Esophago-respiratory fistula (ERF) refers to the formation of a pathological connection between the esophagus and respiratory tract. Acquired ERF is a rare but life-threatening diagnosis in adults. We describe a 79-year-old male who was admitted with an inhalation smoke injury. He was diagnosed with ERF by endoscopic visualization and sampling of the hyaline cartilage within the wall of the esophagus. Percutaneous endoscopic gastrostomy placement and conservative measures were effective in the management of ERF. Esophago-respiratory fistula (ERF) refers to the formation of a pathological connection between the esophagus and respiratory tract. Although rare in adults, it can cause significant morbidity and mortality in patients , 2. Malith day of admission, the patient developed acute melena and anemia while on a heparin drip for atrial fibrillation. Vitals were remarkable for hypotension and tachycardia, and his hemoglobin level was 6.5 g/dL. Esophagogastroduodenoscopy (EGD) was remarkable for a midesophageal ulcer and associated mucosal tear surrounded by granulation tissue. On the anterior wall of the esophagus, there was a yellow-tan foreign body that appeared to be embedded onto the esophageal wall  tube was placed the following day. Eventually, the pathology of the foreign body samples revealed fragments of hyaline cartilage, similar to that found in the tracheobronchial tree, which was concerning for ERF. Chest CT scan without contrast showed a narrowed left mainstem bronchus abutting the esophagus with foci of suspected extraluminal air along the left lateral margin of the esophagus . This waThe fistula had either resolved by the time of bronchoscopy or was too small to be visible during the procedure. The follow-up chest CT scan 11 days after stent placement no longer visualized the previously seen foci of air between the esophagus and left mainstem bronchi.Acquired ERF is a rare but life-threatening diagnosis in adults . A studyAlthough the initial symptoms of ERF can be insignificant, a delayed diagnosis can lead to severe complications such as pneumonia, life-threatening hemoptysis, and respiratory failure , 6, 15. The goal of treatment of ERF is to prevent severe complications and optimize nutritional status . SurgicaIn summary, we present a case of ERF due to inhalation burn injury, which was diagnosed by endoscopic visualization and sampling of hyaline cartilage within the wall of the esophagus. The most likely cause of ERF was inflammation and irritation of the bronchial mucosa secondary to smoke inhalation. PEG placement and conservative measures were effective in the management and healing of ERF. As in our case, the collaboration between pulmonology and gastroenterology services is essential for the diagnosis and management of this condition.


Metastatic neuroendocrine tumour (NET) to brain has been reported in 1.5–5% of patients with NETs. Differentiation between intracranial NET metastasis and meningiomas can cause a diagnostic dilemma. We present a symptomatic case of a 66-year-old male with a history of left-sided skull base mass. The diagnosis of a meningioma was made based on the MRI findings and clinical presentation. The patient received radiation and the mass remained stable on serial MRI images at follow-up visits. Five years after his initial presentation, the patient’s mass showed further growth. He also complained of worsening of his recent diagnosis of irritable bowel syndrome and fluctuations in his blood pressure. Surgical resection was performed, and histopathological features were consistent with moderately differentiated neuroendocrine tumour. Further evaluation with 68 Gallium-DOTATATE positron emission-computed tomography (Ga-68 PET/CT) showed metastatic disease involving the bones, lymph nodes, and liver without convincing evidence of the location of primary malignancy within the bowel loops or the pancreas. The patient was started on combination of capecitabine and temozolomide with partial response and significant improvement of his symptoms. This case highlights the clinical and radiological behaviour of intracranial NET that can mimic the diagnosis of meningioma. NETs can rarely metastasize to the brain with an incidence less than 5% of patients.4 Since meningioma is the most common type of primary intracranial tumors,5 differentiating between a dural metastasis and a meningioma is crucial for patient management.The incidence of neuroendocrine tumors (NETs) has increased in the United States over the last two decades.Herein, we present a case of intracranial NET metastasis that mimics a meningioma in a patient without known history of NET at the time of diagnosis.A 66-year-old, right-handed male presented to the neurology clinic with intermittent paresthesia over the left cheek and chin region. In addition, he experienced an intermittent double vision that was worsening in the 3 months prior. Magnetic resonance imaging (MRI) of the brain showed an enhancing mass centered in the left cavernous sinus, with extension to the brainstem homolaterally, causing mild displacement of the medial left temporal lobe without evidence of brain oedema. The mass was infiltrating the left Meckel’s cave. The clinical and radiological findings were consistent with a meningioma .After multidisciplinary discussion, stereotactic radiotherapy was recommended over surgical resection given the location of the tumour with proximity to carotid artery and cranial nerves, making complete surgical resection a challenge. Therefore, the patient received a total of 5040 cGy of radiation over 28 fractions.During the 6-month follow-up, the patient had significant improvement of his diplopia and facial dysesthesias. Follow-up MRIs over the next 3 years showed stable appearance of the mass, and the patient showed complete clinical neurological recovery. However, almost 5.5 years after radiation therapy, a repeat MRI scan revealed significant enlargement of the prepontine and the cavernous sinus lesion with no associated neurological symptoms . On reviAfter multidisciplinary team discussion, the decision of partial surgical resection was recommended due to brain stem compression. The patient underwent resection of his prepontine mass that had caused brainstem compression with residual mass located at the cavernous sinus. He showed a reasonable post-operative recovery, although he had some difficulties such as persistent ataxia and syncope-like episodes. Surprisingly, the pathology came back as well-differentiated neuroendocrine tumour, with Ki 67 index of 10% and diffusely strong positive immunostaining for synaptophysin, chromogranin, and CDX2. The patient was referred to the neuroendocrine oncology clinic. 68 Gallium-DOTATATE positron emission-computed tomography (Ga-68 PET/CT) scan evaluation revealed metastatic lesions affecting liver, bones, and lymph nodes without evidence of primary tumour . The patThe patient’s mass continued to show response on CAPTEM with partial response in the liver lesions and stable MRI appearance and no evidence of new metastatic lesions. On the patient’s last visit , he denied any new seizures, headache, or cognitive dysfunction. He showed few symptoms of peripheral neuropathy without any neuropathic pain or subsequent falls. The patient is completely independent in all his daily living activities.6 Only 1.3–1.4% of all brain metastases have neuroendocrine tumour origin.7–9 Bronchopulmonary NET is the most frequent subtype to metastasize to the brain.In this report, we present a case of metastatic NET to the dura with radiological features suggestive of meningioma. The majority of NETs arise in the gastrointestinal tract or in the bronchopulmonary system. Most metastases occur in the liver, bones and, lungs, while intracranial metastases are extremely rare.10 Stereotactic biopsy should be recommended in cases that the tumours location allows any neurosurgical operation. Otherwise, repeat MRI imaging to show stability or slow growth over 6 months to a year or systemic imaging with CT or MRI to rule out metastatic disease may be helpful if the diagnosis of meningioma is in doubt. However, low-grade NET metastases may grow at a similar slow rate as is seen with meningiomas, thus in this rare case repeat MRI may have falsely generated confidence in the diagnosis of meningioma. Fludeoxyglucose (FDG) uptake on PET imaging is not useful, due to high physiological baseline uptake of the brain.11 However, FDG PET is a very useful modality for evaluation of metastatic disease in general.Dural metastases in general are not uncommon. Moreover, radiological features of many tumours including dural metastases on CT/MRI imaging are often similar to that of meningioma resulting in a diagnostic dilemma.12 This is because meningiomas routinely express somatostatin receptors and are universally positive on SRI.13 For this reason, SRI is known to be useful in distinguishing residual meningioma tumour from postoperative scarring in the post-operative setting.14,15 However, SRI can show moderate-to-high activity in a variety of tumour types that are somatostatin-receptor positive, such as NETs..16 Therefore, while SRIs might not distinguish NET from meningioma, the presence of extracranial somatostatin receptor positive lesions would point to a diagnosis other than meningioma.The national comprehensive cancer network guidelines recommend considering Somatostatin receptor imaging (SRI), such as octreotide planar and SPECT scans or 68 Gallium-DOTATATE PET scans if diagnostic doubt regarding the diagnosis of meningioma exists based on MRI features.17One of the radiotracers that may be, surprisingly, useful for differentiating meningiomas from other tumours is Pittsburgh compound B (11C-PiB) which is used in PET imaging in dementia. We have previously reported a 100% specificity for meningioma with (11C-PiB), although further research is required before this technique becomes part of clinical practice.18 Deshaies et al also reported a case of meningioma-like mass that turned out to be NET.19 In both our case and their case, carcinoid symptoms were present. This should maintain a high index of suspicion for NET in any patient with carcinoid like symptoms and an intracranial mass. Clinical suspicion should be raised in any intracranial mass including a dural-based lesion or a benign-looking mass that has undergone surveillance for several years. Imaging of NETs is often challenging and usually requires a combination of both anatomic and functional techniques.20 Metastasis of carcinoid tumour to intracranial meningioma is also a possibility and has been reported in the literature21Functional NETs secrete hormones that could result in variety of symptoms such as watery diarrhoea, labile blood pressure, and flushing, also known as carcinoid syndrome. The presence of such symptoms can help in the diagnosis of an indeterminate intracranial mass. However, there is often a long interval between the start of symptoms and diagnosis.22 Our patient’s last follow-up was around 8 years following his initial presentation with dural metastasis. Despite the relative short median survival time of patients with intracranial NET metastases, longer survival over five years has been reported in a few cases.22–24 This prolonged survival may be partially explained by the well-differentiated subtype of the tumours of those who lived longer22In our case, the positive CDX2 seen on pathology that is usually seen in gastrointestinal and pancreatobiliary tract tumours, raised the suspicion of the diagnosis of an intracranial metastasis of unknown origin. This concern was further raised when extensive systemic metastases were seen on imaging. Grade 1/2 NET patients with brain metastasis have an estimated median survival of 15 months from the time of brain metastases diagnosis.25,26 Our patient showed stabilised intracranial tumour and shrinkage of his liver metastases on his temozolomide and capecitabine treatment. Temozolomide can cross-blood brain barrier and it has shown a valuable effect in treating NETs either alone or in combination with capecitabine.6,27 This may highlight the clinical benefit of temozolomide in patients with brain metastases from NETs.22 Several factors should be taken into consideration in tailoring the treatment lines including patient age, neurological state, histological differentiation, tumour extension location of the tumour, and its relation to the surrounding structures . Further research may be needed in prioritising the different treatment lines.There is still no consensus regarding the standard treatment of intracranial NETs. Treatment options include surgical resection, somatostatin analogs including 177 Lutetium DOTATATE, whole-brain radiation or intensity-modulated radiation therapy. A prior study suggested comprehensive treatment including surgical resection and chemo-radiotherapy is expected to offer a longer tumour-free survival than surgical treatment alone.Intracranial NETs metastases are extremely rare. Carcinoid like symptoms and advanced imaging techniques could help in differentiating neuroendocrine metastases from meningioma. Multidisciplinary care should tailor a comprehensive treatment for patients with intracranial NETs for the best possible outcomes.Dural neuroendocrine tumour (NET) metastasis is extremely rare, and its differentiation from meningioma can cause a diagnostic dilemma.Clinical suspicion should include intracranial benign-looking mass; even that had undergone surveillance for several years, in patients with watery diarrhoea, flushing, or labile blood pressure symptoms.While Somatostatin receptor imaging might not distinguish NET from meningioma, the presence of extracranial somatostatin receptor positive lesions would point to a diagnosis other than meningioma. Pittsburgh compound B may play a promising role in the future.Multidisciplinary care should tailor a comprehensive treatment for patients with intracranial NETs for the best possible outcomes.


Teaching Point: Nasal and nasopharyngeal Rosai-Dorfman disease is a rare cause of nasal obstruction. A 33-year-old male presented to the otolaryngologist with persistent complaints of nasal blockage. On clinical examination a nasal septum deviation was seen and was confirmed by subsequent cone-beam computed tomography (CT) of the sinuses, which also showed additional soft tissue masses in the anterior nasal septum, in the left-sided posterior nasal cavity, and at the level of the left torus tubarius . SuboptiSinus histiocytosis with massive lymphadenopathy, also known as Rosai-Dorfman disease, is a rare condition of idiopathic sinus histiocyte proliferation with incorporated phagocytosed lymphocytes. The etiology is not completely understood. A disturbed immunologic response against certain pathogens, more likely viruses, is presumed. This disease presents most frequently in the first two decades of life  and has Rosai-Dorfman disease clinically presents with non-malignant and painless cervical lymphadenopathies or fever , which cNon-contrast-enhanced CT will visualize a mass without further possibility to make a differentiation. The lesions have a high attenuation on FDG-PET CT, have an iso- to hypo-intense signal on T1- and T2-weighted MR images compared to grey matter, and homogeneously enhance after contrast administration. All of these imaging findings were present in this case.Rosai-Dorfman disease is a benign pathology with an overall self-limiting course. Therefore, treatment is not necessary in most cases. Surgical intervention is executed for biopsy – with subsequent histological confirmation of the diagnosis – and rarely at local lesions to reduce concordant symptoms .


A fistula that connects the bowel to other organs, such as the urinary bladder or small intestine, is a relatively frequent complication, often associated with inflammatory diseases such as diverticulitis, Crohn's disease, colorectal cancer, or lymphoma. Splenocolic fistula is an extremely rare condition described in the literature. It can occur in cases of splenic tumors, including splenic diffuse large B cell lymphoma. We report the case of an 82-year-old man who presented with melaena, worsening asthenia, hypotension, and abdominal pain in the left flank and the ipsilateral lumbar region. Ultrasound and computed tomography documented splenomegaly, thickening of the splenic flexure of the colon, and the presence of a fistulous passage between the colon and the splenic hilum. The diagnosis of lymphoma was made following laparotomy and caudal splenopancreatectomy. Due to the aggressive clinical behavior of this type of lymphoma, splenectomy is the main treatment in patients with splenomegaly, abdominal pain, and tumor expansion. A colonic fistula is an abnormal tunnel that connects the colon to the skin or an internal organ. When abnormal communication occurs between the colon and spleen, a splenocolic fistula develops, a rare condition described in the literature . It has BCL6 gene, but translocations involving the BCL2 or MYC genes can also occur (Diffuse large B-cell lymphomas (DLBCLs) are lymphoid neoplasms characterized by rapid, aggressive growth, making up about 40% of B-cell malignancies . They arso occur .DLBCLs are characterized by extensive lymph node involvement, systemic symptoms, and, in 40% of cases, an extranodal localization . Clinically, they determine palpable splenomegaly, abdominal pain, and signs of tumor expansion in cases of hepatic and splenic involvement . In the Intra-abdominal fistula formation is frequent. It is often due to inflammatory gastrointestinal diseases or malignant tumors. It is known that fistulas occur mainly in various kinds of lymphoma rather than carcinomas . We repoAn 82-year-old man presented with worsening asthenia, hypotension, abdominal pain in the left flank and ipsilateral lumbar region, and melaena. He had been reporting for about 1 year the onset of physical discomfort, anemia, abdominal pain in the left quadrants, and tendency to be constipated. He had already undergone an esophagogastroduodenoscopy and a pancolonoscopy, which had not shown any significant pathologies. On physical examination, he was afebrile and had a tender abdomen on deep palpation on both the left side and the ipsilateral lumbar region. Laboratory tests were significant for leukocytosis , anemia (hemoglobin 10.50 mg/dl), and thrombocytosis . The patient was given fluid resuscitation and broad-spectrum antibiotics. Ultrasonography of the abdomen showed splenomegaly (DT 15.32 cm), presence of a hypoechoic area with a pyramidal shape (base to the splenic hilum and apex to the subcapsular site), and intralesional air, indicating the possible presence of a fistula. A thickening of the splenic flexure of the colon with a fistulous tract that infiltrated the hilum of the spleen was evident.An abdomen CT scan showed splenomegaly and concentric wall thickening of the descending colon near the splenic flexure, with fistulous communication between the colon and the medial surface of the spleen. A hypodense area with air bubbles was also evident . BecauseKlebsiella pneumoniae. Specific antibiotics were then administered. Once the infection had cleared, the patient left the clinical center and began chemotherapy in association with immunotherapy for cancer treatment, according to the Clinical Practice Guidelines. The regimen is called R-CHOP . The patient is still being treated. Three months later, an ileostomy closure surgery was done. The patient underwent laboratory tests and radiological examinations every 3/6 month for 1 year and is still undergoing cancer therapy.Immunohistochemical analysis revealed CD20, CD43 , CD22, PDLBCL is classified within the non-Hodgkin lymphomas (NHLs), accounting for about 40% of all NHL cases worldwide , 12. It The definition of primary extranodal lymphoma is controversial, especially in those patients who have both nodal and extranodal involvement. According to Krol et al. , primaryNHL can start anywhere in the body where lymph tissue is found, and for this reason, the digestive tract, where many organs have lymph tissue, is often involved. This is particularly true for DLBCL.Intra-abdominal fistula formation is a rare event. Fistulas are usually the result of intra-abdominal inflammation or infection or the consequence of malignant disease, or they develop as a complication of abdominal surgery or trauma. Our search of the literature led to the finding of a previous report of colonic fistula formation, a gastrocolic fistula , and, suFistula formation can be a consequence of different pathologies, such as Crohn's disease , divertiIt is known that a fistulization is a rare event in malignancies such as gastric or colonic cancer . The devBoth in splenocolic fistula and other cases of colic fistulas associated with malignancies, necrosis is often thought to be the ultimate cause of fistula formation. Necrosis is accompanied by ulceration and extensive coagulation. In most cases, surgery, after preoperative imaging, allows removal of the fistulous tract by resecting the organs, or the affected portions of them, involved in the pathological process.In our case, the patient presented with a rare splenocolic fistula. Surgical intervention was urgent and particularly useful to diagnose the fistula associated with DLBCL and to confirm the splenic localization. Similar cases have been reported in the literature. In particular, Katayama et al. described a case of a fistula of the colon in non-Hodgkin's lymphoma of the spleen . NatschiIn conclusion, we report an exceedingly rare condition in which the striking feature was the presence of a splenocolic fistula associated with DLBCL in an elderly patient. Our findings suggest that lymphoma cells had infiltrated from the spleen to the colon. As with any severe chronic pathology, early detection and recognition of the disease process were necessary to prevent complications. It is crucial to consider splenocolic in the differential diagnosis when a patient present with sepsis, splenomegaly, abdominal pain, and tumor expansion.


Paraphimosis is a urologic emergency that requires prompt diagnosis to avoid potential morbidity. Diagnosis is made clinically and imaging findings have not been described. We will present the case of an 84-year-old man with a history of metastatic prostate cancer and prior pelvic external beam radiation therapy who presented to the emergency department with urinary retention. A urethral Foley catheter was placed for bladder decompression. He subsequently developed painful penile swelling and was found to have iatrogenic paraphimosis. Retrospective review of his contrast-enhanced CT of the abdomen and pelvis performed while in the emergency department before hospital admission revealed the relatively thickened, hypoattenuating prepuce located proximal to the corona of the glans penis, consistent with the clinical diagnosis. We will examine the imaging findings in this case and propose the novel “wet collar” sign to suggest the diagnosis of paraphimosis on CT. Paraphimosis is a urologic emergency and prompt detection and treatment must occur in order to avoid morbidity, which can ultimately include necrosis of penile tissues and the necessity for operative management -2. It isAn 84-year-old man with a complicated medical history to include metastatic prostate cancer previously treated with pelvic external beam radiation therapy whose course was complicated by a right ureteral stricture treated with an internal ureteral stent presented to our emergency department with 14 hours of urinary retention. He was tachycardic but afebrile. A Foley catheter was placed by the nursing staff 18 hours after symptom onset which yielded one liter of urine. Concurrent blood analysis indicated leukopenia and neutropenia. Urinalysis demonstrated pyuria. Blood and urine cultures were obtained. Given his clinical presentation, an antibiotic regimen was initiated for presumptive bacteremia with a urinary source, and a contrast-enhanced CT of the abdomen and pelvis was obtained to assess for any evidence of other complications, including hydronephrosis.CT showed a mildly hydronephrotic right kidney with a delayed nephrogram and asymmetric cortical atrophy, as well as an appropriately positioned internal right ureteral stent and the presumed site of stricture within the mid ureter; however, the acuity of these findings could not be determined as comparison images were not available. The urinary bladder was adequately decompressed. There were otherwise no immediately suspicious genitourinary findings, and eventually the patient was admitted to the internal medicine service to continue antibiotic treatment while awaiting results from the blood and urine cultures.The following afternoon, almost 24 hours after Foley catheter placement, he was noted to have penile swelling and complained of sensitivity to touch, stating that his hospital gown and sheets were causing him discomfort when they rubbed against his genitals. At that time he was documented to have iatrogenic paraphimosis for which the primary team initially planned to prescribe a course of non-steroidal anti-inflammatory medicine to decrease penile swelling and allow the prepuce to reduce. However, early the next morning, nearly 35 hours after Foley catheter placement, urology was consulted due to progressive penile swelling and pain, and his paraphimosis was manually reduced. He was later discharged from the hospital after his blood and urine cultures were negative for greater than 48 hours. His episode of acute urinary retention and pyuria was likely multifactorial but upon further investigation was found to be a recurring problem for which he sought treatment with his primary medical facility.Upon further discussion between urology and radiology, his CT was later reviewed with respect to his diagnosis of paraphimosis. Axial Video , coronalParaphimosis is characterized by entrapment of the preputial skin behind the corona of the glans penis which eventually tightens around the penile tissues, leading to vascular compromise and causing edema and penile pain . IncidenUltrasound and MRI have been utilized successfully for multiple disorders affecting the male external genitalia, such as traumatic penile fractures, penile Mondor’s disease, and Peyronie’s disease . UltrasoParaphimosis is a condition that should be readily and easily diagnosed by physical examination. First-line conservative treatment measures involve an attempt at manual reduction of the prepuce . As a reIn our case, we identified signs of edema as they present elsewhere in the body, mainly evidenced by relative hypoattenuation and thickening compared to adjacent normal tissues. Additionally, with the benefit of retrospective knowledge of the diagnosis, we were able to identify the abnormally retracted position of the prepuce with associated thickening and apparent hypoattenuation which we termed the “wet collar.” This presents a few challenges, however, as normal data have not been defined in this context. For example, preputial entrapment requires a dynamic assessment on the physical examination, and CT represents an image at a single point in time. The degree of preputial retraction which could be considered normal has not been elucidated. The level of relative hypoattenuation that lies within an acceptable range has not been described. Ultimately, the benefits of a comprehensive radiologic investigation into developing an optimal imaging protocol to assess paraphimosis must be weighed against the utility of the low-cost gold standard: the history and physical examination. However, this diagnosis may initially go overlooked in certain patient populations, and a radiologist’s knowledge of paraphimosis may allow its incidental detection for improved clinical outcomes.Paraphimosis is an emergent condition and imaging has no role in its routine evaluation. The authors do not advocate a change in practice at imaging centers or the development of imaging protocols for the detection of paraphimosis on imaging as an initial diagnostic test. However, it is a diagnosis that can initially go unrecognized, and patients will undoubtedly undergo radiologic studies for other reasons which may have a chance of imaging the pathology and allowing the radiologist to suggest the diagnosis. This can potentially lead to improved care and positively affect the patient’s condition. While the imaging findings we have presented and the “wet collar” sign we have proposed would require investigation to detail the breadth of normal imaging appearances of the prepuce and glans penis and to elucidate the performance profile of the “wet collar” sign as a marker for disease, we hope that with our report we have made the first steps toward bringing paraphimosis into the radiologist’s diagnostic awareness and given him or her factors to consider should he or she encounter it at the workstation.


The lesion was completely removed in piecemeal fashion through a tiny incision on the sulcus, which did not cause any new neurological deficits. The modified Rankin Scale improved from 4 before the surgery to 1, and the patient had no recurrence during the 2 years of follow-up. The advantage of this access and the dissection techniques for this challenging lesion are introduced, based on our experience with more than 230 surgeries of brainstem cavernoma.Hemorrhagic brainstem cavernous malformations carry a high risk of progressive neurological deficits owing to recurrent hemorrhages and hence require complete surgical resection while minimizing damage to the dense concentration of nuclei and fibers inside the brainstem. To access lesions inside the lower pons, the senior author (H.B.) has preferred to approach the lesions via the “perifacial zone” through the pontomedullary sulcus from the inferior surface of the pontine bulge for more than 20 years.https://youtu.be/0H_XqkQgQ9I.The video can be found here:  This video will demonstrate the microsurgical resection of a brainstem cavernous malformation via the “perifacial zone,” which is through the pontomedullary sulcus using the retrosigmoid supracondylar approach.The patient is a 32-year-old man presenting with progressive headache, diplopia, sensory impairment on the right side, and gait disturbance. Neurological examination revealed complete left abducens paresis, hemihypesthesia of the entire right side of the face and body, and gait ataxia.MRI displayed a 13-mm lesion inside the lower pons with a hemosiderin rim, and the so-called popcorn pattern of variable image intensities, which indicated hemorrhagic cavernoma.The lesion was too deep to access through the middle cerebellar peduncle from the lateral surface of the pons or through the floor of the fourth ventricle from the dorsal surface of the pons. In such cases, we have preferred to approach the lesion through the pontomedullary junction from the inferior surface of the pontine bulge, which is called perifacial zone, for over 20 years. This route also provides an optimal trajectory of access along the long axis of the lesion, as we often encounter lesions on the superodorsal-to-inferoventral axis along the longitudinal fibers, including the medial lemniscus, as seen in this case.The retrosigmoid supracondylar approach and access through the perifacial zone were planned with the aim of complete lesion removal under MEP, SEP, ABR, and facial electromyograms. The half-sitting position was selected to obtain optimal caudal-to-cranial viewing trajectory.In a half-sitting position with slight head flexion, a C-shaped retroauricular incision was made. A retrosigmoid craniotomy was performed and the condylar fossa was drilled out. The dura was incised along the sigmoid sinus and caudal edge of the craniotomy.After dural opening, the CSF was released from the cerebellomedullary cistern. The PICA, lower cranial nerves, and CNs VII and VIII were carefully dissected. The flocculus was lifted up after its dissection from the lower cranial nerves, and the choroid plexus was coagulated to decrease its size. The root exit zone of the facial nerve was precisely confirmed by electrical stimulation. Since the space between CNs IX and X did not provide adequate exposure in this case, we utilized the space between CNs X and XI. Here you can see the pontomedullary sulcus, on which the vein of the pontomedullary junction coursed. The perforating arteries were carefully separated. Clear exposure of the inferolateral surface of the pontine bulge revealed slight yellowish discoloration.The pia was coagulated through the space between the rootlets of CNs X and XI. About 2-mm cortical incision was made in a puncture fashion just below the exit zone of CN VII and ventral to CN IX.The lesion cavity in the lower pons was located right underneath the incision, and the hematoma, including the fresh clot, was evacuated. The entire surface of the cavernoma was dissected at the appropriate cleavage plane from the healthy parenchyma using bipolar forceps and microscissors, and a cottonoid was placed between the separated portions of the malformation and parenchyma. As in most cases, a brain retractor was not applied to the brainstem. Piecemeal removal of the cavernoma was performed through the tiny cavity. To prevent local vasospasm, nimodipine solution was used for irrigation.The lesion bed was carefully examined to confirm the absence of any residual cavernous malformations, which is a common cause of recurrence. You can see the size of the cortical incision and complete preservation of all the surrounding structures, including the cranial nerves, without any damage. No associated venous anomalies were seen in this case.Extubation was performed the next day and the patient developed no new neurological deficits. His gait disturbance disappeared within 3 weeks after the surgery.Postoperative MRI confirmed complete resection of the lesion and significant reduction of the mass effect. Also, you can see the surgical corridor through the pontomedullary sulcus.The patient had no new neurological deficits or recurrence during the 2 years of follow-up after the operation.The mRS improved from 4 before the surgery to 1 on the final follow-up day.


Sporadic gastric foveolar-type adenomas are extremely rare and are usually small, flat or slightly raised lesions that occur in the oxyntic mucosa.We reported here a case of a 70-year-old female with a sporadic gastric foveolar-type adenoma occurring in the mucosa at the junction of the gastric body/antrum. The adenoma was a protruding lesion of 2 × 1.8 cm sized, causing symptoms of upper gastrointestinal bleeding, and the basal and surrounding mucosa showed pseudopyloric gland metaplasia without atrophy, intestinal metaplasia, H. pylori infection, or active inflammation. It had somatic mutations in both APC and KRAS genes.This is the first reported case of a large sporadic gastric foveolar-type adenoma that occurred in the mucosa of pseudopyloric gland metaplasia and with Gastritis Cystica Profunda, which modify our understanding of the morphological features and molecular underpinnings of this type of lesion. Sporadic gastric foveolar-type adenoma is an extremely rare gastric lesion, usually a small, flat polypoid lesion that occurs in the oxyntic mucosa . CompareA 70-year-old female patient was referred to our hospital for "hematemesis for 10 days". The volume of hematemesis was about 150 ml. The patient had melena, but no fresh blood in the stool. Gastroscopy showed there were two wide-base polyps in the greater curvature of gastric body/antrum junction (about 50 cm from the incisor teeth). The larger one was 2 × 1.8 cm in size, with a papillary or gyrus-like appearance in the surface; the smaller one was 1 × 1 cm in size and was slightly flat  eradication therapy. She had no family history of tumors and six offspring were in good health.The patient underwent endoscopic mucosal dissection (ESD) on the larger polyp at the junction of the gastric body/antrum and endoscopic mucosal resection (EMR) on the other smaller polyps. The operation process went smoothly, and the patient's postoperative condition was stable.The ESD specimen of the larger polyp at the junction of the gastric body/antrum showed a broad-based protruding lesion with a size of 2.3 × 1.3 × 1.3 cm. There were no ulcers and necrosis on the surface. The cut surface was solid, gray-pink, and soft in texture. The lesion was confined to the mucosal layer and the nearest distance to the mucosal lateral margin was 0.5 cm. The smaller polyp at the junction of the gastric body/antrum and the polyp in the subcardia area were both sessile polyps, the size of which was 0.8 × 0.5 × 0.3 cm and 0.5 × 0.4 × 0.2 cm, respectively.The larger polyp at the junction of the gastric body/antrum was a 0-Is protruding type lesion Fig. a. The tuThe immunohistochemistry staining of MUC5AC showed diffuse cytoplasmic staining Fig. a, while A diagnosis of gastric foveolar-type adenoma with low-grade dysplasia was rendered; the base of the tumor was accompanied by gastritis cystica profunda (GCP).The other two polyps at the junction of the gastric body/antrum and the subcardia area were similar in morphology. They were composed of foveolar epithelium without dysplasia, arranged with enlarged and twisted glands accompanied by stroma edema and inflammatory cell infiltration. The morphology of these two lesions was consistent with gastric hyperplastic polyps.A somatic genetic test was performed on the larger polyp at the junction of the gastric body/antrum. We analyzed 20 genes related to gastric tumors and polyps through next-generation gene sequencing (NGS) and found APC and KRAS gene mutations in the adenoma tissue. See “Appendix” for the list of genes tested and specific mutation forms.Most of the gastric foveolar-type adenomas are related to hereditary tumor syndromes, such as familial adenomatous polyposis (FAP) and gastric adenocarcinoma and proximal gastric polyposis (GAPPS), and only a few sporadic cases have been reported , 5. FAP Some researchers believe that most adenomas are mucosal polypoid dysplasia that develops from chronic gastritis . Except Sporadic gastric foveolar adenomas are extremely rare. The cases reported before are usually small, flat, or even depressed lesions located in the mucosa of gastric oxyntic glands. We reported here a protruding lesion with a maximum diameter of 2.3 cm, which caused symptoms of gastrointestinal bleeding. Recently, there is a study about a type of sporadic gastric foveolar-type adenoma with a raspberry-like appearance , which iThe case also showed gastritis cystica profunda at the base of the tumor. The occurrence of gastritis cystica profunda is considered to be related to iatrogenic  infection, chronic ischemia, or inflammation, showing prolapse of the mucosa and glands to the muscularis mucosa and even the submucosa . The deeIn summary, we reported a valuable case of sporadic gastric foveolar-type adenoma which occurred at the junction of gastric corpus/antrum in an elderly patient. The tumor was large in size and caused symptoms of upper gastrointestinal hemorrhage and accompanied by gastritis cystica profunda. This is different from the previously reported cases, which expands the clinical manifestations of gastric foveolar-type adenoma and increases the understanding of its morphology and molecular basis.


Biliary Ascariasis occurs when Ascaris lumbricoides worms invade the biliary system. It may cause biliary obstruction, cholangitis, cholecystitis, or acute pancreatitis. We report a case of a 37-year-old female patient who presented with a history of upper abdominal pain, nausea, vomiting, and weight loss for two weeks. Ultrasound showed dilated common bile duct with linear tubular echogenic structure in the common bile duct and bowel loops. Endoscopic Retrograde Cholangio pancreatography (ERCP) revealed large adult worms confirming the diagnosis of Ascariasis. Ascariasis, a helminthic infection caused commonly by Ascaris lumbricoides, infects about 33% of the world’s population making it one of the most common gastrointestinal tract parasitic infestations . Humans A 37-year-old lady with no chronic medical illness presented with two weeks history of episodes of upper abdominal pain. The pain was dull in nature, non-radiating, and worsening with movements and change in position, which did not get relieved with antacid or analgesics. It started gradually without a specific pattern and sometimes reached 10/10 in severity. She also experienced anorexia for one month, nausea and vomiting for three days duration. There was a history of significant loss of weight of 5 kg over the last month. She recently came from the Philippines and working as a housemaid. The patient denied any history of diarrhea, constipation, hematemesis and melena.Her vitals were within normal limits. On examination, the patient was conscious and oriented. The abdomen was soft, and lax, with severe epigastric tenderness. There was no palpable mass or hepatosplenomegaly. Ultrasound abdomen was done which showed dilated common bile duct (CBD), measuring 9 mm. There was a linear tubular echogenic structure noted in the common bile duct. There was another linear echogenic structure noted within the bowel loops in the epigastric region and the right lumbar area. Other major visualized abdominal organs were unremarkable. These linear echogenicities were likely due to ascariasis Figures -1C.The gastroenterology team was consulted and advised ERCP. On ERCP, the stomach showed a large adult worm, which was stuck to the suction channel and removed out. The first and second parts of the duodenum showed multiple big adult worms of ascariasis. Cholangiogram showed one big adult worm in the left hepatic duct and proximal CBD. The guide wire was passed selectively to the left hepatic duct and the adult worm was removed with balloon weep Figures , 2B. CBDThe patient was given 400 mg albendazole tablet, weekly once for two weeks, intravenous fluid became symptomatically better, and advised to follow up in the GI clinic for stent removal after deworming.Ascariasis infection, a global parasitic infection by the organism “Ascaris lumbricoides”, is most commonly distributed in tropical and subtropical areas. The most common cause of the spread of Ascariasis infection is poor hygienic precautions and poverty . Humans The clinical presentation of Ascariasis infection is either symptomatic or asymptomatic with the vast majority of symptomatic infection occurring in case of a significant parasitic load. The symptoms develop according to the phase of the parasite’s life cycle; larvae in the lungs are manifested by pulmonary symptoms, while adult worms in the bowels are manifested clinically by non-specific abdominal symptoms, including abdominal pain, nausea, vomiting, anorexia and diarrhea, like our case. Adult worms’ transfer through the gastrointestinal tract may lead to complications, host present mostly with a complaint of an acute abdomen. When worms travel through the ampulla of vater to the biliary tract, pancreas, and liver, liver abscess, acute cholecystitis, and acute pancreatitis might occur . The bulThe diagnosis of Ascariasis infection in developed countries cannot depend exclusively on the history, clinical features, and stool analysis. Many medical imaging methods have been well-evolved, such as ultrasonography, considered an initial and reliable diagnostic tool for biliary Ascariasis . The worUltrasound is an easy, highly sensitive, strongly specific, safe, and non-invasive modality and it has been used as the first imaging tool for biliary Ascariasis diagnosis, especially when the worm is moving, the typical shape of it on ultrasound is a linear echoic with no shadowing structure. Three modalities are available for the treatment of Ascariasis: non-surgical management, surgical management for patients with poorly responsive to the medications or those who are complaining of severe infection, and endoscopic management. ERCP represents an endoscopic diagnostic and therapeutic method, which has been efficiently used to visualize and remove the worms from the biliary tract at the time of diagnosis .Literature research reported about a Korean case of biliary Ascariasis infection, ERCP was used as a confirmatory diagnostic and therapeutic method, the worm was extracted through the ampulla of the vater by using the balloon sweeping method, the treatment was completed with the administration of medical management using the drug “albendazole”, the patient was discharged without any complication . Our patThe chief complaints of Ascariasis infection are non-specific abdominal symptoms; as a result, it may be easily misdiagnosed with other diseases. Therefore, in endemic areas, the parasitic infection should be one of the differentials of an acute abdomen. Ultrasonography has achieved great success in confirming the diagnosis. Radiologists need to be aware of the radiologic features and manifestations of parasites. ERCP in association with anthelmintic drugs has been properly documented as the mainstay treatment modality.


Osteosarcoma, a malignant bone tumor, rarely occurs in the sternum, especially the fibroblastic type, and is associated with poor survival. This case report describes a patient with a neoplasm of the sternum who underwent tumor resection 3 times and reconstruction twice because of the high risk of recurrence.A 60-year-old Chinese man presented with a 3-cm palpable bulging mass located in front of his sternum. Chest computed tomography (CT) revealed an anterior chest wall neoplasm with sternal destruction.Pathological examination revealed that the mass was a low-grade malignant primary fibroblastic osteosarcoma.Locking plates were used for chest wall reconstruction, demonstrating good structural stability and economic applicability. Regarding the ineffectiveness of current therapies, whole-exome sequencing was conducted, and no targets matched any of the currently available agents.No recurrence was found on regular reexamination.Surgery is the first choice of treatment for patients with primary fibroblastic osteosarcoma of the sternum. The reconstruction-locking plate is a good alternative for chest wall reconstruction. Whole-exome sequencing can shed new light on this uncommon disease and help identify novel therapeutic targets. However, its occurrence in the chest wall, especially in the sternum, is infrequent. The average age of patients with osteosarcoma of the sternum is approximately 42 years at diagnosis.2A 60-year-old man presented with a 3-cm anterior chest neoplasm, without any positive symptoms. The mass was misdiagnosed as a benign tumor at a local hospital. Local excision was performed, and the pathological results revealed a solitary fibrous tumor. One year later, the neoplasm relapsed and the patient was referred to our department. Physical examination demonstrated a 3-cm palpable bulging mass located directly in front of the sternum. Routine blood tests, biochemical tests, tumor markers, and other laboratory examinations revealed no abnormalities. Chest computed tomography (CT) revealed an 8.5-cm anterior chest wall neoplasm with 3-cm deep infiltration of the sternum Fig. A, and whUnfortunately, the anterior chest wall neoplasm relapsed eight months later. Chest computed tomography (CT) revealed a 3-cm neoplasm in the anterior chest wall and residual mesosternum with a 2-cm deep infiltration , BBS9 (Bardet-Biedl Syndrome 9), Alpha-Sarcoglycan (SGCA), and GALK1 . These 4 SNVs are missense mutations located in exons. Protein function prediction was performed using SHIF, Polyphen2, and MutationTaster, all of which were deleterious. The GERP++ scores of these genes, analyzed by dbNSFP version 3.0, were >2, indicating that these genes may be located in conserved regions.Concerning the negative consequences, whole-exome sequencing was conducted to determine whether there are any well-known gene mutations for which we presently have target agents, with ethics committee approval and informed consent obtained from the patient. The results revealed 314 single-nucleotide variants (SNVs), including 9 synonymous mutations, without currently available targets. This detection revealed 4 potentially meaningful SNVs, including Since the last operation, the patient was accessed every 6 months for one year and a half. The patient recovered well and was able to engage in light labor. Physical examination and CT did not reveal any recurrence.3Osteosarcoma typically occurs between the ages of 10–14 years and affects the long bones, including the femur, tibia, and humerus. Five of the 6 patients underwent extensive surgical resection with or without neo-oradjuvant treatment. Only 1 case chose chemoradiotherapy because of distal metastasis. Overall, surgery is the first choice unless the patient presents with metastasis.We reviewed the existing case reports of osteosarcoma of the sternum Table . A total,6–9 FiveHerein, we report a case of primary fibroblastic osteosarcoma of the sternum. In this case, the patient presented with a superficial painless tumor, chest CT scanning indicated tissue infiltration, and whole-body bone scan showed no bone metastasis. This might be the reason why the local hospital misdiagnosed the case as an SFT. We performed reconstructions twice using locking plates rather than 3D printing materials owing to the financial constraints of patients. Fortunately, these structures were stable. This could be the ideal surgical option for patients without medical insurance.Given the poor prognosis of this rare disease, we performed whole-exome sequencing, which has not been performed previously. The results indicated 314 SNVs, including nine synonymous mutations. Although no targets correspond to currently available agents, we provide novel perspectives and possible targets that require additional study to elucidate their functions in this disease.Data curation: Shaozi Fu.Investigation: Yile Zhou.Writing – original draft: Shaozi Fu, Yile Zhou.Writing – review & editing: Heyun Xu.


CHEK2) p.H371Y germline mutation. A 45‐year‐old woman presented with a uterine mass. The transvaginal sonographic examination confirmed a 4.4 cm × 3.7 cm, cystic and solid, mass located on the right side uterine wall near isthmus. First, laparoscopy with the neoplasm resection was carried out. Based on morphological and immunohistochemical characteristics of tumor cells that expressed glial fibrillary acidic protein (GFAP), S‐100, and vimentin, the tumor was diagnosed as an ependymoma. After that, she underwent a laparotomic total hysterectomy, bilateral salpingo‐oophorectomy, and lymphadenectomy. Furthermore, we performed next‐generation sequencing (NGS) of the patient's resected tumor tissue and peripheral blood and identified a novel CHEK2 p.H371Y germline mutation. Following surgery, the patient received oral tamoxifen (10 mg 2/day) and followed by letrozole (2.5 mg/day) for 6 months. The patient remained disease‐free after 4 years of follow‐up. Conceivably, CHEK2 p.H371Y is a driving gene for the development of extra‐CNS ependymoma.Ependymomas arise from ependymal cells lining the ventricles and central canal of the spinal cord and can occur throughout the whole neuraxis. The lesion rarely occurs in extracranial or extraspinal regions, particularly in the uterine broad ligament. Thus, for the pathogenesis of nonsacral extra‐central nervous system (CNS) ependymomas remains elusive. Here, we describe a rare case of primary uterine broad ligament. ependymoma with cell‐cycle‐checkpoint kinase 2 ( CHEK2, also known as CHK2) p.H371Y germline mutation. The patient was treated with surgery, followed by endocrine therapy.Ependymomas predominantly develop from neuroectodermal organs. Primary extraneural ependymomas are rare and typically defined as a sacrococcygeal, pelvic, or extrapelvic ependymoma based on tumor location. The most common site for extraneural ependymomas remains the sacrococcygeal region, with more than 50 reported cases.GFAP, vimentin, PAX‐8, S‐100 protein, P16, epithelial membrane antigen, and AE1/AE3. Among them, GFAP, S‐100, vimentin expression is characteristic of the tumor  and estrogen receptor (ER) in many tumor cells. Negative staining for P53, calretinin, a‐inhibin, cytokeratins 7 and CD10 was observed. Postoperative positron emission tomography and computed tomography (PET/CT) scans confirmed no residual lesions and distant metastasis. Furthermore, using PCR and NGS, a novel CHEK2 p.H371Y germline mutation, which specifically referred to H371Y hybrid germline mutation caused by the single base substitution of exon 11 of CHEK2 gene, was detected in the DNA obtained from tumor tissue and DNA peripheral blood  arm of chromosome 22. CHEK2 is a tumor suppressor gene that encodes a multifunctional kinase that is activated essentially by the ataxia telangiectasia‐mutated (ATM) protein in response to DNA double‐strand breaks.CHEK2, in turn, phosphorylates several critical cell‐cycle proteins, including p53, Cdc25, and BRCA1, which trigger cell‐cycle arrest, apoptosis, and the activation of DNA repair.CHEK2 p.H371Y mutation is located within the activation loop of the CHEK2 protein kinase domain, which is crucial for the activation of CHEK2 in response to DNA damage. Further functional analysis revealed that the CHEK2 p.H371Y mutation causes a dramatic decline in CHEK2 activity and is a pathogenic mutation. CHEK2 p.H371Y confers a 2.43‐fold increase in breast cancer risk among Chinese women.CHEK2 p.H371Y mutation may share some similarity to BRCA1 mutation; therefore, carriers of CHEK2 H371Y mutation may be considered potential candidates for treatment with poly ADP‐ribose polymerase‐1 inhibitors.NGS of DNA obtained from peripheral blood and tumor tissues samples revealed that CHEK2 p.H371Y mutation in extra‐CNS is novel to the molecular profile of CNS ependymoma. However, due to limited tissue availability and lack of funding for more in‐depth molecular testing, whether CHEK2 p.H371Y is a pathogenic gene for the development of extra‐CNS ependymoma is currently uninvestigated. Thus, future studies are needed to confirm the gene expression profile of extra‐CNS ependymomas, which may provide insight into the cellular origin and molecular drivers of these rare tumors. In summary, primary uterine broad ligament ependymomas are extremely rare kinds of extra central nervous system ependymomas and there is no one unified standard treatment for this. Thus, to better understand the molecular pathogenesis of this rare neoplasm, further molecular profiling studies are needed to guide clinical therapy. The finding of CHEK2 p.H371Y might conceivably become a major breakthrough for the treatment of extra‐CNS ependymomas.The finding of None declared.Jiaxin Yin was mainly responsible for drafting of the manuscript and revising. Min Yao was responsible for supporting pathology images. Xiaofeng Cong was responsible for acquisition of data and follow up. Hongdi Lu contributed analysis and Interpretation of data. Pengfei Cui contributed supervision. Ziling Liu contributed conception and design.Figure S1 Macroscopic photo of this neoplasmClick here for additional data file.Figure S2 Cell‐cycle‐checkpoint kinase 2 (CHEK2) is located on the long (q) arm of chromosome 22. p.H371Y germline mutation is caused by single base permutation in exon 11 of the CHEK2 gene.Click here for additional data file.


Hemangiomas are a benign vascular tumor and the most common benign tumors of the head and neck region. Although they usually present at birth or arise early in life, most of them involute spontaneously before adulthood. However, typically these tumors are located outside of paranasal sinuses and nasal cavity, including the lateral skull base, parotid gland, larynx, tongue and skin.3In this article, we reported a hemangioma originating from the pterygopalatine fossa, treated via the endoscopic approach without any complication. To our knowledge, the cavernous hemangioma of pterygopalatine fossa has not previously been published in the English literature.A 65-year-old female presented with headache and fullness on the face for several months. No other concomitant complaint was present. Nasal endoscopy, otological evaluation and cranial nerves examination were normal. Laboratory examination, complete blood count and routine blood chemistry were within the normal range. CT scan showed a well-circumscribed 3.4 cm × 2.8 cm soft tissue mass extending from the left pterygopalatine fossa to the left cavernous sinus . It causIn MRI, dense-heterogeneous contrasted mass was detected and it was mildly hyperintense on the T2-weighted images and hypointense on the T1-weighted images .Figure 2Endoscopic endonasal resection of the vascular lesion was performed under general anesthesia, without any complications. In operation, first, the medial wall of the maxillary sinus was removed and wide antrostomy was made. Anterior and posterior ethmoidectomy was added for increased visualization to posterior portion of the maxillary sinus. After that, sphenoidotomy was made and opticocarotid recess was identified for evaluation of the relationship with cavernous sinus. Posterior wall of the maxillary sinus was removed partially and the tumor was seen in PPF . At thisHemangiomas are benign vascular tumors most commonly found in the head and neck region, whereas these lesions rarely originate from the sinonasal area. They may arise from osseous, mucosal and submucosal portions of the nasal cavity and paranasal sinuses. In the literature, numerous classification systems were described for hemangiomas, with histological subtyping being the most commonly accepted. They are classified into three subtypes, according to dominant vessel size at the histological evaluation, as capillary, cavernous and mixed.5Capillary hemangiomas are the most common and are more often seen in children; they are usually located in oral mucosa, tongue and skin. These lesions usually may regress spontaneously, in early childhood; for this reason, surgical treatment is not recommended.6The surgical approach to Pterygopalatine Fossa (PPF) is difficult, for anatomical reasons . The PPF9In our case, fine-needle aspiration cytology was not performed, because the mass was located in an area which was not accessible with a fine needle aspiration biopsy. Only blood analysis, imaging modalities and clinical examination were used for diagnosis. At the preoperative radiological examination the mass was reported as a soft tissue tumor. The endoscopic transnasal approach was used for excision of a soft tissue tumor extending from the PPF to the cavernous sinus.Preoperative CT and MRI are useful in defining the location and extension of hemangiomas. CT findings of the cavernous hemangiomas are a soft-tissue density circumscribed mass, and also enhancing after injection of contrast. Hemangiomas of nasal cavity and paranasal sinuses may cause some changes on the adjacent bone. Generally, these changes seem like benign on imaging modalities. Dillon et al. reported three of six patients with cavernous hemangioma of nasal cavity have adjacent bone changes. In addition, all three cases have benign changes consisting of remodeling and expansion.Angiography and transarterial embolization are precious for the diagnosis and treatment of hemangiomas because biopsy or surgery may cause bleeding. Transarterial embolization may be used as an alternative treatment option for cavernous hemangiomas.Suitable treatment of a sinonasal hemangioma is wide excision of the tumor with the underlying soft tissue or mucosa, and cauterisation or ligation of the feeding vessels. Preoperative embolization may be useful for extensive lesion.In conclusion, cavernous hemangiomas are rare, benign lesions of the nasal cavity and paranasal sinuses. The present case is the first well-documented primary cavernous hemangioma of the pterygopalatine fossa. We completely removed this lesion via the endoscopic transnasal approach without preoperative embolization.The authors declare no conflicts of interest.


Angiomatosis is a rare non-neoplastic proliferative vascular lesion that typically develops during childhood or adolescence with a female predominance. Management of angiomatosis is challenging because of the manifestation of a wide variety of lesions as well as their invasive and highly recurrent nature.We report the case of a 74-year-old man who presented with a right lower back mass that persisted for a decade. The mass progressively enlarged and had been painful in the previous month.Computed tomography (CT) revealed suspected lipomatous sarcoma with invasion of the ribs, pleurae, and lung parenchyma. The final pathological examination revealed angiomatosis.The patient underwent wide composite excision of the tumor along with excision of the pleura and lung nodules in the right lower and middle lobes via video-assisted thoracoscopic surgery (VAST). Fasciocutaneous rotational flap reconstruction was performed immediately after the wide composite excision and video-assisted thoracoscopic surgery (VAST).The patient recovered uneventfully, was discharged without complications, and tolerated the daily activities well.Angiomatosis is a rare benign vascular tumor that frequently mimics malignancy. Even if the patient profile does not match the reported epidemiology of this disease, differential diagnosis should be considered. Complete resection is the mainstay of treatment for the prevention of recurrence. It is more common in females in the first 2 decades of life. This case report discusses a rare case of lower back angiomatosis with invasion of the underlying ribs and pleura in an elderly male patient.Angiomatosis is characterized by diffuse proliferation of vessels and adipose tissue and has been reported to occur in a variety of locations, including the extremities, muscles, bones, and, in a few cases, the chest wall.A 74-year-old man presented to our hospital’s clinic in the reconstructive and plastic surgery department complaining of a large mass in the right lower back region for the past 20 years. The main reason for his visit was the recent progressive enlargement of the mass for 1 month. The mass in the right lower back was accompanied by ecchymosis and positional pain. The patient had no medical, surgical, or trauma history.Physical examination revealed an ill-defined, soft, and tender mass with a maximum diameter of 14 cm and ecchymosis of the right lower back , 7th (10.0 cm), and 8th (15.0 cm) ribs. Video-assisted thoracoscopic surgery (VATS) was performed to excise the right pleura and lung nodules in the right lower and middle lobes / phosphoinositide 3-kinases (PI3K) mutations or associated with Klippel–Trenaunay syndrome, Sneddon syndrome, or Gorham disease. The latter is acquired from infections, such as human immunodeficiency virus or bartonellosis.,6 Radiological imaging may reveal nonspecific heterogeneous lesions in different tissue types with vascular involvement. Histopathological features include the proliferation of blood vessels of varying sizes, which are usually associated with large amounts of adipose tissue.Angiomatosis is a benign vascular lesion that most commonly affects females during the first 2 decades of life.This was an unusual case of angiomatosis in an elderly man with no relevant medical history. In addition, computed tomography (CT) revealed a heterogeneously enhanced lesion with extensive fat and focal calcification, primarily involving the lower back, with skeletal and pleural invasion of the chest wall, which are infrequent sites of angiomatosis. As a result, a clinical picture of malignant soft tissue tumor, such as liposarcoma, was highly suspected.,3 36% of patients demonstrate more than 1 recurrence during follow-up.,5 Wide surgical excision is the mainstay of treatment for disseminated angiomatosis. Radiotherapy, embolization, interferon α2a adjuncts, and angiogenesis inhibitors have been reported in the literatures.–3 In this patient, no local recurrence at 3-year follow up was observed, emphasizing the importance of margin-free complete resection.The definitive treatment of angiomatosis requires complete resection due to the high recurrence rate of up to 88%.In conclusion, angiomatosis is a rare soft tissue tumor that appears clinically similar to a variety of lesions such as hemangiomas, angiolipomas, liposarcomas, and metastatic tumors. Clinicians should consider angiomatosis as a differential diagnosis even if the patient profile does not fit the reported epidemiology of this disease. Although complete resection is challenging, particularly for extensively disseminated lesions, margin-free resection may be a definitive treatment.We thank Dr Austin Chen and Dr Savitha Ramachandran for editing the manuscript and for providing critical suggestions.All authors have read and agreed to the published version of the manuscript.Conceptualization: Yu-Chi Wang, Yun-Nan Lin.Data curation: Yu-Chi Wang.Project administration: Yu-Chi Wang.Supervision: Yur-Ren Kuo.Visualization: Yu-Chi Wang.Writing – original draft: Yu-Chi Wang, Yun-Nan Lin.Writing – review & editing: Chee-Yin Chai, Hsien-Pin Li, Yi-Ting Chen, Yur-Ren Kuo.


Safety and efficacy data on intravenous thrombolysis (IVT) for acute ischemic stroke (AIS) in case of known left ventricular thrombus (LVT) are lacking. We present the case of a 35-year-old male with disabling AIS and known LVT that was treated successfully with intravenous alteplase. Apart from neurological improvement, post-procedural full thrombus lysis was also evident. Even though performing IVT in similar instances constitutes a difficult decision for physicians, it may be reasonable in the context of acute disabling stroke. Up to today, straightforward guidelines on the proper management of acute ischemic stroke (AIS) associated with known left cardiac thrombus (LCT) are lacking. European Stroke Association (ESO) guidelines do not comment on whether intravenous thrombolysis (IVT) is contraindicated or not in such instances , whereasA previously healthy 35-year-old Caucasian male was admitted to the coronary care unit of a nearby secondary care hospital due to acute dyspnea, bilateral lower limb oedema, night sweats, and elevated blood pressure (190/140 mmHg). Baseline electrocardiogram revealed sinus rhythm, whereas transthoracic echocardiogram (TTE) demonstrated: (a) left ventricular (LV) hypertrophy and dyskinesis, (b) 30% ejection fraction (EF), (c) a mobile LV thrombus (3.5 × 2.5 cm) with a narrow stalk ; thus, was transferred to the neurology department of our tertiary referral hospital.Serum blood glucose and blood pressure levels at hospital admission were 103 mg/dL and 160/100 mmHg, respectively. Urgent brain CT revealed two preexisting infarcts, one near the left frontal horn and a second within the left cerebellar hemisphere, no evidence of new ischemic area (ASPECTS score: 10) Fig. a, c. BraCoronary angiogram performed four days afterwards was normal, whereas new TTE demonstrated EF of 45% and complete thrombus lysis Fig. e. 1 weekIn our patient, urgent brain CT scan performed before IVT revealed two small infarcts of unknown time of occurrence. Due to the fact that he was neurologically asymptomatic, IVT was not contraindicated as according to ESO expert consensus statement IVT with alteplase is recommended in selected cases of recent infarcts, like those that are small and exhibit good clinical recovery .The presence of those two infarcts in different vascular territories argued in favor of cardioembolic etiology and was consistent with the known large LVT. Proceeding with IVT in the presence of preexisting LVT is a difficult decision the physician must make due to the lack of data regarding the safety of the procedure . Danger Thrombus characteristics that are associated with embolic events include protrusion into the LV cavity, mobility independent of myocardium, patient age > 68, thrombus area, length of the thrombi in the lumen, and LVT recurrence . In our Although our experience cannot be generalized and more studies are needed, our decision may be logical and in keeping with the comment of Tsivgoulis et al. that in potentially severe disability due to AIS, withholding IVT in case of known cardiac thrombus is not justified .


Falciform ligament appendagitis is an extremely rare disorder, which is characterized by hematogenous or nonhematogenous inflammatory changes in the fat appendage that is contiguous with the falciform ligament. The imaging and clinical features of this condition are similar to those of epiploic appendagitis, especially when caused by torsion of the fatty appendage . We report 2 cases of falciform ligament appendagitis with localized epigastric pain. The ultrasound imaging features of the 2 cases presented here were an oval hyperechoic mass contiguous with the falciform ligament and increased echogenicity of the surrounding inflammatory fat. Both patients were managed conservatively with symptomatic treatment alone. Understanding the imaging features of this falciform ligament appendagitis is important, because ultrasound is often the first choice for noninvasive imaging of acute abdomen. As there is limited detailed literature on falciform ligament appendagitis comparing high-frequency linear probes with CT and MRI, we consider this case report to add valuable information on this poorly reported condition. The first diagnosis of falciform ligament appendage torsion (FLAT) using CT and ultrasound was reported by Coulier et al. in 2001 Case 1: A 25-year-old man was referred to our emergency room with persistent epigastric pain. His body mass index (BMI) was 18.5 kg/m2. Ultrasound and abdominal X-ray done at local clinic a few hours prior did not identify the cause. Palpation revealed localized tenderness on the epigastric area without guarding or rebound tenderness. Blood tests revealed a white blood cell (WBC) count of 10,720/ μL and a C-reactive protein (CRP) value of 0.13 mg/dL. For the ultrasound examinations, we used Aplio 400  with a 14L5 linear probe set at 10-MHz frequency. Ultrasound revealed an oval-shaped hyperechoic mass, which had a hypoechoic rim and was adjacent to the falciform ligament, and increased echogenicity and disturbance of the surrounding fat  with a 12L5 linear probe set at 8-MHz frequency. Ultrasound revealed an oval-shaped hyperechoic mass, which had a hypoechoic rim and was adjacent to the falciform ligament, and inflammation of the surrounding fat  the disease itself is rare, 2) it may have been missed on an ultrasound, and (3) it resolves spontaneously and patients do not return to the hospital. Among the studies on FLA, the most common imaging reported to have been used were ultrasound and CT  it may hGiven the anatomic location, a lesion in the falciform ligament is located at a close distance from the ultrasound probe; therefore, the use of a regular 3.5-MHz convex probe alone would make it difficult to notice the lesion. Given that both the patients were slightly leaner than standard body weight, we were able to use a high-frequency linear probe set at 8-10-MHz frequency. For mildly obese patients in whom this linear probe cannot be used, we recommend the 6-MHz high frequency convex probe. Thus, when FLA is suspected, the use of a high-frequency probe may improve diagnostic performance. In case 2, follow-up ultrasound demonstrated gradual shrinkage of the hyperechoic mass and reduction of the surrounding inflammatory fat until complete resolution . There hCT classically demonstrates FLA as an area of fat density with a thin peripheral rim of hyperattenuation, with or without the classical central dot corresponding to a thrombosed vein The diagnosis of infarcted ligamentum teres hepatis lipoma by MRI has only been reported in one case A few cases of surgical treatment for FLAT had been reported Written informed consent was procured from the patients for publication of this case.Ultrasound interpretation and manuscript writing: DM, CT and MRI interpretation: MS, and clinical diagnosis: YFThe Fukuoka Tokushukai Medical Center Research Ethics Committee has confirmed that no ethics approval was required. All procedures were in accordance with the ethical standards of the responsible institutional and national committee on human experimentation and with the Helsinki Declaration of 1975 and its late amendments.Data sharing not applicable to this article as no datasets were generated or analyzed during the current study.Not applicable.


The presence of primary retroperitoneal hydatid cysts is rare, even in endemic areas. The authors report a young man with a retroperitoneal hydatid cyst who underwent total cystectomy. The surgeons should suspect hydatid cysts in case of any abdominal cysts, especially in endemic areas, and avoid any spillage and puncture. Even in endemic locations, primary retroperitoneal hydatid cysts are scarce. However, practitioners, particularly surgeons, should think about hydatid cysts as a possible differential diagnosis for retroperitoneal masses. He had no complaint of abdominal pain. The past medical history was insignificant. During physical examinations, a local abdominal bulge was found in the left upper quadrant area. Other systemic physical examinations were normal. All his laboratory investigations were within the normal range. On abdominal ultrasound imaging, an 18 × 13 cm cystic cavity was found in the retroperitoneum area with a hyperechogenic membrane and clear fluid, suggesting a hydatid cyst. Computed tomography showed a vast (20 × 14 cm) retroperitoneal cystic lesion between the diaphragm and bladder, which shifted the abdominal organs to the right side of the abdomen Figures , 3 and 43Echinococcus granulosus is a 5 mm worm that can live for 5–20 months within the jejunum of dogs. Echinococcal eggs exist in the small intestine of canine animals and excrete with feces. Intermediate hosts such as humans, sheep, or cattle ingest these eggs. Then, these eggs penetrate through the intestinal mucosa, enter the blood and lymphatic circulation, and affect body organs.E. granulosus. It was stated that E. granulosus mostly (95%) affects the lungs and liver, while only 5% involved other organs.Also, in the serologic examination, the detection of Immunoglobulin G antibody by the ELISA (enzyme‐linked immunosorbent assay) method showed high sensitivity and specificity .The gold standard treatment for hydatid cysts is total cystectomy without contamination.The authors meet the four criteria for authorship based on the International Committee of Medical Journal Editors (ICMJE) recommendations.All authors declare no conflicts of interest.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Infantile hypertrophic pyloric stenosis (IHPS) is a common cause of infant vomiting. Emergency department (ED) diagnosis is usually made by pyloric ultrasound and treated by pyloromyotomy.An eight-week-old boy with a history of IHPS about six weeks status post pyloromyotomy presented to the ED with vomiting and failure to thrive, and a critically narrowed pylorus was identified by ultrasound. An upper gastrointestinal series confirmed recurrent pyloric stenosis, necessitating another pyloromyotomy.Prolonged vomiting after pyloromyotomy should be concerning for recurrent IHPS. Upper gastrointestinal series should augment ultrasound to diagnose recurrent IHPS and determine whether a second pyloromyotomy is warranted. Infantile hypertrophic pyloric stenosis (IHPS) is a well-described pediatric surgical emergency often presenting with “projectile” emesis of undigested milk and a hungry baby, not uncommonly with dehydration or even failure to thrive. Ultrasound has generally become more readily available in the emergency setting and has become the mainstay of IHPS diagnosis. With early ultrasound evaluation, it is less common to identify classic clinical findings such as a peristaltic wave, a palpable “olive-shaped” mass, or hypochloremic, hypokalemic metabolic acidosis. Early presentation for vomiting in a young infant, physical exam, and resultant imaging facilitate early surgical intervention and best outcomes. Although an upper gastrointestinal (GI) series may also be used to diagnose IHPS (sensitivity 100% and specificity 100%),2Infantile hypertrophic plyroic stenosis is managed by increasing the size of the pyloric canal such that foods may appropriately pass. Procedures are usually curative, and recent literature suggests that recurrence of pyloric stenosis is exceedingly rare. Although some children receive balloon dilation to alleviate the pathologic obstruction of IHPS, the current mainstay of care is laparoscopic or even open pyloromyotomy.An eight-week-old male patient presented to the ED with his parents for a three-week history of frequent episodes of post-prandial non-bloody and non-bilious emesis .The pareOn further interview with parents and on chart review, the patient was born at term by vaginal delivery with a birth weight of 3,460 grams and a normal physical exam. Six weeks prior, at 17 days of age, he was taken to the ED after two days of persistent, forceful, non-bloody and non-bilious emesis, which occurred after every feed. During that initial ED visit, the patient’s basic metabolic profile was normal, including a potassium of 4.6 milliequivalents per liter (mEq/L) (reference range: 3.7–5.2 mEq/L) and chloride of 103 mEq/L (reference range: 96–106 mEq/L), and an abdominal ultrasound was concerning for an abnormally large pylorus measuring 2 centimeters (cm) in length and 4 millimeters (mm) in width with no passage of food contents. The patient urgently underwent a successful laparoscopic pyloromyotomy without complication. He was discharged home with his parents with an uneventful three-week period including normal oral intake and adequate weight gain for his age.What do we already know about this clinical entity?Hypertrophic pyloric stenosis is a well-described pathology causing vomiting leading to failure to thrive in infants. Diagnosis is usually made by ultrasound.What makes this presentation of disease reportable?This presentation describes a case of a child who had already received a pyloromyotomy with another clinically classic case of pyloric stenosis.What is the major learning point?When pyloric stenosis is diagnosed very early in life, despite appropriate surgical intervention, it could possibly recur. An upper gastrointestinal series may help with diagnosis.How might this improve emergency medicine practice?The clinical signs and symptoms of pyloric stenosis are generally consistent and require appropriate evaluation even if a child has already what is typically definitive intervention.On evaluation, the patient was lethargic with a weak cry. The child appeared small for stated age with dry mucous membranes and a flat fontanelle. Vital signs on presentation included a temperature of 98.5° Fahrenheit, heart rate 130 beats per minute, respiratory rate 35 breaths per minute, oxygen saturation 98%, and blood pressure 81/48 millimeters of mercury (mm Hg). The patient had clear bilateral tympanic membranes and a normal posterior oropharynx. His chest was clear to auscultation, cardiac exam was grossly normal, and his abdomen was soft, without dilatation, and without palpable masses. He had a three second capillary refill  and a normal genital exam for his age. The child produced a weak cry when nurses started a peripheral intravenous line.A basic metabolic panel was significant for a hypokalemic (2.6 mEq/L), hypochloremic (76 mEq/L) metabolic alkalosis. A venous blood gas showed a pH greater than 7.70 (reference range: 7.31–7.41), partial pressure of carbon dioxide 35 mm Hg (reference range: 41–51 mm Hg), and partial pressure of oxygen 53 mm Hg (reference range: 30–40 mm Hg). We were unable to calculate bicarbonate and base excess due to a pH more than 7.70. The patient underwent an abdominal ultrasound in the ED, which suggested IHPS with an enlarged pyloric channel measuring 2.1 cm and a thickened muscle measuring 0.5 cm, again with minimal passage of fluids through the pylorus . The surThe underlying cause of a recurrent hypertrophic pyloric stenosis after a successful pyloromyotomy remains unclear. Emesis after feeds is a common and is the most frequent complaint post pyloromyotomy. It is likely due to pyloric edema, pylorospasm, or reflux.Ankermann et al (2002) described evaluation for recurrent pyloric stenosis. If postprandial projectile emesis occurs after successful operation and a symptom-free interval, the absence of fluid passage through the pyloric channel should be demonstrated radiologically with a swallow of water-soluble contrast before reoperation is considered.10Given that the etiology of recurrent pyloric stenosis after a successful pyloromyotomy is unknown, one consideration is that “recurrence” is actually a result of the progression of the original disease and the initial surgery was probably performed at an early stage after which the pylorus continued undergoing an active process of hypertrophy.Infantile hypertrophic pyloric stenosis is easily identified on abdominal ultrasound as a cause of refractory vomiting. Although rare, despite surgical pyloromyotomy, a minority of infants may develop a recurrence of IHPS, presenting clinically similarly to the sentinel case. Diagnosis can be made by repeated ultrasound but should be augmented by upper gastrointestinal series to avoid false positive diagnoses, which might actually be the result of postoperative edema. Management for recurrent cases is a second pyloromyotomy, which usually has good and definitive outcomes.


A 36-year-old Caucasian, nulliparous patient sought care at a private gynecology clinic after 6 months of attempting to conceive. During the initial consultation, the patient reported severe dysmenorrhea and deep dyspareunia. During the gynecological examination, a nodule in the left lateral vaginal fornix was palpable. The MRI showed a hypointense nodular lesion in the left paracolpium described as an endometriosis nodule. Laparoscopic resection of the nodule was indicated. The patient showed improvement in symptoms after surgical treatment. This case report describes the technique for laparoscopic approach to paracolpium tumors. Uterine leiomyomas are very common in women during menacme . VaginalDue to the low incidence of leiomyomas in the paracolpos, other diagnoses should be considered when a mass is observed in this anatomical region , 3. MagnFor patients whose paracolpal tumors are asymptomatic, initial management can be expectant as the probability of malignancy is low . In the http://www.scareguideline.com).This case report was approved by an institutional review board, the Research Ethics Committee (CAAE 44315920.9.0000.8527). Written patient consent was obtained and is on file at our institution. To improve transparency and reporting quality, the SCARE Guideline  was used as a checklist  in which 0 means no pain and 10 means terrific pain, the scores were 7 for dysmenorrhea and 10 for deep dyspareunia.The patient reported no comorbidities and denied previous surgeries. During the gynecological examination, palpation of a nodule in the left lateral vaginal fornix reproduced the patient's dyspareunia. On specular examination, the nodule did not bulge the vaginal wall; no changes in the vaginal mucosa such as ulceration or bleeding were observed.The ovarian reserve was considered normal; the preoperative anti-Mullerian hormone level was 5.3 ng/mL. The hysterosalpingography and the partner's spermogram were normal. The MRI showed a hypointense nodular lesion on T1- and T2-weighted sequences measuring 1.6 × 1.3 × 1.0 cm in the left paracolpium. The lesion was described as a deep infiltrative endometriosis nodule , the sigmoid was mobilized from the level of Toldt's fascia until the identification of the left ureter where it crosses the left common iliac artery. Left ureterolysis was performed from where it crosses the common iliac artery to its intersection with the uterine artery.The next step was the dissection of the left pararectal space, in order to separate the uterosacral ligament affected by endometriosis from the hypogastric nerve and from the ureter. Dissection of the Latzko space was then performed, with preservation of the left mesoureter. This space is important when approaching the parametrium and paracolpos with endometriotic lesions, as it allows distal control of vascular lesions in these regions and reduces inadvertent lesions to the ureter, pelvic splanchnic nerves, and inferior hypogastric plexus .During dissection of the left paracolpos, a well-defined hardened region was identified below the cardinal ligament. When the cardinal ligament was incised, a plane of cleavage was observed that usually does not occur with lesions of deep infiltrating endometriosis . This clMultimodal approaches to management of pain and postoperative nausea and vomiting were implemented. Postoperative analgesia included infiltration of port sites with bupivacaine with epinephrine 0.5% (approximately 20 mL) at the end of surgery and scheduled systemic analgesia with intravenous ketorolac tromethamine and dipyrone.The patient tolerated an oral diet 2 hours after surgery. She began walking 4 hours after surgery and thus did not require pharmacological thromboprophylaxis. After spontaneous diuresis, the patient was discharged within 24 hours, with no movement limitation or complaints of pain.She was followed up with postoperative pelvic physiotherapy as a routine part of the multidisciplinary treatment of deep dyspareunia in our service. At her postoperative review 30 days after surgery, the patient reported that the dysmenorrhea was considerably less intense (NRS = 1 versus 7 preoperatively) and that she had already had sexual intercourse without dyspareunia (NRS = 0).Histopathological evaluation of the paracolpal tumor revealed that it was a leiomyoma . The patVaginal leiomyomas are uncommon, and their symptoms can mimic endometriosis , 7. WhenAlthough both TVUS and MRI are cited as the imaging studies of choice in the diagnosis of leiomyomas and endometriosis , MRI hasOur technique for approaching the paracolpal nodule was the same used for the treatment of complex deep endometriosis. This approach makes it possible to prevent injuries to the inferior hypogastric plexus and thus avoid functional complications such as changes in bowel habits, urinary retention, and reduction in vaginal lubrication .As for the postoperative recovery, laparoscopy seems to be related to less postoperative pain compared to vaginal surgery . MeasureEgbe et al. described a case of vaginal myomectomy in which the couple interrupted their sexual activity and psychological support was required . ColpotoWhen surgical treatment of a paracolpal tumor is indicated, the location of the lesion should be considered, and the laparoscopic approach could be a good option for lesions of the upper third of the vagina. The systematization for the treatment of paracolpal leiomyomas can be the same as that used for nerve sparing deep infiltrative endometriosis resection. The laparoscopic approach to vaginal lesions can minimize functional complications, especially dyspareunia, and favors a rapid recovery. Until the impact of colpotomy on women's sexual is better elucidated, laparoscopic surgery can be an option in the treatment of vaginal leiomyomas which avoids colpotomy.


Hypertrophic pyloric stenosis is a common cause of vomiting in the first few weeks of life, but in rare cases, it may occur in older subjects with a major risk of delayed diagnosis and complications. We describe the case of a 12-year-and-8-month-old girl who presented to our department for epigastric pain, coffee-ground emesis, and melena, which arose after taking ketoprofen. An abdomen ultrasound showed thickening (1 cm) of the gastric pyloric antrum, while upper-GI endoscopy documented esophagitis and antral gastritis with a non-bleeding pyloric ulcer. During her hospital stay, she had no further episodes of vomiting and was therefore discharged with a diagnosis of “NSAIDs-induced acute upper gastrointestinal tract bleeding”. After 14 days, following recurrence of abdominal pain and vomiting, she was hospitalized again. At endoscopy, pyloric sub-stenosis was found, abdominal CT showed thickening of large gastric curvature and pyloric walls, and an Rx barium study documented delayed gastric emptying. On suspicion of idiopathic hypertrophic pyloric stenosis, she underwent Heineke–Mikulicz pyloroplasty with resolution of symptoms and restoration of a regular caliber of the pylorus. Hypertrophic pyloric stenosis, although occurring rarely in older children, should be taken into account in the differential diagnosis of recurrent vomiting at any age. Hypertrophic pyloric stenosis (HPS) is a common condition in the neonatal period (incidence of 1.5–3 per 1000 live births) . Few casWe report the case of an adolescent girl that, after an extensive gastroenterological and surgical work-up, was successfully treated for late onset HPS.Case description A 12-year-and-8-month-old girl accessed the emergency department for a recent history of epigastric pain associated with multiple episodes of coffee-ground emesis and melena, which arose after taking two doses of ketoprofen for gingivitis. She was the second child of non-consanguineous parents, born at term from vaginal delivery (birth weight: 2810 kg). She had an unremarkable medical history, except for a bilateral inguinal hernia surgically corrected at three years of age. There was no history of cow’s milk or other allergies and no exposure to long-term medications or to active or passive smoking. Initial laboratory exams documented a normal full blood count (hemoglobin levels 13.2 g/dL) and coagulation pattern within normal limits. On abdominal ultrasound, a “marked parietal thickening (approximately 1 cm) of the gastric pyloric antrum supported by submucosal and mucosal edema” was described .The patient was therefore admitted to our Pediatric Department where, upon physical evaluation, she presented good general conditions; her blood pressure was 110/59 mmHg, and the heart rate was 110 bpm. Weight and height were both on the 25° centile for age. At a careful medical recall, she reported a history of intermittent nausea and precocious satiety in the previous year. On admission, she was started on IV fluids and gastroprotective therapy, continuous monitoring of vital parameters, and, after surgical evaluation, she underwent urgent upper GI-endoscopy 2 h after admission. Endoscopy showed distal esophagitis, and abundant coffee-ground fluid (700 mL) was found and aspirated from the stomach. The antral mucosa had a nodular aspect, and a pre-pyloric, non-bleeding ulcer with fibrin-covered fundus was documented. A narrow pyloric channel was noted, but the pediatric gastroscope could pass through .Histologic examination documented hyperplastic-reparative aspects of the distal esophagus mucosa and reactive changes in the gastric body and antral mucosa. Standard stains and immune-histochemistry were negative for CMV and Helicobacter pylori. Celiac serology and fecal calprotectin were negative. During hospitalization, the patient had no further episodes of vomiting and maintained good general clinical conditions and stable vital parameters. The clinical picture was considered compatible with a diagnosis of “NSAIDs-induced acute upper gastrointestinal tract bleeding” and the girl was discharged on oral proton-pump inhibitors. Ten days after discharge, she reported loss of appetite and precocious satiety, which was associated with weight loss of about 1 kg. Three days after, she experienced episodes of abdominal pain and nausea after a large meal, followed by non-bilious vomiting. For the subsequent recurrence of two episodes of profuse and coffee-ground emesis in the following days, she was readmitted in the hospital. At a second endoscopy, a fibrin-covered pyloric ulcer (approximately 8 mm in size and not actively bleeding) and a substenotic appearance of the pylorus were found . A contrast barium Rx study documented delayed gastric emptying with slow passage of fluids through the pylorus. In consideration of the pyloric substenosis, an abdominal CT scan was performed, which excluded neoplastic lesions but confirmed the presence of diffuse wall thickening of the large gastric curvature and pylorus .On suspicion of idiopathic hypertrophic pyloric stenosis, the patient was transferred to the Department of Pediatric Surgery, where she was maintained on total parenteral nutrition. On day 22 from admission, she underwent Heineke–Mikulicz pyloroplasty (HMP) by an open approach. At laparotomic exploration, the pylorus appeared thickened and hypertrophic. Full-thickness biopsies of the pyloric wall were performed and documented foveolar hyperplasia, mild inflammatory lymphocytic infiltrate of the lamina propria, and blood congestion of the submucosa; it also confirmed pyloric muscle hypertrophy. A pyloroplasty was then performed by the technique of Heineke–Mikulicz via a full-thickness longitudinal incision of the muscular wall of the pylorus, which was then sutured transversely. After pyloroplasty, water was infused via a nasogastric tube, which showed patency of the pylorus and absence of perforation. In the postoperative period, the patient gradually was restarted on oral diet, initially with creamy and later with solid foods, with no occurrence of vomiting or other gastrointestinal symptoms. Ultrasound examination at 30 days post-op was within normal limits .At endoscopy performed three months after surgery, a pylorus of regular caliber and normal gastric and duodenal mucosa were documented. At the nine-month medical follow-up, the girl did not complain of any symptom and had regained 2 kg of weight.We described the case of a late-onset pyloric hypertrophy in an adolescent Italian girl and compared this with similar cases from the literature.HPS is the principal cause of GOO in the first months of life . At this age, incidence is of 1.5–3 per 1000 live births ,10,11. TOur extensive literature review identified a total of 21 cases of HPS described in children with onset of symptoms after three months of age (range: seven months–17 years) ,19,20,21In all the reported cases, vomiting was the presenting complaint, accompanied by weight loss or growth failure, abdominal pain, and precocious satiety. Apart from our case, gastrointestinal hemorrhage (hematemesis and/or melena) was reported only in another adolescent girl . InteresThe peculiarity of our case was certainly the age and gender of the patient, the presentation with hematemesis, and the confounding initial endoscopic picture, which showed signs of inflammation compatible with NSAID’s gastritis. Recurrence of symptoms requiring a second endoscopic evaluation led to further investigations and finally allowed us to postulate a diagnosis of HPS.In the management of suspected HPS, first-level investigations include abdominal ultrasound that can document an increased pylorus thickness  or require further investigations as previously indicated. In our case, we performed both CT and a barium study, and two endoscopic assessments were needed before a definitive diagnosis was made. A histological confirmation  is not always necessary, and most of the time, it is not easy to obtain a full-thickness biopsy.In terms of treatment, both endoscopic and surgical strategies have been reported in pediatric-onset HPS. We found endoscopic balloon dilatation attempted in eight cases ,16,19,21The treatment of choice for neonatal HPS is pyloromyotomy, which was traditionally performed under laparotomy and subsequently adapted to laparoscopy ,26,27. MPyloroplasty, initially performed in 1886 by Dr. Heineke and subsequently by Dr. Mikulicz, relies on longitudinally opening the pylorus and transverse closure, and it is the most common technique for the treatment of peptic ulcer disease. The surgical principle on which this technique is based, given its effectiveness, has also been adopted for the treatment of other pathological stenoses, such as intestinal strictures secondary to Crohn’s disease. Given the patient’s age and the thickness of the pylorus, we decided to perform an HMP with an open approach in order to have safer control of the pylorus and to be able to perform a pyloroplasty that would be effective while minimizing the risk of recurrence or iatrogenic injury. In fact, although in our center the approach of choice for pyloroplasty in the infant is laparoscopic, in this case, we felt that the lower tactile feedback of laparoscopy might expose us to a higher risk of perforation or incomplete muscle opening. HMP was the technique of choice in three other cases reported in the literature ,19 and, A more invasive surgical technique was required in the remaining nine cases, including distal gastrectomy with Billroth-I or Roux-en-y reconstruction ,14,16,20All the reported cases and our personal experience indicate a successful outcome of the surgical strategy with a complete restitution ad integrum of pyloric function and symptom resolution, regardless the specific surgical technique. In this setting, the choice of more aggressive surgical strategies is therefore questionable unless specific contra-indication to less invasive procedures exists or there is a specific necessity for diagnostic purposes  (e.g., tHPS should be considered in the differential diagnosis of older children and adolescents with recurrent vomiting or hematemesis, even when other causes seem to be more plausible. A multistep work-up, including upper gastrointestinal endoscopy and abdominal CT scan, should be followed in order to reach the final diagnosis, and treatment by a minimally invasive endoscopic/surgical procedure should be preferred.