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Case Report
Diffuse intra-abdominal splenosis presenting as carcinomatosis
exhibiting positron emitted tomography hypermetabolic activity☆
Brian Kellert
a,
⁎, Michelle Caster
a,1
, Ryan Des Jean
b,2
, Luis Vaccarello
c,3,4
a
The Ohio State University/Mount Carmel Health OB/GYN Residency Program, 395 W State St, 5th Floor, Columbus, OH 43210, United States
b
Mount Carmel Health System, Mount Carmel East Hospital, Department of Pathology, 5959 E Broad Street, Columbus, OH 43213, United States
c
The Zangmeister Cancer Center, 3100 Plaza Properties Blvd, Columbus, OH 43219, United States
article info
Article history:
Received 12 February 2013
Accepted 22 March 2013
Available online 2 April 2013
Keywords:
Splenosis
Carcinomatosis
PET
Case report
CL is a 54 year old perimenopausal, nulliparous, morbidly obese
Caucasian woman who was referred to gynecologic oncology after
discovery of peritoneal implants on CT scan of the abdomen and pelvis,
obtained for persistent epigastric pain. She denied history of weight
loss, bloating, constipation, pelvic pain, or dysuria. Her history included
laparoscopic splenectomy thirteen years prior for idiopathic thrombo-
cytopenic purpura (ITP), and she had no family history of cancer.
Abdominal and rectopelvic examinations were normal. Cancer
antigen 125 and carcinoembryonic antigen levels were within normal
limits. CT review identified multiple small enhancing anterior peritone-
al nodular implants without evidence of pelvic mass (Fig. 1A). Given
pronounced imaging findings, resolution of prior symptoms, and his-
tory of splenectomy, a positron emission tomography (PET) scan was
obtained and showed hypermetabolic activity within some implants
(Fig. 1B).
As the case was highly suspicious for a malignant process, the pa-
tient was consented for exploratory laparotomy and debulking. Intra-
operatively, the greater omentum had multiple small maroon nodules
under 3 cm in size (Fig. 2). Extensive implantation was observed
along the anterolateral abdominal walls, small and large bowel, blad-
der, uterus, ovaries, and tubes. A partial omentectomy was performed
and sent for frozen and permanent section. Pathologist review of fro-
zen sections found lymphoid tissue favoring spleen which confirmed
our clinical suspicions. Complete survey found otherwise normal
abdominopelvic organs, and the abdomen was closed. The final pa-
thology report described unremarkable spleen, confirmed by flow cy-
tometry revealing normal lymphoid populations. When the patient
returned to the office for post-operative examination, she had a pe-
ripheral blood smear that showed no Howell–Jolly bodies.
We reviewed her splenectomy operative report which described
laparosocpic excision with morcellation and removal through an en-
doscopic bag without spillage. No comment was made of splenic nod-
ules. The patient's platelet count four months prior to splenectomy
was 21 K/μL, rising to 383 K/μL the day of splenectomy with medical
therapy, then rising to 583 K/μL one month following surgery. Her
platelet count was 432 K/μL at the time of consultation thirteen
years later.
Discussion
Splenosis is defined as ectopic splenic tissue commonly arising
from splenic trauma-induced autotransplantation, or rarely as con-
genital accessory spleens or polysplenia (Fremont and Todd, 2007;
Lake et al., 2012). Ninety three percent of splenosis cases follow ab-
dominal trauma, and arises after 65% of splenic ruptures (Fremont
and Todd, 2007; Ksiadzyna and Peña, 2011; Malik et al., 2012).
Small (b3 cm) visceroperitoneal implants are typically found, and
secondary splenosis is associated with greater implant quantity
(maximum of 400 reported) compared to congenital origins (up to
10) (Fremont and Todd, 2007; Ksiadzyna and Peña, 2011). Implants
may arise months to years following initial insult, and may be func-
tionalsuchasinrecurrenceofITP(Ksiadzyna and Peña, 2011).
These implants may mimic carcinomatosis upon front line imaging
modalities (Neri et al., 1986; White et al., 1989; Short et al., 2011;
Stovall and Ling, 1988; Mikhael et al., 2009).
Gynecologic Oncology Reports 5 (2013) 46–48
☆This is an open-access article distributed under the terms of the Creative Commons
Attribution License, which permits unrestricted use, distribution, and reproduction in
any medium, provided the original author and source are credited.
⁎Corresponding author at: The Ohio State University/Mount Carmel Health OB/GYN
Residency Program, 395 W State St, 5th Floor, Columbus, OH 43210, United States. Fax:
+1 614 293 5877.
E-mail addresses: brian.kellert@osumc.edu (B. Kellert), rc_desjean@hotmail.com
(R. Des Jean), lvaca@zangcenter.com (L. Vaccarello).
1
Fax: +1 614 293 5877.
2
Fax: +1 614 234 6278.
3
Fax: +1 614 383 6001.
4
Permanent Contact: The Zangmeister Cancer Center, 3100 Plaza Properties Blvd,
Columbus, OH 43219, United States.
2211-338X/$ –see front matter © 2013 The Authors. Published by Elsevier Inc. All rights reserved.
http://dx.doi.org/10.1016/j.gynor.2013.03.004
Contents lists available at SciVerse ScienceDirect
Gynecologic Oncology Reports
journal homepage: www.elsevier.com/locate/gynor
The recurrence risk of ITP secondary to splenosis is unclear.
Meta-analysis of 23 reports of ITP relapse following laparoscopic
splenenectomy (74% performed for ITP) was 43.6 per 1000 patient
years, 1–5 years post-operatively (Mikhael et al., 2009). Khatkouda et
al. compare their post-splenectomy ITP recurrence risk of 6% to the 4–
24.3% range of several studies, and suggest that capsular rupture may
increase recurrence to 12.5% (Katkhouda et al., 1998). The lack of
Howell–Jolly bodies 13 years post-splenectomy suggests functional
splenotic nodules in our patient, thus a risk of ITP recurrence exists.
The majority of splenosis cases are found incidentally, and it has
rarely been reported as a primary pathologic process. As concisely
reviewed by Fremont et al.; “Pain secondary to infarction, intestinal
obstruction due to the adhesive bands of the splenic implants, gastro-
intestinal hemorrhage, hydronephrosis secondary to a growing mass
exerting pressure on the ureter, as well as an enlarging abdominal
mass with associated infection have all been reported (Fremont and
Todd, 2007).”
Imaging modalities such as ultrasound, CT, and MRI do not provide
confirmatory diagnosis. Splenosis may resemble normal splenic tissue
upon these imaging techniques, and none can exclude carcinomatosis
(Lake et al., 2012; Ksiadzyna and Peña, 2011). Many radiologic and nu-
clear imaging techniques have been explored for non-invasive confir-
mation of suspected splenosis, with 99 m Technetium heat-damaged
erythrocyte scintigraphy regarded as ideal due to preferential uptake
in splenic tissue with normal liver uptake (Ksiadzyna and Peña, 2011;
Malik et al., 2012).
PET scans assist oncologists in treatment planning and malignancy
response quantification. Injection of labeled biologically active glu-
cose results in concentration of tracer in many tissues exhibiting
increased metabolic activity, a fundamental process in malignant
tumors (Aluddin, 2012). Quantification of glucose metabolism is de-
pendent upon many variables, including basal glucose levels, stress,
volume of distribution, medications, in addition to standardized and
facility-specificprotocols(Vriens et al., 2010).
A literature review found no reports of splenosis exhibiting PET
hypermetabolic activity. In fact, a recent case report of peritoneal
splenosis mimicking carcinomatosis used negative PET findings in sup-
port of primary laparoscopy instead of laparotomy in a patient with his-
tory of cervical cancer (Ake et al., 2012). While PET signal may be
influenced by habitus and glucoregulation, this morbidly obese patient
was not diabetic, fasted properly prior to the study, had a normal glu-
cose level (93 mg/dL), and underwent a standardized protocol with
fused PET–CT images and appropriate uptake phase of 60 min.
Direct visualization and tissue biopsy are the ideal means of char-
acterizing a suspected malignant intra-abdominal process. This pa-
tient had normal tumor marker levels and resolution of symptoms,
but ominous CT and PET findings. Given this clinical picture, we
proceeded with planned laparotomy for debulking surgery. Her sur-
gery and recovery were uncomplicated and the findings provided
her with great relief. In retrospect, scintigraphy may have added in-
formation to suggest a preoperative diagnosis of splenosis. In this
case, visual and tissue confirmation could have been accomplished
by laparoscopy. In this patient's clinical scenario we feel that visual
and pathologic confirmation is mandatory. The vascular nature of
splenic implants may increase the risk for hemorrhage with CT or
ultrasound-guided biopsy, and this method would not provide for vi-
sual survey of the abdominopelvic cavity.
Conclusions
Splenosis is a rare phenomenon that may be mistaken for carcino-
matosis upon standard imaging techniques. PET is routinely used to bet-
ter characterize an abdominopelvic process suspicious for malignancy.
In workup of novel carcinomatosis in patients with history of abdominal
trauma or splenectomy, splenosis shouldbe part of the differential diag-
nosis, and consideration of nuclear imaging modalities including PET
scan, scintigraphy, and diagnostic laparoscopy should all be considered
prior to proceeding with laparotomy.
Conflict of interest statement
The authors of this manuscript have no conflicts of interests to disclose.
Fig. 1. A: CT scan with IV contrast, coronal section at L5 showing anterior peritoneal
wall implants on the patient’s right. B: PET scan showing metabolic hyperactivity in
these same implants.
Fig. 2. Intraoperative view of anterior surface of greater omentum with splenosis
implants.
47B. Kellert et al. / Gynecologic Oncology Reports 5 (2013) 46–48
Consent
Written informed consent was obtained from the patient for pub-
lication of this case report and accompanying images. A copy of the
written consent is available for review by the Editor-in-Chief of this
journal on request.
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