syndrome

Contact Info

Product

Resources

About