Akishige Kanazawa

D Komatsu

JN Bruneton

Katsunobu Sakurai

Kayo Yoshida

KM Hoda

M Miettinen

M Sarlomo-Rikala

Mamiko Takii

Masashi Takemura

MR Benz

NR Bees

RJ Loffeld

S Prévot

SN Vinhais

WS Melvin

X Altuna

Y Daimaru

Y Otomi

Journal of Medical Case Reports

English

PubMed

Springer - Publisher Connector

CASE REPORT Open Access
Gastric malignant schwannoma presenting with
upper gastrointestinal bleeding: a case report
Masashi Takemura1, Kayo Yoshida2*, Mamiko Takii2, Katsunobu Sakurai2 and Akishige Kanazawa2
Abstract
Introduction: We report a case of gastric malignant schwannoma presenting with gastrointestinal bleeding.
Case presentation: A 70-year-old Japanese man presented with gastrointestinal bleeding to our hospital.
Gastrointestinal endoscopy revealed a protruding lesion in the gastric body. Hematoxylin and eosin staining of
biopsy specimens from this lesion revealed sheets of spindle cells. Immunohistochemistry revealed that these cells
were positive for S-100 protein and negative for c-Kit and smooth muscle actin. Because mitosis was diffusely
visible, this tumor was diagnosed as a gastric malignant schwannoma. Distal gastrectomy with lymph node
dissection was performed and the patient’s postoperative course was uneventful. However, five months after the
surgery, he died from multiple liver metastases.
Conclusion: Cases of gastric malignant schwannoma have rarely been reported. The efficacy of surgical resection
and postoperative prognosis continues to remain unclear and should be investigated further.
Keywords: Gastric malignant schwannoma, S-100 protein, liver metastasis
Introduction
Schwannomas are neurogenic tumors that originate
from different organs as well as other areas throughout
the body. Gastric schwannomas are rare; however, if
they do occur in the gastrointestinal tract, the most
common site is the stomach. Gastric schwannomas
represent 0.2% of all gastric neoplasms [1]. Gastrointest-
inal endoscopy, such as endosonography, is the principal
diagnostic tool for gastric schwannoma [2]. However,
differentiating a schwannoma from other gastric submu-
cosal tumors is often difficult. The definitive diagnosis
of gastric schwannoma is established by pathological
and immunohistochemical examination of resected sur-
gical specimens [3].
Schwannomas are generally benign. Complete surgical
removal is sufficient treatment for a benign gastric
schwannoma because of excellent postoperative prog-
nosis. Gastric malignant schwannomas are extremely
rare and only a few cases have been reported [4,5]. We
present the case of a 70-year-old man who presented
with gastrointestinal bleeding because of gastric malig-
nant schwannoma.
Case presentation
A 70-year-old Japanese man with melena that began five
days before admission was admitted to our hospital. He
complained of abdominal discomfort and epigastralgia.
Five years before, he had undergone a right upper
lobectomy for lung cancer (T1b N0 M0 Stage IA). Phy-
sical examination revealed no abnormal findings asso-
ciated with the abdomen except a surgical scar on the
right side of his chest. Evaluation of laboratory data on
admission revealed that his hemoglobin level was 7.0 g/
dL and hematocrit value was 23.1%. Upper gastrointest-
inal endoscopy revealed a distinctly protruding lesion
(diameter: 5 cm) at the lesser curvature of the middle
third of the gastric body (Figure 1). The surface of the
tumor bled easily on contact with the endoscope.
Microscopic examination of hematoxylin and eosin
(H&E)-stained biopsy specimens of the lesion revealed
sheets of spindle cells. Immunohistochemical studies
showed that the tumor cells were positive for S-100 and
negative for c-kit, CD34, and smooth muscle actin. The
MIB-1 index was 48.5%. Based on these findings, the
tumor was diagnosed as a gastric malignant
* Correspondence: kayo19780419@yahoo.co.jp
2Department of Gastrointestinal Surgery, Osaka City General Hospital, 2-13-
22, Miyakojima Hondori, Miyakojima, Osaka City, Osaka, 534-0021, Japan
Full list of author information is available at the end of the article
Takemura et al. Journal of Medical Case Reports 2012, 6:37
http://www.jmedicalcasereports.com/content/6/1/37 JOURNAL OF MEDICAL
CASE REPORTS
© 2012 Takemura et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative
Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly cited.
schwannoma. Abdominal computed tomography (CT)
revealed a thickened posterior wall of the gastric body
(Figure 2). There was no evidence of lymph node swel-
ling surrounding the stomach or metastatic liver tumors.
He underwent a distal gastrectomy with regional
lymph node dissection. Macroscopically, the elevated
lesion was approximately 6 × 5 cm in diameter and
located at the lesser curvature of the gastric body (Fig-
ure 3). The covering mucosa was ulcerated. The tumor
was located mainly in the proper muscle layer. There
was no lymph node involvement and the surgical mar-
gin was negative for tumor cells. Microscopic examina-
tion of the resected and H&E-stained specimens showed
a spindle cell neoplasm arranged in a palisade manner
that was consistent with a schwannoma (Figure 4).
Mitosis was scattered with 10 mitoses per 50 high-
power fields. Immunohistochemistry revealed that the
tumor cells were positive for S-100 protein and negative
for c-kit and smooth muscle actin (Figure 5A,B). These
histopathological and immunohistochemical findings are
consistent with a gastric malignant schwannoma.
His postoperative course was uneventful and he was
discharged from our hospital on day 12 after surgery.
However, abdominal CT performed three months after
surgery revealed multiple liver metastases and ascites
(Figure 6).. He died five months after surgery without
undergoing any additional treatment.
Figure 1 Upper gastrointestinal endoscopy showing a
protruding lesion at the lesser curvature of the gastric body.
Figure 2 Abdominal computed tomography showing a
thickened posterior wall of the gastric body. No evidence of
lymph node swelling or a metastatic liver tumor is observed.
Figure 3 Macroscopic findings of the resected specimens. The
elevated lesion approximately 6 × 5 cm in diameter is located at
the lesser curvature of the gastric body.
Figure 4 Microscopic findings of the resected and hematoxylin
and eosin-stained specimen showing a spindle cell neoplasm
arranged in a palisade manner.
Takemura et al. Journal of Medical Case Reports 2012, 6:37
http://www.jmedicalcasereports.com/content/6/1/37
Page 2 of 4
Discussion
Schwannomas, also known as neurinomas and neurile-
momas, are rare stromal tumors of spindle cells of the
gastrointestinal tract that arise from the schwann cells
of the gastrointestinal neural plexus. The stomach is the
most common gastrointestinal site of schwannomas,
which constitute 0.2% of gastric neoplasms [1,3]. Gastric
schwannomas frequently occur in individuals 30 to 50
years old and are usually solitary lesions arising from
the lesser curvature of the stomach [1,3,6].
Spindle cell tumors of the gastrointestinal tract are
slow growing and covered with normal mucosa; they
principally involve the submucosa and muscularis pro-
pria. The majority of spindle cell tumors of the gastroin-
testinal tract are believed to originate from the smooth
muscle tissue. However, immunohistochemical staining
has enabled differentiation of various spindle cell tumors
[3]. Schwannomas can be distinguished from other spin-
dle cell tumors of the stomach by S-100 protein stain-
ing. Tumor cells that are positive for S-100 protein and
negative for smooth muscle actin, c-Kit and CD34 sup-
port the diagnosis of a schwannoma [7,8].
Preoperative differential diagnosis of gastric submuco-
sal tumors is generally difficult. Computed tomography
and gastrointestinal endoscopy are of limited use when
attempting a definitive diagnosis of a gastric submucosal
tumor [9]. Hoda et al. reported that endoscopic ultra-
sound (EUS)-guided sampling of gastrointestinal submu-
cosal lesions is helpful [3]. They concluded that the
diagnostic yield of EUS-fine needle aspiration (FNA)
was 83.9% and that FNA can be used to establish a defi-
nite or probable diagnosis in the majority of patients
with spindle cell tumors of the gastrointestinal tract.
18F-fluorodeoxyglucose positron emission tomography
(FDG-PET) is a non-invasive diagnostic technique that
enables quantification of tumor activity on the basis of
altered tissue glucose metabolism. Recently, FDG-PET
has been accepted as a powerful diagnostic tool for eval-
uating various tumors, even spindle cell tumors within
the gastrointestinal tract [10-12]. Benz et al. demon-
strated that FDG-PET can reliably discriminate malig-
nant peripheral nerve sheath tumors from benign ones
[10]. However, reports on the utility of FDG-PET for
diagnosing gastric schwannomas are rare. Komatsu et
al. reported a case of gastric schwannoma reflected by
increased FDG uptake [12]. However, they showed that
gastric schwannomas cannot be differentiated from gas-
trointestinal stromal tumors by FDG accumulation and
that FDG-PET was not helpful for evaluating whether
the tumors are benign or malignant. Therefore, further
studies are required to clarify the utility of FDG-PET in
the diagnosis of gastric schwannoma.
Bruneton et al. reviewed 112 cases of gastric schwan-
noma and found that the majority of the cases (63%)
reported gastrointestinal bleeding as the first symptom
and 42% presented with abdominal pain [13]. Gastroin-
testinal bleeding may be present in a case with deep
ulceration due to gastric activity or ischemic changes in
the covering mucosa and/or a schwannoma. The
Figure 5 Immunohistochemical analysis showing that the
tumor cells were positive for S-100 protein (A), and negative
for c-Kit (B) and smooth muscle actin.
Figure 6 Abdominal computed tomography three months after
surgery showed multiple metastatic lesions in the liver and
ascites.
Takemura et al. Journal of Medical Case Reports 2012, 6:37
http://www.jmedicalcasereports.com/content/6/1/37
Page 3 of 4
remaining patients had non-specific abdominal discom-
fort. In our patient who had gastrointestinal bleeding,
the covering mucosa was found to be ulcerated during
gastrointestinal endoscopy.
Gastric malignant schwannomas can be distinguished
from benign schwannomas on the basis of histological
examination of the resected specimens, not by clinical
symptoms or imaging studies. The treatment strategy
for gastric schwannoma is, therefore, the same regard-
less of whether the tumor is malignant or benign. Surgi-
cal resection is the only possible treatment for gastric
schwannoma [1,14]. The postoperative prognosis for
solitary benign schwannoma is excellent. Recurrent dis-
ease is generally associated with an incomplete surgical
margin. Additionally, recent advances in molecular or
target therapy have an important role for the treatment
of gastrointestinal mesenchimal tumors. However, the
usefulness of molecular therapy for gastric schwannoma
is not clearly established because only a few cases with
gastric schwannoma have been reported. On the other
hand, Xabier et al. reported that there is a high expres-
sion rate of platelet-derived growth factor receptor
(PDGFR) and c-kit in vestibular schwannoma [15]. They
concluded that direct inhibition of these molecules by
Gleevec® (imatinib mesylate)may have relevant thera-
peutic applications for this kind of tumor. From these
results, molecular therapy for gastric schwannoma may
become an additional treatment modality and deserves
further examination.
Conclusions
Gastric malignant schwannomas are extremely rare, and
only a few cases have been reported. Given the paucity
of the currently available published literature, the effi-
cacy of surgical resection and postoperative prognosis
for such cases warrants further study.
Consent
Written informed consent was obtained from the patient
for publication of this case report and any accompany-
ing images. A copy of the written consent is available
for review by the Editor-in-Chief of this journal.
Author details
1Department of Upper Gastrointestinal Surgery, Hyogo College of Medicine,
1-1, Mucogawa-machi, Nishinomiya City, Hyogo, 662-8501, Japan.
2Department of Gastrointestinal Surgery, Osaka City General Hospital, 2-13-
22, Miyakojima Hondori, Miyakojima, Osaka City, Osaka, 534-0021, Japan.
Authors’ contributions
All authors were actively involved in direct patient care and have read and
approved the manuscript. MT is the principal author and was involved in
the collection of data. AK contributed to writing the manuscript. MT, KY and
KS were involved in the collection of relevant literature and proof read the
manuscript.
Competing interests
The authors declare that they have no competing interests.
Received: 1 August 2011 Accepted: 25 January 2012
Published: 25 January 2012
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doi:10.1186/1752-1947-6-37
Cite this article as: Takemura et al.: Gastric malignant schwannoma
presenting with upper gastrointestinal bleeding: a case report. Journal
of Medical Case Reports 2012 6:37.
Takemura et al. Journal of Medical Case Reports 2012, 6:37
http://www.jmedicalcasereports.com/content/6/1/37
Page 4 of 4


Gastric malignant schwannoma presenting with upper gastrointestinal bleeding: a case report

Abstract
          
            Introduction
            We report a case of gastric malignant schwannoma presenting with gastrointestinal bleeding.
          
          
            Case presentation
            A 70-year-old Japanese man presented with gastrointestinal bleeding to our hospital. Gastrointestinal endoscopy revealed a protruding lesion in the gastric body. Hematoxylin and eosin staining of biopsy specimens from this lesion revealed sheets of spindle cells. Immunohistochemistry revealed that these cells were positive for S-100 protein and negative for c-Kit and smooth muscle actin. Because mitosis was diffusely visible, this tumor was diagnosed as a gastric malignant schwannoma. Distal gastrectomy with lymph node dissection was performed and the patient's postoperative course was uneventful. However, five months after the surgery, he died from multiple liver metastases.
          
          
            Conclusion
            Cases of gastric malignant schwannoma have rarely been reported. The efficacy of surgical resection and postoperative prognosis continues to remain unclear and should be investigated further.
          </jats:sec

Crossref

Benign schwannoma of the gastrointestinal tract: a clinicopathologic and immunohistochemical study. Hum Pathol

de Saint-Maur PP: Benign schwannoma of the digestive tract: a clinicopathologic and immunohistochemical study of five cases, including a case of esophageal tumor.

Eilber FC: Quantitative F18-fluorodeoxyglucose positron emission tomography accurately characterizes peripheral nerve sheath tumors as malignant or benign. Cancer

Faigel DO: EUS-guided sampling of suspected GI stromal tumors. Gastrointest Endosc

Gastric malignant schwannoma in a child.

Gastric schwannoma exhibiting increased fluorodeoxyglucose uptake. Gastric Cancer

Gastric schwannoma–a clinicopathological analysis of six cases. Histopathology

Gastrointestinal stromal tumors–definition, clinical, histological, immunohistochemical, and molecular genetic features and differential diagnosis. Virchows Arch

Gastrointestinal stromal tumors. Ann Chir Gynaecol

MG: Gastric schwannoma. Clinical and pathologic considerations. Am Surg

Neurogenic tumors of the stomach. Report of 18 cases and review of the literature. Rofo

Ongkeko WM: Potential role of Imatinib Mesylate (Gleevec°, STI-571) in the treatment of vestibular schwannoma. Otol Neurotol

Relationship between FDG uptake and the pathological risk category in gastrointestinal stromal tumors.

TM: Schwannoma of the esophagus: computed tomography and endosonographic findings of a special type of schwannoma. Acta Radiol

Upper gastrointestinal bleeding due to a malignant Schwannoma of the stomach.

file:///data/remote/core/dit/data/Springer-OA/pdf/faf/aHR0cDovL2xpbmsuc3ByaW5nZXIuY29tLzEwLjExODYvMTc1Mi0xOTQ3LTYtMzcucGRm.pdf

Gastric malignant schwannoma presenting with upper gastrointestinal bleeding: a case report

Abstract

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