Phosphaturic mesenchymal tumor (PMT) is a rare entity that presents as a paraneoplastic syndrome that causes tumor-induced osteomalacia (TIO). Most of these cases were located in the extremities. We report a case of a phosphaturic mesenchymal tumor arising in the left upper thigh. The tumor was discovered during the clinical workup of a patient complaining of osteomalacia symptoms with multiple fractures. Phosphaturic mesenchymal tumor (PMT), also known as tumor-induced osteomalacia (TIO), is a rare paraneoplastic syndrome caused by mesenchymal tumors secreting fibroblast growth factor-23 (FGF-23) -3. In 19A 48-year-old female who was a known case of diabetes mellitus presented to our hospital with right hip pain, inability to bear weight, and a history of multiple unprovoked fractures. Laboratory workup showed hypophosphatemia (0.26 mmol/L), low 1,25-hydroxy vitamin D (65 nmol/L), high FGF-23 , high alkaline phosphatase level (525.0 U/L), normal serum calcium level (2.24 mmol/L), and high 24-h urine phosphate (53 mmol/day)  showed osteopenia with multiple fractures. The patient was evaluated by ultrasonography and was found to have a 3.2 × 2 cmy Figure .No other bone or soft tissue masses were identified. An Octreotide scan showed a hot lesion in the left upper thigh Figure .During the evaluation, the patient was treated empirically with phosphate, calcium, and vitamin D supplements. The lesion on the left upper thigh was surgically excised. Histopathological examination revealed a bland spindle cell proliferation in a background of hemangiopericytomatous-like blood vessels, multinucleated giant cells, smudgy/grungy basophilic matrix, and microcystic changes with rare mitosis (1-2/10 HPF). There was no evidence of pleomorphism, hyper-cellularity, or necrosis Figure -3D.A panel of immunohistochemistry was performed, including vimentin, CD68, CD56, CD34, ERG, and Ki67. The tumor cells showed positive immunoreactivity against ERG and CD56, with variables for CD68, while vimentin and CD34 were negative. Ki67 showed a very low proliferative index (<5%) Figure -4C. In lPhosphaturic mesenchymal tumors are an infrequent entity that is usually present as a paraneoplastic syndrome that causes tumor-induced osteomalacia (TIO) -3. OsteoPhosphaturic mesenchymal tumors are found to be the most common cause of TIO . PMT chaThe laboratory findings observed in PMT-induced osteomalacia are hypophosphatemia and relative hyperphosphaturia, the most consistent biochemical derangements. In addition, increased levels of intact FGF-23, low-normal serum calcium and parathormone levels, and a decrease in 1,25 dihydroxyvitamin D are also noted. However, a non-phosphaturic state has also been described . TherefoDifferent imaging modalities are used to localize the tumor. Whole-body Tc-99m sestamibi scanning, octreotide scintigraphy, 18F-fluorodeoxyglucose (FDG) positron emission tomography (PET), whole-body MRI, and computed tomography (CT) are used for the localization of these tumors. It is difficult to recommend one modality over another, and the choice primarily depends on availability . CT typiMicroscopically, four variants of PMTs exist: osteoblastoma-like variant, non-ossifying fibroma-like variant, ossifying fibroma-like variant, and mixed connective tissue variant ,4. The mPMTs' histological features are characterized by the presence of grungy, smudgy basophilic calcifications, bland-looking normochromic spindle cells with small nuclei, and inconspicuous nucleoli in a myxoid background and very few mitotic figures. Additional morphological features such as osteoid matrix, multinucleated giant cells, hemangiopericytoma-like blood vessels, microcystic changes, and entrapment of mature adipose tissue can be seen. None of the above histological features are pathognomonic in and of themselves, but in the right clinical context and with the laboratory findings, they support PMT diagnosis ,3-5. TheRadiation therapy is used for tumors that are not amenable to surgical resection or incompletely resected tumors with positive margins. Radiofrequency ablation for small bony tumors in critical locations is advised. Medical treatment including phosphorus and calcitriol supplementation is proposed for cases where the tumor cannot be clinically localized. However, hyperparathyroidism, hypercalcemia, and nephrolithiasis could be potential complications of such medical treatment ,4.PMT is characteristically a benign tumor ,5. LocalPhosphaturic mesenchymal tumor is an extremely rare neoplasm. PMT is considered one of the most common causes of tumor-induced osteomalacia (TIO) and is characteristically benign. We report an additional case of PMT-induced osteomalacia in a 48-year-old woman. The presence of hypophosphatemia, hyperphosphaturia, and a high level of fibroblast growth factor-23 with clinical symptoms of osteomalacia are essential clues for PMT diagnosis. The patient did well after surgery and was discharged in stable condition. A follow-up was done after six months, and there was no recurrence.


Background: Pituitary apoplexy results from hemorrhage, infarction, or hemorrhagic infarction within a pituitary tumor. Subclinical or clinical apoplexy is not uncommon in acromegaly, owing to the large size of the tumor at initial detection. Growth hormone excess in acromegaly often persists following surgery. However, in rare instances, pituitary apoplexy may present a spontaneous cure to growth hormone excess.Case Report: A 40-year-old male presented with holocranial headache for the past 16 years that had worsened in severity during the prior year. Two months before presentation, he experienced a severe headache that he described as the worst headache of his life. The patient had prominent acromegaloid features that he ignored, as they seemed to cause no harm. The patient had no signs of clinically active disease. Magnetic resonance imaging of the brain revealed a pituitary macroadenoma with evidence of hemorrhage. Serum insulin-like growth factor 1 and oral glucose–suppressed serum growth hormone levels were normal, suggestive of inactive or silent disease. Pituitary apoplexy causing spontaneous remission of acromegaly was diagnosed, and close follow-up was planned for the evolution of hypopituitarism.Conclusion: This case highlights a rare presentation of acromegaly in which an episode of symptomatic pituitary apoplexy revealed the diagnosis of pituitary adenoma and led to the cure of growth hormone hypersecretion. However, pituitary apoplexy may be subclinical or asymptomatic in up to one-fourth of patients with pituitary adenoma and is incidentally discovered on imaging or at autopsy.4Pituitary apoplexy is a rare but potentially fatal clinical syndrome that results from hemorrhage, infarction, or hemorrhagic infarction within a pituitary tumor. Pituitary apoplexy has been described at an incidence of 0.17 episodes per 100,000 patients per year, affecting approximately 2% to 12% of patients with pituitary adenoma.10 The etiopathogenesis for development of pituitary apoplexy includes the outgrowth of a pituitary tumor in relation to its blood supply and compression of the pituitary gland vascular supply by the expanding tumor mass, resulting in necrosis and infarction.11 Pituitary apoplexy is more often reported with nonfunctioning pituitary adenomas (NFPAs) than with functioning adenomas, an observation attributed to the fact that NFPAs are often diagnosed late when they are already quite large in size.12 However, the association with NFPAs may be slightly overestimated, as functionality may not be apparent because of the pituitary damage caused by apoplexy.13 Pituitary apoplexy has also been described with prolactinomas, corticotropinomas, and growth hormone–producing tumors.Precipitating factors for pituitary apoplexy include head trauma, major surgery , uncontrolled hypertension, diabetes mellitus, anticoagulant use, cranial irradiation, dopamine agonist therapy, and dynamic testing.3 However, studies have demonstrated favorable outcomes with conservative management, suggesting that surgical treatment should be reserved for select patients with visual defects and a reduced level of consciousness.17The diagnosis of pituitary apoplexy is established via computed tomography or magnetic resonance imaging (MRI) of the brain demonstrating a pituitary tumor with evidence of necrosis and/or hemorrhage. Historically, pituitary apoplexy was considered a neurosurgical emergency, and surgical decompression was performed in all cases.19 When it is a complication of functional pituitary adenoma, pituitary apoplexy can sometimes (rarely) lead to the resolution of hormonal hypersecretion and remission of the disease state.22 We describe such an occurrence in a middle-aged male with acromegaly who had spontaneous cure of his growth hormone excess state following an episode of symptomatic pituitary apoplexy.Pituitary apoplexy can result in multiple acute endocrine insufficiencies, the most common and clinically significant being secondary adrenal insufficiency.A 40-year-old male presented to our clinic with a history of holocranial headaches for the past 16 years that had gradually worsened. For the 1 year prior to presentation, he was troubled on an almost daily basis with severe headaches that also caused frequent nocturnal awakenings. He had been treated elsewhere for migraine headaches without significant symptomatic improvement.He had noticed acromegaloid changes in the form of coarsening facial features and an increase in the size of his hands and feet during the prior 10 years. However, he did not seek medical attention for these changes, as they seemed to cause no harm. The patient had no history of seizures, loss of consciousness, recurrent vomiting, or episodes of transient visual obscurations. He denied a history of hypertension or diabetes mellitus. Two months before the current presentation, he had an episode of severe headache that he described as the worst headache of his life. The episode lasted for 4 to 5 hours, and he was treated with intravenous analgesics and fluids. No nausea, vomiting, dizziness, blurring of vision, diplopia, or any other focal neurologic deficit was associated with the episode. He denied a history of head trauma, anticoagulant use, prior surgical intervention, cranial irradiation, and pituitary dynamic testing.2, respectively. The clinical features of acromegaly were confirmed on physical examination; however, no signs of active disease were noted . These investigations suggested normal pituitary function with no evidence of growth hormone hypersecretion. MRI showed enlarged pituitary fossa, a 12-mm macroadenoma in the right half of the pituitary gland, and evidence of hemorrhage within the mass , idiopathic intracranial hypertension, posttraumatic headache, substance use or withdrawal-related headache, migraine, cluster headache, and idiopathic thunderclap headache. Our patient had severe acute headache with clinical features suggestive of acromegaly and neuroimaging evidence of pituitary macroadenoma with hemorrhage, suggestive of pituitary apoplexy. However, clinical and biochemical markers of acromegaly activity were normal, suggestive of inactive disease. The patient had no history of transsphenoidal surgery or cranial irradiation. Pituitary apoplexy causing spontaneous remission of acromegaly was therefore diagnosed.Because the laboratory evaluation did not reveal any endocrine abnormalities, no specific treatment was initiated. The severity of headache reduced significantly during the course of the patient's hospital stay, and he was advised to take oral analgesic paracetamol when needed. He was counseled regarding the need for close follow-up to detect hypopituitarism and was discharged with a plan to repeat pituitary function and neuroimaging at an interval of 3 to 6 months.23 In fact, many patients are diagnosed on the appearance of symptomatic mass effects caused by the tumor or incidentally when seeing a physician for some other ailment. The delay in diagnosis exposes patients to the risk of tumor progression and a state of uncontrolled growth hormone hypersecretion, resulting in increased morbidity and mortality. Our patient had had symptoms of growth hormone excess for 10 years, but he ignored them because the symptoms did not result in any functional limitation. However, the symptom of headache prompted him to see multiple physicians who failed to diagnose the primary disease.The diagnosis of acromegaly is often delayed by many years (average 10 to 12 years) because of the slow and subtle progression of physical findings that patients fail to notice.24 Surgery is the first line of therapy for most patients with acromegaly. However, remission rates are only 80% for patients with microadenoma and <50% for those with macroadenoma,25 implying that a large proportion of patients are left with a persistent growth hormone hypersecretion state, requiring consideration for medical therapy , radiation therapy , or repeat surgery. Our patient had an undiagnosed pituitary macroadenoma that bled, possibly because of its large size, and resulted in the spontaneous cure of acromegaly without therapeutic intervention.In more than 95% of cases of acromegaly, the cause for growth hormone hypersecretion is a somatotroph pituitary adenoma, commonly macroadenoma.30 While a precipitating event such as gastrointestinal hemorrhage or gadolinium-DTPA (diethylenetriaminepentaacetic acid) administration causing an acute change in blood pressure was identified by some authors, no trigger could be identified by others.30 Our patient had no recognizable precipitating factor for the acute event.Clinical remission of acromegaly following apoplexy is extremely rare and to our knowledge has only been reported in 29 cases in the literature.26 This observation highlights the importance of close surveillance for pituitary function in any patient with suspected or confirmed pituitary apoplexy. While we found no pituitary hormone deficits in our patient at the current presentation, he will be closely followed for potential deficits.Pituitary apoplexy is an important cause of hypopituitarism. A literature review reveals that more than two-thirds of patients who had cure of acromegaly following pituitary apoplexy developed other pituitary hormone deficiencies, and a small fraction (<10%) even evolved to panhypopituitarism.Pituitary apoplexy should be considered in the differential diagnosis of a patient presenting with acute severe headache. In rare instances, pituitary apoplexy may result in a spontaneous cure of the pituitary hypersecretion state. Patients with suspected or confirmed pituitary apoplexy should undergo periodic assessment for the development of hypopituitarism. This case highlights a rare presentation of acromegaly in which an episode of symptomatic pituitary apoplexy revealed the diagnosis of pituitary adenoma and led to the cure of growth hormone hypersecretion.


A case of tracheobronchopathia osteochondroplastica (TO) was diagnosed in a 53‐year‐old man, who presented with prolonged cough and recurrent respiratory tract infection. Bronchoscopy revealed exostosis over the anterolateral wall of trachea and main bronchi sparing the posterior membranous wall. The endobronchial biopsy subsequently revealed ossification of the cartilage. To date, the aetiology of this condition remains unknown, and treatment is mainly symptomatic, emphasizing on timely management of recurrent respiratory infections. Bronchoscopy or surgical intervention is usually reserved for symptomatic patients with severe airway narrowing and airflow obstruction. Tracheobronchopathia osteochondroplastica (TO) is a rare benign disorder affecting the trachea and main bronchi characterized by the development of submucosal cartilaginous and bony nodules. We report a case of TO in a patient presented with prolonged cough and recurrent respiratory tract infection. Tracheobronchopathia osteochondroplastica (TO) is a rare benign disorder affecting the trachea and main bronchi. It is characterized by the development of submucosal cartilaginous and bony nodules. These features are typically seen in computed tomography (CT) of the thorax and flexible bronchoscopy, whereas biopsy and histological identification is essential in making the diagnosis. First described by Rokitansky, Luschka, and Wilks in the 19th century , 3, 4, TA 53‐year‐old man presented with chronic productive cough with greenish sputum for the past six months. Further history revealed intermittent fever with no symptom of breathlessness or haemoptysis. He worked as a construction worker for the past 20 years. Otherwise, he had no significant medical illness or history of tobacco use. No family history of lung disease or malignancy was reported as well. Prior to this, he had been treated with multiple courses of antibiotic therapy without clinical improvement.Both systemic and chest examinations revealed no significant abnormalities and there was no lymphadenopathy. Further investigations with chest radiograph showed right middle zone consolidation and computed tomography (CT) of the thorax revealed subsegmental collapse with bronchiectasis of the right middle lobe and nodular calcified excrescences were seen protruding into the lumen of trachea and main bronchi Fig. . There wTO is an uncommon benign large airway disorder characterized by bony or cartilaginous nodules on tracheal and bronchial wall. With only less than 400 cases of TO reported in the literature , it is mTO often follows a benign course  where paIn summary, TO should be considered as a possible differential diagnosis in patients presenting with chronic cough and recurrent respiratory infection. Albeit rare, early recognition of this rare disorder is important in guiding appropriate management for these patients.Appropriate written informed consent was obtained for publication of this case report and accompanying images.Suat Yee Lim and Mohd Faizul Abu Samah conceived the idea and wrote the manuscript with consultation and guidance of Lalitha Pereirasamy and Irfhan Ali Hyder Ali. Bee See Chew contributed to the analysis and description of the histological image. All authors reviewed and gave the final approval of the version to be published.


Vagus nerve schwannoma is a very rare benign nerve tumor. Pheochromocytoma is a rare, mostly benign tumor of the adrenal medulla with a large clinical spectrum. Their association is uncommon. The management of both tumors depends solely on surgery. The surgery of vagal schwannomas is particularly challenging considering the anatomical compositions of the area. Here, we report a case of a 76 year-old patient with cervical vagal schwannoma and benign pheochromocytoma association. We discuss the diagnosis and the surgical management of these tumors. •Vagus nerve schwannoma is a very rare benign nerve tumor.•Pheochromocytoma is a rare catecholamine-secreting tumor that originates in the adrenal medulla and is responsible for secondary hypertension.•We describe the case of a patient with pheochromocytoma, in whom a cervical vagal schwannoma was discovered incidentally. It is majorly asymptomatic and rarely undergoes malignant transformation . The prePheochromocytoma is a rare catecholamine-secreting tumor that originates in the adrenal medulla and is responsible for secondary hypertension . Its diaTo our best knowledge, few cases of their association have been reported in the literature. Here, we describe the case of a patient with pheochromocytoma, in whom a cervical vagal schwannoma was discovered incidentally. Based on a literature review, we discuss the diagnosis, pathogenesis, surgical management and genetic disorders of this association. This case report follows SCARE guidelines 2020 .2We report a case of a 76 year-old man, with a history of Type 2 diabetes on oral antidiabetic drugs. The patient was presented in the emergency department with the classic triad of: headache, sweating, and heart palpitation and hypertension. He was non-smoker and has no other medical or family history. Laboratory tests were done, including 24-h urine fractioned metanephrine and catecholamine, were elevated. Twenty days earlier, he had been hospitalized to the regional hospital for a hypertensive crisis at 240/105 mmHg associated with headaches, palpitations and hot flashes. On physical examination, he had normal cardiopulmonary and neurological systems. Notably absent were symptoms of nausea, vomiting, bloating, and change in bowel habit or weight loss.Cervical and thoraco-abdominal computed tomography (CT scan) revealed a large single mass of the right adrenal gland. It was heterogeneous with areas of necrosis and cystic change, measuring 64 × 57 mm . BesidesBased on the clinical and radiological findings, the therapeutic approach in this case consisted of an urgent open right adrenalectomy. The patient was ready to undergo surgery by an experienced surgeon after medical preparation with α-adrenergics and hypertension control. The exploration of the right adrenal gland confirmed the presence of a 9 cm cystic mass that was carefully extracted. No extra-adrenal nodules were seen. The procedure lasted 90 min, with an intra-operative diuresis of 600 ml and bleeding loss estimated at 100 cc. The post-operative recovery of the patient was without incident, and two weeks later, we noted the normalization of blood pressure and urinary catecholamines.The histopathological examination confirmed the diagnosis of pheochromocytoma, .Fig. 3MiThe patient refused the exploration of his cervical lesion associated with pheochromocytoma and was lost to follow-up. Six months later, the patient presented herself in our department with complaint of headaches and cervical swelling. Vital parameters and all laboratory investigations, including blood exam, kidney function and urinalysis, were within normal limits.Under general anesthesia, the patient underwent a cervicotomy with an excision of the cervical mass, with uneventful postoperative course. The histopathological examination confirmed the diagnosis of schwannoma arising from the vagus nerve .Fig. 4MiThe patient currently continues to be asymptomatic and disease-free 24 months later. There was no evidence of recurrence.Taking the cunt association of the pheochromocytoma and the cervical schwannoma, we suspected a possible genetic alteration. A genomic analysis was indicated, but the patient was refused this genetic testing.3Cervical vagal schwannomas are extremely rare slow-growing tumors originating from the vagus nerve. They are covered with Schwann cell sheath . SchwannThese tumors are usually asymptomatic and present as a neck mass. If there are symptoms, they are majorly represented by hoarseness and occasionally a paroxysmal cough when palpating the mass, which is a pathognomonic sign of vagal schwannoma .Cervical computed tomography (CT) with contrast can help in the diagnosis of this tumor, but magnetic resonance imaging (MRI) remains the tool of choice for the diagnosis and determination of the nerve origin, but also for providing pre-operative information useful in planning surgical treatment . Fine-neThe gold standard of treatment is the surgery that consists of intracapsular enucleating of the tumor in order to minimize nerve damage, guided by a nerve stimulator and the surgical microscope. Conventional extracapsular excision with end to end anastomosis has been done with mixed results, because it can damage the normal fascicles during the dissection of the capsule ,9. The mHistologically, schwannoma typically show two main patterns, a very cellular area made of spindle cells with palisading of cell nuclei (Antoni A) and a less cellular area (Antoni B) with strong staining of S-100 protein.Given the benign nature of most vagal schwannomas and their slow-growing evolution, a conservative approach should always be considered. In our case, the patient started to show cervical pain and hoarseness leading to the decision of surgical excision.Pheochromocytoma is a rare neuroendocrine tumor originating in the adrenal gland medulla and producing catecholamines and other neuropeptides that are responsible for secondary hypertension. They are often seen between the 3rd and 5th decades of life. According to literature, 10–25% of the cases of pheochromocytoma can be associated with genetic and familial syndromes (multiple endocrine neoplasia type 2 (MEN 2), type 1 neurofibromatosis and Von-Hippel-Landau disease in younger ages [The typical triad of headache, sweating and heart palpitations guides the diagnosis and elevated plasmatic or urinary metanephrine and normetanephrine confirms it. Computed tomography (CT) and magnetic resonance imaging (MRI) help locate the tumor .Surgical excision remains the best treatment option after a thorough medical preparation with α adrenergics and eventually β adrenergics.The association of both tumors in a patient is very rare, and to our best knowledge, only few cases have been reported .Given the presence of pheochromocytoma, the surgery should focus first on the adrenal gland. The cervical vagus schwannoma can be postponed. A testing for genetic disorders should be suggested to our patient. The major limitations in term of diagnosis of this association are the determination of the nature of these tumors, and the surgical management.4This case report sheds light on the rarity of cervical vagus schwannoma and its association with benign pheochromocytoma. A thorough study of this association should be done, including researching for genetic disorders in order to comprehend this association.No ethical approval necessary.All sources of funding should be declared as an acknowledgement at the end of the text. Authors should declare the role of study sponsors, if any, in the collection, analysis and interpretation of data; in the writing of the manuscript; and in the decision to submit the manuscript for publication. If the study sponsors had no such involvement, the authors should so state.The author(s) received no financial support for the research, authorship and/or publication of this article.Dr Harhar Marouane: Have written the article, have consulted the patient, prescribed all of the tests and prepared the patient for surgery and participated in the surgery. Dr Harouachi Abdelhakim: have helped writing the article, data collection. Pr Bouhout Tariq (oncology surgery professor): have supervised the writing of manuscript. Pr Serji Badr (oncology surgery professor): have supervised the writing of the paper. Pr El Harroudi Tijani (oncology surgery professor): Writing, review and editing of the manuscript, and had been the leader surgeon of the case.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Our paper is a case report; no registration was done for it.Harhar Marouane.Not commissioned, externally peer reviewed.The authors declared no potential conflicts of interests with respect to research, authorship and/or publication of the article.


Bowel obstructions can have a variety of causes, including impacted feces, adhesions, volvulus, non-internal hernias, and in rare cases internal hernias. We report a 63-year-old woman who presented to the emergency department with severe abdominal pain, nausea, vomiting, and obstructive symptoms that had started 12 hours earlier. A computed tomographic scan of the abdomen and pelvis showed a right internal hernia with a cecal bascule traversing through the foramen of Winslow, concerning for a closed-loop obstruction. The patient underwent an exploratory laparotomy with cecal bascule reduction and cecopexy. Given the increased mortality risk if undiagnosed, it is important to remain aware of internal hernias. Patient outcomes are markedly improved with early diagnosis and surgical intervention.                             A cecal bascule is a rare condition in which the cecum folds anteriorly and superiorly over the ascending colon.                            A 63-year-old woman with a history of gastroesophageal reflux disease (GERD) associated with a small hiatal hernia presented to the emergency department with severe abdominal pain, nausea, vomiting, and obstructive symptoms that had started 12 hours earlier. The pain was epigastric in nature. She reported no passing of flatus or bowel movement for 24 hours prior to presentation. She also reported a history of intermittent abdominal pain and obstipation that would self-resolve without treatment over the last several years. She had no prior surgical history. Her body mass index was 31. She denied any fever, chills, chest pain, or shortness of breath. Her vitals on presentation included a blood pressure of 110/70 mm Hg, heart rate of 105 beats per minute, and respiratory rate of 22 breathes per minutes. On physical examination, the abdomen was distended with focal tenderness in the epigastric and peri-umbilical regions. No peritoneal signs were noted.            Laboratory analysis revealed a leukocytosis to 12.9 K/uL  with an elevation in creatinine to 0.8 mg/dL from 0.4 mg/dL . Her blood urea nitrogen level was elevated to 20 mg/dL from 8 mg/dL . Her lipase, amylase, and liver function tests were within normal limits. Her lactate level was mildly elevated to 3.0 mg/dL .            A computed tomography (CT) scan of her abdomen and pelvis revealed a right internal hernia containing cecal bascule herniation through the foramen of Winslow into the lesser sac, concerning for a closed-loop obstruction . There w            The patient’s symptoms including pain, nausea, vomiting, no flatus or bowel movements, along with her leukocytosis and imaging findings were concerning for an internal hernia with a cecal bascule traversing the foramen of Winslow causing a closed-loop obstruction. The patient was taken to the operating room.            She underwent an exploratory laparotomy. After fixed retractors were placed, the appendix was noted to be in the right upper quadrant near the gallbladder and liver edge . The ter            Given the viability of the cecum and right colon, the decision was made to perform a cecopexy rather than right hemicolectomy to minimize the risk of a recurrent internal hernia. An appendectomy was also performed. Omentum was placed near the foramen of Winslow to also help prevent future risk of a recurrent internal hernia. No attempt was made at suture closure of the foramen of Winslow space. The abdomen was closed in the standard fashion.            The patient’s postoperative course was uncomplicated. Her pain was well-controlled with oral pain medication. Her diet was advanced in a stepwise fashion. She was able to ambulate independently and void spontaneously upon discharge. The patient was discharged 4 days following her operation. She has also been doing well more than 6 months out from surgery.                            ,An internal hernia is a rare cause of intestinal obstruction, accounting for less than 5% of cases.            -Although numerous organs have been known to herniate through the foramen of Winslow, cecal herniation is rare. Cecal herniation through the foramen of Winslow has only been reported in a limited number of cases. These cases include a cecal bascule herniation, cecal herniation along with the ascending colon and terminal ileum, cecal herniation with volvulus, and cecal herniation in the absence of a bascule.            ,,With the advent of computed tomography (CT) imaging, diagnosing cecal herniation through the foramen of Winslow preoperatively has become easier compared to abdominal plain films and barium enemas. A CT scan may help provide the best anatomical delineation and is now the standard method of detection.            ,,,,A cecal bascule is a rare condition where the hypermobile cecum can fold anteriorly and superiorly leading to obstruction . Differe            ,,After a diagnosis of a cecal bascule herniation through the foramen of Winslow has been made, prompt definitive treatment is required to minimize the risk of strangulation. Although some reports describe laparoscopic approaches for internal hernias through the foramen of Winslow,                            Patients who present with acute onset abdominal pain with associated gastrointestinal obstructive symptoms require prompt workup for bowel obstruction secondary to internal hernia or closed-loop obstruction. A cecal bascule through the foramen of Winslow with closed-loop obstruction is a rare condition that warrants emergent surgical management given risk of colon strangulation. The risks and benefits of cecopexy compared to a right hemi-colectomy must be weighed carefully based on intraoperative bowel viability.


Foreign bodies inside the orbital cavity are rare. They may lead to serious complications, depending on their nature, size and mechanism of injury. A 29-year-old male presented with a motorcycle handle embedded in his left orbit, with the vision unaffected. Active wound bleeding, increasing hematoma, a low haemoglobin level, signs of hypovolemic shock, ocular acuity and mobility were investigated. A computed tomography scan revealed a long bent metallic object lodged between the globe and floor of the left orbit with fracture of the medial orbital wall and ethmoidal hemosinus with an intact cribriform plate of the ethmoid. An interdepartmental collaborative effort of Oculoplasty, Oro-Maxillofacial and Neurosurgery were utilized in the removal of the foreign body. The patient recovered well after the surgery and a course of antibiotic therapy.A single large round-tipped foreign body in orbit composed of both metal and plastic is an extremely rare incident and fortunately in our case, was relatively harmless despite its large size. The diagnosis and management of intraorbital foreign bodies must be tailored according to their type and a proper localisation by all possible means, blunt dissection, careful haemostasis aided with good lighting, and exposure helps in their atraumatic removal. Intra-orbital foreign bodies (FBs) are sequelae of facial trauma with an object. The incidence of intra-orbital FB is high in military practices; however, their occurrence in civilian life is uncommon and are predominantly due to road traffic accidents. A high-velocity injury, like a gunshot or an industrial accident, usually precedes the situation ,2.A 29-year-old male patient was brought to the emergency department with a history of road traffic accident Figure , Panel aOn local examination, a motorcycle handle was seen lodged in his left orbit, passing through the lower eyelid. His left upper and lower eyelids were swollen and chemosed. There was a severe limitation of extraocular muscle movements in the left eye. Visual acuity was 6/18 in the left eye with no relative apparent pupillary defect. On indirect ophthalmoscopy, posterior segment examination of both eyes was normal. There was no evidence of any injury to any other parts of the body.A computed tomography (CT) scan of the head and orbit revealed a well-defined iso-dense structure in the left orbit lying in between the globe and the floor (Figure The FB was a metallic handle measuring 10.3 cm in length and 2 cm in diameter (Figure Intra-orbital FBs are infrequent situations; they have an incidence of 2.9%. The common causes for intraorbital FBs are gunshot injuries, industrial mishaps and road traffic accident as in our case . They arThe clinical presentation for orbital FBs varies from being asymptomatic to having visual disturbances, pain and swelling. They may not always be as evident as in our case, a small FB may get hidden in orbit and the surrounding orbital fat can conceal the trajectory. In such cases, the patient may have persistent inflammatory signs, limitation of ocular movements and diplopia.A thorough radiological assessment aids in pinpointing the exact site of the FB. It can help in roughly estimating the nature and size, and the amount of peri-orbital tissue inflammation. It also provides vital information regarding the integrity of the globe. The choice of imaging modality is determined by the suspected nature of the FB .Plain X-ray can be utilized to locate the FBs; however, these films lack the ability to provide the exact location in relation to adjacent tissue response or surrounding damage . UltrasoSurgical removal of the FB is indicated in situations as a large or sharp-edged FB, presence of signs of infection or inflammation, evidence of proptosis, restricted motility, presence of optic nerve compression, etc. . The surThe literature suggests that in cases of inorganic FBs, the need for surgery depends on their location . AnterioMetals and glass are well tolerated, and if not causing any symptoms or signs, may be left in situ . All orgLarge intraorbital foreign bodies may not always be associated with severe vision loss or gross ocular morbidity. A multidisciplinary approach with timely intervention can yield excellent outcomes.


Gastric lipomas are very rare benign tumors. Only around 217 cases have been reported. Most gastric lipomas are found incidentally; however, larger neoplasms can be symptomatic. The presented 64 years old male with incidental finding of pyloric lipoma adds another example to the few documented in the literature. The patient had symptoms of breath shortness and lack of energy two months after COVID 19 pneumonia. A low dose CT scan with iodine contrast enhancement of the chest and upper abdomen led to the diagnose. The diagnosis of gastric lipoma can be achieved through diagnostic imaging or the combination of endoscopic techniques. Treatment can be carried out by endoscopy, robotic or classical surgery. Gastric lipomas are rare tumors, comprising only 1-3% of benign stomach tumors The reported case is of a 64-year-old man. The patient had symptoms of breath shortness and lack of energy two months after COVID 19 pneumonia. 256 Helical CT scan with low dose protocol and iodine contrast enhancement of the chest and upper abdomen is performed. After the scan and the incidental finding of the pyloric lipoma, the patient was questioned about clinical symptoms. He described several instances of heartburn and epigastric pain after meals in the last year. He has not experienced vomiting or other upper dyspeptic symptoms.The CT scan of the upper abdomen shows an oval, well-encapsulated formation in the pylorus of the stomach. Due to the contrast enhancement, the lesion appears non homogeneously contrasted but remains with fatty density. Measurements of CT density after contrast enhancement in Hounsfield Units (HU) are between -85 and -46 in different locations of the lipoma. . The forAfter the incidental finding and CT diagnosis of pyloric lipoma, the patient is advised to consult an abdominal surgeon for histological verification and treatment planning.Gastric lipomas are very rare, slow-growing benign tumors formed by mature adipose tissue. The colon is the most common site of involvement within the gastrointestinal tract, followed by the ileum and jejunum Lipomas close to the pylorus, such as in the presented case, can cause obstructive symptoms. They can be caused by blocking the pylorus or protruding into the duodenum. It is reported that a lipoma of a size greater than 2.0 cm will present with abdominal pain more than 50% of the time; however, 37% of patients will have a presentation of chronic or acute GI bleeding, obstruction, and dyspepsia The most important differential diagnosis of gastric lipoma is liposarcoma. Differential diagnosis can also be made with soft tissue tumors of the gastrointestinal tract, such as leiomyoma or fibroma, neurilemoma adenomyoma, Brunner gland adenoma, and ectopic pancreas Usually, on barium studies, extra mucosal neoplasms, including lipomas, appear as a well-defined filling defect with a 'bull's eye' appearance.3 CT is a precise imaging diagnostic tool for lipoma Upper GI endoscopy will show a submucosal mass and three signs which help diagnosis In some cases, CT may produce a diagnosis without having to perform an endoscopy. More accurate diagnosis of gastric lipoma can be achieved through the combination of diagnostic imaging and endoscopic techniques. Treatment can be carried out by endoscopy, robotic or classical surgery, depending on the specific case.Patient consent has been obtained.


Approximately, there are about 200 cases of polyorchidism in the medical literature. In the past, surgical treatment was done but now with imaging studies, less aggressive approach is recommended.Here we present a case of 40-year-old man who was diagnosed incidentally with polyorchidism in the right hemiscrotum which is quite unusual in this age.A 40-year-old man presented to the emergency department with a swelling and pain in the left hemiscrotum. In palpation, we noticed a scrotal mass in the right hemiscrotum. His parents had first noticed a scrotal mass when he was two years old and was incorrectly diagnosed as hydrocele by an unauthorized practitioner.In the left hemiscrotum, Doppler confirmed acute epididymitis diagnosis that was treated conservatively with antibiotics and NSAIDs. In the right hemiscrotum, MRI showed that the lump had separate epididymis and shared a common vas deferens with the right testis, which confirmed the diagnosis of supernumerary testis and the patient underwent a follow-up ultrasound after a month and after six months of his presentation.Triorchidism is the most common type of polyorchidism. Polyorchidism is diagnosed incidentally hence it is asymptomatic. There are many types of Polyorchidism and tow classification have been described. When the patient is asymptomatic the concentrative treatment is recommended.Polyorchidism is a rare congenital anomaly in the genitourinary tract. It is diagnosed incidentally. Ultrasound or MRI are used to diagnose polyorchidism cases. •This Case Report is about a rare condition (polyorchidism) in a 40-year-old man which is very unusual in this age.•Diagnosis has been made by imaging studies only. No need for aggressive treatment in uncomplicated cases.•This case report provided a brief review about the classification of Polyorchidism and other differential diagnosis. It is defined as the presence of three testes or more.Polyorchidism has many forms; Triorchidism (three testes) is the most common type. Bilateral double testis (4 testes) is a rare type .Ahlfeld first described polyorchidism histologically in 1880. It was first reported by Lane in 1895 as clinical case .Approximately, there are about 200 cases of polyorchidism in the medical literature .Most cases of Polyorchidism are asymptomatic and are diagnosed incidentally. About 65% of polyorchidism cases were reported on left side with median age 17 years [Ultrasound or MRI help in diagnosis and follow polyorchidism patients .Here we present a case of 40-year-old man who was diagnosed incidentally with polyorchidism in the right hemiscrotum which is quite unusual in this age. This case report has been reported in line with the SCARE Criteria [2A 40-year-old man presented to the emergency department with a swelling and pain in the left hemiscrotum with no history of trauma or surgical procedures. Therefore, he was referred to the urology department. Drugs and Family history are unremarkable, the patient does not take any medication and he does not recall any type of allergy. He smokes one pack-year and does not drink alcohol and works as trader.Physical examination indicated an acute left epididymitis. In palpation, we noticed a scrotal mass in the right hemiscrotum.His parents had first noticed scrotal mass when he was two years old and was diagnosed incorrectly as hydrocele by an unauthorized practitioner. He had not undergone any further investigation.We performed US, which showed normal left testis and enlarged left epididymis with hypoechoic. In addition, we performed Doppler evaluation to demonstrate blood flow, which was increased. Therefore, we confirmed acute epididymitis diagnosis that was treated conservatively with antibiotics and NSAIDs. In the right hemiscrotum, Color Doppler ultrasound evaluation revealed oval lump with echotexture and blood flow similar to the ipsilateral testis .Fig. 1CoWe put Triorchidism and adenomatoid tumor as differential diagnosis for this lump.The Serum tumor markers were in the normal range. , and .MRI showed that the lump had separate epididymis and shared a common vas deferens with the right testis, that was not obvious on Doppler ultrasound evaluation, which confirmed the diagnosis of supernumerary testis (Triorchidism). The supernumerary testis had intermediate signal intensity on T1-weighted images and high signal intensity on T2-weighted images and that is the same of normal testes .Fig. 2 classification and type C according to Leung classification.The patient underwent a follow-up ultrasound after a month and after six months of his presentation. Then we put the patient on regular follow-up yearly. The patient is still radiologically stable 6 month after his presentation, with his radiologic follow-up the patient has good prognosis, and he did not have any complications as the clinician expected.3Testis begins to form around 6th week of gestiaon from genital ridge which is a small bulge on the dorsal coelomic wall that contain migratory primordial germ cells. There are many theories that point to the cause of polyorchidism such as anomalous appropriation of cells, duplication or transverse vs longitudinal division of the urogenital ridge, incomplete degeneration of a portion of the mesonephros and development of peritoneal bands ,5.Triorchidism is the most common type of polyorchidism. However, bilateral double polyorchidism (4 testicles) had been reported . The lefThe median age of polyorchidism cases is 17 .Here we present a case of 40-year-old man who was diagnosed incidentally with polyorchidism in the right hemiscrotum which is quite unusual in this age.Polyorchidism is diagnosed incidentally hence it is asymptomatic. However, it could be symptomatic when it is associated with cancer . In our Based on Leung classification there are four types of polyorchidism: Type A. the supernumerary testicle has neither epididymis nor vas deferens and has no connection to the other testicles. Type B. the supernumerary testicle shares the same epididymis and vas deferens of the other testicles. Type C. the supernumerary testicle has separate epididymis but shares a vas deferens with the other testicles. Type D. the supernumerary testicle has separate epididymis and vas deferens .Furthermore, in a meta-analysis study another classification has been described: Type A when it is connected to vas deferens with subgroups: A1 separate epididymis and vas deferens. A2 separate epididymis but shares vas deferens with other testicle A3 shares epididymis and vas deferens with other testicle. A4 separate vas deferens but shares epididymis with other testicle. It is classified as Type B when it has no connection to vas deferens with subgroups: B1 separate epididymis. B2 only testicular tissue. The most common type is A3 [In our case, the classification of the Supernumerary testis was type A2 according to Bergholz and Wenke (2009) classification and type C according to Leung classification.In the same meta-analysis, 73% of the cases were diagnosed histologically which is quite different in our case where we only diagnosed the case with imaging studies . This alDifferential diagnoses for polyorchidism are: encysted hydrocele, spermatocele or varicocele, Morgagnian cyst or testicular neoplasm, therefore, physical examination alone is not enough to diagnose polyorchidism. Imaging study like Ultra Sound, Doppler and MRI are essential for diagnosis, US is the most common imaging study and can confirm the diagnosis, MRI is less common ,8.We did not confirm the diagnosis by histological examination because the patient refused the biopsy and there is no need for it because there was no neoplastic signs and the imagining study alone confirmed polyorchidism .The treatment of polyorchidism is still debated. In the past, Surgical removal of supernumerary testicle was preformed but the presence of imaging study changed the way we treat polyorchidism, when the patient is asymptomatic Type A (our patient) the concentrative treatment is recommended, but the patient should be monitored regularly because there is an increased number of malignancy in case of polyorchidism. If the malignancy was confirmed a surgical removal is recommended . Based o4Polyorchidism is a rare congenital anomaly in the genitourinary tract. However, it should be considered as a differential diagnosis of scrotal mass despite its rarity. It is diagnosed incidentally. Ultrasound or MRI are being using to diagnose polyorchidism cases.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.Data used on this case report are available from corresponding author on reasonable request.AK and MEJ: Revision of the manuscript. MM, MZBA and BB: Drafting of the manuscript. OA: Conception and design of the study. AK: Approval of the final version of the manuscript.All authors confirm that they have read and approved the final manuscript.Owais Aleter.Not commissioned, externally peer-reviewed.The patient signed consent form to publish this case with the figures.The patient was informed and approved the manuscript.None.ApprovedNone.1. Name of the registry:2. Unique Identifying number or registration ID:3. Hyperlink to your specific registration (must be publicly accessible and will be checked):Owais Aleter.No funding was obtained for this case report.Authors declare they have no conflict of interest.


Retroperitoneal sarcoma (RPS) is a rare and heterogeneous tumor. A percutaneous core needle biopsy (PCNB) is required for obtaining a histological diagnosis of the condition and for planning the therapy. Surgery is the standard of care for RPS; it is a standardized procedure, and it should be performed in a referral sarcoma center. Sarcoma surgery is rarely performed on an urgent or emergent basis. In this report, we describe a case of a retroperitoneal leiomyosarcoma that presented with spontaneous rupture and hemoperitoneum, which required surgical treatment in an urgent manner. To our knowledge, this is the first case of hemoperitoneum due to RPS rupture to be reported in the literature. Retroperitoneal sarcomas (RPS) is a rare tumor characterized by wide heterogeneity in histology and behavior. Its annual worldwide incidence is 0.76 new cases per 100,000 people according to the latest findings . In caseA 53-year-old man presented to the emergency department of another hospital with abdominal pain and severe weight loss (12 kilograms). An abdominal CT scan showed a 10-cm left retroperitoneal mass. According to the latest European Society for Medical Oncology (ESMO) Guidelines , a CT-guThe tumor was in direct contact with the left kidney, the pancreatic tail, and the psoas muscle. The superior mesenteric artery was free of infiltration. The chest CT scan was normal. Blood examination revealed severe malnutrition: hemoglobin of 9.6 g/dl, albumin of 24.8 g/l, and prealbumin 0.14 g/l. The coronavirus disease (COVID-19) nasal swab test was negative. Considering the clinical deterioration of the patient and the presence of hemoperitoneum, which proved the spontaneous bleeding of the mass, the decision to conduct surgery as a delayed emergency was made. A median laparotomy extending transversally to the left flank was performed, and 2.5 liters of hemorrhagic ascites were evacuated. A large tumor perforation was identified in the distal part of the tumor with colon perforation. The tumor had extensively invaded the peritoneal layer and it was spontaneously bleeding over the entire surface. The left kidney was completely encompassed by the tumor. The first step of surgical resection was the liberation of the root of the mesentery, contiguous but not infiltrated by the tumor. After coloepiploic separation, transverse colon and rectosigmoid junction were divided. The distal pancreas and spleen were indivisible from the tumor. Given the extension of the mass, we decided to perform a distal splenopancreatectomy. Vascular dissection was performed from the iliac vessels up to the aorta with a sub-adventitial dissection. In the left iliac fossa, the ureter was divided, and the femoral nerve was isolated. The left renal vein and artery were divided by stapler and ligation, respectively. The left psoas fascia was detached from the vertebra and it was left posteriorly to the tumor. Finally, the tumor was removed en-bloc with the left kidney, ipsilateral colon, mesocolon, spleen, and distal pancreas in front, aponeurosis of the psoas in the back, and parietal peritoneum on the side of the mass  and then by an exploratory laparotomy on the 14th POD. During surgery, no active bleeding had been found and peritoneal cavity lavage with saline solution had been performed. After the resolution of the pancreatic fistula, the surgical drains were removed and the patient was discharged on the 42nd POD. The histological exam revealed a 16-cm dedifferentiated leiomyosarcoma (FNCLCC grade 3). The tumor showed high mitotic activity (23 mitoses/10 high-power fields) and areas of necrosis (up to 50%). Resection margins were negative. Tumor rupture and colic perforation were confirmed by the pathologist. The multidisciplinary tumor board composed of surgical oncologists, oncologists, radiotherapists, radiologists, and pathologists discussed the clinical case after surgery and decided to have the patient undergo follow-up. A thoracoabdominal CT scan after four months was recommended. No further adjuvant systemic treatments were deemed to be indicated in this case.Sarcoma surgery is an elective procedure, usually performed by an expert sarcoma team after PCNB. En-bloc resection with involved or adherent organs is a standard procedure with adjustments according to the presentation of sarcoma  ,5. In raIn our patient, the large perforation of the tumor surface and left colon were probably not related to PCNB, but rather to the spontaneous perforation of the necrotic part of the tumor demonstrating the fast-growing of the mass. We also found peritoneal infiltration, spontaneous bleeding of the mass, and hemorrhagic ascites intraoperatively. Peritoneal infiltration should not justify a more limited resection . The bloSpontaneous RPS rupture is an extremely rare event. However, surgical techniques as previously described, which recommend compartmental surgery, should be considered even in this rare presentation. Spontaneous RPS rupture exposes the patient to peritoneal seeding and sarcomatosis and is associated with a poor prognosis.


Lung cancer associated with cystic airspaces, especially solitary multicystic lesion lung cancer, is a rare disease . It is difficult to diagnose owing to the lack of a clear definition; therefore, diagnosis of these neoplastic lesions remains challenging.We outlined two cases of elderly Chinese men who were admitted to the hospital with a solitary multicystic lesion of the lung and subsequent surgical resection, confirming a diagnosis of adenocarcinoma.For solitary pulmonary cystic airspaces , it is important to properly recognise their imaging features. Due to the possibility of malignancies, timely surgery is an effective treatment strategy for early diagnosis. Lung cancer associated with cystic airspaces (LCCA) is an uncommon manifestation, representing only 1–7% of all lung cancers , 3. In cAfter Womack and Graham first suggested that pulmonary cystic lesions are possibly associated with bronchogenic carcinoma in 1941, Anderson and Pierce reported on carcinoma of the bronchus presenting as thin-walled cysts for the first time in 1954 . Since tIn this case report, we present two male patients admitted to the hospital with a solitary multicystic lung lesion who had undergone surgical resection, confirming a diagnosis of adenocarcinoma. A review of the conditions focusing on their diagnosis and treatment is also included.A 66-year-old man, who had smoked 20 cigarettes per day for 40 years, presented to the pulmonary department with sudden onset haemoptysis for 1 week. He had other concurrent symptoms, including a chronic wet cough for 20 years, with no associated dyspnoea, chest pain, or weight loss. The patient had no history of any other disease. On initial physical examination, he had a blood pressure of 154/105 mmHg, with a pulse rate of 77 beats per minute (bpm). He had normal heart sounds and clear lungs with no dry or wet rales on auscultation.Routine laboratory investigations were normal, including complete blood count, serum urea, and electrolyte levels. Tumour markers  were all within normal limits. Chest computed tomography (CT) demonstrated a solitary multicystic lesion with a thin-wall measuring 24 mm × 12 mm in the right upper lobe  of 61% and a ratio of FEV1/forced vital capacity (FVC) reduced to 58.6% (< 70%), suggesting an obstructive ventilatory disturbance due to smoking; this indicates that the patient has chronic obstructive pulmonary disease (COPD).Two days later, flexible bronchoscopy revealed only blood clots, and bronchoalveolar lavage in the right upper lobe bronchus found neither malignant cells nor acid-fast bacilli (AFB). Noting that the patient already had haemoptysis and that other diseases commonly causing haemoptysis  were not initially considered, a percutaneous CT-guided biopsy of the solitary multicystic lesion was performed two days later. Since the cystic airspace was completely thin-walled, two small pieces of lung tissue were penetrated. Following tissue biopsy fixation, processing, sectioning and routine histochemical staining, histological examination showed alveolar epithelial hyperplasia (AEH) with an adherent growth pattern without excluding adenocarcinoma in situ, microinvasive adenocarcinoma, or invasive adenocarcinoma due to the lack of tissue.Due to the small amount of tissue acquired with biopsy under transthoracic puncture, the pathological result did not present an exact diagnosis of LCCA. However, the presence of atypical AEH cells raised speculation regarding early LCCA. Surgery is one of the most reliable therapies for pulmonary adenocarcinomas. We subsequently performed a right upper lobectomy with lymph node dissection using video-assisted thoracoscopic surgery (VATS). Grossly, the sliced resected specimen revealed a tan-whitish cystic tumour, 2.3 cm in length, scattered with small nodules. However, lung cancer was not identified in the frozen section, supporting the diagnosis of a benign mass.Three days later, a histopathology report documented lepidic-predominant adenocarcinoma with 40% invasive acinar adenocarcinoma. Fig. . The lesPatient 2 was a 56-year-old man who had no smoking history and presented to the pulmonary department with no symptoms. Radiological examination revealed a cyst measuring 10 mm × 8 mm in the left upper lung  has been reported since the 1940s; however, it is still currently considered as a relatively rare tumour (a rare imaging performance of NSCLC). In 2017, Fintelmann et al. [As LCCA has an overall low prevalence, it is easy for radiologists and respiratory doctors to miss it . PatientTherefore, CT image features and possible carcinogenic mechanisms may help us understand and diagnose cystic airspace cancers , 9. CurrCystic airspaces are not always present. Most LCCA lesions have new or increased nodular components or thickening of the wall over time. Sheard et al.  reportedAfter a CT scan shows the initial examination of cystic airspaces, appropriate further radiological and invasive operations may be required for the diagnosis of LCCA. Normal clinical inspection such as transbronchial biopsy, percutaneous lung biopsy, and positron emission tomography-computed tomography (PET-CT) are helpful but insufficient for special types such as type IV multicystic lesion, as mentioned in the two cases .Mendoza et al. found that more than 300 cases of LCCA have been described  in the fIn contrast, PET-CT has a number of drawbacks in LCCA. Small and cystic air space lesions, multicystic type IV in particular, may reduce the overall density of metabolically active cells; hence, the uptake of fluorine-18-fluorodeoxyglucose (FDG) in a small pericystic lesion can be difficult to measure, and a negative PET result does not reliably exclude malignancy , 23, 24.We report two cases of type IV LCCA that showed multicystic lesions. With increased physician suspicion and improved CT scan and puncture biopsy techniques, the diagnosis rate of LCCA has increased, but it is still often overlooked and misdiagnosed. The majority of cancers associated with cystic airspaces are of the adenocarcinoma type. Awareness of these CT features is important, and the limitations of preoperative biopsy and PET-CT should be recognised. Timely surgery is essential for early diagnosis.


Eosinophilic granuloma (EG) is the unifocal osseous form of Langerhans cell histiocytosis (LCH), which usually affects the skull and long bones. Although it most commonly affects the pediatric age group, it can rarely be seen in adults. Skeletal involvement is common in adult patients, but isolated rib involvement is extremely rare. Differential diagnosis includes other osteolytic lesions such as Ewing’s sarcoma, tuberculosis, multiple myeloma, lymphoma, and primary bone malignancy.The diagnosis must be confirmed histopathologically. In addition to pathological Langerhans cells, inflammatory cells such as lymphocytes, eosinophils, and macrophages are observed in microscopy. Immunohistochemically, CD1a, S-100, and Langerin positivity are observed in biopsy and/or surgical excision material.Treatment options may vary depending on the localization and extent of the disease. In unifocal EG, close observation of the patient may be preferred, as well as surgical excision, radiotherapy, and intra-lesional steroid administration. The prognosis in patients with a single bone lesion is quite good compared to other groups.In this case report, we present a metachronous EG of rib developed in two different ribs by an interval of seven years, which were both surgically treated. In this mild variant of LCH, surgical resection with clean margins has a favorable outcome without the need for additional adjuvant therapy. Metachronous tumors may develop in isolated unifocal bone EGs, and long-term follow-up is mandatory. Langerhans cell histiocytosis (LCH) is an idiopathic disease that occurs as a result of the localized or widespread accumulation of Langerhans cells originating in the bone marrow in various tissues for an unknown reason . AlthougEosinophilic granuloma (EG) is the unifocal osseous form of LCH, which usually affects the skull and long bones . SkeletaInformed consent was obtained from the patient for publication of this case. A 39-year-old male patient, admitted with chest pain in 2014, was found to have an expansile lesion in the left fifth rib in his radiological examinations, and surgical resection was decided , an expansile hypodense lesion of approximately 3 cm x 2 cm was observed in the lateral arc of the left fourth rib. F-18 fluorodeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) showed pathological uptake  in the lytic lesion in the left fourth rib and also changes secondary to the previous operation in the anterolateral arcs of the left fifth to sixth ribs Figure . SurgicaThe patient was discharged without any problems on the second day after the operation. The patient was evaluated by the multidisciplinary council; no additional treatment was required, and close follow-up was recommended. The patient was doing well in his second month of follow-up.LCH is a disease characterized by monoclonal proliferation of Langerhans cells of bone marrow origin; the exact etiology is unknown. Symptoms vary according to the localization and affected organ. The annual incidence was reported as 2-9/1.000.000 cases in the pediatric group (under the age of 15). But it is seen rarer in adults (1-2/1.000.000 cases per year) ,4.EG, which is the mildest variant and localized form of LCH, may present in two ways: a single bone lesion (monostotic) or multiple bone lesions (polyostotic). Skeletal involvement is more common in men. The most frequent site is mandible 30%) followed by cranium (21%), vertebrae (13%), and extremities (13%). While the rib involvement is rare (6%), it is more frequent in adults than in the pediatric population. In a report by Oh et al. including 211 adults and 330 pediatric patients, the rib involvement ratio was 25% for adults and 12% for the pediatric group [0% followIsolated rib involvement is extremely rare, and its diagnosis is challenging. Differential diagnosis includes other osteolytic lesions such as Ewing’s sarcoma, tuberculosis, multiple myeloma, lymphoma, and primary bone malignancy ,4. Thus,There is no standard treatment for LCH. Treatment options may vary depending on the localization and extent of the disease. In unifocal EG, close observation of the patient may be preferred, as well as surgical excision, radiotherapy, and intra-lesional steroid administration. Systemic chemotherapy is recommended in multisystem involvement . The proIn conclusion, we present an extremely rare metachronous rib EG in an adult patient presenting with an interval of seven years. In this mild variant of LCH, surgical resection with clear margins has a favorable outcome without the need for additional adjuvant therapy. Metachronous tumors may develop in isolated unifocal bone EGs, and long-term follow-up is mandatory.


Pleomorphic xanthoastrocytoma is a rare brain tumor of WHO grade II designation. This case report describes the author's experience with the discovery of the tumor, living with the tumor, and eventual treatment and aftermath of how it affected her life and her understanding of the medical system. Pleomorphic xanthoastrocytoma (PXA) is a type of astrocytoma that is very rare and accounts for <1% of all astrocytomas [DiagnosisI was diagnosed with a brain tumor in my left ventricle incidentally in 2014 at the age of 20, after volunteering to do brain magnetic resonance imaging (MRI) in college for a professor studying Alzheimer’s disease. My only symptom was consistent “déjà vu” moments occurring about three to four times daily. At the time, my professor quickly got me an appointment with a neurologist who accepted the school insurance and I had a more thorough MRI done. The resulting images showed what neurologists assumed was a meningioma that was about 2.0 x 2.1 x 2.2 cm^3 in volume in my left ventricle presumably pressing on my hippocampus, explaining those symptoms. They referred me to get an electroencephalogram (EEG), where I was officially diagnosed with seizure activity. I was prescribed levetiracetam 500 mg twice a day (BID), which decreased these events to about one to two times daily. The dosage was increased to 750 mg BID and the symptoms were well controlled as long as I was compliant with the medication.Over the next two years, I continued to take levetiracetam without any change in symptoms and continued to get an MRI every six months to monitor the status of the tumor. I also established care with a neurosurgeon to be prepared for any advances in symptoms or changes in the status of the tumor. During my gap year between graduating and applying to medical school, I started developing additional symptoms that resembled absence seizures that my neurologist labeled as complex partial seizures associated with amnesia based on my description of the experiences, despite increasing my levetiracetam dosage to 1000 mg BID.SurgeryThus, in the winter of 2017, in between my medical school interviews, I had a left temporal-parietal craniotomy for the left posterior hippocampal brain tumor. The final pathology showed a pleomorphic xanthoastrocytoma of WHO grade II with BRAF v600E mutation.Pre-surgery PreparationI was blessed to have a very comprehensive team of providers to discuss my case and provide me care. My professor, neurologist, and neurosurgeon set up a meeting with the neurologist and neurosurgeon at the facility that I ultimately chose to pursue the surgery at. I had to have another MRI and an ophthalmological exam as well prior to the surgery. My surgeon was also very understanding of my worries about having this surgery done right before one of my medical school interviews. He planned the surgery strategically during a two-month gap between my scheduled interviews at the time.Post-operative ComplicationsPost-operatively, I have a full tonic-clonic seizure including tongue biting that resulted in a full day of neuro ICU stay due to post-operative inflammation from the surgery. Additionally, I had some minor difficulty with reading certain words, spelling, and verbalizing my thoughts coherently. The levetiracetam dose was increased to 1500 mg BID post-operatively. Despite these small issues, I had a relatively unremarkable stay and a smooth recovery. I was discharged on post-operative day two after the drain was removed with six days of enoxaparin and levetiracetam decreased to 1000 mg BID.Follow-upOver the next years, I continue to have MRI and follow up every six months including during medical school. After the seizure, I was deemed unfit to drive for two years by my neurologist, but my classmates and medical school were very supportive in assisting me with accommodations. I continued to slowly decrease my levetiracetam dosage with my neurologist over the course of the next four years. Currently, I am not taking any medication for seizures and have been seizure-free for five years. I still have the scar from the craniotomy, and the only irritation it gives me is flaking skin in dry environments.PXA is a rare WHO grade II CNS tumor of astrocytic origin with a favorable prognosis, especially if resected -3. In faWhile the majority of PXA tumors have a favorable diagnosis, there has been a recent addition to the 2016 World Health Organization classification of tumors in 2016 involving anaplastic pleomorphic xanthoastrocytoma (A-PXA) as grade III glial tumors . The stuThe overall survival rate of those with PXA is very favorable, with those that have a BRAF mutation having a better prognosis than those without . The fivAdditional learning pointsAs a fairly independent first child of first-generation immigrants who did not speak much English, my first instinct after that meeting with my professor was immediately “What do I do next?” In the following months, I learned more about how to navigate the medical system from the patient side more than I learned in even my third year of medical school. From being put on a two-month waitlist to see a neurologist until my professor made some calls for me, to calling my insurance company (thankfully I was still under 26 and covered by my parent’s company insurance) to see which primary care providers, MRI facilities, and neurosurgeons were considered “in-network,” I was able to understand the frustration and confusion many of my patients currently feel today. Since I am the only person in my family with any sort of even a science background, I needed to translate my diagnosis to my parents while also struggling to understand what was happening myself.The moment of the diagnosis also took away some of my independence, now that I could no longer drive for an indefinite amount of time, and forced me to rely on others in a way that I was definitely extremely uncomfortable with. Similarly, I was extremely worried about the financial burden my diagnosis would cause my parents. It was then that I learned about and was very thankful for the “pre-existing conditions” clause that would require insurance companies to cover my condition even when my father changed companies for work. Ultimately, when I saw the final sticker cost of my surgery and the short stint in the ICU, I was extremely grateful to be in a situation where I was under 26 years old and still covered under my parents’ insurance. If I had not gotten that MRI for the Alzheimer’s study and thus gotten diagnosed much later due to my initial extremely nonspecific symptoms, I may not have been so lucky!It was also through this experience that I was able to be more vulnerable to others. As a young person, I used to think I was invincible. Yet, with a single diagnosis, I was suddenly rendered unable to drive. Ultimately, the main impact of this tumor was the limitations it placed on my driving ability. While I was in college and during my gap year, I lived in an area with robust public transportation where driving a vehicle was unnecessary and even possibly a nuisance. However, my first two years of medical school were only possible with the supportive administration and my classmates because it was in an area where having a car was almost an absolute necessity for transportation. Thankfully, I lived with classmates and the administration assigned them similar schedules to mine. By the third year, these same friends basically gave me the driving lessons I so desperately needed due to my four-year hiatus from behind the wheel. Thus, when I was cleared to drive, I was finally able to regain my independence and start my rotations that were located throughout the state.A brain tumor is a life-changing diagnosis that is often devastating. I was lucky enough to have not only an incredibly incidental find before any obvious symptoms, but also a supportive care team, family, classmates, and school administration who supported me through not only the diagnosis but its treatment and the aftermath. While a PXA is a WHO grade II CNS tumor, it is a relatively benign tumor with a five-year survival rate of over 75%. The best treatment for PXA is complete resection, but those with A-PXA may need additional radiotherapy, systemic therapies, or other novel treatment strategies.


Intraosseous lipomas are rare bone lesions that can affect any part of the skeleton. In the calcaneum, they are, generally, asymptomatic, but in some cases, patients may complain of pain, swelling or tenderness. Well-conducted radiography and MRI examinations can lead to an accurate diagnosis. In most cases, patients could benefit from conservative means of treatment, but in long-lasting symptomatic cases, surgical treatment may be a good option. The purpose of this article is to increase clinicians’ awareness of this lesion as a possible cause of heel pain and to describe a case of a symptomatic intraosseous lipoma of the calcaneum who underwent curettage and bone cement filling after failure of conservative treatment. The first indexed description of an intraosseous lipoma dates back to 1976 ,3. At prThe most common location is the lower limb, usually within the calcaneum, femur, tibia, and fibula, but intraosseous lipomas may occur in any bone of the body ,7,8,9.Most cases are asymptomatic and are usually incidentally discovered on radiographs performed for unrelated disorders . HoweverThese lesions are frequently misdiagnosed . ClinicaThe decision whether to treat these lesions surgically or the preference towards a conservative treatment is controversial ,13,15. TIn this report, we describe a case of a symptomatic, painful, intraosseous lipoma of the calcaneum who underwent curettage and cement filling after failure of several months of conservative treatment.A 40-year-old male engineer, former professional rugby player, was referred to our clinic with a left heel inflammatory pain that was worsening during jogging or trailing. The symptoms started seven months before and the patient presented to a regional local hospital for investigations where a plain radiography was performed and a simple bone cyst diagnosis was suspected. Patient was recommended a break from physical activity for six months and non-steroid anti-inflammatories drugs (NSAIDS) to ameliorate pain. The pain increased gradually and he started to complain of swelling. The pain was hardly controlled with NSAIDS and non-morphinic analgesics.Our clinical examination revealed a mild tenderness in the posterior foot, including the ankle and the heel, without evidence of a palpable mass. Ankle and subtalar joint mobilities were limited. The laboratory blood tests and urine analysis results were normal.Radiography revealed a benign-appearing bone lesion of 16 × 19 mm within the anteroinferior part of the calcaneum which was well defined, radiolucent, almost entirely homogeneous with a small central sclerotic focus-“Cockade sign” ,14, descSurgery was performed and a direct lateral approach to the calcaneum was chosen. The saphenous nerve and the long peroneus tendon were reclined superiorly and distally a. A boneThe histopathological exam  and immuPostoperatively, the pain subsided completely. The patient was discharged the second day after the surgery. Total weight bearing was allowed without crutches. No cast or brace was applied. Rehabilitation was conducted for two weeks in order to encourage full ankle mobilities.In the follow-up, the patient was evaluated every six weeks during the first three months and every six months until the first two years after surgery. Starting from the first postoperative consultation at six weeks, the ankle joint mobilities were normal, the patient was painless, and radiological examination showed no modifications. At the time of the last follow-up at 24 months, no modification and no recurrence were observed a,b and tIntraosseous lipomas are very rare lesions ,6. ApproThe average age at the time of diagnosis ranges from 30 to 60 years  and mostThe radiological appearance of intraosseous lipomas is often uncharacteristic and can be confused easily with other bone lesions such as chondroid tumors, aneurysmal bone cysts, fibrous dysplasia, bone infarcts and liposclerosing myxofibrous tumours . MoreoveUsually, diagnosis is easily-defined when MRI and CT scans are performed . If radiConservative treatment consisting of clinical and radiological follow-up is advocated in asymptomatic patients without impending pathologic fracture or suspicion of malignancy ,15. In oThere is no consensus regarding the surgical treatment of symptomatic bone lipomas, nor is there consensus concerning when a surgical procedure should be performed. In case of failure of conservative treatment, common surgical options include curettage with or without filling of the cavity with different substitutes ,11,21. RThis study is a case report and it was subject to inherent limitations and biases. The study was retrospective, the technique of reconstruction was not randomized, and the preference of the surgeon may have contributed to a selection bias. However, bone lipomas are rare lesions, and we report a case with a good follow-up when the literature convinces us about the current controversy regarding the surgical treatment and time when such treatment should be performed.Despite the low prevalence of lipomas of the calcaneum, physicians need to be aware about this lesion that can cause heel pain. Intraosseous lipomas are more often asymptomatic, but patients can present chronic heel pain, local inflammation signs, and even edema. Being able to diagnose an intraosseous lipoma through MRI or CT scan imaging and offering the best treatment options is essential for the health of future patients. It is our opinion that bone cement is cheap, easy to handle, and common enough to be easily available at most hospitals, making it a good option to fill lytic lesions after the curettage of bone lipomas while consenting immediate full-weight bearing.


Hemangiomas are benign tumors that are very common in the head and neck region. However, intravascular hemangiomas are very rare. Hemangiomas are classified as capillary, cavernous or mixed tumors according to the proliferating cells. Ultrasound, computed tomography, MR imaging and angiography are useful diagnostic tools and are generally required when planning surgical treatment. Definitive diagnosis is established by histopathological examination, differentiating hemangiomas from other vascular tumors or malignancies. We present a rare case of capillary hemangioma protruding from the external jugular vein. In our patient, the tumor was totally removed under local anesthesia. No complications and no recurrence were observed in the following two years.Intravascular tumors can present as neck masses and a definitive diagnosis is made by histopathological examination. Imaging tools provide important information about anatomy, the extent of the tumor, and for surgical planning. Hemangiomas are benign lesions characterized by a neoplastic proliferation of vascular endotelial cells; they are common vascular tumors in the head and neck region although their appearance as primitive intravascular proliferations is very unusual. Intraluminal hemangiomas must be differentiated from other vascular tumors and malignancies. In this article we present the case of a rare intravascular centrolobular capillary hemangioma protruding from the external jugular vein. A 69-year-old woman with an unremarkable medical history came for an examination on a painless mass on the left side of her neck, which had slowly and gradually grown in size over the two previous months. She had no history of previous trauma or catheter insertions in the neck. On physical examination everything was normal except for the presence of a left-sided mobile and non-pulsating mass along the course of the external jugular vein (EJV) without any inflammatory symptoms. An echography scan was performed showing a 15 x 8 mm intraluminal lesion, with intrinsic arterial and venous blood flow on the doppler scan  without Vascular anomalies are soft tissue lesions with congenital errors in vascular development. According to the classifications of Mulliken and Glowacki 1982) (982 (1), Intrinsic vascular malformations arising from the wall of the EJV are very rare. In English literature we found only seven articles describing intravascular tumours arising from the EJV ,5-10. The first article describing a vascular intrinsic tumour of the EJV appeared in 1967 where the author wrote about the utility of venography for its diagnosis . SartescWith MRI, intravascular haemangiomas typically show intermediate signal intensity on T1-weighed images, very high signal intensity on T2-weighed images, and variable enhancement after intravenous gadolinium administration. Although ecography, computed tomography, and MR imaging give important details about anatomy and surgical planning, the definitive diagnostic tool is the histopathological examination -9. In our patient, after ecography and a CT scan we performed an excisional biopsy under local anesthesia, ligating the EJV on both sides, and confirmed the histophatologic diagnosis and the total removal of the tumor. The histologic differential diagnosis of intravenous lobular capillary hemangioma includes inflammatory angiomatous nodules, intravascular papillary endothelial hyperplasia, intravenous atypical vascular proliferation, pseudo-Kaposi sarcoma, angiosarcomas, intravascular fasciitis, and an organized thrombus . We did not observe postoperative complications or recurrence after two years. In previous cases no recurrence has been reported after the total removal of the tumor. Intravascular tumors can appear as neck masses and the definitive diagnosis is made by histopathological examination considering the importance of imaging tools to provide information about anatomy, the extent of the tumor, and for surgical planning.The authors declared no conflicts of interest with respect to the authorship and/or publication of this article. The patient has consented to the submission of the case report for submission to the journal.


A 60-year-old man presented with progressive disturbance of consciousness. His father had died of malignant lymphoma, while his mother and sister died of acute leukemia. Magnetic resonance imaging (MRI) revealed multiple high-intensity lesions in the bilateral cerebral hemispheres on diffusion-weighted images. Serum soluble interleukin 2 receptor was 5,640 U/mL. Screenings of blood antibodies known to rise in autoimmune diseases were all normal. Cerebrospinal fluid examinations demonstrated slight elevation of protein and glucose, while the oligoclonal band and myelin basic protein were not elevated. Biopsies of bone marrow and random skin did not show any malignant features. His consciousness gradually deteriorated over a week, with lesions in his right frontal, left temporal, and bilateral parietal lobes shown to be growing. Therefore, open brain biopsy was performed, and one block of the right frontal lesion was harvested. Histological examination revealed atypical large cells only in the capillaries. Although immunohistochemical examinations showed positive staining for CD20, they were negative for CD3. Histopathological diagnosis was intravascular large B-cell lymphoma. After undergoing six cycles of intravenous chemotherapy with rituximab, cyclophosphamide, doxorubicin, and prednisone, his consciousness and neurological symptoms improved, and he appeared to achieve remission. Two years later, there have been no apparent recurrences, and the brain lesions have disappeared. Intravascular large B-cell lymphoma (IVLBCL) is a rare subtype of extranodal diffuse large B-cell lymphoma that is characterized by selective growth of lymphoma cells within the lumina of small blood vessels -3. IVLBCA 60-year-old man had a history of ischemic stroke and was undergoing dialysis because of unexplained chronic kidney disease. There was a history of cancer in his family, with his father dying of malignant lymphoma and his mother and sister dying of acute leukemia. He was transported to our hospital due to subacute progressive disturbance of consciousness. Magnetic resonance imaging (MRI) revealed multiple small high-intensity lesions in the bilateral cerebral hemispheres on diffusion-weighted and fluid-attenuated inversion recovery images Figures -1F. ThesA bone marrow biopsy was performed, but the first pathological results did not show any malignant features. In addition, a random skin biopsy did not find any atypical lymphocytes. In spite of the antiplatelet therapy, his consciousness gradually deteriorated over a week. A subsequent MRI showed increases in the high-intensity lesions in the right frontal, left temporal, and bilateral parietal lobes on T2-weighted images Figures -2C. FurtThe surface of the lesion was covered by hemosiderin Figure . The sulHistological examination revealed atypical large cells only in the capillaries without any of these cells found around the parenchymal tissue Figures , 4B. ImmAfter diagnosis, the patient was treated with six cycles of intravenous chemotherapy with rituximab, cyclophosphamide, doxorubicin, and prednisone (R-CHOP). His consciousness and neurological symptoms improved, and he appeared to achieve remission. Two years later, there have been no apparent recurrences, and the high-intensity lesions of the MRI have almost completely disappeared Figure -2I.BCL6 gene in IVLBCL has also been reported [IVLBCL is now divided into three types  according to the clinical features . The clareported .Although biopsies were performed for each of the affected organs, they were initially only performed for the bone marrow or skin. According to the report, 67% patients were diagnosed with IVLBCL by bone marrow biopsy. . Random Matsue et al. presented MR images that showed abnormal findings in 32 86.4%) of 37 IVLBCL patients . There a6.4% of 3As IVLBCL diagnosis is difficult due to the non-specific symptoms, IVLBCL is often diagnosed by autopsy. However, if it were possible to diagnose IVLBCL, chemotherapy for IVLBCL could potentially be effective ,6. RajyaIn our case, several factors indicated neoplastic brain lesions, such as a family history of cancer, atypical cerebral infarction findings on MRI, lesions that appeared to be growing, deterioration of the patient’s consciousness over a short period, and elevation of the serum soluble interleukin-2 receptor. Although bone marrow and random skin biopsies were promptly performed, the first biopsy results did not show any malignant features. At that time, IVLBCL was given as one of differential diagnosis. It could be pathologically undiagnosed when only using a small specimen obtained via a stereotactic needle biopsy, as IVLBCL diagnosis requires proof of atypical cells in small blood vessels. Therefore, in our case, we planned to perform an open brain biopsy, which could select harvest both small and large size specimens. Even though our initial small specimen revealed no abnormalities, the large size of the specimen subsequently made a diagnosis. As has been reported in other studies, an early pathological diagnosis along with an early initiation of chemotherapy can make it possible for patients to achieve remission for IVLBCL. Our current results show that open brain biopsy can be a useful choice for IVLBCL diagnosis when there are brain lesions present and other biopsy methods cannot definitively determine a correct diagnosis.


The objective of this report is to present a rare case of a recurrence after 20 years of retroperitoneal dedifferentiated liposarcoma after surgical resection and to discuss the lessons learned from this rare phenomenon for patients management and understanding the behavior of these aggressive tumors. A 75-year-old woman presented with recurrent retroperitoneal dedifferentiated liposarcoma who had undergone a surgical resection 20 years earlier and had no evidence of disease on frequent follow-ups during that period. The histopathologic examination revealed different morphologic characteristics between the initial and recurrent presentations. The fluorescence in situ hybridization showed amplification of the mouse double minute 2 homolog (MDM2), a regulator of p53 gene on chromosome 12q15, and positive cyclin-dependent kinase 4 (CDK4) immunostain.Liposarcoma long-term recurrence is a challenging surgical disease to provide the best survival outcome. Incomplete resection could explain the recurrence in anatomic locations where the lesions are intermixed with the neighboring adipose tissue. However, dedifferentiated liposarcoma can rarely recur after 20 years. The molecular transformation and the survival analysis of these tumors predict certain behaviors. The refraction for radiation therapy in our case and the mixed morphology provide some insight into the biology and the clinical management for these aggressive tumors.  Liposarcomas are rare malignant tumors that are derived from mesenchymal cells, specifically adipocytes, and are classified as the most common soft tissue sarcoma making up about 20% of all types of adult sarcomas . As defiThe first reported case of a retroperitoneal liposarcoma in the literature was published in 1949 . PatholoThe gold standard of care for retroperitoneal liposarcoma is aggressive surgical management, however, radiation or chemotherapy are also possible treatment options . In addiFrom a clinical perspective, a retroperitoneal liposarcoma recurrence following the initial treatment plan is not uncommon, as disease progression within six months to two years has been frequently noted . ThroughWe report a case of a 75-year-old female with a past medical history of retroperitoneal liposarcoma, however, although resection was done on this patient, a new retroperitoneal liposarcoma regrowth was found 20 years post-operatively, displaying this rare phenomenon. We discuss the challenges of managing patients with long-term recurrence of retroperitoneal liposarcomas following complete surgical resection in order to share our experience with this rare phenomenon and to explore the biology behind the recurrence to improve the understanding of these aggressive tumors.A 75-year-old woman with a past medical history of retroperitoneal liposarcoma and history of underlying malnutrition and hypoalbuminemia who underwent radical resection in 2000 presented in 2020 with abdominal discomfort and a CT scan showed evidence of retroperitoneal tumor mass Figure . There was a large left abdominal/retroperitoneal mass measuring approximately 20.5 x 22.8 x 26.1 cm  at an outside institution, but cancer was refractory and was therefore admitted to our institution for surgical resection of a new left-sided abdominal mass. The patient underwent a complex operation including the following elements: exploratory laparotomy, lysis of adhesions, radical resection of abdominal mass, total gastrectomy, distal pancreatectomy and splenectomy, roux-en-Y reconstruction with neo-gastric conduit obstruction, partial colectomy, splenic flexure immobilization, appendectomy, liver wedge resection, omentectomy, and feeding jejunostomy placement. Histopathologic examination showed undifferentiated spindle cell sarcoma in the majority of the mass with dedifferentiated retroperitoneal liposarcoma components in some areas Figure . MitoticPost-operatively, the patient was transferred to the surgical intensive care unit and required vasopressors and ventilator support. Due to her underlying malnutrition and hypoalbuminemia, the patient had significant fluid shifts leading to bilateral pleural effusions which necessitated bilateral chest tubes and a tracheostomy. She was successfully weaned off these and ultimately required hemodialysis for management of acute kidney injury and metabolic encephalopathy. Once the patient was hemodynamically stable with significantly improved mental status, the patient was transferred to acute rehab, had her trach removed, and was tolerating a diet.We presented a rare case of long-term recurrence of retroperitoneal dedifferentiated liposarcoma (DDL) after complete surgical resection in a 75-year-old woman. The implication of this rare phenomenon is of clinical and biological significance, as it poses a challenge from the management point of view, but it also sheds light on the biology of these aggressive tumors. Genetically WDL and DDL are similar, but DDL has more genetic alterations, specifically 6q23 and 1p32 co-amplifications . FurtherThe overall rate of local recurrence of DDLs is 80% within five years of a complete surgical resection . In factAlthough surgical resection currently remains the optimal treatment option for patients, for those suffering from unresectable or recurrent growths, chemotherapy remains a viable option as well. In fact, the median survival rate was found to be 29 months (95% CI 24-40 months) for DDL patients following chemotherapy, demonstrating the success of chemotherapy as a treatment option . AlthougThe treatment of choice for DDLs is the complete surgical resection of cancer which, in turn, leads to an increase in overall patient quality of life. However, patients may undergo multiple surgical operations for these tumors due to the recurrence. Parmeggiani et al. mentioned in their case report that their particular patient had to undergo three complete surgical excisions of cancer in a span of three years to increase the overall patient’s survival . This prOur patient’s tumor recurred after 20 years, which is very unlikely to be due to incomplete resection because, within a five-year period, it’s noted that 80% of all DDL recur locally . This diIn summary, retroperitoneal liposarcoma long-term recurrence is a challenging aspect of the disease from a surgical point of view to provide the best outcome and to improve survival. Incomplete resection is a possibility in some anatomic locations, especially if the lesions are intermixed with neighboring adipose tissue. However, dedifferentiated liposarcoma can rarely recur after 20 years. The molecular transformation and the survival analysis of these tumors predict certain behaviors. The refraction for radiation therapy in our case and the mixed morphology provide some insight into the biology and the clinical management for these aggressive tumors.


We report the case of a craftsman who developed a rapidly progressive subcutaneous emphysema of his forearm after a minor stab injury into the palm of his hand. Based on our case report we discuss differential diagnosis and management of acute subcutaneous emphysema. Subcutaneous emphysema (SE) is defined by an accumulation of gas along fascial planes in the subcutis. The combination of an acute swelling with crepitus on physical examination and a subcutaneous gas collection on radiographs points to a necrotizing soft tissue infection (NSTI) . With anA 56-year-old flooring installer sustained a wood splinter injury in his left palm while examining the processed wooden floor surface. He immediately removed the splinter (0.5 × 4 cm) himself. About 20 min later he noticed an increasing swelling with crackling sensations of the affected hand. Within minutes, this spread up to his forearm. The patient was admitted to our clinic 6 h after the injury in good general condition, vital signs were stable and no fever was present. On clinical examination, a distinct painless swelling from the hand to the distal forearm was present. Apart from mild perifocal redness and tenderness around the small penetration wound, there were no signs of infection and no skin changes . No painInterestingly, the same patient had suffered from emphysema of the same arm 7 years ago after a contusion of the left wrist without visible skin lesions or a fracture . The patOnly a few reports of non-infectious SE after minor skin lesions exist . The nonIn BSCE the period between the initial trauma and the development of the swelling and crepitus is significantly shorter than in NSTI . In contThe reason for the spontaneous emphysema without any obvious skin lesion from which our patient had suffered 7 years ago remains unclear. No other comparable case can be found in the current literature. An individual predisposition for subcutaneous emphysema can be discussed but is purely speculative.At an early stage, the distinction between an infectious SE and a BSCE can be difficult. We, therefore, recommend admission of patients with signs of a BSCE for close monitoring and re-evaluation. If in doubt, initiation of a broad-spectrum antibiotic therapy and immediate surgical exploration is imperative. Further imaging (e.g. MRI) should not delay surgery. Usually, limited local exploration of the initial wound or the most prominent swelling is sufficient to rule out an NSTI. Typical intraoperative findings of necrotizing fasciitis would be friability of the fascia, blunt dissection with minimal resistance (‘finger test’) and presence of grey-coloured fluid (‘dishwater fluid’) ,17. If n