Diverticulosis of the appendix (DA) is rare and frequently found incidentally. Some cases are discovered after presenting with similar symptomatology to acute appendicitis, whereas other cases may be completely silent. Fibrous obliteration (FO) is a histologic finding indicative of cellular proliferation secondary to relapses of subclinical inflammatory processes. We report a case of a 75-year-old female with a history of chronic, intermittent abdominal pains who presented to the general surgery clinic after an abnormal thickening of the appendix was discovered on abdominal and pelvic computed tomography imaging. The patient underwent laparoscopic appendectomy for suspicion of malignancy. The histologic evaluation of the specimen demonstrated a diverticulum at the distal end of the appendix with FO of the lumen. We suspect the chronic nature of her disease course may have led to the FO of the diverticulum. An extensive literature search was performed, which revealed no other cases of FO of appendiceal diverticula. This may be the first case of diverticulosis of the appendix with FO in the English medical literature. If DA is discovered early with non-invasive imaging, surgical excision should be performed prophylactically as an association with an increased risk of perforation and neoplastic progression has been found. Diverticulosis of the appendix (DA), or appendiceal diverticular disease (ADD), is a rare occurrence with an incidence ranging from 0.004% to 2.1% . Since iDA can be completely asymptomatic or present similarly to acute appendicitis if the diverticulum becomes inflamed. Most cases of DA are discovered incidentally during post-operative histopathologic evaluation of the specimen or during intra-operative laparoscopy ,5,7. DiaFibrous obliteration (FO) is a proliferative lesion that was first described by Masson in 1928 ,13. He eHere, we report a case of an elderly woman who was incidentally found to have FO of an appendiceal diverticulum after laparoscopic appendectomy for a suspected mucocele neoplasm of the appendix. The patient was found to have a thickened appendix on abdominal and pelvic computed tomography (CT) imaging after several years of non-specific complaints of abdominal pain.A 75-year-old Caucasian female presented to her primary care provider’s (PCP’s) office with complaints of intermittent, mild episodes of non-specific abdominal pains for approximately five years. She also noted occasional post-prandial nausea with the consumption of solid and liquid food products and globus sensation, which she attributed to a history of sliding hiatal hernia. Her past medical history was also significant for gastroesophageal reflux disease, chronic obstructive pulmonary disease, and hypertension. Her surgical history included hysterectomy, cholecystectomy, and esophagogastroduodenoscopy (EGD), which revealed the sliding hiatal hernia four years ago. Her PCP ordered abdominal and pelvic CT imaging, which was significant for a thickened appendix with an approximated diameter of 11 mm Figures -1C. She On arrival at the general surgery clinic, the patient only noted symptoms relating to her hiatal hernia, such as occasional post-prandial nausea and vomiting, but denied current abdominal pain. She also denied fever, chills, loss of appetite, diarrhea, rectal bleeding, and melena. On physical examination, her abdomen was soft, non-distended, and non-tender. McBurney’s point and Rovsing’s sign were both negative. After reviewing the CT imaging, malignant processes, such as mucocele, could not be excluded, and laparoscopic appendectomy was recommended. After discussing options of observation versus surgical intervention, and the risks, benefits, and potential outcomes of each, she consented to the procedure. During the operation, the appendix was found to be grossly enlarged and pale in appearance. The gross specimen was measured at 68 mm in length and 13 mm in diameter. The serosal surface was pink-to-tan in color. Transverse sectioning revealed a grossly edematous mucosa that occluded the lumen. The distal tip of the appendix appeared expanded, giving a gross appearance most consistent with that of a diverticulum. Histologic evaluation of the specimen confirmed diverticulosis of the distal appendix with FO of the diverticulum Figures -2D. The DA is a rare occurrence and most frequently occurs as a pseudodiverticula derived from a weakening of the muscularis propria of the appendiceal wall . In contOur literature search was performed using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) 2020 guidelines with the assistance of Harzing's Publish or Perish® software  . The seaKalyneck described the first case of DA in 1893 . Since tA few studies have demonstrated an association between DA and neoplastic processes. Lim et al. performed a systematic review and meta-analysis, which revealed the prevalence of neoplasia in appendix specimens with DA to be 26.94%, compared to a 1.28% prevalence of neoplasia in specimens without DA . In the In conclusion, DA is a rare entity often discovered incidentally with an asymptomatic presentation or with symptomatology similar to acute appendicitis. Here, we presented the case of FO of a diverticulum of the appendix in a woman who had been experiencing intermittent episodes of subclinical abdominal pain for five years. We suspect the chronic nature of her disease course may have led to the FO of the diverticulum. This may be the first case of DA with FO in the English medical literature. Diagnosis of this entity prior to operative intervention is challenging as it may be completely silent. If DA is found early through the use of barium enema or CT imaging, surgical excision should be performed prophylactically as an association with an increased risk of perforation and neoplastic progression has been found previously.


Complete resolution of symptoms occurred with three cycles of PRRT.Neuroendocrine tumor (NET) is a rare tumor that has been observed in different sites such as lungs and throughout the gastrointestinal tract. Clinical features are usually non-specific and vary considerably depending upon the location of the tumor. Symptoms are similar to those of common conditions such as peptic ulcer disease, gastritis, irritable bowel syndrome, asthma, etc. Thus, an initial diagnosis of a NET usually occurs at an advanced stage. This report describes a case of pancreatic NET  with liver metastasis in a 37-year-old male which was found to be inoperable due to extensive direct involvement of the proximal jejunal branches and superior mesenteric vein. Peptide receptor radionuclide therapy (PRRT) with lutetium-177 dotatate ( Neuroendocrine tumors (NETs) are rare type of neoplasms derived from specialized cells known as enterochromaffin cells. They are most commonly found in the gastro-intestinal tract and lungs and can be benign or malignant . The mos2] presented with chronic diarrhea for one year. He complained of watery, non-bloody diarrhea which was sometimes associated with urgency. He reported 5–8 episodes of moderate-sized bowel movements per day without a nocturnal component. He denied blood in stool, abdominal pain, fever, or fecal incontinence. However, he had anorexia and weight loss for the past year. There was no complaint of nausea, vomiting, jaundice, dyspnea, or wheezing. He had taken antibiotics several times but didn’t have any improvement. His symptoms were not associated with any dietary triggers; however, he had been advised to eliminate milk products, caffeine, and gluten from his diet, which he did but with no improvement. Physical examination revealed normal thyroid and no hepatosplenomegaly. No abnormality was detected on systemic clinical examination.A 37-year-old man [body mass index (BMI) 22.7 kg/m5/cumm. Liver and renal function tests were unremarkable. Thyroid function test and blood glucose were within normal limits. Serum immunoglobulin A (IgA) anti-tissue transglutaminase and IgA anti endomysial antibodies were negative. Abdominal ultrasonography revealed a hypoechoic lesion in the liver with retroperitoneal lymphadenopathy. Triple phase contrast-enhanced computed tomography (CT) of abdomen revealed a well-defined mass lesion of size 40 mm × 47 mm × 48 mm in close relation to the head of pancreas and another well-defined lesion of size 48 mm × 33 mm in segment VII of the right lobe of liver with both lesions showing arterial phase enhancement  PET CT] showed active SSTR expressing lobulated, centrally necrotic mass lesion with intense DOTA-NOC avidity [standardized uptake value (SUV) max-38.2] involving the head of pancreas measuring approximately 43 mm × 50 mm × 48 mm, closely abutting the duodenum and inferior vena cava (The ena cava . Anotherena cava . Biopsy 177Lu-DOTATATE). The patient received three cycles of PRRT with 177Lu-DOTATATE. At 9 months follow-up, the patient reported complete resolution of symptoms with improved quality of life. Biochemical markers status also showed stable disease with serum chromogranin-A and neuron-specific enolase reported values of 97 ng/mL and 15.6 ng/mL respectively. The post-treatment 68Ga DOTA-NOC PET CT scan could not be performed as the patient had already incurred a huge financial burden and could not afford the costly imaging.On diagnostic laparoscopy, there was a single metastatic lesion in segment VII of the liver with the rest of the peritoneal cavity free of metastatic disease. There was a direct invasion of the proximal jejunal branches and superior mesenteric vein by the tumor. The procedure was converted to open laparotomy. Common hepatic lymph node dissection, hepatic metastasectomy, cholecystectomy, and kocherization of the duodenum were done. Jejunal mobilization was tried, but in view of direct extensive invasion by the tumor of proximal jejunal branches, the procedure couldn’t be proceeded and abandoned. Due to the inoperability of the primary tumor, the patient was treated with peptide receptor radionuclide therapy (PRRT) with lutetium-177 dotatate (PNETs comprise approximately 7% of all NETs and only 1–2% of all pancreatic tumors . There h177Lu-labelled somatostatin analogues. Studies with 177Lu-DOTATATE indicate that the therapy is safe, effective, has no serious side effects and the median progression-free survival is longer than 40 months [From a treatment perspective, surgery is the therapy of choice for functional tumors irrespective of the tumor size. The surgical therapy, whenever feasible offers the best option to cure the syndromes and improve the disease outcome . The Nat0 months . It also0 months . The assThis case report highlights the diagnostic and therapeutic challenges in PNETs. Due to the wide heterogeneity of the presentation of the disease, a high index of clinical suspicion is important for early diagnosis. Although surgery is the mainstay of treatment but is not feasible in advanced cases. The treatment of advanced NETs should be individualized based on the prognostic and predictive factors and the availability of novel treatment modalities. PRRT is an emerging treatment option in advanced and inoperable NETs improving patient outcomes and overall quality of life.


Sialolithiasis is a common condition characterized by the formation of salivary stones or calculi, also known as sialoliths, within a salivary gland or its duct. Usually, sialolithiasis presents as salivary gland swelling with intermittent pain around mealtime. All salivary glands can develop sialolithiasis, yet it occurs most commonly in the submandibular gland. In this report, we describe an unusual large sialolith measuring 1.7 cm and uncommonly resembling a canine tooth. The sialolith was removed surgically via intraoral approach, and the salivary secretion was restored. Sialolithiasis is a frequent salivary gland condition in which calcifications or calculi  form inside salivary glands or in their ductal system . It is eA 34-year-old male patient presented to our oral and maxillofacial surgery center complaining of swelling under his tongue Figure . DigitalThe overlying mucosa was stretched and shiny with reddish area on the superior aspect of the swelling. Salivary flow was negligible from the right submandibular duct orifice. On palpation, no regional lymphadenopathy was noticed. The presence of an unusually large sialolith in the region of the submandibular duct was confirmed by a topographic mandibular occlusal radiograph that showed a radiopaque mass resembling a canine tooth, anteriorly projected under the right inferior border of the mandible close to the genial tubercle Figure .The sialolith was removed surgically via intraoral approach under local anesthesia. The procedure consisted in enlarging the ductal orifice with an incision of 10 mm approximately after fixing the sialolith by a distal suture. A gentle pressure was then applied distally provoking the release of the sialolith through the incision. After that, an everted suture of the incised mucosa was performed, thus creating a salivary fistula. The sialolith removed was lightweight and presented many porosities. Its shape was unusually similar to a canine tooth and measured 1.7 cm in length Figure .No postoperative complications were noted. Salivary flow was restored after the removal of the sialolith.The submandibular gland is the second largest of the three major salivary glands, the others being the parotid and sublingual glands. It lies in the submandibular triangle and presents two lobes, superficial and deep, separated by the mylohyoid muscle . The subThe exact pathogenesis of sialolithiasis remains unclear. Some believe that sialoliths result from the deposition of mineral salts on nidi of bacteria, mucus, or desquamated cells ,13. The Sialoliths are yellowish, round or oval masses and can be rough or smooth . They usThe differential diagnosis of sialolithiasis includes oral bacterial infections, Sjögren's syndrome, sarcoidosis, chronic sclerosing sialadenitis (Kuttner's tumor), and neoplastic or non-neoplastic lesions. Appropriate diagnosis depends on a thorough medical history along with clinical and radiological evaluations . RadiogrSialography consists of radiography after injecting a contrast medium into the salivary duct. It helps visualizing the entire glandular system. This technique is contraindicated in patients allergic to contrast medium and suffering from acute infections. Moreover, sialography is not indicated in the case of sialolith suspected in the posterior segment of the duct which could be moved toward the body of the gland thus complicating its removal . ScintigThe most suitable treatment is selected according to the size and location of the sialolith . Small cIn this report, a sialolith of unusual size (1.7 cm long) and shape (resembling a canine tooth) was presented. Very few cases of sialolith mimicking a canine tooth have been reported in the literature. Additionally, sialoliths of this size are usually symptomatic and rarely located in the salivary duct; in our case, the sialolith was located in the anterior segment of the duct and was completely asymptomatic. A topographic mandibular occlusal radiograph was sufficient to confirm its presence, and the appropriate surgical treatment has restored salivary secretion.Sialolithiasis is one of the most common salivary gland pathologies, usually manifesting as swelling with intermittent pain around meals. Hence, painless sialoliths of unusual size and shape located in the submandibular duct are rare. Careful clinical and radiological assessments are crucial for establishing the appropriate diagnosis and treatment. The management of sialolithiasis is based on the size and location of the sialolith. While accessible unusually large calculi may be surgically removed, inaccessible ones require the removal of an entire lobe or the whole gland.


Uterine cervical Müllerian adenosarcoma (MA), a rare malignant tumor of the female reproductive organs, is composed of a benign epithelium and a low-grade malignant stromal component. Because few studies have investigated the clinical management of MA, misdiagnosis often occur. Therefore, we proposed an optimal course of clinical management for patients with MA. MA is possibly a malignant transformation of the cystadenofibroma. In this study, we present a case of a 46-year-old woman who presented with symptoms of MA of the uterine cervix, such as metrorrhagia and a cyst in the cervical canals, after transvaginal excision of the left ovarian mucinous cystadenofibroma. MA was first identified by Clement and Scully in 1974 . Contrar1.1We treated a 46-year-old married woman who presented with a cervical cyst that had been growing for 2 years with irregular vaginal bleeding for the past 6 months in our hospital. She underwent a transvaginal excision of the left ovarian cyst in 2005. The postoperative pathology results of the left ovarian tumor showed a mucinous cystadenofibroma. Subsequently, the patient underwent a laparoscopic excision of the left adnexal pseudocyst about 10 cm and pelvic adhesion lysis in 2007. The postoperative pathology results indicated left adnexa in the fibrous connective tissue.The examination results showed that the diameter of the mass in the cervix was 4 cm and an enlarged uterus, the size of 8-week’s gestation (8.9 × 4.4 × 3.4 cm). Ultrasonography revealed a cystic-solid mass in the cervix measuring 9.1 × 8.1 × 5.8 cm Figure 11.2The specimen of total laparoscopic hysterectomy with right salpingo-oophorectomy was received for histopathological examination. On gross pathological examination, a multilocular cystic mass measuring 5.0 × 4.8 × 4.5 cm was observed in the cervix. The cut surface of the tumor was a mucosal surface. A 1.5-cm myoma was observed in the myometrium of the posterior uterine wall. The endometrial cavity was free of tumors. The right ovary and the right fallopian tube were normal. On microscopic examination, the mass was found to be benign or mildly atypical Müllerian glands and low-grade malignant stroma Figure 22MA is a mixed epithelial and mesenchymal tumor with benign or mildly atypical glandular and malignant stromal cells. In total, 71%, 15%, 12%, and 2% of cases had MA occurring in the uterine corpus, ovaries, pelvis, and uterine cervix, respectively . The etiMixed Müllerian tumors are classified into adenomyomas, adenofibromas, adenosarcomas, and carcinosarcomas  on the basis of the composition of benign or malignant epithelial and mesenchymal elements of Müllerian origin. MA is a low-grade neoplasm along the spectrum of mixed Müllerian tumors, with adenofibromas at the beginning and carcinosarcomas at the end . Some tuThe management of adenofibroma and adenosarcoma is assumed to be similar and includes a hysterectomy. However, the optimal course of therapy for adenosarcomas is also uncertain. This uncertainty may arise from the fact that adenosarcoma appears in the early stages of reproductive life, and its potential for malignancy remains poorly understood. Most authors have recommended hysterectomy accompanied by bilateral salpingo-oophorectomy because treatment plans are made by analogy with treatments for common diseases for which the optimal treatment has already been determined . AlthougThe prognosis of MA is characterized by invasion of the cervical wall and sarcomatous overgrowth (SO). Although MA is regarded as less aggressive and has lower malignancy potential, some unfavorable prognostic factors have been reported, such as a high mitotic rate and the presence of heterologous elements, deep myometrial invasion, necrosis, and extrauterine spread. However, the presence of myometrial invasion and SO has been demonstrated to be associated with poor prognosis, increased risk of postoperative recurrence, and fatal outcome , 19. ForWe conducted a Medline search for articles on MA published in English between 1976 and 2022 using “Müllerian adenosarcoma, uterine cervix” or “adenosarcoma, uterine cervix” or “Müllerian adenosarcoma, cervix” or “adenosarcoma, cervix” as keywords and selected papers reporting data on premenopausal women. The clinical characteristics of these patients are summarized in The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by Peking University First Hospital’s ethical review committee. The ethics committee waived the requirement of written informed consent for participation.XZ: conceptualization and writing-original draft. CP: data analysis. YH: writing-original draft. YZ: conceptualization, writing-reviewing and editing. All authors contributed to the article and approved the submitted version.


True congenital pancreatic cysts are a rare pathological process reported within feline and human literature. To date there has been no documented case of a true congenital cyst affecting a canine patient. The objective of this case report is to document the clinical findings, diagnostic investigations, surgical treatment, histopathological diagnosis and long-term outcome of a dog with a true pancreatic cyst.A 5-month-old crossbreed dog was presented with a six-week history of abdominal pain, apparent bilateral pelvic limb weakness, reluctance to walk and intermittent vomiting and diarrhoea. An abdominal ultrasound examination performed by the dog’s primary care veterinarian identified a large intra-abdominal structure of unclear origin. A computed tomographic examination identified a large ovoid structure measuring 156 mm in length, 95 mm in height and 89 mm in width and apparently originating from the left limb of the pancreas. An exploratory coeliotomy was performed and a partial pancreatectomy was performed to allow complete removal of the cystic structure. Histopathological analysis of sections of the wall of the large fluid-filled cyst identified a thick fibromuscular wall lined by a well regimented hyperplastic tall columnar epithelium with basally located round to ovoid nuclei featuring fine chromatin stippling and abundant apically located and surface mucin, concurrent with a true congenital pancreatic cyst. A long-term follow-up of twenty-nine months identified no clinical signs of recurrence.A partial pancreatectomy and en bloc excision of a true pancreatic cyst provided an excellent long-term outcome in a dog. Cystic lesions of the pancreas are infrequently documented within both the veterinary and human literature. Isolated case reports and series identify pseudocysts , 2, cystA five-month-old entire female crossbreed dog weighing 14.6 kg (32.2 lb) was presented for further investigations of insidious onset of abdominal pain, apparent bilateral pelvic limb weakness, reluctance to walk and intermittent vomiting and diarrhoea for a duration of six weeks prior to referral. Initially, clinical signs were managed with symptomatic therapies by the dog’s primary veterinarian. Due to the persistence and progressive worsening of observed signs, the dog’s primary veterinarian performed abdominal radiography and an abdominal ultrasound examination which identified a large 60 mm × 80 mm ovoid structure in the mid abdominal region. The primary veterinarian suspected mild inflammation of the surrounding mesentery and the remainder of the examination was considered normal at this time. An ultrasound-guided fluid sample was obtained from the structure and cytological analysis identified a proteinaceous fluid with necrotic cellular material and mild, non-septic neutrophilic inflammation with moderate to marked accumulation of uniformly blue-black pigment.The patient was bright, alert and responsive during the examination, and all clinical parameters were within normal reference ranges. A non-reducible, non-painful umbilical hernia was identified. She displayed no obvious signs of pain or discomfort on abdominal palpation. The previously described abdominal cystic structure was not palpable on abdominal palpation.Hematology and biochemistry analysis were grossly unremarkable and identified mild elevations in urea 15 mmol/l (3.6–8.6 mmol/l), creatinine 256 mmol/l (20-120 mmol/l), alkaline phosphatase (ALP) 167 U/L (0-82 U/L), alanine aminotransferase (ALT) 83 U/L (0–36 U/L) and amylase 2642 U/L (400–1300 U/L). Urinalysis of a sample obtained by cystocentesis identified epithelial 1+, red blood cells 1+, white blood cells 4+, and was therefore considered normal.A computed tomographic (CT) examination was performed under general anaesthesia. A combination of butorphanol 0.3 mg/kg 0.13 mg/lb) (torbugesic 10 mg/ml)3 mg/lb  and acellular debris. Biochemical analysis of the fluid sampled identified a total protein level of less than 20 g/l, albumin less than 10 g/l, amylase and lipase values of 15,570 U/L and 1150 U/L were measured, respectively and a glucose concentration of 3.57 mmol/l.On the day of surgical exploration, the dog was pre-medicated with medetomidine hydrochloride 1μg/kg (0.0004 mg/lb) (sedastart 1 mg/ml) and methadone 0.2 mg/kg (0.09 mg/lb) (synthadon 10 mg/ml).A standard ventral midline coeliotomy was performed from the xiphoid process to the pubis. The large ovoid structure was identified in the cranial mid-abdomen, consistent with the computed tomographic description of location and was confirmed to arise from the pancreas, at the junction between the left and the right limbs. The right limb of the pancreas was grossly normal and present within its physiological anatomical location. The left limb of the pancreas was oedematous and confluent with the surface of the mass. There was minimal adhesion of the mass to the remaining intra-abdominal structures. A partial pancreatectomy, the left limb was performed to allow complete removal of the cystic structure, en-bloc Figs.  and 3.FiAdditional procedures performed, included ovariectomy, repair of the non-reducible umbilical hernia and the placement of an oesphagostomy feeding tube, due to risk of likely development of pancreatitis subsequent to surgery. No intra-operative complications were encountered. Abdominal closure was performed using 2.0 polydioxanone The entire mass was submitted to the institutional anatomic pathology department and sections of the wall Fig.  of the mThe dog recovered uneventfully from anaesthesia and was maintained in the hospital intensive care unit overnight. Within 12 h of surgery finish, the dog was eating and drinking voluntarily, and the oesophagostomy feeding tube was not employed. Post-operative analgesia was provided via a continuation of the fentanyl (fentadon 50μg/ml)The dog was discharged from hospital three days post-operatively with orally administered non-steroidal anti-inflammatory medication, meloxicam 0.1 mg/kg (0.045 mg/lb) once daily (metacam 1.5 mg/ml).Congenital pancreatic cysts are a rare condition and infrequently reported in both the veterinary and human literature. A relatively recent article within the human literature identified only 25 reported cases of congenital pancreatic cysts  and as fIn human medicine, congenital pancreatic cysts are found preferentially in the neck or tail (62%) and the head (32%) of the pancreas or can diffusely involve the whole pancreas (6%) . These gThe exact aetiology of congenital pancreatic cysts remains uncertain at this time  but theyThe current recommendation within the human literature regarding diagnostic investigation is computed tomography (CT) with iodinated contrast medium or magnetic resonance (MR) with gadolinium. Both diagnostic imaging modalities can achieve a diagnosis and help identify any evidence of malignant transformation, however, their accuracy in differentiating benign cysts from potentially malignant cysts is limited and varies between 20 and 80% depending on studies . Fine neOnce a pancreatic cyst is identified, cystic fluid analysis can be useful to differentiate between the various types of pancreatic cystic lesions: congenital, retention, duplication, pseudocyst, neoplastic, and parasitic cysts . Within The role of glucose concentration in intracystic fluid as a marker in the differential diagnosis of pancreatic cysts was first proposed by Park et al. in 2013. In particular, glucose concentration can be used to differentiate mucinous from non-mucinous cysts ; mucinouA definitive diagnosis of a congenital pancreatic cyst can be obtained through histopathological analysis. True congenital pancreatic cysts are described to be lined with cuboidal-to-columnar epithelium  or simplWhile a complete cystectomy is the traditionally preferred treatment for a congenital pancreatic cyst  If the lThe rarity of documented cases of congenital pancreatic cysts within the veterinary literature makes it difficult to delineate similarities or inconsistencies when compared to human medicine. The case presented here describes a confirmed histopathologically diagnosed true congenital pancreatic cyst with elevated cystic amylase levels. Further investigations and case reports will be required to assess the relevance and commonality of these findings. However, complete surgical excision of the cyst was deemed curative for this single case, and long-term follow-up yielded no recurrence of clinical signs, no cyst regrowth, and no adverse clinical effects of partial pancreatectomy in a juvenile patient.


Emphysematous gastritis is a form of gastritis characterized by both gastric inflammation and the presence of intramural gas. Its occurrence is rare, and its presentation is non-specific. Consequently, no definitive guidelines for management have been outlined. We herein detail the diagnosis and surgical management of a female with complicated emphysematous gastritis following a cesarean section delivery. In light of the gastric ischemia noted on esophagogastroduodenoscopy, the decision was made to proceed with surgical management to ascertain the extent of necrosis. Following a partial gastrectomy, the patient had an uneventful postoperative course and met feeding milestones. Given the high morbidity rate of emphysematous gastritis and the success of our intervention, we propose the utilization of exploratory laparoscopy/laparotomy in patients with identified or highly suspected gastric ischemia. This aids in the characterization of ischemia and guides decision-making on the extent of gastric resection . Emphysematous gastritis is a rare form of gastritis with a mortality rate of more than 50% . It is cA 39-year-old G3P2102 with a history of type 2 diabetes mellitus, noncompliance with medication, hypertension, and depression was admitted for hyperglycemic control and superimposed preeclampsia with severe features at 33 weeks and three days gestational age. She remained admitted until induction of labor at 34 weeks gestation. A primary cesarean section was pursued due to the maternal inability to accommodate lithotomy positioning for vaginal delivery. A chart review showed multiple presentations to the emergency department four to five months prior to admission with complaints of nausea, emesis, and abdominal pain that were attributed to pregnancy at that time.3/µL) with a left shift (88.5% neutrophils). A chest computerized tomography (CT) angiogram was ordered for concern of a pulmonary embolism which revealed evidence of emphysematous gastritis with air in the gastric wall and no evidence of pulmonary embolism  but afebrile. Laboratory workup was significant for a new leukocytosis  without lactic acidosis. She was started on piperacillin/tazobactam, and fluconazole. Due to concerns of full-thickness necrosis secondary to gastroparesis, given her history of uncontrolled diabetes, she underwent diagnostic laparoscopy with esophagogastroduodenoscopy, which confirmed full-thickness necrosis of the anterior wall of the gastric fundus and part of the body. The decision was made to convert to exploratory laparotomy, and a 15 cm ×15 cm resection of the gastric fundus and body was performed  and tube feedings through the jejunostomy tube.On clinic follow-up one week after discharge, the patient was doing well, tolerating her clear liquid diet and tube feeds with no abdominal pain, nausea, or emesis; therefore, the diet was advanced to a full liquid/puree diet with the plan to repeat esophagogastroduodenoscopy in two weeks.Staphylococcus aureus, Streptococci, E. coli, Clostridium species, Enterobacter species, Pseudomonas, Proteus species, and Candida [Emphysematous gastritis was first described by Frankel in 1889 . The exaAn important distinguishing factor in our report is that the patient demonstrated symptoms of diabetic gastroparesis based on her multiple presentations to the emergency department with emesis and abdominal pain (attributed to pregnancy at that time), which also predisposed her to develop emphysematous gastritis. In the event of gastroparesis and gastric dilation, the intraluminal pressure can exceed the gastric venous pressure, usually greater than 20 mmHg, impairing intramural blood flow and leading to gastric wall ischemia and necrosis ,10.Emphysematous gastritis has a non-specific presentation, including nausea, vomiting, abdominal pain, abdominal tenderness on examination, and leukocytosis. A CT scan is more sensitive and specific than abdominal films, with a classical finding of gastric wall thickening and air steaks or pockets within the gastric wall ,11. In aIt is crucial to distinguish emphysematous gastritis from gastric emphysema due to their overlapping radiological findings. Gastric emphysema results from mechanical injury to the stomach mucosa, either from a direct injury (nasogastric tube or esophagogastroduodenoscopy) or from increased intraluminal pressure within the stomach, i.e., barotrauma, leading to the introduction of air into the stomach wall ,11. As aThe utilization of esophagogastroduodenoscopy remains controversial. While some experts emphasize urgent esophagogastroduodenoscopy to rule out gastric ischemia and the need for laparotomy ,7. OtherIn this report, we present the case of a 39-year-old postpartum female with postpartum emphysematous gastritis and gastric necrosis requiring surgical intervention. While there are many points of discussion, in this case the two most relevance from a surgical perspective are the initial antibiotic choice and the need for operative intervention.The literature review showed two management strategies, non-operative versus operative, based on the clinical stability and evidence of necrosis on esophagogastroduodenoscopy . In our Given the limited available data and the lack of a standardized clinical decision-making pathway for this rare condition, we propose the following strategy. When the diagnosis of emphysematous gastritis is confirmed by CT scan, immediate initiation of medical management is necessary, including nulla per os (NPO) status, a nasogastric tube, broad-spectrum antibiotics , and an antifungal agent if needed. Medical management should be followed by an esophagogastroduodenoscopy to assess for the presence of mucosal ischemia. In the absence of ischemia, we recommend continued non-operative management. However, if ischemia is present, we propose further investigation with an exploratory laparoscopy/laparotomy to further characterize the degree of ischemia and to determine whether a partial or complete gastrectomy is required.By introducing this proposed management strategy, we aim to establish a more delineative, systematized pathway for treatment that eliminates ambiguity and optimizes patient outcomes.


Stromal tumors of the testis are rare. However, among this group, Leydig cell tumors (LCT) are the most frequent, and recent studies suggest that LCTs account for up to 22% of small testicular nodules. It is now accepted that small LCTs can benefit from testis-sparing surgery or in some selected cases radiological surveillance. Since percutaneous testicular biopsy is still not recommended, the diagnosis of LCT rests on multimodal imaging techniques. Therefore, it is essential for the radiologist and the urologist to know the main imaging features of LCTs in ultrasound and MRI.Pre-operative testicular tumor characterization is a challenge for radiologists and urologists. New data concerning imaging approaches or immunochemistry markers improve the management of patients presenting with a testicular tumor, sometimes avoiding radical orchiectomy. In the past 20 years, imaging modalities, especially ultrasound (US) and magnetic resonance imaging (MRI), improved, allowing for great progress in lesion characterization. Leydig cell tumors (LCT) are rare testicular tumors developing from the stromal tissue, with relatively scarce literature, as most of the studies focus on the much more frequent germ cell tumors. However, with the increase in testicular sonography numbers, the incidence of LCT appears much higher than expected, with some studies reporting up to 22% of small testicular nodules. Multimodal ultrasound using Doppler, Elastography, or injection of contrast media can provide crucial arguments to differentiate LCT from germ cell tumors. Multiparametric MRI is a second intention exam, but it allows for extraction of quantifiable data to assess the diagnosis of LCT. The aims of this article are to review the latest data regarding LCT imaging features, using multimodal ultrasound and multiparametric MRI, and to focus on the peculiar aspect of the testis of patients with Klinefelter’s syndrome. The possibility of an LCT should be raised in front of a small hypoechoic tumor with a marked corbelling hypervascularization in an otherwise normal testicular pulp. Ultrasonographic modules, such as ultrasensitive Doppler, contrast-enhanced ultrasonography, or elastography, can be used to reinforce the suspicion of LCT. MRI provides objective data regarding vascularization and enhancement kinetics. Testicular tumors are one of the most frequent cancers in the young. A total of 95% of these lesions derive from the germ cell line with seminomatous and non seminomatous tumors. Sex cord stromal tumors are rare, as they represent only 5% of all testicular tumors. This group is dominated by one subtype of tumor: the Leydig cell tumor (LCT), as Sertoli cell tumors are extremely rare  . Leydig ®, CEUS, contrast-enhanced ultrasound/sonography, elastography, SWE, strain elastography, shear wave elastography, MRI, MR, multiparametric MRI/MR, contrast-enhanced MRI/MR, and diffusion-weighted/DWI MRI/MR), individually associated with the terms testicle/testis and Leydig/Leydigoma. The two senior radiologists reviewed titles and abstracts one by one, excluding posters, comments, and letters to the editor, duplicate articles, pediatric studies, and animal studies. Several articles in a language other than English were included because of their importance to the issue in question.The literature search was performed by two senior radiologists specialized in testicular imaging, using the PubMed, EMBASE, and Cochrane databases, and covering articles from their first availability until December 2021. We considered different methods of ultrasound imaging and MRI and their variations  [Analysis of the adjacent testicular pulp is primordial because most LCTs are found in normal pulp, and only 9.4% of the lesions were accompanied by non-grouped grade I  microlithiasis . These rr—ultrasensitive Doppler yielded 24 references, including 6 case reports.The PubMed search for articles published between 1993 and 2021, using the key words Leydig cell tumour/tumor—ultrasound/sonography—SMI® from Canon or Angioplus RT® from Aixplorer). Maxwell et al. found that close to 95% of lesions were hypervascular compared with the adjacent pulp, with an enveloping vascularization pattern or a mixed  pattern of the vascular architecture [Color Doppler allows for qualitative assessment of tumor vasculature and its organization. Most recent ultrasound devices have a Doppler module optimized for the detection of the microflow associated with a decrease in motion artifacts , (CEUS) allows for qualitative and quantitative analysis of testicular and lesion enhancement. The first studies using CEUS showed that LCTs present early enhancement and that a feeding vessel in the periphery of the tumor could be identified [The PubMed search using contrast-enhanced ultrasound-CEUS/contrast-enhanced ultrasound yielded 15 references, including four case reports. Through the intravenous injection of microbubbles  ,18,19. TThe PubMed search using the key words Leydig cell tumour/tumor—ultrasound/sonography—elastography—SWE—strain elastography—SE—shear wave yielded 8 references between 2012 and 2021, including 2 case reports.The use of testicular elastography spread at the end of the 2000s. Most studies use the technique of strain elastography. In 2012, Goddi et al. reported promising results, with a sensitivity/specificity of 87.5%/98.2% in differentiating benign lesions from malignant lesions, though this study included only 2 LCTs among the 103 nodules studied . The firThe PubMed search using the key words Leydig cell tumour/tumor—MRI—yielded 17 references between 1990 and 2021, including two case reports.With the rising incidence of LCTs and the possibility of conservative surgery or close monitoring, the use of MRI of the scrotum is becoming increasingly widespread. This examination has the advantage that it is reproducible, provides morphological information, and quantifies enhancement. However, given its limited accessibility, MRI is at present a second-line examination.p = 0.001) and of moderate intensity in 60% of seminomas vs. 5.3% of LCTs (p < 0.001). Second, compared with the pulp, 68.4% of LCTs were well-defined vs. 10% of seminomas (p < 0.001). The T1 signal did not reveal a statistically significant difference. These results are to be weighed against the MRI findings of El Sanharawi et al., who analyzed 12 benign tumors, including 11 LCTs and 12 malignant tumors that included 10 seminomas [As in ultrasound examination, sporadic LCTs are well-delimited solid masses with regular margins. They have a low signal intensity on T2-weighted sequences . In 2015eminomas . In this−3 mm2 s−1, with a linear increase in ADC with age [2 and reported ADC mapping of 22 malignant lesions (including 20 seminomas of average size 8 mm) and 25 benign tumors (25 LCTs of average size 6 mm) [The testicle has a high cellular density, which physiologically restricts apparent diffusion coefficient (ADC) mapping. Tsili et al. reported that the mean ADC in men aged 20–39 years was 1.09 × 10with age . In anotwith age . Few stuze 6 mm) . LCTs reLCTs are hypervascular lesions and this characteristic is found by Doppler ultrasound and by CEUS. Dynamic contrast-enhanced MRI can be used to analyze quantitative data in order to distinguish LCTs from seminomas and hence to propose testis-sparing surgery. The 2015 prospective study of Manganaro et al. on 19 LCTs showed that scrotal MRI with injection had a sensitivity of 89.47% and a specificity of 95.65% for the diagnosis of LCTs and a precision of 93% in distinguishing between seminoma and LCT . In thisKlinefelter syndrome (KS) or 47,XXY  affects 0.1–0.2% of the general population, but the incidence rises to 3.1% in the infertile population . PatientImprovement in characterization of sporadic Leydig cell tumors using B Mode US, Color/ultrasensitive Doppler, Contrast-enhanced Ultrasound, Elastography, and multiparametric MRI leads to a better management of such lesions: testis-sparing surgery or even follow up can now be suggested based on advanced imaging. The radiologist must recognize the specific echostructure of Klinefelter syndrome patients due to Leydig cell hyperplasia or small LCT to avoid useless and potentially harmful surgery.


Duplication cysts close to the ileocecal valve are usually treated with ileocecal resection. However, loss of the ileocecal valve will lead to problems, especially in infants. Mucosectomy of the cyst would be a better alternative that preserves the ileocecal valve. We report two cases of duplication cyst in the terminal ileum successfully treated with mucosectomy.Case 1. A 3-month-old boy with bilious emesis and abdominal distention was referred to our hospital with a diagnosis of small bowel obstruction caused by an abdominal cyst. Computed tomography revealed a cystic mass compressing the terminal ileum and causing mechanical small bowel obstruction. His general condition deteriorated quickly; emergency laparotomy was performed. Although the small intestines were dilated and partially twisted, there was no necrosis. Following intestinal decompression, a cystic mass adjacent to the terminal ileum was confirmed on the mesenteric side. Cyst mucosectomy was performed to preserve the ileocecal valve. Case 2. A 5-month-old boy with sudden onset of hematochezia was referred to our hospital with a diagnosis of intussusception. Following unsuccessful contrast enemas, emergency surgery was performed. A cystic mass adjacent to the terminal ileum was confirmed; there was no intussusception. Cyst mucosectomy was performed. Both patients had an uneventful postoperative course.Cyst mucosectomy, which preserves the ileocecal valve, is safe and effective for treating duplication cysts in the terminal ileum. Duplication cyst of the alimentary tract is a rare congenital anomaly with diverse clinical presentation , 2. SurgTo date, there have been few case reports of IC valve-sparing surgery to preserve IC valve function. Most cases have been treated with IC resection and ileocolic anastomosis, even when IC valve-sparing surgery is feasible , 8. HereA previously healthy 3-month-old boy with sudden onset of bilious emesis and abdominal distention for 1 day was transferred to our hospital. Abdominal ultrasound and contrast-enhanced computed tomography showed a cystic mass (60 mm) located in the right lower abdomen .The optimal surgical approach for small intestinal duplication cysts is removal of the lesion. This is generally accomplished by resecting the duplication cyst with adjacent bowel and primary anastomosis , 10. In Catalano et al. first reported the safety and efficacy of IC valve-sparing resection in three pediatric patients with IC duplication cyst . They apConcerning clinical presentation, Patient 1 had volvulus and Patient 2 was initially misdiagnosed with intussusception. A review of the Japanese literature on IC duplication showed that the majority of IC duplications were cystic. One-half of the cases manifested by 1 year of age. Initial symptoms included vomiting and abdominal pain. The unique clinical picture of IC duplication cysts included intussusception (34%), hematochezia (24%), and perforation (4%), but no intestinal volvulus has been documented . In PatiOf note, the choice of surgical approach should be based on the patient’s hemodynamic status, because mucosectomy will require more time than enucleation. The operative times of our cases, which underwent mucosectomy, were 204 and 217 min, respectively. In contrast, operative times in the previous two reports, which underwent enucleation, were 105 min (mean from 3 cases) and 146 min, respectively . EnucleaWe encountered two patients with IC duplication cyst that presented with volvulus and hematochezia, respectively. IC valve-sparing surgery was safe and effective for duplication cysts adjacent to the IC valve.


Growing teratoma syndrome (GTS) is rare and can mimic disease recurrence in patients with a history of immature teratoma. Benign hypermetabolic lymphadenopathy found on staging and surveillance computed tomography (CT) and positron emission tomography (PET) may lead to the presumption of metastatic malignancy. We report a case of a 38 year old with mixed mature and immature teratomas who developed new peritoneal masses after adjuvant chemotherapy despite a normalization of tumor markers. In addition to low FDG uptake observed in these peritoneal masses, a PET scan showed hypermetabolic lymphadenopathy and pulmonary and spleen lesions suggesting widespread metastases. Subsequent surgical resection confirmed a mixed pathology with GTS and sarcoidosis. We reviewed the current literature evidence of GTS and sarcoidosis as a benign cause of lymphadenopathy in cancer patients. We emphasize the importance of a tissue diagnosis before instituting therapy for presumed cancer recurrence to avoid potentially fatal diagnostic traps and management errors. A multiple disciplinary team approach is imperative in managing patients with suspected recurrent immature teratomas.  Immature teratomas of the ovary represent approximately 1% of ovarian tumors . Tumor mCompletion surgery with comprehensive staging is recommended as initial surgery for patients with immature teratoma who do not desire fertility preservation . If patiMost patients with immature teratomas are cured, but a small percentage develop disease recurrence. It has been reported that 90% of recurrence occurs within 24 months of the initial diagnosis . BecauseA 38-year-old female presented to a local hospital with acute abdominal pain in early 2018. She had a history of a left dermoid cyst. Physical examination found a 10 cm tender left-lower abdominal mass. She had an elevated AFP of 160 IU/mL and an undetectable beta-HCG. The provisional diagnosis was a ruptured dermoid cyst and an urgent laparoscopic left salpingo-oophorectomy was performed. Pathology revealed a grade 3 immature teratoma. Her subsequent staging CT scan showed several tiny pulmonary nodules measuring 2 to 3 mm and multiple small ill-defined hypodensities within the splenic parenchyma of unknown etiology. In addition, there was a borderline enlarged pre-carinal lymph node measuring 17 by 12 mm.A completion surgery with comprehensive staging was performed in a tertiary referral hospital. Scattered deposits/implants were observed on the surface of the abdominal/pelvic cavity, which was completely resected macroscopically. The final pathology revealed a staged 3B mixed grade 3 immature teratoma A and matShe received four cycles of BEP adjuvant chemotherapy and tumor markers (AFP) normalized during chemotherapy. Repeat CT at the completion of chemotherapy however demonstrated new peritoneal masses A. The biHer imaging and pathology were reviewed in our Gynecological Oncology multidisciplinary team (MDT) meeting. The peritoneal masses with low FDG uptake were consistent with growth teratoma syndrome (GTS). The hypermetabolic lymphadenopathy and splenic lesions could have been due to metastatic immature teratomas, but other benign conditions, such as infectious etiologies or inflammatory diseases, could not be ruled out. A careful review of FNAC showed scattered group of small, atypical cells that were devoid of mitoses and marked nuclear atypia. Therefore, the differential diagnoses included benign endobronchial cells with cartilage from respiratory tract origin or components of recurrent teratomas. The management options of surgical resection or second-line high-dose chemotherapy with stem cell rescue were debated. The final consensus was to proceed with a laparotomy to completely resect the mildly FDG avid peritoneal masses and highly FDG avid porta hepatis lymph nodes and splenic lesions.In July 2018, the patient had the third procedure, including a laparotomy, tumor debulking and splenectomy. The final pathology confirmed GTS involving the peritoneal lesions. However, the spleen, porta hepatis nodes, vaginal vault and liver surface nodules showed large active non-caseating granulomas with epithelioid and Langerhans giant cells with focal central hyalinization and necrosis C,D consiIn this report, we described a female with resected mixed mature and immature teratomas who had a complete biochemical response to adjuvant BEP chemotherapy, but developed a diagnostic challenge of enlarging peritoneal lesions. Staging PET scan showed hypermetabolic lymphadenopathy and pulmonary nodules and splenic lesions. She was subsequently found to have dual pathology with GTS and sarcoidosis.GTS was first reported by Logothetis in 1982, after observing enlarging solitary mass(es) following chemotherapy, despite normal tumor markers in six patients with metastatic mixed germ-cell tumors . It was The incidence of GTS in males with non-seminomatous germ-cell tumors has been estimated at around 2–8%. Two recent, large case series reported GTS to occur in 20% of patients with immature teratomas and half of GTS occurred during chemotherapy ,14,15. TThe pathogenesis of GTS is still uncertain. Two different mechanisms have been proposed. The first is that chemotherapy induces the malignant cell differentiation of an immature teratoma into a mature teratoma. The second is that chemotherapy selectively destruct immature components that are chemotherapy sensitive, whereas mature components, resistant to chemotherapy, persist and grow as GTS . GTS doeThe risk factors for GTS have not been confirmed, but the presence of residual disease at primary surgery is considered an important contributor . GTS genIn patients with immature teratomas, GTS can be mistaken for disease relapse or recurrence. The diagnosis of GTS can be challenging, but should be suspected after serial imaging showing enlarging mass(es) during or after treatment with chemotherapy with discordant reduction in tumor markers . There aFDG-PET/CT is widely used for cancer staging and surveillance. FDG-avid lymph nodes, however, are not specific for malignancy . InflammSarcoidosis is a multisystemic, non-caseous, benign granulomatous disease of unknown etiology with the incidence estimated to be 50 to 150 per 100,000 population . The diaSeveral small case reports have described the coexistence of sarcoidosis and malignancy, including Hodgkin’s lymphoma and testicular cancer. In a retrospective case review of 565 patients who underwent mediastinoscopy at a Swedish Medical Center and Cancer Institute, the prevalence of biopsy-proven sarcoidosis was 7.5% . Even acIn conclusion, GTS is a rare entity that can cause diagnostic uncertainty, post-treatment of immature teratomas. A multiple disciplinary team approach is imperative in managing patients with suspected recurrent immature teratomas. Hypermetabolic lymphadenopathy on staging or surveillance imaging can present a diagnostic dilemma. To avoid potentially fatal diagnostic traps and management errors, it is important to obtain adequate tissue through a core needle biopsy whenever possible, or even a surgical biopsy if necessary, before instituting therapy for presumed cancer recurrence.


An adrenal incidentaloma is an adrenal mass ≥ 1 cm in size discovered on imaging performed for indications other than suspected adrenal disease. It has variable etiologies, which can be benign or malignant, including primary or metastatic disease. We present a rare case of metastatic lung adenocarcinoma with isolated unilateral adrenal metastases, presenting as an adrenal incidentaloma in an asymptomatic patient with no known history of malignancy. A 76-year-old man with a past medical history of chronic obstructive pulmonary disease (COPD) and heavy tobacco use was admitted for the evaluation and treatment of pneumonia. He was found to have an incidental 4.6 cm unilateral adrenal mass on his CT chest. He underwent a workup for the mass, including further imaging studies that were indeterminate and a hormonal workup that concluded that the mass was nonfunctional. Due to the patient’s comorbidities, it was determined that he was not a surgical candidate. A multidisciplinary team recommended a biopsy, which revealed metastatic lung adenocarcinoma. The primary lung cancer was located using positron emission tomography with 2-deoxy-2-(fluorine-18) fluoro-D-glucose combined with computed tomography (F-FDG-PET/CT). The patient was evaluated by an oncology service and started on chemotherapy. In this case report, we discuss the approach for evaluating adrenal incidentalomas as well as the role the biopsy has in this process based on a literature review. In addition, we draw a comparison between our case and similar cases in the literature while highlighting the differences that make this case unique. An adrenal incidentaloma is an asymptomatic adrenal mass >1 cm in size, detected incidentally on imaging that was performed for another indication, without the suspected adrenal disease . Most adHere, we report the case of an asymptomatic patient with no known history of cancer who presented with a unilateral adrenal incidentaloma. Imaging was indeterminate; a biopsy revealed metastatic lung adenocarcinoma, after which the primary malignancy was located using a positron emission tomography with 2-deoxy-2-(fluorine-18) fluoro-D-glucose combined with a computed tomography (F-FDG-PET/CT) scan. The patient was subsequently diagnosed with oligometastatic lung adenocarcinoma, as metastases were limited to a single adrenal gland.A 76-year-old man with a history of coronary artery disease, congestive heart failure, obstructive sleep apnea, hypertension, peripheral vascular disease, hyperlipidemia, chronic obstructive pulmonary disease (COPD), and a history of heavy tobacco use presented to an outside hospital with a four-day history of cough, shortness of breath, and wheezing. CT chest without contrast revealed bilateral lower lobe pneumonia, diffuse emphysematous changes, and an incidental finding of a 3.5 x 2.9 x 4.6 cm left adrenal mass. The adrenal mass was not present on previous CT imaging from three years ago. He was treated with broad-spectrum antibiotics for pneumonia with clinical improvement. He had a 50-pack tobacco use history per year and quit smoking following this hospitalization.On a repeat CT chest with contrast two months later, the adrenal mass measured 4.1 x 3.4 x 6.7 cm, with heterogeneity and high pre-contrast density. The imaging characteristics, along with the interval increase in size, were suspicious of malignancy. Of note, this imaging showed preexisting pulmonary emphysema with a 6 mm nodule in the superior segment of the left lower lobe that was stable when compared to imaging from three years ago; the imaging also showed improved consolidation in the bilateral lower lobes. He was started on steroids for secondary organizing pneumonia.The patient was referred to the urology clinic for an enlarging left-sided adrenal mass. A repeat CT of the chest, abdomen, and pelvis on the day of the initial visit with urology demonstrated that the left adrenal nodule was stable in size (6.3 cm) compared to the second scan done three months ago Figure . The preAt that time, his shortness of breath had improved since his hospitalization, but his physical activity remained significantly limited due to his chronic cardiopulmonary symptoms. He did not have any weight loss, fevers, chills, or gross hematuria. He was found to be a poor surgical candidate and was referred to endocrinology for a hormonal work-up, with a repeat CT scan planned in six weeks. The work-up revealed a normal complete blood count (CBC) and basic metabolic panel (BMP) , cortisol of 7 ug/dl (5-23 ug/dl), adrenocorticotropic hormone (ACTH): 12.2 pg/ml (7.5-63.3 pg/ml), dehydroepiandrosterone sulfate (DHEAS): 13 ug/dl (28-175 ug/dl), plasma metanephrines: 0.16 nmol/L (0.0-0.49 nmol/L), and plasma renin: 7.4 ng/ml/hr (0.5-4.0 ng/ml/hr). These normal values ruled out Cushing’s syndrome, primary hyperaldosteronism, and pheochromocytoma, indicating that the nodule is nonfunctional.A follow-up CT scan with the renal mass protocol, two months after the urology appointment, revealed that the mass had remained stable in size. At the tumor board meeting, the decision was made to get further assessment via an F-FDG PET/CT scan followed by an adrenal mass biopsy due to the indeterminate CT findings and the patient’s poor surgical candidacy. The patient was initially unable to get a PET-CT scan due to insurance issues, so he underwent a left adrenal biopsy. Pathology revealed a metastatic carcinoma. The immunohistochemical panel was performed, and it showed tumor cells to be diffusely and strongly positive for cytokeratin 7 (CK7) and thyroid transcription factor 1 (TTF-1), while negative for cytokeratin 20 (CK20), melan-A, inhibin, and S100. Based on the morphology and immunohistochemical profile of the tumor cells, a metastatic adenocarcinoma most likely from the lung primary was favored Figures .The patient was eventually able to undergo an F-FDG PET/CT scan, which revealed intense FDG activity fusing to the left renal mass, measuring standardized uptake value (SUV) max 16.2, consistent with the biopsy-proven disease, and moderate to intense FDG activity fusing to left lung upper lobe peripheral reticulonodular changes, measuring SUV max 4.6 Figure . The patMolecular testing of the biopsy specimen by the 22C3 assay revealed a programmed cell death ligand 1 (PD-L1) score of 50%. Based on these results, Keytruda (pembrolizumab) was recommended during the following oncology visit. After that, the patient chose to transfer his care to an oncologist closer to his home to receive this treatment.We reported a rare case of metastatic lung adenocarcinoma with isolated unilateral adrenal metastases, presenting initially as an asymptomatic adrenal incidentaloma. The patient did not have a prior history of lung cancer but had a long history of smoking and COPD. He had two preexisting stable 6 mm nodules on the CT chest that were not suspicious of malignancy, along with emphysematous changes and a few prominent mediastinal paratracheal lymph nodes. In this case, the adrenal incidentaloma was found on a CT chest, and an endocrine workup showed it was nonfunctional. The patient was not a surgical candidate. The diagnosis was established via a biopsy of the adrenal mass, which revealed lung adenocarcinoma. An FDG PET scan detected intense FDG activity in the adrenal incidentaloma and in the upper lobe of the left lung, where the location of primary cancer is likely, which is a different area from where the preexisting lung nodules were located.Most adrenal incidentalomas are nonfunctioning adrenocortical adenomas; other etiologies include but are not limited to adrenocortical carcinoma, pheochromocytoma, aldosterone- or cortisol-producing adenomas, or metastases. It is recommended to establish whether the mass is benign or malignant at the time of the initial detection of the incidentaloma . The iniAs for the hormonal workup of adrenal incidentalomas, it is recommended that all patients undergo a low-dose dexamethasone suppression test to exclude autonomous cortisol secretion. It is also recommended to exclude pheochromocytoma in all patients by measurement of plasma metanephrines or urinary fractionated metanephrines. For patients with hypertension or hypokalemia, plasma aldosterone and plasma renin can be measured to exclude primary aldosteronism. In patients with clinical or imaging features of adrenocortical carcinoma, measurement of sex hormones is recommended .In the case we presented above, the adrenal incidentaloma was found in an asymptomatic patient without a history of malignancy; a biopsy revealed metastatic lung adenocarcinoma; and an FDG PET scan identified the primary malignancy location in the lung, along with the isolated unilateral adrenal metastasis. In a literature review, a similar case was reported where a 67-year-old patient with no history of malignancy was being evaluated for a 4.6 cm adrenal incidentaloma that was found during a workup for nephrolithiasis. The mass was nonfunctional, and he eventually underwent an adrenalectomy, which revealed metastatic papillary thyroid cancer. A thyroid ultrasound later identified a nodule where the primary malignancy was located .In a study that was done to examine the role of FNA in the workup of adrenal incidentalomas, data from 1715 patients referred for evaluation of suspected unknown primary cancer were retrospectively reviewed; 1639 patients were found to have malignancies, and the adrenal gland was involved in 95 patients (5.8%). Only four of the 95 patients (0.2% of the 1639 patients) had isolated adrenal metastases; three of them were found to have lung adenocarcinoma, and only one had a unilateral lesion, but in none of the cases was primary cancer located. It is noteworthy that none of these adrenal masses were true incidentalomas, as all four had abdominal pain, and three of them had anorexia. The study concluded that metastatic cancer presenting as a true incidentaloma was extremely rare (0 out of 1639), so FNA was not recommended as part of the routine workup for a patient presenting with a true unilateral adrenal incidentaloma without a prior history of malignancy .The case we are reporting is unique in that the adrenal mass was a true incidentaloma as the patient was asymptomatic; it was also unilateral, while the majority of adrenal metastases are bilateral . An F-FDIt is also notable that the two preexisting 6 mm nodules found on the CT chest were located in the superior segment of the left lower lobe and in the right middle lobe, while the primary malignancy was located in the upper lobe of the left lung, where the F-FDG PET/CT scan detected intense FDG activity.Due to the rarity of adrenal metastases presenting as unilateral adrenal incidentalomas, previous studies showed no benefit of adrenal biopsies in such cases where the patient lacks any history of prior or current malignancy, and has no symptoms suggestive of occult malignancy. However, in our case, obtaining a biopsy of the adrenal incidentaloma eventually led to a diagnosis of metastatic primary lung adenocarcinoma.


Amyloidosis cutis dyschromica is a rare variant of primary cutaneous amyloidosis characterized by hyper- and hypopigmented macules. In this paper, we reported a case of a 16-year-old Filipino girl with hyper- and hypopigmented to depigmented macules on the upper and lower extremities, which started when she was 9 years of age. Amyloidosis cutis dyschromica (ACD) is a rare variant of primary cutaneous amyloidosis (PCA) which was introduced by Morishima et al. in 1970. ACD is characterized by the presence of hyperpigmented, hypopigmented or depigmented macules of varying sizes, which can present in generalized fashion . It is aA 16-year-old Filipino girl was born to parents with a non-consanguineous marriage. At 9 years of age, hyperpigmented and hypopigmented macules were initially noted to appear on her legs. There was no associated redness, itch, pain or numbness, and no medication or product taken or applied. No consultation was conducted then. Her birth history was unremarkable. During the interim of 5 years, the patient noted a gradual increase in the number of the lesions which later on involved her arms. There was no associated pruritus, erythema, telangiectasia, atrophy or blisters. No prior episodes of photosensitivity, inflammatory cutaneous conditions or systemic illness were also reported. Her birth history and mental developmental milestones were normal. There was a questionable family history of hypopigmented macules on the legs of her maternal grandfather. The general physical examination was unremarkable. Laboratory tests requested were not performed due to financial constraints of the patient. No medication or product was applied to the affected areas prior to consultation. The patient had minimal sun exposure as she stayed mostly indoors. Cutaneous examination revealed bilateral symmetrical involvement of the distal aspect of upper and lower extremities  with lesA skin punch biopsy sample was taken from the hypopigmented macular lesion of her right leg. Histopathological examination stained with hematoxylin–eosin (H & E) stain, and observed under light microscopy, showed compact hyperkeratosis, hypergranulosis, and irregular psoriasiform hyperplasia. Within a widened papillary dermis, there are collections of amorphous pink globules with a perivascular infiltrate of lymphocytes, histiocytes, eosinophils, and melanophages A. The prThere are three major types of primary cutaneous amyloidosis, namely lichenoid, macular, and nodular. Amyloidosis cutis dyschromica belongs to atypical and rare variant of primary cutaneous amyloidosis, which was first discussed by Morishima et al. in 1970 .It is predominantly reported in those of East and South-East Asian ethnicity (63%) . Its maiReticular and dotted pigmentation with hypopigmented spots can spread all over the body, with or without associated pruritus . PathogeSeveral important entities may manifest in generalized pigmentation with hypopigmented macules. Differentials such as dyschromica, including dyschromatosis universalis hereditaria, xeroderma pigmentosum, tuberous sclerosis, idiopathic guttate hypomelanosis and poikiloderma-like amyloidosis are important to be considered. In histopathology of the former four conditions, amyloid deposits are not appreciated. Meanwhile, the latter shares similar ACD but other features such as poikiloderma, clinical features with lichenoid papules, bullae, photosensitivity, short stature, and palmo-plantar keratoderma are also present .2 laser have been studied, and showed varying results. For certain cases of ACD, systemic retinoic acid derivatives, particularly acitretin, have shown good improvement [Avoidance of sun exposure and use of sun protection  are being advised to all the patients. Other treatment options, such as the use of topical corticosteroids, keratolytics, dimethyl sulfoxide, oral vitamin C and E and COrovement ,11.


Pseudomyxoma peritonei (PMP) is a rare manifestation of primary mucinous neoplasms. We report two rare cases of PMP originating from mucinous primary ovarian neoplasms. The case series discusses the cases of female patients aged 86 and 52 years who presented with worsening dyspepsia, abdominal distension, pelvic pain, and altered bowel habits. Both of the patients underwent evaluation comprising cancer antigen-125 (CA-125) levels, ultrasound (US) examination of the abdomen and the pelvis, tumor markers, cytological evaluation, and contrast-enhanced computed tomography (CECT) of the pelvis and abdomen. Patients were diagnosed to have pseudomyxoma peritonei arising from mucinous ovarian tumors. Patients were referred to the surgical department and were successfully managed with repeated removal of mucinous material. The present case report highlights the significant radio-pathologic characteristics of PMP, which originated from mucinous ovarian tumors. Pseudomyxoma peritonei (PMP) is a rare disease arising from primary mucinous tumors, which are usually appendiceal mucinous epithelial neoplasms and rarely mucinous ovarian tumors . PMP is Case report 1An 86-year-old female patient presented with progressively worsening indigestion and abdominal distension for two to three months and was referred to the department of radiology for the evaluation of a pelvic mass. Ultrasound examination of the abdomen and pelvis revealed a multiseptated and large cystic mass in the right ovary with severe ascites in the abdomen. The expression level of the tumor marker cancer antigen-125 (CA-125) was 61.2 U/ml . Contrast-enhanced computed tomography (CECT) of the abdomen and pelvis showed a large, peripherally enhancing cystic mass in the right ovary with thin enhancing septae. A similar cyst was noted, inseparable from the left ovary  with nodular peritoneal thickening and fat stranding. Around 20 ml of ascitic fluid was aspirated for further histopathological evaluation, which revealed mucinous epithelium totally lacking cytologic atypia consistent with PMP . Nodular peritoneal thickening was also noted, especially along the bilateral paracolic gutter. The surface of the liver and spleen adjacent to the diaphragm showed scalloping, which was suggestive of visceral peritoneal seedings. Scalloping of the liver surface was noted along the falciform ligament as well. Nodular deposits were noted on the surface of small bowel loops, especially in the right iliac fossa. There was moderate ascites entrapping bowel loops in the middle. Fluid aspiration was done, which showed acellular pink proteinaceous mucus material with a few RBCs in the background, consistent with pseudomyxoma peritonei Figure .Further, a histopathological examination of the pelvic mass was done, which showed an ovarian lesion composed of thin-walled cysts with mucinous columnar cells having basally oriented nuclei, consistent with benign mucinous ovarian cystadenoma Figure .The patient was referred to the surgery department for further management.PMP appears to be more common in women as a result of the trans-coelomic spread of ovarian tumors. Female patients also tend to present early due to the aggressive enlargement pattern of ovarian mucinous tumors giving rise to early symptoms such as abdominal pain and swelling . Due to The self-contained nature of the tumor rupture which limits microbial contamination by closing communications to the gut is the likely cause of the disease's asymptomatic nature. However; abdominal distension, discomfort, and pain are the late-onset symptoms brought on by the slow-moving fluid build-up caused by omental caking and pelvic masses. Such disease progression leads to malnutrition among patients. Also, bowel obstruction and compromise of the respiratory functions occur setting off a cascade of terminal events .PMP appears to be more common in women as a result of the trans-coelomic spread of ovarian tumors. Female patients also tend to present early due to the aggressive enlargement pattern of ovarian mucinous tumors, giving rise to early symptoms such as abdominal pain and swelling . Due to The self-contained nature of the tumor rupture, which limits microbial contamination by closing communications to the gut, is the likely cause of the disease's asymptomatic nature. However, abdominal distension, discomfort, and pain are the late-onset symptoms brought on by the slow-moving fluid build-up caused by omental caking and pelvic masses. Such disease progression leads to malnutrition among patients. Also, bowel obstruction and compromise of respiratory functions occur, setting off a cascade of terminal events .Several immunohistochemical markers such as CA-125, ER, PR, Pax8, and WT1 are known to be expressed at different levels in primary epithelial ovarian tumors . Ca-125 The CT scan reveals a mucinous ovarian tumor appearing as a large, unilateral, and complex cystic mass with variably attenuating fluid within the cyst loculi and internal calcifications. Features suggestive of an ovarian malignancy include a necrotic soft-tissue component with thick septa, thick irregular walls, and sometimes papillary projections. Other supportive findings include surrounding organ invasion, adenopathy, ascites, and involvement of the peritoneum, omentum, or mesentery. However, these findings are only specific to epithelial neoplasms and not to their subtype, which is mucinous neoplasms. In fact, most mucinous ovarian neoplasms are benign (4 in 5 cases) or borderline on pathologic analysis . AscitesCECT of the pelvis and abdomen is the gold-standard imaging of choice in the early staging of pre-treatment ovarian cancer and for detecting metastases to other organs. CT is supposed to be 94% accurate in the staging of ovarian cancer. However, limitations with CT include <5 mm metastases being undetectable when it involves the bowel surface, mesentery, and/or peritoneum. MRI continues to remain a problem-solving technique that is not used routinely and is not always necessary even though it has shown equal accuracy when compared to CT . DiffusiIt was estimated that the overall five-year survival rate for pseudomyxoma peritonei ranged between 11% and 75%. The median survival is 9.8 years and is independently associated with low-grade tumors , the finPMP is a rare clinical manifestation mostly associated with mucinous tumors. The case report presented is a rare clinical presentation arising from mucinous ovarian tumors, even though the most common cause is appendiceal tumors. These ovarian tumors are mostly benign or borderline, with a rare chance of malignancy. With the knowledge of classic imaging findings on CT, PMP can be diagnosed early on, which has been the aim of this case report. PMP has certain typical characteristics in CT that all radiologists should be aware of in order to increase the sensitivity of its detection. Confirmation of the origin of PMP is done through immunohistochemistry. Following this, the causative tumor is diagnosed histologically. FIGO system-staged mucinous ovarian neoplasms are then managed surgically , thus giving a good long-term prognosis for patients.


Alveolar soft part sarcoma (ASPS) is a rare kind of malignant soft tissue tumor with undefined differentiation, of which the incidence rate accounts for only 0.5–1.0% among all kinds of soft tissue tumors. An even rarer ASPS occurs in kidney.Here we reported a case of a 7-year-old girl diagnosed with nephrogenic ASPS, regarding the analyses of the incidence, clinical manifestation, pathology and genetic diagnosis, in order to deepen the recognition of the disease.ASPS is very rare, and tends to occur to young patients. It is very significant to precisely diagnose ASPS at an early stage, which will be the key point for the following treatment choices and prognosis. Alveolar Soft Part Sarcoma (ASPS), as a malignant tumor with unclear differentiation, tends to occur in juveniles, with the age of peak onset ranging from 15 to 35 and the histology featuring as specific acinus-like or organ-like structures . This stA 7-year-old female patient came to see a doctor in our hospital for low back ache with gross hematuria for one day. No abnormality was detected by physical examination. The result of routine urinalysis indicated 2083 RBCs/µl, while no abnormality was reported in other laboratory examinations.Ultrasonic examination (Mindray Resona 7) indicated: A solid hypoechoic mass with a size of 29.2 × 31.9 mm located in the dorsal parenchyma of upper pole of left kidney, with clear border and being endogenous for most part, slightly protruding from the surface of kidney, partly pressing towards renal collecting system, and annular calcification was observed in its surrounding  showed a circular soft tissue density with a size of 29 × 32 mm, which exhibited non-uniform interior and multiple arc and spot-like shadows of calcification inside of the foci and in the surrounding. The CT value of the solid part was 44HU, which was obviously increased after enhanced scanning. CT value during renal cortex phase was approximately 155 HU which equaled to that during renal medullary phase. Coarse shadows of blood vessels were observed inside of the foci, with a few small spot-like non-intensified focus in the center. The border between the mass and renal tissue was clear, and no apparent symptom of destruction was observed in neighbouring kidney calices , ectopia was detected in t(Xp11.2) TFE-3) genes. Gene sequencing was performed at 3D Med Inc. The results shown that gene arrangement occurred between exons 1–7 of ASPL and exons 6–10 of TFE3  chromosome translocation occurred in ASPS, which meant that a new fusion gene igenesis . Notablybalanced .KMT2C and BRCA2 were found, whereas the gene of ALK, BRAF, CD274, EGFR, FGFR2 and FGFR3 were normal. In BRCA2-mutated cells, the repair ability of DNA double-strand damage was impaired, and the express deficiency of BRCA is related to the occurrence and progression of non-small cell lung cancer, pituitary tumor, thyroid cancer, pancreatic cancer, as well as breast cancer [Otherwise, mutations of t cancer , 18. Howt cancer . In thist cancer . Previout cancer . NeverthCompared with other malignant tumors, ASPS possesses worse prognosis and higher invasion. Factors that affect its prognosis include age of onset, tumor size, primary site and metastasis. It has been considered by most researchers that the key point of treating ASPS is radical surgery. Given its poor prognosis and unremarkable effects of adjuvant chemoradiotherapy, molecular targeted therapy has become a new choice . HoweverTo sum up, ASPS is very rare, and tends to occur to young patients. It is very significant to precisely diagnose ASPS at an early stage, which will be the key point for the following treatment choices and prognosis.


Granulosis rubra nasi is a rare autosomal dominant disease of the eccrine glands. It is clinically characterized by mid-face hyperhidrosis, most prominent at the tip of the nose, and dark erythematous papules on the nose, cheeks, chin, and upper lip. Although it commonly occurs in childhood, it can also occur in adults. This is a case report of two female granulosis rubra nasi patients. This report, to the best of my knowledge, has not before been described in Ethiopian individuals and is hence being reported due to its rarity. Granulosis rubra nasi (GRN), also known as “Acne papulo-rosacea of the nose,” is a childhood inflammatory disorder of the eccrine glands first described by German dermatologist Jadassohn in 1901 [It is an autosomal dominant familial disease of children involving the eccrine glands of the nose, cheeks, and chin. This condition appears to be extremely rare  and geneIt is a chronic condition with a benign outcome. The cause is unknown and usually goes away on its own during adolescence. The treatment is both symptomatic and cosmetic in nature.I am reporting 2 cases of GRN here.The first case is a 19-year-old female who presented with a raised red lesion over the nose since childhood without sensitivity to sunlight. She was otherwise well, and there was no relevant family history. Physical examination revealed multiple dark erythematous papules in the nasal dorsum and alae with violaceous patches over the apex . The resThe second patient is a 25-year-old female who presented with redness and excessive sweating over the nose for more than 5 years. Physical examination of the nose revealed dark erythematous multiple papules that disappear on diascopy . This paAfter discussing the potential side effects of the few treatment options available in our setup (cryotherapy and isotretinoin), and since even those options were only moderately effective and considering the lack of the relatively safe topical treatment options in our setting , both patients were reassured considering the benign nature of the disease.GRN is a rare non-neoplastic autosomal dominant inherited disorder of the eccrine glands. There are also reports of autosomal recessive inheritance; however, the gene locus has not been identified .Etiology and pathogenesis of this disorder have not been elucidated. A marked increase in sweat production on the nose occurs in granulosis rubra nasi, possibly resulting in a unique form of sweat retention. This hyperhidrosis precedes months or years before the development of maculae, papules, vesicles, pustules, and, later, telangiectasia. Excessive sweating may cause cystic dilatation of eccrine ducts and lead to the development of erythema and papules . EddowesHeid et al. reported a case of pheochromocytoma in a 19-year-old woman with diffuse hyperhidrosis, tachycardia, and granulosis rubra nasi in whom surgical removal of the tumor was followed by the regression of nasal dermatosis .Although the exact frequency is unknown, it is believed that men are affected more than women; likewise, 6 out of the 7 patients described by Jadassohna were boys . SymptomIt is clinically characterized by hyperhidrosis of the central part of the face, most conspicuous on the tip of the nose, followed by the appearance of dark erythematous papules that disappear on diascopic pressure, vesicles, and telangiectasia over the nose, cheeks, and upper lips. A few small pustules may occur. The tip of the nose is usually red/violet, cold, and not infiltrated. This condition is usually asymptomatic except for mild pruritus . Heid etAt times, affected patients may have associated poor peripheral circulation and hyperhidrosis of the palms and soles . Summer Granulosis rubra nasi usually resolves spontaneously at puberty; however, it occasionally persists indefinitely, in which case telangiectasia becomes the predominant feature .The clinical picture of granulosis rubra nasi is distinctive, so there is rarely a problem with the diagnosis.Histology is helpful in difficult-to-diagnose cases and demonstrates a perivascular and periductal mononuclear cell infiltrate, cystic dilatation of eccrine sweat glands, and vascular proliferation. Histopathology of a papule showed epidermal atrophy, basal vacuolar degeneration, and nodular collection of lymphocytes; few histiocytes; and multiple blood vessels with swollen endothelial cells .Under polarized contact dermoscopy, Palit et al. reported a diffuse erythematous background with discrete round-to-oval pink and red structureless area, a few of them which showed overlying scattered to grouped brown dots .Possible differentials include papulopustular rosacea, papular sarcoid, trichoepithelioma, and perioral dermatitis. Other differential diagnoses include acne vulgaris, lupus pernio, and lupus erythematosus.It is advised to check for symptoms of pain and itching. Check for family history; the condition typically is autosomal dominant familial. Particular sensitivity to sunlight may suggest an alternative diagnosis. Other symptoms, such as a racing heart or a faint feeling, may indicate an alternative diagnosis  , as diapTo differentiate between these conditions, the distribution and morphology should be considered. The clinical features of perioral dermatitis consist of monomorphic small papules and pustules, erythema, and scaling, with distribution primarily around the mouth. In rosacea, there is erythema of the cheeks and nose, along with telangiectasia and accentuation of the erythema by vasomotor instability. Furthermore, in these two diseases, there is no hyperhidrosis of the central face, a prominent feature of granulosis rubra nasi.Photosensitive dermatoses are usually easy to differentiate from granulosis rubra nasi because they are more extensive and do not show the characteristic hyperhidrosis .Multiple eccrine hidrocystoma also presents with multiple translucent, dome-shaped papules on the periorbital and malar areas, with the frequent presence of craniofacial hyperhidrosis. However, this often occurs in middle-aged women and does not have the background erythema of the granuloma rubra nasi .In the majority of cases, counseling and explaining the self-resolution nature of the condition after puberty are the mainstays of management. However, in rare cases, it occasionally persists indefinitely, and it can have significant cosmetic and psychological impact.Treatments described in the literature, such topical indomethacin, tacrolimus, botulinum toxin A , cryothePiotr and Katarzyna have described a 14-year-old patient that responded well to low-dose isotretinoin , and KumGRN is a rare disorder. One should remember that it could be a complication of hyperhidrosis. Treatment is symptomatic and cosmetic. Counseling the patient about the self-limiting nature of the condition is of paramount importance. To my knowledge, this is the first time it has been reported in Ethiopian patients.


Primary leiomyosarcoma of the ureter is an extremely rare, aggressive malignancy of the urinary tract. This report describes a case of primary leiomyosarcoma of the distal left ureter in a middle-aged male, with no tumor recurrence achieved following resection and end-to-end ureteroureterostomy. Our patient was diagnosed with left distal ureter leiomyosarcoma following workup of an unexplained left hydroureteronephrosis on ultrasound. Wide excision was not required to achieve clear margins and a cure for this patient.Case PD is a 59-year-old man initially admitted for left epididymo-orchitis, treated with a 6-week course of oral ciprofloxacin with resolution on repeat testicular ultrasound.His past medical history includes bipolar disorder on lithium, methamphetamine use and sexually transmitted chlamydial infections. He does not smoke.A renal tract ultrasound 6-week post-admission detected an unexplained 14-mm left hydroureter and hydronephrosis. An uncomplicated prostatomegaly of 38 cc was noted. His estimated glomerular filtration rate had also dropped from 78 to 56.Computed tomography-intravenous pyelogram (CT-IVP) revealed a 1.7-cm intraluminal mass within the left distal ureter that extends beyond the ureteric lumen into peri-ureteric fat, with moderate left hydroureteronephrosis .He was readmitted for a left retrograde pyelogram, ureteroscopy and washings. No filling defect, or luminal lesion was noted intraoperatively, although the ureter was tortuous. Ureteric washings were inconclusive due to insufficient cells.Repeat CT-IVP was conducted 2 months following the initial CT-IVP. The mass had expanded to 1.9 cm with lobulated borders. Left hydroureteronephrosis persisted, however the distal left ureter remained patent. The unit X-ray meeting recommended a relook ureteroscopy, washings and biopsy. A Mag-3 Renogram with Frusemide washout revealed high grade left renal obstruction, and split function of left 41%, right 58%. A magnetic resonance imaging suggested the mass remained stable with no invasion.Case PD returned for repeat ureteroscopy, ureteric washings, biopsy and stent insertion. A large bladder volume was noted intraoperatively. There was no papillary lesion, with compression appearing to arise externally. Biopsy was smooth muscle actin positive on immunohistochemistry, and demonstrated a smooth muscle tumor with some atypia, consistent with grade 2 leiomyosarcoma .CT chest for staging revealed no metastases, but detected an incidental 5.1-cm dilated ascending aortic aneurysm.Case PD consented for a boari flap, psoas hitch, ureteric reimplant ± left nephroureterectomy. Following lithotomy and reflection of bowel, minimal mobilization was required to identify the ureteric mass located at the level of iliac bifurcation and appeared 2 cm in size. Good ureteric length was mobilized, and after excision of the mass, a tension free end-to-end anastomosis of ureter was performed without complication .Tumor histopathology confirmed a grade 3 leiomyosarcoma arising in the wall of the ureter and extending through the wall into the peri-ureteric connective tissue and to within 0.2 mm of the circumferential soft tissue margin. The ureteric margins were well clear .The patient recovered well post-operatively and was discharged home after removal of drain tubes and catheter. Repeat CT-IVP 5 months post-operation showed no evidence of tumor recurrence .Around 20 reports of primary leiomyosarcoma of the ureter are in the literature, with most tumors arising from the ureter being transitional cell carcinomas , 2. SmooNo clinical pattern has been associated with ureter leiomyosarcoma, however predominant presenting symptoms include flank or abdominal pain, urinary tract infection and palpable mass . As withEarly literature reviews describe a short survival of reported cases; a 1988 review noted five patients died within 2 years of the disease out of seven existing cases with detailed follow-up following surgery .Noting the aggressive nature of the condition, treatment of choice has been reported as total nephroureterectomy with en bloc resection of the tumor , 4. ContNo further cases of primary ureter leiomyosarcoma could be identified in the literature since the case reported by Shastri in 2016 to this date.It is important to report primary ureter leiomyosarcoma cases to assist further studies investigating the condition and the best approach to its treatment; in this case no malignant recurrence was observed 6 months following tumor resection and ureteroureterostomy.


Despite numerous reports on ischemic bowel obstruction caused by internal hernia, no case presentation has been reported of an internal hernia caused by a bridge formed between the medial and lateral zones of the liver. Herein, we report the first case of ischemic bowel obstruction caused by a hepatic bridge.A 24-year-old man complaining of abdominal pain was referred to our hospital and admitted. Computed tomography showed formation of a closed loop of small bowel with a hernia orifice near the hilar region, and poor contrast of the prolapsed small bowel. We suspected ischemic bowel obstruction caused by an internal hernia with a fissure of the greater omentum as the hernia orifice, and performed emergency surgery. Laparoscopic observation revealed that the medial and lateral segments of the liver formed a bridge on the dorsal side at the liver portal, and that the small intestine was ischemic in the gap created between the bridge and the medial and lateral liver segments. A Meckel’s diverticulum was also invaginated in the gap. The bridge was dissected out and the hernia orifice was opened to release the bowel obstruction. The small bowel was preserved and the Meckel’s diverticulum was resected. The patient’s postoperative course was uneventful.We experienced a case of ischemic bowel obstruction caused by hepatic bridge formation, which was successfully treated by laparoscopic surgery.The online version contains supplementary material available at 10.1186/s12876-022-02294-x. An internal hernia is defined as the protrusion of abdominal viscera, most commonly small bowel loops, through a peritoneal or mesenteric aperture into a compartment in the abdominal and pelvic cavity , 2. InteSteinke classified internal hernias into two types: fossa hernias, in which organs are inserted into the peritoneal fossa or scrotum of the abdominal cavity, and hiatal hernias, in which organs are inserted into an abnormal hiatus . The gapHerein, we present a novel case of an extremely rare type of primary visceral hernia that presented with ischemia of the small bowel and invagination of a Meckel’s diverticulum caused by formation of a bridge between the medial and lateral segments of the liver.A 24-year-old man with comorbidities of hypertension and atopic dermatitis and no history of abdominal surgery presented to the previous hospital with a chief complaint of upper abdominal pain. Blood tests and CT scan showed no abnormalities. Afterwards, his abdominal pain worsened and he visited our emergency room. He had localized tenderness in the upper abdomen but no signs of peritoneal irritation. The patient was admitted to hospital for follow-up because of severe abdominal pain. The next morning, a further increase in abdominal pain was observed. CT showed the formation of a closed loop of the small bowel with a hernia orifice near the hilar region and poor contrast of the prolapsed small bowel Fig. . We suspLaparoscopic observation revealed that the medial and lateral segments of the liver formed a bridge on the ventral side at the liver portal, and the small bowel was ischemic in the gap created between the bridge and medial and lateral liver segments Fig. . A MeckeThe patient had an uneventful postoperative course and was discharged on the fourth postoperative day. Histopathological examination of the Meckel’s diverticulum showed ectopic gastric mucosa without malignancy.We experienced a previously unreported type of small bowel obstruction caused by an internal hernia. Although preoperative imaging revealed findings characteristic of an internal hernia, we were unable to differentiate it from a hiatal hernia because of the anatomical similarity of the hernia orifice.Bridges between the lateral and medial segments of the liver are often experienced in routine surgery. In an intraoperative search of 104 patients, Sugarbaker et al. reported that 49% had a bridge between the parenchyma of the liver  . The gapLaparoscopic surgery for ischemic bowel obstruction was reported to be safe in patients with a stable general condition and a mild intestinal dilatation, which allows for a clear view of the abdominal cavity . Thus, iThe Meckel’s diverticulum was found incidentally in our patient during surgery, but was not considered to be a direct cause of the internal hernia. Most Meckel’s diverticula are asymptomatic, with only 6.4% of patients developing symptoms . AlthougIn summary, we experienced a case of ischemic bowel obstruction caused by hepatic bridge formation, which was successfully treated by laparoscopic surgery.Additional file 1. Video of laparoscopic surgery. The data is an edited video of the laparoscopic surgery, including the intra-abdominal view, release of the internal hernia, and opening of the hernia orifice.