Biliary cystadenoma  is a rare cystic tumor that arises from the biliary epithelium. The cause of biliary cystadenoma is still unclear. Jaundice is a rare presentation of intrahepatic biliary cystadenoma, which can lead to a diagnostic dilemma. Herein, we present a case of intrahepatic biliary cystadenoma that primarily exhibited as jaundice. A 56-year-old woman has suffered from yellow staining of her skin and sclera for more than 1 month. She had a poor appetite and mild epigastric pain. Laboratory examination showed elevated levels of total bilirubin and elevated carbohydrate antigen 19-9 (CA19-9). A contrast-enhanced computed tomography of the abdomen showed a 7.4 * 5.3-cm, oval, low-density lesion in the left liver parenchyma with a clear boundary and visible septa. The common bile duct was obviously dilated with wall thickening. On magnetic resonance imaging, the lesion in the liver showed a multilocular cystic, unenhanced long T2 signal. There was local thickening of the common bile duct wall with short T2-like filling defects and high signal intensity on diffusion-weighted imaging (DWI). The patient had no history of other malignant tumors and adjuvant therapy such as radiotherapy and chemotherapy. She was clinically suspected of having either biliary cystadenoma or a malignancy; hence, resection was performed. Macroscopically, the excised tissue specimen showed a polypoid mass in the common bile duct, which extended along the bile duct to the intrahepatic bile duct. There was a cystic and solid mass in the left liver with yellow turbid fluid, which was associated with the polypoid mass in the common bile duct. Histopathology suggests mucinous cystadenoma of the liver and hilar bile duct. The differential diagnosis of biliary cystadenoma and treatment selection have been discussed. Obstructive jaundice is a heterogeneous disease characterized by bile duct obstruction and cholestasis. Patients may have typical clinical manifestations such as jaundice of the skin and sclera, dark yellow urine, and lighter or even clay-white stool. It is vital to determine the nature of the lesion for the formulation of the treatment plan. However, the differential diagnosis of obstructive jaundice is usually difficult.Biliary cystadenoma is a rare neoplasm arising from intrahepatic or extrahepatic bile ducts. The majority of biliary cystadenoma arises from the intrahepatic ducts and is usually asymptomatic. The presenting symptoms are usually right hypochondrium pain or abdominal mass. Extrahepatic biliary cystadenoma usually presents with obstructive jaundice either directly by tumor blockage or by mucin secretion. In this paper, we present a patient with biliary cystadenoma involving the left intrahepatic and left main hepatic ducts presenting as obstructive jaundice. We also discussed the differential diagnosis and treatment selection of biliary cystadenoma.12/L; reference range, 3.8 * 1012/L to 5.1 * 1012/L) and a decreased hemoglobin . Autoimmunological test and IgG4 were normal. Hepatitis B serologies demonstrated negative hepatitis B surface antigens and hepatitis C virus antibodies. Serum tumor marker examination showed elevated carbohydrate antigen 19-9 (CA19-9) , while CA125 and carcinoembryonic antigen were within the normal range. A contrast-enhanced computed tomography of the abdomen showed a 7.4 * 5.3-cm, oval, low-density lesion in the left liver parenchyma with a clear boundary and visible septa. The septation was slightly enhanced, and there was no enhancement of its internal fluid components. The intrahepatic bile ducts were mildly dilated. The common bile duct was obviously dilated with wall thickening. The gallbladder was normal in size, but the wall was thickened. In addition, slightly larger retroperitoneal lymph nodes and cystic shadows in the duodenum could be seen. No obvious discontinuity or filling defect was found in the portal vein branch and trunk, and the presence of a cancer thrombus was not considered , direct bilirubin , indirect bilirubin , alanine aminotransferase , aspartate aminotransferase , gamma-glutamyl transferase , and alkaline phosphatase . A blood cell routine showed a decreased red blood cell count : this kind of patient can present with non-specific clinical manifestations such as abdominal pain and jaundice. This disease can be manifested as hepatic cystic lesion and dilatation of the intrahepatic bile duct and common bile duct on CT; its clinical manifestations, laboratory examination, and imaging are not specific. The diagnosis of this disease mainly depends on postoperative pathological examination.Biliary cystadenoma: the patient was a middle-aged woman with progressive jaundice. CT showed a well-defined, low-density intrahepatic mass with internal septa, which showed mild enhancement on contrast-enhanced CT. MRI showed a well-defined multilocular high signal occupation with internal septa on T2WI. Mucinous cystic neoplasm often occurs in the liver. According to the principle of monism, we considered that the mass of the hilar may be caused by partial detachment of the intrahepatic mass, thus causing obstructive jaundice.Cyst of the liver is a common benign disease and generally will not exhibit clinical symptoms, and very few people will be jaundiced due to cyst compression of the bile duct. The most common clinical type is a congenital hepatic cyst, which can be divided into two subtypes: single and multiple. Multiple hepatic cysts can be seen in the liver with multiple dark areas of fluid, and several cysts may be fused to communicate or appear as several septations within a large cyst. In addition, the congenital polycystic liver is often accompanied by cysts in other organs such as the kidneys and lungs. This patient does not exhibit the typical clinical features of a cyst of the liver.Cholangiocarcinoma is a diverse group of malignancies emerging from the biliary epithelium. According to its anatomical site, cholangiocarcinoma can be divided into intrahepatic cholangiocarcinoma (iCCA), perihilar cholangiocarcinoma (pCCA), and distal cholangiocarcinoma (dCCA). They have different clinical features. The most common symptoms of iCCA patients are right upper abdominal pain and weight loss. Jaundice occurs in approximately 25% of patients, and CA199 levels can be elevated in some patients. iCCA appears as a focal hepatic mass on CT and MRI, and the bile duct surrounding the mass may be dilated. The typical finding on an enhanced scan is that the mass has peripheral or central enhancement. Patients with pCCA may have obvious jaundice. The lesion shows delayed enhancement on CT. The characteristic is helpful for the differential diagnosis of pCCA. Histopathology is the gold standard to determine the diagnosis of cholangiocarcinoma.Intraductal papilloma neoplasm of the bile duct is divided into intraductal papillary mucinous neoplasm of the bile duct (IPMN-B) and IPNB-without mucin secretion (IPNB-NM) according to the presence or absence of mucous secretion. While IPMN-B has similar histological features to the corresponding pancreatic lesions, IPNB-NM may represent a distinct type. IPMN-B communicates with the bile duct; mucus or tumor debris can cause intermittent bile duct obstruction, thereby causing jaundice. As the bile duct pressure rises, the mucus can be discharged, and the obstruction is relieved. Thus, jaundice is often fluctuating. Secondary dilatation of the duodenal papilla has been observed in some patients. The presence of extensive bile duct dilatation with or without tumor on imaging and mucus outflow from the opening of the duodenal papilla under endoscopic retrograde cholangiopancreatography (ERCP) are highly suggestive of this disease. Compared with IPMN-B, IPNB-NM has no mucus secretion, the downstream biliary obstruction usually does not dilate, and the degree of biliary dilation is not as obvious as that of IPMN-B. Compared with IPMN-B, IPNB-NM is more aggressive and has a worse prognosis. IPNB may have different imaging findings according to the location of the tumor, the size and morphology of the mass, and the degree of mucin secretion .Biliary cystadenoma is a rare cystic tumor that tends to be more common in middle-aged women. This study reports an intrahepatic biliary cystadenoma partially shedding into the common bile duct, causing obstructive jaundice. Biliary cystadenoma mostly occurs in the liver and is often found due to non-specific abdominal pain or jaundice. On CT, it can appear as a low-density mass in the liver with internal septa and wall nodules, and enhancement can be seen along its wall and internal septum. MRI usually shows multilocular hyperintensity on T2WI with a well-defined internal septum and variable signal intensity on T1WI. Increased thickening and enhancement of the septa, nodules, and capsule with coarse calcifications have been cited as determinants of malignancy  for the publication of any potentially identifiable images or data included in this article.K-SL evaluated the patient. Y-CL and A-DS initiated the case report, reviewed the literature, and drafted the manuscript. K-SL: concept and design, study supervision, and critical revision of the manuscript for important intellectual content. All authors contributed to the article and approved the submitted version.


Tumefactive multiple sclerosis comprises a rare subset of multiple sclerosis that often poses a diagnostic challenge to physicians. It is unique in its presentation as a solitary lesion, usually larger than 2 cm, with surrounding vasogenic edema, commonly mimicking a primary intracranial malignancy. We present a case of a 25-year-old female with no significant past medical history who presented to our institution with homonymous superior quadrantanopia. During her admission, she underwent a magnetic resonance imaging (MRI) of the brain, which revealed a large lesion in the left temporal area surrounded by marked edema. A thorough workup revealed a diagnosis of tumefactive multiple sclerosis. Subsequently, she was initiated on intravenous immunoglobulin rather than stress dose steroids, given the concern for a superimposed infection. Interestingly, the patient had a paradoxical progression of her symptoms as well as expansion of the vasogenic edema on a repeat MRI. In our case, we highlight the key differences in tumefactive multiple sclerosis diagnosis and management. Tumefactive multiple sclerosis (TMS) represents a rare form of multiple sclerosis characterized by a solitary lesion usually larger than 2 cm with surrounding edema. Although its incidence is thought to be 3 out of a million people per year, its recognition is nonetheless important . In contA 25-year-old female without a significant past medical history was referred by her ophthalmologist for new-onset homonymous superior quadrantanopia. Upon arrival at the emergency room, a non-contrast CT of the head revealed a large area of vasogenic edema in the left frontal and temporal lobes, as shown in Figure Further evaluation revealed a diagnosis of Graves' disease, which raised our suspicion for a concomitant autoimmune diagnosis of TMS. The decision was made to begin treatment with IVIG rather than with stress steroids due to concern for possible underlying infection. Interestingly, the patient's initial complaint of homonymous superior quadrantanopia soon progressed to homonymous hemianopia. This was accompanied by worsening right-sided numbness and tingling. A repeat MRI obtained five days after initiation of IVIG revealed extension of the vasogenic edema, as shown in Figure TMS is a rare subset of multiple sclerosis. The prevalence of TMS is estimated to be 1-3/1,000 cases of multiple sclerosis or three cases per million per year in the general population. TMS may occur at any age but is most often seen in ages 20-30 .High-dose corticosteroids are considered the first-line treatment in the subset of patients with TMS. Approximately 86% of patients show improvement with treatment. In those patients without symptomatic improvement, plasmapheresis is considered as second-line therapy . IVIG haThe long-term prognosis of TMS is variable. Approximately one-third of patients will not develop a subsequent demyelination attack. In the subset of patients who do, two-thirds of them will follow the relapsing-remitting course. Early recognition of this rare disease and its appropriate management are paramount in improving outcomes in patients diagnosed with TMS.TMS remains a challenging disease in terms of its pathogenesis and diagnosis. Despite being categorized as a subset of multiple sclerosis, our experience has taught us that it can behave differently than the traditional multiple sclerosis we commonly see. Increased tumefaction with IVIG therapy in our case highlights the need for further research in the field of this often misdiagnosed phenomenon.


A subhepatic appendix and an interlobar hepatic bridge are both rare anatomical variants. To find both entities in the same patient at the time of a laparoscopic cholecystectomy makes for a unique case report. Subhepatic appendicitis has a reported incidence of 0.08%, and there have been only published three case reports of an interlobar hepatic bridge. Their lack of involvement in acute cholecystitis facilitated an easier dissection process and prevented serious complications to the liver or the right hemi-colon.  A subhepatic appendix and an interlobar hepatic parenchymal bridge are rare anatomical entities. A case report with both conditions in the same patient makes for a truly unique presentation.Their lack of involvement in the acute cholecystitis facilitated an easier laparoscopic gallbladder extraction and reduced the risk of more extensive complications to either the liver or the right hemi colon in this case report.A 54-year-old female patient was referred to the acute care surgical team with acute onset abdominal pain over the epigastrium. The pain radiated to her back, and there was a food–pain association with fatty meals. This was her second admission in a month, and she was previously treated for gallstone pancreatitis. Her pain profile was similar over the two hospital admissions. Clinically, her abdomen was tender on deep palpation over the epigastrium. She had no overt signs of peritonism. She remained hemodynamically stable throughout her clinical examination. Significant laboratory investigations included a leucocytosis and an elevated lipase over five times the normal range. Liver functions were normal.Computerized tomography scan of the abdomen showed an acute cholecystitis as well as radiological features of an acute pancreatitis. There was no pancreatic necrosis, abscess or choledocholithiasis.She was managed conservatively with analgesia, diet modification and antibiotics and responded well to medical management. After her clinical symptoms settled, we discussed further management, and she signed an informed consent for a laparoscopic cholecystectomy at this admission.Intraoperatively she had severe inflammation at the Calot’s triangle, and we performed a retrograde, fundus down and cholecystectomy. She was noted to have a subhepatic appendix along with a high riding cecum  and 2. TConcurrently, we identified an interlobar hepatic bridge between the right and left liver lobes isolated from the cholecystitis . The intHer postoperative recovery was uneventful, and she was discharged home.Pathology confirmed an acute cholecystitis.et al. in 1955 [Subhepatic appendicitis was first described by King  in 1955 . Delays  in 1955 . Inciden in 1955 , 8 excluet al. [Contributing to the unusual nature of this case was the presence of a subhepatic bridge containing liver parenchymal tissue. Only three other cases of an interlobar parenchymal bridge could be found in the literature . The firet al.  in a pubWe present a rare case report of a subhepatic appendix and a concomitant interlobar hepatic bridge observed during a laparoscopic cholecystectomy, which we believe to be the first in the published literature.


Sacrococcygeal teratomas (SCT) are known as rare tumors, but they are the most common tumor in fetuses and newborns. This study aims to present fetal magnetic resonance imaging (MRI) findings of SCT diagnosed prenatally and compare them with that of the prenatal Ultrasound (US) findings.Eleven patients diagnosed as SCT prenatally by US and further assessed by MRI are included. MRI was performed via a 1.5-T magnet with a body coil. The presence, size, content extension, and compressive effects of each mass were determined and correlated with US findings and with postnatal examinations, including surgery and pathology. As complications, the presence of ascites and skin edema or pleural or pericardial effusion was diagnosed as hydrops. The amniotic fluid index was calculated for the assessment of oligo- or polyhydramnios.US findings are found strongly correlated with MRI findings. An agreement on the extent of each mass was observed in eight patients, disagreement in one fetus was an extension of the tumor within the spinal canal recognized only at MR and assessment of intrapelvic-abdominal extension was superior in MRI. There were n = 6 type I, n = 2 type II, n = 1 type III and, n = 2 type IV tumors. MRI was superior to US for detecting displacement of the colon (n = 3), intrapelvic-intraabdominal extension (n = 1), urinary tract complication (n = 2), and intraspinal extension (n = 1).MRI is found to be superior to US especially in the assessment of intrapelvic and intraspinal extension of the tumor, colonic displacement, and complications. Sacrococcygeal teratomas (SCT) are known as rare tumors, but they are the most common tumor in fetuses and newborns. The incidence reported in the literature is 1/35,000 to 40,000 live births ,7. The vDiagnosis is usually made during the Ultrasonography (US) scan performed in the second or third trimester of pregnancy ,12. On UThe University Hospital research ethics committee approved this study (ID 2021-340). Written informed consent was waived for the retrospective analyses by the Institutional Review Board. We conducted a retrospective single-center study at our University Hospital to review all fetal SCT cases suspected before birth and confirmed postnatally between January 2011–2021. Data were collected from the digital patient archiving system . Most cases were submitted with a presumed diagnosis of tumor or fetal hydrops. All pregnant women underwent a detailed fetal anomaly scan using high-resolution US equipment.Type I: developing only outside the fetus ; the majority of cases, Type II: extra-fetal with intra-pelvic presacral extension, Type III: extra-fetal with abdominopelvic extension, Type IV: tumor developing entirely in the fetal pelvis. As complications, the presence of ascites and skin edema or pleural or pericardial effusion was diagnosed as hydrops. The presence of oligo- or polyhydramnios was determined by calculating the amniotic fluid index. Descriptive statistics were used due to the small number of cases. Categorical data are given as numbers (n) and percentages (%).Fetal ultrasonographic evaluations were performed by a perinatologist (at least 10 years of experience) and a radiologist (at least 15 years of experience) experienced in the obstetric US, separately. Fetal growth, exact tumor dimensions, morphology, and location, the presence or absence of additional malformations, and assessment of amniotic fluid volume were evaluated. There was no conflict between the experts in the interpretation of the images. It was agreed that MRI should be performed as advanced imaging to evaluate the extent of SCT and additional anomalies. MRI was performed via a 1.5-T magnet  with a body coil. In our center, fetal MRI (1.5 Tesla) is standard for additional evaluation. The mother was positioned either supine or in a partial left lateral decubitus position. The following imaging sequences were performed: T2-weighted Turbo Spin Echo sequences of fetus in sagittal, coronal, and axial planes ; T1-weighted gradient-echo relative to the fetus in the axial plane ; and diffusion-weighted imaging in the axial plane . The scanning time for all was 16–18 min. All MR studies were reviewed and interpreted by two radiologists, who also knew the results of MRI results were compared with US reports and images. The presence, size, content extension, and compressive effects of each mass were determined and correlated with US findings and with postnatal studies, including surgery and pathology. MRI findings were confirmed at surgery or autopsy in all patients. Depending on the radiological findings, the extent of the SCT was classified according to the American Academy of Pediatrics, Surgical Section classification which includes four categories : Type I:Eleven patients diagnosed as SCT prenatally by US and further defined by MRI are included. Diagnostic quality MR images were obtained in all patients. All images taken in MR examination were evaluated. US findings are strongly correlated with MRI findings. An agreement on the extent of each mass was observed in eight patients, disagreement in one fetus was an extension of the tumor within the spinal canal recognized only at MR, and assessment of intrapelvic-abdominal extension was superior in MRI. On MRI, sacrococcygeal teratomas (n = 11) were completely cystic in four fetuses, mixed and solid in seven . AccordiSCT causes displacement of the bladder by the effect of pelvic mass, hydronephrosis, and large convoluted ureters. The bladder displacement was observed in 3 of 11 fetuses (27.2%), and bilateral hydronephrosis in 2 fetus and unilateral hydronephrosis in 1 fetus on MRI, but only hydronephrosis was detected by sonography. In the patient with unilateral hydronephrosis, no other kidney was observed , and oligohydramnios was present. In the pathological examination, the cases were documented as mature (n = 8), immature (n = 2) and mixed teratomas (n = 1).In this study, we aimed to evaluate imaging findings and analyze which imaging approaches are most valuable in fetuses diagnosed with prenatal SCT. SCTs are the most common tumors of the fetus and the neonate, carrying a variable prognosis. The majority tend to be benign (~80%). Alpha-fetoprotein (AFP) and beta HCG may be elevated. A pathology-based classification is as: benign (mature) 70%–80% or malignant (immature). Those presenting in older infants tend to have a higher malignant potential and those presenting in utero have a poor prognosis due to complications. It has been reported that fetal mortality is higher if the gestational week at the time of diagnosis is early in SCT . Mass siComplications include urologic complications (the most common cause of morbidity), high output cardiac failure from AV shunting: which in turn can cause hydrops fetalis, gastrointestinal tract obstruction, compression of underlying nerves leading to urinary/fecal incontinence, anemia, dystocia, tumor rupture. Tumor compression of the bladder outlet caused retention in the urinary system, followed by renal impairment, oligohydramnios, and pulmonary hypoplasia , also tuDifferential diagnoses include sacral chordoma, sacral meningocele, terminal myelocystocoele, and enteric (tailgut) cyst for cystic types and for type IV lesions also low-lying neuroblastoma, rhabdomyosarcoma, small round blue cell tumor in the sacral region. Treatment is with surgical excision inclusive of coccygectomy with additional chemotherapy for malignant ones.MRI is widely used in these fetuses, although there are few studies in the literature that it is effective ,21–23. MThe limitation of our study is that it was designed retrospectively, US were performed by a perinatologist and a radiologist, separately. Although there was no conflict among our researchers, perhaps consensus would be a better alternative.MRI is found to be superior to US, especially in the assessment of intrapelvic and intraspinal extension of the tumor, colonic displacement, and complications. While MRI contributes to the diagnosis, it also serves as a guide for treatment, birth planning, and counseling. In order to optimize pre and postnatal management for the evaluation of fetal sacrococcygeal teratoma, fetuses with SCT detected by US should be referred to MRI to evaluate tumor size, content, and extent.


Background: There are many reconstruction methods after sternal tumor resection, but the method that LARS mesh combines with bone-cement has not been reported.Case report: A 54-year-old female patient and a 55-year-old male patient admitted to our department all presented with sternum masses, but neither presented with respiratory disorders. In women with limited manubrium sternum lesions, we resected the manubrium sternum completely. In men with sternal lesions, we removed part of the sternum and part of the sternocostal joint. The patients recovered well after surgery, and there were no respiratory complications and no tumor recurrence during the 1-year follow-up respectively.Conclusion: We report two cases of sternal defect repair using LARS mesh combined with bone cement. This method is safe and stable, and can achieve satisfactory results. Primary chest tumours are infrequent, accounting for less than 1% of primary bone tumours , and onlSurgery is the primary treatment for sternal tumours when obvious clinical symptoms occur, whether malignant or benign. Radical resection is the optimal therapy for surgeons to guarantee non-tumour margins and low recurrence rates . HoweverReconstruction usually consists of two aspects: skeletal reconstruction and soft tissue reconstruction . Soft tiIn this cases, we support the use of an innovative ligament advanced reinforcement system (LARS)-bone cement sandwich as a prosthesis for reconstruction of the chest wall, which has not yet been reported in the literature. This new method achieved a good therapeutic effect, with uneventful recovery at the 1-year follow-up.We report the cases of a 54-year-old female patient and a 55-year-old male patient who presented to our department with sternal masses, from March to May 2021; both patients were operated on by the same doctor. In the female patient, the lesion was located in the manubrium sternum, and was diagnosed as chondrosarcoma by postoperative pathology. In the male patient, the lesion was located in the middle of the sternum with involvement of the fourth rib, and was diagnosed as bone fibrous dysplasia based on postoperative pathology . Both patients underwent preoperative chest 3-dimention computed tomography  . MeanwhiBoth patients were placed in the supine position during the operation. The patients were anaesthetised using two-chamber endotracheal intubation after routine testing of blood oxygen and blood pressure and evaluating the electrocardiogram findings. The arterial catheter was indwelling for observation of blood gas analysis in surgery.For sternal tumours, we ensured an adequate resection area according to individual principles, which meant the confirmation of incision and excision margin depended on intrinsic properties of tumours. Preoperative printing of the 3D model played a positive role in understanding the lesion scope and determining the surgical plan. For the female patient with a lesion limited to the manubrium sternum, the platysma muscle, sternocleidomastoid muscle, sternoclavicular joint, sternocostal joint, and the joint between the sternocleidomastoid and sternum were sequentially severed after exposure. For the male patient with a lesion located in the sternal body, we separated the sternal body from the manubrium sternum and bilateral third-sixth sternal costal joints. The residual costal stumps were proven to be non-tumourous on intraoperative pathology examination. The tumor histological characteristics of the female patient presented as lobulated hyaluronic cartilage nodules, which resembled normal cartilage to some extent. The margin of the tumor was white and yellow granular. The tumor histological characteristics of the male patient presented as no obvious boundary of osteocele, cystic fibrous changes, and the bone marrow cavity was occupied by grayish white or grayish red hyperplastic fibrous tissue.After removing the sternal lesions, bone cement was poured into a pre-made mould. The mould was fabricated using 3D printing technology based on CT information to ensure its suitability. This mould is generally composed of two parts: the bottom and the cover. During fabrication, we first poured an appropriate amount of paste bone cement into the bottom and then covered it with a cover to ensure that the prosthesis surface was smooth. Subsequently, holes were drilled at the edge of the bone-cement prosthesis. The bone cement prosthesis was implanted in the sternal defect, and it was firmly fixed on the adjacent ribs and the residual sternum with non-absorbable wires. Next, the sternocleidomastoid muscle was sutured to the cement, which stabilised the sternocleidomastoid joint. We then fixed the LARS mesh to the surface of the cement prosthesis and the peripheral ligament on the pectoralis major muscles through interrupted non-absorbable sutures on the holes reserved in the cement. A drainage tube was inserted in both the patients during the operation. The surgical procedures were seen in Both patients were routinely transferred to the ward for primary care after surgery. They were provided with postoperative anti-inflammatory and analgesic therapy, and their respiratory function was closely monitored. When the drainage flow was less than 20 mL for 2 days, the drainage tube was removed. The average drainage time of the two patients was 8.5 days, and the average hospital stay was 16.5 days. No major postoperative complications occurred in either patient. The male patient experienced mild dyspnea on the night of surgery, which may have been caused by incision pain.Postoperative pain management is extremely important in patients undergoing sternal resection. Furthermore, thoracic movement should be limited to avoid implant displacement. A small range of implant forward movements could be accepted, while implant back movements must be avoided, as this could lead to respiratory and circulatory failure. Both patients underwent chest tomography after drainage extubation that revealed sternal prosthesis located at proper position and cover the defect well . A folloThe sternum is a rare site for neoplasms, and surgery is the most important treatment for sternal tumours . ConsideIt has been reported that various materials were used in chest reconstruction. Rigidity is the most important property that can prevent flail chest and protect organs . In thisTo overcome these bottlenecks, we proposed a new sandwich structure, referred to as PMMA, encapsulated with a LARS mesh. PMMA can provide sufficient support to the chest wall , is easyIn addition to PMMA, the LARS mesh is also essential for chest reconstruction. The restoration of respiration and movement of the upper limbs required micro-motion of the sterno-clavicular sternocostal joints, which was possible due to the elastic property of the mesh . TraditiBesides chest wall reconstruction, soft tissue coverage of the prosthesis is necessary to prevent infection. In different situations, primary closure, skin grafts, myocutaneous flaps, and local advancement flaps may be used . In thisSternal tumours are rare, and are generally treated by tumour resection followed by chest defect reconstruction, which remains a challenging procedure. Bony and soft tissue reconstructions are two prominent aspects of maintaining chest cosmesis, respiratory function, and organ protection. The LARS-PMMA-LARS sandwich prosthesis has potential to be a suitable substitute for the sternum. Proper soft tissue reconstruction also promotes restoration of chest function and structure.


Osteoid osteoma is the most common benign osteogenic bone neoplasm. Osteoid osteomas are typically located in the metaphysis and diaphysis of long bones, especially the tibia and femur. However, less common sites of the skeleton can be affected as well, including carpal bones. Among carpal bones, the scaphoid and the capitate are the most affected. Osteoid osteoma of the trapezium is an extremely rare entity, with only seven cases reported in recent literature. We present a case of a 29-year-old male with persistent left wrist pain who was diagnosed with an osteoid osteoma of the trapezium bone. The diagnosis was based on the patient’s history, clinical examination and findings from the CT scan, MRI, and plain radiographs. The patient was treated with an excision biopsy with no additional bone grafting. After a follow-up period of 12 months, no pain or signs of recurrence were present. We conducted a literature review to elucidate the clinical presentation as well as the proper diagnostic tools and therapeutic methods for this rare occurrence. Osteoid osteoma, which Jaffe first reported in 1935 -4. RevieA 29-year-old Caucasian male was referred to our upper limb department after reporting persistent left wrist pain for a period of six months without a history of trauma. Over the past eight weeks, the pain has become progressively worse, limiting his daily activities. From his past medical history, the patient was free of disease. The pain was most prominent at night, disturbing the patient’s sleep, and was occasionally relieved with the use of analgesics and oral anti-inflammatory drugs. Splinting has also been tried to alleviate pain without long-lasting favorable results.On physical examination, local tenderness over the volar area at the carpometacarpal joint of the thumb was evident with mild swelling. The range of movement of the carpometacarpal joint was slightly decreased compared to the contralateral side.A plain radiograph was obtained initially, revealing bone sclerosis Figure . SupportBased on the above clinical features and imaging findings, an excisional biopsy through a volar incision was decided Figure . The nidThe patient showed immediate improvement in his symptoms, and six months postoperatively, he has returned to his previous level of activity without restrictions and with a full range of motion Figures -9. PlainOsteoid osteoma is a common benign lesion, representing 10% to 12% of all benign bone tumors and 3% of all primary bone tumors ,8. The fThe most frequent clinical feature of osteoid osteoma is local pain, which is worsened at night and is typically relieved with non-steroidal anti-inflammatory drugs . Local tStandard diagnostic imaging is routinely obtained and consists of plain radiographs, CT, MRI, and bone scintigraphy. The typical radiolucent nidus can be obvious on plain radiographs in the case of a cortical lesion but may be absent in medullary lesions, especially in locations such as the carpus where radiography alone may not be sufficient to assess the bone. In this case, a sclerotic bone was found on the initial radiographic evaluation, prompting further investigation. MRI is reportedly less useful during the diagnostic procedure as it may lead to misdiagnosis and fail to identify small nidi ,11. HoweTo the best of our knowledge, only a few cases of osteoid osteomas located in the trapezium bone have been published in recent literature. Hundley  reportedWhile in other anatomic locations, different treatment options can be discussed for the management of such lesions, surgical or conservative, the treatment of osteoid osteoma of the carpal bones is surgical excision of the nidus -7. AnothIn conclusion, osteoid osteoma of the trapezium is an almost unknown entity for the unaware physician. A thorough history may reveal the characteristics of the pain, such as chronicity and nocturnal appearance, which should raise awareness. The diagnosis is generally reached by plain radiographs and CT tomography. The definitive diagnosis is set by histological examination. Treatment consists of cutterage of the osteoid osteoma and trapeziectomy in larger tumors. In that case, bone grafting and bone substitutes may be necessary. We present the eighth report of this lesion in the literature with the aim of raising suspicion about the presence of this entity in patients with long-lasting, disabling, undiagnosed wrist pain.


Hemangiomas are the most common vascular tumors in children. Although hemangiomas are common, they are rarely seen in areas like the trachea and larynx.The most common clinical manifestations in tracheal hemangiomas are stridor and respiratory distress. The main diagnostic method is bronchoscopy. Other imaging techniques like computed tomography scans and MRIs are also helpful. Various treatment options are now used for treating the disease, including beta blockers like propranolol, local and systemic steroids, and surgical resection.An 8-year-old boy with the chief complaint of severe progressive dyspnea and a history of neonatal postbreastfeeding cyanosis was admitted. On physical examination, he had tachypnea, and stridor was heard upon auscultation. There was no history of fever, chest pain, or coughing. He underwent a rigid bronchoscopy followed by a neck computed tomography scan. The results indicated a soft tissue mass with a vascular nature. An MRI of the neck confirmed the diagnosis of tracheal hemangioma. The mass was not resectable during surgery; hence, angioembolization was carried out. Treatment was successful and there was no recurrence on the follow-up.Based on the findings in this literature review tracheal hemangiomas present with stridor, progressive respiratory distress, dyspnea, hemoptysis, and chronic coughs. Advanced tracheal hemangiomas commonly do not reduce in size by themselves and need treatment. A close follow-up ranging from 3 months to 1 year is recommended.Although tracheal hemangiomas are rare they should be considered in the differential diagnosis of severe dyspnea and stridor. HighlightsHemangiomas are the most common benign soft tissue tumors in children. Even though their most common sites are the head and neck, they are rarely seen in the trachea and larynx.Tracheal or laryngeal hemangiomas can significantly affect patients’ lives, and even cause respiratory distress and arrest. Hence, proper diagnosis and treatment of these diseases are crucial. Tracheal hemangioma’s most common signs and symptoms are stridor, respiratory distress, progressive dyspnea, chronic cough, and hemoptysis.The main diagnostic tool for this disease is bronchoscopy.Treatment options vary from beta blockers like propranolol, local and systemic steroids, to surgical resection.2. Infantile hemangiomas occur in nearly 4–5% of infants, which may be present at birth or shown later in life3. Hemangiomas go through an involution stage once they have reached their complete growth near one year of age and it may take from months to years; however, a small percentage of hemangiomas never go through this stage and do not become smaller in size4. The most common areas of involvement are the head and neck with the subglottic region being the most involved4. Although hemangiomas usually do not require special intervention, factors such as hemangioma size, growth rate, and location can change this5.Vascular diseases in children and infants include vascular tumors and malformations. Hemangiomas are the most common among vascular tumors and also the most common benign soft tissue tumor in infants6. Several different approaches have been suggested over time for the treatment of tracheal hemangiomas including steroid injections, endoscopic and open excisions, and medications like systemic steroids and vincristine5.Hemangiomas residing in the larynx or trachea, although rare, need close monitoring and intervention as they may lead to irreparable and life-threatening consequences. Tracheal hemangiomas present in various ways in children and adults. Dyspnea, stridor, chronic cough, and obstructive respiratory disease are the most common signs and symptoms of tracheal hemangiomas in children. The main diagnostic tool for tracheal hemangiomas is bronchoscopyHere, we present a case of an 8-year-old with intraluminal tracheal hemangioma presenting with respiratory distress to an academic tertiary center, which was managed with three sessions of angioembolization.7.This manuscript was prepared following the Surgical CAse REport (SCARE) guidelinesAn 8-year-old boy without any documented medical or surgical history was admitted to a tertiary academic center with the chief complaint of progressive dyspnea. His parents mentioned that he used to suffer from postbreastfeeding cyanosis in infancy and also after crying. In the years after infancy and before school age the patient’s symptoms were described as exertional dyspnea becoming worse by exercising and walking accompanied by fast heartbeats and increased shallow breathes. As the patient had reached the school age, his dyspnea had become more severe with stridor heard while breathing. His dyspnea and stridor had progressed in the last 3 months. He had not experienced fever, chest pain, weight loss, and coughing in the course of his illness.2: 85%). Laboratory results were normal and no anemia or leukocytosis was seen.In general appearance, he was a child with a height of 125 cm and a weight of 27 kg, and physical examination revealed tachycardia (heart rate: 120), tachypnea (respiratory rate: 30), mild hypotension ( blood pressure: 90/70), and low oxygen saturation  scan was performed revealing a vascular mass behind the larynx and cervical trachea Figs. –3. SubseHe was transferred to the radiology department for an MRI with contrast. At MRI, a multiloculated nodular lesion in the intraluminal part of the trachea and also partly in the infra-glottic area, measuring 12×8 mm and taking up half of the tracheal lumen, was found. The lesion was hypo-signal in the T1 sequence, hyper-signal in the T2 sequence, and heterogeneous after the contrast injection Figs.  and 5. MThree sessions of angioembolization were carried out for the patient, and the patient’s progress was controlled with MRI. Finally, after hemangioma atrophy tracheostomy was extracted.He was given guaifenesin every 8 h and discharged from the hospital. At 3 month follow-up, the patient’s symptoms were significantly improved and repeated bronchoscopy did not demonstrate any recurrence.8. Hemangiomas are classified by the extent of tissue involvement as focal, segmental, and indeterminate, and by the depth of the tissue affected as deep, superficial, and mixed8.Hemangioma is a soft tissue tumor caused by hyperplasia of the endothelial cells. Hemangiomas are either congenital or infantile. Infantile hemangiomas appear in the first 8 weeks of birth and grow rapidly until 6–12 months of age, and then they usually shrink in a natural and physiological way and disappear completely. Congenital hemangiomas; however, are fully developed lesions at the time of birth9. Various factors may lead to the formation of these hemangiomas consisting of hormonal imbalance in pregnancy, infections, arteriovenous malformations, and vascular growth factors alteration10.Tumors of the tracheobronchial tree are a rare concept in children and are divided in two groups of benign and malignant lesions. The most common benign tumors of the tracheobronchial tree in children are hemangiomas and squamous papilloma11. A tracheal hemangioma can present as intractable asthma and may lead to respiratory failure and even death in severe cases.The low prevalence of this disease causes a delay in its diagnosis, as seen in this case. The presence of a mass in the trachea can cause symptoms such as stridor, cough, shortness of breath exacerbated by exercise, and hemoptysis (in the case of hemangiomas)9. Rigid bronchoscopy and flexible endoscopy are used to retrieve samples and histopathologic assessments confirm the diagnosis; however, some studies start the treatment with imaging findings and do not use pathologic samples for confirmation9.As the signs and symptoms of a tracheal hemangioma are not specific to the disease the initial diagnostic tool used for finding the etiology is a chest radiograph as in all the respiratory diseases. CXR is commonly not diagnostic for the disease but may exclude other differential diagnoses. Further diagnostic tools that have been proven to have appropriate sensitivity and specificity in diagnosing the lesion are the CT scan, MRI, and bronchoscopyThe case presented in the current study is a display of a tracheal mass presenting itself with respiratory distress and severe dyspnea.12. Finally, 15 studies were included in the review including 12 case reports and 3 case series. The summary of the studies are displayed in Table We used three databases of PubMed, Scopus, and Web of Science to conduct a literature review. Keywords used were ‘Tracheal Hemangioma’ and ‘Hemangioma of Trachea’ and all studies from 1 January 2013 to 5 March 2023 were included initially. The initial number of articles included was 368, which was then reduced to 216 after deleting the duplicates. After reviewing the remaining articles, irrelevant articles  were excluded. The remaining articles were assessed for eligibility to be included in this study and 20 articles were excluded due to the age of the patients, as only pediatric case series or case repots are included in the current study. Although one study was conducted on patients with subglottic hemangiomas with reported tracheal stenosis, with the aim of investigating their perioperative airway managements, it did not specify the exact location of the hemangioma and the number of patients with tracheal hemangioma was not reported, hence the study was excluded from this reviewTo summarize, a total of 19 children were reported to have tracheal hemangiomas or other subglottal lesions protruding to the trachea. The country distribution of the cases were as follows: USA 42.1% (8/19), Canada 15.7% (3/19), Turkey 10.5% (2/19), Tunisia 10.5% (2/19), Japan 5.2% (1/19), China 5.2% (1/19), and Korea 5.2% (1/19) . Four studies (eight patients) did not mention the age of the patients and the average age in other studies was 55.4 months (from 6 weeks to 15 years) with 54.54% of the cases being 1 year old or younger. There was a slight prevalence of the disease in female with the male to female ration being 4:7. The reported comorbidities and past medical histories included prematurity 5.2% (1/19), hyperbilirubinemia 5.2% (1/19), laryngomalacia 5.2% (1/19), respiratory distress early in life 15.7% (3/19), PHACE syndrome 36.8% (7/19), and a history of adenotonsillectomy 5.2% (1/19). It should be noted that one case suffered from severe prematurity and respiratory distress syndrome at birth, hyperbilirubinemia, and laryngomalacia at the same time. Two studies did not mention the past medical history of the patients and six patients did not have any comorbidities or prior medical conditions.Among 13 patients whose signs and symptoms were recorded, the most common signs and symptoms were stridor 69.23% ( 9/13), respiratory distress 38.46% (5/13), and hemoptysis 23.07% (3/13). Other complaints consisted of dyspnea, chronic coughing, recurrent respiratory infections, and sporadic fascial and cervical hemangiomas. The diagnostic tools used in all studies were mainly a CT scan, an MRI, bronchoscopy, and histopathologic assessments for confirmation. One study confirmed the accuracy of the 3d-CT/bronchoscopy diagnosis for the tumor.Only one case series did not specify the treatment option used for the patient with tracheal hemangioma. The main treatment option observed in the studies was the use of Propranolol 61.11 (11/18), which in 10 patients led to improvement of the signs, symptoms, and the size of the tumor. There was a failure of the propranolol regimen in one study, which led to electrical excision of the lesion. Laser ablation, steroids therapy, and surgical removal of the lesion were reported in 27.77% ( 5/18), 16.66 (3/18), and 16.66 (3/18), respectively. Natural shrinkage of the tumor was reported in one case.There was no report of a tracheal hemangioma from Iran in the past 10 years, therefore making this case report the only reported tracheal hemangioma in the past 10 years in Iran.The study searching was conducted by two separate individuals; hence, improving the accuracy of the search results. All included studies were obtained in full-text form and were in English language; hence, no exclusion was needed in this part.The limitations of this literature review include a publication bias as all studies are case reports and series. The results of this study depend on the quality of the literature search.Tracheal hemangiomas are a rare concept, especially in children. These tumors present with stridor, progressive respiratory distress, dyspnea, hemoptysis, and chronic coughs. Propranolol has been reported to have great efficacy in improving the symptoms and reducing the size of the lesion. Systemic and localized steroids, surgical removal, and laser ablation are other successful methods for treatment. It is not common for a tracheal hemangioma presenting with severe symptoms to shrink naturally. Although there is not sufficient literature, based on this literature review, recurrence is not a common finding after the mentioned treatments as none of the cases reported showed recurrence. A close follow-up ranging from 3 months to 1 year is recommended.NA.No personal information of patients have been revealed in the study.None.None.R.R., H.S.: study concept or design; H.S., P.B.T., H.A.: data collection; R.R.: supervision; H.S., P.B.T., H.A.: data analysis or interpretation; P.B.T.R., R.R., H.A.: writing the paper.None.None.Reza Rezaei.All the required data are available in the manuscript.Not commissioned, externally peer reviewed. Tracheal hemangioma: a case report and literature review.None.


Multifocal epithelial hyperplasia (MEH), or focal epithelial hypertension (FEH), or Heck’s disease, is an uncommon, benign oral mucosal disease associated with human papillomavirus infection. It is mostly observed in indigenous populations of the world, and has been rarely reported in China. However, previous research suggested there might be a greater prevalence of MEH in the Chinese population. While predominantly involves the lips, buccal mucosa and tongue, MEH was occasionally reported to affect the hard palate and gingiva as well.This paper reports a case of extensive MEH lesions that confined to the interdental papilla of a Chinese male without detection of HPV, and summarizes the published gingiva-involved MEH reports from 1966 until present. The lesions were excised with an Er: YAG laser after scaling and root planning, no recurrence was observed after 6-month follow-up.The present report illustrates the need for clinicians to be aware of rare presentations of MEH to facilitate a prompt diagnosis and proper management. More reports are encouraged to determine a correct prevalence rate of MEH in China.The online version contains supplementary material available at 10.1186/s12903-023-03282-7. Multifocal epithelial hyperplasia (MEH), also known as Heck’s disease, is a rare benign disease that was first described in English literature by Archard et al. 1965) [965 1, 22. Previoth, 2020. His chief complaint was the isolated slow-growing exophytic growth on the anterior gums for over 10 years, and he reported lightening of the lesions after supragingival scaling the previous week. The patient denied significant medical or family history, although he mentioned a history of ablative therapy for a single, non-recurring genital wart. Unfortunately, relevant medical records or pathological evidence were not available to confirm the diagnosis.A 37-year-old married male with a history of smoking 3-5 beedis/day for the 20 years was referred to the Stomatology Hospital, Zhejiang University School of Medicine, on November 30Clinical examination revealed multiple demarcated, exophytic, pedunculated, soft, pale papules on the labial and lingual interdental papilla areas of the anterior and molar teeth. The lesions ranged in size from 1 to 5 mm and were coalescent. There was also significant accumulation of plaque and subgingival calculus  analysis using consensus primers GP5 + /6 + and MY09/11, and type-specific primers for HPV16, 18, 13, and 32 (details of methods are submitted in http://blast.ncbi.nlm.nih.gov) revealed the sequences were human genes and so no HPV was detected in this case.Histological analysis showed hyperplastic squamous epithelium with papillomatosis, acanthosis, hyperkeratosis, and parakeratosis. Elongated rete ridges anastomosed horizontally and koilocytosis were noted within the upper layers of the epithelium with a perinuclear halo, with rare mitosoid bodies Fig. . The tisClinical and histopathological features were consistent with a diagnosis of MEH. Scaling and root planning (SRP) was prescribed along with oral hygiene instructions . TopicalMEH is a rare benign proliferative disease of the oral mucosa associated with HPV infection. Two clinical forms have been described in the literature: papulonodular and papillomatous. The papulonodular variant occurred mainly on the lining mucosa and is more common than the papillomatous type, which occurred on the masticatory mucosae such as the attached gingiva and the tongue , 7. MEH The MEH lesions tend to occur on the buccal and labial mucosa, it rarely affects the gingiva or the hard palate. Sethi et al. shortlisted 95 published MEH cases from 1966 to 2020, and found 8 out of 95 total cases reported gingiva lesions . SeventeThere is little documentation on MEH in Chinese population. Two brief MEH case reports were retrieved in Chinese literature database: Chen et al. reported MEH on the buccal mucosa, labial mucosa, palate and attached gingiva of a 31-year-old male in 1986, attached with histopathological pictures . In 2006MEH is strongly associated with HPV infection. Whilst HPV 13 and 32 are the most frequently detected subtypes, co-infection or infection with other HPV-types like HPV 6, 11, 16, 18, 31, 39, 40, 51, 52, 55, 58, 66, 68, 69, 71, 74 and 90 are also occasionally described , 24–26. Along with HPV etiology, low socioeconomic status, crowded living conditions, malnutrition, tobacco exposure, immunosuppression, and poor hygiene are also associated with MEH , 6. A prConsidered as a benign disease, MEH does not require treatment unless the lesions cause functional or cosmetic concerns . In thisGingiva-involved MEH has been uncommonly reported, particularly when it has widespread lesions confined to the gingiva . This maMEH is reported to be the most frequently diagnosed HPV-related oral lesions in healthy pediatric patients . AlthougThis case highlights a rare presentation of MEH confined to the interdental gingiva in a Chinese male. More awareness about MEH is encouraged in population and in clinicians, especially the Chinese clinicians. Future research is required to gain further understanding of the pathogenesis of MEH, its association with viral infection, oral hygiene, age and smoking.Additional file 1.


Unilateral corectopia is an exceedingly rare congenital defect where the pupil is displaced from its central position. Usually, it presents with normal visual aperture or associated with other diseases. We describe the first reported case of a left ectopic pupil in a healthy four-year-old boy with normal lens structure and total lack of visual aperture. Reporting such instances contributes to the understanding of this condition and guide future research endeavors. Further studies are needed to reveal the underlying pathophysiology, refine treatment approaches, and assess long-term outcomes. Congenital defects of the pupil are uncommon. Corectopia (ectopic pupil) is a congenital pupil abnormality characterized by bilateral pupil displacement from their central position ,2. GenerA four-year-old boy presented to our clinic with a complaint of left eye deviation persisting for six months. There was no history of trauma, and the child had no other relevant medical conditions. Family history was irrelevant.On examination, he exhibited 20/20 visual acuity in the right eye, while the left eye only demonstrated the ability to perceive hand movements. The patient was able to track light in all directions of gaze. A slit lamp examination revealed the absence of a pupil in the left eye Figure . Also, FUnder laryngeal mask anesthesia, the pupil was observed to be located at the 6 o'clock position with no visible aperture. Ultrasonography (A and B scan) demonstrated a normal globe structure as shown in Figures The parents were informed about the potential of a poor visual outcome associated with any surgical intervention, particularly pupiloplasty, due to the anticipated development of sensory-deprived amblyopia. However, the patient did not follow up thereafter.Early reports of unilateral isolated ectopic pupil are inadequate. To our knowledge, only seven patients were reported to have isolated unilateral tractional corectopia since 1930 ,5,6. ThiWhile the exact prevalence remains uncertain due to the scarcity of cases, its uniqueness and clinical implications cannot be understated. Some have suggested genetic defects leading to abnormal membranes formation arranged in radiating folds dragging the pupil, remnants of anterior tunica vasculosa lentis membrane, and the potential of fetal iritis ,4,6,7. TFew cases were reported to be associated with syndromes like ELeP, persistent pupillary membrane, Axenfeld-Reiger anomaly, Rieger syndrome, or Myhre syndrome ,3,4,8-10Limited reports suggested satisfactory results following restricted interventions. Atkinson et al. described three patients with corectopia. The treatment plan included an Nd:YAG laser for one baby, and incisional surgery for another one. The third was treated with medical mydriasis. All children have had favorable visual outcomes . GrienerIn the light of a poor visual outcome associated with any surgical intervention, particularly pupiloplasty, due to the anticipated development of sensory-deprived amblyopia, we assume that performing surgical intervention to this case will not be determinantal.Although unilateral corectopia is a rare occurrence, its clinical significance should not be ignored. This case highlights the importance of reporting such instances to contribute to the understanding of this condition and guide future research endeavors. Further studies are needed to reveal the underlying pathophysiology, refine treatment approaches, and assess long-term outcomes.To conclude, while the clinical picture and the genetic features of ELeP and bilateral corectopia are recognized in the literature, isolated unilateral corectopia has received minute concern. Restricted papers proposed reasonable results with Nd:YAG laser. However, we hypothesise that the surgical methods might be helpful if a radiating membrane or the visual aperture were noted. Despite concerns about surgical interventions and sensory-deprived amblyopia, the need to contribute to the knowledge pool, refine treatments, uncover pathophysiology, and assess outcomes remains pivotal. This report underscores the significance of investigating isolated unilateral corectopia, supplementing existing literature on more recognized pupil abnormalities, and guiding future research directions.


The incidence of bilateral adrenal hemorrhage (BAH) in the postoperative setting is rare, but potentially life threatening. A literature review of postoperative BAH reveals that there is limited data on BAH following abdominal surgery. We present a case of BAH following pancreaticoduodenectomy, which has not been previously documented in the literature. A 70-year-old male patient with no previous history of adrenal disease underwent an uncomplicated pancreaticoduodenectomy and was discharged after a typical postoperative course. He was readmitted with abdominal pain and ileus on POD 8 and a computed tomography (CT) scan was initially unremarkable, but a repeat CT scan on POD 11 demonstrated BAH. He was found to have adrenal insufficiency and was successfully treated with steroids. Clinicians should be aware of the possibility of adrenal hemorrhage postoperatively as it can potentially be a fatal surgical complication. To enhance patient outcomes, early detection and appropriate treatment are essential. Bilateral adrenal hemorrhage (BAH) is an uncommon but serious condition when associated with adrenal insufficiency (AI). Reported precipitating conditions include neoplasm, coagulopathy and sepsis , 2. EvenThough uncommon, BAH can lead to rapid decompensation . HistoriWe present the first reported case of post-Whipple BAH, which manifested with abdominal pain and electrolyte abnormalities.A 70-year-old male nonsmoker with strong family history of pancreatic cancer and personal history of idiopathic pancreatitis was found to have a 17 mm pancreatic head mass with pancreatic ductal dilation on screening endosonography (EUS). Fine needle aspiration returned highly atypical cells; malignancy could not be excluded.He had no prior abdominal operations. He denied alcohol use, jaundice or symptoms of pancreatitis.CA 19-9 and carcinoembryonic antigen (CEA) were normal. Computed tomography (CT) scan findings correlated with EUS. Of note, both adrenal glands were radiographically normal.He was offered open pancreatoduodenectomy for diagnosis and potential cure.A prophylactic dose of heparin was administered on the morning of surgery. The operation was completed in a standard fashion without complication and drains were placed abutting the pancreatojejunostomy and hepaticojejunostomy. Estimated blood loss was 200 mL and total time was 374 min.Pathology revealed intraductal mucinous papillary neoplasm with high grade dysplasia and negative margins. All 14 lymph nodes were normal (pTisN0).His immediate postoperative course was routine. He had no evidence of biochemical pancreatic leak and drains were removed. He was discharged on POD 7 at which time he was afebrile, stable, tolerating diet, with pain well controlled.On POD 8, he presented with severe abdominal pain and CT demonstrated an ileus without evidence of postoperative complication. He was readmitted and gradually improved over the next 3 days but continued to experience intermittent abdominal pain and inconsistent bowel function. Therefore, on POD 11 a CT was repeated, demonstrating a new high density 2.6 × 2.6 cm left adrenal collection consistent with subacute hemorrhage . The rigAlthough normotensive, he had significant electrolyte abnormalities including hyponatremia (128 mEq/L) and hypochloremia (96 mEq/L). Serum potassium and magnesium were normal. A corticotropin stimulation test (CST) revealed no elevation in cortisol after 30 and 60 min with an elevated adrenocorticotropic hormone (ACTH) twice the upper limit of normal, consistent with AI.He received hydrocortisone 25 mg IV and the following day he was transitioned to a hydrocortisone 20 mg per oral (PO) morning dose and 10 mg PO evening dose. He remained normotensive and bowel function normalized. At his 2-week post-operative visit, his abdominal pain had resolved. Repeat CST after 6 weeks again failed to stimulate cortisol. He was continued on hydrocortisone at a reduced dose and care was established with an endocrinologist.Postmortem studies have reported the incidence of AH to range from 0.14 to 1.8%, with higher prevalence in critically ill patients . EtiologThe pathophysiology of AH involves a prothrombotic state; adrenal vein thrombosis causes increased intra-gland pressure. Trauma and surgery may also cause vasoconstriction due to catecholamine release . CytokinPresentation of BAH is often nonspecific, making diagnosis difficult . PresentImmediate intervention should be initiated, including corticosteroid administration, fluid resuscitation and correction of electrolyte imbalances . HypotenBAH has previously been described following abdominal surgery , 8, 12. In the case of our patient, aside from abdominal surgery and age over 55, he had no risk factors for AH such as adrenal masses or hypercoagulability. Similarly to prior reports, he presented with abdominal pain, electrolyte abnormalities and was found to have BAH on CT; AI was subsequently confirmed with CST and treated with steroids.Postoperative BAH is a rare but serious occurrence with unclear mechanism. We present the first incident of BAH after pancreaticoduodenectomy at our institution and in published literature, which was diagnosed and treated expeditiously resulting in a good clinical outcome. This case highlights the need to suspect BAH in the setting of vague postoperative symptoms to facilitate hasty diagnosis and treatment given the potential mortality.The authors have no conflicts of interest to disclose.None.No publicly available datasets were used in the writing of this manuscript.Signed informed consent was obtained from the patient for the use of data and images in this manuscript. A copy may be provided upon request.


Intramuscular hemangiomas are benign lesion that rarely occurs within the skeletal muscle of the head and neck regions. These lesions present with nonspecific symptoms resulting in only a few instances of accurate preoperative diagnosis.A 20-year-old male presented with swelling over the nape of the neck on the right side. On clinical examination, the solitary swelling was 4×4 cm on measurement, soft on consistency with regular margin, fluctuant, with no skin changes over the swelling, nontender, no restriction in range of motion of the neck, and no pulsation felt.Ultrasonography and contrast-enhanced MRI revealed intramuscular hemangioma involving the right splenius capitis muscle with no extension to adjacent muscles and minimal extension to the subcutaneous tissue.Excision of the lesion along with splenius capitis was performed with stable postoperative hemodynamics.Since intramuscular hemangiomas pose a challenge in preoperative diagnosis, it requires the sensible use of imaging modalities. Although several treatment modalities have surfaced, intramuscular hemangiomas require definitive operative management to reduce their recurrence. HighlightsIntramuscular hemangiomas are rare benign tumors that account for 1% of all hemangiomas and are even more rarer and uncommon in the head and neck region.Intramuscular hemangiomas have a variable presentation, and accurate preoperative diagnosis has been reported in less than 8% of cases because bruits, thrills, and pulsations are frequently masked due to muscular fibrosis concealing the vascular nature.The definitive diagnosis can be made through the histological study of surgical or biopsy specimens. However, MRI is the gold standard for preoperative diagnosis as it allows better delineation of the tumor.Many treatment modalities have been used for the management of such cases; however, definitive surgical management is required. Such tumors have a high recurrence rate, and the local recurrence rate can be maintained through wide excision, including the cuff of surrounding muscle.Hemangiomas, although being the most common benign tumor, arising within skeletal muscle account for less than 1% of all hemangiomas and occur in large musculature of the trunk and extremitiesHerein we report a rare case of intramuscular hemangioma of splenius capitis that was managed successfully with the excision of the lesion along with the muscle involved.We reported this case following the updated consensus-based Surgical CAse REport (SCARE) guidelinesA 20-year-old male army personnel belonging to the Kirati ethnicity with no known comorbidities presented to the neurosurgery outpatient department of our hospital (a tertiary care facility) with the chief complaint of swelling over the nape of the neck on the right side. According to the patient, the swelling first presented at 20 days of his life, which was the size of a pea that gradually increased to a size of a tennis ball spontaneously. There was no history of trauma. His past history, family history, drug history, and allergic history are nonremarkable. He is a nonsmoker, does not consume alcohol, and has normal sleep and bowel habit. On clinical examination, the solitary swelling was 4×4 cm on measurement, soft on consistency with regular margin, fluctuant, with no skin changes over the swelling, nontender, no restriction in range of motion of the neck, and no pulsation felt. His vital parameters were normal, with no significant findings in systemic examinations.Ultrasonography was ordered, which showed an ill-defined anechoic cystic lesion with thick echogenic internal septation and strands in the subcutaneous plane. A contrast-enhanced MRI revealed fat saturated heterogeneously high signal intensity intramuscular lesion suggesting intramuscular hemangioma involving the right splenius capitis muscle with no extension to adjacent muscles and minimal extension to the subcutaneous tissue Figs. , 2.All of his laboratory parameters were within the normal range. He was then scheduled for surgery. The patient was informed about the risks and benefits of the procedure, along with an explanation of other alternative procedures. He wanted the lesion to be surgically removed. Thus, he underwent excision of the lesion along with the excision of splenius capitis with minimal blood loss. The specimen Fig.  was sentIntramuscular hemangiomas are noticed around adulthood once visible symptoms occur like swelling, pain, or cosmetic asymmetry, although they occur predominantly in childhoodSince intramuscular hemangiomas are rare, they possess considerable therapeutic challenges regarding their locationsThe definitive diagnosis can be made through a histological examination of the specimenSpontaneous regression of the tumor does not occurIntramuscular hemangiomas, although benign and have not been reported to metastasize, have a higher recurrence rate of about 9–28% of the cases due to the microscopically infiltrative pattern of diffusion into surrounding muscular planes of least resistance, thus requiring long-term clinical and radiological follow-upsIntramuscular hemangiomas usually appear as an enlarging mass without specific clinical features. While a superficial mucosal lesion can be diagnosed on clinical grounds, deep lesions require imaging modalities. This case of intramuscular hemangioma is reported to show the presentation of such a lesion, which might help surgeons be aware of the possibility of such a lesion. Hence, clinicians should have a high degree of clinical suspicion and make judicious use of imaging modalities for the diagnosis of the lesion.Such a lesion requires definitive operative management and regular follow-ups.None.Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy is available for review by the Editor-in-Chief of this journal on request.None.There are no conflicts of interest.Not applicable.Not commissioned, externally peer-reviewed.None.


An uncommon and aggressive type of soft tissue sarcoma that develops from peripheral nerves is known as a malignant peripheral nerve sheath tumor (MPNST). It is typically associated with neurofibromatosis type 1. Hence, this case report presents a case of a 42-year-old woman with a mass over the palmar aspect of her right hand that had been slowly growing over the previous year. Complete resection of the tumor with groin flap was performed followed by adjuvant radiotherapy. Over the last year, the patient has been monitored on an outpatient basis without displaying any signs of a local recurrence. Schwann cell-derived malignant peripheral nerve sheath tumors (MPNST) typically affect peripheral nerves or the nerve sheath. They are extremely rare to arise over the palm of the hand and usually have a gradual progression followed by a sudden rapid increase in growth . These tA 42-year-old female reported with complaints of right palm edema that had been present for a year. The swelling progressively increased in size over the year and was associated with pain for the last couple of months. History of excision attempted three months back was present. Local examination revealed a globular, firm, non-tender, non-compressable, non-mobile swelling over the thenar eminence of the right palm. The overlying skin was however normal Figure . Ultrasound of the subcutaneous plane of the palmar aspect on the right hand showed evidence of a well-defined large multilobulated lesion of size approximately 7.0 x 7.1 x 3.4 cm heterogeneously hypoechoic in the echotexture with high vascularity on color Doppler study. The Magnetic Resonance Imaging (MRI) showed a well-defined multilobulated lesion 7.3 cm x 7.1 cm x 3.5 cm in size over the right palm. On T1-weighted imaging Figure , it was In the hemorrhagic background, fine needle aspiration cytology (FNAC) revealed minimal cellularity, some neural cells, and scattered cells with modest pleomorphism. Complete surgical excision of the mass Figure  with groHowever, sections from the medial, lateral, superior, and inferior borders were free from infiltration by cancerous cells. The histopathological analysis of the excised specimen revealed a 40x image showing monomorphic spindle cells arranged in interlacing fascicles, palisades, and whorls Figure  which chMPNST are extremely rare nervous system tumors that arise from fibroblast or Schwann cells of peripheral nerve sheaths ,7. MPNSTMost cases are clinically present as rapidly enlarging soft-tissue masses with neuropathic symptoms such as paresthesias, radicular pain, or motor deficits. It is difficult to differentiate MPNST from benign tumors as there is both radiological and histological resemblance. According to Demir et al. , the cliDue to their histologic similarity, spindle cell sarcomas must be distinguished from one another using immunologic markers and sufficient pathologic samples . The marIt is quite challenging to identify and manage soft tissue sarcomas occurring over uncommon parts of the body. This case enhances understanding on how to approach and treat similar cases in the future. It also justifies that aggressive management in such cases improves both functional and survival outcome of the patient.


Giant hydronephrosis is mostly caused by ureteropelvic junction obstruction (UPJO). Giant hydronephrosis with concurrent malignancy is less common clinically and is easily misdiagnosed. We report a 77-year-old male who presented with sudden onset progressive abdominal distension in the last month. Abdominal computed tomography showed a right severe hydronephrotic kidney with loss of parenchymal thinning. Cystoscopy showed a 1x1cm papillary lesion protruding from the right ureteric orifice. He underwent a right radical nephroureterectomy with bladder cuff excision with lymph node dissection. Histopathology showed low-grade urothelial carcinoma of the ureter and incidental pleomorphic rhabdomyosarcoma in the right kidney. The patient refused chemotherapy and died 6 months later due to lung metastasis. Incidental pathologic finding of renal rhabdomyosarcoma in adults with giant hydronephrosis and urothelial carcinoma is a rare occurrence with diverse clinical presentations, prognoses, and outcomes. Giant hydronephrosis, referred to as a hydronephrotic kidney with a content volume exceeding 1 liter, is an uncommon entity . Giant hPatient’s information: a 77-year-old male presented with a complaint of breathlessness, which was more pronounced on lying down. It was associated with rapidly progressive abdominal distention from the last 1 month. The patient was a chronic smoker with 25 pack years of smoking. He was hypertensive and on regular antihypertensives for the last 8 years. He had a past history of transurethral resection of a bladder tumor three years back with histology of a superficial low-grade urothelial tumor without further treatment or follow-up. There was no significant family history.Clinical findings: on examination, the patient had a tense distended abdomen with a large abdominal mass, which was occupying the entire right hemiabdomen. The mass was non-tender and cystic in consistency with well-defined borders  scan showed a right grossly hydronephrotic kidney of size 23x20x18 cm with parenchymal thinning and containing around 3.3 liters of fluid  was positive for Desmin. The histopathology of the ureter specimen showed non-invasive, low-grade papillary urothelial carcinoma (arcinoma . Lymph nPatient’s perspective: during treatment, the patient and his family were satisfied with the level of care provided to him.Informed consent: written informed consent was obtained from the patient’s family.et al. and Aljbri et al. reported cases of giant hydronephrosis having 13 litters and 12 litters of urine in the pelvicalyceal system, respectively [The giant hydronephrosis case was first published in 1746, and there are a little over 500 cases that have been published with a wide spectrum of underlying causes. UPJO has been recognized as the most common cause of giant hydronephrosis. Whereas, tumor pathology of the urinary tract is uncommon [ectively .Ultrasonography may identify giant hydronephrosis as the presence of hydronephrosis which extends beyond the abdominal midline or extends to five or six vertebral . Computeet al. [The ideal treatment for giant hydronephrosis is usually a simple nephrectomy, because of the frequent association of foci of dysplasia, tumor changes in the parenchyma, and collecting system as a result of chronic irritation . In our et al. .Incidental pathologic finding of renal rhabdomyosarcoma in adults with giant hydronephrosis and urothelial carcinoma is a rare occurrence with diverse clinical presentations, prognoses, and outcomes.


Colonic lipomas are usually incidental findings during colonoscopy. Lipomas ≥ 4 cm can cause obstruction and intussusception. Surgical resection is often recommended in these instancesA 63-year-old man with a known large colonic lipoma causing recurrent intussusception presented with abdominal pain. He had previously declined surgery. Computed tomography scan of the abdomen demonstrated an 8-cm fat-containing mass in the ascending colon. Colonoscopy showed a large trilobed mass in the ascending colon with areas of ulceration, occluding the entire lumen of the colon . The paA colonoscopy was performed, and attempts to place a PolyLoop  around the mass were unsuccessful due to the size and smoothness of the mass. Therefore, the decision was made to use an endoscopic submucosal dissection knife to perform a mucosal incision on the surface of the lesion to expose the underlying fatty tissue . The liRepeat colonoscopy was performed after 8 weeks, and the lesion was significantly smaller. Debulking was performed again using the same technique. As the size of the lesion was significantly reduced compared with the original lipoma, two PolyLoops could be successfully placed over the lesion, allowing sloughing of the remainder of the lesion.Further colonoscopy after 6 months showed significant improvement in luminal narrowing . The paVideo 1 Endoscopic debulking of a large colonic lipoma causing recurrent intussusception using endoscopic mucosotomy technique.Endoscopy_UCTN_Code_TTT_1AQ_2AD


Pancreatic divisum (PD) is a malformation wherein the majority of affected patients remain asymptomatic or present with complications early in life. Some cases, however, may present in adulthood with recurrent pancreatitis, which makes the diagnosis clinically challenging. Here, we present a rare case of an elderly female with acute-on-chronic epigastric pain secondary to pancreatitis due to PD. During hospitalization, the patient was treated for acute pancreatitis and subsequently discharged with recommendations for corrective surgery. This case is unique particularly due to the older age of onset of symptoms, as well as the lack of exacerbating factors such as drug abuse, alcohol, or obesity. This case highlights the importance of considering PD as a differential diagnosis when managing patients with recurrent pancreatitis regardless of their age. Pancreatic divisum (PD) is a congenital malformation of the pancreas occurring in less than 10% of the general population, in which the ventral and dorsal pancreatic bodies fail to fuse during early gut formation . While o2. Physical examination revealed a soft, nondistended abdomen, with global tenderness on palpation, and normoactive bowel sounds. Her laboratory results showed a normal calcium level, and serum IgG4 was negative; hence, autoimmune pancreatitis was ruled out. Amylase and lipase levels  were elevated, and a lipid panel was within normal limits. The hematologic profile was within normal limits. Noncontrast computed tomography (CT) of the abdomen revealed acute pancreatitis with a small amount of fluid layering anterior to the left anterior pararenal space, Balthazar score D (image not shown). This was the patient’s third episode of pancreatitis, and as such further investigations were ordered. MRCP done before admission revealed the “crossing-duct” sign, wherein the dorsal pancreatic duct was seen crossing the common bile duct to drain the minor papilla. Consequently, a diagnosis of type 1 PD was made (Figure A 74-year-old female with a past medical history of chronic pancreatitis, type 2 diabetes mellitus, and gastroesophageal reflux disease presented to the emergency department with upper abdominal pain. The pain was acute in onset; described as constant, dull-aching, non-radiating in nature; 10/10 in intensity; exacerbated by changes in position and with deep palpation; and associated with nausea and vomiting. There were no relieving factors. The patient denied consuming alcohol or tobacco products and reported that she had experienced similar episodes in the past. Her family history was positive for liver cancer in her mother. Vital signs on admission were as follows: blood pressure was 147/78 mmHg, heart rate was 59 beats/minute, temperature was 98°F, and oxygen saturation was 98% on room air. Her body mass index was 30.1 kg/mDuring the course of hospitalization, the patient was started on hydration therapy, ondansetron 4 mg orally once a day for nausea, and hydromorphone 0.5 mg IV when needed for pain management. The patient’s symptoms subsequently improved, her pain subsided, and lipase levels trended down to 428 U/L. She was then discharged from the hospital. She was advised not to consume more than 20 g of fat a day, undergo surgical correction per gastrointestinal recommendations, and follow up monthly for six months.PD is a common congenital pancreatic anomaly with an overall prevalence of about 6% to 10% . AlthougThe symptoms in patients with PD range from asymptomatic to recurrent attacks of mild or severe acute pancreatitis. In symptomatic patients, diagnosis can be confirmed by a secretin-enhanced MRCP, which accurately demonstrates the anomaly ,7. In ouFor mild symptoms, conservative management is usually sufficient . For patThis case highlights the importance of considering even rare etiologies such as PD in unexplained cases of recurrent pancreatitis. Clinicians should be encouraged not to overlook congenital anomalies as a possible etiology solely based on the age of presenting symptoms. Early confirmation of the underlying etiology and aggravating factors can improve patient outcomes.


Retained surgical item is a known incidental surgical complication that is a mass of a sponge. This cotton matrix is left behind in the body cavity after surgical procedures. It is an incidental, rare medical error.Here, the authors present a case of a 30-year-old woman who had undergone a cesarean section 2 months ago and presented with the cardinal symptoms of small bowel obstruction. A computerized abdominal tomography (CT) scan showed a well-defined tubular hyperdense structure adherent to the anterior abdominal wall that exerts a mass effect on the adjacent small bowel loops. Following the computerized abdominal tomography results, an exploratory laparotomy resection and anastomosis of a small segment of the ileum were performed. The postoperative period was uneventful, and the patient has remained disease-free to date.Because it is not anticipated, and its clinical manifestations are variable, it is frequently misdiagnosed, and often unnecessary radical surgical procedures are performed.It should be considered in the differential diagnosis of any postoperative case with an unresolved or unusual presentation. HighlightsDiagnosing a gossypiboma can be challenging, as symptoms may not appear until weeks or even years after the surgery.Medical errors, such as miscounting surgical sponges, poor documentation, or inadequate communication among surgical team members.These retained sponges can cause a variety of early and late complications.1. Since then, numerous cases of gossypiboma have been reported in medical literature, and the condition has become recognized as a rare but potentially serious complication of surgical procedures. The work has been reported in line with the Surgical CAse REport (SCARE) 2020 criteria2.The first reported case of gossypiboma was documented in 1884 by a British surgeon named Wilson. Wilson reported a case of a patient who underwent surgery for a fibroid tumor and later developed symptoms of abdominal pain and fever, only to discover that a surgical sponge had been left inside the patient’s body during the initial surgery9/l, otherwise unremarkable results. For imaging, the abdominal radiograph showed dilated small bowel loops  2 months back in the other hospital. The patient presented to the emergency department complaining of continuous abdominal pain in the umbilical region for 2 days. The pain was associated with nausea, vomiting, and constipation and started about 2 months ago. Family, drug, and psychological history were unremarkable. Upon examination, the patient was lying on the bed in severe pain, and she was alert, conscious, and oriented. Her heart rate was 120/min, blood pressure was 113/74 mmHg, respiratory rate was 20 breaths/min, and temperature was 36.9°C. Upon inspection of the abdomen, a Pfannenstiel incision scar and abdominal distension were noted; upon palpation, the abdomen was soft and lax, along with periumbilical swelling. Laboratory workups revealed leucocytosis of 16.25×103. It’s considered the most common retained surgical item (RSI), RSI is a never event, meaning that it should never happen4. It is preventable, and by applying proactive measures based on causation only, we can avoid causing harm to the patients. This dilemma has been described by several terms in medical literature, such as retained surgical sponges, textilomas, or cottonoid syndrome. Studies suggested that the incidence of Gossypiboma following abdominal surgery is estimated to be between 1 and 1000 to 1 and 1500 procedures. However, the actual number of cases may be higher due to underreporting, possibly due to associated medicolegal issues. RSI, including gossypibomas, were found to be the third most common cause of malpractice in a retrospective study of surgical malpractice conducted in the United States. The reported incidence in the abdominal cavity (56%), pelvis (18%), and thorax (11%). Its worth to highlighted there are several risk factors; the most significant risk factors were any emergency procedures, unplanned changes in the surgery, the patient’s high BMI, and lack of supervision by a senior surgeon6. In the current case, we are reporting a case of partial small bowel obstruction secondary to intrabdominal Gossypiboma at the anterior abdominal wall, which was anterolateral to the umbilical region that exerted a mass effect that had presented 2 months following a cesarean section as for categorizing the operation, a cesarean section is generally considered an abdominal procedure, since it involves incisions through the abdominal wall and peritoneum. Some might also call it a pelviabdominal procedure involving both the abdominal and pelvic regions. However, it would not be accurate to call it a ‘pure’ pelvic operation, as the surgery directly involves the abdominal cavity. We need to highlight that In a typical cesarean section, the peritoneum is indeed opened. This is necessary to access the uterus and deliver the baby. However, surgeons take care to minimize adhesions (scar tissue) that can form as a result of surgery, which could potentially lead to adhesion and can be got adherent to small bowels subsequently, lead to adhesion and this gotten adherent to the RSI  that is unintentionally left behind at the end of the surgery. Clinically, it is challenging to be diagnosed and most of the patients present with classical cardinal symptoms and signs of any bowel obstruction or an RSI-associated complication : NA.Mohammed Alsuhaimi.Not applicable.Not commissioned, externally peer-reviewed.


A 75-year-old man was admitted to our hospital for isolated serum γ-glutamyl transpeptidase  elevation, with fluctuation in the past 2 years . His alVideo 1 Endoscopic ultrasound showed a hypoechoic nodule in the common bile duct. Contrast-enhanced endoscopic ultrasound showed the enhancement of the lesion, but the degree was weaker than the peripheral pancreas.The patient was delivered for surgery, and intraoperative choledochoscopy indicated a villous tumor located in the terminal portion of the CBD with a soft texture. Finally, histological diagnosis indicated adenocarcinoma  . The cuEndoscopy_UCTN_Code_CCL_1AZ_2AC