Acta Orthopaedica 2014; 85 (2): 181–186 181
The etiology of short stature affects the clinical outcome of
lower limb lengthening using external fixation
A systematic review of 18 trials involving 547 patients
Seung-Ju Kim, Wendy Pierce, and Sanjeev Sabharwal
Department of Orthopaedics, New Jersey Medical School, Newark, NJ, USA.
Correspondence S-J.K: firstname.lastname@example.org
Submitted 13-05-29. Accepted 13-11-22
Open Access - This article is distributed under the terms of the Creative Commons Attribution Noncommercial License which permits any noncommercial use,
distribution, and reproduction in any medium, provided the source is credited.
Background and purpose — Distraction 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.
Methods — We 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.
Results — 18 clinical studies were included, with 547 patients
who underwent 1,581 lower limb segment lengthening proce-
dures. Mean follow-up was 4.3 years. The average age at length-
ening was less for individuals with achondroplasia/hypochondro-
plasia (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).
Interpretation — Patients with A/H tolerated larger amounts
of lengthening with fewer complications than those with other
Distraction osteogenesis (DO) uses a corticotomy or osteot-
omy to allow formation of new bone in the gap created by
controlled distraction of the bone segments. In 1905, Codiv-
illa proposed limb lengthening through distraction (Codivilla
2008), and this concept was reintroduced in 1969 by Ilizarov
(Ilizarov and Deviatov 1969) and in 1977 by Wagner (1977)
using external fixation (EF). Since then, DO has been used
to address non-unions, skeletal defects, and limb deformities,
including limb length discrepancy (Aronson 1997, Liu et al.
More recently, this surgical modality has been used to
increase standing height in individuals of short stature (Kocze-
wski et al. 2002). Although there are several challenges asso-
ciated with DO such as length of treatment, the psychologi-
cal consequences of treatment, and permanent complications,
with advances in DO—including the availability of newer
implants and techniques—more individuals with a variety of
underlying diagnoses will undergo limb lengthening. How-
ever, to our knowledge, there have been no large comparative
studies on the clinical outcome of lower limb lengthening for
short stature based on the underlying diagnosis.
We performed a systematic review of the literature on lower
limb lengthening and assessed whether there were any differ-
ences in the reported clinical parameters, such as lengthening
percentage (LP) and healing index (HI), based on the etiol-
ogy of short stature. From previous literature (Paley 1988), we
hypothesized that patients with achondroplasia and hypochon-
droplasia would have undergone greater degrees of lengthen-
ing with fewer reported complications than those with other
diagnoses for their short stature.
Patients and methods
Search strategy and criteria
On October 18, 2012, we searched PubMed, MEDLINE, the
Cumulative Index to Nursing and Allied Health (CINAHL),
and the Cochrane Library for articles published between 1950
and 2012. The search terms used were “stature lengthen-
ing”, “height lengthening”, “limb lengthening”, and “distrac-
tion osteogenesis”. After discarding all duplicate articles and
restricting the search to manuscripts available in the English
182 Acta Orthopaedica 2014; 85 (2): 181–186
language and pertaining to human subjects, 2 of us (SJK, WP)
reviewed each remaining abstract manually. The search was
further narrowed to include only those articles that focused
on bilateral lower limb lengthening for short stature using EF
only and that included the patient’s underlying diagnosis and
limb segment(s) (femur/tibia) that were lengthened (Figure).
An orthopedic surgeon (SJK) and an orthopedic resident (WP)
independently assessed the eligibility of all relevant studies
based on our inclusion criteria. Disagreements between the
reviewers were resolved by discussion. The inclusion crite-
ria included (1) full-length articles published from January
1, 1950 to August 18, 2012, (2) articles written in the Eng-
lish language about human subjects, (3) cases of lengthening
using EF only, and (4) articles that documented complications.
The exclusion criteria included (1) cases of lengthening over
nail (LON), (2) review articles focusing on the technique of
lengthening, (3) articles without clinical outcomes such as LP
and HI, and (4) evaluation of any segment other than the lower
limb (for example, lengthening of the humerus). In order to
minimize bias related to the technique of DO, we excluded
patients who had undergone lengthening involving intramed-
ullary (IM) nails, with or without EF. Limits regarding the
number of patients in each study or the minimum duration
of follow-up were not used. All study designs of levels I–IV
were included. Bibliographies of the studies included were
also searched to identify other relevant studies. Based on the
above search criteria, 18 clinical studies were available for the
final analysis (Table 1).
Relevant information such as level of evidence, patient demo-
graphics, limb segment lengthened, preoperative height,
device(s) used for limb lengthening, amount of lengthening,
LP (final regenerate length divided by initial bone length,
multiplied by 100) (Givon et al. 2001), HI (number of days
to union divided by the final regenerate length in cm) (Kim
et al. 2011), any reported complications, and mean follow-up
period were extracted from each of these studies. Clinical out-
come measures varied among the studies.
The patient’s underlying diagnosis was categorized under
one of the following categories: achondroplasia/hypochon-
droplasia (A/H), Turner’s syndrome (TS), and constitutional
short stature (CSS). The patients with body dysmorphism/
normal height were excluded because most of them underwent
LON. Achondroplasia and hypochondroplasia were integrated
in 1 group because it is often difficult to clearly discriminate
achondroplasia patients from hypochondroplasia patients
clinically, and patients with these 2 diagnoses are reported
together in most reports (Ng et al. 2003). If there was a chance
that the same patients (patients who were reported in 2 or more
studies and treated by the same surgeon over a similar opera-
tion period) had been included more than once in the present
study, only the patients in the study with a larger cohort were
included for data extraction and statistical analysis in order to
minimize any potential for sampling bias.
It was difficult to use a uniform classification of compli-
cations due to reporting inconsistencies. The study by Paley
(1990) was cited by several research groups (Vaidya et al.
2006, Park et al. 2008, Kim et al. 2012c), but then not used
to evaluate the patients noted in their articles. We therefore
evaluated the total numbers (sum) of complications (prob-
lems, obstacles, and sequelae) (Eralp et al. 2010, Kim et al
2011) and complication rate per segment. It was also chal-
lenging to divide complications into problems and obstacles,
as these were not consistently reported. Thus, we documented
only numbers of sequelae (permanent complications) defined
Table 1. The number of patients from each study and the number of
total segments included in outcome analysis
Total no. of
Aldegheri et al. 2001 J Pediatr Orthop B
Aldegheri et al. 1988 J Bone Joint Surg (Br)
Bidwell et al. 2000
Catagni et al. 2005
Cattaneo et al. 1988
Hahn et al. 2003
Kim et al. 2012
Kim et al. 2012
Kitoh et al. 2007
Lie et al. 2009
Ng BKW et al. 2003
Noonan et al. 1997
Noonan et al. 1998
Park et al. 2008
Trivella et al. 1996
Vaidya et al. 2006
Venkatesh et al. 2009 J Bone Joint Surg (Br)
J Bone Joint Surg (Am) 67
J Bone Joint Surg (Br)
J Bone Joint Surg(Br)
Yonsei Medical Journal
J Bone Joint Surg (Br)
J Pediatr Orthop
Hong Kong Med J
Hong Kong J Paediatr
Iowa Orthop J
J Bone Joint Surg (Am)
J Bone Joint Surg (Am)
J Bone Joint Surg (Br)
J Pediatr Orthop
Flow diagram of search methods and criteria.
6,809 article titles reviewed
3,969 abstracts reviewed 2,840 duplicate articles
3,951 articles excluded
based on exclusion criteria
18 articles met inclusion
“limb lengthening”, and
(Jan, 1950 – Aug, 2012)
Acta Orthopaedica 2014; 85 (2): 181–186 183
as all problems during lengthening that were not resolved by
the end of treatment (Eralp et al. 2010, Kim et al 2011).
We analyzed data using SPSS software. The chi-square test
was used to evaluate differences in the complication rates and
sequelae between groups. Any p-value of < 0.05 was regarded
as significant. It was not possible to conduct a meta-analysis
due to the heterogeneity of the reports. An I2 value of 75%
was indicated according to the test statistic for evaluation of
heterogeneity (Higgins et al. 2003).
18 level-IV studies met our inclusion criteria, yielding data on
547 subjects who underwent 1,581 lower limb segment length-
ening procedures. No level-I or -II studies were identified in
our search. While the underlying diagnoses, limb segments
lengthened, and devices used for lengthening were available
for all subjects, other variables such as patient age, sex, pre-
operative height, and length gained were not found on a con-
sistent 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 undergo-
ing 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. Two-
thirds of the patients (367/547) had underlying diagnoses of
A/H and underwent the first lengthening procedure between 4
and 35 years of age (Table 2).
Based on the information available, the average age at
lengthening was less for individuals with A/H (14.5 years)
than for those with TS (18.2 years) or CSS (21.7 years). The
mean gain in height was greater in patients with A/H (9.5 cm)
than in patients with TS (7.7 cm) or CSS (6.1 cm). The HI
ranged from 24 to 67 days/cm. It was also better in patients
with A/H (31 days/cm) and CSS (32 days/cm) than in those
with TS (45 days/cm) (Table 3).
As noted previously, most studies reported untoward events
and complications inconsistently. 805 “complications” were
reported for 1,057 segments, with 0.76 complications reported
per lengthened segment (Table 4). In 2 studies (Cattaneo et
al. 1988, Aldegheri and Dall’Oca 2001), the distribution of
complications among the patients was reported but the sum of
complications was not reported. The complication rate per seg-
ment lengthened was lower for A/H (0.68) than for TS (0.71)
or CSS (1.06) (p < 0.001). Sequelae were seldom reported, yet
occurred in 19 segments, 5 segments, and 4 segments, respec-
tively, with pooled percentages of 1.7% (19/1,111 segments),
2.1% (5/234 segments), and 1.7% (4/236 segments) (Table 5).
The rate of sequelae was similar between groups.
While many authors have reported on limb lengthening for
increasing standing height (Park et al. 2008, Kim et al 2009,
2012b, Lie and Chow 2009), there is limited information on
whether there are any differences in outcome based on the
patient’s underlying etiology.
Despite the challenges with heterogeneity of the patients,
lack of a consistent method of assessment, and reporting of
patient-centered outcomes, we were able to gather some clini-
cally relevant information regarding lower limb lengthening
Table 2. Demographics of patients undergoing limb lengthening for short stature
No. of available
patients A/H a TS b CSS c Total Studies not included
No. of patients
No. of patients in
each study (range)
Age in years
No. of segments
No. of tibias
No. of femurs
367 77 103 547 547 None
5–100 6–20 3–54 3–100 547 None
Park et al. 2008, Aldegheri 1999, Noonan et al. 1998
Aldegheri and Dall’Oca 2001, Aldegheri et al. 1988,
Noonan et al. 1998, Ng et al. 2003, Kitoh et al. 2007
b Turner’s syndrome
c Constitutional short stature
184 Acta Orthopaedica 2014; 85 (2): 181–186
for stature by pooling the extracted data from the existing
literature. Lower limb lengthening for increasing stature was
reported most often (67%) for patients with A/H (Kim et al.
2012b). These patients also appeared to undergo the great-
est amount of lengthening, to heal faster, and to have fewer
reported complications than individuals with other diagnoses
undergoing stature lengthening. Stature lengthening in patients
with TS is more challenging due to the numerous possible
medical conditions including cardiovascular, renal, and endo-
crine abnormalities and also learning disabilities (Gravholt
2004); thus, the indications for limb lengthening may be more
limited. While patients with CSS are generally healthy, due to
normal muscle to bone length ratio, these individuals can be
more predisposed to development of soft tissue contractures
following limb lengthening (Schoenau et al. 2004). It has been
suggested that patients with achondroplasia tolerate lengthen-
ing very well because of their ligament and soft tissue laxity,
and their muscle length exceeds bone length before lengthen-
ing (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. The HI was also better in patients with A/H and
CSS than in those with TS.
Complications arising from DO can be severe, with long-
term residuals. Recently, in a separate study that was not
included in our analysis (due to lack of documented com-
plications), over half of skeletally immature patients with
achondroplasia who underwent DO for stature lengthening
had substantial growth inhibition and premature physeal clo-
sure 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). It has been suggested that
individuals with achondroplasia who have undergone lower
limb lengthening without any sequelae generally have a
Table 3. Amount of lengthening and healing index
No. of patients
included A/H a TS b CSS c?
Studies not included
Mean length gained per
limb in cm (range)
Mean percentage increase
per limb (range)
Healing index in
9 8 6 9 547
(24–41) (31–57) (30–67) (24–67)
Cattaneo et al. 1988, Catagni et al. 2005, Aldegheri
and Dall’Oca 2001, Noonan et al. 1998, Ng et al. 2003,
Bidwell et al. 2000)
Aldegheri and Dall’Oca 2001, Bidwell et al. 2000
b Turner’s syndrome
c Constitutional short stature
Table 4. Reported complications for limb lengthening
A/H a TS b CSS c? p-value Total Studies not included
Complications/ total segment (%) 487/707 (68)
Sequelae/total segment (%)
209/196 (106) < 0.001
28/1,581 (1.8) None
Cattaneo et al. 1988,
Aldegheri and Dall’Oca 2001
19/1,111 (1.7) 5/234 (2.1) 4/236 (1.7)
b Turner’s syndrome
c?Constitutional short stature
Table 5. Reported sequelae of limb lengthening
A/H a TS b CSS c? Total
Necrosis of the
Total no. of sequelae
11 3 2 16
2 0 0 2
b Turner’s syndrome
c?Constitutional short stature
Acta Orthopaedica 2014; 85 (2): 181–186 185
better quality of life and higher self-esteem than those with
one or more sequelae (Kim et al. 2012c). In the present study,
we noted that patients with A/H underwent a larger amount
of limb lengthening with fewer reported complications than
those with other diagnoses. We found the highest frequency
of complications (1 per segment) in patients with CSS. The
lower rate of complications in patients with A/H may result
from the shortening of treatment time associated with more
robust new bone formation at the distraction gap. However,
we were unable to find any relationship between the etiology
of short stature and sequelae, perhaps because of a limited
number of patients with sequelae in published material. Fur-
thermore, there is a distinct possibility of under-reporting of
such untoward events related to the retrospective study design
and lack of long-term follow-up.
The use of IM implants for DO is gaining popularity (Park et
al. 2008). It is quite likely that while the newer techniques of
lengthening may minimize or avoid the annoyance of having
an overlying external fixator, there may be some device-
related issues (Guichet et al. 2003) that would need further
study before one can recommend one technique over the other.
We excluded cases involving IM devices for lengthening in
order to reduce the number of variables related to the device
We noted several limitations in the existing literature on limb
lengthening, which led to our inability to draw firm conclu-
sions. Firstly, this is a pooled analysis of a several level IV
evidence studies with a heterogeneous cohort of patients.
Consequently, not all the data that we wanted to include in
our analysis was consistently available in the studies chosen.
Certain variables, such as patient age and sex, length of fol-
low-up, preoperative or postoperative height, LP, and HI were
not specified or were not presented in sufficient detail to allow
meaningful statistical inferences and comparisons. Secondly,
clinical outcomes including functional scores, quality of life,
and untoward events were not reported using a validated clini-
cal assessment tool across the majority of the studies. While we
did try to reclassify these “complications” in separate catego-
ries, given the paucity of comparable data, we could not evalu-
ate the complications using a uniform classification system.
Instead, we investigated the rate of complications in each study
as we believe that there is less variability in the definition of
this term. Thirdly, the younger mean age of patients with A/H
(14.5) compared to the average age of remaining patients (19.9)
could also have contributed to the better HI and longer length-
ening in this subgroup of short-stature individuals. In addition,
TS was a female-only group and patients with CSS underwent
mostly tibial lengthening. The use of retrospective studies for
the pooled analysis makes it difficult to confirm the direct asso-
ciation of the underlying etiology of short stature with healing
potential and clinical outcome after stature lengthening.
In conclusion, we found that patients with A/H healed faster
with a lower complication rate compared to other groups of
individuals undergoing lower limb lengthening using EF.
However, the length of follow-up in most available studies
was short, with no uniformity in reporting of patient-centered
clinical outcomes. As the technology for DO advances, the
feasibility of performing limb lengthening using innovative
techniques and implants will probably improve. Based on
our literature review, limb lengthening for short stature needs
further risk-versus-benefit analysis using a more robust study
design and a consistent method of reporting patient-centered
outcomes with long-term follow-up.
SJK: literature review, implementation, and manuscript preparation and
editing. WP: data analysis, literature review, and preparation and editing of
manuscript. SS: study design, implementation, and manuscript preparation
No competing interests declared.
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