Open Journal of Ophthalmology, 2012, 2, 103-109
http://dx.doi.org/10.4236/ojoph.2012.24022 Published Online November 2012 (http://www.SciRP.org/journal/ojoph)
103
Severe Symmetric and Chronic Lower Eyelid
Lymphedema in the Setting of Neck Surgery and Psoriasis
Michael E. Possin, Cat N. Burkat
Oculoplastics Service, Department of Ophthalmology and Visual Sciences, University of Wisconsin-Madison, Madison, USA.
Email: mepossin@gmail.com
Received September 7th, 2012; revised October 18th, 2012; accepted October 26th, 2012
ABSTRACT
Purpose: To present a patient with bilateral severe and symmetric lower lid lymphedema in the setting of previous neck
surgery and chronic psoriasis, and to review the potential relationships of neck surgery, irradiation, psoriasis, and
rosacea to chronic lymphedema. Design: Single case report with literature review. Methods: A 60-year-old female with
long-standing psoriasis presented with a 2-year history of severe, symmetric bilateral lower eyelid edema that deve-
loped after total laryngectomy and selective right neck dissection for recurrent Squamous Cell Carcinoma (SCC). 10
years prior she underwent radiation and radical left neck dissection for metastatic disease. Surgical management en-
tailed transcutaneous debulking of the masses combined with ectropion repair and suture tarsorrhaphy. A comprehen-
sive literature review was performed using Pubmed and Medline. Results: Surgical debulking of the soft tissue masses
via a transcutaneous incision resulted in significant improvement in the patient’s lymphedema without recurrence at 5
months follow-up. Histopathologic findings were consistent with chronic eyelid lymphedema. Conclusions: Isolated
eyelid lymphedema is rare, with many etiologies, and poses a diagnostic challenge. While ophthalmologists are familiar
with the ocular manifestations of rosacea such as conjunctivitis or blepharitis, it is important to consider rosacea as an
etiology of eyelid lymphedema. Reviewing the history for previous surgery or radiation to the head and neck, or other
dermatologic inflammatory disorders is also warranted. Rosaceous lymphedema is typically less severe than in post-sur-
gical/radiation patients, and does not respond well to medical treatment; however, it often shows a favorable response to
debulking blepharoplasty surgery, with or without skin grafting. This patient with a history of severe psoriasis and bi-
lateral neck dissections with radiation for SCC also responded well to surgery without recurrence of lymphedema.
Therefore, surgical debulking can be considered in these patients with severe eyelid lymphedema as an option to markedly
improve visual function and overall cosmetic appearance.
Keywords: Lyphedema; Eyelid Edema; Psoriasis; Blepharoplasty; Rosacea; Eyelid Lymphatics; Facial Lymphatic
System; Facial Edema; Squamous Cell Carcinoma
1. Introduction
Lymphedema occurs as a result of impairment of lym-
phatic drainage, leading to the accumulation of protein-
rich lymphatic fluid within tissues. This protein-rich in-
terstitial fluid leads to inflammation and an accumula-
tion of fibroblasts, adipocytes and keratinocytes that
transforms soft tissue into hard fibrotic tissue with stiff,
thickened skin. The most common etiologies of head and
neck lymphedema are associated with trauma, surgery,
tumor growth, radiation and infection to the head and
neck lymphatic system [1]. Isolated chronic eyelid lym-
phedema is rare, with only a few cases reported in the
literature that are mainly associated with rosacea [1-16].
Lymphedema of the eyelids can be very debilitating to
the patient, not only due to the physical appearance but
also from visual field obstruction. This article presents a
patient with severe lower eyelid lymphedema, reviews
the lymphatic drainage considerations, and the potential
association of certain dermatologic diseases with lym-
phedema.
2. Materials and Methods
This was a single case report. Informed consent was ob-
tained from the patient, the guidelines of the Health In-
surance Portability and Accountability Act were ob-
served, and Institutional Review Board approval was
obtained. Retrospective review of the medical record and
a comprehensive review of the literature was performed
using Pubmed and Medline, with key search words of:
lymphedema, eyelid edema, rosacea, psoriasis, eyelid
lymphatics, facial edema, facial lymphatic system, ps-
oriatic arthritis.
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Severe Symmetric and Chronic Lower Eyelid Lymphedema in the Setting of Neck Surgery and Psoriasis
104
3. Case Report
A 60-year-old female presented with severe, painless,
and symmetric bilateral eyelid edema of 2 years duration.
10 years earlier, she was diagnosed with metastatic squa-
mous cell carcinoma of the left neck, for which she un-
derwent combination chemotherapy and radiation treat-
ment. She then underwent radical left neck dissection for
residual disease that also involved the left jugular vein.
She subsequently did well until recurrence 2 years prior
to presentation, which required total laryngectomy, and
selective right neck dissection. Postoperatively, she de-
veloped neck, facial, and lower eyelid lymphedema. The
facial and neck edema resolved within 2 weeks, while her
lower eyelid edema persisted.
On initial exam, the pupils, motility, and slit-lamp
examinations were normal. The lower lids demonstrated
large bilateral lower lid masses, measuring 5.5 × 5 cm on
the right and 6 × 5 cm on the left, with anterior protru-
sion of 4.5 cm. The masses were firm, with non-pitting
edema and overlying brawny erythema and induration of
the skin (Figure 1). Lower eyelid mechanical ectropion
was also present due to the large masses. Blood work,
including complete blood count, chemistries, creatinine,
and thyroid panel, to evaluate for other systemic causes
of eyelid edema was unremarkable. CT scan confirmed
bilateral non-encapsulated lower lid soft tissue masses
without extension into the orbit. Past medical history was
also significant for severe psoriasis, which had led to
multiple hospitalizations.
Surgical management involved bilateral sequential
transcutaneous mass debulking with concurrent ectropion
repair. Intraoperatively, there were no clear planes for
dissection as the mass diffusely involved the skin, sub-
cutaneous tissue, and orbicularis muscle layers (Figure
2(A)). Dissection was performed in the subcutaneous
tissue plane to best delineate the anterior aspect of the
mass. Posteriorly, dissection was taken to just anterior to
the orbital septum. The soft tissue mass was placed in
formalin for histopathologic evaluation. After horizontal
tightening by a lateral tarsal strip procedure, the lower
eyelid skin was draped superolaterally. The skin was
markedly thickened and abnormal in texture and color
(Figure 2(B)). The redundant skin removed measured
over 5 cm vertically, and was submitted for pathology.
Figure 1. External photographs demonstrating severe bi-
lateral lower eyelid masses: anterior view (A), and left lat-
eral view (B).
Figure 2. Intraoperative photographs of left lower eyelid
surgery. (A) Soft tissue mass involving the skin, subcutane-
ous tissue, and orbicularis muscle layers dissected to the
orbital septum, with overlying skin retracted inferiorly, (B)
Redundant skin draped supe riorly and marke d for excision.
The skin was closed with 6-0 fast-absorbing plain gut
suture, with no vertical lid tension. A frost suture tarsor-
rhaphy was placed for 2 weeks (Figure 3). At 5 days
follow-up; there was a reduction in the eyelid lymphe-
dema, with moderate postoperative swelling (Figure 4).
Eyelid skin remained brawny and thickened in texture.
Surgery was then performed on the contralateral side in
the same fashion. At 5 months postoperatively, the thick-
ened skin texture remained, but there was no recurrence
of lymphedema, and she reported a significant improve-
ment in vision and quality of life.
Histopathology of the excised skin revealed thickened
eyelid skin demonstrating significant capillary prolifera-
tion, dermal fibrosis, reactive fibroblasts, and scattered
areas of collagen breakdown. Multiple foci of chronic pe-
rivascular inflammation resembled follicular formation.
A few non-caseating granulomas with mild inflammation
were also seen. The deeper soft tissue mass demonstrated
connective tissue with unremarkable skeletal muscle and
adipose tissue. Similar to the skin, there was capillary
proliferation, dermal fibrosis, collagen breakdown, and
foci of chronic perivascular inflammation (Figure 5).
Accessory lacrimal glands were also seen. The findings
were consistent with chronic eyelid lymphedema.
4. Discussion
The differential diagnosis of chronic facial edema is ex-
pansive. The possible etiologies are inflammatory (acne
vulgaris, systemic lupus erythematosus, sarcoidosis, al-
lergic dermatitis, angioedema, dermatomyositis), infec-
tious (erysipelas, mononucleosis, herpes zoster), con-
genital (facial hemiatrophy, Apert’s syndrome, McCune-
Albright syndrome), malignant (angiosarcoma, lym-
phoma, mycosis fungoides, lymphosarcoma, myeloma,
Kaposi’s sarcoma), trauma, trichinosis, hypothyroidism
and nephrotic syndrome [2]. Comprehensive discussion
of these diseases and their systemic workup is beyond the
scope of this article. Facial edema typically results as a
complication of the overall disease, rather than present-
ing as isolated edema. The less common localized type of
lymphedema usually results from previous infection,
Copyright © 2012 SciRes. OJOph
Severe Symmetric and Chronic Lower Eyelid Lymphedema in the Setting of Neck Surgery and Psoriasis 105
Figure 3. Immediate postoperative photograph with tar-
sorrhaphy in place, demonstrating significant debulking of
the left lower eyelid mass through a subciliary incision.
Figure 4. Postoperative photograph at 5 days follow up
demonstrating reduction in left lower eyelid lymphedema,
with moderate postoperative edema. Eyelid skin remained
brawny and thickene d in te xture.
tumor growth, radiation, surgery, trauma, infection, or
rosacea [1].
The severe bilateral eyelid edema seen in this patient
developed postoperatively after total laryngectomy and
selective right neck dissection two years prior to presen-
tation. Before that, there was a remote history of left
radical neck dissection with radiotherapy. It would first
be important to consider whether the eyelid edema was
secondary to a secondary or metastatic tumor. Although
the CT images at the time of recurrence were not avail-
able from the outside facility, the report noted recurrence
of disease in the right vocal cord. The left neck demon-
strated normal postsurgical changes and no other abnor-
malities. She also underwent a PET scan prior to presen-
tation that did not show any metastatic disease other than
the hyperbolic activity in the right vocal cord. On pre-
sentation to our service regarding her eyelid issues, an
additional CT of the head, maxiface, and orbits was per-
formed which also confirmed no facial or orbital masses.
The eyelid masses were localized in the preseptal eyelid
tissues without orbital extension.
The histopathologic findings of the excised tissue were
Figure 5. Hematoxylin-eosin: 10× magnification (A), 20×
magnification (B). Histopathologic examination of the soft-
tissue specimen demonstrates significant capillary prolif-
eration, dermal fibrosis, reactive fibroblasts, collagen break-
down, and multiple foci of chronic perivascular inflamma-
tion resembling follicular formation. These changes are
consistent with chronic lymphedema.
consistent with chronic lymphedema, which may have
been secondary to disruption of the lymphatic system
from tumor invasion, neck surgery, and/or radiation.
However, it was interesting that the chronic lymphedema
localized to the eyelids only, rather than throughout the
face as well.
Although the eyelid lymphatic system is still not
clearly understood, it is often believed to consist of two
separate drainage systems, in which the submandibular
lymph nodes drain the medial eyelids, central lower eye-
lid and cheek, while the parotid lymph nodes drain the
upper eyelid and lateral aspects of both eyelids [17]. A
recent study using lymphatic contrast and lymphan-
giograms in cadaver specimens demonstrated additional
anatomic variation, with the lateral eyelid draining into
either the parotid or submandibular nodes, and the medial
eyelid draining into the submandibular, parotid or bucci-
nator lymph nodes [18]. Two distinct levels of lymphatic
vessels that are not interconnected have also been de-
scribed in the lower lid. The superficial level is located
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Severe Symmetric and Chronic Lower Eyelid Lymphedema in the Setting of Neck Surgery and Psoriasis
106
between the skin and orbicularis muscle, while the deep
system lies between the orbicularis and tarsal plate [19].
The parotid nodes also drain the frontal, and part of the
parietal, regions of the head. The nasal, oral and mental
regions of the scalp drain to the submandibular nodes and
the oral and nasal branches drain to the buccinator lymph
nodes. Given that the nodes that drain the eyelids also
drain other regions of the head, it is curious that the
lymphedema was solely isolated to the eyelids. In a study
of 81 patients that received head and neck cancer treat-
ment, only three patients developed edema in the face,
neck and eyes, although details of tumor location, treat-
ment, and severity of the eyelid edema were not given for
comparison. No patients had isolated eyelid edema.
Edema of the neck only or of the face and neck were
most common and often improved, even with bilateral
lymphatic disruption [20]. Other articles also report that
secondary lymphedema after head and neck surgery is
common, however, there are no reports of isolated eyelid
edema [21,22]. Schiefkereported 17% - 36% of patients
had external lymphedema after selective neck dissection,
but did not specify the location [23].
Two other cases of chronic bilateral eyelid lymphe-
dema developing after surgery or radiation to the neck
have been reported in the literature. Bernardini et al. pre-
sented a patient with a 9-month history of progressive
lymphedema of the left upper and bilateral lower eyelids
following right radical neck dissection with radiation for
metastatic squamous cell carcinoma 5 years prior [2].
Clinically, the lymphedema appears similar to our patient;
however, rosacea was reported as the cause of her eyelid
edema rather than the neck surgery. In a series of 15 pa-
tients with chronic eyelid lymphedema, Chalasani et al.
briefly reported a patient with 84 months of eyelid lym-
phedema after radiotherapy, although no further details
were provided [3].
Two cases of unilateral eyelid lymphedema following
surgery were also found in the literature. Jordandescribed
a patient who developed right eyelid swelling following
removal of a right parotid tumor, radical right neck dis-
section, and neck radiation [4]. Three months after neck
irradiation, right upper and lower lid edema developed
that improved with excision of the preseptal and pretarsal
components of the orbicularis muscle. They concluded
the lymphedema was secondary to damage to the parotid
and submaxillary lymph nodes. Silverman et al. reported
a case of severe unilateral, cheek and lower eyelid lym-
phedema after resection of an oropharyngeal tumor with
radiation [5]. He was treated with surgical excision and
split-thickness skin grafting.
Venous disruption may also be a contributing factor to
the lymphedema in the patient presented here, as both
internal jugular veins had been invaded with tumor.
However, Ensari et al. reports that in patients who had
staged bilateral neck dissections, with loss of the internal,
external, posterior external and anterior jugular vein,
postoperative digital subtraction angiography showed
that venous drainage of the head was diverted from the
jugular veins to the vertebral venous plexus [24]. There-
fore, persistence of symmetric eyelid lymphedema would
be unusual.
Certainly it is difficult to determine with any certainty
if the lymphedema seen in our patient was more a result
of the tumor removal, reconstruction surgery and radia-
tion affecting lymphatic drainage, or if there was simila-
rity to the cases presented in the literature with rosacea. It
was initially assumed that neck surgery and irradiation
were the causes; however, upon review of the literature
as well as following consultation with head and neck
surgeons, it appeared that although neck, laryngeal, or
facial edema may be common, this is often transient due
to robust collateral drainage. Isolated eyelid edema as
seen in our patient is unusual and rare, even after signifi-
cant bilateral neck surgery.
As mentioned previously, one of the two patients pre-
sented by Bernardini et al. had a prior history of neck
surgery and radiation, but rosacea was considered to be
the cause of lymphedema in both patients [2]. Rosacea is
a cause of facial and eyelid edema, although is not often
described in the ophthalmic literature. Ocular involve-
ment in acne rosacea affects 3% - 58% of patients, with
20% experiencing ocular manifestations as the initial
presentation of rosacea [25]. The initial and most com-
mon stage of rosacea that is most familiar to ophthal-
mologists consists of erythema, teleangectasia, blepharitis,
and conjunctivitis. The second stage includes papules
and pustules, while the third stage includes inflammatory
nodules, tissue hyperplasia, or rhinophyma. Lymphe-
dema of the face or eyelids can present during any stage
and is most often reported in the dermatologic literature
[6-12] and only rarely in the ophthalmology literature
[2,3,13]. Rosaceous edema is typically a non-pitting,
bilateral, solid edema of the mid-third of the face, mainly
affecting the periorbital tissues, but can also affect the
forehead, glabella, nose or cheeks [6]. Unilateral eyelid
lymphedema in association with rosacea has also been
reported [1,2,7,13]. Facial erythema and telangiectasias
on the nose and cheeks are often seen, in addition to the
non-pitting eyelid edema [8].
Chalasani et al. reported fifteen patients with chronic
eyelid lymphedema caused by: acne rosacea (9 patients),
past radiotherapy (1 patient), trauma (1 patient), post-
vitrectomy silicone oil leak (1 patient), and unknown (4
patients) [3]. There were no measurements of the edema,
but appeared less severe in degree. The patients were
either observed, or treated with doxycycline or surgical
debulking eyelid blepharoplasty. Both patients described
by Bernardini et al. underwent debulking of the lower
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Severe Symmetric and Chronic Lower Eyelid Lymphedema in the Setting of Neck Surgery and Psoriasis 107
eyelid mass with horizontal eyelid tightening, with good
functional improvement at three week follow up [2].
Maisels [14] and Silverman [5] reported successful re-
sults with excision of the lower eyelid masses and split-
thickness skin grafting.
In contrast, Jamesreported a patient with rosacea and
bilateral upper and lower eyelid edema that failed to re-
spond to systemic steroids and tetracycline [15]. Excision
and skin grafting of the affected lids was performed, with
recurrence of lymphedema one year later. Marzanoalso
described recurrence within a few months after upper
eyelid blepharoplasty [6]. Kabir et al. reported a case of
sudden onset, severe bilateral periorbital lymphedema of
unknown etiology [16]. Two episodes of acute edema oc-
curred after staged debulkings, and they concluded that
using a skin graft offered a better cosmetic outcome.
Uhara et al. described a 53-year old man with sym-
metrical non-pitting edema, conjunctivitis, and acneiform
eruptions on the face with biopsies consistent with ro-
sacea [9]. He was treated with fleroxacin (100 mg/day)
for two weeks with a marked reduction of both facial and
periorbital edema. In contrast, most cases of chronic eye-
lid lymphedema in patients with acne rosacea in the der-
matology literature did not improve with medical treat-
ment [10-12].
The pathogenesis of rosaceous lymphedema is unclear,
but chronic inflammation is believed to play a role.
Lymphostasis promotes an influx of inflammatory cells
and release of mediators and cytokines by activated en-
dothelial cells, lymphocytes and macrophages [26]. Due
to the lymphatic dysfunction, these cytokines and che-
mokines, which normally would be cleared from the in-
terstitium, remain in the tissue, recruiting more inflam-
matory cells. This chronic inflammatory state activates
fibroblasts, which destroy elastin that surrounds blood
vessels, leading to transudation of fluids. Also, the re-
placement of elastin with collagen causes fibrosis, which
may obstruct the lymphatic vessels of the deep dermis,
leading to congestion, fluid accumulation and lymphe-
dema [27]. This is supported by the histologic findings in
rosaceous lymphedema, which include dilated blood ves-
sels, perifollicular fibrosis, perivascular lymphocytes,
histiocytes, neutrophils, perilymphatic granulomas and
stromal edema [2,7,27]. Although clinically the presenta-
tion of our patient looked most similar to the two patients
with rosacea presented by Bernardini [2], our patient did
not have any signs or symptoms of rosacea. However,
her history of psoriasis led us to question whether the
inflammatory component and histopathologic changes
found in rosacea that lead to this lymphedematous mani-
festation could also be seen in other dermatologic disor-
ders such as psoriasis. Histopathologic examination of
psoriatic skin lesions has demonstrated abnormalities of
the lymphatics such as dilatation and lack of fenestration
[28]. To our knowledge there are no reports in the litera-
ture of psoriasis causing facial or eyelid lymphedema,
but reports of lymphedema in the extremities, predomi-
nantly involving the upper limbs, are common in asso-
ciation with psoriatric arthritis [29-32]. 11% of people
with psoriasis may eventually develop psoriatic arthritis,
and our patient had a known history of severe psoriasis,
as well as arthritis of the hands, wrists and neck [33].
The exact pathogenesis of psoriatric arthritis lymphe-
dema is unknown, but similar to rosacea, inflammation
plays a role. Yamamoto et al. suggests that the inflam-
matory products from the synovium are deposited in the
adjacent lymphatics, leading to the lymphangitis and
lymphatic obstruction [29]. Quantitative lymphoscinti-
graphy disclosed abnormal lymphatic drainage of the
affected edematous limb [30], whereas another study
found no significant change in lymphatic flow in patients
with rheumatoid or psoriatic arthritis [31].
In conclusion, isolated eyelid lymphedema is rare,
with many etiologies, and poses a diagnostic challenge to
the physician. While ophthalmologists are familiar with
the ocular manifestations of rosacea such as conjunctivi-
tis or blepharitis, it is important to consider rosacea as a
possible etiology of significant eyelid lymphedema. A
complete review of history for previous neck surgery or
radiation to the head and neck regions, or other derma-
tologic inflammatory disorders may also be warranted.
To our knowledge, there is no previous report of eyelid
lymphedema associated with psoriasis. In general, rosa-
ceous lymphedema is typically less severe than in post-
surgical/radiation patients, and does not respond well to
medical treatment; however, it often shows a favorable
response to debulking blepharoplasty surgery, with or
without skin grafting. This patient with a history of both
severe psoriasis and bilateral neck surgery and radiation
for malignancy of the neck also responded well to sur-
gery without recurrence of lymphedema. Therefore, sur-
gical debulking can be considered in these patients with
severe eyelid lymphedema as an option to markedly im-
prove vital visual function as well as overall cosmetic
app earan ce .
5. Acknowledgements
Supported in part by an unrestricted grant to the Univer-
sity of Wisconsin Department of Ophthalmology and
Visual Sciences from the Research to Prevent Blindness,
Inc., New York, NY.
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