Non-endemic skeletal fluorosis: Causes and associated secondary hyperparathyroidism (case report and literature review)
Skeletal fluorosis (SF) is endemic primarily in regions with fluoride (F)-contaminated well water, but can reflect other types of chronic F exposure. Calcium (Ca) and vitamin D (D) deficiency can exacerbate SF. A 51-year-old man with years of musculoskeletal pain and opiate use was hypocalcemic with...
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| description | Skeletal fluorosis (SF) is endemic primarily in regions with fluoride (F)-contaminated well water, but can reflect other types of chronic F exposure. Calcium (Ca) and vitamin D (D) deficiency can exacerbate SF. A 51-year-old man with years of musculoskeletal pain and opiate use was hypocalcemic with secondary hyperparathyroidism upon manifesting recurrent long bone fractures. He smoked cigarettes, drank large amounts of cola beverage, and consumed little dietary Ca. Then, after 5 months of Ca and D3 supplementation, serum 25(OH)D was 21 ng/mL (Nl, 30–100), corrected serum Ca had normalized from 7.8 to 9.4 mg/dL (Nl, 8.5–10.1), alkaline phosphatase (ALP) had decreased from 1080 to 539 U/L (Nl, 46–116), yet parathyroid hormone (PTH) had increased from 133 to 327 pg/mL (Nl, 8.7–77.1). Radiographs revealed generalized osteosclerosis and a cystic lesion in a proximal femur. DXA BMD Z-scores were +7.4 and +0.4 at the lumbar spine and “1/3” radius, respectively. Bone scintigraphy showed increased uptake in two ribs, periarticular areas, and proximal left femur at the site of a subsequent atraumatic fracture. Elevated serum collagen type I C-telopeptide 2513 pg/mL (Nl, 87–345) and osteocalcin >300 ng/mL (Nl, 9–38) indicated rapid bone turnover. Negative studies included hepatitis C Ab, prostate-specific antigen, serum and urine electrophoresis, and Ion Torrent mutation analysis for dense or high-turnover skeletal diseases. After discovering markedly elevated F concentrations in his plasma [4.84 mg/L (Nl, 0.02–0.08)] and spot urine [42.6 mg/L (Nl, 0.2–3.2)], a two-year history emerged of “huffing” computer cleaner containing difluoroethane. Non-decalcified histology of a subsequent right femur fracture showed increased osteoblasts and osteoclasts and excessive osteoid. A 24-hour urine collection contained 27 mg/L F (Nl, 0.2–3.2) and |
| doi_str_mv | 10.1016/j.bone.2021.115839 |
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•Fluorocarbon “huffing” is an under-appreciated cause of skeletal fluorosis (SF).•We present a SF case with hyperparathyroidism, osteosclerosis, and osteomalacia.•SF may go undetected due to variation in symptoms, radiology, and biochemistry.•Dietary calcium, prior bone health, and skeletal F exposure influence SF features.•SF is common in endemic areas. We review unusual, non-endemic causes of SF.</description><identifier>ISSN: 8756-3282</identifier><identifier>EISSN: 1873-2763</identifier><identifier>DOI: 10.1016/j.bone.2021.115839</identifier><identifier>PMID: 33418099</identifier><language>eng</language><publisher>NEW YORK: Elsevier Inc</publisher><subject>Atypical femoral fracture ; Bone Density ; Bone Diseases - chemically induced ; Bone Diseases - diagnostic imaging ; Bone pain ; Bone scan ; Difluoroethane ; DXA ; Elevated bone mass ; Endocrinology & Metabolism ; Fluoride ; Fluorocarbon ; Fluorosis ; Hip fracture ; Huffing ; Humans ; Hyperparathyroidism ; Hyperparathyroidism, Secondary - diagnostic imaging ; Hypertrophic callus ; Hypocalcemia ; Life Sciences & Biomedicine ; Male ; Middle Aged ; Osteolysis ; Osteomalacia ; Osteoporosis ; Osteosclerosis ; Parathyroid Hormone ; Periostitis ; Science & Technology ; Spine ; Tea consumption</subject><ispartof>Bone (New York, N.Y.), 2021-04, Vol.145, p.115839-115839, Article 115839</ispartof><rights>2021 Elsevier Inc.</rights><rights>Copyright © 2021 Elsevier Inc. All rights reserved.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>true</woscitedreferencessubscribed><woscitedreferencescount>19</woscitedreferencescount><woscitedreferencesoriginalsourcerecordid>wos000623120700006</woscitedreferencesoriginalsourcerecordid><citedby>FETCH-LOGICAL-c455t-117cf80d5b9d37c004b7090040f8af1fea1c2ad020eb609c4aaaf8cc0193d71d3</citedby><cites>FETCH-LOGICAL-c455t-117cf80d5b9d37c004b7090040f8af1fea1c2ad020eb609c4aaaf8cc0193d71d3</cites><orcidid>0000-0003-4275-8920</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://dx.doi.org/10.1016/j.bone.2021.115839$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>230,315,781,785,886,3551,27929,27930,39263,46000</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33418099$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Cook, Fiona J.</creatorcontrib><creatorcontrib>Seagrove-Guffey, Maighan</creatorcontrib><creatorcontrib>Mumm, Steven</creatorcontrib><creatorcontrib>Veis, Deborah J.</creatorcontrib><creatorcontrib>McAlister, William H.</creatorcontrib><creatorcontrib>Bijanki, Vinieth N.</creatorcontrib><creatorcontrib>Wenkert, Deborah</creatorcontrib><creatorcontrib>Whyte, Michael P.</creatorcontrib><title>Non-endemic skeletal fluorosis: Causes and associated secondary hyperparathyroidism (case report and literature review)</title><title>Bone (New York, N.Y.)</title><addtitle>BONE</addtitle><addtitle>Bone</addtitle><description>Skeletal fluorosis (SF) is endemic primarily in regions with fluoride (F)-contaminated well water, but can reflect other types of chronic F exposure. Calcium (Ca) and vitamin D (D) deficiency can exacerbate SF. A 51-year-old man with years of musculoskeletal pain and opiate use was hypocalcemic with secondary hyperparathyroidism upon manifesting recurrent long bone fractures. He smoked cigarettes, drank large amounts of cola beverage, and consumed little dietary Ca. Then, after 5 months of Ca and D3 supplementation, serum 25(OH)D was 21 ng/mL (Nl, 30–100), corrected serum Ca had normalized from 7.8 to 9.4 mg/dL (Nl, 8.5–10.1), alkaline phosphatase (ALP) had decreased from 1080 to 539 U/L (Nl, 46–116), yet parathyroid hormone (PTH) had increased from 133 to 327 pg/mL (Nl, 8.7–77.1). Radiographs revealed generalized osteosclerosis and a cystic lesion in a proximal femur. DXA BMD Z-scores were +7.4 and +0.4 at the lumbar spine and “1/3” radius, respectively. Bone scintigraphy showed increased uptake in two ribs, periarticular areas, and proximal left femur at the site of a subsequent atraumatic fracture. Elevated serum collagen type I C-telopeptide 2513 pg/mL (Nl, 87–345) and osteocalcin >300 ng/mL (Nl, 9–38) indicated rapid bone turnover. Negative studies included hepatitis C Ab, prostate-specific antigen, serum and urine electrophoresis, and Ion Torrent mutation analysis for dense or high-turnover skeletal diseases. After discovering markedly elevated F concentrations in his plasma [4.84 mg/L (Nl, 0.02–0.08)] and spot urine [42.6 mg/L (Nl, 0.2–3.2)], a two-year history emerged of “huffing” computer cleaner containing difluoroethane. Non-decalcified histology of a subsequent right femur fracture showed increased osteoblasts and osteoclasts and excessive osteoid. A 24-hour urine collection contained 27 mg/L F (Nl, 0.2–3.2) and <2 mg/dL Ca. Then, 19 months after “huffing” cessation and improved Ca and D3 intake, yet with persisting bone pain, serum PTH was normal (52 pg/mL) and serum ALP and urine F had decreased to 248 U/L and 3.3 mg/L, respectively. Our experience combined with 15 publications in PubMed concerning unusual causes of non-endemic SF where the F source became known (19 cases in all) revealed: 11 instances from high consumption of black tea and/or F-containing toothpaste, 1 due to geophagia of F-rich soil, and 7 due to “recreational” inhalation of F-containing vapors. Circulating PTH measured in 14 was substantially elevated in 2 (including ours) and mildly increased in 2. The severity of SF in the cases reviewed seemed to reflect cumulative F exposure, renal function, and Ca and D status. Several factors appeared to influence our patient's skeletal disease: i) direct anabolic effects of toxic amounts of F on his skeleton, ii) secondary hyperparathyroidism from degradation-resistant fluorapatite bone crystals and low dietary Ca, and iii) impaired mineralization of excessive osteoid due to hypocalcemia.
•Fluorocarbon “huffing” is an under-appreciated cause of skeletal fluorosis (SF).•We present a SF case with hyperparathyroidism, osteosclerosis, and osteomalacia.•SF may go undetected due to variation in symptoms, radiology, and biochemistry.•Dietary calcium, prior bone health, and skeletal F exposure influence SF features.•SF is common in endemic areas. We review unusual, non-endemic causes of SF.</description><subject>Atypical femoral fracture</subject><subject>Bone Density</subject><subject>Bone Diseases - chemically induced</subject><subject>Bone Diseases - diagnostic imaging</subject><subject>Bone pain</subject><subject>Bone scan</subject><subject>Difluoroethane</subject><subject>DXA</subject><subject>Elevated bone mass</subject><subject>Endocrinology & Metabolism</subject><subject>Fluoride</subject><subject>Fluorocarbon</subject><subject>Fluorosis</subject><subject>Hip fracture</subject><subject>Huffing</subject><subject>Humans</subject><subject>Hyperparathyroidism</subject><subject>Hyperparathyroidism, Secondary - diagnostic imaging</subject><subject>Hypertrophic callus</subject><subject>Hypocalcemia</subject><subject>Life Sciences & Biomedicine</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Osteolysis</subject><subject>Osteomalacia</subject><subject>Osteoporosis</subject><subject>Osteosclerosis</subject><subject>Parathyroid Hormone</subject><subject>Periostitis</subject><subject>Science & Technology</subject><subject>Spine</subject><subject>Tea consumption</subject><issn>8756-3282</issn><issn>1873-2763</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>HGBXW</sourceid><sourceid>EIF</sourceid><recordid>eNqNkUuLFDEUhYMoTs_oH3AhWSpDtXnUIyWDIIUvGHSj65BKbtlpq5MiSU3T_96UNTa6EVc3JOc7Sc5B6BklW0po_Wq_7b2DLSOMbimtBG8foA0VDS9YU_OHaCOaqi44E-wCXca4J4TwtqGP0QXnJRWkbTfo-Nm7ApyBg9U4_oARkhrxMM4--Gjja9ypOULEyhmsYvTaqgQGR9DeGRVOeHeaIEwqqLQ7BW-NjQf8QqsIOMDkQ_pFjjZBVsxh2b2zcHz5BD0a1Bjh6f28Qt_ev_vafSxuv3z41L29LXRZVamgtNGDIKbqW8MbTUjZN6TNgwxCDXQARTVThjACfU1aXSqlBqE1oS03DTX8Cr1Zfae5P4DR4FJQo5yCPeTXS6-s_PvE2Z387u-koCXjnGYDthroHEgMMJxZSuRSg9zLpQa51CDXGjL0_M9bz8jv3LPgehUcofdD1BachrMsF1UzThlpyLLMavH_6s4mlax3nZ9dyujNikIOOScf5D1ubACdpPH2Xx_5CQWfvmY</recordid><startdate>20210401</startdate><enddate>20210401</enddate><creator>Cook, Fiona J.</creator><creator>Seagrove-Guffey, Maighan</creator><creator>Mumm, Steven</creator><creator>Veis, Deborah J.</creator><creator>McAlister, William H.</creator><creator>Bijanki, Vinieth N.</creator><creator>Wenkert, Deborah</creator><creator>Whyte, Michael P.</creator><general>Elsevier Inc</general><general>Elsevier</general><scope>BLEPL</scope><scope>DTL</scope><scope>HGBXW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0003-4275-8920</orcidid></search><sort><creationdate>20210401</creationdate><title>Non-endemic skeletal fluorosis: Causes and associated secondary hyperparathyroidism (case report and literature review)</title><author>Cook, Fiona J. ; Seagrove-Guffey, Maighan ; Mumm, Steven ; Veis, Deborah J. ; McAlister, William H. ; Bijanki, Vinieth N. ; Wenkert, Deborah ; Whyte, Michael P.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c455t-117cf80d5b9d37c004b7090040f8af1fea1c2ad020eb609c4aaaf8cc0193d71d3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Atypical femoral fracture</topic><topic>Bone Density</topic><topic>Bone Diseases - chemically induced</topic><topic>Bone Diseases - diagnostic imaging</topic><topic>Bone pain</topic><topic>Bone scan</topic><topic>Difluoroethane</topic><topic>DXA</topic><topic>Elevated bone mass</topic><topic>Endocrinology & Metabolism</topic><topic>Fluoride</topic><topic>Fluorocarbon</topic><topic>Fluorosis</topic><topic>Hip fracture</topic><topic>Huffing</topic><topic>Humans</topic><topic>Hyperparathyroidism</topic><topic>Hyperparathyroidism, Secondary - diagnostic imaging</topic><topic>Hypertrophic callus</topic><topic>Hypocalcemia</topic><topic>Life Sciences & Biomedicine</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Osteolysis</topic><topic>Osteomalacia</topic><topic>Osteoporosis</topic><topic>Osteosclerosis</topic><topic>Parathyroid Hormone</topic><topic>Periostitis</topic><topic>Science & Technology</topic><topic>Spine</topic><topic>Tea consumption</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Cook, Fiona J.</creatorcontrib><creatorcontrib>Seagrove-Guffey, Maighan</creatorcontrib><creatorcontrib>Mumm, Steven</creatorcontrib><creatorcontrib>Veis, Deborah J.</creatorcontrib><creatorcontrib>McAlister, William H.</creatorcontrib><creatorcontrib>Bijanki, Vinieth N.</creatorcontrib><creatorcontrib>Wenkert, Deborah</creatorcontrib><creatorcontrib>Whyte, Michael P.</creatorcontrib><collection>Web of Science Core Collection</collection><collection>Science Citation Index Expanded</collection><collection>Web of Science - Science Citation Index Expanded - 2021</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Bone (New York, N.Y.)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Cook, Fiona J.</au><au>Seagrove-Guffey, Maighan</au><au>Mumm, Steven</au><au>Veis, Deborah J.</au><au>McAlister, William H.</au><au>Bijanki, Vinieth N.</au><au>Wenkert, Deborah</au><au>Whyte, Michael P.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Non-endemic skeletal fluorosis: Causes and associated secondary hyperparathyroidism (case report and literature review)</atitle><jtitle>Bone (New York, N.Y.)</jtitle><stitle>BONE</stitle><addtitle>Bone</addtitle><date>2021-04-01</date><risdate>2021</risdate><volume>145</volume><spage>115839</spage><epage>115839</epage><pages>115839-115839</pages><artnum>115839</artnum><issn>8756-3282</issn><eissn>1873-2763</eissn><abstract>Skeletal fluorosis (SF) is endemic primarily in regions with fluoride (F)-contaminated well water, but can reflect other types of chronic F exposure. Calcium (Ca) and vitamin D (D) deficiency can exacerbate SF. A 51-year-old man with years of musculoskeletal pain and opiate use was hypocalcemic with secondary hyperparathyroidism upon manifesting recurrent long bone fractures. He smoked cigarettes, drank large amounts of cola beverage, and consumed little dietary Ca. Then, after 5 months of Ca and D3 supplementation, serum 25(OH)D was 21 ng/mL (Nl, 30–100), corrected serum Ca had normalized from 7.8 to 9.4 mg/dL (Nl, 8.5–10.1), alkaline phosphatase (ALP) had decreased from 1080 to 539 U/L (Nl, 46–116), yet parathyroid hormone (PTH) had increased from 133 to 327 pg/mL (Nl, 8.7–77.1). Radiographs revealed generalized osteosclerosis and a cystic lesion in a proximal femur. DXA BMD Z-scores were +7.4 and +0.4 at the lumbar spine and “1/3” radius, respectively. Bone scintigraphy showed increased uptake in two ribs, periarticular areas, and proximal left femur at the site of a subsequent atraumatic fracture. Elevated serum collagen type I C-telopeptide 2513 pg/mL (Nl, 87–345) and osteocalcin >300 ng/mL (Nl, 9–38) indicated rapid bone turnover. Negative studies included hepatitis C Ab, prostate-specific antigen, serum and urine electrophoresis, and Ion Torrent mutation analysis for dense or high-turnover skeletal diseases. After discovering markedly elevated F concentrations in his plasma [4.84 mg/L (Nl, 0.02–0.08)] and spot urine [42.6 mg/L (Nl, 0.2–3.2)], a two-year history emerged of “huffing” computer cleaner containing difluoroethane. Non-decalcified histology of a subsequent right femur fracture showed increased osteoblasts and osteoclasts and excessive osteoid. A 24-hour urine collection contained 27 mg/L F (Nl, 0.2–3.2) and <2 mg/dL Ca. Then, 19 months after “huffing” cessation and improved Ca and D3 intake, yet with persisting bone pain, serum PTH was normal (52 pg/mL) and serum ALP and urine F had decreased to 248 U/L and 3.3 mg/L, respectively. Our experience combined with 15 publications in PubMed concerning unusual causes of non-endemic SF where the F source became known (19 cases in all) revealed: 11 instances from high consumption of black tea and/or F-containing toothpaste, 1 due to geophagia of F-rich soil, and 7 due to “recreational” inhalation of F-containing vapors. Circulating PTH measured in 14 was substantially elevated in 2 (including ours) and mildly increased in 2. The severity of SF in the cases reviewed seemed to reflect cumulative F exposure, renal function, and Ca and D status. Several factors appeared to influence our patient's skeletal disease: i) direct anabolic effects of toxic amounts of F on his skeleton, ii) secondary hyperparathyroidism from degradation-resistant fluorapatite bone crystals and low dietary Ca, and iii) impaired mineralization of excessive osteoid due to hypocalcemia.
•Fluorocarbon “huffing” is an under-appreciated cause of skeletal fluorosis (SF).•We present a SF case with hyperparathyroidism, osteosclerosis, and osteomalacia.•SF may go undetected due to variation in symptoms, radiology, and biochemistry.•Dietary calcium, prior bone health, and skeletal F exposure influence SF features.•SF is common in endemic areas. We review unusual, non-endemic causes of SF.</abstract><cop>NEW YORK</cop><pub>Elsevier Inc</pub><pmid>33418099</pmid><doi>10.1016/j.bone.2021.115839</doi><tpages>10</tpages><orcidid>https://orcid.org/0000-0003-4275-8920</orcidid><oa>free_for_read</oa></addata></record> |
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| subjects | Atypical femoral fracture Bone Density Bone Diseases - chemically induced Bone Diseases - diagnostic imaging Bone pain Bone scan Difluoroethane DXA Elevated bone mass Endocrinology & Metabolism Fluoride Fluorocarbon Fluorosis Hip fracture Huffing Humans Hyperparathyroidism Hyperparathyroidism, Secondary - diagnostic imaging Hypertrophic callus Hypocalcemia Life Sciences & Biomedicine Male Middle Aged Osteolysis Osteomalacia Osteoporosis Osteosclerosis Parathyroid Hormone Periostitis Science & Technology Spine Tea consumption |
| title | Non-endemic skeletal fluorosis: Causes and associated secondary hyperparathyroidism (case report and literature review) |
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