Subglosso-palatal membrane (SPM) is an extremely rare entity and only a few neonatal cases have been reported in the literature. Anatomically, SPM is a fibrous tissue, extending from the floor of the mouth to the palate. Majority of the cases are recognized during the first feeding with the inability of opening the mouth and latching, thus feeding difficulty is the major sign that prompt the physician to consider the diagnosis. Simple surgical resection of the fibrous band is sufficient for the treatment; however, the practical importance of this entity is that physicians should keep in mind the possible co-occurrence of other congenital anomalies such as cleft lip and palate, micrognathia, microglossia and temporomandibular joint disorders in these infants. We present the case of a 3230g male infant was born at 40 weeks gestation to a 30-year-old gravida 2 para 1 mother via cesarean section. Upon delivery he was noted to have severely restricted oral opening due to a mucous intra-oral band vertically connecting the floor of the mouth to the midline of the hard palate. Tongue movements were intact. The pregnancy history was unremarkable, the mother denied having taken any drugs, there was no family history of congenital anomalies or consanguinity. Immediate surgical resection of the aberrant tissue was performed in the operating room with minimal hemorrhage following excision. The physical examination of the infant was otherwise unremarkable and detailed genetic assessment was also normal. The infant was discharged home on full oral feedings on day 5.


Primary extra-nodal non-Hodgkin lymphoma (PE-NHL) arising in the region of the buttocks is rare. After reviewing the literature from the last 20 years, we found only 3 reported lymphomas originating from soft tissue of the buttocks. In our case, positron emission tomography/computed tomography (PET/CT) was performed for the first time, both before and after treatment, to determine the initial stage of PE-NHL and the curative effects of treatment.We report the case of a 71-year-old woman who was admitted to our hospital due to pain, skin redness, rising skin temperature, and swelling in the right hip.After an initial misdiagnosis of local infection, a histological examination and PET/CT were performed which revealed evidence of non-Hodgkin marginal zone B cell lymphoma of Ann Arbor stage II.Following unsuccessful treatment with cephalosporin, the patient was successfully treated with rituximab combined with cyclophosphamide, doxorubicin, vincristine, and prednisone (R-CHOP) chemotherapy.Comparison of the PET/CT scans taken before and after treatment showed that the lesion size had decreased, as had the fluorodeoxyglucose (FDG) uptake seen in the subcutaneous tissue of the right buttock with standardized uptake value max (SUVmax) 11.6 versus 2.5, respectively. Subsequently, no relapse or distant metastasis has been detected.Young doctors should suspect PE-NHL in similar cases. PET/CT is valuable in the diagnosis and treatment of PE-NHL, as well as for accurately determining PE-NHL stage and aggressiveness. The most commonly involved extra-nodal organs are the gastrointestinal tract, Waldeyer ring, and nasal cavity, while primary extra-nodal non-Hodgkin lymphoma (PE-NHL) arising from the spleen, soft tissue, mediastinum, and other tissues is rare. PE-NHL arising in buttock soft tissue is extremely rare. Only a few case reports are available in the literature. After reviewing the literature from the last 20 years, we found only 3 reported cases of lymphoma originating from soft tissue of the buttocks.–4 Yang Jing reviewed 3724 cases of lymphoma treated in the First Affiliated Hospital of Sun Yat-sen University from 1999 to 2010. Seven cases of primary soft tissue lymphoma were found. Among these, 3 cases were found in lower extremities, 2 cases in the psoas muscle, 1 case in the back, and 1 case in the sternocleidomastoid muscle. Therefore, this type of lymphoma is likely to be misdiagnosed.Non-Hodgkin lymphoma (NHL) is associated with proliferation and differentiation of various immune cells in the immune response of lymphoid tissues. This type of malignant tumor of the immune system usually grows in the form of solid tumors in abundant lymphoid tissues and organs, including lymph nodes, tonsil, spleen, and bone marrow. These tumors can originate from any part of the body in addition to lymphoid tissues.2A 71-year-old woman was admitted to our hematology department complaining of swelling with pain, skin redness, and increasing skin temperature in the right hip. There was no history of physical or psychological diseases, alcohol abuse, or familial history of similar diseases. Physical examination showed a red mass in the right hip and a long strip of mass in the right groin area; however, no superficial lymph nodes were found. Moreover, there was no enlargement of the liver or spleen. The patient was initially diagnosed with local infection and prescribed cephalosporin which had no obvious beneficial effects.,7 positron emission tomography/computed tomography (PET/CT) scans showed increased non-uniform fluorodeoxyglucose (FDG) uptake, with standardized uptake values (SUV)max of 11.6 in the subcutaneous tissue of the right buttock, SUVmax of 9.1 in the right gluteal muscle space, and SUVmax of 9.9 in multiple nodules on the right pelvic wall and right inguinal region  chemotherapy. Comparison of the PET/CT scans before and after treatment showed that the lesion had decreased, as had FDG-uptake in the subcutaneous tissue of the right buttock, with SUVmax of 11.6 vs 2.5, respectively  by biopsy.Due to lack of both first-hand knowledge and reports in the literature, symptoms of swelling with pain, skin redness, and rising skin temperature are commonly considered typical infection symptoms in the early stages of treatment. Moreover, patients are often initially misdiagnosed as having a local infection and treated with cephalosporins (as was the case with our patient) and other anti-infective drugs, but without obvious effect. We found reports of 3 cases similar to ours through reviewing the literature of the previous 20 years. Some reports have revealed that the sensitivity and specificity of PET/CT for extra-nodal lymphoma were 88% to 97% and 100%, respectively.,13 Therefore, PET/CT may be the most suitable option for the diagnosis of patients who are highly suspected of having PE-NHL but reject invasive examination. PET/CT is vital for improving staging accuracy, and can ensure that patients are neither under- or overtreated. At the same time, PET/CT is one of the best methods for diagnosing the initial stages of PE-NHL and for re-staging after treatment. We concluded that, while destructive tumors originating in buttock soft tissue are more common in soft tissue sarcoma, PE-NHL should be considered in the differential diagnosis. A timely, accurate diagnosis of PE-NHL can clarify the course of treatment and improve the prognosis of these patients.An initial diagnosis of non-canonical PE-NHL may not be made due to lack of effective examination standards. Definitive diagnosis of PE-NHL requires the biopsy of lymph nodes, which is considered the gold standard for diagnosing NHL. Recently, the National Comprehensive Cancer Network (NCCN) guidelines recommended PET/CT for primary staging, early or final response evaluation, and determining the prognosis of lymphoma.Professional English language editing was provided by Editage.Project administration: Xiao Li Dai.Writing – original draft: Xiang Meng Li.Writing – review & editing: Hai Song Zhang, Jian Hong Shi.


Echinococcus granulosus. It is a common health problem in many countries. This condition predominantly affects the liver and the lungs, and the spleen to a less extent . Indeed, it is estimated that SHD occurs in less than 2% of abdominal CE and 0.5–8% of CE cases. Here, we present a case of a 44-year-old Chinese woman with primary giant SHD who experienced pain in the left hypochondrium for 10 days. A combination of abdominal ultrasonography and computed tomography (CT) were used for preoperative diagnosis. Laparoscopic splenectomy was performed without any complications, and albendazole (400 mg per day) was administered postoperatively for 3 months. At 3-, 6-, 12-, and 24-month follow-up, the patient remained symptoms free, and abdominal CT found no signs of recurrence. In addition to this case, we review the previous literature on SHD treated by laparoscopy and reveal that laparoscopic approach is safe and effective for SHD. Particularly, we show that laparoscopic splenectomy is feasible for giant cysts (> 10 cm) at high risk of rupture or compressing other vital structures.Cystic echinococcosis (CE), also known as hydatid cyst, is a zoonosis caused by the tapeworm  Echinococcus granulosus.1 The life cycle of E. granulosus includes a definitive host  and an intermediate host . Humans are also incidental intermediate hosts and are at risk for infection.2Cystic echinococcus (CE), also known as hydatid cyst, is a zoonosis caused by the larval form of tapeworm 1 Splenic hydatid cyst (SHD) is a rare entity, occurring in less than 2% of abdominal CE and 0.5–8% of CE cases.4 In general, the diagnosis of SHD is based on the clinical manifestation, radiological imaging, usually combining abdominal ultrasonography (USG) and computed tomography (CT), and/or complemented by serologic tests. However, a minority of SHD cases can only be confirmed by percutaneous aspiration or exploratory laparotomy and/or histopathological examination. Surgery is the mainstay treatment for this condition, and laparoscopy may be an alternative to open surgery in some cases. Here, we report a 44-year-old female patient with a primary giant SHD excised by laparoscopic surgery and also review the previous literature on SHD treated by laparoscopy. This work is reported in line with the surgical case report (SCARE) criteria.5The initial infection usually occurs in childhood with no symptoms, and the latent periods may be more than several years. This infection predominantly affects the liver followed by lungs at 70% and 25%, respectively.A 44-year-old Han Chinese woman who had pain in the left hypochondrium for 10 days was admitted to the hepatobiliary surgery department at Dazhou Central Hospital, Sichuan Province, China. The patient came from a small county of Dazhou in eastern Sichuan. She moved to Tibet at the age of 32 years where she lived for 10 years. She had a history of contact with dogs and livestock at her hometown and in Tibet. On examination, the left upper abdomen appeared swollen and a mobile tender mass was palpated, but no liver was suspected. In the left upper quadrant, the spleen was enlarged 15 cm below the left costal margin and was slightly tender, without muscular defense areas or signs of peritoneal irritation. The other components of the physical examination were normal.3) and eosinophilia (6.8%). Other biochemical tests were in the normal range.Abdominal USG revealed a 13.5 × 12.1-cm–sized anechoic solitary, thin-wall cyst in the splenic parenchyma . ComputeBased on the clinical manifestation and preoperative imaging findings, an SHD was strongly suspected. However, given the high risk of cyst rupture and obvious pressure symptoms, we did not conduct serologic tests, instead we directly performed laparoscopic splenectomy on the 2nd day of admission after communicating with the patient. A standard four-trocar placement was used to establish access ports. Pneumoperitoneum was produced in the supraumbilical area using carbon dioxide (12 mm Hg), after which a 10-mm port was placed. A 5-mm port was positioned to the left of the falciform ligament below the xiphoid and a 10-mm port was placed on the left midclavicular line as the main manipulation port. An additional 5-mm port was positioned at the inferior pole of the spleen to the left midaxillary line for injection and aspiration. An old rupture was found on the surface of the cyst in the upper pole of the spleen, which adhered to the diaphragm. Any adhesion between the cysts and neighboring organs was lysed. After locating the spleen cyst, gauzes moistened with hypertonic saline were placed around the cyst to prevent potential contamination and anaphylactic reactions. Then, a Veress needle was inserted into the cyst via the incision site, after which about 500 mL of the cyst contents were aspirated and 20% hypertonic saline was injected into the cystic cavity as a scolicidal agent. After a 5-minute waiting period, the hypertonic solution was aspirated from the cystic cavity. This cycle of injection and aspiration was repeated five times. Subsequently, the spleen was totally excised and extracted in an endobag. No bleeding points were seen, and the peritoneum was soaked with 20% hypertonic saline for 10 minutes and washed with 0.9% saline three times. Finally, a tubular drain was introduced through the left port and positioned at each of the splenic fossa and pelvic cavity.The cut section of the cyst capsule appeared curled and soft . The his6 It is a significant public health problem in South America, the Middle East and eastern Mediterranean regions, some sub-Saharan African countries, western China, and the former Soviet Union.8 In China, Tibet and Sichuan highlands are highly endemic for CE, with rates of up to 6.8%.9 Cystic echinococcus affects almost any organ of the body. The liver and lung are affected in approximately 70% and 25% of patients, respectively, and other organs such as the brain, muscle, kidneys, bone, heart, and pancreas in a smaller proportion of patients.1 According to Gupta et al.,10 cysts occur in the liver (55–60%), lungs (30%), kidney (2.5%), heart (2.5%), bones (2%), muscles (1%), brain (0.5%), and spleen (1.5%). Primary involvement of spleen is even rarer and occurs in less than 2% of abdominal CE and 0.5–8% of CE cases.4The WHO has included CE in the list of “neglected tropical diseases.”11 Approximately 30% of patients with SHD are asymptomatic, and the detection of this disease is incidental in most cases. When the size of the cyst is sufficiently large, patients may present with upper left abdominal pain and/or a palpable abdominal mass.13 For CE including SHD, a combination of imaging and serological tests may be effective in diagnosing the disease, especially for patients living in endemic areas.14 Hydatid cysts may be visualized and evaluated with USG, CT, or magnetic resonance imaging (MRI). Ultrasonography is the first-line option for the screening of abdominal hydatidosis,15 its sensitivity for evaluating CE is 90–95% and specificity is 93–100%.18 Many reports suggest that CT has higher overall sensitivity than USG (95–100%).20 Computed tomography is the most effective tool for determining the number, size, and anatomic location of cysts, is better than USG in detecting extra-hepatic cysts, and may also be used to monitor the lesions during therapy and detecting recurrences.22 It is also superior to USG in detecting complications such as infection.23 It has been found that MRI has some advantages in terms of detecting obscure cysts missed by USG and CT and of providing cystic pathological features. However, it is usually avoided because of its high costs.24The clinical presentation of CE depends on the site and size of the cysts. Basically, the cyst grows faster in the lungs, with reaching larger sizes, and slower in the liver, spleen, and kidneys.22 ELISA appears to be the most sensitive and specific among the available assays.25 Force et al.26 compared eight serologic tests among 131 patients with E. granulosus infection. They found that IgG ELISA was the most sensitive (94%) and specific (99%) for the majority of cyst locations. Akbulut et al.,27 who reviewed the 10 previous studies, also demonstrated that ELISA was the best method for diagnosing SHD, with a sensitivity of 71.4–100%. Wuestenberg et al.18 reported that IHA had a sensitivity of 50–100% and specificity of 83–88%. Eris et al.22 pointed that the rate of IHA positivity was 57% in primary SHDs and 100% in secondary cases. Therefore, combining ELISA and IHA may increase the sensitivity. These tests, however, may be negative and limited because of the capsule isolation of parasite from the host’s immune system.28Serology is useful for primary diagnosis and for evaluating response to treatment. Common serologic tests include ELISA, indirect hemagglutination (IHA), immunoelectrophoresis, latex agglutination test, and Western blot.30 According to the WHO diagnostic classification, regarding CE with SHD, stage CE-1 and CE-3a cysts have a single compartment. Such cysts that are < 5 cm may be treated with albendazole alone. Cysts that are > 5 cm may be treated with puncture, aspiration, injection, and reaspiration (PAIR) alone or in combination with albendazole. Management for stage CE-2 and CE-3b cysts, which have many compartments, requires either modified catheterization technique or surgery (with adjunctive drug therapy). Stages CE-4 and CE-5 are inactive cysts that can be managed with observation.31 The optimal choice between these approaches is uncertain because of the low level of evidence. As a traditional therapeutic modality, surgery may be effective in some situations for example, single cyst diameter > 10 cm, superficial cyst at risk of rupture, rupture cyst, cysts compressing vital structures, cysts with secondary infection or hemorrhage, and cysts with many daughter vesicles.32 The radical surgical options used for the management of SHD > 5 cm are mainly divided into splenectomy and spleen-saving procedures .33 Since the first case of SHD treated by laparoscopy was reported in 1997,34 several reports have demonstrated the safety of laparoscopy for SHD treatment compared with an open approach.35 However, sepsis-related mortality rates associated with total splenectomy are 4% and 1.9% in children and adults, respectively.37 Thus, surgical approaches that preserve the spleen are preferred. Some studies have revealed that no obvious differences were observed in disease recurrence, postoperative hospitalization, or complications when total splenectomy or spleen-saving procedures were implemented.38 Arikanoglu et al. and Eris et al. stated that spleen-saving surgery for SHD was more appropriate in young patients and those with peripherally located small cysts, and splenectomy was particularly effective in cysts showing multiple, larger, centrally located, and other organ involvement.33 The goal of surgery is to clear the cyst and eliminate residual cavity. Dervenis et al.39 pointed that laparoscopy may increase the risk of spillage due to elevated intra-abdominal pressure caused by pneumoperitoneum. To avoid fluid spillage, a protoscolicidal agent such as hypertonic saline, cetrimide–chlorhexidine, or polidocanol should be injected into the cyst, the surgical field should be protected with pads soaked in protoscolicidal agents and the peritoneum should be washed with hypertonic saline if spillage occurs. It has been reported that perioperative adjunctive therapy with albendazole (or mebendazole) can soften the cyst, facilitating removal and reducing the risk of disease recurrence by inactivating protoscolices.42 The WHO suggests that albendazole (or mebendazole) therapy should be initiated 4–30 days preoperatively and continued for at least 1 month  or 3 months (mebendazole) postoperatively.44 However, the optimal treatment duration is unclear because of lack of sufficient evidence.Management options for CE include surgery, percutaneous management, drug therapy, and observation stage.56 were included in the analysis, of which 11 articles reported primary isolated SHD and four articles reported spleen and other organs involvement. We searched the PubMed database from the inception through February 20, 2019, using the following keywords “splenic hydatid cyst”, “hydatidosis”, “echinococcosis”, “spleen”, and their synonyms to identify studies in which SHD was treated by laparoscopy and summarize the characteristics of the clinical data. The search strategy initially generated 803 relevant articles. After reading the titles and abstracts, 756 articles were excluded. A total of 47 full-text articles were reviewed in detail. Finally, 15 articles31 the diagnosis of SHD has been established. Thus, we did not conduct serologic tests as they were not considered to be necessary. In terms of management options, because the cyst was giant, superficially located in the upper pole of spleen, and accompanied with high risk of rupture that may cause severe complications ,57 laparoscopic splenectomy was initially performed, which was further supported by intraoperative examination of the old ruptured cyst. Ultimately, pathological examination confirmed the diagnosis of SHD. After surgery, albendazole therapy (400 mg per day) was administered for 3 months in accordance with the WHO recommendations, and the patient had no specific complaints. Postoperative follow-up were regularly performed at 3, 6, 12, and 24 months. The patient was symptoms free and had no signs of disease recurrence. Overall, based on previous literature, WHO guidelines, and this case, we suggest a spleen-saving surgery by laparoscopy or other minimally invasive technologies  for SHD < 5 cm, and ≥ 5 cm with a single compartment or daughter vesicles, or located in the spleen periphery in younger cases. In addition, laparoscopic splenectomy is suitable for SHD > 5 cm, particularly > 10 cm, with superficial cyst at risk of rupture or located in the central segment of the spleen, or cyst compressing vital structures or with secondary infection or hemorrhage and multiple cysts due to inoculation or via secondary spread from other organs.In our case, according to the medical history and imaging findings, which were even though different from the typical CE features described in most previous studies and WHO classification of CE-1 (“snow flake sign” in USG),Although SHD is rare, its diagnosis can usually be established based on the history of the patient such as living in an endemic area, and symptoms of CE examined by abdominal USG and/or CT. For a certain type of CE , it can be diagnosed by an additional serologic test, abdominal exploration, percutaneous aspiration, or biopsy. The present case study and the literature review indicate that laparoscopy for SHD is safe and effective and seems to be a good alternative to open procedure. Moreover, total splenectomy is suitable for giant cysts (> 10 cm) at high risk of rupture or compressing other vital structures.


Interstitial Ectopic Pregnancy (IEP) is an uncommon type of ectopic pregnancy with the risk of rupturing and bleeding. The incidence of IEP is about 2-4% of all EPs. The diagnosis and management are challenging. We present a well-timed and managed case of IEP.The case was a 37-yr-old woman presented at the Royan Institute with a chief complain of sudden onset of pelvic pain and moderate vaginal bleeding, three weeks after her positive pregnancy test. She had got pregnant with in-vitro fertilization procedure. She was admitted for a two-dimensional ultrasound (2DUS). The 2DUS findings showed a gestational sac with live embryo and yolk sac which was located high in the fundus and eccentric to the endometrium. The suspicion of IEP rose after the 2DUS findings, the confirmation of further diagnosis was then done by three-dimensional ultrasound, and the treatment was done by laparoscopy. The patient underwent laparoscopic left corneal resection. She was discharged after two days and her β-hCG achieved complete resolution (According to the life-threatening complications that are associated with IEP, acquaintance and suspicion about IEP is important. Specified information that obtained by three-dimensional ultrasound could be useful for exact locating and detection. Interstitial ectopic pregnancy (IEP) is a type of ectopic pregnancy that occurs in the interstitial or intramural segment of fallopian tubes. The interstitial part is the proximal portion of the fallopian tube (1). The incidence of IEP is about 2-4% of all ectopic pregnancies (EPs) (2). Common etiologies are: a previous EP, in-vitro fertilization, pelvic inflammatory disease, and previous ipsilateral salpingectomy (3). The diagnosis and management are challenging (4). In interstitial pregnancy, the risk of hemorrhage and bleeding is higher than the other types of EP and can occur at any time in early pregnancy, so early diagnosis of IEP is crucial here (5). Ultrasonography especially three-dimensional ultrasound (3DUS) facilitates an early diagnosis and increases the success of conservative management for the prevention of rupture in most cases of IEP (6). Here we present a case of IEP with an aim to note the detection steps in favor of its facile management.A 37-yr-old woman presented at the Royan Institute with a chief complain of sudden onset of pelvic pain. She had a history of 10 yr of infertility and hypothyroid. Her past history of surgery was diagnostic laparoscopy, hysteroscopy, and myomectomy. She became pregnant with in-vitro fertilization procedure. Three weeks after a positive pregnancy test, she came to the Royan Institute with sudden pelvic pain. Although she had moderate vaginal bleeding, she was hemodynamic ally stable and her laboratory test was normal. The β-Human Chorionic Gonadotropin (β-HCG) level on admission was 6550 mIU/mL, so she was admitted for two-dimensional ultrasound (2DUS). The 2DUS findings showed a gestational sac (GS) with live embryo and yolk sac which was located high in the fundus and eccentric to the endometrium . The suspicion of IEP was raised by the 2D ultrasonography findings.Next, images by the 3DUS were obtained with the Medison A30 machine and the findings were reported as follows: GS with regular margin was seen at the left fundal portion of the uterus which was surrounded by myometrium. Also, the decidual reaction of endometrium was seen in the uterine cavity. Fetal pole and yolk sac could be detected in the GS. Gestational age was about six weeks and five days .Based on the patient's condition, it seemed that the case would be managed laparoscopic ally. The patient underwent laparoscopic left corneal resection. There were adhesions around the uterus due to the previous myomectomy and all of the adhesions were resected.Harmonic scalpel used for fallopian tube. Thickness and vascularity were seen in the junction of the uterine and fallopian tube. Bleeding was coagulated with bipolar cautery. Hemostasis was secured with bipolar cautery and the tube removed.The removed tube was put in anterior cul-de-sac and the corner of uterus was stich with 1-0 vicrryl. .Then the specimen was removed with adobos and was saved for pathology examination. Finally, the gas existed through umbilical trocar. All trocars were removed under the vision, the incision were closed with a Monocryl. The procedure lasted 2 hr. Considering that pre- and postoperative hemoglobin and another hemodynamic index of the patient were normal, there was no need for blood transfusion. The patient's postoperative course was uneventful. She was discharged after two days and her β-hCG achieved complete resolution  (7).Coronal plane in the 3DUS improved the spatial orientation of EP in relation to uterine cavity obtained in sonography. Implantation occurs in the interstitial portion of the fallopian tube which has more elasticity potential than another part of the fallopian tube, so this portion could be extended without any symptom typically , hence it could be misdiagnosed in the early scan. IEP occurs at a vascularized portion of the uterine so the probability of life-threatening bleeding should be considered. If the rupture occurs, severe bleeding will happen and it is life-threatening for the patient (5). So early diagnosis by 2DUS and 3DUS as a non-invasive initial approach to diagnosis is very crucial here (9).The most important finding in IEP are: 1- an empty uterine cavity without gestational sac, 2- a chorionic sac which is take placed in lateral corner of uterine (the distance more than 1 cm from uterine cavity), 3-endometrial thickness less than 5 mm.and a thin myometrium surrounding the GS  which is the best diagnostic clue .et al. reported that laparoscopy has both diagnostic and treatment advantage in their case report (7).On the other hand, the IEP must be diagnosed with “septate or bicornuate uterus and from fibroids or myometrial contractions” (13). Laparoscopy has the diagnostic and treatment value in our case. Sharma Recently, using ultrasound and clinical expertise, delay diagnosis of IEP became less frequent. The progress in 3D TVUS and its coronal view makes it a precise diagnostic tool for the early diagnosis of IEP and differentiation of it from another type of pregnancy .All contributing authors declare no conflict of interest.


The association between the Abernethy malformation, a rare vascular anomaly in which the portal blood is diverted into the systemic circulation, and development of hepatic tumours is well established. We present a case of multifocal hepatocellular carcinoma (HCC) in the presence of extrahepatic portosystemic shunt with a diminutive portal vein . Abdominal ultrasound performed on a 72-year-old female presenting with elevated liver function tests found a 5.6 cm right hepatic lobe mass. Subsequent CT and MRI examinations demonstrated multifocal lesions. A diminutive portal vein was present (transverse diameter of 7 mm) with a large tortuous complex shunt (maximum transverse diameter 2.0 cm) arising at the portal vein bifurcation with branches connecting to the left renal vein and inferior vena cava. Review of a CT examination performed 10 years ago demonstrated a normal-sized portal vein (transverse diameter of 1.5 cm) with a smaller calibre portosystemic shunt (maximum transverse diameter 9 mm). To our knowledge, this is one of the first reports to demonstrate the evolution of progressive portosystemic shunting and the development of HCC. There is a documented association between congenital extrahepatic portosystemic shunts (CEPS) and the development of both benign and malignant tumours. While the pathogenesis of these lesions remains unclear, nearly one half of all cases of CEPS are associated with nodular liver tumours, most commonly regenerative nodular hyperplasia and focal nodular hyperplasia.A 72-year-old female was referred for ultrasound evaluation of the liver secondary to worsening liver function tests. The patient’s history included obesity, for which she had undergone sleeve gastrectomy 2 months ago. Her liver function tests had been slightly elevated in 2012, but had progressed by the time of current presentation .Ultrasound examination demonstrated a right hepatic lobe hypoechoic solid mass measuring 5.6 cm in transverse diameter. Further evaluation with contrast-enhanced CT scan showed a mildly nodular hepatic surface contour with two heterogeneously enhancing or hyperdense masses containing central areas of diminished attenuation within segments 6 and 7 of the right hepatic lobe . The larReview of the patient's chart showed that she had a CT examination performed 10 years ago. Upon review of images from that examination, the liver surface was again noted to be mildly nodular, but without any discrete mass being present. The portal vein was normal in calibre (maximum transverse diameter of 1.5 cm). The described portosystemic shunt was present; however, it was significantly smaller in calibre compared to the current examination, measuring 9 mm in the transverse diameter .The patient underwent CT-guided biopsy of the larger mass, with pathology results consistent with hepatocellular carcinoma (HCC). The patient is currently undergoing treatment for her disease. The final outcome is yet to be determined.3 In Type 1 shunts, there is complete agenesis of a true portal vein. The splenic and superior mesenteric veins drain into the systemic circulation either at separate locations (Type 1a) or may form a common trunk with a single systemic anastomosis (Type 1b). These malformations, typically present in the paediatric population, have a very high female predominance (approximately 74%) and are associated with other developmental anomalies, including congenital heart defects, biliary atresia and situs inversus.4 In Type 2 shunts, a diminutive portal vein is present with formation of a side-to-side fistula. In contrast to Type 1 anomalies, these patients have no gender predominance and may present at a later stage of life.6 Several anatomical variations of Type 2 malformations have been described, including portorenal and portocaval connections.4Congenital extrahepatic portosystemic shunts (CEPS) are vascular malformations that cause either partial or complete diversion of the portal blood flow away from the liver into the systemic veins. First described by John Abernethy in 1793, the disorder has been classified according to the portosystemic anastomosis.1 CEPS is usually discovered incidentally during work-up for non- specific liver dysfunction, as in our patient.Presenting symptoms of patients presenting later in life include signs of hepatic dysfunction, as in our case, and encephalopathy. Chronic liver dysfunction and cirrhosis are rare but have been reported. The liver disturbance is caused by a lack of hepatocellular nutrition secondary to poor hepatic inflow of blood.2 Rare cases of malignant degeneration to HCC have been described. Even more rare cases of hepatoblastoma and hepatic sarcoma have been published as well.6 As of yet, the exact pathogenesis of tumour formation has not been confirmed. There are some reports that suggest that tumour development occurs secondary to diversion of splanchnic blood flow from the liver, as substances within the splanchnic venous blood  are essential for hepatocyte function and regeneration.7 Furthermore, the increased compensatory hepatic arterial flow could also play a role in neoplastic generation.One well-established correlation with CEPS is the development of hepatic tumours, which includes both benign and malignant processes. The majority of masses described in patients with CEPS are benign regenerative nodules, followed by focal nodular hyperplasia and benign hyperplastic nodules.8 Intervention  is usually reserved for symptomatic treatment, including patients presenting with encephalopathy, coagulopathy and cardiopulmonary symptoms.There is no consensus as to the proper surveillance of these patients, as well as when to intervene. Some authors suggest routine imaging and biochemical surveillance , similar to surveillance guidelines for patients with hepatitis B/C infection.In our patient, sequential CT examinations over 10 years demonstrated progression of the portal venous compromise  and increased shunting (increased shunt transverse diameter), with subsequent development of worsening liver dysfunction and multifocal HCC. As with other previously reported cases, there was no evidence of portal hypertension or chronic sequela of cirrhosis. To the best of our knowledge, this is the first report documenting the occurrence of HCC in a case of increased portocaval shunting CEPS coming to light on serial CT imaging.Patients found to have CEPS are at a higher risk for developing worsening hepatic dysfunction and tumours. A surveillance program may be beneficial for early detection in these patients. As in our case, progression of portosystemic shunting maybe one early feature associated with deterioration.CEPS are uncommon vascular anomalies classified by the complete absence of the portal vein (Type 1) or partial portal vein perfusion (Type 2).CEPS is associated with hepatic dysfunction, as well as benign and malignant neoplastic development.Progression of portosystemic shunting may be indicative of deterioration and tumour development.Informed consent to publish this case (including images and data) was obtained and is held on record.


We present a case of Meigs syndrome in a 19 year old woman. We suspected metastatic ovarian cancer after she had presented in her first pregnancy at 12 weeks gestation. Ultrasound scan had confirmed a complex solid mass in the left adnexa, measuring 7cm x 8cm, a viable 12 weeks pregnancy and gross ascites. She had elevated Ca 125 and serum beta - HCG. She went on to have a spontaneous miscarriage while being worked up for exploratory laparotomy. At laparotomy, a left sided solid ovarian mass 8cm x 10cm with a smooth surface and intact capsule was found. This was later confirmed to be a fibrothecoma at histology. The patient went on to recover without any further reaccumulation of ascites. Meigs syndrome is the triad of benign ovarian tumor, ascites and pleural effusion that resolves after resection of the tumor. It is a rare diagnosis that can only be made after excluding ovarian cancer, which it closely mimics . HistoloWe present a case of a 19 year old subsistence farmer. She presented in her first pregnancy at 12 weeks gestational age. She had been well until 3 months prior to presentation when she developed progressively worsening abdominal distension. The abdominal distention was not in keeping with the gestational age and was associated with vomiting bilious material. There were no associated diarrhea, constipation, urinary symptoms, vaginal discharge or bleeding. She was initially managed at a district hospital before she was referred to a provincial hospital. She decided not to go to the provincial hospital only to present 2 months later to our tertiary hospital casualty with worsening abdominal distention and severe shortness of breath. She had abdominal paracentesis done that produced 4 litres of straw coloured fluid. A right sided pleural tap of 200mls clear fluid was done after confirming pleural effusion on chest Xray. This temporarily relieved her respiratory distress. She had a microcytic anaemia, with haemoglobin of 6.5 g/dl. She was transfused 3 units of packed red blood cells. Her kidney function was normal. An echocardiography was ordered but was not done because of lack of funds. An ultrasound scan done after 4 days showed a normal singleton pregnancy at 12weeks 3 days gestation. Also noted was a complex solid mass in the left adnexa, 7cm x 8cm and gross ascites and pleural effusion. The rest of the abdomen was normal. Serum B HCG was 237000IU/L and Ca-125 was 125U/ml. She had a spontaneous miscarriage while being worked up for laparotomy. At laparotomy 12 litres of straw coloured fluid were drained and 20mls sent for cytology. There was a left sided solid ovarian mass approximately 8cm x 10cm in size with a smooth surface and intact capsule. The right ovary and fallopian tubes were normal. There were no lesions noted on the liver subdiaphragmatic spaces or paracolic gutters. The omentum was grossly normal and there were no palpable lymph nodes. Left salpingectomy and oophorectomy were done and specimens were sent for frozen section. The frozen section report showed a fibrothecoma. Cytological examination of peritoneal and pleural fluid samples showed absence of malignant cells. Biochemistry and microbiology of the fluids were not done due to lack of funds. Patient recovered very well post operatively and was discharged 3 days later. Follow up for 3 months showed the patient had not reaccumulated ascites and pleural effusion and she had recovered well from the operation. The Ca-125 had gone down to 37U/ml.We have presented a case of a 19 year old woman who had a symptomatic complex adnexal mass at 12 weeks gestation, ascites and a pleural effusion who went on to have a spontaneous miscarriage while being prepared for surgery. Among other differentials, the possibility of a ruptured ectopic pregnancy was considered, with the low haemoglobin, an adnexal mass and fluid in the abdomen. However it was quickly ruled out because the patient was haemodynamically stable which was not in keeping with significant haemoperitomeum. She was managed as a patient with probable metastatic ovarian cancer. Adnexal masses in pregnancy have an incidence of 0.1 to 2.4 percent. While the majority of these tumors are benign, the possibility of ovarian cancer should always be considered . WidesprOrganogenesis is mostly complete, minimizing the risk of drug induced teratogenesis. The placenta has taken over hormone production from the corpus luteum thereby minimizing the risk of pregnancy loss due to possible reduction in progesterone levels after tumor resection. Spontaneous pregnancy loss due to fetal abnormalities is likely to have occurred and will not likely be attributed to surgery done after the first trimester. If the ovarian mass persists throughout pregnancy, the risk of malignancy is up to 10% , 9. A siMost benign tumors in pregnancy are benign and they usually resolve spontaneously during the pregnancy. However all pelvic tumors should be evaluated clinically for malignancy potential. If there is significant risk of malignancy, the patient should have surgery as soon as possible, preferably after the first trimester.The authors declare no competing interests.


Wide-field imaging plays an increasingly important role in ocular oncology clinics. The purpose of this review is to describe the commonly used wide-field imaging devices and review conditions seen in ocular oncology clinic that underwent wide-field imaging as part of the multimodal evaluation.Wide-field or wide-angle imaging is defined as greater than 50° field of view. Modern devices can reach far beyond this reporting fields of view up to 267°, when utilizing montage features, with increasingly impressive resolution. Wide-field imaging modalities include fundus photography, fluorescein angiography (FA), fundus autofluorescence (FAF), indocyanine angiography (ICG), spectral domain optical coherence tomography (SD-OCT), and recently wide-field OCT Angiography (OCTA). These imaging modalities are increasingly prevalent in practice. The wide-field systems include laser, optical, and lens based systems that are contact or non-contact lens systems each with its own benefits and drawbacks. The purpose of this review is to discuss commonly used wide-field imaging modalities for retinal and choroidal tumors and demonstrate the use of various widefield imaging modalities in select ocular oncology cases.Clinical examination remains the gold standard for the evaluation of choroidal and retinal tumors. Wide-field imaging plays an important role in ocular oncology for initial documentation, surgical planning, determining the relationship of the tumor to adjacent ocular structures, following tumor size after treatment, and monitoring for recurrence. Clinical examination is the gold standard for the diagnosis of retinal and choroidal tumors that often involve pathology in the peripheral fundus. Historically, imaging technology could easily and clearly capture photographs of lesions located in the posterior pole to the edge of the equator. The first fundus camera had a 20° field of view, later expanded to 30°, and this was set as the standard for fundus photography. Over the last few decades advances in imaging modalities have vastly expanded the field of view photographed and enabled microscopic evaluation of the peripheral retina. Wide-field or wide-angle imaging is defined as greater than 50° field of view with modern devices that can reach far beyond this reporting fields of view up to 267° when utilizing montage images. Montage imaging requires photographic expertise to capture and align the images and often fails to obtain far peripheral views in focus with the posterior pole images. Wide-field imaging has found a particularly important role in ocular oncology for initial documentation, surgical planning, determining the relationship of the tumor to adjacent ocular structures, following tumor size after treatment, and monitoring for recurrence.The wide-field systems include laser, optical, and lens based systems that are contact or non-contact lens systems each with its own benefits and drawbacks Table . CommonlAs imaging characteristics of fundus tumors have been better characterized, using multiple imaging modalities may enhance the diagnostic accuracy of the clinician . Wide-fiAfter diagnosis, wide-field imaging can assist with surgical planning by documenting lesion margins and evaluate the response to treatment with serial images to quantify interval changes. It can also identify the extent of retinal non-perfusion, radiation retinopathy, and other tumor effects that may not be readily apparent on clinical examination.The purpose of this review is to discuss the most commonly used wide-field imaging modalities for retinal and choroidal tumors and demonstrate the use of various widefield imaging modalities in select ocular oncology cases.Retinoblastoma is the most common intraocular malignancy among children. Most often it presents with leukocoria, strabismus, or ocular inflammation and requires prompt examination under anesthesia and treatment once the diagnosis is confirmed. Retinoblastoma can have a wide range of clinical presentations but is classically a gray-white intraretinal mass fed and drained by retinal vessels and can have various associated ocular findings based on the affected structures Fig. Wide-field imaging offers a reliable and quantitative way to monitor response to treatment. Contact lens systems, such as the RetCam 3, are typically used for retinoblastoma evaluation and imaging because non-contact images are difficult to obtain on these young patients in clinic. The RetCam has a more limited field of view, but increased depth compared to Optos. Scleral depression can aid in obtaining the full retinal periphery in the RetCam field of view  malformations, and choroidal hemangiomas. Wide-field imaging particularly FA and ICG offer key diagnostic information for these lesions.von Hippel disease, however, if it is associated with cerebellar hemangioblastomas or other systemic symptoms it is termed von Hippel-Lindau syndrome. Referral for genetic testing, neurologic, nephrological, and systemic evaluation is critical as patients are at increased risk for cerebellar hemangioblastomas, renal cell carcinoma, and pheochromocytoma. Regular ophthalmic evaluation and early treatment of hemangioblastomas can reduce the late complications of this condition including exudative retinal detachment.Retinal capillary hemangioblastomas are a rare angiomatous tumor arising from the retina that presents as a reddish-orange mass with large, dilated feeding and draining retinal vessels. Wide-field imaging and FA play an important role as the lesions are often peripheral and demonstrate a classic FA pattern of rapid filling of the feeding retinal arteriole, later filling of the intralesional blood vessels, followed by drainage through the dilated venule and massive late leakage into the tumor and vitreous Fig. . In one FA is nearly diagnostic of retinal cavernous hemangiomas that are a rare retinal tumor that clinically presents with a “cluster of grapes” appearance. The tumor consists of dilated, thin-walled vascular channels and within these plasma-erythrocyte separation occurs. As a result, the FA reveals a slowly filling lesion with fluorescein that pools at the top of the space (within the plasma segment) and remains there for a prolonged period of time without leakage of the dye. Treatment of these lesions is rarely required, however, referral for systemic evaluation for dermatologic and neurologic manifestations is warranted.Wyburn-Mason syndrome. FA demonstrates characteristic rapid filling of both the afferent and efferent retinal vessels without an intervening capillary bed and no leakage of the dye.Also known as a racemose hemangioma Fig.  representomato ketchup fundus and is associated with encephalofacial angiomatosis (Sturge-Weber syndrome). FA highlights the large choroidal vessels in these lesions in the early phases with late staining of the tumor. Wide angle fundus photography can help monitor the degree and resolution of associated exudative retinal detachments. Sturge-Weber patients require dermatologic and neurologic evaluation for other potentially life-threatening hemangiomas.There are two distinct forms of choroidal hemangiomas: circumscribed and diffuse. Circumscribed lesions are benign, reddish-orange tumors with high reflectivity on ultrasound and do not have systemic associations. These are typically located within the posterior pole and are captured well with traditional fundus imaging. Diffuse tumors present with a saturated red appearing fundus referred to as These lesions represent a benign vascular mass in the retina often found in otherwise healthy individuals. The lesions can cause damage through direct mass effect, or adjacent exudation and resulting macular edema. They often have borders that extend into the mid-periphery that can be visualized with wide-field imaging and FA.The differential for a melanocytic choroidal lesion include choroidal nevus, melanoma, congenital hypertrophy of the retinal pigment epithelium (CHRPE), melanocytoma, adenomas and adenocarcinomas of the nonpigmented and pigmented ciliary epithelium, combined hamartoma of the RPE and retina, metastasis, atypical retinal or choroidal hemorrhage, hemangiomas. Wide-field imaging particularly FA in conjunction with examination, ultrasound, and if necessary tissue biopsy or CT/MRI can assist in determining the correct diagnosis and treatment plan.When evaluating melanocytic lesions, laser projection systems can leave shadows from any anterior opacity including focal cataracts, vitreous opacities, or the lashes or nose, that give the appearance of pigmented lesions or pseudomelanocytic lesions . FurtherChoroidal nevi are incidentally detected in 6–10% of subjects and represent a relatively common ophthalmic condition detected on routine examination , 10. NevOnce a lesion has been determined to be a choroidal nevus, regular follow-up and photographic documentation is recommended at baseline and follow-up. Wide-field photographs can capture the full margins of a nevus in the periphery as well as its relationship to other structures such as the retinal vessels to facilitate future comparisons . AlthougCHRPE is a sharply demarcated, flat, typically darkly pigmented lesion often with lacunae in young adults. Optomap imaging in the optometric population reported a 1.2% prevalence of CHRPE lesions most commonly in the far temporal peripheral retina . The lesChoroidal melanomas are the most common primary intraocular malignancy in adults. They typically present as large, pigmented, elevated, dome-shaped lesions that may be associated with orange pigment, subretinal fluid, RPE changes, or a serous retinal detachment. Orange pigment is due to the contrast of lipofuscin and melanin and is associated with malignant activity. Wide-field FAF enables detailed view of orange pigment and comparison over time Fig. .Clinical examination, multimodal imaging, tissue biopsy and systemic evaluation are critical in suspected uveal melanomas Figs.  and 7. WWide-field imaging offers the advantage of imaging tumors in the periphery  without Wide-angle imaging of melanomas during initial diagnosis documents the initial size of the lesion and its relationship to adjacent structure to monitor response to therapy. Wide-field photography has been reported to yield similar tumor size measurements as ultrasonography when the tumor is less than 3 mm in height and there is no associated subretinal fluid or detachment . There aThe most common intraocular and intraorbital tumor in adults is metastatic disease and thus the recognition of such lesions is critical. The choroid is frequently the site of involvement because metastatic foci spread hematogenously from distant organs. The most common primary site of the cancer varies by gender. Females most often present with breast carcinoma followed by lung carcinoma while males most often present with lung carcinoma . Wide-anRadiation retinopathy is a dose-dependent complication of radiation exposure with presentation similar to diabetic retinopathy Fig. . ComplicOver the last several decades there have been significant advances in retinal imaging technology that have enriched our understanding of various retinal conditions. High resolution images allow for visualization of pigment characteristics, vascular leakage, ellipsoid zone health, details of each retinal layer, presence or absence of the RPE, and recently blood flow and oxygenation, that provide further insight into choroidal and retinal tumors. Modern devices have rapid image acquisition times, short processing times, and offer transmission of photography data to other units or the electronic health record. Wide-field imaging plays an important role in the diagnosis, management, and follow-up of retinal and choroidal tumors as these lesions often extend or present in the periphery or have important vascular changes in the periphery that can be identified and better characterized with wide-field imaging. Machines with multiple modalities of simultaneous, or at least sequential, imaging aid in the evaluation and management of peripheral lesions. For this reason, it is now the standard of care to obtain multimodal imaging of choroidal and retinal tumors in ocular oncology clinic.The exact utility of each camera and wide-field imaging type remains controversial and further research is required in this field. Technology advances at an exponential rate so shortcomings such as resolution, color correction, and field of view will only continue to improve with time. It is important to recognize that despite their proliferation in ophthalmology, wide-field imaging systems have drawbacks that should be considered when evaluating their utility in clinical practice.As these technologies develop, the increasing field of view and resolution offer potential new insights into the pathophysiology of these tumors and better delineation of these lesions for surgical planning. In the future it is likely that various forms of wide-field imaging systems including OCTA will play an increasingly important role across the spectrum of retinal disease including ocular oncology and these wide-field systems may play a role in screening select high-risk populations. More research into these areas is required to better understand define utility of these images in the treatment of patients with retinal and choroidal tumors. Despite these exciting advances in technology, the gold standard for evaluation of choroidal and retinal tumors remains the clinicians examination.


The incidence of endometrial serous carcinoma (ESC) has been increasing, and ESC is resistant to treatment. We report a patient with ESC who responded to radiotherapy for multiple recurrences. The first recurrence was detected in the vaginal wall and left internal iliac lymph node 5 months after the initial treatment. Concurrent chemoradiotherapy (CCRT) was administered. Radiation was delivered using the intensity modulated radiation therapy technique. The second recurrent tumor was detected in the right internal iliac lymph node after 4 months, and CCRT was conducted. After 4 months, the third recurrence was detected in the right common iliac node, and CCRT was performed. After 8 months, the fourth recurrence was detected in the horizontal portion of the duodenum, and radiotherapy was administered. After 9 months, the fifth recurrence was detected in the vaginal wall. Interstitial brachytherapy was conducted. Grade 2 gastrointestinal injury, nausea and radiodermatitis were observed. During the subsequent 13-month follow-up, there has been no recurrence. Although ESC is resistant to treatment, radiotherapy could be effective in some cases. Even when multiple recurrences occur, radiotherapy may be considered a treatment option if the irradiation level is permissible. In Japan, the incidence of endometrial serous carcinoma (ESC) has been increasing . ESC is max) of 15.4 in the vaginal wall and 5.1 in the left internal iliac lymph node, the first recurrence of ESC was diagnosed  was performed  was performed. Tumor regression was observed and the uptake in the recurrent site decreased considerably on the FDG-PET. Grade 2 gastrointestinal fistula, nausea and radiodermatitis  were observed during the treatment. During the subsequent 13-month follow-up, there has been no recurrence.A 68-year-old woman presented to our department with abnormal vaginal bleeding. Her family history was not contributory, and she had no previous medical history. Transvaginal ultrasonography revealed a tumor in the uterine cavity. On endometrial biopsy, the papillary growth of tumor cells was observed. On magnetic resonance imaging (MRI), T2-weighted images showed thickening of the endometrium and contrast enhancement Fig. . On compsed Fig. a, b. Conp = 0.034) [In this study, we report a patient with ESC with local repeated recurrence who responded to five sessions of radiotherapy to the vaginal wall-, the internal-, common-, and iliac nodes, and the horizontal portion of the duodenum. ESC is chemotherapy-resistant and has a poor prognosis . Managem= 0.034) . Based o= 0.034) –15. Seco= 0.034) . In anot= 0.034) . However= 0.034) . In the


Solid tumors of the spleen are rare, with an incidence of 0.007% in all operating and autopsy specimens. In terms of microscopic structure and function, the spleen consists of two parts: the white pulp, which plays an important role in the immune system and the red pulp, which filters the blood.Primary splenic neoplasms can be classified into lymphoid neoplasms arising from the white pulp, and vascular neoplasms which arise from the red pulp.Primary tumors arising from vascular elements include benign lesions such as hemangioma, lymphangioma and hamartoma, intermediate lesions such as hemangioendothelioma, hemangiopericytoma and littoral cell angioma as well as the frankly malignant hemangiosarcoma.It is usually difficult to distinguish a benign from a malignant lesion with preoperative imaging studies and cytological exam by fine-needle aspiration (FNA), that is not easily obtained because of the risk of bleeding.Therefore a splenectomy should be necessary for a definitive diagnosis of splenic tumors.Martel and all for the first time described the sclerosing angiomatoid nodular transformation (SANT), like a vascular lesion of the spleen, with benign clinical course consisting by altered red pulp tissue that has been entrapped by a non-neoplastic stromal proliferative process.We describe a rare case of benign splenic mass documented with FDG/PET-CT , CT and MRI. A 56-year-old female arrived to IRCCS Istituto Nazionale Tumori Milan for a further diagnostic workup and possible treatment of incidentally hypoechoic 5 cm splenic nodule, found with abdominal ultrasonography during a follow-up, for a previous stomach GIST. The contrast-enhanced ultrasonography (CEUS) report, performed in another Institution, described a lesion with centripetal early enhancement, an anechoic central component, a progressive wash-out 44 s after injection. The nodule was evaluated as “suspicious for neoplastic lesion”.1The patient underwent a contrast-enhanced CT, that demonstrated a solitary, round mass, with lobulated margins. On unenhanced phase the lesion had the same spleen’s density. On arterial phase it was heterogeneous, with a lively rim enhancement associated to slight hyperdensity of radiating lines. On venous and delayed phase there was progressive filling. The central scar persisted hypoenhanced in all phases.Axial diameters were 5 × 4.5 cm and craniocaudal distance was 5.2 cm. There was no splenomegaly or lymphadenopathy .Then she underwent fludeoxyglucose  of 4.3, referred as equivocal. No abnormal FDG accumulation has been found elsewhere in the body. Considering the CT findings, a splenic benign vascular tumor was suspected; thus, hematologist and surgeon colleagues decided to perform an MRI .T1 weighted (T1W), without signal intensity drop on out-of-phase T1 sequence and inhomogeneous and predominantly decreased on T2 weighted (T2W). Morphologically the lesion was rounded, with irregular and smooth margins; there were multiple radiating septa inside, caused by the presence of a central hypointense scar. The diffusion-weighted image (DWI), with a b-value of 800 s mm–2, didn’t show signal restriction of mass, that appeared heterogeneous and reflected T2 morphology , on arterials phase we observed a peripheral and septa enhancement, with progressive and centripetal filling, in particular during the delayed phase, with a persistent non-enhancing central hypointense scar .This lesion was considered as an hamartoma and in relation to its dimensions and superficiality (risk of bleeding) a surgical evaluation was required.The surgeons advised for a laparoscopic/laparotomic splenectomy, which the patient refused.Thus she has gone to 3–4 months of follow-up.After 4 months, the MRI revealed the stability of the lesion in terms of morphology and dimension, but confirmed the surgical indication.Finally, after 3 months a laparoscopic splenectomy was performed. At the same time, the surgeons did a partial gastrectomy for the new incidentally mesenchymal lesion of the gastric wall.Grossly, the lesion measured 4.5 cm, it was sharply demarcated from the surrounding splenic parenchyma and was composed by coalescing red-brown nodules.Histologically, the individual nodules had an angiomatoid comprised of round and irregular-shaped vascular spaces with extravasated red blood cells and scattered inflammatory cells. The internodular stroma consisted of variably myxoid to dense fibrous tissue with scattered plump myofibroblasts).Immunostaining revealed different types of vessels in angiomatoid nodules recapitulating the composition on the normal splenic red pulp .Sclerosing angiomatoid nodular transformation (SANT) of the spleen has been described for the first time as a distinct pathological entity in 2004 by Martel et al.2Macroscopically, SANT is a well-demarcated solitary mass composed of dense fibrous stroma containing red-brown nodules. Microscopically, the main findings are multiple angiomatoid nodules separated by collagenous bundles.3SANT lesions were originally thought to occur predominantly in females. However, a recent review of the literature reports that 44.3% of the cases are males, with greater prevalence. between 30 and 60 years of age.4Most lesions are asymptomatic, found incidentally on imaging, and they sometimes presents with abdominal pain.5 because SANT has variable appearance, variable echogenicity, and sometimes internal septa; there is usually internal vascularity on the Color Doppler.US is capable to detect vascular splenic masses, but the ultrasound findings of SANT are inconclusive,6We have found a few reports concerning contrast-enhancement behavior on CEUS; Cao at al. showed a lesion with a diffuse enhancement pattern from 11 s after injection, homogeneously hyperechoic in comparison with the splenic parenchyma in 21 s. It turned out to be isoechoic in 4 s and hypoechoic in 30 s after the injection, with a persistent enhancement up to about 7 min.7A few authors have described CT and MRI findings of SANT; on CT it is solitary and round, with lobulated or smooth margins, iso- or hypoattenuating on an unenhanced scan. After medium contrast injection, the lesion shows peripheral enhancement in the arterial phase, with progressive centripetal filling in a radiating pattern, especially in the delayed phase, while its center continues to be hypodense.T1, hypointense on T2. Following a gadolinium administration, it shows a peripheral and septal enhancing pattern with a hypointense central scar in all sequences; the enhancement persists on venous and delayed phase.8This “spoke wheel” pattern is recognizable also on an MRI; the lesion is usually heterogeneous, iso- to hypointense on 9 DWI  findings correlate well with the pathological findings of SANT, the multinodular high-intensity area as the angiomatoid nodules and the peripheral low-intensity area as fibrous tissues, in both cases described.For Yoshimura et al.However, in the differential diagnoses also other hypervascular focal lesions of spleen, benign or malign, must be considered.T1, and heterogeneously hyperintense on T2, with heterogeneous enhancement on immediate post-contrast images, uniform and intense enhancement on delayed phase, with or without central hypovascular areas.8Hamartoma on a CT appears iso- or hypodense on unenhanced phase and following contrast administration demonstrates heterogeneous enhancement. On an MRI it is isointense on T1- and T2 weighted images, due to hemosiderin deposition.10 After contrast administration, it shows mild heterogeneous enhancement on the immediate post-contrast images, with homogeneous enhancement on delayed imaging.11Littoral cell angiomas of the spleen are always multiple hypoattenuating lesions with sharp margins on non-contrast CT, with an early arterial enhancement, delayed filling and eventually isointense referred to the surrounding splenic parenchyma. They are in 50% of cases hypointense on both T1- and T2 weighted images relative to the spleen. In larger lesions hemorrhage and tumor necrosis areas can appear.8 These lesions demonstrate early contrast-enhancement with variable non-enhancing areas, related to necrosis or hemorrhage.Angiosarcoma has often bigger dimension and more aggressive behavior, with distant metastasis. On CT it can be either solitary or multiple, nodular, poorly marginated masses with heterogeneous attenuation. However, hyperattenuation can be seen in the setting of acute hemorrhage. Some lesions can show peripheral enhancement. On an MRI, these lesions are hypointense on both Actually differential diagnosis with only pre-operative imaging is difficult, because SANT is a rare entity and there are a few images in the literature. Moreover there is a realistic risk of rupture in cases of large spleen masses, so the current direction is splenectomy and hystological diagnosis.In conclusion, SANT is a rare hypervascular benign lesion, recently classified as an independent entity. Knowledge of the typical “spoke wheel” aspect, and of contrast-enhanced pattern, recognizable on CT or MRI, can be helpful in a preoperative diagnosis, avoiding histological examination. A good diagnostic accuracy of DWI MRI imaging is also reported.SANT of the spleen is a distinct benign pathological entity, microscopically characterized by multiple angiomatoid nodules separated by collagenous bundles.At diagnostic examinations SANT morphology can mimic other benign or malignant splenic lesions.Typical morphology and contrast-enhancement behavior on CT and MRI can be helpful for radiologic diagnosis. SANT is usually a solitary, rounded, with lobulated or smooth margins, multiple radiating septa inside, caused by the presence of hypodense/hypointense a central scar. Following intravenous administration of a contrast medium on arterial phase a peripheral and septa enhancement has been observed, with progressive and centripetal filling, particularly during the delayed phase, with a persistent non-enhancing central scar.


Spirometra species or simply sparganum, generally involves subcutaneous tissue or muscle. But occasionally, sparganum can also invade the human brain, resulting in cerebral sparganosis.Sparganosis, a rare and severe parasitic infection caused by the larvae of A 33-year-old woman presented with a 10-day history of headache. Postcontrast magnetic resonance imaging (MRI) revealed an irregular lesion with enhancement and the tunnel-shaped focus extending to the contralateral hemiphere. Cerebrospinal fluid (CSF) analysis disclosed pleocytosis (166 cells/μL) and an elevated protein concentration (0.742 g/L). Enzyme-linked immunosorbent assay (ELISA) revealed positive sparganum-specific antibody in both blood and CSF. Finally, the diagnosis of cerebral sparganosis was comfirmed. She received praziquantel treatment and got a favorable outcome during six-month follow-up.Irregular enhancement and the tunnel sign that extends to the contralateral hemisphere on postconstrast MRI are unusual presentations of cerebral sparganosis. ELISA for sparganum-specific antibody can help confirm the diagnosis. Although surgery is the preferred treatment for cerebral sparganosis, praziquantel might also achieve satisfying outcomes. Spirometra species, spread eggs produced by Spirometra species via defecating. These eggs grow into coracidia in water. Then coracidia are ingested by copepods (first intermediate host) and grow into procercoid larvae. These larvae are infective to frogs, snakes, birds, and mammals in which procercoids mature into plerocercoid larvae [Sparganosis is a severe and uncommon parasitic disease caused by infestation of sparganum , 2. The d larvae , 6. Therd larvae , 8. The d larvae , 10. Whed larvae . Surgeryd larvae .Here, we present a rare case of cerebral sparganosis with unusual MRI findings that remained a diagnostic challenge for neurologists. The patient achieved a good outcome during follow-up with the treatment of praziquantel without surgery. We also reviewed the literature about previous cases of this disease with complete postcontrast MRI.A 33-year-old woman, a nurse, was referred to our hospital due to headache for 10 days. She had a brain CT scan at a local hospital on June 5, 2018, which revealed a hypodensity lesion in the right frontal lobe , cerebral sparganosis is most likely caused by drinking water contaminated by copepod that contains tiny procercoid stage of larva , 12. WheIt has been reported that the characteristics of cerebral sparganosis on CT include the presence of unilateral involvement, low-density lesions in the white matter with ipsilateral ventricular dilatation and localized cortical atrophy, nodular or irregular enhancement with spotty calcification, and changes in the location of lesions . These fPostcontrast MRI is supposed to be superior to CT scan for the diagnosis of most cerebral parasitic diseases , 15. To The differential diagnosis of cerebral sparganosis should include chronic cerebral ischemia, brain tumors and inflammatory granulomas of various causes, such as mycosis, tuberculosis or other parasitic infections , 11, 14.The preferred treatment for cerebral sparganosis is surgical removal. Oral medication, including praziquantel, is deemed unable to kill a live worm or achieve beneficial clinical effects , 9, 30. In conclusion, we report a case with atypical MRI findings of cerebral sparganosis. To the best of our knowledge, the tunnel sign which extends to the contralateral hemisphere on MRI has never been reported. Once cerebral sparganosis is suspected, ELISA for sparganum-specific antibody should be applied. Although surgical removal is generally regarded as the best treatment for cerebral sparganosis, drug treatment, such as with praziquantel, sometimes can achieve satisfying outcomes as well.


Neuroblastoma is one of the most common tumors found in children, and mostly arises in the adrenal gland and paravertebral regions. Orbital neuroblastoma metastasis is relatively rare, and is associated with poor prognosis. Since the symptoms and signs of orbital neuroblastoma are not specific, its diagnosis remains challenging.A 3-year-old girl presented with periorbital ecchymoses (raccoon eyes) and proptosis for 40 days.Abdominal magnetic resonance imaging (MRI) and sonography analysis revealed a large mass in the left adrenal gland (primary tumor). The computed tomography and MRI further revealed multiple soft tissue masses in the skull and both orbits with erosion of the adjacent bones (the metastasis). The histological analysis of the tumor removed from the right orbit confirmed the diagnosis of neuroblastoma.The mass on the right face was surgically removed.The patient exhibited no deteriorative signs at the 6-month follow-up.Clinical manifestations, such as periorbital ecchymoses and proptosis, in combination with radiological analysis and histological findings, are important for the diagnosis of orbital neuroblastoma metastasis. Neuroblastomas are neuroendocrine tumors that can originate anywhere in the sympathetic nervous system, although these are most commonly found in one of the adrenal glands. The clinical symptoms and signs of neuroblastomas are commonly atypical, depending on the location of the primary tumor and metastases. Although metastasis to the orbits has been well-documented for neuroblastomas presenting with various symptoms, such as periorbital ecchymoses (raccoon eyes) and proptosis, neuroblastoma cases with initial symptoms of orbital involvement are rare, accounting for approximately 8% of all neuroblastomas. In the present study, we report a specific case of a 3-year-old girl with neuroblastoma, who presented with initial signs of periorbital ecchymoses (raccoon eyes) and proptosis. In addition, the clinical, radiological, and histopathological studies of orbital neuroblastoma metastasis reported in the literature were summarized.Neuroblastomas are the most frequently identified extracranial tumors that affect children under the age of 4 years old, with an incidence of approximately 1 to 3 in 100,000 cases.2The present study was approved by the Ethics Committee of the Central Hospital of Wuhan, Tongji Medical College of Huazhong University of Science and Technology. All procedures performed in studies that involved human participants were in accordance with the ethical standards of the institutional and national research committee, and the 1964 Helsinki declaration and its later amendments, or comparable ethical standards. A written informed consent was obtained from the patient's legal guardian.A 3-year-old girl was admitted to our hospital presenting with periorbital ecchymoses and proptosis for 40 days. On examination, multiple hard masses were palpated on both the face and head. Bilateral subconjunctival hemorrhages, periorbital ecchymoses (raccoon eyes), and proptosis were identified Fig. A. FundusThe computed tomography (CT) scan highlighted multiple soft tissue masses in the skull and both orbits, with evident erosion into the adjacent bones Fig. B. The maThe mass on the right face was surgically removed. The tumor was cardinal red, with an irregular surface Fig. F. The he3 Neuroblastoma is an undifferentiated malignant tumor that commonly occurs in children, which can rapidly metastasize and widely spread, resulting in a variety of clinical manifestations, such as proptosis, periorbital ecchymosis, abdominal pain, anemia, pancytopenia, bone pain, and paralysis. In the present study, we present a case of a 3-year-old girl with stage IV neuroblastoma, who presented with periorbital ecchymoses and proptosis as initial symptoms. The diagnosis of neuroblastoma was confirmed by radiological analysis of the orbital tumors and an abdominal mass in the adrenal gland, as well as the characteristic histological findings of neuroblastoma. Since clinical manifestations and imaging features are not specific or defined for neuroblastoma, it remains difficult to differentiate neuroblastoma with orbital involvement from other metastatic orbital tumors, such as malignant lymphoma, rhabdomyosarcoma, granulocytic sarcoma, and Wilms tumors. Relevant literature was reviewed, and 10 cases of neuroblastoma with orbital involvement were found by searching PUBMED for published articles between 2000 and 2017 , followed by proptosis , and lytic defects. In the MRI scans, neuroblastomas usually present with heterogeneous low signal intensity on T1-weighted images and high signal intensity on T2-weighted images.Since orbital neuroblastoma metastasis presents with non-specific clinical manifestations, it is often misdiagnosed as child abuse, orbital fracture, or other tumors such as rhabdomyosarcoma. CT and MRI can provide valuable diagnostic information for neuroblastoma identification, especially for pin pointing the primary site of the tumor. A giant hypoechoic mass with hyperechoic areas is the most common abdominal sonographic finding,,12 Moreover, neural markers, such as neuron-specific enolase, TH, and synaptophysin, were often immunopositive in the tumor tissues, while S100 and leukocyte common antigens were immunonegative (Table The histopathological analyses of biopsied orbital masses were often performed to confirm the diagnosis of orbital neuroblastoma metastasis. The histological staining of the biopsied tissues revealed small round cells with a scanty cytoplasm and darkly stained nuclei. cisplatin, doxorubicin, cyclophosphamide, and ifosfamide, are presently available. Other combined therapeutic strategies include 13-cis-retinoic acid, local radiation, resection of the periorbital tumor, and autologous hematopoietic stem cell transplantation. However, in case of more aggressive tumors, drug resistance should be resolved in the future. Targeting techniques by infusion of meta-iodobenzylguanidine conjugated to 131I and 125I are also promising for future developments.The present patient refused any therapeutic regimen. At present, the primary treatment for neuroblastoma is chemotherapy. Multiple protocols with various agents, including carboplatin, vincristine, etoposide, Among the 11 cases with orbital neuroblastoma metastases, systemic chemotherapy was initiated in 6 cases. Although the symptoms improved by follow up after 4 months to 2 years in 4 patients, 2 children died after chemotherapy. Furthermore, although the survival chances for children diagnosed with neuroblastoma have markedly improved over the last 30 years with the advance of multiple treatment modalities, such as chemotherapy, surgery, stem cell transplantation, and immunotherapy, the survival rate for patients with neuroblastoma with orbital metastasis remains low.The prognosis for children with neuroblastoma is dependent on many factors, such as age at diagnosis, disease stage, and histological grade. Orbital neuroblastoma metastases are commonly associated with poor prognosis.Conceptualization: Wan-Ju Yang, Yuan-Yuan Zhou, Shuang Li, Yi Xiang.Data curation: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.Investigation: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.Project administration: Wan-Ju Yang, Yuan-Yuan Zhou, Shuang Li, Yi Xiang.Resources: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.Supervision: Wan-Ju Yang, Yuan-Yuan Zhou, Shuang Li, Yi Xiang.Validation: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.Visualization: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.Writing – original draft: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao.Writing – review & editing: Wan-Ju Yang, Yuan-Yuan Zhou, Fang Zhao, Zhong-Ming Mei, Shuang Li, Yi Xiang.


Endometriosis of the appendix is a very rare entity and commonly affects females in childbearing age. Clinical presentation might be confusing varying from asymptomatic to acute abdominal pain and often mimicks acute appendicitis or chronic pelvic pain. Diagnosis is generally made after pathological examination as operative findings are usually non-specific. This condition poses a diagnostic challenge to radiologists and surgeons altogether and we therefore report a case of a middle aged female who presented with both right lower quadrant and right lower back pain. Recent literature is reviewed and radiological findings discussed. A 40-year-old female with no relevant medical history other than polycystic ovarian disease and resected endometrial polyps presented with both right lower quadrant and right lower back pain lasting for 2 days. No abdominal guarding was displayed at physical examination. Blumberg maneuver was negative. Blood work up tests were normal and transvaginal ultrasound ruled out gynecological disease.The ultrasound revealed a homogenous hypoechoic and thick-walled tubular lesion with regular forms located at the pericecal area; no internal vascularity was noted . The CT The patient was submitted to a partial resection of the cecum including the appendiceal mass. Pathological analysis of the specimen revealed the diagnosis of appendicular endometriosis.The patient was discharged 4 days after surgery and was gynecologically assessed afterwards. Follow-up MRI depicted two foci of endometriomas at the right ovary.Other potential entities to consider include appendicitis and a tumor of the appendix.1,2 Extragenital endometriosis is generally seen in the pelvic cavity and its location and extent considerably vary. The intestinal tract is frequently affected and the involvement might be intraluminal and/or extraluminal.Endometriosis is defined as the presence of ectopic endometrium-like tissue outside the mucosal lining of the uterus. It is usually seen while exploring for pelvic pain, pelvic masses or infertility. It is estimated to affect about 15% of the fertile and 50% of the infertile females. 3] and its clinical manifestations are usually not different from classical acute appendicitis.1,3,4 It may also present as intussusception, obstruction, lower intestinal bleeding and even perforation, especially during pregnancy.4 The operative findings are often not specific,2 the diagnosis being mostly made after pathological examination of the excised appendix. A long history of pain in the right lower quadrant of the abdomen in a female with an intermittent course, who has been known to have endometriosis is the sole clinical sign that can induce to suspect appendiceal endometriosis. However, one must remember that only 41% of these patients will complain of a “cyclic” right lower quadrant pain.1Intestinal endometriosis may clinically present as regional enteritis, appendicitis, ischaemic enteritis or colitis, diverticulitis or a neoplasm. Appendiceal endometriosis is rare; its incidence rate is less than 1% [T1 weighted images and loss of signal—“shading”—on T2 weighted images), but also in pelvic involvement by endometriotic foci, being extremely useful in demonstrating pelvic distortion caused by fibrosis and adherences.5 One must bear in mind that the intestinal wall usually depicts as thickened, with low signal or density because of the hypertrophy of the muscular layer.6 MRI is helpful in differentiating endometriomas from dermoids. Dermoids may also appear hyperintense on T1 weighted images but contain fat. They will therefore display a signal intensity decrease on fat-suppressed images or show chemical shift artifact. CT plays a secondary role for the evaluation of a suspected endometrioma due to its non-specific imaging features generally appearing as complex cystic pelvic masses with high-density fluid components, findings commonly seen in hemorrhagic cysts and neoplasms. Alternatively, a discrete mass might not be visible and non-specific appendiceal dilatation without evidence of inflammation represents the only finding. If endometrial involvement is isolated to the appendix, the imaging features of endometriosis with secondary appendicitis are most commonly indistinguishable from acute appendicitis. Other more typical findings such as complex adnexal cysts and hydrosalpinx may however suggest the diagnosis of endometriosis.Ultrasound is the first-line imaging technique for the evaluation of suspected endometriosis. However, evaluation is usually limited to the detection of endometriomas involving the ovary. The classic “chocolate cyst” appearance of an endometrioma is a homogeneous, hypoechoic lesion within the ovary with low-level echoes and no internal blood flow. Findings are however highly variable, with a spectrum of appearances related to degradation of blood products over time owing to the chronic nature of these lesions. Less common findings such as septations, a fluid–fluid level, a thickened wall, mural nodularity due to retracting clot, a solid mass, calcifications, and others overlap with entities such as hemorrhagic cysts, dermoids, and cystic ovarian neoplasms. MRI provides additional and more specific information not only in endometrioma characterization (with the classic hyperintensity on 8,9Laparoscopy is currently considered the gold standard for the diagnosis of endometriosis and careful examination of the abdominal cavity is performed in order to fully determine the extent of disease. Treatment consists primarily of surgery and is determined by the age of the patient and the severity of symptoms. Visual inspection alone might miss the foci of appendiceal endometriosis; incidental appendectomy is therefore recommended in patients with severe pain. Post-operative follow-up is mandatory.In conclusion, the present case highlights the radiological ancillary features of appendiceal endometriosis consisting of dilatation of the appendix and lack of inflammatory findings in a female who evoked no history of cyclic right lower quadrant pain. This rare condition is mostly diagnosed by histological examination following an appendicectomy performed for a different indication.Endometriosis of the appendix is a rare entity and its pre-operative diagnosis is extremely difficult.Consider its diagnosis in young females complaining of non-specific recurrent lower abdominal pain, especially when a history of infertility is present.Radiological ancillary features often consist of appendix dilation and lack of inflammatory findings.Histopathological examination provides its definitive diagnosis.Appendectomy simply cures the acute symptoms; gynecological follow-up after surgery is required for the underlying invasive endometriosis


Bezoars are rare but may cause gastrointestinal obstruction and ulcers. To the best of our knowledge, only two cases of bezoars in the reconstructed gastric conduit have been reported, but there has been no report on reconstructed gastric conduit obstruction due to bezoars.A 60-year-old man presented to our clinic with abdominal pain and vomiting that occurred suddenly after dinner. Three years before presentation, he had undergone radical thoracoscopic esophagectomy followed by reconstruction of the gastric conduit through the posterior sternum, for esophageal cancer. Enhanced computed tomography scans showed distension of only the gastric conduit without ischemia and distension of the small intestine. According to our findings, we initially diagnosed the patient with postoperative intestinal obstruction caused by adhesions. After conservative treatment failed, the patient underwent an endoscopic study that showed a bezoar at the pylorus ring. We initially failed to remove the bezoar endoscopically because of its large size; hence, we attempted enzymatic dissolution. Three days after the first endoscopic study, the bezoar was disintegrated using a snare and extracted during a second endoscopy. The patient recovered uneventfully and presented with no complications during the 1-year follow-up interval.In cases wherein the discharge of materials in the reconstructed gastric conduit is delayed, bezoars should be considered in the differential diagnosis, and an endoscopic study should be performed to verify the cause of obstruction. Bezoars are defined as masses of indigestible hard materials that form in the gastrointestinal tract  and occuHere, we report an extremely rare case of reconstructed gastric conduit obstruction caused by a bezoar after esophagectomy for esophageal cancer.A 60-year old man presented to our clinic with abdominal pain and vomiting soon after dinner. Three years prior to this event, he underwent radical thoracoscopic esophagectomy followed by reconstruction of the gastric conduit through the posterior sternum, for esophageal cancer. Past medical history was not significant for any medical condition, such as diabetes, or medication that might cause autonomic disorders.On admission, his vital signs were normal, and a routine blood test did not indicate any abnormalities. Physical examination with direct palpation revealed right upper abdominal pain without rebound tenderness. Enhanced computed tomography (CT) scans showed distension of only the gastric conduit without ischemia and without distension of the small intestine Fig. . Based oAt that time, conservative treatment with nasogastric tube drainage and intravenous fluid supplementation was initiated. The patient’s symptoms gradually subsided and oral feeding was initiated 3 days after the conservative treatment. However, immediately after oral feeding, vomiting recurred. An endoscopic study was then performed for further examination, and a bezoar obstruction at the pylorus ring Fig.  was obseWe initially failed to remove the bezoar endoscopically because of its large size; hence, we attempted enzymatic dissolution. Three days after the first endoscopic study, the bezoar was disintegrated using a snare and extracted during a second endoscopy Fig. . The secWe report an extremely rare case of obstruction of the reconstructed gastric conduit caused by a bezoar, 3 years after esophagectomy for esophageal cancer. The patient was treated successfully and did not experience any complications postoperatively.Bezoars grow with the ingestion and accumulation of indigestible materials. The major predisposing factors for bezoar formation in the gastric conduit are delayed gastric emptying, inadequate food mixing caused by poor function of the pyloric sphincter due to truncal vagotomy, and poor digestive function of the stomach because of its use for reconstruction in esophagectomy. . In factOther risk factors for bezoar formation include eating habits, such as excessive persimmon consumption; diabetic gastropathy; cerebral infarction; and medications that reduce gastrointestinal motility . The patIntestinal obstruction is the most prevalent complication of bezoar formation . In one The first choice of treatment for bezoars is removal by endoscopy or a surgical procedure . In our In theory, bezoar formation might be prevented by alleviating delayed gastric emptying. Pylorus drainage procedures, such as pylorotomy and pyloroplasty, are routinely performed in many centers to prevent delayed gastric emptying due to truncal vagotomy, but the efficacy of these procedures remains controversial. On the other hand, Boshier et al. recently demonstrated the safety and efficacy of an intraoperative pyloric stretch procedure for the prevention of delayed gastric emptying following esophagectomy with reconstruction of the gastric conduit. This procedure was performed by passing a Rampley sponge holding forceps through a gastrostomy that was created proximal to the intended gastric resection margin. , 16 AccoTo conclude, considering the possibility that bezoars can cause reconstructed gastric conduit obstruction, this case report highlights the importance of endoscopy-based diagnosis in cases wherein discharge of materials from the gastric conduit is delayed and considering obstruction due to the bezoar as a differential diagnosis in such cases.