gastric neuroendocrine tumours ( nets ) account for more than 20% of all diagnosed gastric tumours . in recent years
, the incidence of these tumours has increased substantially , probably due to an increase in the number and quality of diagnostic tests .
these tumours can be classified into three main types according to their clinical and histological characteristics .
the most common are type 1 tumours ( accounting for 7080% of all gastric nets ) , which present with achlorhydria - induced hypergastrinemia secondary to chronic atrophic gastritis and are commonly associated with b12 malabsorption and pernicious anemia .
most type 1 nets are grade 1 ( g1 ) , with metastasis present in only 25% of patients at diagnosis .
type 2 tumours are the least common ( 56% of gastric nets ) and are sporadic tumours associated with zollinger - ellison 's syndrome or , more commonly , related to type 1 multiple endocrine neoplasms ( men-1 syndrome ) .
type 3 gastric nets are sporadic , often solitary , and usually higher - grade lesions ( g3 being the most common ) .
type 3 nets , unlike types 1 and 2 , are not associated with elevated gastrin levels .
they are typically larger lesions , may be ulcerated , and are capable of inducing carcinoid syndrome ( producing 5-hydroxytryptophan ) .
treatment of metastatic gastric nets is highly complex , and a multidisciplinary approach is considered essential to assure that all therapeutic options are considered . in oligometastatic cases , nccn and
somatostatin analogues have been used to decrease gastrin levels , and there is evidence to suggest that use of these drugs can also reduce recurrences [ 5 , 6 , 7 , 8 ] . here
we present a highly unusual case involving a patient diagnosed with a well - differentiated g2 , type 3 gastric net with exclusive metastatic bilateral ovarian involvement . to our knowledge
, this is the first such case reported in the literature , as the cause of ovarian involvement is usually due to local invasion in the context of patients with major abdominal involvement rather than metastasis .
a 35-year - old hpv-18-postive female , without any previous personal or family medical history of interest , underwent a routine follow - up examination for cervicitis in december 2013 .
a cone biopsy was performed , and a diagnosis of early infiltrating cervical adenocarcinoma was made .
the maximum tumour thickness was 0.7 mm extending 2 mm horizontally and adjacent to an in situ adenocarcinoma ( 5 mm in the largest diameter ) located 2 mm from the upper border of the endocervix .
the clinical stage was pt1a2 . given these findings , together with the fact that the patient was nulligravida but with reproductive desires , a radical trachelectomy plus sentinel node biopsy was planned . in accordance with established protocol ,
pelvic magnetic resonance imaging was performed ( january 2014 ) prior to surgery , revealing a solid , slightly heterogeneous lesion in the right ovary measuring 27  24 mm and an intracapsular lesion ( 10 mm  8 mm ) in the left ovary .
no pathological findings were observed at the cervical level , and no evidence of lymphadenopathy was observed . given these highly unusual radiological findings 
bilateral involvement of the ovaries due to leiomyomas  the multidisciplinary tumour board recommended a biopsy of both ovaries . a bilateral ovarian laparoscopy ( january 29 , 2014 ) revealed a solid 25-mm lesion in the right ovary and a 15-mm lesion in the left ovary .
based on the pathological examination of the intraoperative biopsy , the preliminary finding was a 
right ovarian cyst with areas of carcinoma. consequently , trachelectomy was ruled out and instead a right adnexectomy was performed .
the definitive pathological findings were an infiltrating low - grade net tumour ( ki-67 = 5% ) of likely gastrointestinal origin .
the gastroscopy revealed a tumour measuring approximately 4 cm in diameter with central ulceration located in the greater curvature of the stomach .
the biopsy revealed a well - differentiated , non - gastrin - producing g2 net ( ki-67 = 7% ) .
in addition to the endoscopic tests , somatostatin receptor scintigraphy ( octreoscan ) and fdg - positron emission tomography ( fdg - pet / ct ) were performed to assess disease dissemination : both tests revealed pathological uptake in the body - fundus junction , in the gastrohepatic lymph nodes , and in the left ovary .
chromogranin levels were 105 ng / ml , while urinary 5-hydroxyindoleacetic acid ( 5-hiaa ) levels were 21 mg / day .
2 show the results of the fdg - pet / ct scans . based on these findings
, the case was presented to the multidisciplinary net board , summarized as follows : 35-year - old female with a history of endocervical microinfiltrating adenocarcinoma leading to an incidental diagnosis of a well - differentiated g2 gastric net with metastatic nodal involvement in a single locoregional area with bilateral involvement of the ovaries , treated previously with right adnexectomy . given the patient 's young age and lack of comorbidities , the tumour board  following clinical guidelines  recommended surgical treatment of the affected structures ( left ovary , stomach , and gastrohepatic lymph nodes ) . in late march 2014 ,
a complete laparoscopic gastrectomy was performed , with esophagojejunal anastomosis and d1 lymphadenectomy expanded to include nodal stations 79 .
the postoperative pathological report showed the following : complete gastrectomy with a well - differentiated g2 net invading the entire gastric wall into the serosa , with metastasis in 1 out of 17 dissected nodes .
on immunohistochemistry , the tumour was positive for ae1ae3 , chromogranin , synaptophysin , cdx-2 , and negative for gastrin . given these findings ,
consequently , a laparoscopic hysterectomy with left adnexectomy was performed in may 2014 without complications . in the postoperative period ,
the patient progressed favourably with acceptable gastrointestinal tolerance except for clinical signs of occasional dumping .
pathological findings from the surgical specimens indicated metastasis in the left ovary from a well - differentiated g2 tumour , no evidence of disease in the uterus , and foci of mild dysplasia in the endocervix . after both surgeries , a thoracoabdominal - pelvic ct and an octreoscan ( fig .
the images showed that the patient was free of macroscopic disease , and laboratory results for chromogranin a and 5-hiaa acid levels were normal . in july 2014 ,
the patient had recovered from both surgeries and was in good general condition with tolerable symptoms associated with early onset menopause and total gastrectomy ( occasional episodes of dumping in addition to hot flushes ) .
currently , the patient is free of disease , with excellent performance status ( 100% ) .
the case reported here involves a young woman with an incidental diagnosis of a type 3 , g2 gastric net with bilateral ovarian involvement reminiscent of krukenberg 's tumour , which is typical in gastric adenocarcinomas . at present , the route of spread from the stomach to the ovaries in such cases is not well understood , although diverse mechanisms have been postulated , including lymphatic dissemination ( case reports have described gastric adenocarcinomas confined to the mucosae with lymphatic infiltration and ovarian metastases ) , and haematogenic or peritoneal dissemination [ 10 , 11 ] .
gastroscopy is the main diagnostic and follow - up technique in patients with a gastric net .
ultrasound - guided endoscopy is indicated to assess the extent of gastric wall invasion . for full staging ,
the present case was managed in accordance with published guidelines , which recommend surgical treatment of oligometastatic disease whenever possible [ 3 , 4 ] .
after surgery , the patient was free of disease , and we elected to perform active surveillance with regular ct and octreoscans .
we did not administer any adjuvant treatment in this case because the current evidence does not support the use of adjuvant therapy in patients without postsurgical evidence of disease .
gastric nets should be managed in a center with an experienced multidisciplinary team that can offer the patient a full range of therapeutic alternatives .
although progress has been made in the systemic treatment of advanced nets in recent years , systemic treatment options remain limited [ 5 , 6 ] .
other reported cases are mostly in the context of very advanced disease in which curative - intent surgery is not feasible . in this case ,
however , in patients with a positive octreoscan following surgery , adjuvant treatment with somatostatin analogues may be beneficial in low - grade tumours such as the one reported here
written informed consent was obtained from the patient for publication of this case report and any accompanying images .
a copy of the written consent is available for review by the editor of this journal .
the translation and editing of the manuscript was funded by ipsen pharma s.a . , who also paid for the article processing charges related to publication .
 treatment of metastatic gastric neuroendocrine tumours ( nets ) is challenging . in oligometastatic cases , 
 surgical resection is recommended whenever possible . 
 somatostatin analogues have been used to decrease gastrin levels , and available evidence suggests that these drugs can also reduce recurrences . here 
 we present a highly unusual case involving a patient with a well - differentiated grade 2 , type 3 gastric net with exclusive metastatic bilateral ovarian involvement . to our knowledge , this is the first such case reported in the literature , as the cause of ovarian involvement is usually due to local invasion rather than metastasis . 
 we believe this case is of interest not only due to the unusual presentation , but also because it makes us consider adjuvant treatment with somatostatin analogues in patients with low - grade tumours and a positive postoperative octreoscan . 
Introduction
Case Presentation
Discussion
Conclusion
Statement of Ethics
Disclosure Statement