Femoral lengthening in achondroplasia

Venkatesh, K. P.; Modi, Hitesh N.; Devmurari, K.; Yoon, Ji Yeol; Anupama, B.; Song, Hae Ryong

doi:10.1302/0301-620x.91b12.22418

Cited by 45 publications

(41 citation statements)

References 16 publications

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“…While the underlying diagnoses, limb segments lengthened, and devices used for lengthening were available for all subjects, other variables such as patient age, sex, preoperative height, and length gained were not found on a consistent basis for all studies. 7 of the 18 studies dealt with tibial lengthening only (Cattaneo et al 1988, Noonan et al 1997, McAllister et al 1999, Hahn et al 2003, Catagni et al 2005, Vaidya et al 2006, Park et al 2008), 2 studies (Venkatesh et al 2009, Kim et al 2012a) dealt with femoral lengthening alone, and the remaining 9 studies included patients undergoing combined tibial and femoral lengthening procedures. The average age of the patients at surgery was 16.3 (4–47) years and the mean follow-up time was 4.3 (0.8–16) years.…”

Section: Resultsmentioning

confidence: 99%

“…It has been suggested that patients with achondroplasia tolerate lengthening very well because of their ligament and soft tissue laxity, and their muscle length exceeds bone length before lengthening (Paley 1988). Lengthening of more than 30% was often reported in A/H patients (Aldegheri et al 1988, McAllister et al 1999, Venkatesh et al 2009, Devmurari et al 2010). In the pooled analysis, the mean gain in height (10cm) and LP (36%) was greater in patients with A/H than in patients with other diagnoses.…”

Section: Discussionmentioning

confidence: 99%

“…Recently, in a separate study that was not included in our analysis (due to lack of documented complications), over half of skeletally immature patients with achondroplasia who underwent DO for stature lengthening had substantial growth inhibition and premature physeal closure of the lengthened femurs and tibias (Song et al 2012). Increasing the magnitude of lengthening is also associated with other complications such as adjacent joint stiffness and fractures (Venkatesh et al 2009). Various authors have explored the complications of extensive limb lengthening and attempted to assess patient satisfaction (Lavini et al 1990, Aldegheri and Dall’Oca 2001).…”

Section: Discussionmentioning

confidence: 99%

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The etiology of short stature affects the clinical outcome of lower limb lengthening using external fixation

2014

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Background and purposeDistraction osteogenesis (DO) has been used to gain height in short statured individuals. However, there have been no studies comparing the clinical outcome of limb lengthening based on the etiology of the short stature. We assessed whether different underlying diagnoses are associated with varied clinical outcomes in these patients.MethodsWe performed a systematic review of the literature pertaining to lower limb lengthening using external fixation for short stature. Clinical outcomes including amount of lengthening, healing index (HI), and complications based on the underlying diagnosis for the short stature were documented.Results18 clinical studies were included, with 547 patients who underwent 1,581 lower limb segment lengthening procedures. Mean follow-up was 4.3 years. The average age at lengthening was less for individuals with achondroplasia/hypochondroplasia (A/H) (14.5 years) than for those with Turner’s syndrome (TS) (18.2 years) or with constitutional short stature (CSS) (21.7 years). Mean height gained was greater in patients with A/H (9.5 cm) than in those with TS (7.7 cm) or CSS (6.1 cm) group. The HI was better in A/H (30.8 days/cm) and CSS (32 days/cm) than in TS (45.1 days/cm). The reported complication rate per segment was lower for A/H (0.68) and TS (0.71) than for CSS (1.06).InterpretationPatients with A/H tolerated larger amounts of lengthening with fewer complications than those with other diagnoses.

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Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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The etiology of short stature affects the clinical outcome of lower limb lengthening using external fixation

2014

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“…We have previously recommended temporary cessation of distraction and the application of gradual compression when concave, lateral or central callus shapes appear in the regenerate bone, until satisfactory regenerate with fusiform callus develops. 12 Some of the dissatisfaction expressed by patients with a lengthened lower limb may be attributed to the effects of upper-and lower-limb mismatch. 25,26 Someone with short arms and lengthened lower limbs will have difficulty in donning and removing socks, shoes or underwear.…”

Section: Discussionmentioning

confidence: 99%

“…11 We have also suggested that careful radiological assessment of the patterns of callus formation is a useful method for the evaluation and monitoring of regenerate bone. 12 Using these methods, we wished to investigate the difference in callus maturation between humeral lengthening and femoral lengthening.…”

mentioning

confidence: 99%

Comparison between upper and lower limb lengthening in patients with achondroplasia

Song

et al. 2012

The Journal of Bone and Joint Surgery. British volume

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Lengthening of the humerus is now an established technique. We compared the complications of humeral lengthening with those of femoral lengthening and investigated whether or not the callus formation in the humerus proceeds at a higher rate than that in the femur. A total of 24 humeral and 24 femoral lengthenings were performed on 12 patients with achondroplasia. We measured the pixel value ratio (PVR) of the lengthened area on radiographs and each radiograph was analysed for the shape, type and density of the callus. The quality of life (QOL) of the patients after humeral lengthening was compared with that prior to surgery. The complication rate per segment of humerus and femur was 0.87% and 1.37%, respectively. In the humerus the PVR was significantly higher than that of the femur. Lower limbs were associated with an increased incidence of concave, lateral and central callus shapes. Humeral lengthening had a lower complication rate than lower-limb lengthening, and QOL increased significantly after humeral lengthening. Callus formation in the humerus during the distraction period proceeded at a significantly higher rate than that in the femur. These findings indicate that humeral lengthening has an important role in the management of patients with achondroplasia.

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Birth prevalence of achondroplasia: A systematic literature review and meta‐analysis

Foreman

Kessel²,

Hoorn³

et al. 2020

American J of Med Genetics Pt A

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Achondroplasia is a genetic disorder that results in disproportionate short stature. The true prevalence of achondroplasia is unknown as estimates vary widely. This systematic literature review and meta‐analysis was conducted to better estimate worldwide achondroplasia birth prevalence. PubMed, Embase, Scielo, and Google Scholar were searched, complemented by manual searching, for peer‐reviewed articles published between 1950 and 2019. Eligible articles were identified by two independent researchers using predefined selection criteria. Birth prevalence estimates were extracted for analysis, and the quality of evidence was assessed. A meta‐analysis using a quality effects approach based on the inverse variance fixed effect model was conducted. The search identified 955 unique articles, of which 52 were eligible and included. Based on the meta‐analysis, the worldwide birth prevalence of achondroplasia was estimated to be 4.6 per 100,000. Substantial regional variation was observed with a considerably higher birth prevalence reported in North Africa and the Middle East compared to other regions, particularly Europe and the Americas. Higher birth prevalence was also reported in specialized care settings. Significant heterogeneity (Higgins I2 of 84.3) was present and some indication of publication bias was detected, based on visual asymmetry of the Doi plot with a Furuya‐Kanamori index of 2.73. Analysis of pooled data from the current literature yields a worldwide achondroplasia birth prevalence of approximately 4.6 per 100,000, with considerable regional variation. Careful interpretation of these findings is advised as included studies are of broadly varying methodological quality.

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Femoral lengthening in achondroplasia

Cited by 45 publications

References 16 publications

The etiology of short stature affects the clinical outcome of lower limb lengthening using external fixation

The etiology of short stature affects the clinical outcome of lower limb lengthening using external fixation

Comparison between upper and lower limb lengthening in patients with achondroplasia

Birth prevalence of achondroplasia: A systematic literature review and meta‐analysis

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