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\begin{document}

\title{Malignant Extrarenal Rhabdoid Tumor Derived from the Greater Omentum: A
Case Report and Literature Review}



\author[1]{Xue Shao}%
\author[1]{Wang Xin}%
\author[1]{Lingling Han}%
\author[2]{Luo Shuang}%
\affil[1]{Chengdu University of Traditional Chinese Medicine}%
\affil[2]{Suining Central Hospital}%


\vspace{-1em}



  
  \date{June 14, 2024}


\begingroup
\let\center\flushleft
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\maketitle
\endgroup





\selectlanguage{english}
\begin{abstract}
\textbf{Background}: Extrarenal malignant rhabdoid tumor (MERT) is a
rare and highly metastatic tumor, which is more than 75\% of patients
dying within 6 months of initial diagnosis of MERT. Due to its rarity in
clinical practice, there is no expert consensus or guidelines for the
diagnosis, and it often leads to misdiagnosis and delayed treatment.
\textbf{Case}: This paper reports a 16-year-old girl who presented with
the chief complaint of acute abdominal pain. She underwent laparoscopic
exploration and excisional biopsy, then pathological examination and
immunohistochemistry revealed ``extrarenal malignant rhabdomyoma''. One
month after operation, she died of intra-abdominal hemorrhage and
multiple organ dysfunction. \textbf{Conclusion}: Reviewing the cases of
MERT admitted with acute abdomen reported in the last 10 years, it was
found that these patients had a poor prognosis. The surgery and
chemotherapy is usually beneficial to prolong the survival time of
patients with MERT.%
\end{abstract}%



\sloppy


\textbf{Malignant Extrarenal Rhabdoid Tumor Derived from the Greater
Omentum: A Case Report and Literature Review}

Xue Shao\textsuperscript{1,2}, Xin Wang\textsuperscript{1,2}, Lingling
Han\textsuperscript{1,2}, Shuang Luo\textsuperscript{2,*}

1 School of Medical and Life Science, Chengdu University of Traditional
Chinese Medicine, Chengdu 610000, China

2 Department of Gynecology, Suining Central Hospital, Suining 629000,
China

* Correspondence: 79622499@qq.com

\textbf{Abstract}

\textbf{Background} : Extrarenal malignant rhabdoid tumor (MERT) is a
rare and highly metastatic tumor, which is more than 75\% of patients
dying within 6 months of initial diagnosis of MERT. Due to its rarity in
clinical practice, there is no expert consensus or guidelines for the
diagnosis, and it often leads to misdiagnosis~and~delayed~treatment.

\textbf{Case} : This paper reports a 16-year-old girl who presented with
the chief complaint of acute abdominal pain. She underwent laparoscopic
exploration and excisional biopsy, then pathological examination and
immunohistochemistry revealed ''extrarenal malignant rhabdomyoma''. One
month after operation, she died of intra-abdominal hemorrhage and
multiple organ dysfunction.

\textbf{Conclusion} : Reviewing the cases of MERT admitted with acute
abdomen reported in the last 10 years, it was found that these patients
had a poor prognosis. The surgery and chemotherapy is usually beneficial
to prolong the survival time of patients with MERT.

\textbf{Key words} : Extrarenal malignant rhabdoid tumor,Greater
omentum, Acute abdomen,Chemotherapy

\textbf{Case report}

A 16-year-old girl was admitted to the hospital with the chief complaint
of acute abdominal pain for more than 10~days. A large mass in the
abdomen with clear boundaries was found on abdomina examination.
Ultrasound showed that a cystic about 13cm \selectlanguage{ngerman}× 11cm × 15cm was found in
front of the uterus. The patient underwent laparoscopic exploration for
symptom management and diagnosis. Operative notes illustrated that dense
adhesion of the greater omentum and intestine formed a fish-like mass
with a size of about 15cm× 15cm, with rupture, hemorrhage and necrosis.
Intraoperatively, there was approximately 1000~ml hemorrhage in the
abdominal cavity. Many scattered purplish red nodules between
1.0cm×0.8cm-4cm×4.5cm were seen in the greater omentum. The patient
underwent omentectomy and ovarian biopsy. Postoperative pathological
examination showed that it was a malignant tumor. And
immunohistochemistry showed that CK (AE1/AE3) and Vimentin were
positive, and Bcl-2, CD34, CK20, SMA and WT1 (Wilms' tumor) were
negative. Further detection of INI-1 was also negative. Finally, the
pathological diagnosis of this greater omental mass was MERT. Serum
tumor markers were CA125:1033 U/ml, CA153:15.3 U/ml, and the rest are
within the normal range. Unfortunately, the patient refused further
treated with surgery and chemotherapy. Finally she died of
intra-abdominal hemorrhage and multiple organ dysfunction one month
after operation.

\textbf{Discussion}

Malignant rhabdoid tumor(MRT) is a highly aggressive malignant tumor,
which was first discovered in the kidneys of infants by members of the
Wilms tumor team in 1978.\textsuperscript{{[}1{]}} The tumor is named
because it is composed of cells resembling rhabdomyoblasts by light
microscopy, but having no myogenic filaments in their cytoplasm by
electron microscopy.\textsuperscript{{[}2{]}}MRT in the kidney are most
common in infants and children, but rare in adults. In recent years,
similar lesions in multiple locations outside the kidney have been
reported, such as the central nervous system, liver, uterus, bladder,
pelvic cavity and vulva, which are called malignant extrarenal rhabdoid
tumor (MERT). The occurrence of MERT is extremely rare, and it is
characterized by high metastatic risk, poor prognosis and high
mortality. Due to the absence of specific clinical manifestations and
sensitive examination methods, there exists a dearth of effective
diagnostic criteria and treatment plans.

Reviewing the cases of MERT in recent 10 years, it was found that
ultrasound and abdominal CT scan lacked specificity. There is usually
hypoechoic or isoechoic masses by ultrasound , and there is a mass with
regular shape, clear boundaries, and uneven
density,\textsuperscript{{[}11{]}} or a dense wall and necrotic center
which were been seen by CT.\textsuperscript{{[}3{]}} The tumor can be
characterized by imaging, with difficult to distinguish between benign
and malignant tumor. The final diagnosis is made by pathological and
histological examination. Histologically, the tumor cells are highly
cellular, consisting of monomorphic round or polygonal cells arranged in
solid sheets or in alveolar or trabecular patterns with abundant
eosinophilic cytoplasm containing globular hyaline-like inclusions, and
resemble rhabdomyoblasts.\textsuperscript{{[}4,5{]}} Immunohistochemical
staining for vimentin, cytokeratin, and cytoplasmic epithelial membrane
antigen has generally revealed positive results in malignant rhabdoid
tumors of the kidney and soft parts.\textsuperscript{{[}6,7,8{]}}
Cytogenetic and molecular analyzes have shown abnormalities in the long
arm of chromosome 22 (22q11.2) which encodes the INI-1 gene which is a
core subunit of the SWI/SNF ATP-dependent chromatin remodeling
complex.\textsuperscript{{[}9{]}} INI-1 is ubiquitously expressed in
nuclei of all normal cells. According to literature, some experimental
studies have shown that INI-1 heterozygous-deficient mouse develop
undifferentiated sarcomas with rhabdoid cytologic features, suggesting
that INI-1 is an important tumor suppressor gene. Germline INI-1
mutations have been identified in up to 35\% of patients with sporadic
MERTs, and these patients present very early in life and carry a worse
prognosis.\textsuperscript{{[}10{]}} The immunohistochemical examination
of the patient reported here also confirmed whether INI1 loss indicates
that the mechanism of this disease is caused by abnormal gene
expression.

The clinical diagnosis and treatment of this disease pose significant
challenges. The case reported in this paper is a 16-year-old adolescent
female who was admitted to the hospital with acute abdominal pain. This
condition is often misdiagnosed as a common acute abdomen. Therefore,
for young women with acute abdominal pain, special tumor should be
considered at the time of initial diagnosis. We performed a systematic
review of the literature regarding cases of MERT admitted with
''abdominal pain'' or ''pelvic abdominal mass''. Case reports and center
series were identified by PubMed ''MeSH'' search up in recent 10 years.
Search items included: extrarenal rhabdoid tumor and malignant rhabdoid
tumor. The cases obtained were from 7 manuscripts plus our case report,
for a total of 8 patients to be analyzed (Table 1). According to
literature, MERT is highly malignant, often accompanied by lymph node
metastasis, and even organ metastasis such as liver, lung and bone
occurs in the early stage. The prognosis of these patients is poor.

Table 1\selectlanguage{english}:Reported cases of admitted to hospital with abdominal pain or
pelvic and abdominal mass in the last decade\selectlanguage{english}
\begin{longtable}[]{@{}lllllllllll@{}}
\toprule
\begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{References}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Age/sex}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Clinical} \textbf{Symptoms}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Primary site}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Metastasis}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{INI1}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Laboratory examination}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Laboratory examination}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Tumor marker}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Treatment}\strut
\end{minipage} & \begin{minipage}[b]{0.09\columnwidth}\raggedright\strut
\textbf{Prognosis}\strut
\end{minipage}\tabularnewline
\midrule
\endhead
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{positive}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{nagative}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Haikun Ye et\textsuperscript{{[}11{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
40/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
right upper abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
liver\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Abdomen and pelvis\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
CK 8/18\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}CD56\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}SMA \begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}Vim\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Surgery and Chinese medicine\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Alive (24 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cho IJ et\textsuperscript{.{[}12{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
73/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
right upper abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
cecum\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
lymph node\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}lymph node\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}liver and bone\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
CK\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}VMT\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}MLH-1\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
SMA\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Surgery\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Died (2 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
D'Amico F et.\textsuperscript{{[}13{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
65/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
diffuse abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
colon\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
lymph node\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Vim\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}EMA\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
CK7\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}CD34/31/20/3/45\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}C-Kit\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}OCT-2\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Surgery\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Alive (48 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Voglino C et\textsuperscript{{[}14{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
76/M\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
diffuse abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
lymph nodes\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
CK,Vim\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
CD20/3/117,HMB45,S100\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Surgery\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Dide(2 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\selectlanguage{ngerman}Öfkeli Ö et.\textsuperscript{{[}15{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
70/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
gastric pain\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Stomach\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
lymph nodes and liver\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Vim\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Surgery and chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Xu QH et.\textsuperscript{{[}16{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
67/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Left upper abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Spleen\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
liver, lungs and bones\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
CK\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}EMA\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}Vim\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Surgery\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Recurrence with metastasis(2 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Parker NA et.\textsuperscript{{[}17{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
56/M\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Left upper abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Retroperitoneum and paratticular\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
No distant metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
CD34\begin{CJK}{UTF8}{gbsn}、\end{CJK}\selectlanguage{english}EMA\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
CK AE1/AE3 \begin{CJK}{UTF8}{gbsn}、 \end{CJK}\selectlanguage{english}S100, desmin\begin{CJK}{UTF8}{gbsn}、 \end{CJK}\selectlanguage{english}MSA\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Surgery and neoadjuvant chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.09\columnwidth}\raggedright\strut
Alive(18 month)\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}

Comprehensive analysis shows that due to its rarity, there is a lack of
effective treatment methods. The prognosis of patients admitted to the
hospital with acute abdomen is generally poor and the survival time is
short, which may be related to the acute condition at admission.
Francesco Damiko \emph{et al.}reported a case of a patient with
long-term survival due to the extended surgical
treatment.\textsuperscript{{[}18{]}} Although pathology confirmed node
involvement (13/38 positive) on microscopy, the absence of organ
metastasis at the presentation should be a positive prognostic factor.
However, more cases have shown that surgical treatment alone is often
difficult to prolong the survival rate of patients. Haikun Ye\emph{et
al.} reported that a patient with MERT in the liver had metastases in
the abdomen and pelvis on abdominal CT review one month after modified
right hemihepatectomy, and the patient personally chose traditional
Chinese medicine (astragalus, scubelia, hedyotis, oyster and other
medicinal materials).\textsuperscript{{[}11{]}} After one year of
treatment with traditional Chinese medicine, based on what the patient
described, abdominal CT reexamination showed that the metastatic tumors
had progressed compared with before, but the patient was generally in
good condition without obvious symptoms and signs. It can be seen that
traditional Chinese medicine treatment has a positive significance in
maintaining clinical symptoms, but there is a lack of evidence for
etiological treatment. Nathaniel A. Parker \emph{et al.} reported a case
of a patient whose chest and abdomen CT reexamination three months after
surgery revealed a rapid development of residual lesions, and further
combination therapy with vincristine, doxorubicin plus ifosfamide, and
mesna achieved a good prognosis.\textsuperscript{{[}17{]}} PET/CT
performed 6 months after chemotherapy indicated complete tumor
resolution, and nearly 12 months after stopping chemotherapy, the
patient is still alive, asymptomatic and in complete remission. Naoko
Yasui \emph{et al.} reported that after using VIDE (vincristine,
ifosfamide, neomycin and etoposide) regimen in three MERT patients, both
primary and metastatic lesions were effectively reduced and disappeared,
and the patients received a better prognosis.\textsuperscript{{[}20{]}}
The results suggest that adjuvant chemotherapy has a positive
significance in improving the prognosis. However, Adam D. Wolfe \emph{et
al.} have reported two patients with neck MRT who have no evidence of
disease at seven and four years from diagnosis, following aggressive
multimodal alkylator-based chemotherapy, local control with XRT, and
consolidative high dose chemotherapy with autologous stem cell rescue
(HDC-ASCR) using CEM conditioning.\textsuperscript{{[}21{]}}

Nowsday,there is no guideline about chemoradiotherapy of the MERT.
Through PubMed review of MERT cases using chemotherapy in the past ten
years (Table 2), it can be seen that chemotherapy is beneficial to
prolong survival and bring more opportunities for surgery. However, the
current curative effect of radiotherapy is not exact. Naoko Yasui et al.
reported a case in which the lesion was improved by radiotherapy before
surgery and then recurred, causing the disease to
worsen.\textsuperscript{{[}20{]}} Chemotherapy and surgery were still
used to control the lesion and obtain the opportunity for surgery.
According to the literature, VIDE chemotherapy regimen has some
potential benefits for the survival of patients, but more reports and
studies are needed to confirm it.

Table 2:Reported cases of MERT treated with chemotherapy in recent ten
years\selectlanguage{english}
\begin{longtable}[]{@{}lllllllll@{}}
\toprule
\begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{author}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Age/sex}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Primary site}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Metastasis}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Treatment}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Chemotherapy regimens}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Side effects}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Efficacy}\strut
\end{minipage} & \begin{minipage}[b]{0.08\columnwidth}\raggedright\strut
\textbf{Prognosis}\strut
\end{minipage}\tabularnewline
\midrule
\endhead
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Asada N et.\textsuperscript{{[}19{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
7-year-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
the left cubital fossa\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
preoperative chemotherapy, surgery, and postoperative chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
VDC+ICE\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
hematological toxicity\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Reduction in tumor volume, disappearance of FDG uptake\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Alive (2 year)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Parker NA et.\textsuperscript{{[}17{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
56-year-old/M\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Left upper abdominal pain\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Surgery and neoadjuvant chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
VIDM\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
neutropenic fever and sepsis\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
PET/CT showed complete resolution of the tumor\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Alive(18 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Naoko Yasui\textsuperscript{{[}20{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
2-year-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
the left scapula\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
lung\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
VIDE\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
hematological toxicity and febrile neutropenia\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
The primary and primary lesions disappeared\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Alive(31 month//27 months after VIDE)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
3-year-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
the fifth thoracic vertebra\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
surgery, and postoperative chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
TE for one cycle, CV for two cycles;VIDE for six cycles\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
hematological toxicity\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
TE+CV:aggravation of illness VIDE:stable condition\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Alive(33 month//13 months after VIDE)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
7-year-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
left thigh\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
preoperative chemotherapy, surgery, and postoperative chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
VAC+VIDE\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
hematological toxicity\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
VAC:The primary tumor continued to grow; VIDE:Reduction in tumor
volume\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Alive(23 month/17 months after VIDE)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Adam D. Wolfe\textsuperscript{{[}21{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
13-year-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
neck\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Chemotherapy+Radiation therapy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
VDCCE+ HDC-ASCR utilizing CEM conditioning\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Sequelae of radiotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
stable condition\strut
\end{minipage} & \begin{minipage}[t]{0.11\columnwidth}\raggedright\strut
Alive(7 year)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
19-month-old/F\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
neck\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Alive (53 month)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Konrad M. Szymanski\textsuperscript{{[}22{]}}\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
9-year-old/M\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Urinary bladder\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
No metastasis\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
preoperative chemotherapy, surgey, and postoperative chemotherap and
Radiation therapy\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
VDC+CCE\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
/\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
stable condition\strut
\end{minipage} & \begin{minipage}[t]{0.08\columnwidth}\raggedright\strut
Alive(6 month)\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}

(VDC:vincristine, doxorubicin, and cyclophosphamide;
ICE:ifosfamide,carboplatin and etoposide ; VIDM:vincristine, doxorubicin
, ifosfamide and mesna; VIDE:vincristine(VCR), ifosfamide (IFO),
doxorubicin (DOX), and etoposide(VP); TE: temozolomide and
etoposide;VC:vinorelbine and cyclophosphamide;
VAC:vincristine,dactinomycin and cyclophosphamide; VDCCEC: vincristine,
doxorubicin, cyclophosphamide, carboplatin and etoposide;
CCE:carboplatin, cyclophosphamide, and etoposide)

\textbf{Conclusion}

In conclusion, MERT are characterized by low incidence, high malignancy,
strong invasiveness, and poor prognosis. Relevant examination should be
performed in patients admitted with acute abdomen. After excluding
common diseases, surgical exploration is recommended. The intraoperative
biopsy was taken for pathological examination, and the scope of
operation was guided according to the results of the examination. The
primary lesion and metastatic lesion should be removed as far as
possible during the operation, and the resection margin should be
negative. Postoperative adjuvant chemotherapy with VIDE regimen or
consolidative HDC-ASCR using CEM conditioning was given according to the
results of immunohistochemistry.~When surgical removal of the tumor is
difficult, chemotherapy may be an option to reduce tumor volume. When
the time is ripe for surgery, aggressive~cytoreductive~surgery and
postoperative chemotherapy should be actively performed to improve
survival for patients.

\textbf{Conflict of Interest Statement}

The authors have stated explicitly that there are no conflicts of
interest in connection with this article

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