Topic
Hyperplastic callus formation
About: Hyperplastic callus formation is a research topic. Over the lifetime, 28 publications have been published within this topic receiving 513 citations.
Papers
TL;DR: Hyperplastic callus formation was assessed in patients with osteogenesis imperfecta type V and occurred predominantly during phases of rapid growth.
Abstract: Hyperplastic callus formation was assessed in 23 patients with osteogenesis imperfecta type V. Hyperplastic callus mostly affected long bones in the lower extremities and occurred predominantly during phases of rapid growth.
Introduction: Hyperplastic callus (HPC) formation is one of the most conspicuous features of osteogenesis imperfecta (OI) type V, but the natural history of HPC has not been well characterized.
Materials and Methods: In this retrospective single-center study, we assessed HPC in 23 OI type V patients (9 females and 14 males).
Results: Fifteen patients (65%) had HPC at 48 skeletal sites, 30 of which affected the lower limbs. The number of HPC sites per patient ranged from 0 to 7, with an average of 2.6 for men and 1.1 for women (p = 0.047 for this sex difference; t-test). New HPC formation was observed both after fractures and outside of the context of fractures. Only a minority of lower limb fractures (26%) precipitated HPC formation. After an initial enlargement phase, HPC lesions usually stabilized, but could also resolve completely (n = 2) or progress and lead to bone deformation. The most common complication of HPC was a fracture through the lesion (n = 7). Neither pamidronate nor indomethacin seemed to influence the course of HPC.
Conclusions: HPC is a potentially serious complication of OI type V. Given the rarity of the disorder, treatment studies will require multicenter collaborations.
78 citations
TL;DR: In this paper, two girls with non-familial osteogenesis imperfecta who subsequently developed osteosarcoma of the femur were described and it was suggested that the tumours arose spontaneously and were not related to the underlying bone disorder.
Abstract: 1. Two girls with non-familial osteogenesis imperfecta who subsequently developed osteosarcoma of the femur are described. One is of special interest in that there were multiple bone metastases. 2. It is suggested that the tumours arose spontaneously and were not related to the underlying bone disorder. 3. Because of the relative frequency of hyperplastic callus formation in osteogenesis imperfecta it is most important that adequate biopsy material of any suspicious lesion is examined because the early clinical picture may be indistinguishable from a tumour.
44 citations
TL;DR: The importance of accurate diagnosis is stressed since, in the twenty-one cases reported in the literature, nine amputations were advised and three were actually performed for what were later proved to be benign conditions.
Abstract: Massive callus formation following fracture in osteogenesis imperfecta may on rare occasions be so exuberant as to produce a clinical picture indistinguishable from osteogenic sarcoma. Recent case reports of proved sarcomatous change in two cases of osteogenesis imperfecta reaffirm the necessity for accurate diagnosis. Twenty-three reported cases of massive callus formation without sarcoma are reviewed and two cases are added.
The only abnormal laboratory values even in advanced cases are elevated sedimentation rate and elevated serum alkaline phosphatase. Radiation therapy afforded rapid symptomatic relief in one patient and in the other it was given early in the process and seemed to halt further exuberant callus formation. Reconstructive surgery may be useful. It was carried out twice in one patient without eliciting abundant callus formation.
Biopsy be necessary on occasion. The importance of accurate diagnosis is stressed since, in the twenty-one cases reported in the literature, nine amputations were advised and three were actually performed for what were later proved to be benign conditions.
37 citations
TL;DR: The authors analyzed tissue and cells from a stationary and a rapidly growing hyperplastic callus from a patient with osteogenesis imperfecta (OI) type IV and compared the results with those of compact bone and skin fibroblasts of an age-matched control.
Abstract: We analyzed tissue and cells from a stationary and a rapidly growing hyperplastic callus from a patient with osteogenesis imperfecta (OI) type IV and compared the results with those of compact bone and skin fibroblasts of an age-matched control. Collagen and protein contents per cell were low in the callus tissues and collagen I and III were overmodified as evidenced by an elevated level of hydroxylysine. The degree of lysyl hydroxylation was highest in those regions that appeared most immature by histological examination. Lysyl hydroxylation approached normal levels in collagen from the stationary callus and from the center of the growing callus. Overmodification of collagen was not seen in compact bone or cell cultures (neither skin fibroblasts nor callus cells) from the patient. Elevation of hydroxylysine in collagen from OI patients is generally attributed to mutations that delay triple helix formation. Our observations suggest that the varying degree of collagen modifications may occur in consequence of regulatory mechanisms during bone development and tissue repair. These mechanisms may be defective in some patients with OI as seen in this case with hyperplastic callus formation.
37 citations
TL;DR: A biopsy is critical to establish the differential diagnosis between osteosarcoma and common hyperplastic callus formation in OI; however, it must be applied with great care.
Abstract: We describe a case history of a 24-year-old male with osteogenesis imperfecta (OI) who developed osteosarcoma of the left thigh. High-dose ifosfamide therapy caused marked tumor regression of multiple lung metastases. Immunohistochemically, the tumor cells were diffusely positive for the p53 protein. Mutation of the p53 gene was not detected by direct genomic sequencing of exons 4–8. The radiographic characteristics, including irregularly distributed osteolytic lesions and cortical discontinuity, should not be confused with hyperplastic callus formation, a benign process. A biopsy is critical to establish the differential diagnosis between osteosarcoma and common hyperplastic callus formation in OI; however, it must be applied with great care.
36 citations
Related Topics (5)
Performance Metrics
| Year | Papers |
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| 2021 | 1 |
| 2018 | 1 |
| 2016 | 1 |
| 2008 | 1 |
| 2007 | 1 |
| 2006 | 1 |