Keywords - Lipedema - Lipoedema - Lipectomy
INTRODUCTION
Lipedema was first described by Allen and Hines as a condition consisting of a pathological
increase in subcutaneous fat and edema in the lower limb [1]. It almost exclusively affects women, and there are very few published case reports
of men with lipedema [2]. Lipedema shows familial clustering; one study proposed an autosomal dominant inheritance
with sex limitation [3].
Clinically, lipedema can be characterized in most cases as a chronic disease that
begins in puberty and takes a progressive course. Typically, there is a symmetric
increase in subcutaneous fat in the upper and lower legs due to both hyperplasia and
hypertrophy [4], which can be seen through magnetic resonance imaging or computed tomography [5]
[6]. The feet are spared, so there is an excess of fat at the ankle, also known as the
“inverse shouldering effect.” The upper limb is affected in up to 30% of cases, however
it is rarely affected in isolation [7].
In addition to the increase in subcutaneous fat, there is an increase in capillary
permeability and fragility, resulting in both extravasation of fluid and easy bruising
[8]. Evidence has also been found of morphologic changes in the lymphatic system, such
as microaneurysms; however, the pathophysiological significance remains unclear [9].
In contrast to primary lymphedema, the lymphatic system remains unimpaired in the
initial stages of lipedema and can keep up with the increased amount of interstitial
fluid [4]. Accordingly, lymphoscintigraphy has shown increased lymphatic transport in some
patients [10]. During the later stages of lipedema, the amount of fluid produced exceeds the transport
capacity of the lymphatic system, and the pressure of the fat tissue itself causes
obstruction of the lymphatic vessels, resulting in secondary lymphedema [10]
[11]. Additionally, the deposition of protein-rich edema causes fibrosis of the tissue,
further impairing lymphatic drainage. The term “lipolymphedema” is used to describe
the combined pathology during these stages.
It is known that deposition of fat is positively correlated with stasis of blood and
lymphatic fluid [12]. Therefore, a mutual interaction between adipose tissue and the lymphatic system
exists.
Lipedema is diagnosed based on a clinical examination and anamnesis. Patients typically
show disproportionality between a normal upper body and symmetrically enlarged lower
limbs, and this is often accompanied by elevated body mass index (BMI) levels. Furthermore,
the amount of fat in the affected areas is almost entirely unaffected by physical
activity or dietary measures.
Typical clinical complaints include feeling of tension, pain upon pressure, and easy
bruising. These symptoms are important in the differential diagnosis of lipedema,
particularly in the common case where lipedema and obesity are present at the same
time.
Lipedema can be classified into 3 clinical stages based on morphological appearance
[13]: Stage I, a smooth skin surface with homogenous thickening of the subcutis; Stage
II, a bumpy, wave-like skin surface with nodular structures in the thickened subcutis;
Stage III, an increase in nodular changes and overhanging masses of tissue.
The condition is a major psychosocial burden for most patients, causing pain that
often limits their capacity for exercise. In addition, standing for long periods of
time and high temperatures are not tolerated well by those with lipedema, and in severe
cases, the condition may cause absence from work or lead to occupational disability.
The most widely applied therapy for lipedema is combined decongestive therapy (CDT),
which consists mainly of manual lymphatic drainage and wearing compression garments.
It aims to reduce orthostatic edema and limit recurrence.
Classic dry liposuction cannot be applied to lipedema patients due to the potential
injury to lymphatic vessels. However, the introduction of the tumescent technique
in the 1980s has made the application of liposuction possible. Cadaver studies have
shown markedly reduced injury to lymphatic structures when the tumescent technique
was used [14].
In 1994, Rudkin and Miller [15] described liposuction in combination with skin and subcutaneous fat excisions as
a treatment option for lipedema, in contrast to lymphedema. In 2002, liposuction alone
was presented as a method to surgically reduce the pathological increase in subcutaneous
fat [15]
[16]
[17].
The literature on lipedema, in general, is limited. Most articles are in German and
fall under the disciplines of dermatology or phlebology. The present study aimed to
examine the long-term results of liposuction in patients with lipedema who were treated
in our plastic surgery clinic.
METHODS
From July 2010 to July 2013, 33 female patients received a total of 104 liposuction
procedures in our clinic as treatment for lipedema. Patients were classified preoperatively
into the 3 clinical stages described above.
At the end of 2013, these patients received a standardized questionnaire composed
of 18 items. Visual analog scales (VAS) from 0 to 10 in increments of 0.5 were used
to assess the severity of spontaneous pain, pain upon pressure, feeling of tension,
bruising, cosmetic impairment, and general impairment of quality of life before and
after liposuction treatment. Questions were also asked regarding weight, the frequency
of manual lymphatic drainage per month, and the number of hours per day the patient
wore compression garments. These last two values were added together to give a CDT
score. In 2015, the assessment was repeated using the same questions but assessed
only the current state of the patients’ symptoms.
Twenty-five patients responded in 2013 and were included in this study, and all 25
patients were available for follow-up in 2015.
The first postoperative follow-up was performed between 4 and 34 months after patients
received their last liposuction procedure, with a mean follow-up time of 16 months.
The second postoperative follow-up was performed between 25 and 56 months after the
last liposuction procedure, with a mean follow-up time of 37 months.
For each patient, lipedema diagnosis had been clinically confirmed by a lymphologist,
ruling out other lymphatic diseases. Each procedure was covered by health insurance
after a medical proposal was accepted, and the patient had typically already received
at least 6 months of CDT without improvement of symptoms.
Tumescent liposuction was performed using saline with epinephrine (1:1,000,000) after
obtaining informed consent. All liposuctions were performed as inpatient procedures,
and general anesthesia was used during the multi-hour procedures to increase patient
comfort and reduce operation time. Antibiotics were administered as a single shot
for perioperative prophylaxis only.
Patients were told to bring their compression garments, which were put on immediately
after liposuction. New garments were measured 3 weeks after liposuction and after
swelling had decreased, and manual lymphatic drainage was allowed after postoperative
day 2.
Statistical analyses of complaint severity and the CDT score were performed in SPSS
ver. 21.0 for Mac (IBM Corp., Armonk, NY, USA) using repeated-measures analysis of
variance (ANOVA) with the Bonferroni correction after meeting the criteria of the
Mauchly test of sphericity. All the tests were 2-sided, with α=0.05 and P<0.05 considered
statistically significant.
RESULTS
The age at the first performed liposuction ranged from 23 to 64 years, with a median
of 45 years. BMI upon presentation ranged from 24.5 to 50.6 kg/m2, with a mean of 35.3 kg/m2. After treatment, BMI ranged from 22.7 to 47.2 kg/m2, with a mean of 33.9 kg/m2.
All patients had lipedema of the lower limb. Additional upper limb involvement was
present in 9 patients (36%). One patient had stage I lipedema, 11 patients had stage
II lipedema, and 13 patients had stage III lipedema.
On average, patients received 3 procedures, with a range of 1 to 7 procedures. A total
of 72 liposuctions were performed on the 25 patients. In 41 liposuctions, a vibration-assisted
device was used, and in 31 liposuctions, a water jet-assisted device was used.
The operation time, which included infiltration, was 116 minutes on average and ranged
from 58 to 251 minutes.
The mean volume of removed fat per liposuction was 3,106 mL (range, 1,450–6,600 mL)
and the mean volume of total removed fat per patient was 9,914 mL (range, 4,000–19,850
mL).
One patient developed erysipelas after liposuction, which required antibiotic treatment.
However, there were no other complications during the study period. Therefore, the
complication rate was 1.39%.
Complaints
[Table 1] shows that patients reported substantial lipedema-associated complaints preoperatively.
Spontaneous pain was reported with a mean VAS score of 7.2 (standard deviation [SD],
1.46); the equivalent of “severe” to “very severe” spontaneous pain. Sensitivity to
pressure and feeling of tension were reported with mean VAS scores of 7.38 (SD, 1.79)
and 7.52 (SD, 1.36), respectively, falling within the “very severe” range. The reported
cosmetic impairment ranged from “severe” to “unbearable,” resulting in a mean VAS
score of 8.98 (SD, 0.81). General impairment to quality of life was also reported
as “very severe,” with a mean VAS score of 8.38 (SD, 1.06).
Table 1.
Results from the analysis of the questionnaires (n=25)
|
Measured variable
|
Preoperative
|
Postoperative 1
|
Postoperative 2
|
|
Mean
|
SD
|
Mean
|
SD
|
Mean
|
SD
|
|
SD, standard deviation; CDT, combined decongestive therapy.
a) Visual analog scale of symptom severity ranging from 0 to 10 in increments of 0.5,
with 10 being the most severe;
b) The CDT score was calculated as the sum of the number of manual lymphatic drainage
sessions per month and the number of hours spent wearing compression garments per
day. Four patients were excluded who did not receive full CDT preoperatively.
|
|
Complaintsa)
|
|
|
|
|
|
|
|
Spontaneous pain
|
7.20
|
1.46
|
3.70
|
1.79
|
4.28
|
2.10
|
|
Stage II (n = 11)
|
7.00
|
1.40
|
3.36
|
2.01
|
4.00
|
2.18
|
|
Stage III (n = 13)
|
7.54
|
1.44
|
4.12
|
1.58
|
4.61
|
2.13
|
|
Sensitivity to pressure
|
7.38
|
1.79
|
3.98
|
1.83
|
4.42
|
2.08
|
|
Stage II (n = 11)
|
7.05
|
1.29
|
3.50
|
1.97
|
3.82
|
2.27
|
|
Stage III (n = 13)
|
7.77
|
1.56
|
4.46
|
1.71
|
4.81
|
1.90
|
|
Feeling of tension
|
7.52
|
1.36
|
3.26
|
2.28
|
4.06
|
2.18
|
|
Stage II (n = 11)
|
7.09
|
1.00
|
3.14
|
2.47
|
4.00
|
2.35
|
|
Stage III (n = 13)
|
8.08
|
1.35
|
3.62
|
2.07
|
4.35
|
2.01
|
|
Bruising
|
6.96
|
1.58
|
4.36
|
1.91
|
4.64
|
1.83
|
|
Stage II (n = 11)
|
6.82
|
1.54
|
3.86
|
1.78
|
4.46
|
1.89
|
|
Stage III (n = 13)
|
7.15
|
1.70
|
4.65
|
2.03
|
4.66
|
1.85
|
|
Cosmetic impairment
|
8.98
|
0.81
|
5.10
|
1.93
|
7.36
|
1.66
|
|
Stage II (n = 11)
|
8.68
|
0.72
|
4.55
|
1.97
|
6.96
|
1.52
|
|
Stage III (n = 13)
|
9.31
|
0.78
|
5.58
|
1.91
|
7.85
|
1.69
|
|
Impairment to quality of life
|
8.38
|
1.06
|
4.30
|
1.80
|
5.16
|
1.60
|
|
Stage II (n = 11)
|
8.22
|
1.35
|
4.09
|
2.12
|
4.64
|
1.89
|
|
Stage III (n = 13)
|
8.62
|
0.71
|
4.42
|
1.63
|
5.46
|
1.23
|
|
CDT scoreb)
|
20.48
|
4.13
|
16.38
|
6.97
|
13.90
|
7.32
|
|
Stage II (n = 9)
|
21.22
|
4.58
|
13.33
|
9.15
|
12.00
|
9.89
|
|
Stage III (n = 11)
|
19.91
|
4.06
|
18.55
|
3.93
|
14.90
|
4.57
|
[Fig. 1] shows that the severity of all analyzed complaints was significantly reduced over
the course of liposuction treatment by the time of the first postoperative follow-up.
All but 1 of the patients reported a reduction in spontaneous pain (the chief complaint
in lipedema), with a mean difference in VAS score of 3.5 (95% confidence interval
[CI], 2.83–4.17). Furthermore, all but 1 of the patients reported a reduction in impairment
of quality of life, with a mean difference in VAS score of 4.08 (95% CI, 3.12–5.04).
Fig. 1. ComplaintsBox plots of the complaints before and after liposuction with accompanying
P-values (n=25). VAS, visual analog scales
The Bonferroni-corrected P-value was <0.001 for all 6 complaints. At the second postoperative
follow-up, only the severity of cosmetic impairment significantly increased since
the first postoperative follow-up, and there was significant improvement in all symptoms
between the preoperative period and the second postoperative follow-up. [Fig. 2] shows a comparative subgroup analysis of general impairment to quality of life for
patients with stage II lipedema and stage III lipedema. This symptom was chosen for
analysis because it was the most important complaint. While a significant reduction
in the severity of the complaint from the preoperative period to the first postoperative
follow-up was observed for both stage II and stage III patients, only stage III patients
experienced a significant increase in the severity of the complaint from the first
postoperative follow-up to the second postoperative follow-up.
Fig. 2. Combined decongestive therapy scores and subgroup analysesBox plots of the combined
decongestive therapy (CDT) scores before and after liposuction (n=21, 4 patients excluded
who did not receive full CDT preoperatively) and subgroup analyses of the visual analog
scale (VAS) scores for general impairment of quality to life by stage of lipedema
(n=11 for stage II lipedema, n=13 for stage III lipedema) and the CDT scores by stage
of lipedema (n=9 for stage II lipedema, n=11 for stage III lipedema).
Conservative therapy
Three patients did not wear compression garments preoperatively and only started wearing
them after liposuction in order to retain the results of the treatment. One patient
who did not receive manual lymphatic drainage preoperatively received it postoperatively.
Twenty-one patients regularly received manual lymphatic drainage and wore compression
garments preoperatively. For these patients, CDT scores were calculated as the sum
of manual lymphatic drainage sessions per month and hours spent wearing compression
garments per day.
At the second postoperative follow-up, the CDT scores of 14 patients had decreased
after liposuction treatment, with 3 patients no longer in need of further conservative
therapy. Three patients showed no change in their CDT scores, while 4 patients showed
an increase in their CDT scores.
For all patients who received full CDT preoperatively, the mean CDT score had decreased
from 20.48 (SD, 4.13) during the preoperative period to 16.38 (SD, 6.97) during the
first postoperative follow-up, and then decreased further to 13.9 (SD, 7.32) during
the second postoperative follow-up. [Fig. 2] shows that in the repeated-measures ANOVA, only the reduction in CDT score from
the preoperative period to the second postoperative follow-up was found to be significant
(P=0.011).
A comparative subgroup analysis of patients with stage II lipedema and stage III lipedema
showed a significant decrease in the CDT score from the preoperative period to the
second postoperative follow-up for stage II patients only. The reduction in CDT score
from the preoperative period to the second postoperative follow-up for stage III patients
was barely non-significant (P=0.051).
DISCUSSION
In most countries, CDT is the first line of therapy for lipedema. Though this treatment
aims to stop the progression of the disease and reduce edema, many patients still
see an increase in subcutaneous fat and aggravation of symptoms.
When the tumescent technique for liposuction was introduced in the late 1980s [18], the safety of liposuction improved and injury to lymphatic vessels was reduced
[14]
[19]. Subsequently, liposuction became an option in treating lipedema and reducing the
amount of fat tissue.
Our study showed that liposuction was an effective treatment for lipedema with good
long-term outcomes. Our findings support the results of previous studies, such as
Schmeller et al. [20], Rapprich et al. [21], and the recent long-term study by Baumgartner et al. [22].
However, unlike the aforementioned studies, the data for the preoperative period were
collected retrospectively, representing a possible bias.
An average operation time for liposuction under tumescent anesthesia that includes
infiltration has been reported as 5.5 hours. In our study, the average operation time
under general anesthesia that included infiltration was roughly 2 hours, yielding
comparable amounts of removed fat [22].
In accordance with the Practice Advisory on Liposuction published by the American
Society of Plastic Surgeons, we believe that general anesthesia has advantages over
tumescent anesthesia for long and complex liposuctions, such as those used to treat
lipedema [23].
General anesthesia has been shown to be a safe procedure and has a decreased risk
of aspiration and airway complications when compared to sedation [24]. This may be needed when operating under tumescent anesthesia to alleviate patient
discomfort.
During our study, there was only 1 minor infectious complication from the 72 liposuctions,
resulting in a complication rate of 1.4%. Schmeller et al. [20] saw the same rate of infection in 349 liposuctions used to treat lipedema, and Rapprich
et al. [21] reported just 1 case of deep vein thrombosis during 15 years of surgical treatment
for lipedema. In general, liposuction using the tumescent technique carries a low
surgical risk and has been shown to be a safe and appropriate procedure [25].
The patient examples in [Figs. 3] and [4] show that liposuction led to a marked reduction in the volume of fatty tissue in
the affected limbs and the disproportionality of these limbs to the rest of the body.
Although this was not measured quantitatively in our study, Rapprich et al. [21] were able to show volume reduction in lipedema patients treated with liposuction
using 3-dimensional volumetry.
Fig. 3. Case example 1A 24-year-old patient with stage II lipedema preoperatively (A) and
10 months after 2 liposuctions (B). A total of 8,800 mL of fatty tissue was removed
from her legs.
Fig. 4. Case example 2A 65-year-old patient with stage III lipedema preoperatively (A) and
6 months after 3 liposuctions (B). A total of 11,600 mL of fatty tissue was removed
from her legs.
The severity of all major symptoms of lipedema was significantly reduced, and there
was a significant reduction in the severity of impairment of quality to life, as reported
by the patients.
This corresponds with our clinical experiences, where we have found that lipedema
patients benefit greatly from liposuction treatment and display a very high level
of gratitude.
After extensive liposuction, the resulting excess skin tissue may harm the long-term
aesthetic outcome. This may be a possible explanation for the increase in the VAS
score for cosmetic impairment that was measured during the second postoperative follow-up
and was perceived to be caused by the disease. Lifting operations should be considered
for these patients, taking all necessary precautions to preserve lymphatic vessels.
We propose a simple score to quantify the need for CDT. From our clinical experience
and that of our cooperating lymphologists, we have found that lipedema patients wear
compression garments for roughly 8 to 10 hours per day on average, and patients will
typically receive 2 manual lymphatic drainage sessions per week. Deviations from these
patterns depend on symptom severity and distribution. Thus, summing the number of
hours spent wearing compression garments per day and the number of manual lymphatic
drainage sessions per month is an easy way to quantify CDT in a single score. Using
this score, we were able to show for the first time that liposuction for patients
with lipedema led to a significant decrease in the need for conservative lipedema
treatment and also improved the quality of life for these patients. Previous studies
that had already shown that liposuction may result in a decreased need for conservative
lipedema treatment did not quantify this need or test for significance [20]
[21].
Our data suggest that liposuction treatment for stage II lipedema provides a more
sustainable reduction in the impairment of quality to life and a larger decrease in
the need for conservative therapy than liposuction treatment for stage III lipedema.
Due to the development of secondary lymphedema and the irreversible damage to the
lymphatic system that occurs in later stages of the disease, liposuction should be
implemented as part of the standard therapy for lipedema at early stages. This will
prevent disease progression, improve quality of life, and reduce the need for decongestive
therapy.
