Tumor-induced osteomalacia: successful treatment by radio-guided tumor surgery.
Identifieur interne : 001B85 ( Main/Exploration ); précédent : 001B84; suivant : 001B86Tumor-induced osteomalacia: successful treatment by radio-guided tumor surgery.
Auteurs : RBID : pubmed:19125132English descriptors
- KwdEn :
- MESH :
- chemical , diagnostic use : Indium Radioisotopes.
- complications : Hemangiopericytoma, Hypophosphatemia.
- etiology : Hypophosphatemia, Osteomalacia.
- radionuclide imaging : Hemangiopericytoma.
- surgery : Hemangiopericytoma.
- Adult, Humans, Male.
Abstract
Tumor-induced osteomalacia is a rare syndrome characterized by urinary phosphate loss with hypophosphatemic osteomalacia. The proposed pathogenetic mechanism is paraneoplastic secretion of phosphaturic factors (so-called phosphatonins).We describe a 34-year-old male patient who presented with severe pain of the spine and ribs for at least 2 years. Bone scintigraphy using Technetium hydroxymethylene diphosphonate (Tc HDP) showed multiple lesions suggesting metastatic disease. Bone biopsy however revealed osteomalacia. The patient had subnormal plasma phosphate levels (0.42 mmol/L; normal range, 0.87-1.45) and markedly increased phosphate clearance (82.8 mL/min; normal range, 5.4-16.2). The patient was treated with phosphate supplementation (up to 5 g daily) along with calcium (1000 mg daily) and calcitriol (1.5 microg daily). Although this therapy did not correct hypophosphatemia, it resulted in complete relief of pain within several months. (111)In pentetreotide scintigraphy showed a tiny lesion of 1-cm diameter, which could be localized to the left femoral neck in close vicinity to the greater trochanter by MRI and image fusion analysis. This lesion had not been visualized by Tc-99m HDP bone scintigraphy. Intraoperatively, use of a hand-held gamma probe after administration of (111)Indium pentetreotide ((111)In pentetreotide) clearly identified the tumor, which was completely removed and was shown to be a hemangiopericytoma. After removal of the tumor, phosphate metabolism normalized within 1 week without requirement of phosphate supplementation. Hypophosphatemic osteomalacia, although rare, raises an important differential diagnosis. An underlying tumor may be detected only by (111)In pentetreotide scintigraphy. Preoperative labeling with (111)In pentetreotide is a useful tool in detecting these tumors during surgery.This 34 year old man with osteomalacia had a small causative hemangiopericytoma detected in the indium pentetreotide scintography.
DOI: 10.1097/RHU.0b013e3181960483
PubMed: 19125132
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The proposed pathogenetic mechanism is paraneoplastic secretion of phosphaturic factors (so-called phosphatonins).We describe a 34-year-old male patient who presented with severe pain of the spine and ribs for at least 2 years. Bone scintigraphy using Technetium hydroxymethylene diphosphonate (Tc HDP) showed multiple lesions suggesting metastatic disease. Bone biopsy however revealed osteomalacia. The patient had subnormal plasma phosphate levels (0.42 mmol/L; normal range, 0.87-1.45) and markedly increased phosphate clearance (82.8 mL/min; normal range, 5.4-16.2). The patient was treated with phosphate supplementation (up to 5 g daily) along with calcium (1000 mg daily) and calcitriol (1.5 microg daily). Although this therapy did not correct hypophosphatemia, it resulted in complete relief of pain within several months. (111)In pentetreotide scintigraphy showed a tiny lesion of 1-cm diameter, which could be localized to the left femoral neck in close vicinity to the greater trochanter by MRI and image fusion analysis. This lesion had not been visualized by Tc-99m HDP bone scintigraphy. Intraoperatively, use of a hand-held gamma probe after administration of (111)Indium pentetreotide ((111)In pentetreotide) clearly identified the tumor, which was completely removed and was shown to be a hemangiopericytoma. After removal of the tumor, phosphate metabolism normalized within 1 week without requirement of phosphate supplementation. Hypophosphatemic osteomalacia, although rare, raises an important differential diagnosis. An underlying tumor may be detected only by (111)In pentetreotide scintigraphy. Preoperative labeling with (111)In pentetreotide is a useful tool in detecting these tumors during surgery.This 34 year old man with osteomalacia had a small causative hemangiopericytoma detected in the indium pentetreotide scintography.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">19125132</PMID><DateCreated><Year>2009</Year><Month>01</Month><Day>12</Day></DateCreated><DateCompleted><Year>2009</Year><Month>05</Month><Day>06</Day></DateCompleted><Article PubModel="Print"><Journal><ISSN IssnType="Electronic">1536-7355</ISSN><JournalIssue CitedMedium="Internet"><Volume>15</Volume><Issue>1</Issue><PubDate><Year>2009</Year><Month>Feb</Month></PubDate></JournalIssue><Title>Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases</Title><ISOAbbreviation>J Clin Rheumatol</ISOAbbreviation></Journal><ArticleTitle>Tumor-induced osteomalacia: successful treatment by radio-guided tumor surgery.</ArticleTitle><Pagination><MedlinePgn>31-4</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1097/RHU.0b013e3181960483</ELocationID><Abstract><AbstractText>Tumor-induced osteomalacia is a rare syndrome characterized by urinary phosphate loss with hypophosphatemic osteomalacia. The proposed pathogenetic mechanism is paraneoplastic secretion of phosphaturic factors (so-called phosphatonins).We describe a 34-year-old male patient who presented with severe pain of the spine and ribs for at least 2 years. Bone scintigraphy using Technetium hydroxymethylene diphosphonate (Tc HDP) showed multiple lesions suggesting metastatic disease. Bone biopsy however revealed osteomalacia. The patient had subnormal plasma phosphate levels (0.42 mmol/L; normal range, 0.87-1.45) and markedly increased phosphate clearance (82.8 mL/min; normal range, 5.4-16.2). The patient was treated with phosphate supplementation (up to 5 g daily) along with calcium (1000 mg daily) and calcitriol (1.5 microg daily). Although this therapy did not correct hypophosphatemia, it resulted in complete relief of pain within several months. (111)In pentetreotide scintigraphy showed a tiny lesion of 1-cm diameter, which could be localized to the left femoral neck in close vicinity to the greater trochanter by MRI and image fusion analysis. This lesion had not been visualized by Tc-99m HDP bone scintigraphy. Intraoperatively, use of a hand-held gamma probe after administration of (111)Indium pentetreotide ((111)In pentetreotide) clearly identified the tumor, which was completely removed and was shown to be a hemangiopericytoma. After removal of the tumor, phosphate metabolism normalized within 1 week without requirement of phosphate supplementation. Hypophosphatemic osteomalacia, although rare, raises an important differential diagnosis. An underlying tumor may be detected only by (111)In pentetreotide scintigraphy. Preoperative labeling with (111)In pentetreotide is a useful tool in detecting these tumors during surgery.This 34 year old man with osteomalacia had a small causative hemangiopericytoma detected in the indium pentetreotide scintography.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Harbeck</LastName><ForeName>Birgit</ForeName><Initials>B</Initials><Affiliation>Department of Medicine, University of Kiel, Kiel, Germany. bharbeck@1med.uni-kiel.de</Affiliation></Author><Author ValidYN="Y"><LastName>Schöcklmann</LastName><ForeName>Harald</ForeName><Initials>H</Initials></Author><Author ValidYN="Y"><LastName>Seekamp</LastName><ForeName>Andreas</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>Czech</LastName><ForeName>Norbert</ForeName><Initials>N</Initials></Author><Author ValidYN="Y"><LastName>Mönig</LastName><ForeName>Heiner</ForeName><Initials>H</Initials></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType>Case Reports</PublicationType><PublicationType>Journal Article</PublicationType></PublicationTypeList></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>J Clin Rheumatol</MedlineTA><NlmUniqueID>9518034</NlmUniqueID><ISSNLinking>1076-1608</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance>Indium Radioisotopes</NameOfSubstance></Chemical></ChemicalList><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Hemangiopericytoma</DescriptorName><QualifierName MajorTopicYN="Y">complications</QualifierName><QualifierName MajorTopicYN="Y">radionuclide imaging</QualifierName><QualifierName MajorTopicYN="N">surgery</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Hypophosphatemia</DescriptorName><QualifierName MajorTopicYN="N">complications</QualifierName><QualifierName MajorTopicYN="Y">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Indium Radioisotopes</DescriptorName><QualifierName MajorTopicYN="Y">diagnostic use</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N">Osteomalacia</DescriptorName><QualifierName MajorTopicYN="Y">etiology</QualifierName></MeshHeading></MeshHeadingList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="entrez"><Year>2009</Year><Month>1</Month><Day>7</Day><Hour>9</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2009</Year><Month>1</Month><Day>7</Day><Hour>9</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2009</Year><Month>5</Month><Day>7</Day><Hour>9</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="doi">10.1097/RHU.0b013e3181960483</ArticleId><ArticleId IdType="pubmed">19125132</ArticleId></ArticleIdList></PubmedData></pubmed></record>Pour manipuler ce document sous Unix (Dilib)
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