Infantile hemangioma (IH) is important to all dentists, particularly dentists providing services to pediatric populations, because significant morbidity and mortality can occur from vascular lesions in children. Specialists of the oral cavity have the responsibility to identify patients with IH, a lesion that could be potentially life threatening. We present a case of IH and will provide a narrative review of the most recent literature. We discuss the diagnosis, risk stratification, treatment, complications, and impact on routine dental procedures. A proper diagnosis is crucial as oral and perioral IH are considered high-risk due to the increased risk of ulceration and feeding impairment. Referral to a hemangioma specialist for comprehensive team treatment is optimal. The natural history of IH consists of a long proliferative phase, which manifests as clinically visible growth. As a result of early encounters with patients, the pediatric dentist may often be considered the primary care provider. Infantile hemangioma (IH) is important to all dentists, particularly pediatric dentists, because injury can occur from this type of vascular lesion in the oral region, even during trivial trauma such as feeding. Bleeding caused by iatrogenic or minor manipulations during treatment or examination could cause bleeding and possibly morbidity or even mortality. Recognition of vascular tumors such as IH is crucial because they are the most common tumor of infancy . While IThe differentiation of IH from a vascular malformation is crucial for accurate diagnosis and treatment. A vascular malformation is present at birth and grows in proportion to the infant. In contrast, IH is absent at birth but may appear and proliferate rapidly for months . IH appeIH is a vascular tumor, which are benign neoplasms that are a result of proliferating endothelial cells. Local complications may include problems such as bleeding, tissue destruction, and pain. Systemic sequelae include thrombocytopenia, congestive heart failure (CHF), and death. Infantile hemangiomas are the most common type of benign vascular tumor, making up approximately 5–10% of all vascular tumors . The lesThe focus of this paper is to provide the reader with a case report for illustration of the condition and its management. Diagnosis and collaboration allow for successful management of patients with infantile hemangiomas by the dentist. At an older sibling’s appointment, the patient’s mother communicated a concern about her infant, and she had the baby present with her at the time. The parent’s chief concern was, “She has a red thing that has been growing in her mouth”. The mother made an appointment for the review of the lesion, and the 5-month-old female presented to the pediatric dentist for evaluation. Her medical history was unremarkable, and she was born at full term with no complications. The lesion was not noted at birth.The mother stated that the patient was not displaying any abnormal signs or symptoms for a child of that age. She fed regularly and had normal sleep patterns. She did not show excessive irritability. There was no history of excessive vomiting, diarrhea, fever, or cough. The mother stated she noticed the lesion was growing, and she did not remember if there had been a diagnosis, although she had inquired of the child’s pediatrician. She was concerned about the location being in the mouth and what the repercussions would be. The intra-oral clinical exam showed  a 2.5 cmThe differential diagnosis included hemangioma, vascular malformation, or telangiectasia lesions, and a consultation with an oral and maxillofacial pathologist was conducted. The patient was placed on a monthly recall for observational appointments with a provisional diagnosis of IH. They were also referred to dermatology and ENT for further evaluation. The patient, since their initial appointment with the pediatric dentist, had visited the ENT and the dermatologist. Both of those specialists agreed that the lesion was an infantile hemangioma and are following the patient. IH is the most common vascular tumor of infancy. A distinguishing characteristic is its growth cycle, usually obtained from the patient’s history. The lesions typically appear during the neonatal period, two weeks after delivery . The firOur patient’s history, as described by her mother, is consistent with the initiation and proliferation phases of IH. at our patient’s age of 5 months at initial observation, it was most likely at the end of the proliferation phase and entering the plateau. The proliferation phase could continue up until the first year of life. IH lesions are typically 0.05–5.0 cm in size and often affect the head and neck area . They caThe diagnosis of IH is based on the findings of the clinical exam and screening questions for history and duration. Most cases can be diagnosed based on growth history and appearance. Diascopy may be a useful screening tool, although caution must be used to avoid glass or plastic breakage that could induce bleeding . SometimHemangiomas are true neoplasms characterized by proliferation and increased rates of endothelial cell turnover. Infantile hemangiomas typically develop during the first 2 months of life and demonstrate rapid proliferation between 6 and 12 months of age . These cThe complications of IH can be life-threatening. Heart failure and respiratory distress have been associated . DependiWhen deciding how to manage patients who present with this lesion, treatment considerations may be divided into non-active and immediate. Non-active refers to watchful waiting if the IH is less than 2 cm and specifically located on the trunk or extremities. During watchful waiting, the patient is followed for sequential appointments for close monitoring . ChronolSpecialized hemangioma teams are the best resource for patients with oral or perioral lesions. Under the care of the team, the first line of treatment for ulcerated or proliferating IH is oral propranolol . A randoIn conclusion, although the lesions of IH are generally benign in nature, there still needs to be a proper diagnosis and understanding of the prognosis, clinical course, and treatment rationale. Smaller lesions less than 2 cm and not located on the face or in the oral cavity have much less risk associated with them. Lesions located in the oral cavity or that are larger should be referred to a specific hemangioma team for evaluation. The role of the dentist or other health care provider is to make an accurate identification of the lesion, provide guidance to the family, and provide proper referrals for expert evaluation such as an oral and maxillofacial pathologist, dermatologist, or ENT.


Muscular and subcutaneous candidiasis is a rare entity in immunocompromised patients, but it should be kept in mind when we see multiple cystic soft tissue masses in addition to target‐shaped hepatosplenic lesions in neutropenic patients. US and MRI are useful imaging modalities for the diagnosis and follow‐up of these patients.Soft tissue candidiasis is an opportunistic infection in immunocompromised patients and must always be diagnosed and treated as soon as possible. In this case report, the patient is a 14‐year‐old boy with acute myeloid leukemia M3‐type who presented with numerous soft tissue and hepatosplenic candidal abscesses. Soft tissue candidiasis is a type of infection that occurs in individuals with compromised immune systems, and it is crucial to promptly diagnose and treat this condition. We report a 14‐year‐old boy with acute myeloid leukemia M3‐type who presented multiple candidal abscesses in the soft tissues and hepatosplenic regions.  However, it has considerable importance and prevalence in immunocompromised patients, such as transplant and cancer patients.The manifestations are usually categorized into mucocutaneous or invasive forms. Typical presentations of the mucocutaneous form can be seen as paronychia, intertrigo, thrush, vulvovaginitis, and esophageal candidiasis. Candidemia is the most easily recognized manifestation of invasive candidiasis, but it can involve virtually any anatomic site and cause widespread visceral dissemination.2The patient is a 14‐year‐old boy who has presented with signs of epistaxis and fatigue since 3 months ago. Laboratory data showed severe pancytopenia. Considering the risk of spontaneous hemorrhage, he was immediately transferred to the tertiary center for comprehensive care. Subsequent investigations through bone marrow biopsy and flow cytometry were consistent with the diagnosis of AML M3 type. The patient received appropriate therapy, first with daunorubicin and ATRA and later with Arsenic trioxide. He then presented to the hospital 3 months later with numerous bulging subcutaneous masses on his buttocks, thighs, calves, and plantar surface of his feet. Similar lesions, albeit fewer, were observed in the back and upper extremities.Candida Albicans . Manifestations of imaging depend on the stage of the disease, but the most frequent ultrasound pattern in the liver and spleen is several small hypoechoic lesions. Four dominant patterns of hepatosplenic involvement have been described. The earliest pattern comprises a peripheral hypoechoic zone that correlates with fibrosis, with a second hyperechoic zone consisting of inflammatory cells. The central hypoechoic nidus relates to necrotic fungal elements. This pattern is called the “wheel within a wheel.” The second pattern is called the “bull's eye” or target pattern, with a peripheral hypoechoic halo encircling a central echogenic core. The third and most common pattern is seen as multiple hypoechoic lesions that can be seen in conjunction with the other three patterns. The fourth pattern, manifesting as echogenic foci, is usually seen at the late stages of the disease and correlates microscopically with central fibrosis, calcifications, or both.MRI seems superior to computed tomography (CT) scan and ultrasonography in identifying hepatosplenic and musculoskeletal lesions associated with candidiasis. In a patient with acute hepatosplenic or soft tissue candidiasis, lesions on MRI are round, measured <1 cm in diameter, markedly hyperintense on T2‐weighted images, and show restriction on diffusion‐weighted imaging (DWI). At the chronic stage, especially with antifungal treatment, a hyposignal rim surrounding the primary lesions and a non‐enhancing center on contrast images are seen, consistent with the necrotic core seen on histologic examination. When the lesions are calcified, they appear hyperdense in CT scan and have low signal in MRI.There aren't many studies that provide imaging results for subcutaneous and intramuscular candidal abscesses. We discovered a few case reports in the literature that contained imaging data such as CT or MRI Table . When deChloromas or myeloid sarcomas comprise immature myeloid cells, most often leukemic blasts.Multifocal bacterial abscesses can occur in immunocompromised patients. Septic emboli can be primarily found in the lungs, especially in AML patients with port catheter.Extra‐pulmonary tuberculosis should always be considered as a differential diagnosis of multiple subcutaneous and hepatosplenic masses in an immunocompromised patient, even though it's a rare finding.Cysticercosis is a kind of endemic parasitic disease that is rare in our country. The most commonly affected tissues are the central nervous system and skeletal muscles. In ultrasonography, the scolex is seen inside the lesion, which may be calcified. When the lesions are cystic, they have similar characteristics to fluid on both CT and MRI, but when these lesions calcify, they appear as hyperdense foci parallel to muscle fibers on CT, giving a characteristic appearance called “rice‐grain” calcification.Soft tissue and skeletal muscle hydatid cysts are very rare and usually secondary. They can occur in the lower extremities, trunk, neck, or legs. The pectoralis major, sartorius, quadriceps, and gluteus muscles can be involved. It usually appears as a focal multi‐vesicular cystic lesion in the muscle(s) that can invade the adjacent bone.Soft tissue mycetomas due to maduramycosis or other fungal infections usually occur in the foot and endemic areas. Mycetoma is a kind of chronic soft tissue inflammation caused by fungi or actinomycetes. They appear as multiple, small, round T2 hyperintense lesions with central hypointense foci in MRI. Central hypointense foci are mycetoma grains in pathology called the “dot in a circle” sign which is specific for this entity.4In conclusion, we presented an AML M3 patient with a multifocal muscular and hepatosplenic abscess caused by Candida albicans, shown in ultrasound and MRI and proved by pathology. He was treated successfully with medical and surgical methods. Also, we reviewed the literature about the imaging manifestations of a few similar cases and finally discussed the imaging features of musculoskeletal candidiasis and its differential diagnosis.Amirreza Jahanshahi: Investigation; supervision; validation; visualization. Sanam Nami: Validation; visualization. Abbas Ali Hosein Pour Feizi: Investigation; validation. Samin Alihosseini: Visualization; writing – original draft; writing – review and editing. Mehran Jaberinezhad: Writing – original draft; writing – review and editing. Mirsaeed Abdollahi: Writing – original draft; writing – review and editing. Faezeh Rahimi: Writing – original draft. Masih Falahatian: Conceptualization; investigation; project administration; visualization; writing – review and editing.None.No author states to have any conflicts of interest.Written informed consent was obtained from the patient for publication of this case report and accompanying images.


Hyperparathyroidism in patients with multiple endocrine neoplasia 1 is attributed to the excessive secretion of parathyroid hormone (PTH) from multiple parathyroid glands. This can be successfully treated through complete resection of the parathyroid glands; however, subsequent surgery is often required because of the presence of supernumerary or ectopic parathyroid glands. Therefore, identifying the locations of all functional glands is crucial for precise resection. Here, we report a case of ectopic mediastinal parathyroid adenoma that was successfully resected using robot-assisted thoracoscopic surgery.99mTc-methoxyisobutylisonitrile scintigraphy indicated a strong accumulation of tracers, suggesting an ectopic lesion in the mediastinum. Persistent hyperparathyroidism after total parathyroidectomy via neck incision was attributed to an ectopic parathyroid tumor in the mediastinum. Thus, we decided to resect the tumor using robot-assisted thoracoscopic surgery to perform the procedure gently and carefully. During surgery, a mediastinal tumor was identified as it was detected radiographically. Because it did not invade the surrounding tissues, it could be completely resected without injuring the capsule. The patient was discharged without complications. Postoperatively, Ca and intact PTH levels decreased back to normal. The final pathological diagnosis confirmed that the mass was an ectopic mediastinal parathyroid adenoma.A 53-year-old woman underwent a total parathyroidectomy with autotransplantation for multiple endocrine neoplasia 1-associated primary hyperparathyroidism. The patient previously underwent laparoscopic distal pancreatectomy for a pancreatic neuroendocrine tumor. She also presented with a mediastinal tumor and nonfunctional pituitary adenoma that could be followed up. Blood tests before total parathyroidectomy showed high levels of intact PTH (183 pg/mL) and calcium ; however, postoperative blood tests still revealed high levels of intact PTH (103 pg/mL) and Ca (11.4 mg/dL). Computed tomography and magnetic resonance imaging revealed a 45-mm-sized mass in the right upper mediastinum as a well-defined solid and cystic lesion, whereas Minimally invasive surgical resection of a remnant ectopic lesion was successfully performed in a patient with multiple endocrine neoplasia 1 using robot-assisted thoracoscopic surgery. Hyperparathyroidism is characterized by an excessive secretion of parathyroid hormone (PTH) from the parathyroid glands, necessitating complete resection of the causative glands for successful treatment. This condition is also known to occur in patients with multiple endocrine neoplasia type 1 (MEN1) and often requires multiple subsequent surgeries because of the presence of supernumerary or ectopic parathyroid glands. Therefore, it is vital to identify the locations of all functional glands for successful resection.Minimally invasive thoracoscopic surgery is performed worldwide to treat various diseases  and incl99mTc-methoxyisobutylisonitrile (MIBI) scintigraphy showing increased uptake in the neck region  with four arms. A robotic endoscope was placed in the seventh intercostal space (ICS) on the middle axillary line, and three robotic operating trocars were placed in the fifth ICS for the right hand, eighth ICS for the left hand, and eighth ICS for the fourth arm. All robotic trocars were 8 mm ports. Fenestrated bipolar forceps, a monopolar spatula, and SynchroSeal  were used. A 12-mm assistant port was also placed in the eighth ICS on the anterior axillary line with carbon dioxide insufflation. The mediastinal tumor was located between the SVC and trachea. The ectopic adenoma was easily detected, and the surgical margin could be clearly identified. The operation time was 76 min, console time was 46 min, and amount of blood loss was minimal. The tumor morphology was consistent with preoperative radiological findings; therefore, it was completely resected using RATS without injuring the capsule. Blood tests performed 1 day postsurgery showed a decrease in calcium level (8.3 mg/dL), and the patient was discharged on the 4th day without postoperative complications. Two weeks after the surgery, the serum intact PTH level decreased to 25.6 pg/mL. The final pathological diagnosis was ectopic parathyroid adenoma with solid and cystic components (Figs. EPAs are caused by migratory behaviors during embryogenesis. The inferior parathyroid glands, which are derived from the third branchial arch, share their origins with the thymus. Therefore, EPAs can be found in the thymus and anterior or upper mediastinum, even down to the aortopulmonary window. Approximately 20% of the parathyroid adenomas are located in ectopic sites, whereas ectopic mediastinal parathyroid adenomas, which require a thoracic approach, are reported in 1–2% of all cases . In thisComplete surgical resection is crucial for successful treatment of hyperparathyroidism. Prior to surgery for EPA, imaging modalities, such as computed tomography, magnetic resonance imaging, and MIBI scintigraphy, indicate the size, morphology, and location of the tumors. One study reported the usefulness of monitoring intraoperative PTH levels to reconfirm complete resection of EPAs . In our RATS is performed worldwide for thoracic surgery and has several advantages. Robotic instruments have novel characteristics, such as a three-dimensional view, tremor filtering, articulated instruments with 360° rotations, and seven degrees of freedom. Because of these innovations, RATS can be used for meticulous and time-consuming surgical procedures. Several case reports have described robotic-assisted mediastinal parathyroidectomy for hyperparathyroidism , 5. In JHerein, we report a case of a mediastinal EPA that was successfully resected using RATS.


Thymomas and thymic carcinomas are rare mediastinal neoplasms arising from thymic epithelial cells, and the presence of synchronous or metachronous primary thymic neoplasms in a single patient is an extremely rare event. Thymoma patients appear to have an inherent predisposition toward developing additional neoplasms. This additionally presents a diagnostic challenge, revealing the importance of multidisciplinary expertise to the management of these patients. This is a case report of a patient with a thymoma and thymic carcinoma, submitted to surgical resection and postoperative radiotherapy. Thymic epithelial cell-derived neoplasms, namely thymomas and thymic carcinomas, are infrequent tumors found in the mediastinum. Thymomas have non-neoplastic lymphocytes in a variable number. When we find epithelial tumor cells with overt cytologic atypia, invasiveness, and lack of thymus-like features of thymomas, we have a thymic carcinoma -4. ThymoA 73-year-old Caucasian male, who was submitted to cholecystectomy because of acute cholangitis, had an incidental finding of a mediastinal mass in the imagiologic evaluation. The patient had no other relevant past medical history. Computerized tomography (CT) scan revealed the presence of a heterogeneous lobulated mass in the anterior mediastinum, with some more hypodense areas, with approximately 68 mm of the transverse axis, 74 mm of the anteroposterior axis, and approximately 63 mm of the longitudinal axis. It presented regular and well-defined contours, without calcifications or fat Figure .Blood workup did not show cytopenias nor elevated inflammation parameters, and serologic tumor markers were negative. Positron emission tomography CT (PET-CT) scan showed an anterior mediastinal mass with moderate-to-intense avidity for fluorodeoxyglucose (FDG) Figure , and no The case was presented and discussed in a thoracic tumor board and decided to proceed to surgical resection of the mass for diagnostic and therapeutic purposes Figure .Tumorectomy via sternotomy was performed and the mass was described as multilobulated with white hemorrhagic areas of necrosis. The anatomopathological evaluation found thymic neoplasm patterns and focus of epidermoid carcinoma, with extension to the thymic fat. In the immunohistochemical study, CK5/6, p63, CK19, and EMA  expression were observed in epidermoid carcinoma. Due to the lack of secure resection margins, further treatment was decided with adjuvant radiotherapy directed to the anterior mediastinum, at a dose of 54 Gy in 27 fractions. The patient had no immediate nor short-term treatment-related complications, and remained asymptomatic. Several studies discuss the potential link between thymoma and other malignancies, both before and after a thymoma diagnosis. Thymomas can present as an incidental finding in asymptomatic individuals in 30% of cases, such as this one. Other than the thymoma, the patient had no other identifiable risk factors for the development of a tumor ,4. They We report an extremely rare case of combined thymoma and thymic carcinoma, both rare mediastinal neoplasms. There appears to exist an inherent risk of developing additional neoplasms in patients with thymomas, and when they are asymptomatic, the diagnostic challenge is even greater. This case also aims to emphasize the importance of the three pillars when approaching a mediastinal mass: imaging, histopathology, and multidisciplinary expertise. Depending on the diagnosis, the recommended treatments and follow-up strategies may differ significantly.


This paper presents a rare case of fetal hydrops detected at just 23 weeks of gestation in a 22-year-old woman’s first pregnancy. The fetal ultrasound revealed severe skeletal anomalies, craniofacial deformities, and thoracic abnormalities, suggesting a complex and severe skeletal dysplasia, potentially type IA Achondrogenesis—a lethal autosomal recessive condition marked by ossification delay. This case highlights the significance of advanced genetic testing, such as next-generation sequencing (NGS) and whole-genome sequencing (WGS), in diagnosing and understanding skeletal dysplasias. Skeletal dysplasias represent a group of genetic disorders that affect osteogenesis. The prevalence of this condition is 1 in 4000 births. Sadly, 25% of affected infants are stillborn, and around 30% do not survive the neonatal period. There is a wide range of rare skeletal dysplasias, each with its own specific recurrence risk, dysmorphic expression, and implications for neonatal survival and quality of life. When skeletal dysplasia is incidentally discovered during routine ultrasound screening in a pregnancy not known to be at risk of a specific syndrome, a systematic examination of the limbs, head, thorax, and spine is necessary to reach the correct diagnosis. Prenatal diagnosis of skeletal dysplasia is crucial for providing accurate counselling to future parents and facilitating the proper management of affected pregnancies. An accurate diagnosis can be a real challenge due to the wide spectrum of clinical presentations of skeletal dysplasia but advances in imaging technologies and molecular genetics have improved accuracy. Additionally, some of these skeletal dysplasias may present clinical overlap, making it especially difficult to distinguish. After the 11th revision of genetic skeletal disorder nosology, there are 771 entities associated with 552 gene mutations. The most common types of skeletal dysplasia are thanatophoric dysplasia, osteogenesis imperfect, achondroplasia, achondrogenesis, and asphyxiating thoracic dystrophy. This is a rare case of a 22-year-old woman referred to our fetal department due to fetal hydrops detected at 23 weeks of gestation. This was her first pregnancy from a non-consanguineous marriage, and she had no significant medical history. The patient initially sought medical attention at 23 weeks of gestation, primarily due to social issues. The ultrasound examination in our department revealed multiple abnormalities in the fetus, prompting further investigation and management.Short limbs with severe micromelia affecting all four limbs.Remarkably bowed femur and humerus.Bilateral clubfoot.Skeletal abnormalities see A,B:ShortHypomineralization of the skull bones.Enlarged head and small face.Craniofacial abnormalities see A,B:HypomNarrow thorax with evidence of pulmonary hypoplasia.Multiple rib fractures.Thorax abnormalities see A,B:NarroPresence of fetal hydrops, characterized by ascites, pleural effusion, and subcutaneous edema.Fetal hydrops See A,BPresenThe constellation of abnormalities observed in this case is highly indicative of complex and severe skeletal dysplasia. The prognosis for the fetus is concerning due to the severity of the abnormalities and associated complications. Further diagnostic investigations and genetic counselling were warranted to determine the underlying etiology and provide appropriate management and counselling for the patient and her family. The abnormalities observed were highly suggestive of type IA Achondrogenesis, which is a rare lethal autosomal recessive form of skeletal dysplasia characterized by a severe delay of ossification . DiagnosUnfortunately, despite our efforts this patient declined all further investigations and tests. She was offered the option of termination considering this is a lethal condition, and after discussing it with her partner she opted for termination on the same day see .In summary, this case report gives us an insight into the complexity of the diagnosis of skeletal dysplasias. These conditions stem from pervasive disruptions in bone development that commence during fetal growth and persist throughout an individual’s lifespan. The prenatal diagnostic process for suspected skeletal dysplasia entails comprehensive ultrasound imaging of various anatomical areas, including the long bones, thoracic region, extremities, cranial area, spinal column, and pelvic region. Achieving a precise diagnosis is reliant upon genetic testing, as it plays a crucial role in accurately identifying and understanding these conditions.


Although most duodenal carcinomas are pathological adenocarcinomas, a small number of cases have been reported of adenosquamous carcinoma, characterized by variable combinations of two malignant components: adenocarcinoma and squamous cell carcinoma. However, owing to the small number of cases of non-ampullary duodenal adenosquamous carcinoma, there have been no reported cases of emergency pancreaticoduodenectomy for gastrointestinal hemorrhage due to non-ampullary duodenal adenosquamous carcinoma.A 66-year-old Japanese male presented to the referring hospital with a chief complaint of abdominal pain, diarrhea, and dark urine that had persisted for 1 month. The patient was referred to our hospital because of liver dysfunction on a blood examination. Laboratory results of the blood on the day of admission showed that total and direct bilirubin levels (12.0 mg/dl and 9.6 mg/dl) were markedly increased. An endoscopic retrograde biliary drainage tube was inserted for the treatment of obstructive jaundice, and imaging studies were continuously performed. Contrast-enhanced computed tomography and endoscopy revealed an ill-defined lesion involving the second portion of the duodenum, predominantly along the medial wall, and measuring 60 mm in diameter. No metastases were observed by positron emission tomography. Pancreaticoduodenectomy was planned based on the pathological findings of poorly differentiated adenocarcinoma. However, 2 days before the scheduled surgery, the patient experienced hemorrhagic shock with melena. Owing to poor hemostasis after endoscopic treatment and poor control of hemodynamic circulation despite blood transfusion, radiological embolization and hemostasis were attempted but were incomplete. An emergency pancreaticoduodenectomy was performed after embolizing the route from the gastroduodenal artery and pseudoaneurysm area to reduce bleeding. The operation was completed using an anterior approach without Kocherization or tunneling due to the huge tumor. The operation time was 4 h and 32 min, and blood loss was 595 mL The pathological diagnosis was adenosquamous carcinoma. The postoperative course was uneventful with 17 day hospital stay and the patient is currently well, with no signs of recurrence 9 months after surgery.This report presents an extremely rare case of successful emergency pancreaticoduodenectomy for gastrointestinal hemorrhage caused by non-ampullary duodenal adenosquamous carcinoma. The incidence of duodenal carcinoma has recently increased due to the widespread use and development of endoscopic examination , 2. AlthA 66-year-old Japanese male presented to the referring hospital with a chief complaint of abdominal pain, diarrhea, and dark urine that had persisted for 1 month. The patient was referred to our hospital because of liver dysfunction on a blood examination. He had a history of treatment for hypertension, diabetes mellitus, and dyslipidemia, but no history of liver dysfunction or habitual alcohol consumption.Laboratory results of the blood on the day of admission showed that total and direct bilirubin levels (12.0 mg/dL and 9.6 mg/dL) were markedly increased. Mild elevations in the aspartate aminotransferase (56 U/L), alanine aminotransferase (50 U/L), gamma-glutamyl transpeptidase (296 U/L), alkaline phosphatase (196 U/L), and amylase levels (249 U/L) were observed. Carbohydrate antigen 19–9 was elevated at 48.9 U/mL, but the carcinoembryonic antigen level was normal at 3.6 ng/mL. Tests for hepatitis B virus surface antigen and hepatitis C virus antibodies were negative.An endoscopic retrograde biliary drainage (ERBD) tube was inserted for obstructive jaundice, and endoscopic and imaging evaluations were performed continuously. Computed tomography (CT) of the abdomen showed a 60-mm lesion in the area of the 2nd portion of the duodenum and pancreatic head, associated with gallbladder enlargement, bile duct dilation due to stenosis of the lower bile duct, and dilation of the main pancreatic duct. Contrast-enhanced CT revealed that the lesion originated from the medial wall of the duodenum, with irregular wall thickening, and extended consecutively to the pancreatic head, with the invasion of the common bile duct and main pancreatic duct. The anterior superior, posterior superior, and inferior pancreaticoduodenal arteries (IPDA), forming the network between the gastroduodenal and superior mesenteric arteries (SMA), were considered the feeding arteries. Positron emission tomography (PET)-CT showed accumulation SUV: 13.3) at the lesion; however, there were no abnormal accumulation findings at other sites  was difficult, the pancreas was gradually divided from the caudal side without a tunneling procedure, and the SMV anterior surface was exposed. Thereafter, the right side of the SMV/SMA/plexus was dissected from the uncinate process of the pancreas in an anterior-to-posterior direction. Finally, the pancreatic head and duodenum containing the tumor were dissected from the retroperitoneum and removed. The operation was completed using an anterior approach without Kocherization or tunneling. The operation time was 4 h and 32 min, and blood loss was 595 mL. The hemoglobin level was elevated to 9.8 g/dl after the surgery with total of 26-unit red blood cells transfusion.The resected specimen showed an 8-cm type 3 tumor that invaded from the duodenum to the pancreatic head and formed a deep ulcer with vascular embolization coils at the base of the ulcer Fig. . PatholoThe patient’s postoperative course was uneventful, and he was discharged on the 17th postoperative day. On postoperative day 58, S-1 (tegafur/gimeracil/oteracil potassium) plus oxaliplatin therapy (SOX) was initiated as adjuvant chemotherapy. The patient is currently well, with no signs of recurrence 9 months after surgery.The pathogenesis and natural history of NADASC are unknown owing to their rarity. The consensus regarding the treatment of these infrequent carcinomas is surgical resection of the tumor with negative margins, irrespective of histology. Furthermore, when duodenal carcinoma is large and deeply invades the pancreatic head, it may cause bleeding due to invasion of the pancreatic head arcade artery, as in the present case. Arteries in this region receive blood flow from both the celiac artery and the SMA, and hemostasis by interventional radiology can be difficult and requires surgical intervention. There are few reports in the literature on ePD for duodenal carcinoma. Only three reports have described ePD as a lifesaving procedure for gastrointestinal hemorrhage from duodenal carcinoma in detail –13  approach without Kocherization or tunneling in front of the SMV due to a large tumor with bleeding. Owing to the rarity of ASC in the duodenum, very limited information exists in the literature regarding radiologic findings, clinical features, and ideal management strategies. Further accumulation of cases and investigations of these uncommon carcinomas are needed.Here, we report an extremely rare case of gastrointestinal hemorrhage due to NADASC, which was successfully treated with ePD.


Calcinosis cutis describes the deposition of calcium in the dermis. A case of a 69-year-old woman with idiopathic calcinosis cutis that presented as a mobile subcutaneous nodule is described. The patient had an asymptomatic, firm, mobile subcutaneous nodule on her right lower leg of at least six months duration. The nodule could be easily moved from one location to another. An incisional biopsy was performed. Microscopic examination of the tissue specimen showed islands of basophilic calcium material in dense sclerotic dermal connective tissue establishing the diagnosis of calcinosis cutis. Mobile solitary calcification is an unusual presentation of idiopathic calcinosis cutis. In addition to idiopathic calcinosis cutis, benign mobile subcutaneous tumors have also been derived from adnexal structures of hair follicles and adipose tissue. Hence, not only idiopathic calcinosis cutis, but also subepidermal calcinosis in the ocular adnexa, proliferating trichilemmal cyst with focal calcification, and mobile encapsulated adipose tissue can present as a mobile subcutaneous nodule. The features of idiopathic calcinosis presenting as a mobile subcutaneous nodule as well as the characteristics of other benign mobile subcutaneous tumors are reviewed. The deposition of calcium in the skin is referred to as calcinosis cutis. Five types of calcinosis cutis have been observed. These include calciphylaxis, dystrophic calcinosis, iatrogenic calcinosis, idiopathic calcinosis, and metastatic calcinosis -4.Mobile subcutaneous tumors are uncommon. They can be derived from adipose tissue, adnexal structures, or calcium. These benign neoplasms can be manipulated to travel from one area to another -14.A woman presented with an asymptomatic mobile subcutaneous nodule; an incisional biopsy revealed a subepidermal calcific nodule. The history of this idiopathic variant of calcinosis cutis is summarized. Also, the features of other mobile subcutaneous nodules are reviewed.A 69-year-old Caucasian woman presented for evaluation of a lesion beneath the skin on her right leg. The patient noticed the new mass after experiencing a fall six months earlier. However, she commented that the lesion could have been present prior to her fall and that she had not been aware of it until after the fall.She had a 50-pack-year history of smoking cigarettes and also had multiple medical comorbidities. These included asthma, chronic pain, depression, gastroesophageal reflux disease, hyperlipidemia, macular degeneration, osteoporosis, and prediabetes; in addition, she recently had a keratoacanthoma removed from her right leg. There was no history of immunosuppression or collagen vascular disease. Her medications included an albuterol inhaler, alendronate, aspirin, flonase, omeprazole, trazodone, tylenol, and simvastatin. Her serum chemistries, including calcium and phosphate, were normal.A complete cutaneous examination, from head to toe, was performed. An 18 x 18 millimeter red scar was present on her right leg at the excisional biopsy site of the keratoacanthoma , scrotal calcinosis, and tumoral calcinosis ,2. Our pThe calcified subepidermal nodule was initially recognized by Winer in 1952 as a solitary congenital nodular calcification of the skin . He repoIn 1963, Woods and Kellaway introduced the nomenclature of subepidermal calcified nodules . Indeed,Subepidermal calcified nodule typically presents as a single, small, asymptomatic, painless, hard, freely mobile yellow-white papule on the head and neck region with the eyelid being the most common site. Rare locations such as fingers, knees, oral mucosa, palms, penis, soles, and toes have been documented. The appearance of the overlying epidermis can be verrucous, hyperkeratinized, or papillomatous, and can vary in color from yellow, white, gray, or pink ,17.There is no history of trauma, systemic disease, or abnormal laboratory findings in patients with the subepidermal calcified nodule. Notably, calcium, phosphate, and parathyroid hormone levels are typically in the normal range. When it presents as a single nodule, it can clinically mimic a calcifying pilomatrixoma, epidermal inclusion cyst, hemangioma, seborrheic keratosis, or trichilemmal cyst . When itThe woman in this report had an acquired asymptomatic subcutaneous mobile nodule. It could readily be moved from a proximal site on her leg to a more distal location and then it could be moved back to the original site of origin. An incisional biopsy not only removed the benign lesion but also established the diagnosis of a mobile subepidermal calcific nodule.In contrast to our patient’s lesion occurring on her leg, a retrospective study of 53 patients observed mobile subepidermal calcinosis to be localized to the ocular adnexa. Men were affected more commonly than women (in a 3:1 ratio) and most (89%) of the patients are 21 years of age or younger. In addition, the majority of patients (63%) were non-Caucasian .The lesions of mobile subepidermal calcinosis localized to the ocular adnexa appeared as a solitary, hard, white, or yellow nodules with papillomatous features and were frequently located on the upper eyelid. Most of the patients (82%) had a single lesion . RecentlSeveral investigators have proposed various mechanisms of pathogenesis for subepidermal calcified nodules. Woods and Kellaway suggested two mechanisms for the formation of the subepidermal calcified nodule. The first mechanism is that calcified granules within the stroma create a larger calcified mass through calcification and degeneration of the stroma. Alternatively, the large calcified lesion occurs first and its subsequent reabsorption leaves the calcified granules that form the calcified nodule with associated tissue changes .Other researchers have proposed calcification of pre-existing skin structures. It was thought that cutaneous calculi might be derived from milia. It has also been postulated that they are hamartomas of sweat duct origin, most likely syringomas, with calcification. Alternatively, they may result from calcium deposits in a preexisting nevus .In 1980, Tezuka explained the mechanism that forms a subepidermal calcified nodule to be secondary calcinosis resulting from the deposition of calcium and phosphate after the degranulation of mast cells ,18. CalcFor a patient with calcinosis cutis, a series of laboratory tests and imaging studies are recommended to identify the type of calcification and determine its etiology. In all patients, serum calcium, inorganic phosphate, alkaline phosphatase, and albumin levels should be measured; serum calcium or phosphate or both are elevated in metastatic calcification. All of these laboratory tests were normal in our patient ,4.Definitive diagnosis of calcinosis cutis is by biopsy. Microscopic examination shows calcium deposition in the dermis, with or without chronic lymphocytic inflammation. Often a histiocytic reaction to the dermal deposits is also present. Indeed, chronic inflammation and foreign body giant cell reactions are prominent in children and adolescents; in contrast, these pathologic changes may be mild or absent in adults ,14.Surgical excision is the first-line therapy for idiopathic calcinosis cutis, as was done in our patient. Indeed, an excisional biopsy -- for diagnostic purposes and treatment -- is an excellent approach when the clinical diagnosis of calcinosis cutis is suspected. Recurrence can occur after incomplete excision .Indications for the treatment of subepidermal calcified nodules are impairment of function due to location and the presence of pain. However, cosmetic concerns are the most common reasons for treatment. In addition to surgical removal, intralesional corticosteroid injection is another treatment modality for subepidermal calcified nodules .The differential diagnosis of benign mobile subcutaneous tumors is summarized in Table Patients with MEAT can present with multiple lesions. MEAT is also referred to by several designations. The adipose tumors of MEAT were initially reported as “abacus tumors,” a name coined to describe a marble-like, subcutaneous mass that is freely mobile, often over distances of several centimeters. Most of the lesions occur in regions vulnerable to trauma, such as the lower and upper extremities. They have also been occasionally described in the peritoneum -7.Subsequently, these adipose tumors have been referred to as mobile encapsulated lipoma, nodular cystic fat necrosis, encapsulated fat necrosis, and encapsulated adiponecrosis ,6. They More recently, MEAT was proposed as a more accurate description . The tumOssifying plexiform tumor is an extraordinarily rare benign cutaneous neoplasm. Although none of the lesions demonstrated reports of progression or metastasis, local recurrence of the tumor was documented in one patient. It presents as subcutaneous nodules on the fingers of women, measuring 1 to 2 centimeters in greatest dimension. The tumor has a distinctive histologic appearance typified by plexiform nests of cytologically bland spindle-to-epithelioid cells in a myxocollagenous stroma with associated foci of central ossification, which may range from osteoid to the mature bone -11.A proliferating trichilemmal cyst is also known as either a pilar cyst or wen. They are slow-growing, rubbery, non-tender, mobile nodules typically located on the scalp. Microscopic examination can reveal focal calcification. Complete excision is recommended since the tumor has a potential for locally aggressive behavior and malignant transformation .The woman in this report presented with an asymptomatic mobile nodule. The tumor was completely removed when an incisional biopsy was performed. Microscopic examination established the diagnosis of a benign subepidermal calcific nodule.Calcinosis cutis is a clinical entity characterized by the deposition of insoluble calcium salts in the skin and subcutaneous tissue. There are five variants of calcinosis cutis: calciphylaxis, dystrophic calcification, iatrogenic calcification, idiopathic calcification, and metastatic calcification. A woman presented with a subcutaneous and mobile nodule on her leg; an incisional biopsy not only removed the entire lesion but also established the diagnosis of the subepidermal calcific nodule type of idiopathic calcinosis cutis. In addition to a subepidermal calcific nodule, MEAT, proliferating trichilemmal cysts, ossifying plexiform tumor, and subepidermal calcinosis in the ocular adnexa can present as a mobile subcutaneous neoplasm. Surgical removal of the lesion and microscopic evaluation of the tissue specimen can differentiate the morphologically similar appearing lesions.


Giant chorangiomas are uncommon yet frequently associated with adverse pregnancy outcomes. A 37-year-old female was referred due to findings of a placental mass during a second-trimester ultrasound. A fetal survey at 26 weeks revealed a 69×97×75 mm heterogenous placental tumor with two prominent feeding vessels. Her prenatal course was complicated by worsening polyhydramnios requiring amnioreduction, gestational diabetes, and transient severe ductal arch (DA) constriction. Placental pathology confirmed the diagnosis of giant chorioangioma following delivery at 36 weeks. To our knowledge, this represents the first case of DA constriction in the setting of a giant chorangioma. Chorangiomas were first described by Clarke in 1798  and are A 31-year-old woman, gravida two, para one, was referred for findings suspicious of placental chorangioma. An ultrasound (US) at 26 weeks gestation showed a live fetus with an estimated fetal weight (EFW) at >99th percentile, polyhydramnios (amniotic fluid index {AFI} = 43.7 cm), and a well-defined, circumferential, heterogenous placental mass at the chorionic surface bulging into the amniotic cavity measuring 69×97×75 mm. On ultrasound, two prominent feeding vessels were located 34 mm away from the placental cord insert Figures -1d.The anatomic survey, including a fetal echocardiogram, and Doppler assessment of middle cerebral artery peak systolic velocity (MCA-PSV) and umbilical artery pulsatility index (UA-PI), was unremarkable. Given worsening abdominal discomfort, hospital admission for monitoring, antenatal steroids, and US-guided amnioreduction (AR) were recommended. Additionally, we discussed the availability of in-utero devascularization in the event of fetal hydrops and/or heart failure. A total of two liters of fluid was removed, and the post-procedure deepest vertical pocket (DVP) was 9 cm. The patient reported significant relief and was ultimately discharged home on hospitalization day 3. An amniotic fluid analysis reported a 46XX karyotype and normal microarray. Biophysical profile (BPP), fetal echocardiogram (FE), MCA-PSV, UA-PI, tumor size, and hydrops assessment were performed weekly thereafter, in addition to serial growth examinations. Her prenatal course was also complicated by gestational diabetes (GDM), which was well-controlled. A follow-up US at 29 weeks noted spontaneous resolution of previously diagnosed polyhydramnios. However, a repeat FE one week later (30 weeks) revealed new-onset severe DA constriction, moderate tricuspid valve regurgitation (TR), and right ventricle (RV) dysfunction, prompting re-admission for monitoring Figures -2d.During her hospitalization, interval US at 32 weeks showed the absence of previously identified blood flow through the tumor’s feeding vessels, shrinking chorangioma 85×51×74 mm) and feeding vessels, and resolution of prior echocardiographic findings. Outpatient follow-up resumed, and labor induction was undertaken at 36 3/7 weeks after a thorough discussion with the patient and family members. A 2770 g baby girl was delivered vaginally with appearance, pulse, grimace, activity, and respiration (APGAR) scores of nine and nine at one and five minutes, respectively. An episode of neonatal hypoglycemia warranted a 24-hour admission to the neonatal intensive care unit. Otherwise, her course was uncomplicated, and she was discharged home on the second day of life. The placenta weighed 650 g (90-95th percentile). Gross examination revealed an 83×82×47 mm peripheral pink-gray-yellow variegated, rubbery, demarcated, subchorionic, infarcted mass, and histopathological examination confirmed the diagnosis of giant chorioangioma .Chorangiomas arise from the chorionic mesenchyme due to aberrant vasculogenesis of mature stem villi. Prenatally identified tumors are most commonly detected in the late second trimester ,8. Our rThe scarcity of evidence has challenged the antenatal management of giant chorangiomas. The likelihood of perinatal death is positively correlated with tumor size and the presence of fetal hydrops . TemporiOur case represents the first report of a giant placental chorangioma and severe DA constriction with favorable outcomes. Although the mechanistic link between these two conditions has yet to be discovered, this report highlights the importance of serial fetal echocardiograms in pregnancies complicated by these tumors to identify changes that may warrant closer monitoring to minimize complications. The rare nature of giant chorangiomas calls for further evidence to delineate their natural history and management strategies that help improve outcomes for these patients.


An eight-year-old boy presented with a one-month history of left eyelid swelling. The patient was diagnosed with periorbital cellulitis at another clinic and was treated with oral antibiotics. However, the swelling did not subside. On initial consultation, the patient had left upper eyelid swelling with erythema. His extraocular muscle motility was normal, and the results of blood tests were unremarkable. A computed tomographic scan demonstrated a mass in the superior orbit with destructive changes in the left frontal bone. Histopathological examinations revealed a dense infiltrate of histiocytic cells. Immunohistochemical staining of the tumors for CD1a and Langerin was positive. A pathological diagnosis of Langerhans cell histiocytosis was made. Since orbital Langerhans cell histiocytosis has a high risk for central nervous system involvement, chemotherapy was the treatment of choice for any residual lesion to prevent sequelae to the central nervous system. At the six-month follow-up, the lesion did not recur, and the patient did not experience any central nervous system sequela. Langerhans cell histiocytosis (LCH) is a rare inflammatory myeloid neoplasm that usually develops during childhood. LCH affects single or multiple organs and can be classified into three distinct types: single-system single site (SS-s), single-system multisite (SS-m), and multisystem (MS). Orbital involvement of LCH is rare, and a previous report showed only one (0.4%) of 250 biopsied orbital lesions in children was LCH . OrbitalSince the prognosis of SS-s LCH is generally good, excisional biopsy or curettage is chosen for the treatment of SS-s LCH . HoweverAn eight-year-old boy presented with a one-month history of swelling in the left upper eyelid with no associated periocular pain or pain on eye movement. The patient was treated at an ophthalmology clinic with oral antibiotics for a few weeks, based on the diagnosis of left periorbital cellulitis. The swelling temporarily subsided but then relapsed shortly after. He had a prior history of ventricular septal defect and asthma. There was no remarkable family history. On the initial consultation, the patient had noticeable swelling and erythema in the left upper eyelid without tenderness on palpation Figure . A subcuA CT scan demonstrated a mass in the superior orbit with destructive changes in the left frontal bone Figure . There wA biopsy of the left orbital lesion was performed under general anesthesia by two of the authors (TY and YT) three days after the patient was referred to our department. The orbital lesion was removed as thoroughly as possible using an ultrasonic aspirator. Pathological examinations of the harvested specimen revealed a dense infiltration of histiocyte-like cells with infiltration of neutrophils, lymphocytes, and eosinophils Figure . ImmunohSince orbital LCH has a high risk of CNS involvement, chemotherapy was the chosen treatment modality to address the remaining lesion and avoid CNS sequelae. Induction therapy, including cytosine arabinoside, vincristine, and prednisolone, was administered for six weeks, and maintenance therapy was given thereafter. At six-month follow-up, the lesion did not recur, and the patient did not experience any CNS sequela.We report a pediatric case of orbital LCH who presented with left eyelid swelling and erythema, which led to a misdiagnosis of orbital cellulitis by the attending ophthalmologist. Although the incidence of LCH and orbital cellulitis are predominantly found in childhood , it is iDuring the incisional biopsy of the left orbital lesion, we attempted to remove as much of the lesion as possible using an aspirator. However, a considerable size of residual tumor was left, which was confirmed on postoperative MRI. Since the patient had no other lesions throughout the rest of the body (SS-s LCH), re-curettage or re-aspiration of the lesion was a treatment option in our case. However, the risk of cerebrospinal fluid leakage outweighed the benefit of such a treatment option. Furthermore, chemotherapy was strongly recommended for cases with lesions at risk for CNS involvement. A previous study reported that more than 70% of patients with CNS-risking lesions received chemotherapy, and 144 of the 146 99%) patients did not have active disease at the last follow-up [9% patienWe report a case of orbital LCH in a pediatric patient. Although eyelid swelling and erythema resembled orbital cellulitis, the typical CT finding of a soft tissue mass in the superolateral orbit with an osteolytic lesion led us to the early performance of the biopsy. Since orbital involvement is recognized as a CNS-risking lesion, chemotherapy was chosen to prevent CNS sequelae. A cooperative multidisciplinary approach involving not only general ophthalmologists and orbit specialists but also radiologists, pathologists, and pediatric oncologists is essential for the proper management of orbital LCH.


We report an atypical granular cell tumour (GCT) presenting in a 6‐year‐old boy. GCTs are of neural origin and most cases arise in patients between the ages of 40 and 60. There are few reported cases in children, in whom malignant presentations are exceptionally rare. This patient presented with a 1 year history of a slowly enlarging nodule on the right anterior abdomen. Examination revealed a firm, nodular dermal skin lesion, which was fully excised. Histology revealed an atypical GCT. We report a rare case of atypical granular cell tumour presenting in a 6‐year‐old boy. We report the presentation, excision, and histological findings of this unique case. Examination revealed a firm, nodular, dermal skin lesion measuring 20x11 mm. The skin overlying was erythematous , suggestive of atypical GCT. The nodule was fully excised with a 3 mm margin, including a cuff of Scarpa's fascia to which the lesion had adhered. Subsequent histology confirmed a well‐circumscribed but non‐encapsulated dermal tumour. The morphological impression of GCT was supported through strong S‐100 staining on immunohistochemistry. There were 7 mitoses per 10 high power fields (x200) and the mib proliferation index was approximately 5% , high nuclear to cytoplasmic ratio, and pleomorphism.The tumour extended deep to the scarpa's fascia, illustrating these tumours can behave in an invasive manner even if otherwise categorised as benign. Wide local excision and long‐term monitoring is recommended, as local recurrence is 2%–18%.This case report highlights the clinical features of an unusual paediatric presentation of an atypical GCT. Granular cell tumour should be amongst the differential for solitary subcutaneous nodule and this case demonstrates the importance of histological sampling. There is need for follow‐up guidelines for GCT.All authors declare no conflicts of interest.Yuma Kitahara: Project administration ; Writing – original draft (Lead); Writing – review & editing . Catherine Elizabeth Hook: Writing – review & editing . Kana Miyagi: Writing – review & editing . Nigel P. Burrows: Conceptualization (Lead); Project administration ; Writing – review & editing .Not applicable.


A cutaneous horn is a rare, hyperkeratotic, projecting lesion that can be mostly found in sun-exposed areas of the skin. The base of the lesions can reveal an underlying malignancy. They can also be associated with several benign or pre-malignant dermatologic conditions. A biopsy of the base of the lesion and histopathological analysis are needed to confirm the diagnosis. Management depends on the underlying disease; however, surgical excision is the preferred treatment method. A cutaneous horn, less commonly described as cornu cutaneum or devil's horn, is a conical, protruding, hyperkeratotic growth, sometimes large enough to resemble an animal horn . CompactA 34-year-old South-Asian male, a small shop owner, visited the general outpatient surgery clinic with a strange history of a horn-like projection from his abdomen Figure  for arouThe patient had normal physical growth with a muscular body on examination. Upon closer inspection of the abdomen, a curved growth of around 12 cm x 5 cm x 4 cm in length, width, and height, respectively, was seen arising from the anterior abdomen, around 10 cm cranial to the level of the umbilicus and one-inch right lateral to the midline (Figure The lesion was painless, firm-to-hard on palpation, and projected more outward as a curved horn on the leg-rising test's contraction of the abdominal wall muscles. No features of malignancy, like adjacent surrounding skin involvement or changes in skin texture, were noted. No similar growth was noticed on his body. Despite thoroughly discussing treatment options and possible outcomes, the patient hesitated for an excisional biopsy as he believed the growth to be auspicious.Patients with cutaneous horns usually present with a conical, projecting growth in predominantly sun-exposed areas. Therefore, it is important to inquire about the patient's nature of work or involvement in outdoor activities ,6. It shUpon palpation, the clinician would find a firm-to-hard, well-rooted structure varying from a few millimeters to several centimeters long. The horns are usually prominent, white to yellowish, and keratinous ,3. The cHistopathological findings of the horn will show thickening of the stratum corneum or hyperkeratosis with or without acanthosis. If the lesion is benign, one can observe parallel, horizontal layers of keratinized tissue . While aManagement of a cutaneous horn varies as per the underlying etiology of the condition. There are three mainstays of treatment: surgical, through total excision, medical therapy, or laser ablation . In the Psychological aspects should also be considered in the case of a cutaneous horn. Some patients may express feelings of embarrassment and believe the lesion to be a hindrance to their social life. The clinician must address these aesthetic complaints and counsel the patient as they can significantly impact their mental health -12.The cutaneous horn of the abdomen is a rare and peculiar occurrence. However, clinical history, examination, and excisional biopsy confirm the diagnosis. Successfully managing a cutaneous horn involves a combination of surgical excision and close follow-up. Regular monitoring is crucial to identify any potential recurrence or malignant transformation. More reporting of such cases and research will contribute to a better understanding of this fascinating phenomenon and aid in optimizing patient care.


The cholecystocolonic fistula (CCF) is an atypical variant of biliary disease, and it is the second most common intestinal fistula after cholecystoduodenal fistula. Intraoperative diagnosis is frequent, which implies challenging surgical management, especially in patients, often aged, with comorbidities. The rarity of this condition, atypical and various presentation, diagnostic and management complexity, makes it a unique surgical entity. We report our experience of an 84-year-old man with a history of chronic cholecystitis who presented with nonspecific symptoms. The imaging tests aroused the suspicion of gallbladder-colic fistula in the preoperative diagnosis, facilitating the subsequent surgical treatment that confirmed the diagnosis. Biliary fistulas to various structures, especially to the gastrointestinal tract  and the abdominal wall , have been disclosed in the literature. Particularly, the cholecystocolonic fistula (CCF) is a late complication of chronic gallbladder disease. It represents the second most common gallbladder-enteric fistula  after cholecystoduodenal fistula. Its incidence is approximately 0.06%-0.14% of patients undergoing cholecystectomy, and more than 90% of these cases are diagnosed intraoperatively 2), dyslipidemia, smoking, CAD, and biliary lithiasis. In our ward, the patient underwent a magnetic resonance cholangiopancreatography (MRCP), which showed the presence of an essential inflammatory process affecting the gallbladder with a fluid layer in the peri-bladder area . Moreover, biphasic contrast medium injection in whole-body computed tomography (WBCT) was assessed. It revealed thickened gallbladder wall with multiple calculi embedded within it and, in the fundal region, a slight soft tissue tract leading to the adherent adjacent part of the hepatic flexure of the colon, which showed thickened and anatomically uneven walls as well . An areaBased on the radiological findings, fistulous communication between the fundal region of the gallbladder and the hepatic flexure of the colon was diagnosed.The patient underwent laparotomic treatment of the gallbladder and the hepatic flexure of the colon. Then, he was followed up over 9 days on inpatient care basis and discharged with a complete resolution of the clinical picture.CCFs are one of the rare complications associated with gallstone disease. A persistent inflammatory process can lead to ulceration and ischemia of gallbladder walls and closed organs due to the erosive and fistulization process. In addition to a chronic inflammatory process, previous surgical treatments represent other possible causes, in particular, gastric surgery Symptomatology comprises a broad spectrum, varying from diarrhea, which is the most frequent symptom (70%) Another uncommon but described presentation of the CCF is acute onset complications like gallstone ileus, a stone located in the sigmoid colon after perforation of the walls, and massive bleeding or liver abscess This report describes a case where abdominal pain and fever were the only symptoms. The patient's medical history was decisive in arousing the suspicion of this rare complication.Even if the patient's clinical situation was stable, laboratory exams showed an increase in the selected cholestasis indices, associated with transaminases as hepatic functional evaluation, requiring further investigation by imaging tests.Generally, the diagnosis of CCF is achieved preoperatively in only 8% of cases. As a first approach, US evaluation offers prompt and rapid diagnosis of chronic cholecystitis, but no obvious signs of a fistula may be present. In most cases, it is useful to screen the abdomen. In fact, ultrasound is not usually diagnostic, while the survey with barium enema has sometimes allowed the identification of the cholecystocolic fistula In this medical case, biphasic contrast medium injection in whole-body computed tomography (WBCT) was able to detect fistulous communication between the fundal region of the gallbladder and the hepatic flexure of the colon, together with chronic cholecystitis. An abdominal CT scan is the gold standard imaging method with a sensitivity and specificity of 93% and 100%, respectively According to the literature, the preoperative diagnosis of CCF is quite challenging. The absence of preoperative diagnosis often forces a more complex procedure due to the intraoperative diagnosis, changing the surgical options from an elective course to an emergency one, which usually involves adhesiolysis and bowel resection too, as main complications, especially in aged and comorbid patients. Alternatively, when it is possible, it is warmly suggested ERCP with endoscopic sphincterotomy as the primary potential option , reducing the pressure in the biliary ducts and promoting the closure of the uncomplicated fistula.Several surgical treatment approaches have been reported for both uncomplicated gallbladder-colic fistula and those associated with complications. However, no “consensus” has been developed to address the optimal surgical management of CCF. Conservative management is also suggested for uncomplicated fistulas in frail patients with limited life expectancy From our experience with this case, we tend to disagree with the conservative management suggested by literature, particularly for frail patients like ours. As there is evidence of biliary obstruction risk with potential biliary sepsis from colonic bacteria translocation or of the passage of gallstone in the sigmoid colon, all with deleterious physiologic changes and unfavorable consequences, we aim to underline the importance of performing surgery when the clinical condition of the patient is sufficiently stable to be undergone.The particular clinical condition of our patient, in addition to the necessity for a shorter operative time, the risk of the hostile operative field, and a higher conversion rate from laparoscopic to open procedure in the setting of CCF, made the laparotomic system safer than the laparoscopic approach. In the end, cholecystectomy with the fistula tract resection should be recommended for that medical case.Gallbladder perforation with a colic fistula is often a rare complication of chronic gallbladder disease. Symptoms are minimal and usually nonspecific, and preoperative instrumental investigations often fail the diagnosis, which is considerably challenging for the surgeons.We hope that outlining this case will encourage the report of further instances to develop guidelines for an optimal diagnosis and surgical management of this rare entity.Written informed consent was obtained from the patient.


The gingival cyst of the adult (GCA) is a rare odontogenic cyst, consisting of 0.3% of all odontogenic cysts. This case report, based on CARE guidelines for case reports, aims to present a case of a 52-year-old female patient with a symptomatic translucent nodule in the upper left anterior gingiva, measuring approximately 6mm. Excisional biopsy was performed, and the histological examination revealed multiple cystic cavities lined by the squamous epithelium of varying thickness with focal areas of nodular thickenings. The presence of clusters of cells with clear cytoplasm within epithelial thickenings was observed. PAS staining was negative in clear cells. The diagnosis of the GCA was established. Despite its rarity, GCA should be considered in the differential diagnosis of gingival lesions. Conservative surgical treatment proved to be effective, with no signs of recurrence. In addition, the possibility of migration of epithelial remnants from the surface epithelium after trauma is discussed.4The gingival cyst of the adult (GCA) is a rare odontogenic cyst with 157 cases reported until 2019.5 Usually, present as nodules in the vestibular gingiva of the mandibular canine and pre-molars of women around the 5th and 6th decades of life.6This lesion is characterized by a capsule composed of fibrous connective tissue with mild inflammation coated by non-keratinized epithelium.Hereby, we report a case of a 52-year-old female patient with a GCA in the in the upper left gingiva.7 A 52-year-old female patient presented to the dentist complaining of a gingival lesion over the last 3 months. The patient denied a history of local trauma. The oral examination revealed an asymptomatic translucent nodule in the upper left gingiva, near tooth #11, measuring approximately 6mm, absent anterior teeth, and generalized gingival resorption , other odontogenic cysts as a lateral periodontal cysts or inflammatory cysts as the radicular cyst.6 Furthermore, gingival metastases commonly resemble benign nodules.11 The GCA is only diagnosed in 50%12 to 55%6 of the cases.GCA usually presents as a firm, well-defined, nodular elevation with a transparent and slightly bluish color. Generally, less than 1 cm in size in the attached gingiva or interdental papilla.8 This cyst has indolent radiographic features,1 however, it may cause radiolucent changes due to the pressure atrophy of the underlying alveolar bone.2 In some cases, the atrophy can be so intense that it generates a more well-defined lesion similar to those of lateral periodontal cysts.13 In the 157 cases found in the literature, no reports of bone expansion, cortical bone perforation, tooth displacement or resorption caused by GCA were observed. In 38 cases (24.2%), bone erosion was observed.1 A series of 20 cases12 demonstrated the presence of radiographic alterations in only two patients. Our case did not present any bone involvement.Among the odontogenic cysts, GCA only originates exclusively from soft tissues.10 It may also show remnants of glycogen-rich clear cells from the dental lamina, cystic enlargement, and degeneration with the formation of microcysts.2Histologic features of GCA include a non-inflammatory fibrous capsule lined by a non-keratinized thin epithelium, eventually with areas of thickening.1 Follow-up ranged from 1 to 92 months. Only 1 case of GCA recurrence after 7 years of the lesion’s excision was found. However, it is not possible to confirm whether it was a true recurrent cyst or a second lesion in the same location of the excision.13 Generally, recurrence is not expected after surgical excision of the lesion with minimal margins.Surgical excision was the treatment in 96.5% of cases reported in the literature. Other forms of therapy included curettage (2.7%) and enucleation (0.9%).Despite its rarity, the GCA should be considered in the differential diagnosis of gingival lesions. A correct diagnosis can exclude lesions with a worse prognosis or that require more aggressive therapy. In the presented case, conservative surgical excision proved to be effective, with no signs of recurrence.


Temporomandibular Disorder (TMD) is a disease according to the International Statistical Classification of Diseases a significant public health problem, which is considered the most common cause of chronic pain of non-dental origin.In this regard due to the anatomical proximity between the ear and the TMJ, many patients report an otological complaint which may be associated with a type of TMD including tinnitus, auricular fullness, vertigo, itching, joint noise sensation and otalgia; therefore, the first evaluation of the otolaryngologist is common,Synovial Chondromatosis (CS), more common in larger joints such as knee and shoulder andrarelyin the TMJ.Thus, the objective of this study is to report a rare case of CS in the TMJ with a complaint of related otological symptoms and with the success through arthroscopic surgical treatment.This study was conducted within ethical criteria and approved by the Research Ethics Committee of the Universidade do Oeste Paulista ‒ UNOESTE of Presidente Prudente, Brazil with protocol number 7770.Patient M.C.R., 37-years old, male, Caucasian, presented with a complaint of pain in the left ear for one year. At the beginning of the symptom, he sought an otolaryngologist who performed all the investigation through clinical and complementary exams, without systemic changes and with otoscopy showing a slightly convex, translucent, mobile, and intact tympanic membrane. He came to the conclusion that the otalgic complaint was not justified, and the patient was forwarded for evaluation by a professional dentist specializing in Temporomandibular Disorders and Orofacial Pain.Through the propaedeutic of clinical examination without obtaining an image, Temporomandibular Disorder (TMD) was diagnosed as the cause of the symptom and conservative clinical treatment was performed for one year at the expense of physiotherapy sessions, photobiomodulation with low intensity laser, electrotherapy, interocclusal devices and occlusal adjustments. However, due to no improvement in the complaint, the patient was forwarded for evaluation by the dental specialty of Oral and Maxillofacial Surgery.Afterwards clinical examination and complementary imaging (magnetic resonance imaging) it was possible to observe the presence of numerous free bodies  in the left Temporomandibular Joint (TMJ) A and B. The loco regional examination showed arthralgia on palpation in the left TMJ region with radiating to the left ear, without the presence of noises or other relevant signs.The patient underwent arthroscopic surgery on the left TMJ with the aim of removing free bodies. Thirty-four free bodies were found and removed , mostly Chondromatosis affecting the temporomandibular joint is considered a rare event.Particularly in the case reported, an important point that we must discuss is that the patient's symptom was isolated, the reflex otalgia, did not present crackling, limitation or deviation in mouth opening, common signs reported in the literature and presented in TMD and CS. Therefore, what we discussed here is that even without common complaints of internal TMJ disorders, CS can cause restricted otological complaints, however, arising from a joint pathology, even without presenting signs and symptoms common to temporomandibular disorders. A possible explanation for this fact is that the greatest amount of chondromatosis free bodies was present in the retrodiscal region, a region of great innervation and TMJ sensitivity, causing this patient's reflex otalgia.The recommended treatment method for CS is surgicalCurrently, arthroscopic surgery for the surgical treatment of TMJ CS has been gaining ground due to the positive results with the technique. Cai et al.An important aspect of arthroscopic surgical treatment is the power of navigating inaccessible spaces in open surgery. Bai et al.Currently, studies seem to suggest arthroscopic surgery when the CS of the TMJ is restricted to the upper intra-articular space and open surgery when the CS embrace the extra-articular space.Despite the TMJ synovial chondromatosis being rare, it is important in the presence of otalgia, especially in cases of non-involvement of otological pathologies, or when considering TMD's, consider it as a differential diagnosis combined with MRI as a complementary exam and decisive for reducing the time for diagnosis. Finally, in cases of intra-articular CS, arthroscopic surgery is a viable, less invasive, and efficient option in the treatment of TMJ CS.The authors declare no conflicts of interest.


A 10-year-old boy was evaluated for intermittent colicky abdominal pain, general malaise, and asthenia. Imaging revealed a solid liver lesion. Subsequent biopsy and extension studies diagnosed the lesion as undifferentiated embryonal sarcoma of the liver, classified as PRETEXT II, group III according to the postoperative staging system of the Intergroup Study for Soft Tissue Sarcomas. He underwent neoadjuvant chemotherapy using alternating cycles of ifosfamide, doxorubicin, vincristine, D-actinomycin, and cyclophosphamide. This was followed by surgical intervention and two additional adjuvant chemotherapy cycles, resulting in a complete disease response. The patient remains in follow-up and shows no signs of relapse 28 months post-diagnosis. Undifferentiated embryonal sarcoma of the liver is a rare and often misdiagnosed condition that can be mistaken for a benign disease. Its prognosis hinges on timely and accurate diagnosis, which is essential for effectively treating patients with this aggressive pathology with a high mortality risk. Notably, there is no standard treatment approach. In our case, we implemented therapeutic strategies from various literature reports, yielding a promising outcome and positive patient progression. Undifferentiated embryonal sarcoma of the liver (UESL) is a rare tumor of mesenchymal origin first identified in 1978 by Stocker and Ishak . Most caA 10-year-old boy with no notable medical background came to us, reporting a month of generalized discomfort, weakness, lethargy, and sporadic colicky abdominal pain. An abdominal ultrasound detected a primarily solid liver mass, suggesting either a hepatic hemangioma or sarcoma. A contrast-enhanced abdominal computed tomography (CT) scan displayed an exophytic lesion in hepatic segment IV, sized 87 x 75 mm on the axial plane. This lesion partially pressed against the gallbladder without invading it and showed clear suprahepatic and main portal veins Figure . A subseUpon assessment, the boy was fever-free, and an 8-cm tender mass was palpable in his right upper quadrant. The complete blood count and liver function tests were within reference ranges. Both α-fetoprotein and β-human chorionic gonadotropin levels were within normal limits, and there was no sign of infectious diseases. A follow-up abdominal ultrasound revealed a growth in the right hepatic lobe due to a mainly solid mass showing peripheral flow on Doppler and dispersed hyperechoic areas without acoustic shadowing in hepatic segment IV. This lesion had an exophytic profile with numerous vessels and arteriovenous malformations, suggesting a sizable hemangioma. Another contrast-enhanced abdominal CT depicted a lesion in hepatic segment IV, measuring 103 x 85 mm, with increased fatty density nearby in the peritoneum and slight compression of the gallbladder without invasion Figure .Our institution's pathology department review described a malignant tumor consisting of spindle and pleomorphic cells with vague cytoplasm. These cells were organized in relaxed sheets with some hemorrhagic necrosis zones. Immunohistochemical tests indicated positive results for cluster of differentiation 56 (CD56), vimentin, B-Cell lymphoma 2, and desmin, with a proliferation rate (as measured by the KI67 index) of 60%. The tumor did not react to myogenic differentiation, myogenic factor 4, glypican 3, or keratin AE1-AAE3, confirming the diagnosis of undifferentiated embryonal hepatic sarcoma.Considering the extensive involvement of liver segments I, III, and IV and the tumor size that initially rendered a full resection infeasible, the patient underwent neoadjuvant chemotherapy with ifosfamide  and doxorubicin (37.5 mg/m2/day on Days 1 and 2 of each cycle), administered every 21 days. After two cycles, a subsequent CT scan showed a mostly liquid mass in hepatic segment IV, measuring 95 x 69 mm on the axial plane, with the stable disease indicated Figure . After rA surgical oncologist then assessed the boy and performed a resection using intraoperative ultrasound guidance. This procedure involved an extra-anatomic segmentectomy of segments IV and part of V, including the gallbladder in the resection. Post-treatment pathology confirmed the presence of undifferentiated embryonal hepatic sarcoma, measuring 75 x 72 mm, with 1% viable tumor and 99% necrosis. The tumor displayed positive markers for glypican 3, vimentin, CD56, and desmin, with a KI67 index of 40%. No tumor was detected at the section margins (Figure The patient began adjuvant chemotherapy 15 days post-resection and completed six cycles. Post-treatment imaging, including an abdominal CT, showed changes resulting from the hepatic segment IV resection but no masses at the surgical margin. The remaining liver segments appeared normal, with no detected lesions Figure . A contrUESL is an infrequent and aggressive malignant tumor originating from primary mesenchymal tissue . It reprClinically, UESL may be detected unexpectedly in an asymptomatic child or might present symptoms such as an abdominal mass, pain, fever, weight loss, and anorexia, like in our case. Other symptoms can include vomiting, diarrhea, lethargy, constipation, and dyspnea ,4. FeverThere are no unique serum markers for UESL. Some patients may show signs of leukocytosis and anemia potentially resulting from intratumoral events like hemorrhage, necrosis, or infection . OccasioOn imaging, X-rays may depict dense, soft tissue in the upper right quadrant with the displacement of adjacent intestinal loops. Ultrasound often reveals a primarily solid, encapsulated tumor with varied echogenicity. Typically, tumors exceed 10 cm at diagnosis . CT may UESL diagnosis is confirmed via histopathology and immunohistochemistry . TypicalDue to UESL's rarity and its potential for misdiagnosis, many patients receive inaccurate preoperative evaluations. Several differential diagnoses exist, ranging from benign conditions like hepatic mesenchymal hamartoma and hydatid cysts to more severe diseases like hepatoblastoma and hepatocellular carcinoma ,3,9.Treatment strategies have evolved over the years. Initially, treatment centered on surgical resection, yielding survival rates below 40% -4. SubseUESL, though rare, poses significant clinical challenges due to its aggressive nature and potential for misdiagnosis. This report underscores the necessity for healthcare practitioners, including pediatric specialists, to recognize UESL when evaluating atypical liver lesions in children aged eight to 18. Heightened awareness can expedite accurate diagnosis and timely intervention, improving patient outcomes.


A false pituitary tumor describes pituitary enlargement due to intracranial hypotension. Reported previously primarily in the neurological literature, we present this case referred to endocrinology for evaluation of a pituitary mass. A 24-year-old male was referred to endocrinology for evaluation of pituitary enlargement without a hypo-enhancing lesion on magnetic resonance imaging (MRI). The main symptom reported was headache that was worse in the standing position and in the afternoon. He had no symptoms or signs of pituitary mass-effect, or hormone excess or deficiencies. Past medical history was relevant for a history of nerve schwannoma status post resection with subsequent spinal fusion. Biochemical evaluation of pituitary hormones was normal. Upon review of his pituitary MRI, other abnormalities seen were suggestive of intracranial hypotension. Based on his history and imaging findings, he was diagnosed with intracranial hypotension causing a “false pituitary tumor” rather than pituitary enlargement or abnormality. Further evaluation revealed multiple spinal leaks that were patched. His symptoms subsided within a few days of repair. Endocrinologists should be aware of the possible misdiagnosis of a pituitary mass due to intracranial hypotension. Referrals for pituitary masses or enlargement are increasingly encountered in the endocrine clinical practice, mostly related to incidental findings due to increased imaging for other reasons . PituitaA 24-year-old male was referred to our clinic for a pituitary tumor/mass discovered during evaluation of progressively worsening headaches over 5 years. He described them as a sensation of increased pressure or diffuse headache occurring in the afternoon, positional or orthostatic (worse while standing), particularly behind his eyes, precipitated by physical activity, and accompanied by photophobia, blurry vision, nausea, and projectile vomiting. Prior to presentation, evaluation for headaches at his local hospital included brain MRI that identified a pituitary tumor by report.Past medical history included a history of C7 to T1 nerve schwannoma status post resection and subsequent spinal fusion from C6 to T1, 8 years prior to his presentation. One year later, due to instrumentation failure, he underwent fusion extension from C4 to T3 as well as wound revision for poor wound healing.Review of systems was remarkable for intentional weight loss of 60 pounds in 1 year due to dietary changes and increased physical activity. He denied visual field deficits and did not have any symptoms or signs of pituitary hormone excess or deficiency. On confrontation, he had no evidence of visual field deficits.A repeat MRI with and without intravenous contrast at our institution revealed a mildly enlarged and diffusely enhancing pituitary with rounded convexity of the superior margin . There wEndocrine laboratory testing, including IGF-1, prolactin, FSH, LH, testosterone, TSH, free T4, ACTH, and cortisol levels, was normal.Upon further evaluation by neurosurgery, the history of positional or orthostatic headaches and prior spine surgeries and imaging findings suggestive of intracranial hypotension, it was suspected that this young man had cerebrospinal fluid (CSF) volume depletion syndrome manifesting with orthostatic headaches. Upon review of prior images, he had evidence of extradural CSF accumulation in the spine suggestive of CSF leak. This finding had been present since his 1-year follow-up imaging after spinal fusion and persisted at his 2-year and 3-year follow-up imaging. He also had evidence of brain sagging as early as 1 year after his spinal fusion. In the setting of diffuse pituitary gland enhancement without hormonal abnormalities, it was concluded that the pituitary gland findings on MRI were not a pituitary tumor but rather a pituitary enlargement due to anatomical adaptation from CSF volume depletion.Our patient underwent a dynamic computed tomography myelogram for leak localization and repair. The myelogram revealed CSF leaks at the T1-T2, T3, and T5 vertebral body levels. The study guided placement of multilevel blood patches at these sites. After the procedure, the patient's headaches completely resolved in 3 days.Although his spinal CSF leaks were likely related to his schwannoma and his prior surgeries, his history of poor wound healing raised the possibility of connective tissue disorder that predisposes to spontaneous CSF leak. He was evaluated by the genetics department to rule out common connective tissue disorders such as Marfan and various types of Ehlers-Danlos syndromes. Genetic testing was completed and was negative for relevant connective tissue disorders. Our patient did not undergo follow-up imaging at our institution to assess resolution of intracranial anatomic changes related to intracranial hypotension.Endocrinologists should be aware of the presentation of false pituitary tumors with enhancing pituitary enlargement without a hypo-enhancing lesion compatible with a pituitary tumor or evidence of pituitary hormone secretion or deficiency. Clinically, patients with CSF volume depletion characteristically present with orthostatic headaches that usually develop in the afternoon . CSF volEqually relevant to endocrinologists, it is important to note that opposite of what happens with intracranial hypotension, intracranial hypertension can present with empty sella. As a matter of fact, in patients with idiopathic intracranial hypertension, empty sella, partial or complete, is the most commonly reported MRI finding .— Empty A “false pituitary tumor” should be considered in patients presenting with pituitary enlargement and other imaging findings of intracranial hypotension but without frank evidence of a pituitary hypo-enhancing lesion.Patients with intracranial hypotension usually present without pituitary laboratory abnormalities since they do not have a pituitary space-occupying lesion.The cardinal symptom of intracranial hypotension is orthostatic headache.Past medical and surgical histories should be reviewed if there is a high suspicion for CSF leak, as localization of the leak site should be a priority before treatment.Treatment for intracranial hypotension includes patching of the leaks after which symptoms and imaging abnormalities resolve.All authors made individual contributions to authorship. M.D.H., A.C., and J.A. participated in the diagnosis and management of the case. M.D.H. and E.T. participated in preparing the manuscript. All authors reviewed and approved the final draft.