Intravascular papillary hemangioendothelioma (IVPH) is a benign lesion previously reported in the nasal cavity, neck, upper extremities, and breast. Diagnosis with cross-sectional imaging can prove difficult, with histopathological examination necessary for diagnosis. IVPH resulting in carpal tunnel symptoms is quite rare.We report the case of a 37-year-old woman who presented with a radial, volar right wrist mass enlarging over the span of 5 years. She noted numbness and tingling in her wrist and thumb, exacerbated by minor accidental collisions and wrist hyperextension. There was no antecedent trauma. On examination, a mildly tender, mobile mass was evident at the volar aspect of the right wrist. Magnetic resonance imaging (MRI) with contrast demonstrated a lobulated, predominantly T2 hyperintense, heterogeneously enhancing mass thought to be a peripheral nerve sheath tumor. The patient elected for surgical excision of the mass, and the histopathological examination showed organizing thrombi with prominent papillary endothelial hyperplasia. At the 2-month follow-up, the patient had full range of motion of her fingers and wrist, with subjectively normal sensation in the distribution of the median nerve.Carpal tunnel syndrome, in exceedingly rare occasions, can result from an IVPH. MRI findings may be confused with more common entities. Histopathological confirmation remains necessary for conclusive diagnosis. Since its original description as a hemorrhoidal vein tumor, intravascular papillary hemangioendothelioma (IVPH) has been established as a benign lesion. It has been reported to be found in the nasal cavity, breast, neck, and hand –5. A rarIVPH can be challenging to diagnose both clinically and with advanced cross-sectional imaging. Histopathological analysis is necessary for a conclusive diagnosis. IVPH of the peripheral nervous system (PNS) can present with a variety of clinical syndromes depending on the affected peripheral nerve. It is known to share common imaging features with neurogenic tumors and tenosynovial giant cell tumors, with varying magnetic resonance imaging (MRI) characteristics for primary and secondary IVPH . The patThe patient is a 37-year-old right-hand dominant woman who first presented at an outside facility for examination of a radial, volar right wrist mass. She initially noted the hard mass in 2016. She experienced nerve-like pain, numbness, and tingling from her wrist to thumb with exercise. She denied any prior trauma. Electromyography did not reveal nerve damage. Magnetic resonance imaging (MRI) suggested a peripheral nerve sheath tumor. At the time, the patient declined surgical intervention as her symptoms did not greatly impede her daily life. She presented to us 3 years later, reporting interval growth of the mass and pain with hyperextension loading and minor accidental collisions.Focused examination of her right hand and wrist revealed a 3.0 × 2.0-cm mobile mass at the volar aspect of the right wrist, just beneath the palmaris longus tendon in the distal forearm, which was mildly tender to palpation. No subjective sensory deficits were noted to light touch. The patient had full strength, and no thenar atrophy was appreciated. There was a negative Tinel sign over the mass.Multiplanar multisequence MRI of the right wrist was obtained before and after 4.5 mL of an intravenous gadolinium-based contrast. This showed a lobulated, predominantly T2 hyperintense, heterogeneously enhancing mass measuring 2.0 cm transverse × 1.2 cm dorsal-volar × 3.1 cm in length along the volar-radial aspect of the distal forearm/wrist. T1-weighted sequences demonstrated the “split fat” and “tail sign” indicating the neural origin of the mass (Fig. The patient elected to undergo elective surgical removal of the symptomatic, enlarging mass. On the day of surgery, she was induced under general anesthesia, prepped, and draped in a typical sterile fashion. An extensive carpal tunnel approach was drawn, and an incision was made in the distal forearm ulnar to palmaris longus. After careful dissection of the superficial soft tissue to the volar forearm fascia with loupe magnification, the palmaris longus was radially retracted to expose the median nerve. A large, fragile, seemingly vascular mass was examined with the median nerve Fig. . Under mHistopathology of the specimen revealed multiple irregularly contoured vascular caverns lined by endothelial cells with dense fibrous connective tissue septae, features suggestive of a cavernous hemangioma Fig. . AdditioThe patient recovered well after her right median nerve release and extensive internal neurolysis with removal of the IVPH. At her 2-month follow-up, her wound was healing well and non-tender to touch. She had full range of motion of her fingers and wrist, and sensation in the median nerve distribution was subjectively normal.Historically, IVPH was commonly referred to as Masson’s vegetant hemangioendothelioma. First described by Masson to be neoplastic, it was later determined to be a benign composition of reactive thrombi encompassed by a proliferative papillary endothelium . IVPH acThere are several theories to the pathogenesis of this variant hemangioma. A prior study suggests the etiology may be hormonal, given its resemblance to other benign vascular masses . AnotherAlthough the composition of the tumor is benign in nature, IVPH must be differentiated from other soft tissue tumors. The differential diagnosis includes venous malformations, angiovenous malformations, pyogenic granulomas, schwannomas, neurofibromas, and Dabska tumors. In addition to the aforementioned differential considerations, key diagnoses to consider are angiosarcomas and Kaposi sarcomas, as both are malignant and aggressive in nature , 18. It MRI imaging characteristics are known to correlate histopathologically to help narrow the differential and offer a preliminary diagnosis. Venous malformations can be characterized as lobulated masses with intermediate to low signal on T1-weighted images, with increased intensity on T2-weighted images . AngioveFor a definitive diagnosis, histopathological examination is required. Histologic features characteristic to IVPH are papillary-like proliferation of the endothelium, proximity to a dilated vessel wall, lack of infiltration of adjacent soft tissue, and organized intraluminal thrombi formation . In retrWe report a rare case with radiologic-pathologic correlation, in which a carpal tunnel release and internal neurolysis performed for a soft tissue mass initially thought to be a peripheral nerve sheath tumor. Surgical resection and subsequent histopathological examination revealed intravascular papillary endothelial hyperplasia and findings consistent with IVPH.


Fasciola hepatica, is a neglected zoonotic food-borne trematodiasis. The Caspian littoral in northern Iran is endemic for the disease, and human fascioliasis is well-known in that region. In the present study, we report the diagnosis, identification, and clinical management of a human case of fascioliasis associated with common bile duct (CBD) obstruction from a non-endemic remote area in southeastern Iran.Fascioliasis, caused by Fasciola, and further molecular investigations, identified the flukes as F. hepatica using both pepck multiplex PCR and cox1 sequencing.A 42-year-old female was admitted to Afzalipour Medical Center hepatobiliary surgery ward in Kerman with abdominal pain for the past three months. Dilated biliary tract and an ill-defined mass in CBD were reported in abdominal ultrasonography and magnetic resonance cholangiopancreatography, respectively. During distal CBD operation, nine leaf-like motile flatworms were isolated. A morphological study confirmed all the isolates as Molecular and morphological findings of the study indicated the presence of human fascioliasis in the southeastern province of Sistan and Baluchestan in Iran. Fascioliasis is among the etiologies of chronic cholecystitis, and physicians should consider chronic cholecystitis associated with fascioliasis in the differential diagnosis. In the present report, endoscopic ultrasound was usefully applied for the accurate diagnosis of biliary fasciolosis.The online version contains supplementary material available at 10.1186/s12879-023-08310-z. Fasciola hepatica, and F. gigantica. Human and livestock infection occurs by ingestion of metacercariae during the consumption of contaminated raw vegetables or drinking water. In humans, maturation of metacercaria into the adult fluke takes approximately 3 to 4 months [Fascioliasis is a zoonotic food-borne parasitic disease, mainly caused by the liver fluke, 4 months . Metacer4 months .F. hepatica is rare and surgical intervention is recommended [Fascioliasis can cause a spectrum of clinical presentation in the acute and chronic phases of the infection including gastrointestinal symptoms, abdominal pain in the right upper quadrant, hepatomegaly, mild hepatitis, chronic cholecystitis, cholangitis, fatigue, anemia, pancreatitis, and weight loss. Biliary tract obstruction by ommended .Fasciola infection [Human fascioliasis is well-known in Iran after two major epidemics in 1989 and 1999 in the northern province of Gilan. Subsequently, World Health Organization (WHO) considered Iran among the six most infected countries with serious concerns about nfection . In Irannfection –7. Moder6/mm3, Hb: 8.7 g/dl, HCT: 28.5%, MCV: 78.3 fl.), and hypoglycemia (FBS = 77 mg/dl). Other serum factors like bilirubin, gamma-glutamyl transpeptidase (GGT), aspartate aminotransferase (AST), alanine aminotransferase (ALT), and alkaline phosphatase (ALP) were within normal range. A history of hypothyroidism was recorded in the patient hospital documents.A 42-year-old female with abdominal pain in the right upper quadrant (RUQ) for the past three months, was admitted to Afzalipour Medical Center hepatobiliary surgery ward in Kerman. The patient was a housewife living in rural areas of the southeastern province of Sistan and Baluchestan in Iran. Fig. a. She reIn abdominal ultrasonography, a dilated biliary tract was reported. Magnetic Resonance Cholangiopancreatography (MRCP) revealed dilated Common Bile Duct (CBD) and an ill-defined mass in CBD. Subsequently, Endoscopic Ultrasound (EUS) was performed by linear echo-endoscope. EUS reported a hyperechoic parenchyma in the liver seen without intrahepatic stone and normal left adrenal. CBD was dilated and measured at 15 mm and 8 mm in proximal and distal parts, respectively. CBD contained a large 14 mm in diameter mass lesion. Gallbladder had a lot of sludge and micro lithiasis. In the pancreas, dilated pancreatic duct was measured at 4 mm at the neck. The patient underwent laparotomy for CBD exploration. Cholecystectomy was performed. Distal CBD was opened up, and nine leaf-like motile flatworms were found with thick bile discharge.  were amplified from nuclear DNA by multiplex PCR assay , 9. The Fasciola isolates were identified as F. hepatica using both pepck multiplex PCR and cox1 sequencing. DNA sequence polymorphism analysis using DnaSP V.6.12.03 software revealed four haplotypes and five polymorphic sites in the 423 bp fragment among the nine sequenced isolates.For phylogenetic analysis, a 500 bp partial mitochondrial cytochrome c oxidase subunit 1 (cox1) gene fragment was amplified by Ita8: ACGTTGGATCATAAGCGTGT and Ita9: CCTCATCCAACATAACC . The PCRF. gigantica as the predominant Fasciola species, and the overall prevalence in humans and livestock in the region was reported as 0.3% and 20.1%, respectively [Fasciola infections [Fasciola species have been found throughout Sistan and Baluchestan province [Fasciola.Fascioliasis is common in many parts of the world, particularly in areas where traditional livestock husbandry is a common practice. In recent years the number of human fascioliasis has increased drastically with the estimates ranging from 2.4 million to 17 million people infected . Fasciolectively , 13. Anifections . Lymnaeiprovince , howeverFascioliasis comprises two main stages: acute or liver phase occurs for about eight weeks when immature juvenile worm migrate from the small intestine to the liver and sometimes other organs through the abdominal cavity. At this stage, the excretory-secretory products synthesized by the fluke are responsible for abdominal pain, anemia, urticaria, diarrhea, and hyper-eosinophilia. The chronic or biliary phase begins following the acute phase during which the juveniles develop into the adult worms and release eggs. , 17.Fasciola worm was found in his gall bladder, however no molecular investigation was performed to identify the helminth at the species level. The patient reported consuming fresh vegetables and no history of travel to the endemic regions of fascioliasis in Iran [Fasciola antibody in 2.4% of the subjects [Suaeda fruticosa), Sowzi (Cardaria draba), and Goshook  from the biliary tract [Right upper quadrant, fever, malaise, leukocytosis, weight loss, urticaria, anemia, epigastric pain, sweating, anorexia, jaundice, and hyper-eosinophilia are the most common presentations of human fascioliasis reported from sporadic cases from various parts of the country , 26. Fasreported . In modereported –32. The reported . Conventry tract , 34, 35.Fascioliasis is among the etiologies of chronic cholecystitis, and physicians should consider chronic cholecystitis associated with fascioliasis in the differential diagnosis. Ultrasonography is believed to be helpful in the diagnosis of biliary fasciolosis, however it may miss distal CBD lesions. EUS is an accurate tool for exploring the lower biliary tree. As described in other studies, the role of MRCP and EUS in the biliary fasciolosis diagnosis is documented in the present report.Below is the link to the electronic supplementary material.Supplementary Material 1


Adenomas are common pathologies of the salivary glands that are often associated with the major salivary glands and occur in the fourth to sixth decades of life. They are seldom seen in the pediatric age group and rarely in the minor salivary glands. Autoimmune sialadenosis of the minor salivary glands is a new phenomenon that has seldom been reported in the literature, with as few as three cases. Histopathological examination of the excised specimen is the definitive diagnosis, and these lesions have to be differentiated from adenomas and low-grade malignancies of the minor salivary glands. Management strategies of these lesions are extremely variable, ranging from wait-and-watch principle to the use of immunosuppressants and excision of the gland. This case report discusses the etiopathogenesis of the autoimmune sialadenosis and the management strategies. Benign salivary gland lesions can manifest as either sialadenitis or sialadenosis. Sialadenitis can be of either an acute viral infectious nature or a more chronic inflammation as a result of obstruction or stricture . On the Reports of sialadenosis have seldom been reported in the literature with most of them occurring in major salivary glands and in adults. However posterior palatal swellings originating from minor salivary glands can be of varied etiology like infections, autoimmune conditions, and neoplasms . Adults A 13-year-old male patient with fair systemic health and no known medical comorbidities presented to us with a complaint of swelling in the right posterior palate extending from first molar to third molar region  in individuals with IgE-mediated disease, which is influenced by genetic, racial, immunological, and environmental variables -12.PHIESD presents with an insidious onset in the pediatric age group of 7-14 years with women being more commonly affected. The presence of comorbid allergic condition has been reported to be among 46.7% of patients with hyper-IgE-related salivary gland disease. However, this incidence may be higher in patients with eosinophilic sialodochitis . Hence, Management of patients with PHIESD is an area of concern as minimal evidence is available in the literature. Very few reports of PHIESD have been reported in the literature, with most of them being reported bilaterally in major salivary glands with inconclusive evidence on management . The optInterventions for these lesions, especially in children, often lean towards a conservative approach. The recommended method, with utmost caution, is to adopt a wait-and-watch strategy. Self-care measures such as daily glandular massage, sialagogues, and chewing gum can help alleviate salivary gland distension. Allergen testing, anti-allergic therapy, and avoidance of the allergens may be helpful if known allergens are established. Immune modulator therapy is advisable in patients with multi-system involvement as reported by Baer et al. .Management of isolated minor salivary gland PHIESD has not been reported in the literature so far. Though wait-and-watch appears to be an ideal option considering the pediatric age group, extreme caution on regular follow-up needs to be emphasized in these cases as the probability of low-grade mucoepidermoid carcinoma or adenoid cystic carcinoma cannot be ruled out, especially in the palatal minor salivary glands . ExcisioThe exact nature of the disease, its underlying mechanisms, and the optimal management approach are yet to be fully determined. Existing literature on this condition is scarce, reflecting the limited knowledge available. Recurrence after excision of the lesion has seldom been reported. Van de Veen W et al. in their series had a follow-up between 12 and 68 months post-excision and reported no recurrence, and no regression was noted in unoperated cases. Anti-allergic treatment was associated with recurrence, warranting long-term medical management . HoweverPHIESD represents a relatively recent pathological entity affecting the salivary glands. Furthermore, the occurrence of this disease specifically in the minor salivary glands of the palate has not been previously documented. In the absence of established guidelines, surgical excision of the affected salivary glands has emerged as an effective treatment option. This approach has shown positive outcomes in managing PHIESD, though further research is needed to ascertain its long-term efficacy and potential alternative therapeutic strategies. Further research endeavors and clinical investigations are warranted to shed light on the optimal management strategies for PHIESD and advance the understanding of this intriguing pathological condition.


Xanthogranulomatous inflammation (XGI) is a rare, benign condition that can affect several organs, including the gallbladder, kidney, skin, gastrointestinal tract, lymph nodes, and soft tissues. It is often misdiagnosed as a malignancy. In this report, we present the case of a 79-year-old male who presented with persistent jaundice for 11 months. Computed tomography and magnetic resonance imaging revealed pancreatic head enlargement, gallbladder thickening, and common bile duct thickening, leading to a preoperative diagnosis of malignant neoplasm of the pancreatic head. During surgery, dense adhesions were found around the portal vein, suggestive of mass invasion. To relieve obstruction, choledochojejunostomy was performed. Postoperative pathological examination revealed xanthogranulomatous cholecystitis (XGCc), xanthogranulomatous cholangitis (XGCg), and xanthogranulomatous pancreatitis (XGP). XGI affecting the bile ducts and pancreas is extremely rare, and there are no reported cases of simultaneous involvement of the gallbladder, bile duct, and pancreas by XGI. This study provides valuable insight into the differential diagnosis of XGI by presenting the imaging features of XGI patients. XGI 2A 79-year-old male was admitted to our hospital with abdominal pain, jaundice and weight loss. No obvious abnormality was found in the physical examination of the patient. Laboratory results: carbohydrate antigen 19-9 (CA19-9)>400 U/ml, total bilirubin (TBIL) 267.1 μmol/L . Other laboratory test results were within the normal range. Because the patient had severe jaundice, he first underwent percutaneous transhepatic cholangio drainage. One month later, he was hospitalized again. Laboratory results: CA19-9 125.2 U/ml, TBIL 96.3 μmol/L. Contrast-enhanced computed tomography (CT) showed that the common bile duct was thickened  can be found in the article/supplementary material.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.JZ and HL are responsible for the writing of this article, and XL collected patient data. QG, PW, XY, and BL for help with reviewing previous articles. SS was responsible for the final review of the article. All authors contributed to the article and approved the submitted version.


Groove pancreatitis (GP) is a chronic type of pancreatitis involving the groove area between the head of the pancreas, the duodenum, and the common bile duct. Alcohol abuse is one of the main pathogenetic factors, although its etiology is not clearly defined. Differential diagnosis of pancreatic disorders remains difficult. The lack of diagnostic management and the restrictive number of patients are the main barriers. This article presents a case of a 37-year-old male diagnosed with GP after several episodes of epigastric pain and vomiting, with a history of chronic alcohol consumption. The patient’s radiological and laboratory results excluded the possibility of malignancy and suggested the diagnosis of groove pancreatitis with duodenal stenosis. After initial conservative treatment failed, surgical management was decided. A gastroenteroanastomosis was made in order to bypass the duodenum aiming for a total resolution of the symptoms and an uneventful recovery of the patient. Although most studies suggest pancreatoduodenectomy (Whipple’s procedure) as the treatment of choice, a less major procedure can be performed in evidence of malignancy absence. Groove pancreatitis (GP), also known as paraduodenal pancreatitis, is a form of chronic disease located in the pancreaticoduodenal groove, defined by the head of the pancreas, the duodenum, and the common bile duct. It is categorized into two forms: pure and segmental. Chronic alcohol consumption and heavy smoking seem to have a strong connection with GP’s pathogenesis, which is not clearly determined yet . GP usuaA 37-year-old male patient presented to the emergency department complaining of postprandial epigastric pain radiating to the back and vomiting episodes relating to food intake over the last fourteen days. Furthermore, he reports recent weight loss and weakness because of his inability to feed due to the severe symptoms mentioned above. The patient has a history of chronic alcohol consumption and heavy smoking over the past 10 years, and it was the only one mentioned from the patient’s medical history. The physical examination showed tenderness in the upper abdomen and the right hypochondric region. According to his laboratory exams, serum amylase was within the normal range, and the level of lipase was 90 U/L . Tumor markers, CA 19.9, and carcinoembryonic antigen (CEA) were found normal. An abdominal Computed Tomography (CT) was performed. It revealed a thickening of the pyloric and the duodenal wall and a 26 mm hypodense mass at the level of the pancreatic head visible in the arterial phase. An upper GI endoscopy was then conducted, which showed luminal stenosis, edema, and redness of the duodenum. In a second CT performed some days later, the pancreatic head appeared enlarged and with fuzzy limits. In addition, cystic lesions (maximum diameter of 12 mm) and fibrous tissue were found in the head of the pancreas near the duodenal wall . Based oGroove pancreatitis (GP), also known as paraduodenal pancreatitis, is a form of chronic pancreatitis located in the pancreaticoduodenal groove . The groIn 1973, Becker was the first to describe a type of chronic focal pancreatitis located in the pancreatoduodenal area, with the German name of “Rinnen pancreatitis” , and theThe pathogenesis of GP is not yet precisely determined, although there seems to be a strong correlation between chronic alcohol consumption and heavy smoking . More prPatients diagnosed with GP are mostly alcoholic men in their fourth or fifth decade of life . The cliRegarding the diagnostic imaging findings of GP, several methods have been used through the years, although their diagnostic management remains challenging. Contrast-enhanced abdominal computed tomography (CT) shows a hypodense poorly enhanced lesion between the pancreatic head and the duodenal wall. Cysts can be found in the groove area or/and the duodenal wall, which is also thickened, causing duodenal stenosis ,17,18. TPancreatic head carcinoma, periampullary cancer, pancreatic groove neuroendocrine tumor, cystic dystrophy of the duodenum, and plastron presence in acute pancreatitis are included in the differential diagnoses of GP ,18,22. DThe treatment options of GP are conservative measures and surgical intervention. As regards the conservative treatment, it aims to substitute the pancreatic function with a pancreatic enzyme supplement and relieve the patient’s pain with sufficient analgesia . Other mGroove Pancreatitis is a very rare type of chronic pancreatitis. Patients may present with non-typical symptoms, such as abdominal epigastrium pain, vomiting, and weight loss. Diagnosis of GP poses a challenge to clinicians due to its similarities with pancreatic carcinoma, which sometimes leads to unnecessary over-treatment of patients undergoing extreme interventions that add morbidity or even mortality to the patients. In all cases of pancreatic head abnormalities and duodenal stenosis, suspicion of GP should be raised, primarily in cases where the patient’s history is associated with prolonged alcohol intake. Initial treatment includes conservative measures or less invasive endoscopic techniques. In the failure of the previously mentioned options, surgery is the management of choice. Although, further diagnostic criteria should be established in order to facilitate the early diagnosis and the targeted treatment of patients.


This study discusses a case of coexistence of two distinct cysts, a nasopalatine duct cyst (NPDC) and a radicular cyst, within the anterior region of the maxilla. NPDC is a prevalent non-odontogenic developmental cyst, while radicular cysts are commonly found in odontogenic inflammatory cysts. The clinical and radiographic characteristics of these cysts are explored, emphasizing the importance of accurate diagnosis and treatment planning. In this case, a 51-year-old male patient presented with swelling and pain in the maxillary anterior region. Radiographic examinations revealed a heart-shaped radiolucent lesion extending from tooth 13 to 23, associated with the NPDC, and a separate radicular cyst. Surgical enucleation and tooth extraction were performed as the treatment of choice. This unique case underscores the significance of meticulous radiographic assessment to detect multiple cystic lesions within the same area. The nasopalatine duct cyst (NPDC) is one of the most common non-odontogenic developmental cysts known as incisive canal cysts -4. MeanwFurthermore, NPDC can be developed at any age but is more common in the fourth to sixth decades of life, and in the case of radicular cysts, it is more frequent between the second and fifth decades of life with more incidence in the male gender for both lesions ,4,7-9.According to some literature works, the cyst clinically appears as swelling, usually asymptomatic in the anterior region of the palate, which is detected by routine clinical check-ups or radiographs -3,6,10. In the case of radicular cysts, although they could be asymptomatic and discovered by periapical radiograph while diagnosing a non-vital tooth, the peri-radicular inflammation can lead to many symptoms, such as swelling, pain, abscess, or intraoral or extraoral fistula .Radiographically, NPDCs show a well-defined rounded or heart-shaped radiolucent area surrounded by a sclerotic margin. Moreover, the intact status of lamina dura around the nearby teeth apices was confirmed; however, according to Allard et al., resorption of maxillary incisors root might be seen in rare cases -3. Even Through the histopathological examination, NPDCs appear as a cavity with an epithelial lining surrounded by a connective tissue wall. Moreover, the presence of mucous glands and neurovascular bundles, in addition to variable types of the epithelium, can be seen, such as columnar, cuboidal, squamous, or even respiratory type of epithelium, depending on the proximity to the nasal cavity ,3,8. By The standard treatment modality of NPDCs is surgical by enucleation, which is indicated for cases with clinical symptoms that interfere with their quality of life or for diagnostic purposes in the case of extensive lesions ,3. MeanwThis case report presents a case of the coexistence of two distinct cysts with different origins and pathogenesis in the anterior maxilla.History and examinationA 51-year-old male patient came to the dental clinic with a chief complaint, “I want to repair my anterior bridge,” with no significant findings regarding medical and social history; however, the patient had a history of trauma on the upper anterior area due to a car accident 10 years ago, which led to multiple extractions and fabrication of the upper anterior bridge from the upper right canine to the upper left canine. Furthermore, the patient complained of pain and swelling that started several weeks ago in the maxillary anterior teeth region  shows a well-defined heart-shaped radiolucent lesion extending from tooth #13, crossing the midline to tooth #23 with loss of the continuity of lamina dura of the teeth related to the lesion Figure . Three-dIncisional biopsyFurther investigation was needed, so an incisional biopsy under local anesthesia from the labial vestibule in the apical area of the upper left canine was done, and the specimen was sent for histopathological examination. It shows dense fibrous connective tissue with chronic inflammatory infiltrates containing predominantly lymphocytes and plasma cells. Moreover, there are few areas of hemorrhage and small blood vessels. The diagnosis was a periapical cyst based on the histopathological findings.ManagementThe preferred therapeutic intervention involved surgical enucleation of the cyst, coupled with the extraction of the nonviable upper left lateral incisor and canine. Under local anesthesia, by performing bilateral greater palatine nerve blocks and bilateral anterior superior alveolar nerve blocks, the previously placed defective anterior bridge was removed. Subsequent to this, the patient was directed to rinse with a chlorhexidine mouthwash. Both crestal and sulcular incisions were executed, accompanied by two vertical releasing incisions. A periosteal elevator was utilized to reflect the flap. The cystic wall was then carefully dissected from both the labial mucosa and the nasal floor with sharp scissors. The cyst was enucleated and preserved in formalin for histopathological evaluation. The compromised teeth were then extracted. Hemostasis was achieved, followed by the nose-blowing test to confirm the integrity of the nasal floor. The surgical site was sutured using a 4-0 polyglactin absorbable suture.Histopathological assessment of the removed lesion unveiled a cystic structure. This was lined by ciliated pseudostratified columnar epithelium. The underlying connective tissue was characterized by fibroblasts, collagen fibers, vasculature, and nerve fibers. Some regions also exhibited hemorrhage. Based on these findings, the final diagnosis was an NPDC. The aforementioned surgical procedures and the histopathological specimen are represented in Figures A meticulous re-evaluation of the periapical radiographs and CBCT revealed the presence of not one, but two cystic structures, as illustrated in Figure The oral cavity, with its complex embryonic development, frequently presents various pathologies. An NPDC emerges as the prevailing non-odontogenic developmental cyst, originating from the remnants of the nasopalatine duct. Radiographically, NPDCs often display a distinctive heart-shaped radiolucency situated between the central incisors . By contIn managing the unique concomitant occurrence of the NPDC and radicular cyst, an integrated approach is imperative. Initial diagnostic steps include comprehensive radiographic examinations, where panoramic views offer a preliminary understanding while advanced modalities, such as CBCT, provide detailed insights into the cysts' size, boundaries, and relationship to adjacent vital structures . Post-suThe simultaneous presence of an NPDC and a radicular cyst, in this case, serves as a testament to the unpredictable nature of oral pathologies. This unprecedented co-occurrence not only challenges the conventional understanding of cystic development but also underscores the importance of comprehensive diagnostic measures and meticulous treatment planning. The rarity of such a contiguity reinforces the importance of continual learning and adaptability in oral pathology diagnostics. As the field continues to evolve, such unique cases emphasize the importance of a holistic approach to patient care, ensuring that both surgical interventions and post-operative monitoring are conducted with utmost precision and diligence.


Odontomas are the most common odontogenic neoplasms. They are generally small and asymptomatic. This article presents an unusual case of a giant maxillary complex odontoma, which obscured a part of the maxillary antrum and impacted a tooth. This was discovered during an episode of maxillofacial cellulitis. In this case, surgical excision of the lesion was performed under general anesthesia, and the closure was performed with a fat pad pedicled flap. A brief review of the literature was performed to analyze the characteristics of this clinical entity and their implication in the treatment. An odontoma is currently considered as a hamartoma because it is highly differentiated. It is a mixed odontogenic benign tumor because it is both epithelial and mesenchymal in origin . AccordiThe aim of this report is to describe a case of giant maxillary intrasinusal complex odontoma revealed by cellulitis and to describe the specifics of treatment resulting from this localization. We have also analyzed similar cases that have been reported in the literature and discussed the importance of imaging.A 27-year-old patient with no allergy presented with right posterior maxillary severe pain . He had no medical or surgical history. No radiological examination was prescribed at the time.On the day of the consultation, facial examination revealed a swelling in the right upper jaw, which was firm, non-indurated, and without eyelid edema. Endobuccal examination revealed a fluctuating swelling from distal to tooth 16 associated with exposure of hard tissue of inhomogeneous appearance extending 2 cm up to the right maxillary tuberosity. The panoramic radiograph revealed a heterogeneous radiopaque image of about 4 cm in its long axis, well delimited by a radiolucent halo  . Indeed,Four relevant articles on the subject were reviewed, which contained four case reports of giant maxillary complex odontomas -10. We hThe clinical presentation of this lesion type is highly variable, depending on the patient. Exo-buccal swelling may or may not be present. During an endobuccal examination, delayed tooth eruption and a buccal exposure of the lesion with an inhomogeneous appearance may be observed.Dental panoramic imaging reveals an inhomogeneous radio-opaque mass. Further analysis must be conducted using CT scans to precisely determine the appearance, extent, and relationship to surrounding structures and to establish a definitive diagnosis and rule out differential diagnoses such as calcifying odontogenic cyst, calcifying odontogenic tumor, ameloblastic fibroma, fibro-odontoma, and fibro-osseous lesion. Furthermore, a biopsy and an anatomopathological examination must be performed. Prior to the removal of a maxillary giant odontoma, a comprehensive treatment plan with oral rehabilitation solutions is necessary.Some authors have shown that an odontoma, giant or not, can interfere with eruption of the adjacent teeth, causing impaction . This reThe etiology of this disease is still unknown, but many factors have been considered to play an important role in its pathogenesis, such as local trauma, infection, genetics, or a family history of conditions such as Gardner syndrome and Hermann syndrome ,7.The 3D-CT examination is important for the correct diagnosis of the expansion and involvement of the tumor. The exact position of all involved structures  was verified by 3D-CT, allowing for the best planning of the surgical intervention . To our Through this article, we understand the importance of systematic complementary examinations  to avoid diagnostic errancy particularly when faced with eruption anomalies. Although these are benign tumors, their removal can be very mutilating and should be combined with other surgical techniques such as closure of the buccal-sinusal communication with a fat pad pedicled flap in this case. When the damage to the adjacent teeth and surrounding bone is severe, complex oral rehabilitation is required; hence, multidisciplinary therapeutic strategies are important.


Benign fibro-osseous lesions are a group of pathological conditions characterized by the replacement of normal bone with cellular fibrous connective tissue that undergoes mineralization. The most common types of benign fibro-osseous lesions include fibrous dysplasia, ossifying fibroma, and osseous dysplasia. However, diagnosing these lesions can be challenging due to their overlapping clinical, radiological, and histological features, which can cause a diagnostic dilemma for surgeons, radiologists, and pathologists. One rare type of benign fibro-osseous lesion is the cemento-ossifying fibroma (COF), which is a definitive form of a benign fibro-osseous tumor that affects the craniofacial region, particularly the jaws (70%). Here, we present a case of COF in a 61-year-old female patient in the maxillary anterior region. Due to a clear distinction between the lesion and healthy bone, the lesion was treated with conservative surgical excision followed by curettage and primary closure. However, differential diagnosis of COF can be highly challenging for clinicians due to its overlapping features with other fibro-osseous lesions like Paget’s disease and fibrous dysplasia. Ossifying fibroma and fibrous dysplasia often present a histopathological, clinical, and radiological overlap. The post-operative follow-up after eight months was unpredictable, with a radiological picture showing the increased thickness of the frontal bone, parietal bone, and maxilla with obliteration of marrow spaces, alteration of the trabecular pattern with a cotton wool/ground glass appearance, and reduced maxillary sinus space. Proper evaluation and diagnosis of fibro-osseous lesions are necessary before arriving at a final conclusion. Cemento-ossifying fibroma in the maxillofacial skeleton is uncommon, and after eight months, the recurrence rate is rare. This case highlights the importance of considering COF as a differential diagnosis for fibro-osseous lesions in the maxillofacial region and the necessity for proper evaluation and diagnosis to determine the appropriate treatment plan and prognosis. In summary, the diagnosis of benign fibro-osseous lesions can be challenging due to their overlapping features, but early diagnosis and proper evaluation are essential for successful treatment outcomes. COF is a rare type of benign fibro-osseous lesion where other fibro-osseous lesions in the maxillofacial region should be considered as a differential diagnosis, and the necessary steps should be taken to confirm the diagnosis before arriving at a final conclusion. The fibro-osseous lesions of bone comprise a heterogeneous group of disorders that include developmental, neoplastic, or reactive (dysplastic) lesions characterized by the replacement of normal bone by fibrous tissue that acquires mineralization. They encompass two major lesions: fibrous dysplasia and ossifying fibroma. Cemento-ossifying fibroma (COF) is a definite form of benign fibro-osseous tumor affecting the craniofacial region (70%), principally in the jaws ,2. Most A 61-year-old female patient reported to the Department of Oral and Maxillofacial Surgery at Saveetha Dental College, Chennai, Tamil Nadu. The patient's chief complaint was swelling over the upper front teeth region for the past five years. History revealed that the swelling was first noticed five years ago and had been causing mobility of the tooth for the past week (Figure Radiographic investigations were taken, and the orthopantomogram Figure  showed aSurgical techniqueUnder general anesthesia, all mobile teeth were extracted and a standard crevicular incision was made. The mucoperiosteal flap was raised, and there was a clear distinction between the lesion and healthy bone. The mass was excised Figure  and sentThe histopathological picture Figure  shows a Regular follow-up was done for eight months, and complete healing had taken place with no recurrence Figure . The ortCemento-ossifying fibroma is a benign fibro-osseous tumor that is thought to have various controversies about its origin. It is believed to arise from the mesenchymal blast cells of the periodontal ligament and is composed of cementum, bone, and fibrous tissue ,7. CliniThe differential diagnosis of this could be highly challenging for the clinician due to the overlap between various fibro-osseous lesions, like Paget's disease, and fibrous dysplasia. Fibrous dysplasia is a benign fibro-osseous lesion that is also characterized by the replacement of normal bone by cellular fibrous connective tissue, leading to the formation of fibrous tissue and bone. Fibrous dysplasia and COF can have similar radiographic and histological features, making it difficult to differentiate between the two. However, fibrous dysplasia usually presents as a diffuse lesion involving the entire bone, whereas COF is usually a well-defined, localized lesion. On the radiological picture, fibrous dysplasia appears as a poorly differentiated lesion with a ground glass attenuation, contrary to cemento-ossifying fibroma. There have been reports in the literature of histological and radiological overlap between ossifying fibroma and fibrous dysplasia . SteinerRadiographically, Paget's disease appears with a cotton-wool-like appearance and cortical thickening, while histologically, it displays disorganized and woven bone. Paget's disease may present with mixed radiolucent-radiopaque lesions; histological examination of the tissue can help differentiate between the two conditions. Paget's disease is typically a diffuse process that affects multiple bones, while cemento-ossifying fibroma is usually localized to a single bone ,10. ReduThe treatment for COF could be complete excision and curettage. There are cases of cemento-ossifying fibromas that have been present for more than 30 years without any trouble or exacerbation . In largIn conclusion, cemento-ossifying fibroma in the maxillofacial region, as presented in this report, is uncommon after eight months, and the differential diagnosis is quite challenging. The diagnosis of benign fibro-osseous lesions is highly challenging as they overlap in clinical, radiological, and histological presentations, thereby providing a diagnostic dilemma for surgeons, pathologists, and radiologists as the treatment plan and prognosis differ in each scenario. Clinical, radiological, and histopathological examinations should be carefully correlated before arriving at the final diagnosis, and adequate treatment protocols should be decided accordingly.


Laparoscopic cholecystectomy with intraoperative cholangiogram is commonly performed, especially when there is suspicion of choledocholithiasis. We present a case of acute pancreatitis post-procedure for management of acute cholecystitis and suspicion of distal common bile duct sludge, potentially caused by the dislodgement of microlithiasis, which resolved on its own with supportive management. This case emphasizes the need to consider acute pancreatitis as a rare but possible post-cholecystectomy complication in a patient who continues to be unwell post-operatively. Acute calculus cholecystitis has a high incidence in the general population. The prevalence of gallstones in the general population is 10-15% . The sta9/L, with neutrophilia of 8.23 × 109/L. C-reactive protein (CRP) was high at 145 mg/L. Serum lipase was normal at 139 (reference range: 20-210). She was started on intravenous ceftriaxone and metronidazole and was further investigated with another abdominal ultrasound on the day of admission. It showed a thickened gallbladder wall of 5 mm with pericholecystic fluid, but no gallstones were seen. CBD measured 7 mm, similar to the previous. The following day, total bilirubin climbed to 45 µmol/L, and therefore, magnetic resonance cholangiopancreatography (MRCP) was performed, which again demonstrated a thickened gallbladder wall with pericholecystic fluid and was also suspicious for biliary sludge in the distal CBD  was 8 mm. Her liver function tests (LFTs) were normal then. She has a history of sleeve gastrectomy and, later, a mini gastric bypass three years ago for weight loss. Her observations were normal; she had right upper quadrant tenderness and a positive Murphy’s sign on examination. Laboratory examination showed deranged LFTs with a total bilirubin of 20 µmol/L, alanine aminotransferase (ALT) of 168 U/L, alkaline phosphatase (ALP) of 222 U/L, and gamma-glutamyl transferase of 166 U/L. The white blood cell count was raised to 11.38 × 10As she had cholecystitis, she then underwent laparoscopic cholecystectomy a few hours after MRCP, with a plan for trans-cystic bile duct exploration. However, the intraoperative cholangiogram showed free flow into the duodenum without any filling defects Figure , and theIntraoperative cholangiogram (IOC) is commonly performed during cholecystectomy, especially when there’s a moderate-to-high risk of a common bile duct stone. It involves inserting a 4-6 Fr catheter  into the cystic duct and injecting contrast; at our center, we use Urografin®  through a 20-ml syringe flushed manually. While it is a common procedure often performed along with laparoscopic cholecystectomy, there have been no instances of pancreatitis as a complication reported in the literature. In a retrospective study by Morgan et al. with 435 patients who underwent IOC, three patients developed pancreatitis, two who underwent ERCP for choledocholithiasis, which pancreatitis was attributed to, and one who had hyperamylasemia seven days post-operatively and was diagnosed with papillary stenosis and microlithiasis during ERCP. The study concluded that IOC was not associated with pancreatitis [Sphincter of Oddi stenosis (SOS) is defined by an abnormal narrowing caused by inflammation or scarring, such as pancreatitis, passage of gallstones, iatrogenic injury, infection, and adenomyosis. It is diagnosed with an elevated basal pressure of >40 mm Hg. SOS has been implicated in recurrent acute idiopathic pancreatitis in up to 20% of patients . While tAnother potential cause of acute pancreatitis is microlithiasis. In patients with recurrent pancreatitis without an obvious cause, microlithiasis is found in 13-70% of patients through bile salt analysis or endoscopic ultrasound (EUS) -8. It isWe report a case of acute pancreatitis post-intra-operative cholangiogram on a background of sludge in the distal common bile duct. While it is unlikely that the cholangiogram on its own caused pancreatitis, surgeons should keep into consideration pancreatitis as a diagnosis in a patient who continues to be unwell after what seemed like a definitive intervention, where interrogation of the biliary tract is involved, especially when there is a history of common bile duct calculi or sludge. It could possibly be caused by papillitis from the passage of calculi or sludge, or turbulence during the flushing of contrast leading to the reflux of sludge into the pancreatic duct.


Burkitt’s lymphoma (BL), an aggressive kind of non-Hodgkin’s B-cell lymphoma. On the other hand, appendiceal carcinoid tumors are uncommon neuroendocrine neoplasms.The authors report a case of a 15-year-old Syrian adolescent who was admitted to our hospital due to a persistent, severe generalized abdomen pain accompanied by nausea, vomiting, loss of appetite, and inability to pass stool or gas. An abdominal radiograph revealed dilated intestinal loops with air-fluid levels. The patient underwent emergency surgery through which a retroperitoneal mass was removed as well as part of the ileum and the appendix. The final diagnosis was consistent with intestinal BL associated with an appendiceal carcinoid tumor.The correlation between gastrointestinal carcinoids and other types of tumors was frequently reported. However, there have been few reports of carcinoid tumors being associated with lymphoreticular system cancers. BLs were classified into three variants: endemic, sporadic, and acquired immunodeficiency-associated BL while appendiceal neuroendocrine tumors were classified as the following: well-differentiated neuroendocrine tumors with benign or uncertain malignant potential; well-differentiated neuroendocrine carcinoma with low malignant potential; and mixed exocrine-neuroendocrine carcinoma.Our article demonstrates an unusual association between BL and an appendiceal carcinoid tumor that highlights the significant role of histological and immunohistochemical staining in confirming the diagnosis, as well as the role of surgery in treating the complications of intestinal BLs. HighlightsBurkitt’s lymphoma is an aggressive kind of non-Hodgkin’s B-cell lymphoma.Appendiceal carcinoid tumors are uncommon benign neuroendocrine neoplasms.There have been few reports of carcinoid tumors being associated with lymphoreticular system cancers.Chemotherapy is the mainstay of Burkitt’s lymphoma.Surgery is the main treatment of appendiceal carcinoid tumor.Burkitt’s lymphoma (BL), an aggressive kind of non-Hodgkin’s B-cell lymphoma, typically identified in children and young adults. It has three clinical-epidemiological variants: endemic, sporadic, and acquired immunodeficiency-associated BL9 /l) and elevated levels of C-reactive protein , otherwise normal.We report a case of a 15-year-old Syrian adolescent who was admitted to our hospital due to a persistent, severe generalized abdomen pain accompanied by nausea, vomiting, loss of appetite, and inability to pass stool or gas. Family history was unremarkable as well as surgical, drug, and allergic histories. Physical examination revealed a soft, tender abdomen without guarding. Lab findings were significant for leukocytosis : Not applicable.Not commissioned, externally peer-reviewed.


Lipoma is a common soft tissue tumour in the human body, but at the same time is very rare in the palm and rarer still in the thenar region. These lipomas in the hand can give rise to various problems such as cosmetic, functional, and neurological compromise among others and removing them becomes important when symptomatic. Diagnosing a hand pathology becomes important as a missed diagnosis can have long-term functional consequences for a patient. In the case report, we discuss a hand palmar prominence which presented as an effusion and later turned out to be a large lipoma. Further, we also present a literature review of published thenar lipoma cases to throw light on the nuances of this rare pathology location, which, to our knowledge, has not been done comprehensively. The World Health Organization classifies soft tissue tumours into lipomatosis, lipomatosis of nerve, angiomyolipoma, lipoblastoma, myolipoma, chondroid lipoma, spindle cell lipoma, hibernoma, and benign simple lipoma . Soft tiLipomas in the hand are a rare occurrence, with thenar eminence subcutaneous region being the most common when it occurs. A subfascial lipoma (deep variety) can also occur in the hand but is generally pushed to the side due to the unyielding nature of aponeurosis . We presA 32-year-old, right-hand dominant white Caucasian male working as a manual labourer presented to the clinic with the main complaint of cosmetic disfigurement and functional impairment due to a palmar tumefaction in the left hand, which had been standing for a few months and progressively increasing. On examination, it was a painless prominence on the thenar eminence which felt soft with diffuse borders. There was no nerve involvement in the hand, verified by checking the dermatomes of the radial, median and ulnar nerves in the hand. There was reduced function due to a mass effect in terms of weak palmar grip and pinch.It was labelled as an effusion or a ganglion in the first instance with further differentials of an infected cyst, soft tissue tumour, and foreign body granuloma. Magnetic resonance imaging (MRI) was obtained due to its higher sensitivity which showed a 5.8 × 2.5 × 1.8 cm lipoma within the flexor carpi radialis muscle (intramuscular) but separate from flexor digital tendons (Figure It was excised uneventfully by an upper limb surgeon under a regional block. The lipoma was found to be arising from within the thenar muscles and was well encapsulated. Histological examination of the excised mass confirmed it to be a lipoma with benign characteristics. The surgical site healed well with full hand function retained by the patient, as checked by the recovered grip and pincer strength. The patient was followed up for six months and eventually discharged with no further recurrence or problems. Figure The first documented hand lipoma case report dates to 1971 when Phalen et al. published a report of 15 hand and wrist lipomas. They did not find any malignancy in any of the cases but almost 25% had a neurological compromise . AnotherThe presence of thick unyielding palmar fascia is believed to cause the larger lipomas to present peripherally on the palm and along the carpal tunnel, and at the same time, the palm due to its unique anatomy and gap under the palmar fascia gives rise to deceptively larger lipomas than other sites ,9. WhileIn the days when MRI was not widely prevalent, a study in 1991 found ultrasound a useful investigation for delineating lipomas from other tumours and foreign bodies. The study concluded that an experienced sonographer was the right person to perform these studies for the results to be reproducible and reliable . Later, Surgical excision is warranted and should be offered if the lipoma is greater than 5 cm, has a functional restriction, muscle dysfunction, reduced strength, triggering, pain, or paresthesia . A lipomThere is a suggestion that a trained hand surgeon operates on benign cases, and in cases of suspicion of malignancy, a cancer surgeon along with a hand surgeon should be present ,15,26. IInterestingly, one case report from 2007 and a publication from 1972 mentioned that the lipoma was seen to erode the metacarpal, which is an unusual presentation, as lipomas do not affect the bone; further, there was full recovery post-excision ,31. AparThe first line of investigation in such cases is usually an ultrasound due to ready availability but is not immune to interoperator variability and difficulty in defining borders; hence, the challenge of differentiating it from other soft tissue masses . Where pGoing through the published literature on palmar and specifically thenar lipomas, it was found that only a handful of thenar lipomas have been published Table . Among tPalmar lipomas are a very rare entity and very few have been reported in the past. Even rarer are lipomas in the thenar eminence, as in this case. Missing a diagnosis in the palm can have serious complications with hand function, as lipomas in the palm can deceptively present with clinical features intermingling with other lesions. There was no associated pain but only cosmetic and functional impairment, with MRI being the investigation of choice to diagnose and determine the type and relations to safely excise the lesion. Surgical excision followed by histologic analysis was the final management which led to good recovery and full function. Reviewing the previously reported thenar lipoma cases gives us a good insight into the different patterns of presentation and factors to keep in mind while managing them.


Heart tumors are rare in dogs. They can be benign or malignant. Clinical signs depend primarily on the location of the tumor and its effect on blood flow.An eleven-year-old crossbreed male dog lethargic and anorectic for previous 3 days was presented to the veterinary clinic. The focused ultrasound assessment with sonograms in trauma (FAST) revealed multiple tumors in the heart which were then confirmed in echocardiographic examination performed by a veterinary cardiologist. Due to the poor general condition and grave prognosis, the dog was humanely euthanized. The autopsy revealed numerous intracardiac tumors in all four heart chambers. No proliferative changes were found in other organs either in thoracic or abdominal cavity. Immunohistochemical examination was performed using formalin-fixed, paraffin-embedded tissue from heart masses. The antibodies against myoglobin, desmin, smooth muscle actin, vimentin, CD34, S100, and pan-cytokeratin (AE1/AE3) were used. Microscopically, the tumor was composed of fascicles of spindle-shaped cells with pale eosinophilic cytoplasm with round, oval, and focally elongated nuclei and one or two prominent nucleoli. The tumor cells showed strong diffuse cytoplasmic immunopositivity for myoglobin and vimentin and focal staining for desmin. Immunostainings for smooth muscle actin-SMA, CD34, pan-cytokeratin, S-100 protein were negative. The immunohistochemical staining pattern confirmed rhabdomyosarcoma.This is the first description of the primary multiple heart rhabdomyosarcoma in a dog. Heart tumors (HT) are uncommon in companion animals, accounting for roughly 0.2% of all neoplasms. Whether primary or metastatic HT occur more often is unclear both in dogs and cats , 2. HemaClinical signs and their severity depend on the type, location, and size of the tumor. As long as there is no effect on the blood flow, HT are most often diagnosed incidentally during a routine echocardiographic examination performed e.g., in search for metastases from the spleen HSA .Rhabdomyosarcoma (RMS) is a soft tissue tumor derived from mesenchymal tissue with myogenic differentiation and associated with the embryogenesis of striated muscle. RMS has been described in many animal species, including dogs –9, cats It can originate from both skeletal and heart striated muscle, which does not mean that these are the most common locations of the tumor . In dogsRMS can be classified histologically as pleomorphic, embryonic or alveolar. Embryonic and follicular forms occur in young patients and are collectively referred to as juvenile RMS, while pleomorphic forms occur mainly in adults. Nodules are “grape-like” in their appearance , 26.These tumors may be mistaken for undifferentiated sarcoma depending on the phenotype of the variant. Immunohistochemical staining or electron microscopy are essential for an accurate classification . RMS is An eleven-year-old crossbreed male dog weighing 35 kg and living in a dog shelter was presented to the veterinary clinic semi-conscious in lateral recumbency. The dog had been lethargic and lacked appetite for previous 3 days. No other clinical sings were reported by the dog’s caretakers. In the clinical examination the rectal body temperature was 38.1 °C, mucous membranes were pink and moist, and lymph nodes were normal. Respiration and heart rate were increased to 45 breaths and 150 beats per minute, respectively. No pathological heart murmurs were audible. In the neurological examination reaction to external stimuli including pupils reaction to light was vastly reduced. The focused ultrasound assessment with sonograms in trauma (FAST) performed using GE Healthcare Logiq F6  with a 10 − 6 MHz microconvex transducer revealed multiple masses in the heart and the patient was referred for a specialist echocardiographic examination. Echocardiographic examination was performed using Mindray M7  ultrasound machine with a 4-2s MHz phased array transducerin in a lateral recumbency. Numerous intracardiac tumors in all four chambers were found. The smallest nodule was approximately 0.5 cm in diameter and the largest was approximately 3.0 × 1.5 cm  as well as with a ready-to-use Masson’s Trichrome staining kit . On immunostaining with myoglobin, desmin, vimentin, the normal myocardium  4 μm tissue sections and an automated immunostaining procedure . CD34, S-100, CD31 stains were performed on the BOND MAX immunohistochemistry apparatus  in a closed system using the BOND Compact polymer antibody RTU detection system (ready to use). Masson’s Trichrome staining was performed on the Artisan apparatus  a closed system, using Masson’s Trichrome reagents, catalog number AR173. SMA, VIM, Desmin and Multicytokeratin stains were performed on the Autostainer Link apparatus  using the EnVision FLEX detection kit . The antibodies from Agilent/Daco  against myoglobin (A 324), desmin (clone D33), smooth muscle actin (SMA) (clone 1 A4), vimentin (clone V9), CD34 (QBEnd 10) S100 , and cytokeratin (clone AE1/AE3). Myoglobin was used at a dilution of 1:30, the rest of antibodies were ready to use (RTU).2 (including atypical mitoses; Fig. Microscopically, the tumor was composed of fascicles of spindle-shaped cells with pale eosinophilic cytoplasm with round, oval and focally elongated nuclei with anisokaryosis and one or two prominent nucleoli Fig. A and B. The tumor cells showed strong diffuse cytoplasmic immunopositivity for myoglobin Fig. A and vimMultiple cardiac pleomorphic RMS was diagnosed based on histopathological and immunohistochemical examination.In veterinary medicine heart masses are diagnosed using the echocardiographic examination, however, the definitive diagnosis of the type of tumor is only possible if postmortem examination has been performed. In addition, in the differential diagnosis, other types of heart masses, such as thrombi, vegetations or tuberculomas, should be taken into account , 28. One


Hepatic masses are relatively common findings, and the diagnostic approach often begins by identifying patient and mass characteristics that are risk factors for malignancy. Chronic immunosuppression is a known risk factor for various malignancies, and azathioprine in particular has been reported in association with solid and hematologic malignancies, including diffuse large B-cell lymphoma.A 46-year-old white woman presented to clinic with several weeks of gastrointestinal symptoms and was found to have a hepatic mass on imaging. Her history was notable for neuromyelitis optica spectrum disorder on chronic immunosuppression with azathioprine. It was initially thought to be an inflammatory adenoma. On 6-month follow-up imaging, the mass had grown rapidly in size and was surgically resected. Further workup determined the mass to be an iatrogenic immunodeficiency-associated Epstein–Barr virus-positive diffuse large B-cell lymphoma confined to the liver. Azathioprine was discontinued and the patient underwent treatment with rituximab with no evidence of recurrence 2 years after the initiation of treatment.This case report describes the first time hepatic Epstein–Barr virus-positive diffuse large B-cell lymphoma has been reported with azathioprine, which highlights the unique sequelae of chronic immunosuppression, including atypical hematologic malignancies, and the importance of considering chronic immunosuppression in the diagnostic evaluation of a hepatic mass. Ultrasound performed for right upper quadrant abdominal pain may uncover an incidental or causal hepatic mass . EvaluatChronic immunosuppression has been identified as a risk factor for various malignancies –5 and imA 46-year-old white woman presented to the internal medicine clinic with several weeks of worsening symptoms of esophageal reflux, a pressure-like sensation in the abdomen, early satiety, intermittent nausea, and bloating. Her medical comorbidities at that time included seronegative neuromyelitis optica spectrum disorder with bilateral optic neuritis resulting in legal blindness and transverse myelitis resulting in neuropathic pain and neurogenic bladder/bowel treated with low-dose prednisone and azathioprine for the past 12 years. She also had Henoch–Schönlein purpura in childhood transiently requiring dialysis, chronic kidney disease, gastro–esophageal reflux disease, obstructive sleep apnea, pernicious anemia, subclinical hypothyroidism, hypertension, asthma, and herpes simplex labialis. Her surgical history included a laparoscopic cholecystectomy 8 years prior. Her family history is limited to lung cancer in a paternal grandfather. Her medications included amlodipine, azathioprine, duloxetine, fluticasone–salmeterol, gabapentin, hydrochlorothiazide, hydroxyzine, norethindrone, omeprazole, oxybutynin, prednisone, simvastatin, and valacyclovir. Physical examination was notable for moderate abdominal tenderness most prominent in the epigastrium and right upper quadrant.A right upper quadrant ultrasound was obtained, which showed increased hepatic echogenicity suggestive of hepatic steatosis and a 1.9 × 2.4 × 1.9 cm hypoechoic solid mass in the right lobe of the liver. An MRI was pursued to further characterize the hepatic mass , biliary cystadenoma, and hepatic abscesses; benign solid masses, including hepatic adenomas, focal nodular hyperplasia, hemangiomas, and angiolipomas; malignant solid masses, including hepatocellular carcinoma, cholangiocarcinoma, primary lymphoma, angiosarcoma, hepatoblastoma, and hepatic metastases; and focal fatty liver, which, while not a true mass, may present as such on imaging , 26, 27.This case also demonstrates the importance of serial imaging, not only to detect interval growth of a lesion, as was found in this case, but also to detect changes in imaging characteristics. The initial MRI image obtained of the lesion in this case appeared compatible with a focal area of infection or inflammation. Follow-up MRI 6 months later was interpreted to most likely represent a hepatic adenoma with possible transformation to hepatocellular carcinoma, ultimately found to be lymphoma. Primary hepatic lymphoma is rare, representing less than 1% of all extra-nodal lymphomas . It has In brief, DLBCL is a B-cell lymphoma that is one of the most common lymphoid malignancies in adults and can arise de novo or as transformation of a less aggressive lymphoma , 7, 35. This case illustrates the unique sequelae that can occur from chronic immunosuppression, in this case from an iatrogenic source. Although rare cases of hematologic malignancies have been seen in chronic azathioprine use, a diagnosis of primary hepatic EBV-positive DLBCL in an immune deficiency/dysregulation setting is also possible. This report bolsters the recommendation for clinicians caring for patients on chronic immunosuppression to consider atypical malignancies when evaluating a new symptom or finding. In summary, this report describes an index case of primary hepatic EBV-positive DLBCL associated with chronic immunosuppressive therapy with azathioprine in a patient with neuromyelitis optica spectrum disorder.


Pyogenic granuloma (lobular capillary hemangioma) is a common reactive tumour-like lesion of the oral cavity arising in response to various stimuli such as chronic local irritation, drug intake, and hormonal changes. The clinical features are similar to other reactive oral lesions such as peripheral giant cell granuloma (PGCG) and therefore the definitive diagnosis depends on histopathologic evaluation. We report a case of a three-year-old Indian boy presenting with a three-month history of a 3.3×1.4×0.8 cm large sessile, lobular, soft-tissue mass of the left mandibular posterior gingival region which was referred to us. An excisional biopsy of the lesion revealed multinucleated giant cells lying in an inflammatory cell infiltrate-rich stroma consisting of plump endothelial cells on histopathologic examination suggestive of lobular capillary hemangioma. The patient was asymptomatic with no new growth on regular follow-up. This is the first reported case of a large pyogenic granuloma in a very young child, which is an uncommon presentation.  The pyogenic granuloma (PG) or lobular capillary hemangioma (LCH), first described by Hartzell, is a non-neoplastic tumour-like growth of the oral cavity . It is mA three-year-old Indian boy reported to our department with a large gingival swelling and difficulty in mastication. The child was prescribed antibiotic courses several times at multiple clinics. Non-resolution of the lesion compelled parents to seek treatment at our hospital. Parents also reported that the child’s negative behaviour hampered proper intraoral examination and hence proper diagnosis. Clinical evaluation of the patient in our hospital was done in a child-friendly environment; intraoral examination revealed the presence of a reddish pink sessile, lobular mass of size 3.3×1.4×0.8 cm in the left posterior mandibular region which was soft to firm on palpation (Figure The lesion involved the gingiva around the mandibular first and second primary molar and obliterated the buccal and lingual vestibule. It had started as a small lobe which gradually increased in size to reach the present size and the patient had difficulty in food consumption as reported by the parent. There was dental plaque and the presence of caries in teeth adjacent to the lesion was noted as well. A differential diagnosis of PG or PGCG based on clinical presentation was made.Laboratory tests and resultsThere was superficial erosion of the alveolar bony crest in relation to the molar teeth. The second primary molar showed a circumscribed radiolucent lesion surrounding the distal root apex despite being caries-free. The striking feature of PGCG is the superficial resorption of the bony crest or cupping defect . The firPain-eliciting pulp sensibility test to ascertain pulp involvement at the clinic was not possible due to young age. Hence, the presumptive diagnosis of PGCG was made.Treatment and follow-upTreatment was initiated after obtaining informed consent from the parents after explaining the condition, side effects, and benefits and risks of treatment. In the initial appointment, restorations were done using the atraumatic restorative technique to improve the otherwise negative behaviour of the child at 2-3 years of age. Oral prophylaxis was done and the area of the lesion was cleaned and irrigated to decrease the inflammation around the lesion. Multiple irrigations were done locally every three days for two weeks to decrease inflammation and improve oral hygiene before attempting treatment. The carious teeth were restored. Later, the lesion was surgically excised under general anaesthesia after evaluating the patient’s general condition and suitability to undergo the treatment. Excisional biopsy was performed and histopathological diagnosis revealed acanthotic epithelium, several dilated, abundant proliferative blood vessels lined by flattened-to-plump endothelial cells, and an inflammatory cell infiltrate-rich stroma, which was suggestive of LCH Figures .In the one-month postoperative follow-up visit, the area showed complete resolution of the lesion Figure .Regular follow-up visits were done at seven days, 15 days, one month, and three months, respectively. The patient will be recalled at six months and every year hereafter. The importance of maintaining good oral hygiene was also emphasised to the parents.PG occurs as a long-term response to factors such as localised irritation, trauma, and hormones. Its occurrence is rather uncommon in the oral cavity of children, with poor oral hygiene being the common causative factor ,6. Peak Differential diagnoses for gingival lesions in children that may mimic the PG are the PGCG, peripheral ossifying fibroma, parulis, hemangioma, and fibrous epulis . Based oThe parulis is an erythematous nodule of the gingiva, which is frequently associated with a foreign body, a pocket, or a non-vital tooth. It is generally painful with purulent exudate, which helps to differentiate this inflammatory disease from PG . The perHemangiomas are congenital lesions, and some vascular malformations increase in size during childhood. Characteristic features are brisk bleeding, increased warmth of the tissue, and blanching upon palpation . These fThere are two histological variants for the PG. The first one is characterised by the proliferating blood vessels organised in lobular aggregates called LCH PG whereas the second type reveals highly vascular proliferation that resembles granulation tissue called non-LCH PG .The treatment of the PG involves the removal of irritating factors and surgical excision of the lesion in order to reduce recurrences. In the present case, the patient was subjected to an excisional biopsy of the lesion. Early detection of the PG results in a more conservative treatment and management. The other treatment options for the management of intraoral PG include laser-assisted excision  and intrCareful medical history followed by a complete clinical, radiological, and histopathological examination is a critical step in the diagnostic process which helps in correct diagnosis and an appropriate treatment plan. In our case, the child was referred to multiple places without treatment as the physicians were unable to correctly identify and treat the lesion as it presented with similarities to PGCG as well. Intraoral PG, although rare, can present in young children as well and if treated promptly reduces the possibility of recurrence and incidence of morbidity.


Paranasal sinus angiosarcoma is an uncommon malignancy, with only a few reported cases worldwide. Although it exhibits multiple symptoms, facial paralysis has not been previously documented as a noticeable presentation.In this case, we report a 40-year-old male who presented with facial numbness and pain for one month, weakness of his facial muscles for 15 days, and recurrent right epistaxis for 1 year. He had a history of nasal inflammatory polyps with chronic sinusitis. Computed tomography and magnetic resonance imaging showed space-occupying lesions in the right nasal cavity and maxillary sinus, with bone destruction occurring in the sinus wall and turbinate. This patient then underwent endoscopic surgery. According to the histopathological and immunohistochemical results, he was eventually diagnosed with paranasal sinus angiosarcoma in April 2021. To date, this patient has not initiated any radiotherapy or chemotherapy and has survived with lymphatic metastasis for at least 3 years.This manuscript suggests that paranasal sinus angiosarcoma can present with facial paralysis. Moreover, pathological and immunohistochemical tests are still vital for diagnosing paranasal sinus angiosarcoma and differential diagnosis. Additionally, regular follow-up is crucial for patients with paranasal sinus angiosarcoma, enabling monitoring of recurrence, metastasis, and recovery while contributing valuable clinical data to understanding this rare disease and associated research endeavours. Paranasal sinus angiosarcoma (PSA) is a rare malignant tumour and aggressive soft tissue sarcoma of endothelial origin . AngiosaDetecting and diagnosing PSA early remains challenging due to the mild initial symptoms. Angiosarcoma occurring in the paranasal sinus can invade surrounding structures, including the lateral nasal wall, anterior wall of the sinus, infraorbital nerves, eyes, and maxilla. This invasion can result in various symptoms, such as epistaxis, nasal congestion, headaches, toothaches, and diplopia , 15. PreHere, we report a case of PSA with unilateral FP as a novel manifestation. The severity of FP was assessed by the House-Brackman (H-B) grading system. We discuss the relationship between FP and PSA, analyse the differential diagnosis of PSA from pathological and immunohistochemical perspectives, and draw valuable lessons from this case.In April 2021, a 40-year-old man came to the Guangdong Provincial Hospital of Chinese Medicine ENT department complaining of facial numbness and pain for one month, weakness of his facial muscles for 15 days, and recurrent right epistaxis for 1 year. Initially, he experienced numbness and pain on the right side of his face, followed by abnormal flushing on the right midface approximately 10 days later. He developed sagging at the corners of the right side of his mouth and a deepening of the right nasolabial groove .CT and Magnetic Resonance imaging (MRI) revealed an increased extent of the space-occupying lesions in the right nasal cavity and maxillary sinus compared to the images from 2020 Fig. a and b. The postoperative pathological result revealed the presence of more adipose spindle cell hyperplasia under the mucosa of the right maxillary sinus mass, and the findings of mitotic figures on the images Fig. a and b sAt the 3-year follow-up, the patient described alleviating his facial numbness and hypoesthesia following surgery. Additionally, after being diagnosed with PSA, he chose to quit smoking. However, in August 2021, MRI scans of the sinus and neck revealed that the tumour may have infiltrated the skin along the bone defect of the anterior wall of the right maxillary sinus Fig. a. ConcurIn November 2022, the patient developed a purplish-red induration on his cheek Fig. , but theAlthough the causes of FP are various, the influence of tumours cannot be ignored. In a series of 300 patients with treatable FP seen by otolaryngologists, 112 of these patients were confirmed to have a tumour as a pathogenesis of FP . HoweverTo our knowledge, perineural invasion (PNI) and perineural tumour spread (PNTS) are well-documented modes of dissemination for head and neck malignancies . SymptomThe connection between FP and PSA may be interpreted by the anatomy of the paranasal sinuses, trigeminal nerve, and facial nerve. The maxillary nerve, as one of the second branches of the trigeminal nerve, plays a significant role in supplying sensory components to the nasal cavity and paranasal sinuses . In addiDuring the initial operation, the masses were diagnosed as inflammatory polyps of the nasal cavity and paranasal sinuses. However, considering the patient’s medical history, we suspected that the masses may not have been solely inflammatory polyps. We thought they might have been tumours that exhibited abnormal differentiation due to unknown factors during the process of a recurrence. Unfortunately, our speculation could not be confirmed at the time due to limitations in medical techniques. Additionally, some antigens were lost in the pathological specimen over time, which hindered the immunohistochemical tests. This dilemma highlights the importance of timely pathological and immunohistochemical tests when diagnosing accurately and distinguishing it from other diseases.PSA should be differentiated from reticulohistocytoma, intravascular papillary endothelial hyperplasia, epithelioid sarcomas, and melanoma. Table There is currently no convincing conclusion regarding the optimal management of PSA. Surgical resection is still the primary therapeutic for angiosarcomas at present . When deStudies have shown that the benefit of radiotherapy and chemotherapy in this disease is indistinctive, but targeted agents and immunotherapy show promise , 48. ForThis case alerts otorhinolaryngologists that although PSA with FP as the symptom is rare in clinical practice, the possibility of PSA should be considered when common causes are excluded, especially in patients with unilateral recurrent epistaxis. ENT doctors need to be fully aware of the risks of facial nerve damage and massive bleeding when formulating a surgical plan for PSA. Moreover, the importance of pathological and immunohistochemical tests in diagnosing PSA should be emphasized.


Bladder paraganglioma is exceedingly rare, accounting for <0.05% of bladder tumors. This is a case of paraganglioma with no symptom other than palpitations during urination, with atypical imaging, resulting in acute respiratory distress syndrome after transurethral resection of the bladder tumor.123Iodine metaiodobenzylguanidine scintigraphy, urinalysis, and pathological examination revealed bladder paraganglioma. Robot‐assisted radical cystectomy and ileal neobladder reconstruction were performed.A 46‐year‐old man underwent transurethral resection of the bladder tumor for a bladder tumor 61 × 52 mm in size on contrast‐enhanced computed tomography. The patient only had micturition attacks and was suspected to have urothelial carcinoma on magnetic resonance imaging. The patient had acute respiratory distress syndrome after the operation which improved conservatively. The This study reported bladder paraganglioma with no symptoms other than micturition attacks in which acute respiratory distress syndrome occurred after transurethral resection of the bladder tumor. ARDSacute respiratory distress syndromeCECTcontrast‐enhanced computed tomographyGAPPgrading of adrenal pheochromocytoma and paragangliomaMRImagnetic resonance imagingTURBTtransurethral resection of the bladder tumorWIweighted imagingIt is important to preoperatively diagnose bladder paraganglioma even if the symptoms are mild and imaging atypical as bladder tumor transurethral resection may result in acute respiratory distress syndrome.Paragangliomas are neural crest‐derived extra‐adrenal neoplasms. Bladder paragangliomas are exceedingly rare, accounting for 10% of paragangliomas and <0.05% of bladder tumors.Here, a paraganglioma case with no symptoms other than micturition attacks and atypical imaging was reported, resulting in ARDS after TURBT.A 46‐year‐old man presented to his physician with anal pain and was found to have a perianal abscess and bladder mass by CECT. After perianal abscess incision and drainage, the patient was referred to our hospital for further bladder mass examination and treatment. Palpitations during urination were observed a few months ago but had no other medical history. Complete blood count and biochemistry tests were within normal ranges; urinalysis showed no hematuria, and urinary cytology showed a class III lesion. CECT showed a 61 × 52 mm bladder tumor at the left bladder sidewall and mild left external iliac lymph node enlargement. MRI showed a faint high signal intensity on T2‐WI and a high signal intensity on T1‐WI and DWI, which suggested urothelial carcinoma with extramural invasion  Fig. . Based oAfter oral doxazosin mesylate administration, the dose gradually increased to 16 mg/day for preoperative preparation. Robot‐assisted radical cystectomy and ileal neobladder reconstruction were performed. The left external iliac lymph node was smaller than before on the CT scan just before surgery. Intraoperative rapid pathological diagnosis at the equivalent site showed no evidence of malignancy. Thus, pelvic lymphadenectomy was omitted. Systolic blood pressure sometimes rose to 180 mmHg during surgery and phentolamine was administered at 2–10 mg/hour as a continuous intravenous and 1–2 mg as a bolus injection when appropriate, but after tumor removal, it remained in the normal range and no special vasopressors or antihypertensives were used. On histopathologic analysis, the bladder paraganglioma showed invasion into the perivesical fat, but the resection margins were negative. The patient had an uneventful postoperative clinical course and was discharged on postoperative day 22. Postoperatively, serum and urinary catecholamine levels normalized. Genetic testing was not performed in this case without the patient's request.Urinary bladder paraganglioma are relatively rare tumors and have been reported to account for <0.05% of bladder tumors.The most common signs and symptoms of bladder paraganglioma are micturition attacks, hypertension, headache, and hematuria.123I‐MIBG scintigraphy and 18F‐fluorodeoxyglucose‐position emission tomography. MRI shows low‐signal intensity on T1‐WI and moderately high‐signal intensity on T2‐WI typically; sensitivity can be as high as 88%.123I‐MIBG scintigraphy also has a high specificity of 96%.In addition to cystoscopy and echography, diagnostic imaging for bladder paraganglioma includes CT, MRI imaging, and nuclear medicine scans, such as A systematic review by Beilan et al. reported that 68.9% of patients who underwent surgery for bladder paraganglioma underwent partial cystectomy, 11.3% underwent total cystectomy, and 19.8% underwent TURBT as surgical procedures for bladder paraganglioma.In conclusion, this study reported a bladder paraganglioma with no symptoms other than micturition attacks, in which ARDS occurred after TURBT.Toshifumi Takahashi: Data curation; formal analysis; supervision; validation; visualization; writing – original draft. Tatsuya Hazama: Data curation. Hideto Ota: Data curation. Yuya Yamada: Data curation. Masakazu Nakashima: Data curation; supervision; validation. Masahiro Tamaki: Data curation; supervision; validation. Noriyuki Ito: Data curation; supervision.Not applicable.Written informed consent to participate in this study and for the publication of this report was obtained from the patient for ethics approval.Not applicable.


Xanthogranulomatous cholecystitis (XGC) is a rare form of chronic gallbladder inflammation that most commonly presents as acute cholecystitis and is often mistaken for carcinoma of the gallbladder. This case details the hospital course and follow-up of a 77-year-old male who developed suspected acute acalculous cholecystitis (AAC) resulting in severe sepsis after elective left total knee arthroplasty (TKA). Histopathological findings after elective cholecystectomy later revealed XGC as the underlying etiology. While xanthogranulomatous cholecystitis (XGC) is generally considered to be a rare form of chronic cholecystitis, patients primarily present with signs and features of acute cholecystitis. There are many factors that can cause cholestasis leading to XGC; however, the majority of cases are a result of gallstone obstruction of the cystic duct. Cholestasis causes increased intraluminal pressures within the gallbladder which can lead to ischemia, bacterial proliferation, sepsis, and shock . XGC is XGC typically occurs in patients over the age of 50 and has a prevalence ranging from 1.3% to 1.9% in Europe and the Americas and 8.8% in India ,3. XGC mSince XGC is typically a histopathological diagnosis, it is often only found incidentally upon postoperative histopathological analysis after cholecystectomy, as we discovered in our case. This report aims to bring awareness to an uncommon acalculous presentation of XGC occurring after orthopedic surgery, as well as to highlight the successful management of an elderly patient with sepsis secondary to suspected acute acalculous cholecystitis (AAC).A 77-year-old Caucasian male with a past medical history significant for hypertension, gastroesophageal reflux disease, chronic gout, and mild neurocognitive impairment presented to our hospital’s emergency department from an inpatient rehabilitation facility with complaints of right upper quadrant abdominal pain, worsening lethargy, and left knee pain one week after elective left total knee arthroplasty (TKA). He underwent TKA due to progressive osteoarthritis but was otherwise in good health and able to complete all activities of daily living without significant issues. The rehabilitation facility from which he was being sent had started empiric vancomycin and cefepime due to concern for sepsis. The patient described the abdominal pain as sharp and feeling like a pulled muscle in his right upper quadrant.The abdominal pain was associated with hypotension (92/63 mmHg), tachycardia 105 bpm), a respiratory rate of 18 respirations/min, and an oxygen saturation of 100% on two liters of oxygen via nasal cannula. On exam, the patient presented with an ill appearance, labored respirations, and obtundation, responsive only to noxious stimuli. He had right upper quadrant abdominal tenderness to palpation. His left knee incisions were clean, dry, and without any signs of infection. Initial laboratory evaluation was notable for leukocytosis, with a WBC count of 26.5 thou/mcl. Blood cultures obtained at admission returned positive results for coagulase-negative staphylococcus, which was suggested by pathology to likely be due to contamination. Additional lab values are shown below in Table 05 bpm, aAbdominal ultrasound showed sludge in the gallbladder without echogenic stone and mild wall thickening, suggesting the possibility of AAC Figure . A hepatApproximately five months later, the patient followed up with outpatient general surgery due to continued right upper quadrant abdominal pain and elected to undergo cholecystectomy. During laparoscopy, a considerable amount of adhesions were noted, especially the omentum which was adhered to the liver, and the hepatic flexure of the colon which was scarred to the gallbladder. Once the gallbladder was resected, the cholecystostomy tube was found to be within a peri-gallbladder hepatic abscess. Upon gross inspection, the gallbladder was markedly fibrotic and contracted in appearance. A subtotal cholecystectomy approach was used, and the gallbladder was successfully removed laparoscopically. Surgical pathology reported no evidence of gallstones. In addition, fungal stains for fungal forms  and calcium (von Kossa) were all negative. After completed pathological analysis, a final diagnosis of XGC was given.Our patient presented with sepsis one week following an elective left total knee replacement. Ultrasound and HIDA scan imaging suggested AAC as the source of infection, which was treated with antibiotics and drainage via a percutaneous cholecystostomy tube. Eventually, the patient elected to undergo elective cholecystectomy, which showed no evidence of gallstones. Pathological analysis revealed the presence of a hepatic abscess and XGC.Our case represents a minority presentation of XGC given the lack of gallstone obstruction. One study showed that cholelithiasis was found in 69% to 96% of cases and obstruction of the cystic duct in 80% of cases [Given the histopathological confirmation of XGC, we suspect that the knee replacement surgery was likely an exacerbating factor in the gallbladder inflammatory disease process. Since there are no clearly documented risk factors for XGC, we looked at risk factors for AAC. Risk factors for AAC are processes that contribute to cholestasis and increased intraluminal pressures, mechanisms that are also thought to contribute to XGC ,10. ThusWe were unable to identify any other documented cases of XGC occurring in a postoperative setting. Similar forms of cholecystitis, such as AAC, have been known to occur postoperatively, and a small number of studies document AAC following orthopedic procedures. One case report had a confirmed histological analysis of acute acalculous gangrenous cholecystitis, and the other reported acute or chronic acalculous cholecystitis with associated hemorrhage on pathological analysis ,13. FurtCholecystectomy remains the recommended treatment of XGC, though there is a high risk of conversion to open laparotomy and other surgical complications. Biliary fistulas are the most common postoperative complication followed by surgical site infections, hepatic abscesses, bleeding, and organ injury. In the management of XGC complications, a partial liver resection may be necessary if local invasion has occurred due to fistula or abscess formation .XGC proves to be a difficult preoperative diagnostic challenge given its similar presentation to other forms of acute and chronic cholecystitis and the necessity for histopathological analysis to confirm its diagnosis. Though XGC is generally considered benign, it can result in cholecystitis leading to life-threatening sepsis. In its acute acalculous presentation, XGC should be treated as AAC until proven otherwise with surgical consultation for cholecystectomy versus percutaneous cholecystostomy and antibiotics. Cholecystectomies involving XGC should be approached with extreme caution and careful planning as they are at high risk of conversion to open due to the inflammatory nature of XGC and its propensity to form fistulae with surrounding organs.