Physeal growth arrest after tibial lengthening in achondroplasia

Song, Sang Heon; Agashe, Mandar; Huh, Young Jae; Hwang, Soon Young; Song, Hae Ryong

doi:10.3109/17453674.2012.678802

Cited by 15 publications

(5 citation statements)

References 28 publications

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“…They showed that continuous compressive forces on the physis result in either temporary or permanent cessation of growth according to the Heuter-Volkmann principle. Sang-Heaon et al in their study showed the growth arrest on the tibia, especially on the medial portion of the proximal physis [22]. They also suggested starting lengthening in achondroplasia patients after 11 years old in their population when the growth of proximal tibia physeal decrease to less than 10 mm/years.…”

Section: Discussionmentioning

confidence: 91%

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Does the technique of limb lengthening affect physeal growth in patient with achondroplasia? Comparison of the simultaneous and consecutive tibia and femur lengthening with external fixators

Balcı

Anarat

Bayram

et al. 2023

Journal of Pediatric Orthopaedics B

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We aimed to retrospectively investigate and compare patients who underwent bilateral simultaneous and consecutive lengthening surgery of the femur and tibia in terms of the effect on physeal growth. We hypothesize that compared with the sequential lengthening technique, simultaneous lengthening of the bilateral femur and tibia affects physeal growth to a greater extent. Twentysix patients with achondroplasia who operated in our clinic between 1995 and 2015 for limb lengthening were included in the study. Fourteen patients with bilateral lengthening of the femur and tibia at the same time were named as simultaneous bilateral lengthening (SBL). Twelve patients with bilateral lengthening of the femur and then bilateral tibia lengthening in other sections or vice versa, at a different time was named consecutive bilateral lengthening (CBL). All patients were followed until at least 18 years old. The physeal arrest was measured with predicted final length at the first visit (investigated with the multiplier method), the total amount of lengthening, and final clinical, and radiological length. Mean limb lengthening was 145 mm (48.5%) and 151 mm (46.6%) for simultaneous and consecutive groups respectively. For lower extremity length, the first group reached 527.6 mm while the expected was 447.3 mm. Considering 151 mm lengthening, the mean growth disturbance for the SBL group was 70.7 mm and for the CBL group was 47.5 mm. For total height comparing between two groups, disturbance for height was 80.5 mm and 65.4 mm, respectively. Although there was no statistical difference, simultaneous bilateral femoral and tibial lengthening has more physiological physeal disturbance effects than consecutive lengthening in patients with achondroplasia.

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Section: Discussionmentioning

confidence: 91%

“…Sang-Heaon et al . in their study showed the growth arrest on the tibia, especially on the medial portion of the proximal physis [22]. They also suggested starting lengthening in achondroplasia patients after 11 years old in their population when the growth of proximal tibia physeal decrease to less than 10 mm/years.…”

Section: Discussionmentioning

confidence: 99%

Does the technique of limb lengthening affect physeal growth in patient with achondroplasia? Comparison of the simultaneous and consecutive tibia and femur lengthening with external fixators

Balcı

Anarat

Bayram

et al. 2023

Journal of Pediatric Orthopaedics B

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“…Out of the 58 included studies, four were randomised control trials [ 22 , 32 , 53 , 54 ], 25 were retrospective case studies [ 5 , 23 – 25 , 27 , 28 , 30 , 34 , 35 , 39 , 41 , 42 , 45 , 46 , 48 , 49 , 57 , 59 , 65 – 69 , 71 , 73 ], 15 were retrospective cohort studies [ 20 , 21 , 26 , 29 , 31 , 36 , 38 , 40 , 47 , 55 , 58 , 60 , 61 , 70 , 74 ], eight were controlled before and after studies [ 15 , 51 , 52 , 56 , 62 – 64 , 72 ], two were prognostic studies [ 19 , 37 ], and one study was a non-randomised control trial [ 50 ]. There were three cross-sectional studies that used patient focused surveys [ 33 , 43 , 44 ].…”

Section: Resultsmentioning

confidence: 99%

Interventions for improving clinical outcomes and health-related quality-of-life for people living with skeletal dysplasias: an evidence gap map

Moy¹,

Flynn²,

Henríquez³

et al. 2023

Qual Life Res

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Purpose Skeletal dysplasias are rare genetic disorders that are characterized by abnormal development of bone and cartilage. There are multiple medical and non-medical treatments for specific symptoms of skeletal dysplasias e.g. pain, as well as corrective surgical procedures to improve physical functioning. The aim of this paper was to develop an evidence-gap map of treatment options for skeletal dysplasias, and their impact on patient outcomes. Methods We conducted an evidence-gap map to identify the available evidence on the impact of treatment options on people with skeletal dysplasias on clinical outcomes (such as increase in height), and dimensions of health-related quality of life. A structured search strategy was applied to five databases. Two reviewers independently assessed articles for inclusion in two stages: titles and abstracts (stage 1), and full text of studies retained at stage 2. Results 58 studies fulfilled our inclusion criteria. The included studies covered 12 types of skeletal dysplasia that are non-lethal with severe limb deformities that could result in significant pain and numerous orthopaedic interventions. Most studies reported on the effect of surgical interventions (n = 40, 69%), followed by the effect of treatments on dimensions of health quality-of-life (n = 4, 6.8%) and psychosocial functioning (n = 8, 13.8%). Conclusion Most studies reported on clinical outcomes from surgery for people living with Achondroplasia. Consequently, there are gaps in the literature on the full range of treatment options (including no active treatment), outcomes and the lived experience of people living with other skeletal dysplasias. More research is warranted to examine the impact of treatments on health-related quality-of-life of people living with skeletal dysplasias, including their relatives to enable them to make preference- and valued based decisions about treatment.

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“…Increased height and longer arms may also reduce some of the limitations in mobility, activities of daily living, and independence that have been reported for adults with achondroplasia [ 3 , 24 ]. Furthermore, improvements in growth may reduce the need for surgical lengthening of lower and upper limbs, which together can take several years to complete [ 29 , 30 ] and may have a substantial negative impact on the patient’s HRQoL during that period.…”

Section: Discussionmentioning

confidence: 99%

Literature review and expert opinion on the impact of achondroplasia on medical complications and health-related quality of life and expectations for long-term impact of vosoritide: a modified Delphi study

Savarirayan

Baratela

Butt

et al. 2022

Orphanet J Rare Dis

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Background Achondroplasia is associated with disproportionate short stature and significant and potentially severe medical complications. Vosoritide is the first medicine to treat the underlying cause of achondroplasia and data from phase 3 and phase 2 extension studies showed effects on growth and body proportions. However, there are currently no long-term data available on the direct impact on endpoints such as medical complications and health-related quality of life (HRQoL). This study explored the perceived impact of achondroplasia on medical complications, HRQoL, healthcare resource use and mortality, and potential modifying effects of vosoritide, based on published evidence and expert opinion. Structured expert opinion was obtained by an international modified Delphi study among 14 experts in managing achondroplasia performed on a virtual platform and consisting of an explorative phase followed by an anonymous individual rating round. Results Overall, the panelists expect that in individuals starting long-term treatment between 2 years of age and puberty, growth velocity increases observed in the clinical trials will be maintained until final height is reached (92% agreement) and will likely result in clinically meaningful improvements in upper-to-lower body segment ratio (85%). Earlier treatment initiation will likely result in a greater final height (100%) and more likely improve proportionality (92%) than later treatment. Although current data are limited, ≥ 75% of panelists find it conceivable that the earlier long-term treatment is started, the greater the probability of a positive effect on the lifetime incidence of symptomatic spinal stenosis, kyphosis, obstructive sleep apnea, and foramen magnum stenosis. These are among the most clinically important complications of achondroplasia because of their high impact on comorbidity, mortality, and/or HRQoL. A positive effect of vosoritide on the incidence of surgeries through lifetime was considered more likely with earlier long-term treatment (90%). Conclusions This explorative study, based on international expert opinion, provides further insight into the medical and functional impacts of achondroplasia and how these might be modified through long-term use of vosoritide. The results can be used to guide the direction and design of future research to validate the assumptions and to discuss potential treatment outcomes with disease modifying therapies with families and clinicians.

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Physeal growth arrest after tibial lengthening in achondroplasia

Cited by 15 publications

References 28 publications

Does the technique of limb lengthening affect physeal growth in patient with achondroplasia? Comparison of the simultaneous and consecutive tibia and femur lengthening with external fixators

Does the technique of limb lengthening affect physeal growth in patient with achondroplasia? Comparison of the simultaneous and consecutive tibia and femur lengthening with external fixators

Interventions for improving clinical outcomes and health-related quality-of-life for people living with skeletal dysplasias: an evidence gap map

Literature review and expert opinion on the impact of achondroplasia on medical complications and health-related quality of life and expectations for long-term impact of vosoritide: a modified Delphi study

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