Publications by authors named "Alan Oestreich"

31 Publications

Homozygous missense variant in BMPR1A resulting in BMPR signaling disruption and syndromic features.

Mol Genet Genomic Med 2019 11 7;7(11):e969. Epub 2019 Sep 7.

Division of Endocrinology, Cincinnati Children's Hospital and University of Cincinnati College of Medicine, Department of Pediatrics, Cincinnati, OH, USA.

Background: The bone morphogenetic protein (BMP) pathway is known to play an imperative role in bone, cartilage, and cardiac tissue formation. Truncating, heterozygous variants, and deletions of one of the essential receptors in this pathway, Bone Morphogenetic Protein Receptor Type1A (BMPR1A), have been associated with autosomal dominant juvenile polyposis. Heterozygous deletions have also been associated with cardiac and minor skeletal anomalies. Populations with atrioventricular septal defects are enriched for rare missense BMPR1A variants.

Methods: We report on a patient with a homozygous missense variant in BMPR1A causing skeletal abnormalities, growth failure a large atrial septal defect, severe subglottic stenosis, laryngomalacia, facial dysmorphisms, and developmental delays.

Results: Functional analysis of this variant shows increased chondrocyte death for cells with the mutated receptor, increased phosphorylated R-Smads1/5/8, and loss of Sox9 expression mediated by decreased phosphorylation of p38.

Conclusion: This homozygous missense variant in BMPR1A appears to cause a distinct clinical phenotype.
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http://dx.doi.org/10.1002/mgg3.969DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6825850PMC
November 2019

Selective rickets from localized advanced maturation-a case report.

Authors:
Alan E Oestreich

Skeletal Radiol 2019 Aug 20;48(8):1289-1291. Epub 2018 Nov 20.

Department of Radiology ML 5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH, 45229-3039, USA.

An unusual cause of rickets is illustrated by a patient with infantile multisystem inflammatory disease who, by age 2 years and 4 months, developed striking radiographic and clinical rickets restricted to those joints involved by the inflammatory process. The locally increased vascularity from his inflammation led to increased maturation at those sites so rapid as to override the usual enchondral calcification, thus causing a rickets pattern. Other sites, such as the proximal humeri, lacking any inflammation, showed no increased maturation rate and did not manifest local rickets. Rapid local bone maturation may cause localized rickets.
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http://dx.doi.org/10.1007/s00256-018-3114-2DOI Listing
August 2019

Distinguishing child abuse fractures from rickets.

Authors:
Alan E Oestreich

Pediatr Radiol 2018 03 24;48(3):305-307. Epub 2017 Nov 24.

Department of Radiology ML5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave., Cincinnati, OH, 45229-3039, USA.

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http://dx.doi.org/10.1007/s00247-017-4037-0DOI Listing
March 2018

Bowdler spur also found in camptomelic dysplasia.

Authors:
Alan E Oestreich

Pediatr Radiol 2016 Feb 6;46(2):300. Epub 2015 Oct 6.

Department of Radiology, Cincinnati Children's Hospital Medical Center, Radiology 5031, 3333 Burnet Ave., Cincinnati, OH, 45229-3039, USA.

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http://dx.doi.org/10.1007/s00247-015-3471-0DOI Listing
February 2016

Normal spinous process metaphyseal-equivalent lucency simulating infant abuse fractures.

Skeletal Radiol 2015 Oct 9;44(10):1519-22. Epub 2015 Jun 9.

Department of Radiology, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH, 45229-3039, USA,

We report a normal radiographic finding that may be mistaken for child abuse trauma in the posterior thoracolumbar spinous processes of young infants after the first week of life. A lucency paralleling the posterior margin of the ossified spinous process is equivalent to the metaphyseal lucent bands seen normally after about a week of the child's age at the ends of long tubular bones. A similar lucency is seen just under the growth plate of vertebral bodies at that early age, giving the bone-in-bone appearance. Our index case was imaged at 3 weeks and then at 5 weeks of age, with no evidence of periosteal reaction or endosteal callus on that follow-up study, confirming the lack of fractures.
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http://dx.doi.org/10.1007/s00256-015-2184-7DOI Listing
October 2015

Concave distal end of ulna metaphysis alone is not a sign of rickets.

Authors:
Alan E Oestreich

Pediatr Radiol 2015 Jul 1;45(7):998-1000. Epub 2015 Feb 1.

Department of Radiology, 5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave., Cincinnati, OH, 45229-3039, USA,

Background: Statements have been made in the literature and in legal testimony that misrepresent the radiographic finding of concave distal end of the ulnar metaphysis.

Objective: To demonstrate that a concave distal end of the ossified ulna in infancy can be normal.

Materials And Methods: Eighty distal wrists of randomly selected infants in the first year of life with radiographic evidence that ruled out rickets were reviewed.

Results: In 16 of the cases (20%), mild or moderate concavity of the distal end of the ulna was seen. An intact metaphyseal collar of distal radius or ulna confirmed the absence of radiographic rickets.

Conclusion: The finding of 20% of concave distal ulnas in the first year of life confirms the widely acknowledged statements that concave distal end of the ulna alone is not indicative of rickets. Statements to the contrary are not justified.
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http://dx.doi.org/10.1007/s00247-014-3268-6DOI Listing
July 2015

RSNA centennial article: ALARA 1912: "As low a dose as possible" a century ago.

Authors:
Alan E Oestreich

Radiographics 2014 Sep-Oct;34(5):1457-60

From the Department of Radiology, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave, Cincinnati, OH 45229-3039.

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http://dx.doi.org/10.1148/rg.345130136DOI Listing
November 2015

Umbilical vein catheter tip - slight rightward or anterior shift and trajectory can indicate liver penetration.

Pediatr Radiol 2014 Nov 7;44(11):1470-1. Epub 2014 Jun 7.

Department of Radiology 5031, Cincinnati Children's Hospital Medical Center,3333, Burnet Ave., Cincinnati, OH, 45229-3039, USA,

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http://dx.doi.org/10.1007/s00247-014-3035-8DOI Listing
November 2014

The metaphyseal collar is also present until later childhood and at birth.

Authors:
Alan E Oestreich

Pediatr Radiol 2014 Aug 5;44(8):1046-7. Epub 2014 Jun 5.

Department of Radiology 5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave., Cincinnati, OH, 45220-3039, USA,

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http://dx.doi.org/10.1007/s00247-014-3034-9DOI Listing
August 2014

Vigorous periosteal reaction secondary to copper deficiency in an infant on total parenteral nutrition.

Pediatr Radiol 2013 Oct 28;43(10):1411-3. Epub 2013 Jul 28.

Department of Radiology, Cincinnati Children's Hospital Medical Center Radiology 5031 CCHMC, 3333 Burnet Ave., Cincinnati, OH, 45229-3039, USA,

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http://dx.doi.org/10.1007/s00247-013-2759-1DOI Listing
October 2013

Ellis-van Creveld syndrome: its history.

Pediatr Radiol 2013 Aug 12;43(8):1030-6. Epub 2013 Jun 12.

Division of Pediatric Surgery, Department of Surgery, Weill Cornell Medical College, New York Presbyterian Hospital, New York, NY 10065, USA.

The story of Ellis-van Creveld syndrome is one of serendipity. By chance, Simon van Creveld and Richard Ellis purportedly met on a train and combined their independently encountered patients with short stature, dental anomalies and polydactyly into one landmark publication in 1940. They included a patient used in work published previously by Rustin McIntosh without naming McIntosh as a coauthor. This patient was followed radiologically by Caffey for nearly two decades. In 1964, Victor McKusick felt compelled to investigate a brief report in an obscure pharmaceutical journal on an unusual geographic cluster of short-statured Amish patients in Pennsylvania. This review highlights the lives of the individuals involved in the discovery of Ellis-van Creveld syndrome in their historic context.
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http://dx.doi.org/10.1007/s00247-013-2709-yDOI Listing
August 2013

Early radiology and the National Medical Association.

Authors:
Alan E Oestreich

J Natl Med Assoc 2011 Jan;103(1):46-9

Cincinnati Children's Hospital Medical Center, Radiology 5031 CCHMC, 3333 Burnet Ave, Cincinnati, OH 45229-3039, USA.

Both the discovery of the x-ray and the founding of the National Medical Association (NMA) occurred in 1895. Radiologists have participated importantly in the NMA since the lecture of Dr Marcus F. Wheatland at the 1909 Annual Meeting in Boston, Massachusetts, which led to his election as president of the NMA for the following year. More recently, Dr Tracy M. Walton was president of the NMA in its centennial year. This article details early and continued participation in the NMA by radiologists, including the founding of the Section on Radiology in 1949, with its first chairman, Dr William E. Allen, Jr, longtime leader in radiology, the section, and the NMA.
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http://dx.doi.org/10.1016/s0027-9684(15)30242-xDOI Listing
January 2011

Umbilical vein catheterization--appropriate and inappropriate placement.

Authors:
Alan E Oestreich

Pediatr Radiol 2010 Dec 2;40(12):1941-9. Epub 2010 Oct 2.

Department of Radiology 5031, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3039, USA.

Because of the dangers of inappropriate placement of an umbilical vein catheter (UVC), knowledge of the proper pathway and tip localization is important. The acceptable ductus venosus/hepatic vein/inferior vena cava confluence location is compared to inappropriate localizations below and above the diaphragm, with emphasis on confusion about the position when the UVC tip is in the umbilical vein itself, including the umbilical vein recess. Small amounts of gas can inadvertently accompany initial UVC placement and provide landmarks for position evaluation.
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http://dx.doi.org/10.1007/s00247-010-1840-2DOI Listing
December 2010

Scenes from the past: "Skeeter": Harlem Renaissance author's description of a young man's impression of a radiology examination room.

Authors:
Alan E Oestreich

Radiographics 2010 Sep;30(5):1411-4

Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3039, USA.

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http://dx.doi.org/10.1148/rg.305105044DOI Listing
September 2010

Preautopsy imaging in cerebro-costo-mandibular syndrome.

Pediatr Radiol 2010 Dec 13;40 Suppl 1:S50. Epub 2010 May 13.

Department of Radiology 5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave., Cincinnati, OH 45229-3039, USA.

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http://dx.doi.org/10.1007/s00247-010-1703-xDOI Listing
December 2010

Systematic evaluation of bone dysplasias by the paediatric radiologist.

Authors:
Alan E Oestreich

Pediatr Radiol 2010 Jun 30;40(6):975-7. Epub 2010 Apr 30.

Radiology Department, 5031 Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave, Cincinnati, OH 45220-1135, USA.

A bone dysplasia is often a difficult diagnosis for the radiologist to achieve. However, principles of interpretation can make the task both interesting and often straightforward. In general, one of the factors of bone growth is in some way impaired, yielding an abnormal skeleton. To analyse what is impaired may greatly assist in narrowing the diagnostic possibilities. In most dysplasias, the affected growth factor is either one of enchondral or membranous bone growth. When interpreting bone radiographs for dysplasia one should be aware that aberrant positioning, that may lead to foreshortening in space, may simulate impaired growth in time. Dysplasia diagnosis for the paediatric radiologist is an art, but it can also be a science (as well as a pattern recognition challenge).
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http://dx.doi.org/10.1007/s00247-010-1594-xDOI Listing
June 2010

Comment on Hedayati, et al.

Authors:
Alan E Oestreich

Skeletal Radiol 2010 Apr;39(4):397; author reply 399

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April 2010

Stress fractures of ankle and wrist in childhood: nature and frequency.

Pediatr Radiol 2010 Aug 24;40(8):1387-9. Epub 2010 Feb 24.

Department of Radiology, Cincinnati Children's Hospital Medical Center, MLC 5031, 3333 Burnet Ave, Cincinnati, OH 45229-3039, USA.

Background: Stress fractures of many etiologies are found not infrequently in various tarsal bones but are less commonly recognized in carpal bones.

Objective: To assess the distribution of tarsal and carpal stress fractures.

Materials And Methods: During the last three decades, the senior author collected locations of tarsal and carpal bone stress fracture callus seen on plain radiographs.

Results: 527 children with tarsal and carpal stress fractures were identified (88 children had multiple bones involved). The totals were: calcaneus 244, cuboid 188, talus 121, navicular 24, cuneiforms 23, capitate 18, lunate 1, and scaphoid 1. Stress fractures were more frequently seen once we became aware each particular bone could be involved.

Conclusion: Tarsal and carpal stress fractures in children are not rare. Careful perusal of these bones is urged in all susceptible children with limping or wrist pain.
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http://dx.doi.org/10.1007/s00247-010-1577-yDOI Listing
August 2010

Letter to the Editors.

Authors:
Alan E Oestreich

Skeletal Radiol 2010 Jan 9. Epub 2010 Jan 9.

Department of Radiology, Cincinnati Children's Hospital, 3333 Burnet Avenue, 5031, Cincinnati, OH, 45229-3039, USA,

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http://dx.doi.org/10.1007/s00256-009-0848-xDOI Listing
January 2010

Interaction between academic radiology and the news media: a potentially powerful and unpredictable process--five stories.

AJR Am J Roentgenol 2009 May;192(5):1382-7

Department of Radiology, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229-3039, USA.

Objective: Although articles have been published describing the relationship between news reports and the general medical literature, to our knowledge little has been published describing the relationship between radiology publications and news coverage. We present five instances of news stories centered on radiology publications or abstracts of presentations at national meetings and the effect of the media coverage within the medical community.

Conclusion: The five examples show that news coverage can have a dramatic effect on advancing research findings and public health information published in radiology journals. Even when news coverage is inaccurate or sensationalized, the attention of the general public can drive change not only generally in medicine but also specifically in radiology.
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http://dx.doi.org/10.2214/AJR.08.1829DOI Listing
May 2009

Worldwide survey of damage from swallowing multiple magnets.

Authors:
Alan E Oestreich

Pediatr Radiol 2009 Feb 20;39(2):142-7. Epub 2008 Nov 20.

Radiology Department 5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Ave., Cincinnati, OH, 45229-3039, USA.

Background: It is increasingly recognized that in children swallowed multiple magnets cause considerable damage to the gastrointestinal tract.

Objective: To emphasize that complications from swallowed magnets are extensive worldwide and throughout childhood.

Materials And Methods: The author surveyed radiologists and researched cases of magnet swallowing in the literature and documented age and gender, numbers of magnets, nature of the magnets, reasons for swallowing, and clinical course.

Results: A total of 128 instances of magnet swallowing were identified, one fatal. Cases from 21 countries were found. Magnet swallowing occurred throughout childhood, with most children older than 3 years of age. Numbers of swallowed magnets ranged up to 100. Twelve children were known to be autistic. Many reasons were given for swallowing magnets, and a wide range of gastrointestinal damage was encountered. Considerable delay before seeking medical assistance was frequent, as was delay before obtaining radiographs or US imaging.

Conclusion: Damage from swallowing multiple magnets is a considerable worldwide problem. More educational and preventative measures are needed.
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http://dx.doi.org/10.1007/s00247-008-1059-7DOI Listing
February 2009

The usefulness of magnification in postgastric magnetopathy.

Authors:
Alan E Oestreich

Pediatr Radiol 2007 Dec 27;37(12):1268-9. Epub 2007 Sep 27.

Department of Radiology #5031, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3039, USA.

Magnification of a radiographic image using the PACS magnification tool or a magnifying glass can establish that an apparently cylindrical foreign body in the abdomen is actually several magnets attached to one another. There are several reports of abdominal complications from the presence of more than one magnet lying beyond the stomach (postgastric magnetopathy). Such groups of attached magnets have been mistaken for solitary cylinders, and this potential misinterpretation also occurred at initial viewing of images from two of seven of our patients with this condition. Because there is generally no risk of complications from one magnet, the distinction is important.
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http://dx.doi.org/10.1007/s00247-007-0617-8DOI Listing
December 2007

Danger of multiple magnets beyond the stomach in children.

Authors:
Alan E Oestreich

J Natl Med Assoc 2006 Feb;98(2):277-9

Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3039, USA.

Examples from our recent experience, as well as several reports in surgical and pediatric journals, proclaim the danger in children whenever more than one swallowed magnet travels beyond the stomach. They may attract across the thin walls of small bowel, causing ischemia, necrosis and perforation into the peritoneum. The radiologist or other healthcare worker seeing magnets in the abdomen on radiographs should consider the situation an emergency and recommend surgical treatment as soon as possible.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2595063PMC
February 2006

Multiple magnet ingestion alert.

Authors:
Alan E Oestreich

Radiology 2004 Nov;233(2):615

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http://dx.doi.org/10.1148/radiol.2332041446DOI Listing
November 2004

Gaucher disease: alendronate disodium improves bone mineral density in adults receiving enzyme therapy.

Blood 2004 Sep 9;104(5):1253-7. Epub 2004 Mar 9.

Division of Human Genetics, Children's Hospital Research Foundation ML 4006, Cincinnati OH 45229-3039, USA.

Symptomatic patients with Gaucher disease (GD) (acid beta-glucosidase [Gcase] deficiency) are treated with injectable human recombinant GCase. Treatment results in significant decreases in lipid storage in liver, spleen, and bone marrow, but the generalized osteopenia and focal bone lesions present in many adult patients are refractory to treatment. A double-blind, 2-arm, placebo-controlled trial of alendronate (40 mg/d) was performed in adults with GD who had been treated with enzyme for at least 24 months. Primary therapeutic endpoints were improvements in (1) bone mineral density (BMD) and content (BMC) at the lumbar spine, and (2) focal lesions in x-rays of long bones assessed by a blinded reviewer. There were 34 patients with GD type 1 (age range, 18-50 years) receiving enzyme therapy who were randomized for this study. After 18 months, DeltaBMD at the lumbar spine was 0.068 +/- 0.21 and 0.015 +/- 0.034 for alendronate and placebo groups, respectively (P =.001). Long-bone x-rays showed no change in focal lesions or bone deformities in any subject in either arm. Alendronate is a useful adjunctive therapy in combination with enzyme replacement therapy (ERT) for the treatment of GD-related osteopenia in adults, but it cannot be expected to improve focal lesions.
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http://dx.doi.org/10.1182/blood-2003-11-3854DOI Listing
September 2004

The acrophysis: a unifying concept for understanding enchondral bone growth and its disorders. II. Abnormal growth.

Authors:
Alan E Oestreich

Skeletal Radiol 2004 Mar 20;33(3):119-28. Epub 2003 Dec 20.

Department of Radiology #5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229-3039, USA.

In order to discuss and illustrate the effects common to normal and abnormal enchondral bone at the physes and at all other growth plates of the developing child, the term "acrophysis" was proposed. Acrophyses include the growth plates of secondary growth centers including carpals and tarsals and apophyses, and the growth plates at the nonphyseal ends of small tubular bones. Abnormalities at acrophyseal sites are analogous to those at the physeal growth plates and their metaphyses. For example, changes relating to the zone of provisional calcification (ZPC) are often important to the demonstration of such similarities. Lead lines were an early example of the concept of analogy from abnormality due to physeal and to acrophyseal disturbance. The ZPC is a key factor in understanding patterns of rickets and its healing. Examples (including hypothyroidism, scurvy and other osteoporosis, Ollier disease, achondroplasia, and osteopetrosis, as well as the family of frostbite, Kashin-Beck disease, and rat bite fever) illustrate the acrophysis principle and in turn their manifestations are explained by that principle.
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http://dx.doi.org/10.1007/s00256-003-0735-9DOI Listing
March 2004

The acrophysis: a unifying concept for enchondral bone growth and its disorders. I. Normal growth.

Authors:
Alan E Oestreich

Skeletal Radiol 2003 Mar 28;32(3):121-7. Epub 2003 Jan 28.

Department of Radiology #H-5031, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, Ohio 45229-3039, USA.

In order to discuss and illustrate the common effects on normal and abnormal enchondral bone at the physes and at all other growth plates of the developing child, the term "acrophysis" is proposed. Acrophyses include the growth plates of secondary growth centers including carpals and tarsals and apophyses, and the growth plates at the non-physeal ends of small tubular bones. The last layer of development of both physes and acrophysis is the cartilaginous zone of provisional calcification (ZPC). The enchondral bone abutting the ZPC shares similar properties at physes and acrophyses, including the relatively lucent metaphyseal bands of many normal infants at several weeks of age. The bone-in-bone pattern of the normal vertebral bodies and bands of demineralization of the tarsal bones just under the ZPC are the equivalent of those bands. The growth arrest/recovery lines of metaphyses similarly have equivalent lines in growth centers and other acrophyseal sites. Nearly the same effects can also be anticipated from the relatively similar growth plate at the cartilaginous cap of benign exostoses ("paraphysis"). The companion article will explore abnormalities at acrophyseal sites, including metabolic bone disease and dysplasias.
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http://dx.doi.org/10.1007/s00256-002-0604-yDOI Listing
March 2003

Prevalence of reduced bone mass in children and adults with spastic quadriplegia.

Dev Med Child Neurol 2003 Jan;45(1):12-6

Division of Pediatric Gastroenterology, Children's Hospital Medical Center, 3333 Burnet Ave, Cincinnati, OH 45229, USA.

This study was designed to test the hypothesis that non-ambulatory patients with spastic quadriplegia will have reduced bone mass which worsens with increasing age. Forty-eight patients (age 5 to 48 years, median age 15 years; 19 females and 29 males) were studied. Anticonvulsants were used in 29 patients (60.4%). Lumbar spine bone mineral density (LS-BMD) was markedly reduced compared with age-and sex-matched control individuals with a z score of -2.37 +/- 0.21. Twenty-eight (58%) had z scores of less than -2. A history of documented previous fracture was present in 19 patients (39%). Patients with a history of fracture had significantly lower (p = 0.05) LS-BMD z scores (-2.81 +/- 0.29) compared with those without a history of fracture (-2.11 +/- 0.26). Mean serum 25-OH vitamin D was 29.6 +/- 1.9ng/mL (normal 9 to 37.6ng/mL) with three patients having serum 25-OH vitamin D levels less than 15ng/mL. These findings indicate that BMD is markedly reduced in non-ambulatory children and adults with neuromuscular disease. Reductions in bone mass put them at greater risk for non-traumatic fractures.
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January 2003