High incidence of significant bone loss in patients with severe congenital neutropenia (Kostmann’s syndrome)
Objective: Clinical observation of bone pain, unusual fractures in two patients, and diffuse osteopenia/osteoporosis led us to assess bone mineral content and density in 30 patients with severe congenital neutropenia who were treated with recombinant-methionyl-human granulocyte colony-stimulating fa...
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| Veröffentlicht in: | The Journal of pediatrics 1997-10, Vol.131 (4), p.592-597 |
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| creator | Yakisan, Elif Schirg, Eckart Zeidler, Cornelia Bishop, Nick J. Reiter, Alfred Hirt, A. Riehm, Hansjörg Welte, Karl |
| description | Objective: Clinical observation of bone pain, unusual fractures in two patients, and diffuse osteopenia/osteoporosis led us to assess bone mineral content and density in 30 patients with severe congenital neutropenia who were treated with recombinant-methionyl-human granulocyte colony-stimulating factor (r-metHuG-CSF).
Study design: We reviewed roentgenograms in 29 of these 30 patients to evaluate bone loss before and during treatment. In addition, in 17 of the 30 patients, bone mineral status could be assessed by both quantitative computed tomography (Q-CT;
n = 16) and dual energy x-ray absorptiometry (DXA;
n = 1). In one patient, Q-CT was not possible because of severe vertebral fractures.
Results: Of the 30 patients investigated, 15 had evidence of osteopenia/osteoporosis observed on spine radiographs (
n = 5), on Q-CT/DXA (
n = 1/
n = 1), or on radiographs and Q-CT (
n = 8). In 13 of the 30 patients, only a lateral radiograph of the lumbar spine was available, 5 of 13 showing either increased kyphosis and wedging of the vertebrae or compression fractures of the vertebral bodies, indicating severe established osteoporosis. In eight patients, the findings of the spinal radiographs were normal. In nine patients, spinal radiographs were taken before r-metHuG-CSF treatment. Osteoporotic vertebral deformation (
n = 3) or reduced bone mass (
n = 3) was seen in six of these nine patients. The levels of serum biochemical markers of bone metabolism were all within normal ranges except for mild elevation of the serum alkaline phosphatase level. The degree of spinal bone mineral loss did not correlate with dose and duration of r-metHuG-CSF treatment or with the age or sex of the patients.
Conclusions: These data indicate a high incidence of bone mineral loss in children with severe congenital neutropenia. The underlying pathogenesis of bone demineralization is not clear. It is more likely that the bone loss was caused by the pathophysiologic features of the underlying disease, but it is possible that r-metHuG-CSF accelerates bone mineral loss. (J Pediatr 1997;131:592-7) |
| doi_str_mv | 10.1016/S0022-3476(97)70068-4 |
| format | Article |
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Study design: We reviewed roentgenograms in 29 of these 30 patients to evaluate bone loss before and during treatment. In addition, in 17 of the 30 patients, bone mineral status could be assessed by both quantitative computed tomography (Q-CT;
n = 16) and dual energy x-ray absorptiometry (DXA;
n = 1). In one patient, Q-CT was not possible because of severe vertebral fractures.
Results: Of the 30 patients investigated, 15 had evidence of osteopenia/osteoporosis observed on spine radiographs (
n = 5), on Q-CT/DXA (
n = 1/
n = 1), or on radiographs and Q-CT (
n = 8). In 13 of the 30 patients, only a lateral radiograph of the lumbar spine was available, 5 of 13 showing either increased kyphosis and wedging of the vertebrae or compression fractures of the vertebral bodies, indicating severe established osteoporosis. In eight patients, the findings of the spinal radiographs were normal. In nine patients, spinal radiographs were taken before r-metHuG-CSF treatment. Osteoporotic vertebral deformation (
n = 3) or reduced bone mass (
n = 3) was seen in six of these nine patients. The levels of serum biochemical markers of bone metabolism were all within normal ranges except for mild elevation of the serum alkaline phosphatase level. The degree of spinal bone mineral loss did not correlate with dose and duration of r-metHuG-CSF treatment or with the age or sex of the patients.
Conclusions: These data indicate a high incidence of bone mineral loss in children with severe congenital neutropenia. The underlying pathogenesis of bone demineralization is not clear. It is more likely that the bone loss was caused by the pathophysiologic features of the underlying disease, but it is possible that r-metHuG-CSF accelerates bone mineral loss. (J Pediatr 1997;131:592-7)</description><identifier>ISSN: 0022-3476</identifier><identifier>EISSN: 1097-6833</identifier><identifier>DOI: 10.1016/S0022-3476(97)70068-4</identifier><identifier>PMID: 9386665</identifier><identifier>CODEN: JOPDAB</identifier><language>eng</language><publisher>New York, NY: Mosby, Inc</publisher><subject>Adolescent ; Adult ; Alkaline Phosphatase - blood ; Back Pain - etiology ; Biological and medical sciences ; Bone Density ; Child ; Female ; Filgrastim ; Follow-Up Studies ; Granulocyte Colony-Stimulating Factor - therapeutic use ; Hematologic and hematopoietic diseases ; Hematopoietic Cell Growth Factors - therapeutic use ; Humans ; Incidence ; Lumbar Vertebrae - diagnostic imaging ; Male ; Medical sciences ; Neutropenia - complications ; Neutropenia - congenital ; Neutropenia - drug therapy ; Neutropenia - epidemiology ; Osteoporosis - diagnostic imaging ; Osteoporosis - epidemiology ; Osteoporosis - etiology ; Other diseases. Hematologic involvement in other diseases ; Recombinant Proteins ; Severity of Illness Index ; Syndrome ; Tomography, X-Ray Computed</subject><ispartof>The Journal of pediatrics, 1997-10, Vol.131 (4), p.592-597</ispartof><rights>1997 Mosby, Inc.</rights><rights>1998 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c441t-234e1e5b0398a55dc8b2a496a1cc3b530690609386a780d6ca6571d968f3a2043</citedby><cites>FETCH-LOGICAL-c441t-234e1e5b0398a55dc8b2a496a1cc3b530690609386a780d6ca6571d968f3a2043</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0022347697700684$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65306</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=2109769$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/9386665$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Yakisan, Elif</creatorcontrib><creatorcontrib>Schirg, Eckart</creatorcontrib><creatorcontrib>Zeidler, Cornelia</creatorcontrib><creatorcontrib>Bishop, Nick J.</creatorcontrib><creatorcontrib>Reiter, Alfred</creatorcontrib><creatorcontrib>Hirt, A.</creatorcontrib><creatorcontrib>Riehm, Hansjörg</creatorcontrib><creatorcontrib>Welte, Karl</creatorcontrib><creatorcontrib>From the Departments of Pediatric Hematology/Oncology and Radiology, Medical School Hannover, D-30625 Hannover, Germany; Shriners Hospitals for Crippled Children, Montréal, Quebec, Canada; and the Department of Pediatrics, University of Bern, Switzerland</creatorcontrib><title>High incidence of significant bone loss in patients with severe congenital neutropenia (Kostmann’s syndrome)</title><title>The Journal of pediatrics</title><addtitle>J Pediatr</addtitle><description>Objective: Clinical observation of bone pain, unusual fractures in two patients, and diffuse osteopenia/osteoporosis led us to assess bone mineral content and density in 30 patients with severe congenital neutropenia who were treated with recombinant-methionyl-human granulocyte colony-stimulating factor (r-metHuG-CSF).
Study design: We reviewed roentgenograms in 29 of these 30 patients to evaluate bone loss before and during treatment. In addition, in 17 of the 30 patients, bone mineral status could be assessed by both quantitative computed tomography (Q-CT;
n = 16) and dual energy x-ray absorptiometry (DXA;
n = 1). In one patient, Q-CT was not possible because of severe vertebral fractures.
Results: Of the 30 patients investigated, 15 had evidence of osteopenia/osteoporosis observed on spine radiographs (
n = 5), on Q-CT/DXA (
n = 1/
n = 1), or on radiographs and Q-CT (
n = 8). In 13 of the 30 patients, only a lateral radiograph of the lumbar spine was available, 5 of 13 showing either increased kyphosis and wedging of the vertebrae or compression fractures of the vertebral bodies, indicating severe established osteoporosis. In eight patients, the findings of the spinal radiographs were normal. In nine patients, spinal radiographs were taken before r-metHuG-CSF treatment. Osteoporotic vertebral deformation (
n = 3) or reduced bone mass (
n = 3) was seen in six of these nine patients. The levels of serum biochemical markers of bone metabolism were all within normal ranges except for mild elevation of the serum alkaline phosphatase level. The degree of spinal bone mineral loss did not correlate with dose and duration of r-metHuG-CSF treatment or with the age or sex of the patients.
Conclusions: These data indicate a high incidence of bone mineral loss in children with severe congenital neutropenia. The underlying pathogenesis of bone demineralization is not clear. It is more likely that the bone loss was caused by the pathophysiologic features of the underlying disease, but it is possible that r-metHuG-CSF accelerates bone mineral loss. (J Pediatr 1997;131:592-7)</description><subject>Adolescent</subject><subject>Adult</subject><subject>Alkaline Phosphatase - blood</subject><subject>Back Pain - etiology</subject><subject>Biological and medical sciences</subject><subject>Bone Density</subject><subject>Child</subject><subject>Female</subject><subject>Filgrastim</subject><subject>Follow-Up Studies</subject><subject>Granulocyte Colony-Stimulating Factor - therapeutic use</subject><subject>Hematologic and hematopoietic diseases</subject><subject>Hematopoietic Cell Growth Factors - therapeutic use</subject><subject>Humans</subject><subject>Incidence</subject><subject>Lumbar Vertebrae - diagnostic imaging</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Neutropenia - complications</subject><subject>Neutropenia - congenital</subject><subject>Neutropenia - drug therapy</subject><subject>Neutropenia - epidemiology</subject><subject>Osteoporosis - diagnostic imaging</subject><subject>Osteoporosis - epidemiology</subject><subject>Osteoporosis - etiology</subject><subject>Other diseases. Hematologic involvement in other diseases</subject><subject>Recombinant Proteins</subject><subject>Severity of Illness Index</subject><subject>Syndrome</subject><subject>Tomography, X-Ray Computed</subject><issn>0022-3476</issn><issn>1097-6833</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>1997</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkcFu1DAQhi0EKkvhESr5gFB7SBnHiR2fEKqgRa3EAThbjjPZGiX2YnuLeuM1-no8SZ3uaq89WdZ89j_zDSEnDM4ZMPHxB0BdV7yR4lTJMwkguqp5QVYMlKxEx_lLsjogr8mblH4DgGoAjsiR4p0Qol0Rf-XWt9R56wb0FmkYaXJr70Znjc-0Dx7pFFIqCN2Y7NDnRP-6fEsT3mFEaoNfo3fZTNTjNsewKTdDT69DyrPx_v-_h0TTvR9imPHsLXk1minhu_15TH59_fLz4qq6-X757eLzTWWbhuWq5g0ybHvgqjNtO9iur02jhGHW8r7lIBQIWIYwsoNBWCNayQYlupGbGhp-TD7s_t3E8GeLKevZJYvTZDyGbdJSlRzFZAHbHWhjmTLiqDfRzSbeawZ68ayfPOtFolZSP3nWS8DJPmDbzzgcXu3Flvr7fd0ka6YxmqI4HbB62ZJQBfu0w7DIuHMYdbJuWcTgItqsh-CeaeQRYs2bMg</recordid><startdate>19971001</startdate><enddate>19971001</enddate><creator>Yakisan, Elif</creator><creator>Schirg, Eckart</creator><creator>Zeidler, Cornelia</creator><creator>Bishop, Nick J.</creator><creator>Reiter, Alfred</creator><creator>Hirt, A.</creator><creator>Riehm, Hansjörg</creator><creator>Welte, Karl</creator><general>Mosby, Inc</general><general>Elsevier</general><scope>IQODW</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>7X8</scope></search><sort><creationdate>19971001</creationdate><title>High incidence of significant bone loss in patients with severe congenital neutropenia (Kostmann’s syndrome)</title><author>Yakisan, Elif ; Schirg, Eckart ; Zeidler, Cornelia ; Bishop, Nick J. ; Reiter, Alfred ; Hirt, A. ; Riehm, Hansjörg ; Welte, Karl</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c441t-234e1e5b0398a55dc8b2a496a1cc3b530690609386a780d6ca6571d968f3a2043</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>1997</creationdate><topic>Adolescent</topic><topic>Adult</topic><topic>Alkaline Phosphatase - blood</topic><topic>Back Pain - etiology</topic><topic>Biological and medical sciences</topic><topic>Bone Density</topic><topic>Child</topic><topic>Female</topic><topic>Filgrastim</topic><topic>Follow-Up Studies</topic><topic>Granulocyte Colony-Stimulating Factor - therapeutic use</topic><topic>Hematologic and hematopoietic diseases</topic><topic>Hematopoietic Cell Growth Factors - therapeutic use</topic><topic>Humans</topic><topic>Incidence</topic><topic>Lumbar Vertebrae - diagnostic imaging</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Neutropenia - complications</topic><topic>Neutropenia - congenital</topic><topic>Neutropenia - drug therapy</topic><topic>Neutropenia - epidemiology</topic><topic>Osteoporosis - diagnostic imaging</topic><topic>Osteoporosis - epidemiology</topic><topic>Osteoporosis - etiology</topic><topic>Other diseases. Hematologic involvement in other diseases</topic><topic>Recombinant Proteins</topic><topic>Severity of Illness Index</topic><topic>Syndrome</topic><topic>Tomography, X-Ray Computed</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Yakisan, Elif</creatorcontrib><creatorcontrib>Schirg, Eckart</creatorcontrib><creatorcontrib>Zeidler, Cornelia</creatorcontrib><creatorcontrib>Bishop, Nick J.</creatorcontrib><creatorcontrib>Reiter, Alfred</creatorcontrib><creatorcontrib>Hirt, A.</creatorcontrib><creatorcontrib>Riehm, Hansjörg</creatorcontrib><creatorcontrib>Welte, Karl</creatorcontrib><creatorcontrib>From the Departments of Pediatric Hematology/Oncology and Radiology, Medical School Hannover, D-30625 Hannover, Germany; Shriners Hospitals for Crippled Children, Montréal, Quebec, Canada; and the Department of Pediatrics, University of Bern, Switzerland</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>The Journal of pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Yakisan, Elif</au><au>Schirg, Eckart</au><au>Zeidler, Cornelia</au><au>Bishop, Nick J.</au><au>Reiter, Alfred</au><au>Hirt, A.</au><au>Riehm, Hansjörg</au><au>Welte, Karl</au><aucorp>From the Departments of Pediatric Hematology/Oncology and Radiology, Medical School Hannover, D-30625 Hannover, Germany; Shriners Hospitals for Crippled Children, Montréal, Quebec, Canada; and the Department of Pediatrics, University of Bern, Switzerland</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>High incidence of significant bone loss in patients with severe congenital neutropenia (Kostmann’s syndrome)</atitle><jtitle>The Journal of pediatrics</jtitle><addtitle>J Pediatr</addtitle><date>1997-10-01</date><risdate>1997</risdate><volume>131</volume><issue>4</issue><spage>592</spage><epage>597</epage><pages>592-597</pages><issn>0022-3476</issn><eissn>1097-6833</eissn><coden>JOPDAB</coden><abstract>Objective: Clinical observation of bone pain, unusual fractures in two patients, and diffuse osteopenia/osteoporosis led us to assess bone mineral content and density in 30 patients with severe congenital neutropenia who were treated with recombinant-methionyl-human granulocyte colony-stimulating factor (r-metHuG-CSF).
Study design: We reviewed roentgenograms in 29 of these 30 patients to evaluate bone loss before and during treatment. In addition, in 17 of the 30 patients, bone mineral status could be assessed by both quantitative computed tomography (Q-CT;
n = 16) and dual energy x-ray absorptiometry (DXA;
n = 1). In one patient, Q-CT was not possible because of severe vertebral fractures.
Results: Of the 30 patients investigated, 15 had evidence of osteopenia/osteoporosis observed on spine radiographs (
n = 5), on Q-CT/DXA (
n = 1/
n = 1), or on radiographs and Q-CT (
n = 8). In 13 of the 30 patients, only a lateral radiograph of the lumbar spine was available, 5 of 13 showing either increased kyphosis and wedging of the vertebrae or compression fractures of the vertebral bodies, indicating severe established osteoporosis. In eight patients, the findings of the spinal radiographs were normal. In nine patients, spinal radiographs were taken before r-metHuG-CSF treatment. Osteoporotic vertebral deformation (
n = 3) or reduced bone mass (
n = 3) was seen in six of these nine patients. The levels of serum biochemical markers of bone metabolism were all within normal ranges except for mild elevation of the serum alkaline phosphatase level. The degree of spinal bone mineral loss did not correlate with dose and duration of r-metHuG-CSF treatment or with the age or sex of the patients.
Conclusions: These data indicate a high incidence of bone mineral loss in children with severe congenital neutropenia. The underlying pathogenesis of bone demineralization is not clear. It is more likely that the bone loss was caused by the pathophysiologic features of the underlying disease, but it is possible that r-metHuG-CSF accelerates bone mineral loss. (J Pediatr 1997;131:592-7)</abstract><cop>New York, NY</cop><pub>Mosby, Inc</pub><pmid>9386665</pmid><doi>10.1016/S0022-3476(97)70068-4</doi><tpages>6</tpages></addata></record> |
| fulltext | fulltext |
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| ispartof | The Journal of pediatrics, 1997-10, Vol.131 (4), p.592-597 |
| issn | 0022-3476 1097-6833 |
| language | eng |
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| source | MEDLINE; Elsevier ScienceDirect Journals |
| subjects | Adolescent Adult Alkaline Phosphatase - blood Back Pain - etiology Biological and medical sciences Bone Density Child Female Filgrastim Follow-Up Studies Granulocyte Colony-Stimulating Factor - therapeutic use Hematologic and hematopoietic diseases Hematopoietic Cell Growth Factors - therapeutic use Humans Incidence Lumbar Vertebrae - diagnostic imaging Male Medical sciences Neutropenia - complications Neutropenia - congenital Neutropenia - drug therapy Neutropenia - epidemiology Osteoporosis - diagnostic imaging Osteoporosis - epidemiology Osteoporosis - etiology Other diseases. Hematologic involvement in other diseases Recombinant Proteins Severity of Illness Index Syndrome Tomography, X-Ray Computed |
| title | High incidence of significant bone loss in patients with severe congenital neutropenia (Kostmann’s syndrome) |
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