Metanephric adenoma (MA) is a rare benign renal tumor that resembles renal cell carcinoma and Wilms’ tumor in radiological as well as pathological appearance. It can present at any age or gender, and it is extremely rare in the pediatric age group with less than 50 reported cases. Fanconi–Bickel syndrome (FBS) is a rare autosomal recessive disorder of carbohydrate metabolism. Herein, we report a rare incidence of MA in a boy with a genetically confirmed FBS who underwent a nephron-sparing surgery.A 21-month-old boy was referred to the pediatric urology clinic for further evaluation of an incidentally discovered left renal mass. His laboratory investigations showed normal renal function, hypophosphatemia, high blood glucose level, markedly elevated serum alkaline phosphatase, and low serum vitamin D. Blood picture showed signs of polycythemia and urinalysis showed glucosuria and aminoaciduria. Genetic testing was positive for Fanconi–Bickel syndrome. Radiological investigations were carried out with abdominal ultrasound and computerized tomography (CT) with intravenous contrast documented a sharply marginated peripheral hypoechoic hypovascular homogeneously enhancing mass at the upper pole of the left kidney measuring 2.0 × 1.8 × 2.0 cm. The child was admitted and started on supportive treatment until his medical condition was stabilized, then underwent elective open left partial nephrectomy via a left upper transverse abdominal transperitoneal incision. The excised renal mass was sent for histopathological assessment and was found to be a tumor composed of tightly packed tubules with no mitotic figures or necrosis and scanty cytoplasm consistent with MA. After good hydration and tumor resection, his polycythemia gradually improved. The patient was discharged home in a good condition with his proper replacement therapies. His follow-up abdominal ultrasound after 12 months showed no signs of recurrence.Metanephric adenoma is extremely rare in the pediatric age group, especially in those who have a FBS. The only way to diagnose and treat this tumor is by surgical resection as most patients are asymptomatic. A nephron-sparing surgery is better for this age group in which the future renal function is considered. Metanephric adenoma (MA) is a rare benign renal tumor that resembles renal cell carcinoma and Wilms’ tumor in radiological and pathological appearance –4. It caFanconi–Bickel Syndrome (FBS) is a rare autosomal recessive disorder of carbohydrate metabolism first described in 1949. It is characterized by nonfunctional glucose transporter 2 (GLUT2) mutation, hepato-renal glycogen accumulation, both fasting hypoglycemia as well as postprandial hyperglycemia and hypergalactosemia indicating an impaired utilization of these two monosaccharides, and proximal renal tubular dysfunction , 7. The Herein, we report a rare occurrence of MA in a boy with a genetically confirmed FBS who underwent a nephron-sparing surgery (NSS).A 21-month-old full-term boy product of normal vaginal delivery, known to have lactose intolerance, has been referred to the pediatric urology clinic for further evaluation of an incidentally discovered left renal mass. His mother noticed a gradually progressive abdominal distension with polyuria, polydipsia, and constipation. The patient's family history includes consanguineous parents with two healthy brothers and ten sisters (2 of them had died during their childhood).On physical examination, his vital signs were in the normal range, height, and weight on the 25% centile with no dysmorphic features. The abdomen was distended, soft, and lax with no palpable abdominal masses or ascites. Genital examination showed a normal circumcised penis and palpable bilateral testes in the scrotum. In addition, there were positive signs of active rickets.The initial laboratory workup revealed normal serum creatinine 37 umol/L, reduced serum phosphorus 0.81 mmol/L, markedly elevated serum alkaline phosphatase 2178 unit/L, low serum vitamin D 16.8 ng/ml, elevated blood glucose level 8.9 mmol/L with normal glycosylated HB 5.5%. Additionally, the level of serum lactate dehydrogenase (LDH) was in its normal range (186 unit/L). CBC showed signs of polycythemia (HGB 18.1 g/dL and HCT 52.6%). Urine analysis showed glucosuria and aminoaciduria. Genetic testing was considered because of the family history and clinical presentation, and it was positive for homozygous for the c.474A > C p.(Arg158Ser) familial variant in the SLC2A2 gene, which supported the clinical diagnosis of Fanconi–Bickel syndrome (FBS).Radiological investigations were done, and the abdominal ultrasound recognized a sharply marginated peripheral hypoechoic hypovascular mass at the upper pole of the left kidney measuring 2.0 × 1.8 × 2.0 cm  and epithelial membrane antigen (EMA). The cells have rounded nuclei with scanty cytoplasm and low mitotic activity. These findings were consistent with our case in addition to the positive WT1 and CD57 staining , 10.Surgical excision is the mainstay of management of MA because of its unknown malignant potentiality. Both radical and partial nephrectomies were reported either by open or laparoscopic approach , 13, 14.In general, a solid renal mass found in a 21-month-old child is most likely to be a Wilms’ tumor. Based on the current guidelines, radical nephrectomy is the standard treatment, but recent data supported the efficacy of NSS in treating Wilms’ tumors even in those without tumor-associated syndromes such as Beckwith–Wiedemann syndrome and Denys–Drash syndrome –17. In aAfter surgical resection of the tumor, no specific therapy is recommended. There have been reports of MA metastases, even though they are considered benign , 18. RelFinally, we think that the combination of MA and FBS in our case was a mere coincidence. To our knowledge, there was no reported association between FBS and childhood tumors except for one report of hepatocellular carcinoma . GLUT geMetanephric adenoma is very rare in the pediatric age group, especially in those diagnosed with FBS. The only way to diagnose and treat this tumor is by surgical resection as most patients are asymptomatic. A NSS is a valid option in this age group in which the future renal function is considered. It will be of great benefit to find a less invasive and accurate diagnostic method for this tumor to conduct a more conservative management protocol.


Myositis ossificans is a benign, non-neoplastic condition characterized by heterotropic bone formation in muscle and other soft tissues, frequently associated with direct and acute trauma.We report a case of a man who developed three lesions compatible with myositis ossificans, thirty years after suffering an injury to his face.A 71-year-old man was admitted with a history of trismus, pain in the upper left labio-gingival sulcus and difficulty in adjusting his partial denture for the past 20 days. He related a gunshot wound suffered around 30 years ago, with an entry point in his right cheek and no exit wound. At the time, he was treated by a trauma service and submitted to facial fracture fixations and bone grafts in the palate and alveolar ridge. History also included non-insulin dependent diabetes mellitus, high blood pressure and a prostatectomy eight years prior for prostate cancer. Laboratory analysis demonstrated normal fidings on serum calcium, alkaline phosphatase, parathyroid hormone levels and renal function.He was in excellent overall health, with a cheek bulge on the left and discrete associated cutaneous hyperemia. Trismus was observed, limiting mouth opening 2 cm. A bulge in the cheek mucosa was also observed near the labiogingival sulcus, with intact oral mucosa, where a 1.5 cm and well delimited firm lesion, fixed to deep structures, could be felt.When submitted to computerized tomography scan of the face, a metallic fragment was identified next to the left masticator and buccal spaces, lateral to the maxilla, in addition to metallic fragments adjacent to the zygomatic arch and in the masticator space. Deformities were identified in the walls of the ipsilateral maxillary sinus with bone loss and healed fractures. Metallic fragments were also found in the right infratemporal fossa with a fracture of the posterior wall of the maxillary sinus, bone loss in the palate and anterior wall of the left maxillary sinus and sclerosis of the zygomatic arch on the same side. Three dense amorphous ossified formations were observed in the subcutaneous tissue of the maxillary region and left masticator space, amongst the temporal, masseter and mimetic musculature, in a cortical/medullary pattern, the largest measuring 3.5 cm × 2.5 cm × 1.5 cm . Lymph nThe lesions were removed through a mucosal incision on the left superior labiogingival mucosa, revealing structures that were hardened and firmly attached to adjacent tissues . ProjectImmediately after surgery the patient experienced a reduction in trismus, despite a progression of paresis of ophthalmic and buccal branches of the facial nerve and the mimetic musculature, expected due to extensive surgical manipulation.A pathological examination identified fragments of compact bone tissue amidst the fibrin and hemorrhaging with fibroplasia and mixed inflammatory infiltrate in periosteal tissues, with no signs of malignancy. Findings were compatible with myositis ossificans.Two months after the operation, an infection occurred in the soft tissues of the left hemiface, probably due to contamination via the maxillary sinus, which improved after antibiotic therapy. The patient has been receiving speech therapy for one year, since the operation, with a substantial reduction in trismus and partial recovery of neural function.We describe a case of a circumscript myositis ossificans or traumatic myositis ossificans, unlike the progressive type – a rare genetic condition that is incurable. The former may be associated or, less frequently, unassociated with trauma. The associated trauma can be perforating or closed, burns, infections, fractures, neurological traumas, etc.Symptoms include an increase in volume and temperature, erythema, pain, paresthesia and restricted local mobility when the affected area is associated with a joint.5Jiang et al.Most cases of circumscript myositis ossificans are self-limited, in which lesions tend to regress spontaneously.Computerized tomography is the imaging exam of choice for the diagnosis of this entity. Magnetic resonance is a more sensitive way of identifying small and initial lesions, though less specific.Its initial differential diagnosis includes inflammatory and/or infectious processes, such as thrombosis, cellulitis and osteomyelitis.Once we established an indication for surgical resection of the lesion due to symptoms and limitations in buccal opening, we did not perform biopsies. We relied on signs of myositis ossificans from the imaging. However, some authorsSurgical resectionTwo aspects required our attention in this case. First, the development of the lesions 30 years after an injury is uncommon, since myositis ossificans is characterized by its development weeks after the trauma. We found one report of myositis ossificans in temporal muscle, 25 years after a severe traumaIn the last three years it could be observed an interesting increase in the reports in the literature of myositis ossificans in head and neck territory. Even so, this is an infrequent situation and physicians must remain alert to it, as well as the possible differential diagnoses, such as sarcomas, to ensure proper treatment and follow up. There are few reports of similar cases in muscles of the face that develop years after the trauma. There are also few reports of multiple lesions. Attention must also be made to the clinical history of the patient, looking for any possible local trauma.The authors declare no conflicts of interest.


Teratomas are rare neoplasms that arise from pluripotent germ cells. Sacrococcygeal teratomas are often diagnosed in infants but are rare in adults; a mature teratoma can contain hair, teeth, bony tissue, and other mature tissue types. Herein, we report for the first time a patient with a teratoma containing intact bones that formed a pseudoarthrosis.A 49-year-old woman was admitted to hospital after a massive life-long sciatic tumor had begun to grow larger over the past year. A 16 cm × 25 cm solid mass with a clear boundary was palpable in the sacrococcygeal region. Radiography, computed tomography, and magnetic resonance imaging indicated a sacrococcygeal teratoma, although blood alpha-fetoprotein levels were normal. The teratoma was completely excised using 3-dimensional reconstruction mixed reality (MR) technology with no notable complications. Postoperative pathological examination of the excised lesion confirmed a mature teratoma. Interestingly, two intact irregular bones that formed a pseudoarthrosis were isolated; one was 11 cm and the other 6 cm. The patient is currently healthy and has experienced no recurrences.Sacrococcygeal teratomas are rare, especially in adults, and often comprised lots of components, such as fat, bony tissue. However, it's first reported that formation of pseudoarthrosis in this case so far. It is difficult for surgeons to achieve complete excision without complications owing to the complex anatomic structure of the sacrum. The 3-dimensional reconstruction and mixed reality (MR) technology based on computed tomography can provide spatial visualization, which allows surgeons to examine the teratoma at different angles preoperatively. Combining 3-dimensional reconstruction and mixed reality (MR) technology in this case facilitated complete resection and prevented recurrence. Teratomas are rare congenital tumors derived from germ cells; they account for 2.5% of all germ cell tumors  and are In this report, we describe a rare sacrococcygeal teratoma diagnosed in an adult that contained 2 intact bones forming a pseudoarthrosis. We also describe how 3D reconstruction and mixed reality (MR) technology were used to ensure precise surgical resection. This report was prepared according to the SCARE checklist .A 49-year-old woman presented at Shengjing Hospital  after noticing that a massive sacrococcygeal tumor she had since birth had been growing over the past year. One month before her visit, the patient felt pain in her hip owing to a rapidly enlarging mass. The patient didn't receive any medical intervention and the symptoms were not released over past year. The patient was previously healthy with no history of chronic cardiovascular diseases, infectious diseases, allergy, or surgery. She did not smoke or consume alcohol, and had no familial diseases.Physical examination revealed a palpable 16 cm long × 25 cm wide solid mass in the sacrococcygeal region, with a clear boundary and good mobility. There was an obvious osteal protuberance in the center of the mass that had poor mobility. Preoperative laboratory workups including complete blood count, urinalysis, liver function test, and renal panel were within normal range. Pelvic radiography revealed a long rod-like bone in the pelvic cavity, while pelvic enhanced computed tomography (CT) revealed a massive space-occupying lesion in the pelvic-sacrococcygeal region that was 14.5 cm × 10.5 cm × 21.5 cm. Pelvic enhanced magnetic resonance imaging revealed a massive sacrococcygeal multiloculated teratoma comprised mainly of a fatty, sebaceous material as well as a long bone lacking any obvious abnormalities on enhanced scanning .via postoperative pathological analysis.A multidisciplinary consultation was arranged to decide on the optimal therapeutic approach based on 3D reconstruction and mixed reality (MR) technology, which were used to ensure precise surgical resection. The 3D reconstruction was performed using the “visual volume” software  with CT-derived data. The 3D model was then uploaded into the mixed reality (MR) image system, whereupon the surgeon could visualize the 3D model through the mixed reality (MR) helmet  . This enThe surgery was performed on March 26, 2021. After administering general anesthesia and disinfecting the area, a 40 cm arced incision was created in the sacrococcygeal region from the posterior superior iliac spine to the contralateral posterior superior iliac spine along with the lower boundary of the tumor . An intact encapsulated mass was found under the deep fascia; its upper boundary reached the sacral 5 vertebra, its lower boundary reached the anococcygeal ligament, and its bilateral boundaries were adjacent to the gluteus and surrounding soft tissue. The tumor protruded into the pelvic cavity and compressed the ampulla recti. Given that it had an intact capsule, the tumor did not invade the surrounding organs and we decided to remove the teratoma integrally to decrease the recurrence rate. The teratoma was rigid and lacked elasticity because of the two bones inside, which may break the capsule leading to incomplete resection if the surgical area was small. Therefore, we removed the coccyx to expose an enough space for surgical operation. Next, we separated the tumor from the pelvic cavity with the guidance of 3D reconstruction and mixed reality (MR) technology. Especially, when separating the deep bottom of the teratoma, meticulous operation was necessary to avoid bladder and rectum injury. Finally, we reconstructed the sacral structure to help recover sciatic function by anchoring a mesh through the suture to the surrounding tissue in order to provide mechanical support. Muscle reconstruction was achieved by suturing the anococcygeal ligament to the coccygeal stump . Surgeryd bosselated. The cut surface exhibited multiple cysts that contained yellow fat, gray viscous sebaceous matter, hair, muscle, and bone tissue. Of particular interest, two intact irregular bones that formed a pseudoarthrosis were isolated from the mass; one was 11 cm long and the other 6 cm  didn't show any signs of recurrence. In the perspective of the patient, the symptoms including pain, disability of defecation and urination was released entirely. The life quality of the patient was significantly improved. Meanwhile, the patient satisfied with the successful surgery and uneventful recovery. She held the view that she was in good condition and enjoyed a happy life like normal person.Teratoma is a rare type of neoplasm that originates from primordial germ cells; its site of occurrence varies with age . In infaSacrococcygeal teratomas can be classified into four types according to their depth of infiltration; these range from type I tumors that are located outside the sacrococcygeal region to type IV lesions that exist exclusively in the presacral region and pelvic cavity . Our patvia thorough analysis of the imaging data. In our patient, 3D reconstruction and mixed reality (MR) technology were used to strategize for and navigate the procedure  technology allowed to predict related intra-operative challenges, assisted the surgeons in devising a meticulous surgical plan to prevent unnecessary damage, and ensured complete tumor removal. Moreover, visualization aided us in explaining the planned procedure to the patient, who was thus able to understand both the surgical process and associated risks  technology in clinical practice. The spatial anatomic structure that mixed reality (MR) provides is based only on CT-derived values; in actuality, the anatomic structure may be more complicated in a narrow surgical view, and may be affected by hemorrhage and other factors . Mixed rPostoperative monitoring is also necessary , as the Sacrococcygeal teratomas rarely occur in adult women. Interestingly, formation of pseudoarthrosis in the teratoma is first reported in this case. Given the risks of surgical complications and incomplete excision with these tumor types, 3D reconstruction and mixed reality (MR) technology are advanced auxiliary tools that surgeons can use to achieve satisfactory outcomes.


The incidence of hepatocellular carcinoma (HCC) associated with extrahepatic primary malignancy (EHPM) is extremely rare, especially for those that involve primary renal cell carcinoma (PRC). Here we present a case of a 66-year-old male who was diagnosed with HCC complicated with lymph node metastasis at posterior pancreas head and PRC. Biopsy results of the liver and the lymph node confirmed the diagnosis of HCC. The disease progressions of both HCC and PRC are controlled effectively following the initiation of comprehensive therapy including pembrolizumab, lenvatinib, radiotherapy, and transcatheter arterial chemo-embolization (TACE). Ultimately, the patient had successfully access to surgery and complete response (CR) of all the tumors were achieved after surgery. The abdominal computed tomography (CT) scan revealed a mass in posterior pancreas head adjacent to IVC which suggested a malignant tumor, a hyperdense mass in right kidney, enlarged gallbladder, and cholelithiasis. Ultrasound-guided percutaneous transhepatic gallbladder drainage (PTGD) was performed upon admission but jaundice remained persistent post-operatively. The patient was then transferred to our hospital for further treatment.On admission, physical examination revealed jaundice of the skin and the sclera accompanied with slight abdominal tenderness over the right hypochondriac region. No rebound tenderness or guarding was felt on palpation of the abdomen. Lab reports were as follow: AFP 9243.33 μg/L, PIVKA-II 897.6 mAU/mL, HBsAg (+), HBeAb (+), HBcAb IgG (+), HBV-DNA 8.81*10^2 IU/mL, Total Bilirubin 167.1 μmol/L, Direct Bilirubin 123.5 μmol/L. The enhanced abdominal CT scan was repeated which confirmed a mass measuring 8.0*4.5 cm at posterior pancreas head which was highly suggestive of malignant tumor. The scan also revealed multiple intrahepatic masses with dimensions ranging 1 to 3 cm, right renal mass which portrayed malignant feature, cholelithiasis, and cholecystitis Figure 1A multidisciplinary team (MDT) discussion was conducted and diagnosis was narrowed down to (1) posterior pancreas head neoplasm with multiple intrahepatic metastases or primary liver cancer associated with posterior pancreas head metastasis; (2) Primary renal cell carcinoma. To further confirm the diagnosis and alleviate the symptoms, endoscopic retrograde cholangiopancreatography (ERCP)-guided biliary stenting and biopsy were performed in the posterior pancreas head mass. BUS-guided liver mass biopsy was also performed and Pathology test result of the liver indicated a poorly differentiated HCC while the immunohistochemistry test result showed HepPar1 (+), Arg-1 partial (+), GPC-3 (+), AFP (+), CK7 partial (+), CK19 (+), ki-67 (+) 50% Figure 2Our team decided to adhere to the first-line treatment suggested by the National Comprehensive Cancer Network (NCCN) Guidelines for liver cancer since there was no definite target indicated for immunotherapy from the genetic testing. To minimize the effect of obstructive jaundice on PIVKA-II assessment, Vitamin K was supplemented immediately after the alleviation of biliary obstruction. Consequently, PIVKA-II level of the patient was successfully reduced to 64.01 mAU/mL after 10 days of vitamin K prescription which reflected that elevation of PIVKA-II was mainly due to biliary obstruction.After the patient recovered from jaundice about 44 weeks ago (baseline + 2 weeks) , combined therapy which consisted of pembrolizumab plus lenvatinib (pembrolizumab 200mg Q3W and lenvatinib 8mg QD) was initiated. Subsequently, radiotherapy for the metastatic lymph node posterior pancreas head was started 1 week after the combined therapy . 40 weeks ago (baseline + 6 weeks), repeated lab test results showed reduced level of AFP and PIVKA-II to 2173.04 μg/L and 63.66 mAU/mL respectively. Follow-up abdominal CT scan revealed multiple intrahepatic lesions which were dimensionally similar to the pre-treatment period. Nevertheless, the mass in the posterior pancreas head showed significant shrinkage and reduced enhancement intensity. The right renal mass was also smaller compared to the previous scan Figure 1Pembrolizumab was administered for the second time about 40 weeks ago (baseline + 6 weeks) and post-therapy follow-up assessments were carried out about 36 weeks ago (baseline + 10 weeks). AFP and PIVKA-II levels were 1096.84 μg/L and 38 mAU/mL respectively. Overall shrinkage in all lesions was observed through CT scan Figure 1HCC complicated with extrahepatic multiple primary cancer (MPC) is rarely reported, albeit high incidence rate of HCC. With the advancements in early cancer detection, the prevalence of MPC has increased gradually over the past decade. According to previous reports, the incidence rate of MPC ranged between 0.7 and 11.7% . The mosAlthough the pathogenesis of MPC remains unclear, the underlying mechanism was known to be attributed to host-, lifestyle-, environment-, gene- as well as treatment-related factors , 19. TheAfter an in-depth discussion by the Molecular Tumor Board (MTB), our patient was diagnosed with HCC complicated with lymph node metastasis at posterior pancreas head, combined with PRC. Different from “HCC only” patients, management for second primary cancer should always be taken into careful consideration for HCC-associated MPC patients. As reported by 9 previous cases of HCC-associated PRC, all of them were solitary masses in both organs without lymph node metastasis. Seven out of the 9 cases received surgical treatment while the other 2 received interventional procedures and targeted therapy. Fortunately, all 9 patients achieved satisfactory outcomes after the treatments. However, our patient suffered from multifocal HCC with giant lymph node metastasis in the posterior pancreas head combined with PRC which has rendered the conventional radical resection implausible. Since there was no similar case reported in the past, the management for this patient is considered exploratory.Due to the complexity of the case, MTB suggested to initiate combined antitumor therapy after the alleviation of biliary obstruction. Also, management of HCC was prioritized over PRC as recommended by urologists and oncologists due to higher malignancy portrayed by HCC. As per the recommendations of the National Comprehensive Cancer Network (NCCN) Guidelines on HCC, pembrolizumab and lenvatinib are preferred as the first-line therapy for advanced-stage liver cancer. And it is possible that the combination of pembrolizumab and lenvatinib can produce a synergistic antitumor effect , 23. ApaThe emergence of stereotactic body radiotherapy (SBRT) has also been playing an important role in preventing radiation-induced liver disease (RILD), the most dreadful complication of conventional radiotherapy . It doesFor advanced HCC, TACE is often regarded as the first-line treatment –35, and In conclusion, we report for the first case of multifocal HCC with giant lymph node metastasis in posterior pancreas head accompanied with PRC. His tumor biomarkers’ level were significantly reduced as well as the tumor sizes after comprehensive treatment including pembrolizumab plus lenvatinib, radiotherapy and TACE. Ultimately, the patient had successfully access to surgery and complete response of all the tumors were achieved. Pembrolizumab plus lenvatinib remains the maintenance therapy in the follow-up. Since there were no precedent cases and treatment guidelines for this type of MPC, we hope to provide a reference for diagnosis and treatment of similar patients in the future through our experience in this case.The original contributions presented in the study are included in the article/Supplementary Material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.JC and ZZ performed the literature search and drafted the manuscript. XX designed and supervised the study. All authors contributed to the article and approved the submitted version.


Actinomyces are a part of the normal flora of the cervicofacial region, gastrointestinal tract, and urogenital tract, but can cause infections when the normal mucosal barrier is lost. Herein, we report a rare case of actinomycosis of the gallbladder in a 60-years-old-female. The patient presented with right hypochondrium pain since three months; ultrasonography showed cholelithiasis with thick oedematous wall. An open cholecystectomy was carried out. Histological examination revealed an inflamed gallbladder with colonies of radiating filamentous structures having numerous sulphur granules which on gram staining showed filamentous gram-positive rods. The diagnosis of Actinomycosis of gallbladder was made. After cholecystectomy, prolonged antimicrobial therapy is recommended for patient with actinomycosis to prevent recurrence and even mortality. It is a part of the normal microflora in cervicofacial region and gastrointestinal tract. However, the disease occurs when normal mucosal barrier is lost due to trauma, surgery or infection. It usually spreads invading tissue planes of the body forming abscess that finally leads to the formation of draining sinuses. Actinomyces israelii is the most common species.A 60-years-old-female presented with dull aching pain at the right hypochondrium since three months with no history of abdominal surgery and no significant medical history. On physical examination, there was tenderness in the right hypochondrium. Ultrasonography of the abdomen revealed cholelithiasis with thick gallbladder wall. Laboratory studies revealed normal liver function tests, amylase and serum electrolytes. Human immunodeficiency virus, Hepatitis B, and Hepatitis C serology were negative. The pre-operative diagnosis was symptomatic cholelithiasis.The patient was admitted and an open cholecystectomy was performed. Gall bladder was received for histopathological examination.Gross examination revealed the gallbladder measuring 9.5x2.5x0.8 cm. The mucosa was bile stained and gall bladder wall was thickened measuring 0.8 to 1 cm. On cut section, a single grayish white calculi measuring 1x1cm was seen. Microscopic examination revealed focally denuded mucosal epithelium with many colonies of radiating filamentous structures having numerous sulphur granules ,2 which Follow-up was advised to the patient after two weeks but the patient did not present on the follow-up. So, we lost contact with the patient and the outcome was unable to assess.1 The bacteria are commensal of the oral cavity, gastrointestinal tract and urogenital tract and have low pathogenicity, but any breech in tissue barriers by trauma, surgery or infection can facilitate the organism to penetrate and cause disease.4 Actinomycosis is also associated with other risk factors like immunosuppression, neoplasia, diabetes and intrauterine contraceptive devices (IUDs).3 It is endemic globally, prevalent more commonly in developing countries with poor socioeconomic conditions like Nepal.Actinomycosis is a chronic infection caused by Actinomyces species which is an anaerobic grampositive filamentous bacterium. There are more than 30 species under the genus Actinomyces, the most common species are Actinomyces israelii, A. gerenseriae and A. naeslundii.3 Mir, et al. reviewed 16 cases with gallbladder and bile duct actinomycosis. They found that 10 cases had the stones in gallbladder or bile duct, two cases had diabetes mellitus and eight cases were associated with other diseases including liver tumor, liver abscess, gallbladder carcinoma, amyloidosis, myocardial infarction, rheumatoid arthritis, glomerulonephritis and heart failure. Only seven cases were not affected by any underlying disease.5 In our case, the patient had gallbladder stone but no other underlying conditions. The stone could have predisposed to actinomycosis following bacterial reflux from the duodenum into the inflamed gallbladder. Most studies show the mean age at presentation to be 65 years and is more common in females. It is consistent with our case who was a 60 years old female.Actinomycosis most commonly presents in the cervicofacial infection followed by abdomino-pelvic region. Gallbladder actinomycosis is a very rare finding and there are very few cases reported in literature. Most cases are associated with gallstone or other underlying diseases.2It is difficult to diagnose gallbladder actinomycosis clinically, it may present as acute or chronic cholecystitis and often mimics carcinoma radiographically. Histolopathological diagnosis of actinomycosis is based on the demonstration of "sulfur granules" a dense aggregate of filamentous Actinomyces associated with suppurative inflammation. However, sulfur granules can occasionally be found in other infections such as Nocardia brasiliensis, Streptomyces madurae, and Staphylococcus aureus. Although sulfur granules are seen only in 50% of patients with actinomycosis, the micro-organism presenting as filamentous gram positive bacteria are pathognomic for diagnosing actinomycosis.6After cholecystectomy, prolonged antimicrobial therapy is recommended for patient with actinomycosis to prevent recurrence. The treatment of choice is high dose intravenous penicillin followed by oral penicillin alone or combined with ß-lactamase inhibitor  to cover both aerobic and anaerobic bacteria.Actinomycosis of gallbladder is a very rare condition and poses a great diagnostic challenge because of its non-specific symptoms and wide variety of clinical presentation as it tends to mimic different diseases such as symptomatic cholelithiasis and gallbladder carcinoma. The diagnosis is usually made by histological examination of the cholecystectomy specimen. The treatment needs to be long term antibiotics to avoid recurrence and even mortality.


A very rare complication of robot-assisted laparoscopic radical prostatectomy (RALP) is bladder stone formation from a hemostatic clip as a nidus. A 70-year-old man presented 11 years after a RALP with recurrent UTI, worsening lower urinary tract symptoms, and visible haematuria. A flexible cystoscopy revealed a large 3.5 cm x 1.5 cm stone at the bladder neck. Computed Tomography (CT) Urogram revealed normal kidneys with no hydronephrosis and a bladder stone. The patient underwent a cystolithotripsy as a day case which was performed by use of Swiss LithoClast® Trilogy lithotripter, which delivered controlled ultrasonic and ballistic energy and simultaneous suction through a single probe. Hemostatic clips were incidentally identified during the procedure and were successfully removed. The patient was discharged on the same day and made an excellent recovery. Hence, migration of clips is a rare occurrence, and good intraoperative techniques should avoid such complications to occur. Prostate cancer is a commonly diagnosed malignant tumor in men . The disA 70-year-old man was reviewed on the two-week wait pathway for visible haematuria and worsening lower urinary tract symptoms (LUTS). He was diagnosed with high-risk Gleason 3+4=7 organ-confined (T3aN0M0) adenocarcinoma of the prostate in 2011 and successfully underwent a robot-assisted laparoscopic radical prostatectomy with no post-operative complications. He remained well for almost 11 years with an undetectable PSA. He was recently referred by the general practitioner with a three-month history of worsening LUTS and new onset visible haematuria. When questioned about his symptoms, he described increased frequency, incomplete bladder emptying, urgency, and dysuria.On examination, the patient had an unremarkable cardiorespiratory system. Abdominal examination revealed a soft, non-tender abdomen. A bladder scan revealed significant residual urine of 150ml, and his serum prostate-specific antigen (PSA) was untraceable at <0.01. Flexible cystoscopy revealed a normal anterior urethra, the sphincter was seen, and immediately proximal to this was a large bladder stone with difficult negotiation of the flexible cystoscope. Both ureteric orifices were identified after the washout of debris in the bladder, and no cancer was identified in the bladder. A digital rectal exam (DRE) revealed no prostate or masses. Abdominal ultrasonographic examination revealed a large bladder stone at the bladder neck and normal kidneys. A CT urogram as part of the evaluation of visible haematuria revealed normal kidneys, simple renal cysts, no hydronephrosis or renal lesions, and a 3.5 cm stone at the level of the bladder neck Figure .The patient was electively admitted to the day surgery unit and underwent stone fragmentation with a Swiss LithoClast® Trilogy lithotripter to a yellow-colored stone Figure  at the lHem-o-Lok® clips (HOLCs)  are widely used for controlling the lateral pedicles in laparoscopic radical prostatectomy . These aThe duration of the symptoms manifesting is variable, with some cases presenting within less than 1-2 years of the surgery . Our patThe literature review identified approximately nine cases. Tugcu V et al. , in 2009George A. TuriniIII et al.  in SepteMigration of hemostatic clip and bladder stone formation after robotic laparoscopic radical prostatectomy can present as irritable lower urinary tract symptoms, visible haematuria, and urinary tract infections irrelevant to time or duration after surgery. However, such occurrences are also rare. Good intraoperative technique and care should be taken to avoid such complications.


Paragangliomas are rare, non-epithelial neuroendocrine neoplasms originating in paraganglia, for instance the adrenal medulla, or at extra-adrenal locations. The aim of this study was to review the literature regarding abdominal extra-adrenal paragangliomas diagnosed pre-operatively with fine-needle biopsy (FNA and/or FNB). The PubMed database was searched to identify such cases, using a specific algorithm and inclusion/exclusion criteria. An unpublished case from our practice was also added to the rest of the data, resulting in a total of 36 cases for analysis. Overall, 24 (67%) lesions were found in females, whereas 12 (33%) in males. Most  were identified around and/or within the pancreatic parenchyma. FNA and/or FNB reached or suggested a paraganglioma diagnosis in 17/36 cases (47.22%). Of the preoperative misdiagnoses, the most common was an epithelial neuroendocrine tumor (NET). Regarding follow-up, most patients were alive with no reported recurrence; however, 5/36 patients exhibited a recurrence or a widespread disease, whereas one patient died 48 months following her diagnosis. In two patients, transient hypertension was reported during the EUS-FNA procedure. In conclusion, this study showed that the preoperative diagnosis of these lesions is feasible and, while diagnostic pitfalls exist, they could significantly be avoided with the application of immunochemistry. Paragangliomas are rare, non-epithelial neuroendocrine neoplasms derived from the neural crest, yet their incidence has increased over recent years. Pheochromocytomas and paragangliomas (often referred together as PPGLs) are histologically similar. Notably, according to the latest WHO guidelines, pheochromocytomas are now officially considered as “intra-adrenal paragangliomas” ,2. PPGLsPPGL patients could present with a clinical picture related to catecholamine hypersecretion, such as headaches, palpitation, and high blood pressure, coupled with high levels of metanephrines in their plasma or urine. In such cases, clinicians could suspect, readily identify, and subsequently manage the lesion ,10,11. HThe aim of this study was to review the literature regarding the reported cases of abdominal extra-adrenal paragangliomas diagnosed pre-operatively with FNA and/or FNB, with the goal to reveal their main demographics, diagnostic pitfalls, and clinical behavior. In addition, we also present an unpublished case of a retroperitoneal paraganglioma from our practice, diagnosed with EUS-FNB.The PubMed database was searched for cases of abdominal extra-adrenal paragangliomas diagnosed with FNA and/or FNB from 1 January 2000, until 26 May 2022, using the following algorithm: (paraganglioma OR pheochromocytoma) AND (FNA OR “fine-needle aspiration” OR FNB OR EUS-FNA OR EUS-FNB).Inclusion criteria involved any studies  reporting abdominal extra-adrenal paragangliomas diagnosed with FNA and/or FNB, where the final diagnosis was reached either preoperatively from the FNA/FNB material or by examining the subsequent surgical excision specimen. Exclusion criteria involved studies reporting solely PPGLs found in the adrenals (pheochromocytomas), outside the abdomen, or diagnosed with modalities other than FNA/FNB. Articles where individual data of the paraganglioma cases described could not be extracted were also excluded at a subsequent step. Three authors  first performed a title–abstract selection, which was followed by a full-text evaluation of all eligible articles. Any discrepancies were resolved by reaching a consensus.® file: first author, year of publication, gender, age, location of the mass in the abdomen, diameter of the mass assessed with endoscopy or radiology , clinical presentation, biopsy type (FNA and/or FNB), preoperative diagnosis derived from the FNA and/or FNB pathologic interpretation, immunochemistry performed on the FNA or FNB material, whether or not the mass was excised with surgery, and follow-up information.The following data regarding individual paraganglioma cases were extracted in an ExcelThe flowchart of our study is shown in The extracted data from the 36 eligible paraganglioma cases are shown in Our case was about a 35-year-old female patient who underwent an EUS-FNB of a retroperitoneal mass, which had previously been detected incidentally. EUS revealed this mass was located in the paraduodenal area and had a diameter of 65 mm, while it was significantly vascular. Two passes with a 22G FNB needle through the duodenum were performed; cores were put directly into formalin, whereas the remaining material was used for conventional and liquid-based cytology.Microscopic examination of the FNB material revealed the presence of neoplastic cells, mostly arranged in syncytial groups . ImmunohThe mass was subsequently excised and surgical pathology confirmed the abovementioned preoperative diagnosis. Immunohistochemistry against SDHA, SDHB, and ATRX was additionally performed, and the neoplastic cells within the mass were found to retain their staining.To our knowledge, this is the first literature review summarizing the published data of abdominal extra-adrenal PPGLs diagnosed with FNA and/or FNB. This study showed that PPGLs preoperative diagnosis using the abovementioned modalities is feasible, especially when pathologists are familiar with the cytomorphologic features and immunochemistry is additionally applied on the cytologic or histologic material. As According to the literature, abdominal extra-adrenal paragangliomas are most often found in the periaortic and pericaval regions ,35. NotaAs shown in our study, neoplasms other than epithelial NETs could also enter the main differential diagnosis, depending on the retroperitoneal location of extra-adrenal PPGLs. For instance, in para-gastric or para-duodenal lesions, a gastrointestinal stromal tumor (GIST) needs to be ruled out; GISTs are positive for DOG1 and CD117 with immunochemistry whereas they are negative for neuroendocrine markers, in contrast to paragangliomas. Furthermore, when found in the perirenal space or the retroperitoneal soft tissue, a renal cell carcinoma or a soft tissue tumor, such as a schwannoma or sarcoma, should also be excluded. In addition, various metastatic carcinomas or a melanoma could often be considered as a potential diagnosis. Metastatic carcinomas would be Keratin positive, while melanomas positive for Melan-A, HMB-45, and diffusely positive for S100 with immunochemistry  ,40,41. LThe accurate diagnosis of a retroperitoneal PPGL is clinically important, especially its distinction from epithelial NETs. First, paragangliomas could be associated with a distinct clinical picture related to the activation of the sympathetic nervous system, while the metanephrines are regarded as biomarkers to monitor their response to therapy and potential recurrence. Furthermore, as paragangliomas often carry germline (more that 40% of the cases) and/or somatic mutations, genetic testing is offered to the affected patients and its results could impact patients’ management, follow-up, and prognosis ,10,43,44This study has some important limitations. Data were extracted from case reports or small case series, which are generally considered of low-quality evidence. This could not be avoided though, as abdominal paragangliomas are rare lesions, while we additionally focused only on the ones diagnosed with FNA and/or FNB. Furthermore, we need to consider the reporting bias, as PPGLs suspected clinically as such were most likely not biopsied. Lastly, in six of the initially eligible studies reporting abdominal extra-adrenal paragangliomas, individual data could not be extracted; thus, these reports were excluded from the review .This study showed that the preoperative diagnosis of abdominal extra-adrenal paragangliomas with FNA or FNB is feasible. Diagnostic pitfalls exist, but could significantly be avoided within a multidisciplinary setting and the application of immunochemistry on the cytologic or histologic material. The retroperitoneal paragangliomas most often aspirated with FNA or FNB were reported to be located around or within the pancreas, and a PanNET was the main differential diagnosis.


Becker's nevus is also known as Becker melanosis. It is a benign lesion which can be presented as congenital or acquired with hairless or hypertrichotic lesions. It's a rare case which affects mainly male individuals. It is often pigmented and gets darker by time and excessive hair growth can be seen over it. A 29-year-old transgender patient presented with hyperpigmentation with the lesion which started at the age of 15 as a small hyperpigmented macule. The lesion increased gradually to form giant patches. On examination a right-side hyperpigmentation involving the anterior chest, shoulder, scapular region, upper arm with hypertrichosis and irregular margins. On histopathological examination, hyperpigmentation of the basal layer and melanophages were present in the upper dermis. A diagnosis of Becker’s nevus was made based on the clinical appearance and histopathological findings. After counseling the patient about the disease, laser therapy was referred and regular follow-up was advised.


The retroperitoneum can host a wide spectrum of soft tissue lesions. Thesetumours pose a challenge to the pathologist as the morphology is not of muchhelp and immunohistochemistry becomes a necessity.Sixty years old male presented with 2 months history of abdominal lump, painand dyspepsia. The MRI revealed a heterogeneous mass in the retroperitoneuminvolving right para spinal muscle, right iliac fossa and right perinephricregion with destruction of right transverse process and erosion of adjacentL3 vertebra. Trucut biopsy of the mass was reported as fibroliposarcoma atan outside lab. Patient underwent a wide local excision. Grossly the tumourgave an impression of a liposarcoma but the microscopy showed areas ofspindle cells, epitheloid cells, focal areas of ganglion like cells andlarge areas of myxoid change. IHC panel of S-100, SMA, caldesmon, myogenin,myoglobin and Alk-1 was negative. MDM2, CDK4 and p16 IHC came positiveproving it to be a dedifferentiated liposarcoma.We report a curious case of retroperitoneal soft tissue tumour with complexmorphology and IHC features diagnosed as dedifferentiated liposarcoma basedon MDM2, CDK and p16 positivity. These tumours pose a challenge to the pathologist as the morphology is not ofmuch help and immunohistochemistry becomes a necessity. One of the characteristic aswell as distinguishing feature of dedifferentiated liposarcoma is that it is usuallylocated in the retroperitoneum unlike well differentiated liposarcoma, which is mostoften seen in limbs. On microscopy, dedifferentiated liposarcoma shows areas ofatypical lipomatous tumours but most of the areas show dedifferentiated neoplastictissue. This dedifferentiated tumour can make this tissue look like complexneoplasms such as malignant fibrous histiocytoma, chondrosarcoma, osteosarcoma,angiosarcoma or sometimes even as melanoma, meningioma, lymphoma.7 Even though it appears thatdedifferentiated liposarcoma would develop from well differentiated liposarcoma,many cases are found de novo as well. Dedifferentiation is a process of progression from well differentiated formto a higher-grade less differentiated form. In most circumstances dedifferentiationworsens the prognosis. However, in cases of dedifferentiated liposarcoma, neitherthe local extent nor the grade has any significant influence on the behaviour orprognosis of this tumour.8 We report such a case of aretroperitoneal mass diagnosed as fibroliposarcoma on biopsy gave a completelydifferent histological picture on final histopathology. It required a whole set ofIHC and FISH to establish the diagnosis.The retroperitoneum can host a wide spectrum of soft tissue lesions. Among these,liposarcoma is the most common soft tissue sarcoma. WHO classification categorizesliposarcoma into 5 categories, out of which well differentiated liposarcomaconstitutes around 40%.A sixty years old male presented with 2 months history of abdominal lump, pain anddyspepsia. On clinical examination around 10 cm × 10 cm non-tender, retroperitonealtumour was palpable. MRI of the abdomen revealed a 15 cm × 12 cm heterogeneousretroperitoneal mass involving right para spinal muscle extending deep intoreteroperitoneum, right iliac fossa and right perinephric region with destruction ofright transverse process and erosion of adjacent L3 vertebra. FNAC of the mass wasperformed and it showed features of fibroliposarcoma . PatientOn gross examination the 15 cm × 12 cm × 7 cm specimen contained a7 cm × 3.5 cm × 3.1 cm tumour with whitish/pale yellow cut surfaces. Grossly thetumour gave an impression of a liposarcoma but the microscopy was altogether adifferent story with areas of spindle cells, epitheloid cells, focal areas ofganglion like cells and large areas of myxoid changes . HaematoFigures 6 As per WHO classification, it has 5 subtypes namely, well differentiated,myxoid, round cell, pleomorphic and dedifferentiated types. Dedifferentiated liposarcoma has histological features of both welldifferentiated and poorly differentiated liposarcoma along with non-lipomatousareas. Mostly, this tumour is asymptomatic and identified incidentally on imaging asa part of routine medical examination. However, it can present with abdominal lump,pain or discomfort, or sometimes obstructive or compressive symptoms. CT scan, canbe used to assess the extent and anatomy. Biopsy is usually performed before surgeryand correlated with radiology to reach a diagnosis.Retroperitoneal tumour is an overall rare entity accounting for less than 0.2% of allmalignant tumours. It is most commonly seen in males in their 4th to 5th decade oflife. Around 40% of all the retroperitoneal tumours is liposarcoma. in their study described that more than 90% dedifferentiated liposarcomashowed a MDM2 and CDK4 staining showing that MDM2 and CDK4 immunohistochemistry is asensitive technique in the diagnosis of dedifferentiated liposarcoma. MDM2amplification has been described in malignant fibrous histiocytoma and othersarcomas as well such as MPNST, PNET, rhabdomyosarcoma, low grade ordedifferentiated osteosarcoma and synovial sarcoma. However, another study involving malignant fibrous histiocytoma have shownthat some of these cases in the past might have been misclassified and were indeeddedifferentiated liposarcoma. Dedifferentiated liposarcoma has also been described to have overexpressionof cell cycle regulator p16. Thway et al described the utility of immunohistochemistry for MDM2, CDK4 and p16 in theroutine diagnosis of dedifferentiated liposarcoma. However, there have beenconflicting studies showing lesser usefulness of p16 as compared to MDM2and CDK4. In our case, all the 3 IHC markers namely, MDM2, CDK4 and p16 were positiveshowing the usefulness of these markers in such complex cases with atypicalfeatures. Techniques like CGH, FISH and quantitative PCR can also be employed tofurther confirm the diagnosis.Even though most retroperitoneal tumours are diagnosed on microscopy using regularstains, sometimes they require special stains to rule out certain pathologies. Thesestains are SMS, S100, actin, desmin, caldesmon, myogenin, myoglobin, Alk-1, etc.Sometimes, IHC is required to get to a diagnosis and thus it increases thecomplexity of the case such as differentiated liposarcoma. Atypical lipomatoustumours and dedifferentiated liposcarcoma shows a characteristic genomicamplification of 12q13 to 15. We could not test the copy number changes inchromosome 12 as the test was not available at our centre. It includes theproto-oncogenes MDM2 and CDK4. So MDM2 and CDK4 staining plays a very important rolein the diagnosis of dedifferentiated liposarcoma. Binh et al


Lipoleiomyoma is a rare neoplasm of the uterus. It is considered a variant of uterine myomas. Its reported incidence varies from 0.03% to 0.2%. Lipoleiomyoma consists of variable proportions of mature lipocytes and smooth muscle cells. These tumors generally occur in asymptomatic obese perimenopausal or postmenopausal women. About 90.7% of lipoleiomyomas arise from the uterine corpus, with only 6.5% arising from the cervix. When it occurs in the cervix, it is difficult to diagnose and treat it. We report an uncommon case of pelviscopic resection of uterine cervical lipoleiomyoma showing continuous growth after menopause.A 55-year-old postmenopausal woman was diagnosed with 40 mm-sized uterine myoma 4 years ago. The size of the mass increased to 58 mm in the last year.An ultrasound scan revealed a 58 × 34-mm-sized round hyperechogenic and barely vascularity mass that appeared to have originated on the left side of the uterine cervix. Final pathologic findings showed lipoleiomyoma.After admission to the hospital, we performed pelviscopic removal of uterine lipoleiomyoma and both tubes. Microscopic examination revealed a significant amount of fat cells between muscle cells.Surgeries were successful. The patient had been followed up regularly for three years after surgery. She did not experience any complications. She remained disease-free.Although lipoleiomyomas mainly occur in postmenopausal women, they can also occur in the uterine cervix. They can increase in size after menopause. They can be removed laparoscopically. If a hyperechoic mass occurred in the uterus after menopause that keeps growing without symptoms, a differential diagnosis of lipoleiomyomas must be performed. Lipomatous uterine tumors are unusual benign neoplasms. Most cases of lipoleiomyoma cannot be distinguished clinically from leiomyoma. They can be diagnosed typically by gross glistening yellow appearance of the tumor. Most patients are asymptomatic. Occasionally they may complain of vaginal bleeding or pelvic pain. Lipoleiomyomas are predominantly located in the uterus, although, extrauterine locations have also been reported. Their ultrasonographic findings include a well-defined, homogeneously echogenic lesion without vascularity. The tumor consists of long intersecting bundles of bland and, smooth muscle cells admixed with mature fat cells. Surgical treatment is suggested for symptomatic lesions or when a diagnosis is in doubt. Here we report a very rare case of lipoleiomyoma of the uterine cervix with a literature review. It was removed pelviscopically. This case report can be helpful for clinicians.“Myolipoma of soft tissue” was thought to be first described in 1991 by Meis and Enzinger.A 55-year-old Asian woman, gravida 2, para 2, presented with cervical mass that were increasing in size. She had no medical or surgical history. She was 163 cm tall with a weight of 62 kg at the time of her visit. She went through menopause 5 years ago. She had not taken any hormonal replacement therapy. She was found to have a 40 mm-sized mass on the cervix in an examination 4 years ago. However, the size suddenly increased to 58 mm last year. She had no symptoms. After menopause, the size of the mass increased, making it necessary to make differential diagnosis from cancer. She was transferred to our hospital. Her Pap (Papanicolaou) smear done 6 months ago was normal. Her cancer antigen (CA) 125 (0–35 U/mL) level was 10.5 U/mL. All other laboratory values including lipid profiles were within normal ranges. Gynecologic examination revealed no abnormalities of the vulva or, cylindrical vaginal portion of the cervix. No evident pathological change was detectable. A well-defined, hyperechogenic mass measuring 58 34 mm was seen in the cervix. Thickness of the endometrium was 3 mm. Both ovaries and tubes were normal on transvaginal ultrasound. There was no vascularity in the mass on color Doppler  shows secondary to the predominant fatty component in the lesion. Hyperintensity is seen on T1 weighted sequences. Hypointense on T2 fat suppression sequences is also noted. Additionally, fat suppression techniques can be used to verify the diagnosis as most lesions show fat suppression. We did not perform additional MRI as the mass was benign on APCT. Lipoleiomyomas when small and asymptomatic usually do not require treatment. They are clinically similar to leiomyomas. Treatment depends on clinical symptoms and the size/location of the lesion. Uterine artery embolization or surgical excision can be performed, as indicated. For uterine fibroids, regular follow-up for changes in size or symptoms has been suggested. However, the rate of growth is higher for lipoleiomyomas than for conventional uterine myomas, the latter of which tend to decrease in size after menopause. It has been reported that the relatively rapid growth rate of lipoleiomyomas mimics that of malignant tumors. For symptomatic lesions or when a diagnosis is in doubt, surgical treatment is recommended. General surgical options for lipoleiomyomas include myomectomy through abdominal, laparoscopy or hysteroscopy, and hysterectomy with or without bilateral salpingo-oophorectomy through the abdomen or vagina. Histological examinations of lipoleiomyomas show a mixture of bland, spindle-shaped smooth muscle cells without nuclear atypia in a whorled pattern with admixed mature adipocytes. A significant amount of fat cells between muscle cells can be seen. The adipose component is entirely mature without any lipoblasts.Uterine lipoleiomyomas resulting from degeneration of smooth muscle cells in an ordinary leiomyoma are rare benign tumors of the uterus. In 2014, Akbulut et al reported that patients with lipoleomyomas ranged in age from 34 years to 77 years (mean: 55.49 years) with sizes ranging from 0.5 cm to 55 cm in diameter (mean: 5.50 cm). Sixty-nine (90.7%) tumors were in the uterine corpus while only 5 (6.5%) were in the cervix.Although the patient’s history and imaging studies play an important role in preoperative diagnosis and site of the lipoleiomyoma, it is the pathological examination that can confirm the diagnosis finally. In the present case, the patient underwent laparoscopic lipoleiomyoma resection. She had no recurrence at 3 years of follow-up. This study is of great value in that few studies have reported lipoleiomyomas removed by laparoscopy so far.The authors are grateful to Soonchunhyang University Cheonan Hospital for their assistance and encouragement.Conceptualization, data curation, investigation, and writing of original draft preparation: Yun Sook Kim. Writing-review, Pathology: Ji Hye Lee. All authors read and approved the final manuscript.


Atrial myxomas account for approximately 50% of all primary cardiac tumors. The size, location, risk of embolic event, and involvement of other cardiac structures, are all factors that contribute to the wide range of presentation for cardiac myxomas. Patients with myxomas may remain asymptomatic, while others may report symptoms such as fatigue and fever, dyspnea, and syncope. It is important to recognize arrhythmias as an uncommon symptom of myxomas.We report a rare case of a 67-year-old man who presented with pre-syncopal episodes, symptomatic bradycardia, and night sweats found to have a 5.5 × 5.1 × 3 cm myxoma in the left atrium. During diastole the mass caused dynamic flow obstruction across the mitral valve. The patient underwent surgical resection of the mass given his symptomatology and risk of embolic events. Removal of the myxoma resulted in resolution of both pre-syncopal episodes and the patient’s sinus bradycardia.Atrial myxomas are a rare cause of pre-syncope and symptomatic bradycardia. It is important to have a clinical suspicion for atrial myxomas given early diagnosis and surgical intervention are key in improving the prognosis of these patients. This case also highlights the importance of taking into account the source of the myxoma’s blood supply in relationship to other cardiac structures, and further correlating these findings with clinical symptoms. Cardiac masses are rare and have a wide range of clinical presentations depending on their location, which makes their diagnosis challenging. Cardiac masses can be primary or secondary depending on their origin, and further categorized as benign and malignant. Secondary, or metastatic tumors to the heart, are more common than primary cardiac tumors. Approximately 90% of primary cardiac masses are benign with the remaining 10% malignant. Myxomas are the most common benign cardiac mass and account for approximately half of all benign cardiac tumors . The sizA 67-year-old male with chronic obstructive pulmonary disease (COPD), hypothyroidism, and asymptomatic sinus bradycardia, presented to the emergency department (ED) after experiencing a pre-syncopal episode. Earlier that week the patient developed sudden onset dizziness and light headedness while at dinner. Similar episodes over the past year were reported with worsening night sweats for several weeks. He denied headaches, vertigo, chest pain, shortness of breath, nausea, changes in vision or focal neurologic deficits. The lightheadedness lasted for about 30 s and then self-resolved, but he experienced persistent fatigue for a few hours afterwards. Prior to the episode of dizziness, he denied any notable changes with body position, flushing of the face/body, anxiety, and was not wearing any tight-fitting clothes. An electrocardiogram (ECG) showed sinus bradycardia and referred him to the emergency department for further evaluation.In the emergency department, he was afebrile with a regular pulse of 39 bpm, blood pressure of 161/81, respiratory rate 18, and oxygen saturation was within normal limits on room air. Blood work was unremarkable. ECG showed sinus bradycardia, notched p-waves in limb lead II, and peaked T-waves in precordial leads V3, V4 and V5 with no evidence of heart block or ischemic changes  showed a Left Ventricular Ejection Fraction (LVEF) of 60–65%, a positive bubble study and a mass in the left atrium estimated to be 3 cm × 4.8 cm, that was observed obstructing the mitral valve during diastole Fig. a, b. TheAfter the diagnosis of left atrial myxoma was made the patient was referred for surgical resection. Prior to surgery, coronary angiography demonstrated no significant coronary artery disease but did demonstrate tumor blushing consistent with myxoma. The patient then underwent left atrial mass resection and patent foramen ovale (PFO) repair. In the operating room, the atrial tumor was a large, solid, friable semi-mobile mass with a wide base stalk adherent to the left atrial septum, approximately 5.5 × 5.1 × 3 cm. The procedure was completed without any intraoperative complications. The gross specimen was dark-tan and red, with irregular borders and overall smooth surfaces Fig. . The patWhile patients are frequently evaluated for causes of pre-syncope and syncope, intracardiac masses represent an unusual and rare culprit. In this case the left atrial myxoma was causing intermittent flow obstruction, mimicking severe mitral stenosis when it descended into the plane of the mitral valve (MV) annulus during diastole leading to the patient’s pre-syncopal event. Using velocity time integral method, the calculated mean pressure gradient across the MV was measured to be 1.31 mmHg Fig. a, b. WheAnother unique aspect of this case is the resolution of the patient’s bradycardia after surgical removal of the myxoma. Conduction and rhythm disorders are rare symptoms from cardiac myxomas, occurring in less than 10% of cases , 5. The This case illustrates the importance of having a high clinical suspicion and considering myxomas when determining the cause of symptomatic bradycardia and pre-syncope. The left atrial myxoma received its blood flow from the RCA, which also provides blood to the sinoatrial node. Although the pathophysiological mechanism behind left atrial tumor sick sinus syndrome remains a topic for further investigation, in our case it is possible that the sinus bradycardia is a result of shunting of blood away from the sinoatrial node and towards the myxoma. In support of this, after the left atrial myxoma was removed, the patient’s sinus bradycardia resolved.


The inversion of the uterus is a rare complication of the puerperium and much rarer during the non-puerperal period. The most common cause of non-puerperal inversion is a submucosa leiomyoma; however, identification can be difficult. Herein, we present a case of pedunculated uterine leiomyoma that causes uterus inversion in a 30-year-old virgin woman with a correct diagnosis; a hysterectomy was prevented for the patient. Uterine leiomyoma (UL) is the most common benign tumor of the female genital tract and has become a major health problem . UL occuUL can have many side effects such as dyspareunia , increasOur case report describes a 30-year-old virgin woman presented at the emergency room of Fatemieh Hospital with vaginal bleeding and a protrusion of a mass from the vagina. The patient had a history of severe bleeding in the form of hypermenorrhea, which had intensified the day before the emergency room visit and also felt a feeling of pressure in the perineum and a mass coming out of the vagina. The patient was conscious during the physical examination.The physical examination revealed a body temperature of 37 °C, a heart rate (HR) of 105 beats per minute, a respiratory rate (RR) of 18 beats per minute, and a blood pressure (BP) of 90/60 mmHg. Pelvic examination revealed the presence of a large mass measuring 6-7 cm in diameter with a necrotic appearance and protruding from the cervix and vagina. Necessary tests were requested for the patient . Due to The vagina was examined in the OR after induction of anesthesia. Due to the abnormal thickness and excessive size of the UL pedicel, the UI was suspected following the protruding of the UL from the vagina, so the patient was asked for an ultrasound. Abdominal ultrasound of the uterus in the pelvic cavity was not observed and the diagnosis of UI was confirmed Figures .Therefore, myomectomy was performed carefully from the fundus level for the patient, and samples were sent for pathological examination . The endThe uterus was returned to the cervix and pelvis, and then the uterine suspension was performed to prevent UI to the round ligament Figures . The abdUI are classified into two groups, including (a) puerperal and (b) non-puerperal inversions . Non-pueThe etiology of UI is not well understood. A thin uterine wall, rapid tumor growth, tumor size, fundic localization of the tumor, tumor attachment to the uterine wall with a thin pedicle, dilatation of the cervix due to uterine cavity distension, and quick expulsion of the tumor are all possible factors . Most noImaging techniques such as ultrasound and magnetic resonance imaging (MRI) help with the diagnosis. However, the diagnostic value of transvaginal ultrasound is limited, especially in cases where large mass protrude into the vagina, because of physical limitations; the probe cannot be inserted into a choked vaginal canal; another issue is that ultrasonic beams could not be transferred beyond the mass, resulting in low-quality sonographic images . TherefoReposition procedures, according to the reproductive desire of the patient, or hysterectomy could be considered for surgical treatment . In our


Secretory otitis media is defined as the presence of fluid in the middle ear space causing aural fullness and hearing loss secondary to Eustachian tube obstruction or incomplete resolution of acute otitis media.A 55 years old, otherwise healthy man presented with a history of hearing loss and fullness in the left ear. Otoscopy showed a hyperemic tympanic membrane with fluid build-up behind it in the left ear, while the right ear was normal. Cranial nerve examination did not show any abnormality except for a slight hypoesthesia of the facial area innervated by the second trigeminal branch. Audiometry showed mild sensorineural hearing loss on the right side and moderate mixed hearing loss on the left side. Tympanometry was type “As” on the right side and type “B” on the left side. Nasopharyngoscopy did not reveal abnormalities in the nasopharynx, hypopharynx and at the level of the glottis. The clinical findings were therefore suggestive for serous otitis media and a treatment with systemic steroids, antibiotic and decongestant nasal spray was established. The patient underwent a myringotomy with insertion of transtympanic drainage tube. Three months later, he presented with vertigo associated with nausea and neurovegetative symptoms, with left side hearing loss not associated with postural changes. Vestibular examination showed grade 2 left-beating spontaneous nystagmus with a positive Romberg test on the left side. The clinical findings were consistent with a diagnosis of vestibular neuritis and a brain MRI scan was then performed . The scaThe capsule was then dissected superiorly, where the lesion encountered the middle cranial fossa dura. The lesion was removed. Histopathological examination reported a CD31+ hemangioma of the II trigeminal branch, with benign features. Brain MRI performed after 6 months did not show any evidence of disease recurrency.Unilateral middle ear effusion may result from Eustachian tube compression or invasion rather than from physiologic dysfunction. Gacek identified the four levels of potential Eustachian tube compromise as being the lumen of the Eustachian tube, the nasopharynx, the infratemporal fossa and the petrous apex.To our knowledge, our case seems to be the first case of recurrent secretory otitis media caused by an hemangioma of the II trigeminal branch of infratemporal and pterygopalatine fossa.ab extrinseco Eustachian tube obstruction. In our opinion, it is therefore necessary to add an imaging tool to the diagnostic algorithm, preferably a MRI, in all those cases of secretory otitis media lasting more than 3 months that present a negative nasopharyngoscopy evaluation.At the moment, the diagnostic algorhythm of recurrent secretory otitis media includes, besides audiometry, nasopharyngoscopy as the only instrumental tool. This determines, in a small amount of cases, inability to find a definite cause of the symptomatology, leading the physician to assign this disorder to a more generic tubaric dysfunction. Actually, even if rare, serous otitis media may be caused by the presence of cranial base masses that determine an The authors declare no conflicts of interest.


Cystic lymphangioma is a benign tumour that occurs secondary to obstruction of lymphatic channels. Its appearance in the paediatric age group is quite common, but adulthood presentation is infrequent. Common locations are head and neck areas, whereas intra-abdominal occurrence is rare. To date, a few retroperitoneal cystic lymphangioma cases have been reported. A pre-operative clinical detection is always confusing, and most often, the diagnosis rests over the intraoperative findings and histopathological examination. The cyst's complete surgical resection remains the treatment of choice in patients with bulky, rapidly growing lesions or symptoms. Herein, we report a large retroperitoneal cystic lymphangioma that mimicked the mesenteric cyst clinically. According to the image findings, primary retroperitoneal masses do not originate from a specific retroperitoneal organ and are divided into two major groups: solid and cystic. Retroperitoneal cysts are rare in incidence and can be neoplastic or non-neoplastic. Neoplastic retroperitoneal lesions include lymphangioma, mature teratoma, cystic mesothelioma, cystadenoma, and cystadenocarcinoma. On the contrary, epidermoid cyst, nonpancreatic pseudocyst, bronchogenic cyst, lymphocele, haematoma, and urinoma are non-neoplastic lesions .The incidence of retroperitoneal lymphangioma is about 1%. Clinical presentation is usually asymptomatic because of its slow growth rate. Symptomatic cases, although few, present with abdominal pain, lump abdomen, and local compressive symptoms. Large cysts may present as acute abdomen with features of intestinal obstruction, torsion, or bleeding . BecausePatient information: a 65-year-old male patient of Indian ethnicity presented in the general surgery outpatient department with a left upper abdominal lump associated with dull aching continuous non-radiating pain around the umbilicus over three months. On questioning, he denied any history of vomiting, haematemesis, constipation, bleeding per rectum, melena, fever, anorexia, loss of weight, or difficult micturition.Clinical findings: physical examination of the patient revealed a moderate built and an apparent fullness over the left hypochondrium that extended towards the umbilicus. An ill-defined lump of 10 x 7 cm, spanning the entire left hypochondrium and extension towards the umbilicus and the epigastrium, could be palpated on abdominal examination. The swelling was non-tender, smooth-surfaced, firm in consistency, moving horizontally but immobile vertically with no local temperature rise. On percussion, a zone of resonance was noteworthy around the lump. Examination of the bilateral renal angles, lumbar regions, and back was unremarkable. No generalized lymphadenopathy was present.Diagnostic assessment: considering mesenteric cyst and retroperitoneal cyst as differential diagnoses, an ultrasound abdomen was done that showed a cystic mass of size 12 x 11 cm with septations noted on the left side above the level of the umbilicus. Spleen, pancreas, and bilateral kidneys are normal. Contrast-enhanced computed tomography (CECT) showed evidence of a well-defined thin-walled lesion of 13x10x12 cm in the left anterior para renal space. The lesion abutted the left kidney and renal vessels posteriorly, the spleen laterally, the left lateral abdominal wall anteriorly, and the bowel loops medially. The adjoining mass had also displaced the left ureter medially in its upper and mid-part without any evidence of hydronephrosis and psoas muscle involvement  and axilla (20%). Approximately 5% are intra-abdominal, where it can arise from mesentery, retroperitoneum, and greater omentum [Lymphangioma is a rare type of benign malformation of lymphatic channels. Its incidence is expected in the paediatric population but rare in the adult population. For the first time, cystic lymphangioma was reported in 1913 by d axilla 0%. ApproClinical presentation is asymptomatic in most cases. When symptomatic, they present as vague abdominal pain, slowly growing mass, nausea/vomiting, and anorexia . Large cRadiological investigations like ultrasound, computed tomography (CT), and magnetic resonance imaging (MRI) complement. An ultrasound shows a sharply delineated fluid-filled cystic lesion with septations. CT and MRI can demonstrate uni or multiseptated lesions, relationships with the adjacent organs, and characteristic water density fluid content . In the Complete excision of the cyst is the standard gold treatment . Other mThe final tissue diagnosis is arrived at after histopathological examination of the excised cyst. The characteristic features include an endothelial lined well-circumscribed cyst wall containing focal lymphoid tissue aggregates and stroma composed of collagen fibrous meshwork . AccordiPatient perspective: the patient shared his perspective on the treatment in terms of pain relief and disappearance of the swelling.Informed consent: the patient gave written informed consent to publish his anonymized information in this article.Cystic Lymphangioma is a rare benign tumour that occurs due to abnormal lymphatic channels. Adult and intra-abdominal incidence of cystic lymphangioma is infrequent. Because of rarity and non-specific clinical presentation, it poses diagnostic challenges. It often requires surgical excision and histopathological examination for a confirmed diagnosis.


Ewing’s sarcoma is a part of a rare group of malignant neoplasms, whose pathological morphological features are small round cells. Extraskeletal Ewing’s sarcoma is a more uncommon primary tumor. Herein, we report the case of a 66-year-old man who complained of chest tightness. Subsequent chest CT scans revealed an irregular and uneven density mass on the right side of the anterior mediastinum with invasion of the superior vena cava, pericardium and right lung. The patient’s clinical symptoms were improved after performing excision of the mediastinal lesions under cardiopulmonary bypass. Based on histological and immunohistochemical findings, the tumor was diagnosed as extraskeletal Ewing’s sarcoma. Extraskeletal Ewing’s sarcoma (EES) is a small round cell malignant tumor that grows outside of bone tissue, which arises from the mediastinum is extremely rare. Compared with patients with bone tumors, patients with EES have an older average age of onset. The clinical features and imaging appearances are non-specific, and patients often present with a rapidly growing mass that causes localized pain . The diaA 66-year-old man with an anterior mediastinal tumor was admitted to the hospital for surgical treatment. He went to the emergency department of the hospital 1 day prior because of chest tightness, and plain chest CT showed a mass on the right side of the anterior mediastinum that compressed the adjacent right lung, accompanied by pleural effusion (Intraoperative exploration revealed that the tumor had invaded the superior vena cava, pericardium and right middle lung, so the surgeon adopted resection of the mediastinal lesions under cardiopulmonary bypass, partial resection of the superior vena cava with artificial blood vessel replacement and lobectomy of the right middle lung with lymph node dissection. The pathological findings confirmed that it was a small round cell malignant tumor (Because the circulatory failure caused by pericardial tamponade was difficult to cure, the family members of the patient finally refused further invasive surgery for the patient; only noninvasive ventilators were used to maintain the patient’s vital signs, and he had entered the end stage of clinical life.MIC gene, is a relatively specific antibody of the ES family. However, CD99 expression is not specific to ES, as it can be expressed by other tumors, such as ependymoma (EWS-FLI-1 fusion gene (EES is a rare entity with high-grade malignancy that commonly involves the soft tissues of the trunk and extremities. It is more common in the lower limbs, paraspinal, retroperitoneum and chest wall and rarely occurs in solid organs. Primary mediastinal EES is extremely rare , 3. Therendymoma . To the ion gene . The imaion gene . Howeverion gene . Treatmeion gene . The pation gene .Most cases of primary EES in the mediastinum appear as masses upon diagnosis with no specific clinical symptoms. On imaging, these tumors are usually large, enveloped, soft tissue density masses that can compress or invade adjacent tissues and easily metastasize, and an enhanced scans can show inhomogeneous moderate or significant enhancement. Imaging can be performed as a diagnostic tool, but the final diagnosis is mainly based on pathology and immunohistochemistry. In terms of treatment, surgical resection combined with chemotherapy, radiotherapy or both is the best solution.The original contributions presented in the study are included in the article/Supplementary Material. Further inquiries can be directed to the corresponding author.Written informed consent was obtained from the participant for the publication of any potentially identifiable images or data included in this article.MC, DZ, XZ, YL, TW, LW, GF, SJ, and WC conceived the idea for the article. MC drafted the manuscript. WC approved the final version of the manuscript. All authors contributed to the article and approved the submitted version.


A traumatic neuroma occurs at the injury site of a peripheral nerve; however, albeit rarely, this variant of a neuroma can involve a nerve that has not experienced penetrating trauma. A lower extremity amputation stump is the most common location of a traumatic neuroma. Traumatic neuromas may be symptomatic; tumor-associated pain can be severe and significantly affect the patient’s quality of life. Several hypotheses have been postulated for the pathogenesis of neuroma-related pain, including alpha-smooth muscle actin, neural fiber structural changes, nerve growth factor, and/or sensitization of the affected nerve. In addition to prevention, non-surgical treatment  and surgical interventions  have been used to manage neuroma-associated pain. A traumatic neuroma of the nose is rare. Indeed, it has only been described in three individuals: two women (including the Caucasian woman in this report and a Turkish woman) and one man. The benign tumor was extremely painful in both women; however, the man’s lesion was non-tender. Prior trauma to the nasal site included either a laceration or elective surgery; however, the reported woman did not experience any penetrating trauma to her nose. The diagnosis was established following an excisional (for the man), incisional (for the Turkish woman), or punch (for the Caucasian woman) biopsy. Follow-up was provided for two of the patients. The man’s neuroma had been completely excised, and he never developed tumor-associated tenderness. However, the pain persisted after the biopsy healed for the reported woman whose neuroma was not entirely removed. The explosive and markedly severe character of the reported patient’s lesion-related tenderness prompted us to propose an acronym for this uncommon yet exquisitely painful variant of a neuroma: tender nasal traumatic (TNT) neuroma. A neuroma is a benign lesion. It is composed of nerve tissue. The nasal mucosa can be the site of neuromas in patients with multiple endocrine neoplasia 2B (MEN2B) -3. A traumatic neuroma is a variant of the tumor that usually develops following injury to the nerve -9. They The characteristics of a woman with a tender nasal traumatic (TNT) neuroma are described. Also, the features of other patients in whom a traumatic neuroma of the nose has been reported are summarized. In addition, the approaches to the management of an individual with a painful traumatic neuroma are reviewed.A 64-year-old Caucasian woman presented with severe tenderness of her nose. She had rosacea, affecting her cheeks and nose, of several years duration. Her nose had been painful--particularly when touched--for approximately three years; the symptoms began as a tender spot on the nasal tip, had progressed to involve her entire nose and currently felt as though there were needles inside her nose that were sticking her. In addition, she was especially uncomfortable when the facial mask she was wearing, as a precautionary measure against coronavirus disease 2019 (COVID-19), would contact her nose. However, at times the severe tenderness would suddenly erupt in an explosive manner without any precipitating factor.She had previously been evaluated by her primary care physician, dermatologists, and otolaryngologists without establishing a diagnosis for her nose pain. She had no history of prior surgery or penetrating trauma to her nose; however, she mentioned that recurrent episodes of redness and soreness--possibly caused by insect bites, inflammation of the hair follicles, or flares of her rosacea--had occurred. Daily treatment with oral doxycycline for one month provided no relief of her symptoms. The medications she took to relieve the pain resulting from some of her medical conditions did not improve her nasal symptoms.Her medical history was significant for diabetes mellitus, fibromyalgia, hyperlipidemia, hypertension, hypothyroidism, and lower back pain. Her daily oral medications included aspirin, atenolol, atorvastatin, gabapentin, metformin. In addition, she would take naproxen or tramadol when the pain from her fibromyalgia or lower back became more severe.Cutaneous examination of her face showed malar erythema. Her nose was large with an erythematous bulbous tip without rhinophymatous changes; there was a prominent papule, which measured four-by-four millimeters, with a central depression at the nasal tip Figure . A threeMicroscopic evaluation of the tissue specimen, after staining with hematoxylin and eosin, showed a patulous follicular opening in the upper portion of the specimen. The follicle structure contained a widely dilated sinus-like epithelial tract lined by acanthotic epidermis. Adjacent to the follicle, there were numerous prominent sebaceous glands Figure .In the deeper dermis, there was a proliferation of bundles of spindle-shaped cells. The cells were characteristic of nerve fibers. They appeared as bundles with an irregular interlacing pattern and were present in the fibrous dermal stroma Figures , 3. Immunoperoxidase staining was performed to confirm the identity of the spindle-shaped cells. The solubility of proteins in 100 percent ammonium sulfate (S100) stain showed strongly positive diffuse staining that highlighted the cells of the nerve bundles in the reticular dermis Figure . The cluCorrelation of the patient’s clinical history, lesion morphology, and pathologic findings established a diagnosis of a traumatic neuroma with an overlying dilated pore. Most of the benign dermal neural neoplasm had been removed during the biopsy. However, residual tumor extended to the lateral margins of the specimen.The patient continued to experience pain in her nasal tip after the biopsy site had healed. A trial of intralesional triamcinolone acetonide was suggested; however, she refused to have any injections into her nose. Her nose pain had occurred while she was using gabapentin; therefore, treatment with pregabalin was not recommended. Oral carbamazepine was suggested; she appreciated the recommendation and decided to follow up with the doctor who managed her fibromyalgia and back pain for alternative non-surgical interventions for her nasal tenderness.A traumatic neuroma is typically associated with either a prior amputation  or an acute injury to the peripheral nerve -9. HowevMost traumatic neuromas are asymptomatic and occur post-amputation on the lower extremity -6,11. HoThe nose is an extraordinarily rare location for a traumatic neuroma. It is remarkable, considering the large number of elective cosmetic procedures performed on the nose, that only three individuals with a symptomatic traumatic neuroma at this site have been described. The characteristics of the patients with a traumatic neuroma of the nose are summarized in Table The patients with a nasal traumatic neuroma included two women and one man. One of the women was Turkish, and her age at neuroma diagnosis was not stated. The other woman was Caucasian, and she was 64 years old when her neuroma was diagnosed. The man was also Caucasian; his neuroma was diagnosed at age 16 years ,15.The Caucasian woman has multiple medical problems. These included diabetes mellitus, fibromyalgia, hyperlipidemia, hypertension, hypothyroidism, and low back pain. The other patients did not have any comorbidities ,15.The onset age of the neuroma varied. The man was four years old when he lacerated his left upper lip, and it was sutured. Neuroma symptoms  began when he experienced blunt trauma to the same lip site at age 11 years. The mass progressively enlarged, beginning at age 14 years; he was 16 years old when he was evaluated [The Turkish woman had an aesthetic rhinoplasty; within four weeks after the surgery, she developed pain localized to the columella and membranous septum of her nose. The pain not only persisted but also continued to increase postoperatively. One year after her operation, the pain had become intolerable .The Caucasian woman’s symptoms began when she was 61 years old. She had not experienced a penetrating injury or surgical operation on her nasal tip; however, the location was the site of recurrent insect bites, inflamed hair follicles, and rosacea flares. She had several evaluations since the onset of her unresolved nasal pain; the diagnosis of neuroma was finally established when she was 64 years old.The duration of the nasal neuroma ranged from one year to 12 years  before confirming the diagnosis. Both women had severe and unabating pain localized to the site of the tumor. The man’s lesion was painless ,15.The morphology of the benign neoplasm varied. The Turkish woman had a normal-appearing nose that contained neuromatous tissue that was discovered after a transfixion incision at the caudal end of the septal cartilage of her nose was performed. The other woman had a four-by-four millimeters umbilicated papule on the erythematous and bulbous tip of her nose. The man had a firm 1.0 by 1.5-centimeter nodule on the left medial crus of his nose ,15.A traumatic neuroma is a benign non-encapsulated neoplasm; a microscopic examination of the lesion shows a disorganized and tangled tumor composed of bundles that contain axons, endoneurial cells, nerve fibers, and perineurial cells surrounded by fibroblasts, collagenous connective tissue, and/or scar -6,10. ThImmunoperoxidase studies were only performed for Caucasian women. Similar to another traumatic neuroma, diffuse, strongly positive staining of the tumor cells was demonstrated with S100 stain -18. In aThe neuroma was completely removed during the excisional biopsy of the man’s tumor. Additional therapy, following the incomplete removal of the Caucasian woman’s neoplasm, was declined by the patient. Neuroma treatment was not described for Turkish woman ,15.The biopsy site on the nasal tip of the Caucasian woman healed without any complications. However, after the biopsy of her neuroma, she continued to have persistent pain. The follow-up for the Turkish woman was not described ,15.The man experienced blunt trauma to the excision site of his neuroma, and a similar-sized asymptomatic nodule developed. An incisional biopsy showed fibrosis accompanied by chronic inflammation and a foreign body reaction. The site completely healed--with a symmetric columella and without any residual mass or discoloration--following two monthly injections of triamcinolone acetonide .The pathophysiologic mechanism for the development of pain associated with a neuroma remains to be established. Several hypotheses--which are not necessarily mutually exclusive--have been postulated. These include not only peripheral and central sensitization of the affected nerve but also the involvement of alpha-smooth muscle actin, nerve growth factor, and/or structural changes in neural fibers ,7.The management of a painful neuroma includes prevention, non-surgical treatment, and surgical intervention -13. PrevNon-surgical treatments include systemic or local pharmacologic agents, local chemical interventions, cryotherapy, and physiotherapy. Oral drug therapies include alpha-receptor agonists, carbamazepine (a sodium channel blocker), pregabalin , and opioids. Injectable intralesional agents that have been used are anesthetics, corticosteroids, etanercept, or onabotulinum toxin A ,5,7,8.Topical medications have included analgesics  and anesthetics (such as lidocaine). In addition to topical regional application, other routes of drug delivery for the management of a traumatic neuroma include intrathecal injection and intravenous infusion. Another treatment for a traumatic neuroma that is not specifically localized to the neuroma involves electrode-implantation into the spinal cord and subsequent pulse current stimulation ,7. Chemical interventions for a traumatic neuroma include not only intralesional corticosteroids but also ablation with neurolytic agents such as alcohol and phenol; however, leakage and local tissue necrosis are potential adverse events associated with ablation ,7. DecreThere are several potential surgical interventions for a traumatic neuroma. Direct repair at the time of injury--before the development of the neuroma--is optimal. Otherwise, the simplest surgical method for treating a traumatic neuroma is ligation of the nerve ending; after proximal ligation, the nerve can remain in a site that will minimize subsequent irritation. Alternatively, there are many options for the management of the distal free end of the proximal nerve: burying it in muscle or bone, attaching it to a vein, capping the nerve end, reconstruction of the nerve continuity, or covering the nerve with a local skin flap ,6-9,13.An acronym is a mnemonic device that is used as a memory aid. Explosives--such as 2,4,6-trinitrotoluene --are used for industrial, military, and mining applications. The dramatic  manifestation of tumor-associated pain from our patient’s traumatic neuroma of the nose prompted us to introduce the acronym for this lesion: TNT neuroma.A traumatic neuroma may be associated with severe pain. It typically occurs at the site of a prior amputation of the lower extremity. However, albeit rarely, a traumatic neuroma has been described on the nose. A Caucasian woman with an extremely painful nasal traumatic neuroma on her nasal tip is reported; the other two patients with this tumor on their nose include a Turkish woman and a Caucasian man. The traumatic neuroma of the nose was extraordinarily tender (in both women) and occurred following penetrating injury to the skin (in the Turkish woman and the man). Evaluation of the tissue specimen obtained after a biopsy of the affected area established the diagnosis. Follow-up was provided for two of the patients; the man never developed tumor-associated tenderness and his neuroma had been entirely removed, whereas even after the biopsy healed, excruciating pain persisted for the reported woman whose neuroma had not been completely excised. Prompted by the reported patient’s explosive and markedly severe lesion-associated pain, we proposed the acronym TNT neuroma to emphasize the symptom (tender), the location , and the variant (traumatic) of this rare type of neuroma.


Oral metastases are rare; nevertheless, they must be considered in the differential diagnosis of lesions in patients with a previous history of malignancy or older adults. The clinical signs of oral metastasis typically comprise pain, dysphagia, ulceration, bleeding, and paresthesia. Soft tissue sarcomas tend to affect the extremities and retroperitoneum. The most common metastases in the oral cavity are carcinomas, and sarcomas rarely metastasize to this area. Undifferentiated pleomorphic sarcoma (UPS) is a mesenchymal malignancy that infrequently affects the head and neck site. It shows a male predilection and occurs in all age groups. The lung is the most common area of distant metastasis in undifferentiated pleomorphic sarcoma. This report presents a 61-year-old female patient with a painful bluish-purple mass of the posterior right mandibular alveolar mucosa. She had a history of thigh UPS about four years ago. An incisional biopsy was performed, and the specimen was stained with hematoxylin and eosin. Immunohistochemical antibodies for CK, S100, desmin, myogenin, MDM2, SOX10, and caldesmon were negative and focally positive for CD68. Ki-67 index was about 70%. This report aimed to increase awareness of a rare lesion by describing the clinical, histopathologic, and immunohistochemical findings of metastatic undifferentiated pleomorphic sarcoma of the oral cavity. Sarcomas are rare malignant tumors with aggressive behavior originating from mesenchymal cells . Soft tissue sarcomas can occur anywhere in the body; however, the most common site is the extremities and retroperitoneum. The most common microscopic subtypes are liposarcoma, pleomorphic sarcoma, leiomyosarcoma, rhabdomyosarcoma, and synovial sarcoma. Metastasis of sarcoma is mainly hematologic, and approximately 90% of distant metastases occur in the lung. In sarcomas’ of extremity, metastasis occurs in about 25% of cases, especially if the primary tumor is deep and close to the fascia  staining and molecular analysis, the classification of sarcomas is still challenging, with approximately 20% of all sarcomas being unclassified . More thMetastatic sarcomas are rare in the oral cavity; carcinomas are the most common metastases. Although oral metastases have a poor prognosis, the timely diagnosis of these malignancies is essential to the patient . UndiffeA 61-year-old woman presented a painful mass of the posterior right lower jaw. The extra-oral examination was normal, whereas she mentioned the pain in the chin area and lip paresthesia. There was no cervical lymphadenopathy. *3/µl). ESR was within the normal range (11 mm/hrs), and CRP was 3+. Intraoral examination showed a painful bluish-purple mass covered by mucosa with soft consistency extended from distal of right second premolar to retromolar pad region measuring about 2cm×1cm with two months duration  for CK, S100, desmin, myogenin, MDM2, SOX10, and caldesmon was negative and focally positive for CD68. Ki-67 index was about 70% .Based findings mentioned above, a diagnosis of metastatic UPS was made, and a whole-body scan was recommended to identify other metastatic lesions. Unfortunately, the patient died of Covid-19 infection before performing a whole-body scan and completing treatment.Soft tissue sarcoma metastasis to the oral cavity is exceedingly rare, with only a few cases reported in the literature ,3,5-7. OOur patient also suffered from a painful mass with lip and chin numbness. A numbed chin is a significant diagnostic symptom of metastatic lesions . In dentKaplan et al.  mentioneIn the current case, local recurrence, lymph node involvement, and distant metastasis were observed. UPS is a diagnosis of exclusion. This neoplasm does not have distinct histological characteristics and may reveal a fascicular, storiform, or sheet-like proliferation and frequently shows an admixture of spindle and pleomorphic, rarely multinucleated cells, with palely eosinophilic or slightly basophilic cytoplasm . The preMetastatic sarcoma to the oral cavity is a rare phenomenon, with few cases reported in the literature. However, the possibility of a metastatic lesion should be considered in patients complaining of paresthesia (numb chin syndrome). Familiarity with these lesions' clinical and microscopic characteristics is critical for clinicians and pathologists to have a proper and timely treatment plan for the patients.