Bullous hemorrhagic dermatosis (BHD) is a systemic side-effect of low molecular weight heparin, characterized by multiple intra-epidermal hemorrhages distant from the site of injection. There have been several small case series and literature reviews on BHD, but none have captured a complete set of reported patients. We sought to describe a case of BHD with late diagnosis and completely summarize the existing English and Spanish literature with searches of Pubmed, Scopus, Ovid Embase and Ovid Medline. After narrowing to 33 relevant reports, we describe 90 reported cases worldwide from 2004 to 2017, in addition to a new case from our institution as a means of comparison. We found that BHD was common in elderly men  and typically occurred within 7 days of administration of anticoagulation (median 7 days ± 6.4) usually with enoxaparin use (66% of cases). Lesions occurred primarily on the extremities only (67.9% of cases). Coagulation testing was most often normal before administration, and the majority of patients had coagulation testing in therapeutic range during treatment. Most practitioners stopped anticoagulation if continued therapeutic intervention was no longer required (57% of cases), or changed therapy to another anticoagulation if continued treatment was required (14.3% of cases). Therapy was continued outright in 23% of patients. The lesions usually resolved within 2 weeks . There was no difference in time to resolution between patients who continued the culprit anticoagulant or changed to a different anticoagulant, and those who discontinued anticoagulation altogether . Four deaths have been reported in this clinical context, two specified as intracranial hemorrhage. These deaths were unrelated to the occurrence of BHD. Continuation of low-molecular weight heparins appeared to be safe in patients with BHD. Low molecular weight heparins (LMWH) are extensively used as anticoagulation for primary and secondary prophylaxis against thrombosis, particularly in patients with malignancy. There are several known dermatological side-effects of LMWH, including injection site hematoma and skin necrosis, eczematous dermatitis, and the lesser known bullous hemorrhage dermatosis (BHD). The latter was first described in 2004 by Dyson et al. . It typiWe performed a literature search of Pubmed, Scopus, Ovid Embase and Ovid Medline. The keywords for the search were “bullous” “hemorrhagic” and “dermatitis”. Of the initial 213 search results, 51 were found to be duplicates. The remaining 162 were reviewed by title and abstract. Of these, 33 were deemed relevant to BHD, and the entire article was reviewed. The majority of the excluded search results either did not include anticoagulant use or were relevant to dermatologic diseases other than BHD.Our patient is a 62-year-old male with multiple comorbidities including morbid obesity, coronary artery disease, lipodermatosclerosis of the lower extremities, chronic peripheral venous insufficiency, and prostate cancer (Gleason 4+5) on long-term androgen deprivation therapy. He was previously treated with docetaxel for pelvic lymph node metastases. The patient also had a small renal tumor for which he was followed with imaging. He had a distant history of varicose vein ligation. While undergoing surveillance imaging to evaluate for spread of his prostate cancer, an incidental pulmonary embolism was discovered. He was started on enoxaparin 120 mg by subcutaneous injection twice daily. Within several days the patient noticed several small black blisters on his hands that resolved spontaneously. These were not reported by the patient or observed by any practitioner during routine clinic visits. The timeline of development of hemorrhagic skin lesions of this patient is outlined in Fig. We describe 90 cases of BHD discovered in our literature search, in addition to our local case Table  1–33]. –33. 1–33Summary statistics were extracted based on our review of the literature Table . The aveThe review of BHD presented here is the largest collection of cases compiled from the English and Spanish literature to our knowledge. BHD has been documented in medical reports, but appears under-recognized in the clinical practice. We also presented a case from our own institution.BHD is a non-immune dermatological eruption related to anticoagulation given for either primary or secondary prophylaxis. Enoxaparin is the most common culprit anticoagulant, followed by fondaparinux. Lesions are distant from the site of injection. Clinical or pathological signs of inflammation are absent. The extremities are the primary sites of eruptions in many cases. BHD caused by DOACs is not specified to date from several examples of large clinical trials for VTE treatment –36. No pOur case is unusual for the delay of 4 months in the appearance of BHD consisting of very large lesions in the lower extremities. Most lesions will appear within 7 days of the first anticoagulation administration. Their appearance is usually of small, dark bullae, rather than large hemorrhagic bullae. Lesions are not painful. In retrospect, our patient did recall having small blisters on his hands that occurred within days of starting enoxaparin. However, he did not report these small skin lesions as they resolved spontaneously. He sought medical attention after developing large dark bullae on both calves.From our viewpoint, if characteristics of BHD are present as outlined above and in Table After the appearance of BHD, maintaining anticoagulation with a LMWH may increase a patient’s risk of recurrent bullae , 22, 31.BHD is sometimes associated with eczematous reaction at sight of injection that may suggest a type IV hypersensitivity reaction . The proOf the 91 cases presented, there were four cases that had bleeding complications, two with fatal intracranial hemorrhage , 28. AvaHemorrhagic bullous dermatosis secondary to LMWH usage has been reported in the literature but is under-recognized. In this case report the diagnosis was not made until the patient presented with large hemorrhagic bullae after 4 months of LMWH. Biopsy may not be required if the clinical characteristics of BHD are present. The anticoagulation of choice may be continued if lesions are small and are resolving. Risk evaluation for bleeding may proceed along standard guidelines. This case series also highlights a need for clinical reporting of BHD when using DOACs.


A rupture of the membranous part of trachea during thoracoscopic and transhiatal resection of esophagus is a rare complication that occurs in 0.4% of cases. This complication often requires thoracotomy and is associated with prolonged pulmonary ventilation, long-term pleural draining due to persistent air leakage and development of a tracheopleural fistula, prolonged hospitalization, and high risk of septic and secondary cardiorespiratory complications.A 52-year-old woman complained of difficulty eating solid food, impaired swallowing, persistent pain behind the sternum, nausea, sickness and weight loss. EGD revealed tumor almost completely obturating the lumen in the middle third of the esophagus. The histopathological examination showed esophageal SCC. CT scan confirmed 33 × 29 × 55 mm tumor. The patient underwent thoracoscopic esophageal resection during which two defects of the tracheobronchial tree with sizes of 15 mm and 30 mm were detected and then successfully sewn using intracorporeal continuous Stratafix 3.0 suture. The patient was diagnosed with cancer of middle third of esophagus pT3N1M0, stage IIIB.Thorough selection of patients undergoing thoracoscopic esophageal resection together with gentle manipulations with the esophagus in the area adjacent to the trachea can prevent intraoperative tracheobronchial damages. Timely diagnosis of such serious complications makes it possible to successfully manage them using thoracoscopic techniques.Intraoperative tracheal and bronchial ruptures can be successfully managed with thoracoscopic intracorporeal suture. The intIn this case report our objective was to present the experience of thoracoscopic closure of the membranous part of the trachea defect, which occurred during thoracoscopic resection for esophageal cancer.The operation was performed in the surgical department of the non-governmental clinical hospital.The work has been reported in line with the SCARE criteria .2A 52-year-old Caucasian woman was admitted to our surgical department. She complained of difficulty eating solid food, impaired swallowing, persistent nagging pain behind the sternum, periodic heartburn and nausea, sickness. The woman had lost 6 kg in the previous 2 months. She had been using antacid medications without prescription for a month with no effect. The patient denied smoking and alcohol intake. The woman had no previous surgical or another medical history.The diagnostic measures included esophagogastroduodenoscopy (EGD) with biopsy and contrast chest spiral computed tomography (contrast CT) scan. EGD revealed tumor almost completely obturating the lumen in the middle third of the esophagus (25 cm from incisors), impassable for the endoscope. A biopsy was taken. The histopathological examination showed esophageal squamous cell carcinoma (SCC).CT scan confirmed the diagnosis, showing 33 × 29 × 55 mm tumor of the middle third of the esophagus intensively and irregularly absorbing contrast . Above tThe patient was diagnosed with cancer of middle third of esophagus T3N1M0, stage IIIB. According to national clinical standards the patient underwent surgical treatment . The timOperative technique (the operative technique of thoracoscopic stage is described in details in our previous case report [e report ):The patient was initially placed in the supine position, and a double lumen endotracheal tube was placed in preparation for the thoracoscopic mobilization of the esophagus. The abdominal stage was open (laparotomy). The gastric tube was formed by dissecting gastrocolic ligament along the greater curvature from the body of the stomach to the subcardial part preserving the right gastroepiploic artery. Then short gastric vessels, gastrophrenic ligament, abdominal part of the esophagus, hepatogastric ligament were cut. The left gastric artery was cut after lymph node dissection in the area of hepatic and splenic arteries, celiac trunk. The gastric tube was formed from the fundus by resecting small curvature by 2 cassettes of 100 mm linear stapler , leaving 35 mm gastric pouch. The phrenic vein together with a part of diaphragmatic cruses was cut, expanding the esophageal opening of the diaphragm for gastric conduit and the tumor withdrawal. Then the laparotomy incision was closed and the patient was turned to the prone position for the thoracoscopic mobilization of the esophagus and creation of the intrathoracic anastomosis.Further four trocars were placed into the right pleural cavity: two 10 mm, one 12 mm and one 5 mm. After the right lung collapse, mediastinal pleura was incised from diaphragm continuing up to 2–4 cm above the carina using EnSeal tissue sealer. The azygos vein was clipped with Hem-o-lock  and cut.The esophagus was mobilized with the fat tissue and the lymph nodes around it up to the planes of the aorta, the pericardial sac, and contralateral pleura. Then the esophageal tumor with sizes of 6.5 × 4 cm tightly adjoined to the first main bronchus and trachea became visible. The tumor was hard to dissect in the plain of the trachea and the bronchus. The esophagus was mobilized up to the level of the pleural dome, taken on the holder and separated from the tracheobronchial tree.Next, the esophagus was cut off using the Endopath Echelon Flex 45 stapler  with a 45 mm yellow cassette at the distance of 5 cm from the tumor. After the specimen was removed two defects of the tracheobronchial tree were detected. The first rupture with sizes of 15 mm was situated in the area of the left main bronchus right below the tracheal bifurcation . AnotherNext, the resected part of the esophagus was placed into a plastic container and moved into the abdominal cavity through the esophageal opening of the diaphragm. Then the gastric tube was pulled into the mediastinum and trial comparison of the edges of the anastomosis without tension was performed. The gastric tube was fixed to the remaining part of the esophagus by Vicryl 2.0 suture. After that, the stomach and the esophagus were opened using electrocauter. The esophagogastroanastomosis was performed using V-loc 2.0 suture at the level of the pleural dome . Then, tA methylene blue test of the anastomosis was performed; there was no data for dehiscence and leakage. Then the pleural cavity was drained on the right side by two drain tubes; trocars were removed and trocar wounds were sutured. The resected specimen was removed from the abdominal cavity and sent to the laboratory for pathological examination.The surgery was performed by the team, consisted of 3 experienced in thoracoscopy surgeons. The overall operative time was 345 min. The patient was extubated 1 h after the end of the operation with no signs of respiratory disorders. There was no air leakage through the pleural drains and they were removed on 5th postoperative day. The woman tolerated the surgery well and started to take in a liquid food on the 6th postoperative day. On the 10th day after the surgery an esophagogram with liquid contrast (Urografin) was performed .Fig. 6PoOn the 12th day after the surgery the patient was discharged from the hospital in a good state of health with standard dietary and medical recommendations. There were no postoperative complications, no symptoms of dysphagia, nausea or vomiting.3The esophageal resection with right-side thoracoscopy and intrathoracic anastomosis of mediastinal esophagus and gastric tube has a number of advantages compared to thoracotomic access. They are less operative trauma, reduced blood loss, convenience in forming a gastroesophageal anastomosis, faster and better straightening of the right collapsed lung. The improvement of thoracoscopic technique leads to decrease of the operative time and makes it comparable to thoracotomic access operation duration. In comparison with transhiatal interventions, the advantage of this operation is the perfect exposure that allows performing complete esophageal dissection and possible en bloc resection ,11.A damage of the membranous part of the trachea and bronchi during esophagectomy is a rare complication which can be caused by the technical difficulties of dissecting the esophageal tumor of a large size, excessive traction of the esophagus in the area adjacent to the membranous part of the trachea, and is facilitated by sclerotic changes of muscle fibers connecting the esophagus and the trachea. The membranous part of the trachea has a delicate, fragile structure, which requires an adequate selection of suture material for its suturing. The use of intracorporeal continuous Stratafix 3.0 suture with subsequent strengthening by the mediastinal pleura makes it possible to achieve impermeability of the respiratory tract. If possible, early postoperative extubation of the patient contributes to the prevention of respiratory failure.More thorough selection of patients undergoing thoracoscopic esophageal resection together with gentle manipulations with the esophagus in the area adjacent to the trachea can prevent intraoperative tracheobronchial damages. Timely diagnosis of such serious complications makes it possible to successfully manage them using thoracoscopic techniques.4We have successfully managed intraoperative tracheal and bronchial rupture during thoracoscopic resection for esophageal cancer with use of the intracorporeal suture.The authors declare no conflicts of interest. The authors have no financial, consultative, institutional and other relationships that might lead to bias or conflict of interest.None.Ethical approval has been exempted by our institution, NGHCI Railway Clinical Hospital at the “Rostov-Glavnyy” station, OAO Russian railways, Rostov-on-Don, Russian Federation.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.Khitaryan А. – conceptualization, funding acquisition, investigation, methodology, project administration, resources, supervision, verification, writing original draft, writing review & editing.Miziev I. – conceptualization, funding acquisition, investigation, methodology, project administration, resources, supervision, verification, writing original draft, writing review & editing.Veliev K. – conceptualization, writing original draft, writing review & editing.Voronova O. – data curation, investigation, writing original draft, writing review & editing.Golovina A. – conceptualization, formal analysis, methodology, project administration, data curation, formal analysis, software, resources, visualization, writing original draft, writing review & editing.Zavgorodnyaya R. – data curation, investigation, writing original draft, writing review & editing.Kovalev S. – data curation, investigation, writing original draft, writing review & editing.Alibekov A. – data curation, investigation, writing original draft, writing review & editing.Orekhov А. – conceptualization, writing original draft, writing review & editing.None.This publication is neither ‘first-in-man study’ nor a research.Khitaryan А.G.Golovina A.A.Not commissioned, externally peer reviewed.


Joubert syndrome is a rare autosomal recessive neurodevelopmental disease characterized by abnormal breathing patterns composed of episodic tachypnea/apnea, hypotonia, ataxia, developmental delay, intellectual impairment, ocular impairment, renal cysts, and hepatic fibrosis. We report the case of a 4-year-old boy who presented with global developmental delay, bilateral nystagmus, and gaze instability with difficulty walking and maintaining an upright posture. A detailed examination revealed facial dysmorphic features with a depressed nasal bridge and deepened orbital sockets. Neurological examination yielded positive results for hypotonia, gait ataxia, bilateral horizontal pendular nystagmus, and a grade 1 ptosis more prominent in the right eye. However, no abnormal breathing patterns were observed in our case. Magnetic resonance imaging revealed the characteristic molar tooth sign and a batwing appearance of the fourth ventricle. Joubert syndrome (JS) is an autosomal recessive neurological disorder named after Marie Joubert in 1969 -4. ClassA 4-year-old boy presented to the radiology department as a referred case from the department of pediatrics, where he was primarily admitted for cough, bilateral nystagmus, and gaze instability. His past medical history revealed his difficulty in walking and maintaining an upright posture. These symptoms emerged at six months of age and gradually worsened. Further questioning of the patient’s mother revealed poor and delayed developmental milestones. He achieved rollover at the age of five months and social smile at the age of three months. Currently, he is barely able to stand on his own and requires permanent support. He was delivered via an uncomplicated cesarean delivery and weighed 2.6 kg at birth. The postnatal history was negative for prolonged admission at the neonatal intensive care unit. Apart from a mild cough, there was no history of asthma, feeding difficulty, or respiratory problems. Detailed family history was taken, revealing a consanguineous marriage with a single male child. No other prior members in their family were affected.On physical examination, the child appeared extremely thin and fragile. Mild facial dysmorphia was noted with a depressed nasal bridge and deepened orbital sockets. He weighed less than the 25th percentile (6.0 kg) on the pediatric growth chart for his age. He appeared to be awake and alert. However, when instructed, he was unable to focus his gaze on specific objects. Ocular examination revealed bilateral horizontal pendular nystagmus without myopia. Grade 1 ptosis that was more prominent in the right eye than the left was noted. The cardiovascular examination proved to be normal. A pulmonary examination revealed a prominent expiratory wheeze without chest indrawing. Examination findings of the cranial nerves, apart from the oculomotor nerve, were normal. The motor exam revealed hypotonia and ataxia with normal tendon reflexes.A full series of magnetic resonance imaging (MRI) scans were conducted at our radiology division to determine the cause of the delayed milestones and to figure out the source of the hypotonia and bilateral nystagmus. The axial T2-weighted MRI revealed total aplasia of the cerebellar vermis with prominent, thickened, and elongated superior cerebellar peduncles forming a characteristic molar tooth appearance. Furthermore, the fourth ventricle appeared enlarged and triangular, giving it a slight batwing appearance Figures -3. BasedJS is known to be underreported as having a prevalence of less than one in 100,000. Until 2009, approximately 200 cases of JS had been reported worldwide, with a further 12 cases being reported to date . It was Cilia are either motile or non-motile organelles that, via ciliogenesis, transport proteins in either direction. A defect in ciliogenesis results in the abruption of various signaling pathways such as Wnt, sonic hedgehog, planar cell polarity, and directional movement. One such major transduction pathway plays a vital role in regulating neuronal cell proliferation and axonal migration . JS is kThe term JS is reserved for individuals who fulfill only the diagnostic criteria of developmental delay, abnormal ocular movements, radiological evidence of molar tooth sign, and cerebellar vermis changes . The terThe examination reveals facial dysmorphic features, including a large head, prominent forehead, rounded eyebrows, epicanthal folds, ptosis, upturned nose with evident nostrils, and low-set tilted ears . HypotonFrom a radiological perspective, a characteristic molar tooth sign can help guide the diagnosis of JS . MTS resManagement strategies are aimed at supportive and symptomatic treatment. Special care needs to be taken to manage respiratory and feeding problems. Cognitive difficulties require appropriate rehabilitation strategy, and regular follow-up . These pOur patient presented with prototypical findings of hypotonia manifested as difficulty maintaining an upright posture since the age of six months. He also presents with features of bilateral nystagmus and oculomotor dysfunction. However, no respiratory pattern dysregulation was noted in our patient; the late diagnosis of the child may account for an improvement in breathing dysregulation with age. Also consistent with the diagnosis of JS were the MRI findings of molar tooth appearance, the batwing appearance of the fourth ventricle, and hypoplasia of the cerebellar vermis. Due to the absence of multi-organ involvement, the diagnosis of JSRD was omitted, and that of pure JS was made. A possible role of consanguinity was also reported by İncecik et al. at a rate of 63.6% . ConsistJS presents with the clinical features of respiratory dysregulation, infantile hypotonia, developmental delay, nystagmus, oculomotor disturbance, and intellectual impairment. Due to the variability in clinical phenotypes, a delayed diagnosis is possible. Diagnosis requires essential diagnostic criteria in clinical history, MRI findings, and a multi-gene panel. The prototypical MRI finding is a molar tooth appearance with concomitant cerebellar vermis hypoplasia and a batwing configuration of the fourth ventricle. Management of such cases includes easing respiratory and feeding difficulties, along with rehabilitation for cognitive and behavioral difficulties.


Intestinal obstruction ascribed to internal hernia is quite rare, especially in adults. There are no differentiating features in the presentation of intestinal obstruction due to internal hernia as compared to other causes. Delay in the diagnosis of this condition carries a considerable risk especially in a virgin abdomen. We report a rare case of internal hernia which presented as acute small and large bowel obstruction.We report a 47- year- old male with generalized abdominal pain associated with vomiting and obstipation. The patient was in hypovolemic shock that only had a transient response to resuscitation. CT scans of the abdomen with contrast was done and showed both large and small bowel obstruction. Exploration laparotomy was done and revealed a concurrent nonviable portion of ileum and twisted sigmoid colon (volvulus) which protruded through a congenital transmesentric defect. Resection was mandatory, and repair of the defect was done.Incidence of internal hernia generally does not exceed 1%. The diagnosis of congenital internal hernia relies on absence history of trauma, inflammatory process and abdominal surgery. Protrusion of simultaneous small and large bowels together through transmesenteric congenital gate is uncommon.Whether the patient presenting with intestinal obstruction has a history of undergoing previous surgeries (for any reason) or not, the diagnosis of internal hernia must be kept in mind. Coexisting involvement of both small and large bowels that need resection poses the question of the need for restoration of bowel continuity with either colostomy or ileostomy. The pre23), hemoglobin (10.4 g/dl), platelets (136 × 103/μl), INR 1.7. Plain abdominal radiographs showed dilated small and large bowel loops , BUN (27.0), Na (143) and normal WBC count  or not, the diagnosis of internal hernia must be kept in mind. Clinical sense and early diagnosis when possible aid tremendously in prevention of catastrophic consequences . Trying to prove the diagnosis of internal hernia radiologically without consideration of patient clinical status may delay surgical intervention thereby increasing mortality and morbidity. Finally, involvement of both Small and large bowels by the obstruction can lead to strangulation and eventually resection need to be performed. This may necessitate colostomy versus ileostomy.The authors declare no conflicts of interests.The authors did not receive any fund.This case report is exempted from ethical approval by our institution.The authors state that they have written and signed consent from the patient to publish this report.Dr. Malek Al-Omari: Conceptualization, Methodology, Validation, Investigation, Writing – original draft, Writing – review and editing, Visualization, Supervision. Dr. Mohammad Al-Doud: Formal Analysis, Resources, Data curation, Writing – review and editing.The authors don’t need to register this work.Dr. Malek A. Al-Omari, the corresponding author of this manuscript accept full responsibility for the work and the conduct of the study, access to the data and controlled the decision to publish.Not commissioned, externally peer-reviewed.


Colonic diverticular disease is a common condition, affecting 50% of the population aged above 80. In contrast, rectal diverticular disease is a rare condition with very few cases reported, while symptomatic rectal diverticular disease is even rarer. We present a case of a symptomatic large rectal diverticulum presenting with a retrorectal abscess. A 49-year-old Caucasian female was brought to the emergency department complaining of abdominal pain and weakness in the lower limbs. She was found to have obstructive uropathy and unilateral sciatic neuropathy. She rapidly developed acute abdomen and emergency laparotomy revealed a giant purulent rectal diverticulum. The patient underwent exploratory laparotomy and a loop colostomy was made to decompress the colon. •Colonic diverticular disease is a common condition, affecting 50% of the population aged above 80.•In contrast, rectal diverticular disease is a rare condition with very few cases reported, while symptomatic rectal diverticular disease is even rarer. We repo2A 49-year-old Caucasian female was brought to the emergency department complaining of abdominal pain and weakness in the lower limbs. She had developed lower abdominal pain radiating to the lumbar region, along with gradually deteriorating paraparesis 24 hours prior to her presentation. The patient's past medical history included a colectomy 17 years previously, due to endometriosis.In the emergency department, the patient was anuric and tachycardic. Physical examination revealed diffuse abdominal tenderness on deep palpation and mild rebound (rigidity/tenderness). Digital rectal examination revealed stenosis of the colectomy's anastomosis. Neurologic examination revealed asymmetric paraparesis and hypoesthesia in the lower limbs, affecting hip extension, knee flexion, ankle dorsiflexion, plantarflexion, eversion and big toe extension, with brisk tendon reflexes in the knees but absent in the ankle, in keeping with bilateral sciatic neuropathy, worse on the left. Laboratory tests revealed leukocytosis and impaired renal function . Ultrasound and CT scan of the abdomen and pelvis was performed, which demonstrated a gas filled cavity rectal dilatation, pyeloureteral dilatation, in addition to free fluid and stranding of the perirenal area bilaterally, in keeping with obstructive uropathy, attributed to bilateral compression of the ureters by the gas filled cavity initially presumed to be dilated rectum .Fig. 1AxBilateral nephrostomies were inserted. In the subsequent 24 hours the patient deteriorated with acute abdomen, bilateral sciatic neuropathy and sepsis. MRI and subsequent CT scan of the pelvis were performed, both of which demonstrated a sizeable (13 × 8 × 8 cm), thick-walled, enhancing, true rectal diverticulum, originating from the right lateral rectal wall, accompanied by extensive inflammation of pelvic structures , Fig. 3.The patient underwent exploratory laparotomy. A loop colostomy in the descending colon was made, in order to confront the acute abdomen and to decompress the obstruction of the upper rectum. The diverticulum was detected, strongly attached to the postperitoneum . A perit3Diverticular disease of the colon is a common condition developed countries, affecting 50% of the population aged above 80. Colonic diverticula are acquired herniations of the mucosa and part of the submucosa through the muscularis propria. Diverticula are more frequently seen in the distal colon, with the sigmoid colon being affected in 90% of the patients .In contrast, rectal diverticular disease is a rare condition with very few cases reported. They make up solely for 0.1% of colonic diverticular disease . Two majIn contrast to colonic diverticula, most rectal diverticula are true diverticula, involving all three layers of the rectal wall. In addition, they largely accompany colonic diverticulosis. Nevertheless, rectal pseudodiverticula and solitary rectal diverticula have been reported ,8. The mTwo main factors contribute to the pathogenesis of rectal diverticula; 1. A pressure gradient between the bowel lumen and the mucosa; 2. Areas of relative weakness of the bowel wall. This theory is supported by the fact that rectal diverticula are true ,4,7. SurRectal diverticulosis is usually an incidental diagnosis. In some rare cases, rectal diverticula become symptomatic, presenting with symptoms of diverticulitis , as well as strictures, ileus, change in bowel habits, rectovesical fistula, rectal prolapse, perineal mass and retrorectal cyst. To our knowledge, sciatic neuropathy and bilateral obstructive uropathy, as in our case, have never been reported before. Asymptomatic patients do not require treatment, whereas symptomatic rectal diverticulosis necessitates surgical intervention, if possible ,3,7,8. AIn conclusion, clinicians should be aware of symptomatic rectal diverticulosis as a rare but potentially clinically significant complication of rectal surgery. Operations in the rectal wall which affect either the continuity or the muscular lining, may form points of least resistance, thus predisposing for the development of rectal diverticula. In our case, it emerged clinically as a large retrorectal abscess, requiring prompt intervention.Authors are aware that Annals of Med and Surg is committed to following the highest standards of publication ethics as promoted by the Committee on.Publication Ethics.No funding.Stefanos Gorgoraptis, Medical Student: contribution in the narration of the manuscript.Sofia Xenaki, MD MSc FACS mESCP : contribution in the narration of the manuscript, assisted in the operation and contributed in the collection of data.Elias Athanasakis, MD PhD: Surgeon and contribution in the narration of the manuscript.Anna Daskalaki, MD: contribution in the narration of the manuscript.Konstantinos Lasithiotakis, MD PhD FEBS: reviewed the manuscript.Evangelia Chrysou, MD PhD: was the specialized Colorectal Radiologist.Emmanuel Chrysos, MD PhD FACS: was the Prof of the Department and reviewed the manuscript.4980.Emmanuel Chrysos MD PhD FACS.Professor of Surgery.Head of the Surgical Department.The patient has given consent for possible publication of this case report.The patient has given consent for possible publication of this case report.Not commissioned, externally peer reviewed.The Authors declare that have no Conflict of Interest.


Relatively cysts that contain papillary carcinoma are large.•We present a 53-year-old with a papilloma in a large cyst of the breast that presented a painless palpable mass.•Excisional biopsy may be needed to make a proper diagnosis for the suspicious lesion. Intracystic lesions of the breast prove to be benign more often than malignancy, most intracystic lesions are benign papillomas. Generally sizes of cysts that contain papilloma are small. Relatively cysts that contain papillary carcinoma are large. A papilloma in a large cyst of the Breast is rare.We report a rare case of a papilloma in a large cyst of the breast that presented a painless palpable mass. A 53-yr-old woman noticed palpable mass in her right breast. Ultrasonography indicated a 7 × 7 cm sized large cyst. There is a 2 × 2.5 cm sized hyper-echoic, polypoid mass in the cyst. On fine-needle aspiration was not obtained malignant cells. Excisional biopsy was done. Pathologic finding was papilloma.Tumors in a large cyst of the breast is difficult in distinguish benign from malignancy by preoperative imaging, cytological and histological study.Excisional biopsy should be performed before cancer surgery such as mastectomy to manage the cysts with intracystic growths. Almost the lesions prove to benign more than malignancy . The sizThe work in this case has been reported in line with the SCARE criteria .2A 53-yr-old woman visited our hospital due to palpable mass in her right breast that had been rapid growing for approximately 2 months. The patient did not complain pain and had no nipple discharge. She had no specific family history and breast cancer risk factors. On physical examination, we palpated a well marginated, round, movable mass in right breast subareolar area.We ordered a check mammography for breast lesion. But patient was not able to test because of the pain during examination. Ultrasonography showed a 7 × 7 cm sized large cyst. There is a 2 × 2.5 cm sized hyper-echoic, polypoid mass in the cyst . On fineWe explained the result of examination to the patient as the cystic mass would be benign rather than malignancy, but malignancy was not ruled out. So we recommended surgical removal of the cystic mass, the patient agreed that. Excision biopsy was performed under general anesthesia. Amount of cystic fluid was 250cc. The resected specimen was a large cyst, measuring 7 × 7 cm and a intracystic papillary tumor, gray-brown color, measuring 2 × 2.5 cm, on the base of the cyst wall . The his3Intracystic breast lesions are associated with a variety of benign, atypical, and malignant pathologic diagnoses and the lesions prove to be benign more often than malignancy, Most intracystic lesions are benign papilloma ,2,6.The cyst develop by secretion and bleeding of an intraductal papilloma which distends and obstructs lactiferous ducts, causing cyst, and papilloma can be visualized inside the cyst . IntracySymptoms of intracystic papillomas are mainly painless, non-tender, palpable mass in subareolar area. Diagnosis is made by mammography, ultrasonography, computed tomography, magnetic resonance imaging and FNA or core needle biopsy. The mammographic finding of intracystic papilloma is non-specific, well marginated mass or negative on small lesion. Sonographically, it usually has a well circumscribed, septated cystic mass including irregular solid, hypo-echoic portion . HoweverThere have been some reports that describe the methods to distinguish between intracystic papillomas and intracystic papillary carcinomas preoperatively . Shah 1 reported4A papilloma in a large cyst of the breast very rarely occurs. Although the size of the cysts with intracystic growths is large, it may be a benign tumor. Therefore, excisional biopsy should be performed before cancer surgery such as mastectomy to manage the cysts with intracystic growths.No potential conflict of interest relevant to this article was reported.None.Not applicable. The study is exempt from ethical approval in our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying figures.Kim Hwan Soo and Yang Hei Kim- Data collection, writing the paper.Sung Bae Park - study concept, writing the paper, advised and designed the report.N/A.Sung Bae Park.Not commissioned, externally peer reviewed.


Pituitary metastasis is a rare condition with a poor prognosis. Very few patients with pituitary metastasis are symptomatic. It is often associated with presence of co-existing metastases to other organs. Isolated pituitary metastasis as the first presentation of primary malignancy is uncommon.nil per os for 10 h prior to EGD, after which she developed alteration of consciousness. Further investigation revealed hypernatremia with sodium level of 161 mmol/L and low urine osmolality of 62 mOsm/kg. Her urine output was 300 mL per hour. Diabetes insipidus (DI) was diagnosed based on evidence of polyuria, hypernatremia, and low urine osmolality. Her urine output decreased and urine osmolality increased to 570 mOsm/kg in response to subcutaneous desmopressin acetate, confirming central DI. Pituitary magnetic resonance imaging showed a heterogeneous gadolinium enhancing lesion at the sellar and suprasellar regions, measuring 2.4 × 2.6 × 3.9 cm compressing both the hypothalamus bilaterally and the inferior aspect of optic chiasm as well as displacing the residual pituitary gland anteriorly. The posterior pituitary bright spot was absent. These MRI findings suggested pituitary macroadenoma. There were also multiple small gadolinium-enhancing lesions up to 0.7 cm in size with adjacent vasogenic brain edema at the subcortical and subpial regions of the left frontal and parietal areas, raising the concern of brain metastases. Pituitary hormonal evaluation was consistent with panhypopituitarism. Histopathological and immunohistochemical studies of the pituitary tissue revealed an adenocarcinoma, originating from the lung. Computed tomography of the chest and abdomen was subsequently performed, showing a 2.2-cm soft tissue mass at the proximal part of right bronchus. There was no evidence of distant metastases elsewhere. The final diagnosis was adenocarcinoma of the lung with pituitary metastasis manifesting as panhypopituitarism and central DI. Palliative care along with hormonal replacement therapy was offered to the patient. She died 4 months after diagnosis.A 72-year-old woman presented with a 2-month history of polyuria, increasing thirst and unexplained weight loss. Esophagogastroduodenoscopy (EGD) was scheduled as part of the investigation. She was kept Diagnosis of pituitary metastasis is challenging, especially in patients with previously undiagnosed primary cancer. It should be considered in the elderly patients presenting with new-onset central DI with or without anterior pituitary dysfunction. Metastasis to pituitary gland is a rare situation in clinical practice with a poor prognosis. The prevalence of metastatic pituitary tumors was approximately 1% among all pituitary tumor resections . Most ofPituitary metastasis is often associated with the presence of multiple additional metastatic sites, especially in bones , 6. IsolA 72-year-old woman with a 5-pack-year smoking history presented with drowsiness after esophagogastroduodenoscopy (EGD). Her past medical history was remarkable for community-acquired pneumonia at right middle lung Fig. a 3 monthnilper os for 10 h prior to the procedure. The EGD findings were mild non-erosive antral gastritis. After EGD, she developed nausea, vomiting, and drowsiness. Physical examination revealed a body temperature of 37 °C, a blood pressure of 100/57 mmHg, a pulse rate of 90/min, and a respiratory rate of 16/min. She was 44.5 kg in body weight, was 148 cm in height, and had a body mass index of 20.3 kg/m2. She had flat neck veins, a normal thyroid gland without nodules, normal breath sounds, no abnormal palpable masses, no hepatosplenomegaly, no breast masses, and no superficial lymphadenopathy. Neurological examination was remarkable for bitemporal hemianopia evaluated by confrontation test.The patient underwent EGD 2 months after the outpatient visit. She was kept She was immediately admitted to the hospital because of the altered mental status. At the first hour after admission, she had polyuria with a urine output of 300 mL/hour (6.7 mL/kg/hour). Laboratory tests showed a serum sodium level of 160 mmol/L; a potassium level of 3.9 mmol/L; a chloride level of 125 mmol/L; a bicarbonate level of 24 mmol/L; a creatinine level of 1.4 mg/dL. Serum osmolality was 325 mOsm/kg. Her urine specific gravity was 1.002 without proteinuria or glucosuria. Urine osmolality was 62 mOsm/kg. Diabetes insipidus was diagnosed based on evidence of polyuria along with hypernatremia and low urine osmolality. Desmopressin acetate (DDAVP) 1 microgram was given by subcutaneous injection. One hour later, her urine output decreased to 70 mL/hour, and urine osmolarity increased to 570 mOsm/kg. Based on decreasing urine output and a more than 50% increase in urine osmolality in response to DDAVP, a diagnosis of central diabetes insipidus was made.Given the diagnosis of central diabetes insipidus, further investigations including magnetic resonance imaging (MRI) of the pituitary gland and evaluation of the anterior pituitary hormones were performed. Pituitary MRI demonstrated a heterogeneous high signal intensity (SI) lesion in T1-weighted (T1W) imaging, which also appeared as a low SI lesion in T2-weighted (T2W) imaging with heterogeneous gadolinium enhancement at the sellar and suprasellar region, measuring 2.4 × 2.6 × 3.9 cm. The lesion was compressing the hypothalamus bilaterally and the inferior aspect of optic chiasm with increased SI in T2W imaging at the right optic nerve and bilateral optic tracts. It was also anteriorly displacing the residual pituitary gland. The pituitary stalk could not be identified, and the bright spot of posterior lobe was absent. These MRI findings suggested pituitary macroadenoma with hemorrhage  but negative for cytokeratin-20, CDX-2 and GFAP Fig. . These fTo identify the primary adenocarcinoma of the lung, a CT scan of the chest was performed which revealed a 2.2 × 2.2 cm enhancing soft tissue mass at the proximal part of the right bronchus that supplies the medial segment of the right middle lung. This was causing atelectasis of the distal part of the medial segment of the right middle lung and diffuse centrilobular emphysema at both upper lobes without pleural effusion. The liver, spleen, kidneys, and adrenal glands appeared unremarkable. No osteolytic lesion of the ribs, spine, and other bony structures were seen Fig. . The finPalliative care for the advanced-stage lung cancer was offered to the patient. She received hormone replacement including desmopressin acetate 10 mcg intranasal solution daily, oral prednisolone 5 mg daily, and levothyroxine 75 mcg daily for central diabetes insipidus, secondary adrenal insufficiency, and central hypothyroidism, respectively. Two months later, she developed bilateral ophthalmoplegia and died 4 months after the diagnosis.Pituitary metastases are usually detected in patients with advanced cancer and associated with widespread metastases, typically affecting elderly patients, especially those aged 60–70 years . They ofThe four pathways of metastases to the pituitary gland –23 incluRegarding the location of metastatic tumors in the pituitary, 84.6% were found at the posterior pituitary either alone or in combination with the anterior pituitary whereas only 15.4% were found at the anterior pituitary alone . Also, tThe majority of pituitary metastases are clinically silent. Symptomatic pituitary metastases were found in only 7% of cases , and it The analysis of our patient’s hormonal profiles showed panhypopituitarism with mild hyperprolactinemia and central diabetes insipidus, corresponding to her MRI findings that both the anterior and posterior pituitary glands were involved by metastasis.Other symptoms of pituitary metastases included visual field deficits due to compression of the optic chiasm in 27.9% of cases, ophthalmoplegia caused by cranial nerve III, IV, or VI palsy in 21.6% of cases, headache and/or post-ocular pain in 15.8% of cases, and fatigue and/or general malaise in 7.9% of cases [Some clues to raise clinical suspicion of pituitary metastasis in our patient were onset of diabetes insipidus at an older age, rapidly progressive weight loss, and fatigue. These were likely symptoms of tumor burden or hypopituitarism. Also, the recent onset of bitemporal hemianopia was a clinical clue of optic chiasm compression.The radiographic findings of pituitary metastases are usually non-specific. In MRI studies, iso- or hypointensity signals on T1W imaging and hyperintensity on T2W imaging with homogeneous enhancement after gadolinium injection are the common findings. Imaging studies may be fruitful for distinguishing metastatic disease when the pituitary stalk is thickened, and invasion of the cavernous sinus along with sclerosis of the sella turcica are seen. Nonetheless, there were no such discriminative characteristics seen in our patient except for the presence of concomitant brain lesions with the same signal intensity as that of the suprasellar mass, which offered a diagnostic clue to metastatic pituitary tumor .The prognosis of patients with pituitary metastasis is poor due to the aggressiveness of the primary malignancy. The mean survival after developing a pituitary metastasis is only 6 months as in our patient, and 1-year survival is less than 10% .In conclusion, metastasis to the pituitary gland is a rare and life-threatening condition that results in poor prognostic outcomes. It is difficult to diagnose, especially in patients with previously undiagnosed primary cancer. Pituitary metastasis should be considered in elderly patients presenting with new-onset diabetes insipidus with or without anterior pituitary dysfunction. MRI findings of pituitary stalk thickening, invasion of cavernous sinus, sclerotic changes around the sella turcica, and coexistence of brain lesions are also important diagnostic clues.


MLH1, MSH2, MSH6, or PMS2) cause this devastating disease. Given the rarity of the syndrome, often-asymptomatic tumors, and overlap with neurofibromatosis-1, diagnosis is frequently unrecognized or delayed. We present a unique case of a 14-year-old female with minimal gastrointestinal symptoms diagnosed with invasive adenocarcinoma secondary to biallelic PMS2 variants.Constitutional  Mismatch Repair Deficiency is a rare autosomal recessive disorder characterized by numerous cancers presenting as early as the first decade of life. Biallelic germline variants in one of four mismatch repair genes ( PMS2 [ PMS2 mutations.Constitutional  Mismatch Repair Deficiency (BMMRD) is a rare autosomal recessive disorder characterized by early-onset cancers as early as the first decade of life. It is caused by inherited biallelic mismatch repair (MMR) gene variants, most commonly PMS2 –4. This  PMS2 , 6. Affe PMS2 . While u PMS2 , 9. Nota PMS2 . We presThe proband is a 14-year-old previously healthy female born to nonconsanguineous healthy parents who was admitted for having fever, fatigue, lower quadrant abdominal pain, and vomiting. Abdominal computerized tomography (CT) revealed significant hydronephrosis consistent with a right ureteropelvic junction (UPJ) obstruction, for which she underwent ureteral stent placement on hospital day 2. Her postoperative course was complicated by continued abdominal pain and fever, as well as an episode of rectal prolapse. Of note, she also endorsed an episode of rectal prolapse months prior to presentation which was manually reduced at home. On hospital day 6, due to continued fevers, abdominal and pelvic MRI was obtained. This study revealed a pelvic fluid collection concerning abscess; CT guided transgluteal drainage performed by Interventional Radiology resulted in 150ml cloudy yellow fluid. The etiology was believed to be an infected urinoma caused by instrumentation during stent placement and she was treated with ceftriaxone and metronidazole. The patient's fevers and pain continued, prompting an abdominal and pelvic CT on hospital day 10. The imaging was notable for diffuse ascites, bowel wall thickening, and organizing fluid collections within the pelvis. She ultimately went for exploratory laparotomy and washout with intraoperative findings of multiple pockets of turbid fluid. The entire bowel was evaluated and there was no evidence of perforations, fistulas, or other causes of gastrointestinal leakage. Peritoneal fluid cultures grew polymicrobial organisms, however, suggestive of gut flora. She was transitioned to meropenem and fevers eventually resolved prior to discharge. MLH1, MSH2, and MSH6. There was complete loss of expression of PMS2; this testing was deemed indeterminate due to lack of staining in both tumor tissues and normal tissue.Two months following discharge the patient underwent outpatient elective appendectomy, right pyeloplasty, and ureterotomy with stent placement due to persistent hydronephrosis. Pathology revealed normal appendix tissue. Ureter pathology was significant only for acute and chronic inflammation, with no evidence of malignancy. Routine screening abdominal ultrasound 2 months later revealed stable hydronephrosis; however, an incidental 3cm soft tissue mass presumed to be near the sigmoid colon was noted. Follow-up MRI showed nearly circumferential thickening of the sigmoid colon which was subsequently evaluated by esophagogastroduodenoscopy and colonoscopy. Findings were significant for 9-10 sessile polyps in transverse, descending, and sigmoid colon with large cluster of irregular appearing polyps in the sigmoid . Histolo MLH1, MSH2, and MSH6, suggesting intact proteins. There was again loss of expression of PMS2 in both tumor and nontumoral cells . Familial testing concluded that one variant was inherited from each parent, with her asymptomatic brother also carrying the father's PMS2 variant.The patient underwent repeat colonoscopy approximately 3 weeks after her first procedure with an adult interventional gastroenterologist. Findings were significant for two tubular adenomas in the colon at 25cm and 30cm from the anus, moderately differentiated adenocarcinoma at 24cm, a moderately differentiated adenocarcinoma in the rectum (10-15cm), and an anorectal mass with moderately differentiated adenocarcinoma arising in the background of tubulovillous adenoma with extensive high-grade dysplasia. Both tubular adenomas were removed by snare polypectomy, while the remaining masses were biopsied. Immunohistochemical staining was positive for CDX2, CK7, CK20, and villin. There was no loss of expression ofal cells  consisteDue to the findings of invasive disease as well as previous pelvic fluid suggestive of a microperforation, the diagnosis of Stage 4 adenocarcinoma was established. Chemotherapy was initiated with Capecitabine  followed by two cycles of FOLFOX6 , as well as 6 weeks of radiation. She finally underwent total colectomy along with total abdominal hysterectomy with bilateral salpingectomy, and pathology revealed no evidence of residual adenocarcinoma in the colon.Although colorectal cancer in children is rare, this case demonstrates that invasive disease can be diagnosed in the pediatric population. While GI manifestations of BMMRD are well described and fit the phenotype of our patient's numerous adenomas and adenocarcinomas, our patient's only true gastrointestinal symptom was rectal prolapse, making her diagnosis even more challenging until incidental findings on ultrasound. More importantly, she lacked the most common features of familial consanguinity or café-au-lait macules which would trigger further investigation into genetic disease. It is extremely important to recognize BMMRD given its association with additional cancers including brain tumors, hematologic malignancies, and rare cases of urologic malignancies . The European consortium “Care 4 CMMRD” published guidelines for diagnosis in 2014 . Data waMLH1, MSH2, MSH6, and PMS2). However, the majority of BMMRD patients with GI cancers report biallelic mutations in PMS2. In classic LS, monoallelic carriers of PMS2 variants have a lower penetrance for GI cancers. The combination of the early-onset cancer in the proband, later-onset cancer in the parents, and incomplete penetrance for PMS2-LS leads to a family history that is often negative [ PMS2 variants would remain unaffected and produced offspring without being aware of his/her own risk of cancer development. Our case is unique in the fact that there was no consanguinity in the family. Forty-six of the 91 known families with BMMRD demonstrate either consanguinity or homozygosity for one mutation [Biallelic mutations have been reported in all four DNA MMR genes (negative . This mamutation . It is p


A 67-year-old woman presented with postmenopausal vaginal bleeding. Full body imaging demonstrated an intrauterine mass with deep myometrial invasion but no nodal or other metastatic disease. Uterine curettage was performed. Histologically, the tumor was an endometrioid adenocarcinoma with sarcomatous element and a hepatoid component, the latter was immunohistochemically positive for alpha-fetoprotein, HepPar-1, and arginase-1. The patient underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy. Serum alpha-fetoprotein level decreased from 31896 ug/l preoperatively to 2063 ug/l postoperatively. Eight weeks later, a rise in serum alpha-fetoprotein was detected, and a biopsy-proven vaginal recurrence was diagnosed. Palliative chemotherapy led to tumor shrinkage and a concurrent decrease in the serum alpha-fetoprotein level. A rise in serum alpha-fetoprotein, refractory to second-line chemotherapy, was accompanied by subsequent development of ureteric obstruction, ascites, and radiological evidence of peritoneal metastases. This is an unusual case of uterine carcinosarcoma with an alpha-fetoprotein-producing hepatoid adenocarcinoma component. Serum alpha-fetoprotein level corresponds to disease recurrence and progression. Hepatoid adenocarcinomas are rare tumors with histologic appearance resembling malignant hepatocytes with expression of alpha-fetoprotein (AFP), both immunohistochemically and serologically. They have been reported in various sites including the lung, colon, renal pelvis, stomach, gallbladder, and the female genital tract  –7. To daA 67-year-old woman, gravida 1, para 1, with a medical history of psoriasis and bipolar affective disorder, presented with postmenopausal vaginal bleeding. Physical examination found an 18-week sized uterus without palpable groin lymph nodes. Both adnexa were unremarkable. Magnetic resonance imaging (MRI) of the pelvis and computed tomography (CT) with contrast of the abdomen and thorax demonstrated a localized anterior intrauterine mass with deep myometrial invasion. There was no pelvic or inguinal lymphadenopathy . The livThe resected specimen weighted 575 g and measured 11.0 x 9.5 x 8.0 cm with an anterior exophytic tumor measuring 7.5 x 6.0 x 4.0 cm with a tan cut surface  and detaThe postoperative clinical course was uneventful, and she was planned to receive taxotere and cyclophosphamide (TC) as adjuvant chemotherapy. Before adjuvant therapy was initiated, a tumor was found in the residual vaginal canal on 8-week postoperative follow-up clinical examination, and biopsy confirming histological recurrence. The patient proceeded to chemotherapy (TC), and the serum AFP level showed a decrease from 6152 ug/l to 4288 ug/l after the first cycle. Serological response was sustained throughout the six cycles of chemotherapy, with a postchemotherapy serum AFP level of 4770 ug/l . Follow-Hepatoid adenocarcinomas have been reported in lung, colon, renal pelvis, stomach, gallbladder, and the female genital tract  –7. The hHeterologous sarcomatous components, including most commonly rhabdomyoid , chondroLiterature review yielded only four cases of uterine carcinosarcoma with hepatoid adenocarcinoma component –10, all The current case highlighted the aggressive clinical course of this rare tumor, with a propensity of local progression over short time span. Furthermore, the disease progression can be monitored by serum AFP level. Increased level indicated possible recurrence/metastases, and failure to achieve normal AFP level also indicated compromised outcome.


Lung large-cell neuroendocrine carcinoma (LCNEC) is an aggressive and a rare type of lung cancer, and the prognosis of LCNEC with distant metastasis is extremely poor, with a five-year survival rate of 0%. Here, we report a case of laparoscopic hepatectomy for liver metastasis of lung LCNEC.A 63-year-old man received a routine physical examination, and abnormal chest radiographic findings were observed; chest computed tomography (CT) in our hospital revealed that the patient had left pneumothorax and a lesion measuring 18 mm in the inferior lingular segment of the lung. The patient underwent thoracoscopic lobectomy, and the final pathological diagnosis was lung LCNEC. Four years after surgery, abdominal CT revealed a mass measuring 27 mm in the liver. The patient underwent laparoscopic partial hepatectomy, and postoperative pathological examination showed liver metastasis of LCNEC. There was no sign of recurrence 6 months after hepatectomy.LCNEC with distant metastasis has a poor response to systemic chemotherapy, and the median survival time of patients with distant metastasis is estimated to be approximately 6 months, with a five-year survival rate of 0%. Although the common site of metastasis from LCNEC is the liver, there are no previous reports of hepatectomy for liver metastasis of LCNEC.We report a case of laparoscopic hepatectomy for liver metastasis of lung LCNEC. It is suggested that surgical resection for solitary distant metastasis of LCNEC may improve prognosis. LCNEC is an aggressive and a rare type of lung cancer that accounts for 3 % of all primary lung malignancies , and the22 of tissue. Moreover, staining of the tumor was positive for chromogranin A (18F-fluorodeoxyglucose positron emission tomography (FDG-PET) revealed a high accumulation of FDG with a maximum standardized uptake value (SUVmax) of 3.4 in the liver tumor (A 63-year-old man was diagnosed with abnormal chest radiographic findings by routine physical examination and was referred to the Department of Thoracic Surgery at our hospital. He had no past medical and surgical history. Chest CT revealed left pneumothorax and a lesion measuring 18 mm in diameter in the inferior lingular segment of the lung . The patgranin A b and syngranin A c, which granin A a. 18F-fler tumor b, and noer tumor . Using ter tumor  and stai3Lung NETs account for approximately 25% of primary lung neoplasms . In the Lung LCNEC is an aggressive and a rare type of lung cancer that accounts for 3% of all primary lung malignancies . Even LCLCNEC with distant metastasis has a poor response to systemic chemotherapy , and theConsidering the high rate of recurrence after hepatectomy, we selected laparoscopic nonanatomical hepatectomy to maximize the remnant liver volume and prepare for future repeat hepatectomy. In laparoscopic hepatectomy, surgeons cannot use their hands to palpate the liver to search for additional tumors or their fingers to mark the resection margin. Recently, fusion ICG-fluorescence imaging, which enables real-time indication of pseudocolor-fluorescence signals on white color images, serve as a simple navigation tool that provides information on tumor location, segmental boundaries, and bile duct anatomy . In part4In summary, we report a case of laparoscopic hepatectomy for liver metastasis of lung LCNEC. Recurrence-free survival for 6 months postoperative is a short duration but is longer than the median survival time previously reported. Although the prognosis of LCNEC with distant metastasis is extremely poor, these results suggest that surgical resection for solitary distant metastasis of LCNEC may improve prognosis.This study did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Ethical approval was not obtained for this case report.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.Hisoka Yamane: Concept and design of study, drafting the manuscript, participation in the treatment and perioperative management of the patient, approval of final manuscript.Sachiko Yoshida: Design of study, approval of final manuscript.Toshihiko Yoshida: Design of study, approval of final manuscript.Masayasu Nishi: Design of study, approval of final manuscript.Takashi Yamagishi: Design of study, approval of final manuscript.Hironobu Goto: Design of study, approval of final manuscript.Dai Otsubo: Design of study, approval of final manuscript.Akinobu Furutani: Design of study, approval of final manuscript.Taku Matsumoto: Design of study, participation in the treatment of the patient, approval of final manuscript.Yasuhiro Fujino: Design of study, approval of final manuscript.Kazuyoshi Kajimoto: Review of pathological findings, approval of final manuscript.Toshiko Sakuma: Review of pathological findings, drafting the manuscript, approval of final manuscript.Masahiro Tominaga: Design of study, revision, participation in the treatment of the patient, approval of final manuscript.Not applicable.Hisoka Yamane accepts full responsibility for the work and the conduct of the case report, had access to the data, and controlled the decision to publish.Not commissioned, externally peer-reviewed.All authors declare that they have no conflicts of interest in relation to this study.


Acute mesenteric ischaemia (AMI) is an uncommon cause of acute hospital admission with high mortality rates (50–90%) that requires early diagnosis and treatment. With the increase in average life expectancy, AMI represents one of the most threatening abdominal conditions in elderly patients. Untreated, AMI will cause mesenteric infarction, intestinal necrosis, an overwhelming inflammatory response and death. Early intervention can reverse this process leading to a full recovery, but the diagnosis of AMI is difficult. The failure to recognise AMI before intestinal necrosis has developed is responsible for the high mortality of the disease. Unfortunately, common CT findings in bowel ischaemia are not specific. Therefore, it is often a combination of nonspecific clinical, laboratory and radiological findings that helps most in the correct interpretation of CT findings. The purpose of this article is to provide an overview of the anatomy, physiology of mesenteric perfusion and discussions of causes, pathogenesis and CT findings in various types of acute bowel ischaemia. Familiarity with various imaging features of mesenteric injury is essential to make a timely diagnosis that will lead to improved patient outcomes.AMI is a potentially life-threatening disorder whose prognosis depends on early recognition, accurate diagnosis and timely intervention.• Arterial inflow occlusion due to thrombosis or embolisation is the most common cause of AMI.• Four aetiological types of AMI have been associated with different characteristics and risk factors .• Physical examination and laboratory findings are not sensitive or specific for diagnosing AMI; therefore, MDCT is still the first-line imaging method in suspected AMI.• Although a number of scoring systems for prognosis have been proposed, these have not been validated in large-scale studies.•  Intestinal ischaemia refers to insufficient blood flow within the mesenteric circulation to meet the metabolic demands in the bowel. It is a potentially catastrophic entity that may require emergent intervention in the acute setting. Although the clinical signs and symptoms of intestinal ischaemia are nonspecific, CT findings can be highly suggestive in the correct clinical setting.To accurately diagnose mesenteric ischaemic disease, the radiologist must be acquainted with both the mesenteric arterial and venous anatomies of the bowel. The three major arteries that supply the small and large bowel are the coeliac trunk, superior mesenteric artery (SMA) and inferior mesenteric artery (IMA) Fig. . The coeMiddle and inferior rectal arteries, branches of the internal iliac arteries, supply the distal rectum.There are numerous important mesenteric collateral pathways that provide a rich vascular safety net for the mesenteric blood supply. The gastroduodenal artery, usually the first branch of the common hepatic artery, provides an important collateral pathway between the coeliac artery and SMA. The marginal artery of Drummond  and arcade of Riolan  connect the SMA and IMA. Four arcades of anastomosis are formed between the IMA and lumbar arteries arising from the aorta, sacral and internal iliac arteries. In addition, the peripheral small mesenteric vessels are anatomically arranged in a parallel series configuration that supplies the mucosa, submucosa and muscularis propria of the bowel .The venous system returns essentially parallel to the arterial supply. The superior and inferior mesenteric veins run parallel to the arteries and drain the respective part of the bowel. The inferior mesenteric vein (IMV) joins the splenic vein, and the splenic vein joins the superior mesenteric vein (SMV) to form the main portal vein. Numerous collateral venous pathways can form between the mesenteric and systemic veins including the gastric and oesophageal, renal, lumbar and pelvic veins.Under normal circumstances, the human bowel receives around 20% of the resting cardiac output, of which two-thirds supplies the intestinal mucosa . In the Autoregulation of intestinal perfusion may maintain tissue viability below a systemic blood pressure of 70 mmHg; however, in cases where systemic blood pressure is below 40 mmHg, local myogenic autoregulation is overruled by systemic autoregulation and local protective mechanisms fail. This results in an increasingly ischaemic bowel wall, similar to what would happen in the case of mesenteric underperfusion due to vascular occlusion .muscularis propria layers (stage II), to dangerous and life-threatening continuous necrosis of all bowel wall layers  (stage III) .Fig.Fig41].FContrarily, hyperenhancement of the bowel wall in shock bowel indicates neither hyperaemia nor hyperperfusion, but typically corresponds to prolonged enhancement of the bowel wall due to reduced arterial perfusion after vasospasm of the mesenteric arteries caused by the effects of angiotensin II or after reduced venous outflow due to contraction of mesenteric veins caused by the effects of adrenaline and noradrenaline Fig. .Mesenteric fat stranding, mesenteric fluid and ascites are also nonspecific CT findings in acute bowel ischaemia, and their presence depends heavily on the cause, pathogenesis and severity of the ischaemia as well as on its location in the small or large bowel.In exclusively arterial occlusive mesenteric ischaemia, the presence of segmental mesenteric fat stranding and free fluid interleaved between the mesenteric folds associated with the poorly enhancing bowel is highly suggestive of transmural infarction .In venous outflow obstruction. The vascular engorgement and oedema of the bowel wall in turn lead to leakage of extravascular fluid into the bowel wall and mesentery Fig. .Pneumatosis and portomesenteric venous gas have been reported as less common but more specific findings of acute bowel ischaemia, being present in 6%–28 and 3%–14% of cases, respectively.Although pneumatosis is not a specific finding of intestinal ischaemia, which may occur in a wide range of non-emergent benign scenarios, when found, bowel ischaemia must be excluded first and foremost.Pneumatosis may manifest with small isolated gas bubbles in a circumferential distribution within an ischaemic bowel wall or as broad rims of air dissecting the entire bowel wall into two layers Fig.  42]. It. It42]. Portomesenteric venous gas may consist only of some small gaseous inclusions within the mesenteric veins or may extend into the intrahepatic branches of the portal vein, where it is typically found in the periphery of the liver Fig. .Blood clot or narrowing of the lumen of the visceral branches of the abdominal aorta or low-attenuation filling defect on contrast-enhanced CT of the superior mesenteric vein has higher specificity values Fig. .The calibre of the vessels is another important criterion. Due to the venous outflow obstruction, engorged mesenteric veins are typically observed Fig. .Infarcts of other visceral organs may suggest this diagnosis Fig. .While the severity of bowel ischaemia is variable, the presence of gas within the peritoneal cavity suggests perforation and peritonitis, which are high-mortality complications of infarction Figs.  and 12.FA number of scoring systems for predicting morbidity and mortality have been proposed for AMI, but these have not been validated in large-scale studies .Radiologists play a critical role in the diagnosis and appropriate triage of patients with mesenteric ischaemia. MDCT has a high specificity and sensitivity to diagnose intestinal ischaemia. Since mesenteric ischaemia may present with many different radiological appearances, an understanding of the intestinal vascular and mesenteric anatomy as well as the pathophysiology of mesenteric ischaemic disease helps improve the diagnosis of this challenging disease.


The differential diagnosis of a congenital cutaneous vascular-appearing mass in a newborn is broad and includes both benign and malignant tumors. We report the case of a newborn who presented with an erythematous exophytic skin nodule on the right upper leg. Excision was performed due to ulceration, concern for bleeding, and for diagnosis. Pathology revealed the mass to be an infantile myofibroma. This case highlights the importance of considering a broad differential diagnosis in a newborn with a cutaneous mass. While history, physical exam, and imaging can help diagnose some cases, a biopsy or excision is often needed to distinguish benign lesions from more concerning lesions. The diagnosis of vascular-appearing cutaneous masses in an infant can be challenging. We present a neonate with a vascular-appearing ulcerated skin lesion that was presumed to be a hemangioma by the referring primary care provider, and pathology later revealed it to be an infantile myofibroma (IM). A brief review of the differential diagnoses considered for this case is provided, along with the suggested management of infantile myofibroma.An 11-day-old male was referred for evaluation of a cutaneous mass of the right upper lateral thigh . The chi3Clinical diagnosis of vascular-appearing congenital skin nodules can be difficult, and often a tissue diagnosis is required. The differential diagnosis in this child included: congenital hemangioma, juvenile xanthogranuloma, pilomatrixoma, myofibroma, and fibrosarcoma.Congenital hemangiomas are fully formed at birth, and then either undergo rapid involution , fail to involute , or undergo initial rapid involution that then stops at some point . They often have a rim of pallor and coarse overlying blood vessels and may ulcerate, as a rare complication. Juvenile xanthogranulomas are yellow, red, or purple colored nodules that are present at birth or appear in the first year of life. They are usually solitary but can be multiple and undergo growth and ulceration. Their natural history is spontaneous regression . PilomatOf note, although neuroblastoma and nasal glioma were not included in the differential diagnosis in this case, they can present as a cutaneous, vascular-appearing masses in neonates. Nasal gliomas are frequently misdiagnosed as hemangiomas, especially by nonpediatric providers. They are collections of heterotopic neuroglial tissue that present as raised red masses on the nasal dorsum. Any midline nasal mass should raise the suspicion for a glioma or encephalocele, and CT or MRI is frequently obtained for further evaluation of the lesion and possible intracranial extension. Neuroblastoma is the most common neonatal malignant tumor, with 2/3 having metastases . CutaneoInfantile myofibromas, although rare, are the most common fibrous tumors of infancy . They caMost cases of IM are thought to be sporadic. Familial forms of IM have been reported with autosomal dominant and recessive inheritance patterns , 14. MutHistopathology can provide a definitive diagnosis. IM have a characteristic histological pattern with an outer zone of spindle-shaped myofibroblasts arranged in fascicles and an inner zone of round cells with enlarged hyperchromatic nuclei surrounding thin walled hemangiopericytoma-like blood vessels. Necrosis, calcification, and vascular extension may be present in the central area. Immunohistochemical stains provide definitive diagnosis with the smooth muscle stains actin and vimentin being positive and S100 (positive in neurofibroma) and GLUT-1 (positive in infantile hemangioma) being negative.The natural history of IM is of gradual regression, possibly due to apoptosis, over the first few years of life, although some lesions exhibit an initial phase of rapid growth . Of noteThis case highlights a common clinical scenario faced by pediatricians caring for newborns: accurately diagnosing a congenital vascular skin nodule. When the diagnosis of a vascular-appearing pediatric mass is not clear, further workup includes imaging studies such as ultrasound and MRI and is done by an experienced radiologist who may differentiate a solid tumor with vascularity from a congenital hemangioma. An incisional or excisional biopsy is often performed with specific immunohistochemical stains for precise diagnosis. If there is concern for systemic disease, appropriate workup should be performed. Prompt referral to an experienced pediatric dermatologist or pediatric plastic surgeon is crucial for the best plan of care.


The extent of clinical misdiagnosis is unknown in Norway, but may be high.2 This is important as delayed treatment has unfavourable consequences.3 We report how a GP, with no clinical ultrasound experience, recorded images with a pocket-sized ultrasound device (PSUD) under supervision to confirm a complete Achilles tendon rupture. This could present a new indication for GP ultrasound.The incidence of complete Achilles tendon rupture is 18 per 100 000 patient-yearsA 36-year-old man experienced acute pain above the right heel accompanied by an audible snap while sprinting. He immediately had difficulty walking and 3 hours later consulted an on-call GP. Posterior ankle swelling with a tender depression 3 cm proximal to the calcaneum was found. Active plantar flexion against resistance was weak and Simmonds–Thompson test was ‘partially positive’ on applying a strong calf-squeeze. Based on these findings, calf muscle rupture was diagnosed as the Achilles tendon was thought to be intact. The patient was advised to elevate the foot and wait 2 weeks for improvement. Two days later a second GP, who was aware of a history of an audible snap, considered complete tendon rupture and reexamined the patient. Findings included an absent right heel raise due to weakness, minimal active plantar flexion against gravity and lying prone, significant right ankle swelling without bruising, and an altered angle of declination. Palpation elicited no ankle bony tenderness, yet a painful gap was identified 6 cm proximal from the calcaneal attachment, along the line of the Achilles tendon. Simmonds–Thompson's test was clearly positive. The positive Simmond’s triad indicated a clinical diagnosis of complete rupture of the Achilles tendon.A 3.4–8 MHz linear array probe PSUD , set at a depth of 3.5 cm, was used under the supervision of a rheumatologist experienced in ultrasound. The tendon was enlarged from 1 cm to 6 cm above the calcaneal insertion, where a clear gap was seen . Two hou3Tromsø Hospital serves a large area with a population of approximately 160 000. Between 2010–2014 an average of 21 patients per year were referred by their GP for suspected Achilles rupture. Approximately one-third (8.2 patients per year) had a complete rupture confirmed by ultrasound. It is unknown how many patients were initially misdiagnosed and, although the numbers are few, this is important to investigate as any delayed diagnosis has serious consequences for patients undergoing either operative or non-operative treatment.2 despite the claim that clinical examination ‘done properly’ has a sensitivity close to 100%.4 Inadequate training5 and infrequent case exposure may be reasons for this. This case highlighted an incorrect interpretation that weak active plantar flexion excluded a complete tendon rupture. The action of deep flexor tendons was not appreciated as the reason why plantar flexion may be partially preserved. Although improved training should minimise such misinterpretation, we feel that ultrasound offers supportive visual evidence which adds value to the diagnostic process. Other benefits include reduced economic and time costs of transport, fewer orthopaedic reviews and radiologist ultrasound, and earlier access to local non-surgical treatment.Misdiagnosis rates have been reported to be as high as 22%7 The value for PSUDs is unknown and possibly lower due to poorer imaging and operator technique and/or interpretation. The VScan did however answer the clinical question about complete rupture. Research should compare both devices on a larger scale for complete rupture and alternative pathologies, such as partial rupture.Hospital ultrasound has a positive predictive value for diagnosing complete Achilles ruptures of 100%.9 yet this should not deter primary care research. This new indication for PSUDs has already benefitted emergency medicine10 and could also save orthopaedic consultation time. The role of GP ultrasound should be to reduce misdiagnosis and provide a safer opportunity to develop generic ultrasound skills. We hope this report will stimulate research collaboration to assess the impact of GP ultrasound on measurable clinical end-points related to Achilles tendon injury.Norwegian GPs already use ultrasound to assess urinary retention, fetal viability, deep venous thrombosis, gall stones, abdominal aortic aneurysm, and skin abscesses. These indications still apply despite no requirement for certified training in Norway. The evidence base for diagnostic accuracy and impact on clinical outcomes is weak,


As an extremely rare malignant neoplasm, only 12 mesenchymal chondrosarcoma (MC) arising in kidney have been reported to date. Herein, we reported a case of primary renal MC resected with robot assistance, which has not been reported before. According to the cases reported in English literature, we analyzed the characteristics of this rare malignancy and systematically review its treatment. Mesenchymal chondrosarcoma (MC), a specific histological pattern consisting of highly undifferentiated small cells and islands of well-differentiated hyaline cartilage, was initially described by Lichtenstein and Bernstein in 1959 . While mHere, we report a case of primary renal MC resected with robotic assistance, which has not been reported before to our knowledge. Along with those reported renal MC cases, the features and management of this rare neoplasm will be discussed.A 62-year-old man, with no significant medical history, was hospitalized with left loin pain and intermittent gross hematuria. Nothing except for mild costovertebral angle tenderness was found abnormal on routine physical examination. The laboratory tests including hematologic studies and urinalysis are shown in With a clinical diagnosis of left renal cell carcinoma with invasion into renal vein and metastasis to the left lung and 7th thoracic vertebra, the patient underwent robot-assisted left radical nephrectomy with renal vein thrombectomy and lymph node dissection after renal arterial embolization.The gross specimen of the removed kidney measured 16 cm × 15 cm × 9.5 cm , Vim (+)During the follow-up, the patient presented severe headache caused by skull metastasis after postoperative 2 weeks. Considering the patient’s progressively weak condition after surgery, supportive care and pain management were pursued until the patient died of widespread metastases at postoperative 1 month, and no adjuvant chemotherapy or radiotherapy was adopted.A literature search was performed in December 2018 without restrictions to regions. The primary source was the electronic PubMed database. The following terms and their combinations were searched:  and (‘chondrosarcoma’ [Title/Abstract]) and . Our computer search was also supplemented with manual searches of reference lists of all retrieved articles. About 12 cases of renal MC have been previously reported in English literature  1–3,5–1.MC comprises 2–9% of all chondrosarcomas , and mosA peripheral distribution of MC is found in patients aged 50 years and older, while the peak incidence for MC is in the 2nd–3rd decade of life . As showAs shown in The presence of calcification, the key to an imaging diagnosis of MC , was obsThe coexistence of benign tumors’ features and imaging performance similar to other malignant soft tissue tumors in extraskeletal MCs made it hard to differentiate from other tumors . The diaThe treatment of primary MC has no widely accepted guidelines due to its rarity and the lack of appropriate studies and clinical trials to assess the best management . IncreasMany studies have found that adjuvant radiotherapy for patients with MC plays a role in local tumor control, but the association with overall, disease-free, or metastasis-free survival is insignificant ,24, thatA consensus on the doxorubicin-based chemotherapy regimens as the adjuvant therapy of MC has been reached ,7,19. HoMany new treatment strategies have been introduced. Shakked et al.  introducPrognosis of MC differs with the variation of the site of tumor origin ,30. The


Sialolipoma is an extremely rare salivary gland tumor characterized by a well circumscribed mass composed of glandular tissue and mature adipose elements. Herein our aim was to present the fifth case of congenital sialolipoma, which was firstly followed up as a parotid gland hemangioma, and underline the fact that sialolipomas should be kept in mind in the differential diagnosis of congenital parotid gland masses.A 10-month old male presented with a left-sided huge neck mass which progressed after birth. Radiologic examination revealed a tumor originating from the parotid gland filling the parapharyngeal space. Histopathologic examination of an incisional biopsy was consistent with sialolipoma. A total parotidectomy with preservation of the facial nerve was performed at the age of 1 year. The postoperative recovery was uneventful with normal facial nerve function. There was no recurrence at the 24-month follow-up.Although it is a very rare benign tumor, congenital sialolipoma should be kept in mind in the differential diagnosis of congenital parotid mass. Sialolipoma was firstly described by Nagao et al. as a new type of benign salivary gland neoplasm characterized by a well circumscribed mass composed of glandular tissue and mature adipose elements . AlthougHerein our aim was to present the fifth case of congenital sialolipoma, which was firstly followed up as a parotid gland hemangioma, and underline the fact that sialolipomas should be kept in mind in the differential diagnosis of congenital parotid gland masses.A 3-month-old male was seen in our pediatric oncology department because of a growing parotid gland mass. The mass was congenital and was followed by the pediatric oncology team with a prediagnosis of hemangioma. The patient’s magnetic resonance imaging (MRI) scan showed an infantile hemangioma in the proliferative phase filling the parapharyngeal space . The child then received propranolol therapy, but despite this treatment the mass continued to grow, and the child was referred to our department at the age of 10 months. The otorhinolaryngological examination revealed an 8×7-cm mass filling the left parotid region, with normal facial nerve motor functions . HistopaWe planned to perform total excision of the mass with a transparotid approach. The facial nerve was monitored intra-operatively, and surgery was performed under magnification using surgical loops. A standard modified Blair incision was used, flaps were raised , then thThe main trunk of the facial nerve was found to be unusually elongated before pes anserinus. A soft lobular mass was encountered. A tumor involving the superficial parotid lobe was first dissected, then the deep lobe of the parotid gland and portion of the tumor filling the parapharyngeal space were gently dissected under the facial nerve . A JacksOn macroscopic examination, the mass was found to be a lipomatous specimen, 9×8×4 cm in size. The cut surface was consistent with the appearance of a lipoma. Histopathologic examination confirmed the diagnosis of a sialolipoma. The lesion was composed of mature adipose tissue mixed with acinar, ductal structures of a normal salivary gland .The postoperative period was uneventful. The patient’s facial nerve motor functions were well preserved, and he was discharged on the third postoperative day. The patient is still followed, and has no signs of recurrence in his 24th postoperative month.The term sialolipoma was first used by Nagao et al. when they described seven sialolipoma cases in their retrospective analysis of the 2,051 primary salivary gland tumor specimens . SialoliTo date, more than 40 cases of sialolipomas have been presented, but the majority are in adult patients. Sialolipoma in children is extremely rare, with only four congenital cases depicted to date –5. To thParotidectomy in small children may present some difficulties. First of all, anatomical structures are much smaller, the facial nerve is located relatively superficially in comparison with adult patients, and the mastoid process is not fully developed. Consequently, operating under magnification with surgical loops or microscopes is mandatory, and facial nerve monitoring may facilitate identification of the nerve. In our case, the main trunk of the facial nerve was found to be 5 cm, unusually elongated before the pes anserinus. We believe that this finding was due to pressure and the stretching effect of the giant tumor. Similar surgical findings were also reported by Kidambi et al . Complete surgical excision  with facial nerve preservation is the treatment of choice, and no recurrence has been reported in the literature regarding congenital sialolipomas. Our case was also followed for 24 months without any signs of recurrence.Although very rarely encountered, congenital sialolipomas should be kept in mind for the differential diagnosis of congenital parotid masses. Definitive treatment of this tumor is obtained with superficial/total parotidectomy depending on the extent of the lesion.


The hydatid cyst of the liver is a parasitic disease due to the development of echinococcosis granulosus. It is common in livestock regions in developing countries but is gaining interest in the West due to migratory flows. If it remains a benign and asymptomatic affection for a long time, its natural evolution is often enamelled of complication which can put at the risk of vital prognosis. Diagnosis and staging are based on morphological examinations, including ultrasound and CT scan. The hydatid serology retains a place especially for the detection of recurrence after hydatid cyst of the liver surgery. In addition to surgery considered up to as the radical treatment of choice, other techniques have appeared in the therapeutic arsenal in combination with oral treatment with albendazol for uncomplicated cases thus reducing the morbidity of surgery. We report a case of giant hydatid cyst associated with exophytic liver development under mesocolic associated with a peritoneal hydatidosis. The hydatic cyst of the liver generates various pathological lesions responsible for various polymorphic clinical pictures; its treatment aims to eliminate the parasite, and to solve the problem of the residual cavity and any associated complications. The therapeutic methods are numerous , medicalThis is a 57-year-old patient with no notable pathological history, originally from rural areas. He presented for 12 months of mild abdominal pain and intermittent aggravated by abdominal distention and feeling of heaviness without other associated signs or deterioration of the general condition. Clinically there is abdominal distension with generalized dullness. Apart from a positive serology, the remainder of the biological examination was unremarkable. The ultrasound shows a large heterogeneous abdominal mass whose origin is difficult to determine. A requested abdominal CT scan finds: bulging abdominal fluid formation not elevated measuring 27cm/26cm/16cm seat of parietal calcification and membranes displacing the digestive structures , this foEpidemiology: echinococcosis is an unevenly distributed zoonosis with areas of high endemicity such as the Mediterranean , the Middle East, Latin America  and China [nd China . It affend China . Factorsnd China .Clinic: circumstances of discovery are very diverse, dominated by: pain of the right hypochondrium, right basithoracic, right lumbar or epigastric. Fortuitly during a consultation for other affection, examination of employment, the balance of extension for other localization. Clinically: the abdominal inspection is most often normal. Sometimes there is a curvature of the right hypochondrium. Palpation and percussion seek hepatomegaly: it is inconstant. The hydatid cyst of the liver is sometimes seen as a rounded or oval mass, well limited, firm or renitant, painless, mobile with the liver when breathing.Abdominal ultrasound: as a key diagnostic test in view of its availability and reliability [iability , it makeiability  was the iability   by eliminating differential diagnoses, namely: a biliary cyst, a hepatic angioma, an adenoma, a hepatocarcinoma, a liver abscess or a hepatic metastasis in its cystic form. Computed tomography is also indicated in case of multiple hydatid localization, complicated hydatid cyst of the liver or in case of hydatid recurrence [currence .Hydatid serology: it finds its interest in case of doubt diagnosis especially for hydatid cyst of the liver type I and IV classification Gharbi and for detection of recurrence after surgical treatment, it is based on the detection of antiparasitic antibodies present in the blood [he blood . Severalhe blood .Objectives: the objectives of the treatment of the hydatid cyst of the liver are the destruction of the parasite, the treatment of the cystic cavity, the treatment of the possible complications and the prevention of the hydatid recurrences.Medical treatment: oral antihelminthics have a direct effect on scolex and perhaps also on the membrane with reduced permeability [eability . Albendaeability . ABZ is eability . Efficaceability . When ABeability .Percutaneous puncture (PAIR): the percutaneous treatment (PAIR) includes the percutaneous puncture of the cysts by means of an echographic or scanner control, the aspiration of the cystic fluid, the injection of a scolicidal agent for a duration of 10 to 15 min and the re-aspiration of the liquid [e liquid . The mose liquid . Standare liquid . It is ie liquid .Which way first? the classical approach is a right subcostal laparotomy that can be enlarged on the left for cysts of the left liver 2. The laparoscopic approach for the management of hydatid cyst of the liver was first reported in 1992 [ in 1992 , A retro in 1992 .What scholic agent? the use of formalin and hydrogen peroxide as an agent scolicide was discontinued following the occurrence of accidents and serious complications such as anaphylactic shock, sclerosing cholangitis and air embolism [embolism . Currentembolism .Radical or conservative surgery? radical techniques include regulated hepatectomies and total perkystectomy, while conservative surgery can be summarized as partial perkystectomy  [nt dome) . The curnt dome) .Prevention: the fight against hydatidosis cannot be conceived without the implementation of control and prophylaxis measures adapted to the local or regional context concerned [oncerned : health The hydatid cyst of the liver is a parasitic condition that can remain latent for a long time. The treatment of hydatid cyst of the liver is mainly surgical. However, it remains difficult to codify because of the diversity of pathological forms of the disease. And despite improvements in surgical techniques, high morbidity remains associated with the disease. Efforts in control, health education and prevention need to be stepped up to reduce parasitic transmission.The authors declare no competing interests.


Additional chest CT (not illustrated) confirmed another large conglomerate of enlarged lymph nodes in the superior mediastinum. Smaller nodes were also found in the cervical thoracic angles. The definite diagnosis of diffuse large B cell non Hodgkin lymphoma suggested by CT was confirmed by surgical biopsy of a cervical node. Complementary Lymphoma arise from the lymphoids cells of the immune system at different stages of differentiation. Thus they may present with a wide range of different clinical, morphological and immunologic features. Currently the term leukemia refers to the primary involvement of bone marrow and blood and the term lymphoma refers to solid tumors of the immune system.The involvement of extranodal sites (ENS) is a common feature in the course of non Hodgkin’s lymphoma (NHLs). Any organ can be involved with a predilection for the gastrointestinal tract (15–20%) and the bone marrow (15–20%). Other potentially affected organs comprise brain, testis, ovary, lung, nasopharynx, soft tissue, thyroid, kidney, liver, breast, skin, etc. Diffuse large B cell lymphoma (DLBCL) is the most common type of NHL with a prevalence of 33%. The patients may present with primary disease of lymph nodes or that of ENS and more than 50% of patients present with some site of ENS at the time of initial diagnosis.Testicular lymphoma is a rare entity accounting for less than 1% of all malignant lymphoma and for 1–9% of testicular tumors. However in patient older than 60 years it is the most common testicular tumor (45%) and the most frequent histological type is diffuse large B cell non Hodgkin lymphoma (DLBCL). Testicular lymphoma presents with three distinct expressions: as a rare primary extranodal lesion, as the primary manifestation of occult generalized lymphoma or a as secondary testicular involvement developing during the clinical course of generalized lymphoma (the most common presentation as reported in our patient). Autopsy studies have found microscopic involvement of the testis in 18,5% of NHLs. Thus when a testicular tumor is found in an elderly male or a patient already affected by malignant lymphoma the hypothesis of testicular lymphoma is strongly obvious.Ultrasound findings include focal or diffusely decreased echogenicity of a generally enlarged and painless testis and diffuse increased vascular flow is found within the mass with Colour Doppler ultrasound. The most important differential diagnosis concerns seminoma and is sometimes difficult even with MRI. Nevertheless lymphoma more commonly also involves the epididymis and the spermatic cord as reported in our patient.18F-Fluorodeoxyglucose positron emission tomography-computed tomography (18F-FDG PET-CT) has now been extensively established in the initial staging and in the evaluation of the therapeutic response of Hodgkin and NHLs. The reported case also highlights the role of 18F-FDG PET-CT in the detection of unusual or unsuspected extranodal NHLs sites that may have an important bearing on the management and prognosis.The role of positron The authors declare that they have no competing interests.