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A ruptured pheochromocytoma: an
unlikely cause of Brown-Sequard syndrome
To the Editor:
A 56 year old woman with severe angina was taken to a
local community hospital. Her past medical history was
significant for uncontrolled hypertension (currently taking
three antihypertensives), deep venous thrombosis, and a
descending aortic aneurysm. Computed tomography (CT)
showed a retroperitoneal bleed. The decision was made to
emergently transfer her to a tertiary-care center for her
persistent tachycardia and hypotension. En route, she
arrested, was resuscitated, and was transfused blood products
on arrival in preparation for the operating room. The patient
underwent an emergent exploratory laparotomy, but later
required an interventional radiology procedure to embolize
the retroperitoneal bleed.
Her intensive care unit stay was complicated by
abdominal compartment syndrome, ischemic bowel, as
well as pneumonia. Six weeks after her initial insult, her
mental status improved, but she had lower extremity
weakness, which was later attributed to Brown-Sequard
syndrome. This was thought to be secondary to spinal cord
ischemia, which likely occurred during her cardiac arrest.
She underwent serial CT scans of the abdomen to evaluate
for resolution of the retroperitoneal bleed, which showed a
right-sided pheochromocytoma. After 8 weeks, she was
discharged to a rehabilitation facility and was started on an
alpha-blocker, phenoxybenzamine, to control her hypertension. She underwent resection of the pheochromocytoma as
well as an ileostomy takedown one year after her inciting
event; her diagnosis was confirmed by pathology. The
patients surgery and recovery were both uneventful and she
was discharged home.
This patient might have benefited from further workup of
her uncontrolled hypertension. Her severe hypertensive
episode precipitated a major retroperitoneal bleed, leading
to spinal cord ischemia, resulting later in a diagnosis of
Brown-Sequard syndrome. This syndrome may be secondary
159
to a spinal cord tumor, ischemia, infectious or inflammation,
multiple sclerosis, or trauma/stabbing. Retroperitoneal
bleeding secondary to pheochromocytoma rupture has not
been well documented [1,2]. There was a case report of
Brown-Sequard syndrome in conjunction with a pheochromocytoma, but the neurological deficit was attributed to a
metastasis of the malignancy [3,4].
While pheochromocytoma comprises only 0.1% of cases
of hypertension, it should be included in the differential
diagnosis for severe hypertension, especially if multiple
antihypertensives are unable to control blood pressure
adequately [5]. While there is some controversy with
current recommendations of whether or not to start alphablockers preoperatively, the cornerstone of management
has been to start these medications preoperatively for blood
pressure control, and to diminish responses to catecholamine secretion. Alpha blockers should be started two
weeks preoperatively to restore intravascular volume; they
should also be started prior to the addition of beta blockers
to avoid precipitating congestive heart failure. Given the
extensive cardiac changes that may occur, it is advisable
to obtain an echocardiogram to further assess cardiac
function. General anesthesia with or without regional
technique has been used [5,6]. Continuous blood pressure
monitoring is required, as well as both vasodilators or
vasopressors to manage hemodynamic changes. Continuous
infusions of phentolamine or sodium nitroprusside may be
used for hypertension intraoperatively [5]. While postoperative hypotension is possible, it is less likely with adequate
volume resucitation and preoperative treatment with alpha
blockers [5]. Lastly, ischemic complications such as BrownSequard syndrome, while rare, may be seen in the
postoperative period. In an emergent setting, vigilance is
key; both hypertension as well as hypotension may be
problematic and preparation for massive hemorrhage
is required.
Megha Karkera MD (Fellow)
Department of Anesthesiology
University of Arkansas for Medical Sciences
Little Rock, AR 72205, USA
E-mail address: mmkarkera@uams.edu
http://dx.doi.org/10.1016/j.jclinane.2013.10.008
References
[1] Hayashi T, Nin M, Yamamoto Y, et al. Pheochromocytoma with
retroperitoneal hemorrhage after abdominal trauma. Hinyokika Kiyo
2009;55:703-6.
[2] Ito K, Nagata H, Miyahara M, Saito S, Murai M, Narimatsu Y.
Embolization for massive retroperitoneal hemorrhage from adrenal
pheochromocytoma: a case report. Hinyokika Kiyo 1997;43:
571-5.
[3] Habib M, Tarazi I, Batta M. Arterial embolization for ruptured adrenal
pheochromocytoma. Curr Oncol 2010;17:65-70.
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Correspondence
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