Orthopaedics & Traumatology: Surgery & Research (2013) 99, 72—79

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ORIGINAL ARTICLE

Fracture following lower limb lengthening in

children: A series of 58 patients
F. Launay a,∗, R. Younsi a, M. Pithioux b, P. Chabrand b, G. Bollini a, J.-L. Jouve a

a
Timone Children Hospital, Department of Pediatric and Orthopaedic Surgery, 264, rue Saint-Pierre, 13385 Marseille cedex 5,
France
b
Interdisciplinary Study Group in Musculoskeletal and Cardiovascular Biomecanics, Human Motion Research Institute, UMR CNRS
7287, Aix Marseille University, Faculty of Sports Sciences CP 910, 163, avenue de Luminy, 13288 Marseille cedex 09, France

Accepted: 21 August 2012

KEYWORDS Summary
Lengthening; Introduction: Fracture is one of the main complications following external fixator removal used
Lower limb; in cases of progressive lower limb lengthening; rates as high as 50% are found in the literature.
Fracture; The aim of this study was to determine the factors influencing this complication.
Children; Materials and methods: One hundred and eleven cases of lower limb lengthening were per-
Limb length formed in 58 patients (40 femurs and 71 tibias). The mean age at surgery was 10.1 years old.
inequality Lengthening was performed in all cases with an external fixator alone, associated in 39.6% of
cases with intramedullary nailing. The patients were divided into three groups according to dis-
ease etiology (congenital, achondroplasia and other). The fractures were classified according
to the Simpson classification.
Results: Twenty fractures were recorded (18%). Sixteen fractures were found in patients with
congenital disease, four with achondroplasia and none in the group of other etiologies. The
fracture was more often in the femur (27.5%) than in the tibia (12.7%).
Discussion: The rate of fracture is influenced by different factors depending on the etiology of
disease. In congenital diseases, the fracture rate is higher when there is lengthening of more
than 15% of the initial length and a delay between surgery and the beginning of lengthening
of less than 7 days. In patients with achondroplasia, the influence of a relative percentage of
lengthening is less important than in those with congenital disease. However, to avoid fractures,
lengthening should not be started in children under the age of nine. Moreover, lengthening
should begin at least 7 days after the fixator has been placed.
Type of study: Retrospective.
Level of evidence: Level IV.
© 2012 Elsevier Masson SAS. All rights reserved.

∗ Corresponding author. Tel.: +33 04 91 38 69 05.

E-mail address: franck.launay@ap-hm.fr (F. Launay).

1877-0568/$ – see front matter © 2012 Elsevier Masson SAS. All rights reserved.
http://dx.doi.org/10.1016/j.otsr.2012.08.005
Fracture after lower limb lengthening in children 73

Introduction

Limb lengthening is associated with numerous

complications: infections [1,2], stiff joints [2,3], pseu-
darthrosis [2—4], early union [3,4] and neurological
sequella [3,4]. Fractures also occur after the fixator is
removed in 3—50% of the lengthened segments [1—5].
Although the principles of distraction are applied and
the callotasis technique has been popular since the
1950s, this rate remains high [6—8]. These principles
are essential to obtain optimal union of the distraction
site, and to limit the risk of fracture after removal of
the external fixator. Bone fixation must be stable [6—8],
while preserving the periosteum as much as possible
during the osteotomy [6—8], and a low energy osteotomy
should be performed [6—8], with a rhythm of lengthen-
ing of 0.5—2 mm/d [6—8] while the delay between the
osteotomy and the start of lengthening should be 4—15 days
[6—8].
Several non-invasive procedures may be used to evalu-
ate bone union and determine when the external fixator
should be removed, such as ultrasound, Dual Energy X-ray Figure 1 Lengthening of the femur with monolateral external
Absorptiometry (DEXA) osteodensitometry which provides fixator.
quantitative and qualitative evaluation of bone, as well as
digital X-ray and CT scan [9—16]. The fracture rate after
• Group 1: congenital lower limb length discrepancies, such
removal of the external fixator is reduced to a rate of 3.6%
as fibular hemimelia or congenital short fibula (58 length-
with the use of osteodensitometry [9,13]. However, in most
enings in 35 patients);
centers, the most common technique is standard AP X-ray
• Group 2: patients with achondroplasia or a similar disease
because it is simple to use, inexpensive and accessible.
operated on for their short stature (34 lengthenings in
Thus, based on the principles of Fischgrund, the fixator is
eight patients);
removed when three continuous cortices at least 2 mm thick
• Group 3: post-traumatic, post-infectious or post-tumoral
are present [17].
lower limb length discrepancies (disease on healthy bone)
The aim of our study was to analyze whether other factors
(19 lengthenings in 15 patients).
influence the risk of these fractures based on a retrospective
series of leg lengthening.
Lengthening was obtained with an external fixator in
all cases (Fig. 1): monolateral fixator in 38 lengthenings
(34.2%), circular in 68 lengthenings (61.3%), and hybrid in
Materials and methods five lengthenings (4.5%). A monolateral fixator was used in
all patients in group 2 (achondroplasia and associated dis-
Between 2000 and 2010, at the Timone Children’s Hospi- eases), bilateral in all cases and crossed lengthening was
tal in Marseille, 111 limb lengthenings were performed in never performed.
58 patients. The femur was lengthened in 40 cases (36%) Lengthening was associated with perioperative
and the tibia in 71 (64%). All clinical files and X-rays were intramedullary nailing (Kirchner wires of different diam-
retrospectively evaluated by an observer who was differ- eters, and Metaizeau nails (Fig. 2)) in 44 segments. The
ent from the practicing surgeon. Patient data was obtained osteotomy was performed with a drill and an osteoma by
from the clinical file (date of birth, sex, type of disease, the ‘‘postal stamp’’ technique (low energy osteotomy) in
limb operated on), the chronology of events in relation to 60 cases (54%) and with a Gigli saw (high energy osteotomy)
the procedure (date of surgery, date of removal of fixator, in 51 other cases (46%). The level of the osteotomy was
date of fracture), as well as the different complications the diaphysis in 92 segments (82.9%) and the metaphysis in
which occurred during distraction and their management. 19 segments (17.1%). The periosteum was opened and the
X-rays were analyzed to measure the initial length of the entire circumference was rasped to perform the osteotomy
lengthened limb, the length of regenerated bone at the end in all cases.
of distraction, the diameter of the lengthened bone at the After a mean delay of 7.2 days (2—14 days) between the
osteotomy site, the diameter of the regenerated bone at the date of surgery and the beginning of lengthening, the rhythm
end of distraction as well as to classify the fractures. Mea- of lengthening was 0.5—1 mm/d. The rigidity of the callus
surements were performed with a centimeter ruler in X-rays was evaluated with standard AP and profile X-rays based
before 2008 and on the computer (PACS) in X-rays performed on Fischgrund criteria (3/4 continuous cortices at least
after 2008. 2 mm thick), to determine when to remove the external
The mean age of patients at surgery was 10.1 years old fixator.
(range 2.1—20.3). Patients were divided into three groups After removal of the external fixator, 56 segments (50.4%)
according to the etiology of disease: were immobilized and 22 (19.8%) were allowed immediate
74
Figure 2 Lengthening of the femur with intramedullary nail-
ing.
partial or total postoperative weight-bearing; the remaining
cases only applied weight later. At the end of distraction,
the diameters and lengths were calculated and recorded
according to the following methods:
• recording of diameters: by comparing the smallest diame-
ter of the callus to the diameter of the former osteotomy
site on standard AP and/or profile X-rays (Fig. 3);
• recording of lengths: expressing the rate of lengthening
by comparing the length of regenerated bone to the initial
length of the lengthened limb (Fig. 4).
Figure 3 Diameters. This is the relationship between the
smallest diameter of regenerated bone (red line) and the diam-
eter of native bone at the osteotomy site (black line).
F. Launay et al.
Figure 4 Lengths. This is the relationship between the length
of the regenerated bone (red line) and the initial length of the
lengthened bone segment (black line).
All of the fractures were classified according to the Simp-
son classification [1] (Figs. 5—7).
Statistical analysis was performed using the Pearson
Chi2 test. When the theoretical participants were fewer
than five, the Fisher test was used. The Student-t test
was used for means. The differences were considered to
be statistically significant when the P value was less than
0.05.
Results
Absolute lengthening obtained was 5.3 cm (range 2—8.6 cm).
Relative lengthening in relation to the initial length of the
operated bone segment was 23% (range 6—57%). Lengthening
lasted a mean 65.7 days (28—152 days) and mean fixation was
228.5 days (98—448 days). The Healing Index was 45.1 days
per centimeter (21—102 d/cm).
There were 20 fractures/111 lengthenings (18%). The
distribution of fractures in relation to the lengthened
bone segments is found in Table 1. The mean delay
between removal of the external fixator and the fracture
was 63.2 days (0—547 days). Seventy-five percent of these
fractures (15/20) occurred within the first month. The distri-
bution of fractures in relation to the Simpson classification is
found in Table 2. Placement of intramedullary nails reduced
the number of fractures after removal of the external fix-
ator, but this was not significant (Table 3). Nevertheless,
it should be noted that the rate of infection at the site
of external fixation when intramedullary nails were used
(25 infections/44 lengthenings or 56.8%) was slightly higher
than the rate of infection without intramedullary nails (34
infections for 67 lengthenings or 50.7%). Once again the
difference was not statistically significant.
We found a greater number of femoral fractures in
patients in group 1 (congenital disease) (Table 4). We also
Fracture after lower limb lengthening in children 75

Figure 5 Simpson classification: Type 1A: deformity without any real continuity of the callus; Type 1B: fracture of the center of
the regenerated bone; Type 2: fracture of the bone/regenerate interface; Type 3: fracture at a distance from the callus in the same
bone segment; Type 4: fracture of another bone segment.

Table 1 Distribution of fractures in relation to the lengthened bone segment and the etiology of disease.

Number of lengthenings Number of fractures Percentage P

Femur 40 11 27.5 S
Tibia 71 9 12.7 P = 0.05
Group 1 58 16 27.6 S between G1 and G3
Group 2 34 4 11.8 (P = 0.01)
Group 3 19 0 0.0
S: significant; NS: non-significant; G1: Group 1; G2: Group 2.

Figure 6 Type 1B femoral fracture. Figure 7 Type 2 femoral fracture.

76
Table 2 Distribution of fractures by type.
Absolute number Percentage
Type 1A 5 25
Type 1B 3 15
Type 2 11 55
Type 3 1 5 These results included lengthening of lower and upper
Type 4 0 0 limbs.
noted that the number of fractures increased with the rel-
ative lengthening was more than 15% of the initial length
of the bone and when the delay between surgery and the
beginning of lengthening was more than 7 days (Table 4). By
combining a rate of lengthening of more than 15% and a delay
before beginning lengthening of no more than 7 days, we
identified 100% of the femoral fractures in group 1 (5 frac-
tures in 5 femurs). The difference was significant in relation
to the other femurs in group 1 (P = 0.01).
We also found more femoral fractures in the patients
in group 2 (achondroplasia) (Table 5). We noted that the
rate of fracture was higher in patients under the age of
nine and when the delay between surgery and the beginning
of lengthening was less than 7 days (Table 5). By com-
bining age less than nine and a delay between surgery
and the beginning of lengthening of less than 7 days, half
of the segments were fractured (4 fractures/8 segments
including 1/2 tibias and 3/6 femurs). The difference was
significant (P = 0.0001) in relation to the other segments in
group 2.
The presence of an infection during the lengthen-
ing program increased the number of fractures after
removal of the external fixator (Table 6). However, the
presence or absence of radiological signs of deep infec-
tion was not a predictive factor of the incidence of
fracture (Table 6).
Discussion
Lengthening and intramedullary nailing
In the past few years, the efficacy of stable elastic
intramedullary nailing has been reported in the treatment of
fractures in children [18,19]. Other authors have evaluated
the association of external fixation and rigid internal fixation
with rigid intramedullary nails [20—22]. Although this asso-
ciation has reduced the duration of fixation and the Healing
Index, several major complications were observed [20,21],
such as osteomyelitis, failure of the intramedullary nail or
the incarceration of the intramedullary nail.
The goal of flexible intramedullary nails is to improve
fixation of the bone callus and limit medullary destruction.
This was observed during lengthening of the upper limbs in
particular the forearm, in the study by Launay et al. [23],
with better union of the bone callus, shorter fixation, fewer
deformities and no infection.
In the studies by Lascombes et al. [24—26], the Heal-
ing Index was reduced in the group with intramedullary
nailing, with a reduction in fixation time and in the rate
of fractures. These results were significant in particular in
F. Launay et al.
patients with congenital diseases of the femur and the fore-
arm. No fractures occurred in their clinical series in 1994
[24], after lengthening in 14 segments (2 upper limbs and
12 lower limbs). The rate of fracture in their compara-
tive series [25] was lower in the group with intramedullary
nailing (1 type 1A fracture/92 cases in the group with
nailing and 21 fractures/194 cases in the group without).
In the series by Launay et al. [27], no fractures occurred
after removal of the external fixator for lengthening of seven
congenital femurs associating Flexible Intramedullary Nail-
ing (FIN) and a monolateral fixator.
In the present series, the Healing Index was reduced
in group 2 when intramedullary nailing was used, what-
ever the bone lengthened. The Healing Index of the femur
was also reduced in group 1 although the difference was
not significant. Fractures occurred in 6/44 bone segments
(13.6%) in the population with intramedullary nailing. Frac-
tures occurred in 14/67 segments (20.9%) in the population
without intramedullary nailing. Although the fracture rate
was reduced with intramedullary nailing, the difference
was not significant. This confirms the results described
above.
Lengthening in patients with achondroplasia
In patients with achondroplasia and associated diseases
(group 2), relative lengthening can be as high as 50% of
the lengthened segment without any increase in the frac-
ture rate. This may be explained by the elasticity of the
ligament and tendon in this disease [28—30]. In our series,
we did not observe any relationship between the rate of
lengthening and the fracture rate. On the other hand, we
did observe that the fracture rate was significantly higher
in patients who were under the age of nine (33.3%), com-
pared to patients who were older than nine (0%), P = 0.003.
We did not find any difference when we varied the age limit.
In their work on progressive lengthening in achondroplasia,
Aldegheri et al. suggest that lengthening should be per-
formed later, between 12 and 16 years old [8,28,29]. Ganel
et al. suggest that lengthening can be performed in boys at
8 years old in boys and at 15 in girls because union of the
callus is poorer in the latter [30]. The fractures in our series
were found in male patients, but there was no significant dif-
ference found in relation to female gender. In the literature,
the delay between the osteotomy and beginning lengthen-
ing is 4—15 days [6—8,27—29,31]. In the series by Aldegheri
et al. [29], a delay of 5 days was proposed in patients with
achondroplasia. We observed an increased risk of fracture
when the delay was less than 7 days (28.6%), while no frac-
tures were observed when the delay was more than 7 days
(P = 0.01). Moreover, the association of lengthening before
the age of nine and less than 7 days delay before beginning
lengthening resulted in a higher risk of fracture (1/2 seg-
ments were broken) and the statistical difference with the
rest of the population (age older than 9 and delay of less
than 7 days) was even greater P = 0.0001. We did not find any
association between the two parameters (age of the patient
and delay before lengthening) and the rate of fracture in
the literature.
Fracture after lower limb lengthening in children 77

Table 3 Distribution of fractures in relation to the presence of Flexible Intramedullary Nailing (FIN) (or stable elastic
intramedullary nailing).

Number of lengthenings Number of fractures Percentage P

With FIN 44 6 13.6 NS

Without FIN 67 14 20.9 P = 0.33
NS: non-significant.

Table 4 Distribution of fractures in group 1 (congenital diseases).

Congenital Number of lengthenings Number of fractures Percentage P

Femur 15 8 53.3 S
Tibia 43 8 18.6 P = 0.009
Lengthening ≤ 15% 13 1 7.7 NS
Lengthening > 15% 45 15 33.3 P = 0.06
Delay ≤ 7 days 21 9 42.9 S
Delay > 7 days 37 7 18.9 P = 0.04
S: significant; NS: non-significant.

Table 5 Distribution of fractures in group 2.

Achondroplasia Number of lengthenings Number of fractures Percentage P

Femur 18 3 16.7 NS
Tibia 16 1 6.3 P = 0.36
Patients < 9 years old 12 4 33.3 S
Patients > 9 years old 22 0 0.0 P = 0.003
Delay < 7 days 14 4 28.6 S
Delay > 7 days 20 0 0.0 P = 0.01
S: significant; NS: non-significant.

Table 6 Distribution of fractures depending on the presence or not of infection.

Infections Number of lengthenings Number of fractures Percentage P

Infected segments 59 15 25.4 S

Non-infected segments 52 5 9.6 P = 0.03
Infection w/ radiological sign 23 6 26.1 NS
Infection w/o radiological sign 36 9 25.0 P = 0.92
S: significant; NS: non-significant.

Lengthening in patients with congenital diseases
For Dahl et al. [32], the suggested rate of lengthening for
the fewest number of complications (all complications com-
bined, including fractures) should not be more than 15%.
For Maffulli et al. [33], it should be less than 18%, and 25%
for Karger et al. [34] and Antoci et al. [3,35]. The series
by Kamlesh et al. [36] found that all fractures occurred
in cases with lengthening of more than 30%. These results
apply, in particular to congenital diseases (fibular hemimelia
and congenital short femur) because in achondroplasia a
rate of lengthening as high as 50% or more can be applied
[27—29].
In our series, we did not find a relationship between
the rate of fractures and the rate of lengthening in the
achondroplasia and associated group, while fracture rate
of 33.3% (15/45) was found in the congenital disease group
when lengthening was more than 15%. On the other hand,
when lengthening was 15% or less, the fracture rate was
7.7% (1/13), although the difference was not significant
(P = 0.06).
We chose a threshold of 7 days for the delay between
surgery and lengthening in patients with congenital disease.
Nine fractures/21 segments (42.9%) were observed when the
delay was less than 7 days and 7/37 fractures when the delay
was 7 days or more. The difference was not significant. On
78
the other hand, by associating a rate of lengthening of more
than 15% and a delay of less than 7 days, we found frac-
tures in all femurs, or 5 fractures/5 femurs. The difference
was significant with the rest of the femurs in the congenital
disease group. P = 0.01. It should be noted that none of these
femurs had associated intramedullary nailing.
Conclusion
The rate of fracture after removal of external fixation in
a lengthening program is still high in particular in patients
with congenital length discrepancies of the lower limb.
There is no single rule to avoid fracture in all cases. How-
ever, certain elements such as relative lengthening, delay
between osteotomy and beginning lengthening or placement
of intramedullary nails can markedly reduce their occur-
rence.
Disclosure of interest
The authors declare that they have no conflicts of interest
concerning this article.
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