Pancreatic mixed serous neuroendocrine neoplasm (PMSNN) is an extremely rare disease. Only a few cases on the surgical treatment of PMSNN have been reported in the literature, and it is unclear whether there is invasion of important peripancreatic vessels.We report the case of a 39-year-old female patient with PMSNN accompanied by invasion of important peripancreatic vessels. She underwent surgery and achieved satisfactory recovery.Abdominal enhanced CT images showed an enhanced mass with a nonenhanced cyst involving the head and body of the pancreas, which invaded important peripancreatic vessels. The lesion had been misdiagnosed and mistreated as a metastatic carcinoma before admission.CT 3-dimensional (3D) visualization reconstruction images showed intact peripancreatic vessels. Radical pancreatoduodenectomy was successfully performed and confirmed that the main blood vessels around the pancreas were only compressed or even wrapped by the mass, but not penetrated.The patient recovered well and was discharged on the 19th day after surgery. Pathological examination reported the diagnosis of PMSNN with the collision type combination and the well-differentiated grade 2 pancreatic neuroendocrine tumor. She was followed up for 18 months without any abnormalities.This case demonstrates that surgical treatment of PMSNN with invasion of peripancreatic vessels can be successful. Preoperative abdominal CT 3D visualization reconstruction is helpful in determining the degree of invasion of important peripancreatic vessels, and plays a key role in formulating an accurate surgical plan and improving patient outcome. In the 2019 World Health Organization (WHO) Digestive System Tumor Classification, NEN was divided into neuroendocrine tumor (NET) and neuroendocrine carcinoma (NEC). Due to the malignant potential of the NEN component, and its close relationship with Von Hippel-Lindau (VHL) syndrome, patients with MSNN usually undergo thorough systemic examinations, surgical treatment, and close clinical follow-up.Mixed serous neuroendocrine neoplasm (MSNN) is a mixed tumor containing 2 components with different pathologies,–5 and it is unclear whether there is invasion of important surrounding vessels. In addition, there are no reports on the surgical treatment of peripancreatic vessels invasion. We report the case of a 39-year-old female PMSNN patient, without obvious symptoms, with invasion of important peripancreatic vessels and the collision type combination manifestation, but not VHL syndrome.Pancreatic mixed serous neuroendocrine tumor (PMSNN) is an extremely rare disease. It is very difficult to determine the benignity and malignancy of its NEN component before surgery. In theory, once the diagnosis of PMSNN is made, surgical treatment is preferred. However, so far only a very few cases of surgical treatment for PMSNN have been reported in the literature,A 39-year-old female patient was admitted to Beijing Electric Power Hospital on December 11, 2020 with a 6-mon history of the inadvertent discovery of an abdominal mass.The study has been approved by the ethics committee of Beijing Electric Power Hospital, State Grid Corporation of China, Capital Medical University, P. R. China. The patient has provided informed consent for publication of the case.Six months ago before admission, the patient inadvertently found a fist-sized abdominal mass, without abdominal pain, diarrhea, fever, or other obvious discomfort. She was hospitalized twice in a Beijing famous hospital. Abdominal enhanced computed tomography (CT) and magnetic resonance imaging (MRI) showed an enhanced mass invading important surrounding blood vessels in the omental sac area, with uneven metabolism on positron emission tomography (PET)-CT. Combined with a history of “left oophorectomy for ovarian borderline mucinous cystadenoma canceration (well differentiated mucinous adenocarcinoma)” 11 months previously, and no postoperative chemotherapy was administered, the metastatic carcinoma of the lesser omental sac was diagnosed. The opinion of the multidisciplinary team was “Metastatic carcinoma of the lesser omental sac has high surgical risk and poor expected effect. Radiotherapy and chemotherapy are recommended”. She was discharged after resolutely refusing radiotherapy and chemotherapy.During the past 6 months, the abdominal mass gradually increased without obvious weight loss, fatigue or other discomfort. She had no history of smoking, drinking, or family disease, and no contact history of COVID-19 pneumonia.After admission, her physical examination found that the skin and sclera did not show yellow staining, no tenderness or rebound pain was observed in the abdomen, the palpable mass under the xiphoid process of the upper abdomen was tough, smooth, inactive, and without tenderness, no ascites were observed. The laboratory examinations showed that blood sugar level was 5.84 mmol/L. Liver function tests showed that alanine aminotransferase was 22 U/L, aspartate aminotransferase was 15 U/L, total protein was 66.3 g/L, albumin was 40.1 g/L, total bilirubin was 8.85 μmol/L, direct bilirubin was 1.27 μmol/L, and γ-glutamyl transpeptidase was 60U/L. The levels of tumor markers were carbohydrate antigen 19–9 8.20 U/mL, carcinoembryonic antigen 0.828 ng/mL, pro-gastrin-releasing peptide 34.9 pg/mL, and neuron specific enolase 12.5 μg/L. The imaging examinations showed that abdominal enhanced CT images showed an enhanced mass with a nonenhanced cyst involving the head and body of the pancreas , and reports then gradually increased.,8 The 2010 WHO classification of pancreatic tumors clearly puts forward the concept of mixed serous neuroendocrine tumor (MSNN), meaning that SCN and NEN occur simultaneously in the pancreas, and MSNN is listed as a new subtype of SCN.,4 However, due to a lack of understanding of this tumor in clinical practice, it is often misdiagnosed and the diagnosis is missed.Keel SB et al,4,10: (1) Diffuse subtype: The entire pancreas is occupied by numerous PSCN associated with one or more PanNET, which could be located in any region of the pancreas; (2) Mixed subtype: The 2 different tumors combined with each other in a mass cannot be distinctly divided; (3) Solitary subtype: The 2 different components coincide in the pancreas without any intermingling area; and (4) Collision subtype: The 2 different lesions are separated from each other in most parts, but with a partially intermixed or overlapping zone. PanNET is further graded on a scale of 1–3 based on mitotic rates and Ki67 proliferation index as follows: PanNET grade 1 (G1) with <2 mitoses/10 HPF and a <3% Ki67 index; PanNET grade 2 (G2) with 2–20 mitoses/10 HPF and a 3–20% Ki67 index; and PanNET grade 3 (G3) with >20 mitoses/10 HPF and a > 20% Ki67 index. Accordingly, after reviewing the pathological images of the previous case reports of PMSNN, all the PanNET components diagnosed as “low-grade/well-differentiated malignant NET”or “well-differentiated NEC” should be reclassified as well-differentiated PanNET rather than a poorly differentiated PanNEC. Therefore, our case belongs to the Collision subtype of PMSNN and the well-differentiated grade 2 PanNET.PMSNN is termed mixed tumors containing 2 components with different pathologies, PSCN and PanNET. Based on the distribution pattern of PSCN and PanNET, PMSNN is categorized into 4 subtypes–13 which can manifest as simple cysts, SCN and NEN. The malignant transformation rate of SCN is low, and asymptomatic patients do not need to be treated. Most NEN have no function but have a tendency for malignant transformation, which requires careful treatment, including regular follow-up, surgical treatment and drug treatment. At present, there are no special guidelines for pancreatic lesions in VHL syndrome. Before hospitalization, although this patient had a history of “left oophorectomy for ovarian borderline mucinous cystadenoma canceration (well differentiated mucinous adenocarcinoma)” 11 months previously, no lesions in other organs were found during preoperative examination, and no abnormalities in other organs were found during the 1 year postoperative follow-up period. In addition, VHL gene detection was normal, therefore VHL syndrome was excluded.PMSNN is an extremely rare disease and is often associated with VHL syndrome, which is a rare and autosomal dominant disease with a tendency to become cancerous. The incidence of VHL syndrome is approximately 1/36,000 to 1/45,500. Approximately 60% of patients with VHL syndrome have pancreatic lesions, suggested that the surgical strategy of nonfunctional PanNET usually depends on the tumor size and pathological grade. For the patients with G1 and G2 PanNET whose maximum tumor diameter is <2 cm, imaging follow-up can be carried out every 6 to 12 months on the premise of full communication with patients and their families; for those with G2 PanNET, surgical treatment should be considered. In the follow-up period, if patients have significant tumor growth , and show evidence of regional lymph node metastasis or signs of local invasion, and pancreatic duct dilatation or obstructive jaundice, immediate surgery should be carried out. The choice of surgical method depends on the general condition of the patient, the location of the tumor, and the number of tumors. For nonfunctional G1 and G2 PanNET with a small maximum diameter, local tumor resection and minimally invasive surgery are recommended, which have advantages in reducing intraoperative bleeding, shortening operation time and protecting pancreatic secretory function, and the long-term prognosis of patients is not statistically different from that of patients receiving conventional pancreatic resection. For PanNET located in the head and uncinate process of the pancreas, or adjacent to the main pancreatic duct, or multiple tumors in the pancreas, it is generally recommended to perform conventional pancreatectomy and dissect the peripancreatic lymph nodes.Since this case belongs to a simple PMSNN unrelated to VHL syndrome, and the NET component is a well-differentiated G2 PanNET, it is necessary to further explore the treatment principles of nonfunctional PanNET. The Chinese guidelines for the diagnosis and treatment of pancreatic neuroendocrine neoplasms (2020) For PanNET of the pancreatic head and uncinate process, pancreaticoduodenectomy is recommended, and pancreatic head resection with organ preservation can also be performed under specific conditions according to the maximum diameter and extent of tumor invasion. Segmental pancreatectomy is feasible for tumors in the body of pancreas. For tumors in the body and tail of the pancreas, distal pancreatectomy (including combined splenectomy) is recommended.For nonfunctional PanNET with a maximum diameter ≥ 2 cm, conventional pancreatectomy and regional lymph node dissection are recommended. Abdominal multi-slice spiral CT (MSCT) enhanced scanning, combined with 3D reconstruction technology can obtain images of anatomical structure around the pancreas, which can accurately reflect the pancreatic mass itself and the main blood vessels around the pancreas. In our patient, abdominal MSCT was used to enhance scanning, combined with 3D reconstruction, including MIP and 3D visualization reconstruction, to examine the main blood vessels around the pancreas. Abdominal enhanced CT images showed the enhanced mass with a nonenhanced cyst involving the head and body of the pancreas, which invaded important surrounding blood vessels. Coronal surface MIP images revealed that the soft tissue mass invaded the common hepatic artery, superior mesenteric artery, inferior mesenteric artery, portal vein, and superior mesenteric vein. However, 3D visualization reconstruction images showed that the vessel walls of the portal vein, superior mesenteric vein, and superior mesenteric artery were intact. Radical pancreatoduodenectomy confirmed that the main blood vessels around the pancreas were compressed or even wrapped by the mass, but the blood vessel walls were not penetrated.Pancreatic cancer easily expands into the retroperitoneal space, involving the main arteries and veins around the pancreas. This is of great significance in determining whether the pancreatic cancer can be surgically removed and for evaluating the prognosis of patients. Therefore, it is very important to accurately evaluate the main blood vessels of the pancreas during preoperative imaging.Therefore, with regard to PMSNN invading peripancreatic vessels, it is worthwhile operating on the basis of preoperative abdominal CT 3D visualization reconstruction evaluation. Even if abdominal enhanced CT shows that important peripancreatic vessels are invaded, this is not an absolute contraindication. In addition, even if there is a history of left ovarian borderline mucinous cystadenoma canceration, it is not necessarily metastatic cancer. If metastatic cancer is present, but not extensive metastasis, surgical exploration in the presence of simple vascular invasion is still possible. If there is vascular compression, surgery is of great significance. If the blood vessels are infiltrated, wrapped and fused, replacement with artificial blood vessels and allogeneic blood vessels may prolong the life of patients. suggested that all PanNET patients should be followed up regularly. For low-risk PanNET patients who did not receive surgical treatment, the follow-up is to monitor the progress of primary tumor and possible tumor metastasis. For PanNET patients undergoing radical surgery, the follow-up is to exclude the tumor recurrence in situ and metachronous tumor metastasis.The prognosis of PMSNN is associated with the classification, grading and staging of tumors. The Chinese guidelines for the diagnosis and treatment of pancreatic neuroendocrine neoplasms (2020)In summary, this case demonstrates that surgical treatment of PMSNN with invasion of peripancreatic vessels can be successful. Preoperative abdominal CT 3D visualization reconstruction is helpful in determining the degree of invasion of important peripancreatic vessels, and plays a key role in formulating an accurate surgical plan and improving patient outcome.Conceptualization: Zongming Zhang, Limin Liu, Youwei Li.Investigation: Zongming Zhang, Limin Liu, Youwei Li, Zhuo Liu, Chong Zhang, Yue Zhao, Mingwen Zhu, Baijiang Wan, Hai Deng, Xiyuan Xie, Kun Tian, Zhentian Guo, Haiyan Yang, Jiahong Liao, Hongyan Zhu, Lili Liu, Man Wang, Xiaoting Ma, Tiantian Liu, Niuniu Huang, Yujiao Gao, Jing Zhao, Fang Liao, Fengyuan Li, Xueting Wang, Ruijiao Yuan, Xinying Liu, Lidan Chang.Funding acquisition: Zongming Zhang.Supervision: Zongming Zhang.Writing—original draft: Zongming Zhang, Limin Liu, Youwei Li.Writing—review & editing: Zongming Zhang.


Proliferative fasciitis is an extremely rare benign myofibroblastic proliferation that typically presents as a rapidly growing subcutaneous mass. Precise histopathological interpretation is required to obtain a proper diagnosis. Due to a symptomatology overlap, discrimination from soft tissue sarcomas is crucial in the prevention of unnecessary excessive treatment that could be potentially harmful to the patients. Here, we present a rare case of atypical localization of proliferative fasciitis. The lesion was predominantly localized in the groin with the invasion of the scrotum and clinically mimicked soft tissue sarcoma. However, according to a proper histopathologic analysis, the diagnosis of proliferative fasciitis was concluded. With a large number of pseudosarcomatous lesions, there is a rising urge to introduce these rare but benign processes to physicians in order to prevent misdiagnosing patients. Proliferative fasciitis and other pseudosarcomatous lesions are rare benign myofibroblastic proliferations that display similar clinical and histological features to soft tissue sarcomas -3. MargiWe present a 70-year-old male patient who was originally reported as a suspected case of soft tissue sarcoma. The lesion presented as a palpable, fast-growing and mildly painful mass predominantly localized in the groin with the invasion of the scrotum. The skin above the lesion remained intact. To evaluate the extent of the lesion, an MRI scan with a contrast dye focused on the thigh, groin, and pelvis was performed Figure .As observed, the lobular tumorous expansion of 60x55x75mm in size was localized in the groin, contained both solid and cystoid components, and extended to the radix of the penis. The lesion was further ongoing to the upper end of the left part of the scrotum dislocating the spermatic cord laterally. However, signs of the infiltration of the spermatic cord or the surrounding tissues were not shown. Lymphadenopathy was ruled out. The next step included a 14G core needle biopsy (CNB). All samples displayed a proliferation of plump spindle fibroblastic/myofibroblastic cells with large nuclei and prominent nucleoli Figure .Among those cells, large cells with irregular shapes and abundant amphophilic to basophilic cytoplasm were patchily spread. These cells are known as ganglion-like elements and bear a singular nucleus or multiple large nuclei with only one prominent nucleolus . MitoticProliferative fasciitis is a benign pseudosarcomatous lesion that was shown to occasionally mimic soft tissue sarcoma . DiagnosIn our unusual case, a 70-year-old male patient with very atypical localization of proliferative fasciitis was presented. The palpable tumor mass appeared unusual throughout the clinical examination and the imaging techniques revealed a lobular tumorous expansion in the groin, resulting in an extensive debate on a possible malignant origin of such finding. The CNB was performed to avoid misinterpretation of the groin-localized process. The CNB, as well as incisional biopsy, is characterized by a high diagnostic accuracy and even though, discussions about the ideal biopsy techniques are still ongoing, CNB was shown to have lower risks of potential complications ,12. TherThe diagnosis of pseudosarcomatous lesions can be challenging. Even though these lesions were previously demonstrated to hold characteristic microscopic features, and were associated with previous physical trauma, the rapid growth of pseudosarcomatous lesions may cause diagnostic confusion and subsequent treatment errors. It is of major importance to discriminate rare lesions, such as proliferative fasciitis, from soft tissue sarcomas to prevent unnecessary excessive treatment. Due to the histological overlap with other disease entities, proliferative fasciitis may be often regarded as malignant and accordingly overtreated. The knowledge of the specific pathologic features described is the best prevention of misdiagnoses. The estimation of the final diagnosis should, thus, depend on the evaluation of cytomorphological features and proper histopathological examination.


Synovial sarcoma is a soft tissue sarcoma of temporarily unknown histologic origin with the ability for biphasic differentiation, occurring mostly in the vicinity of large joints of the extremities. Synovial sarcoma that originates in the liver is extremely rare. Only 7 cases have been reported in the domestic and international literature.We report an 11-year-old female patient who underwent partial hepatectomy for a liver mass. Microscopically, she was diagnosed with hepatic biphasic synovial sarcoma. Cytogenetic examination revealed the fusion gene SS18-SSX1 (+), which confirmed the diagnosis.Synovial sarcoma of the liver is a rare malignancy that is difficult to diagnose. Confirmation of diagnosis is based on histopathological assessment combined with immunohistochemical staining and, if necessary, cytogenetic aids.The online version contains supplementary material available at 10.1186/s13000-022-01233-4. Synovial sarcoma (SS) is a rare but highly malignant soft tissue sarcoma, accounting for approximately 5–10% of soft tissue sarcomas . The mos2, the total volume of the patient’s liver was approximately 2063 cm2, and the volume of the left liver was approximately 415 cm2. If the right half of the liver was removed, the functional liver volume removed was approximately 37%. In addition, the liver function evaluation results showed that the retention rate of indocyanine green was 2.5% in 15 minutes. These results indicated that the patient had good liver function and was expected to tolerate the surgery. Therefore we operated on 11 August 2021. During the surgery, we observed a large mass in the right lobe of the liver that was confined to the liver and did not break through the peritoneum. Exploration of the other abdominal organs showed no abnormalities, so we removed the intact mass together with the right half of the liver. The postoperative pathology specimen (Fig. An 11-year-old female patient was admitted to the hospital on July 29, 2021 with the chief complaint of “intermittent nausea, vomiting and abdominal pain for 6 years, aggravated for 2 months”. The patient was born at full-term, had an uneventful delivery and was previously healthy. Physical examination indicated that the liver area was slightly inflated, and a liver mass could be palpated 3 cm below the rib cage, with a hard texture and positive tenderness to touch. Laboratory tests indicated that the white blood cell count was 12.92 × 10^9/L, ESR was 44 mm/1H, and tumor markers and the stool routine examination were normal. An abdominal angio CT suggested a giant low-density mass in the right lobe of the liver of approximately 13.6 × 11.2 cm in size (Fig. Synovial sarcomas originating in the liver are extremely rare, with only 7 cases –11 reporSynovial sarcoma of the liver has an insidious onset, lacks specific clinical symptoms, and has no reported risk factors associated with its development. Patients generally present with abdominal pain with no obvious cause, and most of the lesions have reached a large size at the time of presentation –11. In tSynovial sarcomas of the liver tend to appear as large aggressive heterogeneous masses on CT. These masses may have hemorrhage and cystic changes, often with calcification, and tend to show uneven moderate or significant enhancement on enhancement CT . MRI is Thus, pathology is the only criterion for the diagnosis of hepatic synovial sarcoma. Microscopically, biphasic hepatic synovial sarcoma is composed of different proportions of both spindle cells and epithelioid cells, of which the spindle cells are often uniform in size and tightly arranged in a striated, fasciculated or herringbone shape. Epithelioid cells are often cubic or tall columnar in shape and may be arranged in a glandular, nest-like or papillary structure , 10. ImmAt present, the main treatment for synovial sarcoma of the liver is a combination of early surgery combined with postoperative chemotherapy, radiotherapy or biological therapy. Because synovial sarcoma of primary origin in the liver is relatively rare, the prognosis for this disease is not yet clear. However, it is promising that as research into the molecular mechanisms of synovial sarcoma continues, biologic therapy may open up new avenues of treatment for this disease.Additional file 1.


Primary seminal vesicle tumors are extremely rare. Several rare pathological types of primary seminal vesicle tumors have been reported, such as adenocarcinoma, but there is no report on adenofibroma. We report the first case of adenofibroma arising from the seminal vesicle. A 50-year-old man, with no history or clinical evidence of any other tumors, accidentally found a pelvic mass during an ultrasound examination. As the mass grew, the patient developed mild constipation, without genitourinary or other symptoms. All laboratory examinations were normal. MRI of the pelvis revealed a mixed density, measuring 11.7×9.9×8.2cm, well circumscribed mass. The rectum, bladder, prostate and lymph nodes were normal. We successfully performed the open surgery and removed the mass. Histopathological results confirmed that the mass was a primary seminal vesicle adenofibroma. In literature, we found that for biphasic differentiated tumors, it is easy to reduce the accuracy of pathological diagnosis because of insufficient puncture. Therefore, preoperative puncture biopsy for seminal vesicle tumors should be investigated further. Male pelvic tumors include bladder tumors, prostate tumors, intestinal tumors, and primary malignant lymphoma among others . SeminalAdenofibroma is common in mammary glands and genital system  of women, and occasionally in other sites such as the lungs , 3 and bvia abdominal ultrasound. Eight months ago, the patient was admitted to our hospital because of progressive growth of the pelvic mass. As the mass grew, the patient developed mild constipation, but no genitourinary or other symptoms. Laboratory examinations, including serum tumor markers and free/total prostate specific antigen (PSA) levels were normal. Magnetic resonance imaging (MRI) of the pelvis revealed a mixed density, measuring 11.7 ×9.9×8.2cm, well circumscribed mass  during an ultrasound examination five years ago. The patient had no clinical symptoms or signs. He chose conservative treatment and regular evaluation of the mass The patient was subjected to puncture biopsy of the pelvic mass, and the lesion turned out to be mesenchymal tumor arising from the seminal vesicle. Immunohistochemistry showed:CK (glandular epithelium+), CR(-), β-catenin(-), SMA, CD 34(+), CD117(-), S100(-), Ki67(about 1%+), which did not support gastrointestinal stromal tumor. A definite diagnosis is recommended after complete resection of the tumor. Given the possibility of malignant lesions and other compression symptoms caused by tumor growth, the patient decided to accept complete tumor resection. Laparoscopic exploration revealed a clear boundary between the tumor and surrounding tissues. However, the root of the mass could not be exposed because the mass was so large and took up the whole pelvic cavity. Therefore, we performed open surgery on the patient and successfully removed the mass. The gross specimen exhibited a white-gray, cauliflower-like appearance, uneven with calcification, well encapsulated, measuring 12.5×9.5×7.0cm Figure 2Primary seminal vesicle tumors are classified as epithelial , mesenchymal , mixed epithelial sromal (cystadenoma) and other rare lesions  . DiffereAdenofibroma is a biphasic differentiated tumor. Microscopically, the tumor is a mixed epithelial stromal tumor, mainly composed of benign fibrous components. Although most adenofibromas are benign, there are a few cases of malignant transformation . Thus, iDespite its important clinical value, puncture biopsy could not identify the true pathological characteristic features of the tumor in our case. Song et al  and BabaThe raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.YL and JW were involved in the surgery. Pathological images are provided by DS. YL and DS did literature search. YL drafted the paper. JW revised the paper. All authors have read and approved the final manuscript.This study was funded by Yantai Science and Technology Bureau .The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Squamous papillomas are benign lesions that are associated with human papillomavirus infection. Oral squamous papilloma of the uvula is uncommon and rarely causes symptoms. In this case report, we present a rare case of symptomatic squamous papilloma of the uvula where the patient complained of mass-related symptoms such as dysphagia and choking sensation. The lesion was surgically excised using electrocautery with excellent outcomes. In addition, we reviewed literature related to the lesion. Oral squamous papillomas (OSPs) are asymptomatic neoplasms of the oral cavity, growing mostly in the soft palate . AlthougA 24-year-old female presented with a complaint of dysphagia to solids and odynophagia for 2 months. These symptoms started progressively and were associated with continuous throat clearing, globus sensation, and on-and-off choking sensation, especially during sleep. Upon examination, a long pedicled lesion was noticed extending from the uvula. Flexible nasopharyngoscopy revealed an elongated uvula touching the posterior part of the tongue. Post-nasal drip and acid reflux were noticed in the pharyngeal cavity. No ulceration or discoloration was seen on the lesion.The patient underwent surgery under general anesthesia, in which the lesion was excised using electrocautery. A macroscopic histopathology examination revealed a soft tissue measuring 2 cm x 0.5 cm x 0.3 cm. Microscopic findings showed a polyp lined by benign stratified squamous epithelium Figure . The strOSPs are benign, asymptomatic soft tissue neoplasms of the oral cavity. Their macroscopic appearance is characterized by pedunculated, usually, solitary exophytic extensions with a cauliflower-like surface . It is rSymptomatic squamous papillomas are rare. The location and size of the lesion play a role in the emergence of symptoms ,4. Five In a case series involving 464 cases of oral papillomas, 34% of the cases were in the palatal complex . Only 4% of the cases were located in the uvula .Another retrospective study was conducted to analyze 207 biopsies of oral papillomas performed from 1996 to 2012. Most of the lesions occurred in the soft palate (23%). The second most common site was the posterior border of the tongue (20.9%). The lesions were more predominant in males than females (ratio = 1.26:1) [Along with the clinical evaluation, the definitive diagnosis of squamous papillomas depends on biopsy and histopathological examination . FeatureSymptomatic squamous papillomas are influenced by the location. For instance, uvular papilloma can be associated with symptoms due to its distinct site of occurrence. Although it is rare, uvular squamous papilloma is reported to be associated with mass-related symptoms such as dysphagia and choking sensation. Although lesions greater than 1 cm are few, even small-sized uvular papillomas appear to cause symptoms. The treatment of choice predominantly relies on electrocautery.


Ulerythema ophryogenes (UO) or keratosis pilaris rubra atrophicans faciei is a disorder of keratinization that primarily affects the face. The inflammatory process in UO may eventually result in alopecia. The incidence of this disorder is still unknown. We present a case of UO in a 28-year-old male, the first of its kind in Saudi Arabia. Ulerythema ophryogenes (UO) is a rare dermatological disorder of keratinization primarily affecting the face. It manifests as erythema and multiple facial papules that result in atrophy, scarring, and alopecia . A clearWe report a case of a 28-year-old Saudi single male who presented to our dermatology clinic for progressive asymptomatic facial erythema and gradual diffuse thinning of eyebrow hair that has been evident since childhood. He was seen by multiple dermatologists and was misdiagnosed on multiple occasions with alopecia areata (AA) but has never received any form of treatment. The patient's past medical history is insignificant, and his surgical history is only positive for cosmetic breast reduction surgery for gynecomastia. He had not been on any medications. His family history is negative for similar conditions including rosacea and AA. Cutaneous examination of the face revealed symmetrical well defined macular erythema with prominent telangiectasia over the cheeks sparing the nasolabial folds Figures , 2, alon UO, also known as Keratosis pilaris rubra atrophicans faciei, is a rare and underreported variant of keratosis pilaris. It is characterized by perifollicular erythema and horny red papules particularly involving cheeks, eyebrows, and the nasal bridge  . It may In conclusion, the incidence and prevalence of UO are still unavailable, and no ethnic, racial, or gender associations have been established. We report a case of UO in a 28-year-old male Saudi, the first of its kind in our country, Saudi Arabia, and even in the middle east region.


Sclerosing Polycystic Adenosis (SPA) is a rare disease of the salivary glands, first described by Smith et al.SPA affects mainly the parotid and submandibular glands, being much less frequent on the minor salivary glandsFemale, African-Brazilian, 82 years old, came to us complaining of pain in the mouth floor when using her full mandible denture. During her intraoral exam, we noticed a misfitting of the upper and lower dentures and an ulcerated, reddish and firm nodule, measuring 2cm in its longest axis. The patient was instructed to refrain from using her dentures and after 15 days she returned for nodule reassessment - which remained unaltered. With clinical suspicion of an erythroplastic lesion, we carried out an incisional biopsy and the material was then referred to the Laboratory of Surgical Pathology. The lesion was characterized by fibrous nodules permeated by interconnecting tubules, either ectasic or with apocrine metaplastia . Some duThe literature has approximately 35 cases of SPA reported so far, with only four of those involving minor salivary glands. Therefore, this is the fifth case of minor salivary gland SPA.Since this is a rarely described lesion, there is no characteristic clinical data for it. Smith et al.,,,,The histomorphological aspects hereby described fit those established for SPA. The lesion was made up of fibrous nodules, permeated by interconnecting tubules, either ectasic or with apocrine metaplasia, also showing cell vacuolization and atypia3,,,Immunohistochemistry for proteins S-100 and smooth muscle actin, from the case presented, showed the presence of myoepithelial cells in the tubule-acinar elements of the lesion,SPA's treatment is based on lesion excision - with safe margins. Follow up must be careful because recurrences are commonSPA's diagnosis must be carefully considered, especially because of its histomorphological similarity with different malignant neoplasias of the salivary gland.


Endometriosis is characterized by the presence of an ectopic endometrial gland. Intestinal endometriosis with small bowel obstruction is uncommon. In this case, a 37-year-old woman with no history of endometriosis presented with a chief complaint of abdominal pain. Computed tomography revealed a distal small bowel obstruction. Surgical intervention was performed because of the emergent abdominal condition and the potential resistance to conservative management. Histopathological examination revealed appendiceal and ileal endometriotic lesions. Preoperative diagnosis was difficult because there were no specific clinical features. Intraoperatively, it is difficult to distinguish intestinal endometriosis and bowel malignancy; thus, oncological resection should be performed. Endometriosis is defined as the presence of ectopic endometrial tissue outside the uterine corpus and is estimated to affect 4–17% of menstruating women , 3. ElecA 37-year-old woman presented with an acute onset of diffuse abdominal pain and vomiting unrelated to menstruation. The patient had no history of abdominal surgery, including a caesarean section. Her faecal occult blood test results were positive for the last 3 years, and the annual colonoscopy did not reveal any abnormalities. She had ileus of unknown cause and had undergone conservative medical management at another hospital 1 year prior. Physical examination revealed abdominal distention and peritoneal irritation. Blood examination revealed a slightly elevated circulating white blood cell count of 10 020/μl, but other laboratory data were within the normal range. Computed tomography (CT) revealed ascites and a small bowel obstruction at the level of the distal ileum . Her abdHerein, we report a rare case of small bowel obstruction with appendiceal and ileal endometriosis. Clinically, the symptoms of intestinal endometriosis are numerous and non-specific , 2. AlthIntestinal endometriosis is mainly treated surgically because of the difficulty of preoperative diagnosis. Patients with intestinal endometriosis may present with acute abdominal pain and require urgent medical attention , 2, 4–8.Endometriosis sometimes also involves the lymph nodes; however, according to some reports, the positive lymph nodes that are left untreated might have no clinical influence , 13. TheOne of the characteristic findings in our case was that endometrial glands were found in the appendix and ileum of both organs. To the best of our knowledge, only three similar cases have been reported , 13, 14.Appendiceal and ileal endometriosis is rare cause of small bowel obstruction. Preoperative diagnosis is difficult because of the non-specific symptoms and CT findings. Exclusion of bowel malignancy is essential; if doubtful, oncological resection should be performed.M.Y. and M.M. performed surgeries. K.K. performed post-operative management. K.K., M.Y. and M.M. were involved in drafting the manuscript, revising it critically for important intellectual content and making substantial contributions to clinical data acquisition. H.T. made substantial contributions to the pathological data acquisition and helped draft the manuscript. All authors have read and approved the final manuscript.


Ewing's sarcoma was identified by James Ewing in 1921 as a perivascular endothelial tumor.Extraskeletal ES rarely occurs in the head and neck localization, account for only 1–4% of all ES, and usually prefers mandibula.An 11 year-old female patient presented to our clinic with the complaint of swelling on her right cheek. Her history revealed that the swelling had emerged 2 weeks prior and had been growing increasingly since then. In the physical examination, there were no other significant findings except an immobile, hard on palpation 2 × 2 cm mass lesion on the right cheek. Laboratory tests showed no abnormalities. Magnetic Resonance Imaging (MRI) revealed a mass lesion centered in the right maxillary sinus extending to the anterior wall and soft tissue . No metaOn suspicion of malignancy, the patient underwent open surgery by approaching through sublabial incision and subsequently biopsy specimen was submitted to pathology.In the histopathological examination, the tumor showed closely packed uniform small round cells with high nuclear/cytoplasmic ratios and finely dispersed chromatin, without prominent nucleoli. Mitotic figures were numerous. Areas of necrosis were present. Immunohistochemically, the tumor cells were strongly positive with CD99 and vimentin as well. Occasional clusters had some cells positive for synaptophysin and CD56. The tumor cells were negative with CD3, CD20, LCA, PanCK, myogenin, myoD1 and desmin. Ki-67 proliferative index was found between 30% and 50% in some areas. Subsequently, ES appeared as the most probable diagnosis. Therefore the patient material submitted to the molecular pathology for the evaluation of classical ESWR1-FLI1 rearrangement. The results of Fluorescence In Situ Hybridization (FISH) for ESWR1-FLI1 rearrangement confirmed the diagnosis ES/PNET .Figure 32; D, doxorobucin 75 mg/m2; C, cyclophosphamide 1.2 g/m2; I, Ifosphamide 1.8 g/m2; E, etoposide 100 mg/m2) Pediatric Oncology Group protocol which were administered 17 cycles over 48 weeks. During the follow-up, after 4 months, local recurrence was detected. The patient underwent open surgery with the same procedure approaching through sublabial incision. The resection was extended to reach safely negative tumor margins. Radiotherapy was added to treatment procedure following the surgery. The metastasis investigation was carried out by Positron Emission Tomography (PET) scan and MRI every 6 months. Also the patient was followed by routine clinical examination every 3 months. There was no finding of recurrence or metastasis at the 13th month of follow-up  which corresponds to a fusion between the EWS gene (22q12) and the FLI1 gene (11q24) which is a transcription factor.Moreover, four different translocations are observed. Those are EWS-ERG t, EWS-ETV1 t, EWS-E1AF t, and EWSFEV t according to their frequency, respectively.The aim of ES treatment is to maintain all normal functions and to prevent recurrences and sequellae in the long term while eliminating the disease. The recommended treatment is a combination of surgery with chemotherapy and radiotherapy. Early diagnosis followed by wide resection, chemotherapy, and radiotherapy can leave the patient disease-free for a long time. It has been reported that the recent use of proton beam therapy provides local control of the disease and prevents possible complications of radiotherapy on surrounding tissues .Ewing's sarcoma is rare in the maxillary sinus. Surgery, chemotherapy, and radiotherapy should be used in combination to treat this lesion, which stems from high-grade tumors. Patients should be closely followed-up with due to the possibilities of local recurrence and distant metastasis.The authors declare no conflicts of interest.


The diagnosis is often difficult and delayed, even after comprehensive work-up. To our knowledge, we report for the first time an ectopic corticotroph tumor of the posterior wall of the sphenoid sinus, leading to false positive results of bilateral inferior petrosal sinus sampling (BIPPS) and which was finally localized by a co-registered11 C Methionine PET/MRI was performed and revealed a hypermetabolic lesion adjacent to the posterior wall of the sphenoidal sinus. After surgical resection, this polypoid mass was identified as an ectopic ATCH-secreting pituitary adenoma expressing ACTH and T-Pit and complete remission of hypercortisolism was observed.A 48-year-old woman was referred for a high clinical suspicion of ACTH-dependent CS. Biological testing comprising low dose dexamethasone suppression and CRH stimulation tests were indicative of pituitary Cushing’s disease, but comprehensive pituitary MRI did not reveal any pituitary adenoma. BIPSS confirmed however a central origin of ACTH secretion  and revealed a significant right-to-left gradient (6.2), leading to a first right-sided exploratory hypophysectomy, that did not cure the patient. BIPSS images were reviewed and revealed preferential drainage of the left pituitary to the right petrosal sinus, leading us to a left sided exploratory hypophysectomy, which was again unsuccessful. A11 C Methionine PET co-registered to high resolution MRI for localizing ectopic adenomas, efficiently guiding surgical removal and leading to complete remission of hypercortisolism.In conclusion, we report a case of ACTH-dependent Cushing’s syndrome, caused by an ectopic corticotroph adenoma located in the sphenoidal sinus, which perfectly mimicked the biological features of a classical pituitary ACTH adenoma on a comprehensive hormonal evaluation including BIPPS, and the features of a benign naso-sinusal polyp at MRI. We report for the first time a key role of The dia2) with buffalo neck, typical skin frailty, peripheral amyotrophy and hypertension (150/90 mmHg).A 48-year-old woman was referred to our Endocrinology Department for suspicion of Cushing’s syndrome (CS). She complained of weight gain (7 kg in 1 year), face swelling and frequent bruising. Menstruations had been irregular for the last year. She also had recently suffered from a spontaneous rib fracture and aseptic osteonecrosis of the shoulder. Clinical examination revealed truncal adiposity  and neutrophilia at 11,390/μl . Hba1c was normal at 5.6%. A recent bone density scan had revealed osteoporosis at the femoral site (T-score − 3.1) and osteopenia in the lumbar spine (T-score − 1.9).Morning cortisol and ACTH concentrations were high Table , as was Pituitary magnetic resonance imaging (MRI) including high resolution T2-weighted sequences, as well as pre- and post-contrast T1-weighted sequences with rapid dynamic acquisition failed to reveal an adenoma . Pituitary function was otherwise fully preserved.Because of the lack of biological remission, a retrospective look on BIPSS images was done which highlighted a preferential cross drainage of the left pituitary towards the right petrosal sinus. The patient therefore underwent a second exploratory surgery now targeting the left side of the pituitary, with removal of a more soft part of the left pituitary. Again, post-operative cortisol level remained elevated at 543 nmol/l at day 2 and histopathological examination showed normal pituitary tissue. Importantly, pituitary function was again preserved without any hormonal deficit. A treatment with ketoconazole was initiated but rapidly discontinued because of nausea and abdominal pain. Other medical treatments such as metyrapone or osilodrostat were considered at this stage but the cost was prohibitive as they are not reimbursed by the Belgian social security.68Ga-Dotatate-positron emission tomography (PET)/CT imaging failed to reveal a potential source of ectopic ACTH or CRH secretion. A 11C Methionine PET co-registered with high resolution 3D MRI was performed next and did not demonstrate a residual pituitary adenoma. This imaging procedure showed a normal uptake in the remaining pituitary gland  located on the posterior wall of the sphenoid sinus  to remove this posterior sphenoidal mass. Serum cortisol decreased to very low levels 30 nmol/l) on post-operative day 2 and the patient was started on hydrocortisone replacement therapy. Histological examination confirmed the presence of pituitary adenomatous tissue with a Ki67 proliferation index at 1–2% and positive immunohistochemical staining for both ACTH and T-Pit  were highly consistent with a pituitary origin of ACTH secretion. We highlight the essential role played by a co-registered A recent review highlighted that ACTH-secreting adenomas are the most frequent ectopic pituitary adenomas . They caBilateral inferior petrosal sinus sampling (BIPSS) is indicated to distinguish ectopic ACTH secretion from Cushing’s disease, particularly in cases of negative MR imaging in patients with biological evidence of ACTH-dependent cortisol excess . False nIn our patient, in spite of negative pituitary MR examination, the very high central/peripheral ACTH gradient at BIPSS (above 100) convinced us to perform exploratory pituitary surgery, initially targeted to the right side and thereafter to the left side, as advocated by recent surgical guidelines . We now MRI has a stronger positive predictive value than BIPSS to determine pituitary tumor lateralization and current guidelines therefore recommend to perform BIPSS only in case of negative MRI to lateralize the lesion . However11C Methionine PET/MRI. As methionine is an amino-acid used for protein synthesis, 11C Methionine is preferentially taken up by protein secreting adenomas and has shown a sensitivity of 70–100% in recent studies involving Cushing’s disease patients [11C Methionine PET/MRI is considered superior to 18FDG-PET/MRI or CT for this purpose [11C Methionine PET/MRI has demonstrated encouraging results in this indication [To our knowledge, we report for the first time localization of such an ectopic corticotroph tumor by co-registered patients . Recent patients , and 11C purpose . Indeed,Ectopic pituitary adenomas originate from cellular remnants of the migration of Rathke’s pouch cells along the pharyngeal tract and have been shown to remain under the regulatory control of the hypothalamus , 12. In 11C Methionine PET co-registered to high resolution MRI for localizing ectopic adenomas, efficiently guiding surgical removal and leading to complete remission of hypercortisolism.In conclusion, we report a case of ACTH-dependent Cushing’s syndrome, caused by an ectopic corticotroph adenoma located in the sphenoid sinus, which perfectly mimicked the features of a classical pituitary ACTH adenoma on a comprehensive hormonal evaluation including BIPPS, and the features of a benign naso-sinusal polyp at MRI. We report for the first time a key role of


A 56-year-old female patient reported the appearance of a nodule on the posterior region of the right arm six months before. During this period, she reported that the lesion showed progressive growth, pain, and friability, with frequent local bleeding. This patient had been submitted to a radical mastectomy and right axillary dissection, with adjuvant chemotherapy and radiotherapy 11 years before, and since then, she had chronic lymphedema in the right upper limb.Dermatological examination revealed a hyperchromic tumor, measuring approximately 5.0 × 4.0 cm, with central areas of ulceration and slight local bleeding on the posterior region of the right arm. Lymphedema was also observed in the right upper limb and violaceous-brown satellite nodules, measuring up to 0.5 cm .Figure 1An incisional biopsy of the tumor and satellite nodules was performed. The patient returned for consultation after one month with a histopathological result that showed a neoplasm characterized by vascular formations permeated by atypical epithelioid cells infiltrating the superficial and deep dermis, with perineural permeation .Figure 2At this consultation the tumor was slightly larger and there was an increase in the number of satellite lesions .Figure 3a)Stewart-Treves syndromeb)Squamous cell carcinomac)Melanomad)Merkel carcinomaImmunohistochemistry examination was requested, which showed CD31 positivity, confirming the hypothesis of Stewart-Treves syndrome .Figure 4Stewart-Treves syndrome (STS) is a rare entity, with a poor prognosis, consisting of the appearance of cutaneous angiosarcoma in areas of chronic lymphedema and accounts for approximately 5% of angiosarcomas.5The etiology and pathophysiology of the syndrome remain unknown. It is debated whether the lymphedema would favor oncogenesis due to the lymphatic drainage failure and interstitial fluid accumulation and stasis, in addition to the possible occurrence of neoplastic transformation during angiogenesis.The diagnosis is based on the clinical picture, along with the biopsy. Histopathology may be suggestive, with irregular vascular spaces lined by mitotic hyperchromatic pleomorphic tumor endothelial cells.The treatment is based on surgical resection with wide margins, and chemotherapy and radiotherapy can also be used.Although it is rare, knowledge of STS is necessary because, due to its aggressiveness, only its early diagnosis can help to increase patient survival.None declared.Roberta Akeme de Oliveira Sato: Design and planning of the study; drafting and editing of the manuscript; critical review of the literature.Clovis Antônio Lopes Pinto: Intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; effective participation in research orientation.Celia Antonia Xavier de Moraes Alves: Effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases.Juliana Arêas de Souza Lima Beltrame Ferreira: Effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the manuscript; approval of the final version of the manuscript.None declared.


Intraosseous lipomas are benign lesions of the bone. In the jaws, they are very rare and in most cases incidentally discovered on panoramic radiographs taken in dental practice. They are usually asymptomatic and appear radiologically as a radiolucent image sometimes including some radio-opacities. Histologically, they consist of mature adipose tissue associated with variable degrees of necrotic fat and calcification. In this report, we describe a case of intraosseous lipoma in the mandibular symphysis region of a 37-year-old female as well as the treatment adopted. Lipomas are common, benign tumors of adipose tissue that may involve any part of the body, especially the trunk and proximal extremities. They generally present as slow-growing soft masses. Usually, lipomas are asymptomatic when they are subcutaneous, and may cause pressure-related symptoms when they are deep . AlthougMilgram has proposed a histological classification of IOLs depending on the degree of the lesion evolution: Stage 1, a tumor with viable fat cells; stage 2, a tumor composed of both viable fat cells and fat necrosis, and calcification; stage 3, a tumor with necrotic fat, calcification of necrotic fat, variable degrees of cyst development, and reactive new bone formation .Radiologically, their appearance can vary according to the stage of the lesion ,6. TypicA 37-year-old female was referred by her dentist to our oral and maxillofacial surgery center for evaluation of a radiolucency incidentally detected on a panoramic radiograph. The lesion was located in the anterior region of the mandible below the teeth apices Figure .Medical and physical examinations revealed a healthy woman with no extra-oral abnormal findings. Intra-orally, no signs of inflammation were found and the mandibular anterior teeth were positive to vital tests.A CT scan was requested for better assessment of the lesion. Panoramic reconstruction, cross-sectional and axial views revealed a well-defined unilocular hypodense image centered by small round radiopacities located in the mandibular symphysis region. No buccal and lingual plates’ expansion or root resorptions or displacement were noticed Figures -2c.Under loco-regional anesthesia, a vestibular mucoperiosteal flap was detached from the underlying bone for better visibility and the cortical plate was drilled for access window through which a thorough curettage of the lesion was made.Macroscopically, yellow fat lobules were observed, separated by bony partitions and vascular septa; the dense structures visible on radiographic examination appeared as intralesional bone calcifications.Histopathological results of the specimen showed a mature adipose tissue, without cellular atypia, associated with variable degrees of necrotic fat and calcification Figure .The diagnosis was in favor of an IOL. Annual follow-up is suggested to prevent any recurrence of the lesion.IOLs are very uncommon benign tumors of the bone that account for about 0.1% of all bone lesions. They are mostly detected in the fourth and fifth decades of life with no gender predilection ,10. IOLsFor many researchers, the etiology of IOLs remains unknown. For others, trauma and nutritional problems may be among the etiological factors. In the mandible, which is supplied principally by the inferior alveolar artery, obliteration of the nutrient arteries may lead to infractions in some areas where fatty cells of bone marrow may collect to form a “lipomatous mass” . For PolMandibular IOLs, generally occur in the posterior segment and very rarely in the symphysis region ,10. AccoThe symptoms of IOLs depend on the location and size of the lesion . If sympThe treatment of choice for IOLs is surgical enucleation. However, extensive lesions may require bone resection followed by grafting ,13,15. RIn our report, we have presented a case of a stage 2 mandibular IOL. A complete surgical enucleation was performed with no recurrence till date.Mandibular IOLs are rare bone tumors. There are few reported cases in the literature located in the anterior section of the mandible. Many lesions may constitute a differential diagnosis challenge for IOLs. For that, a thorough assessment of the clinical, radiological, and histological components of these lesions is mandatory to surmount the diagnostic. Furthermore, their early diagnosis and accurate treatment consisting in a complete surgical enucleation/resection followed by long-term follow-up of the patient are essential to prevent any probable recurrence.


We report a case of localized sarcomesothelioma detected during screening via a low-dose CT (LDCT) scan. The patient is a 71-year-old female, a current 56-pack-year cigarette smoker with a past medical history of myocardial infarction and stroke with a Zubrod score of zero. A screening LDCT revealed a 1.9 cm × 1.8 cm × 1.4 cm right lower lobe lesion with smooth margins and close association with the hemidiaphragm. A wedge resection with biopsy showed high-grade sarcomatoid mesothelioma with extensive desmoplastic morphology and negative margins. The patient opted for imaging surveillance, and at 12 months has shown no evidence of tumor recurrence on positron emission tomography (PET)/CT. The case shows that LDCT screening discovers cancers and saves lives. It also presented a dilemma for the patient and her oncologist because common guidelines do not define a recommended treatment. Malignant pleural mesothelioma (MPM) is an aggressive tumor originating from mesothelial cells lining the lung pleura, peritoneum, tunica vaginalis, and pericardium, listed in descending order of occurrence . The incThe patient is a 71-year-old female, a current 56-pack-year cigarette smoker with a past medical history of myocardial infarction and stroke, with a Zubrod score of zero. She denied fevers, chills, night sweats, hemoptysis, or unexpected weight loss and did not have occupational exposure to asbestos. A screening LDCT revealed a 1.9 cm × 1.8 cm × 1.4 cm right lower lobe lesion with smooth margins and close association with the hemidiaphragm Figures . A wholeGiven the overall clinical history and imaging, it was decided that the patient required additional biopsy via either a repeat CT-guided needle or surgical excision. Because of the proximity to the diaphragm, a repeat needle biopsy was considered too difficult, and she was referred to a cardiothoracic surgeon.Right, video-assisted thoracoscopic surgical (VATS) wedge resection of the mass, incorporating small but adequate margins of lung and diaphragmatic muscle, was performed Figures -5. BecauFinal pathologic analysis revealed a 2 cm high-grade sarcomatoid mesothelioma with extensive desmoplastic morphology and negative margins. The immunostains were positive for calretinin, MCK, GATA3, patchy positivity for CK7 and pan keratin, and rare cells positive for WT-1.The patient was referred to a medical oncologist; the patient opted for imaging surveillance, and at 12 months has shown no evidence of tumor recurrence on PET/CT.The above case is notable because it shows that LDCT scans discover cancers and save lives. This is also demonstrated by National Lung Screening Trial data showing a 20% relative reduction in mortality from non-small cell lung cancer with LDCT compared to chest X-rays . Second,This case highlights the importance of LDCT scanning for early diagnosis of life-threatening illnesses, based on USPSTF guidelines. Also emphasized is the clinical decision-making that prompted the patient’s caretakers to persist in securing a diagnosis based on her risk factors. Given the rare nature of sarcomatoid mesothelioma, we hope the information provided can add to the few other case reports for a better understanding of this cancer.


Astrocytic hamartoma is a benign glial tumor. It may be associated with tuberous sclerosis and can also be found incidentally on retinal examination as an isolated presentation. Here, we describe multimodal imaging characteristics of astrocytic hamartoma in a patient with retinitis pigmentosa. Spectral domain optical coherence tomography of both eyes showed moth-eaten optically empty spaces and hyperreflective dots along with foveal thinning. Multicolor image highlighted mulberry appearance of the lesion with green shift signifying elevated lesion. In infrared reflectance, lesion was hyporeflective with its margins well delineated. Green reflectance and blue reflectance highlighted calcification as multiple hyperreflective dots. Autofluorescence showed typical hyperautofluorescence. Astrocytic hamartoma is a benign and stable tumor that originates from glial astrocytes of the retina or optic nerve. It may be associated with phakomatosis like tuberous sclerosis and neurofibromatosis or found incidentally on retinal examination as an isolated presentation . A rare A 15-year-old male came to our hospital with gradual diminution of vision and night blindness in both eyes since 5 years. There was no family history of phakomatosis. Comprehensive examination did not reveal clinical features of phakomatosis. Best-corrected visual acuity was 20/1200 and 20/600 in right and left eye, respectively. Fundus examination of both eyes showed creamy white, semitranslucent, elevated lesion surrounding the optic disc along with attenuation of blood vessels and bony spicules in the mid-periphery (Figures 1a/b Astrocytic hamartoma should be monitored closely to look for any progression. It should be differentiated from other optic nerve head lesions like optic nerve head meningioma, hemangioma, papilledema, combined hamartoma of the retina and retinal pigment epithelium, and most commonly optic nerve head drusen . MulticoThis report highlights characteristics of multicolor imaging in astrocytic hamartoma and its association with RP.The authors declare that they have no competing interests.


Pneumatosis Cystoides Intestinalis (PCI) is a fairly rare condition that is, however, becoming of increasing incidence. It is difficult to ascertain as many patients are asymptomatic, but, in adults, if it does present it usually presents in the fifty-to-eighty years age groups.It may be primary (15% - idiopathic and benign condition) or secondary  but the aetiology is unclear, with some theories that have been put forward. Many consider its pathogenesis to be due to increased intra-luminal intestinal pressure and, therefore, build-up of gas from commensal gut bacteria. Imaging and histology are important in diagnosis.This case presentation explores the complicated presentation of a patient with PCI and his management, thereof, in order to review the appropriate investigations and the subsequent options of management.Because it is a poorly understood condition with varying presentations, it is often misdiagnosed. The diagnosis is critical to allow the correct and most appropriate management to be carried out: non-surgical supportive care versus surgical intervention with resection and stoma or anastomosis. ●Pneumatosis Cystoides Intestinalis (PCI) is a fairly rare condition and is difficult to diagnose as many patients are asymptomatic.●Imaging and histology are key the in diagnosis and subsequent non-surgical versus surgical management.●This case presentation explores the complicated presentation of a patient with PCI and his management, along with some associated concurrent diagnoses.Pneumatosis cystoides intestinalis: presentation and management of a complicated case. It is characterized by the presence of multiple gaseous cysts in the intestinal wall that contain a mixture of carbon dioxide, nitrogen, and hydrogen. Commonly, the cysts are located beneath the serosa and mucosa of the intestinal wall with majority being in the ileum, followed by the colon, and at times (but rarely) in both small bowel and colon . The peaThe aetiology of PCI is not known, it may be primary or secondary, but has been associated with gastrointestinal surgeries, Chronic Obstructive Pulmonary Disease (COPD), laxative use, connective tissue disorders, malnutrition, alpha-glucosidase inhibitor, and colonoscopies . PrimaryClinical examination may include symptoms of abdominal pain, nausea and vomiting, diarrhoea, haematochezia, or may present complicated with pneumoperitoneum, volvulus, intestinal obstruction, and ischaemia . Non-surIn this case presentation, the complex findings of multiple visceral cystic lesions, a gastric volvulus and gastric outlet obstruction present a challenge to the surgical approach and management of such a patient.22.1A 39-year-old black African male presented to a secondary facility emergency department complaining of a two-week history of progressive abdominal distension with one day of associated severe abdominal pain and post-prandial vomiting. No history of loss of weight, no past surgical history, and no medical history or chronic medication use. There was no family history of note, and the patient had no history of smoking or alcohol intake.2.2General examination: underweight patient, no palpable lymph nodes Heart Rate (HR) = 114 beats per minute; Blood Pressure (BP) = 103/59 mmHg, Temperature = 36.4° Celsius, Respiratory Rate (RR) = 27 breaths per minute.Abdominal examination: massively distended, tense, tympanic abdomen, peritonitic. Succussion splash present. Per rectum examination: soft brown stool with a normal prostate. Cardiovascular and respiratory examinations were normal.2.3Two large bore intravenous (IV) lines were inserted and 1 L crystalloid fluid bolus given with subsequent crystalloid infusion rate of 125ml/hr IVI. Urine catheter inserted drained 600ml clear urine, nasogastric tube (NGT) inserted and 3.5 L of gastric material drained with some relief of abdominal distension and pain .Table 1IChest X-ray: bilaterally elevated diaphragms with small but clear but reduced lung fields and no free air under the diaphragm, Abdominal X-Ray : large r2.41)Pneumoperitoneum noticed on entering the peritoneum2)Massively dilated pelvic stomach with Organo-axial gastric volvulus – viable.3)Narrow pylorus (<0.5cm)4)Multiple extensive cystic mesenteric-para -jejunal-ileal-colonic duplication cysts starting 110cm from Duodenal-Jejunal (DJ) Flexure to proximal ascending colon .Fig. 2An5)Massive supra-hepatic cystic mass of 15cmx12cmTheatre:2.5-Detorsion of gastric volvulus – viable distal oesophagus and stomach – gastropexy performed along the anterior fundus.-All cystic-duplication intestinal cysts left intact: true intestinal lumen viable and peristalsis present throughout.-Supra-hepatic cystic lesion left in situ with pencil drain placed inferiorly2.6IVF support. Enhanced Recovery After Surgery (ERAS) protocol was followed and feeds were progressed from clear fluids to soft diet withing 48 hours post operatively. Patient noted to have post-prandial vomiting within 1–2 hours after eating on day six post-surgery, subsequently oral feeds were stopped and patient kept nil per os on IVF support. Booked for a Gastroscopy and Barium Meal and follow-through. Gastroscopy: large amounts of gastric content – unable to view stomach. Gastric lavage was carried out and repeat Gastroscopy after 48 hours showed: Gastric Outlet Obstruction, pin-hole pylorus visualized and 6× biopsies taken. Barium meal and follow-through on day ten post-surgery demonstrated a large pelvic stomach with delayed transit of contrast into small bowel and subsequent large bowel. At this point the patient tolerating clear sips per os and supported with Total Parenteral Nutrition (TPN). Abdomen otherwise soft and not tender, minimal distention. Histology Report of Biopsies: No Helicobacter Pylori organisms noted; gastric mucosa shows widespread chronic gastritis, no active inflammation, no evidence of lymphoid follicle formation, no evidence of dysplasia or invasive malignancy. Day 19 post-surgery pyloric stenting was attempted but failed. Elective surgery planned involving a distal gastrectomy with Bill Roth 2 (Gastrojejunostomy) procedure, Jejunal and ileum cyst-lesion resection , Computed Tomography (CT) , colonoscopy (polypoid protruding lesions) and histology assist in confirming this diagnosis . The hisMajority of PCI lesions are in fact found in the colon (approximately 46%), followed by the small bowel in roughly 27%, and then stomach in less than 5%. It may occur in both large and small bowel in 7% of patients [Surgical intervention is not necessarily indicated if no complication exists on presentation, and the condition may be managed with oxygen administration, antibiotic treatment if sepsis is proven, cessation of any causative agents or medication, and a trial of nil per os with intravenous fluid support. Surgical intervention depends on localization of the cystic lesions and clinical signs of complication such as bowel obstruction, septicaemia, perforation, or peritonitic abdomen with associated hypotension and/or acute kidney injury.In the above case study, the patient presented with an acute peritonitic abdomen and imminent signs of hypotension and acute kidney injury. The gastric volvulus was managed in the initial laparotomy with detorsion and gastric pexy, however, as there was no further mechanical small bowel or large bowel obstruction, the hepatic and small bowel cystic lesions were managed conservatively. Supportive management was ensued here forth, but, the further development of gastric outlet obstruction from the thickened pylorus necessitated a relook laparotomy involving a distal gastrectomy with a gastrojejunostomy and resection of the hepatic cyst.4PCI is not a common disease but, however, its increasing in incidence makes it important to be aware of as a differential diagnosis. Imaging and histology are of utmost importance in its diagnosis. Conservative non-operative management may be carried out in stable and uncomplicated patients, which involves oxygen administration, antibiotic treatment, maintaining a patient nil-per-os, cessation of alpha-glucosidase inhibitors, and intravenous fluid support, with an optimistic prognosis and outcome in over 70% of case. In the limited cases endoscopic treatment with fine needle aspiration or resection may be done if disease is confined. In complicated cases of obstruction, septicaemia, or perforation surgery may be undertaken with resection of the affected portion of viscera with resultant anastomosis or stoma depending on the case. As demonstrated by this case, both non-operative and eventual operative managements of PCI were undertaken demonstrating the complexity of its management.University of Witwatersrand Human Research Ethics Committee (HREC) approved the case report. Ethics approval was approved by the Ethics Committee.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this Journal.The author(s) received no financial support for the research, authorship, and/or publication of this article.All Authors contributed equally to this case report.Informed Consent given.“Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request”.1. Name of the registry:National Health Research Database.2. Unique Identifying number or registration ID:GP 202 2070 40.3. Hyperlink to your specific registration (must be publicly accessible and will be checked):https://nhrd.health.gov.za.Rudo Pswarayi.Not applicable.The following information is required for submission. Please note that failure to respond to these questions/statements will mean your submission will be returned. If you have nothing to declare in any of these categories then this should be stated.The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.


Spontaneous esophageal perforation, also commonly referred to as Boerhaave's syndrome, is one of the most lethal diseases causing an acute abdomen. Though rare, emergent surgical intervention is often required and management can be various based upon the site of the perforation. This literature has been written in line with the SCARE criteria  A 76-year-old man presented with acute abdominal pain. Computed tomography (CT) revealed and an emergent esophagogastroduodenoscopy (EGD) was performed carefully, which revealed a 7 cm all-layer esophageal laceration in the left lower esophageal wall. In our case, a hiatal hernia was protruding into the mediastinum, and the perforation site was inside of it, but there was no invasion into the thoracic cavity, thus a transabdominal approach was performed without thoracotomy.This type of esophageal perforation within a hiatal hernia is quite rare and provides a unique clinical challenge. In addition, A review reported the average length of spontaneous esophageal perforation to be around 2 cm while our case had a perforation with a length of 7 cm. We chose the combination of the simple suture with omental buttress and wide drainage, but a complete fundoplication was impossible due to its large size of perforation.We chose the open abdominal approach because the case had high inflammation, a hiatal hernia and possibility of retro-gastric perforation. However, MIS should have been considered first if a situation or human resources allow it. •Esophageal perforation combined with a hiatal hernia is quite rare.•Our case had about 7cm of perforation though it is around 2cm on average.•We chose the trans abdominal approach due to the hiatal hernia.•In some conditions, thoracotomy is not required, and it would be beneficial when considering the invasiveness. Its mortality rate is estimated as high as at least 20 % 2The patient was a 76-year-old man with a chief complaint of sudden repetitive hematemesis after lunch, followed by persistent epigastric tenderness. His past medical history was significant for bladder cancer and benign prostatic hyperplasia, for which he had completed the TUR-BT and chemotherapy. He presented to his local hospital where he underwent clinical evaluation followed by a non-contrast computed tomography (CT) of the chest, abdomen, and pelvis revealing a high-density area suggesting bleeding in his stomach. Upper gastrointestinal bleeding was suspected, so an emergent esophagogastroduodenoscopy (EGD) was performed. It revealed a 7 cm esophageal laceration extending into the muscle layer -a, as weHe appeared in distress and had intense tenderness with guarding in the epigastric region when he arrived at our hospital. He was oriented for all, and vital signs are follows; Heart rate: 94 beats/min and regular, blood pressure: 164/111 mmHg, respiratory rate: 30/min, SpO2: 97 % (ambient air), and body temperature: 90.8 F. Laboratory data revealed as follows; WBC: 19,000/μL, C-reactive protein: 0.36 mg/dL, Hemoglobin: 12.8 g/dL, but otherwise were within normal limits. A new CT scan with contrast of the chest, abdomen, and pelvis was performed. This demonstrated lower esophageal dilation accompanied by fluid and free air around it. The abdominal CT scan demonstrated contrast extravasation from the stomach, further suggesting a perforation -a. The hUnfortunately, 5 days after the index operation, inflammatory markers were trending up, and a leak was identified on an upper gastrointestinal fluoroscopy study with contrast. The decision was made to take the patient back to the operating room. During the second operation, we found an approximately 1 cm perforation at the most anal side of the closure and revision was impossible based on the friability of the tissue. Sufficient irrigation was applied followed by the replacement of the drainage tubes into hernia sac. On postoperative day (POD) 38, X-ray with oral contrast was performed which revealed no leakage then drainage tubes were removed. Water intake was resumed on the day. Oral food intake was restarted on POD 44 after confirming the complete healing of the site with repeat EGD. Finally, the patient moved to the original hospital for the physiotherapy 47 days after the first operation.3Even normal esophageal perforation itself is comparatively rare, but a case accompanied with a hiatal hernia is extremely rare. We found only a few reports of similar cases while performing a literature review on Pub Med searching with the words “esophageal perforation” or “Boerhaave's syndrome”, and “hiatal hernia” within the time frame of 1952–2022 Based on the hypothesis that the hiatal hernia contributed to this perforation, it was likely due to a unique and/or unusual physiologic mechanism. Given the perforation site was located exclusively on the abdominal esophagus and not the lower thoracic esophagus in addition to the larger size of the tear, the greater intraabdominal pressure might be concentrated to the abdominal esophagus or the abdominal esophagus might be directly compressed by the fundus of the stomach within the hernia sac. To the best of our knowledge, only one similar case has been reported so far. It was also approached transabdominally and they also lacked clarity on the mechanism of perforation. While the etiology/pathophysiology in inconclusive, it is likely unique in respect to typical spontaneous esophageal perforations without hiatal hernia.Fundic patch is also well-known method to strengthen the perforated site and prevent reflux post-operatively Our patient clearly required surgical intervention, and esophagectomy might have been considered given the length of the lesion. However, it was challenging due to the location combined with the hiatal hernia, hence we chose the combination of the simple suture with omental buttress and wide drainage. In this case, leakage was observed from the repaired part of the lower esophagus after the primary surgery. One of the lessons learned from this experience is that it might be important to perform the fundic patch as much as possible even if covering the entire damaged site is challenging. Several reports mention that minimally invasive surgery (MIS) including laparoscopic, thoracoscopic or both of them could be performed safely for Boerhaave's syndrome, even if it is highly inflamed. They showed some MIS technique such as Toupet's partial fundoplication or omental buttress with flap, which reached preferrable consequences This type of perforation is challenging, but possible to treat with a trans-abdominal approach as long as there is no contamination in the thoracic cavity. An abdominal approach may have the advantage of avoiding some of the morbidity associated with thoracotomy. We believe it was the right approach in this patient and perhaps the correct approach in patients with similar presentations in the future.There are no funding sources.The ethical approval was given by our institution.Our patient and family consented.Hidenori Maki: Primary assistant of the surgery and corresponding authorSeiya Inoue: Primary surgeon of the surgery, supervisor and writerMasakazu Goto: Assistant of the surgery and writerTakeshi Nishino: Assistant of the surgery and writerTakahiro Yoshida: Assistant of the surgery and writerHiromitsu Takizawa: Assistant of the surgery, supervisor and writerNot applicable.Hidenori Maki, M.D.We don't have no conflicts of interest to declare on this literature.


Intracranial inflammatory pseudotumors (IIPT) are one of the differential diagnosis for the central nervous system (CNS) tumors. They represent a rare condition that may mimic clinically and radiologically intracranial tumors and induce their complications. Among their etiologies, neurosarcoidosis is one of the less known and less frequent. To the best of our knowledge, only two cases of posterior fossa IIPT have been reported in the literature. We present here the 3rd case related to a neurosarcoidosis.We report the case of a 55-year-old female patient who presented with an altered state of consciousness associated to severe intracranial hypertension syndrome for four months. Glasgow coma scale on admission was 14/15. Brain imaging revealed bilateral cerebellar micronodular meningeal enhancement regarding the mesencephalon and the pons, as well as a nodular lesion of the 4th ventricle causing a triventricular acute hydrocephalus. The patient had a ventriculo-peritoneal shunt with a favorable outcome. Afterwards, she underwent a salivary gland biopsy which confirmed the diagnosis of neurosarcoidosis.Posterior fossa IIPT are very rare, mainly when located in the posterior fossa, leading to confusion with other pathologies. MRI has an important role in the diagnosis of these lesions, and the determination of their etiology. It shows other than the IIPT itself, many other signs such as leptomeningeal enhancement, nodular lesions or pituitary stalk thickening. These signs can orientate towards the diagnosis. Treatment may associate to symptomatic approach, corticosteroids. Surgical resection may be proposed when the diagnosis remains doubtful. Intracranial inflammatory pseudotumors (IIPT) are one of the differential diagnosis for the central neurologic system (CNS) tumors. They represent a rare condition that can mimic clinically and radiologically intracranial tumors and induce their complications ,2. Most A 55-year-old female patient with a history of hypertension and type 2 diabetes mellitus presented at our hospital emergency with an altered state of consciousness associated to severe intracranial hypertension syndrome made of headaches and vomiting. Occipital headaches, vomiting, visual blur and vertigo were noticed in the past four months without any history of head trauma or fever.On admission, her Glasgow coma scale was 14/15. She had no motor weakness. Brain magnetic resonance imaging (MRI) revealed bilateral cerebellar micronodular meningeal enhancement  and 2. TPreoperative assets were in favor of a posterior fossa granulomatosis. And due to the acute hydrocephalus, a ventriculo-peritoneal shunt was performed.Postoperative, the patient had no more intracranial hypertention syndrome.A sputum stain for mycobacterium, the Mantoux test and culture isolation of mycobacteria were negative, as well as the cerebrospinal fluid assesment and angiotensin enzyme assay which were normal. The patient was discharged one week after surgery and was referred to the internal medicine department. Thereby, she underwent a salivary gland biopsy  which coIIPTs are a rare entity, described as a reactive, inflammatory and non-neoplastic phenomenon that can occur in any area in the human body, but mainly involves lungs and orbit . PosteriThe pathogenesis of inflammatory pseudotumors remains unclear . HistoloSarcoidosis is a chronic granulomatous disease of an unknown origin, characterized by noncaseating granuloma ,6. This thventricule. Cerebrospinal fluid assessment can show high protein content, pleocytosis and rarely low glucose concentration (Hydrocephalus, communicating or noncommunicating, may be seen as a complication of meningeal inflammation . Howeverntration , 16. Thintration . Angiotentration . The treth ventricle. Thus, its surgical resection was more difficult and dangerous. We solved at first the problem of hydrocephalus with perspective of treating the underlying lesion. Afterwards, surgical approach was laid off after discovering the inflammatory nature of the tumour.Our approach was different from those found on the litterature, which were based on surgical resection of the tumor . In factThe prognosis of the IIPT depends on the etiology and the site of the tumor. Even though they are considered as benign entity, they can be aggressive locally or have distant dissemination , 8.In conclusions inflammatory pseudotumors remain one of the most uncommon differential diagnosis of the central nervous system tumors. Among their etiologies, neurosarcoidosis is one of the less known and less frequent, mainly when located in the posterior fossa, leading to confusion with other pathologies. MRI has an important role in the diagnosis of these lesions, and the determination of their etiology. It shows other than the IIPT itself, there are many other signs such as leptomeningeal enhancement, nodular lesions or pituitary stalk thickening. These signs can orientate towards the diagnosis. Treatment may associate to symptomatic approach, and corticosteroids. Surgical resection may be proposed when the diagnosis remains doubtful.


Uterine leiomyosarcoma distant metastasis is common, and lung metastasis has been reported. However, unique cases have been identified either with late onset of metastatic disease or with large size of lung metastasis. A typical approach to avoid metastasis would be a hysterectomy. Nonetheless, metastatic recurrence is common. We encountered a case at our hospital with leiomyosarcoma metastatic to the lungs. Lung metastasis was noted to be 17 cm in diameter. This size has not yet been reported in the literature to the best of our knowledge. The lungs can be involved in many metastases from a variety of tumors. Severe respiratory compromise may ensue when a significant portion of the lung is involved . The mosWe report the case of a 47-year-old woman who presented to the emergency department with shortness of breath that had been progressively worsening over the past three weeks. The patient has been experiencing difficulty breathing upon ambulation and during prolonged speech. She described a feeling of chest tightness associated with her shortness of breath but otherwise denied any cough, peripheral swelling, fever, recent infection, recent travel, or sick contact exposure. She, however, had a medical history of uterine cancer diagnosed one year prior to the presentation. She was initially admitted to the hospital for heavy menstrual bleeding, which was diagnosed as being caused by a tumor noted on computed tomography (CT) Figure .The tumor was 19 cm in diameter with bilateral moderate hydronephrosis and hydroureter. She underwent a total abdominal hysterectomy. Pathology revealed leiomyosarcoma stage 4, for which she underwent chemotherapy and radiation therapy. She has been following up with her primary oncologist without any new symptoms or metastatic disease until the current presentation. She did not experience any symptoms after therapeutic interventions aside from the onset of dyspnea. She visited her primary oncologist, who advised a full-body positron emission tomography (PET) and CT scan, which revealed multiple areas of uptake, mostly within the right lung fields. She then underwent a chest X-ray, which revealed a completely opacified right hemithorax with tracheal deviation to the left Figure .She was admitted to the medical floor for management. A therapeutic pleural tap drained 1750 ml of fluid and revealed exudative findings with a white blood cell count of 34/mcl with 85% lymphocytic predominance. The pH of the pleural fluid was 7.8; lactate dehydrogenase was 874 µ/l; and albumin was 2.7 g/dl. There was no growth in fluid culture. An acid-fast stain was performed and was negative. Cellularity was obtained and confirmed as being cancerous cells of leiomyosarcoma origin. After the pleural tap, the patient did not improve, and due to suspicious lesions on the chest X-ray, a CT chest was performed, and a large leiomyosarcoma mass was noted within the right lung field with compression of the mediastinal structures (Figure The mass was 17 cm in its largest dimension. A delineation revealed the persistence of pleural fluid around the mass. Radiation oncologists advised radiation therapy sessions, and communication with the patient was made to initiate home oxygen therapy. The patient received radiation therapy. However, she passed away two months after her diagnosis.Leiomyosarcoma is a type of rare cancer that arises from smooth muscle cells. Smooth muscle is located in the hollow organs of the body, such as the gastrointestinal tract, blood vessels, uterus, and urinary bladder. Leiomyosarcoma can arise from any location but most commonly occurs in the uterus in females, the retroperitoneum, or the intra-abdominal region . Among aUterine leiomyosarcoma can metastasize to any organ system. The most commonly involved organs are the lungs, abdomen, and brain. The most common distant metastasis is to the lungs . PulmonaPatients with lung metastasis commonly present with cough, dyspnea, and chest pain. Some patients may remain asymptomatic at presentation . Our patThe prognosis of leiomyosarcoma is very poor. In advanced stages, they are likely incurable, as the response to currently available treatment is not satisfactory . The iniPatients should be followed regularly after treatment of uterine leiomyosarcoma since recurrence might occur at any time after treatment completion. Early diagnosis may prevent diffuse metastasis through a surgical approach. While leiomyosarcoma of the uterus has been reported in the literature to metastasize to the lungs, larger metastatic lesions have been rarely reported. Further investigational therapeutic interventions might be required in order to prevent or avoid any metastatic potential. Future research delineating oncogenic receptors involved in metastatic disease might be of benefit to such a patient population.


Translevator perineal hernias are rare pelvic floor defects that can be classified based on their anatomical location as either anterior or posterior relative to the transverse perineal muscle. This report describes an unusual clinical and radiological presentation of a posterior perineal hernia in an elderly lady. The case highlights the limitations of CT when findings are not definitive and the role of MRI as a superior problem-solving tool to better delineate the pelvic anatomy, exclude sinister pathology and confirm the diagnosis. An 84-year-old lady presented with worsening frailty and unintentional weight loss. No altered bowel habits or tenesmus was reported. Her past medical history included Parkinson’s disease and hysterectomy for a non-cancerous cause. Clinical examination and blood tests were unremarkable.The patient underwent CT chest, abdomen and pelvis as a part of the initial workup to exclude any sinister pathology. CT showed eccentrically thickened left posterolateral rectal wall with an exophytic component breaching the meso-rectal fascia and extending into the left ischiorectal fossa at 4 o‘clock position raising the concern for a sinister locally advanced rectal mass . HoweverRectal cancer was the main differential diagnosis in our case which is unique to the already documented benign differential diagnoses in the available literature including pelvic lipomas, fibromas, rectoceles, cystoceles or rectal prolapse.Given the patient’s age, frailty and lack of symptoms, the patient was managed conservatively for this small incidental posterior perineal hernia. Primary acquired perineal hernias occur due to excessive stretching of the pelvic floor muscles including pregnancy, childbirth, constipation, obesity or chronic ascites. In our case, given the patient’s demographics, previous hysterectomy and anatomical location, we propose that the posterior perineal hernia is likely an acquired type. Secondary perineal hernias are incisional hernias related to previous surgeries, for example, abdominoperineal or urological procedures. Anatomical classification of perineal hernias is relative to the transverse perineal muscle, either anterior or posterior in location (Aetiological classification of perineal hernias include primary and secondary types. Primary congenital perineal hernias are rare and have been associated with failure of regression of the peritoneal cul de sac in the embryo.location . Before the era of CT and MRI, diagnosis was either clinical or using barium enema. One case report in 2018 described the utility of 3D pelvic floor ultrasound. Most of the published cases have used CT scan for final diagnosis. To our knowledge, only three cases of perineal hernias have been reported in the literature using MRI as a diagnostic modality. There is no other published case on posterior perineal hernia secondary to only advanced age and previous hysterectomy as risk factors with MRI providing the diagnosis.Approximately 100 cases of posterior perineal hernia have been reported in the literature. Most cases are female with only a few reported in males and paediatrics. A soft and reducible perineal, labial or gluteal mass may be palpable due to bowel, bladder, fat, lymph nodes or vessels in the hernial sac. Occasionally, intestinal obstruction or urinary dysfunction can occur. Strangulation is uncommon as the hernia neck is typically wide with an elastic muscular defect. There is no established ideal surgical approach for symptomatic cases. Surgical reduction and pelvic floor reconstruction are offered to suitable candidates as a definitive treatment option. Surgical approaches described in the literature include abdominal, transperineal or combined operation.In symptomatic cases, common presentations are pain, incontinence or skin ulceration.In conclusion, this is a rare case of an elderly lady with an incidental finding of posterior Richter’s type perineal hernia. Initial CT findings raised the concern of a sinister locally advanced rectal mass. MRI pelvis provided a definitive diagnosis of perineal hernia but no sinister rectal pathology. Our case highlights the limitations of CT when findings are not definitive and the role of MRI as a superior problem-solving tool. Asymptomatic posterior perineal hernias can be managed conservatively and there is no established ideal surgical approach for symptomatic cases.Perineal hernia can be an asymptomatic incidental finding on imaging.CT imaging of perineal hernia can mimic a sinister rectal mass while MRI can better delineate the anatomy and confirm the diagnosis.Understanding of key anatomical landmarks in pelvic floor anatomy and accurate interpretation of MRI imaging can prevent misdiagnosis as a pelvic mass.Written informed consent was obtained from the patient for publication of this case report, including accompanying images.


Her general, cardiovascular and respiratory examinations were all normal. A resting electrocardiogram (ECG) showed a normal sinus rhythm, PR interval 126 ms, prolonged QRS duration 138 ms, and a delta wave (pictured - lead II). Leads I, II, and aVF had inverted T waves. Ventricular pre-excitation, Wolff-Parkinson-White syndrome type B was diagnosed. Echocardiography was unremarkable. The nature of her condition was explained to her. Advice to avoid medications like verapamil and digoxin was offered along with a recommendation to avoid over-strenuous activity. Screening ECGs were advised for her immediate family. As she had been asymptomatic up to this point, cardiology follow-up was recommended for consideration of a prophylactic ablation procedure. This case illustrates the benefit of a high index of suspicion and the utility of electrocardiography in diagnosing a rare condition.A 25-year-old female presented for a medical consultation complaining of sudden central chest pain, pleuritic in nature. She had never experienced such pain before. She denied experiencing coughing, shortness of breath or other respiratory or cardiac symptoms. Notably her grandparents, now in their 80s, were apparently being treated for ischemic heart disease attributed to their old age. She occasionally drank alcohol < 20 units per week. She was not using tobacco. On examination, her body mass index was 24.8 kg/m