Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets
Summary In X-linked hypophosphatemic (XLH) rickets, dual-energy X-ray absorptiometry (DXA) measurements must be analyzed with caution. High-resolution peripheral quantitative computed tomography (HR-pQCT) analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more a...
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| description | Summary
In X-linked hypophosphatemic (XLH) rickets, dual-energy X-ray absorptiometry (DXA) measurements must be analyzed with caution. High-resolution peripheral quantitative computed tomography (HR-pQCT) analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.
Introduction
The purpose of this study is to evaluate bone mineral density (BMD) and microarchitecture in 37 patients (13 children and 24 adults) with XLH confirmed by
PHEX
mutations from a tertiary center compared to healthy controls.
Methods
Areal BMD (aBMD) was evaluated by DXA, whereas volumetric BMD (vBMD) and microarchitectural parameters were analyzed by HR-pQCT.
Results
Adult XLH patients had higher lumbar aBMD (
p
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| doi_str_mv | 10.1007/s00198-017-3949-8 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_1891885876</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2008065599</sourcerecordid><originalsourceid>FETCH-LOGICAL-c405t-b9f10f750070ede6bf59ec71077ac9582ffc3bfd6abd01aa094935f26b7f05c83</originalsourceid><addsrcrecordid>eNqNkc-K1TAUxoMoznX0AdxIwI2b6EnbNMlyuDM6woAoI9xdSNPEZqZNa5Iq9018XHPnjgqC4CqQ73e-8-dD6DmF1xSAv0kAVAoClJNaNpKIB2hDm7omlWzZQ7QBWXMiG7o7QU9SuoFSIyV_jE4qQWXTCNigHxff9Ljq7OeAZ4e7OVg8-WCjHnFvQ_J5j3Xoy5-Js45m8NmavB7kRUc92Wxjwt0en-_O7sDLT2T5uL3GPuCa48KPfbThTtL9OuaEv_s84B0Zfbi1PR72y7wMc1oGnW3pgqM3tzanp-iR02Oyz-7fU_T57cX19pJcfXj3fnt2RUwDLJNOOgqOs7Ia2N62nWPSGk6Bc20kE5Vzpu5c3-quB6o1lDvVzFVtxx0wI-pT9Orou8T562pTVpNPxo6jDnZek6JCUiGY4O1_oK2oJWPACvryL_RmXmMoi6gKQEDLmJSFokeq3DalaJ1aop903CsK6pCwOiasSsLqkLA6zPvi3nntJtv_rvgVaQGqI5CKFL7Y-Kf1v11_Avf5sl4</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2008065599</pqid></control><display><type>article</type><title>Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets</title><source>MEDLINE</source><source>Springer Online Journals Complete</source><creator>Colares Neto, G. P. ; Pereira, R. M. R. ; Alvarenga, J. C. ; Takayama, L. ; Funari, M. F. A. ; Martin, R. M.</creator><creatorcontrib>Colares Neto, G. P. ; Pereira, R. M. R. ; Alvarenga, J. C. ; Takayama, L. ; Funari, M. F. A. ; Martin, R. M.</creatorcontrib><description>Summary
In X-linked hypophosphatemic (XLH) rickets, dual-energy X-ray absorptiometry (DXA) measurements must be analyzed with caution. High-resolution peripheral quantitative computed tomography (HR-pQCT) analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.
Introduction
The purpose of this study is to evaluate bone mineral density (BMD) and microarchitecture in 37 patients (13 children and 24 adults) with XLH confirmed by
PHEX
mutations from a tertiary center compared to healthy controls.
Methods
Areal BMD (aBMD) was evaluated by DXA, whereas volumetric BMD (vBMD) and microarchitectural parameters were analyzed by HR-pQCT.
Results
Adult XLH patients had higher lumbar aBMD (
p
< 0.01) than the controls. At the radius, the vBMD was similar between XLH patients and controls. At the tibia, XLH patients had lower total vBMD (
p
= 0.04), likely resulting from decreased trabecular vBMD (
p
< 0.01), and this difference was observed in the children and adult groups. Analysis based on metabolic status showed that the adult XLH patients with non-compensated disease had lower cortical vBMD at the tibia than the compensated XLH patients (
p =
0.03). The microarchitectural differences at the radius and tibia included lower trabecular number (
p
< 0.01), greater trabecular separation (
p
< 0.01), and higher trabecular network inhomogeneity (
p
< 0.01) in XLH patients compared to their controls. At the radius, adults exhibited greater trabecular deficits than were seen in children.
Conclusions
In XLH patients, DXA measurements must be analyzed with caution due to the interference of anatomic and anthropometric factors. HR-pQCT analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.</description><identifier>ISSN: 0937-941X</identifier><identifier>EISSN: 1433-2965</identifier><identifier>DOI: 10.1007/s00198-017-3949-8</identifier><identifier>PMID: 28194480</identifier><language>eng</language><publisher>London: Springer London</publisher><subject>Adolescent ; Adult ; Adults ; Age Factors ; Aged ; Bone density ; Bone Density - physiology ; Bone mineral density ; Child ; Children ; Computed tomography ; Cortical bone ; Dual energy X-ray absorptiometry ; Endocrinology ; Familial Hypophosphatemic Rickets - diagnostic imaging ; Familial Hypophosphatemic Rickets - pathology ; Familial Hypophosphatemic Rickets - physiopathology ; Female ; Humans ; Hypophosphatemia ; Lumbar Vertebrae - diagnostic imaging ; Lumbar Vertebrae - pathology ; Lumbar Vertebrae - physiopathology ; Male ; Medicine ; Medicine & Public Health ; Middle Aged ; Mineralization ; Mutation ; Original Article ; Orthopedics ; Osteoporosis ; Radius ; Radius - diagnostic imaging ; Radius - pathology ; Radius - physiopathology ; Rheumatology ; Rickets ; Tibia ; Tibia - diagnostic imaging ; Tibia - pathology ; Tibia - physiopathology ; Tomography, X-Ray Computed - methods ; Vitamin D ; Young Adult</subject><ispartof>Osteoporosis international, 2017-05, Vol.28 (5), p.1685-1692</ispartof><rights>International Osteoporosis Foundation and National Osteoporosis Foundation 2017</rights><rights>Osteoporosis International is a copyright of Springer, (2017). All Rights Reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c405t-b9f10f750070ede6bf59ec71077ac9582ffc3bfd6abd01aa094935f26b7f05c83</citedby><cites>FETCH-LOGICAL-c405t-b9f10f750070ede6bf59ec71077ac9582ffc3bfd6abd01aa094935f26b7f05c83</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s00198-017-3949-8$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s00198-017-3949-8$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,776,780,27901,27902,41464,42533,51294</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28194480$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Colares Neto, G. P.</creatorcontrib><creatorcontrib>Pereira, R. M. R.</creatorcontrib><creatorcontrib>Alvarenga, J. C.</creatorcontrib><creatorcontrib>Takayama, L.</creatorcontrib><creatorcontrib>Funari, M. F. A.</creatorcontrib><creatorcontrib>Martin, R. M.</creatorcontrib><title>Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets</title><title>Osteoporosis international</title><addtitle>Osteoporos Int</addtitle><addtitle>Osteoporos Int</addtitle><description>Summary
In X-linked hypophosphatemic (XLH) rickets, dual-energy X-ray absorptiometry (DXA) measurements must be analyzed with caution. High-resolution peripheral quantitative computed tomography (HR-pQCT) analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.
Introduction
The purpose of this study is to evaluate bone mineral density (BMD) and microarchitecture in 37 patients (13 children and 24 adults) with XLH confirmed by
PHEX
mutations from a tertiary center compared to healthy controls.
Methods
Areal BMD (aBMD) was evaluated by DXA, whereas volumetric BMD (vBMD) and microarchitectural parameters were analyzed by HR-pQCT.
Results
Adult XLH patients had higher lumbar aBMD (
p
< 0.01) than the controls. At the radius, the vBMD was similar between XLH patients and controls. At the tibia, XLH patients had lower total vBMD (
p
= 0.04), likely resulting from decreased trabecular vBMD (
p
< 0.01), and this difference was observed in the children and adult groups. Analysis based on metabolic status showed that the adult XLH patients with non-compensated disease had lower cortical vBMD at the tibia than the compensated XLH patients (
p =
0.03). The microarchitectural differences at the radius and tibia included lower trabecular number (
p
< 0.01), greater trabecular separation (
p
< 0.01), and higher trabecular network inhomogeneity (
p
< 0.01) in XLH patients compared to their controls. At the radius, adults exhibited greater trabecular deficits than were seen in children.
Conclusions
In XLH patients, DXA measurements must be analyzed with caution due to the interference of anatomic and anthropometric factors. HR-pQCT analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Adults</subject><subject>Age Factors</subject><subject>Aged</subject><subject>Bone density</subject><subject>Bone Density - physiology</subject><subject>Bone mineral density</subject><subject>Child</subject><subject>Children</subject><subject>Computed tomography</subject><subject>Cortical bone</subject><subject>Dual energy X-ray absorptiometry</subject><subject>Endocrinology</subject><subject>Familial Hypophosphatemic Rickets - diagnostic imaging</subject><subject>Familial Hypophosphatemic Rickets - pathology</subject><subject>Familial Hypophosphatemic Rickets - physiopathology</subject><subject>Female</subject><subject>Humans</subject><subject>Hypophosphatemia</subject><subject>Lumbar Vertebrae - diagnostic imaging</subject><subject>Lumbar Vertebrae - pathology</subject><subject>Lumbar Vertebrae - physiopathology</subject><subject>Male</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Middle Aged</subject><subject>Mineralization</subject><subject>Mutation</subject><subject>Original Article</subject><subject>Orthopedics</subject><subject>Osteoporosis</subject><subject>Radius</subject><subject>Radius - diagnostic imaging</subject><subject>Radius - pathology</subject><subject>Radius - physiopathology</subject><subject>Rheumatology</subject><subject>Rickets</subject><subject>Tibia</subject><subject>Tibia - diagnostic imaging</subject><subject>Tibia - pathology</subject><subject>Tibia - physiopathology</subject><subject>Tomography, X-Ray Computed - methods</subject><subject>Vitamin D</subject><subject>Young Adult</subject><issn>0937-941X</issn><issn>1433-2965</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNqNkc-K1TAUxoMoznX0AdxIwI2b6EnbNMlyuDM6woAoI9xdSNPEZqZNa5Iq9018XHPnjgqC4CqQ73e-8-dD6DmF1xSAv0kAVAoClJNaNpKIB2hDm7omlWzZQ7QBWXMiG7o7QU9SuoFSIyV_jE4qQWXTCNigHxff9Ljq7OeAZ4e7OVg8-WCjHnFvQ_J5j3Xoy5-Js45m8NmavB7kRUc92Wxjwt0en-_O7sDLT2T5uL3GPuCa48KPfbThTtL9OuaEv_s84B0Zfbi1PR72y7wMc1oGnW3pgqM3tzanp-iR02Oyz-7fU_T57cX19pJcfXj3fnt2RUwDLJNOOgqOs7Ia2N62nWPSGk6Bc20kE5Vzpu5c3-quB6o1lDvVzFVtxx0wI-pT9Orou8T562pTVpNPxo6jDnZek6JCUiGY4O1_oK2oJWPACvryL_RmXmMoi6gKQEDLmJSFokeq3DalaJ1aop903CsK6pCwOiasSsLqkLA6zPvi3nntJtv_rvgVaQGqI5CKFL7Y-Kf1v11_Avf5sl4</recordid><startdate>20170501</startdate><enddate>20170501</enddate><creator>Colares Neto, G. P.</creator><creator>Pereira, R. M. R.</creator><creator>Alvarenga, J. C.</creator><creator>Takayama, L.</creator><creator>Funari, M. F. A.</creator><creator>Martin, R. M.</creator><general>Springer London</general><general>Springer Nature B.V</general><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>3V.</scope><scope>7QP</scope><scope>7RV</scope><scope>7TS</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>KB0</scope><scope>M0S</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope></search><sort><creationdate>20170501</creationdate><title>Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets</title><author>Colares Neto, G. P. ; Pereira, R. M. R. ; Alvarenga, J. C. ; Takayama, L. ; Funari, M. F. A. ; Martin, R. M.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c405t-b9f10f750070ede6bf59ec71077ac9582ffc3bfd6abd01aa094935f26b7f05c83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Adolescent</topic><topic>Adult</topic><topic>Adults</topic><topic>Age Factors</topic><topic>Aged</topic><topic>Bone density</topic><topic>Bone Density - physiology</topic><topic>Bone mineral density</topic><topic>Child</topic><topic>Children</topic><topic>Computed tomography</topic><topic>Cortical bone</topic><topic>Dual energy X-ray absorptiometry</topic><topic>Endocrinology</topic><topic>Familial Hypophosphatemic Rickets - diagnostic imaging</topic><topic>Familial Hypophosphatemic Rickets - pathology</topic><topic>Familial Hypophosphatemic Rickets - physiopathology</topic><topic>Female</topic><topic>Humans</topic><topic>Hypophosphatemia</topic><topic>Lumbar Vertebrae - diagnostic imaging</topic><topic>Lumbar Vertebrae - pathology</topic><topic>Lumbar Vertebrae - physiopathology</topic><topic>Male</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Middle Aged</topic><topic>Mineralization</topic><topic>Mutation</topic><topic>Original Article</topic><topic>Orthopedics</topic><topic>Osteoporosis</topic><topic>Radius</topic><topic>Radius - diagnostic imaging</topic><topic>Radius - pathology</topic><topic>Radius - physiopathology</topic><topic>Rheumatology</topic><topic>Rickets</topic><topic>Tibia</topic><topic>Tibia - diagnostic imaging</topic><topic>Tibia - pathology</topic><topic>Tibia - physiopathology</topic><topic>Tomography, X-Ray Computed - methods</topic><topic>Vitamin D</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Colares Neto, G. P.</creatorcontrib><creatorcontrib>Pereira, R. M. R.</creatorcontrib><creatorcontrib>Alvarenga, J. C.</creatorcontrib><creatorcontrib>Takayama, L.</creatorcontrib><creatorcontrib>Funari, M. F. A.</creatorcontrib><creatorcontrib>Martin, R. M.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Calcium & Calcified Tissue Abstracts</collection><collection>Nursing & Allied Health Database</collection><collection>Physical Education Index</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Public Health Database</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Nursing & Allied Health Premium</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><jtitle>Osteoporosis international</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Colares Neto, G. P.</au><au>Pereira, R. M. R.</au><au>Alvarenga, J. C.</au><au>Takayama, L.</au><au>Funari, M. F. A.</au><au>Martin, R. M.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets</atitle><jtitle>Osteoporosis international</jtitle><stitle>Osteoporos Int</stitle><addtitle>Osteoporos Int</addtitle><date>2017-05-01</date><risdate>2017</risdate><volume>28</volume><issue>5</issue><spage>1685</spage><epage>1692</epage><pages>1685-1692</pages><issn>0937-941X</issn><eissn>1433-2965</eissn><abstract>Summary
In X-linked hypophosphatemic (XLH) rickets, dual-energy X-ray absorptiometry (DXA) measurements must be analyzed with caution. High-resolution peripheral quantitative computed tomography (HR-pQCT) analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.
Introduction
The purpose of this study is to evaluate bone mineral density (BMD) and microarchitecture in 37 patients (13 children and 24 adults) with XLH confirmed by
PHEX
mutations from a tertiary center compared to healthy controls.
Methods
Areal BMD (aBMD) was evaluated by DXA, whereas volumetric BMD (vBMD) and microarchitectural parameters were analyzed by HR-pQCT.
Results
Adult XLH patients had higher lumbar aBMD (
p
< 0.01) than the controls. At the radius, the vBMD was similar between XLH patients and controls. At the tibia, XLH patients had lower total vBMD (
p
= 0.04), likely resulting from decreased trabecular vBMD (
p
< 0.01), and this difference was observed in the children and adult groups. Analysis based on metabolic status showed that the adult XLH patients with non-compensated disease had lower cortical vBMD at the tibia than the compensated XLH patients (
p =
0.03). The microarchitectural differences at the radius and tibia included lower trabecular number (
p
< 0.01), greater trabecular separation (
p
< 0.01), and higher trabecular network inhomogeneity (
p
< 0.01) in XLH patients compared to their controls. At the radius, adults exhibited greater trabecular deficits than were seen in children.
Conclusions
In XLH patients, DXA measurements must be analyzed with caution due to the interference of anatomic and anthropometric factors. HR-pQCT analysis suggested that XLH primarily affects the cancellous compartment, with the tibia more affected than the radius. Effective treatment of XLH appears to positively affect bone mineralization, mainly in the bone cortex.</abstract><cop>London</cop><pub>Springer London</pub><pmid>28194480</pmid><doi>10.1007/s00198-017-3949-8</doi><tpages>8</tpages></addata></record> |
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| ispartof | Osteoporosis international, 2017-05, Vol.28 (5), p.1685-1692 |
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| language | eng |
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| source | MEDLINE; Springer Online Journals Complete |
| subjects | Adolescent Adult Adults Age Factors Aged Bone density Bone Density - physiology Bone mineral density Child Children Computed tomography Cortical bone Dual energy X-ray absorptiometry Endocrinology Familial Hypophosphatemic Rickets - diagnostic imaging Familial Hypophosphatemic Rickets - pathology Familial Hypophosphatemic Rickets - physiopathology Female Humans Hypophosphatemia Lumbar Vertebrae - diagnostic imaging Lumbar Vertebrae - pathology Lumbar Vertebrae - physiopathology Male Medicine Medicine & Public Health Middle Aged Mineralization Mutation Original Article Orthopedics Osteoporosis Radius Radius - diagnostic imaging Radius - pathology Radius - physiopathology Rheumatology Rickets Tibia Tibia - diagnostic imaging Tibia - pathology Tibia - physiopathology Tomography, X-Ray Computed - methods Vitamin D Young Adult |
| title | Evaluation of bone mineral density and microarchitectural parameters by DXA and HR-pQCT in 37 children and adults with X-linked hypophosphatemic rickets |
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