Objective: Posterior scleritis in a child is a rare condition. High-resolution imaging techniques in the course of posterior scleritis have not been published extensively in literature. The authors reported a case of posterior scleritis in a 12-year-old child to demonstrate multimodal imaging techniques in the course of development and improvement of the disease.Methods: Case report that included fundus photography, spectral domain optical coherence tomography with enhanced depth imaging, blue-peak autofluorescence, multicolor imaging, fluorescein angiography, indocyanine green angiography, and ultrasonography.Results: A twelve-year-old healthy boy presented with ocular pain and mild vision loss. His visual acuity was 20/ 32. There was no sign of inflammation on the ocular surface. There were no cells in the anterior chamber or vitreous. Ultrasonography revealed the diagnosis of posterior scleritis. When he was seen the next day for multimodal imaging techniques, he presented with exudative retinal detachment with visual acuity of 20/ 100. One week after the beginning of the therapy, ocular symptoms, and findings resolved and visual acuity improved to 20/ 20. Conclusion: Multimodal imaging techniques, which are important for the diagnosis of posterior scleritis, before and after the treatment, are presented in this case report. However, prompt diagnosis and treatment is needed to prevent progression of ocular involvement and subsequent vision loss. We presented the initial and developmental clinical features and characteristics of this condition with multimodal imaging techniques in a child with posterior scleritis. Scleritis is one of the most severe inflammatory disorders affecting the ocular surface. Posterior scleritis is scleral inflammation primarily affecting the scleral-choroidal-retinal layers posterior to equator. Posterior scleritis may involve a localized area (nodular posterior scleritis) or a more common area (diffuse posterior scleritis). In the majority of the cases, posterior scleritis is an autoimmune disease at origin. Sometimes, the diagnosis can be difficult due to variable presentation and ocular findings shared by some other ocular and periocular entities such as Harada’s disease, thyroid orbital inflammatory disease, and optic neuritis. Therefore, the disease can be overlooked or misdiagnosed . Posterior scleritis usually presents itself as ocular pain, vision loss, refractive changes, proptosis, optic disc swelling, exudative retinal detachment, a bulky appearance reminiscent of choroidal mass, retinal pigment epithelial detachments, choroidal effusion, and chorioretinal folds [1-10]. Posterior scleritis in children is rarer than in the adult population [4]. Fundus fluorescein angiography and optic coherence tomography features have been reported previously. Although the diagnosis can be mistaken with some other diseases, ultrasonographic evaluation helps establishing a definite diagnosis. The most common ultrasound feature is high reflective scleral wall thickening. Increased posterior scleral wall thickness more than 2 mm has been reported in more than half of the patients [3]. The other feature is the “T” sign. The “T” sign is seen due to the presence of fluid in the sub-Tenon’s space. The “T” sign was reported in 25% to 41.2% of the cases. Choroidal effusion, localized serous retinal detachment, posterior coats flattening and swelling of the optic disc can also be seen in ultrasonographic imaging [3-6].Posterior scleritis is an inflammation of the sclera posterior to the ora serrata and it is usually under-recognized [3]. No systemic abnormality was detected in our case. However, his parents were given an advice to close observation probably for systemic involvement or recurrence in the future. Posterior scleritis can be misdiagnosed if a comprehensive approach with multimodal imaging is not performed. Our case did not have anterior scleritis findings, which was reported one quarter to 3 quarter of the patients . Initially, the only clinical finding observed was retinal striae in the posterior pole. There were no inflammatory signs in anterior or posterior segments. Approximately 25% of the patients have some degree of anterior chamber reaction, and 10% have vitreous cells [3].Posterior scleritis is generally autoimmune in origin and it is associated with systemic disease in up to 40% of the patients [7]. The clinical picture resolved completely within one month after the beginning of systemic corticosteroid and topical NSAID. Systemic corticosteroid was added to treatment because the majority of cases reported in literature required it for resolution . Recurrence was not observed during the 12-month follow-up. However, it has been reported that the majority of recurrences occurred between month 6 and 18 after discontinuation of the therapy [7-10]. Immunosuppressive therapy may be needed in severe cases associated with systemic diseases and unresponsive to standard treatment. Our case also showed that the immediate diagnosis might be crucial in some patients, because the worsening of the clinical picture and the visual acuity could occur if treatment is delayed even for a few days. Serous retinal detachment has been reported as the most frequent clinical sign in both posterior scleritis and Vogt-Koyanagi-Harada disease. Serous retinal detachment seen on OCT, choroidal thickening by EDI-OCT and fluorescein angiographic features implied a diagnosis of Harada disease, but unilateral involvement, absence of cells in the anterior chamber or vitreous and convex elliptic elevation of RPE contour due to compression of choroidal thickness resulting from scleral swelling contrary to diffuse thickening of choroid in Harada disease strongly suggested posterior scleritis. The symptom of periocular pain and ultrasonographic features confirmed the diagnosis. The pain probably results from tendonitis and stretching of Tenon’s capsule by edema into Tenon’s space or stretching or destruction of the nerves that pass through the sclera [Increasing awareness of clinical and multimodal imaging features in the course of posterior scleritis could lead to a correct diagnosis with a timely treatment.


Penile metastases are very rare and arise most frequently from genitourinary cancers. Penile metastases from rectal adenocarcinoma are less common.We report the case of a 47-year-old North Afican man with penile metastases from a rectal adenocarcinoma, which was discovered 4 months after abdominoperineal resection. A penile biopsy was carried out and established the metastatic nature. He underwent palliative chemotherapy treatment. He was still alive 4 months after diagnosis of penile metastases.The prognosis of metastasis to the penis is very poor; the best results have been achieved with surgery but only for lesions where metastasis is limited to the penis. Despite its rich vascularization and the extensive circulatory communication between the neighboring organs, metastatic involvement of the penis is relatively infrequent . Most ofThe prognosis of such metastasis is very poor regardless of the treatment options. Treatment is more often palliative than curative .In this case report, we describe a case of penile metastasis secondary to a rectal adenocarcinoma.A 47-year-old North Afican man presented with bleeding per rectum and tenesmus of several months’ duration. A rectal examination revealed a mass close to his anal sphincter. Colonoscopy showed a large, ulcerated and multilobulated mass less than 1 cm beyond the anal verge. Multiple biopsies of the rectal mass were obtained. The specimens were sent for pathological examination and returned with findings of moderately differentiated adenocarcinoma.A computed tomography (CT) scan of his abdomen revealed multiple lymph nodes of the mesorectum measuring less than 5 mm but otherwise no overt metastatic disease.He received radiochemotherapy then underwent an abdominoperineal resection; there was no clinical or radiological evidence of distant metastasis at the time of resection.A histopathological examination revealed a moderately differentiated adenocarcinoma of the rectum without lymph node metastasis; the disease was staged as T3 N0 M0. Our patient underwent adjuvant chemotherapy.He remained well until 4 months later, when he presented with bowel occlusion and urogenital complaints.A physical examination showed a nodule of the corpus cavernosum without visible skin lesions Fig. .Fig. 1PhA CT scan was performed that showed: lung metastases; external iliac lymph node metastases; lombo-aortic, celio-mesenteric, and inguinal lymph nodes; penile metastases; and bone metastases Fig. .Fig. 2CoHe received corticotherapy for the bowel occlusion caused by peritoneal carcinomatosis with good evolution then he underwent palliative chemotherapy soon after a biopsy of his penis confirmed the metastatic nature of the lesion. He was still alive 4 months after diagnosis of penile metastases.Penile metastases are very rare despite the rich vascularization of the penis and its extensive circulatory intercommunications with neighboring organs.Metastatic involvement of the penis despite its proximity to the rectum mainly originates from the bladder and prostate. Regional lymph nodes, the liver, lungs, and the vertebral column are most likely to be involved in metastasis from rectal cancer .Retrograde venous transportation suggested the main pathways to penis metastasis. The corpus cavernosum is the most common site of penile metastasis. The glans penis and corpus spongiosum are rarely involved , 5, 6.The most common presenting symptoms in order are perineal pain, induration, urethral obstruction, priapism, and hematuria. Metastases can present as well as plaques, wart-like nodules, ulceration, erythema, or induration of the penis , 8.Penile metastasis from rectal adenocarcinoma usually occurs within 2 years after diagnosis of the primary tumor and is frequently associated with dissemination to multiple organs , 7–9, foPenile metastases are unlikely to be solitary, and treatment should focus on palliative control, which can be achieved by radiation therapy, systemic chemotherapy, or, in selected refractory cases, surgery , 7.However, regardless of the treatment option chosen, prognosis in the setting of penile metastases remains poor. Reported survival usually varies from 7 months to 2 years but some long-term survivals have been seen after aggressive surgical treatment (penile amputation) with the best results noted in patients where penile metastasis was the only site of recurrence , 7–11. PThis report is an additional new case of primary rectal adenocarcinoma with penile metastasis which is rare and usually arises from genitourinary cancers. Despite the advances in cancer therapy within the last decade, the prognosis is still extremely poor. The best results are achieved with surgery when metastasis is limited to the penis.


The trigeminocardiac reflex (TCR) is a brainstem reflex that may be observed in anaesthesia during surgical procedures stimulating the intracranial or peripheral portion of the trigeminal nerve. The peripheral TCR is divided into the oculocardiac reflex and the maxillomandibulocardiac reflex based on the affected sensory branches of the trigeminal nerve. In veterinary medicine the oculocardiac reflex has been described, however the maxillomandibulocardiac reflex has never been reported.A 5-year-old male Epagneul Breton was presented for surgical management of an upper lip mass. During surgery, a sudden severe bradycardia and a decrease in systemic arterial blood pressure developed. The occurrence of a maxillomandibulocardiac reflex was suspected on the basis of the temporary link between surgical stimulation and haemodynamic changes. Three doses of atropine were given before starting a dopamine infusion due to lack of response. The dopamine infusion normalized heart rhythm and blood pressure. The dog recovered uneventfully and he was discharged 24 h later with a sinus rhythm and no sign of recurrence of arrhythmias.The TCR is a rare but potentially life-threatening complication of procedures involving the sensory areas innervated by the three branches of the trigeminal nerve and it may cause bradycardia with hypotension. The use of a β1-adrenergic receptor agonist such as dopamine may be indicated in cases of a refractory response to the conventional treatment with atropine. The trigeminocardiac reflex (TCR) is a brainstem reflex defined as a sudden decrease of at least 20% in heart rate and/or systemic arterial blood pressure during stimulation of any of the sensory branches of the trigeminal nerve . The TCRIn human anaesthesia, TCR is observed frequently during surgical procedures involving the base of the skull, and cases of TCR are reported during procedures to correct strabismus in children or basal cell carcinoma removal from the skin over the zygomatic arch –8. The iThe only type of TCR reported in veterinary clinical practice is the oculocardiac reflex although the real medical relevance of this condition has been questioned , 9. Two We describe a case report of a suspect of maxillomandibulocardiac reflex and its management in an Epagneul Breton undergoing surgical excision of a labial mass.A 5-year-old, male, Epagneul Breton, weighing 15 kg, was admitted for examination and management of a mass located on the internal surface of the upper lip detected by the owner 2 weeks before.Dirofilaria immitis) prophylaxis. No past history of syncope or exercise intolerance were reported by the owner. Rectal temperature was 38.2 °C, heart rate 96 beats/min and respiratory rate 24 breaths/min, mucous membranes were pink and the capillary refill time was 2 s. Thorax auscultation was unremarkable, heart sounds were rhythmic and normal with no audible murmur. A soft tissue mass was palpated on the internal surface of the upper lip, symmetrically developed and straddling the labial frenulum  scan was scheduled to detect any contraindication to palliative surgery.I’O2 = 0.5) administered through a 10 mm ID silicone endotracheal tube.A 20 gauge intravenous (IV) catheter was placed in the right cephalic vein and an infusion of lactated Ringer’s solution 5 mL/kg/h was started. Dexmedetomidine  2 µg/kg and methadone  0.12 mg/kg were administered intravenously as pre-anaesthetic medication. General anaesthesia was induced with propofol  2.5 mg/kg and maintained with sevoflurane  in a mixture of oxygen/air  was applied to maintain normocapnia  and an IV infusion of ketamine  10 µg/kg/min was started with a loading dose of 0.5 mg/kg. At the beginning of the surgery, a heart rate (HR) between 85 and 95 beats/min was recorded with a sinus rhythm. Anaesthesia was uneventful until 90 min after induction, then the HR progressively decreased from 82 to 29 beats/min within 5 min , and high resting vagal tone . No clinTo prevent or minimize the occurrence of the TCR during surgery in human medicine, the use of loco-regional anaesthesia has been proposed . BecauseIn conclusion the occurrence of a maxillomandibulocardiac reflex (a subtype of TCR) although rare may be a potential life-threatening complication of procedures involving the sensory areas innervated by the maxillary and mandibular branches of the trigeminal nerve and it may cause bradycardia with hypotension. As in human medicine, parasympatheticolytic agents like atropine may not reverse the cardiovascular abnormalities, as these are due not only to an over activation of the vagus but also to a sympathetic depression. In case of refractory response to the conventional treatment, the use of a β1 adrenoceptor agonist may be indicated.


The paraumbilical hernia sac often contains the omentum, the small bowel, and less commonly the colon. The herniation of the pancreas through a paraumbilical hernia is extremely rare and has been reported only by two cases in the literature; moreover, acute pancreatitis secondary to this condition is a particularly unusual event.We present a very unusual case of a 67-year-old female patient with a voluminous paraumbilical hernia containing the pancreas, complicated by acute pancreatitis. Laboratory data revealed an elevation of the pancreatic enzymes. An intravenous contrast-enhanced computed tomography (CT) scan of the abdomen demonstrated a large hernia sac containing multiple viscera, including the pancreas. The patient underwent emergency laparotomy with a diagnosis of intestinal obstruction.The clinicians should consider this rare condition in the differential diagnosis of patients presenting with large paraumbilical hernias associated with classical symptoms of acute pancreatitis, particularly in the absence of typical risk factors for pancreatitis. An intravenous contrast-enhanced abdominal CT scan should be performed immediately in these patients. We recommend the patients and the surgeons to consider prompt surgical repair for paraumbilical hernias to avoid further complications and the higher incidence of morbidity and mortality associated with emergency surgeries. •Paraumbilical hernia sac often contains omentum, small bowel, and seldom colon.•Herniation of pancreas through a paraumbilical hernia is an extremely rare event.•Pancreatitis linked to paraumbilical hernias is associated with high mortality.•Prompt surgical repair of umbilical hernias in high risk patients is recommended. The cHerniation of the pancreas through a paraumbilical hernia is extremely rare, with only two cases previously reported in the literature ,5. Moreo22. The patient was eventually transferred from ER to our surgical unit for further investigations.A 67-year-old, Caucasian, multiparous, pensioner woman walked into the emergency room (ER) with a 3-day history of severe epigastric pain radiating to the back, nausea, vomiting and a 5-year history of a large non-reducible paraumbilical hernia superior to the umbilicus. The hernia had gradually increased in size without causing obstructive symptoms; the reason why the patient had not sought prior medical assistance. She had no significant past medical and surgical history and was not on any medication. She denied drug allergies, tobacco, alcohol, and substance use. Her body mass index (BMI) was 24 kg/mOn initial assessment, she appeared hypotensive, tachycardic, and tachypnoeic. On physical examination, a massive circumferential, tender, irreducible supraumbilical mass (transverse diameter of about 30 cm) with ulcerated areas of overlying skin was noted .Fig. 1Ma3/mL, C-reactive protein 1809 nmol/L, pancreatic amylase 24150 nmol/s•L, serum lipase 47900 nmol/s•L, blood urea nitrogen 34 mmol/L, and creatinine 0.16 mmol/L. Other labs included normal liver function tests and normal bilirubin levels. The urinary catheter and nasogastric tube were placed. Urine output was poor and the nasogastric tube drained around 1500 ml of bilio-enteric material. An urgent intravenous (IV) contrast-enhanced computed tomography (CT) scan of the thorax and abdomen was performed. CT scan of the thorax demonstrated mild bilateral pleural effusion and marked distension of the oesophageal lumen with air fluid levels. Abdominal CT revealed a voluminous left paramedian supraumbilical hernia protruding through an abdominal wall defect of about 6,5 cm. Hernia sac contained loops of the small bowel and their mesentery, as well as the segments of ascending, transverse and descending colon. Additionally, the antro-pyloric portion of the stomach and the first portion of the duodenum with air-fluid levels were herniated through the sac, pulling with them a portion of the pancreas. The body and tail of the pancreas appeared oedematous with peri-pancreatic fluid collection and mesenteric stranding as a sign of glandular sufferance , the patient was transferred to the operating theatre on the same day with a diagnosis of intestinal obstruction in order to undergo an emergency laparotomy. The patient was placed in a supine position and general anaesthesia was induced. The surgery was performed by senior general surgeon G.L., experienced in emergency surgery and abdominal wall hernia repair. Following a midline incision, the voluminous hernia sac was identified, isolated, and opened .Four possible mechanisms have been thought to be responsible for a hernia-associated pancreatitis: (1) the recurrent parenchymal trauma due to repetitive trans-hernial sliding of the organ; (2) ischemic insult as a result of intermittent stretching and traction of the vascular pedicle due to repetitive migration of the pancreas into the hernia sac; (3) volvulus or intermittent folding of the pancreatic duct leading to obstruction of pancreatic secretions, intraductal hypertension, and the resultant inflammation; and 4) Anoxic injury induced by Incarceration of the pancreas  Anoxic i,10.nil per os, IV antibiotic therapy, and analgesic control) is preferred, however, severe cases unresponsive to medical therapy, or cases involving perforation, or incarceration such as ours, require immediate surgical management [Due to the paucity of reported cases and experience, the optimal management of acute pancreatitis secondary to pancreatic herniation is not well established. Although due to the elevated risks, avoidance of an urgent surgical approach and a primary conservative management : N/A.Iman Komaei


Ischemic colitis occurs as a result of the decrease of arterial blood supply at the level of the colon and may have consequences which vary from completely reversible lesions to ischemic necrosis. It is a condition that affects both sexes, predominantly people over the age of 50, having multiple etiologies in which the main role is played by atherosclerosis. The symptomatology varies from mild symptoms, which are reversible, to severe ones, with a vital risk. The colonoscopy does not show pathognomonic signs, but it has high sensitivity due to the involvement of the mucosa. There are different treatments according to the clinical form. In general, it goes into remission spontaneously. Emergency surgical treatment is required in acute pancolitis, toxic megacolon, massive hemorrhages, when a subtotal colectomy is performed. It represents an anatomical and pathologic spectrum, which depends on the onset, duration, cause and localization of the segment involved, on its length and on the existence of collateral circulation ;2. atherosclerosis which causes embolism or thrombosis in the superior and inferior mesenteric artery [3. hematologic diseases – coagulopathies caused by AT III deficiency, Protein C, S, deficiency, Factor V Leiden, antiphospholipid syndrome, polycythemia vera [4. vasculitis in Takayasu’s disease, giant cell arteritis, periarteritis nodosa, Churg–Strauss syndrome, thrombotic thrombocytopenic purpura [5. vasculopathies in diabetes and amyloidosis;6. parasitic or viral bacterial infections;7. acute pancreatitis;8. pheochromocytoma;9. obstructive colonic lesions which produce secondary distension in colorectal cancer, strangulated hernia, diverticulitis [Iatrogenic causes:1. Medical causes: following a treatment with flutamide, immunosuppressants, AINS, saline laxatives, gold salts, diuretic medications, digitalis, penicillin, following a colonoscopy and an aortography [2. Surgical causes: insertion of various vascular prostheses, liver transplantation, colonic by-pass [Tollefson Classification: according to the severity of the disease and to the anatomical and pathologic lesions, three stages may be described:•Stage I – in which there are edema, hyperemia and hemorrhages localized at the level of the mucosal and submucosal region, with or without ulcerations. The lesions are limited, with a favorable evolution towards healing.•Stage II - the muscularis is involved, with necrosis at this level and the formation of fibrous tissue, leading to strictures. The lessions have a mean severity [•Stage III – necrosis develops on the entire colonic wall, which leads to severe metabolic disturbances, with a severe prognosis. Mortality is high and can reach 60-70%.Clinical Diagnosis – the symptomatology varies from mild symptoms, which are reversible, to severe ones, with a vital risk. The patients have difficult bowel movement with bloody diarrhea and abdominal pain of high and moderate intensity [The physical examination reveals abdominal distension, meteorism, tenderness at palpation, a palpable abdominal mass in the left iliac fossa due to the edema [- laboratory test results indicate leukocytosis.- The abdominal X-ray on an empty stomach may show hydro air images, the segmental dilatation of the involved area, the presence of air at the level of the portal vein or the mesenteric vein, which represents a sign of severity, the imprinting in the involved colonic segments due to the intramucosal hemorrhages [- The irrigography shows defects of nodular filling, edema of the mucosa, dehaustration of the colon, segmental stenoses, with supraadjacent dilatations [- The colonoscopy does not have pathognomonic signs, but it has high sensitivity due to the involvement of the mucosa. Ischemia also induces secondary inflammatory lesions, so that the mucosa looks erythematous and edematous, with disseminated ulcerations. Black and bluish areas may appear which are secondary to the necrosis of the mucosa. Solitary colonic ulcer appears in 75% of the cases. After 48 hours from the onset, the mucosa becomes necrotic and it is hard to differentiate from Crohn’s disease or from pseudomembranous colitis. A characteristic of ischemic colitis is the non-involvement of the rectum, the segmental distribution of the lesions, the clear-cut delimitation of the involved lesions, the frequently favorable evolution towards healing [- The findings of computed tomography are non-specific, they show the dilatation of the colon, with the segmental thickening of the colonic wall, the irregular narrowing of the lumen as a result of the edema, ascites, the presence of air in the portal vein or in the colonic wall;- The ultrasound examination reveals a moderate thickening of the colonic wall, with the loss of the differentiation between its layers, and the absence of the arterial flow is a sign of severe prognosis [- The mesenteric angiography is not a common investigation and it is performed only in the cases of acute ischemia;- Computerized tomographic angiography detects the changes of the blood flow and of the secondary ischemic lesions.The anatomical pathologic examination – is not pathognomonic, it shows the presence of macrophages loaded with hemosiderin [The differential diagnosis is made between acute colitis caused by dysentery, the inflammatory diseases of the colon, diverticulitis [prognosis is favorable, in 50% of the cases it remits spontaneously within 48 hours, with full recovery within 14 days. There are severe cases with fever, shivering, bloody diarrhea which require an emergency laparatomy [Most frequently, the – may be local with perforations, bleeding, abscesses, peritonitis [– systemic with heart failure, kidney failure, disseminated intravascular, coagulation, acidosis [Treatment differs according to the clinical form. In general, it goes into remission spontaneously. The patients in a good state require feeding pause. The undernourished patients are administered parenteral alimentation, and those with severe forms are prescribed a feeding pause, hydroelectrolytic corrections, broad-spectrum antibiotics, transfusions in severe anemias, nasograstric tubes.Analgesics, antispasmodic drugs and antidiarrheal medication are not used because they may lead to toxic megacolon. Corticotherapy predisposes to perforation of the colon and worsens ischemic lesions [The emergency surgical treatment is required in acute pancolitis, toxic megacolon, massive hemorrhages, when a subtotal colectomy is performed. The surgical treatment may be temporized in the forms which evolve with segmental involvement, when the symptoms persist for more than 3 weeks, or in the cases of apparent healing with sepsis, when a segmental colectomy is performed, with a colostomy with the secondary repair of the continuity of the colon due to the increased risk of dehiscence of the primary anastomosis. In the case of colonic strictures, a segmental resection or an endoscopic dilation is performed [It is a relatively recent entity, well individualized from the clinical and therapeutical points of view, as it is the most frequent symptomatic ischemia, with an excellent prognosis in most of cases, whereas the surgical treatment is reserved only to the severe cases with complications.


Amputation neuroma is difficult to diagnose preoperatively. Amputation neuroma arising from a remnant cystic duct after cholecystectomy is rare. Herein, we present a case of amputation neuroma derived from a remnant cystic duct along with a review of the literature.A 60-year-old woman visited our hospital due to a tumor located in the hepatoduodenal ligament. A gallbladder adenoma was resected by open cholecystectomy 30 years prior. Endoscopic ultrasonography demonstrated branched intraductal papillary mucinous neoplasm of the pancreas and a tumor with a low-echoic pattern in the extrahepatic biliary system. Enhanced computed tomography revealed a 6-mm tumor in the artery phase. Surrounding lymph nodes were not swollen. Magnetic resonance cholangiopancreatography showed that the tumor presented with slightly high intensity on T2 weighted imaging. Operative findings revealed that the whitish nodule was moderately attached to surrounding tissues. The remnant cystic duct and the tumor could not be separated; however, no direct invasion toward common bile duct was observed. Rapid intraoperative pathological examination demonstrated that the tumor was a neuroma. The peration time was 251 min and blood loss was 80 ml. The patient was discharged nine days after surgery with no postoperative complications.It is difficult to distinguish amputation neuroma from malignant tumors because radiological findings of a neuroma mimic findings of malignancy. Intraoperative diagnosis is necessary to select an appropriate surgical procedure due to the difficulty of preoperative diagnosis. ANs are characterized by irregular growth of regenerated nerve bundle and fibrosis. ANs are non-neoplastic disorganized growths . ANs for2A 60-year-old woman visited our hospital for evaluation of a tumor arising in a remnant cystic duct 30 years after cholecystectomy for gallbladder adenoma. Laboratory data, including tumor markers such as carcinoembryonic antigen and carbohydrate antigen 19-9, were within normal ranges. The patient had no chief complaint. Previous medical history included breast cancer that was completely resected three years prior to her visit. Since that time she had taken an aromatase inhibitor. Annual follow-up for breast cancer by contrasted computed tomography (CT) showed an intraductal papillary mucinous neoplasm (IPMN) in the pancreas head and an enhanced tumor image around the hepatoduodenal ligament . Endosco3ANs are traumatic neuromas that result from incomplete healing following trauma to the nervous system and often develop in limbs and neck ,5. ANs tAs a result of widespread use of laparoscopic cholecystectomy (LC), reports of AN after LC have been increasing ,7. The pRadiological features of ANs in magnetic resonance imaging studies reveal tumors with intermediate signal intensity on T1WI and high intensity on T2WI . IncreasSurgical resection would be the recommended approach for diagnosis and cure. In a typical case of AN, it is relatively easy to discount a malignant tumor without immunostaining , so avoiThe biliary tract is richly innervated by the hepatic plexus, and ANs may occur after any operation that causes inflammation to hilar lesion. At this time, biliary stricture (BS) by AN after orthotropic liver transplantation (OLT) is the postoperative complication of greatest concern. Technical issues and ischemic-type lesions are considered as the leading causes of BS after OLT. AN is another cause of BS after OLT. The incidence of ANs after liver transplantation may be 0.5% to 30% ,19. Wide4It is difficult to distinguish ANs from malignant tumors using various radiological methods. POCS and IDUS could be helpful in making a precise diagnosis. A surgical approach is acceptable in the case of biliary ANs for both diagnosis and cure, considering the potential for biliary malignancy.None of the authors have any commercial or financial involvement in connection with this study that represents or appears to represent any conflicts of interestThis research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.Onomichi general hospital ethics review committee, OJH-2019-03Written informed consent was obtained from the patient for publication of this case report and any accompanying images.All authors in this manuscript contributed to the interpretation of data, and drafting and writing of this manuscript. Ryosuke Hirohata is first author of this paper. Tomoyuki Abe is corresponding author of this paper. Hironobu Amano, Masahiro Nakahara, Toshio Noriyuki, and Hideki Ohdan conceived and designed the study and drafted the manuscript. Ryosuke Hirohata first diagnosed. Ryosuke Hirohata, Akinori Shimizu, Keiji Hanada, and Masahiro Nakahara were engaged in patient’s care in our hospital including surgery. Hideki Ohdan and Tsuyoshi Kobayashi contributed to study concept, and review of the final manuscript and submission of the paper. All the authors read and approved the final manuscript.The manuscript does not report the result of an experimental investigation or research on human subjects.Tomoyuki Abe.Not commissioned, externally peer-reviewed.


Gallbladder carcinoma (GBC) is a relatively rare and aggressive malignant tumor with a poor prognosis. A systematic review of current clinical studies illustrates an extreme paucity of second-line therapeutic options following the failure of standard-of-care cisplatin-gemcitabine chemotherapy. The efficacy of apatinib, an highly potent and selective oral inhibitor of VEGFR-2 tyrosine kinase, for refractory advanced GBC has not yet been clarified. Herein, we report a case of advanced GBC that presented a durable partial response to apatinib used as monotherapy after the failure of multiline chemotherapies including S-1 monotherapy, capecitabine monotherapy, gemcitabine plus capecitabine, and irinotecan plus oxaliplatin. The patient achieved an efficacy of partial response within 2 months. By September 23, 2019, the duration of treatment had extended for almost 1 year with a satisfactory quality of life, and the administration of apatinib was continued. Dose reduction of apatinib occurred at week four due to grade 2 hypertension and hand-foot skin reaction (HFSR). No fatigue, proteinuria, mucositis, or thrombocytopenia occurred. To the best of our knowledge, this is the first case of a successful use of apatinib monotherapy for heavily pretreated GBC. Further prospective studies are warranted to confirm the efficacy and safety of apatinib in GBC. Gallbladder carcinomas (GBC), relatively rare malignancies arising from epithelial cells of the gallbladder, account for 80–95% of all biliary tract cancers (BTC) and mainly present as mucin-producing adenocarcinomas (90% of patients) . SurgeryTargeting the vascular endothelial growth factor (VEGF) pathway is a consolidated strategy in many cancer treatments. Apatinib is a small-molecule VEGF receptor 2 (VEGFR-2) tyrosinase inhibitor that has been demonstrated to have both an inhibitive effect on cell growth and to be anti-apoptotic.Apatinib has been clinically proven to be safe and effective in treating advanced gastric cancer that failed to be treated with at least two lines of previous systemic therapy . In addiHerein, we report a case of advanced GBC metastasized to the liver and lungs that presented a durable partial response (PR) to apatinib as monotherapy after the failure of multiline chemotherapies.In January 2013, a 56-year-old female complained of persistent pain in the upper abdomen and back; this followed a reported 3 months of poor appetite, weight loss, and jaundice. T2-weighted magnetic resonance imaging (MRI) of the upper abdomen revealed gallbladder lesions. A surgical resection of the gallbladder was conducted on February 28, 2013, after which her jaundice was resolved. Histologically, the lesion was composed of intraductal papillary neoplasms with high-grade intraepithelial neoplasia together with some complex fusion and focal carcinoma. With regards to radical surgery, a margin negative resection status (R0-status) was reached and no positive lymph node was found. Thus, the patient was diagnosed with GBC at pT1aN0M0, Stage IA. No further chemotherapy or radiotherapy was given after surgery. On November 23, 2015, an MRI displayed a large mass occupying the gallbladder area and several soft tissue nodules in the lower segment of the common bile duct. The patient then received a palliative operation on December 17, 2015, including a total pancreectomy, splenectomy, subtotal gastrectomy, and partial hepatectomy. The pathology was confirmed as moderately differentiated adenocarcinoma. The preoperative serum CA19-9 was 731.30 U/mL  on November 24, 2015 , and theAfter seven cycles of the last chemotherapy regimen, the enhanced total abdominal CT scan on September 1, 2018, revealed a 7.8 × 6.9 cm mass located in the original gallbladder area (or the anastomotic stoma), accompanied by abdominal and retroperitoneal lymph node metastasis . MeanwhiGBC, a malignancy usually associated with a history of gallstones, obesity, multiparity, and chronic infections , is the In the last decade, progress has been made to identify the dose and scheduling of a combination of gemcitabine and cisplatin recommended for first-line standard treatment . The addhttps://www.asco.org/) and the National Institutes of Health trial registry . Relevant studies were searched for using relevant medical terms for subject headings  and abstracts . The cut-off date for studies to be considered was August 24, 2019.Angiogenesis is one of the critical steps in tumor growth and hematogenous metastasis, and it plays a significant role in providing oxygen, nutrients, and growth factors to the tumor. The VEGFR2/RAF/MEK/ERK signaling pathway is involved in the process of angiogenesis and this pathway is frequently studied in a wide number of carcinomas, including GBC. In the last few years, some studies have indicated that VEGF-A is highly expressed in GBC, and that angiogentic microvessel density and the high expression of VEGF-A are correlated with poor prognosis of GBC, which suggests that GBC is actually angiogenesis-dependent –22. A liRegorafenib, sunitinib, and bevacizumab are currently the most frequently studied new-generation drugs that inhibit this specific signaling. Regorafenib is a multitargeted kinase inhibitor that inhibits VEGF signaling. A phase II trial  tested rApatinib is highly potent and a selective inhibitor targeting VEGFR-2 tyrosine kinase, and it is taken orally. With a high binding affinity to VEGFR2, apatinib significantly inhibits the migration and proliferation of endothelial cells, decreases tumor microvascular density, and promotes apoptosis. Based on the beneficial results from a randomized, double-blind, placebo-controlled phase III trial comparing apatinib and a placebo, apatinib was recommended as the third line to treat advanced gastric adenocarcinoma and gastroesophageal junction adenocarcinoma by the Food and Drug Administration of China in 2015. Until now, there has been only one retrospective analysis with a small sample size (21 patients were enrolled) that has evaluated the efficacy and safety of apatinib in advanced BTC . In thisIn this case report, the patient was treated with apatinib monotherapy, and PR was observed within 2 months. Meanwhile, there was obvious cavity formation in the lung metastasis, which was consistent with the characteristics of the effect of anti-vascular targeted drugs . The effIn summary, we, for the first time, reported a successful case of apatinib with acceptable toxicity monotherapy in refractory advanced GBC after the failure of multiline chemotherapies. Targeting the VEGFR signaling pathway may be a beneficial strategy for GBC in the near future. Further prospective studies are warranted to confirm the efficacy and safety of anti-vascular therapy in GBC.All datasets generated for this study are included in the article/supplementary material.The studies involving human participants were reviewed and approved by West China Hospital of Sichuan University Biomedical Research Ethics Committee. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.MZ performed the data acquisition. MZ and KZ performed the data analysis and interpretation. PZ performed the radiological analysis of MRI and CT images. MZ and QL performed the manuscript preparation.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Duodenum inversum (DI), also known as inverted duodenum or duodenum reflexum, is a congenital malformation in which the third portion of the duodenum, instead of continuing leftward to the ligament of Treitz, reverses direction and travels in a superior, posterior track prior to crossing the midline above the pancreas. We present a case of a 62-year-old woman presenting with chronic nausea and vomiting, subsequently found to have DI. Duodenum inversum (DI) was first described in 1940 by Feldman and Morrison who described 14 such cases in 20,000 gastrointestinal X-ray examinations, with an incidence of 0.07 per cent [A 62-year-old female with a medical history of generalized anxiety disorder and hyperlipidemia presented to our clinic for further evaluation of chronic nausea and chronic intermittent abdominal pain ongoing for over 10 years. She also reported occasional nonbloody, nonbilious emesis along with the nausea which was not exacerbated by oral intake. Her symptoms were refractory to oral Ondansetron, Metoclopramide, and Promethazine. She denied bloating, weight loss, or changes in bowel habits. Her past surgical history only included an uncomplicated laparoscopic cholecystectomy.On physical exam her abdomen was soft and nontender with normoactive bowel sounds. Laboratory study results showed a hemoglobin level of 12.2 g/ml , total bilirubin level of 0.8 mg/dL , alkaline phosphatase of 74 U/L , aspartate aminotransferase (AST), and alanine aminotransferase (ALT) levels of 32 and 41 U/L, respectively . A random cortisol level was 12 mcg/dl.She was initially sent for a CT angiogram of the abdomen with intravenous contrast which did not show any radiographic evidence of median arcuate syndrome. She then underwent a diagnostic esophagogastroduodenoscopy which revealed a normal duodenum . An uppe stasis in the duodenum; and (3) rapid passage of the contrast medium through the third stage. While not associated with significant morbidity or mortality, DI remains an important diagnostic consideration as it can present with proximal gastrointestinal obstruction [Duodenum inversum is thought to develop due to persistence of the dorsal mesentery with a mobile duodenum , 6. Othetruction .Medical management includes acid suppression due to risk of duodenitis and remains the mainstay of treatment for patients without complications such as bowel obstruction , 6. Fail


Primary intraosseous cavernous hemangiomas are rare skull lesions that are not typically known to involve the orbital bones or the dura. We describe a rare case of a fronto-orbital bone cavernous hemangioma with extension into the dura.A 68-year-old female presented with a one-year history of diplopia with discomfort around her left orbit. Magnetic resonance images demonstrated a mass in the left frontal skull extending into the orbital rim. The patient underwent a craniotomy for tumor resection. Dural invasion was found intraoperatively. Gross total resection and reconstruction were achieved. On the postoperative follow-up, the patient was asymptomatic. Primary calvarial intraosseous cavernous hemangiomas are most commonly located in the frontal and parietal bones. These lesions typically involve only the outer table of the skull. In lesions involving the orbit and dura, excision with cranioplasty can provide symptomatic relief with good cosmetic outcomes.  Primary intraosseous cavernous hemangiomas are rare, benign vascular lesions, accounting for 0.2% of bone tumors and 7% of skull tumors . AlthougIntraosseous hemangiomas of the skull tend to involve the outer table of the skull and spare the inner table and dura matter. Intradural extension of a cavernous hemangioma has only been reported once in the literature . A few cWe present a unique and rare case of a fronto-orbital bone cavernous hemangioma with an extension of the hemangioma beyond the inner table and with the involvement of the dura.A 68-year-old woman presented with a one-year history of diplopia associated with discomfort and pressure around her left orbit and forehead. Physical exam demonstrated proptosis with a 4 mm downward displacement of the left globe. The left inferior orbital rim was not readily palpable. The remainder of the exam was normal. Magnetic resonance imaging (MRI) demonstrated a 5.7 cm x 3.9 cm x 4.4 cm mass in the left frontal skull extending into the left orbital rim and into the left orbit with a displacement of the superior rectus muscle and downward displacement of the globe Figure . The masThe patient underwent a left orbito-zygomatic (OZ) craniotomy for resection of the involved bone and subsequent reconstruction of the orbital walls. The patient was placed in a Mayfield headrest and stereotactic registration was accomplished. A standard OZ approach was performed. After drilling the first access burr hole, it was noted that the dura was not preserved and could not be separated from the inner table. Two additional burr holes and cuts involving the zygoma and orbit were made for access. A side cutting drill was used to turn a bone flap but this could not be mobilized freely. An osteotome had to be used to remove the outer table with some part of the middle table. Further drilling of the remaining hemangioma was performed with a small cutting burr, followed by a diamond burr under the microscope. The bony tumor was found to extend both extra- and intradurally. Drilling of the abnormal bone was continued medially to the edge of the frontal sinus. The dura was preserved when feasible, but significant areas of dura were not preserved. The dural defect was closed with a suturable dural substitute (Durerepair®)  and some tacking at the bony edges where a watertight closure was not feasible. Upon complete resection, a piece of porous polyethylene was cut and contoured (Medpore®)  to recreate the superior orbital rim and the roof of the orbit. The porous polyethylene was fixated laterally to the zygoma and medially to the frontal bone with standard cranial screws. The closure was done using the remainder of the bone flap, which was attached with standard screws and plates  after drilling the involved portion. A postoperative CT scan showed complete resection of the hemangioma Figure .Gross examination of the left frontal bone specimens sent during surgery demonstrated red-white bony tissue Figure . MicroscThe postoperative MRI demonstrated complete resection of the lesion Figure . The patHemangiomas are normally benign, asymptomatic tumors of blood vessels. They most commonly occur in females with a 3:2 ratio. The mean age of presentation is 46 years , 4-6. Three histological subtypes of hemangiomas (based on the size of vascular channels) have been described: cavernous, capillary, and mixed. Vessels of cavernous hemangiomas are large and closely clustered, in contrast to vessels of the capillary subtype, which are thin-walled and separated by normal bone tissue -11. MostIntraosseous hemangiomas of the orbital bones are exceedingly uncommon. A total of 51 cases have been reported in the literature, and they are usually confined to the orbit , 7. TheyIntracranial involvement of an orbital hemangioma has been reported in five other cases to date , 7. The Cavernous hemangiomas of the calvarium usually originate from vessels in the diploic space with the erosion of the outer table and little to no involvement of the inner table . InvolveThe classic characteristic appearance of the hemangioma on CT shows a “honeycomb” or “sunburst” pattern with fine, radiating, and reticulated lines or trabeculae . MRI of Treatment depends on the presenting signs and symptoms of the lesion or on its behavior on sequential imaging. Primary intraosseous hemangiomas typically are asymptomatic and follow a benign course. Surgery is reserved for patients with mass effect, as was the case for our patient. Other indications are hemorrhage or cosmetic concerns . SurgicaWe present an unusual case of an orbital intraosseous cavernous hemangioma located at the junction to the inferior frontal bone. The patient was found to have some involvement of the inner table, as well as the dura. A skull base approach, combined with cranioplasty, resulted in resolution of symptoms with a good cosmetic outcome.


Clear cell carcinoma (CCC) is a well-known aggressive histological type of carcinoma, predominantly seen in ovary and endometrium. However, CCC arising in abdominal wall is a very rare event. We report a case of a 48-year-old woman with an abdominal wall mass at her cesarean section (c-section) scar, which increased in size and became painful in the last months. Radiology revealed a 7 cm mass in the right inferior rectus muscle sheath, suggestive of endometriosis. An irregular, firm mass was resected, densely adherent to the rectus muscle and pubic bone. Frozen section revealed a multicystic lesion with minimal cytologic atypia, and a benign cystic neoplasm was favored. However, permanent sections showed marked nuclear atypia, hobnail morphology, and areas of infiltrative growth within fibrous stroma. No benign endometrial glands were found, although fibrosis and hemorrhage were present. Napsin-A, racemase, and PAX-8 were positive, consistent with CCC, likely arising within a c-section endometriosis focus. Although CCC usually presents with moderate to marked nuclear atypia, it can be mild and, especially in cases with a predominant cystic pattern, create diagnostic difficulties. An endometriosis-associated malignancy should be considered in the differential with any enlarging nodule or increasing pain within an abdominal wall scar. Clear cell carcinoma (CCC) of the female genital tract can arise in the ovary, endometrium, and cervicovaginal region, as well as in peritoneal and other extrapelvic sites. However, CCC arising within an abdominal wall scar is rare, and when it is found, it is most commonly related to underlying endometriosis. Although the pathophysiology is controversial, it usually affects women that underwent operations such as hysterectomy or c-section, with a probable explanation for this occurrence being malignant transformation of an endometriosis focus within the abdominal scar . MalignaHere we report a case of CCC arising from an abdominal wall c-section scar, with a discussion of diagnostic criteria and potential diagnostic difficulties. We also present a review of the available literature relative to this unusual event.This is a 48-year-old woman with no previous cancer history, who presented with complaint of pain at the inferior edge of her midline c-section scar. Surgical and obstetric histories were significant for three c-sections, excision of endometriosis from c-section scar (20 years ago), and hysterectomy and bilateral salpingo-oophorectomy for leiomyomata and endometriosis of left fallopian tube (4 years ago). No positive family history of cancer was reported. Since her hysterectomy, the patient reported decreased sex drive, dyspareunia, weight gain, vaginal dryness, and abdominal pain. She was taking oral estradiol but admits being noncompliant. Eventually, she was switched to vaginal estradiol (10 mcg). No cancer markers were performed. The patient underwent imaging analysis and surgical excision of the mass as described below. She was scheduled for adjuvant chemotherapy with Paclitaxel and Carboplatin; however, she has been lost to follow-up.Computed tomography scan (CT-scan) revealed a 7.0 × 5.0 × 3.3 cm heterogeneous collection at the right inferior rectus sheath and concluded as most likely a scar endometriosis . The patA follow-up PET imaging done after surgery revealed residual uptake in the anterior right abdominal wall, most likely postsurgical effect; however, residual disease could not be completely excluded.The patient underwent exploratory laparotomy, and an irregular, cystic, firm mass was identified, located deep to the rectus muscle in the midline. Dense adhesions and scarring were noted between the mass, rectus muscles, and pubic bone. The mass was excised and submitted for intraoperative consultation. The specimen was received fragmented, consisting of four irregular shaped soft tissue fragments, the largest measuring 5.0 × 3.0 × 2.0 cm and the smallest 2.0 × 0.7 × 1.2 cm. This tissue was firm and hemorrhagic, composed of similarly sized, microcystic spaces. The other pieces consisted of fibroadipose tissue.Microscopic examination of one representative section submitted for frozen section showed a multicystic lesion with large cystic spaces lined by a single layer of flattened to cuboidal epithelium with bland hyperchromatic nuclei and occasional enlarged nuclei Figures . A “beniThe specimen was entirely submitted for permanent sections. Additional sections demonstrated again a predominantly cystic pattern and some tubules, lined by highly atypical cells admixed with more bland-appearing ones, and areas of infiltrative growth within a fibrous desmoplastic stroma. The malignant cells showed clear to eosinophilic cytoplasm, enlarged, hyperchromatic nuclei, prominent nucleoli, and hobnail morphology in areas Figures . MitoticImmunohistochemical stains were performed . NeoplasScar endometriosis has been known at least since 1942 . AlthougMicroscopically, CCC can have a variety of architectural patterns within the same tumor, including cystic, tubulocystic, solid, and papillary growth with characteristic hyalinized fibrovascular cores . CCC is The clinical differential diagnosis of palpable abdominal wall masses includes benign entities such as hernia, hematoma, abscess, desmoid tumors, and other soft tissue neoplasms. If a malignant neoplasm is found in a patient with a history of gynecologic or obstetric surgery, the possibility of a primary malignancy arising from endometriosis should be considered, although cutaneous metastases from a Mullerian or nongynecological primary should be ruled out .Tumors with a predominant cystic or tubulocystic pattern, lined by flat to cuboidal tumor cells with mild nuclear atypia, may cause diagnostic difficulties, in particular with small biopsies and frozen sections , 14. DurProper diagnosis should also take advantage of immunohistochemical panels as needed. Immunostains can support a gynecological origin versus other tumors with clear cells, such as renal cell carcinoma (RCC) or adrenocortical carcinoma. Napsin A, racemase, and hepatocyte nuclear factor (HNF-1b) strong immunopositivity will support a diagnosis of CCC , whereasTo the best of our knowledge, 29 cases of CCC arising in abdominal wall endometriosis have been reported in the literature between 1986 and 2017 . The aveThe diagnosis of malignant transformation of abdominal wall endometriosis is still a challenge for clinicians. There are no characteristic symptoms or markers of malignant transformation, and imaging may only show endometrioma with fast growth. Similarly, imaging in our case suggested a scar endometriosis, and no malignant transformation was suspected before surgery. Given the increased rate of c-sections registered in the last years, we may expect a parallel increase of endometriosis implants in the c-section scar and, therefore, occurrence of CCC of the abdominal wall should be kept in mind .CCC arising from abdominal wall is a very rare event. However, it should be considered in the differential diagnosis of any patient who presents with an enlarging abdominal wall mass or increasing pain within a surgical scar. Although CCC is a high-grade malignancy, the degree of atypia may vary; it can be mild and, especially in cases with a predominantly cystic pattern, can cause diagnostic difficulties.


Abdominal non-tuberculous mycobacterial infection is a rare condition in healthy patients. When it occurs, it leads to the appearance of typical findings of peritoneal involvement, such as thickening of the peritoneal leaflets and the omentum, ascites and enlargement of lymph nodes and of mesenteric nodules. These findings may be misdiagnosed as tumour peritoneal implants.In case of spontaneous regression of the peritoneal involvement and ascites, as well as in the absence of malignancy, the suspicion of infectious disease, including abdominal nontuberculous mycobacterial infection, should be considered. Peritoneal carcinomatosis is a condition usually associated with cancer originating from the ovary, stomach, colon, pancreas or breast. In the presence of a previous and/or simultaneous cancer, the appearance of peritoneal solid nodules usually raises the suspicion of peritoneal carcinomatosis.However, some non-oncologic pathologies may exhibit peritoneal nodules, and may be therefore misdiagnosed as peritoneal carcinomatosis.Non-tuberculous mycobacteria (NTM) are aerobic, non-motile and acid-fast organisms. These bacteria are widespread in the environment, especially in tap water, shower heads, soil, dust and food products, and are carried by domestic and wild animals , 2. NTM Risk factors for NTM include a wide range of well-known predisposing conditions , while other risk factors remain poorly understood.In immunocompromised patients, typical manifestations include lymphadenopathy, fever and weight loss. In healthy patients, clinical manifestations are unremarkable and sometimes may be completely asymptomatic.Abdominal infection by NTM is a rare manifestation of the disseminated disease and may be associated with peritonitis with a high mortality rate .Nontuberculous peritonitis presents with thickening of the peritoneal leaves and the omentum, ascites, enlargement of low-attenuation lymph nodes and of mesenteric nodules . These fThis report describes the rare case of an asymptomatic and immunocompetent patient with abdominal NTM infection mimicking peritoneal carcinomatosis.A 53-year-old Caucasian woman was referred to our institution because of the suspicion of peritoneal carcinomatosis, raised by the findings of ascites at a transvaginal ultrasound performed as a yearly routine exam; a pre-surgical staging exam with computed tomography (CT) scan show thickening of the gastric walls, multiple omental nodules and ascites .Her previous personal history was unremarkable and she denied any clinical symptom or cancer history.At our hospital, she underwent an esophagogastroduodenoscopy and colonoscopy, with results negative for gastric/colon cancer.Her comprehensive metabolic profile revealed mild liver dysfunction with an alanine transaminase of 77 U/L and aspartate transaminase of 71 U/L. When tumour markers were assessed, CA125 demonstrated increased levels of 290 U/mL , whereas carcinoembryonic antigen, CA 19.9 and other immunohistochemical markers were within the normal ranges. Serological assessment of HIV, hepatitis C virus and hepatitis B virus were negative.Ten days later, the patient underwent an ultrasound-guided biopsy  with a dAfter 2 weeks, the patient received a CT scan of the thorax (to complete pre-operative staging), demonstrating a spontaneous (with no therapy) dimensional and numerical reduction of peritoneal lesions in the upper abdomen, partially included in the chest CT scan, as well as resolution of peri-hepatic and peri-splenic ascites .Since there was no evidence of primary cancer at pre-operative examinations and the second CT scan revealed a partial resolution of peritoneal implants and ascites without therapy, the suspicion of an infectious disease was raised.The pathological evaluation of the biopsies performed on the omentum and peritoneum revealed the presence of lymphoid aggregates with a central core of epithelioid cells with large eosinophilic cytoplasm, without atypia or mitosis  and withBecause of the uncertain result of the biopsy and the conflicting results of the 2 CT scans, in order to rule out malignancy with certainty, the patient underwent a laparoscopic surgery in 2 weeks.Definitive histological diagnosis excluded the presence of malignant cells and reported a necrotising inflammation caused by non-tuberculous mycobacteria. Hence, the patient was sent to a hospital with expertise in infectious diseases.A follow-up CT scan performed 1 year later confirmed a complete recovery of peritoneal findings.Peritoneal carcinomatosis is the intraperitoneal dissemination of any tumour that does not originate from the peritoneum itself, and it represents the most common diffuse peritoneal disease. Therefore, in cases of peritoneal solid nodules, the first diagnosis is usually peritoneal carcinomatosis. Nevertheless, there are quite a number of differential diagnoses to be kept in mind when facing peritoneal disease, such as peritoneal lymphomatosis, malignant peritoneal mesothelioma, pseudomyxoma peritonei and peritoneal infections .Among peritoneal infections, NTM is an uncommon disease, but its diagnosis is mandatory because the treatment is mainly medical and should be initiated as soon as possible .The recognition of patients at high risk for this disease is difficult. While some conditions are recognised risk factors, others are unknown. It is impossible to predict which patient will develop this disease because all people are exposed to NTM on a daily basis.Frequently, this condition is misdiagnosed because the clinical symptoms and physical findings are non-specific, and the final diagnosis is based on histology.Previous studies have demonstrated that the most common signs are fever, ascites and abdominal masses , 8, althThe reported CT findings, which are not more specific after the application of post-processing optimised reconstruction algorithms , 10, incMycobacterium Tuberculosis, C. Albicans and Aspergillus [Based on the location and the morphology of the lesions, CT imaging can mimic many conditions, including: other infections, such as from ergillus ; malignaergillus ; and perergillus .Because of the unspecific signs and symptoms, the diagnosis of abdominal NTM infection may be delayed and the lack of appropriate treatment may lead to a high rate of mortality.The standard treatment for this disease consists of a combination of second line anti-tuberculosis drugs  only aftIn the case reported, the first CT scan showed ascites, multiple enhancing peritoneal nodules, well depicted in greater omentum, hepatic capsule and subphrenic space, suggesting the presence of peritoneal carcinomatosis. The second CT scan showed spontaneous resolution of ascites, as well as reduction of number and size of peritoneal lesions. The changes of the lesions over time without treatment raised the suspicion of something different from malignant peritoneal carcinomatosis.In order to rule out malignancy, a laparoscopy was performed, and the final histological diagnosis confirmed the suspicion of an infectious disease sustained by NTM.In conclusion, this report describes a rare case of an asymptomatic and immunocompetent patient with abdominal nontuberculous mycobacterial infection mimicking malignant peritoneal carcinomatosis.Since the symptoms and CT findings are non-specific, the diagnosis may be difficult. After excluding the presence of a previous or a simultaneous tumour, an infectious aetiology should be suspected, in order to send the patient for the right treatment in a timely manner.Hence, abdominal NTM infection should be taken into account in the presence of peritoneal nodules suspected for malignant carcinomatosis, because the treatments are different and a prompt correct diagnosis may be crucial for prognosis.The authors declare that they have no conflicts of interest.No funding was provided to the authors for this work.


The combination of intrapelvic and extrapelvic endometriosis is a very rare condition in gynecology. Patients with endometriosis involving the sciatic nerve are easily misdiagnosed because they usually present with atypical symptoms of endometriosis. Here, we present a rare case of an endometrioma passing through the left greater sciatic foramen. Removal of the endometriotic lesion was performed with a concomitant laparoscopic and transgluteal approach through the cooperation of gynecologists and orthopedic (neuro)surgeons.A 20-year-old woman presented with complaints of severe dysmenorrhea lasting for more than 6 years and dysfunction of her left lower limb lasting for approximately 4 months. Both CT and MRI demonstrated a suspected intrapelvic and extrapelvic endometriotic cyst (7.3 cm × 8.1 cm × 6.5 cm) passing through the left greater sciatic foramen. Laparoscopic exploration showed a cyst full of dark fluid occupying the left obturator fossa and extending outside the pelvis. A novel combination of transgluteal laparoscopy was performed for complete resection of the cyst and decompression of the sciatic nerve. Postoperative pathology confirmed the diagnosis of endometriosis. Long-term follow-up observation showed persistent pain relief and lower limb function recovery in the patient.When a woman complains of unexplained unilateral sciatica, especially a woman suffering from dysmenorrhea, endometriosis of the sciatica nerve should be considered as a potential etiology. Complete excision of the endometriotic lesion and adequate neurolysis (or decompression) of the sciatic nerve through the multidisciplinary cooperation of experienced gynecologists with proper training in laparoscopic pelvic (neuro)surgery and orthopedic (neuro)surgeons is effective. Endometriosis is classically defined as the presence of endometrial glands and stroma outside the uterine cavity . IntrapeMoreover, extension of endometriosis from within the pelvis to outside the pelvis is very rare. Here, the authors describe the diagnosis and treatment of a 20-year-old woman with an endometriotic cyst exiting the pelvis through the left greater sciatic foramen. More importantly, the surgical team in this case developed a novel technique combining a transabdominal and transgluteal approach to completely excise the lesion. Postoperatively the patient recovered without sequelae.The study received approval from the Ethics Committee of The First Affiliated Hospital, Sun Yat-Sen University. Informed consent was obtained from the patient.A 20-year-old woman (G0P0) presented with severe dysmenorrhea lasting for more than 6 years. In the beginning, the pain started from the first day of her menstruation and disappeared after menstruation ended. One year before presentation, it became increasingly intense, lasting longer than before (more than 1 week) and mainly limited to the left lower abdomen. Four months before presentation, she complained of occasional pain and numbness in the left lower limb, especially the popliteal fossa and the sacroiliac joint. However, the pain was not associated with her menstruation. Then, she visited orthopedic surgeons and underwent computed tomography (CT) and magnetic resonance imaging (MRI) of the pelvis as well as the buttocks. CT showed uterus didelphis as well as a cyst in the left buttock (7.3 cm × 8.1 cm × 6.5 cm). MRI confirmed that there was a cyst full of fluid located in the pelvis and extending into the left buttock through the greater sciatic foramen  to alleviate her pain. The adduction function of her left hip joint was severely affected, and she could not even lift her left limb. Bimanual pelvic exploration revealed a mass in the left pelvis, with tenderness on compression of the left buttock. Transvaginal ultrasound at our hospital showed uterus didelphis, with a hematometra in the left uterus. A cyst with a homogenous dark area of liquid (9.3 cm × 6.6 cm) and a clear margin was also detected in the left pelvis. Ultrasound examination also showed that she had only one kidney. The serum level of CA125 was 87.20 U/mL.A clinically suspected diagnosis of an endometriotic cyst passing through the left greater sciatic foramen was established. A surgical team composed of gynecologists, orthopedic surgeons and microsurgical experts discussed different treatment strategies for the case in detail. Finally, a novel minimally invasive technique combining transabdominal and transgluteal laparoscopy to remove the cyst was proposed. This tentative surgical plan with a transgluteal open approach as an alternative was also accepted by the patient.Laparoscopic surgery was performed by the gynecologists first. Didelphis uterus was found, with the left adnexa attached to the left uterus, and the right adnexa attached to the right uterus Fig. . The lefThe patient was then placed in a ventral decubitus position, to continue the transgluteal laparoscopic surgery. The reflection point of the left sciatic nerve on the skin of the left buttock was located first. Three apexes of a triangle around this point were selected for the insertion of laparoscopic trocars Fig.  a and b.Obvious pain relief in the left lower limb was achieved 3 weeks after the operation. The movement disorder of the left lower limb completely recovered after 6 months, with continuous regular rehabilitation exercises. The patient became pregnant (right uterus) successfully right after 6 months of GnRHa treatment postoperatively and gave birth to a healthy baby. The 5-year follow-up examination showed that she continued to be asymptomatic without pain and with normal lower limb function.Endometriosis is a rare cause of functional disturbance of the lower limb(s). Endometriosis infiltrating the sciatic nerve often causes cyclical sciatica. Since Schlicke reported the first case of endometriosis-induced sciatica in 1946 , more thWomen with symptoms of typical sciatica and/or of unilateral lower limb dysfunction usually visit the orthopedic clinic first. However, when women complain of dysmenorrhea simultaneously, especially when the sciatica is also cyclical and occurs during menstrual periods, orthopedic surgeons should consider endometriosis involving the sciatic nerve. In our case, the pain the patient experienced was occasional at the very beginning of the formation of the endometriotic cyst. When cyclical bleeding of the endometrial tissue inside the cyst occurred and the fluid accumulated gradually, the cyst became increasingly larger, compressing the sciatic nerve progressively. As a result, the pain became increasingly intense, with a pain-free interval that gradually shortened until, after a few months, it became persistent. Fortunately, she was introduced to our department promptly once the imaging examinations revealed a cyst passing through the greater sciatic foramen. More importantly, proper treatment was performed, avoiding permanent damage to the left sciatic nerve.The pathophysiology of endometriosis is still poorly understood but appears to be multifactorial. The “pocket-sign” theory was proposed and has been accepted by some scholars based on Sampson’s retrograde theory , 5. It iVascular damage (such as minor injury or surgery) could lead to hematologic migration of endometriosis, especially in patients without other sites of endometriosis. Menstrual blood reflux is the incentive of endometriosis. The retrograde endometrial debris penetrates neighboring tissues and organs every month, where it triggers locally inflammatory and immunological reaction. Patient with sciatic nerve endometriosis usually suffer from periodic sciatica, sometimes beginning 1 to 2 days before or after the first day of a period. The pain is intense and has a pain-free interval in the beginning. But, as time goes on, it can be progressive and becomes permanent finally.Clearly, endometriosis afflicts women in different ways in terms of the type and severity of symptoms, how badly the pelvis is affected, and the effect on health and quality of life. Therefore, treatment must be individually selected for each woman, taking other factors such as age, response to previous treatment, surgical complication rates, and the desire for pregnancy into account. In this case, achieving complete removal of the endometriotic cyst and relief of compression of the left sciatic nerve were of equal importance. Both the gynecologists and orthopedic surgeons thought that surgical removal of the cyst was necessary, but selection of the surgical approach seemed to be a difficult challenge. Laparoscopic resection of the intrapelvic part of the cyst and neurolysis of the sciatic nerve could be achieved by gynecologists who were well-trained and familiar with pelvic (neuro)anatomy . The traWhen a woman complains of unexplained unilateral sciatica, especially a woman also suffering from dysmenorrhea, endometriosis of the sciatica nerve should be considered a potential etiology. Patients are commonly misdiagnosed because they are often sent to orthopedic surgeons, neurologists or neurosurgeons. CT, MRI and transvaginal/transrectal ultrasound are important for the diagnosis. Complete excision of the endometriotic lesion and adequate neurolysis (or decompression) of the sciatic nerve are extremely important. The multidisciplinary cooperation of experienced gynecologists possessing proper training in laparoscopic pelvic (neuro)surgery and orthopedic (neuro)surgeons should gain interest within the medical field as this condition affects more patients than we realize.


Spontaneous remission (SR) of cancer is a very rare phenomenon of unknown mechanism. In particular, SR of non-small cell lung cancer (NSCLC) has been scarcely reported. We present the case of a 74-year-old woman with advanced, poorly differentiated NSCLC (highly expressing programmed death ligand-1 [PD-L1]) that progressed despite multiple lines of chemotherapy but then spontaneously remitted.The patient presented with hemoptysis and was diagnosed with stage IIIA poorly differentiated NSCLC via bronchoscopic biopsy. She had an unremarkable medical history and moderate performance status. The initial treatment plan was surgery after neoadjuvant chemotherapy. Despite conventional chemotherapy, follow-up chest computed tomography (CT) showed gradual tumor progression and she decided against further treatment after fifth-line chemotherapy. However, the size of lung mass was markedly decreased on follow-up chest CT one year after ceasing chemotherapy. Also, follow-up positron emission tomography images showed decreased metabolic activity in the lung mass and a percutaneous biopsy specimen from the diminished lung mass revealed no viable tumor cells. A diagnosis of SR of NSCLC was confirmed, and the patient was without tumor progression on follow-up nine months later. Later, PD-L1 immunostaining revealed high positivity (> 99%) in initial tumor cells.Our case showing SR of poorly advanced NSCLC refractory to multiple lines of chemotherapy suggested the association between immunity and tumor regression. Spontaneous remission (SR) of cancer is defined as partial or complete disappearance of a malignant tumor without any standard treatment or with inadequate treatment for cancer control. Cole and Everson first reported 47 cases of biopsy-confirmed SR of cancer, proposing the term and criteria for SR in 1956 , SR of n. Initial chest x-ray and computed tomography (CT) scan revealed a 5.1 × 2.9 cm lung mass in the left upper lobe with a surrounding halo of ground-glass attenuation Diagnostic bronchoscopy revealed a polypoid mass with luminal obstruction of the left lingular inferior segmental bronchus Fig. -a. BroncAlthough tumor size decreased after 4 cycles of chemotherapy, she decided to stop chemotherapy due to poor general condition and drug side effects and was scheduled for regular follow-up for tumor surveillance.After brain magnetic resonance imaging for cancer staging, which revealed a non-metastatic lesion, a tentative diagnosis of lung cancer with T3N1M0 (stage IIIA according to TNM seventh edition) was proposed. At first, the patient planned to undergo surgery after two cycles of paclitaxel plus carboplatin as neoadjuvant chemotherapy, inducing a decrease in the size of the lung mass from 5.1 × 2.9 cm to 4.1 × 1.5 cm. However, the patient developed drug-induced hepatitis with aspartate aminotransferase (AST) / alanine aminotransferase (ALT) of 110 / 182 IU/L due to a self-prescribed herbal medication, and the surgery schedule was delayed. For two weeks, waiting for the AST / ALT levels to drop, the lung mass slightly increased (4.1 × 2.6 cm), and she hesitated to undergo surgery. So afterwards she received additional two cycles of paclitaxel plus carboplatin, which means that she received a total of four cycles of paclitaxel plus carboplatin. However, the tumor still progressed, and fifth-line chemotherapy was sequentially administered until the fifth-line as follows: second-line for 4 cycles of gemcitabine plus carboplatin, third-line for 2 cycles of pemetrexed, fourth-line for 4 cycles of weekly docetaxel, fifth-line for 1 cycle of weekly vinorelbine. After a vinorelbine monotherapy, the patient refused further chemotherapy due to general weakness. At 4 months after discontinuation of chemotherapy, the size of the tumor on follow-up chest x-ray  after the discontinuation of chemotherapy. Gladwish et al. reported the case of a patient with SR of stable IIB NSCLC after receiving an herbal remedy (essiac tea) [Orostachys japonicus is a flowering plant, containing several organic solvents including ethyl acetate with anti-cancer effects on human gastric cancer cells [Orostachys japonicus in enhancing immunity by increasing immune cell propagation and production of immunity-related cytokines [Orostachys japonicus in humans, SR in our cases might be influenced by Orostachys japonicus intake.Our patient was taking herbal medication (iac tea) , which siac tea) . Chung eiac tea) . Orostacer cells . An in vytokines . In our Because our patient had received multiple cycles of chemotherapy before the occurrence of SR, there was a possibility of pseudo-progression or delayed response to chemotherapy. However, the chemotherapy regimen had changed several times, and tumor regression was observed one year after the last treatment, suggesting a high probability of SR. In addition, all of the chemotherapeutic agents administered to our patient would be less likely to show pseudo-progression because they are conventional drugs rather than immune checkpoint inhibitors.In conclusion, we document a case of SR in a patient with advanced NSCLC refractory to conventional chemotherapy. Although the precise mechanism of SR in this case is unknown, the alteration of immunity might be an explanation. A single case cannot lead to a definite conclusion; nevertheless, our case indicates the importance of immunity in lung cancer control. Further well-designed animal model studies are needed to explore these findings.


The small intestine is a relatively privileged organ; primary tumors are uncommon among malignancies of the gastrointestinal tract, with an average annual incidence rate of 9.9 per million people. Among these uncommon tumors, duodenal tumors are even rarer with an average incidence rate of 0.4% only. Histopathological examination mostly shows villous type. Tubulovillous histology is extremely rare with an incidence of less than 1% of all duodenal tumors. These tumors are either found incidentally or during screening for familial disorders such as familial adenomatous polyposis or Gardner’s syndrome. Patients usually have symptoms such as diarrhea and abdominal pain along with weight loss over a period of months. Itching over the whole body and progressive yellow discoloration of sclera and skin (jaundice) can also be present when tumor compresses the common bile duct (CBD) and causes obstruction, especially in the region of ampulla of Vater. Examination of the abdomen may or may not reveal a mass lesion. Abdominal radiography and ultrasound show intra- and extra-hepatic biliary dilatation along with distention of CBD and growth at the lower end of CBD. Endoscopic retrograde cholangiopancreatography (ERCP) is, however, the investigation of choice as it not only allows direct visualization of a biliary tree but biopsy specimens can also be taken. Palliative procedures like placing a stent in the common bile duct to relieve obstruction can be done or a drain can be placed to drain bile. The definitive treatment is the Whipple procedure involving gastrojejunostomy, choledochojejunostomy, and cholecystectomy. Our case is unique as the patient had duodenal tubulovillous adenoma, a very rare tumor among the already uncommon duodenal tumors. The patient underwent Whipple procedure, T tube was placed, and feeding jejunostomy was done. An uneventful recovery occurred. Small intestinal primary tumors are quite rare with an incidence equal to one-tenth of the similar tumors of the colon . Less thA 64-year-old female presented to the outpatient clinic with a four-month history of painless progressive jaundice of skin and eyes along with loss of appetite and weight loss. On further inquiry, patient-reported one-month history of generalized itching was gradual in onset, intense at times, and mostly involved hands, forearms, and abdomen. There was also a history of pale stools and dark urine along with on and off melena. On review of systems, the patient denied abdominal pain, fever, epigastric discomfort, nausea, hematemesis, vomiting, constipation, or diarrhea. Family history was negative for any familial pathologies such as familial adenomatous polyposis or colorectal carcinoma in first degree relatives. Physical examination revealed a 4 x 2-cm reducible swelling in the epigastric region upon palpation. The abdomen was soft and non-tender otherwise.Laboratory investigations showed elevated total serum bilirubin level 17.5 mg/dl), alkaline phosphatase , and alanine aminotransferase . Other laboratory parameters including serum electrolytes, serum amylase, serum lipase, and coagulation profile were observed to be normal. Abdominal ultrasonogram showed dilated intra-hepatic biliary channels, distended gall bladder with the normal appearing pancreas. Common bile duct (CBD) was distended up to 2.1 cm with a 1.8 x 1.4 cm growth at its distal end  scan of the abdomen confirmed moderate dilatation of intra & extrahepatic biliary channels and dilated CBD up to its lower end Figure .Endoscopic retrograde cholangiopancreatography (ERCP) along with biopsy was scheduled and a double pigtail plastic stent (10 x 5 cm) was placed at the ampulla to achieve free drainage of the bile. Results of ERCP-guided biopsy showed papillary formation lined by cells showing mild atypia Figure .The diagnosis of tubulovillous adenoma of the duodenum was made, and Whipple procedure  was done along with T-tube placement and feeding jejunostomy Figure .No postoperative complications were observed and the patient was discharged on the eleventh postoperative day. T-tube was removed 10 days after discharge and a follow-up cholangiogram was done which came out to be normal.The small intestine is the longest part of the gastrointestinal tract, accounting for 75% of its length and 90% of the mucosal surface. Of all the gastrointestinal malignancies, benign tumors are approximately 3% to 6%, while malignant tumors are 1% to 3% . Over foAdenomas are commonly found tumors of the colon. However, they can also occur in the upper part of intestine especially in the duodenum with an overall incidence of 0.4% of the lesions found during endoscopic studies - 12. TheTubulovillous adenomas may include small, sessile, or polypoid lesions. They are discovered incidentally on endoscopy. Duodenal villous and tubulovillous tumors are often clinically silent . Though The treatment technique is usually decided on the basis of size, extent, and location of the tumor as well as its histopathology . These tTubulovillous adenomas of the duodenum are very rare with an incidence of 0.4% of all the small intestinal lesions. Although traditionally small bowel tumors are difficult to diagnose, however, a heightened index of suspicion and an increase in employment of recent diagnostic techniques such as upper gastrointestinal endoscopy and ERCP has increased the yield and decrease delays in identification. Due to the high recurrence rate of tubule-villous adenomas, a complete resection of the tumor is necessary and watchful follow-up is needed with immediate intervention in case of recurrence.


Mammary hamartoma is a rare benign breast tumor, composed of ducts, lobules, fibers, and adipose tissue. We describe a mammary hamartoma in a man; this is the fourth case being reported in the literature.A 30-year-old man presented with a 1-month history of a painless mass in his right breast.Ultrasound imaging and mammography revealed a lesion, approximately 2.0 cm × 2.0 cm in size, in the right breast, which was considered to be either a lipomyoma or an adenoma fibrosum.The mass was surgically resected. Pathological examination confirmed the diagnosis of mammary hamartoma.The patient was discharged from the hospital after surgery. There was no sign of reoccurrence during a 1-year follow-up period.At present, mammary hamartoma is considered to be a benign lesion, usually treated by surgical resection. Some reports have suggested a possible association between a hamartoma and the development of breast malignancy. The pathology and biology of an association between a mammary hamartoma and malignancy have not been defined to date. Therefore, the true incidence rate of hamartoma in men might be higher than is currently reported. Mammary hamartoma also presents with unique imaging features which can help clinicians in improving the preoperative diagnosis rate of mammary hamartoma and to distinguish malignant components. Herein, we report on a very rare case of mammary hamartoma in a male patient, focusing on the clinical and pathological characteristics for diagnosis and treatment.Mammary hamartoma in men is a rare disease that accounts for 0.12% to 0.24% of all breast tumors and <4.8% of all breast benign tumors.2We received approval from the Institutional Review Board of Jilin University First Hospital, Changchun, Jilin, China for the publication of this report; the patient also provided informed consent for the publication of this case report.A 30-year-old man accidentally discovered a painless, quail egg-sized, mass in the lateral upper quadrant of the right breast, next to the nipple in February 2018. The patient did not initially seek medical consultation. In March 2018, the patient detected a painless enlargement of the mass and sought medical consultation at his local hospital. A breast ultrasonography examination revealed a mass, 23.7 mm × 7.5 mm in diameter ; no alcohol consumption; no history of trauma to the region; no history of prior surgery or radiation exposure of the region; and no personal or significant family history of cancer. With no important history identified, the clinical diagnosis of a hamartoma was established. The physical examination revealed a soft, mobile, painless mass located in the right breast, measuring approximately 2 cm in diameter, with no abnormality identified in the left breast. On March 27, 2018, the patient underwent surgical resection of the mass at our hospital. Gross examination of the resected mass revealed an oval, well-defined, and encapsulated mass, with gray-yellow coloring and a smooth margin Fig. . The nod3 first proposed the name “mammary hamartoma” and defined it as a well-defined mass formed by abnormal cells intermixed with normal mammary tissue. Most patients with mammary hamartoma are women, typically between the age of 33.5 and 66.5 years, with an average mass size of 5.0 cm. To date, only 3 cases of mammary hamartoma in men have been published. The first case involved a 36-year-old man presenting with a slowly growing mass in his left breast, with no evidence of axillary lymphadenopathy. The diagnosis of myoid hamartoma was confirmed after pathological examination. More recently, a case of hamartoma was reported in a 13-year-old boy, in whom the tumor extended into the thoracic cavity. Malignancy was considered and the mass was surgically resected, including 2 adjacent ribs. The diagnosis of hamartoma was confirmed on pathological examination. There is no documented recurrence of the mass in this patient. Due to the unique features of hamartomas, and of hamartomas in men, in particular, there is currently no standard treatment.In 1971, Arrigoni et al The growth of most mammary hamartomas is relatively slow. Patients with small tumors usually have no apparent clinical symptoms, and the hamartoma is identified incidentally on physical examination. However, some hamartomas can increase significantly in size over a short time, most commonly in pregnant and lactating women, indicating that hormones and endocrine factors may play a role in the development of these tumors. In men, there is a need to specifically differentiate a mammary hamartoma from male mammary development.A hamartoma is usually a solitary, soft, round or oval, lesion, with an envelope, and is generally not tender. and endobronchial hamartoma on radiological examination, with the diagnosis confirmed on pathological examination. Therefore, with more evidence, imaging may serve as a useful diagnostic tool to avoid unnecessary surgery. Approximately 94% of mammary hamartomas present with specific characteristics on mammography, namely as a round or oval mass, with uneven central density, and a smooth edge, accompanied by a transparent ring .–17 Calcification within a hamartoma is relatively rare. When present, massive calcification is considered to be benign, while small sites of silty calcification are usually associated with malignancy. Besides, the presence of micro-calcification shadow, a burr sign and lobulated tumor within the fat halo, on mammography, is also indicative of a possible malignancy. On ultrasound imaging, a mammary hamartoma presents as an oval or quasi-circular mass, with clear boundaries, parallel to the epidermis; an uneven echo structure, and mixed hypoechoic and hyperechoic tissue areas, with the majority of lesions having an intact capsule. Elastography can improve the identification of a hamartoma, based on the measured hardness of adipose tissue, glands, and fibrous connective tissue, which can aid in defining the boundary of the tumor. A myohamartoma reveals specific features on high-density T1-weighted magnetic resonance images, with a low-density shadow on T2-weighted images, although the specificity of these image-based findings has not been fully evaluated.,19Mammary hamartomas have specific imaging characteristics, which can greatly aid with the preoperative diagnosis. Moreover, recent studies have also reported on the unique features of nasal hamartomaA mammary hamartoma is composed of the same elements as normal mammary tissue and, thus, is difficult for pathologists to diagnose. Lumpectomy provides a curative treatment. Maintaining the integrity of the capsule during resection could improve the rate of pathological diagnosis. For rare types of hamartomas, such as myohamartomas, immunohistochemistry plays an important role in the diagnosis, with positive expression of specific markers  being strongly associated with the presence of spindle cells, and expression of s-100 and cytokeratin being negatively associated. Although the tumor is histologically benign and often painless, a hamartoma may develop to be quite large if local excision is not performed in a timely manner. The development of lobular carcinoma and invasive carcinoma has also been reported.,22 However, it is unclear if malignancy associated with a hamartoma results from a transformation of hamartoma tissue or if the malignant cells extend from hamartoma tissue. Therefore, the relationship between a hamartoma and breast cancer needs further discussion.Malignancy originating from a hamartoma is extremely rare, but is possible as these tumors contain epithelial tissue. As an example is the case of a 70-year-old woman treated for a mammary hamartoma, who presented with mammogram changes around the mass 6 years later and was diagnosed with invasive ductal carcinoma.Mammary hamartoma in men is rare, with the underlying pathology and biology remaining to be clearly defined. In addition, as men typically pay less attention to palpable masses within the breast tissue, it is commonly noted that lesions may grow substantively before medical attention is sought. As such, hamartoma in men tend to be detected later than in women, requiring differentiation from a malignant lesion. Our case report adds to the body of knowledge on this rare condition, with the accumulation of case reports being important to improve the diagnostic criteria, based on non-invasive techniques, and to establish, in time, a standard of care for this condition.The authors are grateful to the surgical department of the First Hospital of Jilin University for providing the patient's data.Conceptualization: Song Dong.Data curation: Gu Lin.Formal analysis: Wu You.Investigation: Wang Zhen, Chengzhao Xu.Resources: Du Ye.Project administration: Song DongSoftware: Jinghui Hong.Supervision: Song Dong.Writing – original draft: Mengxin Li.Writing – review & editing: Mengxin Li.Mengxin Li orcid: 0000-0002-3071-1669.


Adrenal cavernous hemangiomas are very rare benign tumors that usually present as incidental findings on abdominal imaging. Preoperative differential diagnosis from other benign or malignant adrenal neoplasms may be challenging.A 70-year old man was referred for an 8-cm abdominal mass incidentally discovered on a contrast-enhanced computed tomography (CT) performed to investigate a pulmonary nodule. Biochemical tests ruled out any endocrine dysfunction and iodine 123 metaiodobenzylguanidine whole body scintiscan single-photon emission CT excluded a pheocromocitoma. Findings on magnetic resonance imaging were non-specific and the patient was elected for a left adrenalectomy. Histopathological diagnosis revealed a cavernous hemangioma. A portion of the resected tissue was tested for drug sensitivity to mitotane, doxorubicin, and sunitinib.Adrenal hemangioma is a rare disease but should be included in the differential diagnosis of adrenal tumors. The surgical resection is generally required to exclude malignant disease, resolve pressure-related symptoms, and prevent retroperitoneal hemorrhage. Although specific features in diagnostic imaging are often lacking, if the diagnosis is established preoperatively a laparoscopic adrenalectomy can be performed due to the benign nature of the lesion. Doxorubicin and sunitinib were both capable of reducing primary culture cell viability, this suggest that similar drugs may be useful in the medical treatment of adrenal hemangiomas. Adrenal cavernous hemangioma is a rare entity first described in the mid-1950 [We report the case of a non-functioning adrenal cavernous hemangioma incidentally discovered on a contrast-enhanced computed tomography (CT) and discuss the diagnostic work-up, surgical treatment, and post-operative findings including drug sensitivity testing.A 70-year-old man was referred to the S. Anna University Hospital in Ferrara  for a left upper quadrant abdominal mass incidentally discovered on a contrast-enhanced CT of the chest performed to investigate a 15-mm right pulmonary nodule.The patient was asymptomatic, his past medical history was positive for essential hypertension, and physical examination was unremarkable.CT scan showed a homogeneous 83-mm left adrenal lesion with an average density of 45 HU; rare peripheral dot-like calcifications were also observed Fig. . The rigBiochemical tests ruled out any endocrine dysfunction . A subsequent iodine 123 metaiodobenzylguanidine whole body scintiscan single-photon emission computed tomography-CT (I123-MIBG-SPECT-CT) ruled out the presence of a pheocromocitoma.Due to the non-specific radiological findings and the size of the lesion, a surgical resection was then elected to establish the final diagnosis. The patient underwent a left adrenalectomy trough a left subcostal incision. Intraoperatively, the mass appeared encapsulated and hypervascularised. No evidence of hepatic as well as other peritoneal lesions was present. The operation was straightforward and the postoperative course was uneventful, with the patient discharged home on postoperative day six.The pathological examination revealed a large lesion of 90 mm × 65 mm × 70 mm with spongy appearance due to large vascular spaces. Histologically, the lesion showed a conglomerate of widely open vascular lumina lined by endothelial cells and separated by thick nearly acellular fibrous septa Fig. . The finp < 0.05 vs. control), but not mitotane, was capable of reducing primary culture cell viability. Similarly, sunitinib significantly reduced cell viability both at 1 and at 10 μM .A portion of the tissues was obtained at time of surgery and a primary culture was obtained, as described previously . Cells wAt 53-month follow-up the patient is doing well and has no evidence of recurrence. However, he underwent a laparoscopic prosthetic repair of incisional, umbilical and left inguinal hernias 20 months following the adrenalectomy.Adrenal cavernous hemangiomas are rare benign tumors arising from endothelial cells lining the blood vessels that are usually discovered incidentally on abdominal imaging. They are unilateral in most cases, only one bilateral case has been reported in the literature , generalAbdominal CT and MRI are valuable diagnostic tools for hemangiomas. On contrast-enhanced CT, the presence of both peripheral patchy and centripetal enhancements and highly dense peripheral rim are characteristic for adrenal hemangioma . HoweverSurgical resection is indicated for enlarging adrenal tumors or suspected carcinoma, regardless of its endocrine activity . LaparosIn conclusion, the case of an asymptomatic 8-cm cavernous hemangioma incidentally discovered and successfully treated by open adrenalectomy is presented. Adrenal hemangioma is a rare disease but should be included in the differential diagnosis of adrenal tumors. The surgical resection is generally required to exclude malignant disease, resolve pressure-related symptoms, and prevent retroperitoneal hemorrhage. Although specific features in diagnostic imaging are often lacking, if the diagnosis is established preoperatively a laparoscopic adrenalectomy may be planned due to the benign nature of such a lesion.