Hypertrophic Callus Formation Leading to High-Output Cardiac Failure in a Patient with Osteogenesis Imperfecta

Burke, Teresa; Crerand, S.; Dowling, F

doi:10.1097/01241398-198809000-00022

Cited by 9 publications

(5 citation statements)

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“…summarizes reported short‐ and long‐term complications of bisphosphonate treatment 19–54 . A complete list of all relevant studies considered in this systematic review is provided in Table SII (supporting information published online) 7,11–28,32,33,35–45,47–80 …”

Section: Methods Of Reviewmentioning

confidence: 99%

Effects of bisphosphonates in children with osteogenesis imperfecta: an AACPDM systematic review

Castillo

Samson‐Fang

2008

Develop Med Child Neuro

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This systematic review of the effects of bisphosphonate treatment in children with osteogenesis imperfecta was conducted using the American Academy for Cerebral Palsy and Developmental Medicine methodology for developing systematic reviews of treatment interventions (Revision 1.1) 2004. Despite a large body of published literature, there have been only eight studies with a sufficiently high level of internal validity to be truly informative. These studies confirm improvement in bone density. Many, but not all studies, demonstrate reduction in fracture rate and enhanced growth. There has been extremely limited evaluation of broader treatment impacts such as deformity, need for orthopedic surgery, pain, functioning, or quality of life. Short-term side effects were minimal. Which medication and dosing regimen is optimal and how long patients should be treated are unclear. This body of evidence would be strengthened by a larger controlled trial, because many studies lacked adequate power to evaluate stated outcomes. These studies do not address the impacts of bisphosphonates in children with milder forms of osteogenesis imperfecta and severe forms that are not due to mutations in the type I pro-collagen gene (e.g. types VII and VIII). Additional research is needed into treatment of infants. More studies evaluating medication choices, optimal dosing, duration of treatment, post-treatment impacts, and long-term side effects are necessary.

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Section: Methods Of Reviewmentioning

confidence: 99%

Effects of bisphosphonates in children with osteogenesis imperfecta: an AACPDM systematic review

Castillo

Samson‐Fang

2008

Develop Med Child Neuro

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“…Hyperplastic callus formation in OI was first described more than 90 years ago (6) . Although generally believed to be rare, (7) a number of case reports can be found in the literature (5,8–13) . The incidence of hyperplastic callus among OI patients is unknown, but in a series of 60 type IV patients, 10 (17%) had developed at least one hyperplastic callus before the age of 20 years (14) .…”

Section: Introductionmentioning

confidence: 99%

Type V Osteogenesis Imperfecta: A New Form of Brittle Bone Disease

Glorieux

Rauch

Plotkin

et al. 2000

Journal of Bone and Mineral Research

456

300

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Osteogenesis imperfecta (OI) is commonly subdivided into four clinical types. Among these, OI type IV clearly represents a heterogeneous group of disorders. Here we describe 7 OI patients (3 girls), who would typically be classified as having OI type IV but who can be distinguished from other type IV patients. We propose to call this disease entity OI type V. These children had a history of moderate to severe increased fragility of long bones and vertebral bodies. Four patients had experienced at least one episode of hyperplastic callus formation. The family history was positive for OI in 3 patients, with an autosomal dominant pattern of inheritance. All type V patients had limitations in the range of pronation/supination in one or both forearms, associated with a radiologically apparent calcification of the interosseous membrane. Three patients had anterior dislocation of the radial head. A radiodense metaphyseal band immediately adjacent to the growth plate was a constant feature in growing patients. Lumbar spine bone mineral density was low and similar to age-matched patients with OI type IV. None of the type V patients presented blue sclerae or dentinogenesis imperfecta, but ligamentous laxity was similar to that in patients with OI type IV. Levels of biochemical markers of bone metabolism generally were within the reference range, but serum alkaline phosphatase and urinary collagen type I N-telopeptide excretion increased markedly during periods of active hyperplastic callus formation. Qualitative histology of iliac biopsy specimens showed that lamellae were arranged in an irregular fashion or had a meshlike appearance. Quantitative histomorphometry revealed decreased amounts of cortical and cancellous bone, like in OI type IV. However, in contrast to OI type IV, parameters that reflect remodeling activation on cancellous bone were mostly normal in OI type V, while parameters reflecting bone formation processes in individual remodeling sites were clearly decreased. Mutation screening of the coding regions and exon/intron boundaries of both collagen type I genes did not reveal any mutations affecting glycine codons or splice sites. In conclusion, OI type V is a new form of autosomal dominant OI, which does not appear to be associated with collagen type I mutations. The genetic defect underlying this disease remains to be elucidated. (J Bone Miner Res 2000;15:1650 -1658)

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“…Hyperplastic callus formation is a rare, but wellrecognized complication of OI [8,9,[12][13][14][15]25,30,31]. Initially, it was described as a result of subperiosteal hemorrhages in scurvy [31,32].…”

Section: Discussionmentioning

confidence: 99%

“…weight loss, malaise, dilatation of the veins, and pain to palpation and motion [4,7,10,13]. This clinical situation occurs with alarming rapidity in a few weeks, with or without a fracture or surgery correlation [7,16].…”

Section: Discussionmentioning

confidence: 99%

“…Immobilization with radiological follow-up, [9], adrenocorticotropic hormone therapy [8,13,14] or radiotherapy at early stages, [8,10,[12][13][14]16,30,36] have been proposed by several different authors as treatments to obtain symptomatic improvement. Immobilization with radiological follow-up, [9], adrenocorticotropic hormone therapy [8,13,14] or radiotherapy at early stages, [8,10,[12][13][14]16,30,36] have been proposed by several different authors as treatments to obtain symptomatic improvement.…”

Section: Discussionmentioning

confidence: 99%

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Osteogenesis imperfecta and hyperplastic callus formation in a family: a report of three cases and a review of the literature

Ramírez

Vilella²,

Colón

et al. 2003

Journal of Pediatric Orthopaedics, Part B

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Osteogenesis imperfecta is one of the most common groups of inherited disorders of connective tissue. Hyperplastic callus formation in patients with osteogenesis imperfecta after fracture or surgery is a rare occurrence that has often been misdiagnosed as osteosarcoma. Previous series reported that hyperplastic callus formation is more often present in osteogenesis imperfecta male patients, with white sclerae, and a negative family history of the disorder. This is the first time that this complication has been presented in three female siblings, with a positive family history of osteogenesis imperfecta type IV. An association between osteogenesis imperfecta type IV and hyperplastic callus formation is unclear. This association might, however, be a separate, specific subtype of osteogenesis imperfecta, with an unknown inheritance pattern.

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Hypertrophic Callus Formation Leading to High-Output Cardiac Failure in a Patient with Osteogenesis Imperfecta

Cited by 9 publications

References 0 publications

Effects of bisphosphonates in children with osteogenesis imperfecta: an AACPDM systematic review

Effects of bisphosphonates in children with osteogenesis imperfecta: an AACPDM systematic review

Type V Osteogenesis Imperfecta: A New Form of Brittle Bone Disease

Osteogenesis imperfecta and hyperplastic callus formation in a family: a report of three cases and a review of the literature

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