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Frontiers in Microbiology 2024The incidence of oropharyngeal cancer (OPC) is increasing. This study used bibliometric analysis and topic modeling to explore the research trends and advancements in...
OBJECTIVES
The incidence of oropharyngeal cancer (OPC) is increasing. This study used bibliometric analysis and topic modeling to explore the research trends and advancements in this disease over the past 10 years, providing valuable insights to guide future investigations.
METHODS
7,355 English articles from 2013 to 2022 were retrieved from the Web of Science Core Collection for bibliometric analysis. Topic modeling was applied to 1,681 articles from high-impact journals, followed by an assessment of topic significance ranking (TSR). Medical Subject Headings (MeSH) terms were extracted using R and Python, followed by an analysis of the terms associated with each topic and on an annual basis. Additionally, genes were extracted and the number of genes appearing each year and the newly emerged genes were counted.
RESULTS
The bibliometric analysis suggested that the United States and several European countries hold pivotal positions in research. Current research is focused on refining treatments, staging and stratification. Topic modeling revealed 12 topics, emphasizing human papillomavirus (HPV) and side effect reduction. MeSH analysis revealed a growing emphasis on prognosis and quality of life. No new MeSH terms emerged after 2018, suggesting that the existing terms have covered most of the core concepts within the field of oropharyngeal cancers. Gene analysis identified TP53 and EGFR as the most extensively studied genes, with no novel genes discovered after 2019. However, CD69 and CXCL9 emerged as new genes of interest in 2019, reflecting recent research trends and directions.
CONCLUSION
HPV-positive oropharyngeal cancer research, particularly treatment de-escalation, has gained significant attention. However, there are still challenges in diagnosis and treatment that need to be addressed. In the future, more research will focus on this issue, indicating that this field still holds potential as a research hotspot.
PubMed: 38919494
DOI: 10.3389/fmicb.2024.1387679 -
Bone Reports Jun 2024Tumor-induced osteomalacia (TIO), is a rare acquired paraneoplastic syndrome characterized by defective bone mineralization, caused by the overproduction of fibroblast... (Review)
Review
INTRODUCTION
Tumor-induced osteomalacia (TIO), is a rare acquired paraneoplastic syndrome characterized by defective bone mineralization, caused by the overproduction of fibroblast growth factor 23 (FGF23) by a tumor.
MATERIAL AND METHODS
We conducted a systematic review to identify all case reports of TIO, focusing on those associated with mesenchymal tumors. We followed the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) consensus, and we included patients with a diagnosis of TIO and histological confirmation of phosphaturic mesenchymal tumors or resolution of the condition after treatment of the tumor. Bibliographical searches were carried out until December 2023 in the Cochrane Library, Medline and Embase, as well as congress abstracts online.
RESULTS
We identified 769 articles with 1979 cases reported. Most patients were adults, with a higher incidence on men. Disease duration before diagnosis is a mean of 4.8 years. Most tumors were histologically classified as PMT. Lower limbs were the predominant location. Hypophosphatemia was present in 99.8 % of patients. The FGF23 was elevated at diagnosis in 95.5 %. Resection of the tumor was the treatment of choice in most of patients. After resection, there was a clinical improvement in 97.6 % of cases, and serum phosphorus and FGF23 levels returned to normal ranges in 91.5 % and 81.4 % of the patients, respectively.
CONCLUSION
TIO is usually misdiagnosed with rheumatological or musculoskeletal disorders. The diagnosis should be suspected in patients with hypophosphatemic osteomalacia, and the measurement of serum FGF23 can be useful for diagnosis and management.
PubMed: 38774264
DOI: 10.1016/j.bonr.2024.101772 -
World Neurosurgery Mar 2024Phosphaturic Mesenchymal Tumors (PMTs) are rare mesenchymal neoplasms known for producing Tumor-induced Osteomalacia (TIO). TIO is an uncommon paraneoplastic syndrome... (Review)
Review
BACKGROUND
Phosphaturic Mesenchymal Tumors (PMTs) are rare mesenchymal neoplasms known for producing Tumor-induced Osteomalacia (TIO). TIO is an uncommon paraneoplastic syndrome characterized by radiographic evidence of inadequate bone mineralization and analytical abnormalites.
METHODS
We sought to present a case of TIO caused by skull base PMT with intracranial extension, manifesting with pain, progressive weakness, and multiple bone fractures. Furthermore, a systematic review was performed, following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines. A search was conducted in PubMed database with title/abstract keywords "Phosphaturic mesenchymal tumor" and "Osteomalacia." Search results were reviewed looking for intracranial or skull base tumors.
RESULTS
Our systematic review included 29 reported cases of intracranial PMT. In the reviewed cases there was a significative female predominance with 22 cases (75,86%). Osteomalacia was presented in 25 cases (86,20%). Bone fractures were present in 10 cases (34,48%). The most common site of involvement was the anterior cranial fossa in 14 cases (48,27%). Surgery was performed in 27 cases (93,10%) with previous tumor embolization in 4 cases (13,79%). Total recovery of the presenting symptoms in the first year was achieved in 21 cases (72,41%). Recurrence of the disease was described in 6 cases (25%).
CONCLUSIONS
Skull base PMTs with intracranial extension are extremely rare tumors. Most patients are middle-aged adults with a PMT predominantly located in anterior cranial fossa. Surgery is the current treatment of choice with optimal outcome at 1-year follow-up, although recurrence could be present in almost 25% of the cases.
PubMed: 38561034
DOI: 10.1016/j.wneu.2024.03.138 -
World Neurosurgery Apr 2024Spinal phosphaturic mesenchymal tumor (PMT) is a rare disorder but can be cured once the diagnosis is clear and a complete removal by surgery is performed. To the best... (Review)
Review
BACKGROUND
Spinal phosphaturic mesenchymal tumor (PMT) is a rare disorder but can be cured once the diagnosis is clear and a complete removal by surgery is performed. To the best of our knowledge, only 22 cases in the spine have been described, and we report a case with the largest number of spinal segments (T12-L5) affected among spine PMT cases.
METHODS
A comprehensive literature search was performed until May 23, 2023, following the Preferred Reporting Items for Systematic Reviews guidelines. Studies were chosen through relevant PubMed, Web of Science, and EMBASE searches to prioritize obtaining the largest studies. The Medical Subject Headings and Boolean operators employed for this search were ("PMT" or "TIO" or "Tumor-induced osteomalacia" or "phosphaturic mesenchymal tumor") and ("spine" or "spinal"). Two researchers (L.S.Z. and D.B.C) independently reviewed and evaluated the included articles. Any differing opinions were discussed until a consensus was reached. A total of 18 studies were included. A case report is also presented.
RESULTS
We report a case of spinal PMT. The full text of the relevant articles was construed. A total of 18 studies were reviewed and consolidated. These articles are roughly divided into the following 5 subcategories: 1) clinical features and baseline distribution, 2) laboratory and imaging findings, 3) pathological manifestations, and 4) surgical methods and treatment options.
CONCLUSIONS
Spinal PMT is very rare with a high rate of misdiagnosis and debilitating complications, so it is of significance to increase awareness of the disease among spine surgeons consulted by patients with spinal PMT. Ga-DOTATOC-PET/CT shows very high sensitivity to the spinal PMT but there is no way to exactly determine the location of the tumor. PMT has unique immunohistochemical characteristics and malignant PMT is rare. Once diagnosed, complete surgical excision is the recommended treatment. Burosumab is one of the available options, especially in cases that are recurrent and difficult to surgically resect.
Topics: Humans; Neoplasms, Connective Tissue; Positron Emission Tomography Computed Tomography; Mesenchymoma; Soft Tissue Neoplasms; Spine
PubMed: 38218444
DOI: 10.1016/j.wneu.2024.01.032 -
The Journal of Clinical Endocrinology... Feb 2024Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome usually caused by oversecretion of fibroblast growth factor 23 (FGF23) from a phosphaturic mesenchymal...
CONTEXT
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome usually caused by oversecretion of fibroblast growth factor 23 (FGF23) from a phosphaturic mesenchymal tumor (PMT). PMTs are usually benign neoplasms but some of them show malignant characteristics.
OBJECTIVE
The aim of this study was to compare the clinical characteristics of benign and malignant PMTs inducing TIO.
METHODS
On March 31, 2023, we performed a systematic review of individual patient data analysis in Medline, Google Scholar, Google book, and Cochrane Library using the terms "tumor induced osteomalacia," "oncogenic osteomalacia," "hypophosphatemia," with no language restrictions and according to Preferred Reporting Items for Systematic reviews and Meta-Analyses criteria.
RESULTS
Overall, we collected data from 837 patients with TIO in which the diagnosis of benign and malignant PMT was specified. Of them, 89 were affected by malignant PMT and 748 by benign PMT. Patients with malignant PMTs were younger and presented bone pain, functional impairment, and bone deformities more frequently. Malignant PMTs showed higher values of intact FGF23 and a higher mortality rate.
CONCLUSION
The study results identify the clinical characteristics of patients with malignant TIO, permitting the early identification of patients with PMT at increased risk of malignancy. This may significantly improve the diagnostic approach to disease. Further experimental studies are mandatory to clarify the role of FGF23 in the pathogenesis of malignancy in PMTs.
Topics: Humans; Osteomalacia; Neoplasms, Connective Tissue; Fibroblast Growth Factors; Paraneoplastic Syndromes; Soft Tissue Neoplasms; Mesenchymoma
PubMed: 38006315
DOI: 10.1210/clinem/dgad690 -
Calcified Tissue International Feb 2024Hereditary hypophosphatemic rickets with hypercalciuria (HHRH) is a rare disorder of phosphate homeostasis. We describe a single-center experience of genetically proven...
Hereditary Hypophosphatemic Rickets with Hypercalciuria Presenting with Enthesopathy, Renal Cysts, and High Serum c-Terminal FGF23: Single-Center Experience and Systematic Review.
Hereditary hypophosphatemic rickets with hypercalciuria (HHRH) is a rare disorder of phosphate homeostasis. We describe a single-center experience of genetically proven HHRH families and perform systematic review phenotype-genotype correlation in reported biallelic probands and their monoallelic relatives. Detailed clinical, biochemical, radiological, and genetic data were retrieved from our center and a systematic review of Pub-Med and Embase databases for patients and relatives who were genetically proven. Total of nine subjects (probands:5) carrying biallelic SLC34A3 mutations (novel:2) from our center had a spectrum from rickets/osteomalacia to normal BMD, with hypophosphatemia and hypercalciuria in all. We describe the first case of genetically proven HHRH with enthesopathy. Elevated FGF23 in another patient with hypophosphatemia, iron deficiency anemia, and noncirrhotic periportal fibrosis led to initial misdiagnosis as tumoral osteomalacia. On systematic review of 58 probands (with biallelic SLC34A3 mutations; 35 males), early-onset HHRH and renal calcification were present in ~ 70% and late-onset HHRH in 10%. c.575C > T p.(Ser192Leu) variant occurred in 53% of probands without skeletal involvement. Among 110 relatives harboring monoallelic SLC34A3 mutation at median age 38 years, renal calcification, hypophosphatemia, high 1,25(OH)D, and hypercalciuria were observed in ~30%, 22.3%, 40%, and 38.8%, respectively. Renal calcifications correlated with age but were similar across truncating and non-truncating variants. Although most relatives were asymptomatic for bone involvement, 6/12(50%) had low bone mineral density. We describe the first monocentric HHRH case series from India with varied phenotypes. In a systematic review, frequent renal calcifications and low BMD in relatives with monoallelic variants (HHRH trait) merit identification.
Topics: Male; Humans; Adult; Familial Hypophosphatemic Rickets; Hypercalciuria; Osteomalacia; Enthesopathy; Nephrocalcinosis; Hypophosphatemia; Kidney Diseases, Cystic
PubMed: 37981601
DOI: 10.1007/s00223-023-01156-2 -
The Journal of Clinical Endocrinology... Oct 2023Tumor-induced osteomalacia (TIO) due to fibroblast growth factor 23 (FGF23) overexpression is becoming recognized in patients with malignancy. The condition may be... (Meta-Analysis)
Meta-Analysis
CONTEXT
Tumor-induced osteomalacia (TIO) due to fibroblast growth factor 23 (FGF23) overexpression is becoming recognized in patients with malignancy. The condition may be underdiagnosed, with a scarce medical literature.
OBJECTIVE
To perform a meta-analysis of case reports to allow a better understanding of malignant TIO and its clinical implications.
METHODS
Full texts were selected according to strict inclusion criteria. All case reports were included where patients had hypophosphatemia, malignant TIO, and FGF23 blood levels. Thirty-two of 275 eligible studies (n = 34 patients) met inclusion criteria. A list of desired data was extracted and graded for methodological quality.
RESULTS
Prostate adenocarcinoma (n = 9) were the most tumors reported. Twenty-five of 34 patients had a metastatic disease and a poor clinical outcome was reported for 15 of 28 patients. The median levels of blood phosphate and C-terminal FGF23 (cFGF23) were 0.40 mmol/L and 788.5 RU/mL, respectively. For most of patients, blood PTH was elevated or within range, and calcitriol levels were inappropriately low or normal. Alkaline phosphatase concentrations were increased for 20 of 22 patients. The cFGF23 values were significantly higher for patients with a poor clinical outcome when compared to other patients (1685 vs 357.5 RU/mL). In case of prostate cancer, cFGF23 levels were significantly lower (429.4 RU/mL) than for other malignancies (1007.5 RU/mL).
CONCLUSION
We report for the first time a detailed description of the clinical and biological characteristics of malignant TIO. In this context, FGF23 blood measurement would be of value for the diagnostic workup, prognostication, and follow-up of patients.
Topics: Humans; Male; Calcitriol; Fibroblast Growth Factors; Hypophosphatemia; Osteomalacia; Paraneoplastic Syndromes; Case Reports as Topic
PubMed: 37235783
DOI: 10.1210/clinem/dgad297 -
Clinical Physiology and Functional... Sep 2023Bone scintigraphy (BS) is an important tool for detecting bone metastasis. BS with diffuse increased skeletal radioisotope uptake with absent or faint urinary tract and... (Review)
Review
INTRODUCTION
Bone scintigraphy (BS) is an important tool for detecting bone metastasis. BS with diffuse increased skeletal radioisotope uptake with absent or faint urinary tract and soft tissue activity is defined as a superscan. In this review, we investigate the different etiologies causing superscan and the reported frequency of superscan among different disease entities.
MATERIALS AND METHODS
The search terms were 'bone' AND 'superscan' OR 'superscan' in the PubMed database from 1980 to November 2020. Eligibility criteria included the following: Peer-reviewed studies containing original data using 99mTc-phosphate-analogue BS reporting a superscan pattern. Unretrievable papers, imaging modalities other than BS or with insufficient information to assess the aetiology were excluded. The abstracts of every paper and full texts of potentially eligible papers were assessed independently by three observers.
RESULTS
Sixty-seven papers were included (48 case reports and 19 cohort studies). Studies conducted in patients with osteomalacia or skeletal fluorosis revealed superscan in all patients. Other benign causes of superscan were hyperparathyroidism and kidney disease. Among papers with malignant cause, prostate cancer was the most common cause, followed by gastric cancer. The frequency of superscans ranged from 1.3% in a cohort of mixed cancer types up to 2.6% in patients with gastric cancer and up to 23% in a cohort of prostate cancer patients.
CONCLUSION
Superscan is most frequently seen in prostate cancer, but numerous other cancers and metabolic bone diseases can cause superscan, which should be kept in mind when encountering an unexpected superscan on BS.
Topics: Male; Humans; Radionuclide Imaging; Stomach Neoplasms; Bone and Bones; Bone Neoplasms; Prostatic Neoplasms
PubMed: 37070619
DOI: 10.1111/cpf.12821 -
Frontiers in Nutrition 2023Vitamin D deficiency causes the bone hypomineralization disorder osteomalacia in humans and is associated with many non-skeletal disorders. We aim to estimate the global...
BACKGROUND
Vitamin D deficiency causes the bone hypomineralization disorder osteomalacia in humans and is associated with many non-skeletal disorders. We aim to estimate the global and regional prevalence of vitamin D deficiency in people aged 1 year or older from 2000 to 2022.
METHODS
We systematically searched Web of Science, PubMed (MEDLINE), Embase, Scopus, and Google databases on December 31, 2021, and updated them on August 20, 2022, without language and time restrictions. Meanwhile, we identified references of relevant system reviews and eligible articles and included the latest and unpublished data from the National Health and Nutrition Examination Survey (NHANES, 2015-2016 and 2017-2018) database. The studies investigating the prevalence of vitamin D deficiency in population-based studies were included. A standardized data extraction form was used to collect information from eligible studies. We used a random-effects meta-analysis to estimate the global and regional prevalence of vitamin D deficiency. We stratified meta-analyses by latitude, season, six WHO regions, the World Bank income groups, gender, and age groups. This study was registered with PROSPERO (CRD42021292586).
FINDINGS
Out of 67,340 records searched, 308 studies with 7,947,359 participants from 81 countries were eligible for this study, 202 (7,634,261 participants), 284 (1,475,339 participants), and 165 (561,978 participants) studies for the prevalence of serum 25(OH)D <30, <50, and <75 nmol/L, respectively. We found that globally, 15.7% (95% CrI 13.7-17.8), 47.9% (95% CrI 44.9-50.9), and 76·6% (95% CrI 74.0-79.1) of participants had serum 25-hydroxyvitamin D levels less than 30, 50, and 75 nmol/l, respectively; the prevalence slightly decreased from 2000-2010 to 2011-2022, but it was still at a high level; people living in high latitude areas had a higher prevalence; the prevalence in winter-spring was 1.7 (95% CrI 1.4-2.0) times that in summer-autumn; the Eastern Mediterranean region and Lower-middle-income countries had a higher prevalence; females were vulnerable to vitamin D deficiency; gender, sampling frame, detection assays, sampling region, time of data collection, season, and other factors contributed to heterogeneity between the included studies.
INTERPRETATION
Globally, vitamin D deficiency remained prevalent from 2000 to 2022. The high prevalence of vitamin D deficiency would increase the global burden of disease. Therefore, governments, policymakers, health workers, and individuals should attach importance to the high prevalence of vitamin D deficiency and take its prevention as a public health priority.
SYSTEMATIC REVIEW REGISTRATION
https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42021292586, PROSPERO CRD42021292586.
PubMed: 37006940
DOI: 10.3389/fnut.2023.1070808 -
The Journal of Clinical Endocrinology... Jul 2022Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome, usually caused by small, benign, and slow-growing phosphaturic mesenchymal tumors. Clinically, TIO is...
CONTEXT
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome, usually caused by small, benign, and slow-growing phosphaturic mesenchymal tumors. Clinically, TIO is characterized by renal phosphate leak, causing hypophosphatemia and osteomalacia. This review was performed to assess the clinical characteristics of TIO patients described worldwide so far.
EVIDENCE ACQUISITION
On June 26, 2021, a systematic search was performed in Medline, Google Scholar, Google book, and Cochrane Library using the terms: "tumor induced osteomalacia," "oncogenic osteomalacia," "hypophosphatemia." There were no language restrictions. This review was performed according to Preferred Reporting Items for Systematic reviews and Meta-Analyses criteria.
EVIDENCE RESULTS
Overall, 1725 TIO cases were collected. TIO was more frequent in adult men, who showed a higher incidence of fractures compared with TIO women. The TIO-causing neoplasms were identified in 1493 patients. The somatostatin receptor-based imaging modalities have the highest sensitivity for the identification of TIO-causing neoplasms. TIO-causing neoplasms were equally located in bone and soft tissues; the latter showed a higher prevalence of fractures and deformities. The surgery is the preferred TIO definitive treatment (successful in > 90% of patients). Promising nonsurgical therapies are treatments with burosumab in TIO patients with elevated fibroblast growth factor-23 levels, and with radiolabeled somatostatin analogs in patients with TIO-causing neoplasm identified by somatostatin receptor-based imaging techniques.
CONCLUSION
TIO occurs preferentially in adult men. The TIO clinical expressiveness is more severe in men as well as in patients with TIO-causing neoplasms located in soft tissues. Treatments with burosumab and with radiolabeled somatostatin analogs are the most promising nonsurgical therapies.
Topics: Adult; Data Analysis; Female; Fibroblast Growth Factors; Humans; Hypophosphatemia; Male; Neoplasms, Connective Tissue; Osteomalacia; Paraneoplastic Syndromes; Receptors, Somatostatin; Somatostatin
PubMed: 35468192
DOI: 10.1210/clinem/dgac253