ATF2 , and a pathological cerebellar change has been reported. Magnetic resonance imaging showed reduced whole-brain size, dilated ventricles, developmental abnormalities of the white matter of the cerebrum, white matter signal abnormalities in the occipital lobe, and abnormal morphology of the cerebellum. Histopathology included previously unrecognized irregular neuronal migration in the subventricular zone around the lateral ventricles in the frontal lobe and white matter rarefaction especially at the level of the occipital lobe in the cerebrum in addition to the cerebellar cortical dysplasia that has been previously described. The findings of this case may highlight the critical role of ATF2 in neurodevelopmental processes in the canine brain.Neonatal encephalopathy with seizures (NEwS) is an epileptic encephalopathy with an autosomal recessive inheritance pattern found in Standard Poodle puppies. The causal genetic variant for NEwS has been identified as a homozygous missense mutation in  A previous study reported clinical information, including electroencephalography (EEG) findings, and cerebellar pathology , an autosomal recessive disease reported in Standard Poodle puppies, is caused by a homozygous missense mutation in the A 64-days-old, 1.9 kg, intact female Standard Poodle dog was presented to the Laboratory of Veterinary Radiology, Nippon Veterinary and Life Science University , with a history of ataxia, generalized convulsive seizures, and weakness. This puppy and the six siblings were born alive at the owner's home, while the other sibling was stillborn. The affected puppy was almost the same size as others at birth, however the abnormalities were apparent by 3 days after birth. The puppy was too weak to suckle without owner's assistance and showed reduced growth compared to the siblings. At the second week after birth, ataxic gait became apparent. Additionally, after the third week of age, the puppy could no longer suckle and forced feeding was carried out by the owner. At that time the puppy did not interact with the siblings and dam, and showed dull responses to external stimuli.This puppy developed generalized convulsive seizures  at 19 daATF2 known to cause NEwS  for DNA testing which confirmed that the puppy was homozygous for a missense mutation  in The body condition was thin. Abnormalities detected by neurological examination included cerebellar ataxia with hypermetria , moderatThe EEG was obtained under sedation using dexmedetomidine (20 μg/kg IV). Three additional administrations of dexmedetomidine (10 μg/kg IV) were given since the dog was not adequately sedated. Electrodes were placed as previously described . Further−3 mm2/s), which was calculated by diffusion-weighted imaging, and a decreased FA value (0.15) when compared with the previously reported values from normal 9-week-old dogs   under general anesthesia with propofol induction and isoflurane maintenance. Details of sequences obtained were described in value 0.1 when comtively)] , which atively)] –9.Whole-body computed tomography was obtained with an 80-slice CT scanner . No obvious abnormalities were found. CSF analysis was not available due to the contamination with blood when obtaining by cisternal puncture. Immediately after the above examinations, the puppy was euthanized, using propofol (30 mg/kg IV) followed by potassium chloride (2 mEq/kg IV).After euthanasia, a complete necropsy was performed. Gross examination showed hypoplasia of the ventral part of the cerebellar vermis  and dilaMild sporadic neuronal atrophy was found in the cerebrum. Undifferentiated cells were multifocally or diffusely distributed in the subventricular zone of the frontal lobe . AdjacenATF2 (c.152T>G) by PCR and Sanger sequencing . Written informed consent was obtained from the owner for the participation of the animals in this study.YY and GJ: conception and design. YY, JC, KK, and RA: acquisition of data. YY: drafting the article. YY, DH, JC, KK, GJ, and KU: analysis and interpretation of data. All authors: revising article for intellectual content and final approval of the completed article.GJ heads a laboratory that conducts fee-for-service DNA testing for NEwS. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Liposarcoma of the spermatic cord is very rare, representing about 7% of para testicular sarcomas. It is considered to be one of the highest malignancy grades.We present a case of a liposarcoma of the spermatic cord in a 45-year-old male complaining of a progressive painless swelling in the right inguinoscrotal region. Ultrasonography and computed tomography findings were compatible with liposarcoma of the spermatic cord. We performed a right radical orchiectomy with a wide resection of the mass. Histological examination confirmed the diagnosis and showed a pleomorphic subtype.The mainstay of management of spermatic cord liposarcoma is wide excision with radical orchiectomy. The most important factors for prognosis are the histologic subtype and surgical margin status. Adjuvant radiotherapy should be considered in cases at high risk for local recurrence. Long-term surveillance is mandatory.Liposarcoma of the spermatic cord is an uncommon para testicular tumor which should be part of the differential diagnosis of inguinoscrotal mass. A radical inguinal orchiectomy with wide resection of the soft tissue mass and the spermatic cord are the key to longest local and systemic disease-free survival. LiposarThis procedure was performed by a junior resident with 5 years of specialized training and it has been reported in line with the SCARE 2020 criteria .2A 45-year-old male with a medical history of right inguinal hernia surgery, presented to our department with a progressive painless swelling in the right inguinoscrotal region that was present for over 6 months, with rapid progression in last 2 months. There was no recent history of trauma, lower urinary tract symptoms, drug intake nor family history of cancer. On clinical examination, there was a firm and nodular scrotal mass. The right testis was not identified during palpation; the trans-illumination test was negative and there was no associated inguinal lymphadenopathy.Ultrasonography (US) revealed a heterogeneous hyperechogenic mass in the right hemi-scrotum with mixed echogenicity. Computed tomography (CT) scan showed a 14 × 8 cm mass lesion in the right hemi-scrotum which contained fat and soft tissue . The US Surgery was performed using a right inguinal approach. A yellowish soft tissue mass with a "bunch of grapes" appearance was closely attached to the spermatic cord; the testis was atrophic. Radical orchiectomy with wide resection of the mass was performed.Gross examination showed a 14 × 12 × 8 cm well-circumscribed, encapsulated and nodular mass , Fig. 3.The patient had a good postoperative clinical course with no complications and was discharged on the third postoperative day. Adjuvant radiotherapy was proposed; however, the patient refused any adjuvant treatment. Follow up with CT scans after 6 months were recommended. There have been no signs of any local recurrence during 1-year follow-up period.3Liposarcomas of the spermatic cord are rare. They occur mostly in adults between 50 and 60 years [The typical clinical characteristics of LSC are painless, non-tender, nodular scrotal mass which progresses slowly and then increases rapidly in size . In our Sometimes the tumors are challenging to diagnose and are often mistaken as inguinal hernia, lipoma, hydrocele, epididymal cyst, or testicular tumors. Initially, inguinal and scrotal masses are evaluated by ultrasound in order to distinguish between intratesticular and extra testicular, solid and cystic masses . Once thTumor staging is based on histological examination and grading, and the presence of metastases. According to the World Health Organization’s International Classification of Disease, there are seven recognized classifications of liposarcoma: well, differentiated, myxoid, round cell, pleomorphic, mixed, dedifferentiated, and not otherwise specified . Low-graDue to its rarity, there is no gold standard treatment for LSC, including pleomorphic subtype. Radical orchiectomy, with wide excision of the soft tissue mass and excision of all potentially contaminated tissues from as close to the deep ring as possible, is the cornerstone of treatment of this neoplasm ,12,13. AThe use of adjuvant treatment (chemotherapy/radiotherapy) remains controversial, due to the paucity of data in the literature. The role of adjuvant radiotherapy is not clear. Two prospective randomized trials about the role of the addition of radiation to surgery in soft tissue sarcoma, showed that adjuvant radiotherapy can improve locoregional control and disease-free survival; however, this advantage did not translate into an overall survival benefit ,12. In aFor patient follow-up, the consensus is to perform close and regular follow-up with imaging at 3, 6, 12 and 24 months [4SCL is an uncommon paratesticular tumor which should be part of the differential diagnosis of inguino-scrotal mass. Up to now radical inguinal orchiectomy with wide resection of the soft tissue mass and the spermatic cord are the key to longest local and systemic disease-free survival. Lymph node dissection is not justified. The role of adjuvant radiotherapy remains controversial; however, it should be considered in cases at high risk for local recurrence, such as pleomorphic SCL. Chemotherapy has not proved its benefice until now.The authors report no declarations of interest.We have no sources of funding for our research.This study is exempt from ethnical approval in our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ebey Babe Nejib: writing the initial draft.Sahbi Naouar: critical revision of the manuscript.Bilel Faidi: critical revision of the manuscript.Rayen Lahouar: writing of the final draft.Badreddine Ben Khalifa: case report design.Rafik El Kamel: final review.Not applicable.Professor El Kamel Rafik.Not commissioned, externally peer-reviewed.


Tumors of the paratesticular region are generally tumors of slow growth, with little symptomatology and, in most cases, benign in nature; in this area, a borderline serous tumor may arise hypothetically from Müllerian metaplasia of the tunica vaginalis, which is histologically identical to its ovarian counterpart. We present a 10-year-old male, with right gynecomastia and ipsilateral hydrocele, showing an enlarged right testicle with a volume of 12 ml and a left testicle with a volume of 10 ml. A right orchiectomy was performed, which presented a poorly defined tan tumor of 1.8 cm that occupied the vaginal and epididymal tunica, and infiltrates the testicular parenchyma. Histological sections revealed a cystic neoplasm, with hierarchical papillary projections, covered by one or several epithelial columnar and hobnail cells with moderate atypia and scant mitosis. Immunohistochemical reactions were performed, resulting positive for PAX-8, epithelial membrane antigen, and CK7, confirming the diagnosis of borderline serous tumor. Since the first reported case in 1986, few have been reported, the majority of these in adults with only three cases in children. In the few cases reported, the prognosis is usually favorable after surgical resection, with disease-free follow-up for up to 18 years. Tumors of the paratesticular region, which includes rete testis, epididymis, mesothelium and vestigial epithelium, and paratesticular soft tissues, are in general tumors of slow growth, with little symptomatology and, in most cases, benign in nature. Epithelial paratesticular tumors with Müllerian characteristics  are rare. The borderline serous tumor is analogous to its ovarian counterpart and was first reported by Young and Scully  in 1986,We present a case of a 10-year-old male patient with this unusual diagnosis.We present a 10-year-old male, son of nonconsanguineous parents, born at 36 weeks of gestation without complications, with adequate growth and development, who presented in 2018 gynecomastia and ipsilateral hydrocele. On the physical examination, both testicles were measured with the Prader orchidometer, showing an enlarged right testicle with a volume of 12 ml and a left testicle with a volume of 10 ml. Hormonal profile was performed, which reported normal results according to his age .Pelvic ultrasound was performed with normal results, computed tomography of the chest reported gynecomastia, and abdominal computed tomography without abnormal findings. In addition, a karyotype of fifty metaphases was analyzed with normal results .The patient underwent surgical correction of the hydrocele. During the procedure, a right paratesticular tumor was found, and orchiectomy was performed. The gross examination showed a testicle with 3.5 cm in diameter, and the cut surface presented a poorly defined tan tumor of 1.8 cm, occupying the vaginal tunica and epididymis and infiltrating the testicular parenchyma .In histological sections, a cystic neoplasm was observed, with hierarchical papillary projections, covered by one or several epithelial layers of columnar and hobnail cells with moderate atypia and scant mitosis Figures –4. There5Paratesticular neoplasms of Mullerian origin have morphological characteristics identical to ovarian Mullerian tumors. However, the anatomical origin is not well defined  In this Due to the oddity of this neoplasm, little is known, not only of its origin but of its natural history. McClure et al.  presentePatients are typically reported with painless masses in the paratesticular region. Most have hydrocele as an initial symptom. Serum markers are usually negative.No specific imaging characteristics are recognized for borderline serous tumors of the paratesticular region . Hsieh eMacroscopically, a multicystic mass surrounded by a dense, pinkish, or whitish capsule can be observed. The average size ranges from 1 to 6 cm, with an average of 3.5 cm of diameter . LocatedHistologically, it is a cystic-looking tumor, with papillary projections with intermingling papillary areas with abundant stroma and micropapillary portions, lined by one or several epithelial layers of columnar or hobnail cells alongside moderate atypia: scarce mitosis, with some psammoma bodies. It is usually positive for the following immunohistochemical markers: MUC-31, estrogen receptors, progesterone receptors, PAX-8, and cytokeratin 7. They have a low Ki67 of 1.3-10% (according to the McClure cohort) . These tIt is of vital importance to establish the differential diagnosis with other entities capable of being located in this area: (1) papillary cystadenoma, which is a benign tumor, the second in frequency in epididymis, usually asymptomatic, associated with Von Hippel Lindau syndrome in 68% of bilateral cases. Histologically, papillary projections in cystic spaces covered by cuboidal/columnar cells of single or double layer are observed, and the cells are clear or have eosinophilic cytoplasm, no mitosis, necrosis, or atypia are present; this histologically differentiates it from the borderline serous tumor (moderate atypia). They are usually positive for CK7, CK AE1/AE3, EMA, vimentin, PAX-8, and carcinoembryonic antigen. (2) Malignant mesothelioma which is a rare tumor in the paratesticular region (66 cases described in the literature) [Most of the cases of paratesticular border line serous tumors were treated with orchiectomy, without proven metastases in the series in which there was follow-up, for up to 18 years . Given tIn conclusion, the borderline paratesticualr serous tumor is a rare tumor in the pediatric age, with apparently benign course; however, more experience with this type of neoplasm is needed to evaluate the biological behavior.


Germ cell tumors account for 15% of anterior mediastinum tumors. Fistulas are abnormal communications between two surfaces covered by the epithelium. A fistula can occur between the bronchial tree and the adjacent anatomical structures secondary to variable etiologies. The main clinical manifestations of bronchial fistulas include hemoptysis, purulent cough, and pneumonia, which might threaten the patient's life. Diagnosis can be established with computed tomography, which shows direct and indirect signs of a fistulous tract. We present the case of a 25-year-old patient, with an embryonic carcinoma of the mediastinum, who developed a fistula between the mediastinal mass and the bronchial tree after chemotherapy and thoracic radiotherapy. We carried out a review of the literature about the epidemiological aspects and the physiopathology and the relevant radiological findings of this pathology. Germ cell tumors correspond to 15% of anterior mediastinum tumors in adults; they occur mainly in the second and fourth decade of life and are more frequent in men . In . In 8]. The 5-year overall survival rate in patients with germ cell tumor treated with induction chemotherapy followed by surgery reported to range from 45.0% to 66.7% .The fistulas require treatment because they are associated with hemoptysis, pneumonia, and acute respiratory distress syndrome compromising the patient's life . The plaThe bronchial tree can develop fistulas with adjacent anatomical structures. A bronchopleural fistula is a communication between the pleura and bronchial tree, this type is the most common. The principal etiology of a bronchopleural fistula is a postoperative complication of a pulmonary resection . Less frThe clinical picture of hemoptysis in a patient with a mediastinal mass should raise suspicion of a fistula of the mass with the bronchial tree. Chest CT allows diagnosis when abnormal communication or indirect signs of this pathology are demonstrated.


Torsion of an accessory spleen is an exceedingly rare cause of abdominal pain in pediatric patients. The diagnosis is frequently challenging as presentation is variable and diagnostic imaging can be aspecific. The current case describes an unusual presentation of a torted accessory spleen in a 5-year-old girl with biliary atresia splenic malformation syndrome who initially presented with non-specific abdominal symptoms and fever. The diagnosis was made following fine-needle aspiration of a suspected intraabdominal abscess. The case highlights the diagnostic challenge of accessory splenic torsion and stresses the importance of its inclusion on the differential diagnosis of pediatric patients, especially those with known splenic or laterality abnormalities, presenting with both acute and sub-acute abdominal symptoms. Accessory spleens or splenules represent a congenital focus of normal splenic tissue separate from the spleen. They are typically asymptomatic and observed in ~10–30% of patients at autopsy . Rarely,A 5-year-old girl was admitted with a 1-day history of generalized malaise, right upper quadrant abdominal pain, vomiting, and fever. Her medical history is significant for mosaic terminal 18q deletion, bronchomalacia, and BASM  status post hepatoportoenterostomy with resulting chronic liver fibrosis.She was initially well appearing with a benign abdominal examination and normal vital signs. Laboratory evaluation including basic metabolic panel, complete blood count with differential, prothrombin time/international normalized ratio, and hepatic function panel were reassuring. Blood cultures were obtained and piperacillin/tazobactam was initiated secondary to concern for possible cholangitis. Metronidazole was added after stool pathogen panel returned positive for the giardia antigen. Interestingly, abdominal x-ray, obtained around time of admission, showed significant colonic stool burden, and a continuous polyethylene glycol infusion via nasogastric tube was run for 24 h with good effect in terms of stool output and relief of abdominal pain.On day 3 of admission, despite broad spectrum antibiotics and negative blood cultures, the patient continued to be febrile and developed marked abdominal distension. Abdominal ultrasound was obtained and demonstrated perihepatic and lower abdominal ascites. Right-sided spleen was visualized with normal parenchymal echotexture. Ultrasound guided paracentesis was performed, with fluid analysis revealing 3,724 total nucleated cells with 71% neutrophils. Peritoneal fluid culture did not speciate an organism, however, at the time, this was believed to be due to antibiotic sterilization. A presumptive diagnosis of spontaneous bacterial peritonitis was made, and after consultation with Infectious Diseases, intravenous ceftriaxone was initiated for better empiric coverage of encapsulated organisms.Despite greater than 48 h of appropriate antibiotics, the patient continued to be intermittently febrile and complain of significant abdominal discomfort and distension. Subsequent abdominal computed tomography (CT) showed a large, rim enhancing fluid collection in the right upper abdomen concerning for abscess see , 2. AfteThe initial treatment approach included conservative management with antibiotics, nasogastric tube feedings and pain control based on the desire to avoid surgical intervention in a patient with a complex medical and surgical history, as well as the noted resolution of splenic infarction without surgery . HoweverThe human spleen is derived from proliferating mesenchyme overlying the dorsal pancreatic endoderm between the tiers of the dorsal mesogastrium during the fifth week of embryogenesis . BetweenAccessory spleens are a frequently observed congenital anomaly occurring in ~10–30% of patients on postmortem studies . Of thosBiliary atresia describes a progressive, idiopathic, fibro-obliterative disease of the extrahepatic biliary tree and is the most common indication for liver transplantation in pediatric patients. Biliary atresia often presents in isolation; however, the condition can present in combination with congenital malformations. In the setting of known splenic abnormalities the condition is referred to as BASM. BASM occurs in ~10–15% of patients with biliary atresia , 20. In Diagnosis of a torted accessory spleen can be challenging, and separating the findings from possible cholangitis, gastroenteritis, bacterial peritonitis, abdominal abscess, and constipation in the above case proved difficult. Maintaining a high index of suspicion, especially in a patient with known splenic anomalies, is paramount as results of diagnostic imaging may be non-specific or unavailable in the emergency setting. As seen in Almost all reported pediatric cases of accessory splenic torsion are diagnosed at time of surgery , 16–18. Accessory splenic torsion is a rare pediatric entity and should remain on the differential for pediatric patients presenting with both acute and non-specific abdomen pain, especially in the setting of known splenic malformations and/or other laterality defects. Once confirmed, conservative management could be considered, but operative management is most common.All datasets analyzed for this study are included in the manuscript.Signed informed consent was obtained from the patient's mother for publication of this case report and any potentially identifying information has been removed.DS, NF, RF, PC, and JF conceptualized, drafted, reviewed, and revised the manuscript. All authors approved the final manuscript as submitted and agree to be accountable to all aspects of the work.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Gastric artery aneurysms are rarely caused by segmental arterial mediolysis (SAM), a condition that often involves multiple vessels. The clinical course of SAM after vessel rupture may vary depending on the involved vessels. For example, the “double-rupture phenomenon” observed following the rupture of the splenic artery aneurysm manifests as a biphasic and relatively slow clinical course. Even in cases of rupture of gastric artery aneurysm, the double-rupture phenomenon has only been reported in two cases so far. However, the rupture was not caused by SAM in either case. Herein, we present the apparent first case of a right gastric artery (RGA) aneurysm rupture caused by SAM that presented with a biphasic clinical course, possibly due to the double-rupture phenomenon.A 54-year-old woman was transferred to the emergency department with severe abdominal pain and a cold sweat for a duration of 3 h. She had developed mild abdominal pain and nausea 3 days earlier. Her vital signs were stable. Physical examination revealed tenderness in the epigastric area. Abdominal contrast-enhanced computed tomography revealed an RGA aneurysm with contrast media extravasation. A diagnosis of hemoperitoneum following a ruptured RGA aneurysm was made, and the patient underwent angiography. However, this modality did not reveal any extravasation from the RGA due to an interruption in the peripheral branch of the artery. Nevertheless, to prevent major bleeding, we performed coil embolization at the point of interruption in the RGA, which we suspected to be a ruptured aneurysm. A distal gastrectomy with Roux-en-Y reconstruction for aneurysm resection was performed the following day. There were no postoperative complications, and the patient was discharged 17 days after surgery. Histologically, the RGA demonstrated multiple vacuoles in the medial muscle layer, which were characteristic of SAM.An RGA aneurysm rupture should be considered a differential diagnosis in patients presenting with hemoperitoneum with a slow or biphasic clinical course. Gastric artery aneurysm caused by segmental arterial mediolysis (SAM) is extremely rare. Rarer still, is the rupture of this type of aneurysm, with only one other reported case to date . SAM is Even in cases of rupture of gastric artery aneurysm, the double-rupture phenomenon was only reported in two cases so far , 8. HoweA 54-year-old woman was transferred to the emergency department with complaints of severe abdominal pain and a cold sweat that had developed 3 h earlier. She had developed mild abdominal pain and nausea 3 days earlier. She had no other relevant medical history. Her vital signs were as follows: blood pressure, 116/84 mmHg; pulse rate, 74 beats/min; and body temperature, 35.7 °C. Physical examination revealed tenderness in the epigastric area. Her laboratory tests were normal except for a low hemoglobin level (10.9 mg/dL). Abdominal contrast-enhanced computed tomography (CT) showed a mass on the lesser omentum with enhancement in the early phase, and the liver surface was surrounded by high-density ascites Fig. a, b. TheA pathological examination of the resected RGA revealed partial dissection of the tunica media and intima and a hematoma that had filled the dissected cavity, indicating rupture of the RGA aneurysm Fig. a, b. MulIn this report, we described the case of a 54-year-old woman who was diagnosed with a ruptured RGA aneurysm and presented with a biphasic clinical course possibly indicative of the double-rupture phenomenon, which is typically observed with splenic artery aneurysm.More recently, advances in diagnostic modalities, including CT and histology, have led to an increased number of reports of visceral artery aneurysms caused by SAM. Based on a study of 117 cases of SAM, Peng et al. reported an average patient age of 57.2 years and a male predominance (67.5%) among affected patients. The most commonly involved vessel was the celiac artery, including the celiac axis and its branches. Overall, 4.3% of patients in that study died, although only one death was SAM-related . This laDespite the rupture of a visceral artery aneurysm, our patient exhibited a biphasic and relatively slow clinical course comparable to a case of splenic artery aneurysm rupture with the double-rupture phenomenon reported by Bokerman in 1930 . Two othIt was difficult to confirm the double-rupture phenomenon in our present case, as imaging studies did not reveal the RGA aneurysm rupture prior to the second bleeding, which spread into the peritoneal cavity. The lack of evidence of a rupture on imaging studies is a limitation of this report. We arrived at a diagnosis of ruptured RGA aneurysm with a double-rupture phenomenon based on the clinical course and the hematoma in the lesser omentum observed on imaging. Few reports have explored this phenomenon other than earlier literature on the ruptures of splenic artery aneurysms. Therefore, the detailed mechanism of the onset of the double–rupture phenomenon remains poorly understood. In this situation, timely diagnosis and management of the ruptured RGA aneurysm prior to hemodynamic instability caused by the second bleeding is crucially important. This case can serve as a reference for the diagnosis of a ruptured RGA aneurysm and the planning of an appropriate treatment strategy, which may include laparotomy, laparoscopic surgery, transcatheter arterial embolization, and other modalities. However, further case reports are needed to clarify the clinical features of a ruptured RGA aneurysm.We reported herein a case of a ruptured RGA aneurysm caused by SAM that exhibited a biphasic clinical course, possibly caused by the double-rupture phenomenon. An RGA aneurysm rupture must be included in the differential diagnosis of a patient with hemoperitoneum who presents with a slow or biphasic clinical course.


Primary hepatic lymphoma (PHL) is a very rare malignancy and constitutes 0.016% of all cases of non-Hodgkin's lymphoma and 0.4% of extranodal non-Hodgkin's lymphoma. We describe a rare case of primary hepatic lymphoma presenting as liver abscess which was complicated with the development of a hepatogastric fistula. A 58-year-old man presented with clinical signs of sepsis, high-grade fever, right upper abdominal pain, and weight loss which had progressed in the past 8 months. Noncontrast abdominal computed tomography (CT) revealed a heterogeneously hypodense lesion in the left lobe of the liver with multiple air foci within, which are seen to extend into the body of the stomach. The patient was initially misdiagnosed as a case of rupture of liver abscess into the stomach. Postoperative liver biopsy examination confirmed a diagnosis of diffuse large B-cell lymphoma. Systemic staging revealed no evidence of nodal or bone marrow involvement, so PHL was diagnosed. Chemotherapy was initiated, but discontinued due to the patient's general condition. Finally, the patient succumbed to neutropenic fever following chemotherapy. Here, we present the exceptional case of a primary hepatic lymphoma with an unusual complication, a hepatogastric fistula, and try through the existing literature to show the difficulties involved in diagnosis and treatment. PHL is a rare form of non-Hodgkin lymphoma (NHL), and it is a tumor confined to the liver without evidence of lymphomatous involvement of the lymph nodes, spleen, bone marrow, or other lymphoid structures. The vast majority of PHL is NHL, most often a diffuse large B-cell lymphoma type. PHL was often misdiagnosed as some other tumor and frequently diagnosed intra- or postoperatively. We report an interesting case of PHL that was difficult to discriminate from a liver abscess, with an unusual complication, hepatogastric fistula, diagnosed after postoperative biopsy report, and try through the existing literature to show the difficulties involved in diagnosis and treatment.3 (neutrophils 82%), and platelet counts . Serum lactate dehydrogenase (LDH), C-reactive protein (CRP), and procalcitonin levels were elevated, but the levels of other tumor markers, such as alpha-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were normal. Serology was negative for hepatitis B virus (HBV) surface antigen, HCV, and human immunodeficiency virus. An urgent noncontrast computed tomography (CT) examination of the abdomen and pelvis was performed which showed a heterogeneously hypodense lesion in the left lobe of the liver with multiple air foci within. The liver lesion was extending directly into the body of the stomach  by CT scan of the thorax did not reveal any lymphadenopathy or mass lesion. FDG-PET scan and bone marrow biopsy were negative. Thus, a diagnosis of primary non-Hodgkin lymphoma of the liver, large cell type, was confirmed. The patient condition did not allow additional complementary surgery, and he was managed with six cycles of R-CHOP regimen, rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone. However, a persistent fever occurred 1 month after three cycles of chemotherapy. The patient succumbed to neutropenic fever following chemotherapy.PHL and hepatogastric fistula are extremely rare. To our knowledge, this is the first report on a primary hepatic lymphoma invading the adjacent stomach, which was complicated with the development of a hepatogastric fistula. PHL was first described in 1965 by Ata and Kamel , 3.Primary hepatic lymphoma is very rare and constitutes about 0.0016% of all cases of non-Hodgkin's lymphoma (NHL) . ExtranoPHL can occur at any age, and the average age of the reported patients is the fifth or sixth decade of life. It affects preferentially men with a male/female ratio of 2–3/1 . PHL maySymptoms are usually nonspecific and include hepatomegaly, gastrointestinal symptoms , right upper quadrant, and epigastric pain. Many cases are diffuse large B-cell lymphoma, and the patients show B-symptomatology of weight loss, fever, and night sweats, as well as fatigue and lethargy. Other rare clinical manifestations include pleural effusion, jaundice, thrombocytopenia, metabolic acidosis, and hypercalcemia . LaboratOn ultrasound imaging, majority are hypoechoic as compared to surrounding normal liver parenchyma. Radiological features of PHL are usually nonspecific, and the most common presentation on the computed tomography (CT) scan is a solitary hypoattenuating lesion, which may have a central area of low intensity indicating necrosis. Other less common radiological findings are multiple lesions and diffuse infiltration patterns . On MRI,Diagnosis is often made upon histopathological and immunohistochemical investigation of the liver biopsy by using percutaneous needle aspiration, laparoscopy, or laparotomy . In our Treatment modalities include surgical resection, chemotherapy, and radiotherapy alone or in combination. Most of the reported cases are diffuse large B-cell lymphomas, but this type is usually aggressive with a relatively poor prognosis . CurrentThe clinical case we present is extremely unusual because there is a coincidence of two very rare features, primary hepatic lymphoma and hepatogastric fistula. Direct invasion to the gastric wall is the basis of the formation of a hepatogastric fistula. Nevertheless, a hepatogastric fistula is a rare complication, even more so for primary liver lymphoma. Such a fistula has been described following transarterial embolization, radiotherapy for hepatocellular carcinoma, percutaneous radiofrequency of hepatocellular carcinoma, pyogenic liver abscess, iatrogenic injury of the stomach, percutaneous catheter drainage of the liver abscess, or by direct infiltration of the stomach by hepatocellular carcinoma .In conclusion, we have reported an exceptional case of PHL with an unusual complication that was difficult to discriminate from a liver abscess with a hepatogastric fistula. The preoperative diagnosis was difficult due to the rarity of the disease, the clinical and imaging manifestations of PHL were nonspecific, and levels of alpha-fetoprotein and carcinoembryonic antigen (CEA) were normal. The diagnosis of PHL should be strictly based on histopathology and immunophenotype. PHL should be considered in the differential diagnosis in a patient with space-occupying liver lesions. Favorable prognosis of PHL can be obtained by early surgery combined with chemotherapy in strictly selected patients.


Cerebral venous sinus thrombosis (CVST) is a rare condition. Symptoms and signs arise from a combination of thrombosis of cerebral veins and increased intracranial pressure. The most common presenting symptom is a non-descriptive headache, but presentation varies with underlying etiology. CVST requires a high index of suspicion to diagnose, particularly in those without apparent risk factors. Evaluation and diagnosis should include a combination of a thorough history, ophthalmoscopic examination, laboratory studies, and imaging. Management is multidimensional and aims to reverse the underlying causes, and prompt treatment with anticoagulation by heparin to decrease thrombotic burden, risk of permanent neurological deficits, and death. In the present study, we report the case of a 61-year-old man referred to the emergency department by an ophthalmologist for bilateral papilledema and eventually diagnosed with CVST secondary to isolated antithrombin III deficiency. Although CVST is uncommon, this case is worthwhile to report as the presentation is unique, and it requires a high index of clinical awareness for timely diagnosis and early therapeutic intervention.  Cerebral venous sinus thrombosis (CVST) is a rare condition that occurs in up to four adults per million per year, has a slightly higher incidence in children aged less than 18 years, and is more common in females given gender-specific situations . The pat2) was referred from an ophthalmology clinic to the emergency department for findings consistent with bilateral papilledema and suspicion for raised intracranial pressure  level at 71%  with IV contrast revealed findings consistent with chronic non-occlusive thrombosis of the left transverse sinus, left sigmoid sinus, and left internal jugular vein at the left internal jugular vein fossa and canal in the posterior skull with extensive collateralization in the head and neck Figure . The patCVST is uncommon, represents less than 1% of all strokes, and is often overlooked due to a non-characteristic headache frequently being the primary symptom at presentation ,7. ThereA number of systematic reviews have described the characteristics of CVST, including its presenting symptoms, risk factors, imaging modalities, and patient outcomes. An overview of recent systematic reviews is useful to further explain the uniqueness of the present case. The presentation of CVST is highly variable; however, it commonly presents with at least one of a few symptoms. The most common presenting symptoms include headache, focal neurological deficits, and seizures ,5,9. HeaAnother challenge to diagnosing CVST is the use of proper imaging to visualize the defect. Head CT is often the initial imaging used, although it is only positive in about 30% of the CVST cases ,9,12. CTTo our knowledge, the present case is unique because it is only one of a few reports of an adult with CVST due to isolated AT III deficiency ,16. The Given the infrequency of reported CVST cases and its non-specific presenting symptoms, more cases that describe risk factors and patients' presentations are necessary to identify patterns that promote early recognition of CVST. Timely recognition and efficient workup, along with prompt treatment with anticoagulation, can prevent potentially avoidable consequences, including death.


Uterine leiomyosarcoma (uLMS) is a rare and aggressive malignancy with poor clinical outcomes. Even when localized, uLMS is associated with high rates of local and distant recurrences that are usually fatal. Common sites of recurrence are lung, liver, pelvic lymph nodes, and vertebral and long bones, though atypical patterns of recurrence have been described. Among them, intracranial recurrence appears as a rare finding, almost exceptional in skull and dura. We describe the case of a solitary skull metastasis from uLMS in a 39-year-old woman, which represents the third reported case of skull recurrence in literature. After multidisciplinary discussion, the patient underwent surgery and received adjuvant radiotherapy. After 4 months, she is currently alive, without evidence of extracranial disease. This case highlights the importance of suspecting and recognizing atypical and extremely rare metastasis to this region. We encourage the need for large case series in order to provide further information about cranial recurrences of uLMS taking into account the paucity of data currently available in literature and the frequently unpredictable behavior of this rare and highly lethal disease. Uterine leiomyosarcoma (uLMS) is a rare malignancy accounting for 1–3% of all uterine cancers and approximately for 65% of all uterine sarcomas (US) . CurrentWe herein report a case of cranial recurrence of uLMS in a young woman, representing to our knowledge the third case of uLMS skull metastasis reported in literature.In January 2018, a 39-year-old woman presented to her gynecologist with menorrhagia and pelvic pain; her past medical history included uterine multinodular fibromatosis. A transvaginal ultrasound showed the presence of an intrauterine mass, for which she underwent a total laparoscopic hysterectomy, bilateral adnexectomy, pelvic and paraaortic lymph node biopsies, and omentectomy. The pathology report described a 7 cm diameter, stage IB uterine mass consistent with grade 3 uLMS. Histology showed smooth muscle actin (SMA) positive and desmin positive spindle cells with marked cytological atypia and 15 mitoses per 10 high power fields (HPF). Tumor necrosis was below 50% and no tumoral invasion was found in the uterine serous and omentum. Postoperatively, the patient received adjuvant chemotherapy with epirubicine plus ifosfamide.Clinical and imaging follow up showed no evidence of recurrence until June 2019, when a total body computed tomography (CT) scan revealed an osteolytic lesion in the right temporal-occipital region, 8 mm in diameter and positive to bone scintigraphy. Physical and neurological examination were normal. The subsequent cranial magnetic resonance imaging (MRI) confirmed the presence of the osteolytic temporal-occipital lesion, with epidural extension and partial compression of the underlying brain parenchyma . MRI shoIn October 2019, she underwent a right craniotomy with removal of the lesion and the involved scalp; epidural extension of the tumor was confirmed. Although surgery resulted in removal of all macroscopic disease, microscopic margins were positive (R1). Histological exam showed the presence of spindle cells with moderate pleomorphism, 15 mitotic figures per 10 HPF , and strAlthough hysterectomy offers the best chance of prolonged survival and it is considered as the standard treatment for uLMS, post-operative recurrence rate ranges from 50 to 70%, with a very poor prognosis . Lungs rTo the best of our knowledge, two cases of skull recurrence of uLMS have been reported in literature to date , 17 Tab. In the Signs and symptoms of skull metastases appears as highly non-specific, depending on tumor extension. This may lead to differential diagnosis, especially after several years of disease-free survival . In thisIf metastatic uLMS is certainly associated with poor prognosis, from a theoretical point of view solitary metastasis may show better clinical outcomes. All the patients reported in literature to date, including our case, showed solitary skull recurrence in the absence of other metastatic foci of uMLS. Given the rarity of metastasis to this region, there is a lack of evidence supporting a specific treatment of intracranial metastases from uLMS, whether metastatic site is brain or skull . ManagemDiagnosis of skull recurrence of uLMS requires a high index of suspicion. Its early recognition can be extremely difficult, especially in asymptomatic, healthy patients in which cranial CT scan is often not performed during post-surgery follow-up. There is lack of evidence for choosing the best therapeutic strategy in these patients and appropriate treatment of intracranial recurrences is yet to be defined.The presented report is the third documented case of skull metastasis from uLMS suggesting the importance of improving awareness of this extremely rare occurrence.Therefore, we encourage the collection of larger series in order to provide further information about cranial recurrences of uLMS, taking into account the paucity of data currently available in literature and the frequently unpredictable behavior of this rare and highly lethal disease.All datasets generated for this study are included in the article/supplementary material.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.AR and MCN have made substantial contributions to conception of the study, interpreted the patient data, and drafted the manuscript. VR performed surgery and has been involved in drafting the manuscript. CG performed the histological and pathologic examination of the surgical tissue and has been involved in drafting figures. AP and PD have been involved in drafting and revising the manuscript. GF, DB, and MD performed radiotherapy and have been involved in drafting and revising the manuscript. JM performed imaging and has been involved in drafting the manuscript. MP has made substantial contributions to conception of the study, interpreted the patient data, and drafted the manuscript. MN was the major contributor in concept and designed and revised the manuscript. All authors read and approved the final version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


A para 4, 35-year-old woman with 3 living issues and farmer by occupation presented to our hospital with symptoms of acute urinary retention for which an indwelling urinary catheter was inserted. There were no associated symptoms of hematuria, frequency, urgency or burning micturition. She also had a history of progressively increasing lump abdomen for past 6 months with dull aching pain. The patient gave a history of having undergone laparotomies twice during her childhood for a liver pathology, but she was unaware of the actual diagnosis. No records were available.A physical examination revealed a 10x8 cm cystic abdominal mass in the left lumbar region. The mass was freely mobile and non-tender. There was another large abdomino-pelvic lump corresponding to 18-20 weeks’ gestation, cystic, mobile from side to side, non-tender, and the lower border could not be discerned. A per vaginal examination revealed a normal-sized uterus with a large 10x15 cm cystic mass felt through the right and posterior fornix.Ultrasound revealed two large multi-septated cystic lesions on the right and left side reaching up to the epigastrium with multiple small cysts seen in both masses with no increase in vascularity, no solid areas, and no free fluid in the abdomen .Contrast-enhanced magnetic resonance imaging (MRI) showed non-enhancing peritoneal and omental-based clusters of non-loculated cystic lesions in the mid abdomen (14x11x11 cm), in the left iliac fossa (6x7x7 cm), and in the pelvis (13x10x10 cm) with multiple small cysts within .The patient was planned for a laparotomy. Intra-operatively, the uterus was normal size. A 10x15 cm clear cyst was seen arising from the left ovary with multiple small cysts with viscous pale, yellow fluid inside them. A similar 2x2 cm cyst was seen arising from the right ovary, and a 20x10 cm cyst and a 5x6 cm cyst were seen arising from the greater omentum . Total aA histopathologic examination revealed hydatid cysts lined by an innermost germinal layer with attached brood capsule and daughter cysts. An avascular, eosinophilic, and refractile laminated membrane was seen. The outer layer was fibrovascular with chronic inflammatory cells and giant cell reaction. To our surprise, even the myometrium had evidence of hydatid cysts .The patient was a farmer and livestock handler by occupation. Ultrasound and MRI findings along with the history of two previous surgeries in the past raised a high index of suspicion for hydatid cyst. Pre-operative echinococcal serology was positive. CA-125 was normal. With a provisional diagnosis of suspected peritoneal and ovarian hydatid cyst, the patient was given a course of albendazole 400 mg twice daily for 6 weeks, following which she was taken for surgery.Echinococcosis or hydatidosis is caused by larvae of tapeworm Echinococcus granulosus, belonging to the family Taeniidae. With dogs being the dominant carriers, Echinococcus often infects humans via echinococcus eggs in contaminated food, water or contact with infected animals . We presHydatid disease is an endemic parasitosis in regions such as China, Russia, South America, the Mediterranean Region, Eastern Europe, and Central Asia . PrevaleThe World Health Organization (WHO) Informal Working Group Classification of cystic echinococcosis (CE) is shown in Treatment modalities include puncture aspiration injection reaspiration, which is reserved for uncomplicated cysts that do not have daughter cysts (e.g. WHO stage CE1 and CE3a) (Our patient presented with cysts belonging to stages C2 and C3b for which the preferred treatment is albendazole followed by surgery, which was implemented for this patient (


A large tumour mass was detected in a 65-year-old patient during a routine gynaecological examination. This patient had been treated for over 10 years with levothyroxine for Hashimoto’s thyroiditis and was also given transdermal oestrogen replacement therapy. Before the operation, detailed imaging by CT scan and MRI was performed. A tumour weighing 1.056 grams and measuring 23x12x7 cm was successfully removed through laparotomy. Histopathology revealed the diagnosis of an extra-uterine, so-called parasitic leiomyoma. Post-surgery recovery was uneventful, but Tibolone treatment was indicated due to disturbing menopausal complaints. Uterine leiomyomas, also called fibroids, are common benign adenomas, which sometimes cause metrorrhagia. The growth of fibroids is hormone- dependant, but it is possible that they can shrink after the menopause. However, they may also gain volume during oestrogen replacement therapy. Whilst the overwhelming majority of fibroids are situated in the uterus, extra-uterine, parasitic leiomyomas do occasionally occur . SeveralTo the best of our knowledge, no cases have been published combining a large extra-uterine leiomyoma with Hashimoto’s thyroiditis in a post-menopausal patient receiving adequate hormone replacement therapy with thyroid hormone (Levothyroxine) and oestrogen (Oestrogel).The patient was a 65-year-old obese woman with a BMI of 34.8 kg/m2. She was known to suffer from Hashimoto’s thyroiditis, with elevated anti- thyroglobulin antibodies (232 IU/mL), which has been adequately treated for over 10 years. Treatment with 100 μg of levothyroxine resulted in normal hormone concentrations in the blood: TSH: 0.5.mud/L, free T4: 20.2pmol/L, free T3: 4.9 pmol/L.There were no other biological markers of autoimmune disease. Due to insulin resistance combined with hyperinsulinemia, the patient was successfully treated with the combination of Momordica charantia extract and alpha-lipoic acid , with a After the birth of her third daughter, she was sterilized by tubal ligature. Due to disturbing menopausal complaints, the patient applied transdermal oestradiol .In July 2019, the patient was examined by her usual gynaecologist who detected a large intra- abdominal mass, detached from the uterus.A computerised tomography (CT) scan of the abdomen was performed followed by a magnetic resonance imaging (MRI) scan of the pelvis Fig. & 2 showThe abdomen was opened by a typical laparotomic incision. The intraperitoneal tumour was attached to the anterior side of the abdominal wall, mostly on the left side. There was no connection with the annexes or the uterus. The uterus itself was moderately enlarged due to a few sub-serosal uterine myomas (<2cm). The peritoneum was opened and the tumour was isolated systematically. The surgical team were able to dissect the tumour from the abdominal wall. Intra-operative frozen section and a gross evaluation of the tumour were performed which revealed a benign stromal tumour of approximately 23 x 12 x 7cm. The left rectus abdominis muscle was slightly damaged during the process of tumour removal, which was immediately restored with Vicryl 2 in separate U-sutures. The peritoneum fascia, subcutis and cutis were closed, and intra-abdominal tubular drainage was placed with the installation of a subfascial Penrose drain.The post-operative course was uneventful, and no complications occurred. Because of this favourable course, the patient was able to leave the hospital after four days. Four weeks later, she was seen at an outpatient consultation (FC) in good health with slight local thickening of the abdominal wall at the left side. No residual tumour residue was detected. The patient complained of disturbing vasomotor hot flushes, cognitive and emotional instability, and sleep disturbance. It was decided to temporarily restart transdermal oestrogen supplementation, which was immediately replaced by long-term Tibolone intake .Macroscopically, the mass was described as a large nodular tumour of 1056 grams and 23 x 12 x 7 cm. On cut section, it appeared as a solid homogenous light-brown, tan mass. Paraffin sections showed a moderately to poorly cellular tumour, composed of spindle cells, predominantly arranged in bundles. The nuclei were spindle-shaped with blunt ends. Mitoses were few, and no abnormal mitotic figures were seen. The cells had an eosinophilic cytoplasm and were separated by a wide and oedematous interstitium. The sections showed dispersed mast cells and plasmocytes. At the periphery, a pseudocapsule of connective tissue was noticed. Arterial and venous vessels in the capsule were found at the site of the adhesion. Immunohistochemical investigation was performed and resulted in: (1) Smooth muscle actine: positive; (2) Desmin: positive; (3) S100 protein: negative; (4) Ki-67 proliferation marker: <5% positive cell nuclei.In conclusion, the mass was described as a giant leiomyoma (max. diameter 23 cm) with benign degenerative changes and no signs of malignancy.Whereas uterine leiomyomas are common gynaecological tumours, extra-uterine so-called parasitic leiomyomas are a rare variant of a pedunculated (uterine) sub-serosal leiomyoma occurring outside the uterus and completely separated from it . Extra-uThis mass usually grows intra-peritoneal, more specifically in the pelvis. However, some parasitic myomata have been described in the upper abdomen, on the omentum, in the vagina, sub- or supra-fascial near the port site, sigmoid colon, cervical, retroperitoneal ,in the tParasitic leiomyomas can be either asymptomatic  or causeThe most accurate diagnostic method is a pelvic MRI allowing a detailed description of the tumour and the anatomical relationship with surrounding structures. In addition to these features, MRI may also detect alternative and/or coexistent pelvic or abdominal pathology.The classic appearance of a typical fibroid on MRI is a well-circumscribed mass with homogeneous T2 hypo-intensity and T1 iso-intensity relative to the myometrium . HoweverAn interesting and rather unique feature of the present case is that the leiomyoma occurred in a patient with Hashimoto’s thyroiditis, but without hypothy roidism. There were no other characteristics of autoimmune disease, but the patient presented with insulin resistance and hyperinsu linemia related to obesity. Insulin is a growth factor and hyperinsulinemiais associated with tumour development . Thus, hThe accepted hypothesis of a parasitic leiomyoma is that they are subserous or stemmed myomata. They may detach from the uterus as a result of traction and torsion and be implanted elsewhere . This isIn this case, the diagnosis leiomyomatosis peritonealis disseminata could be ruled out because its origin is smooth muscle metaplasia in the subcoelomatous mesenchyma, which was not the case in this patient. Furthermore, there were no diffuse vascular peritoneal nodules visualized on imaging .Intravenous leiomyomatosis was discounted as it originates, just like leiomyomatosis peritonealis disseminata, from smooth muscle tissue and should present itself intraluminal in intrauterine or systemic veins (often in the vena cava) .A benign metastatic leiomyoma is normally characterized by solitary extra uterine leiomyoma- like nodules, especially pulmonary, and are often found in women with a pelvic surgical history . This diRetroperitoneal leiomyosarcoma is characterized by extensive central necrosis, invasive growth and a more heterogeneous appearance. These characteristics were not visualized in this case, so was also not a sensible diagnosis. In addition to this differential diagnosis, peritoneal carcinomatosis were also less probable because of the absence of ascites and weight loss .In general, the treatment is a resection: open, laparoscopic or robotic, which depends on the location, the size of the mass and its connection to other organs . AnotherIn the case reported, there was no need for hysterectomy nor ovariectomy since the tumour was completely isolated from the female genital organs.


A 60-year-old woman was diagnosed with isolated mucosa-associated lymphoid tissue (MALT) lymphoma of the ocular adnexa and treated with two years of weekly rituximab for eight doses followed by rituximab maintenance. After nearly two years of maintenance therapy, she developed a tender, indurated mass on the left neck. Biopsy results were consistent with primary cutaneous classical Hodgkin lymphoma (PCCHL). Classical Hodgkin lymphoma (HL) with cutaneous involvement is well described in the literature and usually occurs late in the course of the disease. The incidence has decreased in the recent decades, likely owing to improved chemotherapy and radiation therapy of patients with Hodgkin lymphoma, as well as the advent of stem cell transplantation . PCCHL iNon-Hodgkin lymphoma (NHL) and HL can present in the same host, either synchronously or metachronously and perhaps even in composite specimens. We describe the unique case of a patient initially treated for orbital MALT lymphoma who later presented with PCCHL. The combination of MALT lymphoma in the ocular adnexa followed by the appearance of PCCHL without any nodal or bone marrow involvement is exceedingly rare. To our knowledge, this is the first reported case of these two distinct entities occurring in the same host.A 60-year-old woman presented with a 3-week history of left upper eyelid bruising and swelling without constitutional symptoms. Magnetic Resonance Imaging (MRI) revealed a 2.0 cm × 1.3 cm soft tissue mass in the medial extraconal compartment involving the oblique muscle. Biopsy was consistent with extranodal MALT-type marginal zone B-cell lymphoma. Staging workup with computed tomography (CT) and positron-emission tomography (PET) suggested possible cervical and inguinal lymph node involvement. Bone marrow biopsy did not show definitive evidence of involvement. The patient underwent treatment with weekly rituximab monotherapy for 8 weeks. Restaging PET of the orbital soft tissue mass demonstrated decrease in SUV from 11.2 to 4.7 in the orbital soft tissue mass and resolution of the hypermetabolic lymph nodes, indicating clinical and metabolic response to therapy. She was continued on maintenance rituximab every 2 months for 2 years.The patient presented to clinic for her last dose of maintenance rituximab with a 3-4 cm tender, indurated, and slightly erythematous mass on the left side of her neck. She denied constitutional symptoms. CT of the neck demonstrated a soft tissue mass on the left lateral aspect of her mid-neck extending into the overlying subcutaneous tissue and skin that was not seen on prior imaging. Excisional biopsy revealed tumor composed of large pleomorphic malignant cells and admixed inflammatory cells. The tumor was centered in the deep dermis, with satellite nodules (mainly microabscesses) in the superficial dermis. No evident lymph nodes were seen in the specimen. Immunohistochemical (IHC) staining showed that the malignant cells were strongly positive for CD30, CD15, PAX5, and CD79a and negative for CD45, diagnostic of classical Hodgkin lymphoma . Oct2 stPatients who have NHL have an increased incidence of developing HL years later. NHL has most commonly been associated with DLBCL, followed by follicular lymphoma. Proposed mechanisms include long-term effects of lymphoma treatment, immune defects, genetic susceptibilities, lifestyle or environmental factors, and likely interactions between these factors –6. There13 cases of PCCHL have been reported to date , including hematologic malignancies. However, there is no evidence that rituximab specifically contributes to later development of HL , 16, espThe only reported case of MALT with an extranodal HL was described in 2010. Oka et al. reported a patient with synchronous gastric MALT diagnosed at the same time as primary pulmonary Hodgkin lymphoma. Of note, this patient was infected with EBV, a known predisposing risk factor for Hodgkin lymphoma . Both HRIn accordance with the Lugano Classification System, our patient is considered to have stage IE HL. Treatment rationale was based on National Comprehensive Cancer Network (NCCN) guidelines, which suggests treating early stage HL with extranodal involvement with two cycles of ABVD followed by either two additional cycles or radiation therapy. She tolerated and achieved complete remission with this treatment modality. We will continue to follow her progress.In conclusion, PCCHL is an extremely unusual form of extranodal HL. As more reports begin to surface over the centuries, it becomes important to recognize potential risk factors that may be contributing to its pathogenesis, such as immunomodulating therapy, history of NHL, and viral infection/reactivation. Close longitudinal follow-up is recommended in order to identify systemic recurrence of disease, even after achieving remission. This will also lead to better understanding of treatment outcomes, prognostic factors in PCCHL, and primary extranodal HL as a whole and hopefully allow a more uniform consensus regarding therapy.


A solitary fibrous tumor (SFT) is a rare mediastinal neoplasm associated with a high recurrence rate. Total excision on initial surgery is an established indicator of a positive outcome. Here, we report the case of a 52‐year‐old man who was admitted to our hospital with symptoms of cough, chest pain, and dyspnea for two months. Chest computed tomography (CT) scan revealed a middle mediastinal mass which infiltrated adjacent vital structures, and surgery was performed with the assistance of cardiopulmonary bypass (CPB) and median sternotomy. The mass was completely removed and histopathology confirmed the presence of a mesenchymal tumor. The patient had an uneventful recovery without any perioperative symptoms, hoarseness, or dysfunction of the diaphragm. Sixty‐nine months after surgery, a CT scan confirmed that the patient remained disease‐free without necessitating the introduction of chemotherapy or radiotherapy. Here, to the best of our knowledge, we report the first case of a giant invasive mediastinal SFT that was completely resected during initial surgery under CPB with a remarkable outcome. A solitary fibrous tumor (SFT) is a rare mediastinal neoplasm associated with a high recurrence rate.In 2014, a 52‐year‐old man with symptoms of cough, chest pain, and dyspnea for two months was admitted to our hospital. The patient reported no exposure to asbestos, radon gas, or other occupational toxicants. Chest computed tomography (CT) scan revealed an 8.5 × 9.5 cm middle mediastinal mass which was infiltrating adjacent vital structures  and median sternotomy. A firm and hypervascular mass was found encircling the right pulmonary artery (RPA) with compression and anterolateral displacement of the superior vena cava (SVC), as well as compression of the ascending aorta (AAO) and left main bronchus (LMB), but no invasion of the phrenic nerve or recurrent laryngeal nerve. The mass was completely and meticulously dissected Fig . BecauseAfter reviewing the scarce SFT literature, there are four types of combined therapeutic strategies which are summarized as followsSeveral complications may occur attributed to CPB, with bleeding related to systemic heparinization as the major concern.We chose initial en bloc resection by CPB because incomplete resection is associated with poor prognosis for such a giant invasive mediastinal SFT. The following considerations confirmed our decision: (i) compression of the AAO, SVC and RPA existed, which may result in hemodynamic disorder, especially acute right ventricular dysfunction secondary to the sudden relief of the prolonged obstruction of the SVC; and (ii) given that the LMB was compressed, there would be a risk of hypoxia during surgery, as previously reported.The recurrence rate of benign classification is only 2% after surgical excision. Approximately half of malignant classification often develop recurrences and metastases.The vascular supply to the mass in the present case originated from the thoracic aorta. To date, there has been only one case report on the vascular supply of a mediastinal SFT, which was from the coronary arteries, and no other reports have described the vascular supply.The authors declare no conflicts of interest.


Ruptured abdominal aortic aneurysm (AAA) is a highly lethal condition which requires rapid identification and treatment to improve the chance of survival. Computed tomography is the diagnostic modality of choice for ruptured AAA though it is time-consuming and often requires movement of the patient out of the emergency department (ED). Point-of-care ultrasound in the ED has excellent sensitivity and specificity for the detection of AAA, though less is known about its use to diagnose AAA rupture. We report a case of ruptured AAA identified on ultrasound performed at the bedside in the ED.A 77-year-old woman on warfarin with a known AAA presented to our ED with 2 days of epigastric abdominal pain. Point-of-care ultrasound revealed several findings suggestive of rupture of the AAA, which was confirmed on computed tomography. The patient was subsequently taken for emergent operative repair of the AAA and was later discharged from the hospital.Characteristics suggestive of AAA rupture may be seen on ultrasound. As ED physicians become more familiar with the use of point-of-care ultrasound in the evaluation of abdominal pain, identification of these characteristics may aid in the rapid diagnosis of AAA rupture. Rupture of an abdominal aortic aneurysm (AAA) is often lethal, with a mortality of 85-90% . Delays A 77-year-old woman presented to our ED complaining of non-radiating epigastric abdominal pain starting the previous day and becoming severe approximately 2 h before coming to the hospital. She had some associated non-bloody emesis. Her past medical history was significant for hypertension, peripheral vascular disease, coronary artery disease, chronic kidney disease, and chronic obstructive pulmonary disease with an extensive history of smoking. She was taking warfarin for protein S deficiency complicated by previous deep venous thromboses. She had a known infrarenal AAA measuring 4.4 cm in diameter seen on computed tomography (CT) 3 months earlier. The AAA had not changed in size over the previous 4 years. On arrival she appeared uncomfortable, though in no apparent distress. Her blood pressure was 142/113 mmHg, heart rate 60 bpm, respiratory rate 15 bpm, temperature 35.8 degrees Celsius, and pulse oximetry 97% on room air. She was noted to be tender to palpation in the epigastrium without guarding, and no masses were appreciated in her abdomen.A point-of-care ultrasound was performed revealing an AAA measuring approximately 6 cm in diameter Fig. . There wA transfusion of 2 units of packed red blood cells was started in the ED, and the vascular surgery service was consulted. The patient was taken emergently to the operating room where the ruptured AAA was repaired using a polyester graft. After a difficult postoperative course, the patient was eventually discharged to a rehabilitation facility 5 weeks later.The most commonly used definition of an AAA is a maximum infrarenal abdominal aortic diameter greater than 3 cm on ultrasound or CT imaging . While rRapid identification and treatment of ruptured AAA is paramount to improving survival, though its diagnosis often remains elusive. It is estimated that only 30-50% of cases present with the classic triad of abdominal pain, hypotension, and pulsatile mass . In 1994CT is the diagnostic modality of choice for ruptured AAA, though point-of-care ultrasound is more readily available in most EDs. ED ultrasound screening for ruptured AAA has been found to reduce the time to diagnosis and improve patient outcomes . ED poinEmergent operative repair has been the traditional treatment of ruptured AAAs, though endovascular repair has become another treatment option, even in unstable patients . Regardl


Mesothelial cysts are uncommon benign cysts of the mediastinum. Rarely, they are discovered after complications or unusual presentations. This report describes a rare case of pleural effusion revealing a ruptured mediastinal mesothelial cyst in a 28‐year‐old man. The diagnosis of this complicated mesothelial cyst relied on intraoperative and anatomopathological findings. He underwent videothoracoscopy with resection of the cyst. Outcomes were favourable. We report a rare case of a mediastinal mesothelial cyst discovered after rupture in the pleural cavity in a 28‐year‐old man. The diagnosis of this complicated mesothelial cyst relied on intraoperative and anatomopathological findings. To the best of our knowledge, we report the first case of a pleural effusion revealing a ruptured mediastinal coelomic cyst into the pleural cavity. Mesothelial or coelomic cysts are uncommon unilocular benign cysts of the mediastinum , 2. RareA 28‐year‐old man with an uneventful past medical history admitted to our department for chest pain and dyspnoea that started two days ago. He had six pack‐year smoking history. He was not complaining of any other sign. The physical examination was in favour of the diagnosis of a right‐sided pleural effusion. Chest X‐ray showed an opacity of the lower part of the right hemithorax Fig. ‐a. The pCoelomic cysts are derived mainly from the pericardium. They are due to embryonic tissue development abnormalities . The cysAtypical and rare presentations are described in the English literature including rupture, migration, or uncommon sites. In fact, coelomic cysts arising from the extramediastinum are extremely rare. Only two cases of mesothelial cyst deriving from the chest wall pleura were reported . Walker Chest CT scan allows diagnosis by showing a fluid‐filled, thin‐walled cyst without enhancement after contrast injection . In caseIn conclusion, despite being usually asymptomatic, mediastinal mesothelial cyst can be discovered after complications. The rupture in the pleural cavity is an extreme rare condition and imaging can fail to make the diagnosis. VATS is the best therapeutic approach even for complicated cysts.Appropriate written informed consent was obtained for publication of this case report and accompanying images.


Extra-adrenal localization of pheochromocytoma is rare. Its clinical revelation is paroxysmal but maybe in the form of permanent hypertension resistant to treatment. The main problem with these tumors is to affirm their benignity or malignancy, ectopic pheochromocytomas have a malignant development once in two.We report the observation of a 39-year-old woman hypertensive on Amlodipine. She presents for sweating, headache, and palpation. The dosage of urinary metabolites revealed a marked increase in metanephrine and normetanephrine. Imagery had shown, at the expense of the left lateral wall of the bladder, a budding tissue formation with irregular contours which was significantly enhanced after injection of PDC. The diagnosis of an ectopic pheochromocytoma was accepted. The patient underwent a partial cystectomy, the postoperative consequences of which were unremarkable.Threatening secretory syndromes are rare medical situations that require urgent management. Pheochromocytoma can be seen at any age from childhood to old age. The treatment of pheochromocytoma is surgical, once the stages of positive and topographic diagnosis have been carried out, the patient must be immediately entrusted to a team of trained surgeons and anesthetists. The prognosis remains unpredictable, hence the interest in prolonged monitoring.Pheochromocytoma is a tumor developed at the expense of chromaffin tissues, extra-adrenal localizations exist, but remain rare. It is a pathology that presents a diagnostic and therapeutic dilemma in which patients require regular and prolonged postoperative monitoring to detect a possible metastasis or recurrence. The adrenal site as usual (90%), the extra-adrenal location is rare representing 10% of pheochromocytomas. This can be located in the sympathetic and parasympathetic vertebral nodes. ZuckerKandel's organ involvement is by far the most common ectopic location [Its classic clinical expression is paroxysmal  associated with hypertension (hypertension). These elements may be missing and the condition may take on the mask of permanent treatment-resistant hypertension.Its treatment is surgical. The course is marked by the risk of late recurrence even when the tumor appeared to be benign.We report an observation of bladder pheochromocytoma by studying the clinical aspects, the contribution of imaging in the diagnosis of extra-adrenal pheochromocytoma, and establishing a strategy for the therapeutic management of these tumors. This work was reported in accordance with the 2018 SCARE criteria [2Ms. H.N aged 39, multiparous, without profession followed for hypertension for more than a year on calcium channel blocker: amlodipine 10 mg/d. The onset of symptoms was more than a year before admission by the onset of hypertensive paroxysmal attacks, triggered by emotion, a headache of moderate-intensity, unspecified topography, palpitations, and generalized sweating with redness of the face, without hematuria or other urinary signs or notion of permictional symptoms. The whole has evolved in the context of apyrexia and unencrypted weight gain.2, conjunctiva, color, eupneic, apyretic at 37° 2. The cardiovascular examination noted a paroxysmal hypertension at 220 mmHg for the systolic and 140 mmHg for the diastolic, the pulse was at 100 beats/min. There was no palpable mass, nor lumbar contact, the hepatic arrow was at 11 cm, the spleen was not palpable.The clinical examination on admission found a patient in good general condition, weight = 60 kg, height = 1.52 m, body mass index (BMI) = 25 kg/m3, white blood cells at 5,200/mm3. SV was accelerated to 30 mm in the first hour. The dosage of urinary metabolites, metanephrines 0.40 mg/24 h (0.04 to 0.20), normetanephrines 8.41 mg/24 h (0.07 to 0.38).The ionogram revealed a serum at 140 meq/l, the serum potassium at 5 meq/l, the chlorine at 110 meq/l. the blood urea level was 0.29 g/l, the serum creatinine was 6.3 mg/l. The blood sugar was raised to 2.4 g/l. hemoglobin at 12.9 g/100 mL, Platelets at 507,000/mmUltrasound and abdominal CT, They had highlighted, at the expense of the left side wall of the bladder, a budding tissue formation with irregular contours which was significantly enhanced after injection of PDC, measuring 53 × 50 × 60 mm. This mass is the seat of small calcifications. The adrenals were intact. No intra or retro-peritoneal lymphadenopathy, normal-looking liver , Fig. 2.Meta-iodo-benzyl-guanidine scintigraphy labeled with iodine-131 was not done. C- the chest x-ray did not reveal any anomalies. A check-up for multiple endocrine neoplasia was done and was negative . The diagnosis of vesical pheochromocytoma has been strongly suspected, hence the need for surgical exploration.The patient had received antihypertensive treatment based on Amlodipine 10 mg 1cp/d, Doxazosine 2 mg 1cp/d, Atenolol 100 mg 1cp/d.The operation took place in 2 stages:First step: cystoscopy, the exploration of which has found a thickening of the left lateral wall near the bladder neck, with no visible budding mass in the endovesical.Second step: Tumorectomy, Partial cystectomy passing 2 cm from the tumor. During the manipulation of the bladder, the patient presented well-controlled hypertensive peaks at 220/160 mmHg.Macroscopic anatomopathological examination was a well-encapsulated, firm tumor weighing 40 g and measuring 6.5 cm from the major axis . When cuThe postoperative follow-ups were simple with normalization of the blood pressure on day 1 of the postoperative (100/70 mmHg), the pulse was at 77 beats/min. The dosage of metanephrine and urinary normetanephrine at 1 week postoperative was found to be normal.The clinical, ultrasound, and biological follow-up at 3 months and 6 months was without abnormality.3Threatening secretory syndromes are rare medical situations that require urgent management . PheochrPheochromocytoma can be seen at any age from childhood to old age. The average age of predilection is 40 years with extremes ranging from 10 to 74 years . ExceptiThe majority of pheochromocytomas are symptomatic. The symptoms being linked to the secretion of catecholamines  or tumor extension in the case of malignant pheochromocytomas. The others are accidentally discovered during a radiological examination or screening for familial forms . Others The classic clinical expression of pheochromocytoma is the triad: headache, sweating, and palpitations in a hypertensive patient. However, it can take various clinical forms depending on the site, nature, and tumor volume. Sometimes it is responsible for more unusual cardiovascular manifestations such as acute edema of the lung, myocardial involvement, or acute symptoms.Urinary manifestations, namely The per or post-voiding occurrence of general malaise  which accompanies a hypertensive flare is very suggestive of pheochromocytoma with bladder localization . These sHematuria is also a warning sign and may be accompanied by kidney failure. Pollakiuria and dysuria are sometimes reported .Recent progress, made in the knowledge of the genetic mechanisms responsible for pheochromocytomas, reveals the role of germline mutations of different genes involved and allows to improve the diagnosis and the family care. The rule that 10% of pheochromocytomas are bilateral, 10% extra-adrenal, 10% familial, and 10% malignant is questioned today. Although most pheochromocytomas are sporadic, genetic studies in recent years have shown that 25–30% of them are hereditary, due to a germline mutation in the RET, VHL, NF1 genes or genes encoding the subunits B, D and C of succinate dehydrogenase from the Krebs cycle [The basis for the diagnosis of pheochromocytoma is the demonstration of excessively high secretion and consequently elimination of catecholamines or their metabolites. These compounds however have different diagnostic performances.The dosage of adrenaline and urinary free norepinephrine is less reliable than that of their methoxylated metabolites since their increase is considerable during pheochromocytoma and in many situations: asphyxia, hypoglycemia, cold, stress, and physical exercise. Urinary fractional metanephrines are measured after an acid hydrolysis step that allows deconjugation .The abdominal ultrasound is a harmless examination that visualizes the mass, affirms its tissue nature, and studies its impact on the neighboring organs. It also makes it possible to check whether the tumor is localized or multifocal . This exAbdominopelvic CT has a major interest in the location of the tumor. It allows a non-aggressive and precise analysis. Its sensitivity is 88% for tumors over 1 cm in diameter . PrioritMRI offers more effective results than CT and can be performed during pregnancy . It showMIBG scintigraphy is a method described in 1981 by Sisson et al. It has been proposed in the topographic diagnosis of pheochromocytoma, in particular those ectopic or of size less than 1 cm. Its sensitivity is 95% and its specificity is around 99%. MIBG has a biochemical structure close to norepinephrine, it is captured and stored by tissues rich in catecholamines. It is ideally marked with iodine 123 of iodine 131 [Positron Emission Tomography (PET) This imaging technique using positron-emitting tracers, the accumulation of which is visualized by a PET camera, has the advantage of being able to detect lesions of the order of 5–10 mm. It has been used with many agents or markers  .The genetic diagnosis must be generalized since the rate of germline mutation can be present between 12 and 24% of cases in patients with a priori a sporadic pheochromocytoma. A systematic genetic study is therefore recommended before any pheochromocytoma, even sporadic in appearance and especially in case of early or multiple formsforms .The treatment of pheochromocytoma is surgical, once the stages of positive and topographic diagnosis have been carried out, the patient must be immediately entrusted to a team of trained surgeons and anesthetists. Surgical excision is conditioned by the precision of the location of the tumor and requires a suitable approach.The gesture performed is based on the location of the pheochromocytoma, for extra-adrenal tumors and after checking the other chromaffin territories, a simple lumpectomy is performed, the tumor is generally well limited and encapsulated but this excision can extend to the organs neighborhood if they are reached .In the case of vesical pheochromocytoma, cystectomy taking away the whole tumor with an exploration of the ganglionic chains constitutes the therapeutic indication of choice. Transurethral resection does not appear to be reliable. It is likely to be incomplete and seems to generate stronger hypertensive pressures . Some auThe series is short and there is no controlled trial comparing open and endoscopic surgery, or between the two routes of laparoscopic surgery. Subject to sufficient training, endoscopic surgery can be offered to patients with benign pheochromocytoma less than 6 cm in diameter. It allows a lumpectomy and reduces the length of hospital stay while maintaining good safety .It is unpredictable, from a few months to several years and marked by the risk of metastases or recurrences. Late recurrences can occur even when the tumor appears to be benign. In a series of 98 pheochromocytoma operated patients, Van Heerdan had observed six recurrences, three of which were distant, two local and one both local and distant. Local recurrence occurs in the event of incomplete removal of the tumor . The couAccording to the data in the literature, Chapuis  reported4Pheochromocytoma is a tumor developed at the expense of chromaffin tissues, adrenal localization represents 90%, extra-adrenal localizations exist, but remain rare (10%). The vesical pheochromocytoma constitutes 13% of ectopic localizations.The topographic diagnosis is based on CT or MRI. If these are negative, MIBG scintigraphy is a recent method that can be proposed to locate the site of the tumor. These tumors must be diagnosed and treated early to avoid the consequences of adrenergic hypersecretion. The treatment is based on excision surgery (cystectomy for pheochromocytoma of the bladder). Monitoring of patients should be regular and prolonged to detect a possible progression of the disease in the form of metastasis or recurrence.The authors report no declarations of interest.We have any funding for your research.written proof of consent to publish study participants are available when requested and at any time.The consent to publish this information was obtained from study participants. We confirm that written proof of consent to publish study participants are available when requested and at any time.The consent to publish this information was obtained from study participants. We confirm that Dr. IJ, Dr. AT, Dr. YL Dr. WB analysed and performed the literature research, Pr. MD, Pr. AD, Pr. RA performed the examination and performed the scientific validation of the manuscript. Issam Jandou was the major contributors to the writing of the manuscript. All authors read and approved the manuscript.Not applicable.Issam Jandou.Not commissioned, externally peer reviewed.The datasets in this article are available in the repository of the urology database, Chu Ibn-Rochd Casablanca, upon request, from the corresponding author.


Tissue harvesting for patients with a lung nodule is sometimes unsuitable due to the size and location of the nodule. In such cases, it is unclear whether it is acceptable to proceed to definitive lobectomy without intraoperative frozen section analysis.We retrospectively reviewed patients who underwent definitive lobectomy or wedge resection for frozen section analysis at our institution between 2014 and 2018. The sensitivity, specificity, and accuracies of the clinical and frozen section diagnoses were evaluated against the final pathological diagnosis.There were 141 patients in the definitive lobectomy group and 58 patients in the frozen section analysis group, with the latter having smaller and less deep nodules and a lower rate of malignancy on clinical and final pathological diagnoses. The sensitivity, specificity, and accuracy of the clinical diagnosis were 100%, 82%, and 95%, respectively, in the frozen section analysis group and 99%, 67%, and 97%, respectively, in the definitive lobectomy group; values of frozen section diagnosis were 98%, 82%, and 93%, respectively. On subgroup analysis, all ground‐glass nodules clinically diagnosed as malignant had a final pathological diagnosis of malignancy.The accuracy of the clinical diagnosis was high and was not inferior to the frozen section diagnosis. These data suggest that definitive lobectomy is an acceptable treatment option for carefully selected patients with large or deep nodules and ground‐glass nodules clinically diagnosed as malignant. To avoid unnecessary lobectomy, frozen section diagnosis should be considered for nodules likely to be benign.The accuracy of the clinical diagnosis was high and was not inferior to the frozen section diagnosis.Definitive lobectomy is an acceptable treatment option for carefully selected patients with large or deep nodules and ground‐glass nodules with a clinical diagnosis of malignancy. Computed tomography (CT) is effective for screening patients at a high risk of lung cancer, increasing the likelihood of identifying a solitary pulmonary nodule.For patients with a lung nodule without a preoperative tissue diagnosis, radical surgery, generally lobectomy, is often performed after a diagnosis of malignancy from intraoperative frozen section analysis (FSA), with the tissue specimen obtained by wedge resection or needle biopsy.Therefore, the aim of our study was to clarify whether DL could be an acceptable treatment option in selected cases by evaluation of diagnostic accuracy of clinical and frozen section diagnosis among patients who underwent DL or wedge resection for FSA.We retrospectively reviewed the medical records of consecutive patients who underwent lung resection without a preoperative tissue diagnosis between January 2014 and December 2018 at the Jikei University Hospital in Tokyo, Japan. Of these, patients who underwent DL or wedge resection for FSA with intended follow‐up lobectomy were potential cases for our study, and we excluded patients with coexisting or a past‐history of other malignancy, including lung malignancy (with the intention of eliminating the possibility of metastatic lung tumors), and those who underwent pneumonectomy, bilobectomy, segmentectomy, wedge resection without intraoperative FSA, and surgery without curative intent .The clinical diagnosis was based on a comprehensive review of the clinical information, clinical course, radiological findings , chronological evaluation of radiological findings at multiple times including tumor doubling time, and fluorodeoxyglucose‐positron emission tomography (FDG‐PET). Patients were selected based on the degree of certainty of ensuring a safe margin for wedge resection and on the probability of malignancy. They were subsequently assigned to the group of candidates either for DL or for wedge resection for FSA according to our surgical treatment algorithm for lung nodules without preoperative tissue diagnosis Fig .et al. was calculated by variables of age, smoking, history of extrathoracic cancer, tumor diameter, existence of spiculation, tumor located in the upper lobe, and the results of FDG‐PET.Clinical demographic and perioperative information and pathological diagnosis were included in the analysis using the following variables collected from the medical records and our prospective database: age, sex, smoking history, radiological findings of lung CT and FDG‐PET, any preoperative biopsy, preoperative clinical diagnosis, clinical stage, the result of frozen section diagnosis, number of stapler cartridges used during the wedge resection, margins of the wedge resection, operative time for wedge resection and FSA, surgical approach and procedure, final pathological diagnosis and stage, total operative time, and postoperative complications. From CT findings, types of nodules were classified as pure GGN, part‐solid GGN, or solid nodule. The malignant probability score, which is applicable to cases with solid nodules, reported by Herder Informed consent was obtained from all patients for treatment, after explaining the surgical indication for DL and wedge resection for FSA with intended follow‐up lobectomy, the associated risks and benefits. All procedures were performed in accordance with the ethical standards of the Ethics Committee of Jikei University School of Medicine , and the Declaration of Helsinki and its later amendments. The need for obtaining individual patient consent was waived due to the retrospective design of the study.We compared demographics, perioperative outcomes , and pathological diagnoses between the DL and FSA groups using Fisher's exact test or Mann‐Whitney U test as appropriate for the data type and distribution. Perioperative outcomes of wedge resection without lobectomy in cases of benign frozen section diagnosis were excluded from the analysis to eliminate the effects of the extent of surgical resection. Sensitivity, specificity, accuracy, positive predictive, and negative predictive values were calculated for clinical diagnosis or frozen section diagnosis against the final pathological diagnosis. On a subgroup analysis, these values were also compared between the GGN, including pure and part‐solid and solid nodule groups.P‐value < 0.05 was considered statistically significant.All analyses were performed using JMP , and a In total, 818 patients underwent lung resection between January 2014 and December 2018 at our institute. Of these, 199 patients without preoperative tissue diagnosis met our selection criteria and were included in our analysis: 141 in the DL group and 58 in the FSA group. Table Patient characteristics, perioperative information, and pathological diagnoses for each group are shown in Table The flowchart of frozen section diagnosis, clinical diagnosis, and the final pathological diagnoses in each group is shown in Fig In the DL group, seven cases with a benign clinical diagnosis, for whom wedge resection was unsuitable due to the size or depth of the nodule, were treated by lobectomy to confirm pathologically a definitive benign diagnosis. Their final pathological diagnoses were adenocarcinoma in one case and benign in the remaining six cases. The 134 cases in the DL group with a malignant clinical diagnosis were treated with lobectomy, and three of these cases were benign tumor and the remaining 131 cases were malignant on final pathological assessment. There were 43 cases that were feasible for wedge resection but were omitted and assigned to the DL group because of almost certain malignancy according to our algorithm described above, and they all received a final pathological diagnoses of malignancy.Overall, an error in clinical diagnosis was identified in three cases in the FSA group and four in the DL group, while there were four cases with an error in the FSA by wedge resection. The details of these diagnostic errors are presented in Table Diagnostic accuracies of clinical and frozen section diagnosis are shown in Table There were two major findings in this study. The diagnostic accuracy of clinical diagnosis was high and was not inferior to that of frozen section diagnosis, and the accuracies of frozen section and clinical diagnosis for GGN were both 100%.First, the diagnostic accuracy of clinical diagnosis was high and not inferior to that of frozen section diagnosis in both groups. The FSA group included smaller and less deep nodules and a higher rate of benign clinical diagnosis than the DL group. Based on these factors, FSA by wedge resection was deemed to be feasible and necessary for these patients. Diagnostic accuracy, sensitivity, and specificity between the clinical and frozen section diagnoses were not different in the FSA group. The benign nature of the nodule in the 14 cases with a benign clinical diagnosis was diagnosed by FSA and confirmed by final pathological diagnosis, avoiding unnecessary lobectomy. One patient with a benign frozen section diagnosis had a malignant final pathological diagnosis, and the error was caused by sampling, whereas three cases with a malignant or equivocal frozen section diagnosis had a benign pathological diagnosis, and the error was caused by interpretation. Previous studies reported false‐negative and false‐positive rates of FSA against the paraffin section  diagnosis ranging between 1.9%–13.1% and 0%–0.2%, respectively, with a specifically higher false‐negative rate for small nodules (≤1.1 cm in diameter).Second, the diagnostic accuracy of frozen section and clinical diagnosis for GGNs (pure and part‐solid) were 100%. Therefore, GGNs selected carefully with a clinical malignant diagnosis would be enough reason to proceed to definitive resection such as lobectomy or segmentectomy without the need for confirmation of malignancy by FSA. Diagnostic accuracy of FSA by wedge resection was also 100%. However, because of concerns of the possibility of a positive margin in cases of wedge resection for large or deep GGNs with extensive and unclear boundaries, which carry the risk of dissemination,et al.Intraoperative core needle biopsy for FSA provides an alternative method to wedge resection for large or deep nodules, which are unsuitable for wedge resection. Isaka The limitations of our study should be acknowledged. First, we used a retrospective design, comparing groups with very different backgrounds. The DL group included large and high malignant probability nodules or many GGNs, which would affect the high diagnostic accuracy. Furthermore, because FSA was not performed in all patients, the diagnostic accuracy of frozen section diagnosis was among the patients assigned to the FSA group which included small and low malignant probability nodules or less GGNs. Second, we did not include cases of segmentectomy as an oncologically acceptable resection. However, because oncologically acceptable segmentectomy is generally indicated for GGNs,In conclusion, the accuracies of clinical diagnosis in both groups were high and not inferior to that of frozen section diagnosis by wedge resection in the FSA group. In any case, it is important for surgeons to recognize the possibility of diagnostic error, even with FSA by wedge resection. FSA should be considered for cases likely to be benign to avoid unnecessary lobectomy. Therefore, DL is an acceptable treatment option for large or deep nodules and GGNs selected carefully with malignant clinical diagnosis when performing a preoperative nonsurgical biopsy can expose the patient to unnecessary risks or inadequately lengthen surgical management.The authors have declared that no conflicts of interest exist.