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Misdiagnosis of a chylous cyst as chest wall gouty tophusA case of true pseudogout.

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ARTHRITIS & RHEUMATISM
Vol. 56, No. 11, November 2007, pp 3854–3857
DOI 10.1002/art.22957
© 2007, American College of Rheumatology
Misdiagnosis of a Chylous Cyst as Chest Wall Gouty Tophus
A Case of True Pseudogout
Leena G. Adhikesavan,1 William T. Ayoub,2 and H. Ralph Schumacher3
to the left sternoclavicular joint and extending anterior
to that joint, in the region of the sternal head of the left
sternocleidomastoid muscle. There was no bony erosion.
The patient’s primary physician referred him to a pathologist, who performed a fine-needle aspiration. The
pathologist found birefringent crystals that were interpreted to be monosodium urate.
Another 6 months passed before the patient was
referred to a thoracic surgeon. A physical examination at
that time revealed a 4–5-cm soft, slightly tender mass
located in the sternoclavicular area, slightly toward the
left of midline, although extending across the midline.
His serum uric acid level was 4.9 mg/dl. Excision of the
mass was performed. At the time of surgery, the surgeon
thought that the mass was arising from the sternoclavicular joint. The cystic mass was found to consist of a
yellowish pasty material, and the pathologic diagnosis of
the cyst was histiocytic reaction to foreign material
consistent with gout. There were no inflammatory or
malignant cells. There was no further evaluation or
treatment at that time. The mass reappeared in a few
months.
Approximately 6 months after excision, the patient was referred to the rheumatology department for
treatment of presumed tophaceous gout. He was an
otherwise healthy man who had never had a gout attack.
He denied having any pain. Physical examination revealed 2 masses: one was larger than a golf ball and was
centrally located in the superior aspect of the sternum;
the other was marble sized and was located just inferior
to, and to the left of, the larger mass (Figure 1). No tophi
were noted anywhere else.
Fine-needle aspiration yielded a thick, yellowgreen material in the hub of the needle, but then ⬃15 ml
of off-white liquid (Figure 2) was aspirated from the
center of the larger mass. Microscopic examination of a
dried slide of the aspirate revealed birefringent material,
but no distinct needle-shaped crystals (Figure 3). Part of
A patient referred to us for recurrent chest wall
gouty tophus, but who was determined to actually have
a chylous cyst, is described herein. Chylous cysts of the
neck or chest wall can be caused by thoracic duct injury.
Chyle contains 4–40 gm/liter of lipids, mostly triglycerides, and these can form birefringent crystals upon
drying, leading to a false diagnosis of gout.
The diagnosis of neck masses can be confirmed in
most cases based on the patient’s medical history and a
physical examination, along with imaging studies and
fine needle aspiration. We describe a patient who was
referred to us for recurrent chest wall gouty tophus but
who was actually determined to have a chylous cyst.
CASE REPORT
The patient, a 48-year-old man, was referred to
us to consider treatment for tophaceous gout. He developed a chest wall mass 2 years prior to presentation. The
mass appeared a few days after he noticed some pain in
his chest while squeezing the handles on a pruning tool.
He did not seek medical attention for several months. A
chest radiograph and radiographs of the sternoclavicular
joints revealed no abnormalities. Computed axial tomography (CAT) showed a 5-cm soft tissue density superior
1
Leena G. Adhikesavan, MD: Geisinger Medical Center,
Danville, Pennsylvania; 2William T. Ayoub, MD: Geisinger Medical
Group at State College, State College, Pennsylvania; 3H. Ralph
Schumacher, MD: University of Pennsylvania VA Medical Center,
Philadelphia, Pennsylvania.
Address correspondence and reprint requests to William T.
Ayoub, MD, Associate, Geisinger Medical Group at State College,
Department of Rheumatology, 200 Scenery Drive, State College, PA
16801 (e-mail: wayoub@geisinger.edu); or to Leena G. Adhikesavan,
MD, Rheumatology Fellow, Geisinger Medical Center, 100 North
Academy Avenue, Danville, PA 17822 (e-mail: lgadhikesavan@
geisinger.edu).
Submitted for publication June 4, 2007; accepted in revised
form July 12, 2007.
3854
MISDIAGNOSIS OF A CHYLOUS CYST
3855
Figure 3. Polymorphous, birefringent crystals from the aspirated material (original magnification ⫻ 300).
Figure 1. The patient’s chest mass.
the specimen was sent to a rheumatologist (HRS) for
further investigation. Analysis revealed droplets typical
of neutral lipids (Figure 4), but no needle-shaped crystals. The gross and microscopic appearance was thought
to be characteristic of chyle, and an investigation for
lymphatic obstruction was suggested.
The patient was referred back to the thoracic
surgeon for treatment of the chylous cyst. Magnetic
resonance imaging (MRI) revealed a 7-cm ⫻ 5-cm cystic
mass in the sternal notch area of the lower anterior neck
that was partially displacing the medial margins of the
strap muscles. The patient was diagnosed as having a
chylous cyst secondary to traumatic disruption of the
thoracic duct, possibly initially from the strain of using
the pruning tool and then from the surgery. He under-
Figure 2. Fluid aspirated from the chest mass.
went surgical exploration and division of the thoracic
duct by ligation. Multiple areas of chyle leak were
identified, and major branches of the duct were ligated
and clipped. The main duct was found to be avulsed and
was also ligated. The patient is doing well now, with no
recurrence of the neck swelling.
DISCUSSION
Chylous cysts are rare. Chyle is lymphatic fluid of
intestinal origin, which is carried by the thoracic duct to
the bloodstream. Due to the fact that chyle is found only
in the mesentery of the small intestine and the thoracic
duct, chylous cysts are usually seen within the mesentery
of the small intestine or in the retroperitoneum (1,2).
Very few chylous cysts of the neck have been
reported (2–5). Information on the natural history and
pathogenesis of these cysts is limited (4). Weakness in
the thoracic duct wall could be congenital or degenera-
Figure 4. Droplets of neutral lipid characteristic of chyle (original
magnification ⫻ 300).
3856
tive (6,7). Thoracic duct injury is a rare but known
complication of thoracic surgeries, esophagectomy (8),
jugular vein or subclavian vein catheterization (9,10), or
penetrating or blunt trauma (11,12). This patient probably injured his thoracic duct while squeezing the handles on the pruning tool.
Radiography, ultrasonography, CAT, and MRI
are useful imaging techniques in determining the consistency and the anatomic boundaries of chylous cysts.
Simple aspiration is an unsuccessful therapy due to rapid
reaccumulation, since these cysts are connected to adjacent lymphatic structures (2–4). The incidence of malignancy has been reported to be 22% in one study (1) and
33% in another (13), so complete excision is recommended.
Gout is best diagnosed when negatively birefringent, needle-shaped monosodium urate crystals are
viewed using polarized light microscopy. Gout may
involve the chest wall by affecting sternoclavicular
(14,15), costochondral (14), or manubriosternal (16,17)
articulations. Subcutaneous tophus formation by crystal
deposition is rare in other conditions except gout. There
are a few cases where it has been reported in association
with hydroxyapatite (18), calcium pyrophosphate dihydrate (19,20), and calcium oxalate (21) crystals. Subcutaneous cholesterol “tophi” have been reported in 2
cases, both of which were initially mistaken for a gouty
tophus (22). Cholesterol crystals are found occasionally
in synovial effusions from patients with rheumatoid
arthritis (23), ankylosing spondylitis (23), bursitis (23,
24), and osteoarthritis (25). Although most cholesterol
crystals are flat, rectangular, and rhomboid, and resemble broad plates with a notched corner, needle forms can
also be seen (25–27), which can lead to the false
diagnosis of gout. Neutral lipids can also form birefringent crystals upon drying (28), which probably explains
the initial confusion about the appearance of the aspirate on the dried slide.
In this case, the location and relatively rapid
appearance of the mass are not typical of a gouty tophus.
No other findings suggestive of gouty arthritis were
obtained from the patient’s medical history or physical
examination. The appearance of monosodium urate
crystals when examined by polarizing light microscopy is
quite distinct and diagnostic. In order to accurately
diagnose gout, crystalline material should contain distinct needle-shaped crystals that are strongly negatively
birefringent. In this case, the microscopic appearance
was that of a birefringent irregular material without
diagnostic needle-shaped crystals. Although chylous
ADHIKESAVAN ET AL
cysts are rare, they must be considered as one of the
differential diagnoses of chest masses.
AUTHOR CONTRIBUTIONS
Dr. Adhikesavan had full access to all of the data in the study
and takes responsibility for the integrity of the data and the accuracy
of the data analysis.
Study design. Adhikesavan.
Acquisition of data. Adhikesavan, Ayoub, Schumacher.
Analysis and interpretation of data. Adhikesavan, Ayoub, Schumacher.
Manuscript preparation. Adhikesavan, Ayoub.
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MISDIAGNOSIS OF A CHYLOUS CYST
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