Phosphaturic mesenchymal tumors involving skull bones: Report of two rare cases

Mishra, Toshi; Desouza, Maria Alina; Mazumdar, Girish A

doi:10.4103/ajns.ajns_176_17

Cited by 3 publications

(5 citation statements)

References 12 publications

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“…As in our case, the tumor was detected in other six patients by functional imaging [ 7 – 12 ]. The suspicious lesion was confirmed by MRI in our case and in other five patients [ 7 – 10 ] and by CT scan in another one [ 11 ]. Interestingly, the tumor involved invariably the left side of the occipital bone.…”

Section: Literature Reviewsupporting

confidence: 74%

“…In all surgically treated patients [ 6 – 10 ], the histological diagnosis was consistent with PMT, the most common tumor associated with TIO [ 1 ]. It is likely that even in the case reported by Luthra and colleagues [ 12 ] and in our case this would have been the diagnosis if the tumor had been excised.…”

Section: Discussionmentioning

confidence: 99%

“…Of note, only three patients had a complete recovery after surgery [ 7 , 9 , 10 ]. This percentage is below that reported in the literature [ 15 ] emphasizing that occipital bone is a difficult location to approach and for which combined treatments might be necessary.…”

Section: Discussionmentioning

confidence: 99%

“…Interestingly, the tumor involved invariably the left side of the occipital bone. In five patients [ 6 , 8 – 10 , 12 ], the clivus was also involved. In one patient, data on treatment and follow-up were not available [ 12 ].…”

Section: Literature Reviewmentioning

confidence: 99%

“…In three patients [ 9 – 11 ], a postoperative drop of serum FGF23 was documented after surgery. The complete surgical removal of the tumor was obtained in three patients [ 7 , 9 , 10 ], with at least 3 months of follow-up for the two of them. Surgery was unsuccessful in one case [ 8 ].…”

Section: Literature Reviewmentioning

confidence: 99%

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

et al. 2023

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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.

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Section: Literature Reviewsupporting

confidence: 74%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Literature Reviewmentioning

confidence: 99%

Section: Literature Reviewmentioning

confidence: 99%

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

et al. 2023

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Intracranial Phosphaturic Mesenchymal Tumors: A Systematic Literature Review of a Rare Entity

Sánchez Muñoz,

Sanmillan,

Vidal Sarró

et al. 2024

World Neurosurgery

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Tumor induced osteomalacia in head and neck region: single center experience and systematic review

Shah

Lila

Jadhav

et al. 2019

Endocrine Connections

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Tumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal sinuses (n = 3), intracranial (n = 2) and maxilla (n = 2). The unique features from our series is the management of persistent disease with radiation therapy (n = 2) and peptide receptor radionuclide therapy (PRRT) (n = 1). Cohort two has 163 patients identified from 109 publications for systematic review. Paranasal sinuses, mandible, intracranial disease, maxilla and oral cavity, in descending order, are reportedly common tumor sites. Within this cohort, mean age was 46 ± 14 years at presentation with 44.1% having local symptoms. Duration of symptoms varied from 1 to 240 months. Pre-surgery mean serum phosphorus was 1.4 ± 0.4 mg/dL and median FGF-23 levels were 3.6 (IQR:1.8–6.8) times of normal upper limit of normal. Majority (97.5%) were managed primarily with surgical excision; however, primary radiotherapy (n = 2) and surgery combined with radiotherapy (n = 2) were also reported. Twenty patients had persistent disease while nine patients had recurrence, more commonly noted with intracranial and oral cavity tumors. Surgery was the most common second mode of treatment employed succeeded by radiotherapy. Four patients had metastatic disease. The most common histopathological diagnosis reported is PMT mixed connective tissue, while the newer terminology ‘PMT mixed epithelial and connective tissue type’ has been described in 15 patients.

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Phosphaturic mesenchymal tumors involving skull bones: Report of two rare cases

Cited by 3 publications

References 12 publications

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

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

Intracranial Phosphaturic Mesenchymal Tumors: A Systematic Literature Review of a Rare Entity

Tumor induced osteomalacia in head and neck region: single center experience and systematic review

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